Toscano A, Tsujino S, Vita G, Shanske S, Messina C, Dimauro S
Institute of Neurological and Neurosurgical Sciences, University of Messina, Italy.
Muscle Nerve. 1996 Sep;19(9):1134-7. doi: 10.1002/(SICI)1097-4598(199609)19:9<1134::AID-MUS8>3.0.CO;2-0.
Human muscle phosphoglycerate mutase (PGAM-M) deficiency is associated with exercise intolerance, muscle cramps, chronic serum CK elevation, and recurrent episodes of myoglobinuria. Ten patients have been described: 7 African Americans, 1 African, and 2 Caucasians from the Italian kindred described here. Molecular genetic analysis has revealed three different mutations in the PGAM-M gene. The propositus of the Italian family was homozygous for a unique point mutation at codon 90 in exon 1, a C-to-T transition converting an encoded arginine to tryptophan. His sister, who had similar complaints, was also homozygous for this mutation while the paternal grandfather, both parents, a brother and a nephew of the propositus were heterozygous for the mutation. Our studies exclude that PGAM-M deficiency is limited to African Americans, and suggest that the molecular heterogeneity of this rare disorder may be due to a "founder effect" in different ethnic groups.
人类肌肉磷酸甘油酸变位酶(PGAM-M)缺乏症与运动不耐受、肌肉痉挛、慢性血清肌酸激酶升高以及反复发生的肌红蛋白尿发作有关。已描述了10名患者:7名非裔美国人、1名非洲人以及这里描述的来自意大利家族的2名白种人。分子遗传学分析揭示了PGAM-M基因中的三种不同突变。意大利家族的先证者在第1外显子的第90密码子处存在一个独特的点突变,即C到T的转换,该转换将编码的精氨酸转变为色氨酸,其为该突变的纯合子。他有类似症状的妹妹也是该突变的纯合子,而先证者的祖父、父母、一个兄弟和一个侄子则为该突变的杂合子。我们的研究排除了PGAM-M缺乏症仅限于非裔美国人的可能性,并表明这种罕见疾病的分子异质性可能是由于不同种族中的“奠基者效应”。