Evidence map›Paper›PMID 31160583›Full record

ArticleNature communications2019

Fibroadipogenic progenitors are responsible for muscle loss in limb girdle muscular dystrophy 2B.

Marshall W Hogarth, Aurelia Defour, Christopher Lazarski, Eduard Gallardo, Jordi Diaz Manera, Terence A Partridge, Kanneboyina Nagaraju, Jyoti K Jaiswal

Open access · goldAbstract read
In one paragraph

Article in Nature communications, 2019. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 111 papers, 1 of them a synthesis that pooled it.

0numbers the graph read from it
0cells of the map it votes in
111citing papers in PubMed, 1 pooled it
7.6field-weighted citation impact, top 2% 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

111 citing papers in PubMed, 1 synthesis or guideline pooled it, 145 citations in OpenAlex.

  1. Pooled it
  2. Article
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  4. Review
  5. Article
  6. Article
  7. Progress on cell therapy for skeletal muscle disorders.Advanced drug delivery reviews · 2026
    Review
  8. Article
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  11. Cellular and molecular mechanisms of fatty infiltration in paraspinal muscles in spine-specific sarcopenia.Zhong nan da xue xue bao. Yi xue ban = Journal of Central South University. Medical sciences · 2026
    Review
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  13. Article
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  20. Article

51 more citing papers are in PubMed but not listed here.

4 · The record

Corrections and comments

PubMed lists nothing against this paper. Absence here is not a guarantee, only a check that was made.

5 · Who and what money

Authors and funding

8 authors at 3 institutions in 2 countries.

Marshall W HogarthResearch Center for Genetic Medicine, Children's Research Institute, Children's National Health System, Washington, DC, 20010, USA.
Aurelia DefourResearch Center for Genetic Medicine, Children's Research Institute, Children's National Health System, Washington, DC, 20010, USA.
Christopher LazarskiChildren's Research Institute, Children's National Health System, Washington, DC, 20010, USA.
Eduard GallardoUnitat and Laboratori de Malaltdies Neuromusculars, Institut de Recerca, Hospital de la Santa Creu i Sant Pau, Universitat Autònoma de Barcelona, Barcelona, 08041, Spain.
Jordi Diaz ManeraUnitat and Laboratori de Malaltdies Neuromusculars, Institut de Recerca, Hospital de la Santa Creu i Sant Pau, Universitat Autònoma de Barcelona, Barcelona, 08041, Spain.
Terence A PartridgeResearch Center for Genetic Medicine, Children's Research Institute, Children's National Health System, Washington, DC, 20010, USA.
Kanneboyina NagarajuResearch Center for Genetic Medicine, Children's Research Institute, Children's National Health System, Washington, DC, 20010, USA.
Jyoti K JaiswalResearch Center for Genetic Medicine, Children's Research Institute, Children's National Health System, Washington, DC, 20010, USA. jkjaiswal@cnmc.org.ORCID http://orcid.org/0000-0002-5992-5185
Children's National · USUniversitat Autònoma de Barcelona · ESBinghamton University · US

Funding

NCMRR-DC Core Molecular & Functional Outcome Measures in Rehabilitation MedicineR24HD050846 · NICHD · CHILDREN'S RESEARCH INSTITUTE · PI KNOBLACH, SUSAN M · 2005 to 2014
$8.6M
RNA fidelity and protein function of Becker-like in-frame deletionsP50AR060836 · NIAMS · CHILDREN'S RESEARCH INSTITUTE · PI HOFFMAN, ERIC P. · 2011 to 2015
$7.8M
Research_ProjectU54HD090257 · NICHD · CHILDREN'S RESEARCH INSTITUTE · PI GUAY-WOODFORD, LISA MARIE · 2016 to 2020
$5.9M
Understanding the mechanism and role of cell membrane repair in Miyoshi MyopathyR01AR055686 · NIAMS · ROCKEFELLER UNIVERSITY · PI JAISWAL, JYOTI K · 2008 to 2021
$3.6M
Preclinical drug trial in mouse models of inflammationK26OD011171 · OD · STATE UNIVERSITY OF NY,BINGHAMTON · PI NAGARAJU, KANNEBOYINA · 2012 to 2015
$420k
Muscular Dystrophy Association (Muscular Dystrophy Association Inc.) MDA477331NIAMS NIH HHS P50 AR060836NIAMS NIH HHS R01 AR055686NICHD NIH HHS R24 HD050846NICHD NIH HHS U54 HD090257NIH HHS K26 OD011171U.S. Department of Health & Human Services | NIH | Eunice Kennedy Shriver National Institute of Child Health and Human Development (NICHD) U54HD090257U.S. Department of Health & Human Services | NIH | National Institute of Arthritis and Musculoskeletal and Skin Diseases (NIAMS) R01AR055686U.S. Department of Health & Human Services | NIH | NIH Office of the Director (OD) K26OD011171
6 · The paper itself

Abstract

Muscle loss due to fibrotic or adipogenic replacement of myofibers is common in muscle diseases and muscle-resident fibro/adipogenic precursors (FAPs) are implicated in this process. While FAP-mediated muscle fibrosis is widely studied in muscle diseases, the role of FAPs in adipogenic muscle loss is not well understood. Adipogenic muscle loss is a feature of limb girdle muscular dystrophy 2B (LGMD2B) - a disease caused by mutations in dysferlin. Here we show that FAPs cause the adipogenic loss of dysferlin deficient muscle. Progressive accumulation of Annexin A2 (AnxA2) in the myofiber matrix causes FAP differentiation into adipocytes. Lack of AnxA2 prevents FAP adipogenesis, protecting against adipogenic loss of dysferlinopathic muscle while exogenous AnxA2 enhances muscle loss. Pharmacological inhibition of FAP adipogenesis arrests adipogenic replacement and degeneration of dysferlin-deficient muscle. These results demonstrate the pathogenic role of FAPs in LGMD2B and establish these cells as therapeutic targets to ameliorate muscle loss in patients.

Indexed as

AdipocytesAdipogenesisAdipose TissueAdolescentAge of OnsetAnimalsAnnexin A2Case-Control StudiesDysferlinElapid VenomsFemaleFibrosisHumansIn Vitro TechniquesMaleMiceAnnexin A2Anxa2 protein, mousebatimastatDysferlinDYSF protein, humanDysf protein, mouseElapid VenomsnotexinPhenylalanineProtease InhibitorsThiophenes

Identifiers

PMID31160583
PMCPMC6547715
OpenAlexW2948703564

What OpenQuestion holds

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LicenceCC BY
Read underepoch 390

Registered trials

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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.