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Objective: This study examines the association between psychosocial risk and protective factors and a wide range of psychosocial outcomes including emotional, social, cognitive, and physical domains in childhood cancer survivors (CCS). Methods: CCS from the Dutch Childhood Cancer Survivor Study (DCCSS)‐LATER cohort (diagnosed 1963–2001) part 2 (age ≥ 18 years, diagnosed < 18 years, ≥ 5 years since diagnosis) completed questionnaires on psychosocial risk and protective factors (Benefit and Burden Scale, Illness Cognition Questionnaire, Rosenberg Self‐Esteem Scale, and Impact of Cancer Scale), and psychosocial outcomes (Hospital Anxiety and Depression Scale, Self‐Rating Scale for Post‐Traumatic Stress Disorder, TNO‐AZL Questionnaire for Adult Health‐Related Quality of Life, and Short Form‐36). Associations were assessed with regression analysis, adjusting for attained age, sex, number of health conditions, and time since diagnosis, while correcting for multiple testing (p < 0.004). Results: A total of 1382 CCS participated, all diagnosed ≥ 15 years ago. The mean age of participating CCS was 36 years, and 51% were female. Perceived benefit and burden, acceptance, and helplessness, self‐esteem and social support were associated with the psychosocial outcomes. In the models including all psychosocial factors, most associations with psychosocial outcomes were seen for self‐esteem (10×), and perceived burden (9×). Self‐esteem (all β ≤ 0.47) and perceived burden (all β ≤ 0.38) demonstrated strongest associations of medium/large size. Conclusions: Perceptions of childhood cancer, illness cognitions, self‐esteem, and social support play a role in explaining psychosocial functioning in CCS, outweighing the influence of socio‐demographic and medical variables. Addressing negative perceptions and reducing feelings of helplessness, while promoting acceptance, self‐esteem, and social support, could provide intervention targets for CCS who encounter psychosocial challenges. [ABSTRACT FROM AUTHOR]

Purpose: The KLIK method is a tool to systematically monitor and discuss Health Related Quality of Life (HRQOL) in clinical practice. It has been successfully used in clinical practice in The Netherlands, and has recently been implemented in survivorship care for young adult childhood cancer survivors (CCSs). This study evaluates implementation fidelity and satisfaction of CCSs and healthcare practitioners (HCPs) with the KLIK method in survivorship care. Methods: CCSs' HRQOL was monitored using the KLIK questionnaire (PedsQL generic 18–30 years). In a mixed-methods design, implementation fidelity was based on registrations, and user satisfaction was assessed with evaluation surveys (CCSs) and semi-structured interviews (CCSs, HCPs). Descriptive statistics and qualitative analysis methods were used. Results: A total of 245 CCSs were eligible for the study. Fidelity was 79.2% (194/245) for registration in the KLIK PROM portal, 89.7% (174/194) for completed KLIK questionnaires, 74.7% (130/174) for its discussion during consultation. Of the eligible CCSs, 17.6% (43/245) completed the study evaluation survey. Five CCSs and HCPs were invited for an interview and participated. CCSs (7.7/10) and HCPs (7.5/10) were satisfied with the KLIK method. Reported facilitators included increased insight into CCSs' functioning, improved preparation before, and communication during consultation, without lengthening consultation duration. Barriers included CCSs not always completing KLIK questionnaires, incomplete content of the KLIK questionnaire, and the need for customization for CCSs with cognitive disabilities. Conclusion: The KLIK method is a feasible and valuable tool to systematically monitor and discuss HRQOL in survivorship care. Integration of the KLIK method within the organization is essential, with structural support in reminding CCSs to complete questionnaires. [ABSTRACT FROM AUTHOR]

Young adult childhood cancer survivors (YACCS) are a vulnerable group in need of psychosocial support, but tailored interventions are lacking. To examine feasibility and satisfaction, and to explore preliminary effectiveness of an online group intervention (Op Koers Online for YACCS) aimed at teaching active coping skills and providing peer-contact, thereby reducing and preventing psychosocial problems in YACCS. The intervention is based on psycho-education, cognitive behavioral therapy, and aspects of acceptance and commitment therapy. YACCS completed questionnaires pre- and post-intervention. Feasibility was based on attendance, drop-out, and an evaluation questionnaire was administered to assess satisfaction. Preliminary effectiveness was evaluated with the Mastery Scale, Illness Cognition Questionnaire, Distress Thermometer, Impact of Cancer - Childhood Survivors, and Pediatric Quality of Life Inventory. Preliminary effectiveness was investigated by testing differences on the psychosocial outcomes (coping and psychosocial wellbeing) between T0 and T1 within respondents, using paired samples t tests and Cohen's d. 10 YACCS participated in the intervention and completed all questionnaires. There was no drop-out; 90% of participants attended five out of six sessions. Overall, participants were satisfied with the intervention; 7.6 on a 0-10 scale. Distress (Cohen's d=-.6, p=.030) and feelings of helplessness (Cohen's d=-.8, p=.001) reduced from T0 to T1, while self-efficacy (Cohen's d=.8, p=.013,) improved. Other outcomes displayed small effects, but did not change significantly. This first, small pilot study showed short-term decrease in distress and feelings of helplessness and improvement of self-efficacy. Op Koers Online was positively evaluated by YACCS and course leaders, filling a gap in psychosocial services for YACCS. [ABSTRACT FROM AUTHOR]

Background: This study compares a comprehensive range of psychosocial outcomes of adult childhood cancer survivors (CCS) to general population‐based references and identifies sociodemographic and medical risk factors. Methods: CCS from the Dutch Childhood Cancer Survivor Study (DCCSS)‐LATER cohort (diagnosed 1963–2001) part 2 (attained age ≥18 years, diagnosed <18 years, ≥5 years since diagnosis) completed the Rosenberg Self‐Esteem Scale, Hospital Anxiety and Depression Scale, Distress Thermometer, Self‐Rating Scale for Post‐Traumatic Stress Disorder, and the Short Form‐36 (Health Related Quality of Life). CCS' scores were compared with references using analysis of variances and logistic regression analysis, controlling for age and sex (p <.05). Risk factors for worse psychosocial outcomes were assessed with regression analyses (p <.05). Results: CCS, N = 1797, mean age 35.4 years, 49.0% female, all ≥15 years since diagnosis, participated. Three percent reported posttraumatic stress disorder because of childhood cancer and 36.6% experienced clinical distress. CCS did not differ from references on self‐esteem and anxiety but were less depressed (d = −.25), and scored poorer on all health‐related quality of life scales, except for bodily pain (.01 ≤ d ≥ −.36). Female sex, lower educational attainment, not being in a relationship, and being unemployed were negatively associated with almost all psychosocial outcomes. Except for a central nervous system tumor diagnosis, few medical characteristics were associated with psychosocial outcomes. Conclusion: CCS appear resilient regarding mental health but have slightly poorer health‐related quality of life than references. Sociodemographic characteristics and central nervous system tumors were related to most psychosocial outcomes, but no clear pattern was observed for other medical factors. Future studies should address additional factors in explaining CCS' psychosocial functioning, such as coping, social support, and physical late effects. Adult childhood cancer survivors appear resilient regarding mental health but have slightly poorer health‐related quality of life than reference patients. Sociodemographic characteristics and central nervous system tumors were related to most psychosocial outcomes, but no clear pattern was observed for other medical factors. [ABSTRACT FROM AUTHOR]

Purpose: The study aimed to compare the psychosocial development of young adult survivors of childhood cancer (YACCS) with a norm group of young adults from the general population. Methods: From 2017 to 2020, 558 YACCS (18–30 years, 51% female, 10.9% CNS cancer) who participated in the Dutch Childhood Cancer Survivor Study (DCCSS) LATER cohort (diagnosed 1963–2001) part 2 completed the Course of Life Questionnaire (CoLQ), assessing the achievement of milestones. Items were grouped into the scales autonomy, psychosexual, and social development. Differences between YACCS and norm group were examined with ANOVA and Cohen's d (CoLQ scales) and with logistic regression analysis and odds ratio (OR) (CoLQ items), for the total group and YACCS of CNS cancer. Results: The total group of YACCS did not report a less favorable psychosocial development than the norm group. YACCS of CNS cancer scored lower than the norm group (p < 0.001) on the scales autonomy (d = − 0.36) and psychosexual (d = − 0.46). Additionally, on half of the items of autonomy (0.25 ≤ OR ≤ 0.34), psychosexual (0.30 ≤ OR ≤ 0.48), and social (0.23 ≤ OR ≤ 0.47) development, YACCS of CNS cancer were less likely (p < 0.01) than the norm group to have achieved the milestones. Conclusion: Overall, psychosocial development of YACCS was as favorable as the norm, but YACCS of CNS cancer were at risk of an unfavorable psychosocial development in all domains. Monitoring psychosocial development should be included in the standards of psychosocial care, especially for CNS cancer patients and survivors, to be able to trace delay. Personalized interventions should be offered to improve the psychosocial development in an early stage. [ABSTRACT FROM AUTHOR]

