- Email: [email protected]
Daytime sleepiness in children: When a quiet child is not necessarily a good thing
G Model
YPRRV-1193; No. of Pages 2 Paediatric Respiratory Reviews xxx (2017) xxx–xxx
Contents lists available at ScienceDirect
Paediatric Respiratory Reviews
Mini-Symposium: Pediatric Hypersomnolence Symposium Editorial
Daytime sleepiness in children: When a quiet child is not necessarily a good thing
Excessive daytime sleepiness (EDS) in children is a common but under-recognized symptom. A quiet child is a “good” child who does not cause problems and may be ignored by a busy teacher in an overcrowded classroom, or a harried parent juggling household and work responsibilities. EDS is common; in a community-based sample of school-aged children, the prevalence of parent or teacher-reported EDS was estimated at 15% [1]. However, few of these children will come to medical attention, and it thus behooves the pediatrician to ask questions regarding sleep during regular health maintenance visits. Sleepiness has significant adverse effects on learning, mood and quality of life, and thus warrants early recognition and management [1–3]. While sleep deprivation/disruption may result in attentiondeficit/hyperactivity disorder (ADHD)-like symptoms rather than EDS in some children, this mini symposium focuses on actual somnolence. By far the commonest cause of EDS in children is insufficient sleep, often associated with poor sleep hygiene. No fancy tools are needed to recognize this – a simple history, and perhaps a sleep diary, will identify the issues in the vast majority of cases. A particular challenge is the adolescent, who invariably has some degree of physiologic phase shift (i.e., a tendency to become sleepy only late at night, with associated sleepiness in the morning if forced to get up early for school). Although some adolescents have a pathological degree of delayed sleep phase, others may exploit this late-night alertness to socialize using electronic media, resulting in secondary gain if they are allowed to miss school the next day because they are too sleepy to get up. Actigraphy can be very helpful when the child or adolescent and parent give conflicting, vague or questionable histories. These days, many phone apps and over the counter devices provide crude information which may be sufficient to illustrate to the family that sleep is insufficient [4]. The article by Thomas and Burgers in this symposium in Paediatric Respiratory Reviews describes common behavioral causes of EDS in children, with evidence-based approaches to managing these conditions [5]. Medical disorders, ranging from central nervous system tumors to the use of freely available medications such as antihistamines, may result in EDS. The obstructive sleep apnea syndrome may cause overt sleepiness even in young children [6]. Far less common, but of great interest and medical concern, are the specific sleep disorders resulting in EDS, including narcolepsy and Kleine-Levin syndrome (KLS). These conditions are rare enough that the average
pediatrician may never encounter a case. However, it is imperative that the pediatrician be able to identify a degree of EDS that is clearly pathologic, and refer the patient for further evaluation and treatment, as late diagnosis can have a tremendous negative impact on the patient’s wellbeing. Although narcolepsy is a pediatric condition, presenting during the first or second decade of life in more than half of patients [7], it often remains undiagnosed until adulthood [8]. Children with narcolepsy may be misdiagnosed as having depression or even orthodontic or neuromuscular conditions that in fact are manifestations of cataplexy [9]. Childhood narcolepsy is associated with secondary behavioral problems, anxiety and depression [10,11], and it may be that earlier diagnosis can ameliorate these psychosocial sequelae. Comprehensive reviews of childhood narcolepsy have been published recently [11]. The article in this symposium by Dye, Gurbani and Simakajornboon focuses on recent epidemiological shifts, with emphasis on the role of the influenza virus (specifically, H1N1) and certain H1N1 vaccines, in the pathophysiology of narcolepsy [12]. Kleine-Levin syndrome, which consists of cyclic relapsing periods of EDS associated with other psychiatric symptoms, may be thought of as an esoteric condition and one with very few treatment options, but just being able to provide a diagnosis to the family may provide great relief (“no, you and your child are not going crazy, this is a true medical problem that will eventually get better”). This rare but fascinating condition is reviewed in the article by Afolabi-Brown and Mason [13]. Finally, Kotagal describes current treatment options – not only medications, but also important supportive measures such as sleep hygiene, treatment of psychological comorbidities, the importance of exercise, and vocational counselling [14]. As is so often the case in pediatrics, many of the medications used for narcolepsy have not been approved by the Food and Drug Administration for children, and are lacking pharmacokinetic studies. Further research in this area is desperately needed, but in the interim, clinical experience from seasoned pediatric sleep physicians is helpful in guiding management. The articles in this symposium are thoughtful and comprehensive, and reflect the current state of knowledge of hypersomnolence disorders in children. It is clear, however, that much remains unknown about these conditions. Sleep medicine is a new field. As we do not even fully understand the reason why we sleep for such a large part of our lives, it is not surprising that we do not fully understand the conditions resulting in EDS. This should be seen as an opportunity rather than a problem. Sleep medicine, and
http://dx.doi.org/10.1016/j.prrv.2017.01.002 1526-0542/© 2017 Published by Elsevier Ltd.
