Prevalence and risk factors for low bone density in adults with a Fontan circulation

D’Ambrosio, Paolo; Tran, Derek; Verrall, Charlotte E; Attard, Chantal; Ayer, Julian; d’Udekem, Yves; Twigg, Stephen M.; Celermajer, David S.; Cordina, Rachael

doi:10.1111/chd.12836

Cited by 13 publications

(9 citation statements)

References 33 publications

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“…36 Fontan physiology has been associated with a low bone mineral density z score. [37][38][39] This is a particular concern in young children with Fontan circulation, because early bone mass derangements could lead to osteoporosis later in life. In the present study, we found no significant difference in bone mineral density z scores between our Fontan patients and the matched control group, however, data were limited.…”

Section: Hansson Et Almentioning

confidence: 99%

Vitamin D, liver-related biomarkers, and distribution of fat and lean mass in young patients with Fontan circulation

et al. 2021

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Introduction/aim: Young patients with Fontan circulation may have low serum 25-hydroxyvitamin D levels, an affected liver, and unhealthy body compositions. This study aimed to explore the association between vitamin D intake/levels, liver biomarkers, and body composition in young Fontan patients. Method: We collected prospective data in 2017 to 2018, obtained with food-frequency questionnaires, biochemical analyses of liver biomarkers, and dual-energy X-ray absorptiometry scans in 44 children with Fontan circulation. Body compositions were compared to matched controls (n = 38). Linear regression analyses were used to investigate associations of biomarkers, leg pain, and lean mass on serum levels of 25-hydroxyvitamin D. Biomarkers were converted to z scores and differences were evaluated within the Fontan patients. Results: Our Fontan patients had a daily mean vitamin D intake of 9.9 µg and a mean serum 25-hydroxyvitamin D of 56 nmol/L. These factors were not associated with fat or lean mass, leg pain, or biomarkers of liver status. The Fontan patients had significantly less lean mass, but higher fat mass than controls. Male adolescents with Fontan circulation had a greater mean abdominal fat mass than male controls and higher cholesterol levels than females with Fontan circulation. Conclusion: Vitamin D intake and serum levels were not associated with body composition or liver biomarkers in the Fontan group, but the Fontan group had lower lean mass and higher fat mass than controls. The more pronounced abdominal fat mass in male adolescents with Fontan circulation might increase metabolic risks later in life.

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Section: Hansson Et Almentioning

confidence: 99%

Vitamin D, liver-related biomarkers, and distribution of fat and lean mass in young patients with Fontan circulation

et al. 2021

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“…Five studies assessed bone mineral density (BMD) Z-scores measured by either Dual Energy X-ray Absorptiometry (DEXA) or peripheral Quantitative Computed Tomography (pQCT). Each found BMD is mildly decreased in Fontan patients compared to age-related references ( 68 , 99 , 100 , 102 , 104 ). These studies are summarized in Table 2 .…”

Section: Resultsmentioning

confidence: 81%

“…Our search on bone abnormalities in Fontan patients yielded 9 articles ( 39 , 68 , 78 , 99 – 104 ). Five studies assessed bone mineral density (BMD) Z-scores measured by either Dual Energy X-ray Absorptiometry (DEXA) or peripheral Quantitative Computed Tomography (pQCT).…”

Section: Resultsmentioning

confidence: 99%

“…BMD Z-scores are within normal range for the majority of patients. D'Ambrosio et al found a prevalence of 29% for a BMD in the osteopenic range (i.e., Z ≤ 1) and 4% ( n = 1) in the osteoporotic range (Z ≤ 2.5) ( 99 ). Diab et al found a Z-score of −2 in 20% ( n = 13) of their study population ( 100 ).…”

Section: Resultsmentioning

confidence: 99%

“…Hypoxia and reduced cardiac output are common features of Fontan physiology. Hypoxia may increase osteoclast activity and bone resorption ( 39 , 99 ). Reduced circulation of bone marrow, in the context of reduced cardiac output, may impair bone formation ( 103 ).…”

Section: Resultsmentioning

confidence: 99%

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Fontan Circulation Associated Organ Abnormalities Beyond the Heart, Lungs, Liver, and Gut: A Systematic Review

Ritmeester

Veger

Ven

et al. 2022

Front. Cardiovasc. Med.

