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BACKGROUND Chondromyxoid fibroma (CMF) is a rare benign bone tumour of cartilaginous origin, which usually affects the metaphysis of the long bone. Involvement of the temporal bone is extremely rare. Patients with CMF in the temporal bone can present some neurological deficits due to involvement of surrounding neural structures. CASE SUMMARY We present the first case of histopathologically proven CMF originating in the temporal bone and involving the hypoglossal canal in a 40-year-old woman. Hypoglossal nerve paralysis was identified on the cranial nerve examination. The patient underwent surgical excision and was neurologically normal except for mild left facial palsy on 5-mo follow-up examination after surgery. In the current report, the major characteristics and computed tomography/magnetic resonance imaging features of the lesion are discussed. Furthermore, previous literature regarding this pathology is reviewed. CONCLUSION The current study presents the first case of temporal bone CMF involving the hypoglossal canal.