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While LZTS2 is among the most consistently identified DYRK1A\u2010interacting proteins, its roles in embryonic development remain incompletely understood, and its potential contribution to craniofacial development has not been examined.\n                      <jats:italic>Xenopus laevis<\/jats:italic>\n                      was used to test the role of LZTS2 in craniofacial development and its functional relationship with DYRK1A.\n                    <\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Results<\/jats:title>\n                    <jats:p>\n                      Lzts2 and Dyrk1a showed overlapping expression during craniofacial development, with both proteins present in developing facial tissues. Knockdown of Lzts2 disrupted craniofacial morphogenesis and reduced expression of craniofacial genes. These phenotypes closely resembled those caused by decreasing Dyrk1a function. Sub\u2010phenotypic reductions of Lzts2 and Dyrk1a synergized to produce craniofacial defects, while partial reduction of Lzts2 attenuated aspects of the phenotype caused by Dyrk1a overexpression. Comparative analysis of human phenotypes associated with copy number gains of\n                      <jats:italic>LZTS2<\/jats:italic>\n                      and\n                      <jats:italic>DYRK1A<\/jats:italic>\n                      revealed striking overlap, consistent with a potential functional interaction between these genes in humans.\n                    <\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Conclusions<\/jats:title>\n                    <jats:p>These findings identify Lzts2 as a previously unrecognized regulator of craniofacial development and support a functional interaction with Dyrk1a during embryogenesis. Modulating LZTS2 or related regulatory partners may provide a strategy to selectively tune DYRK1A\u2010dependent developmental pathways.<\/jats:p>\n                  <\/jats:sec>","DOI":"10.1002\/dvdy.70192","type":"journal-article","created":{"date-parts":[[2026,10,8]],"date-time":"2026-10-08T13:53:46Z","timestamp":1791467626000},"update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":0,"title":["Evidence for functional convergence between\n                    <scp>LZTS2<\/scp>\n                    and\n                    <scp>DYRK1A<\/scp>\n                    during craniofacial development"],"prefix":"10.1002","author":[{"ORCID":"https:\/\/orcid.org\/0009-0000-2050-4298","authenticated-orcid":false,"given":"Nicole","family":"Cheng","sequence":"first","affiliation":[{"name":"School of Life Sciences and Sustainability Virginia Commonwealth University  Richmond Virginia USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Santiago","family":"Lima","sequence":"additional","affiliation":[{"name":"School of Life Sciences and Sustainability Virginia Commonwealth University  Richmond Virginia USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-9540-597X","authenticated-orcid":false,"given":"Larisa","family":"Litovchick","sequence":"additional","affiliation":[{"name":"Department of Internal Medicine, Division of Hematology, Oncology and Palliative Care Virginia Commonwealth University  Richmond Virginia USA"},{"name":"Massey Cancer Center Virginia Commonwealth University  Richmond Virginia USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-1427-9140","authenticated-orcid":false,"given":"Amanda J. 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