Distress in parents of children with first-onset steroid-sensitive nephrotic syndrome.

BACKGROUND: Steroid-sensitive nephrotic syndrome (SSNS) is associated with a relapsing-remitting course that can be stressful for parents. As little is known of parental distress at the first onset of SSNS, this study aims to describe parental distress and everyday problems in mothers and fathers of a child with newly diagnosed SSNS participating in a randomized controlled trial of levamisole added to corticosteroids. METHODS: To assess distress, the Distress Thermometer for Parents (DT-P) was used, which includes questions on distress (thermometer score 0-10, ≥ 4 "clinical distress") and presence of everyday problems in six domains: practical, social, emotional, physical, cognitive, and parenting. The DT-P was completed 4 weeks after the onset of SSNS. Total sum and individual items of everyday problems were compared with reference data from mothers and fathers of the Dutch general population. RESULTS: There was no difference in clinically elevated parental distress between SSNS mothers (n = 37) and fathers (n = 25) and reference parents. Compared to reference fathers, fathers of a child with SSNS scored significantly higher on emotional problems (P = 0.030), while mothers experienced more parenting problems (P = 0.002). Regression analyses showed that lower parental age and having a girl with SSNS were significantly associated with more practical problems and higher distress thermometer scores, respectively. CONCLUSIONS: Four weeks after onset, SSNS mothers and fathers experience equal distress as reference parents. However, both parents endorsed significantly more everyday problems. Therefore, monitoring parental distress, even in the first weeks of the disease, could contribute to timely interventions and prevent worsening of problems. CLINICAL TRIAL REGISTRY: Dutch Trial Register ( https://onderzoekmetmensen.nl/en/trial/27331 ). A higher resolution version of the Graphical abstract is available as Supplementary information.

Health-related quality of life of children with first onset steroid-sensitive nephrotic syndrome.

This study assessed HRQoL and emotional and behavioral difficulties (EBD) and associated variables in children with first onset SSNS. While relapsing steroid-sensitive nephrotic syndrome (SSNS) in children is associated with lower health-related quality of life (HRQoL), little is known about first onset. Four weeks after onset, children (2-16 years) and/or their parents who participated in a randomized placebo-controlled trial, completed the Pediatric Quality of Life Inventory 4.0 (PedsQL) and Strengths and Difficulties Questionnaire (SDQ) to measure HRQoL and EBD, respectively. Total and subscale scores and the proportion of children with impaired HRQoL (> 1 SD below the mean of the reference group) or SDQ clinical scores (< 10th and > 90th percentile) were compared to the Dutch general population (reference group). Regression analyses were used to identify associated variables. Compared to the reference group, children 8-18 years reported significantly lower total HRQoL, and physical and emotional functioning. A large proportion (> 45%) of these children had impaired HRQoL. There were no differences in HRQoL between children 2-7 years and the reference group, except for higher scores on social functioning (5-7 years). Similar proportions of SSNS and reference children scored within the clinical range of SDQ subscales. Age, sex, and steroid side-effects were negatively associated with HRQol and/or EBD.   Conclusion: This study showed that HRQoL and EBD are affected in children of different ages with first onset SSNS. This calls for more awareness from healthcare providers and routinely monitoring of HRQoL and EBD in daily clinical care to prevent worsening of symptoms.   Clinical trial registry: Netherlands Trial Register ( https://trialsearch.who.int/ ; NTR7013), date of registration: 02 June 2018. What is Known: •  Health-related quality of life (HRQoL) is lower and emotional and behavioral difficulties (EBD) is more affected in children with frequently-relapsing and steroid-dependent nephrotic syndrome. What is New: •  HRQoL and EBD are affected in children with first onset steroid-sensitive nephrotic syndrome compared to a reference group of the Dutch general population. • To what extent HRQoL and EBD are affected depends on the age of the patient.

Internalizing problems before and during the COVID-19 pandemic in independent samples of Dutch children and adolescents with and without pre-existing mental health problems.

