Obstructive sleep apnoea and hypoventilation in an adult with congenital myasthenic syndrome

BMJ Case Rep. 2018 Nov 14:2018:bcr2018226534. doi: 10.1136/bcr-2018-226534.

Abstract

In this case report, we describe an adult male with congenital myasthenic syndrome due to mutations in muscle-specific receptor tyrosine kinase (MuSK, c.79+2 T>G; IVS1 +2 T>G, c.2368 G>A, Val790Met) presenting with sleep apnoea and hypercapnic respiratory failure. In the intensive care unit, he required intubation followed by tracheostomy which resolved obstructive sleep apnoea. Later, due to persistent sleep-associated hypoventilation, he required nocturnal mechanical ventilation. His case illustrates how respiratory muscle weakness due to mutations in MuSK can lead to various forms of sleep disordered breathing.

Keywords: mechanical ventilation; neuromuscular disease; respiratory system.

Publication types

  • Case Reports

MeSH terms

  • Humans
  • Hypoventilation / etiology*
  • Hypoventilation / therapy
  • Intubation, Intratracheal
  • Male
  • Middle Aged
  • Myasthenic Syndromes, Congenital / complications*
  • Respiration, Artificial
  • Sleep Apnea, Obstructive / etiology*
  • Sleep Apnea, Obstructive / therapy
  • Tracheostomy