Serviço de Genética Médica, Hospital de Clínicas de Porto Alegre, RS, Brazil.
Am J Med Genet A. 2010 Oct;152A(10):2604-8. doi: 10.1002/ajmg.a.33612.
We report on a patient presenting with cognitive delay, prenatal and postnatal growth deficiency, microcephaly, ptosis of eyelids, high and broad nasal root, and camptodactyly. Analysis of a dense whole genome single-nucleotide polymorphism (SNP) array showed a de novo 3.35 Mb deletion on 2p15-p16.1. In order to study the parental origin of the deletion we analyzed selected SNPs in the deleted area in the proband and her parents showing Mendelian incompatibilities suggesting a de novo deletion on the chromosome of paternal origin. Based on the five cases described previously in the literature, we have narrowed the critical region responsible for the 2p15-p16.1 microdeletion syndrome phenotype. The critical region does not include the VRK2 gene that had been speculated to have a role in cortical dysplasia. However, the association of the VRK2 gene with cortical dysplasia remains to be determined, as MRI imaging of the brain and gene content of the 2p15-16 deletion becomes established in more patients.
我们报告了一例表现为认知延迟、产前和产后生长发育不良、小头畸形、眼睑下垂、高而宽的鼻根和掌挛缩的患者。对密集的全基因组单核苷酸多态性(SNP)阵列的分析显示,2p15-p16.1 上存在一个新发的 3.35Mb 缺失。为了研究缺失的亲本来源,我们在先证者及其父母的缺失区域中分析了选定的 SNP,结果显示孟德尔不相容性提示父源染色体上存在新发缺失。基于之前文献中描述的五个病例,我们已经缩小了导致 2p15-p16.1 微缺失综合征表型的关键区域。该关键区域不包括 VRK2 基因,该基因曾被推测在皮质发育不良中起作用。然而,VRK2 基因与皮质发育不良的关联仍有待确定,因为更多患者的大脑 MRI 成像和 2p15-16 缺失的基因含量变得确定。