PUBLICATION

Defective Skeletogenesis with Kidney Stone Formation in Dwarf Zebrafish Mutant for trpm7

Authors
Elizondo, M.R., Arduini, B.L., Paulsen, J., MacDonald, E.L., Sabel, J.L., Henion, P.D., Cornell, R.A., Parichy, D.M.
ID
ZDB-PUB-050413-15
Date
2005
Source
Current biology : CB   15(7): 667-671 (Journal)
Registered Authors
Arduini, Brigitte, Cornell, Robert, Elizondo, Michael, Henion, Paul, MacDonald, Erin, Parichy, David M.
Keywords
none
MeSH Terms
  • Kidney/metabolism
  • Chromosome Mapping
  • DNA, Complementary/genetics
  • Zebrafish Proteins/genetics
  • Animals
  • Bone and Bones/anatomy & histology
  • Osteogenesis/genetics*
  • Larva/metabolism
  • Kidney Calculi/genetics
  • Kidney Calculi/veterinary*
  • TRPM Cation Channels
  • Ion Channels/genetics*
  • Ion Channels/metabolism
  • In Situ Hybridization
  • Base Sequence
  • Molecular Sequence Data
  • Abnormalities, Multiple/genetics
  • Abnormalities, Multiple/metabolism
  • Sequence Analysis, DNA
  • Histological Techniques
  • Zebrafish*
  • Protein Serine-Threonine Kinases
  • Protein Kinases/genetics*
  • Protein Kinases/metabolism
(all 24)
PubMed
15823540 Full text @ Curr. Biol.
Abstract
Development of the adult form requires coordinated growth and patterning of multiple traits in response to local gene activity as well as to global endocrine and physiological effectors. An excellent example of such coordination is the skeleton. Skeletal development depends on the differentiation and morphogenesis of multiple cell types to generate elements with distinct forms and functions throughout the body []. We show that zebrafish touchtone/nutria mutants exhibit severe growth retardation and gross alterations in skeletal development in addition to embryonic melanophore and touch-response defects []. These alterations include accelerated endochondral ossification but delayed intramembranous ossification, as well as skeletal deformities. We show that the touchtone/nutria phenotype results from mutations in trpm7, which encodes a transient receptor potential (TRP) family member that functions as both a cation channel and kinase. We find trpm7 expression in the mesonephric kidney and show that mutants develop kidney stones, indicating renal dysfunction. These results identify a requirement for trpm7 in growth and skeletogenesis and highlight the potential of forward genetic approaches to uncover physiological mechanisms contributing to the development of adult form.
Genes / Markers
Figures
No images available
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Expression
Phenotype
Mutations / Transgenics
Human Disease / Model
Sequence Targeting Reagents
Fish
Antibodies
Orthology
Engineered Foreign Genes
Mapping
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Citation
Elizondo, M.R., Arduini, B.L., Paulsen, J., MacDonald, E.L., Sabel, J.L., Henion, P.D., Cornell, R.A., Parichy, D.M. (2005) Defective Skeletogenesis with Kidney Stone Formation in Dwarf Zebrafish Mutant for trpm7. Current biology : CB. 15(7):667-671.