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Respir Case Rep 2020;9(2): 38-42 DOI: 10.5505/respircase.2020.40316
OLGU SUNUMU CASE REPORT
38
Sol Jin1, Jin-Young Lee1, Jehun Kim
2
Since mid-December 2019, a novel coronavirus
(COVID-19) has spread to many countries around
the world, and the number of critically ill patients with
COVID-19 is increasing as the number infections
increase. The optimum treatment and prognosis of
the disease is still unknown. Here, we present the
clinical course and serial computed tomography of a
critically ill Korean patient with COVID-19. The pro-
gression of COVID-19 infection is fast and aggressive,
and no treatment protocol has yet been established.
Additional clinical data are required to determine
whether or not corticosteroid use is clinically benefi-
cial.
Key words: Coronavirus, viral load, corticosteroids,
pneumonia.
Yeni korona virüs (Covid-19) 2019 Aralık ortaların-
dan beri dünyada pek çok ülkede yayılmakta ve
COVID-19'lu ağır hasta sayısı, enfeksiyon sayısı art-
tıkça artmaktadır. Hastalığın optimum tedavisi ve
prognozu halen bilinmemektedir. Burada, COVİD-
19’lu Kore’li bir ağır hastanın klinik seyri ve seri bilgi-
sayarlı tomografi bulgularını sunduk. COVİD-19
enfeksiyonunun progresyonu hızlı ve agresif olup
henüz tedavi protokolü oluşturulmamıştır. Kortikoste-
roid kullanımının klinik yararı olup olmadığını belir-
lemek için ilave klinik verilere ihtiyaç vardır.
Anahtar Sözcükler: Korona virüs, virüs yükü, kortikos-
teroid, pnömoni.
1Department of Infectious Disease, Kosin University Gospel Hospi-
tal, Busan, South Korea
2Department of Pulmonology, Kosin University Gospel Hospital,
Busan, South Korea
1Kosin Üniversitesi Gospel Hastanesi, Enfeksiyon Hastalıkları
Servisi, Busan, Güney Kore
2Kosin Üniversitesi Gospel Hastanesi, Göğüs Hastalıkları
Servisi, Busan, Güney Kore
Submitted (Başvuru tarihi): 17.04.2020 Accepted (Kabul tarihi): 25.04.2020
Correspondence (İletişim): Jin-Young Lee, Department of Infectious Disease, Kosin University Gospel Hospital, Busan, South Korea
e-mail: [email protected]
RES
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RY
CA
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EPO
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Respiratory Case Reports
Cilt - Vol. 9 Sayı - No. 2 39
Since mid-December 2019, a novel coronavirus (COVID-
19) has spread to many countries around the world. On
March 11, 2020, the World Health Organization de-
clared a pandemic, identifying COVID-19 as a public
health emergency of international concern (1).
In the second week of March 2020, the number of con-
firmed cases in Korea passed the 8,000 mark, although
accurate counting was a difficult task being experienced
worldwide. The numbers of patients who are being cured
and discharged is increasing over time, although the
number of patients classified as critically ill with COVID-
19 that require ventilator or extra-corporeal membrane
oxygenator support is also gradually increasing. While
data on the epidemiology and clinical manifestations of
the disease is accumulating, clinical data on critically ill
patients is lacking. The clinical course changes in com-
puted tomography (CT) findings and cycle threshold (Ct)
values, which means that the cycle number at which the
fluorescent signal of the reaction crosses the threshold
can help to confirm the characteristics of the disease and
support the creation of a treatment plan for the patients.
To offer an overview of the clinical features of COVID-19
infection, we present a case of a patient in Korea.
CASE
A 78-year-old man with no remarkable past or family
medical history presented with chills, myalgia, cough and
sputum on February 28, 2020. The symptoms and clini-
cal course of the patient are presented in Figure 1.
Upper respiratory tract (URT) and lower respiratory tract
(LRT) specimens were collected from the patient. Naso-
pharyngeal and oropharyngeal swabs were collected for
URT, and sputum was used for the LRT specimen (2).
Quantitative real-time polymerase chain reaction amplifi-
cation was carried out using the AllplexTM 2019-nCoV
assay (Seegen, Seoul, Korea) (3). Ct values were checked
for the RNA-dependent RNA polymerase gene (R gene)
and E gene.
The patient was found to be COVID-19 positive (Ct value:
upper R gene, 19.86; upper E gene, 17.1; lower R gene,
21.92; lower E gene, 18.59) upon examination by the
public health center, and the patient was hospitalized in a
community hospital in Busan, Korea. On day 2, a chest
radiography revealed mild haziness in the left lower lobe,
and a chest CT revealed ground-glass opacities in the left
lower lobe (Figure 2). Shows the serial changes in chest
CT and radiography. The patient was started on lop-
inavir/ritonavir (Kaletra, AbbVie); 2 tablets (lopinavir 200
mg/ritonavir 50 mg) were given orally bid. On day 4,
fever and sputum persisted and loose stool started. Chest
radiography findings worsened, and the patient had a
fever of 39.0°. Accordingly, the ceftriaxone antibiotic was
started on day 5, and piperacillin/tazobactam and
levofloxacin were started on day 6. The patient showed
no improvement.
Figure 1: Clinical course of the patient. Ct values: cycle threshold value; URT: upper respiratory tract; LRT: lower respiratory tract; cefa: cephalosporin;
pip/taz: piperacillin/tazobactam; FiO2: fraction of inspired oxygen
The 2019 Novel Coronavirus Disease (COVID-19) causing Severe ARDS: Serial Computed Tomography Findingse | Lee et al.
40 www.respircase.com
On day 6, the patient was transferred to the hospital in
Busan, Korea. Upon presentation, he had no dyspnea,
but required oxygen supplementation via a nasal cannula
(2L/min). Vital signs: blood pressure, 148/88 mmHg;
pulse rate, 85 beats/min; respiratory rate, 13 breaths/min;
and body temperature, 38.3°C. Laboratory tests revealed
a white blood cell count (WBC) of 5,290/μL, lactate de-
hydrogenase (LDH) of 570 U/dL, and high sensitivity C-
reactive protein (Hs-CRP) of 14.8 mg/dL. Table 1 shows
the detailed blood test results. A follow-up chest CT
showed an increase in the extent of the multifocal
peribronchial ground grass opacity in bilateral lungs and
mild pleural effusion. A sustained dose of lop-
inavir/ritonavir was given and hydroxychloroquine and
antibiotics (meropenem 1g tid, vancomycin 1g bid,
levofloxacin 750 mg qd) were administered. Despite the
use of acetaminophen and non-steroidal anti-
inflammatory drugs (NSAIDs), the high fever persisted.
Although the test for influenza was negative, peramivir
was injected clinically. The patient had no underlying
disease, although a chest CT showed underlying pulmo-
nary fibrosis. Methylprednisolone (0.5 mg/kg) was admin-
istered from day 7 to day 9.
Subsequently, the patient’s dyspnea worsened and an
intubation was performed on day 9. A follow-up COVID-
19 test was positive on day 12 and Ct values were: upper
E gene, 25.43; R gene, 7.45; lower E gene, 18.96; R
gene, 20.69.
Serial laboratory tests and a chest radiography were per-
formed. The chest radiography revealed diffuse consoli-
dation in bilateral lungs. On day 15, a follow-up chest
CT showed diffuse ground-grass opacity, consolidation in
the bilateral lungs and increased interstitial thickening.
The COVID-19 test was still positive (Ct values: upper R
gene, 27.07; upper E gene, 25.95; lower R gene, 21.83;
lower E gene, 20.36).
A tracheostomy was performed and injections of
methylprednisolone (1 mg/kg) were started on day 16.
After starting methylprednisolone, the patient’s oxygen
demand decreased and his chest radiography findings
improved. (Figure 3) The clinical situation was improved
through the use of a higher dose of corticosteroid; how-
ever, on day 21 the methylprednisolone was stopped due
to gastrointestinal bleeding. Close monitoring and opti-
mum supportive care were continued, with measurements
of the Ct value.
Figure 2: Radiologic findings of the patient. Chest radiography on day 2
(A), chest radiography on day 6 (B), Chest radiography on day 15 (C),
chest computed tomography on day 2 (D and G), computed tomogra-
phy on day 6 (E and H), chest computed tomography on 15 (F and I)
Figure 3: Chest radiography after methylprednisolone administration.
Chest radiography on day 16 (A), chest radiography on day 18 (B),
chest radiography on 20 (C)
DISCUSSION
As the number of COVID-19 infections increase world-
wide, the number of critically ill patients with COVID-19
is also increasing. COVID-19 infection is particularly risky
for older patients and those with underlying diseases (4).
The patient in the present study was otherwise healthy,
with no specific past history, aside from the 78 years of
age. At the time of the first diagnosis, CT showed mild
pneumonia, while the peribronchial pneumonic consoli-
dation gradually increased on follow-up CT. Pneumonia
progressed rapidly in a short period.
The progression of pneumonia was apparent on a serial
chest radiography, although it was difficult to determine
the exact degree, and so a follow-up chest CT was per-
formed, showing far more severe lesions than the chest
radiography.
Respiratory Case Reports
Cilt - Vol. 9 Sayı - No. 2 41
Table 1: Laboratory test results of the patient
Variables Day 2 Day 6 Day 8 Day 10 Day 12 Day 15 Day 17 Day 19
WBC, /μL 5400 5290 4560 11860 7870 8790 5520 5500
Segment neutrophil, % 66.0 84.9 91.1 96.5 80.0 78.0 74.0 81.0
Lymphocyte, % 2.4 7.5 6.1 1.8 4.0 4.0 7.0 5.0
Eosinophil, % 0.1 0.0 0.0 0.0 0.0 1.0 0.0 0.0
Hemoglobin, g/dL 15.0 13.3 14.3 12.6 14.0 12.3 9.8 10.5
Platelets, ×103/μL 147 104 100 125 79 96 96 117
BUN, mg/dL 16.5 20.8 19.8 29.2 38.6 39.1 62.3 73.9
Creatinine, mg/dL 0.99 0.89 0.78 0.92 0.77 0.68 1.51 1.1
Total bilirubin, mg/dL 0.67 0.77 1.14 1.79 2.79 2.81 2.19 6.00
AST, U/L 19 34 49 34 33 52 32 56
ALT, U/L 20 20 29 27 26 28 25 46
LDH, U/dL 207 570 865 852 885 441 535
Sodium, mEq/L 135.0 125.1 134.2 132.3 136.0 138.0 139.5 142.1
Potassium, mEq/L 4.50 4.17 4.48 4.78 4.27 4.30 4.01 3.96
Chloride, mEq/L 101.0 98.9 104.0 104.5 102.0 102.6 102.6 104.7
Total protein, g/dL 6.8 6.2 5.9 5.4 5.1 5.2 5.4 5.9
Albumin, g/dL 4.1 3.3 3.5 2.8 2.7 2.2 2.2 2.9
Hs-CRP, mg/dl 7.43 14.80 18.30 21.54 20.69 23.76 21.60 4.48
Pro-calcitonin, ng/mL 0.392 0.378
PT, sec 11.4 13.8 17.5 12.8 13.5 14.0 14.1 14.1
PT INR 1.04 1.04 1.41 0.94 1.01 1.06 1.07 1.07
Troponin i 14 370 151 107 72
WBC: white blood cell; BUN: blood urea nitrogen; AST: aspartate aminotransferase; ALT: alanine aminotransferase; LDH: lactate dehydrogenase;
Hs-CRP: high sensitivity C-reactive protein; PT: prothrombin time; INR: international normalized ratio
Another unusual finding was that the patient’s oxygen
demand was not as large as would be expected based on
the chest CT images. On day 6, upon his presentation to
our hospital, the patient had no complaints of dyspnea,
and oxygen saturation was 92~98% with nasal oxygena-
tion of 2L. On day 15, chest CT showed damage to the
entire lung because of the pneumonia, but the fraction of
inspired oxygen on ventilator was 0.5~0.6.
In reports concerning the laboratory tests in early stages
of the disease, lymphocytopenia appears to be a negative
prognostic factor (4). Furthermore, highly elevated LDH
and CRP levels are associated with disease severity (5),
and the patient in the present study showed similar char-
acteristics (Table 1).
Similar to the influenza virus, the amount of COVID-19
output was large in the early phase, and it was confirmed
that the virus output of symptomatic and asymptomatic
people was similar (6,7). In this case, comparing the Ct
values at the time of the first CT (day 2) and at the time of
the last CT (day 15) revealed pneumonia to be more
severe in the CT performed later, although the viral load
decreased. It was thus considered that the viral load was
not related to the patient's lung condition or the severity
of the infection.
In Korea currently, lopinavir/ritonavir and hydroxychloro-
quine are being administered for the treatment of
COVID-19 (6), with antibiotics administered together with
both drugs, considering the possibility of bacterial pneu-
monia. However, it is questionable whether lop-
inavir/ritonavir and hydroxychloroquine are helpful. While
they may help lower the concentrations of the virus, they
have not prevented the rapid clinical progression. These
results are in part consistent with the randomized con-
trolled trials in China comparing the lopinavir/ritonavir
group with a standard care group (8). Despite the medi-
cation, the patient’s fever persisted and the pneumonia
The 2019 Novel Coronavirus Disease (COVID-19) causing Severe ARDS: Serial Computed Tomography Findingse | Lee et al.
42 www.respircase.com
progressed. After peramivir was administered for approx-
imately 6 days, the fever improved.
Although, intravenous glucocorticosteroids were com-
monly used in patients with severe Middle East respiratory
syndrome or severe acute respiratory syndrome, their
effects remain controversial, and their efficacy for the
treatment of COVID-19 is as yet undetermined (9). Inject-
ing methylprednisolone (0.5 mg/kg) on days 7–9 resulted
in no significant changes, while clinical improvement was
noted after injecting methylprednisolone (1 mg/kg) on
day 15. It is not known exactly what it was that affected
the clinical course, but these results may derive from the
dose of methylprednisolone or the timing of the disease
progression. More data on methylprednisolone will be
needed in the future.
CONCLUSION
Severe COVID-19 infection proceeds rapidly, according
to the clinical finding and chest CT findings, although no
effective drug has yet been identified. In such situations,
the use of glucocorticosteroids may be clinically useful.
The number of patients continues to increase worldwide,
while data on the treatment and prognosis of the disease
are still insufficient. Further research is warranted in the
future.
CONFLICTS OF INTEREST
None declared.
AUTHOR CONTRIBUTIONS
Concept - S.J., J.Y.L., J.K.; Planning and Design - S.J.,
J.Y.L., J.K.; Supervision - S.J., J.Y.L.1, J.K.; Funding -;
Materials -; Data Collection and/or Processing - S.J.;
Analysis and/or Interpretation - S.J., J.K.; Literature Re-
view - J.K.; Writing - S.J.; Critical Review - J.K., J.Y.L
YAZAR KATKILARI
Fikir - S.J., J.Y.L., J.K.; Tasarım ve Dizayn - S.J., J.Y.L.,
J.K.; Denetleme - S.J., J.Y.L., J.K.; Kaynaklar -; Malzeme-
ler -; Veri Toplama ve/veya İşleme - S.J.; Analiz ve/veya
Yorum - S.J., J.K.; Literatür Taraması - J.K.; Yazıyı Yazan -
S.J.; Eleştirel İnceleme - J.K., J.Y.L
REFERENCES
1. Gorbalenya AE, Baker SC, Baric RS, de Groot RJ, Dros-
ten C, Gulyaeva AA, et al. The Species Severe Acute
Respiratory Syndrome-Related Coronavirus: Classifying
2019-nCoV and Naming it SARS-CoV-2. Nat Microbiol
2020; 5;536–44. [CrossRef]
2. World Health Organization. Laboratory Testing for 2019
Novel Coronavirus (2019-nCoV) in Suspected Human
Cases: access date: 19 March 2020. Place of access:
https://www.who.int.
3. Guidlines for the Laboratory Diagnosis of 2019 Novel
Coronavirus(2019-nCoV) in Korea. 1 ed: Central for
Disease Control; 2020. [CrossRef]
4. Yang X, Yu Y, Xu J, Shu H, Xia J, Liu H, et al. Clinical
course and outcomes of critically ill patients with SARS-
CoV-2 pneumonia in Wuhan, China: a single-centered,
retrospective, observational study. Lancet Respir Med
2020; pii: S2213-2600(20)30079-5. [CrossRef]
5. Singhal T. A Review of Coronavirus Disease-2019
(COVID-19). Indian J Pediatr 2020; 87:281-6. [CrossRef]
6. Kim JY, Choe PG, Oh Y, Oh KJ, Kim J, Park SJ, et al.
The First Case of 2019 Novel Coronavirus Pneumonia
Imported into Korea from Wuhan, China: Implication for
Infection Prevention and Control Measures. J Korean
Med Sci 2020; 35:e61. [CrossRef]
7. Zou L, Ruan F, Huang M, Liang L, Huang H, Hong Z, et
al. SARS-CoV-2 viral load in upper respiratory specimens
of infected patients. N Engl J Med 2020; 382:1177–9.
[CrossRef]
8. Cao B, Wang Y, Wen D, Liu W, Wang J, Fan G, et al. A
trial of Lopinavir-Ritonavir in adults hospitalized with se-
vere Covid-19. N Engl J Med 2020; 382:1787-99.
[CrossRef]
9. Russell CD, Millar JE, Baillie JK. Clinical evidence does
not support corticosteroid treatment for 2019-nCoV lung
injury. Lancet 2020; 395:473-5. [CrossRef]
Respir Case Rep 2020;9(2): 43-46 DOI: 10.5505/respircase.2020.98360
OLGU SUNUMU CASE REPORT
43
Tayfun Kermenli1, Cebrail Azar
2
One of the potential complications of acupuncture is
pneumothorax, caused by the entry of the needle to
the visceral pleura and damaging the lung paren-
chyma. Our patient suffered a bilateral pneumotho-
rax and sudden respiratory failure. Our intention in
this study is to offer a reminder of this rare and life
threatening complication with this case report. A 51-
year-old male patient presented to the emergency
department with dyspnea after acupuncture treatment
at a hotel spa center. A physical examination re-
vealed no respiratory sound in either lung. A chest X-
ray revealed a bilateral pneumothorax. The patient
was treated with a bilateral tube thoracostomy.
Key words: Iatrogenic, pneumothorax, acupuncture,
tube thoracostomy, chest tube.
Akupunkturun komplikasyonlarından biri, iğnenin
viseral plevraya ulaşması ve akciğer parankimine
zarar vermesi sonucu oluşan pnömotorakstır. Acil
servise başvuran hastamızda bilateral pnömotoraks
ve ani solunum yetmezliği vardı. Bu olgu sunumu ile
nadir görülen ve hayatı tehdit eden komplikasyonu
hatırlatmayı amaçladık. Elli bir yaşında erkek hasta,
bir otel spa merkezinde akupunktur tedavisi sonrası
acil servise nefes darlığı şikayeti ile başvurdu. Fizik
muayenesinde her iki akciğerde de solunum sesi
alınamadı. Çekilen akciğer grafisinde bilateral pnö-
motoraks saptandı ve hasta bilateral tüp torakostomi
ile tedavi edildi.
Anahtar Sözcükler: İyatrojenik, pnömotoraks, aku-
punktur, tüp torakostomi, göğüs tüpü.
1Department of Thoracic Surgery Clinic, Medicalpark Elaziğ Hos-
pital, Elazığ, Turkey
2Department of Chest Diseases Clinics, Medicalpark Elaziğ Hospi-
tal, Elazığ, Turkey
1Medicalpark Elazığ Hastanesi, Göğüs Cerrahisi Kliniği,
Elazığ
2Medicalpark Elazığ Hastanesi, Göğüs Hastalıkları Kliniği,
Elazığ
Submitted (Başvuru tarihi): 04.11.2019 Accepted (Kabul tarihi): 14.02.2020
Correspondence (İletişim): Tayfun Kermenli, Department of Thoracic Surgery Clinic, Medicalpark Elaziğ Hospital, Elazığ, Turkey
e-mail: [email protected]
RES
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CA
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Respiratory Case Reports
Cilt - Vol. 9 Sayı - No. 2 44
Pneumothorax should be considered first in patients pre-
senting to the emergency department with respiratory
distress. It can be classified as spontaneous or traumatic
pneumothorax, depending on its etiology (1). Iatrogenic
pneumothorax is evaluated in the traumatic pneumotho-
rax group, and its incidence is higher than spontaneous
pneumothorax (2). It is most commonly seen after a trans-
thoracic needle biopsy (24%), subclavian vein catheteri-
zation (22%), thoracentesis (20%), a transbronchial lung
biopsy (10%), pleural biopsy (8%) and positive pressure
ventilation (7%) (3). The incidence of iatrogenic pneumo-
thorax following acupuncture is reported to be 0.87 in
every 1,000,000 acupuncture treatments, and 1.75 in
anatomical regions close to the thoracic cavity (4). We
present the case of a patient who was diagnosed with
bilateral pneumothorax due to respiratory distress in our
emergency department.
CASE
A 51-year-old male patient presented to our emergency
department with dyspnea. A physical examination re-
vealed no respiratory sound in either lung and the patient
was cyanosed. Ecchymotic areas were noted on the skin
in the lateral region of the vertebral column and in the
medial region of the scapula resulting from cup therapy
and acupuncture needles (Figure 1a). The pulse count
was 103/min and the respiratory rate was 17. Oxygen
saturation in the room air was 86% and blood pressure
was 95/45 mmHg. An anamnesis taken from the relatives
of the patient revealed that the patient had undergone
cup therapy and acupuncture in a hotel spa 1 hour pre-
viously for the treatment of back pain. Blood samples
were obtained from the patient for cardiac tests and the
EKG was observed. The pH of the patient's blood gas was
7.23, PCO2: 58 mmHg, PO2: 67 mmHg. Budesonide-
fluticasone nebulization was initiated in the emergency
room due to the patient's history of asthma. An urgent
chest X-ray was performed with a portable bedside device,
revealing bilateral pneumothorax (Figure 1b).
The green colored angiocate (18G) was entered into the
thoracic cavity to decompress the air, and 80 mg predni-
solone was given intravenously to prevent re-expansion
edema in the lung. Bilateral chest tubes were inserted by
a thoracic surgeon, and the patient was subsequently
taken to the intensive care unit. There was minimal air
leak from the left thorax tube. No air leakage was noted
from the thorax drain in the 2 days following the insertion
of the thorax tube, and both lungs were expanded (Figure
2). The right thorax drain was removed on the third day
and the left thoracic drain was removed on the fourth day,
and the patient was discharged as cured. A chest x-ray
was found to be normal in an outpatient clinic control
one month later.
DISCUSSION
Acupuncture and cup therapy are popular alternative
therapies around the world for the treatment of various
ailments, although it has no scientific basis. In many case
reports published in literature, side effects such as pain,
fatigue, bleeding, vasovagal syncope and numbness
resulting from acupuncture have been described (5,6).
More serious complications that have been reported in-
clude pneumothorax, central nervous system injury, infec-
tion, epidural hematoma, subarachnoid hemorrhage,
cardiac tamponade, gallbladder perforation, hepatitis
and death. The most common of these serious complica-
tions is iatrogenic pneumothorax (7,8), as with our case,
and sometimes these complications can have fatal con-
sequences. Re-expansion edema following the insertion
of a thorax tube as a pneumothorax treatment is a signifi-
cant problem. The main reason for decompression in our
patient was to prevent any re-expansion edema that may
occur in the lung. In the present case, steroid treatment
for re-expansion edema prophylaxis was also given, alt-
hough there are limited studies in literature on this topic
(9).
Merchart et al. (10) carried out a study of 97,733 pa-
tients, in which serious side-effects were seen in six pa-
tients. Suicidal ideation in a 36-year-old man diagnosed
with chronic depression, hypertensive crisis in a 66-year-
old female patient with ischemic stroke history, vasovagal
syncope in a 51-year-old man, acute asthma attack in a
62-year-old woman with an asthma history, and two
women, aged 43 and 73, reported that pneumothorax
developed during needle processing.
Figure 1: Patient photo shows acupuncture needle entries (black arrow)
and ecchymotic areas resulting from cup therapy (a), AP Chest X-ray
showing the bilateral pneumothorax (yellow arrowhead) (b)
A Life-threatening Complication of Acupuncture Theraphy: Bilateral Pneumothorax | Kermenli et al.
45 www.respircase.com
Figure 2: Chest X-ray showing thorax tubes in the right (black arrow)
and left hemithorax (arrowhead)
A case report published by Hampton et al. (11) detailed a
43-year-old female patient with chronic neck pain for
which a chest tube insertion was not necessary as a result
of high volume oxygen therapy. The authors also stated
that the area between the posterior edge of the scapula
and the vertebra was a risky site for pneumothorax. Jian
et al. (12) reported a case diagnosed with postmortem
pneumothorax after acupuncture application. As stated in
literature, acupuncture can be sufficiently dangerous to
be potentially lethal. This suggests that acupuncture may
have serious complications and should be performed only
by specialist health professionals.
