{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,5,26]],"date-time":"2026-05-26T10:02:15Z","timestamp":1779789735258,"version":"3.53.1"},"reference-count":61,"publisher":"Wiley","issue":"1","license":[{"start":{"date-parts":[[2025,9,29]],"date-time":"2025-09-29T00:00:00Z","timestamp":1759104000000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/onlinelibrary.wiley.com\/termsAndConditions#vor"},{"start":{"date-parts":[[2025,9,29]],"date-time":"2025-09-29T00:00:00Z","timestamp":1759104000000},"content-version":"tdm","delay-in-days":0,"URL":"http:\/\/doi.wiley.com\/10.1002\/tdm_license_1.1"}],"funder":[{"DOI":"10.13039\/100012542","name":"Sichuan Provincial Science and Technology Support Program","doi-asserted-by":"publisher","award":["2024NSFSC0647"],"award-info":[{"award-number":["2024NSFSC0647"]}],"id":[{"id":"10.13039\/100012542","id-type":"DOI","asserted-by":"publisher"}]}],"content-domain":{"domain":["onlinelibrary.wiley.com"],"crossmark-restriction":true},"short-container-title":["Clinical Genetics"],"published-print":{"date-parts":[[2026,1]]},"abstract":"<jats:title>ABSTRACT<\/jats:title>\n                  <jats:p>The nexin\u2013dynein regulatory complex (N\u2010DRC) is a large protein complex composed of at least 11 subunits (DRC1\u2013DRC11) and plays a crucial role in ciliary and flagellar motility. It links adjacent doublets of microtubules (DMTs) between A and B microtubules, regulating dynein motor activity. Genetic defects in N\u2010DRC subunits lead to primary ciliary dyskinesia (PCD) and abnormal flagellar motility. In recent years, an increasing number of genetic mutations in N\u2010DRC subunits have been reported, associated with male infertility, characterized by multiple morphological abnormalities of the flagella (MMAF) and asthenozoospermia. Therefore, genetic diagnosis of N\u2010DRC defects in male infertility is of significant clinical importance, impacting the reproductive health of patients and the well\u2010being of their offspring. In this review, we summarize the gene mutations of N\u2010DRC subunits reported in the literature concerning male infertility, analyze the phenotypes of affected patients, and outline the functions and mechanisms of N\u2010DRC in sperm flagellar motility. Furthermore, we provide an overview of gene knockout (KO) mouse models of N\u2010DRC and their associated phenotypes. Finally, we summarize the outcomes of assisted reproductive technology (ART) in both patients and KO mice, offering references for the diagnosis and treatment of clinical male infertility caused by N\u2010DRC genetic factors.<\/jats:p>","DOI":"10.1111\/cge.70081","type":"journal-article","created":{"date-parts":[[2025,9,30]],"date-time":"2025-09-30T01:40:23Z","timestamp":1759196423000},"page":"28-39","update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":1,"title":["The Genetic Defects of N\u2010\n                    <scp>DRC<\/scp>\n                    in Male Infertility"],"prefix":"10.1111","volume":"109","author":[{"ORCID":"https:\/\/orcid.org\/0000-0001-9326-6091","authenticated-orcid":false,"given":"Jiao","family":"Qin","sequence":"first","affiliation":[{"name":"Department of Andrology\/Sichuan Human Sperm Bank, West China Second University Hospital Sichuan University  Chengdu China"},{"name":"Key Laboratory of Birth Defects and Related Diseases of Women and Children (Sichuan University), Ministry of Education  Chengdu China"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Jinyu","family":"Wang","sequence":"additional","affiliation":[{"name":"Department of Medical Genetics West China Second University Hospital of Sichuan University  Chengdu China"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Dingming","family":"Li","sequence":"additional","affiliation":[{"name":"Department of Andrology\/Sichuan Human Sperm Bank, West China Second University Hospital Sichuan University  Chengdu China"},{"name":"Key Laboratory of Birth Defects and Related Diseases of Women and Children (Sichuan University), Ministry of