Mysterious unilateral midfacial destruction – a diagnostic dilemma

Abstract

We describe a case of progressive, significant, bony destruction of right midface over approximately 27 months from an unknown aetiology. A 58‐year‐old male patient originally presented with a white lesion in the upper right buccal sulcus, which a biopsy confirmed to be a benign hyperplastic inflammatory lesion. However, 16 months later, the patient returned with signs and symptoms suspicious of a malignant aetiology. A series of computed tomography scans over 27‐month period revealed progression of extensive permeative right midfacial bony destruction now with intracranial involvement. Contrary to the clinical and radiological findings, the histology results could not confirm malignancy and have been inconclusive on various occasions. We describe the clinical journey to date of this patient and discuss differential diagnoses: actinomycosis osteomyelitis; avascular necrosis of infective cause, such as mucormycosis; verrucous carcinoma; and Gorham's disease (massive osteolysis). We highlight the importance of a collaborative, multicentre, multidisciplinary approach to explore the results of investigations that conflict with the clinical picture of a complex case like this.