{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,9,1]],"date-time":"2026-09-01T03:40:33Z","timestamp":1788234033257,"version":"build-2803163510"},"reference-count":47,"publisher":"Oxford University Press (OUP)","issue":"9","license":[{"start":{"date-parts":[[2025,7,1]],"date-time":"2025-07-01T00:00:00Z","timestamp":1751328000000},"content-version":"vor","delay-in-days":0,"URL":"https:\/\/creativecommons.org\/licenses\/by-nc\/4.0\/"}],"funder":[{"DOI":"10.13039\/501100000925","name":"National Health and Medical Research Council","doi-asserted-by":"publisher","award":["APP1120356"],"award-info":[{"award-number":["APP1120356"]}],"id":[{"id":"10.13039\/501100000925","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/100000002","name":"National Institutes of Health","doi-asserted-by":"publisher","award":["R01HD078641"],"award-info":[{"award-number":["R01HD078641"]}],"id":[{"id":"10.13039\/100000002","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/100000002","name":"National Institutes of Health","doi-asserted-by":"publisher","award":["P50HD096723"],"award-info":[{"award-number":["P50HD096723"]}],"id":[{"id":"10.13039\/100000002","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/501100009708","name":"Novo Nordisk Foundation","doi-asserted-by":"publisher","award":["NNF210C0069913"],"award-info":[{"award-number":["NNF210C0069913"]}],"id":[{"id":"10.13039\/501100009708","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/501100009708","name":"Novo Nordisk Foundation","doi-asserted-by":"publisher","award":["NNF21C0069969"],"award-info":[{"award-number":["NNF21C0069969"]}],"id":[{"id":"10.13039\/501100009708","id-type":"DOI","asserted-by":"publisher"}]},{"name":"Independent Research Fund","award":["1030-00381B"],"award-info":[{"award-number":["1030-00381B"]}]}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":[],"published-print":{"date-parts":[[2025,9,1]]},"abstract":"<jats:title>Abstract<\/jats:title>\n               <jats:sec>\n                  <jats:title>STUDY QUESTION<\/jats:title>\n                  <jats:p>What is the role of coiled-coil domain-containing protein 89 (CCDC89) in mammalian male fertility?<\/jats:p>\n               <\/jats:sec>\n               <jats:sec>\n                  <jats:title>SUMMARY ANSWER<\/jats:title>\n                  <jats:p>The presence of CCDC89 is required for normal sperm motility and therefore optimal male fertility in mice, while CCDC89 variants affected spermatogenesis in both mice and humans.<\/jats:p>\n               <\/jats:sec>\n               <jats:sec>\n                  <jats:title>WHAT IS KNOWN ALREADY<\/jats:title>\n                  <jats:p>Coiled-coiled domain-containing proteins play a variety of roles in biological processes, including cell division, the production of motile sperm, and the regulation of their motility.<\/jats:p>\n               <\/jats:sec>\n               <jats:sec>\n                  <jats:title>STUDY DESIGN, SIZE, DURATION<\/jats:title>\n                  <jats:p>DNA from infertile men with azoospermia was sequenced to identify genetic variants as per the Genetics of Male Infertility Initiative (GEMINI) study. Genetic variants were identified in CCDC89 in three men, by whole exome sequencing. A testis biopsy from infertile Patient 1 (CCDC89 variant c. G903T) was available and used to inspect tissue pathology. Ccdc89 knockout (Ccdc89\u2212\/\u2212) and Ccdc89E297D\/E297D mutant mouse models were generated to define the role of CCDC89 in male fertility and the role of the specific CCDC89 genetic variant, c. G903T, in the pathogenesis of infertility.<\/jats:p>\n               <\/jats:sec>\n               <jats:sec>\n                  <jats:title>PARTICIPANTS\/MATERIALS, SETTING, METHODS<\/jats:title>\n                  <jats:p>CCDC89 RNA expression and protein localization were investigated in a testis biopsy from a control male with normal spermatogenesis. Male fertility of the mutant mouse lines was assessed via breeding, histology, daily sperm production, electron microscopy, computer-assisted and high-speed sperm motility analysis, and in vitro fertilization.