A 32-year-old man with advanced HIV developed a severe fungal infection of his sinuses caused by a rare fungus called Saksenaea oblongispora, which typically doesn’t affect people with HIV. The patient presented with severe facial swelling that rapidly worsened, and despite doctors identifying the fungus through biopsies and lab tests, he did not receive antifungal treatment or surgery before his condition became critical. This is the first case of this particular fungal infection documented in sub-Saharan Africa and in HIV patients, highlighting how dangerous this rare infection can be and how challenging it is to diagnose quickly enough to save lives.