Gastric mucormycosis in a COVID-19 patient with severe lung involvement: a case report
Fardad Ejtehadi1, Navid Omidifar2, Mohammad hadi Niakan3, Reza Nikandish4, Zohre Khodamoradi5, Akbar Rasekhi Kazerouni1
¹Department of Internal Medicine, Gastroenterohepatology Research Center,
School of Medicine, Shiraz University of Medical Sciences, Shiraz, Iran.
²Biotechnology research center, Department of Pathology, School of Medicine,
Shiraz University of Medical Sciences, Shiraz, Iran.
³Trauma Research Center, Shahid Rajaee (Emtiaz) Trauma Hospital, Shiraz University of Medical Sciences, Fars Province, Shiraz, Iran.
⁴Emergency Medicine department Shiraz University of Medical Sciences, Shiraz, Iran.
⁵Department of Internal Medicine, Shiraz University of Medical Sciences, Shiraz, Iran.
*Corresponding author
*Akbar Rasekhi Kazerouni, Department of Internal Medicine, Gastroenterohepatology Research Center, School of Medicine, Shiraz University of Medical Sciences, Shiraz, Fars, Iran.
DOI: 10.55920/JCRMHS.2023.06.001250
Abstract
Background: Mucormycosis (zygomycosis) is a rare opportunistic and life threatening infection that almost always affect immunocompromised patients. The Covid-19 associated mucormycosis is a new condition that has been described with high mortality rates. Gastrointestinal mucomycosis is a rare condition and stomach is the more common site in gastrointestinal mucomycosis.
Case presentation: Presented case is a 41 year old cigarette smoker and diabetic man, who presented with Covid_19 lung involvement. Consequently, he developed with gastrointestinal bleeding and gastric mucormycosis was detected for him.
Conclusions: Evaluating the clinical data and rare features of COVID-19 patients and studying the behavior of the disease is crucial for early detection and management of diseases.
Keywords: Mucormycosis, Covid-19, Gastrointestinal, Opportunistic infection.
Introduction
Mucormycosis (zygomycosis) is a rare opportunistic and life-threatening infection that almost always affects immunocompromised patients. This infection was caused by the zygomycetes class of fungi, Mucorales order, etc. Rhizopus, Mucor, Rhizomucor and Absidia species. These organisms are commonly found on the floor, in decaying organic matter such as vegetables, in animal feces and in the soil. The most frequent mode of transmission is inhalation of the spores followed by implantation of the fungi into the skin lesion, such as burns and ingestion of the spore, and the rarest mechanism is direct intravenous transmission. Common sites of mucormycosis are rhino-orbito-cerebral and lung (1-3).
Risk factors for mucormycosis include malignancies, transplantation, prolonged neutropenia, uncontrolled diabetes mellitus (DM), iron overload, prolonged corticosteroid therapy, intravenous drug abuse, prematurity, and malnutrition. The most common risk factor for mucormycosis is prolonged uncontrolled diabetes mellitus (3-7).
Gastrointestinal mucomycosis is a rare condition and accounts for only 7% of mucormycosis; the stomach is the most common site in gastrointestinal mucomycosis, followed by the colon and ileum. The prognosis of gastrointestinal mucormycosis is poor, and the mortality rate is approximately 85% (4).
Since the onset of the COVID-19 pandemic in December 2019, new aspects of the disease have been discovered. An increasing number of COVID-19-associated mucormycosis cases, a condition that has been described with high mortality rates, have been reported since 2020 following the COVID-19 pandemic in many countries, especially India; the vast majority of these cases were of the Rhino-orbito-cerebral type (1, 8, 9).
Although systemic corticosteroids lead to decreased mortality in patients with severe COVID-19, they may put patients at risk of opportunistic fungal infections such as mucormycosis (10, 11). In addition to immunosuppressive therapy in COVID-19 patients, multiple inflammatory pathways and inflammatory cytokines predispose COVID-19 patients to opportunistic fungal infections (12).
Treatment of mucormycosis includes the combination of surgical debridement and antifungal therapy(13).
We report a patient with locally advanced gastric mucormycosis during admission due to COVID-19 with severe lung involvement.
Figure 1: Circumferential black‒greenish ulcer in the fundus.
Figure 2: Involvement of the fundus by mucor mycosis with extension to posterior wall of the stomach, diaphragm and splenic hilum in surgical exploration.
Figure 3: Histopathology showed severe infection with wide septate hypha with right angular branching in histopathology in favor of mucormycosis.
