López-Expósito Isabel, Ballesta-Martinez María Juliana, Bafalliu Juan Antonio, Vera-Carbonell Ascensión, Domingo-Jiménez Rosario, López-González Vanesa, Fernández Asunción, Guillén-Navarro Encarna
Sección de Citogenética, Centro de Bioquímica y Genética Clínica, Hospital Clínico Universitario "Virgen de la Arrixaca", El Palmar, Murcia, Spain; Centro de Investigación Biomédica de Red de Enfermedades Raras (CIBERER), Instituto de Salud Carlos III (ISCIII), Madrid, Spain.
Unidad de Genética Médica, Servicio de Pediatría, Hospital Clínico Universitario "Virgen de la Arrixaca", El Palmar, Murcia, Spain; Centro de Investigación Biomédica de Red de Enfermedades Raras (CIBERER), Instituto de Salud Carlos III (ISCIII), Madrid, Spain.
Genomics. 2014 Apr;103(4):288-91. doi: 10.1016/j.ygeno.2014.02.008. Epub 2014 Mar 4.
Complex chromosome rearrangements (CCRs) are extremely rare in humans. About 20% of the apparently balanced CCRs have an abnormal phenotype and the degree of severity correlates with a higher number of breakpoints. Several studies using FISH and microarray technologies have shown that deletions in the breakpoints are common although duplications, insertions and inversions have also been detected. We report a patient with two simultaneous reciprocal translocations, t(3;4) and t(2;14;18), involving five chromosomes and six breakpoints. He showed dysmorphic features, preaxial polydactyly in the left hand, brachydactyly, postnatal growth retardation and developmental delay. The rearrangement was characterized by FISH analysis which detected an interstitial segment from chromosome 14 inserted in the derivative chromosome 2, and by whole genome array which revealed an interstitial deletion of approximately 4.5 Mb at the breakpoint site on chromosome 3. To our knowledge this microdeletion has not been previously reported and includes ~12 genes. The haploinsufficiency of one or several of these genes is likely to have contributed to the clinical phenotype of the patient.
复杂染色体重排(CCRs)在人类中极为罕见。约20%表面上平衡的CCRs具有异常表型,严重程度与更多的断点数量相关。多项使用荧光原位杂交(FISH)和微阵列技术的研究表明,断点处的缺失很常见,尽管也检测到了重复、插入和倒位。我们报告了一名患者,其同时存在两个相互易位,即t(3;4)和t(2;14;18),涉及五条染色体和六个断点。他表现出畸形特征、左手轴前多指、短指、出生后生长发育迟缓以及发育延迟。通过FISH分析对该重排进行了特征描述,检测到一条来自14号染色体的间质片段插入到衍生的2号染色体中,通过全基因组阵列揭示了3号染色体断点处约4.5 Mb的间质缺失。据我们所知该微缺失此前尚未见报道,包含约12个基因。这些基因中的一个或几个单倍剂量不足可能导致了该患者的临床表型。