Heart transplantation in rapidly progressive end-stage heart failure associated with celiac disease

Barrio, J.P.; Cura, Geraldine; Ramallo, Germán; Díez, Mirta; Vigliano, Carlos; Katus, Hugo A.; Mereles, Derliz

doi:10.1136/bcr.12.2010.3624

Cited by 5 publications

(7 citation statements)

References 10 publications

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“…Severely dilated left ventricle, concomitant left ventricular dysfunction, very low ejection fraction, pulmonary hemosiderosis, heart block, and/or heart failure have been reported in cardiomyopathy patients with CD[ 112 , 115 - 120 ]. Severe progressive dilated cardiomyopathy, requiring heart transplantation, can occur[ 121 ]. Dilated cardiomyopathy in CD may be accompanied by congestive heart failure and is also becoming increasingly recognized in the pediatric population[ 122 , 123 ].…”

Section: Publication Summary Statisticsmentioning

confidence: 99%

Cardiovascular involvement in celiac disease

et al. 2017

WJC

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Celiac disease (CD) is an autoimmune response to ingestion of gluten protein, which is found in wheat, rye, and barley grains, and results in both small intestinal manifestations, including villous atrophy, as well as systemic manifestations. The main treatment for the disease is a gluten-free diet (GFD), which typically results in the restoration of the small intestinal villi, and restoration of other affected organ systems, to their normal functioning. In an increasing number of recently published studies, there has been great interest in the occurrence of alterations in the cardiovascular system in untreated CD. Herein, published studies in which CD and cardiovascular terms appear in the title of the study were reviewed. The publications were categorized into one of several types: (1) articles (including cohort and case-control studies); (2) reviews and meta-analyses; (3) case studies (one to three patient reports); (4) letters; (5) editorials; and (6) abstracts (used when no full-length work had been published). The studies were subdivided as either heart or vascular studies, and were further characterized by the particular condition that was evident in conjunction with CD. Publication information was determined using the Google Scholar search tool. For each publication, its type and year of publication were tabulated. Salient information from each article was then compiled. It was determined that there has been a sharp increase in the number of CD - cardiovascular studies since 2000. Most of the publications are either of the type “article” or “case study”. The largest number of documents published concerned CD in conjunction with cardiomyopathy (33 studies), and there have also been substantial numbers of studies published on CD and thrombosis (27), cardiovascular risk (17), atherosclerosis (13), stroke (12), arterial function (11), and ischemic heart disease (11). Based on the published research, it can be concluded that many types of cardiovascular issues can occur in untreated CD patients, but that most tend to resolve on a GFD, often in conjunction with the healing of small intestinal villous atrophy. However, in some cases the alterations are irreversible, underscoring the need for CD screening and treatment when cardiovascular issues arise of unknown etiology.

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Section: Publication Summary Statisticsmentioning

confidence: 99%

Cardiovascular involvement in celiac disease

et al. 2017

WJC

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“…Ischemic CMP was excluded in 13 adult cases (68.4%) with either anatomical or functional testing for coronary artery disease (CA, CCTA). Four cases with two relatively young adults (18-and 20-year-olds) did not report any ischemic work-up [17,28,33,41]. Also, one case had negative troponin on presentation and did not have an official ischemic CMP work-up, while one case underwent a negative stress test without additional testing for coronary artery disease [30,31].…”

Section: Discussionmentioning

confidence: 97%

“…Our systematic review identified 28 unique cases (19 adult and 9 pediatric patients) from 23 case reports and 2 case series, published from 1986 to 2022 [17,18,. The overall age range was from 3 to 70 years, with a mean of 27.4 ± 18.0 years.…”

Section: Demographics and Comorbiditiesmentioning

confidence: 99%

“…Among patients with dilated, non-ischemic CMP, there is a notable prevalence of those with CD [14][15][16]. Celiac cardiomyopathy (CCM) is a serious and potentially life-threatening disease, sometimes requiring heart transplantation [17], and can occur in adult and pediatric populations [18]. A potential connection between CD and CMP has been previously identified; however, the data supporting this association are inconsistent [15,[19][20][21].…”

Section: Introductionmentioning

confidence: 99%

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Cardiomyopathy in Celiac Disease: A Systematic Review

Milutinovic,

Jancic,

Adam

et al. 2024

JCM

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(1) Background: 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) Methods: 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) Results: 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) Conclusions: 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.

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“…In a study of 60 people with celiac disease aged greater than 65 years, 5% died secondary to heart failure, significantly higher than in the nonceliac disease population [ 10 ]. Barrio et al presented a first case of a severe progressive dilated cardiomyopathy that required heart transplantation in young woman with celiac disease [ 14 ].…”

Section: Discussionmentioning

confidence: 99%

Cardiomyopathy Associated with Celiac Disease in Childhood

Boskovic

Kitic

Prokić

et al. 2012

Case Reports in Gastrointestinal Medicine

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Celiac disease is predominantly a disease of the small intestine characterized by chronic malabsorption in genetically susceptible individuals who ingest grains containing gluten, such as wheat, barley, and rye. Although previously believed to be uncommon, celiac disease may be present in up to 1% of the adult and children population. Celiac disease is associated frequently with iron-deficiency anemia, dermatitis herpetiformis, selective IgA deficiency, thyroid disorders, diabetes mellitus, and various connective tissue disorders but is rarely associated with cardiomyopathy.

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Heart transplantation in rapidly progressive end-stage heart failure associated with celiac disease

Cited by 5 publications

References 10 publications

Cardiovascular involvement in celiac disease

Cardiovascular involvement in celiac disease

Cardiomyopathy in Celiac Disease: A Systematic Review

Cardiomyopathy Associated with Celiac Disease in Childhood

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