{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,10,5]],"date-time":"2026-10-05T02:26:09Z","timestamp":1791167169076,"version":"4.1.0"},"reference-count":70,"publisher":"Springer Science and Business Media LLC","issue":"1","license":[{"start":{"date-parts":[[2025,3,17]],"date-time":"2025-03-17T00:00:00Z","timestamp":1742169600000},"content-version":"tdm","delay-in-days":0,"URL":"https:\/\/creativecommons.org\/licenses\/by-nc-nd\/4.0"},{"start":{"date-parts":[[2025,3,17]],"date-time":"2025-03-17T00:00:00Z","timestamp":1742169600000},"content-version":"vor","delay-in-days":0,"URL":"https:\/\/creativecommons.org\/licenses\/by-nc-nd\/4.0"}],"funder":[{"DOI":"10.13039\/501100018627","name":"Science and Technology Support Plan for Youth Innovation of Colleges and Universities of Shandong Province of China","doi-asserted-by":"publisher","award":["202201010631"],"award-info":[{"award-number":["202201010631"]}],"id":[{"id":"10.13039\/501100018627","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/501100018627","name":"Science and Technology Support Plan for Youth Innovation of Colleges and Universities of Shandong Province of China","doi-asserted-by":"publisher","award":["202205030999"],"award-info":[{"award-number":["202205030999"]}],"id":[{"id":"10.13039\/501100018627","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/501100001809","name":"National Natural Science Foundation of China","doi-asserted-by":"publisher","award":["82401875"],"award-info":[{"award-number":["82401875"]}],"id":[{"id":"10.13039\/501100001809","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/501100015507","name":"Shandong First Medical University","doi-asserted-by":"publisher","award":["202201-010"],"award-info":[{"award-number":["202201-010"]}],"id":[{"id":"10.13039\/501100015507","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/501100007129","name":"Natural Science Foundation of Shandong Province","doi-asserted-by":"publisher","award":["ZR2023QH164"],"award-info":[{"award-number":["ZR2023QH164"]}],"id":[{"id":"10.13039\/501100007129","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/501100007129","name":"Natural Science Foundation of Shandong Province","doi-asserted-by":"publisher","award":["ZR2022QC184"],"award-info":[{"award-number":["ZR2022QC184"]}],"id":[{"id":"10.13039\/501100007129","id-type":"DOI","asserted-by":"publisher"}]}],"content-domain":{"domain":["link.springer.com"],"crossmark-restriction":false},"short-container-title":["Cell. Mol. Life Sci."],"DOI":"10.1007\/s00018-025-05616-w","type":"journal-article","created":{"date-parts":[[2025,3,17]],"date-time":"2025-03-17T08:48:32Z","timestamp":1742201312000},"update-policy":"https:\/\/doi.org\/10.1007\/springer_crossmark_policy","source":"Crossref","is-referenced-by-count":3,"title":["Loss of Cep135 causes oligoasthenoteratozoospermia and male infertility in mice"],"prefix":"10.1007","volume":"82","author":[{"given":"Hui","family":"Liu","sequence":"first","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Haozheng","family":"Zhang","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Guanghao","family":"Qin","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Tingting","family":"Song","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Xin","family":"Liu","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Zongzhuang","family":"Wen","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Min","family":"Liu","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Xianmei","family":"Wang","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Xiaolong","family":"Fu","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-6619-7107","authenticated-orcid":false,"given":"Jiangang","family":"Gao","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"297","published-online":{"date-parts":[[2025,3,17]]},"reference":[{"key":"5616_CR1","doi-asserted-by":"publisher","first-page":"319","DOI":"10.1016\/S0140-6736(20)32667-2","volume":"397","author":"A Agarwal","year":"2021","unstructured":"Agarwal A, Baskaran S, Parekh N et al (2021) Male infertility. Lancet 397:319\u2013333. https:\/\/doi.org\/10.1016\/S0140-6736(20)32667-2","journal-title":"Lancet"},{"key":"5616_CR2","doi-asserted-by":"publisher","unstructured":"Aitken