The aim of the study was to assess internalizing problems before and during the pandemic with data from Dutch consortium Child and adolescent mental health and wellbeing in times of the COVID-19 pandemic, consisting of two Dutch general population samples (GS) and two clinical samples (CS) referred to youth/psychiatric care. Measures of internalizing problems were obtained from ongoing data collections pre-pandemic (NGS = 35,357; NCS = 4487) and twice during the pandemic, in Apr-May 2020 (NGS = 3938; clinical: NCS = 1008) and in Nov-Dec 2020 (NGS = 1489; NCS = 1536), in children and adolescents (8-18 years) with parent (Brief Problem Monitor) and/or child reports (Patient-Reported Outcomes Measurement Information System®). Results show that, in the general population, internalizing problems were higher during the first peak of the pandemic compared to pre-pandemic based on both child and parent reports. Yet, over the course of the pandemic, on both child and parent reports, similar or lower levels of internalizing problems were observed. Children in the clinical population reported more internalizing symptoms over the course of the pandemic while parents did not report differences in internalizing symptoms from pre-pandemic to the first peak of the pandemic nor over the course of the pandemic. Overall, the findings indicate that children and adolescents of both the general and clinical population were affected negatively by the pandemic in terms of their internalizing problems. Attention is therefore warranted to investigate long-term effects and to monitor if internalizing problems return to pre-pandemic levels or if they remain elevated post-pandemic.

Heritable connective tissue disorders in childhood: Decreased health-related quality of life and mental health.

The psychosocial consequences of growing up with Heritable Connective Tissue Disorders (HCTD) are largely unknown. We aimed to assess Health-Related Quality of Life (HRQoL) and mental health of children and adolescents with HCTD. This observational multicenter study included 126 children, aged 4-18 years, with Marfan syndrome (MFS, n = 74), Loeys-Dietz syndrome (n = 8), molecular confirmed Ehlers-Danlos syndromes (n = 15), and hypermobile Ehlers-Danlos syndrome (hEDS, n = 29). HRQoL and mental health were assessed through the parent and child-reported Child Health Questionnaires (CHQ-PF50 and CHQ-CF45, respectively) and the parent-reported Strengths and Difficulties Questionnaire. Compared with a representative general population sample, parent-reported HRQoL of the HCTD-group showed significantly decreased Physical sum scores (p < 0.001, d = 0.9) and Psychosocial sum scores (p = 0.024, d = 0.2), indicating decreased HRQoL. Similar findings were obtained for child-reported HRQoL. The parent-reported mental health of the HCTD-group showed significantly increased Total difficulties sum scores (p = 0.01, d = 0.3), indicating decreased mental health. While the male and female MFS- and hEDS-subgroups both reported decreased HRQoL, only the hEDS-subgroup reported decreased mental health. In conclusion, children and adolescents with HCTD report decreased HRQoL and mental health, with most adverse outcomes reported in children with hEDS and least in those with MFS. These findings call for systematic monitoring and tailored interventions.

Health-related quality of life in infants, toddlers and young children with sickle cell disease.

BACKGROUND: Little is known about health-related quality of life (HRQoL) in young children with sickle cell disease living in a European country. METHODS: A retrospective cross-sectional evaluation of TNO-AZL Preschool Children Quality of Life questionnaire (TAPQOL, 0-1 year) and Pediatric Quality of Life Inventory (PedsQL, 2-7 years) data was conducted. Study participants included caregivers of children with sickle cell disease aged 0-7 years attending the sickle cell centre at the Erasmus Medical Center or the Amsterdam University Medical Centers between April 2012 and October 2020. Comparisons were made with normative data on HRQoL in the general paediatric population. RESULTS: The study enrolled 136 caregivers of 136 children. In children aged 0-5 years, no significant differences emerged between children with sickle cell disease and the general population. However, in children aged 5-7 years, children with sickle cell disease scored significantly lower on all subscales except for emotional functioning. Multiple regression models showed a negative association between age and HRQoL. No association was found between HRQoL and disease severity or sociodemographic characteristics. CONCLUSIONS: This study demonstrates that HRQoL is negatively correlated with age in young children with sickle cell disease with a significantly lower HRQoL in 5- to 7-year-olds when compared to the general population. Our study underlines the importance of measuring HRQoL in young children to identify patients with impaired HRQoL early in life in order to be able to intervene accordingly. Future research should focus on deepening the knowledge of factors influencing HRQoL in children with sickle cell disease.

