Different subtypes of chronic fatigue in childhood cancer survivors: A DCCSS LATER study.

INTRODUCTION: The aim of the current study was to investigate whether subtypes of chronic fatigue (CF) can be identified in childhood cancer survivors (CCS), and if so, to determine the characteristics of participants with a specific subtype. METHODS: Participants were included from the nationwide DCCSS LATER cohort. The Checklist Individual Strength (CIS) was completed to assess fatigue. Participants with CF (scored ≥35 on the fatigue severity subscale and indicated to suffer from fatigue for ≥6 months) were divided into subgroups using two-step cluster analysis based on the CIS concentration, motivation, and physical activity subscales. Differences between groups on demographics, psychosocial, lifestyle, and treatment-related variables were determined using ANOVA and chi-square analyses (univariable) and multinomial regression analysis (multivariable). RESULTS: A total of 1910 participants participated in the current study (n = 450 with CF; n = 1460 without CF). Three CF subgroups were identified: Subgroup 1 (n = 133, 29% of participants) had CF with problems in physical activity; Subgroup 2 (n = 111, 25% of participants) had CF with difficulty concentrating; and Subgroup 3 (n = 206, 46% of participants) had multi-dimensional CF. Compared to Subgroup 1, Subgroup 2 more often reported sleep problems, limitations in social functioning, and less often have more than two comorbidities. Subgroup 3 more often reported depression, sleep problems, a lower self-esteem, and limitations in social functioning and a lower educational level compared to Subgroup 1. CONCLUSION: Different subgroups of CCS with CF can be identified based on fatigue dimensions physical activity, motivation and concentration. Results suggest that different intervention strategies, tailored for each subgroup, might be beneficial.

Monitoring health related quality of life in survivorship care of young adult survivors of childhood cancer using web-based patient-reported outcome measures: survivors' and health care practitioners' perspectives on the KLIK method.

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.

Psychosocial functioning of adult siblings of Dutch very long-term survivors of childhood cancer: DCCSS-LATER 2 psycho-oncology study.

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 (ß = 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.

Psychosexual development, sexual functioning and sexual satisfaction in long-term childhood cancer survivors: DCCSS-LATER 2 sexuality substudy.

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.

Positive and negative survivor-specific psychosocial consequences of childhood cancer: the DCCSS-LATER 2 psycho-oncology study.

PURPOSE: Numerous studies investigated generic psychosocial outcomes in survivors of childhood cancer (CCS). The present study aimed to describe survivor-specific psychosocial consequences in CCS, and to identify socio-demographic and medical associated factors. 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 the Benefit & Burden Scale (BBSC) and the Impact of Cancer-Childhood Cancer (IOC-CS). Items were scored on a 5-point Likert scale (range 1-5). We examined outcomes with descriptive statistics, and socio-demographic and medical associated factors with regression analyses, corrected for multiple testing (p < 0.004). RESULTS: CCS, N = 1713, age mean (M) 36 years, 49% female, ≥ 15 years since diagnosis, participated. On average, CCS reported 'somewhat' Benefit (M = 2.9), and 'not at all' to 'a little' Burden (M = 1.5) of childhood cancer. Average scores on IOC-CS' positive impact scales ranged from 2.5 (Personal Growth) to 4.1 (Socializing), and on the negative impact scales from 1.4 (Financial Problems) to 2.4 (Thinking/Memory). Apart from cognitive problems, CCS reported challenges as worries about relationship status, fertility, and how cancer had affected siblings. Female sex was associated with more Personal Growth, and more negative impact. CCS more highly educated, partnered, and employed had higher positive and lower negative impact. CCS older at diagnosis reported more positive impact. CNS tumor survivors and those who had head/cranium radiotherapy had higher negative impact. CNS tumor survivors reported less positive impact. CONCLUSION AND IMPLICATIONS: The majority of CCS reported positive impact of cancer while most CCS reported little negative impact. While this may indicate resiliency in most CCS, health care providers should be aware that they can also experience survivor-specific challenges that warrant monitoring/screening, information provision and psychosocial support.

Frailty and sarcopenia within the earliest national Dutch childhood cancer survivor cohort (DCCSS-LATER): a cross-sectional study.

