921Mohit et al., Int J Med Res Health Sci. 2015;4(4):921-924

Available online at: www.ijmrhs.com DOI: 10.5958/2319-5886.2015.00188.5Case report Open Access

PRIMARY GASTRIC ACTINOMYCOSIS: A RARE CASE REPORTMohit Bhatia1, Archana Thakur2, Bibhabati Mishra3, Vinita Dogra4

INTRODUCTION

Actinomycosis is a chronic disease characterized byabscess formation, tissue fibrosis, draining sinuses andulcers. It is caused by the filamentous, gram-positiveanaerobic or microaerophilic bacterial species of thegenus Actinomyces.[1] Actinomycosis mainly presents inthree forms: cervicofacial (31-65%), abdominopelvic (20-36%) and thoracic (15-30%).[2,3] Abdominal actinomycosishas often been called “one of the greatest imitators inclinical practice”.[4] It often presents as an indolent chronicsuppurative process with atypical symptoms that aremisdiagnosed as neoplasms and other inflammatorydiseases like tuberculosis or Crohn’s disease.[5] There is apredilection for appendix and ileocecal region of the boweland thus, it can easily mimic colonic adenocarcinoma,intestinal tuberculosis, chronic appendicitis or regionalenteritis.[6] When outside the intestine, abdominalactinomycosis generally grows by local spread with rareincidences of haematogenous or lymphaticdissemination.[7] Primary gastric actinomycosis isextremely rare, with only 24 cases reported till date.[8-14]

The rarity of gastric involvement by Actinomyces spp. hasbeen attributed to the high luminal acidity of stomach, as aresult of which, the organisms are either killed or theirgrowth is inhibited.[11] We hereby present a case ofprimary gastric actinomycosis, which to the best of ourknowledge, is the first ever report from India.

CASE REPORT

A thirty five years old female patient was admitted in asuper specialty hospital with complaints of low gradeintermittent fever, abdominal pain and discharging sinuseson anterior abdominal wall of one week duration. The

patient revealed that one month prior to the appearance ofdischarging sinuses, she had abdominal swelling. Therewas also history suggestive of loss of appetite andsignificant weight loss. Although, there was no significantpast history suggestive of use of Intra UterineContraceptive Device (IUCD), however, this patient hadundergone cholecystectomy for cholelithiasis at a privatenursing home six months ago. On examination, the patientwas febrile and had pallor. No icterus, cyanosis, clubbing,lymphadenopathy or edema were observed. Systemicexamination revealed no abnormality except for thepresence of two sinuses discharging frank pus on anteriorabdominal wall. Sinus openings were raised, inflamed andflared up [Figures 1a & 1b].

Fig 1:(a & b). Discharging sinuses on anteriorabdominal wallThe following investigations were carried out, the results ofwhich are as follows: Complete haemogram revealedanaemia (hemoglobin: 5.5 gram%) and leucocytosis (totalleucocyte count: 20,400/cu.mm of blood) respectively.

ABSTRACT

Actinomycosis is a chronic disease characterized by abscess formation, tissuefibrosis, draining sinuses and ulcers caused by the filamentous, gram-positiveanaerobic or microaerophilic bacterial species of the genus Actinomyces.Actinomycosis mainly presents in three forms namely cervicofacial (31-65%),abdominopelvic (20-36%) and thoracic (15-30%) respectively. Primary gastricactinomycosis is extremely rare, with only 24 cases reported till date. Wepresent a case report of a thirty five years old female patient who was admittedin a super specialty hospital with complaints of low grade intermittent fever,abdominal pain and two discharging sinuses on anterior abdominal wall.Clinical, radiological, microbiological and pathological evaluation revealedfindings suggestive of primary gastric actinomycosis with underlying gastricadenocarcinoma in this patient. To the best of our knowledge, this is the firstever report of primary gastric actinomycosis from India.