Aim: This review aimed to provide a comprehensive overview of the psychosocial developmental trajectory of various diseases during childhood and adolescence.Methods: Studies of Dutch young adults aged 18-35 years, who had grown up with a chronic disease, were included if the Course of Life Questionnaire had been used to assess psychosocial developmental milestones in three domains: social, autonomy and psychosexual. Differences between the disease groups and the general population were presented as Cohen's d and odds ratios.Results: We included 17 studies comprising 1899 young adults, who had grown up with 18 different paediatric diseases. Psychosocial development was delayed in all three questionnaire domains. Remarkable findings with regard to specific milestones were as follows: less participation in sports clubs in the social domain, less likely to have had paid jobs in the autonomy domain and later sexual intimacy in the psychosexual domain. End-stage renal disease, galactosaemia (males), childhood cancer and orthotopic liver transplants were the most affected disease groups.Conclusion: Children and adolescents with chronic diseases risked delays in psychosocial development. This should be addressed by healthcare providers, along with the physical aspects of diseases, and they should focus on the optimal psychosocial development of the patient. [ABSTRACT FROM AUTHOR]

Introduction and Aim: Knowledge on patterns of beliefs about the illness (illness cognitions) can provide insight into individual differences in adjustment to haemophilia. The current study aimed to identify (a) which sociodemographic and disease characteristics were associated with illness cognitions and (b) which illness cognitions were associated with health‐related quality of life (HRQOL) in young adult men with haemophilia, besides sociodemographic and disease characteristics. Methods: Young adult men (18‐30 years) with haemophilia in the Netherlands participated in an online multicentre cross‐sectional study. Participants completed the Pediatric Quality of Life Inventory Young Adult version (PedsQL_YA). Potential sociodemographic determinants were assessed with the Course of Life Questionnaire (CoLQ) and illness cognitions with the Illness Cognition Questionnaire (ICQ). Multiple linear regression analyses were performed to assess potential determinants of illness cognitions and HRQOL. Results: Seventy young adult men with haemophilia (mean age 24.7 years, SD 3.5) participated. Born outside the Netherlands (β −0.24) and >1 bleed past 6 months (β −0.32) were associated with less acceptance of the disease. More acceptance was associated with better HRQOL in all domains: β 0.23‐0.39. More helplessness was associated with worse total (β −0.30) and physical (β −0.42) HRQOL. Disease benefits, sociodemographic and disease characteristics were not associated with HRQOL. Conclusion: Illness cognitions are associated with HRQOL in young adult men with haemophilia. Early recognition and identification of illness cognitions are important to facilitate support and psychosocial treatment to optimize young adults' well‐being. Extra attention is needed for young adult men with frequent bleeds because they are at risk of lowered levels of acceptance of the disease. [ABSTRACT FROM AUTHOR]

Introduction and Aim: Suboptimal health‐related quality of life and lowered employment rates found in a previous study in young adults (YA) with congenital coagulation disorders (CCD) in the Netherlands underline the need for more insight into professional functioning of YA with CCD and into determinants of professional functioning. Methods: Young adults (18‐30 years) with CCD participated in a cross‐sectional study. Professional functioning was assessed with the Work Productivity and Activity Impairment questionnaire (WPAI). Potential determinants were assessed with the Course of Life Questionnaire (CoLQ), Pediatric Quality of Life Inventory Young Adult version (PedsQL_YA), Illness Cognition Questionnaire (ICQ) and Haemophilia Activities List (HAL). Logistic regression analyses were performed in the complete sample of YA with CCD, and in YA men with haemophilia separately, to examine determinants of WPAI outcomes. Results: Ninety‐four YA (77 men; mean age 24.1 years, SD 3.5 and 17 women; mean age 24.5 years, SD 3.8) with CCD (74% haemophilia A/B) participated. 74.5% of YA were paid employed for on average 30 hours per week. Of these, more than a quarter reported work impairment. Older age and a non‐severe type of haemophilia (in the sample of YA men with haemophilia) were associated with successful (paid) employment. No variables were associated with professional functioning (expressed as Presenteeism and Overall work impairment) in patients with CCD or haemophilia. Conclusion: Three‐quarters of YA with CCD were successful in finding paid employment. Though absenteeism was low, YA with paid employment needs attention as a considerable part experienced work impairment. [ABSTRACT FROM AUTHOR]

Background: We analysed developmental outcomes from a clinical trial early in life and its follow-up at 10.7 years in 123 children with Down syndrome.Aims: To determine 1) strengths and weaknesses in adaptive functioning and motor skills at 10.7 years, and 2) prognostic value of early-life characteristics (early developmental outcomes, parental and child characteristics, and comorbidity) for later intelligence, adaptive functioning and motor skills.Methods and Procedures: We used standardized assessments of mental and motor development at ages 6, 12 and 24 months, and of intelligence, adaptive functioning and motor skills at 10.7 years. We compared strengths and weaknesses in adaptive functioning and motor skills by repeated-measures ANOVAs in the total group and in children scoring above-average versus below-average. The prognostic value of demographics, comorbidity and developmental outcomes was analysed by two-step regression.Outcomes and Results: Socialisation was a stronger adaptive skill than Communication followed by Daily Living. Aiming and catching was a stronger motor skill than Manual dexterity, followed by Balance. Above-average and below-average scoring children showed different profiles of strengths and weaknesses. Gender, (the absence or presence of) infantile spasms and particularly 24-month mental functioning predicted later intelligence and adaptive functioning. Motor skills, however, appeared to be less well predicted by early life characteristics.Conclusions and Implications: These findings provide a reference for expected developmental levels and strengths and weaknesses in Down syndrome. [ABSTRACT FROM AUTHOR]

Background: Like any child, children with Down syndrome (DS) affect the lives of their families. Most studies focus on the adaptation of parents and families of young children with DS, while relatively few studies include the perspective of fathers.Aims: To determine 1) whether mothers and fathers of 11 to 13-year-olds with DS differ from reference parents in health related quality of life (HRQoL) and family functioning, and 2) whether HRQoL in parents of children with DS changes over time, from when the child was 6-8 years old to when the child was 11-13 years old.Methods: 80 mothers and 44 fathers completed HRQoL and family functioning questionnaires. 58 parents (53 mothers) had completed the HRQoL-questionnaire in a previous study.Results: Mothers differed from reference mothers in one HRQoL-domain (Sexuality), while fathers' HRQoL did not significantly differ from reference fathers. Both mothers and fathers scored in the (sub)clinical range more frequently than reference parents in Total family functioning, and in the domains Partner relation and Social network. Furthermore, fathers scored in the (sub)clinical range more frequently than reference parents in Responsiveness and Organization. HRQoL showed no significant change over time.Conclusions and Implications: Our findings indicate frequent family functioning problems but few HRQoL problems in parents and families of children with DS. In offering care, a family based approach with special attention for partner relation and social functioning is needed. [ABSTRACT FROM AUTHOR]

Objectives: Childhood cancer may negatively impact childhood cancer survivors' (CCS) sexuality. However, this is an understudied research area. We aimed to describe the psychosexual development, sexual functioning and sexual satisfaction of CCS, and identify determinants for these outcomes. Secondarily, we compared the outcomes of a subsample of emerging adult CCS to the Dutch general population. Methods: From the Dutch Childhood Cancer Survivor Study LATER cohort (diagnosed 1963–2001), 1912 CCS (18–71 years, 50.8% male) completed questions on sexuality, psychosocial development, body perception, mental and physical health. Multivariable linear regressions were used to identify determinants. Sexuality of CCS age 18–24 (N = 243) was compared to same‐aged references using binomial tests and t‐tests. Results: One third of all CCS reported hindered sexuality due to childhood cancer, with insecure body the most often reported reason (44.8%). Older age at study, lower education, surviving central nervous system cancer, poorer mental health and negative body perception were identified as determinants for later sexual debut, worse sexual functioning and/or sexual satisfaction. CCS age 18–24 showed significantly less experience with kissing (p = 0.014), petting under clothes (p = 0.002), oral (p = 0.016) and anal sex (p = 0.032) when compared to references. No significant differences with references were found for sexual functioning and sexual satisfaction, neither among female CCS nor male CCS age 18–24. Conclusions: Emerging adult CCS reported less experience with psychosexual development, but similar sexual functioning and sexual satisfaction compared to references. We identified determinants for sexuality, which could be integrated in clinical interventions for CCS at risk for reduced sexuality. [ABSTRACT FROM AUTHOR]

