Sessa Adalberto, Toson Antonella, Nebuloni Manuela, Pallotti Francesco, Giordano Ferdinando, Battini Graziana, Maglio Alessia, Meroni Mietta, Calconi Gilberto, Bertolone Gabriele, Gatti Pierlucio
Division of Nephrology and Dialysis, Vimercate Hospital, Italy.
J Nephrol. 2002 Mar-Apr;15(2):109-12.
Anderson-Fabry disease (AFd) is caused by an X-linked inborn error in the glycosphingoLipid metabolic pathway due to an enzymatic defect in a lysosomal hydrolase: alpha-galactosidase A. The defect results in the progressive accumulation of neutral glycosphingolipids in most body fluids and several tissues. The clinical manifestations of AFd are related to organ damage and, obviously, are more severe in hemizygous males than in heterozygous females. In the third decade of life, the course of the disease involves severe deterioration of kidney function progressing to end-stage renal failure. All kind of cells of renal structures are filled with glycosphingolipid deposits. Electron microscopic studies document typical intracytoplasmic osmiophilic bodies with a characteristic "zebra" or "onion-skin" appearance due to concentric lamellation of alternating clear and dark layers. Clinical interest in Fabry patients is related to recent advances in treatment with an intravenous specific enzyme to modify the biochemical error of the glycosphingolipid catabolic pathway.
安德森-法布里病(AFd)是由糖鞘脂代谢途径中的X连锁先天性缺陷引起的,这是由于溶酶体水解酶α-半乳糖苷酶A存在酶缺陷。该缺陷导致中性糖鞘脂在大多数体液和多种组织中逐渐蓄积。AFd的临床表现与器官损害有关,显然,半合子男性的症状比杂合子女性更为严重。在生命的第三个十年,疾病进程涉及肾功能严重恶化,最终发展为终末期肾衰竭。肾脏结构的所有细胞都充满了糖鞘脂沉积物。电子显微镜研究记录了典型的胞浆嗜锇小体,因其交替的明暗层呈同心层状排列而具有特征性的“斑马”或“洋葱皮”外观。对法布里病患者的临床关注与静脉注射特定酶治疗的最新进展有关,这种治疗可纠正糖鞘脂分解代谢途径中的生化错误。