<\/jats:p>\n               <\/jats:sec>\n               <jats:sec>\n                  <jats:title>MAIN RESULTS AND THE ROLE OF CHANCE<\/jats:title>\n                  <jats:p>Ccdc89\u2212\/\u2212 male mice were sub-fertile, with impaired progressive sperm motility and curvilinear velocity due to a rigid sperm tail midpiece without any overt structural defects. While Ccdc89E297D\/E297D males were fertile, their testis weights and germ cell content were reduced, suggesting a potential role of the c. G903T variant, observed in each of the two men, in the pathogenesis of their spermatogenic impairment. We also identified a new genetic variant in CCDC89 (c.G1024A) in another infertile man, that was in trans with the c. G903T genetic variant.<\/jats:p>\n               <\/jats:sec>\n               <jats:sec>\n                  <jats:title>LARGE SCALE DATA<\/jats:title>\n                  <jats:p>N\/A.<\/jats:p>\n               <\/jats:sec>\n               <jats:sec>\n                  <jats:title>LIMITATIONS, REASONS FOR CAUTION<\/jats:title>\n                  <jats:p>The identification of additional infertile men with genetic variants in CCDC89, and quality clinical data, are required to determine prognostic reliability regarding CCDC89 variants. There are likely to be species-specific differences in gene function.<\/jats:p>\n               <\/jats:sec>\n               <jats:sec>\n                  <jats:title>WIDER IMPLICATIONS OF THE FINDINGS<\/jats:title>\n                  <jats:p>Our data highlight a role for CCDC89, in regulating the sperm tail waveform, that is required for optimal sperm fertilization capacity and male fertility. We highlight CCDC89 as a regulator of male fertility in mammals, where variants in CCDC89 can affect spermatogenesis and may be a risk factor for human male infertility. This study underscores the importance of validating clinical genetic findings.<\/jats:p>\n               <\/jats:sec>\n               <jats:sec>\n                  <jats:title>STUDY FUNDING\/COMPETING INTEREST(S)<\/jats:title>\n                  <jats:p>This work was supported by a National Health and Medical Research Council grant (APP1120356), National Institutes of Health grants (R01HD078641 and P50HD096723), the Novo Nordisk Foundation (grant numbers NNF210C0069913 and NNF21C0069969), the Capital Region of Denmark, the Independent Research Fund (grant number: 1030-00381B), and the Svend Andersen Foundation (grant number: 84-A.08), and the Christian and Ottilia Brorsons (No. 12038-1) and Frimodt-Heineke Foundation travel grants for research exchange. The authors have no conflicts of interest.<\/jats:p>\n               <\/jats:sec>","DOI":"10.1093\/humrep\/deaf126","type":"journal-article","created":{"date-parts":[[2025,6,18]],"date-time":"2025-06-18T07:50:43Z","timestamp":1750233043000},"page":"1616-1628","source":"Crossref","is-referenced-by-count":5,"title":["CCDC89 is required for optimal sperm motility and male fertility in mammals"],"prefix":"10.1093","volume":"40","author":[{"ORCID":"https:\/\/orcid.org\/0000-0001-7298-4940","authenticated-orcid":false,"given":"Moira K","family":"O\u2019Bryan","sequence":"first","affiliation":[{"name":"School of BioSciences and Bio21 Molecular Sciences and Biotechnology Institute, The University of Melbourne , Parkville,","place":["Australia"]},{"name":"International Male Infertility Genomics Consortium, IMIGC"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"G\u00fclizar","family":"Saritas","sequence":"additional","affiliation":[{"name":"Department of Growth and Reproduction, Copenhagen University Hospital\u2014Rigshospitalet , Copenhagen,","place":["Denmark"]},{"name":"International Center for