The objective of this study was to investigate satisfaction with and feasibility of an online cognitive behavioral-based group intervention (OK Onco Online) for adolescent childhood cancer survivors (CCS). The intervention, carried out by pediatric psychologists, aimed to prevent psychosocial problems. High levels of satisfaction were reported by the psychologists (n &equals; 6) and the CCS (n &equals; 11, age 11–17). The dropout rate was very low. Some recommendations for optimizing the intervention were mentioned. In conclusion, the positive evaluations by the CCS and the psychologists indicate that the online chat intervention OK Onco Online is a promising innovative group intervention for adolescent CCS. Further research is needed to confirm feasibility and to establish efficacy. [ABSTRACT FROM PUBLISHER]

Background More and more pediatric patients reach adulthood. Some of them are successfully integrating in adult life, but many others are not. Possibly Illness cognitions (IC) - the way people give meaning to their illness/disability - may play a role in individual differences on long-term adjustment. This study explored the association of IC with disease-characteristics and Health Related Quality of Life (HRQoL), anxiety and depression in young adults with a disability benefit due to childhood-onset chronic condition. Methods In a cross-sectional study, young adults (22-31 years, N = 377) who claimed a disability benefit because of a somatic condition since childhood, completed the Illness Cognition Questionnaire (acceptance-helplessness-benefits), RAND-36 (HRQoL) and HADS (anxiety and depression) online. Besides descriptive statistics, linear regression analyses were conducted to predict (1) illness cognitions by age, gender and disease-characteristics, and (2) HRQoL (Mental and Physical Component Scale), Anxiety and Depression by illness cognitions, controlling for disease-characteristics, age and gender. Results Respectively 90.2%, 83.8% and 53.3% of the young adults with a disability benefit experienced feelings of acceptance, benefits and helplessness. Several disease-characteristics were associated with IC. More acceptance and less helplessness were associated with better mental (β = 0.31; β = -0.32) and physical (β = 0.16; β = -0.15) HRQoL and with less anxiety (β = -0.27; β = 0.28) and depression (β = -0.29; β = 0.31). Conclusions IC of young adult beneficiaries were associated with their HRQoL and feelings of anxiety and depression. Early recognition of psychological distress and negative IC might be a key to the identification of pediatric patients at risk for long-term dysfunction. Identification of maladaptive illness cognitions enables the development of psychosocial interventions to optimise their well-being and adaptation to society. [ABSTRACT FROM AUTHOR]

Highlights: [•] We analyzed predictors of HRQoL in parents of children with Down syndrome. [•] Psychosocial factors showed the strongest relations to parental HRQoL. [•] Mainly a supportive network and leisure time were related to better parental HRQoL. [Copyright &y& Elsevier]

Background: As the treatment of chronic or life-threatening diseased children has dramatically over recent decades, more and more paediatric patients reach adulthood. Some of these patients are successfully integrating into adult life; leaving home, developing psychosocially, and defining a role for themselves in the community through employment. However, despite careful guidance and support, many others do not succeed. A growing number of adolescents and young adults who have had a somatic disease or disability since childhood apply for disability benefits. The purpose of this study was to assess the health-related quality of life (HRQoL), anxiety and depression of young adults receiving disability benefits because of somatic conditions compared to reference groups from the general Dutch population and to explore factors related to their HRQoL, anxiety and depression. Methods: Young adults (N = 377, 22-31 yrs, 64.3% female) claiming disability benefits completed the RAND-36 and an online version of the HADS. Differences between respondents and both reference groups were tested using analysis of variance and logistic regression analysis by group and age (and gender). Regression analyses were conducted to predict HRQoL (Mental and Physical Component Scale; RAND-36) and Anxiety and Depression (HADS) by demographic and disease-related variables. Results: The respondents reported worse HRQoL than the reference group (-1.76 Physical Component Scale; -0.48 Mental Component Scale), and a higher percentage were at risk for an anxiety (29.7%) and depressive (17.0%) disorder. Better HRQoL and lower levels of anxiety and depression were associated with a positive course of the illness and the use of medical devices. Conclusions: This study has found worse HRQoL and feelings of anxiety and depression experienced by young adults claiming disability benefits. Healthcare providers, including paediatric healthcare providers, should pay systematic attention to the emotional functioning of patients growing up with a somatic condition in order to optimise their emotional well-being and adaptation to society during their transition to adulthood. Future research should focus on emotional functioning in more detail in order to identify those patients that are most likely to develop difficulties in emotional functioning and who would benefit from specific psychosocial support aimed at workforce participation. [ABSTRACT FROM AUTHOR]

The long-term survival of children with a central nervous system (CNS) tumour is improving. However, they experience late effects, including altered habits and patterns of sleep. We evaluated the presence and type of sleep disorders and daytime sleepiness in these children, and its associations with clinical characteristics and daily performance (fatigue and psychosocial functioning). In a cross-sectional study at the outpatient clinic of the Emma Children's Hospital AMC (February-June 2010), sleep, fatigue and psychosocial functioning were analysed in 31 CNS tumour patients (mean age: 11.8 years; 20 boys) and compared with 78 patients treated for a non-CNS malignancy (mean age: 9.7 years; 41 boys) and norm data. Questionnaires applied were the Sleep Disorder Scale for Children, the Epworth Sleepiness Scale, the Pediatric Quality of Life Inventory, and the Strengths and Difficulties Questionnaire. Sleeping habits and endocrine deficiencies were assessed with a self-developed questionnaire. Increased somnolence was found in CNS tumour patients compared with those with a non-CNS malignancy (8.8 ± 2.8 versus 7.5 ± 2.7; P < 0.05). Both patient groups reported more problems ( P < 0.01) than the norm with initiating and maintaining sleep. No specific risk factors were identified for a sleep disorder in CNS tumour patients, but their excessive somnolence was correlated with lower fatigue related quality of life (QoL) ( r = −0.78, P < 0.001) and worse psychosocial functioning ( r = 0.63, P < 0.001). In conclusion, children treated for a CNS tumour have increased somnolence, significantly increasing fatigue and worsening daily functioning. Further investigation should focus on possibilities to improve sleep quality and diminish fatigue. [ABSTRACT FROM AUTHOR]

Aim: A growing number of young adults with somatic diseases/disabilities since childhood apply for disability benefits. The achievement of psychosocial milestones while growing up (course of life) is assumed to be related to job participation. This study assessed the course of life of young adult beneficiaries with somatic limitations compared with peers from the general Dutch population. Methods: Young adult beneficiaries (22-31 years, N = 415) completed the Course of Life Questionnaire assessing the achievement of milestones on autonomy, psychosexual and social development and risk behaviour. Differences between respondents and peers were tested using analysis of variance and logistic regression analysis by group, age and gender. Associations were expressed as odds ratios (OR) with confidence intervals. Results: The beneficiaries achieved fewer milestones or achieved the milestones at a later age than peers. The differences were substantial: most effect sizes were moderate to large and most of the ORs lower than 0.5. Conclusion: Young adult beneficiaries with somatic limitations since childhood are at risk of a delayed course of life. Healthcare providers should pay systematic attention to the psychosocial developmental trajectory of their patients in order to optimize their development to adulthood and, consequently, create conditions for an optimal labour market position. [ABSTRACT FROM AUTHOR]

Abstract: Little is known on the impact of growing up with Fabry disease (FD) on psychosocial development. Children with FD may suffer from severe recurrent pains in hands and feet, gastro-intestinal symptoms and heat intolerance. These symptoms may influence quality of life and may interfere with a normal psychosocial development. It is important to evaluate psychosocial outcomes of patients with FD into adulthood to be able to optimize support of children with FD. The current cross-sectional questionnaire study investigated psychosocial development and quality of life of 28 young adults with FD, aged 18–35years (9 males, 19 females), using the Course of Life questionnaire and the Short Form-36 questionnaire for quality of life. The results were compared with an age-matched normative population. We found significant differences in the achievement of milestones in social development of male Fabry patients. The milestones that were affected were ‘going out to bars’ and ‘participation in sports activities’. Other than that FD patients appear to be able to achieve a rather normal psychosocial development until adulthood. Quality of life was decreased in Fabry males in the domains of physical functioning and bodily pain and of general health perception in females. [Copyright &y& Elsevier]

Objective: The present paper reports about the content and evaluation of a psycho-educational group intervention for children growing up with a history of cancer, Op Koers Oncologie (OK Onco). OK Onco is aimed at empowerment of survivors of childhood cancer by teaching disease-related skills. The purpose of this pilot study is to evaluate whether OK Onco was appropriate for use among paediatric survivors of childhood cancer. Methods: Eleven participating children and their parents completed questionnaires about the disease-related skills that were taught in the intervention, before the intervention and 0–4 weeks after the intervention. In addition, parents were interviewed in focus groups. Results: Positive outcomes were found on most items concerning disease-related skills. Remarkable improvements were found with respect to the intervention goals ‘social competence’ and ‘positive thinking’. The parents reported that sharing emotions and experiences with other survivors of childhood cancer was an important element of the OK Onco program, which underscores the benefit of a disease-specific program for cancer survivors. Conclusions: In conclusion, the results of the pilot study indicate that OK Onco is appropriate for paediatric survivors of childhood cancer. Implementation of the intervention is the next challenge. Copyright © 2008 John Wiley & Sons, Ltd. [ABSTRACT FROM AUTHOR]

