본문 바로가기
KERD
논문KCI 등재후보

Clinical and Polysomnographic Characteristics of Childhood Narcolepsy in Korea

1

조회

0

다운로드

0

인용

0

참고문헌

키워드

주제분류신경과학

초록

Purpose: Diagnosis of narcolepsy in childhood is challenging because of ambiguous expression of symptoms and various clinical presentations. The purpose of this study is to delineate the clinical and polysomnographic characteristics of childhood narcolepsy. Methods: This study included 78 patients who were diagnosed with narcolepsy by clinical history and polysomnography (PSG) between February 2002 and June 2015 at Samsung Medical Center. Their medical records were reviewed retrospectively and analyzed between two groups; age of symptom onset and the presence of cataplexy. Results: The age at diagnosis and symptom onset was 15.51±2.73 years and 13.03± 2.79 years. Forty-one patients (52.6%) were diagnosed with narcolepsy with cataplexy. All exhibited excessive daytime sleepiness as the first symptom. In the multiple sleep latency test (MSLT), short sleep latency and more than 3 episodes of mean sleep onset of rapid eye movement were observed. The coexisting sleep disorders varied according to age of onset. There was no significant difference in sleep parameters between the two age groups. The patients with cataplexy had higher scores on the Epworth Sleepiness Scale and shorter mean sleep latencies in the MSLT than those without cataplexy (P<0.05). Conclusion: Pediatric narcolepsy presents with excessive daytime sleepiness and has different co-existing sleep disorders according to age of onset. The degree of sleepiness is higher in the patients with cataplexy. Narcolepsy may be suspected in children with excessive daytime sleepiness even if they do not have cataplexy. This should be confirmed using appropriate sleep questionnaires and PSG with MSLT.

Abstract

Purpose: Diagnosis of narcolepsy in childhood is challenging because of ambiguous expression of symptoms and various clinical presentations. The purpose of this study is to delineate the clinical and polysomnographic characteristics of childhood narcolepsy. Methods: This study included 78 patients who were diagnosed with narcolepsy by clinical history and polysomnography (PSG) between February 2002 and June 2015 at Samsung Medical Center. Their medical records were reviewed retrospectively and analyzed between two groups; age of symptom onset and the presence of cataplexy. Results: The age at diagnosis and symptom onset was 15.51±2.73 years and 13.03± 2.79 years. Forty-one patients (52.6%) were diagnosed with narcolepsy with cataplexy. All exhibited excessive daytime sleepiness as the first symptom. In the multiple sleep latency test (MSLT), short sleep latency and more than 3 episodes of mean sleep onset of rapid eye movement were observed. The coexisting sleep disorders varied according to age of onset. There was no significant difference in sleep parameters between the two age groups. The patients with cataplexy had higher scores on the Epworth Sleepiness Scale and shorter mean sleep latencies in the MSLT than those without cataplexy (P<0.05). Conclusion: Pediatric narcolepsy presents with excessive daytime sleepiness and has different co-existing sleep disorders according to age of onset. The degree of sleepiness is higher in the patients with cataplexy. Narcolepsy may be suspected in children with excessive daytime sleepiness even if they do not have cataplexy. This should be confirmed using appropriate sleep questionnaires and PSG with MSLT.

참고문헌 (34)