CONCLUSION
Iatrogenic pneumothorax should be kept in mind in pa-
tients presenting to the emergency department with dysp-
nea. As the first diagnostic method, anamnesis and physi-
cal examination followed by chest radiography are usual-
ly sufficient. Treatment usually requires chest tube inser-
tion, but in the presence of tension pneumothorax, de-
compression with green or gray angiocet (16-18 G) may
be used to relieve the patient's breathing until the chest
tube is inserted. Close follow-up and high volume oxygen
supplementation may considered as a treatment option in
patients with a small pneumothorax.
CONFLICTS OF INTEREST
None declared.
AUTHOR CONTRIBUTIONS
Concept - T.K., C.A.; Planning and Design - T.K., C.A..;
Supervision - T.K., C.A.; Funding - T.K.; Materials - T.K.,
Y.P.G., N.K.; Data Collection and/or Processing - T.K.,
C.A.; Analysis and/or Interpretation - C.A.; Literature
Review - T.K., C.A.; Writing - T.K.; Critical Review - T.K.,
C.A.
YAZAR KATKILARI
Fikir - T.K., C.A.; Tasarım ve Dizayn - T.K., C.A.; Denet-
leme - T.K., C.A.; Kaynaklar - T.K.; Malzemeler - T.K.;
Veri Toplama ve/veya İşleme - T.K., C.A.; Analiz ve/veya
Yorum - C.A.; Literatür Taraması - T.K., C.A.; Yazıyı Ya-
zan - T.K.; Eleştirel İnceleme - T.K., C.A.
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ated with computed tomography-guided transthoracic
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[CrossRef]
3. Noppen M, De Keukeleire T. Pneumothorax. Respiration
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4. Lin SK, Liu JM, Hsu RJ, Chuang HC, Wang YX, Lin PH.
Incidence of iatrogenic pneumothorax following acu-
puncture treatments in Taiwan. Acupunct Med 2019;
37:332-9. [CrossRef]
5. Öncel M, Tezcan B, Sunam GS. Iatrogenic bilateral
pneumothorax after acupuncture. Respir Case Rep 2013;
2:158-9. [CrossRef]
6. Özgen E, Bozkaya Yücel B, Yücel M, Güzel M, Gürgün
KE, Yürümez Y. A rare complication after acupuncture:
pneumothorax. Geleneksel ve Tamamlayıcı Anadolu
Tıbbı Dergisi 2019; 1:1-4.
7. Tucciarone M, Taliente S, Gómez-Blasi Camacho R,
Souviron Encabo R, González-Orús Álvarez-Morujo R.
Extensive pyomyositis of prevertebral muscles after acu-
puncture: Case report. Turk J Emerg Med 2019;
19:113-4. [CrossRef]
8. Huisma F, Konrad G, Thomas S. Pneumothorax after ac-
upuncture. Can Fam Physician 2015; 61:1071-3.
9. Kepka S, Lemaitre L, Marx T, Bilbault P, Desmettre T. A
common gesture with a rare but potentially severe com-
plication: Re-expansion pulmonary edema following
chest tube drainage. Respir Med Case Rep 2019;
27:100838. [CrossRef]
10. Melchart D, Weidenhammer W, Streng A, Reitmayr S,
Hoppe A, Ernst E, et al. Prospective investigation of ad-
Respiratory Case Reports
Cilt - Vol. 9 Sayı - No. 2 46
verse effects of acupuncture in 97 733 patients. Arch In-
tern Med 2004; 164:104-5. [CrossRef]
11. Hampton DA, Kaneko RT, Simeon E, Moren A, Rowell S,
Watters JM. Acupuncture-Related Pneumothorax. Med
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12. Jian J, Shao Y, Wan L, Zhang M, Liu N, Zhang J, et.al.
Autopsy diagnosis of acupuncture-induced bilateral ten-
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Respir Case Rep 2020;9(2): 47-51 DOI: 10.5505/respircase.2020.86570
OLGU SUNUMU CASE REPORT
47
Dilek Erdem1, İrem Karaman
2, Adem Dirican
3, Şevket Özkaya
4
A phantom tumor of the lung is a localized collection
of transudative interlobar pleural fluid in the settings
of decompensated congestive heart failure that re-
sembles a lung neoplasia on chest X-ray, but that
vanishes after the appropriate diuretic therapy. Here,
we report on a phantom tumor with a real lung neo-
plasia in the same location, both of which were con-
tributing to the complaints and the clinical picture of
the patient. Although phantom tumors are rare and
should not be misdiagnosed as masses, it is worth
making a further diagnosis and being cautious for
possible neoplasia in the same setting. Since the co-
existence of heart failure and lung cancer significantly
changes the management and follow-up procedure
of the patient, a multidisciplinary approach and
elaborative work by the pulmonologist, oncologist
and cardiologist is required on a case-by-case basis.
In such patients, cardiotoxic chemotherapy agents
should be avoided as a treatment to prevent the
progression of heart failure. The present case con-
firms the rare possibility of the co-occurrence of lung
neoplasia and phantom tumor, and recommends the
delicate management of such patients.
Key words: Phantom tumor, lung cancer, congestive
heart failure.
Akciğerde fantom tümörü, dekompanse konjestif kalp
yetmezliği durumunda lokalize transüdatif interlobar
plevral sıvı toplanması sonucu görülür ve akciğer
grafisinde akciğer neoplazisine benzer kitle görünü-
müne sebep olur, ancak uygun diüretik tedavisinden
sonra kaybolur. Burada, aynı lokasyonda gerçek bir
akciğer neoplazisi ve fantom tümörünün görüldüğü
ve bu iki durumun hastanın şikayetlerine ve klinik
tablosuna neden olduğu bir olgu bildirdik. Fantom
tümörleri nadir olarak görülür ve akciğerdeki kitle
görünümüyle karıştırılmamalıdır, ancak olası durum-
larda akciğer kanseri için temkinli olmak ve ileri test-
leri yapmak gerekir. Kalp yetmezliği ve akciğer kanse-
ri birlikteliği hastanın yönetim ve takip prosedürünü
önemli ölçüde değiştirdiğinden, münferit olgu bazın-
da pulmonolog, onkolog ve kardiyologtan oluşan
multidisipliner bir yaklaşım ve özenli bir çalışma ge-
reklidir. Bu hastalarda kalp yetmezliğinin ilerlemesini
önlemek için tedavi sırasında kardiyotoksik kemote-
rapi ajanlarından kaçınılmalıdır. Bu olgu, akciğer
neoplazisinin fantom tümörü ile nadir olarak ortaya
çıkma olasılığını doğrulamaktadır ve bu tür hastaların
hassas yönetimine dikkat çekmektedir.
Anahtar Sözcükler: Fantom tümörü, akciğer kanseri,
konjestif kalp yetmezliği.
1Department of Internal Medicine, Bahçeşehir University Faculty of
Medicine, Division of Medical Oncology; Istanbul, Turkey; De-
partment of Medical Oncology, VM Medical Park Samsun Hospi-
tal, Samsun, Turkey
2Bahcesehir University, School of Medicine, Istanbul, Turkey
3Department of Pulmonary Medicine, Samsun Medical Park Hos-
pital, Samsun, Turkey
4Department of Pulmonary Medicine, Faculty of Medicine,
Bahçeşehir University, Istanbul, Turkey
1Bahçeşehir Üniversitesi Tıp Fakültesi, Tıbbi Biyoloji Anabilim
Dalı, İstanbul
2Bahçeşehir Üniversitesi Tıp Fakültesi, İstanbul
3Samsun Medical Park Hastanesi Göğüs Hastalıkları
Bölümü, Samsun
4Bahçeşehir Üniversitesi Tıp Fakültesi, Göğüs Hastalıkları
Anabilim Dalı
Submitted (Başvuru tarihi): 29.02.2020 Accepted (Kabul tarihi): 16.04.2020
Correspondence (İletişim): Dilek Erdem, Department of Internal Medicine, Bahçeşehir University Faculty of Medicine, Division of
Medical Oncology; Istanbul, Turkey; Department of Medical Oncology, VM Medical Park Samsun Hospital, Samsun, Turkey
e-mail: [email protected]
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A phantom tumor of the lung is a localized collection of
transudative interlobar pleural fluid in the settings of de-
compensated congestive heart failure that resembles a
lung neoplasia on chest X-ray, but which vanishes after
the appropriate diuretic therapy (1). Since it is a rare
condition, awareness of this form of pleural effusion is
crucial in the differential diagnosis of pulmonary masses
(2). Here, we reported on a case with a phantom tumor
and with a real lung tumor beneath it, in which both
masses contributed to the appearance of a pulmonary
mass in a chest X-ray.
CASE
A 78-year-old male patient with no history of hyperten-
sion or diabetes mellitus presented to the clinic with com-
plaints of exertional dyspnea, dry cough, paroxysmal
nocturnal dyspnea and angina. The patient had a history
of three coronary angioplasty operations, and he was
using clopidogrel, acetyl salicylic acid, atorvastatin,
metoprolol, spironolactone and escitalopram at the time
of admission.
In his first examination, his vitals were within the normal
limits, with heart rate of 64 and blood pressure of
120/80 mmHg. After an echocardiography examination,
he was diagnosed with grade III mitral insufficiency, ac-
companying grade III tricuspid insufficiency and pulmo-
nary artery hypertension (pulmonary artery pressure=65
mmHg). A chest examination revealed diminished pulmo-
nary sounds and extensive rales in the base of both lungs.
A pulmonary function test revealed a restrictive pattern. In
posteroanterior chest X-ray, an enlarged cardiac silhou-
ette with a bulky opacity in the right lung that was sharply
marginated was found, consistent with a phantom tumor.
(Figure 1) In a thorax HRCT, a phantom tumor in the
interlobar fissure was seen, as a large mass. In addition,
pericardial effusion with bilateral pleural fluids in the
oblique fissure and in the pleural cavity was also reported
(Figure 2). Laboratory tests showed no abnormal findings,
and the sample from pleural fluid was consistent with
transude. The diagnostic hypothesis was decompensated
congestive heart failure in the presence of a phantom
tumor of the lung.
Following hospitalization, no improvement was seen in
the symptoms with IV diuretic treatment. The chest X-ray
was repeated after the first 24 hours, but the phantom
tumor was still present. Consequently, a thorax CT with
PET scan was performed due to a suspicion of lung neo-
plasia. In the PET scan, a 45x39 mm hypermetabolic
mass was observed in the middle lobe of the right lung
under the phantom tumor (Figure 3A). The phantom
tumor (Figure 3B) did not indicate any FDG-uptake, while
its right side demonstrated an increased FDG-uptake.
Based on the findings, a tru-cut biopsy procedure was
planned. The patient was discharged following the biopsy,
but returned to the emergency room one week later with
decompensated atrial fibrillation and ST elevation. The
patient was admitted to the cardiovascular ICU and an
amiodarone infusion was started. After one week of
treatment with anti-arrhythmics and diuretics, the symp-
toms improved, and the patient was discharged until the
results of the biopsy came through.
Figure 1: In the posteroanterior chest X-ray, enlarged cardiac silhouette
with a bulky opacity in right lung which was sharply marginated was
found, which was consistent with phantom tumor
Figure 2: In thorax CT, a phantom tumor in the interlobar fissure in the
appearance of a big mass has reported. A pericardial effusion with
bilateral pleural fluids in oblique fissure and in pleural cavity has also
reported
Phantom Tumor with Real Tumor of The Lung: An Unreported Case | Erdem et al.
49 www.respircase.com
Figure 3: In PET scan, 45x39 mm hypermetabolic mass observed in
middle lobe of the right lung under hypometabolic phantom tumor with
bilateral pleural fluid (A), The phantom tumor did not indicate any FDG-
uptake, while its right side demonstrated an increased FDG-uptake
which belongs to the tumor tissue (B), Hypermetabolic metastatic lesions
were seen in bilateral adrenal glands (C)
Figure 4: In chest x-ray after four months of immunotherapy and cardiac
monitoring revealed the disappearance of phantom tumor with the real
tumor under it
Figure 5: In PET-CT scan results after four months of treatment. Phan-
tom tumor totally disappeared, and the PET scan revealed diminished
pleural effusion with decreased hypermetabolic focuses
The biopsy results reported grade II adenocarcinoma of
the lung with metastasis to the bilateral adrenal glands
(Figure 3C) and increased PD-L 1 levels. At the same time,
a left ventricular thrombus was identified upon admission
to emergency room with night sweats, hemoptysis, angina
and fever. After a final evaluation, a real tumor of the
lung beneath the phantom tumor was diagnosed. The
patient was prescribed with low molecular weight heparin
for the left ventricular thrombus. The patient was hemo-
dynamically stabilized, after which immunotherapy with
nivolumab was started. After the two months of treatment,
a chest X-ray and PET scan revealed a diminished pleural
effusion with decreased hypermetabolic focus (Figure 4
and 5). In the fifth month, the patient’s general status and
symptoms have improved, and the exertional dyspnea
and cough have resolved.
DISCUSSION
Literature contains few reports of phantom tumors of the
lung, all of which resolved after the appropriate treatment
(1-6). The reported cases occurred predominantly in men,
and commonly involved a transverse fissure of right lung
(2). When the transudative fluid in the extravascular space
is unable to pass into the pleural lymphatics properly due
to pleuritis in decompensated heart failure, phantom
Respiratory Case Reports
Cilt - Vol. 9 Sayı - No. 2 50
tumors may appear, especially in the right lung, due to
the higher hydrostatic pressure on the right side of the
lung (5). A local increase in elastic recoil by the neighbor-
ing partially atelectatic lung, which is yielding the accu-
mulation of fluid, is also considered responsible for the
pathogenesis.
In most cases, phantom tumors appear in settings of
congestive heart failure, although there may be other
conditions, such as hypoalbuminemia, renal insufficiency
or pleuritis (2). Accordingly, in the presence of accumu-
lated pleural fluid within the fissure, a differential diagno-
sis should include transudates due to renal failure, exu-
dates due to malignant effusions, benign asbestos-related
effusions, parapneumonic effusions and hemothorax,
chylothorax and fibrous tumors of the visceral pleura of
the interlobar fissure (1).
Here, we reported on a phantom tumor with a real lung
neoplasia in the same location, both of which contributed
to the complaints and the clinical picture of the patient.
As signs of congestive heart failure, the reported pericar-
dial and pleural effusions in CT-scans led us to believe
that decompensation had caused an accumulation of
pleural fluid in the vascular spaces and the appearance
of the mass. Although most case reports suggested no
further investigation, and indicate an immediate launch of
treatment (2,4), the clinical picture of the patient should
be carefully evaluated for further possible diagnoses, as
in the presented case. We found in the present study that
both pleural interlobar fluid and lung neoplasia were
causing the symptoms and the chest X-ray appearance.
The presented case demonstrates the low possibility of the
existence of lung neoplasia in an acute exacerbation of
congestive heart failure settings.
The phantom tumors seen in congestive heart failure
patients are a particularly important concern if the patient
has a concurrent disease requiring treatment. In this case,
the co-existence of heart failure and lung cancer signifi-
cantly changed the management and follow-up of the
patient. In such cases, the treatment course depends on
crucial considerations and a multidisciplinary approach
involving a pulmonologist, oncologist and cardiologist.
Furthermore, each treatment procedure should be de-
signed on a case-by-case basis. In such patients, cardio-
toxic chemotherapy agents should be avoided in the
course of treatment to prevent the progression of heart
failure. Furthermore, a critical evaluation by a cardiolo-
gist should be carried out at every stage of treatment.
CONCLUSION
Phantom tumors are pseudo-tumors of the lung that are
seen on chest X-rays in the setting of congestive heart
failure. They often require no further investigation since,
since they can resolve with appropriate diuretic treatments.
However, a tight follow-up is required to ensure the dis-
appearance of the mass. As in this the present case, there
is the possibility of a real neoplasia of the lung being
missed if the tumor appearance does not resolve follow-
ing treatment. For this purpose, further investigations and
imaging techniques, as well as biopsy procedures should
be performed to ensure an accurate diagnosis. If lung
cancer is discovered in such patients, the cardiologist and
oncologist should collaborate in the establishment of an
appropriate treatment while preventing the possible car-
diac complications of chemotherapeutic agents. The
present case confirms the rare possibility of the co-
occurrence of lung neoplasia with a phantom tumor, and
recommends the delicate management of such patients.
CONFLICTS OF INTEREST
None declared.
AUTHOR CONTRIBUTIONS
Concept - D.E., İ.K., A.D., Ş.Ö.; Planning and Design -
D.E., İ.K., A.D., Ş.Ö.; Supervision - D.E., İ.K., A.D., Ş.Ö.;
Funding - A.D., Ş.Ö., D.E.; Materials - Ş.Ö., D.E.; Data
Collection and/or Processing - D.E., İ.K.; Analysis and/or
Interpretation - D.E., İ.K.; Literature Review - D.E., Ş.Ö.;
Writing - D.E., İ.K., A.D., Ş.Ö.; Critical Review - D.E., İ.K.,
A.D., Ş.Ö.
YAZAR KATKILARI
Fikir - D.E., İ.K., A.D., Ş.Ö.; Tasarım ve Dizayn - D.E.,
İ.K., A.D., Ş.Ö.; Denetleme - D.E., İ.K., A.D., Ş.Ö.; Kay-
naklar - A.D., Ş.Ö., D.E.; Malzemeler - Ş.Ö., D.E.; Veri
Toplama ve/veya İşleme - D.E., İ.K.; Analiz ve/veya Yo-
rum - E D.E., İ.K., N.K.; Literatür Taraması - D.E., Ş.Ö.;
Yazıyı Yazan - D.E., İ.K., A.D., Ş.Ö.; Eleştirel İnceleme -
D.E., İ.K., A.D., Ş.Ö.
REFERENCES
1. Lozo M, Lozo Vukovac E, Ivancevic Z, Pletikosic I. Phan-
tom tumor of the lung: localized interlobar effusion in
congestive heart failure. Case Rep Cardiol 2014;
2014:207294. [CrossRef]
Phantom Tumor with Real Tumor of The Lung: An Unreported Case | Erdem et al.
51 www.respircase.com
2. Tesloianu DN, Chioarta M, Corduneanu D, Ignat AM,
Petris AO, Tesloianu A. Does phantom tumor really exist?!
Maedica (Buchar) 2017; 12:281-5.
3. Ardic I, Yarlioglues M, Celik A, Kaya MG. Vanishing or
phantom tumor of the lung. Tex Heart Inst J 2010;
37:730-1.
4. Melo BS, Serra AC, Belo MT, Belo Neto E, Melo SM.
Phantom tumor of the lung. Rev Assoc Med Bras (1992)
2012; 58:517-8. [CrossRef]
5. Mikaeili H, Mehdizadeh Baghbani J. Multiple phantom
tumor of the lung: A complex appearance resolving with
appropriate intervention. Tanaffos 2016; 15:243-5.
6. Shaikh S, Shaikh S. Pleural effusion resembling a lung
tumor: phantom tumor of the lung. Egypt J Intern Med
2016; 28:174-5. [CrossRef]
Respir Case Rep 2020;9(2): 52-55 DOI: 10.5505/respircase.2020.82435
OLGU SUNUMU CASE REPORT
52
İbrahim Güven Çoşğun1, Düriye Öztürk
2, Çiğdem Özdemir
3, Şule Çilekar
1, Filiz Yavaşoğlu
4,
Ersin Günay1
Diffuse Large B-cell lymphoma is a subgroup of non-
Hodgkin's lymphoma. Lymphoma with endobronchial
pulmonary involvement is a rarely reported condition.
A 64-year-old male patient was admitted to our clinic
with complaints of swellings in the neck and in the
bilateral upper limbs, along with shortness of breath.
A computed tomography of the thorax revealed dif-
fuse mediastinal enlargement with a superior vena
cava obstruction, and a huge right hilar mass for-
mation obliterating the anterior segment of the right
upper lobe. A diagnostic fiberoptic bronchoscopy
showed mucosal irregularity and an endobronchial
lesion on the anterior segment of the right upper lobe.
A diagnosis of endobronchial lymphoma was made
following bronchial biopsies.
Key words: Diffuse Large B-cell Lymphoma, Endob-
ronchial, Non-Hodgkin.
Diffüz büyük B hücreli lenfomalar non-Hodgkin len-
fomaların subtipini oluştururlar. Lenfomaların endob-
ronşial invazyonu oldukça nadirdir. Nefes darlığı,
boyun ve üst ekstremitede şişlik şikayeti olan 64 ya-
şında erkek hasta kliniğimize başvurdu. Toraks to-
mografisinde üst mediastende büyümüş lenf nodları,
vena cava süperiorda daralma ve sağ hiler büyümüş
lenf nodları izlendi. Fiberoptik bronkoskopisinde sağ
akciğer üst lob anterior segmentte endobronşiyal
lezyon izlendi. Fiberoptik bronkoskopik biyopsi ile
endobronşiyal lenfoma tanısı doğrulandı.
Anahtar Sözcükler: Diffüz Büyük B-Hücreli, Lenfoma,
Endobronşiyal Non-Hodgkin.
1Department of Pulmonology, Afyonkarahisar University of Health
Sciences, Medical Faculty, Turkey
2Department of Radiation Oncology, Afyonkarahisar University of
Health Sciences, Medical Faculty, Turkey
3Department of Pathology, Afyonkarahisar University of Health
Sciences, Medical Faculty, Turkey
4Department of Hematology, Afyonkarahisar University of Health
Sciences, Medical Faculty, Turkey
1Afyonkarahisar Sağlık Bilimleri Üniversitesi, Tıp fakültesi
Hastanesi, Göğüs Hastaliklari Ana Bilim Dalı,Afyonkarahisar
2Afyonkarahisar Sağlık Bilimleri Üniversitesi, Tıp fakültesi
Hastanesi, Radyasyon Onkolojisi Ana Bilim
Dalı,Afyonkarahisar
3Afyonkarahisar Sağlık Bilimleri Üniversitesi, Tıp fakültesi
Hastanesi, Patoloji Ana Bilim Dalı,Afyonkarahisar
4Afyonkarahisar Sağlık Bilimleri Üniversitesi, Tıp fakültesi
Hastanesi, Hematoloji Ana Bilim Dalı, Afyonkarahisar
Submitted (Başvuru tarihi): 27.09.2019 Accepted (Kabul tarihi): 07.01.2020
Correspondence (İletişim): İbrahim Güven Çoşğun, Department of Pulmonology, Afyonkarahisar University of Health Sciences,
Medical Faculty, Turkey
e-mail: [email protected]
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Pulmonary lymphomas may occur as a primary pulmo-
nary malignancy or as a secondary manifestation derived
from systemic lymphoma. Primary pulmonary lymphomas
are defined as malignant lymphomas that originate in the
pulmonary parenchyma or bronchi, with or without hilar
lymph node involvement and the absence of any other
tissue localization for 3 months following the diagnosis
(1). In literature, the prevalence of primary pulmonary
lymphoma has been reported in 0.5–1% of malignancies,
accounting for <1% of all malignant lymphomas (2).
Primary pulmonary lymphoma is rare, as pulmonary tissue
contains less lymphoid tissue than other sites. The most
common type of primary pulmonary lymphoma is muco-
sa-associated lymphoid tissue (MALT) lymphoma (2).
Secondary pulmonary lymphoma is more common than
primary pulmonary lymphoma. Diffuse Large B-cell lym-
phoma (DLBCL) is a subgroup of non-Hodgkin's lympho-
ma. Lymphoma with pulmonary involvement has been
rarely reported, and endobronchial lymphoma is an ex-
ceptional finding. Mediastinal lymphoma has an aggres-
sive behavior due to the close relationship with blood
vessels (3). We present here the case of a patient with
endobronchial lymphoma, diagnosed with a bronchial
biopsy.