Research and Research Training in Endocrine Disruption of Male Reproduction and Child Health, Copenhagen University Hospital\u2014Rigshospitalet , Copenhagen,","place":["Denmark"]},{"name":"Department of Cellular and Molecular Medicine, Faculty of Health and Medical Sciences, University of Copenhagen , Copenhagen,","place":["Denmark"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Joseph","family":"Nguyen","sequence":"additional","affiliation":[{"name":"School of BioSciences and Bio21 Molecular Sciences and Biotechnology Institute, The University of Melbourne , Parkville,","place":["Australia"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Sofia B","family":"Winge","sequence":"additional","affiliation":[{"name":"Department of Growth and Reproduction, Copenhagen University Hospital\u2014Rigshospitalet , Copenhagen,","place":["Denmark"]},{"name":"International Center for Research and Research Training in Endocrine Disruption of Male Reproduction and Child Health, Copenhagen University Hospital\u2014Rigshospitalet , Copenhagen,","place":["Denmark"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Anne E","family":"O\u2019Connor","sequence":"additional","affiliation":[{"name":"School of BioSciences and Bio21 Molecular Sciences and Biotechnology Institute, The University of Melbourne , Parkville,","place":["Australia"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Helen","family":"Castillo-Madeen","sequence":"additional","affiliation":[{"name":"Oregon National Primate Research Center, Oregon Health and Science University , Beaverton, OR,","place":["USA"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-3828-8970","authenticated-orcid":false,"given":"Donald F","family":"Conrad","sequence":"additional","affiliation":[{"name":"International Male Infertility Genomics Consortium, IMIGC"},{"name":"Oregon National Primate Research Center, Oregon Health and Science University , Beaverton, OR,","place":["USA"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Maddison","family":"Graffeo","sequence":"additional","affiliation":[{"name":"School of BioSciences and Bio21 Molecular Sciences and Biotechnology Institute, The University of Melbourne , Parkville,","place":["Australia"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Reza","family":"Nosrati","sequence":"additional","affiliation":[{"name":"Department of Mechanical and Aerospace Engineering, Monash University , Clayton, VIC,","place":["Australia"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Jessica E M","family":"Dunleavy","sequence":"additional","affiliation":[{"name":"School of BioSciences and Bio21 Molecular Sciences and Biotechnology Institute, The University of Melbourne , Parkville,","place":["Australia"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-1832-0307","authenticated-orcid":false,"given":"Kristian","family":"Almstrup","sequence":"additional","affiliation":[{"name":"International Male Infertility Genomics Consortium, IMIGC"},{"name":"Department of Growth and Reproduction, Copenhagen University Hospital\u2014Rigshospitalet , Copenhagen,","place":["Denmark"]},{"name":"International Center for Research and Research Training in Endocrine Disruption of Male Reproduction and Child Health, Copenhagen University Hospital\u2014Rigshospitalet , Copenhagen,","place":["Denmark"]},{"name":"Department of Cellular and Molecular Medicine, Faculty of Health and Medical Sciences, University of Copenhagen , Copenhagen,","place":["Denmark"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-1078-756X","authenticated-orcid":false,"given":"Brendan J","family":"Houston","sequence":"additional","affiliation":[{"name":"School of BioSciences and Bio21 Molecular Sciences and Biotechnology Institute, The University of Melbourne , Parkville,","place":["Australia"]}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"286","published-online":{"date-parts":[[2025,7,1]]},"reference":[{"key":"2025090401512759200_deaf126-B1","doi-asserted-by":"crossref","first-page":"2793","DOI":"10.1016\/j.cellsig.2014.08.025","article-title":"Coiled-coil