  1. Liblau RS, "Hypocretin (orexin) biology and the pathophysiology of narcolepsy with cataplexy", Lancet Neurol, 14, pp.318~328 (2015)
  2. Tanaka S, "Detection of autoantibodies against hypocretin, hcrtrl, and hcrtr2 in narcolepsy: anti- Hcrt system antibody in narcolepsy", Sleep, 29, pp.633~638 (2006)
  3. 방양원, "소아 기면증의 진단과 치료", 대한소아신경학회지, 17(2), pp.107~117 (2009), 대한소아신경학회
  4. Babiker MO, "Narcolepsy in children: a diagnostic and management approach", Pediatr Neurol, 52, pp.557~565 (2015)
  5. Dias Costa F, "Narcolepsy in pediatric age - Experience of a tertiary pediatric hospital", Sleep Sci, 7, pp.53~58 (2014)
  6. Nevsimalova S, "Narcolepsy: clinical differences and association with other sleep disorders in different age groups", J Neuro, 260, pp.767~775 (2013)
  7. Szakács A, "Increased childhood incidence of narcolepsy in western Sweden after H1N1 influenza vaccination", Neurology, 80, pp.1315~1321 (2013)
  8. Partinen M, "Increased incidence and clinical picture of childhood narcolepsy following the 2009 H1N1 pandemic vaccination campaign in Finland", PLoS One, 7, pp.e33723~ (2012)
  9. Morrish E, "Factors associated with a delay in the diagnosis of narcolepsy", Sleep Med, 5, pp.37~41 (2004)
  10. Inocente CO, "Depressive feelings in children with narcolepsy", Sleep Med, 15, pp.309~314 (2014)
  11. Thorpy MJ, "Delayed diagnosis of narcolepsy: characterization and impact", Sleep Med, 15, pp.502~507 (2014)
  12. Nakamura M, "Differences in brain morphological findings between narcolepsy with and without cataplexy", PLoS One, 28, pp.e81059~ (2013)
  13. Medicine AAoS, "International classification of sleep disorders: diagnostic and coding manual" (2005), American Academy of Sleep Medicine
  14. Robert MK, "Nelson Textbook of Pediatrics", pp.926~ (2015), Elsevier
  15. Calhoun SL, "Prevalence and risk factors of excessive daytime sleepiness in a community sample of young children: the role of obesity, asthma, anxiety", depression, and sleep, 2011, pp.34, 503~507
  16. Johns MW, "A new method for measuring daytime sleepiness: the Epworth sleepiness scale", Sleep, 14, pp.540~545 (1991)
  17. Hori T, "Proposed supplements and amendments to 'A Manual of Standardized Terminology, Techniques and Scoring System for Sleep Stages of Human Subjects', the Rechtschaffen & Kales (1968) standard", Psychiatry Clin Neurosci, 55, pp.305~310 (2001)
  18. American Sleep Disorders Association and Sleep Research Society, "EEG arousals: scoring rules and examples: a preliminary report from the Sleep Disorders Atlas Task Force of the American Sleep Disorders Association", Sleep, 15, pp.173~184 (1992)
  19. American Academy of Sleep Medicine, "The AASM manual for the scoring of sleep and associated events; rules terminology and technical specifications", pp.1~59 (2007)
  20. Carskadon MA, "Guidelines for the multiple sleep latency test (MSLT): a standard measure of sleepiness", Sleep, 9, pp.519~524 (1986)
  21. Hintze JP, "Sleep in the Pediatric Population", Sleep Med Clin, 11, pp.91~103 (2016)
  22. Dauvilliers Y, "Age at onset of narcolepsy in two large populations of patients in France and Quebec", Neurology, 57, pp.2029~2033 (2001)
  23. Parkes JD, "The narcoleptic syndrome", J Neurol Neurosurg Psychiatry, 59, pp.221~224 (1995)
  24. Frauscher B, "Delayed diagnosis, range of severity, and multiple sleep comorbidities: a clinical and polysomnographic analysis of 100 patients of the innsbruck narcolepsy cohort", J Clin Sleep Med, 9, pp.805~812 (2013)
  25. Carter LP, "Patients' journeys to a narcolepsy diagnosis: a physician survey and retrospective chart review", Postgrad Med, 126, pp.216~224 (2014)
  26. Peterson PC, "Pediatric narcolepsy", Brain Dev, 30, pp.609~623 (2008)
  27. Jambhekar SK, "Periodic limb movements during sleep in children with narcolepsy", J Clin Sleep Med, 7, pp.597~601 (2011)
  28. Heier MS, "Incidence of narcolepsy in Norwegian children and adolescents after vaccination against H1N1 influenza A", Sleep Med, 14, pp.867~871 (2013)
  29. Rocca FL, "Narcolepsy during Childhood: An Update", Neuropediatrics, 46, pp.181~198 (2015)
  30. Kauta SR, "Cases of pediatric narcolepsy after misdiagnoses", Pediatr Neurol, 47, pp.362~365 (2012)
  31. Leu-Semenescu S, "Hallucinations in narcolepsy with and without cataplexy : contrasts with Parkinson’s disease", Sleep Med, 12, pp.497~504 (2011)
  32. Sasai T, "Comparison of clinical characteristics among narcolepsy with and without cataplexy and idiopathic hypersomnia without long sleep time, focusing on HLA-DRB1*1501", DQB1*0602 finding, 2009, pp.10, 961~966
  33. Andlauer O, "Predictors of hypocretin (orexin) deficiency in narcolepsy without cataplexy", Sleep, 35, pp.1247~1255F (2012)
  34. Šonka K, "Narcolepsy with and without cataplexy, idiopathic hypersomnia with and without long sleep time: a cluster analysis", Sleep Med, 16, pp.225~231 (2015)

관련 논문AI추천

이 간행물의 다른 논문 (16건)