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Loss of functional FMRP leads to Fragile X syndrome (FXS), a neurodevelopmental disorder characterized by sensory dysfunction including abnormal auditory processing. While the central mechanisms of FMRP regulation have been studied in the brain, whether FMRP is expressed in the auditory periphery and how it develops and functions remains unknown. In this study, we characterized the spatiotemporal distribution pattern of FMRP immunoreactivity in the inner ear of mice, rats, gerbils, and chickens. Across species, FMRP was expressed in hair cells and supporting cells, with a particularly high level in immature hair cells during the prehearing period. Interestingly, the distribution of cytoplasmic FMRP displayed an age\u2010dependent translocation in hair cells, and this feature was conserved across species. In the auditory ganglion (AG), FMRP immunoreactivity was detected in neuronal cell bodies as well as their peripheral and central processes. Distinct from hair cells, FMRP intensity in AG neurons was high both during development and after maturation. Additionally, FMRP was evident in mature glial cells surrounding AG neurons. Together, these observations demonstrate distinct developmental trajectories across cell types in the auditory periphery. Given the importance of peripheral inputs to the maturation of auditory circuits, these findings implicate involvement of FMRP in inner ear development as well as a potential contribution of periphery FMRP to the generation of auditory dysfunction in FXS.<\/jats:p>","DOI":"10.1002\/cne.25420","type":"journal-article","created":{"date-parts":[[2022,10,12]],"date-time":"2022-10-12T13:19:47Z","timestamp":1665580787000},"page":"149-169","update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":7,"title":["Cellular distribution of the <b>Fragile X<\/b> mental retardation protein in the inner ear: <b>a<\/b> developmental and comparative study in the mouse, rat, gerbil, and chicken"],"prefix":"10.1002","volume":"531","author":[{"ORCID":"https:\/\/orcid.org\/0000-0003-2104-7089","authenticated-orcid":false,"given":"Xiaoyu","family":"Wang","sequence":"first","affiliation":[{"name":"Division of Histology and Embryology, Key Laboratory for Regenerative Medicine of the Ministry of Education, College of Medicine Jinan University  Guangzhou China"},{"name":"Program in Neuroscience, Department of Biomedical Sciences, College of Medicine Florida State University  Tallahassee Florida USA"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Qiwei","family":"Fan","sequence":"additional","affiliation":[{"name":"Division of Histology and Embryology, Key Laboratory for Regenerative Medicine of the Ministry of Education, College of Medicine Jinan University  Guangzhou China"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-4877-0114","authenticated-orcid":false,"given":"Xiaoyan","family":"Yu","sequence":"additional","affiliation":[{"name":"Program in Neuroscience, Department of Biomedical Sciences, College of Medicine Florida State University  Tallahassee Florida USA"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-4347-7063","authenticated-orcid":false,"given":"Yuan","family":"Wang","sequence":"additional","affiliation":[{"name":"Program in Neuroscience, Department of Biomedical Sciences, College of Medicine Florida State University  Tallahassee Florida USA"}],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"311","published-online":{"date-parts":[[2022,10,12]]},"reference":[{"key":"e_1_2_10_2_1","first-page":"46","article-title":"Auditory brain\u2010stem responses in the fragile X syndrome","volume":"43","author":"Arinami T.","year":"1988","journal-title":"American Journal of Human Genetics"},{"key":"e_1_2_10_3_1","doi-asserted-by":"publisher","DOI":"10.1172\/JCI63141"},{"key":"e_1_2_10_4_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.neuroscience.2014.05.006"},{"key":"e_1_2_10_5_1","doi-asserted-by":"publisher","DOI":"10.1371\/journal.pone.0058296"},{"key":"e_1_2_10_6_1","doi-asserted-by":"publisher","DOI":"10.1038\/nn.2563"},{"key":"e_1_2_10_7_1","doi-asserted-by":"publisher","DOI":"10.1002\/dvdy.23978"},{"key":"e_1_2_10_8_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.heares.2016.07.004"},{"key":"e_1_2_10_9_1","doi-asserted-by":"publisher","DOI":"10.1186\/s12915-020-00857-6"},{"key":"e_1_2_10_10_1","doi-asserted-by":"publisher","DOI":"10.1371\/journal.pone.0026120"},{"key":"e_1_2_10_11_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.cell.2011.06.013"},{"key":"e_1_2_10_12_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.tig.2017.07.008"},{"key":"e_1_2_10_13_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.neuron.2012.12.018"},{"key":"e_1_2_10_14_1","doi-asserted-by":"publisher","DOI":"10.1523\/JNEUROSCI.2021-11.2011"},{"key":"e_1_2_10_15_1","doi-asserted-by":"publisher","DOI":"10.1371\/journal.pgen.1003890"},{"key":"e_1_2_10_16_1","doi-asserted-by":"publisher","DOI":"10.1093\/hmg\/2.8.1147"},{"key":"e_1_2_10_17_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.brainres.2006.11.078"},{"key":"e_1_2_10_18_1","doi-asserted-by":"publisher","DOI":"10.1111\/j.1460-9568.2007.05401.x"},{"key":"e_1_2_10_19_1","doi-asserted-by":"publisher","DOI":"10.1523\/JNEUROSCI.0839-18.2019"},{"issue":"185","key":"e_1_2_10_20_1","first-page":"e64187","article-title":"Dissecting cell\u2010autonomous function of Fragile X mental retardation protein in an auditory circuit by in ovo electroporation","volume":"6","author":"Fan Q.","year":"2022","journal-title":"JoVE (Journal of Visualized Experiments)"},{"key":"e_1_2_10_21_1","first-page":"977","article-title":"Brain\u2010stem auditory evoked potentials in the fragile X syndrome","volume":"45","author":"Ferri R.","year":"1989","journal-title":"Am J Hum Genet"},{"key":"e_1_2_10_22_1","doi-asserted-by":"publisher","DOI":"10.1113\/JP270675"},{"key":"e_1_2_10_23_1","first-page":"351","article-title":"General pattern and morphological specializations of the avian cochlea","volume":"8","author":"Fischer F. 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