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Study Objectives Sleep-related head jerks (SRHJ) are often considered as a physiological motor phenomenon, occurring mainly during rapid eye movement (REM) sleep. Their clinical relevance and links with other sleep parameters are unclear.... more
Study Objectives Sleep-related head jerks (SRHJ) are often considered as a physiological motor phenomenon, occurring mainly during rapid eye movement (REM) sleep. Their clinical relevance and links with other sleep parameters are unclear. We characterized the clinical and polysomnographic features of patients with excessive SRHJ and compare them with healthy controls and patients with isolated REM sleep behavior disorder (iRBD). Methods A total of 30 patients (19 males, 27.5 y.o., 16.0–51.0) with a REM-HJ index >30/h were identified over a period of 5 years. All had a video-polysomnographic (PSG) recording to characterize the SRHJ, to assess associations with other sleep parameters and to quantify phasic and tonic electromyographic activity during REM sleep, compared with 30 healthy controls and 30 patients with iRBD. Results Five among the 30 patients had a primary complaint of involuntary nighttime head movements associated with sleepiness or non-restorative sleep. The mean REM...
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Study Objectives Hypersomnolence, defined by excessive daytime sleepiness (EDS) or excessive quantity of sleep (EQS), has been associated with increased morbidity. The aim of this study was to determine the clinical and polysomnographic... more
Study Objectives Hypersomnolence, defined by excessive daytime sleepiness (EDS) or excessive quantity of sleep (EQS), has been associated with increased morbidity. The aim of this study was to determine the clinical and polysomnographic characteristics associated with EQS and EDS assessed objectively during extended polysomnography recording. Methods A total of 266 drug-free subjects (201 women; mean age: 26.5 years [16.08; 60.87]) underwent 32-h bed-rest polysomnography recording preceded by polysomnography and modified multiple sleep latency test (mMSLT). Participants were categorized according to their total sleep time (bed-rest TST ≥19 h, hypersomnia), objective EDS (mean sleep latency on MSLT ≤8 min), and self-reported EDS (Epworth sleepiness scale score >10) and EQS (≥9 h/24 h per week). Results Subjects with hypersomnia were often younger, with normal sleep architecture, high nighttime sleep efficiency, and severe objective EDS. No association with sex, body mass index, Ep...
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Research Interests: Medicine and Gynecology
Objectif Decrire les caracteristiques cliniques et polysomnographiques (PSG) d’enfants et d’adultes presentant un trouble des mouvements rythmiques du sommeil (TMR) en comparaison a des sujets sains. Methodes Cinquante patients avec TMR... more
Objectif Decrire les caracteristiques cliniques et polysomnographiques (PSG) d’enfants et d’adultes presentant un trouble des mouvements rythmiques du sommeil (TMR) en comparaison a des sujets sains. Methodes Cinquante patients avec TMR (23 enfants) ont beneficie d’une evaluation clinique detaillee et d’une PSG. Les caracteristiques PSG etaient comparees a celles de 75 sujets temoins apparies en âge et sexe (41 enfants). Resultats Les patients avec TMR rapportaient des plaintes de mauvais sommeil de nuit (82 %) et de fatigue/somnolence (80 %). Un total de 43,2 % avaient une histoire familiale de rythmies, 46 % des troubles neurodeveloppementaux, et 32 % un syndrome des jambes sans repos (SJSR). 82 % des patients ont presente au moins un episode de rythmies en vPSG (mediane 28, de 1 a 381 episodes), sans differences sur les caracteristiques des rythmies chez les enfants et les adultes. Comparativement aux temoins, les patients presentaient un index apnees-hypopnees (IAH), de mouvements periodiques (IMP) plus eleve, une augmentation de la latence d’endormissement, de la veille intrasommeil, des micro-eveils, et une plus faible efficacite de sommeil. Ces parametres etaient correles a diverses caracteristiques cliniques des rythmies apres ajustement pour l’âge, l’IAH, l’IMP et le SJSR. Conclusion Les TMR s’associent a des alterations du sommeil de nuit, en lien avec des troubles du sommeil associes d’une part, et avec la severite des rythmies, d’autre part. Une evaluation PSG de ce trouble potentiellement severe est requise pour une guider une prise en charge souvent multimodale.
