912 CID 2001:33 (15 September) BRIEF REPORTS

B R I E F R E P O R T

Probable Atypical Cat Scratch DiseasePresenting as Isolated PosteriorPancreatic Duodenal Lymphadenitisand Abdominal Pain

Boris Dzelalija,1 Miro Petrovec,2 and Tatjana Avsic-Zupanc2

1Department of Infectious Diseases, General Hospital Zadar, Zadar, Croatia;and 2Medical Faculty, Institute of Microbiology and Immunology, Ljubljana,Slovenia

We report a case involving a 15-year-old girl with atypical,

clinically unsuspected cat scratch disease (CSD) presenting

as isolated posterior pancreatic duodenal lymphadenitis, fe-

ver, and abdominal pain. The serological, abdominal ultra-

sonographic, and CT findings, as well as clinical and epi-

demiological data, indicate that B. henselae was likely an

etiologic agent of CSD in our patient.

Cat scratch disease (CSD) is a syndrome that is characterized

by regional lymphadenopathy after a cat scratch or bite distal

to the involved lymph node. In addition, many patients have

atypical presentations other than regional adenopathy, such as

Parinauds syndrome [1], neurological syndromes (mainly en-

cephalopathy and neuroretinitis) [2, 3], self-limited granulo-

matous hepatitis [46], splenitis [4, 5], osteitis [7], atypical

pneumonitis [8], endocarditis [9], and a syndrome of pro-

longed fever of unknown origin (in children) [10]. On the basis

of the numerous lines of evidence reported since 1990, it is

reasonable to ascribe the majority of CSD cases to Bartonella

henselae [6, 7]. However, it remains likely that occasional typ-

ical CSD cases can be caused by other agents, such as Afipia

felis [11, 12] and Bartonella clarridgeiae [13]. In this article, we

present what we believe is the first case of isolated posterior

pancreatic duodenal lymphadenitis and abdominal pain asso-

ciated with B. henselae infection.

Case report. A previously healthy 15-year-old girl was ad-

mitted to the Department of Infectious Disease of the General

Hospital Zadar (Croatia) in July 1999. She had a high fever

Received 23 January 2001; revised 3 April 2001; electronically published 21 August 2001.

Reprints or correspondence: Boris Dzelalija, General Hospital Zadar, B. Percica 5, 23 000Zadar, Croatia (boris.dzelalija@zd.tel.hr).

Clinical Infectious Diseases 2001; 33:9124 2001 by the Infectious Diseases Society of America. All rights reserved.1058-4838/2001/3306-0027$03.00

(temperature, 39.2C) and abdominal discomfort, accompanied

by anorexia, nausea, vomiting, and severe rightupper-quad-

rant abdominal pain. During the week before the patient was

admitted to the hospital, the symptoms were much less intense.

She had no cutaneous lesions and/or peripheral adenopathy,

and the remainder of the physical examination findings were

normal.

Two months earlier, the girl had received a 10-week-old kit-

ten, which she often kissed on the mouth, but she did not recall

any local infection from scratches. Five days after admission,

her 10-year-old brother presented with left axial adenopathy

and fever (temperature, 39.5C). These symptoms appeared

4 weeks after a cat scratch on the left wrist, and CSD was

suspected. The diagnosis of CSD was supported by serological

testing.

