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• W3204152092 abstract "Free AccessScientific InvestigationsIdiopathic Hypersomnia Severity Scale to better quantify symptoms severity and their consequences in idiopathic hypersomnia Anna Laura Rassu, MD, Elisa Evangelista, MD, Lucie Barateau, MD, PhD, Sofiene Chenini, MD, Régis Lopez, MD, PhD, Isabelle Jaussent, PhD, Yves Dauvilliers, MD, PhD Anna Laura Rassu, MD CHU Montpellier, Hôpital Gui-de-Chauliac, Service de Neurologie, Unité du Sommeil, Centre National de Référence pour la Narcolepsie, Montpellier, France Institute Neurosciences of Montellier, Université de Montpellier, INSERM, Montpellier, France Search for more papers by this author , Elisa Evangelista, MD CHU Montpellier, Hôpital Gui-de-Chauliac, Service de Neurologie, Unité du Sommeil, Centre National de Référence pour la Narcolepsie, Montpellier, France Institute Neurosciences of Montellier, Université de Montpellier, INSERM, Montpellier, France Search for more papers by this author , Lucie Barateau, MD, PhD CHU Montpellier, Hôpital Gui-de-Chauliac, Service de Neurologie, Unité du Sommeil, Centre National de Référence pour la Narcolepsie, Montpellier, France Institute Neurosciences of Montellier, Université de Montpellier, INSERM, Montpellier, France Search for more papers by this author , Sofiene Chenini, MD CHU Montpellier, Hôpital Gui-de-Chauliac, Service de Neurologie, Unité du Sommeil, Centre National de Référence pour la Narcolepsie, Montpellier, France Institute Neurosciences of Montellier, Université de Montpellier, INSERM, Montpellier, France Search for more papers by this author , Régis Lopez, MD, PhD CHU Montpellier, Hôpital Gui-de-Chauliac, Service de Neurologie, Unité du Sommeil, Centre National de Référence pour la Narcolepsie, Montpellier, France Institute Neurosciences of Montellier, Université de Montpellier, INSERM, Montpellier, France Search for more papers by this author , Isabelle Jaussent, PhD Institute Neurosciences of Montellier, Université de Montpellier, INSERM, Montpellier, France Search for more papers by this author , Yves Dauvilliers, MD, PhD Address correspondence to: Yves Dauvilliers, MD, PhD, Service de Neurologie, Hôpital Gui-de-Chauliac, 80 avenue Augustin Fliche, 34295 Montpellier cedex 5, France; Tel: (33) 4 67 33 72 77; Fax: (33) 4 67 33 72 85; Email: E-mail Address: [email protected] CHU Montpellier, Hôpital Gui-de-Chauliac, Service de Neurologie, Unité du Sommeil, Centre National de Référence pour la Narcolepsie, Montpellier, France Institute Neurosciences of Montellier, Université de Montpellier, INSERM, Montpellier, France Search for more papers by this author Published Online:February 1, 2022https://doi.org/10.5664/jcsm.9682Cited by:5SectionsAbstractEpubPDF ShareShare onFacebookTwitterLinkedInRedditEmail ToolsAdd to favoritesDownload CitationsTrack Citations AboutABSTRACTStudy Objectives:To assess the responsiveness of the Idiopathic Hypersomnia Severity Scale (IHSS) to medications and estimate the minimum clinically important difference, to report clinically relevant score ranges, and to confirm its psychometric properties and whether items need to be weighted in drug-free and treated patients with idiopathic hypersomnia (IH).MethodsTwo-hundred twenty-six (166 drug-free and 60 treated) patients with IH (cross-sectional sample) completed the 14-item IHSS to quantify the severity of the 3 major IH symptoms (excessive daytime sleepiness, prolonged nighttime sleep, and sleep inertia) and consequences; 77 untreated patients were evaluated again after treatment (longitudinal sample). Patients filled in the Epworth Sleepiness Scale, Beck Depression Inventory II, and European Quality of Life questionnaires.Results:The IHSS confirmed adequate psychometric properties