Drosophila lacking dfmr1 activity show defects in circadian output and fail to maintain courtship interest
Drosophila lacking dfmr1 activity show defects in circadian output and fail to maintain courtship interest
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DOI：
10.1016/s0896-6273(02)00724-9
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发表时间：
2002-06-13
期刊：
影响因子：
16.2
通讯作者：
Jongens, TA
中科院分区：
文献类型：
--
作者：
Dockendorff, TC;Su, HS;Jongens, TA
脆性X智力低下是一种由缺乏FMR 1基因产物（一种已知的RNA结合蛋白）引起的突出遗传性疾病。由FMR 1功能调节的特定生理途径尚未确定。成年dfmr 1（也称为dfxr）突变果蝇显示昼夜节律活动，并具有不稳定的运动活动模式，而dFMR 1的过表达导致延长的时间。DFMR 1突变体雄性也表现出降低的求偶活动，这似乎是由于它们不能维持求偶兴趣。分子分析未能揭示时钟成分表达的任何缺陷;然而，CREB输出受到影响。正常昼夜行为所需的神经元的形态学分析揭示了微妙的异常，这表明轴突寻路或突触形成的缺陷可能导致观察到的行为缺陷。
Fragile X mental retardation is a prominent genetic disorder caused by the lack of the FMR1 gene product, a known RNA binding protein. Specific physiologic pathways regulated by FMR1 function have yet to be identified. Adult dfmr1 (also called dfxr) mutant flies display arrhythmic circadian activity and have erratic patterns of locomotor activity, whereas overexpression of dFMR1 leads to a lengthened period. dfmr1 mutant males also display reduced courtship activity which appears to result from their inability to maintain courtship interest. Molecular analysis fails to reveal any defects in the expression of clock components; however, the CREB output is affected. Morphological analysis of neurons required for normal circadian behavior reveals subtle abnormalities, suggesting that defects in axonal pathfinding or synapse formation may cause the observed behavioral defects.