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Disturbance of muscle fiber differentiation in congenital hypomyelinating neuropathy caused by a novel myelin protein zero mutation

  • Kinga Szigeti
  • , Gulam Mustafa Saifi
  • , Dawna Armstrong
  • , John W. Belmont
  • , Geoffrey Miller
  • , James R. Lupski
  • Texas Children's Hospital Houston
  • Baylor College of Medicine

Research output: Contribution to journalArticlepeer-review

31 Scopus citations

Abstract

We report a case of congenital hypomyelination associated with cranial nerve dysfunction, respiratory failure, and hypertrophic cardiomyopathy confounding the clinical picture. Molecular genetic testing showed a complex de novo myelin protein zero (MPZ) mutation consisting of a 3bp deletion of CTA from nucleotide 550 to 552 and insertion of G at nucleotide 550 that by conceptual translation results in a frameshift mutation. Muscle biopsy findings are presented that allude to the effect of abnormal innervation on early postnatal muscle differentiation.

Original languageEnglish
Pages (from-to)398-402
Number of pages5
JournalAnnals of Neurology
Volume54
Issue number3
DOIs
StatePublished - Sep 1 2003

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