Hei Yeun Koo, Jae Hwan Oh, María Beatriz Durán Alonso, Iris López Hernández, Margarita González-Vallinas, María Teresa Alonso, Juan J Tena, Alejandro Gil-Gálvez, Fernando Giraldez, Jinwoong Bok, Thomas Schimmang
{"title":"对 Meis2 基因敲除小鼠的分析揭示了由音速刺猬介导的耳蜗导管模式化。","authors":"Hei Yeun Koo, Jae Hwan Oh, María Beatriz Durán Alonso, Iris López Hernández, Margarita González-Vallinas, María Teresa Alonso, Juan J Tena, Alejandro Gil-Gálvez, Fernando Giraldez, Jinwoong Bok, Thomas Schimmang","doi":"10.1002/dvdy.747","DOIUrl":null,"url":null,"abstract":"<p><strong>Background: </strong>The mechanisms underlying the formation of complex structures such as during the outgrowth of the cochlear duct are still poorly understood.</p><p><strong>Results: </strong>We have analyzed the morphological and molecular changes associated with cochlear development in mouse mutants for the transcription factor Meis2, which show defective coiling of the cochlea. These morphological abnormalities were accompanied by the formation of ectopic and extra rows of sensory hair cells. Gene profiling of otic vesicles from Meis2 mutants revealed a dysregulation of genes that are potentially involved in Sonic hedgehog (Shh)-mediated patterning of the cochlear duct. Like in Shh mutants, Meis2 defective mice showed a loss of genes that are expressed in the apical part of the cochlear duct.</p><p><strong>Conclusions: </strong>Taken together, these data reveal that the loss of Meis2 leads to a phenotype that resembles Shh mutants, suggesting that Meis2 is instrumental for cochlear Shh signaling. The modulation of the same subset of genes provides an interesting insight into which Shh responsive genes are essential for outgrowth and patterning of the cochlear duct.</p>","PeriodicalId":11247,"journal":{"name":"Developmental Dynamics","volume":" ","pages":""},"PeriodicalIF":2.0000,"publicationDate":"2024-10-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":"0","resultStr":"{\"title\":\"Analysis of Meis2 knockout mice reveals Sonic hedgehog-mediated patterning of the cochlear duct.\",\"authors\":\"Hei Yeun Koo, Jae Hwan Oh, María Beatriz Durán Alonso, Iris López Hernández, Margarita González-Vallinas, María Teresa Alonso, Juan J Tena, Alejandro Gil-Gálvez, Fernando Giraldez, Jinwoong Bok, Thomas Schimmang\",\"doi\":\"10.1002/dvdy.747\",\"DOIUrl\":null,\"url\":null,\"abstract\":\"<p><strong>Background: </strong>The mechanisms underlying the formation of complex structures such as during the outgrowth of the cochlear duct are still poorly understood.</p><p><strong>Results: </strong>We have analyzed the morphological and molecular changes associated with cochlear development in mouse mutants for the transcription factor Meis2, which show defective coiling of the cochlea. These morphological abnormalities were accompanied by the formation of ectopic and extra rows of sensory hair cells. Gene profiling of otic vesicles from Meis2 mutants revealed a dysregulation of genes that are potentially involved in Sonic hedgehog (Shh)-mediated patterning of the cochlear duct. Like in Shh mutants, Meis2 defective mice showed a loss of genes that are expressed in the apical part of the cochlear duct.</p><p><strong>Conclusions: </strong>Taken together, these data reveal that the loss of Meis2 leads to a phenotype that resembles Shh mutants, suggesting that Meis2 is instrumental for cochlear Shh signaling. The modulation of the same subset of genes provides an interesting insight into which Shh responsive genes are essential for outgrowth and patterning of the cochlear duct.</p>\",\"PeriodicalId\":11247,\"journal\":{\"name\":\"Developmental Dynamics\",\"volume\":\" \",\"pages\":\"\"},\"PeriodicalIF\":2.0000,\"publicationDate\":\"2024-10-01\",\"publicationTypes\":\"Journal Article\",\"fieldsOfStudy\":null,\"isOpenAccess\":false,\"openAccessPdf\":\"\",\"citationCount\":\"0\",\"resultStr\":null,\"platform\":\"Semanticscholar\",\"paperid\":null,\"PeriodicalName\":\"Developmental Dynamics\",\"FirstCategoryId\":\"99\",\"ListUrlMain\":\"https://doi.org/10.1002/dvdy.747\",\"RegionNum\":3,\"RegionCategory\":\"生物学\",\"ArticlePicture\":[],\"TitleCN\":null,\"AbstractTextCN\":null,\"PMCID\":null,\"EPubDate\":\"\",\"PubModel\":\"\",\"JCR\":\"Q2\",\"JCRName\":\"ANATOMY & MORPHOLOGY\",\"Score\":null,\"Total\":0}","platform":"Semanticscholar","paperid":null,"PeriodicalName":"Developmental Dynamics","FirstCategoryId":"99","ListUrlMain":"https://doi.org/10.1002/dvdy.747","RegionNum":3,"RegionCategory":"生物学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":null,"EPubDate":"","PubModel":"","JCR":"Q2","JCRName":"ANATOMY & MORPHOLOGY","Score":null,"Total":0}
Analysis of Meis2 knockout mice reveals Sonic hedgehog-mediated patterning of the cochlear duct.
Background: The mechanisms underlying the formation of complex structures such as during the outgrowth of the cochlear duct are still poorly understood.
Results: We have analyzed the morphological and molecular changes associated with cochlear development in mouse mutants for the transcription factor Meis2, which show defective coiling of the cochlea. These morphological abnormalities were accompanied by the formation of ectopic and extra rows of sensory hair cells. Gene profiling of otic vesicles from Meis2 mutants revealed a dysregulation of genes that are potentially involved in Sonic hedgehog (Shh)-mediated patterning of the cochlear duct. Like in Shh mutants, Meis2 defective mice showed a loss of genes that are expressed in the apical part of the cochlear duct.
Conclusions: Taken together, these data reveal that the loss of Meis2 leads to a phenotype that resembles Shh mutants, suggesting that Meis2 is instrumental for cochlear Shh signaling. The modulation of the same subset of genes provides an interesting insight into which Shh responsive genes are essential for outgrowth and patterning of the cochlear duct.
期刊介绍：
Developmental Dynamics, is an official publication of the American Association for Anatomy. This peer reviewed journal provides an international forum for publishing novel discoveries, using any model system, that advances our understanding of development, morphology, form and function, evolution, disease, stem cells, repair and regeneration.
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