Prevalence and risk factors for low bone density in adults with a Fontan circulation.
Adult
Bone Density
Bone Diseases, Metabolic
/ diagnostic imaging
Cross-Sectional Studies
Female
Fontan Procedure
/ adverse effects
Health Status
Heart Defects, Congenital
/ diagnostic imaging
Humans
Male
New South Wales
/ epidemiology
Osteoporosis
/ diagnostic imaging
Prevalence
Prospective Studies
Risk Assessment
Risk Factors
Treatment Outcome
Young Adult
Fontan
biochemistry
bone density
congenital heart disease
hormones
single ventricle
Journal
Congenital heart disease
ISSN: 1747-0803
Titre abrégé: Congenit Heart Dis
Pays: United States
ID NLM: 101256510
Informations de publication
Date de publication:
Nov 2019
Nov 2019
Historique:
received:
10
04
2019
revised:
29
07
2019
accepted:
02
08
2019
pubmed:
21
8
2019
medline:
1
7
2020
entrez:
21
8
2019
Statut:
ppublish
Résumé
This study aimed to characterize bone mineral density abnormalities and pathophysiological associations in young adults living with a Fontan circulation. Participants underwent bone mineral density measurement using dual-energy X-ray absorptiometry and serum biochemical analysis, cardiopulmonary exercise and strength testing and transthoracic echocardiography. In our cohort (n = 28), 29% had osteopenic-range bone mineral density and one patient was osteoporotic (average hip t score: -0.6 ± 1.1; spine t score: -0.6 ± 0.9). Four patients (14%) had z scores < -2.0. Parathyroid hormone levels were increased compared with laboratory median (6.1 ± 3.5 vs 4 pmol/L, P = .01) and 27% had 25-hydroxy-vitamin D < 50 nmol/L. 25-hydroxy-vitamin D negatively correlated with parathyroid hormone (ρ = -0.53, P = .01) suggesting secondary hyperparathyroidism. Atrioventricular valve systolic to diastolic duration ratio, an echocardiographic measure of diastolic dysfunction, inversely correlated with hip t and z scores (P < .01). Hip t scores were positively associated with oxygen saturations (ρ = 0.45, P = .05) and tended to be inversely associated with parathyroid hormone levels (ρ = -0.44, P = .07) and N-Terminal pro b-type natriuretic peptide (ρ = -0.42, P = .08). Many young adults with a Fontan circulation have abnormal bone mineral density. The underlying pathophysiology is likely multifactorial. Possible contributors include secondary hyperparathyroidism, hypoxemia, diastolic cardiac dysfunction and neurohormonal activation. As low bone mineral density is clinically relevant and potentially treatable, assessment of bone mineral density should be part of routine care in this cohort.
Types de publication
Journal Article
Langues
eng
Pagination
987-995Subventions
Organisme : National Health and Medical Research Council
ID : 1065794
Organisme : National Health and Medical Research Council
ID : 1076849
Informations de copyright
© 2019 Wiley Periodicals, Inc.
Références
Driscoll DJ, Danielson GK, Puga FJ, Schaff HV, Heise CT, Staats BA. Exercise tolerance and cardiorespiratory response to exercise after the Fontan operation for tricuspid atresia or functional single ventricle. J Am Coll Cardiol. 1986;7(5):1087-1094.
Engelfriet P, Boersma E, Oechslin E, et al. The spectrum of adult congenital heart disease in Europe: morbidity and mortality in a 5 year follow-up period. The Euro Heart Survey on adult congenital heart disease. Eur Heart J. 2005;26(21):2325-2333.
Gewillig M, Goldberg DJ. Failure of the fontan circulation. Heart Fail Clin. 2014;10(1):105-116.
Mori M, Aguirre AJ, Elder RW, et al. Beyond a broken heart: circulatory dysfunction in the failing Fontan. Pediatr Cardiol. 2014;35(4):569-579.
Avitabile CM, Goldberg DJ, Zemel BS, et al. Deficits in bone density and structure in children and young adults following Fontan palliation. Bone. 2015;77:12-16.
Longmuir PE, Russell JL, Corey M, Faulkner G, McCrindle BW. Factors associated with the physical activity level of children who have the Fontan procedure. Am Heart J. 2011;161(2):411-417.
McCrindle BW, Williams RV, Mital S, et al. Physical activity levels in children and adolescents are reduced after the Fontan procedure, independent of exercise capacity, and are associated with lower perceived general health. Arch Dis Child. 2007;92(6):509-514.
Avitabile CM, Leonard MB, Zemel BS, et al. Lean mass deficits, vitamin D status and exercise capacity in children and young adults after Fontan palliation. Heart. 2014;100(21):1702-1707.
Kuwata S, Takanashi M, Hashimoto M, et al. Thyroid function in patients with a Fontan circulation. Am J Cardiol. 2019;123(6):979-983.
Hillsley MV, Frangos JA. Bone tissue engineering: the role of interstitial fluid flow. Biotechnol Bioeng. 1994;43(7):573-581.
