1. Academic Validation
  2. Disturbance of muscle fiber differentiation in congenital hypomyelinating neuropathy caused by a novel myelin protein zero mutation

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

  • Ann Neurol. 2003 Sep;54(3):398-402. doi: 10.1002/ana.10681.
Kinga Szigeti 1 Gulam Mustafa Saifi Dawna Armstrong John W Belmont Geoffrey Miller James R Lupski
Affiliations

Affiliation

  • 1 Department of Molecular and Human Genetics, Baylor College of Medicine and Texas Children's Hospital, One Baylor Plaza, Houston, TX 77030, USA.
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.

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