{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,5,14]],"date-time":"2026-05-14T18:15:46Z","timestamp":1778782546497,"version":"3.51.4"},"reference-count":26,"publisher":"Public Library of Science (PLoS)","issue":"5","license":[{"start":{"date-parts":[[2026,5,14]],"date-time":"2026-05-14T00:00:00Z","timestamp":1778716800000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/creativecommons.org\/licenses\/by\/4.0\/"}],"funder":[{"DOI":"10.13039\/100000053","name":"National Eye Institute","doi-asserted-by":"publisher","award":["R01EY025536"],"award-info":[{"award-number":["R01EY025536"]}],"id":[{"id":"10.13039\/100000053","id-type":"DOI","asserted-by":"publisher"}]}],"content-domain":{"domain":["www.plosone.org"],"crossmark-restriction":false},"short-container-title":["PLoS One"],"abstract":"<jats:p>\n                    In vertebrates, two genes,\n                    <jats:italic>Musashi1 (Msi1)<\/jats:italic>\n                    and\n                    <jats:italic>Musashi2 (Msi2)<\/jats:italic>\n                    , encode for highly similar\n                    <jats:italic>Musashi<\/jats:italic>\n                    protein paralogs. The\n                    <jats:italic>Musashi<\/jats:italic>\n                    proteins are known to bind to 3\u2019-UTRs and control translation. In photoreceptor cells, the\n                    <jats:italic>Musashi<\/jats:italic>\n                    proteins promote the inclusion of photoreceptor-specific alternative exons by binding to the proximal downstream of their introns. While the\n                    <jats:italic>Musashi<\/jats:italic>\n                    proteins are expressed in various cell types, their role in regulating splicing appears to be confined to photoreceptor cells, where the two proteins have exceptionally high expression levels. To test if the photoreceptor-specific role of MSI1 and MSI2 in splicing is due to their expression levels in photoreceptor cells, we generated combined\n                    <jats:italic>Msi1<\/jats:italic>\n                    and\n                    <jats:italic>Msi2<\/jats:italic>\n                    knockouts that progressively reduced the number of\n                    <jats:italic>Musashi<\/jats:italic>\n                    alleles in photoreceptor cells. We analyzed the splicing of photoreceptor-specific exons in the\n                    <jats:italic>Cc2d2a<\/jats:italic>\n                    ,\n                    <jats:italic>Cep290<\/jats:italic>\n                    ,\n                    <jats:italic>Prom1<\/jats:italic>\n                    , and\n                    <jats:italic>Ttc8<\/jats:italic>\n                    genes and the function of photoreceptor cells in the knockouts. We found that a single allele from either\n                    <jats:italic>Msi1<\/jats:italic>\n                    or\n                    <jats:italic>Msi2<\/jats:italic>\n                    is sufficient to maintain photoreceptor function and support high inclusion levels of the photoreceptor-specific exons.\n                  <\/jats:p>","DOI":"10.1371\/journal.pone.0338576","type":"journal-article","created":{"date-parts":[[2026,5,14]],"date-time":"2026-05-14T17:43:48Z","timestamp":1778780628000},"page":"e0338576","update-policy":"https:\/\/doi.org\/10.1371\/journal.pone.corrections_policy","source":"Crossref","is-referenced-by-count":0,"title":["A single Musashi gene allele is sufficient to maintain mouse photoreceptor cells"],"prefix":"10.1371","volume":"21","author":[{"given":"Bohye","family":"Jeong","sequence":"first","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-1108-7271","authenticated-orcid":true,"given":"Peter","family":"Stoilov","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"340","published-online":{"date-parts":[[2026,5,14]]},"reference":[{"issue":"2","key":"pone.0338576.ref001","doi-asserted-by":"crossref","first-page":"230","DOI":"10.1006\/dbio.1996.0130","article-title":"Mouse-Musashi-1, a neural RNA-binding protein highly enriched in the mammalian CNS stem cell","volume":"176","author":"S Sakakibara","year":"1996","journal-title":"Dev Biol"},{"issue":"1","key":"pone.0338576.ref002","doi-asserted-by":"crossref","first-page":"67","DOI":"10.1016\/0896-6273(94)90460-X","article-title":"Musashi, a