RJ, Drevet JR, Moazamian A et al (2022) Male infertility and oxidative stress: a focus on the underlying mechanisms. Antioxid (Basel) 11. https:\/\/doi.org\/10.3390\/antiox11020306","DOI":"10.3390\/antiox11020306"},{"key":"5616_CR3","doi-asserted-by":"publisher","first-page":"102393","DOI":"10.1016\/j.bpobgyn.2023.102393","volume":"90","author":"D Ameratunga","year":"2023","unstructured":"Ameratunga D, Gebeh A, Amoako A (2023) Obesity and male infertility. Best Pract Res Clin Obstet Gynaecol 90:102393. https:\/\/doi.org\/10.1016\/j.bpobgyn.2023.102393","journal-title":"Best Pract Res Clin Obstet Gynaecol"},{"key":"5616_CR4","doi-asserted-by":"publisher","first-page":"61","DOI":"10.1038\/s41585-024-00856-0","volume":"21","author":"Anonymous","year":"2024","unstructured":"Anonymous (2024) Focusing on male infertility. Nat Rev Urol 21:61. https:\/\/doi.org\/10.1038\/s41585-024-00856-0","journal-title":"Nat Rev Urol"},{"key":"5616_CR5","doi-asserted-by":"publisher","first-page":"110987","DOI":"10.1016\/j.mce.2020.110987","volume":"518","author":"T Avidor-Reiss","year":"2020","unstructured":"Avidor-Reiss T, Carr A, Fishman EL (2020) The sperm centrioles. Mol Cell Endocrinol 518:110987. https:\/\/doi.org\/10.1016\/j.mce.2020.110987","journal-title":"Mol Cell Endocrinol"},{"key":"5616_CR6","doi-asserted-by":"publisher","first-page":"1454","DOI":"10.1002\/jemt.24009","volume":"85","author":"M Bizkarguenaga","year":"2022","unstructured":"Bizkarguenaga M, Gomez-Santos L, Madrid JF et al (2022) Zona Pellucida sperm-binding protein 3 receptor distribution during Gopc(-\/-) globozoospermic spermatogenesis. Microsc Res Tech 85:1454\u20131464. https:\/\/doi.org\/10.1002\/jemt.24009","journal-title":"Microsc Res Tech"},{"key":"5616_CR7","doi-asserted-by":"publisher","unstructured":"Blaurock J, Baumann S, Grunewald S et al (2022) Metabolomics of human semen: a review of different Analytical methods to unravel biomarkers for Male Fertility disorders. Int J Mol Sci 23. https:\/\/doi.org\/10.3390\/ijms23169031","DOI":"10.3390\/ijms23169031"},{"key":"5616_CR8","doi-asserted-by":"publisher","first-page":"422","DOI":"10.1126\/science.1209037","volume":"335","author":"M Bornens","year":"2012","unstructured":"Bornens M (2012) The centrosome in cells and organisms. Science 335:422\u2013426. https:\/\/doi.org\/10.1126\/science.1209037","journal-title":"Science"},{"key":"5616_CR9","doi-asserted-by":"publisher","unstructured":"Brukman NG, Nakajima KP, Valansi C et al (2023) A novel function for the sperm adhesion protein IZUMO1 in cell-cell fusion. J Cell Biol 222. https:\/\/doi.org\/10.1083\/jcb.202207147","DOI":"10.1083\/jcb.202207147"},{"key":"5616_CR10","doi-asserted-by":"publisher","first-page":"412","DOI":"10.1016\/j.devcel.2012.06.001","volume":"23","author":"Z Carvalho-Santos","year":"2012","unstructured":"Carvalho-Santos Z, Machado P, Alvarez-Martins I et al (2012) BLD10\/CEP135 is a microtubule-associated protein that controls the formation of the flagellum central microtubule pair. Dev Cell 23:412\u2013424. https:\/\/doi.org\/10.1016\/j.devcel.2012.06.001","journal-title":"Dev Cell"},{"key":"5616_CR11","doi-asserted-by":"publisher","first-page":"710","DOI":"10.1136\/jmedgenet-2021-107919","volume":"59","author":"J Cong","year":"2022","unstructured":"Cong J, Wang X, Amiri-Yekta A et al (2022) Homozygous mutations in CCDC34 cause male infertility with oligoasthenoteratozoospermia in humans and mice. J Med Genet 59:710\u2013718. https:\/\/doi.org\/10.1136\/jmedgenet-2021-107919","journal-title":"J Med Genet"},{"key":"5616_CR12","doi-asserted-by":"publisher","unstructured":"Crapster JA, Rack PG, Hellmann ZJ et al (2020) HIPK4 is essential for murine spermiogenesis. Elife 9. https:\/\/doi.org\/10.7554\/eLife.50209","DOI":"10.7554\/eLife.50209"},{"key":"5616_CR13","doi-asserted-by":"publisher","first-page":"2591","DOI":"10.1016\/j.cub.2015.08.039","volume":"25","author":"KD Dahl","year":"2015","unstructured":"Dahl KD, Sankaran DG, Bayless BA et al (2015) A short CEP135 splice isoform controls Centriole Duplication. Curr Biol 