Primary invasive gastric mucormycosis presenting as meleney's gangrene: A case report and review of literature

Mohamad Safwan; Shafy Ali Khan; Anni Belthazar; Madhu Sasidharan

doi:10.4103/ljms.ljms_45_21

CASE REPORT

Year : 2021 | Volume : 5 | Issue : 4 | Page : 171-173

Primary invasive gastric mucormycosis presenting as meleney's gangrene: A case report and review of literature

Mohamad Safwan¹, Shafy Ali Khan¹, Anni Belthazar², Madhu Sasidharan³
¹ Department of General Surgery, Kerala Institute of Medical Sciences, Thiruvananthapuram, Kerala, India
² Department of Laboratory Medicine, Kerala Institute of Medical Sciences, Thiruvananthapuram, Kerala, India
³ Department of Gastroenterology, Kerala Institute of Medical Sciences, Thiruvananthapuram, Kerala, India

Date of Submission	16-Jul-2021
Date of Acceptance	16-Dec-2021
Date of Web Publication	28-Feb-2022

Correspondence Address:
Dr. Mohamad Safwan
Department of General Surgery, Kerala Institute of Medical Sciences, Thiruvananthapuram, Kerala
India

Source of Support: None, Conflict of Interest: None

DOI: 10.4103/ljms.ljms_45_21

Abstract

Mucormycosis is an uncommon but potentially lethal fungal infection in immunocompromised individuals. The natural history of the disease is vascular invasion followed by thrombosis and necrosis of infected tissues. It can affect any organ system. Gastrointestinal (GI) mucormycosis is quite rare. Here, we report a case of primary invasive gastric mucormycosis in a 45-year-old male with diabetes and ethanol-related chronic liver disease who presented with necrotizing fascitis of the left thigh, groin, and lower abdominal wall. At presentation, he was hemodynamically unstable, anuric, and febrile with a high blood glucose level. He was stabilized with aggressive debridement and critical care management. During hospitalization, he developed sudden episode of upper GI bleed. Gastroscopy revealed extensive ulcerations with thick mucus in the fundus and body of the stomach. Biopsy from the lesions and special stain examination was consistent with invasive gastric mucormycosis. Delayed presentation of our patient and rapid progression to fungemia resulted in mortality.

Keywords: Black fungus, gastric mucormycosis, mucormycosis, upper gastrointestinal bleed

How to cite this article:
Safwan M, Khan SA, Belthazar A, Sasidharan M. Primary invasive gastric mucormycosis presenting as meleney's gangrene: A case report and review of literature. Libyan J Med Sci 2021;5:171-3

How to cite this URL:
Safwan M, Khan SA, Belthazar A, Sasidharan M. Primary invasive gastric mucormycosis presenting as meleney's gangrene: A case report and review of literature. Libyan J Med Sci [serial online] 2021 [cited 2023 Mar 28];5:171-3. Available from: https://www.ljmsonline.com/text.asp?2021/5/4/171/338629

Introduction

Mucormycosis is re-emerging invasive filamentous fungal pathogen which can affect pulmonary, rhinocerebral, skin and soft tissue, central nervous system, and gastrointestinal (GI) system. GI mucormycosis is the least common form and accounts for only 7% of all reported cases of mucormycosis with 85% mortality.^[1] Only 25% of GI mucormycosis has been diagnosed antemortem.

Therefore, the diagnosis of primary gastric mucormycosis can be missed due to the rarity of the disease. We report this case to contribute in part to the better understanding and awareness of this condition.

Case Report

A 45-year-old male who is known case of chronic liver disease (Child-Pugh C) and Diabetes mellitus presented to the emergency department with pain, swelling, and blackish discoloration of the skin over the left thigh following a trivial trauma 1 month ago. He had taken a herbal treatment for the same. Clinical examination revealed necrotizing fasciitis involving the left thigh extending to the groin, lower abdominal wall, left lumbar region, and scrotal edema with features of septic shock. He was resuscitated and taken for emergency exploration, wound debridement, and necrosectomy under anesthesia. Postoperatively, he was shifted to a multidisciplinary intensive care unit (MDICU) and managed with inotropes, broad-spectrum antibiotics, hemodialysis, glycemic control, wound care, and anti-hepatic encephalopathy measures. Tissue culture grew Klebsiella pneumonia and it was negative for fungus. On the postoperative day (POD) 5, the patient was extubated, and dialysis was stopped. He was recovering and was transported from the MDICU to the general ward on POD 7.