Case presentation
A 41-year-old male cigarette smoker with uncontrolled DM, hypertension (HTN) and hyperlipidemia (HLP) was admitted to Namazee Hospital, a referral center in Shiraz, Iran, with dyspnea and malaise and a diagnosis of COVID-19 infection. His high-resolution computed tomography (HRCT) showed approximately 90% lung involvement. At the time of admission, he was intubated due to respiratory distress, and COVID-19-approved management according to the national guidelines at that time, including dexamethasone 8 mg intravenous daily, was started. One week later, he presented with melena and had a hemoglobin drop from 12.1 to 9.4 mg/dl in serial laboratory data checking; after initial resuscitation with intravenous saline, pantoprazole and packed red blood cell transfusion, esophagogastroduodenoscopy was performed, which showed a circumferential black‒greenish ulcer in the fundus (Figure 1). Biopsy was taken, and mucormycosis was confirmed in the biopsy report. An abdominal CT scan showed diffuse wall thickening of the stomach. Amphotericin B liposomal IV was started, and then he was shifted to the operating room. At surgical exploration, involvement of the fundus by mucormycosis with extension to the posterior wall of the stomach, diaphragm and splenic hilum was seen (Figure 2); total gastrectomy, splenectomy, Roux-en-Y esophagojejunostomy and jejunojejunostomy were performed. Histopathology showed severe infection of wide septate hyphae with right angular branching consistent with simultaneous mucormycosis of the stomach and diaphragm (Figure 3). Unfortunately, despite liposomal amphotericin, surgery and initial recovery in the ICU, our patient died two weeks later due to disseminated sepsis with bacterial superinfection with Acinetobacter baumannii.
Discussion and Conclusion
Mucormycosis is a rare and fatal opportunistic infection that occurs in the vast majority of cases in immunocompromised patients. The most common risk factor for mucormycosis is prolonged uncontrolled diabetes mellitus with or without DKA. Other predisposing factors, including solid organ or stem cell transplantation, hematologic malignancies, major trauma, burn, steroid use, iron overload, severe neutropenia, long-term antibiotic use, renal disease, AIDS, SLE, malnutrition, injection drug use and low birth weight infants, are also mentioned in the literature (7, 14, 15). Gastrointestinal mucormycosis accounts for only 7% of cases. The most common site of gastrointestinal involvement is the stomach, followed by the colon, ilium and esophagus. Haider A. Naqvi and colleagues presented gastric mucormycosis in a patient with DM and CKD; esophagogastroduodenoscopy revealed an ulcerative mass with exudate in the fundus (14). Kulkarni et al. reported a case of gastric mucormycosis in a 50-year-old diabetic man with chronic alcoholism; he presented with abdominal pain and distention, vomiting and fever. There was generalized guarding and rigidity. During the operation, there was a large 4x4 cm perforated ulcer in the gastric body that was covered with greenish-gray exudate (16). In rare cases, the disease has been reported in people without immune deficiencies or any risk factor. Prabudh Goel and colleagues reported gastrointestinal mucormycosis with ascending colon involvement and perforation in a 10-year-old boy without any risk factors. He presented with RLQ abdominal pain, abdominal distension and bilious vomiting (13). Additionally, Gurbir Sehmbey and colleagues reported a case of gastric mucormycosis in a 48-year-old healthy man without any significant past medical problems; he was admitted with transpelvic gunshot and underwent abdominal surgery. Approximately two weeks later, he developed coffee-ground output from the nasogastric tube, so esophagogastroduodenoscopy was performed, which revealed a grayish coating large ulcer with an irregular border at the body and fundus of the stomach (17).
After the COVID-19 pandemic, many cases of mucormycosis concurrently with COVID-19 or in the post-COVID-19 period were reported (1). The vast majority of them presented with rhino-orbito-cerebral involvement. Only a few cases of COVID-19-associated gastrointestinal mucormycosis have been reported, which were all in the form of ilium and colon involvement. Mayank Jain et al. reported a 57-year-old diabetic woman with simultaneous ileocolic trunk thrombosis causing acute mesenteric ischemia and ileocolic mucormycosis approximately three weeks after COVID-19 pneumonia (18). Additionally, Ravinder Pal Singh et al. presented a 48-year-old immunocompetent intensive care physician with sigmoid mucormycosis and perforation approximately three weeks after COVID-19 pneumonia (19).
Malakar reported an 82-year-old man who presented with upper gastrointestinal bleeding after complete recovery from COVID-19. He received high-dose oral prednisolone for 21 days 2 months before this event. His upper gastrointestinal endoscopy and biopsy were suggestive of gastric mucormycosis. This Indian case was successfully managed with antifungal therapy without any surgery (20).
Our patient had COVID-19-associated gastric mucormycosis that occurred during COVID-19 pneumonia management. Our patient's risk factors were diabetes melilotus and the use of corticosteroids and broad-spectrum antibiotics. The role of the SARS-CoV-2 virus directly in the development of mucormycosis is unclear and needs more studies in the future.
Acknowledgements: Not applicable.
Funding: Not applicable.
Ethics declarations: Informed consent was obtained from the patient to publish this case report.
Contributions: FE, RN, ZKh, ARK were contributors of writing the first draft of the manuscript. MhN completed surgical excision in this case. NO performed the histological examination of the lesion. All authors read and approve the final manuscript.