CASE
A 64-year-old male patient presented with a history of
neck and bilateral upper limb swelling, as well as short-
ness of breath for the past 2 weeks. Upon physical exam-
ination, swelling to the neck and the bilateral upper ex-
tremities, and bilateral dilated superficial veins on the
chest were observed. The patient was using inhaler drugs
for chronic obstructive pulmonary disease (COPD). The
laboratory findings (hemogram and blood biochemistry)
were within normal limits. A postero-anterior chest X-ray
showed mediastinal and right hilar enlargement (Figure
1A). A contrast-enhanced computed tomography (CT) of
the thorax revealed diffuse mediastinal enlargement with
a superior vena cava obstruction and a right hilar mass
formation obliterating the anterior segment of the right
upper lobe (Figure 1C and D). A diagnostic fiberoptic
bronchoscopy was performed, revealing mucosal irregu-
larity and an endobronchial lesion in the anterior seg-
ment of the right upper lobe (Figure 1B). A histopatholog-
ical examination revealed diffuse large B-cell lymphoma
(DLBCL) infiltration. A bone marrow aspiration biopsy was
negative for lymphoma infiltration. Immunohistochemistry
showed an expression of vimentin, and a clustering of
differentiation 20 (CD20), CD45 and CD19, but not of
CD3, CD15 or CD30 (Figure 2). Due to the bulky lymph
node and superior vena cava syndrome, the patient ini-
tially received a 300 cGy dose fraction per day, with a
total dose of 3000cGy radiation. A chemotherapy proto-
col with rituximab, cyclophosphamide, doxorubicin hy-
drochloride, vincristine and prednisolone (R-CHOP) was
started for the treatment of the patient.
DISCUSSION
We report here on a rare case of lymphoma with endo-
bronchial involvement, diagnosed by bronchoscopic
biopsy. Lymphoma refers to malignant tumors that origi-
nate in the lymph nodes or in other lymphoid tissues (4).
DLBCL is a subgroup and an aggressive form of non-
Hodgkin lymphoma, and accounts for approximately 30%
of all lymphomas (5). In a retrospective study of 1,221
patients with extranodal DLBCL, Takahashi et al. (6) re-
ported lung involvement in 3.7% of cases. Lymphoma
may present as mediastinal lymphadenopathy and as an
isolated mediastinal mass. Pulmonary lymphoma with
endobronchial involvement usually presents as a bulky
lesion in the mediastinum, and is associated with symp-
toms related to local compression (7). The pathogenesis
of endobronchial Hodgkin lymphoma is unclear. The
mechanism is presumed to be a contiguous transmural
spread from the adjacent lymph nodes, but may also
arise in mucosa-associated lymphoid tissue (8). The dif-
ferential diagnosis of lymphoma with pulmonary involve-
ment includes mycobacterial and fungal infections, We-
gener granulomatosis, Langerhans cell histiocytosis and
other pulmonary malignancies (9). Endobronchial lym-
phoma is classified into two types, according to the pat-
tern of involvement: diffuse submucosal infiltration (type I),
and localized solitary mass (type II) (10). Type I includes
diffuse submucosal infiltrates originating from hematog-
enous or lymphangitic spread in the presence of systemic
lymphoma; while type II includes airway involvement by a
localized mass due to the direct spread of lymphoma
from the adjacent lymph nodes, or arising out of bron-
chus-associated lymphoid tissue (11). The pattern of in-
volvement in the current patient was a localized solitary
mass.
Routinely, mediastinoscopy or other invasive surgical
procedures are preferred for the obtaining of an exact
diagnosis, although Endobronchial Ultrasound-
transbronchial needle aspiration (EBUS-TBNA) can be
used as an alternative option (12). Due to the difficulties
in confirming a diagnosis of the lymphoma subtype from
a small volume specimen in EBUS-TBNA, the recommen-
Diffuse Large B-cell Lymphoma Exhibiting Endobronchial Involvement: A Case Report | Coşğun et al.
54 www.respircase.com
dation of EBUS-TBNA for the evaluation of suspected
lymphoma is controversial. The diagnostic sensitivity of
EBUS-TBNA for lymphoma is lower than for lung cancer,
although to avoid invasive surgical procedures, EBUS-
TBNA may still be considered as the initial investigative
technique for suspected lymphoma (13). Recently, EBUS-
TBNA has been reported to be useful in the diagnosis of
mediastinal lymphoma. Of 1,471 cases that underwent
EBUS-TBNA for isolated mediastinal masses and/or lym-
phadenopathy, 27 patients (1.8 %) were diagnosed with
lymphoma (14). In the present case, endobronchial lym-
phoma was diagnosed via bronchoscopic biopsy rather
than EBUS-TBNA. By performing a fiberoptic bronchos-
copy and obtaining biopsy samples showing tumor infil-
tration on the right upper lobe of the right lung, the diag-
nosis of lymphoma was made without the use of EBUS-
TBNA.
Figure 1: Chest X-ray showing enlargement of the right mediastinal and
hiler region (A), Bronchoscopic image endobronchial vegetation right
upper lobe of the right lung (B), Thorax tomography enhancing lymph
node with superior vena cava obstruction (C), Enhancing right hilar
lymph node (D)
Figure 2: Hematoxylin and eosin (H&E) staining of the lung biopsy
demonstrate diffuse proliferation (H&EX40) (A), Lung biopsy demonstrate
diffuse proliferation, high power (H&EX200) (B), Immunohistochemistry
staining of the lung biopsy shows positive for CD20 (X200) (C)
Lymphoma with endobronchial involvement is rare, and a
diagnosis of endobronchial lymphoma with bronchoscop-
ic biopsy has been rarely reported in literature. A system-
atic, careful examination of the bronchial system for a
diagnosis of mediastinal mass lesions is crucial. Broncho-
scopic biopsies for a diagnosis of endobronchial lym-
phoma with huge mediastinal lesions may contribute to
histological diagnoses through the use of minimally inva-
sive procedures, avoiding the need for EBUS or mediasti-
noscopic interventions.
CONFLICTS OF INTEREST
None declared.
AUTHOR CONTRIBUTIONS
Concept - İ.G.Ç., D. Ö., Ç.Ö., Ş.Ç.1, F.Y., E.G.; Plan-
ning and Design - İ.G.Ç., D. Ö., Ç.Ö., Ş.Ç.1, F.Y., E.G.;
Supervision - İ.G.Ç., D. Ö., Ç.Ö., Ş.Ç.1, F.Y., E.G.;
Funding - İ.G.Ç., E.G.; Materials - İ.G.Ç., E.G.; Data
Collection and/or Processing - İ.G.Ç., E.G.; Analysis
and/or Interpretation - İ.G.Ç., E.G.; Literature Review -
İ.G.Ç., E.G.; Writing - İ.G.Ç., E.G.; Critical Review -
İ.G.Ç., E.G.
YAZAR KATKILARI
Fikir - İ.G.Ç., D. Ö., Ç.Ö., Ş.Ç.1, F.Y., E.G.; Tasarım ve
Dizayn - İ.G.Ç., D. Ö., Ç.Ö., Ş.Ç.1, F.Y., E.G.; Denet-
leme - İ.G.Ç., D. Ö., Ç.Ö., Ş.Ç.1, F.Y., E.G.; Kaynaklar
- İ.G.Ç., E.G.; Malzemeler - İ.G.Ç., E.G.; Veri Toplama
ve/veya İşleme - İ.G.Ç., E.G.; Analiz ve/veya Yorum -
İ.G.Ç., E.G.; Literatür Taraması - İ.G.Ç., E.G.; Yazıyı
Yazan - İ.G.Ç., E.G.; Eleştirel İnceleme - İ.G.Ç., E.G.
REFERENCES
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Respiratory Case Reports
Cilt - Vol. 9 Sayı - No. 2 55
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6. Takahashi H, Tomita N, Yokoyama M, Tsunoda S, Yano
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Igawa S, et al. Endobronchial involvement of mantle cell
lymphoma: A case report. Respir Med Case Rep 2016;
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12. Yang H, Zhao H, Garfield DH, Teng J, Han B, Sun J. En-
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13. Steinfort DP, Conron M, Tsui A, Pasricha SR, Renwick WE,
Antippa P, et al. Endobronchial ultrasound-guided trans-
bronchial needle aspiration for the evaluation of suspect-
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Respir Case Rep 2020;9(2): 56-59 DOI: 10.5505/respircase.2020.68542
OLGU SUNUMU CASE REPORT
56
Emine Afşin1, Ayperi Ozturk
2
Karsinoid tümörler nöroendokrin tümör grubunda
değerlendilirler. Gastrointestinal traktusta yerleşimi
daha sık iken nadiren bronşial orjinli de olabilir.
Akciğer kanserlerinin %0,5-1’ini oluşturur. Genellikle
genç yaş grubunda görülür. Yetmiş sekiz yaşında,
erkek hastada rastlantısal olarak istenilen toraks
bilgisayarlı tomografide trakeal polipoid lezyon izlen-
di. Atipik karsinoid tümör olarak tanı alan hasta ileri
yaşı ve ağır KOAH' ı (FEV1 %25) olması nedeniyle
inoperabl kabul edildi. Radyolojik olarak ekstralumi-
nal uzanımı ve metastatik lenf nodu olmaması üzerine
Nd: YAG lazer ve ardından lezyon köküne koterizas-
yon uygulandı. Endobronşial tedavi yaklaşımı ile tam
kür sağlandı.
Anahtar Sözcükler: Atipik karsinoid tümör, endobron-
şial tedavi, trakea.
Carcinoid tumors are evaluated in the
neuroendocrine tumor group. While they are more
common in the gastrointestinal tract, they may rarely
be of bronchial origin. They account for 0.5–1% of
all lung cancers, and are usually seen in the young
age group. A 78-year-old male patient with a
tracheal polypoid lesion was observed at thorax CT.
The patient was diagnosed with an atypical carcinoid
tumor, and was considered to be inoperable due to
his advanced age and severe COPD (FEV1 25%).
Due to the extraluminal extension and no metastatic
lymph nodes identified radiologically, an Nd: YAG
laser was applied and the root of the lesion was
cauterized. Complete cure was achieved with an
endobronchial treatment approach.
Key words: Atypical carcinoid tumor, endobronchial
therapy, trachea.
1İzzet Baysal Devlet Hastanesi, Göğüs Hastalıkları Kliniği, Bolu
2Ankara Atatürk Göğüs Hastalıkları ve Göğüs Cerrahisi Eğitim ve
Araştırma Hastanesi, Girişimsel Pulmonoloji Kliniği, Ankara
1Department of Chest Diseases, İzzet Baysal State Hospital,
Bolu, Turkey
2Department of Interventional Pulmonology, Ankara Ataturk
Chest Diseases and Chest Surgery Training and Research
Hospital, Health Science University, Ankara, Turkey
Başvuru tarihi (Submitted): 06.10.2019 Kabul tarihi (Accepted): 06.12.2019
İletişim (Correspondence): Emine Afşin, İzzet Baysal Devlet Hastanesi, Göğüs Hastalıkları Kliniği, Bolu
e-mail: [email protected]
RES
PIR
ATO
RY
CA
SE R
EPO
RTS
Respiratory Case Reports
Cilt - Vol. 9 Sayı - No. 2 57
Bronşial karsinoid tümörler bronş muköz bezlerindeki
Kultchitsky hücrelerinden köken alırlar. Düşük malignite
potansiyeline sahip, yavaş büyüyen, lokal gelişme göste-
ren, nadiren lenf nodu ve uzak organ metastazı yapan ve
bazen hormonal aktivite gösteren bir tümör grubudur (1).
Karsinoid tümörlerin çoğu ana bronş veya lob bronşu gibi
büyük solunum yollarından köken alır. Atipik ve tipik kar-
sinoid tümör olarak 2 grupta incelenir. Karsinoid tümörle-
rin %10-20’si atipik, %80-90'ı tipik karsinoiddir (2). Genç
yaş grubunda daha sık görülen bu tümörler bizim olgu-
muzda olduğu gibi nadiren ileri yaşta rastlantısal olarak
da görülebilir. Yazımızda atipik karsinoid tümör tanısı
koyup endobronşial tedavi uyguladığımız bir hasta su-
nulmaktadır. Hastamızı sunma amacımız; ileri yaşta nadir
karşılaşılan bir olgu olması ve eşlik eden hastalıkları ne-
deniyle cerrahi yapılamayıp endobronşial tedavi uygu-
lanması nedeniyle literatür eşliğinde tartışmaktır.
OLGU
Yetmiş sekiz yaşında erkek hasta, ağır KOAH, opere la-
rinks karsinomu (skuamöz hücreli karsinom) tanısıyla
polikliniğimizde takip edilmekteydi. Elli paket/yıl sigara
öyküsü vardı. Fizik muayenede ekspiryum uzun olup solu-
num sesleri azalmıştı. Oda havasında oksijen saturasyo-
nu %80 idi. İki yıl önceki toraks bilgisayarlı tomografisin-
de sağ paratrakeal lenfadenomegali olması nedeniyle
kontrol amaçlı toraks bt istendiğinde: trakeal polipoid
lezyon izlendi (Şekil 1 ve 2). Lokal anestezi altında yapılan
fiberoptik bronkoskopisinde: trakeada karinaya 3 cm
mesafede, sağ lateral duvarda, havayolunu %80 oranın-
da daraltan polipoid lezyonlar izlendi (Şekil 3) ve buradan
biyopsi alındı. Patoloji sonucu: nöroendokrin tümör ola-
rak raporlandı. Hastanın solunum sıkıntısı olup havayolu
açıklığını sağlamak üzere endonronşial tedavi planlandı.
Genel anestezi altında rijid bronkoskop ile Nd:YAG lazer
ile lezyonun eksizyonu ve ardından lezyonun köklerine
koterizasyon yapıldı. Patoloji sonucu atipik karsinoid tü-
mör olarak raporlandı. İleri yaşı, ağır KOAH ve opere
larenks karsinomu olmasından dolayı cerrahi tedavi dü-
şünülmedi.
TARTIŞMA
Primer trakeobronşiyal tümörler tüm pulmoner tümörlerin
yaklaşık %0,1’ni oluşturmaktadırlar (3). Karsinoid tümör,
epitel hücrelerinden köken alan akciğerin nöroendokrin
tümörleri arasındadır. Dünya Sağlık Örgütü akciğerin
nöroendokrin tümörlerini: karsinoid tümör (tipik/atipik),
büyük hücreli nöroendokrin karsinom (BHNEK) ve küçük
hücreli karsinom (KHK)olarak sınıflandırmıştır (4). Akciğer
karsinomlarının yaklaşık %2’ sini oluştururlar (5,6). Bron-
kopulmoner karsinoidlerin %75-90’ı santral %10-25’i
periferik yerleşimlidir. Pulmoner karsinoidler çocuklarda
ve gençlerde en sık görülen primer akciğer tümörleridir
(7).
Karsinoid tümörler genellikle 45-55 yaş grubu arasında
görülmektedir. Tipik karsinoidler (TK) atipik (AK) olanlara
göre daha erken yaş grubunda görülme eğilimi gösterirler.
Genel olarak erkek ve kadın cinsiyet için eşit dağılım
izlenmektedir.
Şekil 1: Axial kesitte trakeadaki polipoid lezyonlar.
Şekil 2: Coronal kesitte trakeadaki polipoid lezyonlar
Şekil 3: Bronkoskopide lümeni daraltan polipoid lezyonlar
Trakeal Yerleşimli Atipik Karsinoid Tümör | Afşin et al.
58 www.respircase.com 58
Atipik karsinoid tümörler malign histolojik özellikler ve
agresif tavır gösteren tümörlerdir. TK’in tersine bu tümör-
ler sıklıkla periferal yerleşimli olup daha ileri yaş grubun-
da görülür. Bizim olgumuz ileri yaşta (78 yaşında) olup
lezyonu santral yerleşimli idi. Atipik karsinoidli hastalarda
yüksek oranda metastaz gelişme riski vardır (8,9).
Proksimal lokalizasyon gösteren tümörler kısmi veya tam
bronş obstrüksiyonu oluşturabilirler. Bizim olgumuzda da
bronkoskopik olarak trakea lümeni %80 oranında daral-
mış olarak izlendi. Öksürük, hemoptizi, tekrarlayan enfek-
siyon bulguları klasik semptomlarındandır. Trakea veya
ana bronş yerleşimi olan olgularda stridor gelişebilir.
Pulmoner karsinoid olgularında tanı anında karsinoid
sendrom görülmesi oldukça nadirdir ancak; büyük tümö-
rü olan veya yaygın karaciğer metastazı olan olgularda
izlenebilmektedir (10,11). Olgumuzda olduğu gibi karsi-
noid tümörlerin yaklaşık %40’ı belirgin bir klinik bulgu
olmadan insidental olarak radyolojik bulgularla saptan-
maktadır (12). Radyolojik olarak iyi sınırlı, yuvarlak veya
ovoid, hafifçe lobüle nodüller olarak tomografilerde izle-
nebildiği gibi sadece hava yolunda saplı polipoid lezyon-
lar olarak da izlenebilir (13).
Tanı için günümüzde nöroendokrin tümörlerde somatos-
tatin analogları ile yapılan PET görüntülemede sıklıkla
Galyum-68 kullanılır (14). Tüm karsinoidlerin yakla-
şık %75’i bronkoskopik olarak görülebilir. Bronkoskopik
olarak vaskülaritesi fazla, pembe-mor intakt epitel ile
örtülü polipoid lezyonlar şeklinde izlenirler. Bazı karsinoid
tümörler polipoid ve saplı iken bazıları da sapsız olarak
izlenirler. Tümörün çoğunluğu ekstraluminal yerleşimli
olabileceği için “buz dağı tümörler” olarak da isimlendiri-
lirler (10). Vaskülaritesi oldukça fazla olan ve submukozal
yerleşimli olan bu tümörlerde derin biyopsiler tanı koya-
bilmek için gerekli olup, sıklıkla ciddi kanamalara yol
açabilir. Ancak olgumuzda biyopsi sonrasında kanama
sorunu yaşanmadı. Bazı olgularda genel anestezi altında
rijid bronkoskopi ile işlemin yapılması kanama kontrolünü
sağlamak için tercih edilebilir (15). AK ile TK ayırımı an-
cak cerrahi ve bronkoskopik olarak çıkarılan kitleden
yapılabilir. Karsinoid tümörlerde evreleme için TNM sis-
temi kullanılmaktadır. Ancak multipl karsinoid tümör
mevcudiyetinde metastaz yerine senkron primer tümörler
olarak düşünmek gerekir (16). TK’ler düşük dereceli ma-
lign davranış gösterirler ve beş yıllık sağkalım %87-
100’dür. AK’ler ise daha fazla malign potansiyele sahip-
tirler ve beş yıllık sağkalım oranı %56-75’dir (17,18).
Pulmoner karsinoidlerde tedavinin esası; akciğer dokusu
korunarak tümörün komplet rezeksiyonudur. Ancak tümö-
rün distalinde geri dönüşümsüz değişikliklerin saptandığı
olgularda akciğer rezeksiyonu da gerekebilmektedir.
Ayrıca, cerrahi olarak komple lenfadenektomi yapılması
da önerilmektedir (16).
Son yıllarda doğru seçilmiş olgularda yapılan bronkosko-
pik rezeksiyon sonuçlarının da iyi olduğu bildirilmektedir
(17,19). Bronkoskopik olarak Argon plazma koagülasyo-
nu, elektrokoter, kriyoterapi ya da lazer teknikleri kulla-
nılmaktadır (20). Santral yerleşimli pulmoner karsinoidli
hastalara başlangıçta endobronşiyal tedavi yapılması,
ekstralüminal komponenti belirgin olanlarda da tedavinin
cerrahi rezeksiyon ile tamamlanmasının uygun bir yakla-
şım olacağı belirtilmektedir. Endobronşiyal tedavi sonrası
altıncı haftada bronkoskopik ve görüntüleme yöntemleri
ile lezyonun (özellikle ekstralüminal komponentin) yeniden
değerlendirilmesi önerilir. Ekstralüminal komponenti be-
lirgin olan, tümörün distalinde geri dönüşümsüz paranki-
mal lezyonu olan, endobronşiyal tedavi sonrası nüks olan,
atipik karsinoid histolojisi olan veya belirgin mediastinal
lenfadenopatileri olan olgularda cerrahi tedavi uygulan-
ması önerilir (17). Yirmi yıl sonra bile nüks olabileceğin-
den 20 yıla kadar takip önerilir.
Bizim olgumuzun radyolojik olarak ekstraluminal uzanımı
ve metastatik lenf nodu olmaması üzerine, Nd:YAG lazer
ve ardından lezyon köküne kriyoterapi uygulandı. Hasta-
nın ileri yaşı ve ağır KOAH'ı (FEV1: %25) olması nedeniy-
le inoperabl kabul edildi. Postop bronkoskopik kontro-
lünde lezyon izlenmedi (Şekil 4) ve alınan kontrol biyopsi-
sinde malign hücre izlenmedi.
Şekil 4: Tedavi sonrası altıncı hafta bronkoskopik görünüm
Sonuç olarak; karsinoid tümörler düşük gradlı malign ve
yavaş büyüyen lokal invazif tümörlerdir. Endobronşial
tedavi ile başarılı sonuçlar alınmaktadır. Bizim hastamız
ileri yaşta rastlantısal olarak atipik karsinod tümör tanısı
konulması ve endobronşial rezeksiyon ile tam kür sağ-
Respiratory Case Reports
Cilt - Vol. 9 Sayı - No. 2 59
lanmış olması nedeniyle sunularak ilgili literatür gözden
geçirilmiştir.
ÇIKAR ÇATIŞMASI
Bu makalede herhangi bir çıkar çatışması bildirilmemiştir.
YAZAR KATKILARI
Fikir - E.A., A.O.; Tasarım ve Dizayn - E.A., A.O.; Denet-
leme - E.A., A.O.; Kaynaklar - E.A.; Malzemeler - E.A.;
Veri Toplama ve/veya İşleme - E.A.; Analiz ve/veya Yo-
rum - E.A.; Literatür Taraması - E.A.; Yazıyı Yazan - E.A.;
Eleştirel İnceleme - E.A.
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Respir Case Rep 2020;9(2): 60-65 DOI: 10.5505/respircase.2020.01328
OLGU SUNUMU CASE REPORT
60
Yasemin Arı Yılmaz, Meral Gulhan
Pulmoner tromboemboli daha çok alt ekstremite
venlerinden kaynaklanmaktadır. Ana pulmoner yata-
ğın aniden tıkanması kardiyak dekompansasyona
neden olabilir. Elektrokardiyografi (EKG)de sağ vent-
rikül yüklenme bulguları olan V1-4 T negatifliği,
V1’de QR paterni, komplet veya inkomplet sağ dal
bloğu ve S1Q3T3 görülebilir. Acil ve hızlı tedavi
hayat kurtarıcı olabilmektedir. Streptokinaz ve alteplaz
verilişi saatler sürerken tenekteplaz verilişi puşe şek-
linde olmaktadır. Bu da dakikaların önemli olduğu
acil müdahale gerektiren durumlarda hayati önem
arzetmektedir. Ayrıca riskli ilaçların uygulanması
esnasında sorumlu doktorun hasta başında bekleme
süresini ve iş gücü kaybını kısaltmaktadır. Hastanın
daha kısa sürede normale dönmesini sağlamaktadır.
Bu konuda literatürde çok sayıda çalışma olmasına
rağmen ülkemizde kullanım endikasyonu olmadığın-
dan verilerimiz yetersizdir. Bu olguda alternatif ilaçlar
elde olmadığı için tenekteplaz kullanılmak zorunda
kalınmıştır.
Anahtar Sözcükler: Pulmoner tromboemboli, tromboli-
tik Tedavi, tenekteplaz.
Pulmonary thromboembolisms occur mostly in the
lower extremity veins. Sudden occlusions of the main
pulmonary bed may result in cardiac
decompensation. Electrocardiography (ECG) shows
right ventricular overload findings, V1-4 T negativity,
Q1 pattern in V1, complete or incomplete right
bundle branch block and accompanying S1Q3T3.
Emergency and rapid treatment can be life-saving.
Streptokinase and alteplase administration lasts for
hours while tenecteplase administration is in the form
of push. This is vital in patients requiring immediate
interventions, where minutes can be critical. It also
reduces the waiting time of the responsible doctor
and loss of labor during the administration of risky
drugs, and enables the patient to return to normal in
a shorter time. Although there have been many
studies in the literature addressing this subject, the
available data is insufficient, since there is no
indication for use in our country. In the present case,
as no alternative drugs were available, tenecteplase
had to be used.
Key words: Pulmonary thromboembolism, thrombolyt-
ic therapy, tenecteplase.
Hitit Üniversitesi Göğüs Hastalıkları Ana Bilim Dalı, Çorum Department of Chest Diseases, Hitit University, Çorum, Turkey
Başvuru tarihi (Submitted): 01.10.2019 Kabul tarihi (Accepted): 10.02.2020
İletişim (Correspondence): Yasemin Arı Yılmaz, Hitit Üniversitesi Göğüs Hastalıkları Ana Bilim Dalı, Çorum
e-mail: [email protected]
RES
PIR
ATO
RY
CA
SE R
EPO
RTS
Respiratory Case Reports
Cilt - Vol. 9 Sayı - No. 2 61
Pulmoner tromboemboli daha çok alt ekstremite venle-
rinden kaynaklanmaktadır. Ana pulmoner yatağın aniden
tıkanması kardiyak dekompansasyona neden olabilir.
