Ganguly Bani Bandana, Kadam Vijay, Kadam Nitin N
MGM Center for Genetic Research & Diagnosis, MGM's New Bombay Hospital, Navi Mumbai 400703, India.
Case Rep Genet. 2011;2011:396450. doi: 10.1155/2011/396450. Epub 2011 Sep 25.
Unbalanced chromosomal rearrangements are not common; however, they have a significant clinical expression. The parental balanced translocation produces unbalanced chromosome, which is transmitted to next generation through fertilization of gametes carrying the derivative chromosome. The carriers of balanced rearrangements mostly do not have recognizable phenotypic expression. We report a family comprising of healthy and non-consanguineous young parents and their preemie newborn severely affected with congenital anomalies and systemic disorders. Conventional Gbanding analysis of somatic chromosomes identified a balanced translocation, t(6;10)(p23;q24), in mother and an unbalanced rearrangement, der(6)t(6:10)(p23;q24)mat, in the child. The child has inherited a derivative chromosome 6 with partial deletion of 6(p23-pter) and partial trisomy 10(q24-qter), which has resulted in fusion of genes of two different chromosomes. The prominent phenotypic features of del(6p), including high forehead, flat nasal bridge, agenesis of left ear, atrial septal defect (ASD), craniosynostosis, and growth retardation, are overlapping with specific Axenfeld-Reiger-, Larsen-, and Ritscher-Sinzel/3-C syndromes, however, lacking in ocular anomalies, skeletal laxity, or cerebellar malformation. Therefore, this paper rules out the isolated effect of del(6p23) or trisomy 10(q24) on distinct previously reported syndromes and proposes the combined effect of unbalanced chromosomal alteration.
染色体不平衡重排并不常见;然而,它们具有显著的临床表型。亲代的平衡易位会产生不平衡染色体,该染色体通过携带衍生染色体的配子受精传递给下一代。平衡重排的携带者大多没有可识别的表型表达。我们报告了一个家庭,其父母年轻、健康且非近亲结婚,但其早产儿患有严重的先天性异常和全身性疾病。对体细胞染色体进行常规G显带分析发现,母亲存在平衡易位t(6;10)(p23;q24),而孩子存在不平衡重排der(6)t(6:10)(p23;q24)mat。孩子继承了一条衍生的6号染色体,该染色体部分缺失6(p23 - pter)且部分三体10(q24 - qter),这导致了两条不同染色体的基因融合。del(6p)的显著表型特征,包括高额、鼻梁扁平、左耳发育不全、房间隔缺损(ASD)、颅骨缝早闭和生长发育迟缓,与特定的阿克森费尔德 - 里格尔综合征、拉森综合征和里切尔 - 辛泽尔/3 - C综合征有重叠,但缺乏眼部异常、骨骼松弛或小脑畸形。因此,本文排除了del(6p23)或三体10(q24)对先前报道的不同综合征的单独影响,并提出了染色体不平衡改变的联合效应。