with a factor analysis indicating a 3-component solution. IHSS total score was lower in treated than untreated patients, with a mean difference of 4–5 points in the cross-sectional and longitudinal samples. Distribution-based methods were used to estimate that 4 points represented the minimum clinically important difference. Four severity levels were defined with between-group differences related to treatment. The probability of having severe sleepiness, depressive symptoms, and low quality of life increased with the severity level. Our results showed that IHSS item-weighting was not necessary.Conclusions:The IHSS is a valid and reliable tool to quantify IH symptoms, with 4 severity score levels of clinical importance. The IHSS has adequate psychometric properties and can detect symptom changes after treatment. These findings should stimulate its use in clinical settings and in research studies.Citation:Rassu AL, Evangelista E, Barateau L, et al. Idiopathic Hypersomnia Severity Scale to better quantify symptoms severity and their consequences in idiopathic hypersomnia. J Clin Sleep Med. 2022;18(2):617–629.BRIEF SUMMARYCurrent Knowledge/Study Rationale: The Idiopathic Hypersomnia Severity Scale is a valid and reliable tool to quantify symptoms of idiopathic hypersomnia and their consequences. This study aimed to assess the responsiveness of the Idiopathic Hypersomnia Severity Scale to medications and estimate the minimum clinically important difference between untreated and treated conditions, to report clinically relevant score levels, and to confirm the scale’s psychometric properties in adult patients with idiopathic hypersomnia.Study Impact: This scale has adequate psychometric properties and sensitivity to detect clinical changes in symptoms after treatment. We recommend its use in clinical settings for initial and follow-up evaluations, to monitor and optimize management of patients with idiopathic hypersomnia, and as a tool in future trials.INTRODUCTIONIdiopathic hypersomnia (IH) is a rare central hypersomnolence disorder characterized by 3 major symptoms: excessive daytime sleepiness (EDS), prolonged nighttime sleep, and sleep inertia.1,2 Based on the International Classification of Sleep Disorders, third edition (ICSD-3),3 criteria, EDS is required for IH diagnosis and is the most common and often the most debilitating symptom. However, most patients with IH also report prolonged nighttime sleep, long unrefreshing naps, sleep inertia (ie, great difficulties in awakening from sleep in the morning and after naps), impaired daytime alertness, cognitive difficulties, and brain fog.4–6 In patients with IH, health-related quality of life is reduced,7–10 social and work functioning are impaired, productivity is reduced, and car accidents are more frequent.11 No treatment is currently approved for IH; however, patients are often treated with off-label stimulants and wake-promoting agents approved for use in narcolepsy.12,13Several tools have been developed to evaluate EDS severity in the general population, including the widely used Epworth Sleepiness Scale14 (ESS), which is often chosen to assess EDS in narcolepsy, in obstructive sleep apnea, as well as in IH. In 2019, we developed the Idiopathic Hypersomnia Severity Scale (IHSS), a 14-item self-assessment questionnaire, to measure the severity, frequency, and functional impact of the 3 key IH symptoms. In 218 participants (including 100 patients with IH), IHSS showed good psychometric properties, particularly internal consistency and content validity, and some responsiveness to treatment in patients with IH.15 However, due to the potential global health consequences of IH, we still needed to: define clinically relevant IHSS score ranges that more precisely described