25:2591\u20132596. https:\/\/doi.org\/10.1016\/j.cub.2015.08.039","journal-title":"Curr Biol"},{"key":"5616_CR14","doi-asserted-by":"publisher","first-page":"e16501","DOI":"10.15252\/emmm.202216501","volume":"14","author":"J Dai","year":"2022","unstructured":"Dai J, Li Q, Zhou Q et al (2022) IQCN disruption causes fertilization failure and male infertility due to manchette assembly defect. EMBO Mol Med 14:e16501. https:\/\/doi.org\/10.15252\/emmm.202216501","journal-title":"EMBO Mol Med"},{"key":"5616_CR15","doi-asserted-by":"publisher","first-page":"100395","DOI":"10.1016\/j.xcrm.2021.100395","volume":"2","author":"S Di Persio","year":"2021","unstructured":"Di Persio S, Tekath T, Siebert-Kuss LM et al (2021) Single-cell RNA-seq unravels alterations of the human spermatogonial stem cell compartment in patients with impaired spermatogenesis. Cell Rep Med 2:100395. https:\/\/doi.org\/10.1016\/j.xcrm.2021.100395","journal-title":"Cell Rep Med"},{"key":"5616_CR16","doi-asserted-by":"publisher","first-page":"2210","DOI":"10.1038\/s41467-018-04678-8","volume":"9","author":"EL Fishman","year":"2018","unstructured":"Fishman EL, Jo K, Nguyen QPH et al (2018) A novel atypical sperm centriole is functional during human fertilization. Nat Commun 9:2210. https:\/\/doi.org\/10.1038\/s41467-018-04678-8","journal-title":"Nat Commun"},{"key":"5616_CR17","doi-asserted-by":"publisher","first-page":"754054","DOI":"10.3389\/fcell.2021.754054","volume":"9","author":"S Floriot","year":"2021","unstructured":"Floriot S, Bellutti L, Castille J et al (2021) CEP250 is required for maintaining centrosome cohesion in the germline and fertility in male mice. Front Cell Dev Biol 9:754054. https:\/\/doi.org\/10.3389\/fcell.2021.754054","journal-title":"Front Cell Dev Biol"},{"key":"5616_CR18","doi-asserted-by":"publisher","first-page":"1230","DOI":"10.1091\/mbc.E18-10-0674","volume":"30","author":"D Ganapathi Sankaran","year":"2019","unstructured":"Ganapathi Sankaran D, Stemm-Wolf AJ, Pearson CG (2019) CEP135 isoform dysregulation promotes centrosome amplification in breast cancer cells. Mol Biol Cell 30:1230\u20131244. https:\/\/doi.org\/10.1091\/mbc.E18-10-0674","journal-title":"Mol Biol Cell"},{"key":"5616_CR19","doi-asserted-by":"publisher","first-page":"142","DOI":"10.1016\/j.ydbio.2009.03.020","volume":"330","author":"CB Geyer","year":"2009","unstructured":"Geyer CB, Inselman AL, Sunman JA et al (2009) A missense mutation in the Capza3 gene and disruption of F-actin organization in spermatids of repro32 infertile male mice. Dev Biol 330:142\u2013152. https:\/\/doi.org\/10.1016\/j.ydbio.2009.03.020","journal-title":"Dev Biol"},{"key":"5616_CR20","doi-asserted-by":"publisher","first-page":"e13586","DOI":"10.1111\/and.13586","volume":"53","author":"S Gunes","year":"2021","unstructured":"Gunes S, Esteves SC (2021) Role of genetics and epigenetics in male infertility. Andrologia 53:e13586. https:\/\/doi.org\/10.1111\/and.13586","journal-title":"Andrologia"},{"key":"5616_CR21","doi-asserted-by":"publisher","first-page":"100256","DOI":"10.1016\/j.xhgg.2023.100256","volume":"5","author":"J Guo","year":"2023","unstructured":"Guo J, He WB, Dai L et al (2023) Mosaic variegated aneuploidy syndrome with tetraploid, and predisposition to male infertility triggered by mutant CEP192. HGG Adv 5:100256. https:\/\/doi.org\/10.1016\/j.xhgg.2023.100256","journal-title":"HGG Adv"},{"key":"5616_CR22","doi-asserted-by":"publisher","first-page":"e1003928","DOI":"10.1371\/journal.pgen.1003928","volume":"9","author":"EA Hall","year":"2013","unstructured":"Hall EA, Keighren M, Ford MJ et al (2013) Acute versus chronic loss of mammalian Azi1\/Cep131 results in distinct ciliary phenotypes. PLoS Genet 9:e1003928. https:\/\/doi.org\/10.1371\/journal.pgen.1003928","journal-title":"PLoS Genet"},{"key":"5616_CR23","doi-asserted-by":"publisher","first-page":"129","DOI":"10.1530\/REP-21-0042","volume":"162","author":"M Hoque","year":"2021","unstructured":"Hoque M, Chen D, Hess RA et al (2021) CEP164 is essential for efferent duct multiciliogenesis and male fertility. Reproduction 