An adult osteopetrosis model in medaka reveals the importance of osteoclast function for bone remodeling in teleost fish

Comparative Biochemistry and Physiology. Toxicology & Pharmacology : CBP
Thuy Thanh ToChristoph Winkler

Abstract

Osteoclasts play important roles during bone growth and in maintaining bone health and bone homeostasis. Dysfunction or lack of osteoclasts leads to increased bone mass and osteopetrosis phenotypes in mouse and human. Here we report a severe osteopetrosis-like phenotype in transgenic medaka fish, in which membrane bound EGFP (mEGFP) was expressed in osteoclasts under control of the cathepsin K promoter (ctsk:mEGFP). In contrast to reporter lines with GFP expression in the cytoplasm of osteoclasts, adult fish of the mEGFP line developed bone defects indicative for an osteoclast dysfunction. Activity of tartrate-resistant acid phosphatase (TRAP) was down-regulated and excess bone was observed in most parts of the skeleton. The osteopetrotic phenotype was particularly obvious at the neural and haemal arches that failed to increase their volume in growing fish. Excess bone caused severe constriction of the spinal cord and the ventral aorta. The continuation of tooth development and the failure to shed teeth resulted in severe hyperdontia. Interestingly, at the vertebral column vertebral body arches displayed a severe osteopetrosis, while vertebral centra had no or only a mild osteopetrotic phenotype. This confirms previous reports ...Continue Reading

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Oct 27, 2015·Comparative Biochemistry and Physiology. Toxicology & Pharmacology : CBP·Lynne A Fieber
Nov 1, 2016·Proceedings. Biological Sciences·Tim RolvienBjörn Busse
Sep 7, 2018·Journal of Morphology·Adelbert De ClercqP Eckhard Witten
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Jul 22, 2019·Developmental Biology·L Lleras-ForeroS Schulte-Merker
Oct 22, 2020·American Journal of Physiology. Regulatory, Integrative and Comparative Physiology·Alex M ZimmerSteve F Perry

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