Background: Brainstem, hypothalamic and cerebral symptoms may occur in neuromyelitis optica spectrum disorders (NMOSD). However, pathologic yawning has not been previously described in NMOSD patients.

Methods: Nine AQP4-IgG seropositive NMOSD patients experienced excessive yawning not related to sleep deprivation or fatigue.

Results: Patients were female, aged 19-57 years (median, 39 years) at disease onset. Excessive yawning spells were the presenting symptom of the disease in five patients, lasted 2-16 weeks, and usually occurred in association with nausea, vomiting and hiccups. Brain MRI was abnormal in all patients and most frequently showed brainstem and hypothalamic lesions.

Conclusions: Pathologic yawning may be a neglected although not a rare symptom in NMOSD.