Impairment in Functioning and Quality of Life in Patients with Idiopathic Hypersomnia: The Real World Idiopathic Hypersomnia Outcomes Study (ARISE)

Abstract

Purpose

Idiopathic hypersomnia is a debilitating neurologic sleep disorder characterized by excessive daytime sleepiness, sleep inertia, and prolonged sleep. Its impact on patients’ quality of life and daily functioning has not been fully elucidated. The Real World Idiopathic Hypersomnia Outcomes Study (ARISE) evaluated the daily functioning, relationships, cognition, emotional well-being, and productivity/employment of participants with idiopathic hypersomnia.

Patients and Methods

ARISE was a US-based virtual cross-sectional survey comprising multiple patient-reported outcome measures (Functional Outcomes of Sleep Questionnaire, short version [FOSQ-10], Quality of Life in Neurological Disorders [Neuro-QoL] Social Roles and Stigma domains, British Columbia Cognitive Complaints Inventory [BC-CCI], Patient Health Questionnaire [PHQ-9], and the Work Productivity and Activity Impairment Questionnaire: Specific Health Problem [WPAI:SHP]). Participants were adults 21–65 years of age with idiopathic hypersomnia. Data were analyzed for all participants and for subgroups with/without long sleep time (LST; self-reported sleep ≥11 hours in 24 hours).

Results

Of 75 participants enrolled, most were female (81.3%) and the mean (SD) age was 34.1 (10.7) years. Participants’ scores on the FOSQ-10 (mean [SD] score: 10.7 [2.8]) and the Neuro-QoL Social Roles (43.4 [4.2]) and Stigma (57.3 [5.9]) domains reflected impairments in daily functioning and quality of life. More than half of participants reported moderate to severe cognitive complaints (BC-CCI; 62.7%) and moderate to severe depressive symptoms (PHQ-9; 66.7%). Scores on the WPAI:SHP showed substantial impairments in absenteeism, presenteeism, overall work productivity, and overall regular daily activity (mean percent [SD]: 12.3 [23.6], 47.6 [22.7], 51.4 [24.7], and 64.0 [21.9], respectively). These considerable impairments were found in participants with and without LST.

Conclusion

ARISE participants with idiopathic hypersomnia demonstrated poor quality of life and impaired functioning across multiple symptom domains.

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Keywords:

• relationships
• social
• work productivity
• cognitive function
• hypersomnolence
• long sleep time

Abbreviations

AASM, American Academy of Sleep Medicine; ARISE, Real World Idiopathic Hypersomnia Outcomes Study; BC-CCI, British Columbia Cognitive Complaints Inventory; FOSQ-10, Functional Outcomes of Sleep Questionnaire, short version; ICSD-2, International Classification of Sleep Disorders, 2nd Edition; ICSD-3, International Classification of Sleep Disorders, 3rd edition; LST, long sleep time; Neuro-QoL, Quality of Life in Neurological Disorders; PHQ-9, Patient Health Questionnaire-9; QoL, quality of life; SF-36, Short Form-36; WAFV, Perception and Attention Functions test battery—Vigilance; WPAI:SHP, Work Productivity and Activity Impairment Questionnaire: Specific Health Problem.

Data Sharing Statement

All relevant data are provided within the article and its Supplemental Material. Jazz has established a process to review requests from qualified external researchers for data from Jazz-sponsored clinical trials in a responsible manner that includes protecting patient privacy, assurance of data security and integrity, and furthering scientific and medical innovation. Additional details on Jazz Pharmaceuticals data sharing criteria and process for requesting access can be found at: https://www.jazzpharma.com/science/clinical-trial-data-sharing/.

Acknowledgments

The authors thank the study participants. This study was supported by Jazz Pharmaceuticals. Under the direction of the authors, Emily C. Bruggeman, PhD of Peloton Advantage, LLC, an OPEN Health company, provided medical writing and editorial support for this manuscript, which was funded by Jazz Pharmaceuticals.

Some of the findings from this study have been presented in posters at Psych Congress (2021) and the 36th Annual Meeting of the Associated Professional Sleep Societies (2022), and in an oral presentation at the 74th Annual Meeting of the American Academy of Neurology (2022). An abstract was also published in Neurology. 2022;98(18 Supplement).

Author Contributions

All authors made a significant contribution to the work reported, whether in conception, study design, execution, acquisition of data, analysis and interpretation, or all these areas; took part in drafting, revising, or critically reviewing the article; gave final approval of the version to be published; agreed on the journal to which the article has been submitted; and agreed to be accountable for all aspects of the work.

J Stevens: Conceptualization of the study (equal); Methodology of the study (equal); Project administration of the study (lead); Supervision of the study (lead); Writing of the article – review and editing (equal).

LD Schneider: Conceptualization of the study (equal); Methodology of the study (equal); Writing of the article – review and editing (equal).

AM Husain: Conceptualization of the study (equal); Methodology of the study (equal); Writing of the article – review and editing (equal).

D Ito: Conceptualization of the study (equal); Methodology of the study (equal); Writing of the article – review and editing (equal).

DS Fuller: Data curation of the study (lead); Formal analysis of the study results (lead); Methodology of the study (equal); Visualization of the study results (equal); Writing – review and editing (equal).

PC Zee: Writing of the article – review and editing (equal).

W Macfadden: Conceptualization of the study (supporting); Methodology of the study (equal); Supervision of the study (supporting); Writing of the article – review and editing (equal).

Disclosures

J Stevens is a full-time employee of Jazz Pharmaceuticals who, in the course of this employment, has received stock options exercisable for, and other stock awards of, ordinary shares of Jazz Pharmaceuticals, plc. LD Schneider is an employee of Alphabet, Inc. and is a compensated member of advisory boards and speakers bureaus for Jazz Pharmaceuticals, Eisai, and Harmony Biosciences. AM Husain has received consultancy fees and/or research funding from Jazz Pharmaceuticals, UCB, BlackThorn, Merck, Pipeline, Sage, Eisai, Marinus, and Neurelis, as well as royalties from Springer, Demos Medical, and Wolters Kluwer, and holds an editorship role with Wolters Kluwer. D Ito is an employee of Stratevi, a consulting firm that received research funding from Jazz Pharmaceuticals to conduct this study. DS Fuller is a full-time employee of Jazz Pharmaceuticals who, in the course of this employment, has received stock options exercisable for, and other stock awards of, ordinary shares of Jazz Pharmaceuticals, plc. PC Zee serves on scientific advisory boards for Jazz, Eisai, Idorsia, and Harmony Biosciences. She is also a consultant for CVS Caremark, Septerna and reports institutional grant to Northwestern University from Sleep Number. She owns stock from Teva. W Macfadden is a full-time employee of Jazz Pharmaceuticals who, in the course of this employment, has received stock options exercisable for, and other stock awards of, ordinary shares of Jazz Pharmaceuticals, plc. The authors report no other conflicts of interest in this work.