Mouse Wnt8B is expressed in the developing forebrain and maps to Chromosome 19

Richardson, Melville; Redmond, D. L.; Watson, Christine J.; Mason, John O.

doi:10.1007/s003359901115

Cited by 27 publications

(22 citation statements)

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“…Similar to Wnt8b, Rspo2 activity is also normally detected in the diencephalic region of developing mouse embryos (Nam et al, 2007; Richardson et al, 1999). Accordingly, it is also possible that similar to Wnt8b , expression of Rspo2 should also be maintained outside the putative eye territory for neuroretina differentiation to take place.…”

Section: Resultsmentioning

confidence: 96%

“…Accordingly, taking into account their expression profiles and reported phenotypic alterations we decided to initially focus our analysis on Rspo2 . It is important to mention that in the developing mouse brain this gene is expressed in the diencephalic region overlapping that of Wnt8b (Nam et al, 2007; Richardson et al, 1999), a gene whose expression is negatively regulated by Six3 during neuroretina specification (Liu et al, 2010). …”

Section: Resultsmentioning

confidence: 99%

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An Eye Organoid Approach Identifies Six3 Suppression of R-spondin 2 as a Critical Step in Mouse Neuroretina Differentiation

et al. 2017

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SUMMARY Recent advances in self-organizing, 3-dimensional tissue cultures of embryonic stem cells (ESCs) and induced pluripotent stem cells (iPSCs) provided an in vitro model that recapitulates many aspects of the in vivo developmental steps. Using Rax-GFP expressing ESCs, newly generated Six3−/− iPSCs and conditional null Six3delta/f;Rax-Cre ESCs we identified Six3 repression of R-spondin 2 (Rspo2) as a required step during optic vesicle morphogenesis and neuroretina differentiation. We validated these results in vivo by showing that transient ectopic expression of Rspo2 in the anterior neural plate of transgenic mouse embryos was sufficient to inhibit neuroretina differentiation. Additionally, using a chimeric eye organoid assay we determined that Six3-null cells exert a non-cell autonomous repressive effect during optic vesicle formation and neuroretina differentiation. Our results further validate the organoid culture system as a reliable and fast alternative to identify and evaluate genes involved in eye morphogenesis and neuroretina differentiation in vivo.

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Section: Resultsmentioning

confidence: 96%

Section: Resultsmentioning

confidence: 99%

An Eye Organoid Approach Identifies Six3 Suppression of R-spondin 2 as a Critical Step in Mouse Neuroretina Differentiation

et al. 2017

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“…The Wnt ligand involved here could be Wnt8b, a specific marker of the MAM in all vertebrates (Cui et al, 1995;Hollyday et al, 1995;Kelly et al, 1995;Lako et al, 1998;Richardson et al, 1999;Garda et al, 2002;Houart et al, 2002). Wnt8b downregulates Foxb1 in the MAM of zebrafish (Kim et al, 2002), controls mammillary neurogenesis (Lee et al, 2006), and could be a local organizer (Erter et al, 2001;Houart et al, 2002).…”

Section: Neural Shh In the Pth And Emtmentioning

confidence: 99%

Role of NeuroepithelialSonic hedgehogin Hypothalamic Patterning

et al. 2009

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The hypothalamus is a region of the diencephalon with particularly complex patterning. Sonic hedgehog (Shh), encoding a protein with key developmental roles, shows a peculiar and dynamic diencephalic expression pattern. Here, we use transgenic strategies and in vitro experiments to test the hypothesis that Shh expressed in the diencephalic neuroepithelium (neural Shh) coordinates tissue growth and patterning in the hypothalamus. Our results show that neural Shh coordinates anteroposterior and dorsoventral patterning in the hypothalamus and in the diencephalon-telencephalon junction. Neural Shh also coordinates mediolateral hypothalamic patterning, since it is necessary for the lateral hypothalamus to attain proper size and is required for the specification of hypocretin/orexin cells. Finally, neural Shh is necessary to maintain expression of differentiation markers including survival factor Foxb1.

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“…Similarly to Pax6 and Ngn2 , Lhx2 expression has lost its expression gradient, but is still maintained in the malformed cortex, and not ectopically expressed in the protruded dorsomedial structure in severely affected Ofd1 Δ 4–5/+ mutant embryos (red arrows in Figure 2F). Finally, we assessed the expression of Wnt8b , which is expressed in dorsomedial cortical structures, but not in the cortical primordium (Figure 2G) [33], [34]. Severely affected O fd1 Δ 4–5/+ mutant embryos showed only a slight expansion of Wnt8b expression dorsomedially (red arrows in Figure 2H), indicating that the abnormally protruded dorsomedial structure in mutant embryos has not a cortical origin.…”

Section: Resultsmentioning

confidence: 99%

Ofd1 Controls Dorso-Ventral Patterning and Axoneme Elongation during Embryonic Brain Development

et al. 2012

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Oral-facial-digital type I syndrome (OFDI) is a human X-linked dominant-male-lethal developmental disorder caused by mutations in the OFD1 gene. Similar to other inherited disorders associated to ciliary dysfunction OFD type I patients display neurological abnormalities. We characterized the neuronal phenotype that results from Ofd1 inactivation in early phases of mouse embryonic development and at post-natal stages. We determined that Ofd1 plays a crucial role in forebrain development, and in particular, in the control of dorso-ventral patterning and early corticogenesis. We observed abnormal activation of Sonic hedgehog (Shh), a major pathway modulating brain development. Ultrastructural studies demonstrated that early Ofd1 inactivation results in the absence of ciliary axonemes despite the presence of mature basal bodies that are correctly orientated and docked. Ofd1 inducible-mediated inactivation at birth does not affect ciliogenesis in the cortex, suggesting a developmental stage-dependent role for a basal body protein in ciliogenesis. Moreover, we showed defects in cytoskeletal organization and apical-basal polarity in Ofd1 mutant embryos, most likely due to lack of ciliary axonemes. Thus, the present study identifies Ofd1 as a developmental disease gene that is critical for forebrain development and ciliogenesis in embryonic life, and indicates that Ofd1 functions after docking and before elaboration of the axoneme in vivo.

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Mouse Wnt8B is expressed in the developing forebrain and maps to Chromosome 19

Cited by 27 publications

References 0 publications

An Eye Organoid Approach Identifies Six3 Suppression of R-spondin 2 as a Critical Step in Mouse Neuroretina Differentiation

An Eye Organoid Approach Identifies Six3 Suppression of R-spondin 2 as a Critical Step in Mouse Neuroretina Differentiation

Role of NeuroepithelialSonic hedgehogin Hypothalamic Patterning

Ofd1 Controls Dorso-Ventral Patterning and Axoneme Elongation during Embryonic Brain Development

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