biostudies-literatureLu YKAKENHIBill & Melinda Gates Foundation - Grand Challenges Explorations InitiativeEunice Kennedy Shriver National Institute of Child Health and Human DevelopmentNICHD NIH HHSLotte FoundationJapan Agency for Medical Research and DevelopmentBill and Melinda Gates Foundation501-511https://www.ebi.ac.uk/biostudies/studies/S-EPMC6735960biostudies-literatureUnknown101(2)More than 1000 genes are predicted to be predominantly expressed in mouse testis, yet many of them remain unstudied in terms of their roles in spermatogenesis and sperm function and their essentiality in male reproduction. Since individually indispensable factors can provide important implications for the diagnosis of genetically related idiopathic male infertility and may serve as candidate targets for the development of nonhormonal male contraceptives, our laboratories continuously analyze the functions of testis-enriched genes in vivo by generating knockout mouse lines using the CRISPR/Cas9 system. The dispensability of genes in male reproduction is easily determined by examining the fecundity of knockout males. During our large-scale screening of essential factors, we knocked out 30 genes that have a strong bias of expression in the testis and are mostly conserved in mammalian species including human. Fertility tests reveal that the mutant males exhibited normal fecundity, suggesting these genes are individually dispensable for male reproduction. Since such functionally redundant genes are of diminished biological and clinical significance, we believe that it is crucial to disseminate this list of genes, along with their phenotypic information, to the scientific community to avoid unnecessary expenditure of time and research funds and duplication of efforts by other laboratories.Biology of reproductionCRISPR/Cas9-mediated genome editing reveals 30 testis-enriched genes dispensable for male fertility in mice†.PMC6735960JP18K14715R01HD088412JP17J09669JP17K17852OPP1160866R01 HD095341JP16H06276JP18K14612JPA18J116750JP18K16735R01HD095341JP15H05573JP17H01394R01 HD088412JP17H06840P01 HD087157JP18gm5010001P01HD087157JP16KK0180JP17H04987Young SAMFujihara YLu YMatsumura TShimada KZhang QKodani MIsotani AAitken RJOura SIkawa MRobertson MJCastaneda JMOji AKiyozumi DCoarfa CSakurai NMiyata HHuddleston CATobita TOkabe MLarasati TGarcia TXNoda TMatzuk MMfalseCRISPR/Cas9-mediated genome editing reveals 30 testis-enriched genes dispensable for male fertility in mice†.More than 1000 genes are predicted to be predominantly expressed in mouse testis, yet many of them remain unstudied in terms of their roles in spermatogenesis and sperm function and their essentiality in male reproduction. Since individually indispensable factors can provide important implications for the diagnosis of genetically related idiopathic male infertility and may serve as candidate targets for the development of nonhormonal male contraceptives, our laboratories continuously analyze the functions of testis-enriched genes in vivo by generating knockout mouse lines using the CRISPR/Cas9 system. The dispensability of genes in male reproduction is easily determined by examining the fecundity of knockout males. During our large-scale screening of essential factors, we knocked out 30 genes that have a strong bias of expression in the testis and are mostly conserved in mammalian species including human. Fertility tests reveal that the mutant males exhibited normal fecundity, suggesting these genes are individually dispensable for male reproduction. Since such functionally redundant genes are of diminished biological and clinical significance, we believe that it is crucial to disseminate this list of genes, along with their phenotypic information, to the scientific community to avoid unnecessary expenditure of time and research funds and duplication of efforts by other laboratories.2019-01-01T00:00:00Z2019 Aug2024-11-12T12:05:15.172Z2019-09-24T07:09:37ZS-EPMC67359603120141910.1093/biolre/ioz103