At the time of admission, the erythrocyte sedimentation rate

was 103 mm/h. The peripheral WBC count was cells/911.9 10

L (62% segmented neutrophils, 15% band forms, 16% lym-

phocytes, and 7% monocytes), the hemoglobin concentration

was 12.8 g/dL, and the platelet count was platelets/9563 10

L. Blood chemistry values (creatinine, blood urea nitrogen, total

protein, albumin, globulin, immunoglobulin, amylase, serum

aspartate aminotransferase, alanine aminotransferase, bilirubin,

creatinine phosphokinase, alkaline phosphatase, glucose, cal-

cium, etc.) were normal. Routine urinalysis yielded unremark-

able findings. The results of multiple bacterial cultures of blood

and urine, performed before the administration of antibiotics,

were negative. Ultrasonography revealed a peripancreatic mass

that was cm in diameter.1.6 4.2 3.8

Two days after admission, abdominal CT showed that the

liver and spleen appeared to be normal, but it also revealed a

low-density mass, cm in diameter, located at the2.5 3 5

inferior vena cava, posterior surface of the head of the pancreas,

and descending part of the duodenum and portal vein, con-

sistent with enlargement of the posterior pancreatic duodenal

lymph nodes (figure 1). Findings of chest radiography, upper

endoscopy, and iv urography were normal. To exclude possible

causes of lymphadenopathy, a tuberculin skin test was per-

formed, but the results were negative. Paired serum samples,

which were obtained 11 and 25 days after the onset of illness,

were retrospectively tested by indirect immunofluorescence as-

say (IFA) for antibodies to Epstein-Barr virus/viral capsid an-

tigen, Toxoplasma gondii, cytomegalovirus, B. henselae, and Bar-

tonella quintana. Results of serological testing (table 1)

indicated past immunity to Epstein-Barr virus and cytomega-

by guest on August 4, 2014

http://cid.oxfordjournals.org/D

ownloaded from

http://cid.oxfordjournals.org/

BRIEF REPORTS CID 2001:33 (15 September) 913

Figure 1. In the reported case of cat scratch disease, abdominal CTrevealed a low-density mass ( cm in diameter; arrow) con-2.5 3 5sistent with enlargement of the posterior pancreatic duodenal lymphnodes.

Table 1. Results of serological examination with regard to thedifferential diagnosis of atypical cat scratch disease.

Test or serum sample

Result or titer

Sample 1 Sample 2

VCA/IgG EBV Positive Positive

VCA/IgM EBV Negative Negative

CMV IgG Positive Positive

CMV IgM Negative Negative

Toxoplasma gondii IgG Negative Negative

T. gondii IgM Negative Negative

Bartonella henselae IgG 11:4096 1:4096

B. henselae IgM 1:1280 1:640

Bartonella quintana IgG 1:1024 1:1024

B. quintana IgM 1:320 1:320

NOTE. CMV, cytomegalovirus; EBV, Epstein-Barr virus; VCA, viral capsidantigen.

lovirus, as well as high titers of IgG and IgM antibody to B.

henselae and B. quintana.

The clinical impression was of acute cholecystitis, and the

patient was treated intravenously with cefuroxime (1.5 g q8h)

and gentamicin (160 mg q.d.) for 1 week. Defervescence oc-

curred on day 4 of antibiotic therapy, and the patient was

discharged from the hospital on day 12. Two days after with-

drawal of antibiotics, the laboratory findings showed an eryth-

rocyte sedimentation rate of 91 mm/h and a WBC count of

cells/L with normal differential count. Six weeks after98.9 10

discharge, the erythrocyte sedimentation rate, WBC count, and

hemoglobin, aspartate aminotransferase, alanine aminotrans-

ferase, and lactate dehydrogenase levels, as well as the other

blood chemistry values, were normal.

At a follow-up visit 2 weeks after discharge, the patient ap-

peared to be healthy, and abdominal ultrasonography showed

mild regression of the lymph node enlargement. Eleven weeks

after the onset of the illness, abdominal ultrasonography was

repeated, and it revealed significant regression; 3 weeks later,

resolution of the pancreatic duodenal lymph node enlargement

was evident.

Discussion. CSD was not the initial diagnosis for our pa-

tient. Severe, sudden, intermittent, cramping abdominal pain

and fever were the chief complaints, and because of these symp-

toms, the initial diagnosis was acute cholecystitis. The main

diagnostic indicators of CSD in our patient were the epide-

miological and clinical findings for her brother that were typical

of CSD.

