Milutinovic Stefan, Jancic Predrag, Adam Adam, Radovanovic Milan, Nordstrom Charles W, Ward Marshall, Petrovic Marija, Jevtic Dorde, Delibasic Maja, Kotseva Magdalena, Nikolajevic Milan, Dumic Igor
Internal Medicine Residency Program at Lee Health, Florida State University College of Medicine, Tallahassee, FL 32301, USA.
School of Medicine, University of Belgrade, 11000 Belgrade, Serbia.
J Clin Med. 2024 Feb 12;13(4):1045. doi: 10.3390/jcm13041045.
(1) : Cardiomyopathy in celiac disease or celiac cardiomyopathy (CCM) is a serious and potentially life-threatening disease that can occur in both adults and children. However, data supporting the causal relationship between celiac disease (CD) and cardiomyopathy (CMP) are still inconsistent. The aim of this study was to review and synthesize data from the literature on this topic and potentially reveal a more evidence-based causal relationship. (2) : The Preferred Reporting Items for Systematic Reviews and Meta-Analyses (PRISMA) guidelines were used to search Medline, Embase, and Scopus databases from database inception until September 2023. A total of 1187 original articles were identified. (3) : We identified 28 CCM patients (19 adult and 9 pediatric) with a mean age of 27.4 ± 18.01 years. Adult patients with CCM were predominantly male (84.2%) while pediatric patients were predominantly female (75%). The most common comorbidities associated with CCM were anemia (75%) and pulmonary hemosiderosis (20%). In 35% of patients, CCM occurred before the diagnosis of CD, while in 48% of patients, CCM and CD were diagnosed at the same time. Diagnosis of CD preceded diagnosis of CCM in only 18% of patients. Diagnosis of CCM is often delayed with an average, from the onset of symptoms to diagnosis, of 16 months. All patients were treated with a gluten-free diet in addition to guideline-directed medical therapy. At 11-month follow-up, cardiovascular improvement was seen in 60.7% of patients. Pediatric mortality was 33.3%, while adult mortality was 5.3%. (4) : Clinicians should be aware of the possible association between CD and CMP, and we recommend CD work-up in all patients with CMP who have concomitant anemia. While we identified only 28 cases in the literature, many cases might go unreported due to a lack of awareness regarding CCM. A high degree of clinical suspicion and a prompt diagnosis of CCM are essential to minimizing the risks of morbidity and mortality, as the combination of a gluten-free diet and guideline-directed medical therapy can improve clinical outcomes.
(1)：乳糜泻相关性心肌病或乳糜泻性心肌病（CCM）是一种严重且可能危及生命的疾病，可发生于成人和儿童。然而，支持乳糜泻（CD）与心肌病（CMP）之间因果关系的数据仍然不一致。本研究的目的是回顾和综合该主题的文献数据，并可能揭示更基于证据的因果关系。(2)：采用系统评价和Meta分析的首选报告项目（PRISMA）指南，检索Medline、Embase和Scopus数据库，检索时间从数据库建立至2023年9月。共识别出1187篇原始文章。(3)：我们识别出28例CCM患者（19例成人和9例儿童），平均年龄为27.4±18.01岁。CCM成年患者以男性为主（84.2%），而儿童患者以女性为主（75%）。与CCM相关的最常见合并症是贫血（75%）和肺含铁血黄素沉着症（20%）。35%的患者在CD诊断之前发生CCM，而48%的患者CCM和CD同时被诊断。仅18%的患者CD诊断先于CCM诊断。CCM的诊断通常延迟，从症状出现到诊断的平均时间为16个月。所有患者除接受指南指导的药物治疗外，均接受无麸质饮食治疗。在11个月的随访中，60.7%的患者心血管状况得到改善。儿童死亡率为33.3%，而成人死亡率为5.3%。(4)：临床医生应意识到CD与CMP之间可能存在的关联，我们建议对所有伴有贫血的CMP患者进行CD检查。虽然我们在文献中仅识别出28例病例，但由于对CCM缺乏认识，许多病例可能未被报告。高度的临床怀疑和对CCM的及时诊断对于将发病和死亡风险降至最低至关重要，因为无麸质饮食和指南指导的药物治疗相结合可改善临床结局。
