Knockout of cyclin-dependent kinases 8 and 19 leads to depletion of cyclin C and suppresses spermatogenesis and male fertility in mice

CDK8 and CDK19 paralogs are regulatory kinases associated with the transcriptional Mediator complex. We have generated mice with the systemic inducible Cdk8 knockout on the background of Cdk19 constitutive knockout. Cdk8/19 double knockout (iDKO) males, but not single Cdk8 or Cdk19 KO, had an atroph...

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Main Authors: Alexandra V Bruter, Ekaterina A Varlamova, Nina I Stavskaya, Zoia G Antysheva, Vasily N Manskikh, Anna V Tvorogova, Diana S Korshunova, Alvina I Khamidullina, Marina V Utkina, Viktor P Bogdanov, Iuliia P Baikova, Alyona I Nikiforova, Eugene A Albert, Denis O Maksimov, Jing Li, Mengqian Chen, Gary P Schools, Alexey V Feoktistov, Alexander A Shtil, Igor B Roninson, Vladislav A Mogila, Yulia Y Silaeva, Victor V Tatarskiy
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Language:English
Published: eLife Sciences Publications Ltd 2025-04-01
Series:eLife
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Online Access:https://elifesciences.org/articles/96465
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author Alexandra V Bruter
Ekaterina A Varlamova
Nina I Stavskaya
Zoia G Antysheva
Vasily N Manskikh
Anna V Tvorogova
Diana S Korshunova
Alvina I Khamidullina
Marina V Utkina
Viktor P Bogdanov
Iuliia P Baikova
Alyona I Nikiforova
Eugene A Albert
Denis O Maksimov
Jing Li
Mengqian Chen
Gary P Schools
Alexey V Feoktistov
Alexander A Shtil
Igor B Roninson
Vladislav A Mogila
Yulia Y Silaeva
Victor V Tatarskiy
author_facet Alexandra V Bruter
Ekaterina A Varlamova
Nina I Stavskaya
Zoia G Antysheva
Vasily N Manskikh
Anna V Tvorogova
Diana S Korshunova
Alvina I Khamidullina
Marina V Utkina
Viktor P Bogdanov
Iuliia P Baikova
Alyona I Nikiforova
Eugene A Albert
Denis O Maksimov
Jing Li
Mengqian Chen
Gary P Schools
Alexey V Feoktistov
Alexander A Shtil
Igor B Roninson
Vladislav A Mogila
Yulia Y Silaeva
Victor V Tatarskiy
author_sort Alexandra V Bruter
collection DOAJ
description CDK8 and CDK19 paralogs are regulatory kinases associated with the transcriptional Mediator complex. We have generated mice with the systemic inducible Cdk8 knockout on the background of Cdk19 constitutive knockout. Cdk8/19 double knockout (iDKO) males, but not single Cdk8 or Cdk19 KO, had an atrophic reproductive system and were infertile. The iDKO males lacked postmeiotic spermatids and spermatocytes after meiosis I pachytene. Testosterone levels were decreased whereas the amounts of the luteinizing hormone were unchanged. Single-cell RNA sequencing showed marked differences in the expression of steroidogenic genes (such as Cyp17a1, Star, and Fads) in Leydig cells concomitant with alterations in Sertoli cells and spermatocytes, and were likely associated with an impaired synthesis of steroids. Star and Fads were also downregulated in cultured Leydig cells after iDKO. The treatment of primary Leydig cell culture with a CDK8/19 inhibitor did not induce the same changes in gene expression as iDKO, and a prolonged treatment of mice with a CDK8/19 inhibitor did not affect the size of testes. iDKO, in contrast to the single knockouts or treatment with a CDK8/19 kinase inhibitor, led to depletion of cyclin C (CCNC), the binding partner of CDK8/19 that has been implicated in CDK8/19-independent functions. This suggests that the observed phenotype was likely mediated through kinase-independent activities of CDK8/19, such as CCNC stabilization.