neural RNA-binding protein required for Drosophila adult external sensory organ development","volume":"13","author":"M Nakamura","year":"1994","journal-title":"Neuron"},{"issue":"23","key":"pone.0338576.ref003","doi-asserted-by":"crossref","first-page":"15194","DOI":"10.1073\/pnas.232087499","article-title":"RNA-binding protein Musashi family: roles for CNS stem cells and a subpopulation of ependymal cells revealed by targeted disruption and antisense ablation","volume":"99","author":"S Sakakibara","year":"2002","journal-title":"Proc Natl Acad Sci U S A"},{"issue":"22","key":"pone.0338576.ref004","doi-asserted-by":"crossref","first-page":"8402","DOI":"10.1073\/pnas.0600906103","article-title":"The RNA-binding protein Musashi is required intrinsically to maintain stem cell identity","volume":"103","author":"NA Siddall","year":"2006","journal-title":"Proc Natl Acad Sci U S A"},{"issue":"11","key":"pone.0338576.ref005","doi-asserted-by":"crossref","DOI":"10.1371\/journal.pone.0049810","article-title":"Drosophila Rbp6 is an orthologue of vertebrate Msi-1 and Msi-2, but does not function redundantly with dMsi to regulate germline stem cell behaviour","volume":"7","author":"NA Siddall","year":"2012","journal-title":"PLoS One"},{"key":"pone.0338576.ref006","doi-asserted-by":"crossref","DOI":"10.7554\/eLife.03915","article-title":"Musashi proteins are post-transcriptional regulators of the epithelial-luminal cell state","volume":"3","author":"Y Katz","year":"2014","journal-title":"Elife"},{"key":"pone.0338576.ref007","doi-asserted-by":"crossref","first-page":"2792","DOI":"10.1038\/sj.emboj.7601159","article-title":"Musashi regulates the temporal order of mRNA translation during Xenopus oocyte maturation","volume":"25","author":"A Charlesworth","year":"2006","journal-title":"EMBO J"},{"issue":"3","key":"pone.0338576.ref008","doi-asserted-by":"crossref","first-page":"554","DOI":"10.1182\/blood-2010-12-322081","article-title":"Musashi 2 is a regulator of the HSC compartment identified by a retroviral insertion screen and knockout mice","volume":"118","author":"L de Andr\u00e9s-Aguayo","year":"2011","journal-title":"Blood"},{"issue":"3","key":"pone.0338576.ref009","doi-asserted-by":"crossref","first-page":"318","DOI":"10.1002\/glia.20615","article-title":"Musashi1 RNA-binding protein regulates oligodendrocyte lineage cell differentiation and survival","volume":"56","author":"NR Dobson","year":"2008","journal-title":"Glia"},{"key":"pone.0338576.ref010","doi-asserted-by":"crossref","first-page":"15","DOI":"10.1186\/s12861-015-0064-y","article-title":"Neural stem and progenitor cell fate transition requires regulation of Musashi1 function","volume":"15","author":"AM MacNicol","year":"2015","journal-title":"BMC Dev Biol"},{"issue":"12","key":"pone.0338576.ref011","doi-asserted-by":"crossref","first-page":"3888","DOI":"10.1128\/MCB.21.12.3888-3900.2001","article-title":"The neural RNA-binding protein Musashi1 translationally regulates mammalian numb gene expression by interacting with its mRNA","volume":"21","author":"T Imai","year":"2001","journal-title":"Mol Cell Biol"},{"key":"pone.0338576.ref012","doi-asserted-by":"crossref","first-page":"1355","DOI":"10.1242\/jcs.115.7.1355","article-title":"Musashi: a translational regulator of cell fate","volume":"115","author":"H Okano","year":"2002","journal-title":"J Cell Sci"},{"issue":"6833","key":"pone.0338576.ref013","doi-asserted-by":"crossref","first-page":"94","DOI":"10.1038\/35075094","article-title":"Translational repression determines a neuronal potential in Drosophila asymmetric cell division","volume":"411","author":"M Okabe","year":"2001","journal-title":"Nature"},{"issue":"1","key":"pone.0338576.ref014","doi-asserted-by":"crossref","first-page":"12363","DOI":"10.1038\/s41598-025-97188-9","article-title":"Musashi-dependent mRNA translational activation is mediated through association with the Scd6\/Like-sm family member, LSM14B","volume":"15","author":"K Bronson","year":"2025","journal-title":"Sci Rep"},{"issue":"28","key":"pone.0338576.ref015","doi-asserted-by":"crossref","first-page":"10969","DOI":"10.1074\/jbc.RA119.007220","article-title":"Musashi interaction with poly(A)-binding protein is required for activation of target mRNA