Neonatal lingual and gastrointestinal mucormycosis in a case of low anorectal malformation-a rare presentation.

ABSTRACT

We report a 1.7 kg male infant with a low anorectal malformation treated at an outside facility and referred to us on postoperative day 11. At presentation, his upper abdomen was distended, and he had perianal mucoid discharge. The tongue had a blackish discoloration. An erect abdominal radiograph showed a few fluid-filled bowel loops in the upper abdomen with a gasless lower abdomen and pelvis, suggestive of upper small bowel obstruction. There were no specific radiological features of necrotizing enterocolitis. He underwent laparotomy and bowel resection for perforated jejunum. Histopathology of the tissue specimen was suggestive of mucormycosis. Postoperatively, he received intravenous amphotericin B (liposomal) and was started on liquid enteral nutrition after 2 weeks. However, the anterior two thirds of his tongue gradually sloughed off. He is awaiting reconstruction of the tongue. The purpose of this report is to emphasize that physicians should have a high index of suspicion for oral and gastrointestinal tract mucormycosis in neonates with metabolic disturbances who present with a discolored oral mucosa and an abdominal mass with intestinal obstruction. Early diagnosis and an aggressive approach of combined medical and surgical treatment may improve the outcome of patients with this potentially lethal invasive disease.