Longitudinal Development of Health-related Quality of Life and Fatigue in Children on Home Parenteral Nutrition.

OBJECTIVES: The aim of the study was to describe the longitudinal development of health-related quality of life (HRQOL) and fatigue in children with chronic intestinal failure (CIF) on home parenteral nutrition (PN) and compare these children to the general population. METHODS: Prospective, observational study conducted over 7 years in patients suffering from CIF receiving home PN from 2 tertiary hospitals in the Netherlands. Every 6 months, parents (if child <8 years old) or patients (if child ≥8 years old) completed 2 questionnaires: Pediatric Quality of Life Inventory 4.0 (PedsQL) Generic and Fatigue on the KLIK (kwaliteit van leven in kaart [Dutch Acronym for Quality of Life in Clinical Practice]) Patient Reported Outcome Measures portal, which were compared with the general population. Linear mixed models (LMMs) were constructed to investigate the course of HRQOL over time. RESULTS: Thirty-five patients were included (40% girls). At time of last KLIK contact, patients received HPN for a median of 5.3 years (interquartile range [IQR]: 2.9-9.7). In total, 272 questionnaires were completed. PedsQL generic total score for ages 5 to 7 and 8 to 12 years was significantly lower than the general population (P < 0.01 for both age groups) with effect sizes of 0.73 and 0.71, respectively. PedsQL fatigue total score for ages 5 to 7 years was also significantly lower (P = 0.01; effect size 0.70). LMMs for PedsQL Generic and Fatigue total score 2 to 7 and 8 to 18 years showed no significant coefficient for duration of home PN. CONCLUSIONS: Children suffering from CIF receiving home PN ages 5 to 12 years report lower HRQOL scores than the general population. HRQOL and fatigue do not change during long-term treatment with home PN in these children.

Psychosocial developmental milestones of young adult survivors of childhood cancer.

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.

Patients' and parents' perspective on the implementation of Patient Reported Outcome Measures in pediatric clinical practice using the KLIK PROM portal.

INTRODUCTION: The KLIK Patient Reported Outcome Measures (PROM) portal ( www.hetklikt.nu ) has been implemented since 2011 in clinical practice in over 20 Dutch hospitals. Patients and/or parents complete PROMs before the outpatient consultation and answers are subsequently discussed by clinicians during consultation. This study aims to provide insight into patients' and parents' perspective on the use of the KLIK PROM portal in order to optimize its implementation in pediatric clinical practice. METHODS: Patients (12-19 years) and parents (of children 0-19 years) from the Emma Children's Hospital were invited to participate. A mixed-method design was used; (1) Focus groups were held and analyzed using thematic analysis in psychology, (2) a questionnaire was sent out and analyzed using descriptive statistics. RESULTS: (1) Eight patients and 17 parents participated. Patients mentioned that KLIK has an attractive layout. However, PROMs were sometimes considered irrelevant and repetitive. Parents valued that KLIK provides insight into their child's functioning, but they were not satisfied with the extent to which PROMs were discussed by clinicians. (2) 31 patients and 130 parents completed the questionnaire. Overall, patients and parents reported a satisfaction score of 7.9/10 and 7.3/10, respectively. 81% of patients and 74% of parents indicated that KLIK is easy to use. CONCLUSION: Patients and parents are generally satisfied with KLIK, however, points of improvement were mentioned. These are currently being addressed by e.g., upgrading the KLIK website, implementing PROMIS item banks in KLIK to reduce irrelevancy and repetitiveness of PROMs, and implementation strategies to improve the discussion-rate. In this way, implementation of the KLIK PROM portal can be further optimized, with the ultimate goal to improve quality of care.

