Predictors of narcolepsy and hypersomnia due to medical disorder in pediatric craniopharyngioma

Mandrell, Belinda N.; LaRosa, Kayla; Hancock, Donna; Caples, Mary; Sykes, April; Lu, Zhaohua; Wise, Merrill S.; Khan, Raja B.; Merchant, Thomas E.; McLaughlin-Crabtree, Valerie

doi:10.1007/s11060-020-03519-3

Cited by 24 publications

(29 citation statements)

References 20 publications

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“…Impaired attention, slow processing speed, and hypersomnia are known complications of childhood brain tumor and its treatment, particularly radiotherapy 2,4,15,16 . There are multiple ways by which RT influences cognitive function, including gray and white matter volume loss, 2,5,17 and disruption of astroglial connexins 18 .…”

Section: Discussionmentioning

confidence: 99%

Height, weight, and cardiovascular effects of stimulants on children with brain tumors

Khan

Bano

Wang

et al. 2020

Pediatric Blood & Cancer

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Introduction: Children with brain tumors may develop inattention, slow processing, and hypersomnia. Stimulant medications improve these problems, but their effect on growth, heart rate, and blood pressure (BP) are inadequately explored. Procedure: We retrospectively studied children with brain tumors treated at our institution that had data available for 1 year pre and 2 years on stimulant treatment. Tumor location, gender, radiation treatment (RT), age at RT, drug type, and hormone therapy were variables of interest. Results: We identified 65 children (35 males) that fulfilled eligibility criteria. Focal RT was utilized in 58; 11 additionally had whole brain RT; and seven received no RT. Thirty were treated for hypersomnia and inattention, eight for hypersomnia alone, and rest for inattention. Modafinil was the first drug in 18 (27.7%), and methylphenidate in the others. Forty-seven (72.3%), 45 (69.2%), and 49 (75.4%) were on thyroxine, cortisone, and growth hormones, respectively. There was no difference in pre-and post-stimulant body mass index (BMI), heart rate, and BP. There was also no difference between modafinil and methylphenidate groups. Rate of height acquisition slowed on stimulants (P = .0096). Thyroxine treatment correlated with increase in BMI after stimulants (P = .04). Younger age (P = .0003) and higher prestimulant BMI (P = .0063) correlated with increased heart rate on stimulants, while higher age at RT (P =.016) correlated with elevated systolic BP on stimulants. No associations were found with height acquisition and diastolic BP. Conclusion: Stimulants are well tolerated by children with brain tumors that are appropriately managed for endocrine deficiencies, but may reduce the trajectory of height attainment. K E Y W O R D S blood pressure, brain tumors, childhood, heart rate, stimulants, weight 1 INTRODUCTION A significant number of childhood brain tumor survivors develop neurocognitive impairment, particularly attention span, processing speed, Abbreviations: BMI, body mass index; BP, blood pressure; RT, radiation treatment and working memory. 1,2 Cognitive deficits may result from effect of radiation and chemotherapy on the brain, or due to the direct tumor effect. 3 Latter may represent involvement of eloquent areas of brain by the tumor or disruption of brain connectivity. Younger age and radiation treatment (RT) are the most important risk factors for cognitive impairment in childhood brain tumor survivors, and may be related to loss of gray and white matter volume. 1,2,4,5

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Section: Discussionmentioning

confidence: 99%

Height, weight, and cardiovascular effects of stimulants on children with brain tumors

Khan

Bano

Wang

et al. 2020

Pediatric Blood & Cancer

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“…Sleep disorders (SDs) are classified according to the third edition of the International Classification of Sleep Disorders (ICSD-3) of the American Academy of Sleep Medicine (19), which identifies seven major categories of disorders: insomnia disorders, SBDs, central disorders of hypersomnolence, circadian rhythm sleepwake disorders (CRSWDs), sleep-related movement disorders, parasomnias, and other SDs. The most frequently reported SDs in CP are EDS (11,20), central hypersomnia and secondary narcolepsy (13,14,(21)(22)(23)(24)(25)(26)(27), SBDs (7,24), and CRSWD (28)(29)(30), which require appropriate characterization and understanding of their underlying causes. Since clinical pictures may be complex and different elements may co-exist in the same patient, this may be achieved through a specific expertise in sleep diseases.…”

Section: Classification and Pathogenesis Of Sleep Disorders In Patien...mentioning

confidence: 99%

“…Since clinical pictures may be complex and different elements may co-exist in the same patient, this may be achieved through a specific expertise in sleep diseases. Although the real prevalence of SDs before and after CP surgery/radiotherapy should be clarified, Mandrell et al ( 13 ) showed that 45% of a large sample of pediatric patients with CP were affected by hypersomnia due to a medical disorder and 35% by narcolepsy, and that the main predictor of sleepiness was obesity. In addition, 80% of this cohort complained of EDS at diagnosis or after neurosurgery.…”

Section: Classification and Pathogenesis Of Sleep Disorders In Patien...mentioning

confidence: 99%

“…Overall, SDs have received more attention in pediatric than in adult patients with CP, who include long-term survivors of childhood-onset CP and patients with an adult-onset disease. In pediatric cohorts, SDs have been heterogeneously reported as daytime sleepiness/hypersomnia, sleep disturbances such as difficulty to fall asleep or waking up during the night, and variably evaluated by self-assessment—specific questionnaires ( 11 ), items as a part of QoL assessment ( 12 )—and more recently by means of sleep medicine tools aiming to better define entities such as SBDs, like obstructive sleep apnea (OSA) ( 7 ) or secondary narcolepsy ( 13 ). Within the methodological limits and heterogeneity of reported studies, the prevalence of SDs and/or excessive daytime sleepiness (EDS) approaches 70–80% ( 11 , 13 ), with an adult prevalence of OSA around 40% ( 6 ).…”

