ZFIN ID: ZDB-PUB-191022-3
Loss of rps9 in Zebrafish Leads to p53-Dependent Anemia
Chen, C., Huang, H., Yan, R., Lin, S., Qin, W.
Date: 2019
Source: G3 (Bethesda)   9(12): 4149-4157 (Journal)
Registered Authors: Huang, Haigen, Lin, Shuo, Qing, Wei
Keywords: anemia, p53, ribosomal protein, rps9, zebrafish
MeSH Terms:
  • Anemia/metabolism*
  • Animals
  • Down-Regulation/genetics
  • Embryo, Nonmammalian/abnormalities
  • Embryo, Nonmammalian/metabolism
  • Erythroid Cells/metabolism
  • Erythroid Cells/pathology
  • Gene Expression Regulation, Developmental
  • Hemoglobins/metabolism
  • Mutation/genetics
  • Phenotype
  • Tumor Suppressor Protein p53/metabolism*
  • Up-Regulation/genetics
  • Zebrafish/embryology
  • Zebrafish/genetics
  • Zebrafish/metabolism*
  • Zebrafish Proteins/deficiency*
  • Zebrafish Proteins/genetics
  • Zebrafish Proteins/metabolism
PubMed: 31619461 Full text @ G3 (Bethesda)
Ribosome is a vital molecular machine for protein translation in the cell. Defects in several ribosomal proteins including PRS19, RPL11 and RPS14 have been observed in two types of anemia: Diamond Blackfan Anemia and 5q- syndrome. In zebrafish, deficiency of these ribosomal proteins shows similar anemia phenotype. It remains to be determined if any other ribosome proteins are similarly involved in regulating erythropoiesis. Here we generated mutations in zebrafish rps9, a rarely studied ribosomal protein gene, and investigated its function. Analysis of this mutant demonstrates that rps9 disruption leads to impairment of erythrocyte maturation, resulting in anemia. In addition, the overall phenotype including the anemic state is p53-dependent in rps9 mutants. Furthermore, this anemic state can be partially relieved by the treatment of L-leucine, and dexamethasone, which have been previously used in rescuing the phenotype of other ribosomal protein mutants. Finally, by comparing the phenotype, we showed that there are considerable differences in morphology, cytomorphology, and hemoglobin levels for four ribosomal protein mutants in zebrafish. Based on the observed difference, we suggest that the level of anemic severity correlates with the delayed status of erythrocyte maturation in zebrafish models.