Institute of Inherited Metabolic Disorders, Charles University, 1st Faculty of Medicine and Teaching Hospital, Prague, Czech Republic.
J Inherit Metab Dis. 2010 Dec;33 Suppl 3(Suppl 3):S297-300. doi: 10.1007/s10545-010-9160-0. Epub 2010 Jul 14.
Ultrastructural and histochemical studies of bioptic and postmortem tissue samples from ten Fabry hemizygotes showed lysosomal storage in adipocytes as a constant feature of the classic phenotype of α-galactosidase (GLA) deficiency. The storage was represented by a crescent-shaped line of storage lysosomes of varying thicknesses restricted to the perinuclear subplasmalemmal area. The ultrastructure of the storage lysosomes was dominated by concentric lipid membranes modified by simultaneous deposition of autofluorescent ceroid. Storage was widely expressed in adipose tissue. The number of storage lysosomes was increased, and the lysosomes were more clustered in adipocytes with less voluminous lipid content. The findings should attract interest to studies of adipose tissue biology in Fabry disease, a topic that has not been studied so far. In terms of cell biology, the observations represent indirect evidence of significant lysosomal turnover of α-galactose lipid conjugates in adipocytes demasked by GLA deficiency. The results extend the thus far limited information on the adipocyte lysosomal system and its participation in lysosomal storage disorders.
对 10 例法布里半合子的活检和尸检组织样本的超微结构和组织化学研究表明,溶酶体贮存在脂肪细胞中是 α-半乳糖苷酶(GLA)缺乏症经典表型的一个恒定特征。贮存在受核周质膜下区域限制的新月形不同厚度的贮留溶酶体中。贮存溶酶体的超微结构主要由同心脂质膜组成,同时伴有自发荧光类脂的沉积。贮存广泛表达于脂肪组织中。贮存溶酶体的数量增加,且溶酶体在脂含量较少的脂肪细胞中更加聚集。这些发现应该会引起人们对法布里病脂肪组织生物学的研究兴趣,迄今为止,这一课题尚未得到研究。就细胞生物学而言,这些观察结果代表了间接证据,表明 GLA 缺乏症使α-半乳糖脂类缀合物的溶酶体发生了大量周转。这些结果扩展了迄今为止关于脂肪细胞溶酶体系统及其在溶酶体贮积病中的参与的有限信息。