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PMID:18582859

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Citation

Dakubo, GD, Mazerolle, C, Furimsky, M, Yu, C, St-Jacques, B, McMahon, AP and Wallace, VA (2008) Indian hedgehog signaling from endothelial cells is required for sclera and retinal pigment epithelium development in the mouse eye. Dev. Biol. 320:242-55

Abstract

The development of extraocular orbital structures, in particular the choroid and sclera, is regulated by a complex series of interactions between neuroectoderm, neural crest and mesoderm derivatives, although in many instances the signals that mediate these interactions are not known. In this study we have investigated the function of Indian hedgehog (Ihh) in the developing mammalian eye. We show that Ihh is expressed in a population of non-pigmented cells located in the developing choroid adjacent to the RPE. The analysis of Hh mutant mice demonstrates that the RPE and developing scleral mesenchyme are direct targets of Ihh signaling and that Ihh is required for the normal pigmentation pattern of the RPE and the condensation of mesenchymal cells to form the sclera. Our findings also indicate that Ihh signals indirectly to promote proliferation and photoreceptor specification in the neural retina. This study identifies Ihh as a novel choroid-derived signal that regulates RPE, sclera and neural retina development.

Links

PubMed Online version:10.1016/j.ydbio.2008.05.528

Keywords

Animals; Biological Markers/metabolism; Choroid/embryology; Endothelial Cells/metabolism; Gene Expression Regulation, Developmental; Hedgehog Proteins/genetics; Hedgehog Proteins/metabolism; Homeodomain Proteins/metabolism; Hypopigmentation/pathology; Kruppel-Like Transcription Factors; Mesoderm/embryology; Mesoderm/metabolism; Mice; Mice, Inbred C57BL; Mice, Knockout; Mutation/genetics; Orbit/metabolism; Pigment Epithelium of Eye/abnormalities; Pigment Epithelium of Eye/embryology; Pigment Epithelium of Eye/ultrastructure; Retina/embryology; Retina/pathology; Sclera/abnormalities; Sclera/embryology; Sclera/ultrastructure; Signal Transduction; Trans-Activators/metabolism

Significance

Annotations

Gene product Qualifier GO Term Evidence Code with/from Aspect Extension Notes Status

MOUSE:IHH

acts_upstream_of_or_within

GO:0002053: positive regulation of mesenchymal cell proliferation

ECO:0000315: mutant phenotype evidence used in manual assertion

MGI:MGI:1934258

P

Seeded From UniProt

complete

MOUSE:IHH

acts_upstream_of_or_within

GO:0001708: cell fate specification

ECO:0000315: mutant phenotype evidence used in manual assertion

MGI:MGI:1934258

P

Seeded From UniProt

complete

MOUSE:IHH

acts_upstream_of_or_within

GO:0008284: positive regulation of cell population proliferation

ECO:0000315: mutant phenotype evidence used in manual assertion

MGI:MGI:1934258

P

Seeded From UniProt

complete

MOUSE:IHH

acts_upstream_of_or_within

GO:0003406: retinal pigment epithelium development

ECO:0000315: mutant phenotype evidence used in manual assertion

MGI:MGI:1934258

P

Seeded From UniProt

complete

MOUSE:IHH

acts_upstream_of_or_within

GO:0003382: epithelial cell morphogenesis

ECO:0000315: mutant phenotype evidence used in manual assertion

MGI:MGI:1934258

P

Seeded From UniProt

complete

MOUSE:IHH

acts_upstream_of_or_within

GO:0009880: embryonic pattern specification

ECO:0000315: mutant phenotype evidence used in manual assertion

MGI:MGI:1934258

P

Seeded From UniProt

complete

MOUSE:IHH

acts_upstream_of_or_within

GO:0048596: embryonic camera-type eye morphogenesis

ECO:0000315: mutant phenotype evidence used in manual assertion

MGI:MGI:1934258

P

Seeded From UniProt

complete

MOUSE:IHH

acts_upstream_of_or_within

GO:0060220: camera-type eye photoreceptor cell fate commitment

ECO:0000315: mutant phenotype evidence used in manual assertion

MGI:MGI:1934258

P

Seeded From UniProt

complete

MOUSE:IHH

acts_upstream_of_or_within

GO:0043010: camera-type eye development

ECO:0000315: mutant phenotype evidence used in manual assertion

MGI:MGI:1934258

P

Seeded From UniProt

complete

MOUSE:IHH

acts_upstream_of_or_within

GO:0090136: epithelial cell-cell adhesion

ECO:0000315: mutant phenotype evidence used in manual assertion

MGI:MGI:1934258

P

Seeded From UniProt

complete

MOUSE:SHH

acts_upstream_of_or_within

GO:0043010: camera-type eye development

ECO:0000316: genetic interaction evidence used in manual assertion

MGI:MGI:95729

P

Seeded From UniProt

complete

MOUSE:GLI3

acts_upstream_of_or_within

GO:0043010: camera-type eye development

ECO:0000316: genetic interaction evidence used in manual assertion

MGI:MGI:98297

P

Seeded From UniProt

complete


See also

References

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