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TSN accompanies specific mRNA transport in male germ cells through intercellular bridges. A testis\u2010expressed protein TSNAXIP1 was reported to interact with TSNAX. However the role of TSNAXIP1 in spermatogenesis remained unclear. This study aimed to elucidate the role of TSNAXIP1 in spermatogenesis and male fertility in mice.<\/jats:p><\/jats:sec><jats:sec><jats:title>Methods<\/jats:title><jats:p>TSNAXIP1 knockout (KO) mice were generated using the CRISPR\u2010Cas9 system. The fertility, spermatogenesis, and sperm of TSNAXIP1 KO males were analyzed.<\/jats:p><\/jats:sec><jats:sec><jats:title>Results<\/jats:title><jats:p>TSNAXIP1, and especially its domains, are highly conserved between mouse and human. <jats:italic>Tsnaxip1<\/jats:italic> was expressed in testis, but not in ovary. TSNAXIP1 KO mice were generated, and TSNAXIP1 KO males were found to be sub\u2010fertile with smaller testis and lower sperm count. Although no overt abnormalities were observed during spermatogenesis, lack of TSNAXIP1 induced sperm head malformation, resulting in a unique flower\u2010shaped sperm head. Moreover, abnormal anchorage of the sperm neck was frequently observed in TSNAXIP1 null sperm.<\/jats:p><\/jats:sec><jats:sec><jats:title>Conclusion<\/jats:title><jats:p>A testis\u2010expressed gene TSNAXIP1 has important roles in sperm head morphogenesis and male fertility. Moreover, TSNAXIP1 could be a causative gene for human infertility.<\/jats:p><\/jats:sec>","DOI":"10.1002\/rmb2.12520","type":"journal-article","created":{"date-parts":[[2023,6,28]],"date-time":"2023-06-28T13:05:52Z","timestamp":1687957552000},"update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":4,"title":["<scp>TSNAXIP1<\/scp> is required for sperm head formation and male fertility"],"prefix":"10.1002","volume":"22","author":[{"given":"Tasrin","family":"Sultana","sequence":"first","affiliation":[{"name":"Laboratory of Zoology, Graduate School of Bioresource and Bioenvironmental Sciences Kyushu University  Fukuoka Japan"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Tokuko","family":"Iwamori","sequence":"additional","affiliation":[{"name":"Laboratory of Zoology, Graduate School of Agriculture Kyushu University  Fukuoka Japan"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-5903-8386","authenticated-orcid":false,"given":"Naoki","family":"Iwamori","sequence":"additional","affiliation":[{"name":"Laboratory of Zoology, Graduate School of Bioresource and Bioenvironmental Sciences Kyushu University  Fukuoka Japan"},{"name":"Laboratory of Zoology, Graduate School of Agriculture Kyushu University  Fukuoka Japan"}],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"311","published-online":{"date-parts":[[2023,6,28]]},"reference":[{"key":"e_1_2_8_2_1","doi-asserted-by":"publisher","DOI":"10.1111\/and.13615"},{"key":"e_1_2_8_3_1","doi-asserted-by":"publisher","DOI":"10.1095\/biolreprod.103.026880"},{"key":"e_1_2_8_4_1","doi-asserted-by":"publisher","DOI":"10.4161\/21565562.2014.979623"},{"key":"e_1_2_8_5_1","doi-asserted-by":"publisher","DOI":"10.1021\/acs.jproteome.9b00351"},{"key":"e_1_2_8_6_1","doi-asserted-by":"publisher","DOI":"10.1006\/geno.2002.6779"},{"key":"e_1_2_8_7_1","doi-asserted-by":"publisher","DOI":"10.1002\/mrd.10035"},{"key":"e_1_2_8_8_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.devcel.2007.03.018"},{"key":"e_1_2_8_9_1","doi-asserted-by":"publisher","DOI":"10.1096\/fj.202100960R"},{"key":"e_1_2_8_10_1","doi-asserted-by":"publisher","DOI":"10.1016\/S0014-5793(02)03095-8"},{"key":"e_1_2_8_11_1","doi-asserted-by":"publisher","DOI":"10.1006\/dbio.2002.0679"},{"key":"e_1_2_8_12_1","doi-asserted-by":"publisher","DOI":"10.1128\/MCB.23.18.6419-6434.2003"},{"issue":"8","key":"e_1_2_8_13_1","first-page":"999","article-title":"Improved methodology for the detection and quantification of the acrosome reaction in mouse spermatozoa","volume":"24","author":"Lybaert P","year":"2009","journal-title":"Histol Histopathol"},{"key":"e_1_2_8_14_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.gene.2018.03.059"},{"key":"e_1_2_8_15_1","doi-asserted-by":"publisher","DOI":"10.1146\/annurev.biochem.74.082803.133219"},{"key":"e_1_2_8_16_1","doi-asserted-by":"publisher","DOI":"10.1002\/cm.21338"},{"key":"e_1_2_8_17_1","doi-asserted-by":"publisher","DOI":"10.1111\/andr.13378"},{"key":"e_1_2_8_18_1","doi-asserted-by":"publisher","DOI":"10.1111\/nyas.13690"},{"key":"e_1_2_8_19_1","doi-asserted-by":"publisher","DOI":"10.1242\/jcs.115.1.207"},{"key":"e_1_2_8_20_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.celrep.2014.10.021"}],"container-title":["Reproductive Medicine and Biology"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/pdf\/10.1002\/rmb2.12520","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2024,2,22]],"date-time":"2024-02-22T17:26:03Z","timestamp":1708622763000},"score":1,"resource":{"primary":{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/10.1002\/rmb2.12520"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2023,1]]},"references-count":19,"journal-issue":{"issue":"1","published-print":{"date-parts":[[2023,1]]}},"alternative-id":["10.1002\/rmb2.12520"],"URL":"https:\/\/doi.org\/10.1002\/rmb2.12520","archive":["Portico"],"relation":{},"ISSN":["1445-5781","1447-0578"],"issn-type":[{"value":"1445-5781","type":"print"},{"value":"1447-0578","type":"electronic"}],"subject":[],"published":{"date-parts":[[2023,1]]},"assertion":[{"value":"2023-01-20","order":0,"name":"received","label":"Received","group":{"name":"publication_history","label":"Publication History"}},{"value":"2023-05-21","order":1,"name":"accepted","label":"Accepted","group":{"name":"publication_history","label":"Publication History"}},{"value":"2023-06-28","order":2,"name":"published","label":"Published","group":{"name":"publication_history","label":"Publication History"}}],"article-number":"e12520"}}