GONUTS has been updated to MW1.31 Most things seem to be working but be sure to report problems.

Have any questions? Please email us at ecoliwiki@gmail.com

PMID:24767999

From GONUTS
Jump to: navigation, search
Citation

Coxam, B, Sabine, A, Bower, NI, Smith, KA, Pichol-Thievend, C, Skoczylas, R, Astin, JW, Frampton, E, Jaquet, M, Crosier, PS, Parton, RG, Harvey, NL, Petrova, TV, Schulte-Merker, S, Francois, M and Hogan, BM (2014) Pkd1 regulates lymphatic vascular morphogenesis during development. Cell Rep 7:623-33

Abstract

Lymphatic vessels arise during development through sprouting of precursor cells from veins, which is regulated by known signaling and transcriptional mechanisms. The ongoing elaboration of vessels to form a network is less well understood. This involves cell polarization, coordinated migration, adhesion, mixing, regression, and shape rearrangements. We identified a zebrafish mutant, lymphatic and cardiac defects 1 (lyc1), with reduced lymphatic vessel development. A mutation in polycystic kidney disease 1a was responsible for the phenotype. PKD1 is the most frequently mutated gene in autosomal dominant polycystic kidney disease (ADPKD). Initial lymphatic precursor sprouting is normal in lyc1 mutants, but ongoing migration fails. Loss of Pkd1 in mice has no effect on precursor sprouting but leads to failed morphogenesis of the subcutaneous lymphatic network. Individual lymphatic endothelial cells display defective polarity, elongation, and adherens junctions. This work identifies a highly selective and unexpected role for Pkd1 in lymphatic vessel morphogenesis during development.

Links

PubMed Online version:10.1016/j.celrep.2014.03.063

Keywords

Animals; Animals, Genetically Modified; Cells, Cultured; Embryo, Mammalian/metabolism; Embryonic Development; Endothelial Cells/cytology; Endothelial Cells/metabolism; Humans; Intercellular Junctions/metabolism; Lymph Nodes/growth & development; Lymph Nodes/metabolism; Lymphangiogenesis; Lymphatic Vessels/metabolism; Mice; Mice, Inbred C57BL; Mice, Transgenic; Mutation; Phenotype; Polycystic Kidney, Autosomal Dominant/genetics; Polycystic Kidney, Autosomal Dominant/metabolism; Polycystic Kidney, Autosomal Dominant/pathology; TRPP Cation Channels/antagonists & inhibitors; TRPP Cation Channels/genetics; TRPP Cation Channels/metabolism; Zebrafish; Zebrafish Proteins/genetics; Zebrafish Proteins/metabolism

Significance

Annotations

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

DANRE:A0A3G1GT95

involved_in

GO:0001946: lymphangiogenesis

ECO:0000315: mutant phenotype evidence used in manual assertion

ZFIN:ZDB-GENO-140801-1

P

Seeded From UniProt

complete

DANRE:A0A3G1GT95

involved_in

GO:0001946: lymphangiogenesis

ECO:0000316: genetic interaction evidence used in manual assertion

ZFIN:ZDB-GENO-140801-1,ZFIN:ZDB-MRPHLNO-100707-3

P

Seeded From UniProt

complete

DANRE:F1QIA9

involved_in

GO:0001946: lymphangiogenesis

ECO:0000315: mutant phenotype evidence used in manual assertion

ZFIN:ZDB-GENO-140801-1

P

Seeded From UniProt

complete

DANRE:F1QIA9

involved_in

GO:0001946: lymphangiogenesis

ECO:0000316: genetic interaction evidence used in manual assertion

ZFIN:ZDB-GENO-140801-1,ZFIN:ZDB-MRPHLNO-100707-3

P

Seeded From UniProt

complete

MOUSE:PKD1

acts_upstream_of_or_within

GO:0030155: regulation of cell adhesion

ECO:0000315: mutant phenotype evidence used in manual assertion

MGI:MGI:3612341

P

  • regulates_o_occurs_in:(CL:0002138)

Seeded From UniProt

complete

MOUSE:PKD1

acts_upstream_of_or_within

GO:0030010: establishment of cell polarity

ECO:0000315: mutant phenotype evidence used in manual assertion

MGI:MGI:3612341

P

  • occurs_in:(CL:0002138)

Seeded From UniProt

complete

DANRE:PKD2

involved_in

GO:0001946: lymphangiogenesis

ECO:0000315: mutant phenotype evidence used in manual assertion

ZFIN:ZDB-MRPHLNO-070129-1

P

Seeded From UniProt

complete

DANRE:U3JAI2

involved_in

GO:0001946: lymphangiogenesis

ECO:0000316: genetic interaction evidence used in manual assertion

ZFIN:ZDB-GENO-140801-1,ZFIN:ZDB-MRPHLNO-100707-3

P

Seeded From UniProt

complete

MOUSE:PKD1

acts_upstream_of_or_within

GO:0030010: establishment of cell polarity

ECO:0000315: mutant phenotype evidence used in manual assertion

MGI:MGI:3612341

P

occurs_in:(CL:0002138)

Seeded From UniProt

complete

MOUSE:PKD1

acts_upstream_of_or_within

GO:0030155: regulation of cell adhesion

ECO:0000315: mutant phenotype evidence used in manual assertion

MGI:MGI:3612341

P

regulates_o_occurs_in:(CL:0002138)

Seeded From UniProt

complete

MOUSE:PKD1

acts_upstream_of_or_within

GO:0036303: lymph vessel morphogenesis

ECO:0000315: mutant phenotype evidence used in manual assertion

MGI:MGI:3612341

P

Seeded From UniProt

complete

DANRE:Q6IVV8

involved_in

GO:0001946: lymphangiogenesis

ECO:0000315: mutant phenotype evidence used in manual assertion

ZFIN:ZDB-MRPHLNO-070129-1

P

Seeded From UniProt

complete

DANRE:Q2VF27

involved_in

GO:0001946: lymphangiogenesis

ECO:0000315: mutant phenotype evidence used in manual assertion

ZFIN:ZDB-MRPHLNO-070129-1

P

Seeded From UniProt

complete

Notes

See also

References

See Help:References for how to manage references in GONUTS.