Characterization of a 16 Mb interstitial chromosome 7q21 deletion by tiling path array CGH

Am J Med Genet A. 2007 Feb 15;143(4):333-7. doi: 10.1002/ajmg.a.31601.

Abstract

We report on a 42-year-old female patient with an interstitial 16 Mb deletion in 7q21.1-21.3 and a balanced reciprocal translocation between chromosomes 6 and 7 [karyotype 46,XX,t(6;7)(q23.3;q32.3)del(7)(q21.1q21.3)de novo]. We characterized the size and position of the deletion by tiling path array comparative genomic hybridization (CGH), and we mapped the translocation breakpoints on chromosomes 6 and 7 by FISH. The clinical features of this patient-severe mental retardation, short stature, microcephaly and deafness-are in accordance with previously reported patients with 7q21 deletions. Chromosome band 7q21.3 harbors a locus for split hand/split foot malformation (SHFM1), and part of this locus, including the SHFM1 candidate genes SHFM1, DLX5, and DLX6, is deleted. The absence of limb abnormalities in this patient suggests either a location of the SHFM1 causing factor distal to this deletion, or reduced penetrance of haploinsufficiency of a SHFM1 factor within the deleted interval.

Publication types

  • Case Reports
  • Research Support, Non-U.S. Gov't

MeSH terms

  • Adult
  • Chromosome Deletion*
  • Chromosomes, Human, Pair 6
  • Chromosomes, Human, Pair 7*
  • Deafness / genetics
  • Deafness / pathology
  • Dwarfism / genetics
  • Female
  • Homeodomain Proteins / genetics
  • Humans
  • In Situ Hybridization, Fluorescence
  • Intellectual Disability / genetics
  • Intellectual Disability / pathology
  • Microcephaly / genetics
  • Microcephaly / pathology
  • Models, Genetic
  • Oligonucleotide Array Sequence Analysis*
  • Proteasome Endopeptidase Complex / genetics
  • Transcription Factors / genetics
  • Translocation, Genetic

Substances

  • DLX5 protein, human
  • DLX6 protein, human
  • Homeodomain Proteins
  • SEM1 protein, human
  • Transcription Factors
  • Proteasome Endopeptidase Complex