Objectives: The aim of this study is to investigate parental emotional functioning during the first five years of continuous remission after the end of their child's treatment and to identify predictors of parental emotional functioning. Methods: Psychological distress and situation-specific emotional reactions were assessed in 122 mothers and 109 fathers from 130 families. Longitudinal mixed model analyses were performed to investigate to what extent generic and disease-related coping, family functioning and social support were predictive of parental emotional functioning over time. Results: Initial elevated levels of distress, disease-related feelings of uncertainty and helplessness returned to normal levels during the first two years after the end of treatment. Being more optimistic about the further course of the child's disease (predictive control) was correlated with lower psychological distress and less negative disease-related feelings, while more passive reaction patterns were correlated with higher psychological distress and more negative disease-related feelings. Conclusions: Although in general the parents of children with successfully treated cancer showed adequate emotional resilience, support for these parents should not stop when treatment ends. Parents in need of help can be identified on the basis of their coping abilities. Copyright © 2007 John Wiley & Sons, Ltd. [ABSTRACT FROM AUTHOR]

The aims of this study were to assess the following: (1) The impact of medical determinants on the course of life of survivors of childhood cancer and (2) the impact of the course of life on Quality of Life (Q0L) in young adulthood. A total of 353 Dutch cancer survivors, aged 18-30, completed the RAND-36 Health Survey and the Course-of-Life questionnaire. Multiple linear regression analyses were performed. Survivors of brain tumors and survivors having been treated with radiotherapy reported the achievement of significantly fewer milestones in the psychosexual and social domain than the other survivors. Survivors who achieved fewer milestones in the social domain scored worse on QoL. Health-care providers should help to minimize the harm for children who grow up with cancer by encouraging social and psychosexual development. Children should be encouraged to make friends and to participate in peer activities. [ABSTRACT FROM AUTHOR]

Children with chronic illness (CI) are at risk for internalizing problems, which reduce their quality of life, hamper treatment, and increase family stress. Accordingly, behavioral interventions are provided at the family level. However, the effects of parental involvement on child outcomes are not consistently beneficial. Therefore, it is relevant to study the working mechanisms. In the present study, we tested child coping and parenting stress as underlying mechanisms of the effect of an intervention for children and an additional group intervention for parents. Data were analyzed from a randomized controlled trial. Families of children with chronic illness (N = 120, child M age = 12.11 years, range 7.98–18.07) participated in a cognitive-behavioral-based group intervention and were randomized in the child-only intervention or parent–child intervention. Primary outcomes were parent- and child-reported internalizing problems, whereas the mediators were the use of child active coping skills and parenting stress. The causal model was tested with multilevel mediation analysis. Active coping skills and parenting stress stood out as significant mediators of the effect of the intervention on parent- and child-reported internalizing behavior (Cohen's d effect size range 0.29–1.57). When parents were involved in the intervention, children increased their use of active coping skills and parents decreased in parenting stress, which in turn improved child internalizing problems. Knowing that coping skills and parenting stress underlie the benefit of involving parents can be used for optimizing interventions for children with CI and addressing the risk of internalizing problems. Highlights: Parental involvement in interventions for children with chronic illness can be beneficial, however explanations are still unclear. In this randomized efficacy trial, child use of active coping skills and parenting stress appeared significant temporal mediators. Parental involvement supported child coping and diminished parenting stress, which in turn decreased child internalizing problems. Interventions for children with internalizing problems might be optimized by focusing on determined active components. [ABSTRACT FROM AUTHOR]

Background: This study was aimed at assessing fathers' and mothers' distress 6 months after a pediatric cancer diagnosis and at determining whether this is related to the level of family psychosocial risk 1 month after the diagnosis.Methods: A sample of 192 families completed the electronic Psychosocial Assessment Tool (ePAT) 1 month after the diagnosis. At 6 months after the diagnosis, 119 mothers and 98 fathers completed the Distress Thermometer for Parents (DT-P; of which n=132 had also completed the ePAT at baseline). The DT-P consists of a thermometer score ranging from 0 to 10 (with a score ≥ 4 indicating clinical distress), problem domains (total, practical, social, emotional, physical, cognitive, and parenting for children < 2 years old and for children ≥ 2 years old), and a desire for a referral. The DT-P scores of mothers and fathers were compared with the scores of a reference group of 671 mothers and 463 fathers with healthy children. Within the pediatric cancer group, the DT-P scores of families with elevated total ePAT-scores were compared with the DT-P scores of parents with universal ePAT scores.Results: Parents of children with cancer more often reported clinical distress on the DT-P than parents of healthy children (fathers, 59.2% vs 32.3%; P < .001; mothers, 63% vs 42.3%; P < .001) and reported more problems on all DT-P domains (P < .001 to P = .042) except for the parenting domain for children < 2 years old. Furthermore, the ePAT predicted parental distress 6 months after the diagnosis because parents with elevated ePAT scores reported more problems than parents with universal scores on the DT-P thermometer and most of the DT-P domains (P < .001 to P = 1.00).Conclusions: Initial ePAT risk scores at diagnosis are predictive of future mean levels of parental distress. Cancer 2018;124:381-90. © 2017 American Cancer Society. [ABSTRACT FROM AUTHOR]

Objective: To describe psychosocial outcomes among adult siblings of very long‐term childhood cancer survivors (CCS), to compare these outcomes to reference populations and to identify factors associated with siblings' psychosocial outcomes. Methods: Siblings of survivors (diagnosed <18 years old, between 1963 and 2001, >5 years since diagnosis) of the Dutch Childhood Cancer Survivor Study DCCSS‐LATER cohort were invited to complete questionnaires on HRQoL (TNO‐AZL Questionnaire for Adult's HRQoL), anxiety/depression (Hospital Anxiety and Depression Scale), post‐traumatic stress (Self‐Rating Scale for Post‐traumatic Stress Disorder), self‐esteem (Rosenberg Self‐Esteem Scale) and benefit and burden (Benefit and Burden Scale for Children). Outcomes were compared to a reference group if available, using Mann‐Whitney U and chi‐Square tests. Associations of siblings' sociodemographic and CCS' cancer‐related characteristics with the outcomes were assessed with mixed model analysis. Results: Five hundred five siblings (response rate 34%, 64% female, mean age 37.5, mean time since diagnosis 29.5) of 412 CCS participated. Siblings had comparable HRQoL, anxiety and self‐esteem to references with no or small differences (r = 0.08−0.15, p < 0.05) and less depression. Proportions of symptomatic PTSD were very small (0.4%−0.6%). Effect sizes of associations of siblings' sociodemographic and CCS cancer‐related characteristics were mostly small to medium (β = 0.19−0.67, p < 0.05) and no clear trend was found in the studied associated factors for worse outcomes. Conclusions: On the very long‐term, siblings do not have impaired psychosocial functioning compared to references. Cancer‐related factors seem not to impact siblings' psychosocial functioning. Early support and education remain essential to prevent long‐term consequences. [ABSTRACT FROM AUTHOR]

Abstract: Background: Phenylketonuria (PKU) is a rare inborn error of metabolism caused by phenylalanine hydroxylase enzyme (PAH) deficiency. Treatment constitutes a strict Phe restricted diet with unpalatable amino acid supplements. Residual PAH activity enhancement with its cofactor tetrahydrobiopterin (BH4) is a novel treatment which increases dietary tolerance in some patients and permits dietary relaxation. Relaxation of diet may improve health related quality of life (HRQoL). This prospective cohort study aims to evaluate HRQoL of patients with PKU and effects of BH4 treatment on HRQoL. Methods: Patients aged 4years and older, diagnosed through newborn screening and early and continuously treated, were recruited from eight metabolic centers. Patients and mothers completed validated generic and chronic health-conditions HRQoL questionnaires (PedsQL, TAAQOL, and DISABKIDS) twice: before and after testing BH4 responsivity. Baseline results were compared to the general population. Data collected after BH4 testing was used to find differences in HRQoL between BH4 unresponsive patients and BH4 responsive patients after one year of treatment with BH4. Also a within patient comparison was performed to find differences in HRQoL before and after treatment with BH4. Results: 69/81 (85%) patients completed the questionnaires before BH4 responsivity testing, and 45/69 (65%) participated again after testing. Overall PKU patients demonstrated normal HRQoL. However, some significant differences were found when compared to the general population. A significantly higher (thus better) score on the PedsQL was reported by children 8–12years on physical functioning and by children 13–17years on total and psychosocial functioning. Furthermore, adult patients reported significantly lower (thus worse) scores in the TAAQOL cognitive domain. 10 patients proved to be responsive to BH4 treatment; however improvement in their HRQoL after relaxation of diet could not be demonstrated. [Copyright &y& Elsevier]

