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Pediatr Res：成骨不全的胶原α1型（COL1A1）突变小鼠的肺和膈肌病理学

2018-03-16 MedSci MedSci原创

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成骨不全（OI）通常是由I型胶原基因突变引起的。呼吸系统并发症主要是由于脊柱和肋骨畸形所致。本研究假设可能会发生肺部实质和/或膈肌直接受累。 使用Col1a1Jrt/+小鼠，一种严重显性OI模型，使用平均线性截距长度（Lm）来评估远端空域大小。确定膈肌纤维的横截面积（CSA）和肌球蛋白重链（MyHC）表型以及收缩性质。抗TGF-β的中和抗体处理OI小鼠。 结果显示，OI小鼠出现远端空域扩大（Lm

成骨不全（OI）通常是由I型胶原基因突变引起的。呼吸系统并发症主要是由于脊柱和肋骨畸形所致。本研究假设可能会发生肺部实质和/或膈肌直接受累。

使用Col1a1Jrt/+小鼠，一种严重显性OI模型，使用平均线性截距长度（Lm）来评估远端空域大小。确定膈肌纤维的横截面积（CSA）和肌球蛋白重链（MyHC）表型以及收缩性质。抗TGF-β的中和抗体处理OI小鼠。

结果显示，OI小鼠出现远端空域扩大（Lm + 27％）。膈肌厚度和纤维数量减少，而快速抽搐型IIx/IIb MyHC纤维增加。隔膜的离体力产生（标准化为CSA）也明显降低。抗TGF-β抗体处理未能阻止OI小鼠中Lm值增加。

综上所述，Col1a1Jrt/+的OI小鼠模型表明：1）不受TGF-β驱使的肺部空间扩大；2）减少膈肌的肌肉质量和固有的收缩无力。这些结果表明OI呼吸并发症复杂且多面，不能完全归因于骨表现。

原始出处：

Baglole CJ, Liang F, et al., Pulmonary and diaphragmatic pathology in collagen alpha1 type I (COL1A1) mutant mice with osteogenesis imperfect. Pediatr Res. 2018 Mar 14. doi: 10.1038/pr.2018.36. [Epub ahead of print]

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    2018-09-15 feather89

    #PE#

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    2018-11-13 ms9608593228839890

    #DIA#

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    2018-03-18 zhaojie88

    #成骨#

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    2018-03-18 jktdtl

    #病理学#

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    2018-03-18 爆笑小医

    #肌病#

    0

  6. [GetPortalCommentsPageByObjectIdResponse(id=1727158, encodeId=50861e271583a, content=<a href='/topic/show?id=986913980dd' target=_blank style='color:#2F92EE;'>#PE#</a>, beContent=null, objectType=article, channel=null, level=null, likeNumber=54, replyNumber=0, topicName=null, topicId=null, topicList=[TopicDto(id=13980, encryptionId=986913980dd, topicName=PE)], attachment=null, authenticateStatus=null, createdAvatar=null, createdBy=f06c33759375, createdName=feather89, createdTime=Sat Sep 15 11:23:00 CST 2018, time=2018-09-15, status=1, ipAttribution=), GetPortalCommentsPageByObjectIdResponse(id=1638489, encodeId=0dbf1638489f3, content=<a href='/topic/show?id=afda591646' target=_blank style='color:#2F92EE;'>#DIA#</a>, beContent=null, objectType=article, channel=null, level=null, likeNumber=58, replyNumber=0, topicName=null, topicId=null, topicList=[TopicDto(id=5916, encryptionId=afda591646, topicName=DIA)], attachment=null, authenticateStatus=null, createdAvatar=null, createdBy=987022654425, createdName=ms9608593228839890, createdTime=Tue Nov 13 08:23:00 CST 2018, time=2018-11-13, status=1, ipAttribution=), GetPortalCommentsPageByObjectIdResponse(id=1351684, encodeId=c66a13516849f, content=<a href='/topic/show?id=e95954093cd' target=_blank style='color:#2F92EE;'>#成骨#</a>, beContent=null, objectType=article, channel=null, level=null, likeNumber=64, replyNumber=0, topicName=null, topicId=null, topicList=[TopicDto(id=54093, encryptionId=e95954093cd, topicName=成骨)], attachment=null, authenticateStatus=null, createdAvatar=, createdBy=4c37265, createdName=zhaojie88, createdTime=Sun Mar 18 05:23:00 CST 2018, time=2018-03-18, status=1, ipAttribution=), GetPortalCommentsPageByObjectIdResponse(id=1473455, encodeId=b06e14e3455ed, content=<a href='/topic/show?id=4fd1e102257' target=_blank style='color:#2F92EE;'>#病理学#</a>, beContent=null, objectType=article, channel=null, level=null, likeNumber=68, replyNumber=0, topicName=null, topicId=null, topicList=[TopicDto(id=71022, encryptionId=4fd1e102257, topicName=病理学)], attachment=null, authenticateStatus=null, createdAvatar=, createdBy=c8ce7226273, createdName=jktdtl, createdTime=Sun Mar 18 05:23:00 CST 2018, time=2018-03-18, status=1, ipAttribution=), GetPortalCommentsPageByObjectIdResponse(id=1622321, encodeId=502c1622321c9, content=<a href='/topic/show?id=b9578094833' target=_blank style='color:#2F92EE;'>#肌病#</a>, beContent=null, objectType=article, channel=null, level=null, likeNumber=60, replyNumber=0, topicName=null, topicId=null, topicList=[TopicDto(id=80948, encryptionId=b9578094833, topicName=肌病)], attachment=null, authenticateStatus=null, createdAvatar=, createdBy=395f25, createdName=爆笑小医, createdTime=Sun Mar 18 05:23:00 CST 2018, time=2018-03-18, status=1, ipAttribution=), GetPortalCommentsPageByObjectIdResponse(id=296824, encodeId=e17d296824bb, content=谢谢分享学习, beContent=null, objectType=article, channel=null, level=null, likeNumber=65, replyNumber=0, topicName=null, topicId=null, topicList=[], attachment=null, authenticateStatus=null, createdAvatar=https://wx.qlogo.cn/mmopen/Q3auHgzwzM4o2XVpJ6449yqDsjibydI5HnJ8MibGxNn4HZiaZib0bdfibbwoz3KQrIyN9AGxq36ZlZRNicYlPfOOWMtwgsajY6mGNFiaPclFdowjD0/0, createdBy=97bd1338592, createdName=清风拂面, createdTime=Fri Mar 16 15:42:50 CST 2018, time=2018-03-16, status=1, ipAttribution=)]
    2018-03-16 清风拂面

    谢谢分享学习

    0

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