Clinical and mutational characteristics of spinal muscular atrophy with respiratory distress type 1 in The Netherlands

Neuromuscul Disord. 2013 Jun;23(6):461-8. doi: 10.1016/j.nmd.2013.03.002. Epub 2013 Apr 6.

Abstract

Spinal muscular atrophy with respiratory distress type 1 is an autosomal recessive disorder with early respiratory difficulties, distal muscle weakness, and contractures leading to foot deformities as the most striking clinical symptoms. Mutations of the gene encoding the immunoglobulin heavy chain μ-binding protein 2, mapped on chromosome 11q13, are the cause of the disease. We present the clinical and mutational characteristics of ten patients in the Netherlands who showed considerable clinical variability; they carried six novel mutations, including a deletion of exon 2. However, there were no clear phenotype-genotype correlations.

MeSH terms

  • Child, Preschool
  • Chromosome Mapping
  • Female
  • Genetic Predisposition to Disease / genetics
  • Humans
  • Infant
  • Male
  • Muscle Weakness / genetics*
  • Muscular Atrophy, Spinal / diagnosis
  • Muscular Atrophy, Spinal / genetics*
  • Mutation / genetics*
  • Niederlande
  • Respiratory Distress Syndrome, Newborn / diagnosis
  • Respiratory Distress Syndrome, Newborn / genetics*
  • Spinal Muscular Atrophies of Childhood / diagnosis
  • Spinal Muscular Atrophies of Childhood / genetics*
  • Transcription Factors / genetics

Substances

  • Transcription Factors

Supplementary concepts

  • Spinal muscular atrophy with respiratory distress 1