BACKGROUND: Childhood cancer survivors appear to be at increased risk of frailty and sarcopenia, but evidence on the occurrence of and high-risk groups for these aging phenotypes is scarce, especially in European survivors. The aim of this cross-sectional study was to assess the prevalence of and explore risk factors for pre-frailty, frailty, and sarcopenia in a national cohort of Dutch childhood cancer survivors diagnosed between 1963 and 2001. METHODS: Eligible individuals (alive at the time of study, living in the Netherlands, age 18-45 years, and had not previously declined to participate in a late-effects study) from the Dutch Childhood Cancer Survivor Study (DCCSS-LATER) cohort were invited to take part in this cross-sectional study. We defined pre-frailty and frailty according to modified Fried criteria, and sarcopenia according to the European Working Group on Sarcopenia in Older People 2 definition. Associations between these conditions and demographic and treatment-related as well as endocrine and lifestyle-related factors were estimated with two separate multivariable logistic regression models in survivors with any frailty measurement or complete sarcopenia measurements. FINDINGS: 3996 adult survivors of the DCCSS-LATER cohort were invited to participate in this cross-sectional study. 1993 non-participants were excluded due to lack of response or a decline to participate and 2003 (50·1%) childhood cancer survivors aged 18-45 years were included. 1114 (55·6%) participants had complete frailty measurements and 1472 (73·5%) participants had complete sarcopenia measurements. Mean age at participation was 33·1 years (SD  7·2). 1037 (51·8%) participants were male, 966 (48·2%) were female, and none were transgender. In survivors with complete frailty measurements or complete sarcopenia measurements, the percentage of pre-frailty was 20·3% (95% CI 18·0-22·7), frailty was 7·4% (6·0-9·0), and sarcopenia was 4·4% (3·5-5·6). In the models for pre-frailty, underweight (odds ratio [OR] 3·38 [95% CI 1·92-5·95]) and obesity (OR 1·67 [1·14-2·43]), cranial irradiation (OR 2·07 [1·47-2·93]), total body irradiation (OR 3·17 [1·77-5·70]), cisplatin dose of at least 600 mg/m2 (OR 3·75 [1·82-7·74]), growth hormone deficiency (OR 2·25 [1·23-4·09]), hyperthyroidism (OR 3·72 [1·63-8·47]), bone mineral density (Z score ≤-1 and >-2, OR 1·80 [95% CI 1·31-2·47]; Z score ≤-2, OR 3·37 [2·20-5·15]), and folic acid deficiency (OR 1·87 [1·31-2·68]) were considered significant. For frailty, associated factors included age at diagnosis between 10-18 years (OR 1·94 [95% CI 1·19-3·16]), underweight (OR 3·09 [1·42-6·69]), cranial irradiation (OR 2·65 [1·59-4·34]), total body irradiation (OR 3·28 [1·48-7·28]), cisplatin dose of at least 600 mg/m2 (OR 3·93 [1·45-10·67]), higher carboplatin doses (per g/m2; OR 1·15 [1·02-1·31]), cyclophosphamide equivalent dose of at least 20 g/m2 (OR 3·90 [1·65-9·24]), hyperthyroidism (OR 2·87 [1·06-7·76]), bone mineral density Z score ≤-2 (OR 2·85 [1·54-5·29]), and folic acid deficiency (OR 2·04 [1·20-3·46]). Male sex (OR 4·56 [95%CI 2·26-9·17]), lower BMI (continuous, OR 0·52 [0·45-0·60]), cranial irradiation (OR 3·87 [1·80-8·31]), total body irradiation (OR 4·52 [1·67-12·20]), hypogonadism (OR 3·96 [1·40-11·18]), growth hormone deficiency (OR 4·66 [1·44-15·15]), and vitamin B12 deficiency (OR 6·26 [2·17-1·81]) were significantly associated with sarcopenia. INTERPRETATION: Our findings show that frailty and sarcopenia occur already at a mean age of 33 years in childhood cancer survivors. Early recognition and interventions for endocrine disorders and dietary deficiencies could be important in minimising the risk of pre-frailty, frailty, and sarcopenia in this population. FUNDING: Children Cancer-free Foundation, KiKaRoW, Dutch Cancer Society, ODAS Foundation.

Psychosocial outcomes in long-term Dutch adult survivors of childhood cancer: The DCCSS-LATER 2 psycho-oncology study.

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.

Clinical evaluation of late outcomes in Dutch childhood cancer survivors: Methodology of the DCCSS LATER 2 study.

