Topical Review: A Biopsychosocial Framework for Pediatric Narcolepsy and Idiopathic Hypersomnia.

OBJECTIVE: Provide an overview of current research findings in pediatric central disorders of hypersomnolence (CDH) and propose a biopsychosocial model for clinical management, with a focus on interdisciplinary care and future directions for research and clinical practice. METHODS: Literature review drawing from pediatric and adult narcolepsy, as well as pediatric sleep and chronic pain research to develop an integrative biopsychosocial model for pediatric CDH. RESULTS: Youth with CDH are vulnerable to impairments in academics, emotional, and behavioral functioning, activity engagement and quality of life (QOL). There is a complex interrelationship between neurobiological features of disease, treatment-related factors, and psychological, sleep-related, and contextual factors across development. Research is limited largely to adults and pediatric narcolepsy type 1 and the mechanisms and evolution of morbidity remain poorly understood. CONCLUSIONS: In addition to first-line treatment (pharmacotherapy), routine screening of bio-behavioral and psychosocial functioning and QOL is needed to identify risk for compromised functioning warranting adjunctive interventions with behavioral health specialists.

Utility of the Sleep Disorders Inventory for Students in Clinically Referred Youth With Insomnia: Risk Identification and Relationship With Polysomnographic Measures.

Objective/Background: This study evaluated the overall performance of the Sleep Disorders Inventory for Students (SDIS) in identifying sleep disorders risk and the sensitivity and specificity of specific SDIS subscales in a clinically referred sample of youth with insomnia. Participants: Youth (N = 1,329, M = 2.5-18.99 years) with insomnia, of whom 392 underwent clinically indicated diagnostic PSG within ± 6 months of SDIS screening. Methods: Risk identification for sleep disturbance warranting sleep specialist evaluation was determined based on elevation on any SDIS scale for the entire sample. Patients with obstructive sleep apnea (OSA) and periodic limb movement disorder (PMLD) based on PSG were evaluated in relation to SDIS-OSA and SDIS-PLMD subscales (T-scores ≥ 60), with sensitivity, specificity, and receiver operator characteristic curves computed. Results: Clinical elevation correctly identified 74.0% to 83.6% referred to a sleep specialist. Nearly 30% of the subsample undergoing PSG met criteria for OSA and 20.2% had PLMs. The SDIS-OSA subscale demonstrated low to fair sensitivity and specificity. The SDIS-PLMD subscale displayed low sensitivity and low (children) to fair (adolescents) specificity, with the area under the curve suggesting good classification accuracy for adolescents. Conclusions: The SDIS performed well in identifying overall sleep disturbance risk in a large pediatric insomnia sample, suggesting careful examination of all subscales and total score to increase confidence in referral to a sleep specialist. Sensitivity and specificity indices highlight challenges in identifying primary sleep disorder risk and raise potential research questions regarding measurement of parent perceptions in children with insomnia. Additional research in nationally representative samples is warranted.

Sleep and mood during hospitalization for high-dose chemotherapy and hematopoietic rescue in pediatric medulloblastoma.

OBJECTIVE: Disrupted sleep is common in pediatric cancer, which is associated with psychological distress and may impact neural recovery. Information regarding sleep during pediatric brain tumor treatment is limited. This study aimed to describe objective sleep-wake patterns and examine the sleep-mood relation in youth hospitalized for intensive chemotherapy and stem cell rescue. METHODS: Participants included 37 patients (M age = 9.6 ± 4.2 years) enrolled on a medulloblastoma protocol (SJMB03) and their parents. Respondents completed a mood disturbance measure on 3 days, and patients wore an actigraph for 5 days as an objective estimate of sleep-wake patterns. General linear mixed models examined the relation between nocturnal sleep and next-day mood, as well as mood and that night's sleep. RESULTS: Sleep duration was deficient, sleep efficiency was poor, and daytime napping was common, with large between-subjects variability. There were minimal mood concerns across all days. The sleep and next-day mood relationship was nonsignificant (P > .05). Greater parent-reported child mood disturbance on day 2 was associated with decreased same-night sleep (P < .001) and greater patient-reported mood disturbance was associated with greater same-night sleep latency (P = .036). CONCLUSIONS: Patients with medulloblastoma are vulnerable to disturbed sleep during hospitalization, and mood may be an important correlate to consider. Sleep and mood are modifiable factors that may be targeted to maximize daytime functioning.

