Bardosi A, Eber S W, Roessmann U
Clin Neuropathol. 1985 Mar-Apr;4(2):72-6.
Ultrastructural and enzyme histochemical muscle abnormalities are described in a case with a new variant (type Homburg) of glucosephosphate isomerase (GPI) deficiency, associated with congenital nonspherocytic hemolytic anemia and muscle weakness. The enzyme is thermostable in contrast to other described variants. The muscle fibers showed decreased GPI activity, ultrastructural abnormalities, including giant mitochondria, and a diffuse increase of glycogen. The functional alteration of muscle tissue is due to a stable enzyme protein with decreased specific activity.
本文描述了一例葡萄糖磷酸异构酶(GPI)缺乏症新变体(洪堡型)患者的超微结构和酶组织化学肌肉异常情况,该患者伴有先天性非球形细胞溶血性贫血和肌肉无力。与其他已描述的变体相比,该酶具有热稳定性。肌肉纤维显示GPI活性降低、超微结构异常,包括巨大线粒体,以及糖原弥漫性增加。肌肉组织的功能改变是由于一种具有降低的比活性的稳定酶蛋白所致。