162:129\u2013139. https:\/\/doi.org\/10.1530\/REP-21-0042","journal-title":"Reproduction"},{"key":"5616_CR24","doi-asserted-by":"publisher","first-page":"144","DOI":"10.1136\/jmedgenet-2021-108249","volume":"60","author":"T Hu","year":"2023","unstructured":"Hu T, Meng L, Tan C et al (2023) Biallelic CFAP61 variants cause male infertility in humans and mice with severe oligoasthenoteratozoospermia. J Med Genet 60:144\u2013153. https:\/\/doi.org\/10.1136\/jmedgenet-2021-108249","journal-title":"J Med Genet"},{"key":"5616_CR25","doi-asserted-by":"publisher","first-page":"2307","DOI":"10.1093\/hmg\/ddad061","volume":"32","author":"J Hua","year":"2023","unstructured":"Hua J, Xu B, Liu W et al (2023) Homozygous frameshift variant in POC1B causes male infertility with oligoasthenoteratozoospermia in human and mice. Hum Mol Genet 32:2307\u20132317. https:\/\/doi.org\/10.1093\/hmg\/ddad061","journal-title":"Hum Mol Genet"},{"key":"5616_CR26","doi-asserted-by":"publisher","first-page":"871","DOI":"10.1016\/j.ajhg.2012.03.016","volume":"90","author":"MS Hussain","year":"2012","unstructured":"Hussain MS, Baig SM, Neumann S et al (2012) A truncating mutation of CEP135 causes primary microcephaly and disturbed centrosomal function. Am J Hum Genet 90:871\u2013878. https:\/\/doi.org\/10.1016\/j.ajhg.2012.03.016","journal-title":"Am J Hum Genet"},{"key":"5616_CR27","doi-asserted-by":"publisher","first-page":"2645","DOI":"10.1091\/mbc.E13-03-0149","volume":"24","author":"B Inanc","year":"2013","unstructured":"Inanc B, Putz M, Lalor P et al (2013) Abnormal centrosomal structure and duplication in Cep135-deficient vertebrate cells. Mol Biol Cell 24:2645\u20132654. https:\/\/doi.org\/10.1091\/mbc.E13-03-0149","journal-title":"Mol Biol Cell"},{"key":"5616_CR28","doi-asserted-by":"publisher","first-page":"113","DOI":"10.1016\/j.semcdb.2020.04.019","volume":"110","author":"S Jaiswal","year":"2021","unstructured":"Jaiswal S, Singh P (2021) Centrosome dysfunction in human diseases. Semin Cell Dev Biol 110:113\u2013122. https:\/\/doi.org\/10.1016\/j.semcdb.2020.04.019","journal-title":"Semin Cell Dev Biol"},{"key":"5616_CR29","doi-asserted-by":"publisher","unstructured":"Jin HJ, Ruan T, Dai S et al (2023) Identification of CFAP52 as a novel diagnostic target of male infertility with defects of sperm head-tail connection and flagella development. Elife 12. https:\/\/doi.org\/10.7554\/eLife.92769","DOI":"10.7554\/eLife.92769"},{"key":"5616_CR30","doi-asserted-by":"publisher","first-page":"104675","DOI":"10.1016\/j.ebiom.2023.104675","volume":"93","author":"HJ Jin","year":"2023","unstructured":"Jin HJ, Wang JL, Geng XY et al (2023) CFAP70 is a solid and valuable target for the genetic diagnosis of oligo-astheno-teratozoospermia in infertile men. EBioMedicine 93:104675. https:\/\/doi.org\/10.1016\/j.ebiom.2023.104675","journal-title":"EBioMedicine"},{"key":"5616_CR31","doi-asserted-by":"publisher","first-page":"190","DOI":"10.1016\/j.devcel.2007.07.002","volume":"13","author":"J Kleylein-Sohn","year":"2007","unstructured":"Kleylein-Sohn J, Westendorf J, Le Clech M et al (2007) Plk4-induced centriole biogenesis in human cells. Dev Cell 13:190\u2013202. https:\/\/doi.org\/10.1016\/j.devcel.2007.07.002","journal-title":"Dev Cell"},{"key":"5616_CR32","doi-asserted-by":"publisher","first-page":"1358","DOI":"10.1016\/j.str.2016.06.011","volume":"24","author":"S Kraatz","year":"2016","unstructured":"Kraatz S, Guichard P, Obbineni JM et al (2016) The human centriolar protein CEP135 contains a two-stranded coiled-Coil Domain critical for Microtubule binding. Structure 24:1358\u20131371. https:\/\/doi.org\/10.1016\/j.str.2016.06.011","journal-title":"Structure"},{"key":"5616_CR33","doi-asserted-by":"publisher","first-page":"369","DOI":"10.1038\/s41585-018-0003-3","volume":"15","author":"C Krausz","year":"2018","unstructured":"Krausz C, Riera-Escamilla A (2018) Genetics of male infertility. Nat Rev Urol 15:369\u2013384. https:\/\/doi.org\/10.1038\/s41585-018-0003-3","journal-title":"Nat Rev Urol"},{"key":"5616_CR34","doi-asserted-by":"publisher","first-page":"101","DOI":"10.1186\/1477-7827-8-101","volume":"8","author":"YF Li","year":"2010","unstructured":"Li YF, He W, Kim YH et al (2010) CABYR isoforms expressed in late