Abstract
Mucormycosis isolated to the mandible is a rare presentation occurring generally after dental procedures. The case we report presented with discharging sinuses over facial region with radiological appearance of isolated osteomyelitis of the mandible. The patient used to apply an addictive dental powder over his teeth leading to caries. Following this, he pulled out all his teeth, which probably led to his condition. Invasive sampling revealed mucormycosis. An extensive search for an underlying immunodeficiency revealed that the patient had chronic granulomatous disease (CGD). Despite a prolonged course of L-Amphotericin B, the patient continued to have intermittent pus discharge and surgical debridement and curettage was eventually required. The patient had a chronic course with minimal soft tissue involvement which initially did not raise the suspicion of mucormycosis. The main learning point is that an unusual invasive fungal infection in an otherwise healthy host can be the first symptom of an underlying primary immunodeficiency, like CGD. Invasive fungal infections in patients with CGD often have an indolent course.