Parenting a child with Marfan syndrome: Distress and everyday problems.

Marfan syndrome (MFS) is a multisystemic, autosomal dominant connective tissue disorder that occurs de novo in 25%. In many families, parent and child(ren) are affected, which may increase distress in parents. To assess distress, 42 mothers (29% MFS) and 25 fathers (60% MFS) of 43 affected children, completed the validated screening-questionnaire Distress thermometer for parents of a chronically ill child, including questions on overall distress (score 0-10; ≥4 denoting "clinical distress") and everyday problems (score 0-36). Data were compared to 1,134 control-group-parents of healthy children. Mothers reported significantly less overall distress (2, 1-4 vs. 3, 1-6; p = .049; r = -.07) and total everyday problems (3, 0-6 vs. 4, 1-8; p = .03; r = -.08) compared to control-group-mothers. Mothers without MFS reported significantly less overall distress compared to mothers with MFS, both of a child with MFS (1, 0-4 vs. 3.5, 2-5; p = .039; r = -.17). No significant differences were found between the father-groups, nor between the group of healthy parents of an affected child living together with an affected partner compared to control-group-parents. No differences in percentages of clinical distress were reported between mothers and control-group-mothers (33 vs. 42%); fathers and control-group-fathers (28 vs. 32%); nor between the other groups. Distress was not associated with the children's MFS characteristics. Concluding, parents of a child with MFS did not show more clinical distress compared to parents of healthy children. However, clinical distress was reported in approximately one-third and may increase in case of acute medical complications. We advise monitoring distress in parents of a child with MFS to provide targeted support.

Health-Related Quality of Life and Distress of Parents of Children With Avoidant Restrictive Food Intake Disorder.

OBJECTIVES: Health-related quality of life (HRQOL) of children with avoidant restrictive food intake disorder (ARFID) is impaired. AIM: To measure HRQOL and distress of parents of children with ARFID. METHODS: Cross-sectional cohort study. Parents of children with ARFID, visiting our multidisciplinary feeding team, completed questionnaires on the online Quality of Life in Clinical Practice portal; the Questionnaire for Adult's Health Related Quality of Life to assess parental HRQOL and the Distress Thermometer for Parents. Reference groups of parents of healthy (HC) and chronically ill children (CIC) were used. RESULTS: Eighty-five mothers and 62 fathers of 89 children with ARFID (58% female, median age 1.9 years) were included (response rate 68%). No differences were found regarding HRQOL in 11 of 12 domains between parents of children with ARFID and HC. Mothers of children with ARFID reported significantly higher HRQOL regarding pain and fathers a significantly lower HRQOL on depressive emotions compared to HC. No differences were found in overall and clinical distress scores between parents of children with ARFID and HC/CIC. Mothers of children with ARFID had significantly higher distress scores regarding cognitive problems compared to HC and parenting problems in children <2 years compared to HC/CIC. Significantly higher distress scores on parenting problems in children <2 years were found in fathers of children with ARFID compared to HC/CIC. CONCLUSION: Most HRQOL and distress scores of parents of children with ARFID were comparable to reference groups. Since parents of children with ARFID perceive a lack of understanding and support from the environment, professionals should suggest peer support through patient's organizations. Furthermore, it is important to offer professional support since parents indicated that they would like to talk to a professional about their situation.

Using an implementation science approach to implement and evaluate patient-reported outcome measures (PROM) initiatives in routine care settings.

