First steps to a clinical research unit for developmental research in paediatric cardiology: conception and progress of the LEADER project (Long Term Early Development Research) in CHD.

OBJECTIVE: We developed the Long-term Early Development Research (LEADER) project to investigate the development of children with CHD and/or after cardiopulmonary resuscitation. Both populations are at risk for delays in motor, cognitive, and language development. However, few studies to date have investigated the longitudinal development in these children. METHODS: To establish a clinical research unit, we planned three studies: a cross-sectional study in children after cardiopulmonary resuscitation (LEADER-REA Pilot Study), a longitudinal study in children after cardiopulmonary resuscitation, with a focus on evaluating various biomarkers as predictors for developmental outcome (LEADER-CPR study), and a longitudinal study in children with ventricular septal defect, tetralogy of Fallot, or transposition of the great arteries after cardiac surgery (LEADER-CHD study). RESULTS: Implementation of all three LEADER studies was successful and study protocols were conducted as planned. Findings from the LEADER-REA Pilot study have been recently published and data collection for both prospective trials is ongoing. Descriptive analysis of the first 20 assessments of the LEADER-CHD study showed no severe deficits in overall cognitive, motor, and language developments in the children. CONCLUSIONS: Children with CHD and/or after cardiopulmonary resuscitation are at risk for developmental delay. Therefore, a detailed developmental assessment is necessary as a pre-requisite for individual developmental support. Our LEADER project has been shown to be feasible in a clinical setting and is the first step towards the establishment of a clinical research unit in our clinic with a focus on longitudinal research.

Developmental Outcome in Infants with Cardiovascular Disease After Cardiopulmonary Resuscitation: A Pilot Study.

Unfavorable neurological outcome in children after cardiopulmonary resuscitation in infancy is frequent. However, few studies have investigated the development of these patients using comprehensive developmental tests and the feasibility of the Bayley Scales of Infant Development, 3rd Edition (BSID-III) has not been reported for this population. In this cross-sectional pilot study, we assessed the cognitive, language, and motor development in infants after cardiopulmonary resuscitation of ≥ 5 min with the BSID-III at the age of 12 or 24 months, depending on recruitment age. For analysis, 11 patients with in-hospital (n = 8) and out-of-hospital (n = 3) cardiac arrest were included. BSID-III results could not be quantified in three patients because of visual/hearing and/or motor impairment. In patients with quantifiable scores, 50.0% scored average in composite BSID-III scores, while the other 50.0% showed developmental delays, scoring distinctly below average. We conclude that the BSID-III is feasible for developmental assessment in the majority of the study population, but the use of instruments suitable for hearing/visually impaired and/or severely disabled infants is crucial to avoid biased results. Accurate characterization of developmental deficits is important to facilitate early identification and therapy of deficits.

Retraining of automatic action tendencies in individuals with obesity: A randomized controlled trial.

Obesity is a major health concern, characterized by an automatically activated tendency to (over)-eat. Recent research suggests that an effective way to counteract automatic approach tendencies in unhealthy consumption behavior might be approach bias modification. Therefore, we investigated an approach-avoidance training for unhealthy food cues in 189 patients with obesity of a psychosomatic inpatient clinic who were participating in a nutrition advice program. Patients in the active training group were trained to make avoidance movements (pushing a joystick) in response to unhealthy food pictures and approach movements (pulling the joystick) in response to positive pictures, while the control group received sham training (approaching and avoiding both picture types). Approach-avoidance bias, body mass index, eating pathology and food-specific implicit associations were assessed before and after the training. In line with our hypothesis, approach-avoidance bias improved in the active training group after the training, in comparison to the sham training group. Moreover, this effect generalized to new, untrained stimuli. However, no effects of the training were found in a food-specific Single-Target Implicit Association Test, or on eating pathology questionnaires or body mass index. While the training results are promising, the effect of approach-avoidance bias modification on relevant behavior in obesity has yet to be established before it may be implemented as an add-on treatment.

Educational level and employment status in adults with congenital heart disease.

