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PMID:18836173

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Citation

Nuckels, RJ, Ng, A, Darland, T and Gross, JM (2009) The vacuolar-ATPase complex regulates retinoblast proliferation and survival, photoreceptor morphogenesis, and pigmentation in the zebrafish eye. Invest. Ophthalmol. Vis. Sci. 50:893-905

Abstract

The vacuolar (v)-ATPase complex is a key regulator of the acidification of endosomes, lysosomes, and the luminal compartments of several cell types, tissues, and organs; however, little is know about the in vivo function of the v-ATPase complex or its roles during eye development. This study was conducted to characterize ocular defects in five zebrafish mutants in which core components of the v-ATPase complex were affected (atp6v1h, atp6v1f, atp6v1e1, atp6v0c, and atp6v0d1), as well as a sixth mutant in which a v-ATPase associated protein (atp6ap1) was affected.

Links

PubMed Online version:10.1167/iovs.08-2743

Keywords

Albinism, Oculocutaneous/genetics; Albinism, Oculocutaneous/pathology; Animals; Bromodeoxyuridine/metabolism; Cell Cycle; Cell Proliferation; Cell Survival/physiology; DNA/biosynthesis; Embryo, Nonmammalian/pathology; Fluorescent Antibody Technique, Indirect; Genes, Recessive; In Situ Nick-End Labeling; Melanosomes/pathology; Microphthalmos/genetics; Microphthalmos/pathology; Microscopy, Confocal; Morphogenesis; Photoreceptor Cells, Vertebrate/pathology; Retina/embryology; Retina/ultrastructure; Retinal Pigment Epithelium/pathology; Reverse Transcriptase Polymerase Chain Reaction; Stem Cells/pathology; Vacuolar Proton-Translocating ATPases/physiology; Zebrafish; Zebrafish Proteins/metabolism

Significance

Annotations

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

DANRE:A2AR67

involved_in

GO:0008057: eye pigment granule organization

ECO:0000315: mutant phenotype evidence used in manual assertion

ZFIN:ZDB-GENO-100518-3

P

Seeded From UniProt

complete

DANRE:A2AR67

involved_in

GO:0032438: melanosome organization

ECO:0000315: mutant phenotype evidence used in manual assertion

ZFIN:ZDB-GENO-100518-3

P

Seeded From UniProt

complete

DANRE:A2AR67

involved_in

GO:0003406: retinal pigment epithelium development

ECO:0000315: mutant phenotype evidence used in manual assertion

ZFIN:ZDB-GENO-100518-3

P

Seeded From UniProt

complete

DANRE:Q5U3H5

involved_in

GO:0008057: eye pigment granule organization

ECO:0000315: mutant phenotype evidence used in manual assertion

ZFIN:ZDB-GENO-100518-3

P

Seeded From UniProt

complete

DANRE:Q5U3H5

involved_in

GO:0032438: melanosome organization

ECO:0000315: mutant phenotype evidence used in manual assertion

ZFIN:ZDB-GENO-100518-3

P

Seeded From UniProt

complete

DANRE:Q5U3H5

involved_in

GO:0003406: retinal pigment epithelium development

ECO:0000315: mutant phenotype evidence used in manual assertion

ZFIN:ZDB-GENO-100518-3

P

Seeded From UniProt

complete

DANRE:VA0D1

involved_in

GO:0008057: eye pigment granule organization

ECO:0000315: mutant phenotype evidence used in manual assertion

ZFIN:ZDB-GENO-100518-5

P

Seeded From UniProt

complete

DANRE:VA0D1

involved_in

GO:0032438: melanosome organization

ECO:0000315: mutant phenotype evidence used in manual assertion

ZFIN:ZDB-GENO-100518-5

P

Seeded From UniProt

complete

DANRE:VA0D1

involved_in

GO:0003406: retinal pigment epithelium development

ECO:0000315: mutant phenotype evidence used in manual assertion

ZFIN:ZDB-GENO-100518-5

P

Seeded From UniProt

complete

DANRE:Q8JIU5

involved_in

GO:0008057: eye pigment granule organization

ECO:0000315: mutant phenotype evidence used in manual assertion

ZFIN:ZDB-GENO-100518-3

P

Seeded From UniProt

complete

DANRE:Q8JIU5

involved_in

GO:0032438: melanosome organization

ECO:0000315: mutant phenotype evidence used in manual assertion

ZFIN:ZDB-GENO-100518-3

P

Seeded From UniProt

complete

DANRE:Q8JIU5

involved_in

GO:0003406: retinal pigment epithelium development

ECO:0000315: mutant phenotype evidence used in manual assertion

ZFIN:ZDB-GENO-100518-3

P

Seeded From UniProt

complete

DANRE:B0R0V8

GO:0061075: positive regulation of neural retina development

ECO:0000315:

P

Fig. 7 and Fig. 8 show that the v-ATPase mutants display defects in cell proliferation and increased levels of apoptosis which demonstrates its role in the proper development of the neural retina.

complete
CACAO 7275

DANRE:B0R0V8

GO:0048069: positive regulation of eye pigmentation

ECO:0000315:

P

Fig 5

complete
CACAO 7276

DANRE:B0R0V8

involved_in

GO:0048069: eye pigmentation

ECO:0000315: mutant phenotype evidence used in manual assertion

P

Seeded From UniProt

complete

DANRE:B0R0V8

involved_in

GO:0061075: positive regulation of neural retina development

ECO:0000315: mutant phenotype evidence used in manual assertion

P

Seeded From UniProt

complete


See also

References

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