PURPOSE: Patient-reported outcome and experience measures (PROMs/PREMs) are well established in research for many health conditions, but barriers persist for implementing them in routine care. Implementation science (IS) offers a potential way forward, but its application has been limited for PROMs/PREMs. METHODS: We compare similarities and differences for widely used IS frameworks and their applicability for implementing PROMs/PREMs through case studies. Three case studies implemented PROMs: (1) pain clinics in Canada; (2) oncology clinics in Australia; and (3) pediatric/adult clinics for chronic conditions in the Netherlands. The fourth case study is planning PREMs implementation in Canadian primary care clinics. We compare case studies on barriers, enablers, implementation strategies, and evaluation. RESULTS: Case studies used IS frameworks to systematize barriers, to develop implementation strategies for clinics, and to evaluate implementation effectiveness. Across case studies, consistent PROM/PREM implementation barriers were technology, uncertainty about how or why to use PROMs/PREMs, and competing demands from established clinical workflows. Enabling factors in clinics were context specific. Implementation support strategies changed during pre-implementation, implementation, and post-implementation stages. Evaluation approaches were inconsistent across case studies, and thus, we present example evaluation metrics specific to PROMs/PREMs. CONCLUSION: Multilevel IS frameworks are necessary for PROM/PREM implementation given the complexity. In cross-study comparisons, barriers to PROM/PREM implementation were consistent across patient populations and care settings, but enablers were context specific, suggesting the need for tailored implementation strategies based on clinic resources. Theoretically guided studies are needed to clarify how, why, and in what circumstances IS principles lead to successful PROM/PREM integration and sustainability.

A retrospective assessment of the KLIK PROM portal implementation using the Consolidated Framework for Implementation Research (CFIR).

PURPOSE: The KLIK Patient-Reported Outcome Measure (PROM) portal is an evidence-based intervention implemented in clinical practice in > 25 Dutch hospitals for patients (children and adults) who regularly visit the outpatient clinic. Implementation science frameworks can be used to understand why implementation succeeded or failed, to structure barriers and enablers, and to develop implementation strategies to overcome barriers. This paper aimed to (A) retrospectively describe determinants of successful KLIK PROM implementation using the Consolidated Framework for Implementation Research (CFIR), and (B) identify current barriers and match implementation strategies. METHODS: (A) The KLIK implementation process was described retrospectively based on literature and experience, using the 39 CFIR constructs organized in five general domains: intervention characteristics, outer setting, inner setting, characteristics of individuals, and implementation process. (B) The CFIR-Expert Recommendations for Implementing Change (ERIC) Implementation Strategy Matching tool identified current barriers in the KLIK implementation and matched implementation strategies that addressed the identified barriers. RESULTS: (A) The most prominent determinants of successful KLIK PROM implementation lie in the following CFIR domains: intervention characteristics (e.g., easy to use), characteristics of individuals (e.g., motivation), and process of implementation (e.g., support). (B) 13 CFIR constructs were identified as current barriers for implementing the KLIK PROM portal. The highest overall advised ERIC strategy for the specific KLIK barriers was to identify and prepare champions. CONCLUSION: Using an implementation science framework, e.g., CFIR, is recommended for groups starting to use PROMs in clinical care as it offers a structured approach and provides insight into possible enablers and barriers.

Clinicians' perspective on the implemented KLIK PROM portal in clinical practice.

