Sh3pxd2b Mice Are a Model for Craniofacial Dysmorphology and Otitis Media 英文参考文献.docVIP

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Sh3pxd2b Mice Are a Model for Craniofacial Dysmorphology and Otitis Media 英文参考文献.doc

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Sh3pxd2b Mice Are a Model for Craniofacial Dysmorphology and Otitis Media 英文参考文献

Sh3pxd2bMiceAreaModelforCraniofacial DysmorphologyandOtitisMedia BinYang1,CongTian2,Zhi-guangZhang1*,Feng-chanHan2,RamiAzem2,HepingYu2,YeZheng2 ,Ge Jin3,JamesE.Arnold2,QingY.Zheng2* 1DepartmentofOralandMaxillofacialSurgery,GuanghuaSchoolofStomatology,SunYat-SenUniversity,Guangzhou,China,2DepartmentofOtolaryngology-Head andNeckSurgery,SchoolofMedicine,CaseWesternReserveUniversity,Cleveland,Ohio,UnitedStatesofAmerica,3SchoolofDentalMedicine,CaseWesternReserve University,Cleveland,Ohio,UnitedStatesofAmerica Abstract Craniofacial defects that occur through gene mutation during development increase vulnerability to eustachian tube dysfunction.Thesedefectscanleadtoanincreasedincidenceofotitismedia.Weexaminedtheeffectsofamutationinthe Sh3pxd2bgene(Sh3pxd2bnee)ontheprogressionofotitismediaandhearingimpairmentatvariousdevelopmentalstages. We found that all mice that had the Sh3pxd2bnee mutation went on to develop craniofacial dysmorphologies and subsequently otitis media, by as early as 11 days of age. We found noteworthy changes in cilia and goblet cells of the middle ear mucosa in Sh3pxd2bnee mutant mice using scanning electronic microscopy. By measuring craniofacial dimensions,wedeterminedforthefirsttimeinananimalmodelthatthismousehasalteredeustachiantubemorphology consistent withamore horizontalposition oftheeustachian tube.All mutantswerefoundtohavehearing impairment. ExpressionofTNF-aandTLR2,whichcorrelateswithinflammationinotitismedia,wasup-regulatedintheearsofmutant micewhenexaminedbyimmunohistochemistryandsemi-quantitativeRT-PCR.Themousemodelwithamutationinthe Sh3pxd2bgene(Sh3pxd2bnee)mirrorscraniofacialdysmorphologyandotitismediainhumans. Citation:YangB,TianC,ZhangZ-g,HanF-c,AzemR,etal.(2011)Sh3pxd2bMiceAreaModelforCraniofacialDysmorphologyandOtitisMedia.PLoSONE6(7): e22622.doi:10.1371/journal.pone.0022622 Editor:BruceRiley,TexasAMUniversity,UnitedStatesofAmerica ReceivedApril2,2011;AcceptedJune26,2011;PublishedJuly27,2011 Copyright: ? 2011 Yang et al. This i