Little attention has been paid to the long-term development of idiopathic hypersomnia symptoms and idiopathic hypersomnia comorbidities. The aim of this study was to describe the general health of patients with idiopathic hypersomnia years after the initial diagnosis, focusing on current subjective hypersomnolence and the presence of its other possible causes. Adult patients diagnosed with idiopathic hypersomnia ≥ 3 years ago at sleep centres in Prague and Kosice were invited to participate in this study. A total of 60 patients were examined (age 47.3 ± SD = 13.2 years, 66.7% women). In all participants, their hypersomnolence could not be explained by any other cause but idiopathic hypersomnia at the time of diagnosis. The mean duration of follow-up was 9.8 + 8.0 years. Fifty patients (83%) reported persisting hypersomnolence, but only 33 (55%) had no other disease that could also explain the patient's excessive daytime sleepiness and/or prolonged sleep. In two patients (3%), the diagnosis in the meantime had changed to narcolepsy type 2, and 15 patients (25%) had developed a disease or diseases potentially causing hypersomnolence since the initial diagnosis. Complete hypersomnolence resolution without stimulant treatment lasting longer than 6 months was reported by 10 patients (17%). To conclude, in a longer interval from the diagnosis of idiopathic hypersomnia, hypersomnolence may disappear or may theoretically be explained by another newly developed disease, or the diagnosis may be changed to narcolepsy type 2. Thus, after 9.8 years, only 55% of the examined patients with idiopathic hypersomnia had a typical clinical picture of idiopathic hypersomnia without doubts about the cause of the current hypersomnolence.

Department of Neurology and Centre of Clinical Neuroscience 1st Faculty of Medicine Charles University and General University Hospital Prague Czech Republic

Department of Neurology Faculty of Medicine University of P J Šafárik and University Hospital of L Pasteur Košice Slovak Republic

National Institute of Mental Health Klecany and 3rd Faculty of Medicine Charles University Prague Czech Republic

St Elisabeth University Bratislava Slovak Republic

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Allen, R. P., Walters, A. S., Montplaisir, J., Hening, W., Myers, A., Bell, T. J., & Ferini-Strambi, L. (2005). Restless legs syndrome prevalence and impact: REST general population study. Archives of Internal Medicine, 165(11), 1286-1292. https://doi.org/10.1001/archinte.165.11.1286

American Academy of Sleep Medicine (2005). International classification of sleep disorders. 2nd ed.: Diagnostic and coding manual. American Academy of Sleep Medicine.

American Academy of Sleep Medicine (Ed.). (2014). International classification of sleep disorders (3rd ed.). American Academy of Sleep Medicine.

Anderson, K. N., Pilsworth, S., Sharples, L. D., Smith, I. E., & Shneerson, J. M. (2007). Idiopathic hypersomnia: A study of 77 cases. Sleep, 30(10), 1274-1281. https://doi.org/10.1093/sleep/30.10.1274

Arnulf, I., Leu-Semenescu, S., & Dodet, P. (2022). Precision medicine for idiopathic hypersomnia. Sleep Medicine Clinics, 17(3), 379-398. https://doi.org/10.1016/j.jsmc.2022.06.016

Arnulf, I., Thomas, R., Roy, A., & Dauvilliers, Y. (2023). Update on the treatment of idiopathic hypersomnia: Progress, challenges, and expert opinion. Sleep Medicine Reviews, 69, 101766. https://doi.org/10.1016/j.smrv.2023.101766

Bassetti, C., & Aldrich, M. S. (1997). Idiopathic hypersomnia. A series of 42 patients. Brain, 120(Pt 8), 1423-1435. https://doi.org/10.1093/brain/120.8.1423

Berger, K., Luedemann, J., Trenkwalder, C., John, U., & Kessler, C. (2004). Sex and the risk of restless legs syndrome in the general population. Archives of Internal Medicine, 164(2), 196-202. https://doi.org/10.1001/archinte.164.2.196

Billiard, M., Merle, C., Carlander, B., Ondze, B., Alvarez, D., & Besset, A. (1998). Idiopathic hypersomnia. Psychiatry and Clinical Neurosciences, 52(2), 125-129. https://doi.org/10.1111/j.1440-1819.1998.tb00987.x

Billiard, M., & Sonka, K. (2022). Idiopathic hypersomnia: Historical account, critical review of current tests and criteria, diagnostic evaluation in the absence of biological markers and robust electrophysiological diagnostic criteria. Nature and Science of Sleep, 14, 311-322. https://doi.org/10.2147/NSS.S266090

Bruck, D., & Parkes, J. D. (1996). A comparison of idiopathic hypersomnia and narcolepsy-cataplexy using self report measures and sleep diary data. Journal of Neurology, Neurosurgery, and Psychiatry, 60(5), 576-578. https://doi.org/10.1136/jnnp.60.5.576