ARTICLE INFOReceived: 26th July 2015Revised: 20th Aug 2015Accepted: 20th Sep 2015

Authors details: 1Senior Resident,2,4Director Professor, 3DirectorProfessor & Head, Department ofMicrobiology, Govind Ballabh PantInstitute of Post Graduate MedicalEducation and Research, New Delhi,IndiaCorresponding author: Mohit BhatiaDepartment of Microbiology, GovindBallabh Pant Institute of PostGraduate Medical Education andResearch, New Delhi, India.Email: docmb1984@gmail.com

Keywords: Actinomyces spp.,Primary gastric actinomycosis, Gastricadenocarcinoma

922Mohit et al., Int J Med Res Health Sci. 2015;4(4):921-924

Ultrasonography of abdomen revealed eccentricasymmetric wall thickening of gastric antro-pyloric regionof maximum thickness 3 cm with loss of muralstratification. Few enlarged perigastric lymph nodes of size12 mm×10 mm each were also seen. Contrast enhancedcomputerized tomography (CECT) of abdomen revealedthickening of gastric mucosa with formation of a massextending to anterior abdominal wall and present up totransverse colon. Upper gastro intestinal endoscopyrevealed the presence of a large area of elevated surfaceof size 4 cm×5cm near lesser curvature of stomach. Twoopenings each of size 1cm×1cm with smooth marginsprobably containing some necrotic material werevisualized in this area. The overlying mucosa of elevatedsurface appeared to be slightly nodular and abnormal.Gastric biopsy samples collected during this procedurewere subjected to histopathological examination whichrevealed the presence of neoplastic glands (present inlamina) lined by cells showing nuclear pleomorphism,stratification, high Nuclear: Cytoplasmic ratio andmoderate amount of eosinophilic cytoplasm suggestive ofmoderately differentiated gastric adenocarcinoma.Pus and necrotic material collected from sinuses wassubjected to Gram stain, Ziehl-Neelson stain and aerobicculture and sensitivity. On macroscopic examination, thepus was yellow to brown in color, thick, fowl smelling withpresence of numerous granules which were yellow towhite in color and approximately 0.5-1mm in size. Gramstain of pus sample revealed the presence of numerouspus cells, occasional Gram negative bacilli and numerousGram positive non-fragmented, non-branching, thinfilamentous bacteria [Figure 2].

Fig 2: Gram stain of pus sample showing Grampositive, non-branching, thin filamentous bacteria andGram negative bacilli (1000X)No acid fast bacilli were seen on Ziehl-Neelson stain using3% acid-alcohol as decolorizer. Taking all sterileprecautions, the granules present in pus sample of thispatient were transferred to several glass slides andcrushed with the help of cover slips and wooden handle ofinoculating wire. Potassium hydroxide mount examinationof crushed granules revealed the presence of numerousaseptate filamentous structures approximately 0.5 to 1µmin size [Figure 3]. Gram stain and modified Ziehl-Neelsonstain (using 1% sulfuric acid as decolorizer) of crushedgranules respectively revealed the presence of numerousGram positive and non-acid fast, non-fragmented, non-branching, thin filamentous bacteria without any chains ofspores suggestive of Actinomyces spp [Figures 4 & 5].

Fig 3: KOH mount of crushed granules showingaseptate, non-branching, thin filamentous structuressuggestive of Actinomyces spp. (400X)

Fig 4: Gram stain of crushed sulfur granules revealingGram positive non-branching, thin filamentousbacteria (1000X)

Fig 5: Modified Ziehl-Neelson stain showing non acidfast structures morphologically resemblingActinomyces spp. (1000X)

Escherichia coli sensitive to amikacin, imipenem,meropenem and ertapenem was isolated after twenty fourhours of aerobic incubation of pus sample. In lieu of theclinical picture, aforementioned macroscopic andmicroscopic findings, special requisition was sent from thelaboratory to the clinicians for sending some more pus andnecrotic debris, collected from sinuses of this patient, forperforming anaerobic culture for confirmation ofactinomycosis. However, by the time this requisition wasreceived by the clinicians, the patient had deteriorated

923Mohit et al., Int J Med Res Health Sci. 2015;4(4):921-924

clinically and finally expired due to cardiac arrest owing towhich a second sample could not be collected.Suggestive history, predisposing factors in the form ofsurgery and presence of gastric adenocarcinoma,endoscopic findings and presence of discharging sinuses,macroscopic and microscopic microbiological findingssuggest that it was a case of primary gastricactinomycosis.

