{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,3,27]],"date-time":"2026-03-27T02:44:22Z","timestamp":1774579462721,"version":"3.50.1"},"reference-count":85,"publisher":"Frontiers Media SA","license":[{"start":{"date-parts":[[2024,2,20]],"date-time":"2024-02-20T00:00:00Z","timestamp":1708387200000},"content-version":"vor","delay-in-days":0,"URL":"https:\/\/creativecommons.org\/licenses\/by\/4.0\/"}],"content-domain":{"domain":["frontiersin.org"],"crossmark-restriction":true},"short-container-title":["Front. Cell Dev. Biol."],"abstract":"<jats:p>Mutations in <jats:italic>PRDM15<\/jats:italic> lead to a syndromic form of holoprosencephaly (HPE) known as the Galloway\u2013Mowat syndrome (GAMOS). While a connection between PRDM15, a zinc finger transcription factor, and WNT\/PCP signaling has been established, there is a critical need to delve deeper into their contributions to early development and GAMOS pathogenesis. We used the South African clawed frog <jats:italic>Xenopus laevis<\/jats:italic> as the vertebrate model organism and observed that <jats:italic>prdm15<\/jats:italic> was enriched in the tissues and organs affected in GAMOS. Furthermore, we generated a morpholino oligonucleotide\u2013mediated <jats:italic>prdm15<\/jats:italic> knockdown model showing that the depletion of Prdm15 leads to abnormal eye, head, and brain development, effectively recapitulating the anterior neural features in GAMOS. An analysis of the underlying molecular basis revealed a reduced expression of key genes associated with eye, head, and brain development. Notably, this reduction could be rescued by the introduction of <jats:italic>wnt4<\/jats:italic> RNA, particularly during the induction of the respective tissues. Mechanistically, our data demonstrate that Prdm15 acts upstream of both canonical and non-canonical Wnt4 signaling during anterior neural development. Our findings describe severe ocular and anterior neural abnormalities upon Prdm15 depletion and elucidate the role of Prdm15 in canonical and non-canonical Wnt4 signaling.<\/jats:p>","DOI":"10.3389\/fcell.2024.1316048","type":"journal-article","created":{"date-parts":[[2024,2,20]],"date-time":"2024-02-20T05:30:11Z","timestamp":1708407011000},"update-policy":"https:\/\/doi.org\/10.3389\/crossmark-policy","source":"Crossref","is-referenced-by-count":5,"title":["Prdm15 acts upstream of Wnt4 signaling in anterior neural development of Xenopus laevis"],"prefix":"10.3389","volume":"12","author":[{"given":"Ernestine","family":"Saumweber","sequence":"first","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Slim","family":"Mzoughi","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Arin","family":"Khadra","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Anja","family":"Werberger","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Sven","family":"Schumann","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Ernesto","family":"Guccione","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Michael J.","family":"Schmeisser","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Susanne J.","family":"K\u00fchl","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"1965","published-online":{"date-parts":[[2024,2,20]]},"reference":[{"key":"B1","doi-asserted-by":"publisher","first-page":"147","DOI":"10.1186\/s12886-018-0820-4","article-title":"Extending the ophthalmological phenotype of Galloway-Mowat syndrome with distinct retinal dysfunction: a report and review of ocular findings","volume":"18","author":"Al-Rakan","year":"2018","journal-title":"BMC Ophthalmol."},{"key":"B2","doi-asserted-by":"publisher","first-page":"167","DOI":"10.1042\/BC20070072","article-title":"Wnt4 inhibits beta-catenin\/TCF signalling by redirecting beta-catenin to the cell membrane","volume":"100","author":"Bernard","year":"2008","journal-title":"Biol. 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