RESUMEN
AIMS: Trigeminal neuropathy is more likely to suggest neuronal damage and occur due to secondary pathology than trigeminal neuralgia. Evaluation of underlying etiologies are necessary. CASE: A 29-year-old female patient presented with left sided continuous burning pain likened to pins and needles at maxillary distribution for about a year. Her examination was normal except left-sided buccal swelling without any skin or mucosal change. Cranial MRI revealed asymmetrical dilation of left Meckel's cave, bilateral cerebral developmental venous anomaly and left sided slow flow venous malformation from superior temporal fossa to masseter muscle. Cerebral angiography confirmed widespread venous return anomaly in both cerebral hemispheres and slow-flow venous malformation that does not fill in the early arterial phase in the left buccal space and superficial temporal fossa. Cerebrofacial venous metameric syndrome is diagnosed. Percutaneous sclerotherapy with alcohol is planned in three separate sessions, the first of the three planned sessions is performed yet and the patient stated that her neuropathic pain decreased by 40% afterwards. SIGNIFICANCE: Clinical manifestation of the cerebrofacial venous metameric syndrome depends on the localization of the lesions; therefore, venous anomalies in relation with the trigeminal branches can present with painful trigeminal neuropathy.