Purpose Through this study we aimed to assess the educational level and employment status of adults with CHD in Germany. METHODS: Data were acquired from an online survey carried out in 2015 by the German National Register for Congenital Heart Defects. A total of 1458 adults with CHD participated in the survey (response rate: 37.6%). For 1198 participants, detailed medical information, such as main cardiac diagnosis and information from medical reports, was available. RESULTS: Of the participants surveyed (n=1198), 54.5% (n=653) were female, and the mean age was 30 years. The majority of respondents (59.4%) stated that they had high education levels and that they were currently employed (51.1%). Patients with simple CHD had significantly higher levels of education (p<0.001) and were more likely to be employed (p=0.01) than were patients with complex CHD. CONCLUSIONS: More than half of the participants had high education levels and the majority were employed. The association between CHD and its severity and individuals' educational attainment should be investigated more closely in future studies.

The German EMPATHIC-30 Questionnaire Showed Reliability and Convergent Validity for Use in an Intermediary/General Pediatric Cardiology Unit: A Psychometric Evaluation.

Background: Family-Centered Care is a useful framework for improving care for hospitalized children with congenital heart disease. The EMpowerment of PArents in THe Intensive Care-30 (EMPATHIC-30) questionnaire is a widely accepted tool to measure parental satisfaction with Family-Centered Care. Psychometric properties of the EMPATHIC-30 have been evaluated in neonatal and pediatric intensive care units, but not in pediatric cardiac care units. Therefore, our aim was to assess the psychometric properties of the German EMPATHIC-30 in an intermediary/general pediatric cardiology unit. Methods: We used data from a quality management survey comprising the German EMPATHIC-30, a sociodemographic questionnaire and four general satisfaction items. Data were collected at the intermediary/general pediatric cardiology unit of a specialized heart center in Germany (n = 366). We split the data randomly into two subsets. In the first subset, we assessed internal consistency reliability with McDonald's omega and Cronbach's alpha, and convergent validity using Spearman's rank correlation. Furthermore, we explored the internal structure with Principal Component Analysis (PCA). In the second subset, we validated the resulting structure using Confirmatory Factor Analysis (CFA). Results: The reliability estimates exceeded 0.70 for all five domain scores and 0.90 for the full-scale score. Convergent validity between EMPATHIC-30 domain scores/ the full-scale score and the four general satisfaction items was adequate (rs = 0.40-0.74). The PCA suggested three components, accounting for 56.8% of the total variance. Cross-validation via CFA showed poor model fit (χ2 = 1545.78, χ2/df = 3.85, CFI = 0.70, TLI = 0.66, RMSEA = 0.13), indicating that the EMPATHIC-30 shows no clear and generalizable factor structure in this sample. Discussion: The German version of the EMPATHIC-30 exhibited reasonable psychometric properties in an intermediary/general pediatric cardiology unit. Follow-up studies should investigate the factor structure of the EMPATHIC-30 in other pediatric inpatient care settings.

Family-Centered Care at Pediatric Cardiac Intensive Care Units in Germany and the Relationship With Parent and Infant Well-Being: A Study Protocol.

Rationale and Aim: Infants with Congenital Heart Disease (CHD) are at risk for neurodevelopmental delays, emotional, social and behavioral difficulties. Hospitalization early in life and associated stressors may contribute to these challenges. Family-centered Care (FCC) is a health care approach that is respectful of and responsive to the needs and values of a family and has shown to be effective in improving health outcomes of premature infants, as well as the mental well-being of their parents. However, there is limited empirical data available on FCC practices in pediatric cardiology and associations with parent and infant outcomes. Methods and Analysis: In this cross-sectional study, we will explore FCC practices at two pediatric cardiac intensive care units in Germany, assess parent satisfaction with FCC, and investigate associations with parental mental well-being and parenting stress, as well as infant physical and mental well-being. We will collect data of 280 infants with CHD and their families. Data will be analyzed using multivariate statistics and multilevel modeling. Implications and Dissemination: The study protocol was approved by the medical ethics committees of both partner sites and registered with the German registry for clinical trials (NR DRKS00023964). This study serves as a first step to investigate FCC practices in a pediatric cardiology setting, providing insight into the relationship between FCC and parent and infant outcomes in a population of infants with CHD. Results will be disseminated in peer-reviewed journals.

