Functional genomics of human skeletal development and the patterning of height heritability

Underlying variation in height are regulatory changes to chondrocytes, cartilage cells comprising long-bone growth plates. Currently, we lack knowledge on epigenetic regulation and gene expression of chondrocytes sampled across the human skeleton, and therefore we cannot understand basic regulatory mechanisms controlling height biology. We first rectify this issue by generating extensive epigenetic and transcriptomic maps from chondrocytes sampled from different growth plates across developing human skeletons, discovering novel regulatory networks shaping human bone/joint development. Next, using these maps in tandem with height genome-wide association study (GWAS) signals, we disentangle the regulatory impacts that skeletal element-specific versus global-acting variants have on skeletal growth, revealing the prime importance of regulatory pleiotropy in controlling height variation. Finally, as height is highly heritable, and thus often the test case for complex-trait genetics, we leverage these datasets within a testable omnigenic model framework to discover novel chondrocyte developmental modules and peripheral-acting factors shaping height biology and skeletal growth.

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Publication Details

Journal
Cell
Published
2024-11-15
DOI
https://doi.org/10.1016/j.cell.2024.10.040
Citations
24
Primary Topic
Adipose Tissue and Metabolism
Type
article
Field-Weighted Citation Impact
7.84

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article

Functional genomics of human skeletal development and the patterning of height heritability

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24 citations
Cell
Adipose Tissue and Metabolism
7.84
article

Functional genomics of human skeletal development and the patterning of height heritability

Joel N. Hirschhorn, Terence D. Capellini, Mariel Young, Loïc Yengo, Pushpanathan Muthuirulan, Sailaja Vedantam, Eric Bartell, Daniel Richard, Eirini Marouli
article en
24 citations

Abstract

Underlying variation in height are regulatory changes to chondrocytes, cartilage cells comprising long-bone growth plates. Currently, we lack knowledge on epigenetic regulation and gene expression of chondrocytes sampled across the human skeleton, and therefore we cannot understand basic regulatory mechanisms controlling height biology. We first rectify this issue by generating extensive epigenetic and transcriptomic maps from chondrocytes sampled from different growth plates across developing human skeletons, discovering novel regulatory networks shaping human bone/joint development. Next, using these maps in tandem with height genome-wide association study (GWAS) signals, we disentangle the regulatory impacts that skeletal element-specific versus global-acting variants have on skeletal growth, revealing the prime importance of regulatory pleiotropy in controlling height variation. Finally, as height is highly heritable, and thus often the test case for complex-trait genetics, we leverage these datasets within a testable omnigenic model framework to discover novel chondrocyte developmental modules and peripheral-acting factors shaping height biology and skeletal growth.

CellVol. 188(1)
Broad Institute (US), Boston Children's Hospital (US), Harvard University (US), The University of Queensland (AU), Queen Mary University of London (GB), William Harvey Research Institute (GB)
Harvard University, University of Washington, National Institutes of Health, National Institute of Child Health and Human Development, Eunice Kennedy Shriver National Institute of Child Health and Human Development
Gender equality
Openalex Percentile: Top 2%
Adipose Tissue and Metabolism
7.84
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