Abstract
BACKGROUND: Central hypoventilation syndrome due to unilateral cerebellomedullary infarction is rare and difficult to manage using conventional treatment. Diaphragm pacing can support ventilation in patients with hypoventilation syndrome, mainly because of the high risk of cervical injury. There have been few reports of diaphragm pacing for central hypoventilation syndrome due to unilateral cerebellomedullary infarction. OBSERVATIONS: A 77-year-old man presented with dizziness, truncal ataxia, and dysarthria. MRI showed cerebral infarction in the right lateral medulla oblongata and right cerebellar hemisphere. The patient presented with respiratory failure immediately after admission. The patient required mechanical ventilation and tracheostomy support. Spontaneous breathing was insufficient, particularly during sleep. After a 2-month interval, the ventilator-dependent patient underwent diaphragm pacing. He was weaned from the ventilator 13 days after diaphragm pacing. LESSONS: The authors reported successful treatment with diaphragm pacing for central hypoventilation syndrome due to unilateral cerebellomedullary infarction. Diaphragm pacing is a good treatment option for patients with central hypoventilation syndrome. https://thejns.org/doi/10.3171/CASE25172.