Searle Claire, Johnson Diana
Clinical Genetics, Nottingham University Hospital, United Kingdom.
Clinical Genetics, Sheffield Children's Hospital, United Kingdom.
Am J Med Genet A. 2016 Feb;170A(2):466-470. doi: 10.1002/ajmg.a.37442. Epub 2015 Nov 3.
Five cases of recognizable "Intra-uterine" dwarfism with cranio-facial dysostosis, disproportionately short arms, and other anomalies were written up by endocrinologist, Dr. Alex Russell in 1954. The syndrome he described went on to be named Russell-Silver syndrome. This report gives further information about the life and medical history of the most pictorially documented boy that Dr. Russell originally described in his seminal paper. The gentleman is now 69 years old and we believe him to be the oldest person known to date to have Russell-Silver syndrome. The article includes information relating to his development of type 2 diabetes mellitus, osteopenia, testosterone deficiency, and hypercholesterolemia. Methylation studies have now confirmed the diagnosis of Russell-Silver syndrome by demonstrating partial hypomethylation at the H19 Locus at IGF2P0 on chromosome 11.
1954年,内分泌学家亚历克斯·拉塞尔博士记录了5例可识别的“子宫内”侏儒症病例,这些病例伴有颅面骨发育不全、手臂不成比例地短小以及其他异常情况。他所描述的综合征后来被命名为罗素-西尔弗综合征。本报告提供了关于拉塞尔博士在其开创性论文中最初描述的、照片记录最为详尽的男孩的生活和病史的更多信息。这位先生现已69岁,我们认为他是迄今为止已知的患有罗素-西尔弗综合征的年龄最大的人。文章包括了与他患2型糖尿病、骨质减少、睾酮缺乏和高胆固醇血症的病情发展相关的信息。甲基化研究现已通过证明11号染色体上IGF2P0处H19位点的部分低甲基化,证实了罗素-西尔弗综合征的诊断。