Cardiomyopathy in Celiac Disease: A Systematic Review
(1) <b>Background</b>: 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) a...
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MDPI AG
2024-02-01
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| Series: | Journal of Clinical Medicine |
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| Online Access: | https://www.mdpi.com/2077-0383/13/4/1045 |
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| author | Stefan Milutinovic Predrag Jancic Adam Adam Milan Radovanovic Charles W. Nordstrom Marshall Ward Marija Petrovic Dorde Jevtic Maja Delibasic Magdalena Kotseva Milan Nikolajevic Igor Dumic |
| author_facet | Stefan Milutinovic Predrag Jancic Adam Adam Milan Radovanovic Charles W. Nordstrom Marshall Ward Marija Petrovic Dorde Jevtic Maja Delibasic Magdalena Kotseva Milan Nikolajevic Igor Dumic |
| author_sort | Stefan Milutinovic |
| collection | DOAJ |
| description | (1) <b>Background</b>: 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) <b>Methods</b>: 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) <b>Results</b>: 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) <b>Conclusions</b>: 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. |
| first_indexed | 2024-03-07T22:26:35Z |
| format | Article |
| id | doaj.art-5444195ca58b4bfea2b03daadf6f3afc |
| institution | Directory Open Access Journal |
| issn | 2077-0383 |
| language | English |
| last_indexed | 2024-03-07T22:26:35Z |
| publishDate | 2024-02-01 |
| publisher | MDPI AG |
| record_format | Article |
| series | Journal of Clinical Medicine |
| spelling | doaj.art-5444195ca58b4bfea2b03daadf6f3afc2024-02-23T15:22:08ZengMDPI AGJournal of Clinical Medicine2077-03832024-02-01134104510.3390/jcm13041045Cardiomyopathy in Celiac Disease: A Systematic ReviewStefan Milutinovic0Predrag Jancic1Adam Adam2Milan Radovanovic3Charles W. Nordstrom4Marshall Ward5Marija Petrovic6Dorde Jevtic7Maja Delibasic8Magdalena Kotseva9Milan Nikolajevic10Igor Dumic11Internal Medicine Residency Program at Lee Health, Florida State University College of Medicine, Tallahassee, FL 32301, USASchool of Medicine, University of Belgrade, 11000 Belgrade, SerbiaCardiology Fellowship Program, Cook County, Chicago, IL 60612, USASection of Hospital Medicine, Dartmouth-Hitchcock Medical Center, Lebanon, NH 03766, USAMayo Clinic College of Medicine and Science, Rochester, MN 55905, USASection of Hospital Medicine, Dartmouth-Hitchcock Medical Center, Lebanon, NH 03766, USACardiology Fellowship Program, Icahn School of Medicine at Mount Sinai, New York, NY 10029, USAElmhurst Hospital, New York, NY 11373, USAInternal Medicine Residency Program at Lee Health, Florida State University College of Medicine, Tallahassee, FL 32301, USAFrancisian Health, Olympia Fields, IL 60461, USASchool of Medicine, University of Belgrade, 11000 Belgrade, SerbiaMayo Clinic College of Medicine and Science, Rochester, MN 55905, USA(1) <b>Background</b>: 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) <b>Methods</b>: 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) <b>Results</b>: 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) <b>Conclusions</b>: 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.https://www.mdpi.com/2077-0383/13/4/1045cardiomyopathyceliac diseasegluten-sensitive enteropathyextraintestinal manifestations of celiac diseaseLane–Hamilton syndromeidiopathic pulmonary hemosiderosis |
| spellingShingle | Stefan Milutinovic Predrag Jancic Adam Adam Milan Radovanovic Charles W. Nordstrom Marshall Ward Marija Petrovic Dorde Jevtic Maja Delibasic Magdalena Kotseva Milan Nikolajevic Igor Dumic Cardiomyopathy in Celiac Disease: A Systematic Review Journal of Clinical Medicine cardiomyopathy celiac disease gluten-sensitive enteropathy extraintestinal manifestations of celiac disease Lane–Hamilton syndrome idiopathic pulmonary hemosiderosis |
| title | Cardiomyopathy in Celiac Disease: A Systematic Review |
| title_full | Cardiomyopathy in Celiac Disease: A Systematic Review |
| title_fullStr | Cardiomyopathy in Celiac Disease: A Systematic Review |
| title_full_unstemmed | Cardiomyopathy in Celiac Disease: A Systematic Review |
| title_short | Cardiomyopathy in Celiac Disease: A Systematic Review |
| title_sort | cardiomyopathy in celiac disease a systematic review |
| topic | cardiomyopathy celiac disease gluten-sensitive enteropathy extraintestinal manifestations of celiac disease Lane–Hamilton syndrome idiopathic pulmonary hemosiderosis |
| url | https://www.mdpi.com/2077-0383/13/4/1045 |
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