the severity of IH symptoms and their impact on daily functioning; and compare the IHSS scores in IH patients with/without long sleep time, as well as with other clinical measures of EDS, with depressive symptoms, and with quality of life. We needed also to confirm that IHSS could detect clinical changes in symptoms following treatment and to estimate the minimum clinically important difference (MCID) in order to provide guidelines for what constitutes meaningful within-person change between the with and without treatment conditions. Also, we had assumed that all IHSS items would be equal: however, the number of IHSS items per symptom is different as well as the scoring per item. Therefore, we needed to determine whether the different IHSS items require weighting to better assess IH symptoms, as well as the effects of age, disease duration, and sex on the global score.The aims of the present study in consecutive untreated and treated adult patients with IH were: (1) to assess the responsiveness of IHSS to medications and estimate the MCID, (2) to report IHSS clinically relevant score levels, (3) to confirm its psychometric properties, and (4) to test whether items require weighting to improve IHSS performance.METHODSPatientsConsecutive patients aged 16 years and older with IH (n = 226; n = 166 drug-free and n = 60 treated) followed at the Reference National Center for Narcolepsy–Rare Hypersomnias of Montpellier, France, completed the IHSS, from January 2016 to September 2020 (cross-sectional sample). The sample included 100 patients from the first study (57 untreated, 43 treated) and 126 new patients (109 untreated, 17 treated). Among the 166 untreated patients (118 women, 71.08%; mean age: 30.29 ± 11.17 years), 77 (59 women, 76.62%; mean age 28.89 ± 9.19 years) filled in again the IHSS in the French language after treatment (longitudinal sample) following a median delay of 0.86 years [interquartile range = 0.47–1.35]. Among the 166 untreated patients, 8 (4.8%) had been receiving treatment by antidepressants that was stopped at least 1 month before the study inclusion.IH was diagnosed in drug-free condition according to the ICSD-3 criteria.3 All patients were evaluated by a sleep expert physician who collected also their demographic and clinical data. No participant had history of cataplexy. Patients with sleep-deprivation (< 7 hours of sleep per night), with moderate-to-severe sleep-disordered breathing, or with significant medical or neurologic comorbidity were excluded. Diagnosis was documented by nocturnal polysomnography (PSG) and Multiple Sleep Latency Test for 223 patients (166 in untreated condition and 57 in treated condition) and 32-hour polysomnography bed-rest was also performed in 172 patients (127 in untreated and 45 in treated condition). Overall, 144 patients (64.60%) had a Multiple Sleep Latency Test with a mean latency ≤ 8 minutes. Among the 172 patients who performed the 32-hour recording, 167 (98.8%) had a total sleep time (TST) ≥ 11 hours on the first 24-hour recording, and 147 patients (87.0%) had a TST ≥ 19 hours on the 32-hour recording (ie, alternative diagnostic criterion for IH).16 Cerebrospinal fluid hypocretin-1 levels were available for 55 patients: All had normal level > 200 pg/ml, except 1 patient with intermediate level (131 pg/ml).This study was approved by a French ethics committee (Comité de Protection des Personnes, France: “Constitution of a cohort and of a clinical, neurophysiological and biological bank of rare hypersomnolence disorders”–PHRC 07-138). Consent to participate was provided by all patients.MeasuresPatients were instructed to evaluate the severity of their symptoms during the previous month using the IHSS. The IHSS includes 2 items (1 and 2) on nighttime sleep