Survival rates in pediatric hematopoietic stem cell transplantation (HSCT) for nonmalignant diseases have improved due to advances in conditioning regimens, donor selection, and prophylaxis and... Show moreSurvival rates in pediatric hematopoietic stem cell transplantation (HSCT) for nonmalignant diseases have improved due to advances in conditioning regimens, donor selection, and prophylaxis and treatment of infections and graft-versus-host disease. Insight into the long-term patient-reported outcomes (PROs) after pediatric HSCT for nonmalignant disease is lacking but essential for optimal shared decision making, counseling, and quality of care. The purpose of this research was to determine long-term patient-reported outcomes in allogeneic pediatric HSCT for nonmalignant diseases and to compare these results with Dutch reference data. This single-center cohort study evaluated PROs (PedsQL 4.0, PROMIS item banks), self- or proxy-reported, among patients at >= 2 years after pediatric allogeneic HSCT for nonmalignant disease. Mean scores were compared with those of the Dutch general population. Of 171 eligible patients, 119 participated, for a 70% response rate. The median patient age was 15.8 years (range, 2 to 49 years), and the median duration of follow-up was 8.7 years (range, 2 to 34 years). Indications for HSCT included inborn errors of immunity (n = 41), hemoglobinopathies (n = 37), and bone marrow failure (n = 41). Compared with reference data, significantly lower scores were found in adolescents (age 13 to 17 years) on the Total, Physical Health, and School Functioning PedsQL subscales. Significantly more Sleep Disturbance was reported in children (age 8 to 18 years). On the other hand, significantly better scores were seen on PROMIS Fatigue (age 5 to 7 years) and Pain Interference (age 8 to 18 years) and, in adults (age 19 to 30 years), on Depressive Symptoms and Sleep Disturbance. This study showed better or comparable very long-term PROs in patients after pediatric HSCT for nonmalignant diseases compared with the reference population. Children and adolescents seem to be the most affected, indicating the need for supportive care to prevent impaired quality of life and, more importantly, to amplify their long-term well-being. (c) 2022 The American Society for Transplantation and Cellular Therapy. Published by Elsevier Inc. This is an open access article under the CC BY license (http://creativecommons.org/licenses/by/4.0/) Show less
Fischer, K.; Tieskens, J.M.; Luijten, M.A.J.; Zijlmans, J.; Oers, H.A. van; Groot, R. de; ... ; Popma, A. 2022
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... Show moreThe 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 (N-GS = 35,357; N-CS = 4487) and twice during the pandemic, in Apr-May 2020 (N-GS = 3938; clinical: N-CS = 1008) and in Nov-Dec 2020 (N-GS = 1489; N-CS = 1536), in children and adolescents (8-18 years) with parent (Brief Problem Monitor) and/or child reports (Patient-Reported Outcomes Measurement Information System (R)). 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. Show less
Heesterbeek, M.R.; Luijten, M.A.J.; Gouw, S.C.; Limperg, P.F.; Fijnvandraat, K.; Coppens, M.; ... ; Haverman, L. 2022
Background: The COVID-19 lockdown increases psychological problems in children and adolescents from the general population. Here we investigate the mental and social health during the COVID-19... Show moreBackground: The COVID-19 lockdown increases psychological problems in children and adolescents from the general population. Here we investigate the mental and social health during the COVID-19 lockdown in children and adolescents with pre-existing mental or somatic problems. Methods: We included participants (8-18 years) from a psychiatric (N = 249) and pediatric (N = 90) sample, and compared them to a general population sample (N = 844). Measures were assessed during the first lockdown (April-May 2020) in the Netherlands. Main outcome measures were Patient-Reported Outcomes Measurement Information System (PROMIS (R)) domains: Global Health, Peer Relationships, Anxiety, Depressive Symptoms, Anger, and Sleep-Related Impairment, as reported by children and youth. Additionally, socio-demographic variables, COVID-19-related questions, changes in atmosphere at home from a parent and child perspective, and children's experiences of lockdown regulations were reported by parents. Results: On all measures except Global Health, the pediatric sample reported least problems. The psychiatric sample reported significantly more problems than the general population sample on all measures except for Anxiety and Peer Relationships. Having a COVID-19 affected friend/relative and a COVID-19 related change in parental work situation negatively moderated outcome, but not in the samples with pre-existing problems. All parents reported significant decreases in atmosphere at home, as did children from the general population. Conclusion: We observed significant differences in mental and social health between three child and adolescent samples during the COVID-19 pandemic lockdown and identified COVID-19-related factors influencing mental and social health. Show less