Please cite this article in press as: Marcus CL. Daytime sleepiness in children: When a quiet child is not necessarily a good thing. Paediatr. Respir. Rev. (2017), http://dx.doi.org/10.1016/j.prrv.2017.01.002
G Model
YPRRV-1193; No. of Pages 2 2
Editorial / Paediatric Respiratory Reviews xxx (2017) xxx–xxx
particularly pediatric sleep medicine, presents great opportunities for clinicians and researchers entering the field. References [1] Calhoun SL, Vgontzas AN, Fernandez-Mendoza J, et al. Prevalence and risk factors of excessive daytime sleepiness in a community sample of young children: the role of obesity, asthma, anxiety/depression, and sleep. Sleep 2011;34(4):503–7. [2] Calhoun SL, Fernandez-Mendoza J, Vgontzas AN, et al. Learning, attention/ hyperactivity, and conduct problems as sequelae of excessive daytime sleepiness in a general population study of young children. Sleep 2012;35(5):627–32. [3] Gustafsson ML, Laaksonen C, Aromaa M, et al. Association between amount of sleep, daytime sleepiness and health-related quality of life in schoolchildren. J Adv Nurs 2016;72(6):1263–72. [4] Toon E, Davey MJ, Hollis SL, Nixon GM, Horne RS, Biggs SN. Comparison of Commercial Wrist-Based and Smartphone Accelerometers, Actigraphy, and PSG in a Clinical Cohort of Children and Adolescents. J Clin Sleep Med 2016;12 (3):343–50. [5] Thomas JH, Burger DE. Sleep is an eye-opener: Behavioral causes and consequences of hypersomnolence in children. Paediatr Respir Rev 2016. http://dx. doi.org/10.1016/j.prrv.2016.11.004 [6] Paruthi S, Buchanan P, Weng J, et al. Effect of Adenotonsillectomy on ParentReported Sleepiness in Children with Obstructive Sleep Apnea. Sleep 2016;39 (11):2005–12. [7] Ohayon MM, Ferini-Strambi L, Plazzi G, Smirne S, Castronovo V. How age influences the expression of narcolepsy. J Psychosom Res 2005;59(6):399–405. [8] Thorpy MJ, Krieger AC. Delayed diagnosis of narcolepsy: characterization and impact. Sleep Med 2014;15(5):502–7. [9] Kauta SR, Marcus CL. Cases of pediatric narcolepsy after misdiagnoses. Pediatr Neurol 2012;47(5):362–5.
[10] Stores G, Montgomery P, Wiggs L. The psychosocial problems of children with narcolepsy and those with excessive daytime sleepiness of uncertain origin. Pediatrics 2006;118(4):e1116–23. [11] Aran A, Einen M, Lin L, Plazzi G, Nishino S, Mignot E. Clinical and therapeutic aspects of childhood narcolepsy-cataplexy: a retrospective study of 51 children. Sleep 2010;33(11):1457–64. [12] Dye T, Gurbani N, Simakajornboon N. Epidemiology and Pathophysiology of Childhood Narcolepsy. Paediatr Respir Reviews 2017. http://dx.doi.org/10.1016/ j.prrv.2016.12.005 [13] Afolabi-Brown O, Mason TBA (II), Kleine-Levin Syndrome, in press. [14] Kotagal S, Treatment of narcolepsy and other organic hypersomnias in children, in press.
Carole L. Marcus* Sleep Center, Children’s Hospital of Philadelphia, and the Perelman School of Medicine at the University of Pennsylvania, Philadelphia, Pennsylvania *Corresponding author. Children’s Hospital of Philadelphia, Sleep Center, Suite 9NW50, Main Hospital, 3401 Civic Center Boulevard, Philadelphia, PA 19104. Phone: 267-426-5842, Fax: 267-426-9234 E-mail address: [email protected] (C.L. Marcus).