Purpose: The Psychosocial Assessment Tool (PAT) is a brief family screener, identifying families at universal or elevated risk for psychosocial problems. This study aimed to determine the feasibility and usability of the electronic PAT (ePAT) in pediatric cancer care.Methods: Eighty-six parents of newly diagnosed children with cancer (0-18 years) agreed to participate and registered at the website www.hetklikt.nu (58%). Seventy-five families completed the ePAT at approximately 1 month post-diagnosis. Answers were transformed into an electronic PROfile (PAT ePROfile) and fed back to the psychosocial team. Team members completed a semi-structured evaluation questionnaire. Feasibility was measured as the percentage of website registrations, completed ePATs, and PAT ePROfiles reviewed or discussed by the team. Usability included perceived match of the PAT ePROfile with the team's own risk estimation, perceived added value, and perceived actions undertaken as a result of the PAT ePROfile.Results: Feasibility was 70% for website registration, 87% for completed ePATs, 85% for PAT ePROfile reviewing, and 67% for ePROfile discussion. Team members reported that the PAT ePROfile matched with their own risk estimation (M = 7.92, SD = 1.88) and did not provide additional information (M = 2.18, SD = 2.30). According to the team, actions were undertaken for 25% of the families as a result of the PAT ePROfile. More actions were undertaken for families with elevated risk scores compared to universal risk scores (p = .007).Conclusions: Implementation of the ePAT seems generally feasible, but it is not always clear how this screener adds to current clinical practice. Strategies should be developed together with team members to improve quick exchange of ePAT results and allocate care according to the needs of the families. [ABSTRACT FROM AUTHOR]

Background: To provide targeted support to parents of children with DS, knowledge of their distress and everyday problems is crucial. For this purpose, psychosocial screening instruments can be a valuable addition to routine clinical practice.Aims: To determine differences on a psychosocial screener concerning distress and everyday problems in parents of young adolescents (YAs) with DS versus control parents and in mothers of YAs with DS versus fathers.Methods and Procedures: We compared outcomes of the Distress Thermometer for Parents in 76 mothers and 44 fathers of 11-13-year-olds with DS versus 64 mothers and 52 fathers of age-matched children without DS (comparing mothers and fathers separately). Additionally, we compared mothers and fathers within 34 parent couples of YAs with DS.Outcomes and Results: Clinical distress was not more frequent than in control parents. Mothers further did not report more everyday problems and only differed from their controls on one problem domain and some problem items. Fathers, however, reported more problems than their controls across most domains and wished to talk to a professional about their situation more frequently. Outcomes in mothers and fathers within parent couples did not differ significantly.Conclusions and Implications: This is one of few studies to report on the use of psychosocial screening instruments in parents of children with DS. Our results suggested that attention for fathers of YAs with DS is required. Psychosocial screening instruments that inquire about specific problems and the wish for referral can play an important role in achieving this. [ABSTRACT FROM AUTHOR]

Introduction: Adverse events (AE) of treatment are prevalent and diverse in head and neck rhabdomyosarcoma (HNRMS) survivors. These AEs are often reported by physicians; however, patients' perceptions of specific AE are not well known. In this study, we explored patient‐reported outcomes measuring appearance, health‐related quality of life (HRQOL), and facial function in HNRMS survivors. Second, we assess the relationship between physician grading of AE and patient reporting. Materials and Methods: Survivors of pediatric HNRMS, diagnosed between 1993 and 2017, who were at least 2 years after completing treatment were invited to an outpatient clinic as part of a multicenter cross‐sectional cohort study. At the outpatient clinics, survivors aged ≥8 years filled out the FACE‐Q Craniofacial module; a patient‐reported outcome instrument measuring issues specific to patients with facial differences. AE were systematically assessed by a multidisciplinary team based on the Common Terminology Criteria of Adverse Events system. Results: Seventy‐seven survivors with a median age of 16 years (range 8–43) and median follow‐up of 10 years (range 2–42) completed the questionnaire and were screened for AEs. Patient‐reported outcomes varied widely between survivors. Many survivors reported negative consequences: 82% on appearance items, 81% on HRQOL items, and 38% on facial function items. There was a weak correlation between physician‐scored AEs and the majority of patient‐reported outcomes specific for those AEs. Conclusions: Physician‐graded AEs are not sufficient to provide tailored care for HNMRS survivors. Findings from this study highlight the importance of incorporating patient‐reported outcome measures in survivorship follow‐up. Physician graded adverse effects are not sufficient to provide tailored care for head and neck cancer survivors. Patient reported outcome scores for appearance, health related quality of life and facial function was reported negatively by many survivors. This study highlights the importance of incorporating patient reported outcome measures in follow‐up clinic. [ABSTRACT FROM AUTHOR]

Objective: To describe health‐related quality of life (HRQoL), post‐traumatic stress and post‐traumatic growth of parents of long‐term survivors of childhood cancer (CCS) and study associated factors. Methods: Parents of survivors of the Dutch Childhood Cancer Survivor Study LATER cohort below 30 years and diagnosed 1986–2001 were invited to complete the TNO‐AZL Questionnaire for Adult's HRQoL (e.g., sleep and aggressive emotions), Self‐Rating Scale for Post‐traumatic Stress Disorder, Post‐traumatic Growth Inventory, and Illness Cognition Questionnaire. HRQoL domain scores were compared to references using Mann‐Whitney U tests. Correlations between post‐traumatic stress, growth and HRQoL were evaluated. Medical characteristics of their child and illness cognitions were studied as associated factors of HRQOL, post‐traumatic stress and growth. p < 0.05 was considered statistically significant. Results: Parents (n = 661 of n = 448 survivors, 56% female, mean time since child's diagnosis: 21.3 [SD: 3.3] years) reported better HRQoL in social functioning and aggressive emotions than references (r =.08–0.17). Mothers additionally reported better HRQoL in pain, daily activities, sexuality, vitality, positive and depressive emotions (r =.07–0.14). Post‐traumatic stress was symptomatic in 3%, and associated with worse HRQoL (r = −0.27–0.48). Post‐traumatic growth was positively associated to post‐traumatic stress and better HRQoL (r = 0.09–0.12). Cancer recurrence was associated to better HRQoL (β = 0.37–0.46). Acceptance illness cognitions were associated to better (β = 0.12–0.25), and helplessness to worse outcomes (β = 0.14–0.38). Conclusions: HRQoL of parents of young adult survivors of CCS is comparable to references or slightly better. Only a small proportion reports symptomatic post‐traumatic stress. Improving acceptance and reducing feelings of helplessness may provide treatment targets for parents with psychosocial problems. [ABSTRACT FROM AUTHOR]

Simple Summary: Survivors of childhood cancer have an increased risk to experience symptoms of severe and persistent fatigue. We studied how fatigue might affect the health-related quality of life of these survivors. Questionnaire items asking about a broad range of daily life aspects were compared between fatigued survivors, survivors without fatigue and the general Dutch population. A total of eleven aspects were studied which were all negatively affected by fatigue, with the largest impact seen for Vitality (how much energy does a person have), General Health (perception of current and future health) and Role Limitations (work-related activities). Results show the negative impact fatigue can have on the daily lives of survivors and why it is important to treat fatigue adequately. Background: Early detection and management of late effects of treatment and their impact on health-related quality of life (HRQOL) has become a key goal of childhood cancer survivorship care. One of the most prevalent late effects is chronic fatigue (CF). The current study aimed to investigate the association between CF and HRQOL in a nationwide cohort of CCS. Methods: Participants were included from the Dutch Childhood Cancer Survivor Study (DCCSS) LATER cohort, a nationwide cohort of CCS. Participants completed the Checklist Individual Strength (CIS) to indicate CF (CIS fatigue severity subscale ≥ 35 and duration of symptoms ≥6 months) and the Short Form-36 (SF-36) and TNO (Netherlands Organization for Applied Scientific Research) and AZL (Leiden University Medical Centre) Adult's Health-Related Quality of Life questionnaire (TAAQOL) as measures for HRQOL. Differences in mean HRQOL domain scores between CF and non-CF participants were investigated using independent samples t-tests and ANCOVA to adjust for age and sex. The association between CF and impaired HRQOL (scoring ≥ 2 SD below the population norm) was investigated using logistic regression analyses, adjusting for confounders. Results: A total of 1695 participants were included in the study. Mean HRQOL domain scores were significantly lower in participants with CF. In addition, CF was associated with impaired HRQOL on all of the domains (except physical functioning) with adjusted odds ratios ranging from 2.1 (95% CI 1.3–3.4; sexuality domain) to 30.4 (95% CI 16.4–56.2; vitality domain). Conclusions: CF is associated with impaired HRQOL, urging for the screening and regular monitoring of fatigue, and developing possible preventative programs and interventions. [ABSTRACT FROM AUTHOR]