Research Interests: Medicine and Gynecology
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Objective To investigate the relationship between sensory discomfort/motor component and cardiovascular autonomic response by continuous beat-to-beat blood pressure monitoring (CBPM) during the suggested immobilization test (SIT) in... more
Objective To investigate the relationship between sensory discomfort/motor component and cardiovascular autonomic response by continuous beat-to-beat blood pressure monitoring (CBPM) during the suggested immobilization test (SIT) in patients with restless legs syndrome (RLS). Methods Thirty-two drug-free patients with primary RLS (10 men; mean age 60.29 ± 10.81 years) and 17 healthy controls (2 men; mean age 58.82 ± 11.86 years) underwent a 1-hour SIT starting at 8 pm with concomitant CBPM to measure the heart rate (HR) and systolic/diastolic blood pressure (SBP, DBP). In all subjects, the presence of sensory discomfort and motor component during the SIT (S-SIT+ and M-SIT+, respectively) was quantified. Mixed regression models were used to compare the SBP, DBP, and HR profiles during the SIT by taking into account the repeated measures (6 time periods of 10 minutes). Results In patients with S-SIT+ (n = 17), SBP (p < 0.0001), DBP (p = 0.0007), and HR (p = 0.03) increased during t...
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Study Objectives(1) To compare the presence of autonomic symptoms using the validated SCOPA-AUT questionnaire in untreated patients with narcolepsy type 1 (NT1) to healthy controls, (2) to study the determinants of a high total SCOPA-AUT... more
Study Objectives(1) To compare the presence of autonomic symptoms using the validated SCOPA-AUT questionnaire in untreated patients with narcolepsy type 1 (NT1) to healthy controls, (2) to study the determinants of a high total SCOPA-AUT score in NT1, and (3) to evaluate the effect of drug intake on SCOPA-AUT results in NT1.MethodsThe SCOPA-AUT questionnaire that evaluates gastrointestinal, urinary, cardiovascular, thermoregulatory, pupillomotor, and sexual dysfunction was completed by 92 consecutive drug-free adult NT1 patients (59 men, 39.1 ± 15.6 years old) and 109 healthy controls (63 men, 42.6 ± 18.2 years old). A subgroup of 59 NT1 patients completed the questionnaire a second time, under medication (delay between two evaluations: 1.28 ± 1.14 years).ResultsCompared to controls, NT1 patients were more frequently obese, had more dyslipidemia, with no difference for age and gender. The SCOPA-AUT score of NT1 was higher than in controls in crude and adjusted models. Patients exper...
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ObjectiveTo validate the Idiopathic Hypersomnia Severity Scale (IIHSS), a self-report measure of hypersomnolence symptoms, consequences, and responsiveness to treatment.MethodsThe 14-item IHSS (developed and validated by sleep experts... more
ObjectiveTo validate the Idiopathic Hypersomnia Severity Scale (IIHSS), a self-report measure of hypersomnolence symptoms, consequences, and responsiveness to treatment.MethodsThe 14-item IHSS (developed and validated by sleep experts with patients' feedback) was filled in by 218 participants (2.3% missing data). Among the 210 participants who fully completed the IHSS, there were 57 untreated and 43 treated patients with idiopathic hypersomnia (IH) aged 16 years or older, 37 untreated patients with narcolepsy type 1 (NT1), and 73 controls without sleepiness. IHSS psychometric properties, discriminant diagnostic validity, and score changes with treatment were assessed.ResultsThe IHSS showed good internal consistency and content validity. Factor analysis indicated a 2-component solution with good reliability expressed by satisfactory Cronbach α values. IHSS scores were reproducible without changes in the test–retest evaluation (13 treated and 14 untreated patients). Convergent val...
