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Neuromyelitis optica spectrum disorder (NMOSD) is an autoimmune demyelinating disorder of the CNS that frequently affects brainstem functions.1 Trigeminal neuralgia occurs in 2.5% of patients with NMOSD1; however, the occurrence of trigeminal autonomic cephalalgia (TAC) is rarely reported.2,3 Here, we describe a case of NMOSD with anti–aquaporin-4 (AQP4) antibodies who had a relapse with brainstem and cervical spinal cord lesions manifesting as short-lasting unilateral neuralgiform headache attacks with cranial autonomic symptoms (SUNA), a rare form of TAC.

Short-lasting unilateral neuralgiform headache attacks with cranial autonomic symptoms in NMOSD