Evidence map›Paper›PMID 25801126›Full record

ReviewTrends in molecular medicine2015

Emerging preclinical animal models for FSHD.

Angela Lek, Fedik Rahimov, Peter L Jones, Louis M Kunkel

Abstract readReview
PubMed Full text at PMC Publisher
In one paragraph

Review in Trends in molecular medicine, 2015. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 31 papers.

0numbers the graph read from it
0cells of the map it votes in
31citing 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

31 citing papers in PubMed.

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  14. Mouse models for muscular dystrophies: an overview.Disease models & mechanisms · 2020
    Review
  15. Zebrafish Models ofFrontiers in molecular neuroscience · 2020
    Article
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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

4 authors.

Angela LekDivision of Genetics and Genomics, Manton Center for Orphan Disease Research, Children's Hospital, Boston, MA 02115, USA; Department of Genetics, Harvard Medical School, Boston, MA 02115, USA; The Wellstone Program, Departments of Neurology and Cell and Developmental Biology, University of Massachusetts Medical School (UMMS), Worcester, MA 01655, USA. Electronic address: alek@enders.tch.harvard.edu.
Fedik RahimovDivision of Genetics and Genomics, Manton Center for Orphan Disease Research, Children's Hospital, Boston, MA 02115, USA; Department of Genetics, Harvard Medical School, Boston, MA 02115, USA; The Wellstone Program, Departments of Neurology and Cell and Developmental Biology, University of Massachusetts Medical School (UMMS), Worcester, MA 01655, USA.
Peter L JonesThe Wellstone Program, Departments of Neurology and Cell and Developmental Biology, University of Massachusetts Medical School (UMMS), Worcester, MA 01655, USA; The Eunice Kennedy Shriver National Institute of Child Health and Human Development (NIHCD) Senator Paul D. Wellstone Muscular Dystrophy Cooperative Research Center, University of Massachusetts Medical School, Worcester, MA, USA.
Louis M KunkelDivision of Genetics and Genomics, Manton Center for Orphan Disease Research, Children's Hospital, Boston, MA 02115, USA; Department of Genetics, Harvard Medical School, Boston, MA 02115, USA; The Wellstone Program, Departments of Neurology and Cell and Developmental Biology, University of Massachusetts Medical School (UMMS), Worcester, MA 01655, USA.

Funding

Myogenesis Studies for FSHD BiomarkersU54HD060848 · NICHD · UNIV OF MASSACHUSETTS MED SCH WORCESTER · PI EMERSON, CHARLES P. · 2008 to 2017
$16.1M
Mechanisms of DUX4 mediated FSHD pathologyR01AR062587 · NIAMS · UNIV OF MASSACHUSETTS MED SCH WORCESTER · PI JONES, PETER L · 2013 to 2024
$5.3M
NIAMS NIH HHS #1R01AR062587NIAMS NIH HHS R01 AR062587NICHD NIH HHS U54 HD060848NICHD NIH HHS U54HD060848
6 · The paper itself

Abstract

Facioscapulohumeral dystrophy (FSHD) is a unique and complex genetic disease that is not entirely solved. Recent advances in the field have led to a consensus genetic premise for the disorder, enabling researchers to now pursue the design of preclinical models. In this review we explore all available FSHD models (DUX4-dependent and -independent) for their utility in therapeutic discovery and potential to yield novel disease insights. Owing to the complex nature of FSHD, there is currently no single model that accurately recapitulates the genetic and pathophysiological spectrum of the disorder. Existing models emphasize only specific aspects of the disease, highlighting the need for more collaborative research and novel paradigms to advance the translational research space of FSHD.

Indexed as

AnimalsHomeodomain ProteinsHumansModels, AnimalMuscular Dystrophy, FacioscapulohumeralHomeodomain ProteinsDUX4facioscapulohumeral dystrophymuscular dystrophy

Identifiers

PMID25801126
PMCPMC4424175

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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.