Education of Children With Cyanotic Congenital Heart Disease After Neonatal Cardiac Surgery.

BACKGROUND: The aim of the study was to evaluate the educational achievement of patients diagnosed with univentricular heart physiology (UVHP) or transposition of the great arteries (TGA) after neonatal cardiac surgery. METHODS: An exploratory online survey was performed with patients registered with the National Register for Congenital Heart Defects in Germany. For this publication, a subgroup analysis was conducted among patients diagnosed with TGA (n = 173; 36.3%) and UVHP (n = 304; 63.7%). RESULTS: Median age of the sample at school enrollment was 6 years (range, 5-8 years). The majority were enrolled at a standard elementary school (n = 368 of 477; 77.1%), although patients with UVHP were enrolled significantly more often at a special needs school (n = 52 of 304; 17.1%, TGA patients n = 11/ of 173; 6.4%, P < .001). A total of 45.8% (n = 66 of 144) of the patients graduated with a high school diploma. A substantial number of patients had been diagnosed with behavioral or learning disorders (TGA patients n = 63 of 173 [36.4%], UVHP patients n = 148 of 304 [48.7%]) and received early supportive therapy or remedial teaching before (TGA patients n = 89 of 173 [51.4%], UVHP patients n = 209 of 304 [68.8%]) and/or during their school careers (TGA patients n = 54 of 173 [31.2%], UVHP patients n = 120 of 304 [39.5%]). CONCLUSIONS: A large proportion of patients who underwent neonatal cardiac surgery graduated with a high school diploma. These results are of great importance to congenital heart defect patients, affected families, and treating physicians. Nevertheless, study participants, especially patients with UVHP, face some academic challenges. We conclude that long-term follow-up examinations and regular developmental assessments may be beneficial.

Appraisal-based cognitive bias modification in patients with posttraumatic stress disorder: a randomised clinical trial.

Background: Negative appraisals of the trauma and its sequelae play a crucial role in the development and maintenance of Posttraumatic Stress Disorder (PTSD). Experimental studies have shown promise in reducing negative appraisal through Cognitive Bias Modification (CBM) training. Objective: To determine whether an online CBM training designed to modify dysfunctional appraisals is successful in reducing appraisal bias in PTSD patients. Method: In this double-blinded 2-arm randomised clinical trial, 107 patients with PTSD were randomly allocated to active (n = 49) or control online CBM training (n = 57). Training comprised the completion of four sessions of online CBM training within one week. Change in bias, as measured by a scenario task and questionnaire (i.e. PostTraumatic Cognition Inventory), was the primary outcome. Secondary outcome included change in PTSD symptoms. Assessments took place prior to training, during training sessions, post-training and at 1- and 6-month follow-up. Results: Intent-to-treat analysis indicated that there was no interaction effect of condition by time. Regardless of training condition, participants showed a small to moderate decline in appraisal bias and PTSD symptoms from pre- to post-training. In both conditions, bias change during training sessions was related to decline in PTSD symptomatology following training. No moderators of outcome were found. Conclusions: There was no evidence that active training was more effective than control training in reducing dysfunctional appraisals. In both conditions, participants showed a decline in dysfunctional appraisals and PTSD symptoms following training. Importantly, bias reduction during training was related to PTSD symptom decline following training. Explanations and future research directions are discussed.

