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迟发性法布里病:一种表现为肌阵挛综合征的成人变异型。

Later-onset Fabry disease: an adult variant presenting with the cramp-fasciculation syndrome.

作者信息

Nance Christopher S, Klein Christopher J, Banikazemi Maryam, Dikman Steven H, Phelps Robert G, McArthur Justin C, Rodriguez Moses, Desnick Robert J

机构信息

Peripheral Neuropathy Research Center and Multiple Sclerosis Research Center, Mayo Clinic, Rochester, Minn, USA.

出版信息

Arch Neurol. 2006 Mar;63(3):453-7. doi: 10.1001/archneur.63.3.453.

Abstract

BACKGROUND

Classic Fabry disease, an X-linked recessive lysosomal storage disease due to the deficient activity of alpha-galactosidase A, typically presents in early childhood with acroparesthesias, angiokeratomas, hypohidrosis, and corneal dystrophy. The neuropathic pain presumably results from glycosphingolipid accumulation in the vascular endothelium and in small-caliber nerve fibers, and is treatable by enzyme replacement therapy. Later-onset variants with residual alpha-galactosidase A activity lack vascular endothelial involvement and classic symptoms, which lead to the development of cardiac and/or renal disease after the fourth decade of life.

OBJECTIVE

To expand the later-onset Fabry phenotype to include cramp-fasciculation syndrome without small-fiber neuropathy.

METHODS

A 34-year-old man who presented with chronic exercise-induced pain, fasciculations, and cramps of the feet and legs, and his similarly affected mother, were evaluated. Clinical, biochemical, and molecular studies were performed.

RESULTS

Clinical evaluation suggested the diagnosis of Fabry disease, which was confirmed by reduced plasma and leukocyte alpha-galactosidase A activities (8.8% and 13.4% of normal, respectively) due to a missense A143T mutation. His mother was heterozygous for the A143T mutation.

CONCLUSION

The presentation of cramps and fasciculations without apparent small-fiber neuropathy expands the phenotype of later-onset Fabry disease.

摘要

背景

典型的法布里病是一种X连锁隐性溶酶体贮积病,因α-半乳糖苷酶A活性缺乏所致,通常在儿童早期出现肢端感觉异常、血管角质瘤、少汗症和角膜营养不良。神经病理性疼痛可能是由于糖鞘脂在血管内皮和小口径神经纤维中蓄积所致,可通过酶替代疗法治疗。具有残余α-半乳糖苷酶A活性的晚发型变异型缺乏血管内皮受累及典型症状,导致在生命的第四个十年后出现心脏和/或肾脏疾病。

目的

扩大晚发型法布里病的表型,使其包括无小纤维神经病变的痉挛-肌束震颤综合征。

方法

对一名34岁男性及其同样受累的母亲进行评估,该男性表现为慢性运动诱发的足部和腿部疼痛、肌束震颤和痉挛。进行了临床、生化和分子研究。

结果

临床评估提示为法布里病,血浆和白细胞α-半乳糖苷酶A活性降低(分别为正常水平的8.8%和13.4%),经错义A143T突变证实。他的母亲为A143T突变的杂合子。

结论

出现无明显小纤维神经病变的痉挛和肌束震颤扩大了晚发型法布里病的表型。

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