Raben N, Sherman J B
Arthritis and Rheumatism Branch, National Institute of Arthritis, Musculoskeletal, and Skin Diseases, National Institutes of Health, Bethesda, Maryland 20892, USA.
Hum Mutat. 1995;6(1):1-6. doi: 10.1002/humu.1380060102.
Mutations in the muscle phosphofructokinase gene (PFK-M) result in a metabolic myopathy characterized by exercise intolerance and compensated hemolysis. PFK deficiency, glycogenosis type VII (Tarui disease) is a rare, autosomal, recessively inherited disorder. Multiple mutations, including splicing defects, frameshifts, and missense mutations, have recently been identified in patients from six different ethnic backgrounds establishing genetic heterogeneity of the disease. There is no obvious correlation between the genotype and phenotypic expression of the disease. PFK-M deficiency appears to be prevalent among people of Ashkenazi Jewish descent. Molecular diagnosis is now feasible for Ashkenazi patients who share two common mutations in the gene; the more frequent is an exon 5 splicing defect, which accounts for approximately 68% of mutant alleles in this population.
肌肉磷酸果糖激酶基因(PFK-M)突变会导致一种以运动不耐受和代偿性溶血为特征的代谢性肌病。PFK缺乏症,即糖原贮积症VII型(Tarui病),是一种罕见的常染色体隐性遗传疾病。最近在来自六个不同种族背景的患者中发现了多种突变,包括剪接缺陷、移码突变和错义突变,这证实了该疾病的遗传异质性。该疾病的基因型与表型表达之间没有明显的相关性。PFK-M缺乏症在德系犹太人后裔中似乎较为普遍。对于该基因存在两种常见突变的德系犹太患者,现在可以进行分子诊断;更常见的是外显子5剪接缺陷,约占该人群突变等位基因的68%。