Preparing your results

Our searching services are busy right now. Your search will reload in five seconds.

Forgot Password

If you have forgotten your password you can enter your email here and get a temporary password sent to your email.

Zebrafish mutants calamity and catastrophe define critical pathways of gene-nutrient interactions in developmental copper metabolism.

PLoS genetics | Nov 14, 2008

Nutrient availability is an important environmental variable during development that has significant effects on the metabolism, health, and viability of an organism. To understand these interactions for the nutrient copper, we used a chemical genetic screen for zebrafish mutants sensitive to developmental copper deficiency. In this screen, we isolated two mutants that define subtleties of copper metabolism. The first contains a viable hypomorphic allele of atp7a and results in a loss of pigmentation when exposed to mild nutritional copper deficiency. This mutant displays incompletely penetrant skeletal defects affected by developmental copper availability. The second carries an inactivating mutation in the vacuolar ATPase that causes punctate melanocytes and embryonic lethality. This mutant, catastrophe, is sensitive to copper deprivation revealing overlap between ion metabolic pathways. Together, the two mutants illustrate the utility of chemical genetic screens in zebrafish to elucidate the interaction of nutrient availability and genetic polymorphisms in cellular metabolism.

Pubmed ID: 19008952 RIS Download

Mesh terms: Adenosine Triphosphatases | Alleles | Animals | Cells, Cultured | Copper | Embryo, Nonmammalian | Humans | Melanosomes | Menkes Kinky Hair Syndrome | Mutation | Phenotype | Protein Transport | Zebrafish | Zebrafish Proteins

Research resources used in this publication

None found

Research tools detected in this publication

Data used in this publication

None found

Associated grants

  • Agency: NIGMS NIH HHS, Id: T32 GM07200
  • Agency: NIGMS NIH HHS, Id: T32 GM007200
  • Agency: NIDDK NIH HHS, Id: DK61763
  • Agency: NIDDK NIH HHS, Id: DK44464
  • Agency: NIDDK NIH HHS, Id: R01 DK061763
  • Agency: NIDDK NIH HHS, Id: R01 DK044464
  • Agency: NIDDK NIH HHS, Id: R37 DK044464

ZFIN (Data, Gene Expression)

Publication data is provided by the National Library of Medicine ® and PubMed ®. Data is retrieved from PubMed ® on a weekly schedule. For terms and conditions see the National Library of Medicine Terms and Conditions.