Antecedentes: Las valoraciones negativas del trauma y sus secuelas juegan un rol crucial en el desarrollo y mantención del Trastorno de Estrés Postraumático (TEPT). Estudios experimentales han mostrado promesa en reducir las valoraciones negativas a través de un entrenamiento de modificación de sesgo cognitivo (MSC).Objetivo: Determinar si un entrenamiento MSC en línea diseñado para modificar valoraciones disfuncionales es exitoso en reducir sesgos de valoración en pacientes con TEPT.Método: En este ensayo clínico randomizado doble ciego de 2 ramas, 107 pacientes con TEPT fueron asignados a entrenamiento MSC en línea activo (n=49) o control (n=57). El entrenamiento incluyó la realización de cuatro sesiones de entrenamiento MSC en línea dentro de una semana. El cambio en el sesgo, medido por un escenario de tareas y cuestionario (por ej. Inventario de Cogniciones Postraumáticas), fue el resultado primario. El resultado secundario incluyó cambios en los síntomas de TEPT. Las evaluaciones fueron realizadas antes del entrenamiento, durante las sesiones de entrenamiento, y posterior al tratamiento al mes y a los 6 meses de seguimiento.Resultados: El análisis del tipo intención de tratar indicó que no hubo efecto en la interacción de la condición según el tiempo. Pese a la condición de entrenamiento, los participantes mostraron una disminución leve a moderada en el sesgo de valoración y síntomas de TEPT desde el periodo anterior y posterior al entrenamiento. En ambas condiciones el cambio en el sesgo durante las sesiones de entrenamiento se relacionó con la disminución de la sintomatología de TEPT tras el entrenamiento. No se encontraron moderadores de resultados.Conclusiones: No hubo evidencia de que el entrenamiento activo fuera más efectivo que el entrenamiento control en reducir las valoraciones disfuncionales. En ambas condiciones, los participantes mostraron una disminución en las valoraciones disfuncionales y síntomas de TEPT tras el entrenamiento. De forma importante, la reducción del sesgo se relacionó con la disminución de sintomatología de TEPT tras el entrenamiento. Explicaciones y orientaciones sobre futura investigación fueron discutidas.

Exploring the developmental tasks of emerging adults after paediatric heart transplantation: a cross-sectional case control study.

OBJECTIVE: Paediatric heart transplantation (PHTX) comprises 12% of all cardiac transplants and many of the children now survive into adulthood. Only a few studies have investigated the long-term psychosocial well-being of young adult patients after PHTX; no studies have investigated developmental tasks of emerging adulthood in different domains (family, social environment, education and profession, partnership, social environment). SETTING: Specialised heart centre in Germany. PARTICIPANTS: Thirty-eight young adults aged 22.11 years (SD=4.7) who underwent PHTX and a control group of 46 participants with no known chronic diseases, aged 22.91 years (SD=1.8), participated in the study. OUTCOME MEASURES: All participants completed the following questionnaires: sociodemographic, the F-SozU, to measure perceived social support, the Gießener Beschwerde-Bogen to measure subjective complaints experienced by patients, the KIDSCREEN-27 to measure well-being and the SF-36 to measure health-related quality of life (QoL). RESULTS: 'Family': the quality of the relationship with the parents was found to be equal in both groups, while PHTX patients stayed in closer spatial proximity to their parents. 'Social environment': PHTX patients reported lower social support by peers than the control group. 'Education and profession': PHTX patients most often worked full-time (23%), had no job and/or received a pension (21%). In comparison, most of the healthy controls did an apprenticeship (40%) and/or worked part time (32%). 'Partnership': fewer of the PHTX patients had a partner than the control group while relationship duration did not differ. In exploratory regression analyses, social support by peers predicted physical QoL, whereas physical complaints and the physical role predicted mental QoL in PHTX patients. CONCLUSIONS: Our exploratory findings highlight important similarities and differences in specific developmental tasks between PHTX patients and healthy controls. Future studies should focus on developmental tasks of PHTX patients in this age group more systematically, investigating their role in physical and mental well-being in a confirmatory manner.

Long-term early development research in congenital heart disease (LEADER-CHD): a study protocol for a prospective cohort observational study investigating the development of children after surgical correction for congenital heart defects during the first 3 years of life.

