Literature DB >> 7702096

"Balanced" karyotypes in six abnormal offspring of balanced reciprocal translocation normal carrier parents.

S L Wenger1, M W Steele, L Y Boone, S G Lenkey, J H Cummins, X Q Chen.   

Abstract

Among 6800 consecutive blood samples studied for clinical cytogenetic diagnosis, we identified 30 families in which one parent of the proband had a balanced reciprocal autosomal translocation (excluding Robertsonian rearrangements). Twenty-eight of the 30 families had a malformed and/or mentally retarded proband: 19 with an unbalanced derived chromosome, 3 with abnormalities involving chromosomes other than those in the translocation, 5 with a "balanced" reciprocal translocation, and 1 with a normal karyotype. We hypothesize that the latter 6 affected probands with "balanced" karyotypes could be abnormal due to submicroscopic deletions and duplications as was originally suggested by Jacobs [1984]. Particularly in these 6 families, 83% of translocation breakpoints were associated with fragile sites, more than expected by chance (P < 0.025). This supports the report of an association between fragile sites and constitutional chromosome breakpoints by Hecht and Hecht [1984]. To explain these findings, we propose that autosomal fragile sites are unstable areas which predispose to breaks and unequal crossing over near the fragile site breakpoints creating minute duplications and deletions. Consequently, newborn infants inheriting a seemingly "balanced" karyotype from a normal parent with a balanced reciprocal translocation may still be at an increased risk of being malformed and/or developmentally delayed because of submicroscopic chromosomal imbalances.

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Year:  1995        PMID: 7702096     DOI: 10.1002/ajmg.1320550114

Source DB:  PubMed          Journal:  Am J Med Genet        ISSN: 0148-7299


  3 in total

1.  Paternally derived translocation t(8;18)(q22.1;q22)pat associated in a patient with developmental delay: Case report and review.

Authors:  Lakshmi Rao; Murthy Kanakavalli; Venkata Padmalatha; Pratibha Nallari; Lalji Singh
Journal:  J Pediatr Neurosci       Date:  2010-01

2.  Accurate Breakpoint Mapping in Apparently Balanced Translocation Families with Discordant Phenotypes Using Whole Genome Mate-Pair Sequencing.

Authors:  Constantia Aristidou; Costas Koufaris; Athina Theodosiou; Mads Bak; Mana M Mehrjouy; Farkhondeh Behjati; George Tanteles; Violetta Christophidou-Anastasiadou; Niels Tommerup; Carolina Sismani
Journal:  PLoS One       Date:  2017-01-10       Impact factor: 3.240

3.  Cryptic genomic imbalances in patients with de novo or familial apparently balanced translocations and abnormal phenotype.

Authors:  Carolina Sismani; Sofia Kitsiou-Tzeli; Marios Ioannides; Christodoulos Christodoulou; Violetta Anastasiadou; Goula Stylianidou; Eleftheria Papadopoulou; Emanuel Kanavakis; Zoe Kosmaidou-Aravidou; Philippos C Patsalis
Journal:  Mol Cytogenet       Date:  2008-07-21       Impact factor: 2.009

  3 in total

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