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Zebrafish Tric-b is required for skeletal development and bone cells differentiation
Francesca Tonelli1, Laura Leoni1, Valentina Daponte1
1Department of Molecular Medicine, Biochemistry Unit, University of Pavia, Pavia, Italy.
Frontiers in Endocrinology
|February 9, 2023
Summary
Trimeric intracellular potassium channel B (TRIC-B) is crucial for skeletal development and cell homeostasis. Zebrafish models reveal TRIC-B deficiency impairs collagen modification and bone cell function, offering insights into osteogenesis imperfecta.
Area of Science:
- Cellular Biology
- Developmental Biology
- Genetics
Background:
- Trimeric intracellular potassium channels (TRIC-A and TRIC-B) are ER proteins regulating calcium release.
- TRIC-B deficiency is linked to osteogenesis imperfecta (OI Type XIV) due to calcium flux dysregulation.
- The precise role of TRIC-B in cell homeostasis and disease mechanisms remains unclear.
Purpose of the Study:
- To investigate the function of TRIC-B in skeletal tissue using zebrafish models.
- To elucidate the molecular mechanisms underlying TRIC-B deficiency-related disorders.
Main Methods:
- Generated zebrafish mutants for the tmem38b gene using CRISPR/Cas9.
- Analyzed skeletal phenotypes and collagen modification in mutant zebrafish.
- Assessed osteoblast and osteoclast activity during caudal fin regeneration.
Main Results:
- Demonstrated maternal and zygotic expression of tmem38b in zebrafish.
- Observed under-modified and ER-retained collagen type I in tmem38b mutants.
- Identified impaired osteoblast and osteoclast activity and mineralization defects.
Conclusions:
- TRIC-B is essential for early development and bone cell differentiation.
- Zebrafish models effectively recapitulate aspects of OI Type XIV.
- Findings highlight TRIC-B's critical role in maintaining skeletal integrity and cell homeostasis.
