The development of a tracheo-arterial fistula is a rare but severe complication of tracheotomy. The reported patient presented with such a fistula involving an aberrant right subclavian artery, termed an arteria lusoria, an aortic arch abnormality which is usually asymptomatic.

A 30-year-old man was admitted to our institution for bleeding through his tracheotomy. He had been treated for advanced Duchenne muscular dystrophy, involving invasive assisted ventilation, since the age of 10 years. Surgical exploration and computed tomography scanning revealed a tracheo-arterial fistula involving an aberrant right subclavian artery, associated with severe scoliosis. Emergency, transient haemostasis was achieved by over-inflation of the tracheostomy tube cuff. Aneurysm ablation was successfully achieved as the result of an endovascular interventional radiology procedure.

Arteria lusoria is one of the most common aortic arch abnormalities. The occurrence of an aneurysm of this artery in a tracheotomised patient has not previously been described. In Duchenne muscular dystrophy patients, spinal deformities may result in thoracic compression, which may alter the anatomical relations of mediastinal vessels. Such deformities are slowly progressive. Thus, vigilance is required in the long term management of Duchenne muscular dystrophy patients with a tracheostomy.