丝素a相互作用蛋白（FILIP）的缺失导致握力弱和对伤害性刺激的异常反应。

IF 2.5 4区医学 Q3 NEUROSCIENCES

Neuroscience Letters Pub Date : 2025-02-15 DOI:10.1016/j.neulet.2025.138158

Hideshi Yagi , Keizo Takao , Satoko Hattori , Yusuke Minato , Sachi Kuwahara-Otani , Seishi Maeda , Koichi Noguchi , Tsuyoshi Miyakawa , Makoto Sato

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引用次数: 0

摘要

丝蛋白a相互作用蛋白（Filamin a - interaction protein，小鼠中的FILIP，人类中的FILIP1）首先被确定为一种负性控制啮齿动物神经元迁移的蛋白，随后被证明对新皮层的发育至关重要。在之前的研究中，我们培育了FILIP敲除小鼠来研究FILIP在皮层发育中的体内功能。由于FILIP mRNA在体内广泛表达，我们系统地检测了敲除FILIP的小鼠，以确定FILIP在全身的功能。我们的结果显示，filip基因敲除小鼠表现出握力弱和感觉异常。有趣的是，我们还发现FILIP在背根神经节（DRG）的一个神经元亚群中表达。最近的研究报道，FILIP1突变导致严重的神经和肌肉骨骼异常，从而提出了一种新的疾病实体，称为FILIP1病。期望我们的filip基因敲除小鼠可以作为filip1病变病理研究的模型。

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Deletion of filamin A-interacting protein (FILIP) results in a weak grip strength and abnormal responses to nociceptive stimulation

Filamin A-interacting protein (FILIP in mice, FILIP1 in humans) was first identified as a protein that negatively controls neuronal migration in rodents, and was subsequently demonstrated to be pivotal for the development of the neocortex. In the previous study, we generated FILIP knockout mice to investigate the in vivo functions of FILIP in cortical development. Since FILIP mRNA is widely expressed in the body, we systematically examined FILIP-knockout mice to determine the functions of FILIP throughout the body. Our results showed that FILIP-knockout mice exhibited weak grip strength and sensory abnormalities. Interestingly, we also found that FILIP was expressed in a subset of neurons in the dorsal root ganglion (DRG). Recent research has reported that FILIP1 mutations lead to severe neurological and musculoskeletal abnormalities, resulting in the proposal of a new disease entity, termed FILIP1opathy. It is expected that our FILIP-knockout mice could be used as a model for the pathological investigation of FILIP1opathy.

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来源期刊

Neuroscience Letters 医学-神经科学

CiteScore

5.20

自引率

0.00%

发文量

408

审稿时长

50 days

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