| Literature DB >> 28965848 |
Wolfram Demaerel1, Matthew S Hestand1, Elfi Vergaelen1, Ann Swillen1, Marcos López-Sánchez2, Luis A Pérez-Jurado2, Donna M McDonald-McGinn3, Elaine Zackai3, Beverly S Emanuel3, Bernice E Morrow4, Jeroen Breckpot1, Koenraad Devriendt1, Joris R Vermeesch5.
Abstract
Inversion polymorphisms between low-copy repeats (LCRs) might predispose chromosomes to meiotic non-allelic homologous recombination (NAHR) events and thus lead to genomic disorders. However, for the 22q11.2 deletion syndrome (22q11.2DS), the most common genomic disorder, no such inversions have been uncovered as of yet. Using fiber-FISH, we demonstrate that parents transmitting the de novo 3 Mb LCR22A-D 22q11.2 deletion, the reciprocal duplication, and the smaller 1.5 Mb LCR22A-B 22q11.2 deletion carry inversions of LCR22B-D or LCR22C-D. Hence, the inversions predispose chromosome 22q11.2 to meiotic rearrangements and increase the individual risk for transmitting rearrangements. Interestingly, the inversions are nested or flanking rather than coinciding with the deletion or duplication sizes. This finding raises the possibility that inversions are a prerequisite not only for 22q11.2 rearrangements but also for all NAHR-mediated genomic disorders.Entities:
Keywords: 22q11.2 deletion syndrome; 22q11.2DS; DiGeorge syndrome; Genomic disorder; VCFS; fiber-FISH; inversion polymorphism; low-copy repeats; microdeletion; segmental duplications
Mesh:
Year: 2017 PMID: 28965848 PMCID: PMC5630191 DOI: 10.1016/j.ajhg.2017.09.002
Source DB: PubMed Journal: Am J Hum Genet ISSN: 0002-9297 Impact factor: 11.025