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spelling doaj-art-9f6588e41adb46dba88f7d1c24c55da42025-08-20T01:55:48ZengeLife Sciences Publications LtdeLife2050-084X2025-04-011310.7554/eLife.96465Knockout of cyclin-dependent kinases 8 and 19 leads to depletion of cyclin C and suppresses spermatogenesis and male fertility in miceAlexandra V Bruter0Ekaterina A Varlamova1https://orcid.org/0000-0002-8451-1216Nina I Stavskaya2Zoia G Antysheva3Vasily N Manskikh4Anna V Tvorogova5Diana S Korshunova6Alvina I Khamidullina7Marina V Utkina8Viktor P Bogdanov9Iuliia P Baikova10Alyona I Nikiforova11Eugene A Albert12Denis O Maksimov13Jing Li14Mengqian Chen15https://orcid.org/0000-0003-1706-9509Gary P Schools16Alexey V Feoktistov17Alexander A Shtil18Igor B Roninson19https://orcid.org/0000-0002-9211-1327Vladislav A Mogila20https://orcid.org/0000-0003-2398-0331Yulia Y Silaeva21Victor V Tatarskiy22https://orcid.org/0000-0002-9080-5683Center for Precision Genome Editing and Genetic Technologies for Biomedicine, Institute of Gene Biology, Russian Academy of Sciences, Moscow, Russian Federation; Blokhin National Medical Research Center of Oncology, Moscow, Russian FederationCenter for Precision Genome Editing and Genetic Technologies for Biomedicine, Institute of Gene Biology, Russian Academy of Sciences, Moscow, Russian FederationInstitute of Gene Biology, Russian Academy of Sciences, Moscow, Russian FederationInstitute of Gene Biology, Russian Academy of Sciences, Moscow, Russian FederationBelozersky Institute of Physico-Chemical Biology, Lomonosov Moscow State University, Moscow, Russian FederationCenter for Precision Genome Editing and Genetic Technologies for Biomedicine, Institute of Gene Biology, Russian Academy of Sciences, Moscow, Russian FederationCenter for Precision Genome Editing and Genetic Technologies for Biomedicine, Institute of Gene Biology, Russian Academy of Sciences, Moscow, Russian FederationCenter for Precision Genome Editing and Genetic Technologies for Biomedicine, Institute of Gene Biology, Russian Academy of Sciences, Moscow, Russian Federation; Institute of Gene Biology, Russian Academy of Sciences, Moscow, Russian FederationEndocrinology Research Centre, Moscow, Russian FederationLife Sciences Research Center, Moscow Institute of Physics and Technology, Dolgoprudny, Russian FederationCenter for Precision Genome Editing and Genetic Technologies for Biomedicine, Institute of Gene Biology, Russian Academy of Sciences, Moscow, Russian FederationInstitute of Mitoengineering MSU, Moscow, Russian FederationLife Sciences Research Center, Moscow Institute of Physics and Technology, Dolgoprudny, Russian FederationLife Sciences Research Center, Moscow Institute of Physics and Technology, Dolgoprudny, Russian FederationDepartment of Drug Discovery and Biomedical Sciences, University of South Carolina, Columbia, United StatesDepartment of Drug Discovery and Biomedical Sciences, University of South Carolina, Columbia, United States; Senex Biotechnology, Inc, Columbia, United StatesDepartment of Drug Discovery and Biomedical Sciences, University of South Carolina, Columbia, United StatesThe Engelhardt Institute of Molecular Biology, Russian Academy of Sciences, Moscow, Russian FederationBlokhin National Medical Research Center of Oncology, Moscow, Russian Federation; Institute of Gene Biology, Russian Academy of Sciences, Moscow, Russian FederationDepartment of Drug Discovery and Biomedical Sciences, University of South Carolina, Columbia, United StatesInstitute of Gene Biology, Russian Academy of Sciences, Moscow, Russian Federation; Department of Drug Discovery and Biomedical Sciences, University