Bruno C, Manfredi G, Andreu A L, Shanske S, Krishna S, Ilse W K, DiMauro S
H. Houston Merritt Clinical Research Center for Muscular Dystrophy and Related Diseases, Department of Neurology, Columbia College of Physicians and Surgeons, New York, New York 10032, USA.
Biochem Biophys Res Commun. 1998 Aug 28;249(3):648-51. doi: 10.1006/bbrc.1998.9211.
In a 28-year-old man with myopathy and phosphorylase kinase (PhK) deficiency, we found a G-to-C substitution at the 5' end of an intron in the muscle-specific alpha-subunit gene. The mutation destroys the high-consensus GT sequences at the 5' splice junction of the intron, which causes skipping of the preceding exon. This is the second molecular genetic defect identified in the myopathic variant of PhK deficiency.
在一名患有肌病和磷酸化酶激酶(PhK)缺乏症的28岁男性中,我们在肌肉特异性α亚基基因的一个内含子5'端发现了一个G到C的替换。该突变破坏了内含子5'剪接位点处高度保守的GT序列,导致前一个外显子跳跃。这是在PhK缺乏症的肌病变体中鉴定出的第二个分子遗传缺陷。