domain containing 3 (CCDC3) represses tumor necrosis factor-alpha\/nuclear factor kappaB-induced endothelial inflammation","volume":"26","author":"Azad","year":"2014","journal-title":"Cell Signal"},{"key":"2025090401512759200_deaf126-B2","doi-asserted-by":"crossref","first-page":"e1009388","DOI":"10.1371\/journal.pgen.1009388","article-title":"Ccdc113\/Ccdc96 complex, a novel regulator of ciliary beating that connects radial spoke 3 to dynein g and the nexin link","volume":"17","author":"Bazan","year":"2021","journal-title":"PLoS Genet"},{"key":"2025090401512759200_deaf126-B3","doi-asserted-by":"crossref","first-page":"82","DOI":"10.1016\/S0962-8924(00)01898-5","article-title":"Coiled coils: a highly versatile protein folding motif","volume":"11","author":"Burkhard","year":"2001","journal-title":"Trends Cell Biol"},{"key":"2025090401512759200_deaf126-B4","doi-asserted-by":"crossref","first-page":"476","DOI":"10.1093\/molehr\/gau018","article-title":"The human testis-specific proteome defined by transcriptomics and antibody-based profiling","volume":"20","author":"Djureinovic","year":"2014","journal-title":"Mol Hum Reprod"},{"key":"2025090401512759200_deaf126-B5","doi-asserted-by":"crossref","first-page":"dev201956","DOI":"10.1242\/dev.201956","article-title":"The katanin A-subunits KATNA1 and KATNAL1 act co-operatively in mammalian meiosis and spermiogenesis to achieve male fertility","volume":"150","author":"Dunleavy","year":"2023","journal-title":"Development"},{"key":"2025090401512759200_deaf126-B6","doi-asserted-by":"crossref","first-page":"dev199922","DOI":"10.1242\/dev.199922","article-title":"KATNB1 is a master regulator of multiple katanin enzymes in male meiosis and haploid germ cell development","volume":"148","author":"Dunleavy","year":"2021","journal-title":"Development"},{"key":"2025090401512759200_deaf126-B7","doi-asserted-by":"crossref","first-page":"e1007078","DOI":"10.1371\/journal.pgen.1007078","article-title":"Katanin-like 2 (KATNAL2) functions in multiple aspects of haploid male germ cell development in the mouse","volume":"13","author":"Dunleavy","year":"2017","journal-title":"PLoS Genet"},{"key":"2025090401512759200_deaf126-B8","doi-asserted-by":"crossref","first-page":"193","DOI":"10.1038\/s41586-019-1064-z","article-title":"Genetic compensation triggered by mutant mRNA degradation","volume":"568","author":"El-Brolosy","year":"2019","journal-title":"Nature"},{"key":"2025090401512759200_deaf126-B9","doi-asserted-by":"crossref","first-page":"397","DOI":"10.1074\/mcp.M113.035600","article-title":"Analysis of the human tissue-specific expression by genome-wide integration of transcriptomics and antibody-based proteomics","volume":"13","author":"Fagerberg","year":"2014","journal-title":"Mol Cell Proteomics"},{"key":"2025090401512759200_deaf126-B10","doi-asserted-by":"crossref","first-page":"693258","DOI":"10.3389\/fcell.2021.693258","article-title":"CRISPs function to boost sperm power output and motility","volume":"9","author":"Gaikwad","year":"2021","journal-title":"Front Cell Dev Biol"},{"key":"2025090401512759200_deaf126-B11","doi-asserted-by":"crossref","first-page":"155","DOI":"10.1007\/s00439-020-02159-x","article-title":"A framework for high-resolution phenotyping of candidate male infertility mutants: from human to mouse","volume":"140","author":"Houston","year":"2021","journal-title":"Hum Genet"},{"key":"2025090401512759200_deaf126-B12","doi-asserted-by":"crossref","first-page":"e202302452","DOI":"10.26508\/lsa.202302452","article-title":"Genetic mutation of Cep76 results in male infertility due to abnormal sperm tail composition","volume":"7","author":"Houston","year":"2024","journal-title":"Life Sci