of South Carolina, Columbia, United StatesCenter for Precision Genome Editing and Genetic Technologies for Biomedicine, Institute of Gene Biology, Russian Academy of Sciences, Moscow, Russian Federation; Institute of Gene Biology, Russian Academy of Sciences, Moscow, Russian FederationCenter for Precision Genome Editing and Genetic Technologies for Biomedicine, Institute of Gene Biology, Russian Academy of Sciences, Moscow, Russian Federation; Institute of Gene Biology, Russian Academy of Sciences, Moscow, Russian FederationCDK8 and CDK19 paralogs are regulatory kinases associated with the transcriptional Mediator complex. We have generated mice with the systemic inducible Cdk8 knockout on the background of Cdk19 constitutive knockout. Cdk8/19 double knockout (iDKO) males, but not single Cdk8 or Cdk19 KO, had an atrophic reproductive system and were infertile. The iDKO males lacked postmeiotic spermatids and spermatocytes after meiosis I pachytene. Testosterone levels were decreased whereas the amounts of the luteinizing hormone were unchanged. Single-cell RNA sequencing showed marked differences in the expression of steroidogenic genes (such as Cyp17a1, Star, and Fads) in Leydig cells concomitant with alterations in Sertoli cells and spermatocytes, and were likely associated with an impaired synthesis of steroids. Star and Fads were also downregulated in cultured Leydig cells after iDKO. The treatment of primary Leydig cell culture with a CDK8/19 inhibitor did not induce the same changes in gene expression as iDKO, and a prolonged treatment of mice with a CDK8/19 inhibitor did not affect the size of testes. iDKO, in contrast to the single knockouts or treatment with a CDK8/19 kinase inhibitor, led to depletion of cyclin C (CCNC), the binding partner of CDK8/19 that has been implicated in CDK8/19-independent functions. This suggests that the observed phenotype was likely mediated through kinase-independent activities of CDK8/19, such as CCNC stabilization.https://elifesciences.org/articles/96465CDK8CDK19Spermatogenesis
spellingShingle Alexandra V Bruter
Ekaterina A Varlamova
Nina I Stavskaya
Zoia G Antysheva
Vasily N Manskikh
Anna V Tvorogova
Diana S Korshunova
Alvina I Khamidullina
Marina V Utkina
Viktor P Bogdanov
Iuliia P Baikova
Alyona I Nikiforova
Eugene A Albert
Denis O Maksimov
Jing Li
Mengqian Chen
Gary P Schools
Alexey V Feoktistov
Alexander A Shtil
Igor B Roninson
Vladislav A Mogila
Yulia Y Silaeva
Victor V Tatarskiy
Knockout of cyclin-dependent kinases 8 and 19 leads to depletion of cyclin C and suppresses spermatogenesis and male fertility in mice
eLife
CDK8
CDK19
Spermatogenesis
title Knockout of cyclin-dependent kinases 8 and 19 leads to depletion of cyclin C and suppresses spermatogenesis and male fertility in mice
title_full Knockout of cyclin-dependent kinases 8 and 19 leads to depletion of cyclin C and suppresses spermatogenesis and male fertility in mice
title_fullStr Knockout of cyclin-dependent kinases 8 and 19 leads to depletion of cyclin C and suppresses spermatogenesis and male fertility in mice
title_full_unstemmed Knockout of cyclin-dependent kinases 8 and 19 leads to depletion of cyclin C and suppresses spermatogenesis and male fertility in mice
title_short Knockout of cyclin-dependent kinases 8 and 19 leads to depletion of cyclin C and suppresses spermatogenesis and male fertility in mice
title_sort knockout of cyclin dependent kinases 8 and 19 leads to depletion of cyclin c and suppresses spermatogenesis and male fertility in mice
topic CDK8
CDK19
Spermatogenesis
url https://elifesciences.org/articles/96465
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