BACKGROUND: Childhood cancer survivors face late health problems; despite advances in research, details on risk remain unclear. We describe the methodological aspects of the Dutch Childhood Cancer Survivor Study (DCCSS) cross-sectional clinical study (LATER 2 study). PROCEDURE: From the multi-center DCCSS LATER cohort of 6165 five-year survivors diagnosed during 1963-2001, we invited 4735 eligible survivors in 2016, as well as siblings and parents of survivors. Gaps in evidence identified during development of surveillance guidelines were translated into clinical research questions for 16 outcome-specific subprojects. The regular care visit to the LATER outpatient clinic forms the backbone of outcome assessment complemented with research-defined measurements (physical examination, clinical tests, questionnaires). Furthermore, blood/saliva samples were taken for deoxyribonucleic acid (DNA) extraction. RESULTS: In total, 2519 (53.2%) survivors participated in the LATER 2 study. When comparing participants with nonparticipants, we observed that males, CNS survivors, and those treated with surgery only were less likely to participate. Of the participating survivors, 49.3% were female. Median time since childhood cancer diagnosis was 26.9 years (range 14.8-54.7 years) and median attained age was 34.4 years (range 15.4-66.6 years). CONCLUSIONS: The high-quality data generated in the LATER 2 study will provide valuable insights into risks of and risk factors for clinical and physical and psychosocial health outcomes and factors for early recognition of those health outcomes in long-term childhood cancer survivors. This will contribute to fill in important gaps in knowledge and improve the quality of life and care for childhood cancer survivors.

Patient-reported outcomes in childhood head and neck rhabdomyosarcoma survivors and their relation to physician-graded adverse events-A multicenter study using the FACE-Q Craniofacial module.

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.

Online cognitive-behavioral group intervention for young adult survivors of childhood cancer: a pilot study.

BACKGROUND: Young adult childhood cancer survivors (YACCS) are a vulnerable group in need of psychosocial support, but tailored interventions are lacking. AIM: 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. METHODS: 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. RESULTS: 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. CONCLUSIONS: 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.

Psychosocial functioning of parents of Dutch long-term survivors of childhood cancer.

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 (ß = 0.37-0.46). Acceptance illness cognitions were associated to better (ß = 0.12-0.25), and helplessness to worse outcomes (ß = 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.

The Impact of Cancer-Related Fatigue on HRQOL in Survivors of Childhood Cancer: A DCCSS LATER Study.

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.

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.

Psychosocial function of Dutch children with cancer and their caregivers during different phases of the COVID-19 pandemic.

We compared psychosocial functioning of children with cancer and their caregivers in several phases of the coronavirus disease 2019 (COVID-19) pandemic to before COVID-19. One or more questionnaires on health-related quality of life (HRQoL) or fatigue of children or distress of their caregivers was available from 1644 families. In children with cancer, HRQoL was stable throughout the COVID-19 pandemic. Fatigue was slightly lower and sleep somewhat better during the pandemic than before. Caregiver distress was lower in the first pandemic phase, but increased to pre-COVID-19 levels in later phases, indicating that the length and consequences of the pandemic may be weighing on them.

Increased health-related quality of life impairments of male and female survivors of childhood cancer: DCCSS LATER 2 psycho-oncology study.

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.

Online psychosocial group intervention for adolescents with a chronic illness: A randomized controlled trial.

OBJECTIVE: The present study aimed to evaluate the efficacy of Op Koers Online, a protocolled online psychosocial group intervention for adolescents with a chronic illness (CI). METHODS: Adolescents (12-18 years) with different types of CI (N = 59; Mean age = 15.1 years, SD = 1.7; 54% female) participated in a parallel multicenter randomized controlled trial comparing Op Koers Online (N = 35) with a waitlist control group (N = 24). Assessments (online questionnaires) took place at baseline (T0), 6-months (T1) and 12-months follow-up (T2). Primary outcomes were internalizing and externalizing behavioral problems and disease-related coping skills. Health-Related Quality of Life was secondary. Efficacy was tested with linear mixed models. RESULTS: Compared to the control group, the intervention had a significant positive effect (p < .05) on disease-related coping skills T1 vs T0 (use of relaxation, ß = 0.68; social competence, ß = 0.57) and T2 vs T0 (information seeking, ß = 0.61), and on HRQoL (social-, school-, psychosocial functioning and total HRQoL) T1 vs T0 (ß = 0.52 to ß = 0.60). No intervention effects on internalizing and externalizing behavioral problems were found. CONCLUSION: The results of this randomized controlled trial indicate a positive effect of Op Koers Online. The intervention had beneficial effects on disease-related coping skills and HRQoL. PRACTICE IMPLICATIONS: The next step is to implement Op Koers Online for adolescents in clinical practice.

