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[法布里病;迈向一种治疗方法]

[Fabry's disease; towards a treatment].

作者信息

Linthorst G E, Hollak C E, Bosman D K, Heymans H S, Aerts J M

机构信息

Afd. Inwendige Geneeskunde, onderafd. Klinische Hematologie, Academisch Medisch Centrum, Meibergdreef 9, 1105 AZ Amsterdam.

出版信息

Ned Tijdschr Geneeskd. 2000 Dec 9;144(50):2391-5.

Abstract

Fabry's disease, deficiency of the enzyme alpha-galactosidase A, is an X-linked lysosomal storage disorder. Clinical symptoms are caused by continuous deposition of specific glycolipids in endothelial cells, neural cells, skin and cornea. These depositions give rise to skin (angiokeratoma) and eye abnormalities (cornea verticillata), acroparaesthesias and anhidrosis and later in life cause renal insufficiency and cardiovascular complications. Hemizygous males suffer from Fabry's disease, whereas female carriers (heterozygotes) are usually asymptomatic. Recently, an atypical presentation of Fabry's disease was described in males who only presented with cardiac involvement. Therefore, the actual number of Fabry patients in the Netherlands could be higher than the predicted 300. Diagnosis in males can be established by measuring alpha-galactosidase enzyme activity in plasma, leukocytes or fibroblasts. Apart from kidney transplantation only symptomatic therapy is available today. Enzyme supplementation therapy (as shown in Gaucher's disease) and substrate deprivation are possible ways of treatment in the future.

摘要

法布里病,即α - 半乳糖苷酶A缺乏症,是一种X连锁溶酶体贮积症。临床症状是由特定糖脂在内皮细胞、神经细胞、皮肤和角膜中持续沉积引起的。这些沉积会导致皮肤(血管角质瘤)和眼部异常(涡状角膜)、肢端感觉异常和无汗,后期会引发肾功能不全和心血管并发症。半合子男性会患法布里病,而女性携带者(杂合子)通常无症状。最近,在仅表现为心脏受累的男性中描述了法布里病的一种非典型表现。因此,荷兰法布里病患者的实际数量可能高于预测的300例。男性患者的诊断可通过测量血浆、白细胞或成纤维细胞中的α - 半乳糖苷酶活性来确定。如今,除了肾移植外,仅能进行对症治疗。酶替代疗法(如戈谢病中所示)和底物剥夺是未来可能的治疗方法。

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