Education  Chengdu China"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-5183-4214","authenticated-orcid":false,"given":"Fuping","family":"Li","sequence":"additional","affiliation":[{"name":"Department of Andrology\/Sichuan Human Sperm Bank, West China Second University Hospital Sichuan University  Chengdu China"},{"name":"Key Laboratory of Birth Defects and Related Diseases of Women and Children (Sichuan University), Ministry of Education  Chengdu China"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"311","published-online":{"date-parts":[[2025,9,29]]},"reference":[{"key":"e_1_2_11_2_1","doi-asserted-by":"publisher","DOI":"10.1515\/medgen-2024-2040"},{"key":"e_1_2_11_3_1","doi-asserted-by":"publisher","DOI":"10.1210\/jc.79.6.1525"},{"key":"e_1_2_11_4_1","doi-asserted-by":"publisher","DOI":"10.1038\/s41585-018-0003-3"},{"key":"e_1_2_11_5_1","doi-asserted-by":"publisher","DOI":"10.21037\/tau-20-1434"},{"key":"e_1_2_11_6_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.rbmo.2021.01.011"},{"key":"e_1_2_11_7_1","doi-asserted-by":"publisher","DOI":"10.3389\/fendo.2023.1122004"},{"key":"e_1_2_11_8_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.ajhg.2017.04.012"},{"key":"e_1_2_11_9_1","doi-asserted-by":"publisher","DOI":"10.1111\/cge.13644"},{"key":"e_1_2_11_10_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.gene.2017.08.033"},{"key":"e_1_2_11_11_1","doi-asserted-by":"publisher","DOI":"10.1111\/cge.14197"},{"key":"e_1_2_11_12_1","doi-asserted-by":"publisher","DOI":"10.1136\/jmedgenet-2019-106011"},{"key":"e_1_2_11_13_1","doi-asserted-by":"publisher","DOI":"10.1196\/annals.1389.017"},{"key":"e_1_2_11_14_1","doi-asserted-by":"publisher","DOI":"10.1126\/science.1128618"},{"key":"e_1_2_11_15_1","doi-asserted-by":"publisher","DOI":"10.1091\/mbc.e12-11-0801"},{"key":"e_1_2_11_16_1","doi-asserted-by":"publisher","DOI":"10.1038\/s41467-023-41480-7"},{"key":"e_1_2_11_17_1","doi-asserted-by":"publisher","DOI":"10.1091\/mbc.E12-05-0357"},{"key":"e_1_2_11_18_1","doi-asserted-by":"publisher","DOI":"10.1183\/09031936.00052014"},{"key":"e_1_2_11_19_1","doi-asserted-by":"publisher","DOI":"10.1093\/humrep\/dew262"},{"key":"e_1_2_11_20_1","doi-asserted-by":"publisher","DOI":"10.1038\/nature07520"},{"key":"e_1_2_11_21_1","doi-asserted-by":"publisher","DOI":"10.1091\/mbc.E14-09-1367"},{"key":"e_1_2_11_22_1","doi-asserted-by":"publisher","DOI":"10.1074\/jbc.M114.626556"},{"key":"e_1_2_11_23_1","doi-asserted-by":"publisher","DOI":"10.1038\/s41586-023-06140-2"},{"key":"e_1_2_11_24_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.advms.2019.10.003"},{"key":"e_1_2_11_25_1","doi-asserted-by":"publisher","DOI":"10.3390\/cells8080900"},{"key":"e_1_2_11_26_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.ajhg.2015.08.012"},{"key":"e_1_2_11_27_1","doi-asserted-by":"publisher","DOI":"10.1038\/s10038-021-00985-z"},{"key":"e_1_2_11_28_1","doi-asserted-by":"publisher","DOI":"10.1111\/cge.14459"},{"key":"e_1_2_11_29_1","doi-asserted-by":"publisher","DOI":"10.3389\/fgene.2022.940292"},{"key":"e_1_2_11_30_1","doi-asserted-by":"publisher","DOI":"10.1093\/humrep\/deab099"},{"key":"e_1_2_11_31_1","doi-asserted-by":"publisher","DOI":"10.1093\/hmg\/ddab171"},{"key":"e_1_2_11_32_1","doi-asserted-by":"publisher","DOI":"10.1038\/ng.2533"},{"key":"e_1_2_11_33_1","doi-asserted-by":"publisher","DOI":"10.1038\/s10038-023-01122-8"},{"key":"e_1_2_11_34_1","doi-asserted-by":"publisher","DOI":"10.1111\/andr.13837"},{"key":"e_1_2_11_35_1","doi-asserted-by":"publisher","DOI":"10.1007\/s10815-023-02755-6"},{"key":"e_1_2_11_36_1","doi-asserted-by":"publisher","DOI":"10.7150\/ijbs.69332"},{"key":"e_1_2_11_37_1","doi-asserted-by":"publisher","DOI":"10.1016\/S0065-3233(05)70003-6"},{"key":"e_1_2_11_38_1","doi-asserted-by":"publisher","DOI":"10.1128\/aem.57.3.804-811.1991"},{"key":"e_1_2_11_39_1","doi-asserted-by":"publisher","DOI":"10.1038\/s41392-022-01293-4"},{"key":"e_1_2_11_40_1","doi-asserted-by":"publisher","DOI":"10.1038\/s10038-024-01253-6"},{"key":"e_1_2_11_41_1","doi-asserted-by":"publisher","DOI":"10.1083\/jcb.200303019"},{"key":"e_1_2_11_42_1","doi-asserted-by":"publisher","DOI":"10.1002\/humu.23005"},{"key":"e_1_2_11_43_1","doi-asserted-by":"publisher","DOI":"10.1002\/cm.20196"},{"key":"e_1_2_11_44_1","doi-asserted-by":"publisher","DOI":"10.1074\/jbc.M111.234666"},{"key":"e_1_2_11_45_1","doi-asserted-by":"publisher","DOI":"10.1111\/cge.14450"},{"key":"e_1_2_11_46_1","doi-asserted-by":"publisher","DOI":"10.1073\/pnas.1608458113"},{"key":"e_1_2_11_47_1","doi-asserted-by":"publisher","DOI":"10.1016\/S0959-440X(01)00266-4"},{"key":"e_1_2_11_48_1","doi-asserted-by":"publisher","DOI":"10.1038\/s41431-022-01095-w"},{"key":"e_1_2_11_49_1","first-page":"E5370","article-title":"TCTE1 Is a Conserved Component of the Dynein Regulatory Complex and Is Required for Motility and Metabolism in Mouse Spermatozoa","volume":"114","author":"Castaneda J. M.","year":"2017","journal-title":"Proceedings of the National Academy of Sciences of the United States of America"},{"key":"e_1_2_11_50_1","doi-asserted-by":"publisher","DOI":"10.1091\/mbc.e10-04-0331"},{"key":"e_1_2_11_51_1","doi-asserted-by":"publisher","DOI":"10.1002\/pmic.201200350"},{"key":"e_1_2_11_52_1","doi-asserted-by":"publisher","DOI":"10.3390\/ijms242015363"},{"key":"e_1_2_11_53_1","doi-asserted-by":"publisher","DOI":"10.1038\/s41419-025-07506-2"},{"key":"e_1_2_11_54_1","doi-asserted-by":"publisher","DOI":"10.1371\/journal.pgen.1006220"},{"key":"e_1_2_11_55_1","doi-asserted-by":"publisher","DOI":"10.1371\/journal.pgen.1008585"},{"key":"e_1_2_11_56_1","doi-asserted-by":"publisher","DOI":"10.1371\/journal.pone.0098053"},{"key":"e_1_2_11_57_1","doi-asserted-by":"publisher","DOI":"10.1038\/ncomms4811"},{"key":"e_1_2_11_58_1","first-page":"304","article-title":"Expression of IQCG in the Human Testis and Its Correlation With Asthenospermia","volume":"24","author":"Zhu Y.","year":"2018","journal-title":"Zhonghua Nan Ke Xue"},{"key":"e_1_2_11_59_1","doi-asserted-by":"publisher","DOI":"10.1534\/g3.113.009563"},{"key":"e_1_2_11_60_1","doi-asserted-by":"publisher","DOI":"10.3390\/jcm12020671"},{"key":"e_1_2_11_61_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.theriogenology.2022.10.014"},{"key":"e_1_2_11_62_1","doi-asserted-by":"publisher","DOI":"10.1007\/s10815-019-01509-7"}],"container-title":["Clinical Genetics"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/pdf\/10.1111\/cge.70081","content-type":"application\/pdf","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/full-xml\/10.1111\/cge.70081","content-type":"application\/xml","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/pdf\/10.1111\/cge.70081","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2026,5,26]],"date-time":"2026-05-26T09:03:06Z","timestamp":1779786186000},"score":1,"resource":{"primary":{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/10.1111\/cge.70081"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2025,9,29]]},"references-count":61,"journal-issue":{"issue":"1","published-print":{"date-parts":[[2026,1]]}},"alternative-id":["10.1111\/cge.70081"],"URL":"https:\/\/doi.org\/10.1111\/cge.70081","archive":["Portico"],"relation":{"has-review":[{"id-type":"doi","id":"10.1111\/CGE.70081\/v1\/review2","asserted-by":"object"},{"id-type":"doi","id":"10.1111\/CGE.70081\/v1\/review1","asserted-by":"object"},{"id-type":"doi","id":"10.1111\/CGE.70081\/v1\/decision1","asserted-by":"object"}]},"ISSN":["0009-9163","1399-0004"],"issn-type":[{"value":"0009-9163","type":"print"},{"value":"1399-0004","type":"electronic"}],"subject":[],"published":{"date-parts":[[2025,9,29]]},"assertion":[{"value":"2025-07-31","order":0,"name":"received","label":"Received","group":{"name":"publication_history","label":"Publication History"}},{"value":"2025-09-19","order":2,"name":"accepted","label":"Accepted","group":{"name":"publication_history","label":"Publication History"}},{"value":"2025-09-29","order":3,"name":"published","label":"Published","group":{"name":"publication_history","label":"Publication History"}}]}}