Karolijn Dulfer

Systematic review of the effects of physical exercise training programmes in children and young adults with congenital heart disease

BACKGROUND Most patients with congenital heart disease (ConHD) do not perform regular physical exercise. Consensus reports have stated that exercise should be encouraged and regularly performed in these patients, but this is not common practise. We reviewed the literature on actual evidence for either negative or positive effects of physical exercise training programmes in children and young adults with ConHD. METHODS Using the Medline database, we systematically searched for articles on physical exercise training programmes in ConHD. RESULTS A total of 31 articles met all inclusion criteria; in total, 621 subjects (age range 4 to 45 years) were included. Most studies used training programmes with a duration of 12 weeks. On average, the number of training sessions was 3 times per week. In 12 studies, training intensity was set at a percentage of peak heart rate. Outcome measures reported were PeakVO2, activity levels and muscle strength. Twenty-three studies (72%) found a significant positive change in the main outcome measure after the physical exercise training period. None of the studies reported negative findings related to physical exercise training in ConHD. Cardiac effects have hardly been studied. CONCLUSION In most studies, participation in a physical exercise training programme was safe and improved fitness in children and young adults with ConHD. We recommend that patients with ConHD participate in physical exercise training. Cardiac effects need to be studied more extensively.

Aerobic Exercise Influences Quality of Life of Children and Youngsters With Congenital Heart Disease: A Randomized Controlled Trial

PURPOSE To evaluate effects of an exercise program on health-related quality of life (HRQoL) in children and adolescents with tetralogy of Fallot (ToF) or a Fontan circulation. METHODS Stratified, randomized, controlled intervention study conducted in five participating centers of pediatric cardiology in The Netherlands. In total, 93 patients, aged 10-25 years, with surgical repair for tetralogy of Fallot or with a Fontan circulation for single-ventricle physiology were included. They were randomly allocated with a ratio of 2:1 to: (1) a 12-week period with an exercise program for 3 times per week or (2) to a control group. Randomization was stratified by age, gender, and cardiac diagnosis. At baseline and follow-up after 12 weeks, all participants completed Web-based age-appropriate HRQoL questionnaires. Primary analyses involved change in HRQoL during follow-up. Secondary analyses concerned influence of cardiac diagnosis and comparison with normative data. RESULTS Forty-eight (86%) and 32 (86%) patients in the exercise-group and control-group respectively completed all questionnaires at baseline and follow-up. Compared with the control-group, children, aged 10-15 years, in the exercise-group improved significantly on self-reported cognitive functioning, p < .05, r = .30, and parent-reported social functioning, p < .05, r = .30. Youngsters aged 16-25 years did not change their HRQoL. Cardiac diagnosis had no influence on pre/post changes. Children and youngsters in this study reported comparable or better HRQoL than norm groups. CONCLUSIONS Participation in an exercise program improved HRQoL of children with ToF or a Fontan circulation, especially in those with low baseline QoL.

Associations between exercise capacity, physical activity, and psychosocial functioning in children with congenital heart disease: a systematic review:

Background Children and adolescents operated upon for congenital heart disease (ConHD) may show reduced exercise capacity and physical activity, possibly associated with lowered self-esteem and quality of life (QoL). The studies into associations between these parameters have not been reviewed before. Objective Review of studies into associations between exercise capacity, physical activity, respectively exercise training, and psychosocial functioning of ConHD youngsters. Data sources PubMed, Embase and reference lists of related articles. Study selection Articles published between January 2000 and December 2012 into exercise capacity and/or physical activity, and a measure of psychosocial functioning in children with ConHD. Data extraction Two investigators independently reviewed the identified articles for eligibility, and one author extracted the data. Results Although exercise capacity was strongly related to physical domains of parent-reported and self-reported QoL, it was almost never associated with psychosocial domains of QoL. Physical activity was rarely associated with physical or psychosocial domains of QoL. Remarkably, self-reported depressive symptoms were associated with both physical and psychosocial QoL. The few studies into exercise-training programmes showed promising results in QoL and emotional and behavioral problems, but they contained methodological flaws. Conclusions No clear relationships were found between exercise capacity, physical activity, and QoL in children and adolescents with ConHD. Therefore we recommend assessing QoL separately, preferably both self-reported and parent-reported. Since depressive symptoms were associated with reduced physical and psychosocial QoL, screening on these symptoms is also recommended.