Elektrokardiyografi (EKG)’de sağ ventrikül yüklenme bul-
guları olan V1-4 T negatifliği, V1’de QR paterni, komplet
veya inkomplet sağ dal bloğu ve S1Q3T3 görülebilir (1-
3). Yüksek riskli emboli olarak adlandırılan bu duruma
hipotansiyon hipoksi gibi şok bulguları eşlik eder. Acil ve
hızlı tedavi hayat kurtarıcı olabilmektedir. Streptokinaz ve
alteplaz verilişi saatler sürerken tenekteplase verilişi ise
puşe şeklinde olmaktadır. Bu da dakikaların önemli oldu-
ğu acil müdahale gerektiren durumlarda hayati önem arz
etmektedir. Ayrıca riskli ilaçların uygulanması esnasında
sorumlu doktorun hasta başında bekleme süresini ve iş
gücü kaybını da kısaltmaktadır. Hastanın daha kısa süre-
de normale dönmesini sağlamaktadır. Bu konuda litera-
türde çok sayıda çalışma olmasına rağmen ülkemizde
kullanım endikasyonu olmadığından bizim verilerimiz
yetersizdir. Bu olguda, alternatif ilaçlar elde olmadığı için
pulmoner embolide tenekteplaz kullanılmak zorunda
kalınmıştır ve yaşanan deneyimin ülke verisi olarak payla-
şılması amaçlanmıştır.
OLGU
Göğüs ağrısı ve nefes darlığı şikâyeti ile acil servisimize
başvuran 65 yaşındaki kadın hastanın özgeçmişinde di-
yabetes mellitus ve obezite dışında bilinen hastalığı yoktu.
Hastanın oda havasında geliş satürasyonu (SaO2) %80
idi. Laboratuvar parametreleri tablo 1 de görülmektedir.
Akciğer grafisinde, (film kalitesi kötü olmakla beraber)
ekspiryum filmi olup mediastende hafif dolgunluk vardı.
Yatarak hasta başı çekilmişti. Belirgin parankimal patoloji
izlenmedi (Şekil 2).
Hasta bu bulgularla miyokard enfaktüsü açısından kardi-
yolojiye danışıldı. Hastanın 1 hafta öncesinde de göğüs
ağrısı ve nefes darlığı ile kardiyoloji tarafından değerlen-
dirildiği ve yapılan koroner anjiografi ve ekokardiyografi
(eko) bulgularının normal olduğu öğrenildi. Yapılan eko-
sunda daha önceki ekosunda olmayan ileri triküspit yet-
mezliği izlendi. Sağ boşluklarda genişleme olduğu belir-
tildi. Pulmoner arter basıncı ölçülemedi. Hastada mevcut
bulgular ile miyokard enfarktüsü düşünülmedi.
Hasta tarafımıza konsülte edildi. Hastanın başvuru saati
gece olduğundan o sırada nöbetçi radyoloji uzmanı ol-
madığından alt ekstremite venöz dopler ultrasonografi
(USG) çekilemedi. Ancak sol bacakta derin ven trombo-
zunu düşündürür şekilde, çap farkı, şişlik ve kızarıklık
mevcuttu. Geliş öyküsünde daha önce nefes darlığı ve
göğüs ağrısının olmadığını, şikâyetlerinin bir hafta önce
başladığını ve giderek ciddi şekilde arttığını ifade etti.
Belirgin öksürük ve balgamı yoktu. Kreatinin yüksek oldu-
ğundan kontrastsız toraks BT çekilebildi. BT'de belirgin
parankimal patoloji saptanmadı. EKO bulguları ile uyum-
lu olarak kardiyak şift, sağ boşluklarda genişleme ve
pulmoner arterin dilate olduğu gözlendi (Şekil 3, 4 ve 5).
Şekil 1: Tedavi öncesi EKG (atriyal fibrilasyon).
Tablo 1: Hastanın başvurudaki laboratuvar parametreleri
Parametre Ölçüm Normal
Aralık
Beyaz küre 13850 10⁹/ul 4-10
Hemoglobin 12,6 gr/dl 11-16
Trombosit 193 103/ul 100-300
Glukoz 214 mg/dl 74-106
Kreatin 1,7 mg/dl 0,5-0,9
C-Reaktif Protein 23 mg/l 0-5
D-Dimer: 5 mg/l 0,063-0,701
Troponin 0,074 ng/ml 0,0-0,02
Pro-BNP Çalışılmadı
Venöz kan gazı
• PO2 22,4 mmHg 83-108
• PCO2 40,4 mmHg 32-45
• ph 7,28 7,35-7,45
• O2 sat 21,6 % 95-99
Kardiyak Arrestle Seyreden ve Tenekteplaz Kullanılan bir Masif Pulmoner Tromboemboli Olgusu | Arı Yılmaz et al.
62 www.respircase.com 62
Şekil 2: Akciğer grafisi, ekspiryum filmi olup mediastende hafif dolgun-
luk vardı. Belirgin parankimal patoloji izlenmedi.
Şekil 3: Tedavi öncesi kontrastsız toraks BT.
Şekil 4: Tedavi öncesi kontrastsız toraks BT.
Şekil 5: Tedavi öncesi kontrastsız toraks BT (Sağ ventriküler genişleme
ve kardiyak shift kırmızı ok).
Hasta klinik laboratuvar ve tetkik sonuçları ile rehberlere
göre yüksek riskli pulmoner tromboemboli olarak kabul
edildi. Trombolitik tedavi amaçlı kardiyoloji yoğun bakım
ünitesine göğüs hastalıkları adına yatırıldı. Ancak hasta
yatağına alındıktan hemen sonra kardiyak arrest oldu. Bu
nedenle hastaya transözefajial eko ya da dopler ultraso-
nografi için beklenilemedi. Acil karar verilmesi gereken
bir durumdu. Elimizde diğer trombolitik ajanlar yoktu.
Sistemden istem yapılması, eczaneden alınıp gelinmesi
yaklaşık 30 dakikalık bir zaman kaybı olacağından ve
hastanın bu süreyi bekleyecek zamanı olmadığından,
hasta yakınlarına durum ile ilgili bilgi verilip onamları
alındıktan sonra koroner yoğun bakım ünitesinde hazır
bulunan tenekteplaz hastaya puşe olarak verildi. Hastanın
yaklaşık 90-100kg civarı olduğu tahmin edilerek
50mg=10ml dozunda uygulandı Resüsitasyona devam
edilen hasta bir süre sonra sinüs ritminde döndü (Şekil 6).
Satürasyonları takibinde 10 dk. içinde %90'a kadar yük-
seldi. Koroner anjio yapılan bölgeye kum torbası ile bası
uygulandı. Takipte düşük moleküler ağırlıklı heparin
(DMAH) tedavisine geçildi. Koroner anjio yapılan bölgede
hematom gelişti. Hb:12,6 gr/dl’den 9,6 gr/dl'e geriledi.
Hastaya 1 ünite eritrosit replasmanı yapıldı. Kanaması
duran, satürasyonu %99'a kadar yükselen hasta servise
alındı. Serviste 1 ünite daha eritrosit replase edildi. War-
farin başlandı. INR'si 2-3 arasına gelen ve ek problem
saptanmayan hasta toplam 1 hafta sonrasında yürüyerek
taburcu edildi. Kontrolünde alt ekstremite venöz dopler
USG'de sol popliteal vende trombüs izlendi. Kreatinin
normale gerileyen hastanın Spiral Toraks BT anjiografi-
sinde tamamen düzelme saptandı (Şekil 7 ve 8). Warfarin
ile takibine devam edilmektedir.
Şekil 6: Tedavi sonrası EKG’si (Sinüs ritmi).
Respiratory Case Reports
Cilt - Vol. 9 Sayı - No. 2 63
TARTIŞMA
Bir doku plazminojen aktivatörü olan tenekteplazın miyo-
kard enfaktüsünde kullanım onayı olmasına rağmen he-
nüz pulmoner tromboembolide kullanım onayı mevcut
değildir. ST elevasyonu olan miyokard enfarktüsünde ilaç
dozu vücut ağırlığı baz alınarak hesaplanmaktadır. Öne-
rilen dozlar; <60 kg: 6.000 U, 30 mg, 6 ml. >=60-
<70 kg: 7.000 U, 35 mg, 7 ml. >=70-<80 kg: 8.000
U, 40 mg, 8 ml. >=80-<90: 9.000 U, 45 mg, 9
ml. >=90: 10.000 U, 50 mg, 10 ml’dir. Gerekli doz, 5-
10 saniye içerisinde, tek intravenöz bolus şeklinde uygu-
lanmaktadır (4). Pulmoner tromboembolide (PTE) de
kullanım dozu da benzerdir (5,6).
Pulmoner tromboembolide tenekteplaz kullanımı ile ilgili
literatürdeki ilk veriler 2001 yılına dayanmaktadır (7,8).
O dönemden beri tenekteplazın pulmoner embolideki
kullanımı olgu sunumları ve çalışmalara konu olmaktadır.
Akut miyokard enfarktüsünde alteplaz ve streptokinaz
karşısındaki güvenilirliğini ve etkinliğini kanıtlamış ve onay
almıştır. Aktif olarak kullanılmaktadır. Majör kanamaların
daha az olduğuna yönelik yayınlarda mevcuttur (9).
Şekil 7: Tedavi sonrası Kontrastlı Toraks BT Anjio (Sağ boşluklar ve
kardiyak septum normal).
Şekil 8: Tedavi sonrası Kontrastlı Toraks BT Anjio (Sağ boşluklar ve
kardiyak septum normal).
Ancak yüksek riskli pulmoner embolide kullanıma ilişkin
halen yeterli sayıda çalışma olmadığından rehberlere
girmeyi başaramamıştır ve çalışmalar devam etmektedir.
Nitekim 2019 ERS raporunda da çalışılmaya devam edi-
len ajanlar arasında gösterilmiştir (2).
Literatürde yüksek riskli pulmoner embolide alteplaz ile
tenekteplazı karşılaştıran bir çalışmaya rastlanılamadı.
Ancak orta ve yüksek riskli pulmoner embolide tenektep-
lazın plesebo heparin ya da streptokinaz ile karşılaştırmalı
yayınları mevcuttur:
Bu yayınların en büyüğü Meyer ve ark. (10) tarafından
yapılan orta riskli pulmoner embolili hastalarda tenektep-
laz+heparin ile plasebo+heparini karşılaştıran çok mer-
kezli, çift kör, randomize kontrollü çalışmadır. Trombolitik
tedavinin ilk 7. veya 30. günde mortaliteyi önemli ölçüde
azaltmadığı, ancak hemodinamik bozulmayı önlediği
bulunmuştur. Ancak, majör kanama tenekteplaz grubun-
da anlamlı olarak daha sıktı. Kline ve ark. (11) tarafından
yapılan bir başka randomize çalışmada da düşük mole-
küler ağırlıklı heparin (DMAH) ve DMAH + tenekteplaz
karşılaştırılmıştır. Üç aylık takipte tenekteplaz grubu daha
iyi prognoz, yaşam kalitesi ve fonksiyonel kapasite de artış
göstermiştir.
Becattini ve ark. (12) ise sağ ventrikül disfonksiyonu olan
hemodinamik olarak stabil pulmoner embolili hastalarda
tenekteplaz+heparin ile plesebo+heparini karşılaştırmıştır.
Miyokard enfarktüsü için kullanılan dozlarda kullanıldı-
ğında ciddi kanamalara neden olmadan sağ ventrikül
disfonksiyonunu 24 saatte belirgin şekilde düzelttiği gös-
terilmiştir.
Agrawal ve ark. (13) yaptıkları çalışmada 33 masif, 50
submasif, 20 nonmasif pulmoner embolili, 103 hastanın
62’sine tenekteplaz, 17’sine streptokinaz, 24’üne heparin
uygulanmıştır. Tenekteplaz’ın dispneyi daha belirgin azalt-
tığı ve hatta sağ dal bloğunu geri çevirmede %100 başa-
rılı olduğu saptanmıştır. Tenekteplaz grubunda tedavi
öncesi satürasyonu (SaO2) %88,79’dan tedavi sonra-
sı %96,90, 6 ay sonra %97,91’e çıkarken; streptokinaz
grubunda, tedavi öncesi SaO2 %91,90 tedavi sonra-
sı %94,90 6 ay sonra %90,09; heparin grubunda ise
tedavi öncesi SaO2 % 91,4 tedavi sonrası %92,75 6 ay
sonra %91,83 olarak saptanmıştır (13).
Shukla ve ark. (14), 30 pulmoner embolili hastanın dâhil
edildiği çalışmalarında, tenekteplaz’ı etkili ve güvenilir
olarak saptamışlardır. Yine bu çalışmada 4 sağ dal bloğu
olan hastanın 4’ününde dal blokları düzelmiş olarak
taburcu edilmiştir. Nitekim bizim olgumuzda da sağ dal
bloğu düzeldi ve entübasyona gerek kalmadan sinüs
ritmine döndü.
Kardiyak Arrestle Seyreden ve Tenekteplaz Kullanılan bir Masif Pulmoner Tromboemboli Olgusu | Arı Yılmaz et al.
64 www.respircase.com 64
Tenekteplaz kullanımı inravenöz bolus şeklinde olduğun-
dan ve oda sıcaklığında stabil olup sulandırılmadan kul-
lanılabildiğinden çoğu kez arrest vakalarında da kullanıl-
mış olup çok sayıda başarılı sonuçlar alınmıştır (15,16).
İleri yaş hastalarda ve gebelerde kullanımında bile ciddi
komplikasyon saptanmamıştır (17,18). Alteplaz ile karşı-
laştırıldığında, etkide azalma olmaksızın kanama riskinin
daha az olduğuna dair yayınlar mevcuttur (9,19).
Yüksek ve orta riskli pulmoner embolilerde kullanımına
ilişkin yurtdışı yayınlar giderek artmakta olmasına karşın,
ülkemizde pulmoner tromboembolide kullanımına yönelik
yayın bulunamamıştır. Sadece yurtdışı benzer olgu su-
numları mevcuttur (20-22). Bu nedenle olgumuzun litera-
türe katkı sağlayacağı düşünülmüştür.
Sonuç olarak; bizim olgumuzda elde edilen başarılı yanıt
ve diğer çalışmalara bakıldığında, tenekteplazın ileride
rehberlere girmesi için randomize kontrollü çalışmalara
ihtiyaç olduğu düşünülmektedir.
ÇIKAR ÇATIŞMASI
Bu makalede herhangi bir çıkar çatışması bildirilmemiştir.
YAZAR KATKILARI
Fikir - Y.A.Y., M.G.; Tasarım ve Dizayn - Y.A.Y., M.G.;
Denetleme - Y.A.Y., M.G.; Kaynaklar - Y.A.Y., M.G.;
Malzemeler - Y.A.Y., M.G.; Veri Toplama ve/veya İşleme
- Y.A.Y., M.G.; Analiz ve/veya Yorum - Y.A.Y., M.G.;
Literatür Taraması - Y.A.Y., M.G.; Yazıyı Yazan - Y.A.Y.,
M.G.; Eleştirel İnceleme - Y.A.Y., M.G.
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Respir Case Rep 2020;9(2): 66-69 DOI: 10.5505/respircase.2020.81489
OLGU SUNUMU CASE REPORT
66
Halil İbrahim Yakar1, Asiye Kanbay
2
Pulmonary endarterectomy (PEA) is a surgical treat-
ment approach for the treatment of patients with
chronic thromboembolic pulmonary hypertension
(CTEPH). The surgery is considered high risk in terms
of mortality and morbidity. We present here a patient
at high operative risk due to severe pulmonary hyper-
tension (PH) and chronic renal failure (CKD). The 22-
year-old male patient was undergoing hemodialysis
treatment for three years due to bilateral hydro-
nephrosis-induced CKD, and was admitted to the
hospital with increasing dyspnea. The diagnosis of
CTEPH was made clinically and radiologically. Dur-
ing the follow-up period, an endarterectomy was
planned, although the patient was considered high
risk due to insufficient clinical response, despite med-
ical therapy. Bilateral PEA was applied by thoracic
surgery team. sPAP decreased from 105 to 35 mmHg
on echocardiography, and dyspnea was improved
and functional capacity recovered after the PEA sur-
gery. A renal transplantation was contraindicated in
the preoperative period due to severe pulmonary
arterial hypertension. PEA surgery was enabled to
patient kidney transplantation due to an improvement
in pulmonary functions and pulmonary artery pres-
sure. We present here a case of CTEPH who had a
chance of kidney transplantation after undergoing
successful PEA surgery.
Key words: Chronic Thromboembolic Pulmonary
Hypertension, Chronic Renal Failure, Pulmonary En-
darterectomy.
Pulmoner endarterektomi (PEA), kronik tromboembo-
lik pulmoner hipertansiyon (KTEPH) hastalarında
cerrahi tedavi yöntemi olarak kullanılmaktadır. Diğer
yandan mortalite ve morbiditesi yüksek bir cerrahidir.
Burada ileri derece pulmoner hipertansiyonu (PH)
bulunan, kronik böbrek yetmezliği (KBY) nedenli
hemodialize giren ve bu nedenle yüksek riskle PEA
yapılan bir olgu sunuldu. Yirmi iki yaşında erkek
hasta, bilateral hidronefroza bağlı KBY nedeni ile 5
yıldır haftada 3 gün hemodialize girmekteydi. Gide-
rek artan nefes darlığı nedeniyle hastaneye başvurdu.
Klinik ve radyolojik olarak KTEPH tanısı konuldu.
Takiplerinde medikal tedaviye rağmen yeterli klinik
yanıt alınamaması nedeniyle hastaya yüksek riske
rağmen pulmoner endarterektomi planlandı. Göğüs
cerrahisi tarafından bilateral PEA operasyonu uygu-
landı. Hastanın kontrol ekokardiografisinde sPAP:
105 mmHg’dan 35 mmHg’ a geriledi. Cerrahi son-
rası takiplerinde hastanın istirahat dispnesi kayboldu,
eforla dispne şikâyeti belirgin azaldı ve fonksiyonel
kapasitesi arttı. Operasyon öncesi pulmoner hiper-
tansiyon nedeniyle hastada renal transplantasyon
kontrendike iken, postoperatif şikayetlerinin gerileme-
si ve pulmoner arter basıncı düşmesi nedeniyle hasta-
ya transplantasyon imkânı oluştu. Bu olgu sunumun-
da, başarılı bir PEA geçirdikten sonra böbrek nakli
imkânı oluşan bir KTEPH hastası sunuldu.
Anahtar Sözcükler: Kronik Tromboembolik Pulmoner
Hipertansiyon, Kronik Böbrek Yetmezliği, Pulmoner
Endarterektomi.
1Tokat Gaziosmanpaşa Üniversitesi Tıp Fakültesi Göğüs Has-
talıkları Anabilim Dalı, Tokat
2İstanbul Medeniyet Üniversitesi Tıp Fakültesi Göğüs Hastalıkları
Anabilim Dalı, İstanbul
1Department of Chest Disease, Tokat Gaziosmanpasa
University, Faculty of Medicine, Tokat, Turkey
2Department of Chest Disease, İstanbul Medeniyet University,
Faculty of Medicine, İstanbul, Turkey
Submitted (Başvuru tarihi): 15.12.2019 Accepted (Kabul tarihi): 04.02.2020
Correspondence (İletişim): Halil İbrahim Yakar, Tokat Gaziosmanpaşa Üniversitesi Tıp Fakültesi Göğüs Hastalıkları
Anabilim Dalı, Tokat
e-mail: [email protected]
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Chronic thromboembolic pulmonary hypertension
(CTEPH) is a disease with poor survival rates and increas-
ing frequency (1). Recurrent and organized thrombi oblit-
erate the pulmonary vascular system and cause progres-
sive pulmonary hypertension and right heart failure. Pul-
monary thromboendarterectomy (PEA) is the leading cu-
rative approach to CTEPH, and can be life-saving in
patients with severe pulmonary hypertension and proximal
thrombus in the pulmonary arteries. In pulmonary
endarterectomy operations, residual thrombus and fi-
brous obstructive tissue in the pulmonary arteries can be
extracted under conditions of deep hypothermic circulato-
ry arrest (2). In the postoperative period, pulmonary re-
vascularization, a decrease in pulmonary artery pressure
and clinical improvement can be expected due to the
removal of thrombus tissue. That said, it is a surgical
method associated with a high rate of mortality and mor-
bidity (3,4). We present here a patient considered a high
surgical risk due to CRF prior to PEA, and who gained the
possibility of renal transplantation after successful PEA
surgery.
CASE
A 22-year-old male patient who had undergone hemodi-
alysis three times a week for five years due to bilateral
hydronephrosis, was admitted to our hospital with exer-
cise dyspnea that had developed over the prior three
months (Functional Capacity 3-4). Prominent pulmonary
arteries and cardiomegaly were seen on chest X-ray (Fig-
ure 1), while p-pulmonale, right ventricular hypertrophy
and right bundle branch block were seen on ECG (Figure
2). Echocardiography (ECO) revealed enlargement of the
right heart cavity, severe pulmonary hypertension (sPAP:
105 mmHg) and paradoxical movement due to increased
pressure in the interventricular septum. A Thorax CT an-
giography showed an enlargement of the main pulmo-
nary artery and its branches, a nearly complete thrombus
that obstructed the lumen, extending from the bilateral
main pulmonary arteries to the segment branches, and
also pneumonic infiltration in the right upper lobe (Figure
3). Anticoagulant therapy and antibiotherapy were initiat-
ed for CTEPH and pneumonia.
As a result of the insufficient clinical response to antico-
agulant therapy, despite the three months of anticoagu-
lant therapy, no clinical improvement was observed, and
a PEA operation was planned, despite the high surgical
risk. A bilateral pulmonary endarterectomy with median
sternotomy under systemic hypothermia was successfully
performed by the thoracic surgery team (Figure 4). In the
control ECO, sPAP decreased to 35 mmHg.
In the postoperative follow-up, the patient's dyspnea
complaint reduced considerably and functional capacity
increased. The patient was referred to the Nephrology
Unit for renal transplantation for CRF.
DISCUSSION
CTEPH is one of the leading causes of PHT (5). In pa-
tients with pulmonary artery pressure (PAP) exceeding 50
mmHg, average five-year survival is reported to be 10%
(6). CTEPH should be considered in the differential diag-
nosis of patients presenting with chronic dyspnea and/or
chest pain. CTEPH should be investigated with a CT thor-
ax angiography and echocardiography in such cases.
Surgery is the optimum treatment for patients diagnosed
with CTEPH (7), although PEA has a high risk of morbidity
and mortality (8-10). Accordingly, medical treatment (oral
anticoagulant, CCB, ERA, Riociguat, etc.) is preferred in
patients with PAP> 50mmHg and in the presence of an
additional disease that may increase the risk of postoper-
ative mortality and morbidity (11,12). OAC was initiated
in our case due to sPAP: 105 (> 50) mmHg and CRF.
However, the PEA operation was planned due to the
patient’s inability to respond to medical treatment and the
patient's young age. While a renal transplant was contra-
indicated in the preoperative period due to PHT, a renal
transplantation was possible due to the regression of the
complaints, and the decrease in pulmonary artery pres-
sure in the postoperative period.
Figure 1: Initial Posteroanterior Chest X-ray (Bilateral pulmonary arteries
were prominent, and pneumonic infiltration surrounded the vascular
structures in the right upper lobe)
Pulmonary Thromboendarterectomy Enabling Renal Transplantation in Patient of Chronic Kidney Disease: A Case Report | Yakar et al.
68 www.respircase.com
Figure 2: ECG: Right bundle branch block and right ventricular hyper-
trophy (V1-V2 RSR pattern, ST depression and T wave inversion on right
precordial leads [V1-3])
Figure 3: Axial and coronal section on thorax CT angiography. En-
largement of the main pulmonary artery and its branches, widespread
thrombus obstructing the lumens, extending from the bilateral pulmonary
arteries to the segment branches, and pneumonic infiltration of the right
upper lobe were also observed
Figure 4: Postoperative view of thrombus material in the pulmonary
artery branches
CONCLUSION
Pulmonary endarterectomy is a curative treatment for
CTEPH. While renal transplantation was contraindicated
in the preoperative period due to PHT, it became possible
due to PEA in the postoperative period in our case. As
such, PEA is thought to be important for survival in CTEP
patients, and so all patients should carefully be evaluated
for PEA.
