The simultaneous occurrence of an aneurysmal bone cyst (ABC) on a zygomatic arch with bilateral inferior turbinate gasification is extremely rare, and no previous studies are available. Here we report the case of a 34-year-old Chinese Han female who presented with hyperplasia of the left maxillary bone for one and a half years. The patient was observed to have an ABC on the left zygomatic arch concomitant with bilateral inferior turbinate gasification, as indicated by X-ray computed tomography, contrast-enhanced computed tomography and three-dimensional maxillofacial reconstruction. The patient underwent surgical resection of the cyst, and no postoperative symptoms were observed during the 4 years of follow-up. The etiology of this case is considered to be associated with a gene abnormality.