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法布里病与肾脏受累:从儿童期开始了解未来

Fabry disease and kidney involvement: starting from childhood to understand the future.

作者信息

Chimenz Roberto, Chirico Valeria, Cuppari Caterina, Ceravolo Giorgia, Concolino Daniela, Monardo Paolo, Lacquaniti Antonio

机构信息

Pediatric Nephrology and Dialysis Unit, University Hospital "G. Martino", Messina, Italy.

Unit of Pediatric Emergency, Department of Adult and Childhood Human Pathology, University Hospital of Messina, Messina, Italy.

出版信息

Pediatr Nephrol. 2022 Jan;37(1):95-103. doi: 10.1007/s00467-021-05076-x. Epub 2021 Apr 30.

Abstract

The accumulation of globotriaosylceramide (Gb-3) in multiple organs, such as the heart, kidney, and nervous system, due to mutations in the galactosidase alpha (GLA) gene, represents the key point of Fabry disease (FD). The common symptoms appear in childhood or adolescence, including neuropathic pain, angiokeratoma, acroparesthesia, and corneal opacities. A multi-organ involvement induces a significant deterioration in the quality of life with high mortality in adulthood. The accumulation of Gb-3 involves all types of kidney cells beginning at fetal development, many years before clinical manifestations. A decline in the glomerular filtration rate is rare in children, but it can occur during adolescence. Pediatric patients rarely undergo kidney biopsy that could assess the efficacy of enzyme replacement therapy (ERT) behind its diagnostic role. To date, diagnosis is achieved by detecting reduced α-Gal-A activity in leukocytes and plasma, allowing for the early start of ERT. This review focuses on pediatric kidney involvement in FD, analyzing in depth its diagnostic processes and treatment options.

摘要

由于α-半乳糖苷酶(GLA)基因突变,导致多器官(如心脏、肾脏和神经系统)中球三糖神经酰胺(Gb-3)的蓄积,是法布里病(FD)的关键特征。常见症状出现在儿童期或青少年期,包括神经性疼痛、血管角质瘤、肢端感觉异常和角膜混浊。多器官受累会导致成年期生活质量显著下降且死亡率很高。从胎儿发育开始,早在临床表现出现多年之前,Gb-3的蓄积就累及所有类型的肾细胞。儿童期肾小球滤过率下降很少见,但可能在青少年期发生。儿科患者很少进行肾脏活检,肾脏活检除了具有诊断作用外,还可以评估酶替代疗法(ERT)的疗效。迄今为止,通过检测白细胞和血浆中α-Gal-A活性降低来实现诊断,从而能够尽早开始ERT。本综述重点关注FD患儿的肾脏受累情况,深入分析其诊断过程和治疗选择。

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