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用于杜兴氏肌营养不良基因座的互补DNA探针显示,一名患有营养不良性肌病、甘油激酶缺乏症和先天性肾上腺发育不全的患者存在以前无法检测到的缺失。

Complementary DNA probes for the Duchenne muscular dystrophy locus demonstrate a previously undetectable deletion in a patient with dystrophic myopathy, glycerol kinase deficiency, and congenital adrenal hypoplasia.

作者信息

McCabe E R, Towbin J, Chamberlain J, Baumbach L, Witkowski J, van Ommen G J, Koenig M, Kunkel L M, Seltzer W K

机构信息

Institute for Molecular Genetics, Baylor College of Medicine, Houston, Texas 77030.

出版信息

J Clin Invest. 1989 Jan;83(1):95-9. doi: 10.1172/JCI113890.

Abstract

Genomic DNA from a patient with dystrophic myopathy, glycerol kinase deficiency, and congenital adrenal hypoplasia was investigated using cDNA probes for the Duchenne muscular dystrophy (DMD) locus. Genomic probes had not detected a deletion in this patient. Southern analysis of Hind III-digested genomic DNA from this patient identified a deletion when the three distal Hinc II DMD cDNA fragments were used as probes. The deletion began in the genomic region corresponding to the 1.05-kb Hinc II cDNA fragment and extended through the 3' end of the DMD gene. This represents a centromeric breakpoint that corresponds to a position approximately 10.2-10.6 kb from the 5' end of the 14-kb DMD cDNA. These investigations demonstrate the value of the DMD cDNA probes for improved diagnoses in patients with molecular lesions involving the DMD locus. Furthermore, this novel deletion involving the coding portion of the 3' end of the DMD gene assists in the ordering of exons in this region and will provide insight into the functional role of the carboxy terminus of the DMD gene product, dystrophin.

摘要

使用针对杜兴氏肌营养不良症(DMD)基因座的cDNA探针,对一名患有营养不良性肌病、甘油激酶缺乏症和先天性肾上腺发育不全的患者的基因组DNA进行了研究。基因组探针未检测到该患者存在缺失。当使用三个远端Hinc II DMD cDNA片段作为探针时,对该患者经Hind III消化的基因组DNA进行的Southern分析确定了一个缺失。该缺失始于对应于1.05 kb Hinc II cDNA片段的基因组区域,并延伸至DMD基因的3'端。这代表了一个着丝粒断点,其对应于距14 kb DMD cDNA 5'端约10.2 - 10.6 kb的位置。这些研究证明了DMD cDNA探针在改善涉及DMD基因座分子病变患者诊断方面的价值。此外,这种涉及DMD基因3'端编码部分的新型缺失有助于该区域外显子的排序,并将为深入了解DMD基因产物抗肌萎缩蛋白的羧基末端的功能作用提供线索。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/ae39/303648/4d987955771d/jcinvest00082-0108-a.jpg

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