Literature DB >> 27923434

Listening to the Patient Voice in Narcolepsy: Diagnostic Delay, Disease Burden, and Treatment Efficacy.

Kiran Maski1,2, Erin Steinhart2, David Williams1,2, Thomas Scammell1,3, Julie Flygare4, Kimberly McCleary5, Monica Gow6.   

Abstract

STUDY
OBJECTIVES: Describe common symptoms, comorbidities, functional limitations, and treatment responsiveness among patients with narcolepsy. Investigate the effect of pediatric onset of narcolepsy symptoms on time to diagnosis of narcolepsy and presence of comorbid depression.
METHODS: Cross-sectional survey of 1,699 people in the United States with self-reported diagnosis of narcolepsy. We utilized mixed-methods data analyses to report study findings.
RESULTS: Most participants reported receiving a diagnosis of narcolepsy more than 1 y after symptom onset. We found that the strongest predictor of this delayed diagnosis was pediatric onset of symptoms (odds ratio = 2.4, p < 0.0005). Depression was the most common comorbidity but we detected no association with pediatric onset of narcolepsy symptoms. Overall, participants reported that fatigue and cognitive difficulties were their most burdensome symptoms in addition to sleepiness and cataplexy. The majority of participants reported residual daytime fatigue and/or sleepiness despite treatment. Most participants reported they could not perform at work or school as well as they would like because of narcolepsy symptoms.
CONCLUSIONS: This study provides unique insight into the narcolepsy disease experience. The study quantifies the problem of diagnostic delay for narcolepsy patients in the United States and highlights that symptoms are more likely to be missed if they develop before 18 y of age. These results suggest that narcolepsy awareness efforts should be aimed at parents, pediatric health care providers, school professionals, and children/adolescents themselves. Disease burden is high because of problems with fatigue, cognition, and persistence of residual symptoms despite treatment.
© 2017 American Academy of Sleep Medicine

Entities:  

Keywords:  cataplexy; children; delayed diagnosis; narcolepsy; symptoms; treatment

Mesh:

Year:  2017        PMID: 27923434      PMCID: PMC5337589          DOI: 10.5664/jcsm.6494

Source DB:  PubMed          Journal:  J Clin Sleep Med        ISSN: 1550-9389            Impact factor:   4.062


  30 in total

1.  Early IVIg treatment has no effect on post-H1N1 narcolepsy phenotype or hypocretin deficiency.

Authors:  Stine Knudsen; Bo Biering-Sørensen; Birgitte R Kornum; Eva R Petersen; Jette D Ibsen; Steen Gammeltoft; Emmanuel Mignot; Poul J Jennum
Journal:  Neurology       Date:  2012-06-20       Impact factor: 9.910

Review 2.  Narcolepsy: clinical features, new pathophysiologic insights, and future perspectives.

Authors:  S Overeem; E Mignot; J G van Dijk; G J Lammers
Journal:  J Clin Neurophysiol       Date:  2001-03       Impact factor: 2.177

3.  Challenges in diagnosing narcolepsy without cataplexy: a consensus statement.

Authors:  Christian R Baumann; Emmanuel Mignot; Gert Jan Lammers; Sebastiaan Overeem; Isabelle Arnulf; David Rye; Yves Dauvilliers; Makoto Honda; Judith A Owens; Giuseppe Plazzi; Thomas E Scammell
Journal:  Sleep       Date:  2014-06-01       Impact factor: 5.849

4.  Antibodies to influenza nucleoprotein cross-react with human hypocretin receptor 2.

Authors:  Syed Sohail Ahmed; Wayne Volkmuth; José Duca; Lorenzo Corti; Michele Pallaoro; Alfredo Pezzicoli; Anette Karle; Fabio Rigat; Rino Rappuoli; Vas Narasimhan; Ilkka Julkunen; Arja Vuorela; Outi Vaarala; Hanna Nohynek; Franco Laghi Pasini; Emanuele Montomoli; Claudia Trombetta; Christopher M Adams; Jonathan Rothbard; Lawrence Steinman
Journal:  Sci Transl Med       Date:  2015-07-01       Impact factor: 17.956

5.  HLA DQB1*0602 is associated with cataplexy in 509 narcoleptic patients.

Authors:  E Mignot; R Hayduk; J Black; F C Grumet; C Guilleminault
Journal:  Sleep       Date:  1997-11       Impact factor: 5.849

Review 6.  The epidemiology of narcolepsy.

Authors:  W T Longstreth; Thomas D Koepsell; Thanh G Ton; Audrey F Hendrickson; Gerald van Belle
Journal:  Sleep       Date:  2007-01       Impact factor: 5.849

7.  Health, social, and economic consequences of narcolepsy: a controlled national study evaluating the societal effect on patients and their partners.

Authors:  Poul Jennum; Rikke Ibsen; Eva Rosa Petersen; Stine Knudsen; Jakob Kjellberg
Journal:  Sleep Med       Date:  2012-07-25       Impact factor: 3.492

Review 8.  Hyperactivity and sensation seeking as autoregulatory attempts to stabilize brain arousal in ADHD and mania?

