Unusual manifestation of gastric mucormycosis in a patient with rheumatoid arthritis

  • Marija Milić Perović University Medical Center Zvezdara, Department of Pathology, Belgrade, Serbia
  • Nenad Šolajić University of Novi Sad, Faculty of Medicine, Department of Pathology, Novi Sad, Serbia
  • Lidija Vučković Hardi General Hospital Subotica, Department of Pathology, Subotica, Serbia
  • Tamara Knežević Ivanovski University Medical Center Zvezdara, Department of Gastroenterology, Belgrade, Serbia
  • Jovan Juloski University Medical Center Zvezdara, Surgery Clinic “Nikola Spasić”, Belgrade, Serbia
Keywords: arthritis, rheumatoid, diagnosis, diagnosis, differential, digestive system, histological techniques, mucormycosis

Abstract


Introduction. Mucormycosis is a life-threatening opportunistic infection whose incidence has significantly risen during the last two decades. The gastrointestinal form is very rare, with the stomach as the most common site of infection, followed by the colon and ileum. Risk factors include uncontrolled diabetes mellitus, corticosteroid use, and organ transplantation. We report a patient with a history of rheumatoid arthritis who has developed gastrointestinal mucormycosis. To the best of our knowledge, this is the first such case reported in the literature. Case report. A 53-year-old female patient with a prior medical history of rheumatoid arthritis was admitted to the hospital due to persisting diarrhea. Physical examination revealed diffuse abdominal tenderness to palpation without meteorism and peritoneal signs. Laboratory results demonstrated systemic inflammation, so antibiotic therapy was administered. Abdominal computed tomography findings revealed inflammation of the rectum and the left colon. Colonoscopy findings were indicative of Crohn’s disease. Additionally, the patient had developed profuse rectal bleeding and consequently underwent emergency surgery. Subtotal colectomy with ileostomy and partial gastrectomy was performed. The patient’s condition rapidly worsened after the operation, and she died due to multi-organ failure. Histologic findings of resection specimens discovered chronic active colitis and extensive gastric necrosis associated with dense mixed inflammatory infiltration and numerous non-septate and 90-degree branching hyphae. Diagnosis of invasive gastric mucormycosis was obtained, but unfortunately, several days after the patient’s death. Conclusion. It is very important to obtain high awareness among clinicians of this deadly infection to achieve a prompt diagnosis and effective therapy.

References

Kontoyiannis DP, Lionakis MS, Lewis RE, Chamilos G, Healy M, Perego C, et al. Zygomycosis in a tertiary care cancer center in the era of aspergillus active antifungal therapy: a case control observational study of 27 recent cases. J Infect Dis 2005; 191(8): 1350‒60.

Marr KA, Carter RA, Crippa F, Wald A, Corey L. Epidemiolo-gy and outcome of mould infections in hematopoietic stem cell transplant recipients. Clin Infect Dis 2002; 34(7): 909‒17.

Gleissner B, Schilling A, Anagnostapolous I, Siehl I, Thiel E. Im-proved outcome of zygomycosis in patients with hematologi-cal diseases? Leuk Lymphoma 2004; 45(7): 1351‒60.

Gomes MZR, Lewis RE, Kontoyiannis DP. Mucormycosis caused by unusual mucormycetes, non-Rhizopus, -Mucor, and -Lichtheimia species. Clin Microbiol Rev 2011; 24(2): 411‒45.

Kontoyiannis DP, Lewis RE. Invasive zygomycosis: update on pathogenesis, clinical manifestations, and management. Infect Dis Clin North Am 2006; 20(3): 581‒607, vi.

Martinello M, Nelson A, Bignold L, Shaw D. "We are what we eat!" Invasive intestinal mucormycosis: A case report and re-view of the literature. Med Mycol Case Rep 2012; 1(1): 52‒5.

Spellberg B, Edwards J Jr, Ibrahim A. Novel perspectives on mucormycosis: pathophysiology, presentation, and manage-ment. Clin Microbiol Rev 2005; 18(3): 556‒69.

Agha FP, Lee HH, Boland CR, Bradley SF. Mucormycoma of the colon: early diagnosis and successful management. Am J Roentgenol 1985; 145(4): 739‒41.

Ibrahim AS, Spellberg B, Walsh TJ, Kontoyiannis DP. Pathogene-sis of mucormycosis. Clin Infect Dis 2012; 54(Suppl 1): S16‒22.

