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Front Cell Dev Biol 2024 Jan 01;12:1316048. doi: 10.3389/fcell.2024.1316048.
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Prdm15 acts upstream of Wnt4 signaling in anterior neural development of Xenopus laevis.

Saumweber E , Mzoughi S , Khadra A , Werberger A , Schumann S , Guccione E , Schmeisser MJ , Kühl SJ .

Mutations in PRDM15 lead to a syndromic form of holoprosencephaly (HPE) known as the Galloway-Mowat 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 Xenopus laevis as the vertebrate model organism and observed that prdm15 was enriched in the tissues and organs affected in GAMOS. Furthermore, we generated a morpholino oligonucleotide-mediated prdm15 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 wnt4 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.

PubMed ID: 38444828
PMC ID: PMC10912572
Article link: Front Cell Dev Biol

Species referenced: Xenopus tropicalis Xenopus laevis
Genes referenced: alcam celf1 cryba1 dvl1 egr2 emx1 foxd3 fzd3 gli2 jun lrp2 mapk8 osgep otx2 pax2 pax6 pou4f1 prdm15 prox1 rac1 rax rgma rho rho.2 ripk4 rpe six3 smad2 smad4 snai2 sox13 sox3 tgif1 tmprss2 tp53rk tprkb tubb2b twist1 umod.1 vsx1 wnt4
GO keywords: eye development [+]
Antibodies: Neuronal Ab4
Morpholinos: prdm15 MO2

Disease Ontology terms: Galloway-Mowat syndrome [+]

Article Images: [+] show captions