Search

Background: Survivors of childhood brain tumors are prone to sleep and neurocognitive problems. Effective interventions to improve neurocognitive functioning are largely lacking. In general, sleep problems are negatively related to neurocognitive functioning, but this relationship is unclear in survivors of childhood brain tumors. Therefore, the occurrence of sleep problems, potential risk factors, and the relation between sleep and executive functioning were evaluated., Procedure: Baseline data of a randomized controlled trial on the effectiveness of neurofeedback were used. Childhood brain tumor survivors 8-18 years of age with parent-reported neurocognitive complaints ≥2 years after treatment were eligible. Parents completed the Sleep Disturbance Scale for Children. Executive functioning was assessed by parents and teachers (Behavior Rating Inventory of Executive Functioning). Multiple linear regression analyses were used to examine sociodemographic and medical characteristics and emotional difficulties and hyperactivity/inattention (Strength and Difficulties Questionnaire) as potential risk factors for sleep problems, and to assess the association between sleep and executive functioning., Results: Forty-eight percent of survivors (n = 82, 7.0 ± 3.6 years post diagnosis, age 13.8 ± 3.2 years) had sleep problems and scored significantly worse than the norm on the subscales Initiating and Maintaining Sleep, Excessive Somnolence, and the total scale (effect sizes 0.58-0.92). Emotional problems and/or hyperactivity/inattention were independent potential risk factors. Sleep problems were associated with worse parent-reported executive functioning., Conclusions: Sleep problems occur among half of childhood brain tumor survivors with neurocognitive problems, and are associated with worse executive functioning. Future studies should focus on the development of sleep interventions for this population, to improve sleep as well as executive functioning., (© 2019 Wiley Periodicals, Inc.)

Background: The achievement of age-specific developmental milestones in youth is of great importance to the adjustment in adult life. Young adults who were born preterm, might go through a different developmental trajectory and transition into adulthood than their peers. This study aimed to compare the psychosocial developmental trajectory of young adults who were born preterm with peers from the general population. Young adults from the POPS (Project On Preterm and Small for gestational age infants) cohort study, born in 1983 in the Netherlands, completed online the Course of Life Questionnaire (CoLQ - achievement of psychosocial developmental milestones) at 28 years of age. Analysis of variance by group, age and gender was performed to test differences on the CoLQ scale scores between the POPS-group and 211 peers (25-30 years) from the general population (Ref-group). Differences on item level, representing the achievement of individual milestones, were analyzed with logistic regression analyses by group, age and gender., Results: The POPS-group (n = 300, 32,3% biased response) scored significantly lower than the Ref-group on the scales Psychosexual Development (effect size - 0.26, p < 0.01), Antisocial Behavior (ES - 0.44, p < 0.001) and Substance Use & Gambling (ES - 0.35, p < .001). A further exploration on item-level revealed, among others, that the POPS-group had their first boyfriend/girlfriend at later age, were more often single, misbehaved less at school and smoked, drank and gambled less than the Ref-group. On the scales Autonomy Development and Social Development no differences were found between the POPS-group and the Ref-group., Conclusions: A relatively less vulnerable respondent group of young adults born preterm showed some psychosocial developmental trajectory delays and might benefit from support at teenage age. Because of the non-response bias, we hypothesize that the total group of young adults born preterm will show more severe psychosocial developmental problems.

Background: Head and neck rhabdomyosarcoma (HNRMS) survivors are at risk to develop adverse events (AEs). The impact of these AEs on psychosocial well-being is unclear. We aimed to assess psychosocial well-being of HNRMS survivors and examine whether psychosocial outcomes were associated with burden of therapy., Procedure: Sixty-five HNRMS survivors (median follow-up: 11.5 years), treated in the Netherlands and the United Kingdom between 1990 and 2010 and alive ≥2 years after treatment visited the outpatient multidisciplinary follow-up clinic once, in which AEs were scored based on a predefined list according to the Common Terminology Criteria for Adverse Events. Survivors were asked to complete questionnaires on health-related quality of life (HRQoL; PedsQL and YQOL-FD), self-perception (KIDSCREEN), and satisfaction with appearances (SWA). HRQoL and self-perception scores were compared with reference values, and the correlation between physician-assessed AEs and psychosocial well-being was assessed., Results: HNRMS survivors showed significantly lower scores on PedsQL school/work domain (P ≤ 0.01, P = 0.02, respectively), YQOL-FD domains negative self-image and positive consequences (P ≤ 0.01, P = 0.04, respectively) compared with norm data; scores on negative consequences domain were significantly higher (P = 0.03). Over 50% of survivors negatively rated their appearances on three or more items. Burden of AEs was not associated with generic HRQoL and self-perception scores, but was associated with disease-specific QoL (YQOL-FD)., Conclusion: In general, HRQoL in HNRMS survivors was comparable to reference groups; however, survivors did report disease-specific consequences. We therefore recommend including specific questionnaires related to difficulties with facial appearance in a systematic monitoring program to determine the necessity for tailored care., (© 2018 The Authors. Pediatric Blood & Cancer Published by Wiley Periodicals, Inc.)

