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PMID: 16179221 Published · ppublish English Journal Article Validation Study

Application of a comprehensive subtelomere array in clinical diagnosis of mental retardation.

European journal of medical genetics ·Vol. 48 ·No. 3 ·2005-00-00 ·Pages 250-62

Kok K, Dijkhuizen T, Swart YE, Zorgdrager H, van der Vlies P, Fehrmann R, te Meerman GJ, Gerssen-Schoorl KB, van Essen T, Sikkema-Raddatz B, Buys CH

Abstract

In 2-8% of patients with mental retardation, small copy number changes in the subtelomeric region are thought to be the underlying cause. As detection of these genomic rearrangements is labour intensive using FISH, we constructed and validated a high-density BAC/PAC array covering the first 5 Mb of all subtelomeric regions and applied it in our routine screening of patients with idiopathic mental retardation for submicroscopic telomeric rearrangements. The present study shows the efficiency of this comprehensive subtelomere array in detecting terminal deletions and duplications but also small interstitial subtelomeric rearrangements, starting from small amounts of DNA. With our array, the size of the affected segments, at least those smaller than 5 Mb, can be determined simultaneously in the same experiment. In the first 100 patient samples analysed in our diagnostic practice by the use of this comprehensive telomere array, we found three patients with deletions in 3p, 10q and 15q, respectively, four patients with duplications in 9p, 12p, 21q and Xp, respectively, and one patient with a del 6q/dup 16q. The patients with del 3p and 10q and dup 12p had interstitial rearrangements that would have been missed with techniques using one probe per subtelomeric region chosen close to the telomere.

MeSH Terms
Adult Child Child, Preschool Chromosome Aberrations Chromosomes, Artificial, P1 Bacteriophage/genetics Chromosomes, Bacterial/genetics DNA/analysis Female Gene Dosage Humans Intellectual Disability/diagnosis Male Nucleic Acid Hybridization/methods Oligonucleotide Array Sequence Analysis/methods Telomere/genetics
Chemicals
DNA
Authors & Affiliations
11 authors, click to expand affiliations / ORCID
Kok Klaas
Department of Clinical Genetics, University Medical Centre Groningen, Antonius Deusinglaan 4, 9713 AW Groningen, The Netherlands. k.kok@medgen.umcg.nl
Dijkhuizen Trijnie
Swart Yolanthe E
Zorgdrager Hanny
van der Vlies Pieter
Fehrmann Rudolf
te Meerman Gerard J
Gerssen-Schoorl Klasien B J
van Essen Ton
Sikkema-Raddatz Birgit
Buys Charles H C M
Article Info
Journal
European journal of medical genetics
Abbr.
Eur J Med Genet
ISSN
1769-7212
Published
2005-00-00
Pages
250-62
Language
English
Region
Netherlands
NLM ID
101247089
Subset
IM
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