Subtelomeric study of 132 patients with mental retardation reveals 9 chromosomal anomalies and contributes to the delineation of submicroscopic deletions of 1pter, 2qter, 4pter, 5qter and 9qter

BMC Med Genet. 2005 May 17:6:21. doi: 10.1186/1471-2350-6-21.

Abstract

Background: Cryptic chromosome imbalances are increasingly acknowledged as a cause for mental retardation and learning disability. New phenotypes associated with specific rearrangements are also being recognized. Techniques for screening for subtelomeric rearrangements are commercially available, allowing the implementation in a diagnostic service laboratory. We report the diagnostic yield in a series of 132 subjects with mental retardation, and the associated clinical phenotypes.

Methods: We applied commercially available subtelomeric fluorescence in situ hybridization (FISH). All patients referred for subtelomeric screening in a 5-year period were reviewed and abnormal cases were further characterized clinically and if possible molecularly.

Results: We identified nine chromosomal rearrangements (two of which were in sisters) corresponding to a diagnostic yield of approx. 7%. All had dysmorphic features. Five had imbalances leading to recognizable phenotypes.

Conclusion: Subtelomeric screening is a useful adjunct to conventional cytogenetic analyses, and should be considered in mentally retarded subjects with dysmorphic features and unknown cause.

Publication types

  • Multicenter Study

MeSH terms

  • Abnormalities, Multiple / genetics
  • Adolescent
  • Adult
  • Bone Diseases, Developmental / genetics
  • Child
  • Child, Preschool
  • Chromosome Aberrations*
  • Chromosome Deletion*
  • Chromosome Painting / methods
  • Chromosomes, Human, Pair 2 / genetics
  • Chromosomes, Human, Pair 22 / genetics
  • Craniofacial Abnormalities / genetics
  • Female
  • Growth Disorders / genetics
  • Humans
  • In Situ Hybridization, Fluorescence / methods
  • Intellectual Disability / genetics*
  • Karyotyping
  • Male
  • Syndrome
  • Telomere / genetics*
  • Translocation, Genetic / genetics