DISCUSSION

Actinomyces spp. are often found as saprophytes in theoral cavity, gastrointestinal and female genital tract. Thedestruction of the muscular barrier by trauma in the formof surgeries, endoscopic manipulation, immunesuppression as in leukemia, lymphoma and othermalignancies, renal insufficiency, renal transplant,diabetes and chronic inflammatory diseases arerecognized as predisposing factors for penetrationby these organisms.[15] Upon penetration, theseorganisms in turn result in characteristic clinicalmanifestations in different parts of the body at varyingfrequencies. Primary gastric actinomycosis frequentlypresents as low-grade fever, epigastric pain, weight loss,upper GI bleeding and rarely symptoms of gastric outletobstruction.[3,12,14] The patient under study presented withsimilar clinical features suggestive of actinomycosis. Shehad undergone cholecystectomy six months ago and wasdiagnosed on histopathological examination as a case ofgastric adenocarcinoma later on, both of which arepredisposing factors for acquiring abdominalactinomycosis.There are no specific radiological or endoscopic findingssuggestive of this condition. CT findings mostlydemonstrate an infiltrative lesion with diffuse gastric wallthickening suggestive of gastric adenocarcinoma orlymphoma.[2,14] Endoscopic findings of the disease maysimulate a gastric neoplasm and include submucosaltumor-like or infiltrative lesions and occasionally, mucosalulceration. In the present case, endoscopic findingsrevealed elevated surface and two openings containingnecrotic material near the lesser curvature of stomach.Because of the submucosal localization of theinflammatory process, endoscopic biopsy specimensusually reveal nonspecific inflammatory changes.[14] Out ofthe twenty four reported cases of primary gastricactinomycosis, only three cases have been diagnosedpre-operatively by histopathological examination ofendoscopic biopsy specimens.[9,10,14] In a case reported byKhaleel Al-Obaidy et al, the patient was subjected to uppergastrointestinal endoscopy twice, carried out one weekapart. Actinomycosis was detected by histopathologicalexamination only in the second biopsy which was obtainedfrom an area containing brownish fibrinopurulentinflammatory exudate.[14] In the present case also it ispossible that biopsy samples may not have been obtainedfrom the necrotic area and therefore, the diagnosis ofactinomycosis was missed. However, histopathologicalexamination revealed gastric adenocarcinoma as the mostprobable underlying cause of acquiring this infection incontrast to most reported cases of gastric actinomycosis,

in which it was impossible to trace the mechanism bywhich Actinomyces spp. had reached the gastric wall.[14]

Actinomycosis is often but not always characterized by thepresence of sulfur granules which occur in 50% of thecases. Although the presence of sulfur granules stronglysuggests a diagnosis, these are not pathognomonic for thedisease.[16,17] The differential diagnosis of sulphur granulesincludes nocardiosis, streptomycosis, chromomycosis,eumycetoma and botryomycosis.[18] In the case understudy, preliminary microbiological examination of pus andnecrotic material obtained from discharging sinusesrevealed the presence of organisms morphologicallyresembling Actinomyces spp and largely ruled out theaforementioned conditions.Culture is negative in >76% cases of gastricactinomycosis.[14] In our case, aerobic culture of pus andnecrotic material obtained from discharging sinusesyielded Escherichia coli. Generally typical actinomycoticlesions contain one to ten bacterial species in addition tothe pathogenic actinomycetes. These bacteria areresponsible for early symptoms of the disease andtreatment failures in addition to acting as probablesynergistic pathogens that strengthen the comparativelylow invasive power of Actinomyces spp.[19]

Whether Escherichia coli, which is known to causebotryomycosis[20], had contributed to the development ofmycetoma in our case remains unclear. Unfortunately,anaerobic culture for isolation and speciation ofActinomyces spp. could not be attempted due to earlydeath of the patient.

CONCLUSION

Primary gastric actinomycosis is an extremely raredisease and often missed by routine diagnostic tests if thesample is not specifically collected from the affected area.A high level of suspicion is required both bygastroenterologists and pathologists in order to correctlydiagnose this condition. Actinomycosis should beconsidered in the differential diagnosis of radiological andupper gastrointestinal endoscopic findings of gastric wallthickening, particularly in patients with history ofabdominal surgery, trauma or immune compromisedstatus. In order to avoid the possibility of missing thediagnosis, the pathologists should be more vigilant andemploy appropriate staining techniques when gastricendoscopic biopsy samples reveal the presence of subtlechanges such as inflammatory exudates.