Sleep and Pain in Pediatric Illness: A Conceptual Review.

BACKGROUND: Sleep disruption is a common comorbidity of pediatric pain. Consequences of pain and disrupted sleep, evidence for the pain-sleep relation, and how aspects of illness, treatment, and pharmacological pain management may contribute to or exacerbate these issues are presented. AIMS: This conceptual review explored the relation between pain and sleep in children diagnosed with chronic medical or developmental conditions. The goal of this review is to expand upon the literature by examining common themes in sleep disturbances associated with painful conditions across multiple pediatric illnesses. Populations reviewed include youth with intellectual and developmental disabilities (IDD), migraines, cystic fibrosis (CF), sickle cell disease (SCD), cancer, juvenile idiopathic arthritis (JIA), juvenile fibromyalgia (JFM), and functional gastrointestinal disorders (FGIDs). RESULTS: Consistent evidence demonstrates that children with medical or developmental conditions are more vulnerable to experiencing pain and subjective sleep complaints than healthy peers. Objective sleep concerns are common but often under-studied. Evidence of the pain-sleep relationship exists, particularly in pediatric SCD, IDD, and JIA, with a dearth of studies directly examining this relation in pediatric cancer, JFM, CF, and FGIDs. Findings suggest that assessing and treating pain and sleep disruption is important when optimizing functional outcomes. CONCLUSION: It is essential that research further examine objective sleep, elucidate the pain-sleep relationship, consider physiological and psychosocial mechanisms of this relationship, and investigate nonpharmacological interventions aimed at improving pain and sleep in vulnerable pediatric populations.

Sleepiness, Fatigue, Behavioral Functioning, and Quality of Life in Survivors of Childhood Hematopoietic Stem Cell Transplant.

OBJECTIVES: To examine subjective fatigue and sleepiness as predictors of functional outcomes in long-term pediatric hematopoietic stem cell transplant (HSCT) survivors. METHODS: Participants included 76 survivors assessed 5-14 years post-HSCT. Self-report and parent-proxy (i.e., N = 38) measures of fatigue, excessive daytime sleepiness (EDS), emotional and behavioral functioning, executive functioning, and quality of life (QOL) were completed. Health-related correlates were obtained from medical records. RESULTS: Survivors exhibited significant fatigue for self (M = 69.21 ± 20.14) and parent-proxy (M = 72.15 ± 20.79) report. EDS was endorsed for 20-33% of survivors, depending on the respondent. EDS was not significant for parent-proxy outcomes, but was associated with poorer self-reported QOL and internalizing problems (p < .0016). Fatigue was associated with poorer functioning across all domains (p's < .0016). CONCLUSIONS: A substantial number of pediatric HSCT survivors exhibit sleepiness and fatigue. Fatigue is associated with statistically and clinically greater functional difficulties, highlighting the importance of examining sleep and fatigue and considering interventions to improve alertness.

Health-related quality of life in youth with Crohn disease: role of disease activity and parenting stress.

OBJECTIVES: Health-related quality of life (HRQOL) is an important, but understudied construct in pediatric inflammatory bowel disease. Family level predictors of HRQOL have been understudied as are the mechanisms through which disease activity affects HRQOL. The present study examines the relation between a family level factor (parenting stress) and HRQOL in youth with Crohn disease. Parenting stress is examined as a mechanism through which disease activity affects HRQOL. METHODS: A total of 99 adolescents with Crohn disease and their parents were recruited across 3 sites. Adolescents completed the IMPACT-III (inflammatory bowel disease-specific HRQOL). Parents completed the Pediatric Inventory for Parents, a measure of medically related parenting stress that assesses stress because of the occurrence of medical stressors and stress because of the perceived difficulty of stressors. Disease activity was obtained from medical records. RESULTS: Parenting stress because of the occurrence of medical stressors partially mediated the disease severity-HRQOL relation, reducing the relation between these variables from 49.67% to 31.58% (B= -0.56, P < 0.0001). Bootstrapping analysis confirmed that the indirect effect of disease severity on HRQOL via parenting stress significantly differed from zero. Parenting stress because of the perceived difficulty of medical stressors partially mediated the disease severity-HRQOL relation, reducing the relation from 49.67% to 30.29% (B= -0.55, P < 0.0001). The indirect effect was confirmed via bootstrapping procedures. CONCLUSIONS: As disease severity increased, parenting stress also increased, and adolescent HRQOL decreased. Parenting stress should be considered and assessed for along with medical factors as part of a comprehensive approach to improve HRQOL in adolescents with Crohn disease.