steps of spermiogenesis bind with AKAPs and ropporin in mouse sperm fibrous sheath. Reprod Biol Endocrinol 8:101. https:\/\/doi.org\/10.1186\/1477-7827-8-101","journal-title":"Reprod Biol Endocrinol"},{"key":"5616_CR35","doi-asserted-by":"publisher","first-page":"684","DOI":"10.1093\/biolre\/ioad180","volume":"110","author":"Z Liang","year":"2024","unstructured":"Liang Z, Dai C, He F et al (2024) AKAP3-mediated type I PKA signaling is required for mouse sperm hyperactivation and fertilitydagger. Biol Reprod 110:684\u2013697. https:\/\/doi.org\/10.1093\/biolre\/ioad180","journal-title":"Biol Reprod"},{"key":"5616_CR36","doi-asserted-by":"publisher","first-page":"460","DOI":"10.1038\/ncpuro0879","volume":"4","author":"LI Lipshultz","year":"2007","unstructured":"Lipshultz LI, Lamb DJ (2007) Risk of transmission of genetic diseases by assisted reproduction. Nat Clin Pract Urol 4:460\u2013461. https:\/\/doi.org\/10.1038\/ncpuro0879","journal-title":"Nat Clin Pract Urol"},{"key":"5616_CR37","doi-asserted-by":"publisher","first-page":"478","DOI":"10.1038\/s41419-021-03755-z","volume":"12","author":"Q Liu","year":"2021","unstructured":"Liu Q, Guo Q, Guo W et al (2021) Loss of CEP70 function affects acrosome biogenesis and flagella formation during spermiogenesis. Cell Death Dis 12:478. https:\/\/doi.org\/10.1038\/s41419-021-03755-z","journal-title":"Cell Death Dis"},{"key":"5616_CR38","doi-asserted-by":"publisher","first-page":"7676","DOI":"10.1038\/ncomms8676","volume":"6","author":"M Marjanovic","year":"2015","unstructured":"Marjanovic M, Sanchez-Huertas C, Terre B et al (2015) CEP63 deficiency promotes p53-dependent microcephaly and reveals a role for the centrosome in meiotic recombination. Nat Commun 6:7676. https:\/\/doi.org\/10.1038\/ncomms8676","journal-title":"Nat Commun"},{"key":"5616_CR39","doi-asserted-by":"publisher","first-page":"731","DOI":"10.1038\/ncb2746","volume":"15","author":"V Marthiens","year":"2013","unstructured":"Marthiens V, Rujano MA, Pennetier C et al (2013) Centrosome amplification causes microcephaly. Nat Cell Biol 15:731\u2013740. https:\/\/doi.org\/10.1038\/ncb2746","journal-title":"Nat Cell Biol"},{"key":"5616_CR40","doi-asserted-by":"publisher","first-page":"113","DOI":"10.5534\/wjmh.180055","volume":"37","author":"AD Martins","year":"2019","unstructured":"Martins AD, Majzoub A, Agawal A (2019) Metabolic syndrome and male fertility. World J Mens Health 37:113\u2013127. https:\/\/doi.org\/10.5534\/wjmh.180055","journal-title":"World J Mens Health"},{"key":"5616_CR41","doi-asserted-by":"publisher","first-page":"1197","DOI":"10.1038\/nm.f.1895","volume":"14","author":"MM Matzuk","year":"2008","unstructured":"Matzuk MM, Lamb DJ (2008) The biology of infertility: research advances and clinical challenges. Nat Med 14:1197\u20131213. https:\/\/doi.org\/10.1038\/nm.f.1895","journal-title":"Nat Med"},{"key":"5616_CR42","doi-asserted-by":"publisher","first-page":"50","DOI":"10.1007\/s11934-018-0804-1","volume":"19","author":"M Mima","year":"2018","unstructured":"Mima M, Greenwald D, Ohlander S (2018) Environmental toxins and male fertility. Curr Urol Rep 19:50. https:\/\/doi.org\/10.1007\/s11934-018-0804-1","journal-title":"Curr Urol Rep"},{"key":"5616_CR43","doi-asserted-by":"publisher","first-page":"375","DOI":"10.1016\/j.freeradbiomed.2020.03.008","volume":"152","author":"MK Panner Selvam","year":"2020","unstructured":"Panner Selvam MK, Agarwal A, Henkel R et al (2020) The effect of oxidative and reductive stress on semen parameters and functions of physiologically normal human spermatozoa. Free Radic Biol Med 152:375\u2013385. https:\/\/doi.org\/10.1016\/j.freeradbiomed.2020.03.008","journal-title":"Free Radic Biol Med"},{"key":"5616_CR44","doi-asserted-by":"publisher","first-page":"217","DOI":"10.1111\/cge.12905","volume":"91","author":"PF Ray","year":"2017","unstructured":"Ray PF, Toure A, Metzler-Guillemain C et al (2017) Genetic abnormalities leading to qualitative defects of sperm morphology or function. Clin Genet 91:217\u2013232. https:\/\/doi.org\/10.1111\/cge.12905","journal-title":"Clin Genet"},{"key":"5616_CR45","doi-asserted-by":"publisher","first-page":"380","DOI":"10.1016\/j.molmed.2020.01.011","volume":"26","author":"A