McCarthy ID. Fluid shifts due to microgravity and their effects on bone: a review of current knowledge. Ann Biomed Eng. 2005;33(1):95-103.
Levey AS, Stevens LA, Schmid CH, et al. A new equation to estimate glomerular filtration rate. Ann Intern Med. 2009;150(9):604-612.
Gill TK, Hill CL, Shanahan EM, et al. Vitamin D levels in an Australian population. BMC Public Health. 2014;14:1001.
Cordina R, Ministeri M, Babu-Narayan SV, et al. Evaluation of the relationship between ventricular end-diastolic pressure and echocardiographic measures of diastolic function in adults with a Fontan circulation. Int J Cardiol. 2018;259:71-75.
Balady GJ, Arena R, Sietsema K, et al. Clinician's Guide to cardiopulmonary exercise testing in adults: a scientific statement from the American Heart Association. Circulation. 2010;122(2):191-225.
Jones NL, Makrides L, Hitchcock C, Chypchar T, McCartney N. Normal standards for an incremental progressive cycle ergometer test. Am Rev Respir Dis. 1985;131(5):700-708.
Wasserman KH, Sue D, Stringer W, Sietsema K, Sun X, Whipp B. Principals of Exercise Testing and Intrepretation. Philadelphia, PA: Lippincott Williams and Wilkins; 2012.
Henry MJ, Pasco JA, Pocock NA, Nicholson GC, Kotowicz MA. Reference ranges for bone densitometers adopted Australia-wide: geelong osteoporosis study. Australas Radiol. 2004;48(4):473-475.
Henry MJ, Pasco JA, Korn S, Gibson JE, Kotowicz MA, Nicholson GC. Bone mineral density reference ranges for Australian men: geelong osteoporosis study. Osteoporos Int. 2010;21(6):909-917.
Binkley N, Bilezikian JP, Kendler DL, Leib ES, Lewiecki EM, Petak SM. Summary of the international society for clinical densitometry 2005 position development conference. J Bone Miner Res. 2007;22(5):643-645.
Goldberg DJ, Dodds K, Avitabile CM, et al. Children with protein-losing enteropathy after the Fontan operation are at risk for abnormal bone mineral density. Pediatr Cardiol. 2012;33(8):1264-1268.
Bendaly EA, DiMeglio LA, Fadel WF, Hurwitz RA. Bone density in children with single ventricle physiology. Pediatr Cardiol. 2015;36(4):779-785.
Cordina R, von Klemperer K, Kempny A, et al. Echocardiographic predictors of mortality in adults with a Fontan circulation. JACC Cardiovasc Imaging. 2017;10(2):212-213.
Anker SD, Ponikowski PP, Clark AL, et al. Cytokines and neurohormones relating to body composition alterations in the wasting syndrome of chronic heart failure. Eur Heart J. 1999;20(9):683-693.
Liu LCY, Voors AA, van Veldhuisen DJ, et al. Vitamin D status and outcomes in heart failure patients. Eur J Heart Fail. 2011;13(6):619-625.
Gandhi S, Myers RB. Can parathyroid hormone be used as a biomarker for heart failure? Heart Fail Rev. 2013;18(4):465-473.
Arnett TR, Gibbons DC, Utting JC, et al. Hypoxia is a major stimulator of osteoclast formation and bone resorption. J Cell Physiol. 2003;196(1):2-8.
Witzel C, Sreeram N, Coburger S, Schickendantz S, Brockmeier K, Schoenau E. Outcome of muscle and bone development in congenital heart disease. Eur J Pediatr. 2006;165(3):168-174.
Holler F, Hannes T, Germund I, et al. Low serum 25-hydroxyvitamin D levels and secondary hyperparathyroidism in Fontan patients. Cardiol Young. 2016;26(5):876-884.
Tsampalieros A, Kalkwarf HJ, Wetzsteon RJ, et al. Changes in bone structure and the muscle-bone unit in children with chronic kidney disease. Kidney Int. 2013;83(3):495-502.
Chin AJ, Stephens P, Goldmuntz E, Leonard MB. Serum alkaline phosphatase reflects post-Fontan hemodynamics in children. Pediatr Cardiol. 2009;30(2):138-145.
Marino BS, Goldberg DJ, Dorfman AL, et al. Abnormalities in serum biomarkers correlate with lower cardiac index in the Fontan population. Cardiol Young. 2017;27(1):59-68.
Martins MJ, Azevedo I. Cardiac physiopathology and alkaline phosphatase. Pediatr Cardiol. 2009;30(1):91.
Schiff A, Yang J, Winner LH, Schwartz MC. Bone-specific alkaline phosphatase in patients who have undergone the Fontan operation. Pediatr Cardiol. 2016;37(7):1370-1376.
Janz KF, Letuchy EM, Eichenberger Gilmore JM, et al. Early physical activity provides sustained bone health benefits later in childhood. Med Sci Sports Exerc. 2010;42(6):1072-1078.