duration and quality, 3 items (3, 4, and 5) on sleep inertia and sleep drunkenness after nighttime sleep and 1 (8) after daytime nap, and 3 items (6, 7, and 9) on diurnal symptoms (nap occurrence, daytime sleepiness). Items 10–14 assess daytime functioning alterations due to hypersomnolence. Six items are scored on a 3-point Likert scale and 8 items on a 4-point Likert scale; the Likert scale is a point scale that is used to allow the individual to express how much they agree or disagree with a particular statement. The total IHSS score is the sum of all item scores (range: 0–50), and higher scores indicate more severe symptoms. The definite presence of each of the 3 main symptoms (EDS, long nighttime sleep, and sleep inertia) is defined by a score > 1 to at least 1 of the items related to that symptom. All treated patients had stable drug dosages for at least 1 month before IHSS completion. The original IHSS was developed in French and validated in a French-speaking population; forward and back translations were performed to develop a certified English translation. The MAPI Research Institute (Lyon, France) hosts and distributes the scale and provides a central clearinghouse for all current and future copyrighted translations that may be used after appropriate permissions or licensure.EDS severity was assessed with the ESS (n = 224, score ≤ 10/24: no EDS, 11–15: EDS, ≥ 16: severe EDS). The severity of depressive symptoms was evaluated with the Beck Depression Inventory II (BDI-II) scale17 (n = 207, score ≤ 19: none or mild symptoms; ≥ 20/63: moderate-to-severe symptoms). The quality of life was evaluated in a subgroup (n = 196) using the European Quality of Life-5 Dimensions18 that includes a descriptive system of 5 dimensions (EQ-5D utility score) and a visual analog scale (EQ-5D-VAS) categorized into tertiles. The lowest tertile (score < 60) indicating poorer health quality of life was compared to the other 2 tertiles.Statistical analysisDemographic characteristics and clinical data were described using means and standard deviations for continuous variables, and numbers and percentages for categorical variables. For demographic, clinical, and polysomnography characteristics and IHSS item scores, the independent Student’s t test and analysis of variance were used to compare continuous variables, and the chi-square and Fisher exact tests to compare categorical variables between untreated and treated patients. The dependent t test was used to compare differences between continuous variables at 2 different time points or in 2 different conditions, and the McNemar test or McNemar-Bowker test of symmetry for paired categorical data. Associations between continuous variables were assessed with the Pearson correlation coefficient. To analyze the IHSS factor structure, a principal components factor analysis was performed using a Varimax rotation. The number of factors was determined on the basis of the obtained factor loadings and eigen values. Sampling adequacy was assessed by calculating the Kaiser-Meyer-Olkin index. The internal consistency (reliability) of the item scores was estimated using the Cronbach coefficient α. To compare the different IHSS items with a higher ESS score (ESS ≥ 16), all the IHSS items were included in a single logistic regression model. From this model, the weighted-item total score was calculated for each item with the respective β coefficients. To make the score approach an integer and be more intuitive, all β coefficients were standardized in such a way that the lowest one had a value of 1. As the lowest β value was −0.1209, it was multiplied by 8 and was rounded to the closest integer. The weighted total score for each patient was obtained by summing the scores for the appropriate level of