PURPOSE: Since 2011, the evidence-based KLIK Patient Reported Outcome Measure (PROM) portal has been implemented in clinical practice in > 20 Dutch hospitals. Patients and/or parents complete PROMs on Health Related Quality of Life, symptoms and psychosocial functioning before their outpatient consultation. Answers are converted into an ePROfile and discussed by clinicians during consultation to monitor well-being over time and detect problems early. This study aims to get insight into the KLIK implementation from the clinician's perspective. METHODS: As part of the KLIK implementation process, annual meetings were held with multidisciplinary teams to evaluate the use of KLIK. An online questionnaire was sent regarding (1) overall satisfaction, (2) feeling competent to discuss PROMs, (3) use of KLIK during the consultation, (4) influence of KLIK on the consultation, (5) usability of the KLIK PROM portal, (6) satisfaction with PROMs and feedback, and (7) support of the KLIK expert team. Open questions about (dis)advantages were included. Descriptive analyses were used. RESULTS: One hundred and forty-eight clinicians (response-rate 61%) from 14 hospitals in the Netherlands participated. Results show that: (1) clinicians report an overall satisfaction of median = 69/100 (visual analogue scale), (2) 85.8% feel competent discussing the ePROfile, (3) 70.3% (almost) always discuss the ePROfile, (4) 70.3% think that KLIK improves consultation, (5) 71.6% think KLIK is easy to use, (6) 80.4% are satisfied with the feedback of the overall KLIK ePROfile, (7) 71.6% experience sufficient support of the KLIK team. CONCLUSION: Participating clinicians are generally satisfied with KLIK. Improvements to the KLIK PROM portal are now realized based on the mentioned disadvantages (e.g., shorten PROM completion by use of PROMIS and integrating KLIK with Electronic Health Records).

The impact of lockdown during the COVID-19 pandemic on mental and social health of children and adolescents.

PURPOSE: During the COVID-19 pandemic in the Netherlands, governmental regulations resulted in a lockdown for adults as well as children/adolescents. Schools were closed and contact with other people was limited. In this cross-sectional, population-based study, we aimed to investigate the mental/social health of children/adolescents during COVID-19 lockdown. METHODS: Two representative samples of Dutch children/adolescents (8-18 years) before COVID-19 (2018, N = 2401) and during lockdown (April 2020, N = 844) were compared on the Patient-Reported Outcomes Measurement Information System (PROMIS) domains: global health, peer relationships, anxiety, depressive symptoms, anger, sleep-related impairment by linear mixed models and calculating relative risks (RR (95% CI)) for the proportion of severe scores. Variables associated with worse mental/social health during COVID-19 were explored through multivariable regression models. The impact of COVID-19 regulations on the daily life of children was qualitatively analyzed. RESULTS: Participants reported worse PROMIS T-scores on all domains during COVID-19 lockdown compared to before (absolute mean difference range 2.1-7.1 (95% CI 1.3-7.9). During lockdown, more children reported severe Anxiety (RR = 1.95 (1.55-2.46) and Sleep-Related Impairment (RR = 1.89 (1.29-2.78) and fewer children reported poor Global Health (RR = 0.36 (0.20-0.65)). Associated factors with worse mental/social health were single-parent family, ≥ three children in the family, negative change in work situation of parents due to COVID-19 regulations, and a relative/friend infected with COVID-19. A large majority (> 90%) reported a negative impact of the COVID-19 regulations on daily life. CONCLUSION: This study showed that governmental regulations regarding lockdown pose a serious mental/social health threat on children/adolescents that should be brought to the forefront of political decision-making and mental healthcare policy, intervention, and prevention.

Paediatric patients report lower health-related quality of life in daily clinical practice compared to new normative PedsQLTM data.

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 (ß = -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.

Matching Psychosocial Support Needs of Parents of a Child with a Chronic Illness to a Feasible Intervention.

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.

Options for the Interpretation of and Recommendations for Acting on Different PROMs in Daily Clinical Practice Using KLIK.