The absence of peripheral lymphadenopathy and cat

scratches make diagnosis of CSD difficult. There have been

several recent reports that describe hepatosplenic CSD and ab-

dominal pain as the clinical entity [5, 6]. Due to the insidious

and nonspecific nature of the fever and abdominal pain of CSD

hepatitis and splenitis, diagnosis may be delayed until a history

of cat exposure prompts ultrasonographic or CT abdominal

imagingwhich usually demonstrates multiple lesions (mi-

croabscesses)and serological testing [1417].

Our patient had neither hepatosplenomegaly nor abnormal

liver function test values. Abdominal ultrasonographic and CT

findings of a low-density mass consistent with enlargement of

the posterior pancreatic duodenal lymph nodes indicated a di-

agnosis of lymphoma or acute abscess. We did not explain the

mechanism of the abdominal pain in this case. Possible mech-

anisms that we have considered include peritoneal irritation

and ischemia. CSD is thought to be caused by the inoculation

of B. henselae by the scratch of a cat or kitten [6]. Fleas have

also been suggested as a vector for this organism, which might

explain the absence of cat scratches in some cases [18]. We

believe that the spread of infection to the lymph nodes was

most likely hematogenous.

The patient appeared to improve clinically very quickly.

Three days after antibiotic treatment was started, clinical im-

provement occurred. This observation suggests that antibiotics

shorten the course of the illness, although the effect of anti-

biotics in the treatment of typical as well as atypical CSD is

controversial [7, 12, 1921].

Our patient had elevated IFA titers of species-specific IgG

and IgM to both B. henselae and B. quintana in acute and

convalescent-phase serum samples. However, titers of antibody

to the former pathogen were significantly higher. This may be

explained by the recognized cross-reactivity among different

species of Bartonella [1416]. The role of B. henselae and B.

by guest on August 4, 2014

http://cid.oxfordjournals.org/D

ownloaded from

http://cid.oxfordjournals.org/

914 CID 2001:33 (15 September) BRIEF REPORTS

clarridgeiae in the clinical syndrome known as CSD is not clear.

B. quintana has not been recovered from any patient with CSD

or from any associated cats, although seroreactivity to B. quin-

tana antigen has been observed in several patients with CSD

[14, 22]. This evidence suggests that B. henselae and B. quintana

share common antigenic determinants, but that B. henselae and

not B. quintana is associated with CSD [14, 17, 18]. On the

basis of this evidence and the result of our serological IFA

testing, we presume that the patients illness was due to B.

henselae infection.

The differential diagnosis of typical CSD includes many types

of lymphadenopathy: typical or atypical mycobacterial infec-

tion, yersiniosis, tularemia, plague, brucellosis, leptospirosis,

syphilis, lymphogranuloma venereum, sporotrichosis, histo-

plasmosis, toxoplasmosis, infectious mononucleosis, cytomeg-

alovirus infection, and neoplasms [6, 15, 16, 20]. The diagnosis

of CSD can easily be overlooked in cases with atypical syn-

dromes, such as the one we describe in the current report.

A recognition of the association between CSD and findings

of enlarged posterior pancreatic duodenal lymph nodes with

abdominal pain and fever has important clinical implications.

We suggest that CSD should now be included in the differential

diagnosis of isolated intra-abdominal lymphadenopathy, even

when peripheral adenopathy and an inoculation papule are

absent, as in our patient. Clinical presentation, epidemiological

data, the abdominal ultrasonographic and/or CT appearance

of the lymph nodes, and serological IFA for B. henselae antigen

can all help establish the diagnosis of CSD.

References

1. Parinaud H. Conjunctivitis infectious par les animaux. Ann Ocul1889; 101:2523.

2. Ulrich GG, Waecker NJ, Meister SJ, Peterson TJ, Hooper DG. Catscratch disease associated with neuroretinitis in a 6-year-old girl. Oph-thalmology 1992; 99:2469.

3. Revol A, Vighetto A, Jouver A, Aimard G, Trillet M. Encephalitis incat scratch disease with persistent dementia. J Neurol Neurosurg Psy-chiatry 1992; 55:1335.