Remo","year":"2020","unstructured":"Remo A, Li X, Schiebel E et al (2020) The Centrosome Linker and its role in Cancer and Genetic disorders. Trends Mol Med 26:380\u2013393. https:\/\/doi.org\/10.1016\/j.molmed.2020.01.011","journal-title":"Trends Mol Med"},{"key":"5616_CR46","doi-asserted-by":"publisher","first-page":"1133222","DOI":"10.3389\/fendo.2023.1133222","volume":"14","author":"T Ruan","year":"2023","unstructured":"Ruan T, Yang Y, Jiang C et al (2023) Identification of biallelic variations of CEP70 in patients with male infertility. Front Endocrinol (Lausanne) 14:1133222. https:\/\/doi.org\/10.3389\/fendo.2023.1133222","journal-title":"Front Endocrinol (Lausanne)"},{"key":"5616_CR47","doi-asserted-by":"publisher","first-page":"264","DOI":"10.1093\/biolre\/ioaa064","volume":"103","author":"AM Salicioni","year":"2020","unstructured":"Salicioni AM, Gervasi MG, Sosnik J et al (2020) Testis-specific serine kinase protein family in male fertility and as targets for non-hormonal male contraceptiondagger. Biol Reprod 103:264\u2013274. https:\/\/doi.org\/10.1093\/biolre\/ioaa064","journal-title":"Biol Reprod"},{"key":"5616_CR48","doi-asserted-by":"publisher","first-page":"181","DOI":"10.1007\/978-3-030-89340-8_9","volume":"1358","author":"P Sengupta","year":"2022","unstructured":"Sengupta P, Roychoudhury S, Nath M et al (2022) Oxidative stress and idiopathic male infertility. Adv Exp Med Biol 1358:181\u2013204. https:\/\/doi.org\/10.1007\/978-3-030-89340-8_9","journal-title":"Adv Exp Med Biol"},{"key":"5616_CR49","doi-asserted-by":"publisher","first-page":"48","DOI":"10.1016\/j.gene.2017.08.033","volume":"633","author":"YW Sha","year":"2017","unstructured":"Sha YW, Xu X, Mei LB et al (2017) A homozygous CEP135 mutation is associated with multiple morphological abnormalities of the sperm flagella (MMAF). Gene 633:48\u201353. https:\/\/doi.org\/10.1016\/j.gene.2017.08.033","journal-title":"Gene"},{"key":"5616_CR50","doi-asserted-by":"publisher","first-page":"433","DOI":"10.1038\/s41467-018-08182-x","volume":"10","author":"Y Shen","year":"2019","unstructured":"Shen Y, Zhang F, Li F et al (2019) Loss-of-function mutations in QRICH2 cause male infertility with multiple morphological abnormalities of the sperm flagella. Nat Commun 10:433. https:\/\/doi.org\/10.1038\/s41467-018-08182-x","journal-title":"Nat Commun"},{"key":"5616_CR51","doi-asserted-by":"publisher","first-page":"4984","DOI":"10.1096\/fj.201701096RR","volume":"32","author":"D Sinha","year":"2018","unstructured":"Sinha D, Kalimutho M, Bowles J et al (2018) Cep55 overexpression causes male-specific sterility in mice by suppressing Foxo1 nuclear retention through sustained activation of PI3K\/Akt signaling. FASEB J 32:4984\u20134999. https:\/\/doi.org\/10.1096\/fj.201701096RR","journal-title":"FASEB J"},{"key":"5616_CR52","doi-asserted-by":"publisher","first-page":"464","DOI":"10.1095\/biolreprod.109.083238","volume":"83","author":"S Tardif","year":"2010","unstructured":"Tardif S, Akrofi AS, Dass B et al (2010) Infertility with impaired zona pellucida adhesion of spermatozoa from mice lacking TauCstF-64. Biol Reprod 83:464\u2013472. https:\/\/doi.org\/10.1095\/biolreprod.109.083238","journal-title":"Biol Reprod"},{"key":"5616_CR53","doi-asserted-by":"publisher","first-page":"811","DOI":"10.1093\/oxfordjournals.humrep.a137433","volume":"6","author":"P Thonneau","year":"1991","unstructured":"Thonneau P, Marchand S, Tallec A et al (1991) Incidence and main causes of infertility in a resident population (1,850,000) of three French regions (1988\u20131989). Hum Reprod 6:811\u2013816. https:\/\/doi.org\/10.1093\/oxfordjournals.humrep.a137433","journal-title":"Hum Reprod"},{"key":"5616_CR54","doi-asserted-by":"publisher","unstructured":"Turner KA, Caswell DL, Mcgrady BM et al (2023) CP110 and CEP135 localize near the proximal and distal centrioles of cattle and human spermatozoa. MicroPubl Biol 2023. https:\/\/doi.org\/10.17912\/micropub.biology.000951","DOI":"10.17912\/micropub.biology.000951"},{"key":"5616_CR55","doi-asserted-by":"publisher","first-page":"12","DOI":"10.1007\/s11825-018-0181-7","volume":"30","author":"F