Evidence map›Paper›PMID 33422031›Full record

ArticleBMC musculoskeletal disorders2021

Adapting MRI as a clinical outcome measure for a facioscapulohumeral muscular dystrophy trial of prednisone and tacrolimus: case report.

Leo H Wang, Laura M Johnstone, Michael Bindschadler, Stephen J Tapscott, Seth D Friedman

Open access · goldAbstract readCase Reports
In one paragraph

Article in BMC musculoskeletal disorders, 2021. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 3 papers.

0numbers the graph read from it
0cells of the map it votes in
3citing papers in PubMed
0.5field-weighted citation impact, top 38% 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

3 citing papers in PubMed, 5 citations in OpenAlex.

  1. 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

5 authors at 3 institutions in 1 country.

Leo H WangDepartment of Neurology, University of Washington, Seattle, Washington, USA. leowang@uw.edu.ORCID http://orcid.org/0000-0002-5079-1416
Laura M JohnstoneDepartment of Rehabilitative Medicine, University of Washington, Seattle, Washington, USA.
Michael BindschadlerRadiology Clinical Research Imaging Core/Neurology, Seattle Children's Hospital, Seattle, Washington, USA.
Stephen J TapscottHuman Biology Division, Fred Hutchinson Research Center, Seattle, Washington, USA.
Seth D FriedmanRadiology Clinical Research Imaging Core/Improvement and Innovation, Seattle Children's Hospital, Seattle, Washington, USA.
Seattle Children's Hospital · USUniversity of Washington · USFred Hutch Cancer Center · US

Funding

No grant is acknowledged in the PubMed record.

6 · The paper itself

Abstract

backgroundFacioscapulohumeral muscular dystrophy (FSHD) is a patchy and slowly progressive disease of skeletal muscle. MRI short tau inversion recovery (STIR) sequences of patient muscles often show increased hyperintensity that is hypothesized to be associated with inflammation. This is supported by the presence of inflammatory changes on biopsies of STIR-positive muscles. We hypothesized that the STIR positivity would normalize with targeted immunosuppressive therapy. CASE PRESENTATION: 45-year-old male with FSHD type 1 was treated with 12 weeks of immunosuppressive therapy, tacrolimus and prednisone. Tacrolimus was treated to a goal serum trough of > 5 ng/mL and prednisone was tapered every month. Quantitative strength exam, functional outcome measures, and muscle MRI were performed at baseline, week 6, and week 12. The patient reported subjective worsening as reflected in quantitative strength exam. The MRI STIR signal was slightly increased from 0.02 to 0.03 of total muscle; while the T1 fat fraction was stable. Functional outcome measures also were stable.

conclusionsImmunosuppressive therapy in refractive autoimmune myopathy in other contexts has been shown to reverse STIR signal hyperintensity, however this treatment did not reverse STIR signal in this patient with FSHD. In fact, STIR signal slightly increased throughout the treatment period. This is the first study of using MRI STIR and T1 fat fraction to follow treatment effect in FSHD. We find that STIR might not be a dynamic marker for suppressing inflammation in FSHD.

Indexed as

Muscular Dystrophy, FacioscapulohumeralHumansMagnetic Resonance ImagingMaleMiddle AgedMuscle, SkeletalOutcome Assessment, Health CarePrednisoneTacrolimusPrednisoneTacrolimusAll neuromuscular diseaseCase reportFacioscapulohumeral muscular dystrophy (FSHD)Muscle diseaseOutcome measures

Identifiers

PMID33422031
PMCPMC7797109
OpenAlexW3119240545

What OpenQuestion holds

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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.