Aviv Goldbart1, Paul Peppard2, Laurel Finn2, Chad M Ruoff3, Jodi Barnet4, Terry Young2, Emmanuel Mignot3. 1. Department of Pediatrics and Sleep Wake Disorders Unit, Faculty of Health Sciences, Ben Gurion University, Soroka University Medical Center, Beer Sheva, Israel ; Center for Sleep Sciences and Medicine, and Department of Psychiatry and Behavioral Sciences, Stanford University School of Medicine, Palo Alto, CA. 2. Department of Population Health Sciences, University of Wisconsin-Madison, Madison, WI. 3. Center for Sleep Sciences and Medicine, and Department of Psychiatry and Behavioral Sciences, Stanford University School of Medicine, Palo Alto, CA. 4. Department of Pediatrics and Sleep Wake Disorders Unit, Faculty of Health Sciences, Ben Gurion University, Soroka University Medical Center, Beer Sheva, Israel.
Abstract
STUDY OBJECTIVES: To study whether positive multiple sleep latency tests (MSLTs, mean sleep latency [MSL] ≤ 8 minutes, ≥ 2 sleep onset REM sleep periods [SOREMPs]) and/or nocturnal SOREMP (REM sleep latency ≤ 15 minutes during nocturnal polysomonography [NPSG]) are stable traits and can reflect incipient narcolepsy. DESIGN AND SETTING: Cross-sectional and longitudinal investigation of the Wisconsin Sleep Cohort Study. PARTICIPANTS: Adults (44% females, 30-81 years) underwent NPSG (n = 4,866 in 1,518 subjects), and clinical MSLT (n = 1,135), with 823 having a repeat NPSG-MSLT at 4-year intervals, totaling 1725 NPSG with MSLT studies. Data were analyzed using linear mixed-effects models, and the stability of positive MSLTs was explored using κ statistics. MEASUREMENTS AND RESULTS: Prevalence of a nocturnal SOREMP on a NPSG, of ≥ 2 SOREMPs on the MSLT, of MSL ≤ 8 minutes on the MSLT, and of a positive MSLT (MSL ≤ 8 minutes plus ≥ 2 SOREMPs) were 0.35%, 7.0%, 22%, and 3.4%, respectively. Correlates of a positive MSLT were shift work (OR = 7.8, P = 0.0001) and short sleep (OR = 1.51/h, P = 0.04). Test-retest for these parameters was poor, with κ < 0.2 (n.s.) after excluding shift workers and short sleepers. Excluding shift-work, short sleep, and subjects with negative MSLTs, we found one undiagnosed subject with possible cataplexy (≥ 1/month) and a NPSG SOREMPs; one subject previously diagnosed with narcolepsy without cataplexy with 2 NPSG SOREMPs and a positive MSLT, and two subjects with 2 independently positive MSLTs (66% human leukocyte antigen [HLA] positive). The proportions for narcolepsy with and without cataplexy were 0.07% (95% CI: 0.02-0.37%) and 0.20% (95% CI: 0.07-0.58%), respectively. CONCLUSIONS: The diagnostic value of multiple sleep latency tests is strongly altered by shift work and to a lesser extent by chronic sleep deprivation. The prevalence of narcolepsy without cataplexy may be 3-fold higher than that of narcolepsy-cataplexy.
STUDY OBJECTIVES: To study whether positive multiple sleep latency tests (MSLTs, mean sleep latency [MSL] ≤ 8 minutes, ≥ 2 sleep onset REM sleep periods [SOREMPs]) and/or nocturnal SOREMP (REM sleep latency ≤ 15 minutes during nocturnal polysomonography [NPSG]) are stable traits and can reflect incipient narcolepsy. DESIGN AND SETTING: Cross-sectional and longitudinal investigation of the Wisconsin Sleep Cohort Study. PARTICIPANTS: Adults (44% females, 30-81 years) underwent NPSG (n = 4,866 in 1,518 subjects), and clinical MSLT (n = 1,135), with 823 having a repeat NPSG-MSLT at 4-year intervals, totaling 1725 NPSG with MSLT studies. Data were analyzed using linear mixed-effects models, and the stability of positive MSLTs was explored using κ statistics. MEASUREMENTS AND RESULTS: Prevalence of a nocturnal SOREMP on a NPSG, of ≥ 2 SOREMPs on the MSLT, of MSL ≤ 8 minutes on the MSLT, and of a positive MSLT (MSL ≤ 8 minutes plus ≥ 2 SOREMPs) were 0.35%, 7.0%, 22%, and 3.4%, respectively. Correlates of a positive MSLT were shift work (OR = 7.8, P = 0.0001) and short sleep (OR = 1.51/h, P = 0.04). Test-retest for these parameters was poor, with κ < 0.2 (n.s.) after excluding shift workers and short sleepers. Excluding shift-work, short sleep, and subjects with negative MSLTs, we found one undiagnosed subject with possible cataplexy (≥ 1/month) and a NPSG SOREMPs; one subject previously diagnosed with narcolepsy without cataplexy with 2 NPSG SOREMPs and a positive MSLT, and two subjects with 2 independently positive MSLTs (66% human leukocyte antigen [HLA] positive). The proportions for narcolepsy with and without cataplexy were 0.07% (95% CI: 0.02-0.37%) and 0.20% (95% CI: 0.07-0.58%), respectively. CONCLUSIONS: The diagnostic value of multiple sleep latency tests is strongly altered by shift work and to a lesser extent by chronic sleep deprivation. The prevalence of narcolepsy without cataplexy may be 3-fold higher than that of narcolepsy-cataplexy.
Entities:
Keywords:
HLA; MSLT; REM sleep; cataplexy; narcolepsy; polysomnography
Authors: M M Mitler; J Van den Hoed; M A Carskadon; G Richardson; R Park; C Guilleminault; W C Dement Journal: Electroencephalogr Clin Neurophysiol Date: 1979-04
Authors: Aditya Ambati; Ryan Hillary; Smaranda Leu-Semenescu; Hanna M Ollila; Ling Lin; Emmanuel H During; Neal Farber; Thomas J Rico; Juliette Faraco; Eileen Leary; Andrea N Goldstein-Piekarski; Yu-Shu Huang; Fang Han; Yakov Sivan; Michel Lecendreux; Pauline Dodet; Makoto Honda; Natan Gadoth; Sona Nevsimalova; Fabio Pizza; Takashi Kanbayashi; Rosa Peraita-Adrados; Guy D Leschziner; Rosa Hasan; Francesca Canellas; Kazuhiko Kume; Makrina Daniilidou; Patrice Bourgin; David Rye; José L Vicario; Birgit Hogl; Seung Chul Hong; Guiseppe Plazzi; Geert Mayer; Anne Marie Landtblom; Yves Dauvilliers; Isabelle Arnulf; Emmanuel Jean-Marie Mignot Journal: Proc Natl Acad Sci U S A Date: 2021-03-23 Impact factor: 11.205
Authors: Christopher A Kosky; Anastasios Bonakis; Arthee Yogendran; Gihan Hettiarachchi; Paul I Dargan; Adrian J Williams Journal: J Clin Sleep Med Date: 2016-11-15 Impact factor: 4.062