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.

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., (Copyright © 2016 Elsevier Ltd. All rights reserved.)

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., (Copyright © 2016 Elsevier Ltd. All rights reserved.)

Objective: To evaluate socioprofessional outcomes after 30 years of renal replacement therapy (RRT) and explore predictors of these outcomes., Study Design: The cohort comprised all Dutch patients, born before 1979, who started RRT at age <15 years in 1972-1992. Outcomes including family life, educational attainment, and professional life were obtained in 2000 and 2010 in 80 out of 152 survivors. Participants also completed the Course of Life Questionnaire in 2000, which retrospectively assessed the achievement of developmental milestones while growing up. Socioprofessional outcomes in 2010 were compared with the age-matched general population and with outcomes obtained in 2000. Logistic regression analysis was performed to identify determinants of socioprofessional outcomes., Results: Mean age and time on RRT in 2010 were 40.6 years (range 32.1-52.4) and 28.9 years (range 18.1-39.7), respectively. Patients were less likely to be employed (62.5% vs 81.0%) and have children (28.8% vs 64.8%) compared with the age-matched general population. Comorbidities, dialysis, short stature, and fewer milestones on autonomy were associated with adverse outcomes. Compared with 2000, in 2010 more patients lived with a partner (68.8% vs 43.0%), and more patients had completed a high level educational degree (22.5% vs 13.9%). However, more patients were unable to work on medical grounds in 2010 (36.3% vs 16.3%)., Conclusions: Survivors of pediatric end-stage renal disease may gain social autonomy and optimal educational attainment at an older age compared with their general population counterparts. Awareness among health care professionals of the potential of these children and tailored psychosocial interventions might improve socioprofessional development., (Copyright © 2016 Elsevier Inc. All rights reserved.)

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.

Background: Radiotherapy is a well-known cause of adverse events (AEs). To reduce AEs, an innovative local treatment was developed in Amsterdam: Ablative surgery, MOuld brachytherapy and surgical REconstruction (AMORE)., Aims: (1) to determine the prevalence of AEs in HNRMS survivors and (2) to compare AEs between survivors treated with the international standard: external beam radiotherapy (EBRT-based: London) and survivors treated with AMORE if feasible, otherwise EBRT (AMORE-based: Amsterdam)., Methods: All HNRMS survivors, treated in London or Amsterdam between January 1990 and December 2010 (n = 153), and alive ⩾ 2 years post-treatment were eligible (n = 113). A predefined list of AEs was assessed in a multidisciplinary clinic and graded according to the Common Terminology Criteria for Adverse Events., Results: Eighty HNRMS survivors attended the clinic (median follow-up 10.5 years); 63% experienced ⩾ 1 severe or disabling event, and 76% had ⩾ 5 AEs (any grade). Survivors with EBRT-based treatment were, after adjustment for site, age at diagnosis, and follow-up duration, at increased risk to develop any grade 3/4 event or ⩾ 5 AEs (any grade) compared with survivors with AMORE-based treatments (p = 0.032 and 0.01, respectively). Five year overall survival (source population) after EBRT-based treatment was 75.0%, after AMORE-based treatment 76.9%, p = 0.56., Conclusion: This study may serve as a baseline inventory and can be used in future studies for prospective assessments of AEs following the introduction of novel local treatment modalities. AMORE-based local treatment resulted in similar overall survival and a reduction of AEs secondary to local treatment., (Copyright © 2015 Elsevier Ltd. All rights reserved.)