Tuttelmann","year":"2018","unstructured":"Tuttelmann F, Ruckert C, Ropke A (2018) Disorders of spermatogenesis: perspectives for novel genetic diagnostics after 20 years of unchanged routine. Med Genet 30:12\u201320. https:\/\/doi.org\/10.1007\/s11825-018-0181-7","journal-title":"Med Genet"},{"key":"5616_CR56","doi-asserted-by":"publisher","unstructured":"Umer N, Phadke S, Shakeri F et al (2022) PFN4 is required for manchette development and acrosome biogenesis during mouse spermiogenesis. Development 149. https:\/\/doi.org\/10.1242\/dev.200499","DOI":"10.1242\/dev.200499"},{"key":"5616_CR57","doi-asserted-by":"publisher","first-page":"136","DOI":"10.1093\/molehr\/gau097","volume":"21","author":"X Wang","year":"2015","unstructured":"Wang X, Wei Y, Fu G et al (2015) Tssk4 is essential for maintaining the structural integrity of sperm flagellum. Mol Hum Reprod 21:136\u2013145. https:\/\/doi.org\/10.1093\/molehr\/gau097","journal-title":"Mol Hum Reprod"},{"key":"5616_CR58","doi-asserted-by":"publisher","first-page":"4736","DOI":"10.1242\/dev.139626","volume":"143","author":"M Weidemann","year":"2016","unstructured":"Weidemann M, Schuster-Gossler K, Stauber M et al (2016) CFAP157 is a murine downstream effector of FOXJ1 that is specifically required for flagellum morphogenesis and sperm motility. Development 143:4736\u20134748. https:\/\/doi.org\/10.1242\/dev.139626","journal-title":"Development"},{"key":"5616_CR59","doi-asserted-by":"publisher","first-page":"988","DOI":"10.1093\/biolre\/ioaa016","volume":"102","author":"B Wu","year":"2020","unstructured":"Wu B, Gao H, Liu C et al (2020) The coupling apparatus of the sperm head and taildagger. Biol Reprod 102:988\u2013998. https:\/\/doi.org\/10.1093\/biolre\/ioaa016","journal-title":"Biol Reprod"},{"key":"5616_CR60","doi-asserted-by":"publisher","first-page":"91","DOI":"10.1038\/s41585-023-00816-0","volume":"21","author":"MJ Wyrwoll","year":"2024","unstructured":"Wyrwoll MJ, Van Der Heijden GW, Krausz C et al (2024) Improved phenotypic classification of male infertility to promote discovery of genetic causes. Nat Rev Urol 21:91\u2013101. https:\/\/doi.org\/10.1038\/s41585-023-00816-0","journal-title":"Nat Rev Urol"},{"key":"5616_CR61","doi-asserted-by":"publisher","unstructured":"Xu K, Yang L, Zhang L et al (2020) Lack of AKAP3 disrupts integrity of the subcellular structure and proteome of mouse sperm and causes male sterility. Development 147. https:\/\/doi.org\/10.1242\/dev.181057","DOI":"10.1242\/dev.181057"},{"key":"5616_CR62","doi-asserted-by":"publisher","first-page":"750","DOI":"10.1093\/biolre\/ioz223","volume":"102","author":"K Yamatoya","year":"2020","unstructured":"Yamatoya K, Kousaka M, Ito C et al (2020) Cleavage of SPACA1 regulates assembly of sperm-egg membrane fusion machinery in mature spermatozoadagger. Biol Reprod 102:750\u2013757. https:\/\/doi.org\/10.1093\/biolre\/ioz223","journal-title":"Biol Reprod"},{"key":"5616_CR63","doi-asserted-by":"publisher","first-page":"E430","DOI":"10.1073\/pnas.1424648112","volume":"112","author":"S Yuan","year":"2015","unstructured":"Yuan S, Stratton CJ, Bao J et al (2015) Spata6 is required for normal assembly of the sperm connecting piece and tight head-tail conjunction. Proc Natl Acad Sci U S A 112:E430\u2013439. https:\/\/doi.org\/10.1073\/pnas.1424648112","journal-title":"Proc Natl Acad Sci U S A"},{"key":"5616_CR64","doi-asserted-by":"publisher","unstructured":"Zegers-Hochschild F, Adamson GD, Dyer S et al (2017) The International Glossary on Infertility and Fertility Care, 2017. Fertil Steril 108:393\u2013406. https:\/\/doi.org\/10.1016\/j.fertnstert.2017.06.005","DOI":"10.1016\/j.fertnstert.2017.06.005"},{"key":"5616_CR65","doi-asserted-by":"publisher","first-page":"4926","DOI":"10.1038\/s41467-021-25227-w","volume":"12","author":"Y Zhang","year":"2021","unstructured":"Zhang Y, Liu C, Wu B et al (2021) The missing linker between SUN5 and PMFBP1 in sperm head-tail coupling apparatus. Nat Commun 12:4926. https:\/\/doi.org\/10.1038\/s41467-021-25227-w","journal-title":"Nat Commun"},{"key":"5616_CR66","doi-asserted-by":"publisher","first-page":"1395","DOI":"10.1038\/s41467-022-29109-7","volume":"13","author":"X