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日本特定疾病治疗研究事业中 Fabry 病受惠者的描述性流行病学研究。

Descriptive epidemiology of Fabry disease among beneficiaries of the Specified Disease Treatment Research Program in Japan.

机构信息

Department of Hematology, Nagoya Central Hospital, Nagoya, Japan.

出版信息

J Epidemiol. 2012;22(4):370-4. doi: 10.2188/jea.je20110101. Epub 2012 Jun 9.

Abstract

BACKGROUND

Fabry disease (FD) is a rare X-linked lysosomal storage disorder and is included in the Specified Disease Treatment Research Program in Japan, which subsidizes medical care for beneficiaries with rare and other, designated diseases. However, no report on the epidemiologic features of Fabry disease has been published in Japan.

METHODS

We used clinical research data reports submitted to the program between 2003 and 2008 to assess the epidemiologic features of 315 beneficiaries with FD.

RESULTS

Of the 315 program beneficiaries, 198 were men (mean age, 37.4 years) and 117 were women (mean age, 51.2 years). The overall incidence in Japan was 0.25 cases per 100,000 individuals, and prevalence among men was 1.78 times that among women. More than 80% of beneficiaries were capable of working, going to school, or doing housework; however, 46 beneficiaries (14.6%) required home care, and 9 (2.9%) were living in hospitals or other medical facilities. As compared with the previous year, the clinical course of FD at beneficiary registration was unchanged for 178 of 290 beneficiaries (61.4%), worse for 81 (27.9%), and improved or cured for 31 (10.7%). The distribution of beneficiary-related characteristics was similar between men and women, and no significant difference was observed.

CONCLUSIONS

The high percentage (>80%) of individuals with FD who were able to work, attend school, and perform tasks such as housework could reflect an improvement in the clinical course of FD after enzyme replacement therapy. We must continue data collection and conduct further studies to improve our understanding of the descriptive epidemiology of FD.

摘要

背景

法布瑞病(Fabry 病,FD)是一种罕见的 X 连锁溶酶体贮积症,已被纳入日本特定疾病治疗研究计划,为患有罕见病和其他指定疾病的受益人群提供医疗补贴。然而,目前在日本尚未发表关于 Fabry 病流行病学特征的报告。

方法

我们使用该计划在 2003 年至 2008 年期间提交的临床研究数据报告,评估了 315 名 FD 受益人的流行病学特征。

结果

在 315 名计划受益人群中,男性 198 人(平均年龄 37.4 岁),女性 117 人(平均年龄 51.2 岁)。日本的总发病率为 0.25 例/10 万人,男性的患病率是女性的 1.78 倍。超过 80%的受益人群能够工作、上学或做家务;然而,有 46 名(14.6%)需要家庭护理,9 名(2.9%)居住在医院或其他医疗机构中。与前一年相比,290 名登记受益人群中,178 名(61.4%)的 FD 临床病程保持不变,81 名(27.9%)恶化,31 名(10.7%)改善或痊愈。男性和女性受益人群的特征分布相似,无显著差异。

结论

大多数(>80%)FD 患者能够工作、上学和做家务,这反映了酶替代疗法后 FD 临床病程的改善。我们必须继续收集数据并开展进一步研究,以提高我们对 FD 描述性流行病学的理解。

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