Authors:  Julia Geissler; Marcel Romanos; Ulrich Hegerl; Tilman Hensch
Journal:  Atten Defic Hyperact Disord       Date:  2014-07-06

9.  Predictors of hypocretin (orexin) deficiency in narcolepsy without cataplexy.

Authors:  Olivier Andlauer; Hyatt Moore; Seung-Chul Hong; Yves Dauvilliers; Takashi Kanbayashi; Seiji Nishino; Fang Han; Michael H Silber; Tom Rico; Mali Einen; Birgitte R Kornum; Poul Jennum; Stine Knudsen; Sona Nevsimalova; Francesca Poli; Giuseppe Plazzi; Emmanuel Mignot
Journal:  Sleep       Date:  2012-09-01       Impact factor: 5.849

10.  Chronic alterations in monoaminergic cells in the locus coeruleus in orexin neuron-ablated narcoleptic mice.

Authors:  Natsuko Tsujino; Tomomi Tsunematsu; Motokazu Uchigashima; Kohtarou Konno; Akihiro Yamanaka; Kazuto Kobayashi; Masahiko Watanabe; Yoshimasa Koyama; Takeshi Sakurai
Journal:  PLoS One       Date:  2013-07-29       Impact factor: 3.240

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  23 in total

Review 1.  Neurobiological and immunogenetic aspects of narcolepsy: Implications for pharmacotherapy.

Authors:  Steven T Szabo; Michael J Thorpy; Geert Mayer; John H Peever; Thomas S Kilduff
Journal:  Sleep Med Rev       Date:  2018-11-08       Impact factor: 11.609

2.  Defining disrupted nighttime sleep and assessing its diagnostic utility for pediatric narcolepsy type 1.

Authors:  Kiran Maski; Fabio Pizza; Shanshan Liu; Erin Steinhart; Elaina Little; Alicia Colclasure; Cecilia Diniz Behn; Stefano Vandi; Elena Antelmi; Edie Weller; Thomas E Scammell; Giuseppe Plazzi
Journal:  Sleep       Date:  2020-10-13       Impact factor: 5.849

Review 3.  Immunotherapy in Narcolepsy.

Authors:  Maria Pia Giannoccaro; Giombattista Sallemi; Rocco Liguori; Giuseppe Plazzi; Fabio Pizza
Journal:  Curr Treat Options Neurol       Date:  2020-01-30       Impact factor: 3.598

4.  Nocturnal REM Sleep Without Atonia Is a Diagnostic Biomarker of Pediatric Narcolepsy.

Authors:  Saadoun Bin-Hasan; Aleksandar Videnovic; Kiran Maski
Journal:  J Clin Sleep Med       Date:  2018-02-15       Impact factor: 4.062

Review 5.  Disrupted nighttime sleep and sleep instability in narcolepsy.

Authors:  Kiran Maski; Emmanuel Mignot; Giuseppe Plazzi; Yves Dauvilliers
Journal:  J Clin Sleep Med       Date:  2022-01-01       Impact factor: 4.062

6.  Long-term safety and maintenance of efficacy of sodium oxybate in the treatment of narcolepsy with cataplexy in pediatric patients.

Authors:  Michel Lecendreux; Giuseppe Plazzi; Yves Dauvilliers; Carol L Rosen; Chad Ruoff; Jed Black; Rupa Parvataneni; Diane Guinta; Y Grace Wang; Emmanuel Mignot
Journal:  J Clin Sleep Med       Date:  2022-09-01       Impact factor: 4.324

7.  Treatment of central disorders of hypersomnolence: an American Academy of Sleep Medicine systematic review, meta-analysis, and GRADE assessment.

Authors:  Kiran Maski; Lynn Marie Trotti; Suresh Kotagal; R Robert Auger; Todd J Swick; James A Rowley; Sarah D Hashmi; Nathaniel F Watson
Journal:  J Clin Sleep Med       Date:  2021-09-01       Impact factor: 4.324

8.  Family functioning among adolescents with narcolepsy.

Authors:  Arpita Parmar; Eluen Ann Yeh; Daphne J Korczak; Shelly K Weiss; Zihang Lu; Allison Zweerink; Alene Toulany; Brian J Murray; Indra Narang
Journal:  Paediatr Child Health       Date:  2019-01-11       Impact factor: 2.253

Review 9.  Reviewing the Clinical Implications of Treating Narcolepsy as an Autoimmune Disorder.

Authors:  Maria Pia Giannoccaro; Rocco Liguori; Giuseppe Plazzi; Fabio Pizza
Journal:  Nat Sci Sleep       Date:  2021-05-11

10.  Stability of nocturnal wake and sleep stages defines central nervous system disorders of hypersomnolence.

Authors:  Kiran P Maski; Alicia Colclasure; Elaina Little; Erin Steinhart; Thomas E Scammell; William Navidi; Cecilia Diniz Behn
Journal:  Sleep       Date:  2021-07-09       Impact factor: 6.313

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