Molck Miriam C, Monteiro Fabíola P, Simioni Milena, Gil-da-Silva-Lopes Vera L
Department of Medical Genetics, University of Campinas (Unicamp), São Paulo, Brazil.
J Dev Behav Pediatr. 2015 Sep;36(7):544-8. doi: 10.1097/DBP.0000000000000197.
Copy number variation studies of known disorders have the potential to improve the characterization of clinical phenotypes and may help identifying candidate genes and their pathways. The authors described a child with congenital heart disease, microcephaly, facial dysmorphisms, developmental delay, learning difficulties, and behavioral problems. There was initially a clinical suspicion of 22q11.2 deletion syndrome (22q11.2 DS), but molecular cytogenetic analysis (array genomic hybridization [aGH]) showed the presence of a de novo 3.6-Mb interstitial microdeletion in 8p23.1. The main features of 8p23.1 DS include congenital heart disease and behavioral problems, in addition to minor dysmorphisms and mental delay. Therefore, this article highlights the application of aGH to investigate 8p23.1 deletion in nonconfirmed 22q11.2 DS patients presenting neurobehavioral disorders, congenital cardiopathy, and minor dysmorphisms.
对已知疾病的拷贝数变异研究有潜力改善临床表型的特征描述,并可能有助于识别候选基因及其通路。作者描述了一名患有先天性心脏病、小头畸形、面部畸形、发育迟缓、学习困难和行为问题的儿童。最初临床怀疑为22q11.2缺失综合征(22q11.2 DS),但分子细胞遗传学分析(阵列基因组杂交[aGH])显示在8p23.1存在一个新发的3.6 Mb间质性微缺失。除了轻微畸形和智力发育迟缓外,8p23.1 DS的主要特征包括先天性心脏病和行为问题。因此,本文强调了aGH在调查表现出神经行为障碍、先天性心脏病和轻微畸形的未确诊22q11.2 DS患者中8p23.1缺失的应用。