Tintelnotia destructans invasive rhinosinusitis in an immunocompromised child: A case-report and review of the literature

Abstract

Tintelnotia destructans is a recently described fungus within the family Phaeosphaeriaceae, previously reported only in superficial human infections such as keratitis, onychomycosis, and non-invasive sinus fungus ball. We describe the first case of invasive rhinosinusal infection due to this pathogen in a child with severe aplastic anemia. Diagnosis was established by histopathology showing submucosal fungal invasion and broad-range fungal PCR targeting 18S rRNA identifying T. destructans. Combined endoscopic surgical debridement, antifungal therapy (liposomal amphotericin B and isavuconazole) and immune reconstitution after emergency haplo-identical hematopoietic stem cell transplant ultimately led to complete recovery without recurrence 18 months after treatment, despite continued therapeutic immunosuppression for GvHD. This case expands the known pathogenic spectrum of T. destructans, demonstrating its potential to cause invasive disease in severely immunocompromised hosts. Advances in molecular diagnostic tools may expand the spectrum of fungi causing invasive fungal infections by identification of novel pathogenic species.