ABBREVIATIONS
CTEPH: Chronic thromboembolic pulmonary hyperten-
sion, sPAB: systolic pulmonary artery pressure, PTE: Pul-
monary thromboendarterectomy, CRF: Chronic renal
failure, ECO: Echocardiography, ECG: Electrocardiog-
raphy, OAC: Oral anticoagulant, PHT: Pulmonary
Hipertension, ERA: Endothelin receptor antagonist, CKB:
blockers.
ACKNOWLEDGEMENT
We thank Mr. Bedrettin Yildizeli for the patient's successful
surgical operation.
CONFLICTS OF INTEREST
None declared.
AUTHOR CONTRIBUTIONS
Concept - H.İ.Y., A.K.; Planning and Design - H.İ.Y., A.K.;
Supervision - H.İ.Y., A.K.; Funding - H.İ.Y.; Materials -
H.İ.Y.; Data Collection and/or Processing - H.İ.Y., A.K.;
Analysis and/or Interpretation - H.İ.Y., A.K.; Literature
Review - H.İ.Y.; Writing - H.İ.Y.; Critical Review - H.İ.Y.,
A.K.
YAZAR KATKILARI
Fikir - H.İ.Y., A.K.; Tasarım ve Dizayn - H.İ.Y., A.K.; De-
netleme - H.İ.Y., A.K.; Kaynaklar - H.İ.Y.; Malzemeler -
H.İ.Y.; Veri Toplama ve/veya İşleme - H.İ.Y., A.K.; Analiz
ve/veya Yorum - H.İ.Y., A.K.; Literatür Taraması - H.İ.Y.;
Yazıyı Yazan - H.İ.Y.; Eleştirel İnceleme - H.İ.Y., A.K.
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2. Sunar H, Yıldızeli B, Taş S, Yanartaş M, saçlı H, Kış M, et
al. Pulmonary endarterectomy in chronic thromboembolic
pulmonary hypertension. Turk Gogus Kalp Damar 2013;
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3. Olman MA, Auger WR, Fedullo PF, Moser KM. Pulmo-
nary vascular steal in chronic thromboembolic pulmonary
hypertension. Chest 1990; 98:1430-4. [CrossRef]
4. Lee KC, Cho YL, Lee SY. Reperfusion pulmonary edema
after pulmonary endarterectomy. Acta Anaesthesiol Sin
2001; 39:97-101.
5. Simonneau G, Robbins IM, Beghetti M, Channick RN,
Delcroix M, Denton CP, et al. Updated clinical classifica-
Respiratory Case Reports
Cilt - Vol. 9 Sayı - No. 2 69
tion of pulmonary hypertension. J Am Coll Cardiol 2009;
30 (1 Suppl):S43-54. [CrossRef]
6. Riedel M, Stanek V, Widimsky J, Prerovsky I. Longterm
follow-up of patients with pulmonary thromboembolism.
Late prognosis and evolution of hemodynamic and res-
piratory data. Chest 1982; 81:151-8. [CrossRef]
7. Galié N, Humbert M, Vachiery JL, Gibbs S, Lang
I,Torbicki A, et al. 2015 ESC/ERS Guidelines for the di-
agnosis and treatment of pulmonary hypertension: The
Joint Task Force for the Diagnosis and Treatment of Pul-
monary Hypertension of the European Society of Cardi-
ology (ESC) and the European Respiratory Society (ERS):
Endorsed by: Association for European Paediatric and
Congenital Cardiology (AEPC), International Society for
Heart and Lung Transplantation (ISHLT) Eur Heart J
2016;37:67-119. [CrossRef]
8. D'Armini AM, Zanotti G, Viganò M. Pulmonary endarter-
ectomy: the treatment of choice for chronic thromboem-
bolic pulmonary hypertension. Ital Heart J 2005; 6:861-
8.
9. Hagl C, Khaladj N, Peters T, Hoeper MM, Logemann F,
Haverich A, et al. Technical advances of pulmonary
thromboendarterectomy for chronic thromboembolic
pulmonary hypertension. Eur J Cardiothorac Surg 2003;
23:776-81. [CrossRef]
10. Mellemkjaer S, Ilkjaer LB, Klaaborg KE, Christiansen CL,
Severinsen IK, Nielsen-Kudsk JE, et al. Pulmonary
endarterectomy for chronic thromboembolic pulmonary
hypertension. Ten years experience in Denmark. Scand
Cardiovasc J 2006; 40:49-53. [CrossRef]
11. Rubin LJ, Hoeper MM, Klepetko W, Galiè N, Lang IM,
Simonneau G. Current and future management of chron-
ic thromboembolic pulmonary hypertension: from diag-
nosis to treatment responses. Proc Am Thorac Soc 2006;
3:601-7. [CrossRef]
12. Ghofrani HA, D'Armini AM, Grimminger F, Hoeper MM,
Jansa P, Kim NH, et al. Riociguat for the treatment of
chronic thromboembolic pulmonary hypertension. N Engl
J Med 2013; 369:319-29. [CrossRef]
Respir Case Rep 2020;9(2): 70-73 DOI: 10.5505/respircase.2020.13540
OLGU SUNUMU CASE REPORT
70
Derya Yenibertiz, Berna Akıncı Özyürek, Yurdanur Erdoğan
The most common and important side-effect of war-
farin treatment is bleeding. Erythema nodosum, in the
form of painful, erythematous nodules in the dermal
and subcutaneous tissues, is not a known side-effect
of warfarin. In this case, we report on erythema
nodosum occurring as a side-effect of warfarin treat-
ment. A 49-year-old female patient was treated for
pulmonary thromboembolism after multiple lesions
emerged identified as erythema nodosum in a der-
matology consultation that were interpreted as a
possible side-effect of warfarin treatment. It was con-
cluded that erythema nodosum may present as a
side-effect of warfarin.
Key words: Pulmonary thromboembolism, warfarin,
erythema nodosum.
Warfarin tedavisine bağlı en sık ve en önemli yan etki
kanamadır. Dermal ve subkutan dokularda ağrılı,
eritemli nodüllerden oluşan eritema nodozum warfa-
rinin bilinen bir yan etkisi değildir. Warfarine tedavisi-
ne bağlı gelişen eritema nodozum olgumuzu sunmayı
amaçladık. Pulmoner tromboemboli tedavisi başla-
nan 49 yaşında kadın hastada warfarin kullanımına
bağlı dermatoloji konsültasyonu ile eritema nodozum
olarak tanımlanan çok sayıda lezyon saptandı. War-
farin tedavisine bağlı bir yan etki olarak eritema no-
dozum görülebileceğini vurgulamak istedik.
Anahtar Sözcükler: Pulmoner tromboemboli, warfarin,
eritema nodozum.
Sağlık Bilimleri Üniversitesi, Atatürk Göğüs Hastalıkları ve Göğüs
Cerrahisi Eğitim Araştırma Hastanesi, Ankara
University of Health Sciences, Ataturk Chest Diseases and
Chest Surgery Training and Research Hospital, Ankara, Turkey
Submitted (Başvuru tarihi): 21.09.2019 Accepted (Kabul tarihi): 16.01.2020
Correspondence (İletişim): Berna Akıncı Özyürek, Sağlık Bilimleri Üniversitesi, Atatürk Göğüs Hastalıkları ve Göğüs Cerrahisi Eğitim
Araştırma Hastanesi, Ankara
e-mail: [email protected]
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Pulmonary thromboembolism (PTE) is the result of a clot
in the pulmonary artery or in one of its branches, and is
associated with high morbidity and mortality. Patient-
specific treatment is guided by signs and symptoms,
bleeding risk and comorbidities. Warfarin sodium – a
vitamin K antagonist – is an effective option for the treat-
ment of PTE, despite its narrow therapeutic index, its wide
inter-patient dosing variability, its predisposition to drug
and food interactions, and the need for close monitoring
of the intensity of the anticoagulation effect using the
international normalized ratio (INR). The most common
and important side effect of warfarin treatment is bleeding
(1). Several adverse skin manifestations have been asso-
ciated with the use of oral anticoagulants, ranging from
ecchymosis and purpura, hemorrhagic necrosis and
maculopapular vesicular urticarial eruptions, to purple
toes (2). While skin necrosis has been mentioned as the
most common dermatological side-effect of warfarin in
literature (2-7), we could find no studies in literature iden-
tifying erythema nodosum as a warfarin-related side-
effect.
In this case, we report that erythema nodosum may occur
as a side effect of warfarin treatment.
CASE
A 49-year-old, non-smoking, female patient applied to
our hospital emergency department with a sudden onset
of dyspnea and cough. The patient reported no other
symptoms, such as fever, malaise, fatigue, weight loss,
dysuria or sputum, and there were no signs of infection in
the patient. Upon hospitalization, thrombus was detected
in the subsegmentary arteries at the level of both postero-
basal segments in the lungs in a thorax computed tomog-
raphy angiography (Figure 1). The patient had neither
identified history of chronic medical disease nor a re-
markable family medical history. Her physical examina-
tion was normal and all laboratory parameters, include
C-reactive protein and white blood cells, were normal
with the exception of a raised d-dimer. Low molecular
weight heparin and warfarin treatment were started simul-
taneously; the patient was treated with no other drugs.
When the INR value reached the desired range due to the
effective dose of warfarin, the low molecular weight
heparin was stopped and treatment continued with warfa-
rin. On the 7th day of warfarin treatment, multiple painful,
swollen, nodular indurations emerged all over the body
(Figure 2 and 3). The patient was passed to the derma-
tology and allergy clinic, and the lesions were linked to
the warfarin treatment. Warfarin was stopped, and treat-
ment for erythema nodosum was carried out, as per the
dermatologist’s suggestion. The skin lesions regressed on
follow-up.
DISCUSSION
Erythema nodosum is a form of acute nodular septal
panniculitis, characterized by the sudden onset of ery-
thematous, firm, solid, deep nodules or plaques that are
painful on palpation, and localized mainly on the exten-
sor surfaces of the legs. It occurs more often in women
aged 25–40 years, but can be observed at any age (8).
Erythema nodosum may be linked to a variety of causes,
such as infection, medications, sarcoidosis, pregnancy,
inflammatory bowel disease, vaccination, autoimmune
disease and malignancy, among others. The condition is
idiopathic in approximately 50% of cases. Diagnosis is
generally made clinically, but a biopsy may be required in
atypical cases (9). A skin biopsy is generally not necessary
if the history and physical signs are suggestive of EN, and
the treatment of EN depends on the suspected or docu-
mented etiology, if known (10).
Our patient, a woman aged 49-year-old, presented with
lesions that were diagnosed as erythema nodosum by a
dermatologist. No biopsy was performed as the lesions
were considered typical. Most cases of erythema
nodosum are self-limited and require no treatment. Bed
rest and leg elevation are generally recommended to
reduce discomfort. Nonsteroidal anti-inflammatory drugs
are the first-line treatment for pain management. The
dermatologist started the patient on Colchium Dispert
and nonsteroidal anti-inflammatory drugs.
The present study emphasizes the rarity of erythema
nodosum as a side-effect of warfarin.
CONCLUSION
This article reports on a case of erythema nodosum that
emerged due to warfarin treatment for a pulmonary
thromboembolism.
Figure 1: Computed tomography angiography of the thorax
Erythema Nodosum due to Warfarin Treatment in Pulmonary Thromboembolism: A Case Report | Akıncı Özyürek et al.
72 www.respircase.com
Figure 2: Erythema nodosum on the leg
CONFLICTS OF INTEREST
None declared.
AUTHOR CONTRIBUTIONS
Concept - D.Y., B.A.Ö., Y.E.; Planning and Design - D.Y.,
B.A.Ö., Y.E.; Supervision - D.Y., B.A.Ö., Y.E.; Funding -
D.Y., B.A.Ö.; Materials - D.Y., B.A.Ö.; Data Collection
and/or Processing - D.Y., B.A.Ö.; Analysis and/or Inter-
pretation - E D.Y., B.A.Ö.; Literature Review - D.Y.,
B.A.Ö.; Writing - D.Y.; Critical Review - D.Y., B.A.Ö.
YAZAR KATKILARI
Fikir - D.Y., B.A.Ö., Y.E.; Tasarım ve Dizayn - D.Y.,
B.A.Ö., Y.E.; Denetleme - D.Y., B.A.Ö., Y.E.; Kaynaklar -
D.Y., B.A.Ö.; Malzemeler - D.Y., B.A.Ö.; Veri Toplama
ve/veya İşleme - D.Y., B.A.Ö.; Analiz ve/veya Yorum -
D.Y., B.A.Ö.; Literatür Taraması - D.Y., B.A.Ö.; Yazıyı
Yazan - D.Y.; Eleştirel İnceleme - D.Y., B.A.Ö.
Figure 2: Erythema nodosum on the arm
Respiratory Case Reports
Cilt - Vol. 9 Sayı - No. 2 73
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1. Ageno W, Gallus AS, Wittkowsky A, Crowther M, Hylek
EM, Palareti G. Oral anticoagulant therapy: antithrom-
botic therapy and prevention of thrombosis, 9th ed:
American College Of Chest Physicians Evidence- Based
Clinical Practice Guidelines. Chest 2012; 141: e44S-
e88S. [CrossRef]
2. Chan YC, Valenti D, Mansfield AO, Stansby G. Warfarin
induced skin necrosis. Br J Surg 2000; 87: 266-72.
[CrossRef]
3. Eichhoff G. Warfarin induced skin necrosis within psoriat-
ic plaques Dermatol Online J 2019; 25(6): pii:
13030/qt4gf5r5qk.
4. Tilton C, Livengood S, Hodges J, Marshall J. Warfarin-
induced skin necrosis in the presence of acute hepatic in-
jury and May-Thurner Syndrome. Hosp Pharm 2019; 54:
130- 4. [CrossRef]
5. Fraga R, Diniz LM, Lucas EA, Emerich PS. Warfarin-
induced skin necrosis in a patient with protein S deficien-
cy. An Bras Dermatol 2018; 93: 612- 3. [CrossRef]
6. Sklar LR, Messman A. An atypical case of warfarin-
induced skin necrosis. Clin Pract Cases Emerg Med 2017;
1: 359-61. [CrossRef]
7. Hamada T, Miyake T, Otsuka M, Iwatsuki K. Warfarin-
induced skin necrosis accompanied by aggravation of
vasculitis in a patient with cutaneous arteritis. Int J Der-
matol 2017; 56:779-81. [CrossRef]
8. Hafsi W, Badri T. Erythema Nodosum. [Updated 2019
Dec 20]. In: StatPearls [Internet]. Treasure Island (FL):
StatPearls Publishing; 2020 Jan-. Available from:
https://www.ncbi.nlm.nih.gov/books/NBK470369/
9. Leung AKC, Leong KF, Lam JM. Erythema nodosum.
World J Pediatr 2018; 14:548-54. [CrossRef]
10. Allen RA. Erythema Nodosum. Clinical Gate Dermatolo-
gy 18/ 03/ 2015. Available from:
https://clinicalgate.com/erythema-nodosum-2/
Respir Case Rep 2020;9(2): 74-78 DOI: 10.5505/respircase.2020.46547
OLGU SUNUMU CASE REPORT
74
Ezgi Çimen Çelik1, Serkan Yazgan
1, Soner Gürsoy
1, Ahmet Ucvet
1, Zekiye Aydoğdu
2
Amyloidosis is a disease that is characterized by an
extracellular accumulation of fibril proteins called
amyloid in tissues, and organ dysfunction. There are
various types of amyloidosis, with nodular pulmonary
amyloidosis usually considered a subtype of AL amy-
loidosis. Surgical excision is usually curative and the
prognosis is excellent. This case is presented to em-
phasize the rare occurrence of pulmonary amyloido-
sis and the need to keep malignancies in mind in
differential diagnosis.
Key words: Amiloidosis, breast cancer, metastasis.
Amiloidoz, dokularda amiloid adı verilen fibril yapı-
sındaki proteinlerin ekstrasellüler birikimi ve organ-
larda işlev bozukluğu ile seyreden hastalıktır. Çeşitli
tipleri olup, nodüler pulmoner amiloidoz, genellikle
AL amiloidozun bir subtipi olarak görülür. Cerrahi
eksizyon genellikle küratiftir ve prognoz mükemmel-
dir. Bu olgu, pulmoner amiloidozun nadiren görül-
mesi, ayırıcı tanıda malignitelerin akılda tutulması
gerekliliğini vurgulamak amacıyla sunulmuştur.
Anahtar Sözcükler: Amiloidoz, meme karsinomu,
metastaz.
1Department of Thoracic Surgery, University of Health Sciences,
İzmir Dr Suat Seren Chest Diseases and Surgery Medical Practice
and Research Center, İzmir, Turkey
2Department of Pathology, University of Health Sciences, İzmir Dr
Suat Seren Chest Diseases and Surgery Medical Practice and Re-
search Center, İzmir, Turkey
1SBÜ İzmir Dr. Suat Seren Göğüs Hasalıkları ve Cerrahisi
SUAM, Göğüs Cerrahisi Kliniği Kliniği, İzmir
2SBÜ İzmir Dr. Suat Seren Göğüs Hasalıkları ve Cerrahisi
SUAM, Patoloji Bölümü, İzmir
Submitted (Başvuru tarihi): 24.12.2019 Accepted (Kabul tarihi): 24.02.2020
Correspondence (İletişim): Ezgi Çimen Çelik, Department of Thoracic Surgery, University of Health Sciences, İzmir Dr Suat Seren
Chest Diseases and Surgery Medical Practice and Research Center, İzmir, Turkey
e-mail: [email protected]
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Amyloidosis is characterized by an accumulation of con-
gophilic amyloid fibrin deposits in the extracellular matrix
or organs (1). The most common forms are systemic AL
amyloidosis, systemic AA amyloidosis, systemic wild-type
ATTR amyloidosis, systemic hereditary ATTR amyloidosis
and localized AL amyloidosis (2). Nodular pulmonary
amyloidosis is a usual subtype of localized AL amyloidosis
(3) that may be associated with underlying inflammatory
or malignant conditions. Patients are generally asympto-
matic (4,5). Lesions are usually solitary, and may be mis-
taken for pulmonary malignancies. Biopsy material is
stained with Congo-Red to give a green color under a
polarized microscope, which is diagnostic (6). Excision of
the nodules is usually curative and the prognosis is excel-
lent (7). The case we present here is intended to empha-
size that pulmonary amyloidosis is rare and may be mis-
taken for malignancies.
CASE
A 76-year-old female patient who underwent a right mas-
tectomy 10 years previously for breast apocrine carcino-
ma followed by chemotherapy and radiotherapy, was
diagnosed with pulmonary nodules in radiological follow-
up and referred to us. No pathology was detected aside
from the old operation scar on the right breast upon
physical examination. Increased opacity was noted in the
left paracardiac area on chest radiography. A thorax
computed tomography (CT) showed a 13x15 mm solid
nodule in the left upper lobe (Figure 1 and 2), a 15x20
mm nodule in the lingual (Figure 3 and 4), and an 6x8
mm nodule in the left lower lobe (Picture 5). A positron
emission tomography/computed tomography (PET-CT)
revealed a 1.5x1 cm nodule (SUVmax: 1.6) in the left
upper lobe and a nodule of 2.2x1.4 cm (SUVmax: 1.4) in
the lingula (Figures 6, 7 and 8). Since the patient could
not be diagnosed through bronchoscopy and transthorac-
ic, a fine needle aspiration biopsy (TTFNAB), operation
was selected for diagnosis and treatment. In an explora-
tion with a left thoracotomy, four nodules in the left upper
lobe and one nodule in the lower lobe were palpated. A
wedge resection was performed on the three peripheral
nodules in the left upper lobe and in the lower lobe, and
the frozen section was examined. The frozen result report
read “The nodules may be breast carcinoma metastasis,
but a definitive diagnosis should be confirmed with further
investigations.” A left upper lobectomy was performed,
since the other central lesion in the upper lobe could not
be completely removed due to its closeness to the superi-
or pulmonary vein. The final pathology was reported as
nodular pulmonary amyloidosis (Figures 9, 10 and 11).
The patient was discharged on the 7th postoperative day
and is now in the 4th postoperative month.
Figure 1: Thorax computed tomography showing a 13x15 mm solid
nodule in left lung upper lobe
Figure 2: Thorax computed tomography showing a 13x15 mm solid
nodule in left lung upper lobe (Coronal section)
Figure 3: Thorax computed tomography showing a 15x20 mm nodule
in left lung lingual lobe
Nodular Pulmonary Amyloidosis Mimicking Breast Carcinoma Metastasis | Çimen Çelik et al.
76 www.respircase.com
Figure 4: Thorax computed tomography showing a 15x20 mm nodule
in left lung lingual lobe (sagittal section)
Figure 5: Thorax computed tomography showing a 6x8 mm nodule in
left lung lower lobe
Figure 6: PET /CT showing a 1.5x1 cm nodule (SUVmax: 1.6) in left
lung upper lobe
DISCUSSION
Nodular pulmonary amyloidosis, known also as nodular
parenchymal amyloidosis or nodular amyloidoma, is a
disease that is characterized by the presence of one or
more tumor-like amyloid deposits in the lung (1). It was
first described by Virchow in 1857 (7). It is rarely seen
among the amyloid diseases (3,6), and is characterized
primarily by a congophilic light chain amyloid (AL) depo-
sition in the extracellular matrix, and may be solitary or,
as with tour patient, multiple (1,8). It is usually diagnosed
in the sixth decade and is more common in men (1,9).
Patients are generally asymptomatic, as in our patient
(4,5), although it may cause cough, hemoptysis, dyspnea,
pleural effusion and pulmonary arterial hypertension,
depending on the location (10). It has no specific radio-
logical findings and mimics malignancies radiologically.
The present case emphasizes that pulmonary amyloidosis
is rare and may be mistaken for malignancies. Our pa-
tient was operated on due to the suspicion of breast car-
cinoma metastasis.
18-Fluoro-deoxyglucose (18F-FDG) PET-CT has emerged
as a tool for the diagnosis of pulmonary nodules to re-
duce invasive diagnostic examination. However, 18F-
FDG shows a small amount of uptake in malignancies
with low metabolic activity, such as bronchoalveolar can-
cer, carcinoid tumor and mucinous adenocarcinoma. As
such, despite low involvement on PET-CT (SUVmax: 1,6),
malignancies can be considered in a pre-diagnosis. Fur-
thermore, it has high metabolic rates alongside such non-
malignant conditions as tuberculosis, sarcoidosis and
rheumatoid nodules. In our case, and as in other rarely
reported cases, pulmonary nodular amyloidosis with low
or moderate 18F-FDG involvement can be seen (11). As
such, the results of an 18F-FDG PET-CT should be inter-
preted with caution in the differentiation of pulmonary
amyloidosis from other malignant or benign lesions. A
definitive diagnosis of localized pulmonary amyloidosis
requires histological confirmation (12). In cases in which
patient cannot be diagnosed via a CT-guided fine needle
aspiration biopsy, an invasive surgical resection may be
necessary. The patient in the present study could not be
diagnosed via TFNAB, and so exploratory thoracotomy
was decided upon. Furthermore, in the frozen examina-
tion, no differential diagnosis from malignancies could be
made.
Respiratory Case Reports
Cilt - Vol. 9 Sayı - No. 2 77
Figure 7: PET /CT showing a calcific nodul in left lung upper lobe
Figure 8: PET /CT showing a 2.2x1.4 cm nodule (SUVmax: 1.4) in left
lung lingula lobe
Figure 9: Congo red X 200
Figure 10: Acellular hyalinized material (H&E X200)
Figure 11: Acellular eosinophilic material (H&E X200)
Pulmonary amyloidosis may occur as a component of
localized or systemic amyloidosis (12,13). Localized AL
amyloidosis has a better prognosis than systemic amyloi-
dosis. The 10-year survival rate after surgery is reported
to be 97%. A surgical resection can be performed safely,
and the prognosis is excellent (13). Our patient continues
to be followed-up without problem in the postoperative
4th month.
In conclusion, local nodular pulmonary amyloidosis is a
rare and unusual tumor of the lung, and surgical treat-
ment is curative. Nodules may be solitary or multiple. It
should be kept in mind that amyloidosis may mimic both
benign and malignant pathologies, and should be con-
sidered in a differential diagnosis.
CONFLICTS OF INTEREST
None declared.
AUTHOR CONTRIBUTIONS
Concept - E.Ç.Ç., S.Y., S.G., A.U., Z.A.; Planning and
Design - E.Ç.Ç., S.Y., S.G., A.U., Z.A.; Supervision -
E.Ç.Ç., S.Y., S.G., A.U., Z.A.; Funding - E.Ç.Ç., Z.A.;
Materials – S.Y., E.Ç.Ç., Z.A.; Data Collection and/or
Processing - E.Ç.Ç., Z.A.; Analysis and/or Interpretation -
S.Y., S.G., E.Ç.Ç.; Literature Review - E.Ç.Ç.; Writing -
E.Ç.Ç.; Critical Review - S.Y., A.U., S.G.
