Breathing abnormalities in a female mouse model of Rett syndrome.
Breathing abnormalities in a female mouse model of Rett syndrome.
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DOI：
10.1007/s12576-015-0384-5
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发表时间：
2015-09
期刊：
影响因子：
--
通讯作者：
Jiang C
中科院分区：
文献类型：
--
作者：
Johnson CM;Cui N;Zhong W;Oginsky MF;Jiang C
Rett综合征（RTT）是一种女性神经发育疾病，伴有呼吸异常。为了了解呼吸缺陷是否发生在一组雌性Mecp 2 +/−小鼠（RTT小鼠模型）的早期生命中，以及显示RTT样呼吸异常的小鼠百分比，使用体积描记法测量清醒小鼠的呼吸活动。一组Mecp 2 +/−雌性小鼠的呼吸频率变化显示出与Mecp 2 −/Y雄性小鼠相似的分布模式，而其余小鼠则与野生型小鼠相似。使用k-均值聚类统计分析获得了类似的结果。通过这两种独立的方法，大约20%的雌性Mecp 2 +/−小鼠表现出RTT样呼吸异常，这种异常早在Mecp 2 +/−小鼠3周龄时就开始了，似乎没有随着生长而恶化，并且在3%CO2下受到抑制。只有一小部分Mecp 2 +/−小鼠出现RTT样呼吸异常，这一发现表明RTT模型Mecp 2 +/−小鼠中的等位基因失活不完全。
Rett Syndrome (RTT) is a female neurodevelopmental disease with breathing abnormalities. To understand whether breathing defects occur in the early lives of a group of female Mecp2+/− mice, a mouse model of RTT, and what percentage of mice show RTT-like breathing abnormality, breathing activity was measured with plethysmography in conscious mice. Breathing frequency variation in a group of Mecp2+/− females displayed a distribution pattern similar to Mecp2−/Y males, while the rest resembled the wild-type mice. Similar results were obtained using the k-mean clustering statistics analysis. With these two independent methods, about 20% of female Mecp2+/− mice showed RTT-like breathing abnormalities that began as early as 3 weeks of age in the Mecp2+/− mice, did not seem to deteriorate with growth, and were suppressed with 3% CO2. The finding that only a small proportion of Mecp2+/− mice develop RTT-like breathing abnormalities suggests incomplete allele inactivation in the RTT-model Mecp2+/− mice.
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