translation","volume":"294","author":"CE Cragle","year":"2019","journal-title":"J Biol Chem"},{"key":"pone.0338576.ref016","doi-asserted-by":"crossref","first-page":"14239","DOI":"10.1074\/jbc.M114.548271","article-title":"Musashi protein-directed translational activation of target mRNAs is mediated by the poly(A) polymerase, germ line development defective-2","volume":"289","author":"C Cragle","year":"2014","journal-title":"J Biol Chem"},{"issue":"4","key":"pone.0338576.ref017","doi-asserted-by":"crossref","first-page":"639","DOI":"10.1083\/jcb.200708004","article-title":"Neural RNA-binding protein Musashi1 inhibits translation initiation by competing with eIF4G for PABP","volume":"181","author":"H Kawahara","year":"2008","journal-title":"J Cell Biol"},{"key":"pone.0338576.ref018","article-title":"Musashi exerts control of gonadotrope target mRNA translation during the mouse estrous cycle","volume":"164","author":"ARS Moreira","year":"2023","journal-title":"Endocrinol"},{"issue":"1","key":"pone.0338576.ref019","doi-asserted-by":"crossref","first-page":"1011","DOI":"10.1038\/s42003-022-03990-w","article-title":"The Musashi proteins direct post-transcriptional control of protein expression and alternate exon splicing in vertebrate photoreceptors","volume":"5","author":"F Matalkah","year":"2022","journal-title":"Commun Biol"},{"key":"pone.0338576.ref020","doi-asserted-by":"crossref","first-page":"100048","DOI":"10.1074\/jbc.RA120.015714","article-title":"The Musashi proteins MSI1 and MSI2 are required for photoreceptor morphogenesis and vision in mice","volume":"296","author":"J Sundar","year":"2021","journal-title":"J Biol Chem"},{"issue":"8","key":"pone.0338576.ref021","doi-asserted-by":"crossref","DOI":"10.1371\/journal.pgen.1006256","article-title":"The Musashi 1 Controls the Splicing of Photoreceptor-Specific Exons in the Vertebrate Retina","volume":"12","author":"D Murphy","year":"2016","journal-title":"PLoS Genet"},{"issue":"1","key":"pone.0338576.ref022","doi-asserted-by":"crossref","first-page":"201","DOI":"10.7150\/thno.35895","article-title":"Musashi-1 promotes stress-induced tumor progression through recruitment of AGO2","volume":"10","author":"H-Y Chen","year":"2020","journal-title":"Theranostics"},{"issue":"11","key":"pone.0338576.ref023","doi-asserted-by":"crossref","first-page":"2440","DOI":"10.1016\/j.celrep.2015.11.022","article-title":"The Msi Family of RNA-Binding Proteins Function Redundantly as Intestinal Oncoproteins","volume":"13","author":"N Li","year":"2015","journal-title":"Cell Rep"},{"issue":"1","key":"pone.0338576.ref024","doi-asserted-by":"crossref","first-page":"71","DOI":"10.1084\/jem.20130736","article-title":"Musashi-2 controls cell fate, lineage bias, and TGF-\u03b2 signaling in HSCs","volume":"211","author":"S-M Park","year":"2014","journal-title":"J Exp Med"},{"issue":"3","key":"pone.0338576.ref025","doi-asserted-by":"crossref","DOI":"10.1371\/journal.pone.0120415","article-title":"M\u00fcller glia activation in response to inherited retinal degeneration is highly varied and disease-specific","volume":"10","author":"C Hippert","year":"2015","journal-title":"PLoS One"},{"issue":"1","key":"pone.0338576.ref026","doi-asserted-by":"crossref","first-page":"1","DOI":"10.1159\/000328979","article-title":"M\u00fcller glial cells in retinal disease","volume":"227","author":"A Bringmann","year":"2012","journal-title":"Ophthalmologica"}],"container-title":["PLOS One"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/dx.plos.org\/10.1371\/journal.pone.0338576","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2026,5,14]],"date-time":"2026-05-14T17:43:54Z","timestamp":1778780634000},"score":1,"resource":{"primary":{"URL":"https:\/\/dx.plos.org\/10.1371\/journal.pone.0338576"}},"subtitle":[],"editor":[{"given":"Gerrit","family":"Hilgen","sequence":"first","affiliation":[],"role":[{"role":"editor","vocabulary":"crossref"}]}],"short-title":[],"issued":{"date-parts":[[2026,5,14]]},"references-count":26,"journal-issue":{"issue":"5","published-online":{"date-parts":[[2026,5,14]]}},"URL":"https:\/\/doi.org\/10.1371\/journal.pone.0338576","relation":{},"ISSN":["1932-6203"],"issn-type":[{"value":"1932-6203","type":"electronic"}],"subject":[],"published":{"date-parts":[[2026,5,14]]}}}