Dnali1 is required for sperm motility and male fertility in mice

Abstract Background The sperm flagellum is an evolutionarily conserved specialized organelle responsible for sperm motility and male fertility. Deleterious mutations in genes involved in the sperm flagellum assembly can often cause sperm motility defects and male infertility. The murine Dnali1 gene...

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Main Authors: Yiling Zhou, Yaling Wang, Jingwen Chen, Bangguo Wu, Shuyan Tang, Feng Zhang, Chunyu Liu, Lingbo Wang
Format: Article
Language:English
Published: BMC 2023-11-01
Series:Basic and Clinical Andrology
Subjects:
Online Access:https://doi.org/10.1186/s12610-023-00205-y
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author Yiling Zhou
Yaling Wang
Jingwen Chen
Bangguo Wu
Shuyan Tang
Feng Zhang
Chunyu Liu
Lingbo Wang
author_facet Yiling Zhou
Yaling Wang
Jingwen Chen
Bangguo Wu
Shuyan Tang
Feng Zhang
Chunyu Liu
Lingbo Wang
author_sort Yiling Zhou
collection DOAJ
description Abstract Background The sperm flagellum is an evolutionarily conserved specialized organelle responsible for sperm motility and male fertility. Deleterious mutations in genes involved in the sperm flagellum assembly can often cause sperm motility defects and male infertility. The murine Dnali1 gene encodes a protein that is known to interact with the cytoplasmic dynein heavy chain 1. Results A Dnali1-mutated mouse model was generated by inducing a nonsense mutation in the Dnali1 gene. The Dnali1-mutated male mice presented impaired sperm motility and were completely infertile. Although no obviously abnormal sperm morphology was observed in Dnali1-mutated male mice, the ultrastructural structure of sperm flagellum was disrupted, displaying as an asymmetrical distribution of the longitudinal columns (LCs). Notably, infertile Dnali1-mutated male mice were able to obtain offspring via ICSI. Conclusions Our results uncover a role of DNALI1 in sperm motility and male fertility in mice, and demonstrate that ICSI overcomes Dnali1-associated male infertility, thus providing guidance for the diagnosis and genetic counseling of DNALI1-associated human infertility.
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spelling doaj.art-26553b1549204163bc353458e4b55ca42023-11-26T12:35:43ZengBMCBasic and Clinical Andrology2051-41902023-11-0133111110.1186/s12610-023-00205-yDnali1 is required for sperm motility and male fertility in miceYiling Zhou0Yaling Wang1Jingwen Chen2Bangguo Wu3Shuyan Tang4Feng Zhang5Chunyu Liu6Lingbo Wang7Shanghai Key Laboratory of Metabolic Remodeling and Health, Institute of Metabolism and Integrative Biology, Institute of Reproduction and Development, Obstetrics and Gynecology Hospital, Fudan UniversityShanghai Key Laboratory of Metabolic Remodeling and Health, Institute of Metabolism and Integrative Biology, Institute of Reproduction and Development, Obstetrics and Gynecology Hospital, Fudan UniversityShanghai Key Laboratory of Metabolic Remodeling and Health, Institute of Metabolism and Integrative Biology, Institute of Reproduction and Development, Obstetrics and Gynecology Hospital, Fudan UniversityShanghai Key Laboratory of Metabolic Remodeling and Health, Institute of Metabolism and Integrative Biology, Institute of Reproduction and Development, Obstetrics and Gynecology Hospital, Fudan UniversityShanghai Key Laboratory of Metabolic Remodeling and Health, Institute of Metabolism and Integrative Biology, Institute of Reproduction and Development, Obstetrics and Gynecology Hospital, Fudan UniversityShanghai Key Laboratory of Metabolic Remodeling and Health, Institute of Metabolism and Integrative Biology, Institute of Reproduction and Development, Obstetrics and Gynecology Hospital, Fudan UniversityShanghai Key Laboratory of Metabolic Remodeling and Health, Institute of Metabolism and Integrative Biology, Institute of Reproduction and Development, Obstetrics and Gynecology Hospital, Fudan UniversityShanghai Key Laboratory of Metabolic Remodeling and Health, Institute of Metabolism and Integrative Biology, Institute of Reproduction and Development, Obstetrics and Gynecology Hospital, Fudan UniversityAbstract Background The sperm flagellum is an evolutionarily conserved specialized organelle responsible for sperm motility and male fertility. Deleterious mutations in genes involved in the sperm flagellum assembly can often cause sperm motility defects and male infertility. The murine Dnali1 gene encodes a protein that is known to interact with the cytoplasmic dynein heavy chain 1. Results A Dnali1-mutated mouse model was generated by inducing a nonsense mutation in the Dnali1 gene. The Dnali1-mutated male mice presented impaired sperm motility and were completely infertile. Although no obviously abnormal sperm morphology was observed in Dnali1-mutated male mice, the ultrastructural structure of sperm flagellum was disrupted, displaying as an asymmetrical distribution of the longitudinal columns (LCs). Notably, infertile Dnali1-mutated male mice were able to obtain offspring via ICSI. Conclusions Our results uncover a role of DNALI1 in sperm motility and male fertility in mice, and demonstrate that ICSI overcomes Dnali1-associated male infertility, thus providing guidance for the diagnosis and genetic counseling of DNALI1-associated human infertility.https://doi.org/10.1186/s12610-023-00205-yInfertilitySperm motilityAsthenozoospermiaSperm flagellumICSI
spellingShingle Yiling Zhou
Yaling Wang
Jingwen Chen
Bangguo Wu
Shuyan Tang
Feng Zhang
Chunyu Liu
Lingbo Wang
Dnali1 is required for sperm motility and male fertility in mice
Basic and Clinical Andrology
Infertility
Sperm motility
Asthenozoospermia
Sperm flagellum
ICSI
title Dnali1 is required for sperm motility and male fertility in mice
title_full Dnali1 is required for sperm motility and male fertility in mice
title_fullStr Dnali1 is required for sperm motility and male fertility in mice
title_full_unstemmed Dnali1 is required for sperm motility and male fertility in mice
title_short Dnali1 is required for sperm motility and male fertility in mice
title_sort dnali1 is required for sperm motility and male fertility in mice
topic Infertility
Sperm motility
Asthenozoospermia
Sperm flagellum
ICSI
url https://doi.org/10.1186/s12610-023-00205-y
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