Smith-Magenis syndrome: a new contiguous gene syndrome. Report of three new cases

J Med Genet. 1991 Sep;28(9):627-32. doi: 10.1136/jmg.28.9.627.

Abstract

Interstitial deletion of the short arm of chromosome 17 was detected in three patients. They all had a similar phenotype with mental retardation, behavioural problems, facial dysmorphism, brachycephaly, a broad face with a flat midface, and short and broad hands. All three cases were ascertained over a six month period by two neuropaediatricians aware of this specific anomaly, which suggests that this microdeletion is not particularly rare. Comparison of the clinical and cytogenetic findings in a total of 24 patients allows a new contiguous gene syndrome to be defined that only high resolution analysis can detect. In two cases, molecular analysis confirmed the cytogenetic results. The Charcot-Marie-Tooth type Ia gene has recently been localised to the 17p11.2 sub-band.

Publication types

  • Case Reports
  • Review

MeSH terms

  • Abnormalities, Multiple / genetics*
  • Cells, Cultured
  • Child Behavior Disorders / genetics*
  • Child, Preschool
  • Chromosome Aberrations / genetics*
  • Chromosome Aberrations / pathology
  • Chromosome Banding
  • Chromosome Deletion*
  • Chromosome Disorders
  • Chromosomes, Human, Pair 17 / ultrastructure*
  • Face / abnormalities*
  • Female
  • Hand Deformities, Congenital / genetics
  • Humans
  • Intellectual Disability / genetics*
  • Lymphocytes / ultrastructure
  • Male
  • Phenotype
  • Syndrome