References
- Saadi MHG, Hosseini SA, Khodamoradi Z, Mokhtaryan M, Omidifar N, Moghadami M. Comparison of mucormycosis infection between patients with and without a history of COVID-19 infection: a retrospective cohort study. Transactions of The Royal Society of Tropical Medicine and Hygiene. 2022;117(3):174-8.
- Tahiri G, Lax C, Cánovas-Márquez JT, Carrillo-Marín P, Sanchis M, Navarro E, et al. Mucorales and mucormycosis: Recent insights and future prospects. Journal of Fungi. 2023;9(3):335.
- Muthu V, Agarwal R, Rudramurthy SM, Thangaraju D, Shevkani MR, Patel AK, et al. Multicenter case–control study of COVID-19–associated mucormycosis outbreak, India. Emerging Infectious Diseases. 2023;29(1):8.
- Roden MM, Zaoutis TE, Buchanan WL, Knudsen TA, Sarkisova TA, Schaufele RL, et al. Epidemiology and outcome of zygomycosis: a review of 929 reported cases. Clinical infectious diseases. 2005;41(5):634-53.
- Petrikkos G, Skiada A, Lortholary O, Roilides E, Walsh TJ, Kontoyiannis DP. Epidemiology and clinical manifestations of mucormycosis. Clinical Infectious Diseases. 2012;54(suppl_1):S23-S34.
- Rammaert B, Lanternier F, Zahar J-R, Dannaoui E, Bougnoux M-E, Lecuit M, et al. Healthcare-associated mucormycosis. Clinical Infectious Diseases. 2012;54(suppl_1):S44-S54.
- Özbek L, Topçu U, Manay M, Esen BH, Bektas SN, Aydın S, et al. COVID-19-associated mucormycosis: A systematic review and meta-analysis of 958 cases. Clinical Microbiology and Infection. 2023.
- Shahriarirad R, Khodamoradi Z, Erfani A, Hosseinpour H, Ranjbar K, Emami Y, et al. Epidemiological and clinical features of 2019 novel coronavirus diseases (COVID-19) in the South of Iran. BMC Infectious Diseases. 2020;20(1):427.
- Naqvi HA, Nadeem Yousaf M, Chaudhary FS, Mills L. Gastric Mucormycosis: An Infection of Fungal Invasion into the Gastric Mucosa in Immunocompromised Patients. Case Reports in Gastrointestinal Medicine. 2020;2020:8876125.
- Ahmadikia K, Hashemi SJ, Khodavaisy S, Getso MI, Alijani N, Badali H, et al. The double-edged sword of systemic corticosteroid therapy in viral pneumonia: A case report and comparative review of influenza-associated mucormycosis versus COVID-19 associated mucormycosis. Mycoses. 2021;n/a(n/a).
- Ranjbar K, Moghadami M, Mirahmadizadeh A, Fallahi MJ, Khaloo V, Shahriarirad R, et al. Methylprednisolone or dexamethasone, which one is superior corticosteroid in the treatment of hospitalized COVID-19 patients: a triple-blinded randomized controlled trial. BMC Infectious Diseases. 2021;21(1):337.
- Hoenigl M, Seidel D, Carvalho A, Rudramurthy SM, Arastehfar A, Gangneux JP, et al. The Emergence of COVID-19 Associated Mucormycosis: Analysis of Cases From 18 Countries. 2021.
- Goel P, Jain V, Sengar M, Mohta A, Das P, Bansal P. Gastrointestinal mucormycosis: a success story and appraisal of concepts. Journal of infection and public health. 2013;6(1):58-61.
- Naqvi HA, Yousaf MN, Chaudhary FS, Mills L. Gastric mucormycosis: An infection of fungal invasion into the gastric mucosa in immunocompromised patients. Case reports in gastrointestinal medicine. 2020;2020.
- Abreu BFBBd, Duarte ML, Santos LRd, Sementilli A, Figueiras FN. A rare case of gastric mucormycosis in an immunocompetent patient. Revista da Sociedade Brasileira de Medicina Tropical. 2018;51:401-2.
- Kulkarni RV, Thakur SS. Invasive gastric mucormycosis—a case report. Indian journal of surgery. 2015;77:87-9.
- Sehmbey G, Malik R, Kosa D, Srinivasan I, Chuang KY, Bellapravalu S. Gastric Ulcer and Perforation due to Mucormycosis in an Immunocompetent Patient. ACG case reports journal. 2019;6(8):e00154.
- Jain M, Tyagi R, Tyagi R, Jain G. Post-COVID-19 Gastrointestinal Invasive Mucormycosis. Indian Journal of Surgery. 2022;84(3):545-7.
- Singh RP, Gupta N, Kaur T, Gupta A. Rare case of gastrointestinal mucormycosis with colonic perforation in an immunocompetent patient with COVID-19. BMJ Case Reports CP. 2021;14(7):e244096.
- Malakar S, Elhence A, Prasad P, Ghoshal UC, Pandey G, Mohindra S. Primary gastric mucormycosis presenting with post-Coronavirus disease-19 upper gastrointestinal bleed. Indian Journal of Gastroenterology. 2022:1-2.



