{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"institution":[{"name":"bioRxiv"}],"indexed":{"date-parts":[[2026,4,11]],"date-time":"2026-04-11T07:56:37Z","timestamp":1775894197817,"version":"3.50.1"},"posted":{"date-parts":[[2025,3,13]]},"group-title":"Molecular Biology","reference-count":70,"publisher":"openRxiv","license":[{"start":{"date-parts":[[2025,3,13]],"date-time":"2025-03-13T00:00:00Z","timestamp":1741824000000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/creativecommons.org\/licenses\/by-nc\/4.0\/"}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":[],"accepted":{"date-parts":[[2026,1,14]]},"abstract":"<jats:title>Abstract<\/jats:title>\n                <jats:p>\n                  ATRX is a member of the SWI\/SNF family of ATP-dependent chromatin remodellers. In humans, loss of ATRX function leads to ATRX syndrome, a neurodevelopmental disorder. ATRX mutation in human cell lines is associated with multiple phenotypes including activation of the alternative lengthening of telomere (ALT) pathway, upregulation of retrotransposons and increased sensitivity to replication stress. However, the principal role of ATRX and the reason why its mutation causes such diverse phenotypes is currently unclear. To address this, we studied the role of ATRX in the model organism\n                  <jats:italic>Caenorhabditis elegans<\/jats:italic>\n                  . We find that loss of XNP-1, the\n                  <jats:italic>C. elegans<\/jats:italic>\n                  homologue of ATRX, recapitulates many human phenotypes. Loss of XNP-1 causes ectopic activation of germline genes in somatic cells, indicating a loss of cellular identity control. Strikingly, mutation of the germline transcription factor\n                  <jats:italic>gsox-1<\/jats:italic>\n                  suppresses both this misexpression and multiple\n                  <jats:italic>xnp-<\/jats:italic>\n                  1 phenotypes, including developmental delay and telomeric defects. These findings suggest that ectopic germline gene expression underlies the majority of XNP-1-dependent phenotypes, consistent with a role for XNP-1 in maintaining cellular identity, offering insights into the functions of ATRX in humans.\n                <\/jats:p>","DOI":"10.1101\/2025.03.11.641662","type":"posted-content","created":{"date-parts":[[2025,3,14]],"date-time":"2025-03-14T00:40:17Z","timestamp":1741912817000},"source":"Crossref","is-referenced-by-count":0,"title":["ATRX safeguards cellular identity during\n                  <i>C. elegans<\/i>\n                  development"],"prefix":"10.64898","author":[{"given":"Janie","family":"Olver","sequence":"first","affiliation":[{"name":"School of Biology, University of St Andrews, St Andrews, UK"},{"name":"Currently at Rare and Imported Pathogens Laboratory, UK Health Security Agency, Porton Down, Salisbury, UK"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Mariya","family":"Shtumpf","sequence":"additional","affiliation":[{"name":"School of Biology, University of St Andrews, St Andrews, UK"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Karim","family":"Hussain","sequence":"additional","affiliation":[{"name":"School of Biology, University of St Andrews, St Andrews, UK"},{"name":"Currently at Ingenza Ltd, Midlothian, UK"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Stephen","family":"Methot","sequence":"additional","affiliation":[{"name":"Friedrich Miescher Institute for Biomedical Research, Basel, Switzerland"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-0279-6199","authenticated-orcid":false,"given":"Peter","family":"Sarkies","sequence":"additional","affiliation":[{"name":"Department of Biochemistry, University of Oxford, Oxford, 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