Suppr超能文献

近端15号染色体长臂缺失与非典型普拉德-威利综合征表型

Deletions of proximal 15q and non-classical Prader-Willi syndrome phenotypes.

作者信息

Schwartz S, Max S R, Panny S R, Cohen M M

出版信息

Am J Med Genet. 1985 Feb;20(2):255-63. doi: 10.1002/ajmg.1320200208.

Abstract

A deletion of the long arm of chromosome 15 (usually involving bands 15q11-q12) has been seen in approximately 50% of Prader-Willi syndrome (PWS) patients [Ledbetter et al, 1982]. However, 14 patients with non-PWS (or atypical PWS) phenotype with 15q deletion indicate great clinical variability. A deletion was found in a propositus with a de novo translocation [45,XY, -15, -22, +rec(15;22) (22pter----22q13.2::15q14----15qter)], who had anomalies not normally observed in PWS patients. Activities of several enzymes mapped to the involved chromosomes were studied in the patient and control individuals. A 50% decrease in the level of arylsulfatase-A confirmed a small deletion in 22q(22q13.2----qter), and additional studies localized more precisely the loci for alpha-mannosidase (cytoplasmic) and beta-galactosidase.

摘要

在大约50%的普拉德-威利综合征(PWS)患者中发现了15号染色体长臂缺失(通常涉及15q11-q12带)[莱德贝特等人,1982年]。然而,14例具有15q缺失的非PWS(或非典型PWS)表型患者显示出很大的临床变异性。在一名患有新发易位[45,XY, -15, -22, +rec(15;22) (22pter----22q13.2::15q14----15qter)]的先证者中发现了缺失,该先证者具有PWS患者通常未观察到的异常。在患者和对照个体中研究了几种定位于受累染色体的酶的活性。芳基硫酸酯酶-A水平降低50%证实了22q(22q13.2----qter)存在小的缺失,进一步的研究更精确地定位了α-甘露糖苷酶(细胞质)和β-半乳糖苷酶的基因座。

文献检索

告别复杂PubMed语法,用中文像聊天一样搜索,搜遍4000万医学文献。AI智能推荐,让科研检索更轻松。

立即免费搜索

文件翻译

保留排版,准确专业,支持PDF/Word/PPT等文件格式,支持 12+语言互译。

免费翻译文档

深度研究

AI帮你快速写综述,25分钟生成高质量综述,智能提取关键信息,辅助科研写作。

立即免费体验