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

Vera Radici; Eolia Brissot; Suzanne Chartier; Juliette Guitard; Bettina Fabiani; Mara Memoli; Anne Banet; Laurence Heuberger; Simona Lapusan; Sarah Atallah; Ollivier Legrand; Alexis Genthon

doi:10.1007/s44228-022-00009-3

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

Clin Hematol Int. 2022 Jun 13;4(1-2):60-64. doi: 10.1007/s44228-022-00009-3. eCollection 2022 Jun.

Authors

Affiliations

¹ Service d'Hématologie Clinique et Thérapie Cellulaire, Hôpital Saint-Antoine, Sorbonne Université, AP-HP, Paris, France.
² Clinical Hematology and Bone Marrow Transplant Centre, S.Maria della Misericordia University Hospital, University of Udine, Udine, Italy.
³ Service d'Anatomie Pathologique, Sorbonne Université, AP-HP, Hôpital Saint-Antoine, 75012 Paris, France.
⁴ Sorbonne Université, Inserm, Centre de Recherche Saint-Antoine, CRSA, AP-HP, Hôpital Saint-Antoine, Service de Parasitologie-Mycologie, 75012 Paris, France.
⁵ Department of Medicine and Surgery, Hematology and Hematopoietic Stem Cell Transplant Center, University of Naples Federico II, Naples, Italy.
⁶ Service d'Hématologie, Centre Hospitalier de la Polynésie Française, Pirae, Tahiti France.
⁷ Department of ENT-Head and Neck Surgery, Tenon Hospital, APHP, Sorbonne University, 4 Rue de La Chine, 75020 Paris, France.

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.