Zhang","year":"2022","unstructured":"Zhang X, Wang L, Ma Y et al (2022) CEP128 is involved in spermatogenesis in humans and mice. Nat Commun 13:1395. https:\/\/doi.org\/10.1038\/s41467-022-29109-7","journal-title":"Nat Commun"},{"key":"5616_CR67","doi-asserted-by":"publisher","first-page":"eabn0968","DOI":"10.1126\/sciadv.abn0968","volume":"8","author":"X Zhang","year":"2022","unstructured":"Zhang X, Zheng R, Liang C et al (2022) Loss-of-function mutations in CEP78 cause male infertility in humans and mice. Sci Adv 8:eabn0968. https:\/\/doi.org\/10.1126\/sciadv.abn0968","journal-title":"Sci Adv"},{"key":"5616_CR68","doi-asserted-by":"publisher","first-page":"e18215","DOI":"10.1111\/jcmm.18215","volume":"28","author":"H Zheng","year":"2024","unstructured":"Zheng H, Gong C, Li J et al (2024) CCDC157 is essential for sperm differentiation and shows oligoasthenoteratozoospermia-related mutations in men. J Cell Mol Med 28:e18215. https:\/\/doi.org\/10.1111\/jcmm.18215","journal-title":"J Cell Mol Med"},{"key":"5616_CR69","doi-asserted-by":"publisher","DOI":"10.1016\/j.jgg.2024.06.008","author":"D Zhou","year":"2024","unstructured":"Zhou D, Wu H, Wang L et al (2024) Deficiency of MFSD6L, an acrosome membrane protein, causes oligoasthenoteratozoospermia in humans and mice. J Genet Genomics. https:\/\/doi.org\/10.1016\/j.jgg.2024.06.008","journal-title":"J Genet Genomics"},{"key":"5616_CR70","doi-asserted-by":"publisher","unstructured":"Zhu T, Zhang Y, Sheng X et al (2023) Absence of CEP78 causes photoreceptor and sperm flagella impairments in mice and a human individual. Elife 12. https:\/\/doi.org\/10.7554\/eLife.76157","DOI":"10.7554\/eLife.76157"}],"container-title":["Cellular and Molecular Life Sciences"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/link.springer.com\/content\/pdf\/10.1007\/s00018-025-05616-w.pdf","content-type":"application\/pdf","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/link.springer.com\/article\/10.1007\/s00018-025-05616-w\/fulltext.html","content-type":"text\/html","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/link.springer.com\/content\/pdf\/10.1007\/s00018-025-05616-w.pdf","content-type":"application\/pdf","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2025,3,17]],"date-time":"2025-03-17T08:48:35Z","timestamp":1742201315000},"score":1,"resource":{"primary":{"URL":"https:\/\/link.springer.com\/10.1007\/s00018-025-05616-w"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2025,3,17]]},"references-count":70,"journal-issue":{"issue":"1","published-online":{"date-parts":[[2025,12]]}},"alternative-id":["5616"],"URL":"https:\/\/doi.org\/10.1007\/s00018-025-05616-w","relation":{},"ISSN":["1420-9071"],"issn-type":[{"value":"1420-9071","type":"electronic"}],"subject":[],"published":{"date-parts":[[2025,3,17]]},"assertion":[{"value":"21 October 2024","order":1,"name":"received","label":"Received","group":{"name":"ArticleHistory","label":"Article History"}},{"value":"10 January 2025","order":2,"name":"revised","label":"Revised","group":{"name":"ArticleHistory","label":"Article History"}},{"value":"4 February 2025","order":3,"name":"accepted","label":"Accepted","group":{"name":"ArticleHistory","label":"Article History"}},{"value":"17 March 2025","order":4,"name":"first_online","label":"First Online","group":{"name":"ArticleHistory","label":"Article History"}},{"order":1,"name":"Ethics","group":{"name":"EthicsHeading","label":"Declarations"}},{"value":"All experimental protocols involving animals were granted approval by the Ethics Committee of Shandong First Medical University (Approval Number: W202211250348, Jinan, China), ensuring strict adherence to Chinese animal protection laws and regulations.","order":2,"name":"Ethics","group":{"name":"EthicsHeading","label":"Ethical approval"}},{"value":"Not applicable.","order":3,"name":"Ethics","group":{"name":"EthicsHeading","label":"Consent for publication"}},{"value":"The authors have declared that no competing interest exists.","order":4,"name":"Ethics","group":{"name":"EthicsHeading","label":"Conflict of interest"}}],"article-number":"117"}}