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PMID:17651719

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Citation

Perner, B, Englert, C and Bollig, F (2007) The Wilms tumor genes wt1a and wt1b control different steps during formation of the zebrafish pronephros. Dev. Biol. 309:87-96

Abstract

The Wilms tumor protein WT1 is an essential factor for kidney development. In humans, mutations in WT1 lead to Wilms tumor, a pediatric kidney cancer as well as to developmental anomalies concerning the urogenital tract. Inactivation of Wt1 in mice causes multiple organ defects most notably agenesis of the kidneys. In zebrafish, two paralogous wt1 genes exist, wt1a and wt1b. The wt1 genes are expressed in a similar and overlapping but not identical pattern. Here, we have examined the role of both wt1 genes in early kidney development employing a transgenic line with pronephros specific GFP expression and morpholino knockdown experiments. Inactivation of wt1a led to failure of glomerular differentiation and morphogenesis resulting in a rapidly expanding general body edema. In contrast, knockdown of wt1b was compatible with early glomerular development. After 48 h, however, wt1b morphant embryos developed cysts in the region of the glomeruli and tubules and subsequent pericardial edema at 4 days post-fertilization. Thus, our data suggest different functions for wt1a and wt1b in zebrafish nephrogenesis. While wt1a has a more fundamental and early role in pronephros development and is essential for the formation of glomerular structures, wt1b functions at later stages of nephrogenesis.

Links

PubMed Online version:10.1016/j.ydbio.2007.06.022

Keywords

Animals; Animals, Genetically Modified; Body Patterning/physiology; Embryo, Nonmammalian/metabolism; Gene Expression Regulation, Developmental; Nephrons/embryology; Oligonucleotides, Antisense/pharmacology; WT1 Proteins/genetics; WT1 Proteins/metabolism; Zebrafish/embryology; Zebrafish/metabolism; Zebrafish Proteins/metabolism

Significance

Annotations

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

DANRE:Q08CU4

involved_in

GO:0048793: pronephros development

ECO:0000315: mutant phenotype evidence used in manual assertion

ZFIN:ZDB-MRPHLNO-071107-3

P

Seeded From UniProt

complete

DANRE:Q08CU4

involved_in

GO:0048793: pronephros development

ECO:0000316: genetic interaction evidence used in manual assertion

ZFIN:ZDB-MRPHLNO-071107-1,ZFIN:ZDB-MRPHLNO-071107-3

P

Seeded From UniProt

complete

DANRE:Q1L884

involved_in

GO:0048793: pronephros development

ECO:0000315: mutant phenotype evidence used in manual assertion

ZFIN:ZDB-MRPHLNO-071107-3

P

Seeded From UniProt

complete

DANRE:Q1L884

involved_in

GO:0048793: pronephros development

ECO:0000316: genetic interaction evidence used in manual assertion

ZFIN:ZDB-MRPHLNO-071107-1,ZFIN:ZDB-MRPHLNO-071107-3

P

Seeded From UniProt

complete

DANRE:Q2QBW1

involved_in

GO:0048793: pronephros development

ECO:0000315: mutant phenotype evidence used in manual assertion

ZFIN:ZDB-MRPHLNO-071107-3

P

Seeded From UniProt

complete

DANRE:Q2QBW1

involved_in

GO:0048793: pronephros development

ECO:0000316: genetic interaction evidence used in manual assertion

ZFIN:ZDB-MRPHLNO-071107-1,ZFIN:ZDB-MRPHLNO-071107-3

P

Seeded From UniProt

complete

DANRE:Q98SJ4

involved_in

GO:0032835: glomerulus development

ECO:0000315: mutant phenotype evidence used in manual assertion

ZFIN:ZDB-MRPHLNO-071107-1

P

Seeded From UniProt

complete

DANRE:Q98SJ4

involved_in

GO:0048793: pronephros development

ECO:0000315: mutant phenotype evidence used in manual assertion

ZFIN:ZDB-MRPHLNO-071107-1

P

Seeded From UniProt

complete

DANRE:Q98SJ4

involved_in

GO:0048793: pronephros development

ECO:0000316: genetic interaction evidence used in manual assertion

ZFIN:ZDB-MRPHLNO-071107-1,ZFIN:ZDB-MRPHLNO-071107-3

P

Seeded From UniProt

complete

DANRE:Q9PUT7

involved_in

GO:0032835: glomerulus development

ECO:0000315: mutant phenotype evidence used in manual assertion

ZFIN:ZDB-MRPHLNO-071107-1

P

Seeded From UniProt

complete

DANRE:Q9PUT7

involved_in

GO:0048793: pronephros development

ECO:0000315: mutant phenotype evidence used in manual assertion

ZFIN:ZDB-MRPHLNO-071107-1

P

Seeded From UniProt

complete

DANRE:Q9PUT7

involved_in

GO:0048793: pronephros development

ECO:0000316: genetic interaction evidence used in manual assertion

ZFIN:ZDB-MRPHLNO-071107-1,ZFIN:ZDB-MRPHLNO-071107-3

P

Seeded From UniProt

complete


See also

References

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