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IntroductionPatients with a Fontan circulation are at risk for sequelae of Fontan physiology during follow-up. Fontan physiology affects all organ systems and an overview of end-organ damage is needed.MethodsWe performed a systematic review of abnormalities in multiple organ systems for patients with a longstanding Fontan circulation. We searched online databases for articles describing abnormalities in multiple organ systems. Cardio-pulmonary abnormalities, protein losing enteropathy, and Fontan associated liver disease have already extensively been described and were excluded from this systematic review.ResultsOur search returned 5,704 unique articles. After screening, we found 111 articles relating to multiple organ systems. We found abnormalities in, among others, the nervous system, pituitary, kidneys, and musculoskeletal system. Pituitary edema—relating to the unique pituitary vasculature- may affect the thyroid axis. Renal dysfunction is common. Creatinine based renal function estimates may be inappropriate due to myopenia. Both lean muscle mass and bone mineral density are decreased. These abnormalities in multiple organ systems may be related to Fontan physiology, cyanosis, iatrogenic factors, or lifestyle.ConclusionsHealth care providers should be vigilant for hypothyroidism, visual or hearing deficits, and sleep disordered breathing in Fontan patients. We recommend including cystatin C for assessment of renal function. This review may aid health care providers and guide future research.Systematic Review Registration:https://www.crd.york.ac.uk/prospero/display_record.php?ID=CRD42021232461, PROSPERO, identifier: CRD42021232461.

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Low bone mineral density and its related factors in adults with congenital heart disease in Vietnam: A cross‐sectional study

Truong

Nguyen

Kim

et al. 2022

Health Science Reports

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Background and Aims Recent studies have highlighted the increased risk of low bone mineral density (BMD) in adults with cardiovascular disease. However, little is known about BMD in adults with congenital heart disease (CHD), particularly in developing countries. We hypothesized that factors related to BMD would lead to a high prevalence of low BMD in adults with CHD. This study aimed to determine the prevalence of low BMD and its related factors in Vietnamese adults with CHD. Methods We conducted a cross‐sectional study of 73 adults diagnosed with CHD in Vietnam. Low BMD was classified based on their site‐specific Z ‐scores and T ‐scores at the posteroanterior lumbar spine and left proximal femur. Logistic regression analyses were performed to evaluate factors related to low BMD. Results Low BMD was confirmed in one‐third of the adults with CHD. There were trends of more bone loss in certain parts of the body than in others, with the prevalence of low BMD at the sites of the lumbar vertebrae (L1‒L4) and left proximal femur (femoral neck, trochanteric femur, and intertrochanteric area) of 43.9%, 31.8%, 28.8%, 33.3%, 8.8%, 1.5%, and 6.1%, respectively. The prevalence of low BMD in the lumbar spine was significantly higher than that in the left proximal femur (34.3% vs. 2.9%, p < 0.001). Moreover, the prevalence of low BMD was significantly higher in adults with CHD than in those without polycythemia and vitamin D deficiency (55.6% vs. 20.9%, p = 0.001 and 46.2% vs. 19.4%, p = 0.002, respectively). A stratified multivariate logistic regression analysis revealed that low BMD was associated with polycythemia (odds ratio: 4.72; 95% confidence interval: 1.64–13.58, p = 0.004). Conclusions Low BMD is common among adults with CHD in Vietnam and related to polycythemia.

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Prevalence and risk factors for low bone density in adults with a Fontan circulation

Cited by 13 publications

References 33 publications

Vitamin D, liver-related biomarkers, and distribution of fat and lean mass in young patients with Fontan circulation

Vitamin D, liver-related biomarkers, and distribution of fat and lean mass in young patients with Fontan circulation

Fontan Circulation Associated Organ Abnormalities Beyond the Heart, Lungs, Liver, and Gut: A Systematic Review

Low bone mineral density and its related factors in adults with congenital heart disease in Vietnam: A cross‐sectional study

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