Evidence map›Paper›PMID 41044600›Full record

ArticleGenome biology2025

GWAS-informed data integration and non-coding CRISPRi screen illuminate genetic etiology of bone mineral density.

Mitchell Conery, James A Pippin, Yadav Wagley, Khanh Trang, Matthew C Pahl, David A Villani, Lacey J Favazzo, Cheryl L Ackert-Bicknell, Michael J Zuscik, Eugene Katsevich and 6 more

Abstract read
In one paragraph

Article in Genome biology, 2025. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 6 papers.

0numbers the graph read from it
0cells of the map it votes in
6citing papers in PubMed
–field-weighted citation impact
1 · What the graph read from it

What it found

Each row is one number read from the abstract, on the scale the paper reported it, with its interval. Left of the dashed line favours the treatment, right favours the comparator. Under each row is the sentence it came from. New to these charts? A ten-minute tutorial.

The abstract states no effect estimate the extractor could read, or names no intervention and outcome on the map, so this paper lights no cell and moves no belief. It is still indexed, cited and linked below.

2 · The registry

The trial behind it

Trials whose registry record cites this paper, or whose number appears in the abstract. A trial that started after this paper was published is citing it as background, not reporting it.

Neither the registry nor the abstract names a trial number. If this is a trial report, that itself is worth knowing.

3 · Its place in the literature

Who cites it

6 citing papers in PubMed.

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  5. Loss ofJBMR plus · 2025
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  6. Loss ofbioRxiv : the preprint server for biology · 2024
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4 · The record

Corrections and comments

5 · Who and what money

Authors and funding

16 authors.

Mitchell Conery *Center for Spatial and Functional Genomics, Children's Hospital of Philadelphia, Philadelphia, PA, 19104, USA.
James A Pippin *Center for Spatial and Functional Genomics, Children's Hospital of Philadelphia, Philadelphia, PA, 19104, USA.
Yadav Wagley *Department of Orthopaedic Surgery, University of Michigan Medical School, Ann Arbor, MI, 48109, USA.
Khanh Trang *Center for Spatial and Functional Genomics, Children's Hospital of Philadelphia, Philadelphia, PA, 19104, USA.
Matthew C PahlCenter for Spatial and Functional Genomics, Children's Hospital of Philadelphia, Philadelphia, PA, 19104, USA.
David A VillaniColorado Program for Musculoskeletal Research, University of Colorado Anschutz Medical Campus, Aurora, CO, USA.
Lacey J FavazzoColorado Program for Musculoskeletal Research, University of Colorado Anschutz Medical Campus, Aurora, CO, USA.
Cheryl L Ackert-BicknellColorado Program for Musculoskeletal Research, University of Colorado Anschutz Medical Campus, Aurora, CO, USA.
Michael J ZuscikColorado Program for Musculoskeletal Research, University of Colorado Anschutz Medical Campus, Aurora, CO, USA.
Eugene KatsevichDepartment of Statistics and Data Science, The Wharton School, University of Pennsylvania, Philadelphia, PA, 19104, USA.
Andrew D WellsCenter for Spatial and Functional Genomics, Children's Hospital of Philadelphia, Philadelphia, PA, 19104, USA.
Babette S ZemelDepartment of Pediatrics, Perelman School of Medicine, University of Pennsylvania, Philadelphia, PA, 19104, USA.
Benjamin F Voight *Department of Systems Pharmacology and Translational Therapeutics, Perelman School of Medicine, University of Pennsylvania, Philadelphia, PA, 19104, USA.
Kurt D Hankenson *Department of Orthopaedic Surgery, University of Michigan Medical School, Ann Arbor, MI, 48109, USA.
Alessandra Chesi *Center for Spatial and Functional Genomics, Children's Hospital of Philadelphia, Philadelphia, PA, 19104, USA.
Struan F A Grant *Center for Spatial and Functional Genomics, Children's Hospital of Philadelphia, Philadelphia, PA, 19104, USA. grants@chop.edu.

