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Small intestinal biopsy findings consistent with celiac disease in patients with idiopathic inflammatory myopathy: Review of existing literature.

Seminars in arthritis and rheumatism
October 1, 2021
Gunhild Alvik Nyborg et al. (2 authors)
Journal ArticleResearch Support, Non-U.S. Gov'tReviewHuman Study
Study Details

Study Goal

The researchers aimed to explore the connection between idiopathic inflammatory myopathies (IIM) and celiac disease (CeD) and assess the effects of a gluten-free diet (GFD) on IIM symptoms.

Results Summary

The study found that in 58% of cases, IIM symptoms improved after starting a GFD, with 29% achieving remission using GFD alone. Some patients showed improvement even without classical CeD symptoms or positive serology.

Population

Patients with idiopathic inflammatory myopathies (IIM) and concomitant small intestinal biopsy findings consistent with celiac disease (CeD).

Effective Dosage

Not specified

Duration

Not specified

Interactions

None mentioned

Extracted Claims (4)
InterventionDirectionEndpointPopulationDosageImpactClaim #
gluten-free diet
decrease
muscle inflammation (myositis) component of IIM
patients with idiopathic inflammatory myopathies (IIM) and a concurrent diagnosis of celiac disease (CeD)
-
improves
#1
gluten-free diet
decrease
IIM
patients with IIM and concomitant small intestinal biopsy findings consistent with CeD
14/24 (58%)
signs of improvement
#2
gluten-free diet
decrease
IIM
patients with IIM and concomitant small intestinal biopsy findings consistent with CeD
7/24 (29%)
remained in clinical IIM remission
#3
gluten-free diet
decrease
IIM
IIM cases with small intestinal biopsy findings consistent with celiac disease
majority of cases
improved
#4
Abstract

BACKGROUND: Case reports have described patients with idiopathic inflammatory myopathies (IIM) and a concurrent diagnosis of celiac disease (CeD) for whom the muscle inflammation (myositis) component of IIM improves after the patients start standard treatment with gluten-free diet (GFD). A connection between IIM and CeD is not commonly recognized. AIM: In this first systematic review of the topic, we aimed to explore all peer-reviewed publications of IIM cases and concomitant small intestinal biopsy findings consistent with CeD, published after 1975. METHODS: Systematic literature searches were performed in MEDLINE, PubMed, and EMBASE, supplemented by screening of references and non-systematic searches via Google and Google Scholar. RESULTS: Altogether 30 cases published between 1976 and 2017 were uncovered. Information about gastrointestinal symptoms prior to CeD diagnosis was available for 19 patients, with 6/19 (32%) reporting no GI symptoms. CeD-related serological data were available in 23/30 patients. Endomysial antibodies were present in 10/18 (56%), while only 2/9 (22%) had antibodies against tissue transglutaminase. Serum antibodies to native gliadin were present in 16/18 (89%). Clinical effects of a GFD on the IIM were reported for 24 patients, with signs of improvement in 14/24 (58%), including three patients with otherwise therapy-resistant inclusion body myositis. Longitudinal follow-up data available from the published studies indicated that 7/24 (29%) remained in clinical IIM remission with GFD as the sole therapeutic intervention. CONCLUSION: In the IIM cases presented here, duodenal biopsy findings consistent with celiac disease was sometimes present without classical CeD symptoms or positive traditional CeD serology, and in the majority of cases, the IIM improved after introduction of a gluten-free diet. While extra vigilance towards CeD in IIM patients seems warranted, there is need for more research to clarify if GFD has effects on organ systems other than the small intestine in patients with IIM and small intestinal biopsy findings consistent with CeD.

Medical Subject Headings (MeSH)
AutoantibodiesBiopsyCeliac DiseaseGliadinHumansMyositis
Study Links
Quality Scores
SafetyNot Assessed
Efficacy70/10
Quality65/10
Citation Metrics
Total Citations2
Citations/Year0.5
Relative Citation Ratio0.29
NIH Percentile15.1%
Research Impact Scores
APT Score0.25
Weight Score1.96
Normalized Score0.61
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Small intestinal biopsy findings consistent with celiac dise... | Panacea Index