Buskova, J., Novak, T., Miletinova, E., Kralova, R., Kost Alova, J., Klikova, M., & Veldova, K. (2022). Self-reported symptoms and objective measures in idiopathic hypersomnia and hypersomnia associated with psychiatric disorders: A prospective cross-sectional study. Journal of Clinical Sleep Medicine, 18(3), 713-720. https://doi.org/10.5664/jcsm.9702

Dauvilliers, Y., Bogan, R. K., Arnulf, I., Scammell, T. E., St Louis, E. K., & Thorpy, M. J. (2022). Clinical considerations for the diagnosis of idiopathic hypersomnia. Sleep Medicine Reviews, 66, 101709. https://doi.org/10.1016/j.smrv.2022.101709

Dostálová, S., Šusta, M., Nepožitek, J., Peřinová, P., Příhodová, I., Ibarburu Lorenzo y Losada, V., DallAntonia, I., Bezdicek, O., Nikolai, T., & Šonka, K. (2020). Polysomnographic findings in individuals over 50 years of age lacking subjective signs of sleep disturbance (In Czech). Ceska a Slovenska Neurologie a Neurochirurgie, 83, 57-63.

Evangelista, E., Lopez, R., & Dauvilliers, Y. (2018). Update on treatment for idiopathic hypersomnia. Expert Opinion on Investigational Drugs, 27(2), 187-192. https://doi.org/10.1080/13543784.2018.1417385

Fronczek, R., Arnulf, I., Baumann, C. R., Maski, K., Pizza, F., & Trotti, L. M. (2020). To split or to lump? Classifying the central disorders of hypersomnolence. Sleep, 43(8), zsaa044. https://doi.org/10.1093/sleep/zsaa044

Galuskova, K., & Sonka, K. (2021). Idiopathic hypersomnia and depression, the challenge for clinicians and researchers. Prague Medical Report, 122(3), 127-139. https://doi.org/10.14712/23362936.2021.13

Gaskin, C. J., & Happell, B. (2014). On exploratory factor analysis: A review of recent evidence, an assessment of current practice, and recommendations for future use. International Journal of Nursing Studies, 51(3), 511-521. https://doi.org/10.1016/j.ijnurstu.2013.10.005

Heinzer, R., Vat, S., Marques-Vidal, P., Marti-Soler, H., Andries, D., Tobback, N., Mooser, V., Preisig, M., Malhotra, A., Waeber, G., Vollenweider, P., & Haba-Rubio, J. (2015). Prevalence of sleep-disordered breathing in the general population: The HypnoLaus study. The Lancet Respiratory Medicine, 3(4), 310-318. https://doi.org/10.1016/S2213-2600(15)00043-0

Inoue, Y., Tabata, T., & Tsukimori, N. (2021). Efficacy and safety of modafinil in patients with idiopathic hypersomnia without long sleep time: A multicenter, randomized, double-blind, placebo-controlled, parallel-group comparison study. Sleep Medicine, 80, 315-321. https://doi.org/10.1016/j.sleep.2021.01.018

Javaheri, S., & Javaheri, S. (2020). Update on persistent excessive daytime sleepiness in OSA. Chest, 158(2), 776-786. https://doi.org/10.1016/j.chest.2020.02.036

Johns, M. W. (1991). A new method for measuring daytime sleepiness: The Epworth sleepiness scale. Sleep, 14(6), 540-545. https://doi.org/10.1093/sleep/14.6.540

Kagstrom, A., Alexova, A., Tuskova, E., Csajbok, Z., Schomerus, G., Formanek, T., Mladá, K., Winkler, P., & Cermakova, P. (2019). The treatment gap for mental disorders and associated factors in The Czech Republic. European Psychiatry, 59, 37-43. https://doi.org/10.1016/j.eurpsy.2019.04.003

Kim, T., Lee, J. H., Lee, C. S., & Yoon, I. Y. (2016). Different fates of excessive daytime sleepiness: Survival analysis for remission. Acta Neurologica Scandinavica, 134(1), 35-41. https://doi.org/10.1111/ane.12504

Lammers, G. J., Bassetti, C. L. A., Dolenc-Groselj, L., Jennum, P. J., Kallweit, U., Khatami, R., Lecendreux, M., Manconi, M., Mayer, G., Partinen, M., Plazzi, G., & Dauvilliers, Y. (2020). Diagnosis of central disorders of hypersomnolence: A reappraisal by European experts. Sleep Medicine Reviews, 52, 101306. https://doi.org/10.1016/j.smrv.2020.101306

