Surveying the Down syndrome mouse model resource identifies critical regions responsible for chronic otitis media.

Surveying the Down syndrome mouse model resource identifies critical regions responsible for chronic otitis media.
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DOI:
10.1007/s00335-013-9475-x
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发表时间:
2013-12
期刊:
影响因子:
2.5
通讯作者:
Brown, Steve D. M.
Brown, Steve D. M.
中科院分区:
生物学4区
文献类型:
--
作者:
Bhutta, Mahmood F.;Cheeseman, Michael T.;Herault, Yann;Yu, Yuejin E.;Brown, Steve D. M.

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慢性中耳炎(OM)在唐氏综合征(DS)中很常见,但潜在的病因尚不清楚。我们分析了所有可用的DS部分三体模型小鼠资源,寻找慢性中耳炎症的组织学证据。我们在Dp(16)1Yey小鼠中发现了一个高渗透性的OM,它携带一个完整的MMU16三体。Dp(17)1Yey小鼠和Dp(10)1Yey小鼠均未发现OM,提示疾病位点仅位于MMU16上。Ts1Cje、Ts1RhR、Ts2Yah和Ts65Dn三体和Tc1三体小鼠未发生OM。在这些发现的基础上,我们提出了一种基于Tc1小鼠HSA21非三体区域的寄存的DS慢性中耳炎症的双位点模型。我们还得出结论,环境因素可能在疾病发病中起重要作用。
Chronic otitis media (OM) is common in Down syndrome (DS), but underlying aetiology is unclear. We analysed the entire available mouse resource of partial trisomy models of DS looking for histological evidence of chronic middle-ear inflammation. We found a highly penetrant OM in the Dp(16)1Yey mouse, which carries a complete trisomy of MMU16. No OM was found in the Dp(17)1Yey mouse or the Dp(10)1Yey mouse, suggesting disease loci are located only on MMU16. The Ts1Cje, Ts1RhR, Ts2Yah, and Ts65Dn trisomies and the transchomosomic Tc1 mouse did not develop OM. On the basis of these findings, we propose a two-locus model for chronic middle-ear inflammation in DS, based upon epistasis of the regions of HSA21 not in trisomy in the Tc1 mouse. We also conclude that environmental factors likely play an important role in disease onset.
唐氏综合症小鼠模型中的中耳炎。
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