Department of Rheumatology, Oslo University Hospital - Rikshospitalet, P.O. box 4950 Nydalen, 0424 Oslo, Norway.
Department of Rheumatology, Oslo University Hospital - Rikshospitalet, P.O. box 4950 Nydalen, 0424 Oslo, Norway; Department of Rheumatology and Infectious Diseases, Institute of Clinical Medicine, University of Oslo, Oslo, Norway.
Semin Arthritis Rheum. 2021 Oct;51(5):1033-1044. doi: 10.1016/j.semarthrit.2021.07.012. Epub 2021 Jul 25.
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.
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.
Systematic literature searches were performed in MEDLINE, PubMed, and EMBASE, supplemented by screening of references and non-systematic searches via Google and Google Scholar.
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.
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.
病例报告描述了一些特发性炎性肌病（IIM）患者同时被诊断为乳糜泻（CeD），这些患者在开始无麸质饮食（GFD）的标准治疗后，其肌炎（肌病）成分得到改善。IIM 和 CeD 之间的联系并不常见。
在本研究中，我们首次对这一主题进行了系统回顾，旨在探索自 1975 年以来发表的所有经过同行评审的 IIM 病例和伴有乳糜泻小肠活检结果的出版物。
在 MEDLINE、PubMed 和 EMBASE 中进行系统文献检索，并用 Google 和 Google Scholar 进行非系统检索和参考文献筛选进行补充。
共发现 1976 年至 2017 年期间发表的 30 例病例。在 CeD 诊断前有胃肠道症状的患者有 19 例，其中 6/19（32%）患者无胃肠道症状。23/30 例患者有 CeD 相关的血清学数据。18 例中有 10 例（56%）存在肌内膜抗体，而只有 9 例中的 2 例（22%）存在抗组织转谷氨酰胺酶抗体。18 例中有 16 例（89%）存在天然麦胶蛋白血清抗体。24 例患者报告了 GFD 对 IIM 的临床影响，14/24（58%）患者的症状得到改善，包括 3 例治疗抵抗性包涵体肌炎患者。从已发表的研究中获得的纵向随访数据表明，7/24（29%）例患者在单独使用 GFD 作为唯一治疗干预的情况下，仍处于临床 IIM 缓解状态。
在本研究中报告的 IIM 病例中，十二指肠活检结果符合乳糜泻，有时没有典型的 CeD 症状或阳性的传统 CeD 血清学，并且在大多数情况下，引入无麸质饮食后，IM 得到改善。虽然在 IIM 患者中对 CeD 保持额外警惕似乎是合理的，但仍需要更多的研究来明确 GFD 是否对 IIM 患者和伴有乳糜泻小肠活检结果的患者的小肠以外的器官系统有影响。
