Liposomal amphotericin B in the treatment of pediatric gastrointestinal basidiobolomycosis.
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BACKGROUND: Gastrointestinal basidiobolomycosis is a rare fungal infection caused by Basidiobolus ranarum, increasingly recognized as an intestinal pathogen in children. Its clinical features closely mimic malignancy or inflammatory bowel disease, leading to frequent diagnostic delays. Although endemic in regions such as Saudi Arabia, Iran, and Oman, sporadic cases occur worldwide. Amphotericin B-liposomal remains a cornerstone therapy for invasive fungal disease, but no standardized treatment protocol for pediatric gastrointestinal basidiobolomycosis has been established. CASE PRESENTATION: We report a 6-year-old Asian girl, born preterm with very low birth weight, who presented with persistent abdominal pain without organomegaly, alternating diarrhea and constipation, fever, and anorexia. Initial ultrasonography revealed colonic wall thickening with aneurysmal dilation at the splenic flexure. Colon biopsy confirmed Basidiobolus ranarum. The patient received intravenous liposomal amphotericin B (90 mg daily), with partial radiologic improvement but persistent symptoms, prompting exploratory laparotomy. Adhesiolysis, left hemicolectomy, and resection of jejunal and colonic masses were performed. Histopathology demonstrated dense eosinophil-rich granulomatous inflammation with multinucleated giant cells and a solitary fungal hypha, without classic Splendore-Hoeppli phenomenon, likely due to prior antifungal therapy. Postoperatively, the patient was discharged on oral itraconazole for 28 days with adjunctive cotrimoxazole. At 1-year follow-up, she remained asymptomatic with no evidence of recurrence. CONCLUSION: This case highlights the diagnostic challenges of pediatric gastrointestinal basidiobolomycosis and demonstrates successful management with liposomal amphotericin B followed by itraconazole. It underscores the ongoing gap in establishing standardized treatment and supports further prospective studies to define optimal antifungal and surgical strategies for this rare entity.