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PMID:17542649

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Citation

Yang, CT, Hindes, AE, Hultman, KA and Johnson, SL (2007) Mutations in gfpt1 and skiv2l2 cause distinct stage-specific defects in larval melanocyte regeneration in zebrafish. PLoS Genet. 3:e88

Abstract

The establishment of a single cell type regeneration paradigm in the zebrafish provides an opportunity to investigate the genetic mechanisms specific to regeneration processes. We previously demonstrated that regeneration melanocytes arise from cell division of the otherwise quiescent melanocyte precursors following larval melanocyte ablation with a small molecule, MoTP. The ease of ablating melanocytes by MoTP allows us to conduct a forward genetic screen for mechanisms specific to regeneration from such precursors or stem cells. Here, we reported the identification of two mutants, eartha(j23e1) and julie(j24e1) from a melanocyte ablation screen. Both mutants develop normal larval melanocytes, but upon melanocyte ablation, each mutation results in a distinct stage-specific defect in melanocyte regeneration. Positional cloning reveals that the eartha(j23e1) mutation is a nonsense mutation in gfpt1 (glutamine:fructose-6-phosphate aminotransferase 1), the rate-limiting enzyme in glucosamine-6-phosphate biosynthesis. Our analyses reveal that a mutation in gfpt1 specifically affects melanocyte differentiation (marked by melanin production) at a late stage during regeneration and that gfpt1 acts cell autonomously in melanocytes to promote ontogenetic melanocyte darkening. We identified that the julie(j24e1) mutation is a splice-site mutation in skiv2l2 (superkiller viralicidic activity 2-like 2), a predicted DEAD-box RNA helicase. Our in situ analysis reveals that the mutation in skiv2l2 causes defects in cell proliferation, suggesting that skiv2l2 plays a role in regulating melanoblast proliferation during early stages of melanocyte regeneration. This finding is consistent with previously described role for cell division during larval melanocyte regeneration. The analyses of these mutants reveal their stage-specific roles in melanocyte regeneration. Interestingly, these mutants identify regeneration-specific functions not only in early stages of the regeneration process, but also in late stages of differentiation of the regenerating melanocyte. We suggest that mechanisms of regeneration identified in this mutant screen may reveal fundamental differences between the mechanisms that establish differentiated cells during embryogenesis, and those involved in larval or adult growth.

Links

PubMed PMC1885281 Online version:10.1371/journal.pgen.0030088

Keywords

Animals; Cell Differentiation/genetics; Glutamine-Fructose-6-Phosphate Transaminase (Isomerizing)/genetics; Melanocytes/cytology; Melanocytes/enzymology; Melanocytes/physiology; Molecular Sequence Data; Mutation; RNA Helicases/genetics; Regeneration/genetics; Zebrafish/embryology; Zebrafish/genetics; Zebrafish/growth & development; Zebrafish Proteins/genetics

Significance

Annotations

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

DANRE:F1QD61

involved_in

GO:0031101: fin regeneration

ECO:0000315: mutant phenotype evidence used in manual assertion

ZFIN:ZDB-GENO-071114-2

P

Seeded From UniProt

complete

DANRE:F1QD61

involved_in

GO:0051781: positive regulation of cell division

ECO:0000315: mutant phenotype evidence used in manual assertion

ZFIN:ZDB-GENO-071114-2

P

Seeded From UniProt

complete

DANRE:RPZ

involved_in

GO:0033333: fin development

ECO:0000315: mutant phenotype evidence used in manual assertion

ZFIN:ZDB-GENO-071114-3

P

Seeded From UniProt

complete

DANRE:Q3S344

involved_in

GO:0030318: melanocyte differentiation

ECO:0000315: mutant phenotype evidence used in manual assertion

ZFIN:ZDB-GENO-071114-1

P

Seeded From UniProt

complete

DANRE:Q3S344

involved_in

GO:0002063: chondrocyte development

ECO:0000315: mutant phenotype evidence used in manual assertion

ZFIN:ZDB-GENO-071114-1

P

Seeded From UniProt

complete

DANRE:Q3S344

involved_in

GO:0048703: embryonic viscerocranium morphogenesis

ECO:0000315: mutant phenotype evidence used in manual assertion

ZFIN:ZDB-GENO-071114-1

P

Seeded From UniProt

complete

DANRE:Q6P7X6

involved_in

GO:0031101: fin regeneration

ECO:0000315: mutant phenotype evidence used in manual assertion

ZFIN:ZDB-GENO-071114-2

P

Seeded From UniProt

complete

DANRE:Q6P7X6

involved_in

GO:0051781: positive regulation of cell division

ECO:0000315: mutant phenotype evidence used in manual assertion

ZFIN:ZDB-GENO-071114-2

P

Seeded From UniProt

complete

DANRE:X1WGR7

involved_in

GO:0030318: melanocyte differentiation

ECO:0000315: mutant phenotype evidence used in manual assertion

ZFIN:ZDB-GENO-071114-1

P

Seeded From UniProt

complete

DANRE:X1WGR7

involved_in

GO:0002063: chondrocyte development

ECO:0000315: mutant phenotype evidence used in manual assertion

ZFIN:ZDB-GENO-071114-1

P

Seeded From UniProt

complete

DANRE:X1WGR7

involved_in

GO:0048703: embryonic viscerocranium morphogenesis

ECO:0000315: mutant phenotype evidence used in manual assertion

ZFIN:ZDB-GENO-071114-1

P

Seeded From UniProt

complete

See also

References

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