Mosaic trisomy of a small r(1) with an abnormal phenotype

Am J Med Genet. 2001 Sep 15;103(1):32-5. doi: 10.1002/ajmg.1494.

Abstract

Cytogenetic studies of a mildly dysmorphic 10-year-old male with mild developmental delay and learning difficulties revealed mosaicism for a supernumerary ring chromosome in approximately half of the cells. The karyotype of this patient was established as 47,XY,+r[15]/46,XY[15].ish r(1)(D1Z7+,wcp1-). Although the presence of euchromatic material was shown by C banding, the lack of hybridization with the whole chromosome paint 1 (wcp1) probe suggests that few unique sequences are contained in the ring and that these sequences likely explain the child's dysmorphic features and developmental delay. A review of the literature, including the present case, suggests that the significance of euchromatin in supernumerary r(1) as determined by both C banding and fluorescence in situ hybridization (FISH) with chromosome 1 painting probes can be used as a prognostic indicator for potential severity of the clinical phenotype.

Publication types

  • Case Reports

MeSH terms

  • Child
  • Chromosome Banding
  • Chromosomes, Human, Pair 1 / genetics*
  • Humans
  • In Situ Hybridization, Fluorescence
  • Karyotyping
  • Male
  • Mosaicism
  • Phenotype
  • Ring Chromosomes*
  • Trisomy*