Fatigue in children and adolescents perinatally infected with human immunodeficiency virus: an observational study.

BACKGROUND: Fatigue is common among adults living with human immunodeficiency virus (HIV) as well as children with a chronic disease (CCD). Fatigue can have disastrous effects on health status, including health related quality of life (HRQOL). Even so, fatigue is underexplored in children and adolescents perinatally infected with HIV (PHIV+) in the Netherlands. The objective of this observational study is to explore fatigue in PHIV+ and its association with their HRQOL. METHODS: We measured HRQOL and fatigue using the Pediatric Quality of Life Inventory™ (PedsQL 4.0) and the PedsQL Multidimensional Fatigue Scale (MFS). The PedsQL MFS encompasses three subscales: general fatigue, sleep/rest fatigue and cognitive fatigue, and a total fatigue score. We compared outcomes of PHIV+ children and adolescents in the Amsterdam University Medical Centre with three groups: 1) HIV-uninfected controls (HIV-) matched for age, sex, region of birth, socioeconomic status and adoption status, 2) CCD, and 3) the general Dutch population. Within the PHIV+ group we explored associations between fatigue and HRQOL. RESULTS: We enrolled 14 PHIV+ (median age 10.2 years [IQR 9.2-11.4]) and 14 HIV-. Compared to CCD, PHIV+ significantly reported less general fatigue (mean difference 13.0, 95% CI 1.3 to 24.8). PHIV+ did not score significantly different on any of the other PedsQL MFS scales compared to HIV-, CCD or the general Dutch population. PHIV children scored relatively low on the cognitive fatigue scale in comparison to HIV-uninfected matched controls, CCD and the general population, although these differences did not reach significance. Among PHIV+, a lower score on total fatigue, general fatigue and cognitive fatigue was associated with a lower HRQOL score. CONCLUSIONS: The results of this study suggest that PHIV children and adolescents do not experience more symptoms of fatigue than their healthy peers. However, PHIV children and adolescents may be more likely to experience cognitive fatigue. Fatigue in PHIV also appears to be associated with children's HRQOL. Further research should confirm these exploratory findings.

The importance of specialized sleep investigations in children with a suprasellar tumor.

PURPOSE: Disruption of sleep has great impact on quality of life. In children with a suprasellar tumor and hypothalamic-pituitary dysfunction, the circadian rhythm may be disturbed causing sleep problems. However, also other factors may influence sleep. Awareness of these different etiologies and careful history taking with appropriate additional diagnostics will aid in restoring sleep quality. METHODS: We present the workup of 4 cases with a suprasellar tumor and disturbances of sleep initiation, sleep maintenance, and daytime sleepiness. In parallel, we developed a flowchart, to aid clinicians in the diagnostics of sleep problems in children after treatment for a (supra) sellar brain tumor. RESULTS: All four patients, known with hypopituitarism, presented with sleep complaints and increased daytime sleepiness. In all four, the cause of sleep problems showed to be different. In the first case, sleep evaluation revealed a severe obstructive sleep apnea, whereupon nocturnal ventilation was started. The second case revealed poor sleep hygiene in combination with an obsessive compulsive disorder. Sleep hygiene was addressed and psychiatric consultation was offered. Dexamphetamine treatment was started to reduce her obsessive compulsive complaints. The third case showed a delayed sleep phase syndrome, which improved by educational support. The fourth case revealed a secondary organic hypersomnia for which modafinil treatment was started. CONCLUSION: Sleep disturbances in children with hypopituitarism due to a (supra) sellar tumor can have different entities which require specific therapy. Awareness of these different entities is important to enable appropriate counseling. Referral to an expertise sleep center may be advised, if standard educational support is insufficient.

The impact of maintenance therapy on sleep-wake rhythms and cancer-related fatigue in pediatric acute lymphoblastic leukemia.