YAZAR KATKILARI
Fikir - E.Ç.Ç., S.Y., S.G., A.U., Z.A.; Tasarım ve Dizayn -
E.Ç.Ç., S.Y., S.G., A.U., Z.A.; Denetleme - E.Ç.Ç., S.Y.,
S.G., A.U., Z.A.; Kaynaklar - E.Ç.Ç., Z.A.; Malzemeler -
S.Y., E.Ç.Ç., Z.A.; Veri Toplama ve/veya İşleme - E.Ç.Ç.,
Z.A.; Analiz ve/veya Yorum - S.Y., S.G., E.Ç.Ç.; Literatür
Taraması - E.Ç.Ç.; Yazıyı Yazan - E.Ç.Ç.; Eleştirel İnce-
leme - S.Y., A.U., S.G.
Nodular Pulmonary Amyloidosis Mimicking Breast Carcinoma Metastasis | Çimen Çelik et al.
78 www.respircase.com
REFERENCES
1. Khoor A, Colby TV. Amyloidosis of the Lung. Arch Pathol
Lab Med 2017; 141:247-54. [CrossRef]
2. Sipe JD, Benson MD, Buxbaum JN, Ikeda S, Merlini G,
Saraiva MJ, et al. Nomenclature 2014: amyloidfibril pro-
teins and clinical classification of the amyloidosis. Amy-
loid 2014; 21:221-4. [CrossRef]
3. Kaplan B, Martin BM, Boykov O, Gal R, Pras M, Shecht-
man I, et al. Co-deposition of amyloidogenic immuno-
globulin light and heavy chains in localized pulmonary
amyloidosis. Virchows Arch 2005; 447:756-61.
[CrossRef]
4. Beer TW, Edwards CW. Pulmonary nodules due to reac-
tive systemic amyloidosis (AA) in Crohn's disease. Thorax
1993; 48:1287-8. [CrossRef]
5. Roden AC, Aubry MC, Zhang K, Brady JO, Levin D,
Dogan A, et al. Nodular senile pulmonary amyloidosis: a
unique case confirmed by immunohistochemistry, mass
spectrometry, and genetic study. Human Pathology 2010;
41:1040-5. [CrossRef]
6. Howie AJ, Brewer DB. Optical properties of amyloid
stained by Congo red: history and mechanisms. Micron
2009; 40:285-301. [CrossRef]
7. Utz JP, Swensen SJ, Gertz MA. Pulmonary amyloidosis
The Mayo Clinic experience from 1980 to 1993. Ann In-
tern Med 1996; 124;4, 407-13. [CrossRef]
8. Milani P, Basset M, Russo F, Foli A, Palladini G, Merlini
G. The lung in amyloidosis. Eur Respir Rev 2017;
26(145): pii: 170046. [CrossRef]
9. Yang MC, Blutreich A, Das K. Nodular pulmonary amy-
loidosis with an unusual protein composition diagnosed
by fine-needle aspiration biopsy: a case report. Diagn
Cytopathol 2009; 37:286-9. [CrossRef]
10. Scala R, Maccari U, Madioni C, Venezia D, La Magra LC.
Amyloidosis involving the respiratory system: 5-year's ex-
perience of a multi-disciplinary group's activity. Ann
Thorac Med 2015, 10:212-6. [CrossRef]
11. Standaert C, Herpels V, Seynaeve P. A solitary pulmonary
nodule: pulmonary amyloidosis. J Belg Soc Radiol 2018;
102:20. [CrossRef]
12. Chen KT. Amyloidosis presenting in the respiratory tract.
Pathol Annu 1989; 24:253-273.
13. Baumgart JV, Stuhlmann-Laeisz C, Hegenbart U, Nat-
tenmüller J, Schönland S, Krüger S, et al. Local vs. sys-
temic pulmonary amyloidosis-impact on diagnostics and
clinical management. Virchows Arch 2018; 473:627-37.
[CrossRef]
Respir Case Rep 2020;9(2): 79-82 DOI: 10.5505/respircase.2020.99267
OLGU SUNUMU CASE REPORT
79
Ayse Baccioglu1, Ayse Füsun Kalpaklioglu
1, Tuba Devrim
2
Ultraviolet recall is a photodermatitis reaction that
can occur in prior ultraviolet burned skin during the
administration of systemic medication. No such reac-
tion has been reported with pirfenidone. We report
here on a 75-year-male patient who developed acute
erosive erythema on his face, forearms and hands
under pirfenidone treatment for idiopathic pulmonary
fibrosis after 4 months. The initial diagnosis was drug
eruption, since it developed after the initiation of
pirfenidone, in accordance with hypereosinophilia
and solar dermatitis on a skin biopsy, all of which
improved with discontinuation. However, the patient
tolerated the rechallange test with pirfenidone. The
presence of necrotic keratinocytes in a skin biopsy
and exaggerated dermatitis was unlikely for photo-
dermatitis, but supported an ultraviolet recall reaction.
Pirfenidone was resumed in a tapered dose, and the
patient was successfully followed up for 5 months for
a relapse of skin reaction, as well as IPF disease
activity. This case is important in indicating that the
drug can be tolerated with dose adjustment in the
presence of an ultraviolet recall reaction in contrast
to discontinuation need in drug allergy.
Key words: Drug eruption, phototoxicity, pirfenidone,
ultraviolet recall phenomen.
Ultraviyole hatırlama reaksiyonu, önceden ultraviyole
ışınına bağlı yanık gelişmiş kişinin sistemik ilaç kulla-
nımı sonrası benzer kliniğin gelişmesiyle karakterize
bir fotodermatittir. Pirfenidonla bu reaksiyon daha
önce bildirilmemiştir. Yetmiş beş yaşındaki erkek
hastada idiopatik pulmoner fibrozis için pirfenidon
başlandıktan 4 ay sonra güneş gören deri alanların-
da akut erosiv eritem gelişmişti. Önce pirfenidona
bağlı ilaç erüpsiyonu olduğu düşünüldü, çünkü; eşlik
eden hipereozinofilisi, deri biyopsisinde solar dermati-
tis sonucu vardı ve belirtiler pirfenidondan sonra
gelişmiş ve ilacın kesilmesinden sonra düzelmişti.
Ancak, oral provokasyon testinde hastanın ilacı tolere
etmesi, deri lezyonlarının basit bir solar dermatitis için
çok şiddetli olması ve deri biyopsisinde nekrotik kera-
tinositlerin görülmesi ultraviyole hatırlama reaksiyo-
nunu düşündürdü. Pirfenidon dozu düşürülerek yeni-
den başlandı, 5 aylık takip süresinde deri lezyonları
nüks etmedi ve İPF hastalığında atak gelişmedi. İlaç
allerjisinden farklı olarak pirfenidon ultraviyole hatır-
lama reaksiyonunda, tedaviye doz ayarlamasıyla
devam edilebileceğini göstermesi açısından bu olgu
önemlidir.
Anahtar Sözcükler: İlaç erupsiyonu, fototoksisite,
pirfenidon, ultraviyole hatırlama reaksiyonu.
1Department of Pulmonary Diseases, Kırıkkale University Faculty of
Medicine, Division of Immunology and Allergy, Kırıkkale, Turkey
2Department of Pathology, Kırıkkale University Faculty of Medi-
cine, Kırıkkale, Turkey
1Kırıkkale Üniversitesi Tıp Fakültesi, Göğüs Hastalıkları
Anabilim Dalı, İmmünoloji ve Allerji Bilim Dalı, Kırıkkale
2Kırıkkale Üniversitesi Tıp Fakültesi, Patoloji Anabilim Dalı,
Kırıkkale
Submitted (Başvuru tarihi): 04.12.2019 Accepted (Kabul tarihi): 25.02.2020
Correspondence (İletişim): Ayse Baccioglu, Department of Pulmonary Diseases, Kırıkkale University Faculty of
Medicine, Division of Immunology and Allergy, Kırıkkale, Turkey
e-mail: [email protected]
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Cilt - Vol. 9 Sayı - No. 2 80
Ultraviolet recall reaction is a type of photodermatitis that
occurs in ultraviolet-burned skin after the administration
of systemic medications (1). Such reactions are rare, and
have no report by pirfenidone, which is an oral antifibrot-
ic agent approved for patients with idiopathic pulmonary
fibrosis (IPF) (2). We present here a case of ultraviolet
recall photodermatitis by pirfenidone.
CASE
A 75-year-old Caucasian man was admitted with an
extraordinary sunburn like scaly erythema on his face,
forearms and hands that had emerged a few days earlier.
He had no recent sun exposure, and limited physical
movement that made it almost impossible for him to go
outside. He had been undergoing treatment with
pirfenidone (2403 mg/day) for IPF besides inhaled tiotro-
pium, and budesonide/formoterol for 4 months, and
subsequently developed a desquamative, burning and
erosive erythema limited on sun exposed areas such as
the forehead, scalp, face, neck, dorsa of the forearms
and hands (Figure 1). He had also vitiligo patches on his
face, neck and forearms surrounded by a reddish ul-
tralight-colored skin.
Laboratory tests revealed elevated eosinophil counts
(1400/mL) and a normal total leukocyte count
(8.600/mL), in addition to vitamin D deficiency
(10.55ng/dL, normal; 25–80ng/dL). Renal and hepatic
function tests were within normal limits. Serum ANA, ccp,
ANCA, ACE, anti-ds-DNA/jo1/scl70/sm/ssa/ssb were
also negative. Chest x-ray and pulmonary function tests
were stable when compared to previous values.
Figure 1: Severe erythema of the face (a), desquamative and erosive
erythema on the back of hands (b)
Histopathological findings of the biopsied forearm skin
revealed solar dermatitis with keratinocyte necrosis,
spongiosis with a lichenoid reaction, focal parakeratosis,
and irregular acanthosis in the epidermis, vacuolization,
perivascular lymphocytic cell infiltration and no specific
deposition of immunoglobulin or complements on the
basement membrane or vessel walls (Figure 2).
Pirfenidone was discontinued, and the patient was treated
with 40 mg/day (0.5 mg/kg/day) of intravenous methyl-
prednisolone for 10 days, with slight improvement. The
reaction was concluded to be a phototoxicity reaction in
the sun-exposed areas, in addition to solar dermatitis on
a skin biopsy. It was also considered to be a drug erup-
tion, having developed during pirfenidone treatment, in
accordance with serum hypereosinophilia, and after im-
provement was noted upon discontinuation. However, the
reaction time as of March was unexpected for photoreac-
tivity given the rarity of sunny days, and the solar dermati-
tis clinic was so severe with rapid progression. According-
ly, to confirm the diagnosis, an oral provocation test was
carried out 4 weeks after recovery in May, and the patient
was found to tolerate pirfenidone. The reaction was thus
concluded to be an ultraviolet recall reaction. The pres-
ence of necrotic keratinocytes in the skin biopsy was un-
likely for photodermatitis, but also supported this diagno-
sis. Even though the patient denied sun exposure, the
weather had been slightly sunny a few days before the
reaction, and this limited sun may have recalled the solar
dermatitis experienced in previous years to his extremely
white skin with vitiligo.
Pirfenidone was resumed in a tapered dose (1602
mg/day) following the complete regression of skin lesions
and the suggestion of ultraviolet recall dermatitis rather
than of drug eruption. The patient was successfully fol-
lowed up for 5 months for relapses of the skin reaction,
as well as disease activity of IPF.
DISCUSSION
Even though phototoxicity due to pirfenidone is a relative-
ly common reaction, this case report is important in its
rarity in terms of the tolerability of the drug after recovery.
The most common skin side-effects of pirfenidone have
been reported as 7.5% rash, 4.2% photosensitivity and
generalized pruritis in a single case, but no report of
ultraviolet recall reaction (3-6).
Photodermatitis following the Use of Pirfenidone in a Patient with Idiopathic Pulmonary Fibrosis: An Ultraviolet Recall Reaction | Baccioglu et al
81 www.respircase.com
Figure 2: Focal parakeratosis and irregular acanthosis in the superficial
layer, hydropic degeneration and necrotic keratinocytes in the epidermis
(H&E x40)
A diagnosis of ultraviolet recall reaction was made in the
presence of previous sunburn history accompanied by
systemic pirfenidone use, exaggerated clinic, skin biopsy
and tolerance in a rechallange test. Necrotic keratino-
cytes, epidermal spongiosis and vesiculation were sensi-
tive to ultraviolet recall reaction. Even though the history
of sun exposure was unremarkable, but had lived many
sunburn events in the past leaving behind photo aging
findings. The duration between sunburn and drug intake
varies, between days associated with chemotherapy med-
ications, and months in ampicillin (7,8). The mean dura-
tion between the initiation of pirfenidone and the devel-
opment of skin manifestations was 5.5 months in litera-
ture, and 4 months in the present case (3-6).
Photosensitivity reactions take two forms: phototoxicity,
occurring in sun exposed areas, whereas a generalized
rash is accepted as a photoallergic reaction. Photoaller-
gic reactions represent type IV hypersensitivity responses,
requiring a specific sensitization of the drug that makes
intolerance in rechallange (9). Phototoxicity has non-
immunological mechanism as direct cellular damage by
ultraviolet light (10). Ultraviolet recall refers to a photo-
toxicity reaction that initiates with irradiation and exacer-
bates with systemic medication (1). The reintroduction of
the drug or light exposure may not necessarily cause a
recurrence. Pirfenidone is known to become reactive
through the absorption of light, and the generation of
reactive oxygen radicals mediates to photo irritant results
(10). Another underlying mechanism is likely that
pirfenidone’s antifibrotic effect through suppressing some
inflammatory cytokines that are associated with viral reac-
tivation inducing cutaneous manifestation in some pa-
tients (4). Cofactors for photosensitivity are medications,
lupus and some vitamin deficiencies (9). The patient re-
fused systemic drugs other than pirfenidone before 4
weeks of the reaction time, and lupus was ruled out
based on clinical and negative collagen markers. The
patient’s low vitamin D level may be a result of long-time
sun avoidance, but it has been reported that photosensi-
tivity is not correlated with vitamin D levels (9). It may,
however, be possible to become more sensitive to sun-
light after winter.
Treatments of pirfenidone phototoxicity are topical emol-
lients and in mild-moderate events, while in severe cases,
systemic steroids may be required. A reduction in
pirfenidone dose may be a preventive action, since it has
been reported to be non-phototoxic in 30 mg/kg, but
phototoxic in 160 mg/kg (10). Even though ultraviolet
recall does not necessitate a withdrawal of therapy, many
adverse skin-related events related to pirfenidone may
indicate a discontinuation of the drug, and the appliance
of strict sun exposure may be helpful. In the event of a
reappearance of an adverse event or exacerbation
and/or progression of the disease, nintedanib, as an
alternative anti-fibrotic medication, would have been
started instead of pirfenidone. However, nintedanib was
not the first choice therapy in the present case as the
patient had been using an anticoagulation drug for car-
diac disease, and nintedanib may be associated with an
increased risk of bleeding.
Conclusion
This case report is interesting in that it reminds that a
photosensitive reaction experienced under pirfenidone
may not be due only to a drug allergy or solar dermatitis,
as it may also indicate an ultraviolet recall reaction,
which the drug can be tolerated in rechallange. Although
it has been reported to be a rare and moderate adverse
event, photosensitivity reaction can reduce quality of life
and to potentially increase the risk of the development of
skin cancer. Clinicians should pay particular attention to
risk factors for photosensitization, such as light skin color
and vitiligo, when making a decision for the treatment of
IPF.
CONFLICTS OF INTEREST
None declared.
AUTHOR CONTRIBUTIONS
Concept - A.B., A.F.K., T.D.; Planning and Design - A.B.,
A.F.K., T.D.; Supervision - A.B., A.F.K., T.D.; Funding -;
Materials - A.B.; Data Collection and/or Processing -
.Respiratory Case Reports
Cilt - Vol. 9 Sayı - No. 2 82
A.B., T.D.; Analysis and/or Interpretation - A.B.; Literature
Review - A.B.; Writing - A.B.; Critical Review - A.B., A.F.K.
YAZAR KATKILARI
Fikir - A.B., A.F.K., T.D.; Tasarım ve Dizayn - A.B., A.F.K.,
T.D.; Denetleme - A.B., A.F.K., T.D.; Kaynaklar -; Mal-
zemeler - A.B.; Veri Toplama ve/veya İşleme - A.B., T.D.;
Analiz ve/veya Yorum - A.B.; Literatür Taraması - A.B.;
Yazıyı Yazan - A.B.; Eleştirel İnceleme - A.B., A.F.K.
REFERENCES
1. Shiohara T, Mizukawa Y. Recall phenomenon: some
skin-resident cells remember previous insults. Dermatolo-
gy 2003; 207:127-9. [CrossRef]
2. Noble PW, Albera C, Bradford WZ, Costabel U, Glass-
berg MK, Kardatzke D, et al. Pirfenidone in patients with
idiopathic pulmonary fibrosis (CAPACITY): two random-
ized trials. Lancet 2011; 377:1760-9. [CrossRef]
3. Costabel U, Bendstrup E, Cottin V, Dewint P, Egan JJ,
Ferguson J, et al. Pirfenidone in idiopathic pulmonary fi-
brosis: expert panel discussion on the management of
drug-related adverse events. Adv Ther 2014; 31:375-91.
[CrossRef]
4. Droitcourt C, Adamski H, Polat A, Polard E, Kerjouan M,
Arnouat B, et al. Pirfenidone photosensitization in pa-
tients with idiopathic pulmonary fibrosis a case series. Br
J Dermatol 2018; 178:e222-3. [CrossRef]
5. Papakonstantinou E, Prasse A, Schacht V, Kapp A, Raap
U. Pirfenidone-induced severe phototoxic reaction in a
patient with idiopathic lung fibrosis. J Eur Acad Dermatol
Venereol 2016; 30:1354-6. [CrossRef]
6. Tsuruta A, Washio K, Fukunaga A, Nishigori C.
Pirfenidone-induced photoleukomelanoderma in a pa-
tient with idiopathic pulmonary fibrosis. J Dermatology
2016; 43:207-9. [CrossRef]
7. Basile FG, Creamer S. Docataxel/cylophomide induced
ultraviolet recall dermatitis. J Clin Oncology 2011;
29:e840-1. [CrossRef]
8. Krishnan RS, Lewis AT, Kass JS, Hsu S. Ultraviolet recall-
like phenomenon occurring after piperacillin, tobramycin,
and ciprofloxacin therapy. J Am Acad Dermatol 2001;
44:1045-7. [CrossRef]
9. Mang R, Stege H, Krutmann J. Mechanisms of phototoxic
and photoallergic reactions. In: Frosch PJ, Menné T, Le-
poittevin JP. (eds) Contact Dermatitis Springer, Berlin,
Heidelberg. 2006: 97-104. [CrossRef]
10. Seto Y, Inoue R, Kato M, Yamada S, Onoue S. Pho-
tosafety assessments on pirfenidone: photochemical,
photobiological, and pharmacokinetic characterization. J
Photochem Photobiol B 2013; 120:44-51. [CrossRef]
Respir Case Rep 2020;9(2): 83-86 DOI: 10.5505/respircase.2020.04796
OLGU SUNUMU CASE REPORT
83
Fatma Tokgoz Akyil1, Ahmet Topbas
1, Mustafa Akyıl
2
Congenital bronchial atresia (CBA) is a rare congeni-
tal airway malformation that is caused by an interrup-
tion to a proximal lobar, the segmental or subseg-
mental bronchus, hyperinflation and mucoid impac-
tion distal to the atresic bronchus. Patients may be
asymptomatic, or a cough, shortness of breath or
recurrent infection may be encountered. We present
here the case of a 21-year-old male who presented
with exertional dyspnea and cough on exertion, and
who was diagnosed with congenital bronchial atresia.
Key words: Congenital bronchial atresia, dyspnea,
mucocele.
Konjenital bronş atrezisi lober, segmenter veya sub-
segmenter bronşların atrezik sonlanması ve bu ne-
denle distalde oluşan havalanma artışı ve mukus
birikimi ile karakterizedir. Hastalar asemptomatik
olabileceği gibi; nefes darlığı, öksürük ve rekürren
enfeksiyonlar ile başvurabilir. Bu olgu sunumunda,
ağır eforla ortaya çıkan nefes darlığı ve öksürük ne-
deniyle başvuran ve konjenital bronş atrezisi tanısı
konulan 21 yaşında bir erkek hasta nadir görülmesi
nedeniyle sunulmuştur.
Anahtar Sözcükler: Dispne, konjenital bronş atrezisi,
mucocele.
1Department of Chest Diseases, Çanakkale Mehmet Akif Ersoy
State Hospital, Çanakkale, Turkey
2Department of Thoracic Surgery, Çanakkale Mehmet Akif Ersoy
State Hospital, Çanakkale, Turkey
1Çanakkale Mehmet Akif Ersoy Devlet Hastanesi, Göğüs
Hastalıkları Kliniği, Çanakkale
2Çanakkale Mehmet Akif Ersoy Devlet Hastanesi, Göğüs
Cerrahisi Kliniği, Çanakkale
Submitted (Başvuru tarihi): 31.10.2019 Accepted (Kabul tarihi): 24.01.2020
Correspondence (İletişim): Fatma Tokgoz Akyil, Department of Chest Diseases, Çanakkale Mehmet Akif Ersoy State Hospital,
Çanakkale, Turkey
e-mail: [email protected]
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Cilt - Vol. 9 Sayı - No. 2 84
Congenital bronchial atresia (CBA) is a rare congenital
airway malformation caused by the interruption of a prox-
imal lobar, segmental or subsegmental bronchus and
mucoid impaction, distal to the atresic bronchus. It is
more commonly reported in males, with an estimated
prevalence of 1.2 cases per 100,000 (1-3).
The most frequently involved segments are the left
apicoposterior segment and the left lower lobe, with esti-
mated rates of 64% and 14%, respectively. Radiologically,
bronchocele is a rounded branching opacity showing a
mucus-filled bronchus, and adjacent airway trapping or
emphysematous change may suggest is a suspicion of
bronchial atresia. In bronchoscopy, a blinding-ending
bronchus with classic radiographic features is diagnosti-
cally indicative of bronchial atresia (4,5).
Patients are mostly diagnosed incidentally in their second
or third decades. If the case is symptomatic, cough,
shortness of breath and recurrent infections may present
(6). We report here on a symptomatic young male diag-
nosed with bronchial atresia during his military service.
CASE
A 21-year old male presented with shortness of breath,
cough and sputum on exertion. The patient had been
fulfilling his conscripted military service for the past three
months, and was found to experience dyspnea during
heavy exercise. The patient had no additional diseases or
previously diagnosed lung disease. Over the previous five
years, he had been prescribed antibiotics for bronchitis
on three occasions. A physical examination was normal.
Spirometer forced expiratory volume in one second
(FEV1)/forced vital capacity (FVC) was 83% and FEV1
was 4.66 liters (81% of predicted). Bronchodilator revers-
ibility was negative. On a chest roentgenogram, a finger
in glove sign originating in the right hilum was noted
(Figure 1). A thorax high-resolution computed tomogra-
phy revealed mucoid impaction and peripheral hyperlu-
cency (Figure 2).
In an analysis of a complete blood count, white blood
cells were 13.8 (4.0-10.5) (µl/mlK/ml), and C-reactive
protein was 15 (0-5 mg/dl). Routine laboratory values
were within normal limits.
Upon suspicion of a bronchial abnormality, a fiberoptic
bronchoscopy was performed, and the right intermediate
bronchus was divided into sole lower lobe segments. The
patient was diagnosed with bronchial atresia (Figure 3),
and his military service was terminated with a report of
congenital bronchial atresia. The patient was educated
for possible complications and close follow-up was
scheduled.
DISCUSSION
This case report presents a unique cause of shortness of
breath, and is highly demonstrative of bronchial atresia.
The first case of CBA was defined by Ramsay et al. in
1953, and around 100 cases is have been reported in
literature to date (4,7). The leading locations of involved
bronchi are the apicoposterior segmental bronchus of the
left upper lobe, the right upper lobe, the middle lobe and
the right lower lobe, respectively [6]. Bronchial atresia
may present along with other congenital lung malfor-
mations, such as congenital cystic adenomatoid malfor-
mation, bronchopulmonary sequestration, congenital
lobar emphysema and lesions of mixed pathology (1,2,6).
In the present case, the middle lobe bronchus was affect-
ed and no coincident malformation was detected.
Figure 1: Chest X-ray with finger in glove sign, originating in the right
hilum
Figure 2: High-resolution computed tomography of the chest showing
mucoid impaction and peripheral hyperlucency
An Extremely Rare Cause of Dyspnea on Exertion: Bronchial Atresia | Tokgoz Akyil et al.