each item. The same methodology was used for depressive symptoms ( BDI-II score ≥ 20) and poor health quality of life (EQ-5D-VAS score < 60). To evaluate the effectiveness of a treatment, the MCID was estimated using 2 distribution-based methods: Cohen’s effect size (0.5 × standard deviation [SD] delta) and the empirical rule effect size (0.08 × 6 × SD delta), where delta represents the IHSS total score change between untreated and treated patients. Statistical significance was set at P < .05. Statistical analyses were performed using SAS version 9.4 (SAS Institute, Cary, NC).RESULTSAmong the 60 treated patients with IH (46 women, 76.67%; mean age 37.51 ± 13.74 years) of the cross-sectional sample, 54 were taking 1 stimulant or wake-promoting agent (n = 31 modafinil, n = 19 methylphenidate, and n = 4 pitolisant), 6 were taking 2 drugs (n = 2 methylphenidate and pitolisant, n = 2 methylphenidate and modafinil, and n = 2 modafinil and pitolisant), and 6 (10%) were also taking antidepressants together with a wake-promoting agent. Compared with the 166 untreated patients, treated patients were older, had shorter TST on the polysomnography bed rest recording, self-reported better quality of life, and fewer had severe EDS and depressive symptoms (Table 1). Among the 77 patients of the longitudinal sample, 68 were taking 1 stimulant or wake-promoting agent (n = 30 modafinil, n = 24 methylphenidate, n = 14 pitolisant), 3 were taking 2 drugs (n = 1 modafinil and pitolisant, n = 1 methylphenidate and pitolisant, and n = 1 methylphenidate and venlafaxine), 6 were taking only sodium oxybate, and 5 were also taking antidepressants together with a wake-promoting agent. Compared with the untreated condition, the treated condition was associated with lower ESS scores and less severe depressive symptoms (Table 1).Table 1 —Demographic, clinical, and polysomnographic characteristics of untreated and treated adult patients with idiopathic hypersomnia.VariablesCross-sectional SampleLongitudinal SampleUntreated PatientsTreated PatientsPUntreated PatientsTreated PatientsP(n = 166), n (%)(n = 60), n (%)(n = 77), n (%)(n = 77), n (%)Demographic and clinical characteristics Sex, women118 (71.08)46 (76.67) .4159 (76.62)– Age,a y166; 30.29 (± 11.17)60; 37.51 (± 13.74)< .000177; 28.89 (± 9.19)77; 29.93 (± 9.28)< .0001 Body mass index,a kg/m2166; 23.54 (± 4.27)60; 24.14 (± 4.83) .5177; 23.32 (± 4.16)77; 23.57 (± 4.73) .61 Age at disease onset,a y165; 17.72 (± 7.14)60; 19.44 (± 10.36) .7477; 18.55 (± 7.26)–Subjective rating scales ESS total scorea164; 15.26 (± 4.41)60; 12.53 (± 5.04)< .000174; 15.77 (± 3.75)74; 12.85 (± 5.49)< .0001 ESS total score, ≥ 16143 (87.20)37 (61.67)< .000168 (91.89)52 (70.27) .0002 BDI-II total scorea155; 13.06 (± 9.02)52; 9.08 (± 7.94)< .000142; 14.83 (± 9.31)42; 11.50 (± 8.81) .01 BDI-II total score, ≥ 2036 (23.23)7 (13.46) .1314 (33.33)7 (16.67) .05 EQ-5D utility scorea145; 0.77 (± 0.20)50; 0.80 (± 0.18) .2039; 0.73 (± 0.19)39; 0.78 (± 0.19) .05 EQ-5D-VASa142; 61.96 (± 19.47)54; 68.26 (± 18.75) .0357; 59.58 (± 22.10)57; 62.86 (± 18.24) .14Neurophysiological data TST,a min163; 450.43 (± 39.03)49; 449.27 (± 42.51) .8677; 457.43 (± 38.30)77; 455.90 (± 39.54) .63 Sleep efficiency,a %163; 89.50 (± 5.75)49; 90.65 (± 5.55) .1577; 89.76 (± 5.91)77; 89.79 (± 5.89) .99 24-h bed rest TST ≥ 11 h124 (99.20)43 (97.73) .2466 (98.51)– 32-h bed rest TST ≥ 19 h114 (89.76)33 (73.33)< .000160 (89.55)– MSLT sleep latency,a,b min166; 7.71 (± 3.61)57; 7.35 (± 4.01) .3777; 7.93 (± 3.70)– MSLT sleep latency ≤ 8 mina,b107 (64.46)37 (64.91) .9546 (59.74)–aQuantitative variables are described with numbers, mean (± SD). bAssessed in untreated condition. BDI-II = Beck Depression Inventory II, EQ-5D = European Quality of Life-5 Dimensions questionnaire, ESS = Epworth Sleepiness Scale, MSLT = Multiple Sleep Latency Test, TST = total sleep time, VAS = visual analog scale.IHSS: construct validityIHSS construct validity was evaluated in the whole sample (n = 226: 166 untreated, and 60 treated). The internal consistency of the entire scale was good as indicated by the Cronbach’s α = 0.89. The correlation of each IHSS item with the total score was satisfactory (from 0.55 to 0.89, except for items 6, 7, and 14 that showed lower correlations: 0.25, 0.48, and 0.46, respectively) (Table 2).Table 2 —Factor structure of the Idiopathic Hypersomnia Severity Scale in patients with idiopathic hypersomnia.QuestionsKMO Item by ItemIn the Whole Sample (n = 226 patients)CommunalitiesFactorsIIIIII10.900.630.140.710.3420.900.660.310.750.0730.920.480.200.66–0.00340.920.580.390.650.0750.860.320.430.37–0.0260.710.760.18–0.100.8570.860.680.090.370.7380.900.520.240.68–0.0790.930.370.510.200.26100.880.750.750.370.21110.890.700.820.140.08120.910.580.720.260.06130.910.680.690.390.22140.710.410.53–0.340.08Cronbach’s α0.89––––KMO measure of sampling adequacy0.89––––Percentage of cumulative variance explained–0.58KMO = Kaiser-Meyer-Olkin index.The Kaiser-Meyer-Olkin index was 0.89, confirming the sampling adequacy. The factor analysis and the scree plot indicated a 3-factor solution with eigen values > 1 that explained 58% of the total variance (Table 2). Component I was composed of 7 items on daytime functioning (items 5, 9, 10, 11, 12, 13, 14), component II included 5 items on long sleep duration and sleep inertia (items 1, 2, 3, 4, 8), and component III included 2 items on napping (items 6 and 7). Of note, for the 13.3% of patients who did not drive, item 14 was scored 0 (no problem). The item loading values, which represent how strongly each item is associated with the underlying component, ranged from 0.43 to 0.85 (Table 2). Communalities, which refer to the percentage of variance for each item, ranged from 0.32 to 0.76.Concerning the construct convergent validity, IHSS total score correlated in untreated and treated patients with the ESS (r = .49, P < .0001; r = .62, P < .0001 respectively), BDI-II (r = .44, P < .0001; r = .60, P < .0001), and EQ-5D-VAS scores (r = –.44, P < .0001; r = –.53, P < .0001). The IHSS components I and II scores also correlated with the ESS, BDI-II, and EQ-5D-VAS scores in untreated and treated patients, whereas the component III score correlated with the BDI-II and EQ-5D-VAS scores only in the untreated population (data not shown).Number of symptoms and IHSS scores in treated and untreated patientsIn the cross-sectional sample, the number of symptoms (ie, EDS, long nighttime sleep, and sleep inertia) defined by their presence on at least 1 of the IHSS items related to that symptom was lower in treated than untreated patients (Table 3). Among the 166 untreated patients, 80.7% had all 3 symptoms, 12.7% 2 symptoms (mostly EDS with long nighttime sleep or sleep inertia), and 6.6% only 1 symptom (EDS). Among the 60 treated patients, 63.3% had all 3 symptoms, 20.0% 2 symptoms (n = 6 long nighttime sleep and sleep inertia, n = 4 EDS and long nighttime sleep, and n = 2 EDS and sleep inertia), and 16.7% only 1 symptom (mostly EDS). Similar distributions were found in the longitudinal sample (n = 77 patients), but the differences between untreated and treated condition were not significant (Table 3). In both cross-sectional and longitudinal samples, the number of patients with EDS was lower in the treated groups, although no difference was found concerning prolonged nighttime sleep and sleep inertia. In both samples, between-group differences (treated vs untreated) were found for 7 items in the cross-sectional sample and for 6 items for the longitudinal sample. Five of