{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2025,9,9]],"date-time":"2025-09-09T22:03:17Z","timestamp":1757455397744,"version":"3.41.2"},"reference-count":45,"publisher":"Wiley","issue":"9","license":[{"start":{"date-parts":[[2021,8,17]],"date-time":"2021-08-17T00:00:00Z","timestamp":1629158400000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/creativecommons.org\/licenses\/by-nc-nd\/4.0\/"}],"funder":[{"DOI":"10.13039\/501100004826","name":"Natural Science Foundation of Beijing Municipality","doi-asserted-by":"publisher","award":["7172176"],"award-info":[{"award-number":["7172176"]}],"id":[{"id":"10.13039\/501100004826","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/501100001809","name":"National Natural Science Foundation of China","doi-asserted-by":"publisher","award":["81470698"],"award-info":[{"award-number":["81470698"]}],"id":[{"id":"10.13039\/501100001809","id-type":"DOI","asserted-by":"publisher"}]}],"content-domain":{"domain":["onlinelibrary.wiley.com"],"crossmark-restriction":true},"short-container-title":["Genesis"],"published-print":{"date-parts":[[2021,9]]},"abstract":"<jats:title>Summary<\/jats:title><jats:p><jats:italic>Atoh8<\/jats:italic>, also named <jats:italic>Math6<\/jats:italic>, is a bHLH gene reported to have important functions in the developing nervous system, pancreas and kidney. However, the expression pattern and function of Atoh8 in the inner ear are still unclear. To study the function of Atoh8 in the developing mouse inner ear, we performed targeted deletion of <jats:italic>Atoh8<\/jats:italic> by intercrossing <jats:italic>Atoh8<\/jats:italic><jats:sup><jats:italic>lacZ\/+<\/jats:italic><\/jats:sup> mice. We studied the expression pattern of <jats:italic>Atoh8<\/jats:italic> in the inner ear and found interesting results that <jats:italic>Atoh8<\/jats:italic>\u2010null (<jats:italic>Atoh8<\/jats:italic><jats:sup><jats:italic>lacZ\/lacZ<\/jats:italic><\/jats:sup>) mice were viable but smaller than their littermates and they were severely hearing impaired, which was confirmed by hearing tests (ABR, DPOAE). We collected 129 viable newborns from 18 litters by crossing <jats:italic>Atoh8<\/jats:italic><jats:sup>lacZ\/+<\/jats:sup> mice and found that the distributions of <jats:italic>Atoh8<\/jats:italic><jats:sup>lacZ\/+<\/jats:sup>, <jats:italic>Atoh8<\/jats:italic><jats:sup>lacZ\/lacZ<\/jats:sup> and wild type were very close to their expected Mendelian ratio by \u03c7<jats:sup>2<\/jats:sup> testing. However, no remarkable morphological changes in cochleae in mutant mice were detected under plastic sectioning and electron microscopy. No remarkable differences in the expression of Myosin6, Prestin, TrkC, GAD65, Tuj1, or Calretinin were detected between the mutant mice and the control mice. These findings indicate that <jats:italic>Atoh8<\/jats:italic> plays an important role in the development of normal hearing, while further studies are required to elucidate its exact function in hearing.<\/jats:p>","DOI":"10.1002\/dvg.23442","type":"journal-article","created":{"date-parts":[[2021,8,17]],"date-time":"2021-08-17T10:35:23Z","timestamp":1629196523000},"update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":5,"title":["Targeted deletion of <i>Atoh8<\/i> results in severe hearing loss in mice"],"prefix":"10.1002","volume":"59","author":[{"ORCID":"https:\/\/orcid.org\/0000-0002-0959-1295","authenticated-orcid":false,"given":"Qi","family":"Tang","sequence":"first","affiliation":[{"name":"Department of Otolaryngology, Peking Union Medical College Hospital Chinese Academy of Medical Sciences and Peking Union Medical College  Beijing China"},{"name":"Translational Medicine Center, Peking Union Medical College Hospital Chinese Academy of Medical Sciences and Peking Union Medical College  Beijing China"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Meng\u2010Yao","family":"Xie","sequence":"additional","affiliation":[{"name":"Department