85 www.respircase.com
Figure 3: Endobronchial view from distal end of the right intermediate
bronchus
The exact mechanism of atresia is not yet known, but the
most accepted hypothesis is that the proliferating cells
lose their connection with the developing respiratory bud
during normal lung maturation. Another hypothesis is that
a repetitive vascular insult to lung parenchyma during
early fetal development leads to the obliteration of an
already completed bronchus (1,2).
Atretic bronchi do not communicate with the bronchial
tree. Instead, the bronchoalveolar channels of Lambert,
the pores of Kohn and the interbronchiolar channels
permit the entry of air, but prevent air escape, acting like
a one-way check valve. This results in hyperlucency and
hyperinflation in the distal of the atretic segment. Gener-
ally, mucus amasses, accumulating distal to the atretic
bronchus and creating a mucocele, and this accumula-
tion may lead to recurrent infections (7,8).
On a chest roentgenogram, hilar opacity in a tubular,
round, ovoid or branching structure may be observed.
Thorax CT reveals branching tubular or nodular opacities
radiating from the hilum with a “finger-in-glove” appear-
ance from the formation of mucoid impaction of the dis-
tal bronchus, known as a mucocele (2). Cavitary lesions
and air-fluid levels may be seen distal to the atresic bron-
chus (8–10). In present case, a finger-in-glove sign and
hyperinflation were noted.
Only one-third of diagnosed patients present with symp-
toms, with the most common symptoms being cough,
dyspnea and recurrent infection, although wheezing,
hemoptysis, chest pain and pneumothorax may also be
encountered. Hyperinflation and obstructive pulmonary
defects may cause dyspnea, although dyspnea is mostly
reported as exertional (4,10,11,12). The subject of the
present case study also suffered from dyspnea on exertion.
Prior to starting his military service, he had no symptoms.
Only during heavy exercise did he experience shortness of
breath.
Pathologically, a diagnosis of bronchial atresia is made
based on macroscopic findings, being the mucus plug-
ging of the prominent alveoli. There are usually no acute
or chronic inflammatory changes associated with bron-
chial atresia unless concomitant infection occurs
(1,10,12).
Differential diagnoses are bronchogenic cyst, lung apla-
sia, congenital lobar emphysema, congenital cystic ade-
nomatiod malformation, anomalous pulmonary venous
return, pulmonary sequestration, cystic fibrosis, allergic
bronchopulmonary aspergillosis (ABPA), and other benign
and neoplastic processes, (2) while radiological findings
may suggest CBA. Bronchoscopy is not compulsory for
diagnosis, but it may be necessary for the exclusion of
other bronchial abnormalities (4). In the present case,
radiological findings were suggestive of bronchial atresia,
and were verified from the bronchoscopic findings of the
atretic bronchus.
Follow-up is adequate for asymptomatic patients, while
recurrent infections and complications may require sur-
gery (2). The reported complications are recurrent infec-
tions, spontaneous pneumothorax and degradation of the
pulmonary parenchyma in the long term (1). The patient
reported no symptoms or infections prior to military ser-
vice, and military service was terminated after the diagno-
sis. At 6-months follow-up, the patient is asymptomatic,
and follow-up is continuing.
In conclusion, CBA is a rare airway malformation with
specific exceptional radiologic findings. Other than inci-
dental findings, patients with recurrent infections and
exertional dyspnea, the clinician should consider CBA as
a differential diagnosis.
CONFLICTS OF INTEREST
None declared.
AUTHOR CONTRIBUTIONS
Concept - F.T.A., A.T., M.A.; Planning and Design -
F.T.A., A.T., M.A.; Supervision - F.T.A., A.T., M.A.; Fund-
ing -; Materials - F.T.A., A.T., M.A.; Data Collection
and/or Processing - F.T.A., A.T., M.A.; Analysis and/or
Interpretation - F.T.A.; Literature Review - F.T.A.; Writing -
F.T.A.; Critical Review - F.T.A., A.T., M.A.
Respiratory Case Reports
Cilt - Vol. 9 Sayı - No. 2 86
YAZAR KATKILARI
Fikir - F.T.A., A.T., M.A.; Tasarım ve Dizayn - F.T.A., A.T.,
M.A.; Denetleme - F.T.A., A.T., M.A.; Kaynaklar -; Mal-
zemeler - F.T.A., A.T., M.A.; Veri Toplama ve/veya İşleme
- F.T.A., A.T., M.A.; Analiz ve/veya Yorum - F.T.A.; Lite-
ratür Taraması - F.T.A.; Yazıyı Yazan - F.T.A.; Eleştirel
İnceleme - F.T.A., A.T., M.A.
REFERENCES
1. Gipson MG, Cummings KW, Hurth KM. Bronchial atresia.
Radiographics 2009; 29:1531-5. [CrossRef]
2. Berrocal T, Madrid C, Novo S, Gutiérrez J, Arjonilla A,
Gómez-León N. Congenital anomalies of the tracheo-
bronchial tree, lung, and mediastinum: embryology, ra-
diology, and pathology. Radiographics 2004; 24:e17.
[CrossRef]
3. Schittny JC. Development of the lung. Cell Tissue Res
2017; 367:427-44. [CrossRef]
4. Mahajan AK, Rahimi R, Vanderlaan P, Folch E, Gan-
gadharan S, Majid A. Unique approach to diagnosing
and treating congenital bronchial atresia: a case series. J
Pulm Respir Med 2017; 7:2. [CrossRef]
5. Murat A, Ozdemir H, Yıldırım H, Kursad Poyraz A, Artas
H. Bronchial Atresia of Right Lower Lobe. Acta Radiol
2005; 46:480-3. [CrossRef]
6. Hutchison MJ, Winkler L. Bronchial Atresia. [Updated
2019 Jan 14]. In: StatPearls [Internet]. Treasure Island
(FL): StatPearls Publishing; 2019 Jan-.Available from:
https://www.ncbi.nlm.nih.gov/books/NBK537142/
7. Zylak CJ, Eyler WR, Spizarny DL, Stone CH. Develop-
mental lung anomalies in the adult: radiologic-
pathologic correlation. Radiographics 2002; 22:S25-43.
[CrossRef]
8. Psathakis K, Eleftheriou D, Boulas P, Mermigkis C,
Tsintiris K. Congenital bronchial atresia presenting as a
cavitary lesion on chest radiography: a case report. Cas-
es J 2009; 2:17. [CrossRef]
9. Karaman S, Deveci R, Bahçeci Erdem S, Karkiner A,
Alper H, Can D. Unusual radiological sign in bronchial
atresia. Turk Thorac J 2016; 17:79-81. [CrossRef]
10. Batchelor TJP, Rasburn NJ, Abdelnour-Berchtold E, Bru-
nelli A, Cerfolio RJ, Gonzalez M, et al. Guidelines for
enhanced recovery after lung surgery: recommendations
of the Enhanced Recovery After Surgery (ERAS®) Society
and the European Society of Thoracic Surgeons (ESTS).
Eur J Cardiothorac Surg 2019; 55:91-115. [CrossRef]
11. Traibi A, Seguin-Givelet A, Grigoroiu M, Brian E, Gossot
D. Congenital bronchial atresia in adults: thoracoscopic
resection. J Vis Surg 2017; 3:174. [CrossRef]
12. Wang Y, Dai W, Sun Y, Chu X, Yang B, Zhao M. Con-
genital bronchial atresia: diagnosis and treatment. Int J
Med Sci 2012; 9:207-12. [CrossRef]
Respir Case Rep 2020;9(2): 87-90 DOI: 10.5505/respircase.2020.62687
OLGU SUNUMU CASE REPORT
87
Ahmet Emre Hatır1, Büşra Özyalvaç
2, Sevgi Pekcan
2, Necdet Poyraz
3
Partial anomalous pulmonary venous return is a rare
congenital cardiac defect characterized by a flow of
blood from a few of the pulmonary veins returning to
the right atrium rather than the left atrium. Diagnosis
of this anomaly can be quite difficult. The condition is
usually diagnosed with echocardiography, and mag-
netic resonance imaging may also be useful. The
clinical manifestation and progression of the disease
varies depending on the amount of intra-cardiac
shunt. Signs or symptoms may include dyspnea,
coughing and fatigue. This defect usually causes no
negative symptoms and the child can develop nor-
mally. This research presents the case on an 8-year-
old female patient who complained of coughing and
whose abnormal findings on a chest X-ray led to a
further investigation. The patient was diagnosed with
partial anomalous pulmonary venous return. Diagno-
sis of this anomaly at an early stage can lead to the
patient gaining an awareness of the disease and
being able to deal with any complications that arise
in the adolescent period.
Key words: Partial anomalous pulmonary venous
connection, Atrial septal defects, Magnetic Resonance
Imaging.
Parsiyel anormal pulmoner venöz dönüş, pulmoner
venlerin bir kaçının sol atriyum yerine sağ atriyuma
geri dönmesi ile karakterize nadir görülen konjenital
bir kardiyak defekttir. Bu anomalinin tanısını koymak
oldukça zordur. Bu durum genellikle ekokardiyografi
ile teşhis edilir, ayrıca manyetik rezonans görüntüsü
de yararlı olabilir. Hastalığın klinik belirtisi ve seyri,
intrakardiyak şantın miktarına bağlı olarak değişir.
Belirti veya semptomlar; dispne, halsizlik ve öksürüğü
içerebilir. Bu anomali genellikle ciddi semptomlara
neden olmaz ve çocuk normal olarak büyür ve gelişir.
Bu çalışmada akciğer grafisinde anormal bulguları
olan ve öksürük şikayeti olan sekiz yaşında bir kız
hasta sunulmuş ve ileri araştırma sonucunda parsiyel
anormal pulmoner venöz dönüş tanısı konulmuştur.
Bu hastalığın tanısının erken konulması, hastanın bu
anomaliye olan farkındalığını artırarak ergenlik dö-
neminde ortaya çıkabilecek komplikasyonlar ile baş
etmesine olanak tanır.
Anahtar Sözcükler: Parsiyel anormal pulmoner venöz
dönüş anomalisi, Atriyal septal defektler, Manyetik
Rezonans Görüntüleme.
1Department of Family Medicine, Necmettin Erbakan University,
Konya, Turkey
2Department of Pediatrics, Necmettin Erbakan University, Konya,
Turkey
3Department of Radiology, Necmettin Erbakan University, Konya,
Turkey
1Necmettin Erbakan Üniversitesi Meram Tıp Fakültesi, Aile
Hekimliği Anabilim Dalı, Konya
2Necmettin Erbakan Üniversitesi Meram Tıp Fakültesi,
Çocuk Hastalıkları Anabilim Dalı, Konya
3Necmettin Erbakan Üniversitesi Meram Tıp Fakültesi,
Radyoloji Anabilim Dalı, Konya
Submitted (Başvuru tarihi): 27.12.2019 Accepted (Kabul tarihi): 26.02.2020
Correspondence (İletişim): Ahmet Emre Hatır, Department of Family Medicine, Necmettin Erbakan University, Konya, Turkey
e-mail: [email protected]
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Partial anomalous pulmonary venous return (PAPVR) is a
developmental disorder in which one or more pulmonary
vein cannot connect to the left atrium. PAPVR is usually an
acyanotic lesion (1), and while patients with this anomaly
usually have an asymptomatic course during childhood,
they may begin to show signs at an advanced age. This is
due to volume overload, and leads to complaints such as
fatigue, shortness of breath and coughing (2).
In this study, we present the case of an 8-year-old girl
who was admitted to our hospital with fever and vomiting,
and who had a previous history of hospitalization for
pneumonia. The patient underwent further investigations
after a suspicious lesion was identified on chest X-ray. As
a result, partial anomalous pulmonary venous return was
detected. We present this case report due to the sponta-
neous detection of PAPVR.
CASE
An 8-year-old female patient was brought to our hospital
who, it was learned, complained of coughing, especially
during the winter months, but that did not suffer severely
and did not have frequent illnesses. There was no pathol-
ogy related to birth, although there was a history of hos-
pitalization due to a urinary tract infection at 6 months
and pneumonia at 1 year of age. Her vaccines were
completed. She had no known disease and had no regu-
lar medication. She had 2 healthy siblings and there was
no kinship between her parents.
The height and weight percentiles were in the appropriate
range. An examination of the oropharynx was normal,
respiratory sounds were bilaterally equal and respiratory
sounds were normal. No murmur was heard in the cardi-
ovascular system.
There were no pathological features in laboratory tests. A
computed tomography (CT) was carried out as a suspi-
cious area was identified form the chest X-ray (Figure 1).
The pulmonary veins located in the upper lobe of the left
lung and the flow into the left brachiocephalic vein fol-
lowing the anterior aortic arch was reported (Figure 2).
Left lateral upper partial abnormal pulmonary venous
return was considered.
The patient consulted a pediatric cardiologist. An echo-
cardiography (Echo) was performed and a small atrial
septal defect (ASD) was detected, in addition to a partial
pulmonary venous return anomaly. A 4 mm left-to-right
shunt between the atria was observed. It was found that
the vertical vein collecting the flow originating from the
upper zone of the left lung was entering the innominate
vein. There was also mild insufficiency in the tricuspid
valve (4th degree). As a result of these findings and clini-
cal picture, the patient was not planned to be operated
on by the pediatric cardiologist, and medical follow-up
was recommended.
DISCUSSION
PAPVR was first described by Winslow in 1739 (3). When
an ASD is detected on an Echo, it is always necessary to
look for an associated PAPVR. It is difficult to differentiate
the symptoms, signs, electrocardiographic and radiologi-
cal findings of isolated ostium secundum ASD findings.
An Echo usually confirms the diagnosis. PAPVR is rare,
and has a good prognosis, similar to ostium secundum
ASD (1). PAPVR is examined in five different types, with
the type that connects to the superior vena cava (SVC)
being the most common. Abnormal pulmonary veins
connect to the lower side of the SVC or SVC-right atrial
junction in the most common type. Anomalies are fre-
quently seen with sinus venosus-atrial septal defects (4).
Our case had the most common type of PAPVR, and a
small ASD was detected. In a retrospective multi-slice
computer tomography (MSCT) study performed by Ho et
al. (5), PAPVR anomalies were encountered at a rate of
0.1% in 45,538 live cases.
Figure 1: The suspicious area indicated in blue is remarkable on the
Posterior-Anterior Chest X-ray
Figure 2: Axial MSCT images show that the left superior pulmonary vein,
indicated by the blue arrow in A, opens into the left brachiocephalic vein
at the point indicated by the blue arrow in B
Incidental Diagnosis of Partial Anomalous Pulmonary Venous Return: A Case Report | Hatır et al.
89 www.respircase.com
linical findings may vary in the left-to-right shunt ratio, in
the number and localization of abnormal veins, in the
presence of possible complications such as infection, in
the accompaniment of ASD, and in the size of such ASDs.
If the volume of the shunt is high, secondary pulmonary
hypertension may develop. Cases in which less than half
of the lung volume join the systemic circulation are
asymptomatic (6,7). Common signs and symptoms may
include dyspnea, coughing, fatigue, chest pain, palpita-
tions and tachycardia (5). Heart murmurs and arrhythmi-
as are also likely. In contrast to PAPVR, patients with scim-
itar syndrome are more likely to develop symptoms at an
early age and are more likely to have cyanosis (8).
The radiographical diagnosis of PAPVR is very difficult. If
PAPVR is accompanied by hypogenetic lung syndrome,
radiography plays an important role in the diagnosis (9).
Chest radiography is usually the first imaging method.
Radiographic findings depend on abnormal drainage, the
affected lobe and the left to right shunt. A chest X-ray may
be normal, or dilated SVC, cardiomegaly, right ventricu-
lar dilatation, right atrial dilatation, pulmonary edema or
right ventricular dilatation may be seen (10).
If a significant left-to-right shunt is identified in Echo,
there is a paradoxical interventricular septal movement,
and a right ventricular volume overload may develop.
Abnormal venous drainage to the right SVC may be more
difficult to detect unless a systematic approach is per-
formed. Surgical interventions should be considered in
cases with a volume-loaded right ventricle. Surgery is not
necessary when a single abnormally drained vein does
not load the right ventricle volume. Surgery is typically
performed at a similar time to ASD repair, when the pa-
tient is 3–5 years of age. The type of surgery depends on
the location of the drainage, but generally involves re-
connecting the abnormal vessels directly to the left. Pa-
tients with a repaired PAPVR have good prognosis (11).
In our case, minimal ASDs were seen in Echo. The patient
had no common symptom or infection, and so no cardio-
logic operation was planned, with follow-up recommend-
ed instead. In this study, we conclude that a chest X-ray
taken as the first approach, together with the patient's
clinic and history, provided us with important clues. Our
patient had a history of hospitalization due to pneumonia,
coughing especially in winter, and a suspicious area on a
chest radiography, leading to further investigations.
CONCLUSION
Patients can cope with the complications that may arise in
the adolescent period and at a more advanced age be-
cause of the diagnosis of partial anomalous pulmonary
venous return and patients also may be under control.
We can also learn that knowing the cause of the symp-
toms of the disease can prevent the patient from applying
to the hospital continuously and unnecessary hospitaliza-
tion can be prevented.
CONFLICTS OF INTEREST
None declared.
AUTHOR CONTRIBUTIONS
Concept - A.E.H., B.Ö., S.P., N.P.; Planning and Design
- A.E.H., B.Ö., S.P., N.P.; Supervision - A.E.H., B.Ö., S.P.,
N.P.; Funding - S.P., N.P.; Materials - S.P.; Data Collec-
tion and/or Processing - S.P., A.E.H.; Analysis and/or
Interpretation - S.P., N.P., A.E.H., B.Ö.; Literature Review
- A.E.H., S.P., B.Ö.; Writing - A.E.H., S.P., B.Ö.; Critical
Review - S.P.
YAZAR KATKILARI
Fikir - A.E.H., B.Ö., S.P., N.P.; Tasarım ve Dizayn -
A.E.H., B.Ö., S.P., N.P.; Denetleme - A.E.H., B.Ö., S.P.,
N.P.; Kaynaklar - S.P., N.P.; Malzemeler - S.P.; Veri Top-
lama ve/veya İşleme - S.P., A.E.H.; Analiz ve/veya Yorum
- S.P., N.P., A.E.H., B.Ö.; Literatür Taraması - A.E.H.,
S.P., B.Ö.; Yazıyı Yazan - A.E.H., S.P., B.Ö.; Eleştirel
İnceleme - S.P.
REFERENCES
1. Bernstein D. Partial anomalous pulmonary venous return.
In: Kliegman RM, St. Geme JW, Blum NJ et al, eds. Nel-
son textbook of pediatrics. 21th edition. Elsevier;
2019:2376.
2. Yüksekkaya R, Çelikyay F, Yılmaz A, Deniz Ç, Gökçe E.
Parsiyel anormal pulmoner venöz dönüş anomalisinin çok
kesitli bilgisayarlı tomografi anjiografi bulguları: İki ol-
gunun sunumu. Dicle Med J 2013; 40:128-30.
[CrossRef]
3. Winslow J. Mem Acad Roy Sci 1739:113.
4. Kirklin JW, Barrett-Boyes BG. Atrial septal defect and par-
tial anomalous pulmonary venous connection. Cardiac
surgery. 2nd edition, Vol 1. New York, NY: Churchill Liv-
ingstone 1993; 1:627-30.
5. Ho LM, Bhalla S, Bierhals A, Gutierrez F. MDCT of par-
tial anomalous pulmonary venous return (PAPVR) in
adults. J Thoracic Imaging 2009; 24:89-95. [CrossRef]
Respiratory Case Reports
Cilt - Vol. 9 Sayı - No. 2 90
6. Miller S, Waltman A. The pulmonary circulation. In:
Tavares JM, Ferrucci JT eds. Radiology: Diagnosis-
Imaging Intervention. Philedelphia: JB Lippincott;
1995;1-19.
7. Miller S, Waltman A. The pulmonary circulation. In:
Tavares JM, Ferrucci JT eds. Radiology: Diagnosis-
Imaging Intervention. Philedelphia: JB Lippincott;
1995;1-19. [CrossRef]
8. Vyas HV, Greenberg SB, Krishnamurthy R. MR imaging
and CT evaluation of congenital pulmonary vein abnor-
malities in neonates and infants. Radiographics 2011;
32:87-98. [CrossRef]
9. Neill CA, Ferencz C, Sabiston DC, Sheldon H. The famil-
ial occurrence of hypoplastic right hung with systemic ar-
terial supply and venous drainage "Scimitar syndrome".
Bull Johns Hopkins Hosp 1960; 107:1-21.
10. Katre R, Burns SK, Murillo H, Lane MJ, Restrepo CS.
Anomalous pulmonary venous connections. Semin Ultra-
sound CT MR 2012; 33:485-99. [CrossRef]
11. Webb GD, Smallhorn JF, Therrien J, Redington AN.
Congenital heart diseases. In: Bonow RO, Mann DL,
Zipes DP, Libby P eds. Braunwald's heart disease: a text-
book of cardiovascular medicine. 9th edition. Philadelph-
ia: Elsevier Saunders; 2011:1464-5.
Respir Case Rep 2020;9(2): 91-95 DOI: 10.5505/respircase.2020.04557
OLGU SUNUMU CASE REPORT
91
Esra Aktiz Bıçak, Zeki Korhan, Mustafa Bıçak, Osman Uzundere, Cem Kıvılcım Kaçar, Abdulkadir Yektaş
Negatif basınçlı pulmoner ödem üst solunum yolunda
meydana gelen akut tıkanıklık sonrası veya kronik
tıkanıklığın kalkmasına sekonder gelişebilen bir du-
rumdur. Biz bu olgu sunumunda apendektomi sonrası
gelişen negatif basınçlı pulmoner ödem sonrası geli-
şen şiddetli hipoksi için APRV modunun başarılı yöne-
timini bildiriyoruz. Bilinen sistemik bir hastalık öyküsü
olmayan, 33 yaşında, 85 kilo ağırlığında, erkek has-
taya akut apandisit ön tanısı nedeniyle genel cerrahi
kliniğince apendektomi operasyonu planlandı. Apen-
dektomi ameliyatına alındı ve sonrasında ciddi larin-
gospazm ve şiddetli inspiratuvar efor gelişti. Negatif
basınçlı akciğer ödemi gelişen hasta APRV moduyla
tedavi edilerek operasyon sonrası 5. günde servise
verildi. İnvaziv mekanik ventilasyonda hipoksinin
düzeltilemediği olgularda APRV modu kullanımının
hipoksiyi dramatik şekilde düzelttiğini düşünmekteyiz.
Anahtar Sözcükler: Negatif basınçlı akciğer ödemi,
airway pressure release ventilation, aspirasyon pnö-
monisi, apandektomi.
Negative pressure pulmonary edema is a condition
that may develop after an acute obstruction in the
upper respiratory tract, or may occur secondary to the
elimination of a chronic obstruction. In this case
study, we report on the successful management of the
APRV mode for severe hypoxia developing after a
negative pressure pulmonary edema that developed
after an appendectomy. An appendectomy operation
was planned by the general surgery clinic for a 33-
year-old male patient weighing 85 kg with no known
systemic disease history, due to a preliminary
diagnosis of acute appendicitis. The patient
underwent an appendectomy, after which severe
laryngospasm and severe inspiratory effort
developed. The patient, who developed a negative
pressure pulmonary edema, was treated with the
APRV mode and taken to the service on day 5
following the operation. We believe that the use of
the APRV mode can dramatically improve hypoxia in
cases where hypoxia cannot be corrected in invasive
mechanical ventilation.
Key words: Negative pressure pulmonary edema,
airway pressure release ventilation, aspiration pneu-
monia, appendectomy.
TC. SBÜ. Gazi Yaşargil Eğitim ve Araştorma Hastanesi, Diyarbakır,
Anesteziyoloji ve Reanimasyon Kliniği, Diyarbakır
Department of Anesthesiology and Reanimation, TR. HSU.
Gazi Yaşargil Training and Research Hospital, Diyarbakır,
Turkey
Başvuru tarihi (Submitted): 23.01.2020 Kabul tarihi (Accepted): 16.03.2020
İletişim (Correspondence): Abdulkadir Yektaş, TC. SBÜ. Gazi Yaşargil Eğitim ve Araştorma Hastanesi, Diyarbakır,
Anesteziyoloji ve Reanimasyon Kliniği, Diyarbakır
e-mail: [email protected]
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Negatif basınçlı pulmoner ödem (NBAÖ) üst solunum
yolunda meydana gelen akut tıkanıklık sonrası veya kro-
nik tıkanıklığın kalkmasına sekonder gelişebilen bir du-
rumdur. Ender olmasına karşın ciddi bir komplikasyondur
(1). NBAÖ’nün patofizyolojisinde üst hava yolu tıkanıklığı
sonucu zorlu-kuvvetli inspiryum çabasının, intratorasik
basıç artışına neden olduğu ve bunun da pulmoner in-
terstisyuma non-kardiyojenik sıvı geçişine yol açtığı bildi-
rilmektedir (2-4).
