结节性硬化症

IF 1.1 Q4 MEDICINE, RESEARCH & EXPERIMENTAL
Mari Wataya-Kaneda
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引用次数: 1

摘要

结节性硬化症(TSC)是一种常染色体显性遗传性疾病,其特征为全身性错构瘤、TAND (TSC相关神经精神疾病)和白癜风。这些症状是由于致病基因TSC1或TSC2的基因突变引起的雷帕霉素复合体1 (mTORC1)的机制靶点的持续激活。该病发病机制的阐明和诊断技术的进步使TSC的诊断和治疗发生了巨大变化。诊断标准已在全球范围内建立,mTORC1抑制剂已成为该疾病的治疗剂。以前,治疗策略仅限于对症治疗,如手术。mTORC1抑制剂对TSC的所有症状都有效,但它们也有全身副作用。因此,对一个跨学科、协作的医疗保健系统的需求增加了,从而建立了一个被称为“TSC委员会”的实践结构。此外,为了减少全身给药mTORC1抑制剂的副作用,日本开发了一种局部mTORC1抑制剂制剂,用于治疗TSC引起的皮肤病变。本报告总结了TSC的发病机制和现状,以及神经皮肤综合征政策研究小组对厚生劳动省有关这一罕见难治性疾病政策的贡献。
本文章由计算机程序翻译,如有差异,请以英文原文为准。
Tuberous Sclerosis Complex.

Tuberous sclerosis complex (TSC) is an autosomal dominant inherited disease characterized by systemic hamartomas, neuropsychiatric symptoms known as TAND (TSC-associated neuropsychiatric disorders), and vitiligo. These symptoms are attributed to the constant activation of mechanistic target of rapamycin complex 1 (mTORC1) caused by genetic mutations in the causative genes TSC1 or TSC2. The elucidation of the pathogenesis of this disease and advances in diagnostic technologies have led to dramatic changes in the diagnosis and treatment of TSC. Diagnostic criteria have been created at a global level, and mTORC1 inhibitors have emerged as therapeutic agents for this disease. Previously, the treatment strategy was limited to symptomatic treatments such as surgery. Inhibitors of mTORC1 are effective against all symptoms of TSC, but they also have systemic side effects. Therefore, the need for a cross-disciplinary, collaborative medical care system has increased, resulting in the establishment of a practice structure known as the "TSC Board." Furthermore, to reduce the side effects of systemic administration of mTORC1 inhibitors, a topical formulation of mTORC1 inhibitor was developed in Japan for the treatment of skin lesions caused by TSC. This report summarizes the pathogenesis and current status of TSC and the contribution of the Neurocutaneous Syndrome Policy Research Group to the policies of the Ministry of Health, Labor, and Welfare with respect to this rare, intractable disease.

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来源期刊
KEIO JOURNAL OF MEDICINE
KEIO JOURNAL OF MEDICINE MEDICINE, RESEARCH & EXPERIMENTAL-
CiteScore
3.10
自引率
0.00%
发文量
23
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