Sleep patterns of a primarily obese sample of treatment-seeking children.

STUDY OBJECTIVES: To examine the sleep patterns and the role of day of the week and school break in these patterns within a primarily obese sample of children. METHODS: Participants included 143 obese children (8-12 years) and their parents initiating treatment in a weight-management study in a community-based setting. Demographics, anthropometrics, and objectively measured sleep (i.e., with use of Sensewear Armbands) were collected prior to treatment. RESULTS: Sleep duration was insufficient in our sample, as approximately 88% obtained less than 8 hours of sleep (mean = 6.92, standard deviation = 0.85). Those with lower total sleep time included older children, those identified as African American (compared to those identified as Caucasian), and those identified as Non-Hispanic (compared to those identified as Hispanic). Children on school break initiated sleep later than those in school the week of measurement. Children woke later on weekends and when on school break. There were no differences in day of the week or school break in predicting child sleep duration and total wake time (p's > 0.05). CONCLUSIONS: This study is one of the first to examine sleep patterns within a primarily obese sample of treatment-seeking rural children. There is a need for research to develop a better understanding of how sleep may affect health functioning and weight management, as well as quality of life and psychosocial functioning of children who are overweight or obese.

An exploration of family problem-solving and affective involvement as moderators between disease severity and depressive symptoms in adolescents with inflammatory bowel disease.

Little is known about how family functioning relates to psychosocial functioning of youth with inflammatory bowel disease (IBD). The study aim was to examine family problem solving and affective involvement as moderators between adolescent disease severity and depressive symptoms. Participants were 122 adolescents with IBD and their parents. Measures included self-reported and parent-reported adolescent depressive symptoms, parent-reported family functioning, and physician-completed measures of disease severity. Disease severity was a significant predictor of adolescent-reported depressive symptoms, but not parent-reported adolescent depressive symptoms. Family affective involvement significantly predicted parent-reported adolescent depressive symptoms, while family problem-solving significantly predicted adolescent self-report of depressive symptoms. Neither affective involvement nor problem-solving served as moderators. Family affective involvement may play an important role in adolescent emotional functioning but may not moderate the effect of disease severity on depressive symptoms. Research should continue to examine effects of family functioning on youth emotional functioning and include a sample with a wider range of disease severity to determine if interventions aimed to enhance family functioning are warranted.

Parenting stress in pediatric IBD: relations with child psychopathology, family functioning, and disease severity.

OBJECTIVE: Parenting stress in pediatric inflammatory bowel disease (IBD) has been under-examined. Data validating use of the Pediatric Inventory for Parents (PIP), a measure of parenting stress associated with caring for a chronically ill child, in chronic diseases with intermittent, unpredictable disease courses, such as IBD, are needed. This study presents validity data in support of the PIP in pediatric IBD and examines relations between parenting stress and important psychosocial and medical outcomes. METHODS: Adolescents (N = 130) with IBD and their caregivers across 3 sites completed measures of parenting stress, family functioning, and emotional/behavioral functioning. Disease severity was also assessed for each participant. RESULTS: The PIP demonstrates excellent internal consistency. Parenting stress was significantly higher among those with unhealthy general family functioning and those with children with borderline or clinically elevated internalizing symptoms. Caregiving stress was greater among parents of youth with more active Crohn's disease. CONCLUSION: Results supported the reliability and validity of the PIP for assessing caregiving stress in pediatric IBD. Routine assessment of parenting stress is recommended, particularly among parents reporting unhealthy family functioning and parents of youth with borderline or clinically elevated internalizing symptoms and more active disease.