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Of note, germline conditional knockout of Cdyl can lead to asthenoteratozoospermia and infertility in male mice. However, the role of\n                      <jats:italic>CDYL<\/jats:italic>\n                      gene in human fertility remains uncharacterized.\n                    <\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Materials and methods<\/jats:title>\n                    <jats:p>Data were collected through in silico analysis for an infertile man with asthenoteratozoospermia of Han Chinese descent by performing whole\u2010exome sequencing. Light and electron microscopy were used to characterize the sperm cells of the proband, and the pathogenicity of the genetic factors was determined by functional experiments. To overcome fertility problems, intracytoplasmic sperm injections were performed in the couple.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Main results<\/jats:title>\n                    <jats:p>\n                      Here, we recruited an infertile proband, born to first\u2010cousin parents, displaying idiopathic asthenoteratozoospermia. Whole\u2010exome sequencing identified a splicing mutation (c.103+1G&gt;A) in CDYL, recessively cosegregating in the family. In vitro minigene assays demonstrated that the mutation resulted in aberrant alternative splicing. We found that CDYL co\u2010localizes with Ac\u2010tubulin along the flagella of human spermatozoa. In addition, the expression of Ac\u2010tubulin was severely reduced in spermatozoa from the patient with\n                      <jats:italic>CDYL<\/jats:italic>\n                      mutation. Disruption in CDYL results in thin mid\u2010piece related abnormal flagella morphology and decreased sperm motility. The primary manifestation of sperm ultrastructural abnormalities under the electron microscope is primarily characterized by disorder of axonemal protein complex and anulus.\n                    <\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Discussion and conclusion<\/jats:title>\n                    <jats:p>\n                      We demonstrated that a homozygous\n                      <jats:italic>CDYL<\/jats:italic>\n                      splicing mutation specifically induces a decrease in microtubule acetylation, resulting in thin mid\u2010piece related asthenoteratozoospermia, providing a novel marker for genetic counseling and diagnosis of male infertility.\n                    <\/jats:p>\n                  <\/jats:sec>","DOI":"10.1111\/andr.13825","type":"journal-article","created":{"date-parts":[[2025,1,17]],"date-time":"2025-01-17T00:58:32Z","timestamp":1737075512000},"page":"2091-2100","update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":1,"title":["Aberrant acetylation caused by a\n                    <i>CDYL<\/i>\n                    splicing mutation contributes to thin mid\u2010piece related asthenoteratozoospermia and male infertility"],"prefix":"10.1111","volume":"13","author":[{"given":"Fan","family":"Yang","sequence":"first","affiliation":[{"name":"Department of Obstetrics and Gynecology The First Affiliated Hospital of Anhui Medical University  Hefei China"},{"name":"NHC Key Laboratory of Study on Abnormal Gametes and Reproductive Tract (Anhui Medical University)  Hefei China"},{"name":"Engineering Research Center of Biopreservation and Artificial Organs  Hefei 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