Depression and anxiety symptoms as predictors of mortality in PCI patients at 10 years of follow-up.

Background Depression has been shown to be an independent risk factor for short-term mortality in patients with coronary artery disease (CAD). There are studies suggesting that depression might also be associated with long-term mortality. Anxiety has also been associated with mortality. This study aimed to further investigate the predictive value of depression and anxiety symptoms on all-cause mortality, 10 years after percutaneous coronary intervention (PCI). Methods The study population comprised a consecutive series of CAD patients (n = 1411) treated with PCI between September 2001 and October 2002 at the Erasmus Medical Centre, Rotterdam. The Hospital Anxiety and Depression Scale (HADS) was completed by 1112 patients at baseline to assess levels of depression and anxiety. The endpoint was defined as all-cause mortality. Results The prevalence of depression and anxiety was 24.8% and 27.7%, respectively. The cumulative all-cause mortality rate in depressed patients was 37% versus 20% in non-depressed patients (log-rank p < 0.001). After adjustment, depression remained a predictor of all-cause mortality (hazard ratio (HR) 1.77; 95% confidence interval (CI) 1.36–2.29). Cumulative survival rates did not differ for anxious versus non-anxious patients (log-rank p = .79). However, after adjustment, anxiety was associated with an increased risk for all-cause mortality (HR 1.50; 95% CI 1.14–1.98). A sub-analysis showed that cumulative survival rates did not differ for depressed and anxious patients versus depressed but non-anxious patients (log-rank p = 0.46). Conclusions Depression is associated with an increased risk of 77% for all-cause mortality, 10 years post-PCI, independently of anxiety. Although anxiety was associated with all-cause mortality, it has no additional value in the case of co-occurring depression.

The effect of exercise training on cardiac remodelling in children and young adults with corrected tetralogy of Fallot or Fontan circulation: A randomized controlled trial

BACKGROUND Exercise can improve physical fitness in children and adults with congenital heart disease. We hypothesized that exercise training would not lead to adverse cardiac remodelling in this population. METHODS AND RESULTS This multi-centre randomized controlled trial included children and young adults (10 to 25 years) with either corrected tetralogy of Fallot or Fontan circulation. The exercise-group was enrolled in a 12 week standardized aerobic dynamic exercise training program. The control-group continued their life-style and received care as usual. Both groups underwent cardiopulmonary exercise testing, cardiac magnetic resonance imaging (MRI), echocardiography and neurohormonal assessment, within 2 weeks before and 2 weeks after the intervention period. Fifty-six patients were randomized to the exercise-group and 37 to the control-group. We assessed changes between the pre- and the post-intervention period for the exercise group compared to the changes in the control-group. Peak load increased significantly in the exercise-group compared to the control-group (exercise-group 6.9 ± 11.8 W; control-group 0.8 ± 13.9 W; p=0.047). There were no adverse events linked to the study. Ventricular systolic parameters, cardiac dimensions and neurohormonal markers during follow-up did not change in patients allocated to the exercise-group and control-group. Although there were some isolated minor changes in inflow parameters, there was no consistent pattern of changes, indicating a lack of true change in the diastolic function. CONCLUSION We demonstrated that no clinically relevant adverse cardiac remodelling occurred after 12 weeks of exercise training in patients with either corrected tetralogy of Fallot or Fontan circulation. CLINICAL TRIAL REGISTRATION www.trialregister.nl, identification NTR2731.

