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Mauro Manconi, MD; Susanna Mondini, MD; Andrea Fabiani, MD; Paolo Rossi, MD; Paolo Ambrosetto, MD; Fabio Cirignotta, MD

Arch Neurol. 2003;60:1787-1790.

Background  Anterior spinal artery (ASA) syndrome results in motor palsy and dissociated sensory loss below the level of the lesion, accompanied by bladder dysfunction. When the cervical spine is involved, breathing disorders may be observed.

Objective  To describe the polysomnographic findings in a patient with cervical ASA syndrome complicated by a sleep breathing disorder.

Setting  Unit of neurology at a sleep center.

Patient  A 30-year-old man had an ischemic lesion that affected the anterior cervical spinal cord (C2-C6) bilaterally because of an ASA thrombosis. He developed ASA syndrome associated with respiratory impairment during sleep.

Results  The polysomnographic study during sleep showed a severe sleep disruption caused by continuous central apneas that appeared immediately after falling asleep. Treatment by intermittent positive pressure ventilation normalized the respiratory pattern and sleep architecture.

Conclusions  The sleep breathing pattern was compatible with central alveolar hypoventilation due to automatic breathing control failure caused by a lesion of the reticulospinal pathway, which normally activates ventilatory muscles during sleep. This autonomic sleep breathing impairment resembles that found as a complication in patients who undergo spinothalamic tract cervical cordotomy for intractable pain. This surgical complication is known as the Ondine curse.

From the Units of Neurology (Drs Manconi, Mondini, and Cirignotta) and Respiratory Physiopathology (Dr Fabiani), St Orsola-Malpighi Hospital, and the Department of Neurological Sciences, University of Bologna, (Dr Ambrosetto), Bologna, and the Department of Anaesthesiology and Reanimation, St Benedetto Hospital, St Benedetto del Tronto (Dr Rossi), Italy.

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