However on POD 11, he suddenly had two episodes of coffee ground vomitus and melena. The patient underwent emergency upper GI endoscopy, which revealed extensive ulcerations with thick adherent mucus in the fundus and body of the stomach [Figure 1]. Biopsies were obtained for histopathological examination and culture. On POD 13, his general condition deteriorated suddenly and progressed to septic shock. He was shifted back to MDICU and underwent aggressive critical care management, On POD 14, he succumbed to multi-organ failure. Gastric biopsy revealed gastric mucosa with ulceration and nonseptate fungal hyphae invading the gastric mucosa [Figure 2], [Figure 3], [Figure 4]. Special stains like periodic acid–Schiff [Figure 5] and Gomori's methenamine silver stains [Figure 6] highlighting pauciseptate fungal hyphae with right-angle branching which were consistent with gastric mucormycosis.

Figure 1: Gastroscopy showing extensive ulcerations with thick adherent mucus

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Figure 2: Showing ulcerated gastric mucosal fragment and pauciseptate fungal hyphae (Histopathology microscopy, ×20 power)

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Figure 3: Images of the pauciseptate fungal hyphae within tissue (Histopathology microscopy, ×40 power)

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Figure 4: Demonstrating angioinvasion (Histopathology microscopy, ×20 power)

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Figure 5: Highlighting pauciseptate fungal hyphae with right-angle branching (Periodic acid Schiff stain, ×40 power)

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Figure 6: Highlighting pauciseptate fungal hyphae with right angle branching (Gomori's methenamine silver stain, ×40 power)

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Discussion

GI mucormycosis is a rare life-threatening opportunistic infection due to the invasion of Mucorales into the gastric mucosa. It is caused by the ingestion of sporangiospores in foods such as fermented milk, fermented porridge, dried bread products, and alcoholic beverages.^[1],[2] The most common underlying risk factors are prolonged uncontrolled diabetes mellitus, malignancy, organ transplantation, immunosuppression, long-term steroid usage, penetrating trauma or burns, acquired immune deficiency syndrome, prolonged neutropenia, chronic alcoholism and cirrhosis, and use of deferoxamine-based therapy.^[2] An iatrogenic gastric mucormycosis associated with the use of wooden tongue depressors for mixing medications for critically ill patients on tube feeding and ingestion of herbal medicines have also been reported.^[3],[4]

The common site of GI mucormycosis is the stomach (67%), followed by the colon (21%), small intestine (4%), and esophagus (2%).^[5] Common presenting features are abdominal pain, fever, hematemesis, melena, and ulcer which can perforate and cause peritonitis and shock.^[5],[6] The phagocytic dysfunction, impaired chemotaxis, and defective intracellular destruction of Mucorales in the presence of an acidic environment of the stomach have been implicated in the pathogenesis of gastric mucormycosis. Uncontrolled diabetes and metabolic acidosis may produce a slightly acidic environment in the body and predispose patients to mucormycosis.^[7]

Isolation of organisms by endoscopic biopsy followed by histopathologic examination and fungal culture is the gold standard for diagnosing gastric mucormycosis.^[8] Characteristic gastroscopy findings are patchy ulcerated mucosal lesions with overlying greenish or grayish exudate. Histopathology demonstrates nonseptate, broad, obtuse-angled fungal hyphae with a ribbon-like appearance. Invasiveness and morphology were confirmed by special stains like Periodic acid–Schiff and Gomori's methenamine silver.^[8]

Newer molecular techniques like in situ hybridization and polymerase chain reaction from the biopsied specimen may help in prompt diagnosis and treatment.^[8] A multimodal approach like eliminating underlying risk factors, metabolic support, antifungal therapy with Intravenous amphotericin B or intravenous posaconazole, and surgical resection of necrosed tissue remains mandatory for preventing complications such as gastric perforation, peritonitis, and massive bleeding.^[8]

Conclusion

Invasive gastric mucormycosis is often rapidly progressive disease with high mortality in untreated patients. Most cases are still diagnosed late or at postmortem. A strong clinical suspicion and awareness must arise while managing immunocompromised patients with acute onset of abdominal pain, hematemesis, or massive malena. Early gastroscopic biopsy for histopathology examination and fungal culture should be done in such cases. Timely antifungal initiation and necrosectomy are required for improving patient outcomes.

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form the patient(s) has/have given his/her/their consent for his/her/their images and other clinical information to be reported in the journal. The patients understand that their names and initials will not be published and due efforts will be made to conceal their identity, but anonymity cannot be guaranteed.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.

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