Parental Mental Health Moderates the Efficacy of Exercise Training on Health-Related Quality of Life in Adolescents with Congenital Heart Disease

Abstract To evaluate the moderating influence of parental variables on changes in health-related quality of life (HRQoL) in adolescents with Tetralogy of Fallot (ToF) or a Fontan circulation after participation in standardized exercise training. A multicenter randomized controlled trail in which 56 patients, aged 10–15, were randomly allocated (stratified by age, gender, and congenital heart disease) to a 12-week period with either: (a) 3 times per week standardized exercise training or (b) care-as-usual (randomization ratio 2:1). Adolescents and their parents filled in online questionnaires at baseline and at 12-week follow-up. In this randomized controlled trail, primary analyses involved influence of parental mental health and parental social support for exercise on changes in the TNO/AZL Child Quality of Life Questionnaire Child Form at follow-up. Secondary analyses concerned comparing levels of parental characteristics with normative data. Compared with controls, adolescents in the exercise group reported a decrease in social functioning when their parents had more anxiety/insomnia or severe depression themselves. Adolescents also reported a decrease in social functioning when their parents showed poorer overall mental health themselves. Parents reported comparable or even better mental health compared with normative data. The effect of a standardized exercise program on HRQoL changes in adolescents with ToF or a Fontan circulation is moderated by parental mental health, more specifically by parental anxiety/insomnia and severe depression. The trial registration number of this article is NTR2731 (www.trialregister.nl).

Effect of exercise training on sports enjoyment and leisure-time spending in adolescents with complex congenital heart disease: the moderating effect of health behavior and disease knowledge.

OBJECTIVE The aim of this study was to evaluate the effects of a standardized exercise program on sports enjoyment and leisure-time spending in adolescents with congenital heart disease and to know what the moderating impact of their baseline health behavior and disease knowledge is. METHODS Included were 93 patients, aged 10 to 25, with surgical repair for tetralogy of Fallot or with a Fontan circulation for single-ventricle physiology, of 5 participating centers of pediatric cardiology in The Netherlands. They were randomly allocated, stratified for age, gender, and type of congenital heart disease to a 12-week period with either: (1) three times per week standardized exercise training or (2) care as usual (randomization ratio 2:1). At baseline and after 12 weeks, participants completed Web-based questionnaires and were interviewed by phone. OUTCOME MEASURES Primary analyses tested changes from baseline to follow-up in sports enjoyment and leisure-time spending in the exercise group vs. control group. Secondary analyses concerned the moderating influence of baseline health behavior and disease knowledge on changes from baseline to follow-up, and comparison with normative data. RESULTS At follow-up, the exercise group reported a decrease in passive leisure-time spending (watching television and computer usage) compared with controls. Exercise training had no effect on sports enjoyment and active leisure-time spending. Disease knowledge had a moderating effect on improvement in sports enjoyment, whereas health behavior did not. Compared with normative data, patients obtained similar leisure time scores and lower frequencies as to drinking alcohol and smoking. CONCLUSIONS Exercise training decreased passive, but not active, leisure-time spending. It did not influence sports enjoyment.

Does functional health status predict health-related quality of life in children after Fontan operation?

PURPOSE It is important to identify those children with a Fontan circulation who are at risk for impaired health-related quality of life. We aimed to determine the predictive value of functional health status - medical history and present medical status - on both physical and psychosocial domains of health-related quality of life, as reported by patients themselves and their parents. METHODS We carried out a prospective cross-sectional multi-centre study in Fontan patients aged between 8 and 15, who had undergone staged completion of total cavopulmonary connection according to a current technique before the age of 7 years. Functional health status was assessed as medical history - that is, age at Fontan, type of Fontan, ventricular dominance, and number of cardiac surgical procedures - and present medical status - assessed with magnetic resonance imaging, exercise testing, and rhythm assessment. Health-related quality of life was assessed with The TNO/AZL Child Questionnaire Child Form and Parent Form. RESULTS In multivariate prediction models, several medical history variables, such as more operations post-Fontan completion, lower age at Fontan completion, and dominant right ventricle, and present medical status variables, such as smaller end-diastolic volume, a higher score for ventilatory efficiency, and the presence of sinus node dysfunction, predicted worse outcomes on several parent-reported and self-reported physical as well as psychosocial health-related quality of life domains. CONCLUSIONS Medical history and worse present medical status not only predicted worse physical parent-reported and self-reported health-related quality of life but also worse psychosocial health-related quality of life and subjective cognitive functioning. These findings will help in identifying patients who are at risk for developing impaired health-related quality of life.

