PUBLICATION

Genetic ablation of Pth4 disrupts calcium-phosphate balance, bone development, and kidney transcriptome in teleosts

Authors
Méndez-Martínez, L., Suarez-Bregua, P., Guerrero-Peña, L., Barreiro-Docío, E., Costas-Prado, C., Cobelo-García, A., Rotllant, J.
ID
ZDB-PUB-250829-7
Date
2025
Source
General and comparative endocrinology : 114805114805 (Journal)
Registered Authors
Rotllant, Josep
Keywords
Bone, CRISPR, Calcium, Fish, Kidney, Phosphate, Pth4
MeSH Terms
  • Homeostasis/genetics
  • Zebrafish/genetics
  • Zebrafish/metabolism
  • Phosphates*/metabolism
  • Animals
  • Parathyroid Hormone*/genetics
  • Parathyroid Hormone*/metabolism
  • Bone Development*/genetics
  • Bone Development*/physiology
  • Transcriptome*/genetics
  • Calcium*/metabolism
  • Kidney*/metabolism
PubMed
40876765 Full text @ Gen. Comp. Endocrinol.
Abstract
Parathyroid hormone 4 (Pth4) is an evolutionarily conserved member of the PTH family, expressed in hypothalamic neurons and lost in eutherian mammals. In order to elucidate its role in mineral homeostasis and skeletal development, a pth4 knockout (pth4KO) zebrafish line was generated using CRISPR/Cas9 and transcriptomic profiling was conducted across six key tissues: brain, kidney, intestine, gills, scales, and bone. The results obtained demonstrated that the loss of Pth4 led to pronounced disturbances in calcium and phosphate homeostasis, skeletal deformities, and widespread tissue-specific transcriptional alterations. Notably, dysregulation of mineral regulatory genes-such asfgf23,phex, andslc34a1a was particularly evident in the kidney, suggesting disruption of the FGF23-Klotho axis. In parallel, differential expression of extracellular matrix genes (col1a1a,col10a1a,col11a1) and matrix remodeling enzymes (mmp9,mmp13a,mmp2) in bone and scales indicated impaired skeletal remodeling. Together, these findings highlight a pivotal role for Pth4 in the endocrine and local regulation of mineral metabolism and skeletal integrity, expanding our understanding of PTH family functions in vertebrate physiology.
Genes / Markers
Figures
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Expression
Phenotype
Mutations / Transgenics
Human Disease / Model
Sequence Targeting Reagents
Fish
Antibodies
Orthology
Engineered Foreign Genes
Mapping