title: Assessing sleepiness and cataplexy in children and adolescents with narcolepsy: a review of current patient-reported measures.
abstract: OBJECTIVE
  The objective of this study was to review patient-reported outcome measures assessing excessive daytime sleepiness (EDS) or cataplexy in children or adolescents to determine their usefulness and limitations in pediatric narcolepsy assessment.
  
  
  METHODS
  Searches were performed in Embase and Medline for pediatric measures of EDS and cataplexy that are either patient- or proxy-reported, and searches of http://www.clinicaltrials.gov/ were conducted for studies in narcolepsy that included at least one patient-reported measure. Further review was performed if sleepiness questionnaires (child or proxy-reported), sleep questionnaires that may contain sleepiness questions, proxy-reported child behavior questionnaires, or information on cataplexy measures were mentioned.
  
  
  RESULTS
  All self-reported cataplexy questionnaires from among 27 citations were study-specific diaries and were not identifiable as a recognized validated questionnaire. For EDS, 118 of 401 abstracts were further reviewed and the names of 21 questionnaires identified, of which eight questionnaires did not return additional citations of their validation. The Epworth Sleepiness Scale (ESS) or a modified version was the most frequently used measure of EDS. Although all measures were associated with limitations for use in the pediatric population, the ESS has been successfully used in adolescents and was deemed readily amenable to further modification for children.
  
  
  CONCLUSIONS
  There remains a dearth of validated measures for assessing EDS and cataplexy in children and adolescents with narcolepsy. The need for these measures may be filled by modification or adaptation of existing adult measures; a daily cataplexy diary and the ESS may be readily modified to make them child-friendly with regard to wording and settings, but should still undergo psychometric validation.
authors.affiliation: Mapi, Lyon, France. Electronic address: kbenmedjahed@mapigroup.com.
authors.forename: Khadra
authors.initials: K
authors.lastname: Benmedjahed
authors.affiliation: Jazz Pharmaceuticals, Palo Alto, CA, USA.
authors.forename: Y Grace
authors.initials: YG
authors.lastname: Wang
authors.affiliation: Mapi, Lyon, France.
authors.forename: Jérémy
authors.initials: J
authors.lastname: Lambert
authors.affiliation: Endpoint Outcomes, Boston, MA, USA.
authors.forename: Christopher
authors.initials: C
authors.lastname: Evans
authors.affiliation: Endpoint Outcomes, Boston, MA, USA.
authors.forename: Steve
authors.initials: S
authors.lastname: Hwang
authors.affiliation: Jazz Pharmaceuticals, Palo Alto, CA, USA; Stanford Sleep Medicine Center, Redwood City, CA, USA.
authors.forename: Jed
authors.initials: J
authors.lastname: Black
authors.affiliation: Epworth Sleep Centre, Melbourne, VIC, Australia.
authors.forename: Murray W
authors.initials: MW
authors.lastname: Johns
mesh_terms: D000293:Adolescent; D002385:Cataplexy / Q000175:diagnosis* / Q000209:etiology; D002648:Child; D006970:Disorders of Excessive Somnolence / Q000175:diagnosis / Q000209:etiology*; D005260:Female; D006801:Humans; D008297:Male; D009290:Narcolepsy / Q000150:complications*; D000071066:Patient Reported Outcome Measures; D011594:Psychometrics; D011795:Surveys and Questionnaires
keywords: Adolescents; Assessment; Cataplexy; Children; Narcolepsy; Sleepiness