Anti-IgLON5 disease combines autoimmune and neurodegenerative features, often presenting with movement disorders, bulbar dysfunction, cognitive impairment, and sleep disturbances. Severe motor disability is frequent, but full recovery after prolonged paralysis is rarely reported.

We reviewed clinical, imaging, and laboratory data from a 61-year-old Brazilian woman with confirmed anti-IgLON5 antibodies, analyzing therapeutic interventions and functional outcomes over seven years.

Symptoms began in late 2019 with ataxia, dysphagia, cognitive decline, parkinsonism, and myoclonus, rapidly progressing to tetraparesis, gastrostomy feeding, and ventilatory dependence by mid-2020. MRI showed diffuse cerebral atrophy; CSF analysis revealed lymphocytic pleocytosis and elevated protein levels. Initial therapy with intravenous immunoglobulin and plasma exchange produced limited improvement. In July 2020, azathioprine 50 mg daily was introduced as maintenance immunotherapy. Concurrently, she underwent daily motor physiotherapy, respiratory therapy, and speech/swallowing rehabilitation. Over the next 18 months, she underwent progressive limb strength recovery, improved swallowing and speech, and regained independent gait after nearly two years of complete motor disability. 

This Brazilian case exposes that azathioprine combined with intensive multidisciplinary rehabilitation can achieve substantial motor and functional recovery even after prolonged tetraparesis in anti-IgLON5 encephalitis. Sustained immunotherapy and high-intensity rehabilitation should be considered even in chronic, severe cases.