Objectives. It is generally recognized that for all children the fulfilling of age-specific psychosocial developmental tasks in childhood is of great importance to adjustment in adult life, including participation in society. For young adults with JIA this is more difficult. We assume that the achievement of psychosocial milestones while growing up (psychosocial developmental trajectory) is also related to labour participation. A proportion of all young adults with JIA have to apply for disability benefits. This study assessed the health-related quality of life (HRQOL) and the psychosocial developmental trajectory of young female beneficiaries with JIA compared with peers from the Dutch general population.Methods. Young females with disability benefits because of JIA completed the RAND-36 (HRQOL) and the Course of Life Questionnaire (psychosocial developmental trajectory). Differences between respondents and the peer group were tested using analysis of variance and logistic regression analysis by group and age.Results. The beneficiaries reported worse HRQOL than the peer group and achieved fewer milestones, or achieved the milestones at a later age than the peer group in the autonomy, social and psychosexual domain.Conclusions. Young females with JIA who have to apply for disability benefits are at risk for impaired HRQOL and a delay in their psychosocial developmental trajectory. Parents, physicians and other health-care providers should pay systematic attention to the development of social and independent functioning of children with JIA in order to optimize their adaptation to society at the time of transition to adulthood. [ABSTRACT FROM PUBLISHER]

Background: Much is written about cognitive and motor development; less is known about social and emotional consequences of growing up with congenital hypothyroidism (CH). The objectives of the study were: (1) to compare health related quality of life (HRQoL) and self-worth of 10 year old patients with CH with the general population; (2) to explore associations of disease factors, IQ and motor skills with the outcomes. Methods: Children with CH and their parents completed several questionnaires. Patients were classified to 'severe CH, n = 41' or 'moderate/mild CH, n = 41' based on pre-treatment FT4 concentration. Differences between CH and the general population were tested by analysis of covariance and one sample t-tests (mean scale scores HRQoL and self-worth), chi-square tests and binomial tests (% at risk of impaired HRQoL and self-worth). Linear regression analyses corrected for gender were conducted to explore associations of the outcomes with disease factors, IQ and motor skills. Results: Patients with CH reported lower mean HRQoL on motor, cognitive and social functioning, and on autonomy and positive emotions (p < 0.0001). Patients were also more often at risk for impaired HRQoL and self-worth. No differences were found between the severity groups. Lower IQ was only significant associated with worse cognitive HRQoL. Initial FT4 plasma, age at onset of therapy, initial T4 dose and motor skills were not significantly associated with HRQoL and self-worth. Conclusions: Negative consequences in terms of HRQoL and self-worth are prevalent in children with CH, independent of disease factors, IQ and motor skills. Physicians should to be attentive to these consequences and provide attention and supportive care. [ABSTRACT FROM AUTHOR]

Background: Children with functional constipation report impaired Health-related Quality of Life (HRQoL) in relation to physical complaints and long duration of symptoms. In about one third of children with constipation, symptoms continue into adulthood. Knowledge on HRQoL in adults with constipation persisting from childhood is lacking. Objectives: To assess HRQoL in adults with constipation from early childhood in comparison to that of their peers. Furthermore to gain insight into the specific social consequences related to continuing symptoms of constipation and/or fecal incontinence at adult age. Methods: One HRQoL questionnaire and one self-developed questionnaire focusing on specific consequences of symptoms of constipation continuing into adulthood were administrated to 182 adults with a history of childhood constipation. Successful clinical outcome was defined as a defecation frequency three or more times per week with less than two episodes of fecal incontinence per month, irrespective of laxative use. HRQoL of both adults with unsuccessful and successful clinical outcome were compared to a control group of 361 peers from the general Dutch population. Results: No differences in HRQoL were found between the whole study population and healthy peers, nor between adults with successful clinical outcome (n = 139) and the control group. Adults with an unsuccessful clinical outcome (n = 43) reported significantly lower HRQoL compared to the control group with respect to scores on bodily pain (mean ± SD 77.4 ± 19.6 versus 85.7 ± 19.5, p = 0.01) and general health (67.6 ± 18.8 versus 74.0 ± 18.1, p = 0.04). Adults with an unsuccessful clinical outcome reported difficulties with social contact and intimacy (20% and 12.5%, respectively), related to their current symptoms. Current therapy in these adults was more often self-administered treatment (e.g. diet modifications) (60.4%) than laxatives (20.9%). Conclusion: Overall, young adults with constipation in childhood report a good quality of life, as HRQoL of adults with successful clinical outcome was comparable to that of their peers. However, when childhood constipation continues into adulthood, it influences HRQoL negatively with social consequences in 20% of these adults. [ABSTRACT FROM AUTHOR]

Background: The objective of this study was to compare the health‐related quality of life (HRQOL) of Dutch adult male and female childhood cancer survivors (CCSs) to general population references and to study medical determinants. Methods: CCSs from the Dutch Childhood Cancer Survivor Study LATER cohort (1963‐2001) part 2, who were 18 years old or older (time since diagnosis ≥ 5 years), were invited to complete the TNO‐AZL Questionnaire for Adult Health‐Related Quality of Life. Domain scores and proportions of CCSs with impaired HRQOL (score < 25th percentile of the reference scores) were compared with references via Mann‐Whitney U tests and logistic regression analyses corrected for age and sex (P <.004). Interactions of group with sex were included if they were significant (P <.05). Moreover, medical determinants were analyzed with multivariable logistic regression analyses. Results: HRQOL scores for 1766 CCSs (mean age, 35.9 years [standard deviation, 9.4 years]; male, 51%; response rate, 71%) differed from references on most domains with small effect sizes. Both male and female CCSs were more often impaired in gross and fine motor functioning, cognitive functioning, sleep, and vitality with odds ratios (ORs) > 1.4. In addition, female CCSs were more often impaired in daily activities, pain, and sexuality (ORs, 1.4‐1.9) and were less often aggressive (OR, 0.6). CCCs of central nervous system (CNS) tumors, bone tumors, and retinoblastoma and those with cranial, abdominopelvic, or lower extremity radiotherapy were at increased risk of impairment in 1 or more domains. Conclusions: Dutch adult CCSs, especially females, have impaired HRQOL in several domains; this is most pronounced in cognitive functioning. The vulnerabilities of subgroups at risk, such as CCSs of CNS tumors, were confirmed. Surveillance of HRQOL and multidisciplinary survivor care are recommended. Lay Summary: The health‐related quality of life in a Dutch nationwide cohort of 1766 survivors of childhood cancer was studied.Survivors of childhood cancer were found to have lower health‐related quality of life in several domains (eg, motor functioning and vitality) in comparison with the general population.They most often reported low cognitive functioning (eg, memory and attention).Females had low health‐related quality of life in more domains than males.Survivors of brain tumors had low health‐related quality of life in most domains.Monitoring health‐related quality of life regularly and collaborating between disciplines in survivor care is recommended. Dutch adult survivors of childhood cancer, especially females and central nervous system tumor survivors, have impaired health‐related quality of life in several domains; this is most pronounced in cognitive functioning. Surveillance of health‐related quality of life and multidisciplinary survivor care are recommended. [ABSTRACT FROM AUTHOR]

Aim: To compare Health‐Related Quality of Life (HRQOL) of paediatric patients with newly collected HRQOL data of the general Dutch population, explore responses to individual items and investigate variables associated with HRQOL. Methods: Children (8–12y) and adolescents (13–17y) from the general population (N = 966) and from a paediatric population (N = 1209) completed the Pediatric Quality of Life Inventory (PedsQLTM) online via the KLIK Patient‐Reported Outcome Measures portal. PedsQLTM scale scores were compared between groups with independent t tests, by age group and gender. Responses to PedsQLTM items were explored using descriptive analyses. Linear regression analyses were performed to determine which variables were associated with HRQOL. Results: Paediatric patients reported worse HRQOL than the general population on all PedsQLTM scales (p ≤.001, d = 0.20–1.03), except social functioning, and a high proportion reported problems on PedsQLTM items, for example, 'I have trouble sleeping'. Younger age, female gender and school absence were negatively associated with HRQOL (β = −0.37–0.10, p ≤.008). Conclusion: Paediatric patients reported lower HRQOL than the general population, and school absence, female gender and younger age were associated with lower HRQOL. The results underline the importance to structurally monitor paediatric patients' HRQOL in clinical practice to detect problems and offer the right help on time. [ABSTRACT FROM AUTHOR]