Funding

Phenotypic Diversity in COVID-19UL1TR001878 · NCATS · UNIVERSITY OF PENNSYLVANIA · PI FITZGERALD, GARRET A · 2016 to 2025
$102.4M
Functional Interrogation of T2D-associated genes in human stem cell-derived models and miceUM1DK126194 · NIDDK · UNIVERSITY OF PENNSYLVANIA · PI GRANT, STRUAN F A, KAESTNER, KLAUS H · 2020 to 2024
$8.8M
Functional Mechanisms of T1D Risk Variants and their Target Genes using 3D Epigenomics and Single Cell ApproachesR01DK122586 · NIDDK · UNIVERSITY OF VIRGINIA · PI GRANT, STRUAN F A, RICH, STEPHEN S. · 2019 to 2022
$4.8M
Mechanisms of autoimmune disease risk in IL2/IL2RA-dependent immune toleranceR01AI154773 · NIAID · CHILDREN'S HOSP OF PHILADELPHIA · PI CAMPBELL, DANIEL J, WELLS, ANDREW D · 2021 to 2025
$3.8M
Genomics of bone and body composition traits in childrenR01HD100406 · NICHD · CHILDREN'S HOSP OF PHILADELPHIA · PI GRANT, STRUAN F A, ZEMEL, BABETTE S · 2020 to 2024
$3.4M
Discovery of osteoblast and osteoclast bone mass effector genes using advanced genomicsR01AG072705 · NIA · UNIVERSITY OF MICHIGAN AT ANN ARBOR · PI Struan F A Grant, Kurt David Hankenson · 2022 to 2026
$3.3M
Children's Hospital of Philadelphia Daniel B. Burke Endowed Chair for Diabetes ResearchNational Institute of Allergy and Infectious Diseases R01AI154773National Institute of Diabetes and Digestive and Kidney Diseases,United States R01DK122586National Science Foundation, United States DMS 2113072NCATS NIH HHS UL1 TR001878NIAID NIH HHS R01 AI154773NIA NIH HHS R01 AG072705NICHD NIH HHS R01 HD100406NIDDK NIH HHS R01 DK122586NIDDK NIH HHS UM1 DK126194University of Colorado Gates Grubstake AwardUniversity of Michigan Henry Ruppenthal Family Professorship for Bioengineering and Orthopaedic Surgery
6 · The paper itself

Abstract

backgroundOver 1100 independent signals have been identified with genome-wide association studies (GWAS) for bone mineral density (BMD), a key risk factor for mortality-increasing fragility fractures; however, the effector gene(s) for most remain unknown.

resultsWe execute a CRISPRi screen in human fetal osteoblasts (hFOBs) with single-cell RNA-seq read-out for 89 non-coding elements predicted to regulate osteoblast gene expression at BMD GWAS loci. The BMD relevance of hFOBs is supported by heritability enrichment from stratified LD-score regression involving 98 cell types grouped into 15 tissues. Twenty-three genes show perturbation in the screen, with four (ARID5B, CC2D1B, EIF4G2, and NCOA3) exhibiting consistent effects upon siRNA knockdown on three measures of osteoblast maturation and mineralization. Lastly, additional heritability enrichments, genetic correlations, and multi-trait fine-mapping unexpectedly reveal that many BMD GWAS signals are pleiotropic and likely mediate their effects via non-bone tissues.

conclusionsOur results provide a roadmap for how single-cell CRISPRi screens may be applied to the challenging task of resolving effector gene identities at all BMD GWAS loci. Extending our CRISPRi screening approach to other tissues could play a key role in fully elucidating the etiology of BMD.

Indexed as

Bone DensityCRISPR-Cas SystemsGenome-Wide Association StudyHumansOsteoblastsQuantitative Trait LociSingle-Cell Analysis

Identifiers

PMID41044600
PMCPMC12492525

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Read under generation 80e0d062 · epoch 390. Bibliography from PubMed, PubMed Central and OpenAlex; grants from NIH RePORTER; trial links from ClinicalTrials.gov; estimates, votes and beliefs from the OpenQuestion graph.