A trans-eQTL network regulates osteoclast multinucleation and bone mass.

Pereira, Marie; Ko, Jeong-Hun; Logan, John; Protheroe, Hayley; Kim, Kee-Beom; Tan, Amelia Li Min; Croucher, Peter I; Park, Kwon-Sik et al. · Elife · 2020

basic_science · Level V

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Abstract

Functional characterisation of cell-type-specific regulatory networks is key to establish a causal link between genetic variation and phenotype. The osteoclast offers a unique model for interrogating the contribution of co-regulated genes to in vivo phenotype as its multinucleation and resorption activities determine quantifiable skeletal traits. Here we took advantage of a <i>trans</i>-regulated gene network (MMnet, macrophage multinucleation network) which we found to be significantly enriched for GWAS variants associated with bone-related phenotypes. We found that the network hub gene <i>Bcat1</i> and seven other co-regulated MMnet genes out of 13, regulate bone function. Specifically, global (<i>Pik3cb<sup>-/-</sup></i>, <i>Atp8b2<sup>+/-</sup></i>, <i>Igsf8<sup>-/-</sup></i>, <i>Eml1<sup>-/-</sup></i>, <i>Appl2<sup>-/-</sup></i>, <i>Deptor<sup>-/-</sup></i>) and myeloid-specific <i>Slc40a1</i> knockout mice displayed abnormal bone phenotypes. We report opposing effects of MMnet genes on bone mass in mice and osteoclast multinucleation/resorption in humans with strong correlation between the two. These results identify MMnet as a functionally conserved network that regulates osteoclast multinucleation and bone mass.

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