Evidence map›Paper›PMID 36153796›Full record

ArticleJournal of bone and mineral research : the official journal of the American Society for Bone and Mineral Research2022

Functional Testing of Bone Morphogenetic Protein (BMP) Pathway Variants Identified on Whole-Exome Sequencing in a Patient with Delayed-Onset Fibrodysplasia Ossificans Progressiva (FOP) Using ACVR1

Kelly L Wentworth, Robert L Lalonde, Jay C Groppe, Niambi Brewer, Tania Moody, Steven Hansberry, Kimberly E Taylor, Eileen M Shore, Frederick S Kaplan, Robert J Pignolo and 2 more

Open access · hybridAbstract read
In one paragraph

Article in Journal of bone and mineral research : the official journal of the American Society for Bone and Mineral Research, 2022. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 7 papers.

0numbers the graph read from it
0cells of the map it votes in
7citing papers in PubMed
0.4field-weighted citation impact, top 35% of its field
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

7 citing papers in PubMed, 7 citations in OpenAlex.

  1. Gut microbiome-dependent IL-1 signaling is a mediator of ACVR1bioRxiv : the preprint server for biology · 2026
    Article
  2. Review
  3. Review
  4. Animal models of tendon calcification: Past, present, and future.Animal models and experimental medicine · 2024
    Review
  5. Matrix metalloproteinase-9 deficiency confers resilience in fibrodysplasia ossificans progressiva in a man and mice.Journal of bone and mineral research : the official journal of the American Society for Bone and Mineral Research · 2024
    Article
  6. Article
  7. Functional comparison of human ACVR1 and zebrafish Acvr1l FOP-associated variants in embryonic zebrafish.Developmental dynamics : an official publication of the American Association of Anatomists · 2023
    Article
4 · The record

Corrections and comments

5 · Who and what money

Authors and funding

12 authors at 8 institutions in 1 country.

Kelly L WentworthDepartment of Medicine, Division of Endocrinology and Metabolism, Zuckerberg San Francisco General Hospital, San Francisco, CA, USA.ORCID 0000-0002-3711-1121
Robert L LalondeTufts University School of Dental Medicine, Division of Craniofacial and Molecular Genetics, Boston, MA, USA.ORCID 0000-0002-1803-0983
Jay C GroppeDepartment of Biomedical Sciences, Texas A&M University College of Dentistry, Dallas, TX, USA.ORCID 0000-0003-1058-2772
Niambi BrewerDepartment of Orthopedic Surgery and The Center of Research for FOP & Related Disorders, Perelman School of Medicine, University of Pennsylvania, Philadelphia, PA, USA.ORCID 0000-0002-7627-6594
Tania MoodyInstitute for Human Genetics, the Program in Craniofacial Biology, the UCSF Eli and Edythe Broad Institute for Regeneration Medicine, and the Department of Medicine, Division of Endocrinology and Metabolism, University of California, San Francisco, CA, USA.ORCID 0000-0002-5964-8691
Steven HansberrySan Francisco State University, California Institute of Regenerative Medicine Bridges to Stem Cell Research Program, San Francisco, CA, USA.ORCID 0000-0003-2802-7419
Kimberly E TaylorRussell/Engleman Rheumatology Research Center, University of California, San Francisco, CA, USA.ORCID 0000-0001-6848-9074
Eileen M ShoreDepartment of Orthopedic Surgery and The Center of Research for FOP & Related Disorders, Perelman School of Medicine, University of Pennsylvania, Philadelphia, PA, USA.ORCID 0000-0003-2609-6971
Frederick S KaplanDepartment of Orthopedic Surgery and The Center of Research for FOP & Related Disorders, Perelman School of Medicine, University of Pennsylvania, Philadelphia, PA, USA.
Robert J PignoloDepartment of Medicine, Mayo Clinic, Rochester, MN, USA.ORCID 0000-0002-8533-9438
Pamela C YelickTufts University School of Dental Medicine, Division of Craniofacial and Molecular Genetics, Boston, MA, USA.
Edward C HsiaoInstitute for Human Genetics, the Program in Craniofacial Biology, the UCSF Eli and Edythe Broad Institute for Regeneration Medicine, and the Department of Medicine, Division of Endocrinology and Metabolism, University of California, San Francisco, CA, USA.ORCID 0000-0001-8924-106X
University of Pennsylvania · USBroad Institute · USTufts University · USCalifornia Institute for Regenerative Medicine · USMayo Clinic in Arizona · USSan Francisco General Hospital · USTexas A&M University · USUniversity of California, San Francisco · US

Funding

Bioengineered Composite Alveolar Bone-Tooth Constructs for Tooth RegenerationR01DE026731 · NIDCR · TUFTS UNIVERSITY BOSTON · PI YELICK, PAMELA C · 2017 to 2021
$3.6M
Innate immune regulation of stem cells in bone formationR01AR066735 · NIAMS · UNIVERSITY OF CALIFORNIA, SAN FRANCISCO · PI HSIAO, EDWARD C · 2015 to 2019
$1.8M
Novel Zebrafish Models for Human Fibrodysplasia Ossificans ProgressivaR21AR072085 · NIAMS · TUFTS UNIVERSITY BOSTON · PI YELICK, PAMELA C · 2017 to 2018
$399k
National Institutes of Health-Loan Repayment Program 1L30DE029063-01NIAMS NIH HHS R01 AR066735NIAMS NIH HHS R01AR066735NIAMS NIH HHS R21 AR072085NIAMS NIH HHS R21AR072085NIDCR NIH HHS L30 DE029063NIDCR NIH HHS R01 DE026731
6 · The paper itself

Abstract

Bone morphogenetic protein (BMP) signaling is critical in skeletal development. Overactivation can trigger heterotopic ossification (HO) as in fibrodysplasia ossificans progressiva (FOP), a rare, progressive disease of massive HO formation. A small subset of FOP patients harboring the causative ACVR1

Indexed as

Myositis OssificansOssification, HeterotopicActivin Receptors, Type IAnimalsBone Morphogenetic ProteinsExome SequencingHumansLigandsMutationZebrafishActivin Receptors, Type IACVR1 protein, humanBone Morphogenetic ProteinsLigandsBMP/TGFβDEVELOPMENTAL MODELINGFIBRODYSPLASIA OSSIFICANS PROGRESSIVAGENETIC ANIMAL MODELSPRECLINICAL STUDIES

Identifiers

PMID36153796
PMCPMC9950781
OpenAlexW4297199173

What OpenQuestion holds

Textmetadata
LicenceCC BY-NC
Read underepoch 390

Registered trials

None linked

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.