Leu-Semenescu, S., Nittur, N., Golmard, J. L., & Arnulf, I. (2014). Effects of pitolisant, a histamine H3 inverse agonist, in drug-resistant idiopathic and symptomatic hypersomnia: A chart review. Sleep Medicine, 15(6), 681-687. https://doi.org/10.1016/j.sleep.2014.01.021

Lopez, R., Doukkali, A., Barateau, L., Evangelista, E., Chenini, S., Jaussent, I., & Dauvilliers, Y. (2017). Test-retest reliability of the multiple sleep latency test in central disorders of Hypersomnolence. Sleep, 40(12), zsx164. https://doi.org/10.1093/sleep/zsx164

Mayer, G., Benes, H., Young, P., Bitterlich, M., & Rodenbeck, A. (2015). Modafinil in the treatment of idiopathic hypersomnia without long sleep time-A randomized, double-blind, placebo-controlled study. Journal of Sleep Research, 24(1), 74-81. https://doi.org/10.1111/jsr.12201

Nevsimalova, S., Susta, M., Prihodova, I., Horvat, E. M., Milata, M., & Sonka, K. (2021). Idiopathic hypersomnia: A homogeneous or heterogeneous disease? Sleep Medicine, 80, 86-91. https://doi.org/10.1016/j.sleep.2021.01.031

Ohayon, M. M. (2008). From wakefulness to excessive sleepiness: What we know and still need to know. Sleep Medicine Reviews, 12(2), 129-141. https://doi.org/10.1016/j.smrv.2008.01.001

Penninx, B. W., Pine, D. S., Holmes, E. A., & Reif, A. (2021). Anxiety disorders. Lancet, 397(10277), 914-927. https://doi.org/10.1016/S0140-6736(21)00359-7

Roth, B. (1976). Narcolepsy and hypersomnia: Review and classification of 642 personally observed cases. Schweizer Archiv für Neurologie, Neurochirurgie Und Psychiatrie, 119(1), 31-41.

Ruoff, C., Pizza, F., Trotti, L. M., Sonka, K., Vandi, S., Cheung, J., Pinto, S., Einen, M., Simakajornboon, N., Han, F., Peppard, P., Nevsimalova, S., Plazzi, G., Rye, D., & Mignot, E. (2018). The MSLT is repeatable in narcolepsy type 1 but not narcolepsy type 2: A retrospective patient study. Journal of Clinical Sleep Medicine, 14(1), 65-74. https://doi.org/10.5664/jcsm.6882

Susta, M., Sonka, K., Bizik, G., Petranek, S., & Nevsimalova, S. (2022). Idiopathic hypersomnia-a dynamic simulation model. Frontiers in Neurology, 13, 902637. https://doi.org/10.3389/fneur.2022.902637

Trenkwalder, C., Allen, R., Hogl, B., Paulus, W., & Winkelmann, J. (2016). Restless legs syndrome associated with major diseases: A systematic review and new concept. Neurology, 86(14), 1336-1343. https://doi.org/10.1212/WNL.0000000000002542

Trotti, L. M. (2017a). Idiopathic hypersomnia. Sleep Medicine Clinics, 12(3), 331-344. https://doi.org/10.1016/j.jsmc.2017.03.009

Trotti, L. M. (2017b). Waking up is the hardest thing I do all day: Sleep inertia and sleep drunkenness. Sleep Medicine Reviews, 35, 76-84. https://doi.org/10.1016/j.smrv.2016.08.005

Trotti, L. M., Saini, P., Bliwise, D. L., Freeman, A. A., Jenkins, A., & Rye, D. B. (2015). Clarithromycin in gamma-aminobutyric acid-related hypersomnolence: A randomized, crossover trial. Annals of Neurology, 78(3), 454-465. https://doi.org/10.1002/ana.24459

Trotti, L. M., Staab, B. A., & Rye, D. B. (2013). Test-retest reliability of the multiple sleep latency test in narcolepsy without cataplexy and idiopathic hypersomnia. Journal of Clinical Sleep Medicine, 9(8), 789-795. https://doi.org/10.5664/jcsm.2922

Vernet, C., & Arnulf, I. (2009). Idiopathic hypersomnia with and without long sleep time: A controlled series of 75 patients. Sleep, 32(6), 753-759. https://doi.org/10.1093/sleep/32.6.753

Wang, J., Wu, X., Lai, W., Long, E., Zhang, X., Li, W., Zhu, Y., Chen, C., Zhong, X., Liu, Z., Wang, D., & Lin, H. (2017). Prevalence of depression and depressive symptoms among outpatients: A systematic review and meta-analysis. BMJ Open, 7(8), e017173. https://doi.org/10.1136/bmjopen-2017-017173

Winkelman, J. W. (2022). Treating severe refractory and augmented restless legs syndrome. Chest, 162(3), 693-700. https://doi.org/10.1016/j.chest.2022.05.014