Section: Introductionmentioning

confidence: 99%

“…In pediatric cohorts, SDs have been heterogeneously reported as daytime sleepiness/hypersomnia, sleep disturbances such as difficulty to fall asleep or waking up during the night, and variably evaluated by self-assessment—specific questionnaires ( 11 ), items as a part of QoL assessment ( 12 )—and more recently by means of sleep medicine tools aiming to better define entities such as SBDs, like obstructive sleep apnea (OSA) ( 7 ) or secondary narcolepsy ( 13 ). Within the methodological limits and heterogeneity of reported studies, the prevalence of SDs and/or excessive daytime sleepiness (EDS) approaches 70–80% ( 11 , 13 ), with an adult prevalence of OSA around 40% ( 6 ). A common observation is the higher prevalence of SDs in the presence of hypothalamic involvement, with a bi-directional interplay between obesity and SDs.…”

Section: Introductionmentioning

confidence: 99%

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Sleep Disorders in Patients With Craniopharyngioma: A Physiopathological and Practical Update

et al. 2022

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Sleep disorders (SDs) represent an important issue in patients with craniopharyngioma (CP). Nearly 70% of these patients complain of sleep-wake cycle alterations and/or excessive diurnal somnolence due to sleep-related breathing disorders, such as obstructive sleep apnea (OSA) and/or central hypersomnia, including secondary narcolepsy. SDs may severely reduce quality of life, increase disease-related cardiorespiratory and cardiovascular morbidity, and finally play a major role in increased long-term mortality reported on patients with CP. A major risk factor for SDs is represented by the hypothalamic syndrome, which may develop because of direct hypothalamic damage by the tumor itself and/or complications of the treatments, neurosurgery and/or radiotherapy, and typically includes permanent neuroendocrine dysfunctions, morbid obesity, and secondary metabolic disorders. Despite increasing attention to SDs in the general population, and in particular to OSA as a risk factor for cardio-metabolic diseases and excessive daytime somnolence, sleep evaluation is still not routinely proposed to patients with CP. Hence, SDs are often underdiagnosed and undertreated. The aim of this paper is to update current knowledge of the pathogenesis and prevalence of SDs in patients with CP and propose practical algorithms for their evaluation and management in clinical practice. Particular attention is paid to screening and diagnostic tools for appropriate characterization of SDs, identification of risk factors, and potential role of hypothalamic sparing surgery in the prevention of morbid obesity and SDs. Available tools in sleep medicine, including lifestyle interventions, drugs, and respiratory devices, are discussed, as well as the importance of optimal hormone replacement and metabolic interventions. Current limits in the diagnosis and treatment of SDs in patients with CP and possible future avenues for research agenda are also considered.

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Health‐related quality of life, obesity, fragmented sleep, fatigue, and psychosocial problems among youth with craniopharyngioma

et al. 2021

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Objective: Youth with craniopharyngioma experience weight gain, fragmented sleep, excessive daytime sleepiness (EDS), fatigue, and psychosocial problems that negatively impact their overall health-related quality of life (HRQoL). Greater hypothalamic tumor involvement (HI) may be associated with higher rates or severity of these impairments; however, the direct and indirect impact of HI on the physical and psychosocial consequences associated with pediatric craniopharyngioma remain unclear. The purpose of the current study was to examine relations between HI, body mass index (BMI), fragmented sleep, EDS, fatigue, psychosocial problems, and HRQoL among youth with craniopharyngioma. Methods: Eighty-four youth with craniopharyngioma (M age = 10.27 � 4.3 years, 53.6% female, 64.3% White) were assessed with actigraphy, nocturnal polysomnography, and multiple sleep latency tests prior to proton therapy, when indicated. Caregivers completed measures of fatigue, psychosocial functioning, and HRQoL. Results: Hypothalamic tumor involvement was associated with greater BMI (Est. = 2.97, p = 0.003) and daytime sleepiness (Est. = 2.53, p = 0.01). Greater fatigue predicted more psychosocial problems (Est. = 0.29, p < 0.001) and lower HRQoL (Est. = 0.23, p = 0.001). Psychosocial problems also predicted lower HRQoL (Est. = −0.34, p = 0.004). Fragmented sleep (Est. = 0.03, p = 0.04) and fatigue (Est. = 0.10, p = 0.02) indirectly predicted lower HRQoL through psychosocial problems. Conclusions: Youth with craniopharyngioma with greater HI may benefit from weight reduction interventions and management of excessive sleepiness. Patients should be prospectively monitored for sleep problems, fatigue, and psychosocial problems, as these patients may benefit from interventions targeting fatigue and psychosocial health to improve HRQoL.

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Predictors of narcolepsy and hypersomnia due to medical disorder in pediatric craniopharyngioma

Cited by 24 publications

References 20 publications

Height, weight, and cardiovascular effects of stimulants on children with brain tumors

Height, weight, and cardiovascular effects of stimulants on children with brain tumors

Sleep Disorders in Patients With Craniopharyngioma: A Physiopathological and Practical Update

Health‐related quality of life, obesity, fragmented sleep, fatigue, and psychosocial problems among youth with craniopharyngioma

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