Alliance"},{"key":"2025090401512759200_deaf126-B13","doi-asserted-by":"crossref","first-page":"922","DOI":"10.1002\/dvdy.299","article-title":"The Sertoli cell expressed gene secernin-1 (Scrn1) is dispensable for male fertility in the mouse","volume":"250","author":"Houston","year":"2021","journal-title":"Dev Dyn"},{"key":"2025090401512759200_deaf126-B14","doi-asserted-by":"crossref","first-page":"bqab269","DOI":"10.1210\/endocr\/bqab269","article-title":"Human INHBB gene variant (c.1079T&gt;C: p.Met360Thr) alters testis germ cell content, but does not impact fertility in mice","volume":"163","author":"Houston","year":"2022","journal-title":"Endocrinology"},{"key":"2025090401512759200_deaf126-B15","doi-asserted-by":"crossref","first-page":"15","DOI":"10.1093\/humupd\/dmab030","article-title":"A systematic review of the validated monogenic causes of human male infertility: 2020 update and a discussion of emerging gene-disease relationships","volume":"28","author":"Houston","year":"2021","journal-title":"Hum Reprod Update"},{"key":"2025090401512759200_deaf126-B16","doi-asserted-by":"crossref","first-page":"111","DOI":"10.1093\/molehr\/gay001","article-title":"Epididymal cysteine-rich secretory proteins are required for epididymal sperm maturation and optimal sperm function","volume":"24","author":"Hu","year":"2018","journal-title":"Mol Hum Reprod"},{"key":"2025090401512759200_deaf126-B17","doi-asserted-by":"crossref","first-page":"583","DOI":"10.1038\/s41586-021-03819-2","article-title":"Highly accurate protein structure prediction with AlphaFold","volume":"596","author":"Jumper","year":"2021","journal-title":"Nature"},{"key":"2025090401512759200_deaf126-B18","doi-asserted-by":"crossref","first-page":"e43966","DOI":"10.7554\/eLife.43966","article-title":"Unified single-cell analysis of testis gene regulation and pathology in five mouse strains","volume":"8","author":"Jung","year":"2019","journal-title":"Elife"},{"key":"2025090401512759200_deaf126-B19","doi-asserted-by":"crossref","first-page":"310","DOI":"10.1038\/ng.2892","article-title":"A general framework for estimating the relative pathogenicity of human genetic variants","volume":"46","author":"Kircher","year":"2014","journal-title":"Nat Genet"},{"key":"2025090401512759200_deaf126-B20","doi-asserted-by":"crossref","first-page":"672592","DOI":"10.3389\/fcell.2021.672592","article-title":"New insights into sperm ultrastructure through enhanced scanning electron microscopy","volume":"9","author":"Korneev","year":"2021","journal-title":"Front Cell Dev Biol"},{"key":"2025090401512759200_deaf126-B21","doi-asserted-by":"crossref","first-page":"369","DOI":"10.1038\/s41585-018-0003-3","article-title":"Genetics of male infertility","volume":"15","author":"Krausz","year":"2018","journal-title":"Nat Rev Urol"},{"key":"2025090401512759200_deaf126-B22","doi-asserted-by":"crossref","first-page":"267","DOI":"10.1016\/j.ajhg.2017.01.004","article-title":"InterVar: clinical interpretation of genetic variants by the 2015 ACMG-AMP guidelines","volume":"100","author":"Li","year":"2017","journal-title":"Am J Hum Genet"},{"key":"2025090401512759200_deaf126-B23","doi-asserted-by":"crossref","first-page":"67","DOI":"10.1095\/biolreprod.112.106450","article-title":"Melatonin prevents postovulatory oocyte aging in the mouse and extends the window for optimal fertilization in vitro","volume":"88","author":"Lord","year":"2013","journal-title":"Biol Reprod"},{"key":"2025090401512759200_deaf126-B24","doi-asserted-by":"crossref","first-page":"1275","DOI":"10.1093\/biolre\/ioz008","article-title":"Investigation into the presence and functional significance of proinsulin C-peptide in the female germlinedagger","volume":"100","author":"Martin","year":"2019","journal-title":"Biol Reprod"},{"key":"2025090401512759200_deaf126-B25","doi-asserted-by":"crossref","first-page":"679","DOI":"10.1038\/s41592-022-01488-1","article-title":"ColabFold: making protein folding accessible to