Niyazov Dmitriy M, Nawaz Zafar, Justice April N, Toriello Helga V, Martin Christa Lese, Adam Margaret P
Department of Human Genetics, Emory University School of Medicine, Decatur, GA 30033-5207, USA.
Am J Med Genet A. 2007 Nov 15;143A(22):2700-5. doi: 10.1002/ajmg.a.32005.
Subtelomeric imbalances have been implicated in developmental delay and mental retardation (MR) and described for most chromosomes. This study reports the first detailed description of two individuals with de novo 12q subtelomere deletions and high-resolution mapping of their deletion size with oligonucleotide array CGH for genotype/phenotype comparisons. Patient 1 is an 8-year-old male with borderline mild MR, food-seeking behavior, obesity, no significant dysmorphic facial features, abnormal hair whorl pattern, brachydactyly and mild clinodactyly. Patient 2 is a 12-year-old male with mild MR, food-seeking behavior, obesity, short stature, mild dysmorphic facial features, multicystic kidney and unilateral cryptorchidism. Both patients share a deleted region of approximately 1.6 Mb, including 14 known genes, which perhaps contributed to their similar phenotypes. However, Patient 2 has more severe MR and organ system involvement, possibly due to the larger deletion size ( approximately 4.5 Mb) including an additional eight genes, although it is difficult to make phenotype/genotype correlations based on only two patients. Due to the relatively mild presentation of both of our patients, we propose that a proportion of individuals with subtelomeric imbalances may go undetected and therefore, recommend subtelomeric studies be carried out for cases of unexplained mild MR or isolated learning disability (LD) with behavioral problems in the absence of major dysmorphic features or birth defects. In addition, 12q subtelomeric deletions should be considered in the differential diagnosis of patients presenting with food-seeking behavior and resultant obesity, as well as those referred to rule out Prader-Willi syndrome.
亚端粒失衡与发育迟缓及智力低下(MR)有关,并且在大多数染色体中都有描述。本研究首次详细描述了两名患有新发12号染色体亚端粒缺失的个体,并使用寡核苷酸阵列比较基因组杂交技术对其缺失大小进行了高分辨率定位,以进行基因型/表型比较。患者1是一名8岁男性,有边缘性轻度智力低下、觅食行为、肥胖、无明显面部畸形特征、异常发旋模式、短指畸形和轻度小指内弯。患者2是一名12岁男性,有轻度智力低下、觅食行为、肥胖、身材矮小、轻度面部畸形特征、多囊肾和单侧隐睾。两名患者共享一个约1.6 Mb的缺失区域,包括14个已知基因,这可能导致了他们相似的表型。然而,患者2的智力低下和器官系统受累更严重,可能是由于缺失更大(约4.5 Mb),还包括另外8个基因,尽管仅根据两名患者很难进行表型/基因型相关性分析。由于我们两名患者的表现相对较轻,我们提出一部分亚端粒失衡的个体可能未被发现,因此,对于原因不明的轻度智力低下或伴有行为问题的孤立性学习障碍(LD)且无主要畸形特征或出生缺陷的病例,建议进行亚端粒研究。此外,在对有觅食行为及由此导致肥胖的患者,以及为排除普拉德-威利综合征而转诊的患者进行鉴别诊断时,应考虑1