Invasive Fungal Rhinosinusitis Due to Co-infection with Mucormycosis and Exserohilum rostratum in a Patient with Acute Lymphoblastic Leukemia

Abstract

Invasive fungal infections remain an important cause of complication and morbidity in the management of acute leukemias. Here we report the case of a 27-year-old patient from French Polynesia who was diagnosed with Philadelphia chromosome-negative B-cell acute lymphoblastic leukemia. After induction chemotherapy, she developed rhinosinusitis with extensive bone lysis. The context and clinical presentation quickly made us suspect an invasive mucormycosis infection. However, a multidisciplinary investigation including mass spectrometry techniques also revealed the presence of Exserohilum rostratum, a pathogen member of the genus Exserohilum that is ubiquitous in tropical and subtropical regions but rarely implicated in invasive sinusitis. Antifungal treatment combined with an early surgical approach resulted in a favorable clinical response.

Keywords: Acute lymphoblastic leukemia; Exserohilum rostratum; Invasive fungal infections; Mucormycosis; Setosphaeria.

Fig. 1

Initial palatine lesion (A, withe arrow) and CT scan before surgery showing palatine and septum lysis (B). Massive necrosis of the palate biopsy (C), hyphae identified by routine hematoxylin-eosin stain broad and ribbon-like hyphea in blood vessel stained with Grocott (C, insert)