Abstract
RATIONALE: Peripheral odontogenic myxoma (POM) is a rare mesenchymal tumour and it is the first case report of POM involving orbital and zygoma region. PATIENT CONCERNS: A 16-year-old male presented with a painless, slow-growing swelling over his left infratemporal region. DIAGNOSIS: The histopathological examination of the tumour was diagnosed as POM. TREATMENT: The patient was treated by surgical removal of tumour under general anaesthesia. OUTCOMES: The patient has been under follow-up for the past 2.5 years and there has been no recurrence. TAKE-AWAY LESSONS: POM is a rare mesenchymal tumour. To our knowledge, this is only the second report of a POM of the infratemporal region and the first report of a myxoma, which extends into the zygomatic region and lateral wall of the orbit.