PURPOSE: To assess the impact of maintenance therapy and the additional impact of dexamethasone treatment on cancer-related fatigue and sleep-wake rhythms in pediatric acute lymphoblastic leukemia (ALL) patients and to determine the association between these outcomes. METHODS: A national cohort of pediatric ALL patients (≥ 2 years) was included (± 1 year post-diagnosis). Patients receiving dexamethasone were assessed twice (assessment with and without dexamethasone). Actigraphy assessments were used to calculate sleep-wake outcomes with nonparametric methods. Cancer-related fatigue was assessed with the PedsQL Multidimensional Fatigue Scale. Sleep-wake rhythms and cancer-related fatigue were compared between patients participating in the assessment without dexamethasone and healthy children (linear regression) and between assessments with and without dexamethasone (mixed models). Using linear regression, associations between sleep-wake outcomes and cancer-related fatigue were determined during assessments with and without dexamethasone. RESULTS: Responses were collected for 125 patients (113 assessments with and 81 without dexamethasone). The sleep-wake rhythm was less stable (p = 0.03) and less robust (p = 0.01), with lower physical activity levels (p < 0.001) and higher cancer-related fatigue levels (p < 0.001) in ALL patients compared to healthy children. Physical activity was lower (p = 0.001) and cancer-related fatigue more severe (p ≤ 0.001) during assessments with dexamethasone compared to without dexamethasone. Sleep-wake outcomes were significantly associated with cancer-related fatigue during periods without dexamethasone, but not during periods with dexamethasone. CONCLUSION: Sleep-wake rhythms are disturbed, physical activity levels lower, and cancer-related fatigue levels higher during maintenance therapy. Interventions aimed to enhance sleep-wake rhythms during maintenance therapy could improve cancer-related fatigue. Families should be supported in coping with the additional burden of dexamethasone treatment to improve well-being of ALL patients.

Psychometric properties and norm scores of the sleep self report in Dutch children.

BACKGROUND: Psychometrically robust questionnaires to assess self-reported sleep problems in children are important since sleep problems can have a major impact on child development. The Sleep Self Report (SSR) is a 26-item self-report tool measuring different sleep domains in children aged 7-12 years. This study aims to evaluate the psychometric properties of the SSR and to provide Dutch norm scores. METHODS: Children aged 7-12 years from the general population were recruited through a professional market research agency. In this population, structural validity was assessed with confirmatory and exploratory factor analyses, internal consistency was assessed with the Cronbach's alpha coefficient and norm scores were provided. Additionally, children attending outpatient sleep clinics (clinical population) were invited to participate. SSR scores of the general population and the clinical population were compared to establish discriminative validity. RESULTS: In total, 619 children (mean age: 9.94 ± 1.72 years) from the general population and 34 children (mean age: 9.21 ± 1.63 years) from sleep clinics participated. The 1-factor structure of the SSR was not confirmed with factor analysis. Exploratory analyses did also not yield an appropriate multidimensional structure. Internal consistency of the total score was adequate (Cronbach's alpha: 0.76). The total score distinguished the clinical population from the general population (39.07 ± 5.31 versus 31.61 ± 5.31; P < 0.01). CONCLUSIONS: An appropriate structure of the SSR was not found with factor analyses in this Dutch population. The adequate internal consistency indicates that the total score can be interpreted as a measure of overall sleep problems. The SSR also shows good discriminative validity. We recommend the total score to assess overall sleep problems and item scores to evaluate specific sleep issues and to follow up children's sleep longitudinally, as opposite changes in different item scores may not reflect in the total score. Further research on the development of multidimensional psychometrically sound pediatric sleep self-reports is of major importance.

Cost-effectiveness of a combined physical exercise and psychosocial training intervention for children with cancer: Results from the quality of life in motion study.

This study was performed to estimate the cost-effectiveness of a combined physical exercise and psychosocial intervention for children with cancer compared with usual care. Sixty-eight children, aged 8-18 years old, during or within the first year post-cancer treatment were randomised to the intervention (n = 30) and control group (n = 38). Health outcomes included fitness, muscle strength and quality adjusted life years; all administered at baseline, 4- and 12-month follow-up. Costs were gathered by 1 monthly cost questionnaires over 12 months, supplemented by medication data obtained from pharmacies. Results showed no significant differences in costs and effects between the intervention and control group at 12-month follow-up. On average, societal costs were €299 higher in the intervention group than in the control group, but this difference was not significant. Cost-effectiveness acceptability curves indicated that the intervention needs large societal investments to reach reasonable probabilities of cost-effectiveness for quality of life and lower body muscle strength. Based on the results of this study, the intervention is not cost-effective in comparison with usual care.