斑马鱼 trpm7 突变体在自由运动时运动能力下降。

IF 1.7 4区 生物学 Q4 CELL BIOLOGY Development Growth & Differentiation Pub Date : 2024-07-06 DOI:10.1111/dgd.12937
Kenta Watai, Kenichiro Sadamitsu, Seiji Wada, Makoto Kashima, Hiromi Hirata
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引用次数: 0

摘要

帕金森病是一种以运动功能减退、抑郁和痴呆为特征的神经系统疾病。关岛帕金森病痴呆伴肌萎缩性硬化症是西太平洋关岛和罗塔岛以及日本纪伊半岛报告的帕金森病的一个地方病例。先前的一项遗传学研究表明,关岛帕金森病可归因于 TRPM7 基因的变异,该基因编码与美司他丁相关的瞬态受体电位(TRP)离子通道。但是,帕金森症与 TRPM7 基因之间的联系仍然难以捉摸。之前的研究发现,TRPM7 基因缺陷的斑马鱼胚胎在色素沉着和触觉诱发的运动反应方面表现出缺陷。在本研究中,我们发现了一个新的可存活的等位基因trpm7突变体,该突变体的第一个跨膜结构域发生了I756N氨基酸置换。行为分析表明,trpm7突变体的运动能力受到影响,其运动距离比野生型幼虫短。与野生型幼虫相比,trpm7突变体的运动速度明显降低。与之前在斑马鱼trpm7突变体中发现的多巴胺能神经元减少的现象一样,trpm7突变体运动能力的降低也表明了trpm7表型与帕金森病症状之间的另一种相似性。
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Zebrafish trpm7 mutants show reduced motility in free movement

Parkinson's disease is a neurological disorder characterized by reduced motility, depression and dementia. Guamanian parkinsonism dementia with amyotrophic sclerosis is a local case of Parkinson's disease reported in the Western Pacific Islands of Guam and Rota as well as in the Kii Peninsula of Japan. A previous genetic study has suggested that Guamanian parkinsonism is attributable to a variant of the TRPM7 gene, which encodes for melastatin-related transient receptor potential (TRP) ion channels. But the link between parkinsonism and the TRPM7 gene remains elusive. Previous studies have addressed that trpm7-deficient zebrafish embryos showed defects in pigmentation and touch-evoked motor response. In this study, we identified a new viable allele of trpm7 mutant causing an I756N amino acid substitution in the first transmembrane domain. Behavioral analyses revealed that trpm7 mutants showed compromised motility with their movement distance shorter than wild-type larvae. The velocity of the movement was significantly reduced in trpm7 mutants than in wild-type larvae. Along with a previous finding of reduced dopaminergic neurons in zebrafish trpm7 mutants, reduced motility of trpm7 mutants can suggest another similarity between trpm7 phenotypes and Parkinson's disease symptoms.

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来源期刊
Development Growth & Differentiation
Development Growth & Differentiation 生物-发育生物学
CiteScore
4.60
自引率
4.00%
发文量
62
审稿时长
6 months
期刊介绍: Development Growth & Differentiation (DGD) publishes three types of articles: original, resource, and review papers. Original papers are on any subjects having a context in development, growth, and differentiation processes in animals, plants, and microorganisms, dealing with molecular, genetic, cellular and organismal phenomena including metamorphosis and regeneration, while using experimental, theoretical, and bioinformatic approaches. Papers on other related fields are also welcome, such as stem cell biology, genomics, neuroscience, Evodevo, Ecodevo, and medical science as well as related methodology (new or revised techniques) and bioresources. Resource papers describe a dataset, such as whole genome sequences and expressed sequence tags (ESTs), with some biological insights, which should be valuable for studying the subjects as mentioned above. Submission of review papers is also encouraged, especially those providing a new scope based on the authors’ own study, or a summarization of their study series.
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