Airway pressure releasing ventilation (APRV), akut solu-
num sıkıntısı sendromu olan hastalarda düşük tidal ha-
cimli, yardımcı kontrol ventilasyonuna göre çeşitli avantaj-
lara sahip yeni bir pozitif basınçlı ventilasyon modudur
(5,6).
Biz bu olgu sunumunda, apendektomi sonrası, negatif
basınçlı pulmoner ödeme ikincil gelişen şiddetli hipoksi
için APRV modunun başarılı yönetimini bildiriyoruz.
OLGU
Bilinen sistemik bir hastalık öyküsü olmayan, 33 yaşında,
85 kilo ağırlığında, erkek hastaya akut apandisit ön tanısı
nedeni ile genel cerrahi kliniğince apendektomi operas-
yonu planlandı. Preoperatif değerlendirmede ASA 1 sını-
fında, sigara anamnezi bulunmayan, kronik bir hastalığı
ve geçirilmiş cerrahi öyküsü bulunmayan hastaya genel
anestezi altında operasyon planlandı. Hastanın preop
laboratuvar değerlerinde, lökosit: 15,7 103/UL, hemog-
lobin: 14,1 g/dL, trombosit: 260 103/UL, PT: 13,0 sn,
INR: 1,21 üre: 31 mg/dl, kreatinin: 0,93 mg/dl, AST: 32
U/L ALT: 21 U/L CRP: 4,6 mg/dl olarak saptandı. Hasta-
nın 8 saat açlık süresi sağlandıktan sonra operasyon
masasına alındı. Hasta premedikasyon uygulanmadan,
elektrokardiyogram, non invazif kan basıncı, periferik
oksijen satürasyonu ölçümü yapılacak şekilde monitörize
edildi. Hastanın kalp atım sayısı:84 /dk, arteryel tansiyon:
130/85 mmHg, SpO2: %98 olarak kaydedildi. Hastaya
18 G intraket ile antekubital venden intravenöz yol sağla-
nılarak 15 ml/kg’den serum fizyolojik verildi. Daha sonra
2 mg midazolam, 2 mg/kg propofol, 1mcg/kg fentanil ve
0,6 mg/kg rokuronyum yapılarak 8,0 F çaplı trakeal tüple
orotrakeal entübasyon gerçekleştirildi. Anestezi idame-
si %50 N2O/O2 ve %1-2 sevofluran ile sağlandı. Kırk beş
dakika süren operasyon sürecinde herhangi bir hemodi-
namik ve solunumsal komplikasyon yaşanmadı. Operas-
yon süresince hastaya toplam 1000 ml kristalloid sıvı
verildi. Cerrahinin tamamlanmasını takiben, spontan
solunum aktivitesi görülen hasta 1 mg neostigmin ve
0,5mg atropinle reverse edildi. Ekstübasyon aşamasında
herhangi bir sorunla karşılaşılmayan hastada birkaç daki-
ka içerisinde ciddi laringospazm ve şiddetli inspiratuvar
efor gelişti. SpO2 %85 altına düşmesi üzerine hasta %100
FİO2 ve non invazif CPAP ile ventile edildi. Hastaya 1
mg/kg prednol ve 1 mg/kg lidokain yapıldı. CPAP ile
SpO2 %85’lerde seyreden hasta yoğun bakım ünitesine
alındı. Hasta 8F entübasyon tüpü ile tekrar 0,9 mg/kg
rokuronyum, 2 mg/kg propofol ve 100 mcg fentanil ile
entübe edilerek 0,1 mcg/kg/dk remifentanil infüzyonu ile
sedasyon sağlandı. Sağ internal juguler venden 7F santral
venöz kateter ve sağ radial arterden 20 G intraket ile
arterial kateter takıldı. Hastanın endotrakeal tüpünde hafif
kanlı köpüklü balgam mevcuttu. Hastanın fizik muayene-
sinde dinleme bulgusu olarak her iki hemitoraksta yaygın
krepitan raller bulunması üzerine arteryal kan gazı alındı.
Hastaya AP akciğer filmi çekildi (Şekil 1). FiO2 %100 iken
ve SIMV modunda PEEP 12 cmH2O, Solunum sayısı
16/dk ve PEEP üstü basınç 20 cmH2O iken arter kan
gazında pH: 7,15, PCO2: 58 mmHg PO2: 57mmHg
SpO2: %82 olması üzerine toraks BT çekildi (Şekil 2).
Bunun üzerine hasta PRVC moduna alındı solunum sayısı
16/dk, TV 390 mL, PEEP 12 cmH2O, PEEP üstü basınç
20 cmH2O ve Ptepe alarm üst limiti 35 cmH2O olarak
ayarlandı. İki saat bu şekilde takip edilen hastanın SpO2
si FiO2 %100 iken %85 in üzerine çıkmadı. Hastadan
kardiyoloji konsültasyonu istendi ve yapılan yatak başı
EKO da hastada kalp yetmezliği olmadığı teyit edildi.
Toraks BT’de bilateral konsolidasyon-buzlu cam görüntü-
leri mevcut olan hastada solunum sıkıntısının laringos-
pazm sonrası gelişmesi, fizik muayene, klinik süreç ve
laboratuvar incelemeleri ile tanı, negatif basınçlı akciğer
ödemi ve aspirasyon pnömonisi olarak düşünüldü. İnha-
ler bronkodilatatör, metilprednizolon 250 mg, aminofilin
240 mg ve 40 mg furosemid yapıldı. Ampirik antibiyote-
rapi (meropenem 3X1gr ve vankomisin 2X1gr) başlandı.
İki saat sonra çalışılan kan gazında oksijenasyonu düzel-
meyen hasta APRV modunda takip edilmeye başlandı.
Aynı zamanda sedasyon olarak midazolam 5
mg/kg/h’den ve ketamin 0,5 mg/kg/h’den başlandı.
APRV başlangıç ayarları Phigh: 30 cmH2O, Plow: 0
cmH2O Thigh: 5 sn Tlow: 1sn olarak ayarlandı. Hastanın
takip süreçlerinde APRV moduna alındıktan 2 saat sonra
çalışılan arter kan gazında FiO2: %70 iken pH: 7,33,
PCO2: 40 mmHg, PO2: 105 mmHg SpO2: %95’e yük-
seldi. Hastanın 2. gün mekanik ventilatör ayarları
FiO2: %50 iken Phigh: 25 cmH2O, Plow: 8 cmH2O,
Thigh: 1sn Tlow: 1sn’e düşüldü. Yatışının 2. gününde
çekilen AP akciğer grafisinde minimal bir düzelme vardı
(Şekil 3). Yatışının 3. gününde arter kan gazında pH:
7,43, PCO2: 37 mmHg, PO2: 158 mmHg olan hasta
Negatif Basınçlı Akciğer Ödemi ve İnvazif Mekanik Ventilasyonda APRV Modu: Olgu Sunumu | Yektaş et al.
93 www.respircase.com
CPAP’a alındı. FiO2: %50, PEEP: 8 cmH2O ve PEEP üstü
basıncı: 20 cmH2O olarak ayarlandı. CPAP değerleri
kademeli olarak düşürülen hasta 4. gününde extübe
edildi. Ekstübasyon sonrası AP-akciğer filmi tamama ya-
kın düzeldi (Şekil 4). Hasta 5. gününde genel cerrahi
servisine transfer edildi.
TARTIŞMA
NBAÖ’ye özellikle sağlıklı genç erişkinlerde, göğüs içi
yüksek negatif basınç oluşturabildikleri için daha sık rast-
lanır. Erkeklerde daha sıktır (%80) ve ASA fiziksel durum
P1 ve P2 de %73’dür. Ayrıca obez, üst hava yolu darlığı
olan, kısa boyun, üst hava yolları ile ilişkili operasyon,
uyku apne sendromu ve mediyastinal kitlesi bulunan has-
talarda diğer risk grubunu oluşturmaktadır (7).
Şekil 1: PA akciğer grafisinde bilateral parakardial heterojen parankimal
infiltrasyonlar.
Şekil 2: Toraks tomografisinde bilateral konsolidasyon-buzlu cam görün-
tüleri.
Şekil 3: İkinci gün PA akciğer grafisinde minimal regresyon.
Şekil 4: Ekstübasyon sonrası PA akciğer grafisinde tama yakın regresyon.
Yapılan bir çalışmada hastalar iki gruba ayrılmış ve has-
taları ekstübe etmek için sugammadex kullanan grupta
negatif basınçlı pulmoner ödem genel anestezi pratiğin-
de % 0,009, sugammadex kullanmayan grupta % 0 ora-
nında görülmüştür. Hastaların gruplara ayrılmadığı başka
bir çalışmada ise NBAÖ sıklığı % 0,1-% 11 aralığında
bulunmuş ve laringospazm sonrası ise NBAÖ sıklığının %
3’e ulaştığı bildirilmiştir (7,8).
Respiratory Case Reports
Cilt - Vol. 9 Sayı - No. 2 94 94
NBAÖ, tekrarlanan obstrüksiyonlarla zorlu inspiriyum
yapılması sonucu hızlı negatif intraplevral basınç artışıyla
ilişkilidir. NBAÖ ile ilişkili akut hava yolu obstrüksiyonu-
nun nedeni laringospazmdır ve orofaringeal, baş ve bo-
yun cerrahisi bu riski arttırır. İntratorasik negatif basıncın
artışı venöz dönüşü arttırır ve bu da pulmoner venöz ba-
sıncı yükseltir. Artan vasküler permeabilite de sıvının pul-
moner kapillerlerden alveoler boşluğa geçişine katkıda
bulunur. Solunum yetmezliğine bağlı hipoksi ve asidozis
alveoler memran kapiller yaralanmayla ilişkili olarak
pulmoner vasküler rezistansı arttırır, oluşan dispne, asido-
zis ve hiperadrenerjik cevabı arttırarak diffüz alveoler
hemoraji ve kardiyak sorunlara da neden olabilir (3,4).
İnspiratuvar plevral basınç normalde (-2) – (-5) cmH2O
aralığında olup, tıkanıklığa karşı zorlu inspiryum eforu
sırasında intraplevral negatif basınç -100 cmH2O civarı-
na kadar çıkabilmektedir (3,4). NBAÖ’de ödemin daha
çok postoperatif dönemde gelişen laringospazm veya
preoperatif üst hava yolu patolojilerine bağlı gelişen akut
üst hava yolu obstrüksiyonundan sonra hızlı bir şekilde
ortaya çıktığı görülmüştür (9).
NBAÖ’nün klinik seyri tipiktir. Ayırıcı tanıda aspirasyon,
kardiyojenik pulmoner ödem ve sıvı yüklenmesi düşünül-
melidir (10). Bizim olgumuzda intraoperatif olarak hasta-
ya verilen sıvının normal değerlerde olması, ek bir kardi-
yak sorunun olmaması, dinlemekle yeni oluşan rallerin
bulunması ve EKO’sunun normal olması bizi nonkardiyo-
jenik akciğer ödemine yöneltti. Ayrıca ekstübasyon sonrası
laringospazm yaşamamız ve takiben tablonun oluşması,
akciğer grafisi ve akciğer tomografi bulgularının tanımızı
desteklemesi NBAÖ tanısını güçlendirdi.
Olgumuzda, pulmoner ödemin medikal tedaviye cevap
vermemesi, noninvazif solunum desteğinin yetersiz kalma-
sı reentübasyon ihtiyacının olması, konvansiyonel invazif
modlara yanıt vermemesi ve invazif mekanik ventilasyon
modu olarak kullanılan APRV modunun hipoksiyi drama-
tik olarak düzeltmesi dikkat çekicidir.
NBAÖ’de tanı konulması, yeterli oksijenasyonun sağlan-
ması ve pozitif hava yolu basıncı uygulanması tedavide
esastır. Ciddi NBAÖ tedavisinde agresif hemodinamik
monitörizasyon, reentübasyon ve invazif mekanik ventilas-
yon düşünülmelidir (11). Bu olguda ciddi bir NBAÖ tab-
losu ile karşılaşıldı ve tedavi protokolü ve yapılması gere-
ken agresif monitorizasyon yapıldı. Burada özellikle dikkat
çekmek istediğimiz nokta, bizim olgumuzda da olduğu
gibi non-invazif solunum desteğinin yetersiz kaldığı hasta-
larda tekrar entübasyon kararı vermekte gecikilmemelidir.
Yeterli sedasyon altında invazif solunum desteği sağlanan
hastalar, kısa sürede tedaviye cevap vermektedirler.
APRV modu, gaz dağılım volümü ile akciğer volümünün
uyumunu optimalleştirerek alveoler aşırı gerilimini engel-
ler, ek olarak spontan ventilasyon eforlarında intermittant
release periodda Plow esnasında alveoler ventilasyonu
arttırır ve CO2’i uzaklaştırır. APRV konvansiyonel modlara
göre verilen tidal volümle Pplato ve Ppeak basıncını daha
fazla düşürür. APRV daha az sedasyon ve paralizise izin
verir. Bu mod zorunlu spontan ventilasyon modu oldu-
ğundan farmakolojik paraliziden kaçınılmış olur. Diafram
hareketlerine izin verdiği için de ventilasyon/perfüzyon
oranı iyileşmektedir (5).
Postoperatif NBAÖ nedenli APRV uygulaması hakkında
çok fazla sayıda literatür yoktur. APRV, transalveolar ba-
sınç gradyendini sınırlandırırken alveoler rekruitment en
üst düzeye çıkararak pulmoner disfonksiyon yönetimine
yardımcı olur ve böylece barotravma riskini de azaltır
(5,9,10,11).
Sonuç olarak; NBAÖ nadir görülen fakat yüksek morbidi-
te ve mortalite ile sonuçlanabilen bir durumdur. Riskli
olguların belirlenmesi, özellikle ekstübasyon işlemi esna-
sında laringospazm gelişen genç-erişkin ve erkek hasta-
larda dikkatli olunması ve akciğer ödemi bulguları oluşur-
sa ayırıcı tanıda NBAÖ tanısının mutlaka göz önünde
bulundurulması gerekmektedir. NBAÖ tedavisinde üst
hava yolu obstrüksiyonunun erken tanınması, hipoksinin
önlenmesi, gerekirse reentübasyon ve invazif mekanik
ventilasyonun sağlanması unutulmamalıdır. İnvazif meka-
nik ventilasyonda hipoksinin düzeltilemediği olgularda
APRV modu kullanımının hipoksiyi dramatik şekilde düzel-
teceğini düşünmekteyiz.
ÇIKAR ÇATIŞMASI
Bu makalede herhangi bir çıkar çatışması bildirilmemiştir.
YAZAR KATKILARI
Fikir - E.A.B., Z.K., M.B., O.U., C.K.K., A.Y.; Tasarım ve
Dizayn - E.A.B., Z.K., M.B., O.U., C.K.K., A.Y.; Denetle-
me - E.A.B., Z.K., M.B., O.U., C.K.K., A.Y.; Kaynaklar -;
Malzemeler -; Veri Toplama ve/veya İşleme - A.Y.; Analiz
ve/veya Yorum - A.Y.; Literatür Taraması - A.Y.; Yazıyı
Yazan - A.Y.; Eleştirel İnceleme - A.Y.
KAYNAKLAR
1. Oswalt CE, Gates GA, Holmstrom MG. Pulmonary ede-
ma as a complication of acute airway obstruction. JAMA
1977; 238:1833-5. [CrossRef]
2. Schwartz DR, Maroo A, Malhotra A, Kesselman H. Nega-
tive pressure pulmonary hemorrhage. Chest 1999;
115:1194-7. [CrossRef]
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95 www.respircase.com
3. Liu R, Wang J, Zhao G, Su Z. Negative pressure pulmo-
nary edema after general anesthesia: A case report and
literature review. Medicine (Baltimore) 2019; 98:e15389.
[CrossRef]
4. Xiong J, Sun Y. Negative pressure pulmonary edema: a
case report. BMC Anesthesiol. 2019; 19:63. [CrossRef]
5. Fredericks AS, Bunker MP, Gliga LA, Ebeling CG,
Ringqvist JR, Heravi H, et al. Airway pressure release ven-
tilation: a review of the evidence, theoretical benefits,
and alternative titration strategies. Clin Med Insights Circ
Respir Pulm Med 2020; 14:1179548420903297.
[CrossRef]
6. Morimoto Y, Sugimoto T, Arase H, Haba F. Successful
management using airway pressure release ventilation for
severe postoperative pulmonary edema. Int J Surg Case
Rep 2016; 27:93-5. [CrossRef]
7. Silva LAR, Guedes AA, Salgado Filho MF, Chaves LFM,
Araújo FP. Negative pressure pulmonary edema: report
of case series and review of the literature. Rev Bras
Anestesiol 2019; 69:222-6. [CrossRef]
8. Kao CL, Kuo CY, Su YK, Hung KC. Incidence of nega-
tive-pressure pulmonary edema following sugammadex
administration during anesthesia emergence: A pilot au-
dit of 27,498 general anesthesia patients and literature
review. J Clin Anesth 2020; 62:109728. [CrossRef]
9. Choi E, Yi J, Jeon Y. Negative pressure pulmonary ede-
ma after nasal fracture reduction in an obese female pa-
tient: a case report. Int Med Case Rep J 2015; 8:169-71.
[CrossRef]
10. Harmon E, Estrada S, Koene RJ, Mazimba S, Kwon Y.
Concurrent Negative-Pressure Pulmonary Edema (NPPE)
and Takotsubo Syndrome (TTS) after Upper Airway Ob-
struction. Case Rep Cardiol 2019; 2019:5746068.
[CrossRef]
11. Tsai PH, Wang JH, Huang SC, Lin YK, Lam CF. Charac-
terizing post-extubation negative pressure pulmonary
edema in the operating room-a retrospective matched
case-control study. Perioper Med (Lond) 2018; 7:28.
[CrossRef]
Respir Case Rep 2020;9(2): 96-98 DOI: 10.5505/respircase.2020.78300
LETTER TO EDITOR EDİTÖRE MEKTUP
96
To the Editor,
A 22-year old woman presented to the chest dis-
eases clinic with complaints of non-productive
cough and left side chest pain, ongoing for two
days. She had been admitted to the hospital with a
sudden abdominal pain three days earlier. She was
diagnosed with an abdominopelvic abscess with a
ruptured dermoid cyst, and a salpingo-
oopherectomy was performed. The left side chest
pain initiated three days later.
Upon physical examination, vital signs were normal
and room air oxygen saturation was 98%. Respira-
tory sounds in the bilateral lower chest were de-
creased. An instant chest radiograph revealed
blunt costophrenic angles suggestive of a bilateral
pleural effusion (Figure 1).
Upon ED admission, biochemistry values were
within normal limits, complete blood count param-
eters revealed a mild leukocytosis of 12.6 K/L,
hemoglobin was decreased to 8.9 g/dl, and C-
reactive protein (CRP) was 154 mg/L. On the day
of the consultation, a complete blood count re-
vealed leukocytosis of 9.6 K/L, hemoglobin of
10.1 g/dl and CRP of 58 mg/L. She had neither
fever nor sputum. D-dimer was 812 ng/mL.
Upon ED admission, no pleural effusion was seen
at the proximal sections in an abdominal computer
tomography (CT), although ascites and a minimal
pericardial effusion were detected (Figure 2). The
pleural fluid was exudative according to Light’s
criteria (1); the pleural fluid to serum ratio for pro-
tein was 0.62 (3.6 vs. 5.8 g/dl, respectively); and
the pleural fluid to serum ratio for lactate dehydro-
genase was 0.89 (197 vs. 219 U/L, respectively). A
cytological examination identified the usual pleural
fluid cells, and no proliferation of microbiologic
agents.
Figure 1: Chest radiograph at the time of admission
Figure 2: Preoperative abdominal computer tomography
Figure 3: Two months after admission
RES
PIR
ATO
RY
CA
SE R
EPO
RTS
Respiratory Case Reports
Cilt - Vol. 9 Sayı - No. 2 97
A cardiac examination revealed also a 2-centimeter
pericardial effusion. No ascites were found in a
novel abdominal ultrasonography.
The rapid occurrence of a post-operative pleural
effusion led us to consider a reactional pleuro-
pericardial effusion. A chest radiograph two days
later showed a decreased left side pleural effusion.
The patient was informed that the fluid would regress
in a few weeks.
The patient was discharged the following day and
prescribed antibiotics and anti-inflammatory drugs.
One week later, she was re-admitted to the hospital
with continuous side pain. A cardiac evaluation
detected a regressed pericardial effusion with no
tamponade finding. This time, to exclude a pulmo-
nary embolism, she underwent a thorax CT angi-
ography, revealing no thrombus. Excluding the other
possible diagnoses, the patient was pre-diagnosed
with non-classic Meigs-like syndrome. All symptoms
were resolved two months later, and a chest radio-
graph showed complete regression (Figure 3).
Meigs’ syndrome is a rare finding in coexistence with
a benign ovarian tumor of fibroma, or a fibroma-
like tumor, ascites, pleural effusion and a curative
resection of the tumor (2). A relationship with other
benign ovarian tumors and other symptoms is de-
fined as non-classic Meigs’ syndrome (3).
Only a few studies examining the coexistence of
pericardial effusion alongside Meigs’ syndrome have
been published to date, in which the condition is
referred to as “Meigs-like syndrome”. A postopera-
tive increase in pleural effusion has been reported in
only a single patient with an 8-year history of fibro-
ma (4). In line with this case report, our case had
pericardial effusion and ascites, and developed
pleural effusion along with pericardial effusion after
surgery.
A pleural effusion may occur just immediately after
the operation, in which regressed ascites and peri-
cardial effusion may have overflowed into the pleu-
ral cavity. We believe that our patient differs from
those analyzed in literature in her acute presentation
of a ruptured dermoid cyst. An accurate diagnosis of
non-classic Meigs-like syndrome was accomplished
2 months after the operation, when the effusion had
completely regressed.
This case report is important in revealing that the
development of a pleural effusion following opera-
tions on benign ruptured ovarian tumors is an en-
countered phenomenon. While pericardial effusion
regresses rather early, the complete regression of a
pleural effusion may take longer. Taking this syn-
drome into account, informing the patient and a
close follow-up may allow needless investigations
and examinations to be avoided.
Fatma Tokgoz Akyil1
, Mustafa Akyıl2
1Department of Chest Diseases, Çanakkale Mehmet Akif
Ersoy State Hospital, Çanakkale, Turkey
2Department of Thoracic Surgery, Çanakkale Mehmet Akif
Ersoy State Hospital, Çanakkale, Turkey
Correspondence (İletişim): Fatma Tokgoz Akyil, Department
of Chest Diseases, Çanakkale Mehmet Akif Ersoy State
Hospital, Çanakkale, Turkey
e-mail: [email protected]
CONFLICTS OF INTEREST
None declared.
AUTHOR CONTRIBUTIONS
Concept - F.T.A., M.A.; Planning and Design -
F.T.A., M.A.; Supervision - F.T.A., M.A.; Funding -;
Materials - F.T.A, M.A.; Data Collection and/or
Processing - F.T.A., M.A.; Analysis and/or Interpre-
tation - F.T.A.; Literature Review - F.T.A.; Writing -
F.T.A.; Critical Review - F.T.A., M.A.
YAZAR KATKILARI
Fikir - F.T.A., M.A.; Tasarım ve Dizayn - F.T.A., M.A.;
Denetleme - F.T.A., M.A.; Kaynaklar -; Malzemeler -
F.T.A., M.A.; Veri Toplama ve/veya İşleme - F.T.A.,
M.A.; Analiz ve/veya Yorum - F.T.A.; Literatür Tara-
ması - F.T.A.; Yazıyı Yazan - F.T.A.; Eleştirel İncele-
me - F.T.A., M.A.
REFERENCES
1. Light RW, Macgregor MI, Luchsinger PC, Ball WC Jr.
Pleural effusions: the diagnostic separation of transu-
dates and exudates. Ann Intern Med 1972; 77:507-
13. [CrossRef]
2. Meigs JV. Fibroma of the ovary with ascites and
hydrothorax‐ Meigs syndrome. Am J Obstet Gyne-
col 1954; 67:962-85. [CrossRef]
Respiratory Case Reports
98 www.respircase.com
3. Krenke R, Maskey-Warzechowska M, Korczynski P,
Zielinska-Krawczyk M, Klimiuk J, Chazan R, Light RW.
Pleural Effusion in Meigs' Syndrome-Transudate or
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