Literature DB >> 26436922

Mosaic deletion of 20pter due to rescue by somatic recombination.

Megan M Martin1, Rena J Vanzo1, Mallory R Sdano1, Adrianne L Baxter1, Sarah T South2,3.   

Abstract

We report on a unique case of a mosaic 20pter-p13 deletion due to a somatic repair event identified by allele differentiating single nucleotide polymorphism (SNP) probes on chromosomal microarray. Small terminal deletions of 20p have been reported in a few individuals and appear to result in a variable phenotype. This patient was a 24-month-old female who presented with failure to thrive and speech delay. Chromosomal microarray analysis (CMA) performed on peripheral blood showed a 1.6 Mb deletion involving the terminus of 20p (20pter-20p13). This deletion appeared mosaic by CMA and this suspicion was confirmed by fluorescence in situ hybridization (FISH) analysis. Additionally, the deletion interval at 20p was directly adjacent to 15 Mb of mosaic copy-neutral loss of heterozygosity (LOH). The pattern of SNP probes was highly suggestive of a somatic repair event that resulted in rescue of the deleted region using the non-deleted homologue as a template. Structural mosaicism is rare and most often believed to be due to a postzygotic mechanism. This case demonstrates the additional utility of allele patterns to help distinguish mechanisms and in this case identified the possibility of either a post-zygotic repair of a germline deletion or a post-zygotic deletion with somatic recombination repair in a single step.
© 2015 Wiley Periodicals, Inc.

Entities:  

Keywords:  20p terminal deletion; chromosomal mosaicism; del 20pter-p13; microarray analysis; single nucleotide polymorphism array; somatic repair

Mesh:

Year:  2015        PMID: 26436922     DOI: 10.1002/ajmg.a.37407

Source DB:  PubMed          Journal:  Am J Med Genet A        ISSN: 1552-4825            Impact factor:   2.802


  5 in total

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Journal:  Chromosome Res       Date:  2018-05-12       Impact factor: 5.239

2.  Clinical Performance of an Ultrahigh Resolution Chromosomal Microarray Optimized for Neurodevelopmental Disorders.

Authors:  Karen S Ho; Hope Twede; Rena Vanzo; Erin Harward; Charles H Hensel; Megan M Martin; Stephanie Page; Andreas Peiffer; Patricia Mowery-Rushton; Moises Serrano; E Robert Wassman
Journal:  Biomed Res Int       Date:  2016-11-16       Impact factor: 3.411

3.  Analytical and Clinical Validity Study of FirstStepDx PLUS: A Chromosomal Microarray Optimized for Patients with Neurodevelopmental Conditions.

Authors:  Charles Hensel; Rena Vanzo; Megan Martin; Sean Dixon; Christophe Lambert; Brynn Levy; Lesa Nelson; Andy Peiffer; Karen S Ho; Patricia Rushton; Moises Serrano; Sarah South; Kenneth Ward; Edward Wassman
Journal:  PLoS Curr       Date:  2017-02-27

4.  Chromosomal Microarray Analysis of Consecutive Individuals with Autism Spectrum Disorders Using an Ultra-High Resolution Chromosomal Microarray Optimized for Neurodevelopmental Disorders.

Authors:  Karen S Ho; E Robert Wassman; Adrianne L Baxter; Charles H Hensel; Megan M Martin; Aparna Prasad; Hope Twede; Rena J Vanzo; Merlin G Butler
Journal:  Int J Mol Sci       Date:  2016-12-09       Impact factor: 5.923

5.  Deletion of 20p13 and Duplication of 20p13p12.3 in a Patient with Delayed Speech and Development.

Authors:  Soon Sung Kwon; Jieun Kim; Saeam Shin; Seung Tae Lee; Kyung A Lee; Jong Rak Choi
Journal:  Ann Lab Med       Date:  2018-01       Impact factor: 3.464

  5 in total

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