Huang W J, Zhang X, Chen W W
Department of Neurology, Xuzhou Central Hospital, Xuzhou, Jiangsu, China.
Eur Rev Med Pharmacol Sci. 2015 Apr;19(7):1219-26.
Gaucher disease is a multisystemic disorder that affects men and woman in equal numbers and occurs in all ethnic groups at any age with racial variations and an estimated worldwide incidence of 1/75,000. It is caused by a genetic deficient activity of the lysosomal enzyme glucocerebrosidase due to mutations in the β-glucocerebrosidase gene, and resulting in lack of glucocerebroside degradation. The subsequent accumulation of glucocerebroside in lysosomes of tissue macrophages primarily in the liver, bone marrow and spleen, causes damage in haematological, skeletal and nervous systems. The clinical manifestations show a high degree of variability with symptoms that varies according to organs involved. In many cases, these disorders do not correlate with mutations in the β-glucocerebrosidase gene. Although several mutations have been identified as responsible for the deficient activity of glucocerebrosidase, mechanisms by which this enzymatic defect leads to Gaucher disease remain poorly understood. Recent reports indicate the implication of complex mechanisms, including enzyme deficiency, substrate accumulation, unfolded protein response, and macrophage activation. Further elucidating these mechanisms will advance understanding of Gaucher disease and related disorders.
戈谢病是一种多系统疾病,男女发病率相等,可发生于任何年龄的所有种族群体,存在种族差异,全球估计发病率为1/75000。它是由β-葡萄糖脑苷脂酶基因突变导致溶酶体酶葡萄糖脑苷脂酶活性遗传缺陷引起的,导致葡萄糖脑苷脂降解缺乏。随后,葡萄糖脑苷脂主要在肝脏、骨髓和脾脏的组织巨噬细胞溶酶体中积累,对血液、骨骼和神经系统造成损害。临床表现具有高度变异性,症状因受累器官而异。在许多情况下,这些病症与β-葡萄糖脑苷脂酶基因突变无关。尽管已鉴定出几种突变是导致葡萄糖脑苷脂酶活性缺陷的原因,但这种酶缺陷导致戈谢病的机制仍知之甚少。最近的报告表明复杂机制的参与,包括酶缺乏、底物积累、未折叠蛋白反应和巨噬细胞激活。进一步阐明这些机制将增进对戈谢病及相关疾病的理解。