INTRODUCTION: Congenital heart disease (CHD) is the most common birth defect. Studies on the development of children with CHD point towards deficits in motoric, cognitive and language development. However, most studies are cross-sectional and there is a gap in the knowledge concerning developmental trajectories, risk and protective factors and a lack of research concerning environmental predictors. Specifically, no studies have so far considered the importance of early caregiving experiences and child temperament for the development of children with CHD. METHODS: In a single-centre prospective cohort study, cognitive, motoric and language development of 180 children after corrective surgery for a simple transposition of the great arteries (TGA), tetralogy of Fallot (TOF) or ventricular septal defect (VSD) will be assessed at ages 12, 24 and 36 months with the Bayley Scales of Infant Development 3rd Edition (BSID-III). At age 12 months, a free-play video observation will be conducted to investigate the relationship between primary caregiver and child, and child temperament will be assessed with the Infant Behavior Questionnaire-Revised Short Version. Medical information will be obtained from patient records and demographic information via questionnaires. ANALYSIS: Frequency and severity of developmental delays will be reported descriptively. Differences between groups (TGA, TOF, VSD) will be subjected to repeated-measures analysis across time points. Multiple regressions will be applied for the analysis of predictors at each time point. For the analysis of differential developmental trajectories, mixed-model analysis will be applied. ETHICS AND DISSEMINATION: The study has been approved by the local medical ethics committee. Written informed consent will be obtained from all participants. Parents have the option to be debriefed about BSID-III results after each assessment and about the study results after project completion. Results will be disseminated in peer-reviewed journals and presented at conferences. TRIAL REGISTRATION NUMBER: DRKS00011006; Pre-results.

Changing prevalence of severe congenital heart disease: Results from the National Register for Congenital Heart Defects in Germany.

OBJECTIVE: To assess the prevalence of congenital heart disease (CHD) in Germany in relation to phenotypes, severity and gender. DESIGN: Cross-sectional registry study. SETTING: We analyzed data from patients with CHD born between 1996 and 2015. PATIENTS: A total of 26 630 patients, registered with the NRCHD, were born between 1996 and 2015. 10 927 patients were excluded from the current analysis due to prior registration with the NRCHD under the German PAN Prevalence Study, which showed a potential bias in the inclusion of this patient population (proportion of mild cardiac lesions was comparatively high due to improved diagnostic capability for earlier identifying minor lesions). At least 15 703 patients with demographic data and detailed medical information were included in the current study. INTERVENTIONS: None. OUTCOME MEASURES: Prevalence of CHD in Germany differentiated into gender, severity, and phenotype. RESULTS: In total, 15 703 patients with CHD (47.1% female) were included in this study. The five most common phenotypes were found to be ventricular septal defect (19.2%), atrial septal defect (13.0%), Tetralogy of Fallot (9.3%), univentricular heart (9.4%), and coractation of the aortae (7.0%). The prevalence of CHD in regard to severity changed over the duration of the observation period. From 1996 to 2007, the number of simple CHD rose steadily (P < .001), whereas the number of severe CHD has grown significantly since 2008/2009 (P < .001). In regard to gender, the prevalence of simple CHD was higher in females, whereas complex lesions were more common in males (P < .001). CONCLUSIONS: Our study shows a growing number of registered severe CHD in the recent decade in Germany. This development is noteworthy as it implicates a growing demand for first intensive hospital care, expert pediatric cardiologic aftercare, and consequently higher economic impact for this patient population.

Always Approach the Bright Side of Life: A General Positivity Training Reduces Stress Reactions in Vulnerable Individuals.

Emotional disorders are characterized by cognitive biases towards negative stimuli, and a lack of biases towards positive ones. Therefore, we developed a cognitive bias modification training, modifying approach-avoidance tendencies to diverse emotional pictures. In Study 1, a negative training (pull negative, push positive pictures) was compared to a positive training (vice versa) in 141 students. The pre-existing positivity bias remained after positive training, but reversed into a negativity bias after negative training. This effect transferred to an attentional bias. The training affected neither mood nor emotional vulnerability to stress. In Study 2, we investigated the effects of the positive training in 102 dysphoric and non-dysphoric students, all in a sad mood state. Compared to placebo training, the positive training strengthened a positivity bias, and it reduced emotional vulnerability in dysphoric students. This suggests potential therapeutic value of the training, but further studies are needed.