Please cite this article in press as: Marcus CL. Daytime sleepiness in children: When a quiet child is not necessarily a good thing. Paediatr. Respir. Rev. (2017), http://dx.doi.org/10.1016/j.prrv.2017.01.002
YPRRV-1193; No. of Pages 2 Paediatric Respiratory Reviews xxx (2017) xxx–xxx
Contents lists available at ScienceDirect
Paediatric Respiratory Reviews
Mini-Symposium: Pediatric Hypersomnolence Symposium Editorial
Daytime sleepiness in children: When a quiet child is not necessarily a good thing
Excessive daytime sleepiness (EDS) in children is a common but under-recognized symptom. A quiet child is a “good” child who does not cause problems and may be ignored by a busy teacher in an overcrowded classroom, or a harried parent juggling household and work responsibilities. EDS is common; in a community-based sample of school-aged children, the prevalence of parent or teacher-reported EDS was estimated at 15% [1]. However, few of these children will come to medical attention, and it thus behooves the pediatrician to ask questions regarding sleep during regular health maintenance visits. Sleepiness has significant adverse effects on learning, mood and quality of life, and thus warrants early recognition and management [1–3]. While sleep deprivation/disruption may result in attentiondeficit/hyperactivity disorder (ADHD)-like symptoms rather than EDS in some children, this mini symposium focuses on actual somnolence. By far the commonest cause of EDS in children is insufficient sleep, often associated with poor sleep hygiene. No fancy tools are needed to recognize this – a simple history, and perhaps a sleep diary, will identify the issues in the vast majority of cases. A particular challenge is the adolescent, who invariably has some degree of physiologic phase shift (i.e., a tendency to become sleepy only late at night, with associated sleepiness in the morning if forced to get up early for school). Although some adolescents have a pathological degree of delayed sleep phase, others may exploit this late-night alertness to socialize using electronic media, resulting in secondary gain if they are allowed to miss school the next day because they are too sleepy to get up. Actigraphy can be very helpful when the child or adolescent and parent give conflicting, vague or questionable histories. These days, many phone apps and over the counter devices provide crude information which may be sufficient to illustrate to the family that sleep is insufficient [4]. The article by Thomas and Burgers in this symposium in Paediatric Respiratory Reviews describes common behavioral causes of EDS in children, with evidence-based approaches to managing these conditions [5]. Medical disorders, ranging from central nervous system tumors to the use of freely available medications such as antihistamines, may result in EDS. The obstructive sleep apnea syndrome may cause overt sleepiness even in young children [6]. Far less common, but of great interest and medical concern, are the specific sleep disorders resulting in EDS, including narcolepsy and Kleine-Levin syndrome (KLS). These conditions are rare enough that the average
pediatrician may never encounter a case. However, it is imperative that the pediatrician be able to identify a degree of EDS that is clearly pathologic, and refer the patient for further evaluation and treatment, as late diagnosis can have a tremendous negative impact on the patient’s wellbeing. Although narcolepsy is a pediatric condition, presenting during the first or second decade of life in more than half of patients [7], it often remains undiagnosed until adulthood [8]. Children with narcolepsy may be misdiagnosed as having depression or even orthodontic or neuromuscular conditions that in fact are manifestations of cataplexy [9]. Childhood narcolepsy is associated with secondary behavioral problems, anxiety and depression [10,11], and it may be that earlier diagnosis can ameliorate these psychosocial sequelae. Comprehensive reviews of childhood narcolepsy have been published recently [11]. The article in this symposium by Dye, Gurbani and Simakajornboon focuses on recent epidemiological shifts, with emphasis on the role of the influenza virus (specifically, H1N1) and certain H1N1 vaccines, in the pathophysiology of narcolepsy [12]. Kleine-Levin syndrome, which consists of cyclic relapsing periods of EDS associated with other psychiatric symptoms, may be thought of as an esoteric condition and one with very few treatment options, but just being able to provide a diagnosis to the family may provide great relief (“no, you and your child are not going crazy, this is a true medical problem that will eventually get better”). This rare but fascinating condition is reviewed in the article by Afolabi-Brown and Mason [13]. Finally, Kotagal describes current treatment options – not only medications, but also important supportive measures such as sleep hygiene, treatment of psychological comorbidities, the importance of exercise, and vocational counselling [14]. As is so often the case in pediatrics, many of the medications used for narcolepsy have not been approved by the Food and Drug Administration for children, and are lacking pharmacokinetic studies. Further research in this area is desperately needed, but in the interim, clinical experience from seasoned pediatric sleep physicians is helpful in guiding management. The articles in this symposium are thoughtful and comprehensive, and reflect the current state of knowledge of hypersomnolence disorders in children. It is clear, however, that much remains unknown about these conditions. Sleep medicine is a new field. As we do not even fully understand the reason why we sleep for such a large part of our lives, it is not surprising that we do not fully understand the conditions resulting in EDS. This should be seen as an opportunity rather than a problem. Sleep medicine, and
http://dx.doi.org/10.1016/j.prrv.2017.01.002 1526-0542/© 2017 Published by Elsevier Ltd.
