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Mutations in axoneme\u2010associated genes cause male infertility. Dynein arm proteins are essential in sustaining normal axonemes and promote flagellar motility. However, the function of DNAH7 in male fertility in vivo remains unclear. Herein, we showed that <jats:italic>DNAH7<\/jats:italic> disruption in humans results in male infertility, which was characterised by multiple morphological abnormalities of sperm flagella. The axoneme structure of the sperm from a DNAH7\u2010deficient patient revealed the loss of inner dynein arms. Moreover, the mitochondria of the sperm flagella detached and dispersed outside the axoneme, leading to abnormalities in the mitochondrial sheath in the mid\u2010piece region. Live birth was achieved via intracytoplasmic sperm injection. Thus, DNAH7 is critical for axoneme and mitochondrial development in human sperm. These findings further clarify the spectrum of DNAH7 biology and provide new insights for diagnosing infertility and treating patients harbouring <jats:italic>DNAH7<\/jats:italic> mutations.<\/jats:p>","DOI":"10.1111\/cge.14146","type":"journal-article","created":{"date-parts":[[2022,5,11]],"date-time":"2022-05-11T15:49:02Z","timestamp":1652284142000},"page":"130-135","update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":40,"title":["Loss of function mutation in <i>DNAH7<\/i> induces male infertility associated with abnormalities of the sperm flagella and mitochondria in human"],"prefix":"10.1111","volume":"102","author":[{"ORCID":"https:\/\/orcid.org\/0000-0002-9068-9993","authenticated-orcid":false,"given":"Yang","family":"Gao","sequence":"first","affiliation":[{"name":"Department of Obstetrics and Gynecology The First Affiliated Hospital of Anhui Medical University  Hefei Anhui China"},{"name":"NHC Key Laboratory of Study on 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