Marital quality and loneliness as predictors for subjective health status in cardiac rehabilitation patients following percutaneous coronary intervention

Background Low marital quality is associated with adverse health outcomes and lower personal well-being. Loneliness increases the risk of cardiovascular disease and mortality and predicts poor quality of life. The aim of this study was to investigate the association between marital quality and loneliness and subjective health status in primary percutaneous coronary intervention (pPCI) patients who underwent cardiac rehabilitation (CR). Design/methods In a prospective cohort study, pPCI patients that followed CR were included between 2009–2011. A total of 223 patients responded to the Short Form 12 (SF-12) (subjective health status), Maudsley Marital Questionnaire (MMQ-6) (marital quality) and University of California, Los Angeles – Revised (UCLA-R) questionnaires at baseline (pre-CR) and at three months (post-CR) or at 12 months follow-up. Subjective health status is displayed by a physical component summary (PCS) score and a mental component summary (MCS) score. Generalized estimating equation (GEE) analyses were performed to test improvements in subjective health status. Results Changes over time in subjective health status scores were similar between patients with optimal marital quality vs patients with less optimal marital quality and non-lonely patients vs lonely patients. The MCS level at one-year follow-up of both patients with less optimal marital quality and lonely patients was lower compared with a healthy Dutch population (respectively; mean MCS score 47.3 (standard deviation (SD) 10.5); p = 0.013 and mean MCS score 46.1 (SD 11.2); p = 0.010). Conclusion Both patients with less optimal marital quality and lonely patients did not reach the MCS level of a healthy Dutch population. Therefore, extra care and support should be given to these patients in a CR programme.

Predictive value of depression and anxiety for long-term mortality: differences in outcome between acute coronary syndrome and stable angina pectoris

BACKGROUND Since the early 2000s the treatment of choice for an acute myocardial infarction has moved from thrombolytic therapy to primary PCI (pPCI). As a result, the majority of patients undergoing PCI shifted from stable angina pectoris (SA) to acute coronary syndrome (ACS). Additionally the previously observed association between depression and anxiety and long-term outcome in patients who underwent a PCI may have been changed. The main objective of this study was to investigate the predictive value of depression and anxiety for 10-year mortality, in a cohort with post-PCI patients treated for SA versus patients treated for ACS. METHODS This prospective single center cohort consists of a consecutive series of patients (n=528) treated with PCI. At 1month post-PCI, patients completed the Dutch version of the Hospital Anxiety and Depression Scale (HADS). RESULTS After adjustment for baseline characteristics depression was associated with higher 10-year mortality post-PCI (HR 1.58 95% Confidence Interval [95% CI] 1.04-2.40). In the ACS population no association between depression and 10-year mortality was found (HR 1.05 95% CI 0.62-1.79), in contrast to the SA population (HR 1.97 95% CI 1.09-3.57). After additional adjustment for anxiety, depression was no longer associated with higher mortality. CONCLUSIONS Anxiety at baseline was associated with an increased 10-year mortality rate after PCI. Depression was also associated with higher 10-year mortality, however the association disappeared after additional adjustment for anxiety. This finding was more pronounced in patients presenting with SA as compared to those presenting with ACS, which might be a result of the increasing number of ACS patients treated with pPCI.