all","volume":"19","author":"Mirdita","year":"2022","journal-title":"Nat Methods"},{"key":"2025090401512759200_deaf126-B26","doi-asserted-by":"crossref","first-page":"726","DOI":"10.1016\/j.jmb.2008.11.028","article-title":"A periodic table of coiled-coil protein structures","volume":"385","author":"Moutevelis","year":"2009","journal-title":"J Mol Biol"},{"key":"2025090401512759200_deaf126-B27","doi-asserted-by":"crossref","first-page":"7953","DOI":"10.1038\/s41467-022-35661-z","article-title":"Diverse monogenic subforms of human spermatogenic failure","volume":"13","author":"Nagirnaja","year":"2022","journal-title":"Nat Commun"},{"key":"2025090401512759200_deaf126-B28","doi-asserted-by":"crossref","first-page":"e62524","DOI":"10.7554\/eLife.62524","article-title":"Flagellar energetics from high-resolution imaging of beating patterns in tethered mouse sperm","volume":"10","author":"Nandagiri","year":"2021","journal-title":"Elife"},{"key":"2025090401512759200_deaf126-B29","doi-asserted-by":"crossref","first-page":"12866","DOI":"10.1038\/s41598-019-49177-y","article-title":"Characterisation and localisation of the endocannabinoid system components in the adult human testis","volume":"9","author":"Nielsen","year":"2019","journal-title":"Sci Rep"},{"key":"2025090401512759200_deaf126-B30","doi-asserted-by":"crossref","first-page":"208","DOI":"10.1016\/j.ydbio.2016.01.042","article-title":"Coiled-coil domain containing 42 (Ccdc42) is necessary for proper sperm development and male fertility in the mouse","volume":"412","author":"Pasek","year":"2016","journal-title":"Dev Biol"},{"key":"2025090401512759200_deaf126-B31","doi-asserted-by":"crossref","first-page":"8127","DOI":"10.1093\/nar\/gkae554","article-title":"CilioGenics: an integrated method and database for predicting novel ciliary genes","volume":"52","author":"Pir","year":"2024","journal-title":"Nucleic Acids Res"},{"key":"2025090401512759200_deaf126-B32","doi-asserted-by":"crossref","first-page":"e2101089","DOI":"10.1002\/smtd.202101089","article-title":"Unraveling the kinematics of sperm motion by reconstructing the flagellar wave motion in 3D","volume":"6","author":"Powar","year":"2022","journal-title":"Small Methods"},{"key":"2025090401512759200_deaf126-B33","doi-asserted-by":"crossref","first-page":"2725","DOI":"10.1007\/s43032-021-00595-2","article-title":"Coiled-coil domain-containing (CCDC) proteins: functional roles in general and male reproductive physiology","volume":"28","author":"Priyanka","year":"2021","journal-title":"Reprod Sci"},{"key":"2025090401512759200_deaf126-B34","doi-asserted-by":"crossref","first-page":"24","DOI":"10.1038\/nbt.1754","article-title":"Integrative genomics viewer","volume":"29","author":"Robinson","year":"2011","journal-title":"Nat Biotechnol"},{"key":"2025090401512759200_deaf126-B35","doi-asserted-by":"crossref","first-page":"19089","DOI":"10.1038\/s41598-021-98267-3","article-title":"Integration and gene co-expression network analysis of scRNA-seq transcriptomes reveal heterogeneity and key functional genes in human spermatogenesis","volume":"11","author":"Salehi","year":"2021","journal-title":"Sci Rep"},{"key":"2025090401512759200_deaf126-B36","doi-asserted-by":"crossref","first-page":"2244","DOI":"10.1172\/JCI66466","article-title":"CCDC22 deficiency in humans blunts activation of proinflammatory NF-kappaB signaling","volume":"123","author":"Starokadomskyy","year":"2013","journal-title":"J Clin Invest"},{"key":"2025090401512759200_deaf126-B37","doi-asserted-by":"crossref","first-page":"dev186452","DOI":"10.1242\/dev.186452","article-title":"Transcriptional adaptation: a mechanism underlying genetic robustness","volume":"147","author":"Sztal","year":"2020","journal-title":"Development"},{"key":"2025090401512759200_deaf126-B38","doi-asserted-by":"crossref","first-page":"151","DOI":"10.3389\/fcell.2019.00151","article-title":"CCDC42 localizes to Manchette, HTCA and tail and interacts