Please cite this article in press as: Marcus CL. Daytime sleepiness in children: When a quiet child is not necessarily a good thing. Paediatr. Respir. Rev. (2017), http://dx.doi.org/10.1016/j.prrv.2017.01.002
G Model
YPRRV-1193; No. of Pages 2 2
Editorial / Paediatric Respiratory Reviews xxx (2017) xxx–xxx
particularly pediatric sleep medicine, presents great opportunities for clinicians and researchers entering the field. References [1] Calhoun SL, Vgontzas AN, Fernandez-Mendoza J, et al. Prevalence and risk factors of excessive daytime sleepiness in a community sample of young children: the role of obesity, asthma, anxiety/depression, and sleep. Sleep 2011;34(4):503–7. [2] Calhoun SL, Fernandez-Mendoza J, Vgontzas AN, et al. Learning, attention/ hyperactivity, and conduct problems as sequelae of excessive daytime sleepiness in a general population study of young children. Sleep 2012;35(5):627–32. [3] Gustafsson ML, Laaksonen C, Aromaa M, et al. Association between amount of sleep, daytime sleepiness and health-related quality of life in schoolchildren. J Adv Nurs 2016;72(6):1263–72. [4] Toon E, Davey MJ, Hollis SL, Nixon GM, Horne RS, Biggs SN. Comparison of Commercial Wrist-Based and Smartphone Accelerometers, Actigraphy, and PSG in a Clinical Cohort of Children and Adolescents. J Clin Sleep Med 2016;12 (3):343–50. [5] Thomas JH, Burger DE. Sleep is an eye-opener: Behavioral causes and consequences of hypersomnolence in children. Paediatr Respir Rev 2016. http://dx. doi.org/10.1016/j.prrv.2016.11.004 [6] Paruthi S, Buchanan P, Weng J, et al. Effect of Adenotonsillectomy on ParentReported Sleepiness in Children with Obstructive Sleep Apnea. Sleep 2016;39 (11):2005–12. [7] Ohayon MM, Ferini-Strambi L, Plazzi G, Smirne S, Castronovo V. How age influences the expression of narcolepsy. J Psychosom Res 2005;59(6):399–405. [8] Thorpy MJ, Krieger AC. Delayed diagnosis of narcolepsy: characterization and impact. Sleep Med 2014;15(5):502–7. [9] Kauta SR, Marcus CL. Cases of pediatric narcolepsy after misdiagnoses. Pediatr Neurol 2012;47(5):362–5.
[10] Stores G, Montgomery P, Wiggs L. The psychosocial problems of children with narcolepsy and those with excessive daytime sleepiness of uncertain origin. Pediatrics 2006;118(4):e1116–23. [11] Aran A, Einen M, Lin L, Plazzi G, Nishino S, Mignot E. Clinical and therapeutic aspects of childhood narcolepsy-cataplexy: a retrospective study of 51 children. Sleep 2010;33(11):1457–64. [12] Dye T, Gurbani N, Simakajornboon N. Epidemiology and Pathophysiology of Childhood Narcolepsy. Paediatr Respir Reviews 2017. http://dx.doi.org/10.1016/ j.prrv.2016.12.005 [13] Afolabi-Brown O, Mason TBA (II), Kleine-Levin Syndrome, in press. [14] Kotagal S, Treatment of narcolepsy and other organic hypersomnias in children, in press.
Carole L. Marcus* Sleep Center, Children’s Hospital of Philadelphia, and the Perelman School of Medicine at the University of Pennsylvania, Philadelphia, Pennsylvania *Corresponding author. Children’s Hospital of Philadelphia, Sleep Center, Suite 9NW50, Main Hospital, 3401 Civic Center Boulevard, Philadelphia, PA 19104. Phone: 267-426-5842, Fax: 267-426-9234 E-mail address: [email protected] (C.L. Marcus).
Please cite this article in press as: Marcus CL. Daytime sleepiness in children: When a quiet child is not necessarily a good thing. Paediatr. Respir. Rev. (2017), http://dx.doi.org/10.1016/j.prrv.2017.01.002