INTRODUCTION: This paper describes the different ways in which information is being fed back to health care providers (HCPs) using the Dutch evidence-based KLIK Patient-Reported Outcome Measures (PROMs) portal (www.hetklikt.nu). KLIK was initially developed for children with a chronic illness and their parents, and recently expanded for use in adult health care. Patients complete online PROMs at home about their health-related quality of life (HRQOL), symptoms and/or psychosocial functioning before an outpatient consultation with their HCP. PROMs are subsequently converted into an individual KLIK electronic (e)PROfile and discussed during the consultation to facilitate systematic attention for HRQOL. METHODS FOR PRO SCORE INTERPRETATION: KLIK includes a variety of different PROMs. The KLIK ePROfile initially consisted of literal representations of the individual PROM items in the European traffic light colors (red, orange, green) and only one graph. Over the years, the KLIK ePROfile evolved into a broader spectrum of feedback options; (1) literal representation of individual items, (2) summary scores, and (3) graphic representations (4 options). DEVELOPING RECOMMENDATIONS FOR ACTING ON PRO RESULTS: The primary goal of KLIK is to longitudinally monitor patient and parent self-reported or proxy-reported health outcomes, which means that the focus of KLIK is not mainly on recommending actions for PRO results. However, there are several aids that help interpretation of the KLIK ePROfile and recommendations are given for actions based on PROM scores. The main aid is the KLIK training, which includes a theoretical and a practical part. In the training, 2 tools-a decision tree and a summary of information about the KLIK ePROfile-are given to the HCPs to assist them in using KLIK. DISCUSSION: Customization of a PROM portal is needed for each different patient group and clinical setting. Because the KLIK website is flexible, every PROM (with good psychometric qualities and permission of the publisher) can be built in. However, implementing and feeding back PROMs in pediatric health care encompasses different challenges, because different versions of the same PROM are needed for children of different ages, as well as proxy and self-report versions.

Health-related quality of life in children with Robin sequence.

This patient-reported outcome (PRO) study reports on 102 children with Robin sequence (RS) and their parents. There has been differentiated between those with isolated RS and those with RS as part of a syndrome, and take various treatments into account. All RS families from an earlier described cohort were invited to participate. Parents and RS children completed online questionnaires regarding health-related quality of life (HRQoL), satisfaction with appearance, parental distress, and RS specific topics. Results were compared with the Dutch norm population if available. There was no major difference in HRQoL in RS children and the Dutch norm population, nor between children with isolated RS and those with RS as part of a syndrome. The latter is likely due to the large percentage of children with Stickler syndrome, and small number of RS children with intellectual disability. Parental distress was higher in RS children with syndromes compared to parents of isolated RS children. When comparing various treatments, the subgroup treated by mandibular distraction showed a tendency of lower HRQoL scores, less satisfaction with appearance, and more parental distress. Also in the NPA group parents showed a tendency of more parental distress. Subgroups for each treatment were very small, however, and firm conclusions cannot be drawn. In this study, HRQoL in RS children is demonstrated comparable to the norm population, despite variations in treatment, possibly with less favorable outcome for children who received mandibular distraction. Markedly larger studies are needed to allow more reliable comparison of PROs in various treatments, and to incorporate PROs in management guidelines to obtain optimal patient care. © 2016 Wiley Periodicals, Inc.

Dutch normative data and psychometric properties for the Distress Thermometer for Parents.

PURPOSE: The aim of this study was to provide Dutch normative data for the Distress Thermometer for Parents (DT-P) and to assess internal consistency and known-groups validity. METHODS: A sample of 1421 parents (60.7 % mothers), representative of the Dutch population, completed online sociodemographic questionnaire and the DT-P, which includes a thermometer (0 (no distress) to 10 (extreme distress), ≥4 clinically elevated distress) and everyday problems across six problem domains (practical, social, emotional, physical, cognitive, and parenting). Internal consistency was calculated using Cronbach's alphas. Known-groups validity was assessed by comparing parents of a child with a chronic condition (N = 287, 20.2 %) with parents of healthy children, using Mann-Whitney U tests and Chi-square tests. RESULTS: The DT-P showed acceptable internal consistency (Cronbach's alphas = .52-.89). Parents of a child with a chronic condition more often reported clinically elevated distress than parents of healthy children (53.0 versus 38.2 %, p < .001). Also, on all domains they reported more problems (p = .000-.022). Normative scores for mothers and fathers separately were provided. CONCLUSION: The DT-P distinguishes well between parents of a child with and without a chronic condition. With the current norms available, distress can be evaluated in parents of a child with a chronic condition compared to parents of healthy children in pediatric clinical practice.