Vascular compression is the most common cause of classic trigeminal neuralgia. However, other causes should be excluded. We describe a rare case of a patient presenting with typical trigeminal neuralgia associated with an arachnoid cyst in the petrous apex. A 22-year-old female patient with right trigeminal neuralgia for several years was treated with carbamazepine, which relieved the pain temporarily but was completely ineffective for the 3 months prior to presentation. Thin-slice computed tomography and magnetic resonance imaging showed a cephalocele in the petrous apex. Eventually, surgical aspiration of the cyst was performed using an endoscopic endonasal approach, which resulted in complete resolution of her pain. Petrous apex cephalocele should be considered in the differential diagnosis of trigeminal neuralgia, as it can be easily resolved by surgical aspiration.
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