Ullrich K, Gröbe H, Korinthenberg R, von Bassewitz D B
Pathol Res Pract. 1986 Oct;181(5):627-32. doi: 10.1016/S0344-0338(86)80164-9.
Glycogen storage disease type II Pompe (GSD II) is a lysosomal storage disease caused by an inherited deficiency of acid alpha-glucosidase. In addition to the classical infantile form of GSD II, several clinical variants are known. We describe an infant with the classical course of the disease. Our patient differs from the classical variant by the lack of cardiomegalia and the high residual activity of acid alpha-glucosidase in cultivated skin fibroblasts and muscle tissue. In the present case, however, glycogen storing lysosomes were found in peripheral lymphocytes and skeletal muscle cells. This finding underlines the particular value of ultrastructural investigation in the diagnosis of GSD II.
II型糖原贮积病(庞贝病,GSD II)是一种溶酶体贮积病,由遗传性酸性α-葡萄糖苷酶缺乏引起。除了经典的婴儿型GSD II,还有几种临床变异型。我们描述了一名患有该病经典病程的婴儿。我们的患者与经典变异型不同之处在于没有心脏肥大,且培养的皮肤成纤维细胞和肌肉组织中酸性α-葡萄糖苷酶的残余活性较高。然而,在本病例中,在外周淋巴细胞和骨骼肌细胞中发现了储存糖原的溶酶体。这一发现强调了超微结构研究在GSD II诊断中的特殊价值。