these items were the same in both groups (Table 3).Table 3 —Number of IH symptoms, IHSS items, and total scores in the cross-sectional and longitudinal samples of patients with IH.VariablesCross-sectional SampleLongitudinal SampleUntreated PatientsTreated PatientsPUntreated PatientsTreated PatientsP(n = 166), n (%)(n = 60), n (%)(n = 77), n (%)(n = 77), n (%)Number of IH symptoms .02 .12 0–111 (6.63)10 (16.67)6 (7.79)8 (10.39) 221 (12.65)12 (20.00)6 (7.79)13 (16.88) 3134 (80.72)38 (63.33)65 (84.42)56 (72.73)Number of IH symptomsa2.72 (± 0.66)2.43 (± 0.85)< .012.73 (± 0.72)2.57 (± 0.82) .10Diurnal symptoms (nap occurrence, sleepiness), yes159 (95.78)50 (83.33)< .0173 (94.81)65 (84.42) .01Nighttime sleep duration and quality, yes146 (87.95)49 (81.67) .2367 (87.01)67 (87.01) .99Sleep inertia and drunkenness, yes146 (87.95)47 (78.33) .0770 (90.91)66 (85.71) .25Item #1: Ideal duration of nighttime sleep, ≥ 9 hours127 (76.51)37 (61.67) .0363 (81.82)58 (75.32) .17Item #2: Feeling of not getting enough sleep in the morning, often/always140 (84.34)47 (78.33) .2964 (83.12)60 (77.92) .32Item #3: Need of several alarm calls to wake up in the morning, often/always94 (56.63)26 (43.33) .0847 (61.04)44 (57.14) .47Item #4: Time to feel fully functional after waking up in the morning, 1 hour59 (35.54)20 (33.33) .7631 (40.26)25 (32.47) .16Item #5: Doing/saying irrational things or clumsiness upon awakening, often/always41 (24.70)16 (26.67) .7621 (27.27)17 (22.08) .29Item #6: Naps during the day, often/very often89 (53.61)18 (30.00) .00244 (57.14)31 (40.26) .003Item #7: Ideal length of your naps, ≥ 1 hour107 (64.46)26 (43.33) .00548 (62.34)41 (53.25) .11Item #8: Feeling after a nap, sleepy/very sleepy118 (71.08)37 (61.67) .1859 (76.62)55 (71.43) .25Item #9: Struggle to stay awake during monotonous tasks, often/very often119 (71.69)26 (43.33) .000157 (74.03)37 (48.05) .0002Item #10: Impact of hypersomnolence on general health, significant/very significant impact130 (78.31)35 (58.33) .00365 (84.42)50 (64.94) .002Item #11: Impact of hypersomnolence on intellectual functioning, significant/very significant impact109 (65.66)28 (46.67) .0157 (74.03)44 (57.14) .003Item #12: Impact of hypersomnolence on mood, significant/very significant impact87 (52.41)26 (43.33) .2346 (59.74)42 (54.55) .71Item #13: Impact of hypersomnolence on daily tasks, significant/very significant impact85 (51.20)22 (36.67) .0545 (58.44)33 (42.86) .02Item #14: Impact of hypersomnolence on driving performance, significant/very significant impact64 (38.55)8 (13.33) .000640 (51.95)20 (25.97)< .0001IHSS total scorea30.95 (± 8.64)26.07 (± 10.54)< .00332.77 (± 8.17)28.38 (± 9.31)< .0001IHSS total score, ≥ 26130 (78.31)34 (56.67) .00164 (83.12)54 (70.13) .03IHSS component I (daytime functioning) scorea16.23 (± 5.20)13.53 (± 6.64) .00617.53 (± 4.77)14.73 (± 6.04)< .0001IHSS component II (long sleep duration/ sleep inertia) scorea10.27 (± 3.71)9.20 (± 4.02) .1010.75 (± 3.66)9.99 (± 3.30) .04IHSS component III (napping) scorea4.45 (± 2.00)3.33 (± 1.75)< .00014.48 (± 1.94)3.66 (± 1.91) .0002aQuantitative variables are expressed as mean (± SD). IH = idiopathic hypersomnia, IHSS = Idiopathic Hypersomnia Severity Scale.IHSS total score was higher in untreated patients than in treated patients with a mean difference of 4.88 points (SD = 9.18, P < .01) in the cross-sectional sample and of 4.38 points (SD = 7.41, P < .0001) in the longitudinal sample. The estimated MCID of the IHSS score between untreated and treated patients in the longitudinal sample was 3.71 with the Cohen’s effect size, and 3.56 with the empirical rule effect size. According to the Youden’s Index ([specificity + sensitivity] − 1), the IHSS cut-off value for discriminating between the 166 untreated and the 60 treated