Kennedy KJ, Daveson K, Slavin MA, Van Hal SJ, Sorell TC, Lee A. et al. Mucormycosis in Australia: contemporary epidemiol-ogy and outcomes. Clin Microbiol Infect 2016; 22(9): 775‒81.

Kulkarni RV, Thakur SS. Invasive gastric mucomycosis-a case report. Indian J Surg 2015; 77(Suppl 1): 87‒9.

Abidi ZM, Coelho-Prabhu N, Hargreaves J, Weiland T, Van Dyken I, Tande A, et al. Mucormycosis in patients with inflammatory bowel disease: case series and review of the literature. Case Rep Med 2014; 2014: 637492.

Shankaralingappa S. Unsuspected invasive gastrointestinal mu-cormycosis masquerading as inflammatory bowel disease: A pathologist's perspective. Indian J Pathol Microbiol 2019; 62(2): 332‒4.

Echo A, Hovsepian RV, Shen GK. Localized cecal zygomycosis following renal transplantation. Transpl Infect Dis 2005; 7(2): 68‒70.

Alghamdi A, Lutynski A, Minden M, Rotstein C. Successful treatment of gastrointestinal mucormycosis in an adult with acute leukemia: case report and literature review. Curr Oncol 2017; 24(1): 61‒4.

Forrester JD, Chandra V, Shelton AA, Weiser TG. Gastrointes-tinal mucormycosis requiring surgery in adults with hemato-logic malignant tumors: literature review. Surg Infect (Larchmt) 2015; 16(2): 194–202.

Debata PK, Panda SK, Dash A, Mohanty R, Mallick BN. An unu-sual presentation of colonic mucormycosis mimicking carci-noma colon – a surgeon’s perspective. Int J Surg Case Rep 2015; 10: 248‒51.

Lee SH, Son YG, Sohn SS, Ryu SW. Successful treatment of in-vasive gastric mucormycosis in a patient with alcoholic liver cirrhosis: A case report. Exp Ther Med 2014; 8(2): 401‒4.

Kgomo MK, Elnagar AA, Mashoshoe K, Thomas P, Van Hougen-houck-Tulleken WG. Gastric mucormycosis: A case report. World J Clinl Infect Dis 2018; 8(1): 1‒3.

Pruthvi BC, Anuradha Rao CK, Rupashree S, Deepak S, Vikram S, Jayaprakash B, et al. Gastric mucormycosis masquerading as malignancy in an immunocompetent host. Arab J Gastroen-terol 2010; 11(4): 227‒9.

Roden MM, Zaoutis TE, Buchanan WL, Knudsen TA, Sarkisova TA, Schaufele RL, et al. Epidemiology and outcome of zygo-mycosis: a review of 929 reported cases. Clin Infect Dis 2005; 41(5): 634‒53.

Verma GR, Lobo DR, Walker R, Bose SM, Gupta KL. Dissemi-nated mucormycosis in healthy adults. J Postgrad Med 1995; 41(2): 40‒2.

Cornely OA, Cuenca-Estrella M, Meis JF, Ullmann AJ. European Society of Clinical Microbiology and Infectious Diseases (ESCMID) Fungal Infection Study Group (EFISG) and Eu-ropean Confederation of Medical Mycology (ECMM) 2013 joint guidelines on diagnosis and management of rare and emerging fungal diseases. Clin Microbiol Infect 2014; 20 Suppl 3: 1‒4.

Skiada A, Pagano L, Groll A, Zimmerli S, Dupont B, Lagrou K, et al. Zygomycosis in Europe: analysis of 230 cases accrued by the registry of the European Confederation of Medical My-cology (ECMM) Working Group on Zygomycosis between 2005 and 2007. Clin Microbiol Infect 2011; 17(12): 1859–67.

Cornely OA, Bassetti M, Calandra T, Garbino J, Kullberg BJ, Lortholary O, et al. ESCMID* guideline for the diagnosis and management of Candida diseases 2012: non-neutropenic adult patients. Clin Microbiol Infect 2012; 18 Suppl 7: 19‒37.

Einav S, Raveh D, Lachish T, Baumstarck K, Martin C, Martin-Loeches I, et al. Candida prophylaxis and treatment in critically ill patients after abdominal surgery: a survey of practice. Surg Infect (Larchmt) 2019; 20(6): 510‒8

Published
2022/09/21
Section
Case report