Fibrous dysplasia (FD) is a benign bone disorder presenting with a variety of clinical manifestations. This is the first reported case of anosmia as presenting symptom of FD. We present the case of a 72-year-old female patient with a progressive olfactory dysfunction. Clinical examination revealed evidence of chronic rhinosinusitis; therefore the patient was treated with a course of oral corticosteroids. The patient had no improvement in her olfactory ability and imaging studies were ordered. Bony lesions characteristic of craniofacial FD were found, causing obstruction of the central olfactory pathway. This case emphasises the need to conduct further investigations in patients with rhinosinusitis and olfactory dysfunction especially when they present no response to oral steroid treatment.