of Otolaryngology, Peking Union Medical College Hospital Chinese Academy of Medical Sciences and Peking Union Medical College  Beijing China"},{"name":"Translational Medicine Center, Peking Union Medical College Hospital Chinese Academy of Medical Sciences and Peking Union Medical College  Beijing China"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Yong\u2010Li","family":"Zhang","sequence":"additional","affiliation":[{"name":"Department of Otolaryngology, Peking Union Medical College Hospital Chinese Academy of Medical Sciences and Peking Union Medical College  Beijing China"},{"name":"Translational Medicine Center, Peking Union Medical College Hospital Chinese Academy of Medical Sciences and Peking Union Medical College  Beijing China"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Ruo\u2010Yan","family":"Xue","sequence":"additional","affiliation":[{"name":"Department of Otolaryngology, Peking Union Medical College Hospital Chinese Academy of Medical Sciences and Peking Union Medical College  Beijing China"},{"name":"Translational Medicine Center, Peking Union Medical College Hospital Chinese Academy of Medical Sciences and Peking Union Medical College  Beijing China"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Xiao\u2010Hui","family":"Zhu","sequence":"additional","affiliation":[{"name":"Department of Otolaryngology, Peking Union Medical College Hospital Chinese Academy of Medical Sciences and Peking Union Medical College  Beijing China"},{"name":"Translational Medicine Center, Peking Union Medical College Hospital Chinese Academy of Medical Sciences and Peking Union Medical College  Beijing China"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Hua","family":"Yang","sequence":"additional","affiliation":[{"name":"Department of Otolaryngology, Peking Union Medical College Hospital Chinese Academy of Medical Sciences and Peking Union Medical College  Beijing China"},{"name":"Translational Medicine Center, Peking Union Medical College Hospital Chinese Academy of Medical Sciences and Peking Union Medical College  Beijing China"}],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"311","published-online":{"date-parts":[[2021,8,17]]},"reference":[{"key":"e_1_2_9_2_1","doi-asserted-by":"publisher","DOI":"10.1242\/dev.00881"},{"key":"e_1_2_9_3_1","doi-asserted-by":"publisher","DOI":"10.1126\/science.284.5421.1837"},{"key":"e_1_2_9_4_1","doi-asserted-by":"publisher","DOI":"10.1007\/s00418-015-1368-5"},{"key":"e_1_2_9_5_1","doi-asserted-by":"publisher","DOI":"10.1371\/journal.pone.0023005"},{"key":"e_1_2_9_6_1","doi-asserted-by":"publisher","DOI":"10.1242\/dev.129.10.2495"},{"key":"e_1_2_9_7_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.neuron.2008.02.028"},{"key":"e_1_2_9_8_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.bbagrm.2013.08.003"},{"key":"e_1_2_9_9_1","doi-asserted-by":"publisher","DOI":"10.1371\/journal.pone.0146273"},{"key":"e_1_2_9_10_1","doi-asserted-by":"publisher","DOI":"10.1523\/JNEUROSCI.3951-07.2007"},{"key":"e_1_2_9_11_1","doi-asserted-by":"publisher","DOI":"10.1186\/1756-6606-3-36"},{"key":"e_1_2_9_12_1","doi-asserted-by":"publisher","DOI":"10.1121\/1.2401226"},{"key":"e_1_2_9_13_1","doi-asserted-by":"publisher","DOI":"10.1002\/dvdy.20370"},{"key":"e_1_2_9_14_1","doi-asserted-by":"publisher","DOI":"10.1006\/dbio.1999.9280"},{"key":"e_1_2_9_15_1","doi-asserted-by":"publisher","DOI":"10.1073\/pnas.93.9.3920"},{"key":"e_1_2_9_16_1","doi-asserted-by":"publisher","DOI":"10.1046\/j.1365-2443.2001.00476.x"},{"key":"e_1_2_9_17_1","doi-asserted-by":"publisher","DOI":"10.1371\/journal.pone.0011661"},{"key":"e_1_2_9_18_1","doi-asserted-by":"publisher","DOI":"10.1038\/nature03487"},{"key":"e_1_2_9_19_1","doi-asserted-by":"publisher","DOI":"10.1242\/dev.128.3.417"},{"issue":"125","key":"e_1_2_9_20_1","first-page":"77s","article-title":"Assessment 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