with ODF1 and ODF2 in the formation of the male germ cell cytoskeleton","volume":"7","author":"Tapia Contreras","year":"2019","journal-title":"Front Cell Dev Biol"},{"key":"2025090401512759200_deaf126-B39","doi-asserted-by":"crossref","first-page":"26","DOI":"10.1016\/j.addr.2020.12.012","article-title":"Coiled coil-based therapeutics and drug delivery systems","volume":"170","author":"Utterstrom","year":"2021","journal-title":"Adv Drug Deliv Rev"},{"key":"2025090401512759200_deaf126-B40","doi-asserted-by":"crossref","first-page":"D368","DOI":"10.1093\/nar\/gkad1011","article-title":"AlphaFold Protein Structure Database in 2024: providing structure coverage for over 214 million protein sequences","volume":"52","author":"Varadi","year":"2024","journal-title":"Nucleic Acids Res"},{"key":"2025090401512759200_deaf126-B41","doi-asserted-by":"crossref","first-page":"nwad181","DOI":"10.1093\/nsr\/nwad181","article-title":"CCDC189 affects sperm flagellum formation by interacting with CABCOCO1","volume":"10","author":"Wang","year":"2023","journal-title":"Natl Sci Rev"},{"key":"2025090401512759200_deaf126-B42","first-page":"817","article-title":"Ccdc87 is critical for sperm function and male fertility","volume":"99","author":"Wang","year":"2018","journal-title":"Biol Reprod"},{"key":"2025090401512759200_deaf126-B43","first-page":"1030","article-title":"MetaDome: pathogenicity analysis of genetic variants through aggregation of homologous human protein domains","volume":"40","author":"Wiel","year":"2019","journal-title":"Hum Mutat"},{"key":"2025090401512759200_deaf126-B44","doi-asserted-by":"crossref","first-page":"1454","DOI":"10.1002\/humu.23313","article-title":"Aggregation of population-based genetic variation over protein domain homologues and its potential use in genetic diagnostics","volume":"38","author":"Wiel","year":"2017","journal-title":"Hum Mutat"},{"key":"2025090401512759200_deaf126-B45","doi-asserted-by":"crossref","first-page":"976","DOI":"10.1091\/mbc.e10-04-0331","article-title":"Regulation of flagellar motility by the conserved flagellar protein CG34110\/Ccdc135\/FAP50","volume":"22","author":"Yang","year":"2011","journal-title":"Mol Biol Cell"},{"key":"2025090401512759200_deaf126-B46","doi-asserted-by":"crossref","first-page":"101646","DOI":"10.1016\/j.xcrp.2023.101646","article-title":"The cooperative impact of flow and viscosity on sperm flagellar energetics in biomimetic environments","volume":"4","author":"Yazdan Parast","year":"2023","journal-title":"Cell Rep Phys Sci"},{"key":"2025090401512759200_deaf126-B48","doi-asserted-by":"crossref","first-page":"dev200516","DOI":"10.1242\/dev.200516","article-title":"CCDC38 is required for sperm flagellum biogenesis and male fertility in mice","volume":"149","author":"Zhang","year":"2022","journal-title":"Development"}],"container-title":["Human Reproduction"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/academic.oup.com\/humrep\/article-pdf\/40\/9\/1616\/63638459\/deaf126.pdf","content-type":"application\/pdf","content-version":"vor","intended-application":"syndication"},{"URL":"https:\/\/academic.oup.com\/humrep\/article-pdf\/40\/9\/1616\/63638459\/deaf126.pdf","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2025,9,4]],"date-time":"2025-09-04T05:51:38Z","timestamp":1756965098000},"score":1,"resource":{"primary":{"URL":"https:\/\/academic.oup.com\/humrep\/article\/40\/9\/1616\/8180657"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2025,7,1]]},"references-count":47,"journal-issue":{"issue":"9","published-online":{"date-parts":[[2025,7,1]]},"published-print":{"date-parts":[[2025,9,1]]}},"URL":"https:\/\/doi.org\/10.1093\/humrep\/deaf126","relation":{},"ISSN":["0268-1161","1460-2350"],"issn-type":[{"value":"0268-1161","type":"print"},{"value":"1460-2350","type":"electronic"}],"subject":[],"published-other":{"date-parts":[[2025,9]]},"published":{"date-parts":[[2025,7,1]]}}}