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.

Determinants of impairments in functioning, fatigue, and participation ability in pediatric brain tumor survivors.

BACKGROUND: Pediatric brain tumor survivors (PBTS) experience disease- and treatment-related sequelae. We aimed to investigate the occurrence of participation limitations, impairments in functioning, fatigue, and the association between patient, tumor- and treatment-related factors and these outcomes. METHODS: Children (4-18 years) after treatment for a brain tumor between 2005 and 2014 at the Erasmus Medical Center, Rotterdam, the Netherlands, were eligible. The parent-reported Child and Family Follow-up Survey developed to measure participation and impairments in functioning in youth with acquired brain injury, was used. Fatigue was assessed using the Pediatric Quality of Life Inventory Multidimensional Fatigue Scale. Associations with patient, tumor- and treatment-related factors were explored using univariable analyses. RESULTS: Ninety-one PBTS (median age: 11.3 years [range: 9.5-14.1], time since treatment: 3.9 years [range: 4-6.2]) were included (response rate: 55%). Participation limitations were reported in 53% and were associated with impairments in functioning (15-67%) (P ≤ .01) and fatigue (P ≤ .03).Parent- and child-reported fatigue was increased compared to normative values (P ≤ .02). History of hydrocephalus was associated with increased fatigue (P ≤ .04). Younger age at diagnosis and longer time since diagnosis were associated with impairments in functioning and cognitive fatigue (P < .05).Participation limitations, impairments in functioning and fatigue were similar in PBTS who were <3 or ≥3 years since completion of treatment. CONCLUSION: More than half of PBTS reported limited participation ability, which is associated with impairments in functioning and fatigue. The complication hydrocephalus seems to lead to more fatigue. Participation limitations, impairments in functioning and fatigue appear not to diminish in the longer term.

No increase in psychosocial stress of Dutch children with cancer and their caregivers during the first months of the COVID-19 pandemic.

We studied the psychosocial impact of the start of the COVID-19 pandemic on Dutch children with cancer in outpatient care and their caregivers (n = 799) using regular monitoring and screening outcomes. No differences were observed between the pre-COVID-19 and early-COVID-19 periods in health-related quality of life and fatigue of children. Fewer caregivers were distressed during the COVID-19 period than pre-COVID-19. In conclusion, the additional stress of COVID-19 did not deteriorate psychosocial functioning of children with cancer and their caregivers. Results may be explained by alleviating daily life changes, experience in coping with medical traumatic stress, and appropriate care and support.

Online psychosocial group intervention for parents: Positive effects on anxiety and depression.

OBJECTIVE: To evaluate the efficacy of an online psychosocial group intervention for parents of children with a chronic illness, in terms of anxiety and depression, and disease-related coping skills. METHODS: Parents (N = 73) participated in a parallel multicenter randomized controlled trial comparing an intervention group to a waitlist control group. In the group intervention Op Koers Online (English: On Track Online) parents learned how to use adaptive coping strategies taught with cognitive behavioral therapy and acceptance and commitment therapy techniques. Assessments (online questionnaires) took place at baseline (T0), 6-months (T1), and 12-months (T2) follow-up. Mixed-model analyses were performed to test the difference in change in outcomes between intervention (N = 34) and waitlist control group (N = 33). RESULTS: When compared with the waitlist control group, the intervention had a significant positive effect (p < .05) on changes in anxiety, depression, and total score T1 versus T0 (ß = -.47 to -.51) and T2 versus T0 (ß = -.39 to -.46), the coping skills open communication, relaxation, social support, acceptance, predictive control (ß = .42-.88) and helplessness (ß = -.47) T1 versus T0 and relaxation and positive thinking T2 versus T0 (ß = .42-.53). CONCLUSIONS: Parental anxiety and depression decreased, and use of adaptive coping skills improved after the intervention. The online character, the focus on parents themselves instead of on their child and the possibility for parents of children with rare illnesses to participate, are innovative and unique aspects of Op Koers Online for parents. The next step is to implement the intervention in clinical practice.