patients was 26 (area under the curve 62.8%, 95% confidence interval 54.2–71.2): 78.3% of untreated and 56.7% of treated patients had a score ≥ 26. Concerning the 3 IHSS dimensions, the components I and III scores were significantly lower in the treated groups of the cross-sectional and longitudinal samples, and the component II scores only in the treated group of the longitudinal sample (Table 3).Among untreated patients of the cross-sectional sample, the IHSS total score was higher in women than in men (32.1 ± 8.4 vs 28.1 ± 8.6, P = .006) and also the components II and III scores (P < .01). In the whole population, the IHSS total and component II scores were negatively correlated with age (r = –.20, P = .0027; r = –.34, P < .0001). IHSS total score was lower in patients with mean sleep latency > 8 minutes than in those with mean sleep latency below this cut-off (29.81 ± 8.25 vs 33.00 ± 9.02, P = .01), with similar results for the component I (P = .04) and component II scores (P = .05). No association was found between IHSS total score and TST longer or shorter than 11 hours during the first 24 hours, or longer or shorter than 19 hours during the 32-hour bed rest protocol. No correlation was found between IHSS total score, disease duration, and body mass index.Crude and weighted IHSS total scoresEach item and the crude IHSS scores were compared with several outcomes related to sleepiness, depressive symptoms, and quality of life. Overall, 104 (46.43%) patients had severe EDS (ESS score ≥ 16), 43 (20.77%) moderate/severe depressive symptoms (BDI-II score ≥ 20), and 62 (31.63%) poor health status (EQ-5D-VAS < 60). In univariate analysis, the scores of most IHSS items were higher in patients with severe EDS (except for items 1, 2, 3, 7, and 8). The scores of items 1, 5, 9, 10, 11, 12, and 13 and of items 4, 5, 10, 11, 12, and 13 also were higher in patients with moderate/severe depressive symptoms and poor health status, respectively. Multivariate analysis showed that items 9, 10, and 14 were independently associated with severe EDS, items 1, 5, and 12 with depressive symptoms, and items 5 and 13 with poor health status (Table 4).Table 4 —Multivariate logistic regression model for excessive daytime sleepiness, high level of depression, and low health status according to the IHSS items and risk scores derived from the β coefficients.VariableESS Score ≥ 16 vs < 16 BDI-II Score ≥ 20 vs < 20EQ-5D-VAS Score < 60 vs ≥ 60β Coef.OR [95% CI] PScoreβ Coef.OR [95% CI] PScoreβ Coef.OR [95% CI] PScoreItem #1: Ideal duration of nighttime sleep .11 .01 .22 < 9 hours010010010 ≥ 9 hours–0.83560.43 [0.16;1.20]–7–1.47000.23 [0.07;0.75]–56–0.64510.52 [0.19;1.47]–295Item #2: Feeling of not getting enough sleep in the morning .18 .11 .31 Never/sometimes010010010 Often/always0.91612.50 [0.66;9.54]81.33613.80 [0.74;19.7]510.75232.12 [0.50;9.00]344Item #3: Need of several alarm calls to wake up in the morning .31 .60 .22 Never/sometimes010010010 Often/always–0.38950.68 [0.32;1.44]–3–0.22820.80 [0.34;1.88]–9–0.49330.61 [0.28;1.35]–225Item #4: Time to feel fully functional after waking up in the morning .30 .48 .23 < 1 hour010010010 ≥ 1 hour0.40291.50 [0.70;3.21]3–0.32080.73 [0.30;1.77]–120.48041.62 [0.74;3.53]219Item #5: Doing/saying irrational things or clumsiness upon awakening .16 .04 .005 Never/sometimes010010010 Often/always0.61051.84 [0.79;4.28]50.90682.48 [1.02;5.99]351.16403.20 [1.43;7.16]532Item #6: Naps during the day .12 .91 .87 Never/rarely/sometimes010010010 Ofte" @default.
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• W3204152092 title "Idiopathic Hypersomnia Severity Scale to better quantify symptoms severity and their consequences in idiopathic hypersomnia" @default.
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