Objectives: Parents of children with a chronic illness (CI) are at risk for psychosocial problems. The aim of this study was to refine an existing face-to-face intervention into an online psychosocial group intervention for parents by (1) exploring which themes are important, (2) determine what type of intervention parents would like and (3) assess parents' practical preferences. Methods: Parents of children with a CI (0–18 years) were invited to complete an online questionnaire. To acquire more in-depth information, focus groups and telephone interviews were conducted. Descriptive statistics were used. Results: 272 parents (mean age = 43.1 years, 85% female) participated. Three focus groups (15 parents) and seven telephone interviews were conducted. Most important themes were: the CI of the child, family functioning, taking care of yourself, relationships with others and practical support. Parents preferred a group with parents of children in the same age category. At first, parents preferred face-to-face contact. After an explanation and demonstration of an online intervention, parents became more positive about online support, mostly because they could participate from home. Conclusions for Practice: Parents have a need for psychosocial support focusing on different themes. Professionals should explain and demonstrate an online intervention to parents. Based on these results, Op Koers Online for parents was developed. An RCT to assess feasibility and effectiveness of the intervention is currently running. [ABSTRACT FROM AUTHOR]

Objective: With increasing numbers of children growing up with conditions that are associated with acquired brain injury, efficient neuropsychological screening for cognitive deficits is pivotal. Brief self-report measures concerning daily complaints can play an important role in such screening. We translated and adapted the pediatric perceived cognitive functioning (PedsPCF) self- and parent-report item bank to Dutch. This study presents (1) psychometric properties, (2) a new short form, and (3) normative data for the short form. Methods: A general population sample of children and parents was recruited. Dimensionality of the PedsPCF was assessed using confirmatory factor analyses and exploratory bifactor analyses. Item response theory (IRT) modeling was used to evaluate model fit of the PedsPCF, to identify differential item functioning (DIF), and to select items for the short form. To select short-form items, we also considered the neuropsychological content of items. Results: In 1441 families, a parent and/or child participated (response rate 66% at family level). Assessed psychometric properties were satisfactory and the predominantly unidimensional factor structure of the PedsPCF allowed for IRT modeling using the graded response model. One item showed meaningful DIF. For the short form, 10 items were selected. Conclusions: In this first study of the PedsPCF outside the United States, studied psychometric properties of the translated PedsPCF were satisfactory, and allowed for IRT modeling. Based on the IRT analyses and the content of items, we proposed a new 10-item short form. Further research should determine the relation of PedsPCF outcomes with neurocognitive measures and its ability to facilitate neuropsychological screening in clinical practice. [ABSTRACT FROM AUTHOR]

Objective: The impact of childhood differentiated thyroid carcinoma (DTC) on psychosocial development has not yet been studied. The aim of this study was to evaluate the achievement of psychosocial developmental milestones in long-term survivors of childhood DTC. Design and methods: Survivors of childhood DTC diagnosed between 1970 and 2013 were included. Reasons for exclusion were age <18 or >35 years at follow-up, a follow-up period <5 years or diagnosis with DTC as a second malignant neoplasm. Survivors gathered peer controls of similar age and sex (n = 30). A comparison group non-affected with cancer (n = 508) and other childhood cancer survivors (CCS) were also used to compare psychosocial development. To assess the achievement of psychosocial milestones (social, autonomy and psychosexual development), the course of life questionnaire (CoLQ) was used. Results: We included 39 survivors of childhood DTC (response rate 83.0%, mean age at diagnosis 15.6 years, and mean age at evaluation 26.1 years). CoLQ scores did not significantly differ between survivors of childhood DTC and the two non-affected groups. CoLQ scores of childhood DTC survivors were compared to scores of other CCS diagnosed at similar ages (n = 76). DTC survivors scored significantly higher on social development than other CCS, but scores were similar on autonomy and psychosexual developmental scales. Conclusions: Survivors of childhood DTC showed similar development on social, autonomy, and psychosexual domains compared to non-affected individuals. Social development was slightly more favorable in DTC survivors than in other CCS, but was similar on autonomy and psychosexual domains. [ABSTRACT FROM AUTHOR]

With more children surviving a brain tumor, insight into the late effects of the disease and treatment is of high importance. This study focused on profiling the neurocognitive functions that might be affected after treatment for a pediatric brain tumor, using a broad battery of computerized tests. Predictors that may influence neurocognitive functioning were also investigated. A total of 82 pediatric brain tumor survivors (PBTSs) aged 8–18 years (M = 13.85,SD = 3.15, 49% males) with parent-reported neurocognitive complaints were compared to a control group of 43 siblings (ageM = 14.27, SD = 2.44, 40% males) using linear mixed models. Neurocognitive performance was assessed using measures of attention, processing speed, memory, executive functioning, visuomotor integration (VMI), and intelligence. Tumor type, treatment, tumor location, hydrocephalus, gender, age at diagnosis, and time since diagnosis were entered into regression analyzes as predictors for neurocognitive functioning. The PBTSs showed slower processing speeds and lower intelligence (range effect sizes .71–.82,p < .001), as well as deficits in executive attention, short-term memory, executive functioning, and VMI (range effect sizes .40–.57,p < .05). Older age at assessment was associated with better neurocognitive functioning (B = .450,p < .001) and younger age at diagnosis was associated with lower intelligence (B = .328,p < .05). Medical risk factors, e.g., hydrocephalus, did not show an association with neurocognitive functioning. Late effects in PBTSs include a broad range of neurocognitive deficits. The results suggest that even PBTSs that were traditionally viewed as low risk for neurocognitive problems (e.g., surgery only, no hydrocephalus) may suffer from decreased neurocognitive functioning. [ABSTRACT FROM AUTHOR]

Background Many paediatric brain tumour survivors (PBTS) suffer from neurocognitive impairments. Promising effects of neurofeedback (NF) on neurocognitive functioning have been reported, however research into NF for PBTS has not been conducted. We investigated the effects of NF on neurocognitive functioning in PBTS using a double-blind randomised placebo-controlled trial with a parallel-group design (Pediatric Research on Improving Speed, Memory, and Attention; the PRISMA study). Methods Eligible for inclusion were PBTS with neurocognitive complaints, aged 8–18 years, >2 years post-treatment. They were recruited from five medical centres in the Netherlands. A randomisation table assigned participants to 30 sessions (two per week) of either NF or placebo feedback (PF) (ratio 1:1). Participants, parents, trainers, and researchers handling the data were blinded to group assignment. Participants were assessed pre-, post- and 6 months post-training to determine whether NF training would lead to improved functioning as compared with PF training. Primary outcome measures were attention, processing speed, memory, executive functioning, visuomotor integration, and intelligence. Linear mixed models analyses were used to test differences between NF and PF training over time. Results A total of 82 children were enrolled (mean age 13.9 years, standard deviation = 3.2, 49% males); 80 participants were randomised (NF: n = 40, PF n = 40); 71 participants completed the training (NF: n = 34, PF: n = 37); 68 participants completed training and 6 months post-training assessment (NF: n = 33, PF: n = 35). Similar improvements were found over time for the two treatment groups on the primary outcomes (all p 's > 0.15). Conclusion Results indicated no specific treatment-effects of NF on neurocognitive functioning of PBTS. [ABSTRACT FROM AUTHOR]

Purpose: With more children surviving a brain tumor, neurocognitive consequences of the tumor and its treatment become apparent, which could affect psychosocial functioning. The present study therefore aimed to assess psychosocial functioning of pediatric brain tumor survivors (PBTS) in detail.Methods: Psychosocial functioning of PBTS (8-18 years) with parent-reported neurocognitive complaints was compared to normative data on health-related quality of life (HRQOL), self-esteem, psychosocial adjustment, and executive functioning (one-sample t tests) and to a sibling control group on fatigue (independent-samples t test). Self-, parent-, and teacher-report questionnaires were included, where appropriate, providing complementary information.Results: Eighty-two PBTS (mean age 13.4 years, SD 3.2, 49 % males) and 43 healthy siblings (mean age 14.3, SD 2.4, 40 % males) were included. As compared to the normative population, PBTS themselves reported decreased physical, psychological, and generic HRQOL (d = 0.39-0.62, p < 0.008). Compared to siblings, increased fatigue-related concentration problems (d = 0.57, p < 0.01) were reported, although self-reported self-esteem and psychosocial adjustment seemed not to be affected. Parents of PBTS reported more psychosocial (d = 0.81, p < 0.000) and executive problems (d = 0.35-0.43, p < 0.016) in their child than parents of children in the normative population. Teachers indicated more psychosocial adjustment problems for female PBTS aged 8-11 years than for the female normative population (d = 0.69, p < 0.025), but they reported no more executive problems.Conclusions: PBTS with parent-reported neurocognitive complaints showed increased psychosocial problems, as reported by PBTS, parents, and teachers.Implications For Cancer Survivors: Systematic screening of psychosocial functioning is necessary so that tailored support from professionals can be offered to PBTS with neurocognitive complaints. [ABSTRACT FROM AUTHOR]

