Hudgson P, Fulthorpe J J
J Pathol. 1975 Jul;116(3):139-47. doi: 10.1002/path.1711160303.
Muscle biopsies were obtained from three infants under the age of 12 mth, each of whom was diagnosed as having Pompe's disease. The biopsies revealed a severe vacuolar myopathy with accumulation of large amounts of PAS positive material within the muscle fibres, changes similar to those in adult cases of the disease. In addition large amounts of metachromatic material were found within the muscle fibres in all three cases and in two of them scattered, rather sparse perivascular lymphocytic infiltrates were seen in the interstitial tissue. Review of material previously obtained from two adult cases showed no accumulation of metachromatic material in the older case and only moderate amounts in the younger. However, dense interstitial lymphocytic infiltrates were seen in the former, some concentrated around small vessels. These observations suggest that the pathogenesis of the muscle disorder in acid maltase deficiency may not depend on abnormal glycogen storage only.
对3名12个月以下的婴儿进行了肌肉活检,他们均被诊断为患有庞贝氏病。活检显示为严重的空泡性肌病,肌纤维内有大量PAS阳性物质积聚,病变与该疾病的成人病例相似。此外,在所有3例病例的肌纤维内均发现大量异染性物质,其中2例在间质组织中可见散在、较为稀疏的血管周围淋巴细胞浸润。回顾之前从2例成人病例中获取的材料,发现年龄较大的病例中无异染性物质积聚,年龄较小的病例中仅有少量。然而,在前者中可见密集的间质淋巴细胞浸润,一些集中在小血管周围。这些观察结果表明,酸性麦芽糖酶缺乏症所致肌肉疾病的发病机制可能不仅仅取决于异常糖原储存。