Case Presentation: A 25-year-old female with a history of hypertension and diabetes mellitus (DM) type 1 presented with altered mental status, abdominal pain, and tenderness. On presentation, she was afebrile, hemodynamically stable. Further workup revealed leukocytosis 27000/ml, acute kidney injury with creatinine 2.47 mg/dl, blood glucose 1000 + mg/dl; glycosylated hemoglobin A1c (HbA1c 11.5%); amylase 195 U/L. She was started on treatment for DKA and empiric antibiotics for gastroenteritis with ceftriaxone and metronidazole, however, her symptoms continued to worsen. Computerized tomography (CT) abdomen showed inflammatory changes and pneumatosis of the cecum and distal small bowel. An exploratory laparotomy (ex-lap) was done showing ischemia of small bowel and right colon which was resected. Subsequently, she developed septic shock with Pseudomonas bacteremia, got intubated for airway protection and AKI worsened. She was started on broad-spectrum antibiotics with piperacillin-tazobactam and levofloxacin. However, her symptoms did not resolve, and a revisit ex-lap was performed. The pathology specimen showed broad invasive fungal infection for which she was started on empiric amphotericin-B. GMS and PAS staining were compatible with mucormycosis showing ribbon-like aseptate hyphae with wide angled branching. Cultures tested positive for mucormycosis. A repeat ex-lap for further resection was done to remove the necrotic bowel segment. The AKI worsened while being on amphotericin, and as the sensitivities came back she was switched to isavuconazole for a total 6 weeks of antibiotic treatment. Later, she got extubated, the AKI resolved and was transferred to a long-term health care facility for rehabilitation.
Discussion: Mucormycosis is an opportunistic angioinvasive fungal infection with the nasal cavity as the most commonly affected site. Gastrointestinal mucormycosis (GIM) though rare is associated with high mortality as high as 87%. It usually manifests in immunocompromised hosts with hematological malignancies, transplant recipients or with use of immunosuppressive therapies. Their pathogenesis in our patient might have involved the transient immunosuppression resulting from the DKA allowing the opportunistic fungus to invade the blood vessels. Usually infected sites involve stomach small intestine and colon. The symptoms may be subtle in case of immunosuppressed individuals or involve gastrointestinal bleeding, perforation or unexplained sepsis. GIM is diagnosed on the pathological analysis of the specimen after surgical excision. Aggressive surgical excision of the necrotic bowel segment is essential for source control followed by systemic antifungal therapy. Amphotericin B is the mainstay of treatment with new azoles showing the promising result in some cases. Isavuconazole is the only azole approved for the treatment of invasive mucormycosis.
Conclusions: Gastrointestinal mucormycosis (GIM) is a rare disease entity mainly found in immunocompromised patients or neonates. In a DKA patient presenting with signs or symptoms of gastroenteritis or acute abdomen, GIM should be in differentials,