ACKNOWLEDGEMENTS

Department of Gastroenterology, Govind Ballabh PantInstitute of Post Graduate Medical Education andResearch, New Delhi

Conflict of Interest: Nil

REFERENCES

1. Ferrari TC, Couto CA, Murta-Oliveira C, ConceicaoSA, Silva RG. Actinomycosis of the colon: A rarepresentation. Scand J Gastroenterol, 2000;35:108-9

924Mohit et al., Int J Med Res Health Sci. 2015;4(4):921-924

2. Isik B, Aydin E, Sogutlu G, Ara C, Yilmaz S,Kirimlioglu V. Abdominal actinomycosis simulatingmalignancy of the right colon. Dig DisSci. 2005;50:1312–14

3. Choi MM, Beak JH, Lee JN, Park S, Lee WS. Clinicalfeatures of abdominopelvic actinomycosis: report oftwenty cases and literature review. Yonsei MedJ. 2009;50:555–59

4. Dayan K, Neufeld D, Zissin R, Bernheim J, Paran H,Schwartz I et al. Actinomycosis of the large bowel:Unusual presentations and their surgical treatment.Eur J Surg,1996;162:657-60

5. P. Acquaro, F. Tagliabue, G. Confalonieri, P. Faccioli,and M. Costa. Abdominal wall actinomycosissimulating a malignant neoplasm: case report andreview of the literature. The World Journal ofGastrointestinal Surgery, 2010;2(7):247–50

6. Richards RJ, Grayer D. Actinomycosis: A rare causeof vesicocolic fistula. Am J Gastroenterol, 1989;84:677-9

7. Meyer P, Nwariaku O, McClelland RN, Gibbons D,Leach F, Sagalowsky AI, et al. Rare presentation ofactinomycosis as an abdominal mass: report of acase. Dis Colon Rectum, 2000;43:872-5

8. Oksüz M, Sandikçi S, Culhaci A, Egesel T, Tuncer I.Primary gastric actinomycosis: a case report. Turk JGastroenterol, 2007;18:44–46

9. Lee SH, Kim HJ, Kim HJ, Chung IK, Kim HS, ParkSH, et al. Primary gastric actinomycosis diagnosed byendoscopic biopsy: case report. GastrointestEndosc., 2004;59:586–89

10. Minamino H, Machida H, Tominaga K, Kameda N,Okazaki H, Tanigawa T, et al. A case report onprimary gastric actinomycosis. GastroenterolEndosc., 2011;53(2):262–69

11. Skoutelis A, Panagopoulos C, Kalfarentzos F,Bassaris H. Intramural gastric actinomycosis. SouthMed J.,1995;88:647–50

12. Lee CM, Ng SH, Wan YL, Tsai CH. Gastricactinomycosis. J Formos Med Assoc., 1996;95:66–68

13. Mazuji MK, Henry JS. Gastric actinomycosis: casereport. Arch Surg., 1967;94:292–93

14. Khaleel Al-Obaidy, Fatimah Alruwaii, Areej AlNemer, Raed Alsulaiman, Zainab Alruwaii, MohamedA Shawarby. Primary gastric actinomycosis: report ofa case diagnosed in a gastroscopic biopsy.BMC ClinPathol., 2015; 15: 2

15. D. C. Dominguez, S. J. Antony. Actinomyces andnocardia infections in immunocompromised and nonimmunocompromised patients. Journal of the NationalMedical Association, 1999; 91(1): 35–39

16. Yeguez J.F., Martinez S., Sands L.R., Hellinger M.D.Pelvic actinomycosis as malignant large bowelobstruction: A case report and a review of theliterature. Am Surg, 2000;66(1):85-90

17. Cintron J.R., Del Pino A., Duarte B., Wood D.Abdominal actinomycosis. Dis ColonRectum,1996;39(1):105-8

18. Omsby AH, Bauer TW, Hall GS. Actinomycosis of thecholecystic duct: case report and Review Pathology,1998;30:65-7

19. Schaal KP, Lee HJ. Actinomycete infections inhumans -- a review. Gene, 1992;115:201-11

20. B Devi, B Behera, ML Dash, MR Puhan, SS Pattnaik,S Patro. Botryomycosis. Indian J Dermatol., 2013Sep-Oct; 58(5): 406