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平衡易位正常携带者父母的六个异常后代中的“平衡”核型

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

作者信息

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

机构信息

Department of Pediatrics, University of Pittsburgh, Pennsylvania, USA.

出版信息

Am J Med Genet. 1995 Jan 2;55(1):47-52. doi: 10.1002/ajmg.1320550114.

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.

摘要

在6800份连续的用于临床细胞遗传学诊断的血样中,我们鉴定出30个家系,其中先证者的一位亲本有一条常染色体平衡相互易位(不包括罗伯逊易位)。30个家系中有28个家系的先证者有畸形和/或智力发育迟缓:19个带有一条不平衡的衍生染色体,3个有涉及易位染色体以外其他染色体的异常,5个有“平衡”的相互易位,1个核型正常。我们推测,正如雅各布斯[1984年]最初所提出的,后6个核型“平衡”的受累先证者可能由于亚显微缺失和重复而异常。特别是在这6个家系中,83%的易位断点与脆性位点相关,比预期的偶然发生率更高(P<0.025)。这支持了赫克特和赫克特[1984年]关于脆性位点与先天性染色体断点之间存在关联的报告。为了解释这些发现,我们提出常染色体脆性位点是不稳定区域,易在脆性位点断点附近发生断裂和不等交换,从而产生微小的重复和缺失。因此,从有平衡相互易位的正常亲本那里继承看似“平衡”核型的新生儿,可能仍因亚显微染色体不平衡而有更高的畸形和/或发育延迟风险。

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