Purpose: The aim of this study was to assess internal consistency and construct validity (known-groups validity) and to provide Dutch norm data for the Dutch Pediatric Quality of Life Inventory Multidimensional Fatigue Scale for Young Adults ages 18-30 years (PedsQL fatigue_YA). Methods: A Dutch sample of 649 young adults completed online a sociodemographic questionnaire and the PedsQL fatigue_YA including three subscales: general fatigue, sleep/rest fatigue and cognitive fatigue (0-100: Higher scores indicate less fatigue symptoms). Results: The PedsQL fatigue_YA showed satisfactory to good internal consistency (Cronbach's alpha = .70-.94), except for one scale (.68). The mean scale scores were 68.23 (SD 19.15) for 'general fatigue,' 67.04 (SD 15.54) for 'sleep/rest fatigue' and 74.62 (SD 19.02) for 'cognitive fatigue.' Men reported significantly higher scores on 'general fatigue' and 'sleep/rest fatigue' than women. The PedsQL fatigue_YA distinguished between healthy young adults and young adults with chronic health conditions, with higher scores on all scales in healthy young adults than in those with a chronic health condition. Conclusion: The results demonstrate good psychometric properties of the PedsQL fatigue_YA in a sample of Dutch young adults. With the current norms available, it is possible to evaluate fatigue in the Netherlands from childhood to adulthood with the PedsQL Multidimensional Fatigue Scale. [ABSTRACT FROM AUTHOR]

Aim Kawasaki disease ( KD) is an acute paediatric vasculitis. The psychosocial consequences of this sudden illness for parents are unknown. This study aimed to evaluate health related quality of life ( HRQOL) and parental perceptions of child vulnerability ( PPCV) in parents of children with KD, and to identify variables associated with PPCV. Methods This cross-sectional study included 288 parents (83% mothers) of KD patients (mean age 8.7 years). HRQOL was assessed using the TNO- AZL Questionnaire for Adult's HRQOL ( TAAQOL) and PPCV using the Child Vulnerability Scale ( CVS). Scores of KD parents were compared with reference groups of Dutch parents. Logistic regression analyses were performed to examine associated variables. Results The HRQOL of KD parents was comparable to the HRQOL of parents of healthy children. However, KD parents showed significantly higher PPCV, regarding both the median CVS total score and the percentage in the clinical range. No differences were found in CVS outcomes between KD parents and parents of a chronically ill child. None of the studied parental, child and disease characteristics were significantly associated with PPCV. Conclusion Parents perceived their KD child more vulnerable to illness than healthy children, while in reality the majority had fully recovered from KD. [ABSTRACT FROM AUTHOR]

Background The purpose of this study is to provide Dutch norm data and to assess internal consistency and construct validity for the Pediatric Quality of Life Inventory Young Adult Generic Core Scales (PedsQL_YA) in Dutch young adults aged 18–30 years. Methods A sample of 649 young adults from the general Dutch population aged 18–30 years, stratified by age, sex, marital status and education, completed a socio-demographic questionnaire and the Dutch version of the PedsQL_YA online. Internal consistency of the PedsQL_YA scales was determined with Cronbach's alphas. Norm scores were obtained by calculating the mean PedsQL scale scores by gender, age and health status. Differences in scale scores were analyzed for gender, age and health status (construct validity) using two-sample t-tests and effect sizes were calculated. Construct validity was determined by testing differences in PedsQL scores between healthy young adults and young adults with chronic health conditions. Results All scales of the PedsQL_YA showed satisfactory to excellent internal consistency, with Cronbach's alphas between .77 and .94. Men reported higher scores (indicating better HRQOL) than women on all scales (p < .01), except for school/work functioning. No age differences were found. Young adults with chronic health conditions scored lower on all scales (p < .001) than healthy young adults, indicating good construct validity. Effect sizes varied from medium to large. Conclusions The Dutch version of the PedsQL_YA has adequate psychometric properties. With the availability of reliable norm data, the PedsQL_YA can be used as a tool in the evaluation of health related quality of life in healthy young adults and those with a chronic health condition. [ABSTRACT FROM AUTHOR]

Purpose: Perceiving favourable changes from one's illness may go hand in hand with experiencing harmful psychosocial effects. Each of these constructs should be considered when examining children's levels of psychological adjustment following stressful life events. A paediatric instrument that accounts for both positive and negative impact of stressful events has not been investigated in The Netherlands before. The aim of the study was to investigate psychometric properties of the Dutch version of the Benefit and Burden Scale for Children (BBSC), a 20-item questionnaire that intends to measure potential benefit and burden of illness in children.Methods: Dutch paediatric survivors of childhood cancer aged 8-18 (N = 77) completed the BBSC and other psychological questionnaires: Pediatric Quality of Life Inventory (health-related quality of life), State-Trait Anxiety Inventory for Children (anxiety), Children's Revised Impact of Event Scale (posttraumatic stress) and Strengths and Difficulties Questionnaire (behavioural functioning). Reliability and validity were evaluated.Results: Internal consistency (Cronbach's alpha, benefit 0.84, burden 0.72), test-retest reliability (benefit r = 0.74, burden r = 0.78) and homogeneity (mean inter-item correlation, benefit r = 0.34, burden r = 0.22) were satisfactory. Burden was associated with HRQoL (-), anxiety (+), posttraumatic stress symptoms (+) and behavioural problems. Benefit did not correlate with the psychological outcomes.Conclusions: The Dutch version of the BBSC shows promising psychometric properties. Perceived benefit and disease-related burden are distinct constructs; both should be considered when examining children's psychological adjustment to potentially traumatic experiences. The BBSC may be useful as monitoring and screening instrument. [ABSTRACT FROM AUTHOR]

Objective: To identify and appraise all published quality of life (QOL) measures developed for use with children with cancer and childhood cancer survivors.Methods: MEDLINE, CINAHL, EMBASE, PsycINFO, CancerLit, and Sociological Abstracts were searched from the inception of each database to 15 June 2009. Included articles were ones that described the development and/or psychometric evaluation of a QOL measure developed for use with children with cancer or childhood cancer survivors. Articles were appraised for adherence to internationally recommended guidelines for item generation, item reduction, and psychometric evaluation.Results: Thirteen QOL questionnaires were identified by our search. Eleven measures are applicable to measuring QOL in children with any type of cancer, and two are specific to children with brain cancer. Four measures can be used to measure QOL in children undergoing cancer treatment, six can be used with children on or off treatment, and three are specific to childhood cancer survivors. While all measures underwent some degree of formal development and validation, item generation often did not involve children with cancer or their parents, and a number of measures did not describe or utilize recommended methods for item reduction and psychometric evaluation.Conclusions: Most of the measures identified in this review were designed to measure QOL concerns of children with any type of cancer and at any time, during treatment or survivorship. Our findings can help researchers and clinicians identify scientifically sound measures. [ABSTRACT FROM AUTHOR]

Background: The purpose of this study is to provide Dutch norm data and to assess internal consistency and construct validity for the Pediatric Quality of Life Inventory Young Adult Generic Core Scales (PedsQL_YA) in Dutch young adults aged 18-30 years.Methods: A sample of 649 young adults from the general Dutch population aged 18-30 years, stratified by age, sex, marital status and education, completed a socio-demographic questionnaire and the Dutch version of the PedsQL_YA online. Internal consistency of the PedsQL_YA scales was determined with Cronbach's alphas. Norm scores were obtained by calculating the mean PedsQL scale scores by gender, age and health status. Differences in scale scores were analyzed for gender, age and health status (construct validity) using two-sample t-tests and effect sizes were calculated. Construct validity was determined by testing differences in PedsQL scores between healthy young adults and young adults with chronic health conditions.Results: All scales of the PedsQL_YA showed satisfactory to excellent internal consistency, with Cronbach's alphas between .77 and .94. Men reported higher scores (indicating better HRQOL) than women on all scales (p < .01), except for school/work functioning. No age differences were found. Young adults with chronic health conditions scored lower on all scales (p < .001) than healthy young adults, indicating good construct validity. Effect sizes varied from medium to large.Conclusions: The Dutch version of the PedsQL_YA has adequate psychometric properties. With the availability of reliable norm data, the PedsQL_YA can be used as a tool in the evaluation of health related quality of life in healthy young adults and those with a chronic health condition. [ABSTRACT FROM AUTHOR]