4. Lenoir AA, Deschryver-Kecskemeti K, Shackelford GD, et al. Granu-lomatous hepatitis associated with cat scratch disease. Lancet 1988; 1:1326.

5. Delaboussaye PM, Osborne BM. Cat-scratch disease presenting as ab-dominal visceral granulomas. J Infect Dis 1990; 161:718.

6. Dunn MW, Berkowitz FE, Miller JJ, Snitzer JA. Hepatosplenic cat-scratch disease and abdominal pain. Pediatr Infect Dis J 1997; 16:26972.

7. Muszynski MJ, Eppes S, Riley HD. Granulomatous osteolytic lesion ofthe skull associated with cat-scratch disease. Pediatr Infect Dis J 1987;6:199201.

8. Chesney PJ, Abbasi S. Pulmonary manifestations of cat-scratch disease:a case report and review of the literature. Pediatr Infect Dis J 1995;14:5478.

9. Raoult D, Fournier PE, Drancourt M, et al. Diagnosis of 22 new casesof Bartonella endocarditis. Ann Intern Med 1996; 125:64652.

10. Jacobs RF, Schultze GE. Bartonella henselae as a cause of prolongedfever of unknown origin in children. Clin Infect Dis 1998; 26:804.

11. Alkan S, Morgan MB, Sandin RL, Moscinski LC, Ross CW. Dual rolefor Afipia felis and Rochalimaea henselae in cat-scratch disease. Lancet1995; 345:385.

12. Brenner DJ, Hollis DG, Moss CW, et al. Proposal of Afipia gen.nov.,with Afipia felis sp.nov. (formerly the cat scratch disease bacillus), Afipiaclevelandensis sp.nov. (formerly the Cleveland Clinic Foundationstrain), Afipia broomeae sp.nov., and three unnamed genospecies. J ClinMicrobiol 1991; 29:245060.

13. Kordick DL, Hilyard EJ, Hadfield TL, et al. Bartonella clarridgeiae, anewly recognized zoonotic pathogen causing inoculation papules, fever,and lymphadenopathy (cat scratch disease). J Clin Microbiol 1997; 35:18138.

14. Maurin M, Eb F, Etienne J, Raoult D. Serological cross-reactions be-tween Bartonella and Chlamydia species: implications for diagnosis. JClin Microbiol 1997; 35:22837.

15. La Scola B, Raoult D. Serological cross-reaction between Bartonellaquintana, Bartonella henselae, and Coxiella burnetii. J Clin Microbiol1996; 34:22704.

16. Dalton MJ, Robinson LE, Cooper J, Regenery RL, Olson JG, ChildsJE. Use of Bartonella antigens for serologic diagnosis of cat-scratchdisease at a national referral center. Arch Intern Med 1995; 155:16706.

17. Szelc-Kelly CM, Goral S, Perez-Perez GI, Perkins BA, Regenery RL,Edwards KM. Serologic responses to Bartonella and Afipia antigens inpatients with cat scratch disease. Pediatrics 1995; 96:113742.

18. Zangwill KM, Hamilton DH, Perkins BA, et al. Cat scratch disease inConnecticut. N Engl J Med 1993; 329:813.

19. Bass JW, Freitas BC, Freitas AD, et al. Prospective randomized doubleblind placebo-controlled evaluation of azithromycin for treatment ofcat-scratch disease. Pediatr Infect Dis J 1998; 17:44752.

20. Carithers HA. Cat-scratch disease. Am J Dis Child 1985; 139:112433.21. Holley PH. Successful treatment of cat-scratch disease with ciproflox-

acin. JAMA 1991; 265:15635.22. Anderson B, Sims K, Regenery R, et al. Detection of Rochalimaea

henselae DNA in specimens from cat scratch disease patients by PCR.J Clin Microbiol 1994; 32:9427.

by guest on August 4, 2014

http://cid.oxfordjournals.org/D

ownloaded from

http://cid.oxfordjournals.org/