Zijlmans, J.; Teela, L.; Ewijk, H. van; Klip, H.; Mheen, M. van der; Ruisch, H; ... ; Polderman, T.J.C. 2021
Background: The COVID-19 lockdown increases psychological problems in children and adolescents from the general population. Here we investigate the mental and social health during the COVID-19... Show moreBackground: The COVID-19 lockdown increases psychological problems in children and adolescents from the general population. Here we investigate the mental and social health during the COVID-19 lockdown in children and adolescents with pre-existing mental or somatic problems. Methods: We included participants (8-18 years) from a psychiatric (N = 249) and pediatric (N = 90) sample, and compared them to a general population sample (N = 844). Measures were assessed during the first lockdown (April-May 2020) in the Netherlands. Main outcome measures were Patient-Reported Outcomes Measurement Information System (PROMIS (R)) domains: Global Health, Peer Relationships, Anxiety, Depressive Symptoms, Anger, and Sleep-Related Impairment, as reported by children and youth. Additionally, socio-demographic variables, COVID-19-related questions, changes in atmosphere at home from a parent and child perspective, and children's experiences of lockdown regulations were reported by parents. Results: On all measures except Global Health, the pediatric sample reported least problems. The psychiatric sample reported significantly more problems than the general population sample on all measures except for Anxiety and Peer Relationships. Having a COVID-19 affected friend/relative and a COVID-19 related change in parental work situation negatively moderated outcome, but not in the samples with pre-existing problems. All parents reported significant decreases in atmosphere at home, as did children from the general population. Conclusion: We observed significant differences in mental and social health between three child and adolescent samples during the COVID-19 pandemic lockdown and identified COVID-19-related factors influencing mental and social health. Show less
Luijten, M.A.J.; Terwee, C.B.; Oers, H.A. van; Joosten, M.M.H.; Berg, J.M. van den; Schonenberg-Meinema, D.; ... ; Haverman, L. 2020
Objective To assess the psychometric properties of 8 pediatric Patient-Reported Outcomes Measurement Information System (PROMIS) item banks in a clinical sample of children with juvenile idiopathic... Show moreObjective To assess the psychometric properties of 8 pediatric Patient-Reported Outcomes Measurement Information System (PROMIS) item banks in a clinical sample of children with juvenile idiopathic arthritis (JIA).Methods A total of 154 Dutch children (mean +/- SD age 14.4 +/- 3.0 years; range 8-18 years) with JIA completed 8 pediatric version 1.0 PROMIS item banks (anger, anxiety, depressive symptoms, fatigue, pain interference, peer relationships, physical function mobility, physical function upper extremity) twice and the Pediatric Quality of Life Inventory (PedsQL) and the Childhood Health Assessment Questionnaire (C-HAQ) once. Structural validity of the item banks was assessed by fitting a graded response model (GRM) and inspecting GRM fit (comparative fit index [CFI], Tucker-Lewis index [TLI], and root mean square error of approximation [RMSEA]) and item fit (S-X-2 statistic). Convergent validity (with PedsQL/C-HAQ subdomains) and discriminative validity (active/inactive disease) were assessed. Reliability of the item banks, short forms, and computerized adaptive testing (CAT) was expressed as the SE of theta (SE[theta]). Test-retest reliability was assessed using intraclass correlation coefficients (ICCs) and smallest detectable change.Results All item banks had sufficient overall GRM fit (CFI >0.95, TLI >0.95, RMSEA <0.08) and no item misfit (all S-X-2 P > 0.001). High correlations (>0.70) were found between most PROMIS T scores and hypothesized PedsQL/C-HAQ (sub)domains. Mobility, pain interference, and upper extremity item banks were able to discriminate between patients with active and inactive disease. Regarding reliability, PROMIS item banks outperformed legacy instruments. Post hoc CAT simulations outperformed short forms. Test-retest reliability was strong (ICC >0.70) for all full-length item banks and short forms, except for the peer relationships item bank.Conclusion The pediatric PROMIS item banks displayed sufficient psychometric properties for Dutch children with JIA. PROMIS item banks are ready for use in clinical research and practice for children with JIA. Show less