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Objectif Comparer le profil d’evolution de la pression arterielle (PA) et de la frequence cardiaque (FC) des 24 h et la fonction endotheliale chez des patients atteints d’un syndrome des jambes sans repos idiopathique (SJSRi) compares a... more
Objectif Comparer le profil d’evolution de la pression arterielle (PA) et de la frequence cardiaque (FC) des 24 h et la fonction endotheliale chez des patients atteints d’un syndrome des jambes sans repos idiopathique (SJSRi) compares a des temoins. Methodes Quatre-vingt-quatre patients avec un SJSRi non traites (53 femmes, 55,1 ± 12,3 ans) et 76 temoins (47 femmes, 52,2 ± 15,3 ans) ont beneficie d’un entretien semi-structure, d’une polysomnographie et d’une mesure ambulatoire de la pression arterielle (MAPA), et 61 patients et 69 temoins d’une etude de la fonction endotheliale. Resultats La PA et la FC moyennes (24-heures, jour, nuit), la presence d’une HTA (mesuree par la MAPA) et la fonction endotheliale ne differaient pas entre groupes. Toutefois, les trajectoires de la PA systolique et diastolique sur les 24 heures etaient differentes entre patients et temoins. L’HTA etait retrouvee chez 11,9 % des patients par une mesure tensionnelle classique et chez 46,4 % sur la MAPA, avec deux fois plus d’hypertension nocturne que diurne. Les mouvements periodiques des jambes, les marqueurs de fragmentation du sommeil et le statut « non-dipper » de la PA systolique et moyenne etaient plus frequents chez les patients. Le profil non-dipper etait associe a un âge plus eleve, un âge d’apparition des symptomes plus tardif, des symptomes plus severes et une plus grande fragmentation du sommeil. Conclusion Les patients atteints d’un SJSRi presentent une dysregulation du profil tensionnel sur les 24 heures et une plus grande frequence du profil non-dipper de la PA systolique et moyenne, suggerant un sur-risque de morbi/mortalite cardiovasculaire.
Research Interests: Medicine and Gynecology
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The pathophysiology of rapid eye movement sleep behavior disorder (RBD) associated with narcolepsy type 1 (NT1) is still poorly understood, potentially distinct from idiopathic RBD (iRBD), but may share affected common pathways. We... more
The pathophysiology of rapid eye movement sleep behavior disorder (RBD) associated with narcolepsy type 1 (NT1) is still poorly understood, potentially distinct from idiopathic RBD (iRBD), but may share affected common pathways. We investigated whether MIBG cardiac uptake differs between iRBD and NT1 comorbid with RBD. Thirty-four patients with NT1-RBD and 15 patients with iRBD underwent MIBG cardiac scintigraphy. MIBG uptake was measured by calculating the early and delayed heart to mediastinum (H/M) ratios. A delayed H/M ratio lower than 1.46 was considered abnormal based on a population of 78 subjects without neurological or cardiac diseases. Patients with iRBD were older, had an older RBD onset age and higher REM sleep phasic and tonic muscular activities than NT1-RBD. Lower delayed and early H/M ratios were associated with iRBD, but not with NT1-RBD, in crude and adjusted associations. The delayed H/M ratio differed between iRBD and controls, after adjustment, but not between p...
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Idiopathic hypersomnia (IH) is a poorly characterized orphan central disorder of hypersomnolence responsible for excessive daytime sleepiness (EDS), prolonged nighttime sleep and sleep inertia that often require long-term symptomatic... more
Idiopathic hypersomnia (IH) is a poorly characterized orphan central disorder of hypersomnolence responsible for excessive daytime sleepiness (EDS), prolonged nighttime sleep and sleep inertia that often require long-term symptomatic stimulant medication. To date, no drug has currently the authorization for the treatment of IH patients worldwide. Areas covered: The authors reviewed data on pharmacological treatment of IH obtained from published literature (Medline/PubMed/Web of Science) and Clinicaltrial.gov database from 1997 to 2017. Most of data on treatment of IH derived from observational studies and case series with only three well-designed clinical trials available. Expert opinion: In two recent randomized, double-blind, placebo-controlled trials, modafinil improves EDS in IH. Most of other wakefulness-promoting agents labeled for narcolepsy have similar efficacy in cases series of IH patients. Pitolisant and sodium oxybate show promising results in two retrospective studies....
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To assess video-polysomnographic (vPSG) criteria and their cut-off values for the diagnosis of disorders of arousal (DOA; sleepwalking, sleep terror). One hundred and sixty adult patients with DOA and 50 sex- and age-matched healthy... more
To assess video-polysomnographic (vPSG) criteria and their cut-off values for the diagnosis of disorders of arousal (DOA; sleepwalking, sleep terror). One hundred and sixty adult patients with DOA and 50 sex- and age-matched healthy participants underwent a clinical evaluation and vPSG assessment to quantify slow wave sleep (SWS) interruptions (SWS fragmentation index; slow/mixed and fast arousal ratios and indexes per hour) and the associated behaviors. First, a case-control analysis was performed in 100 patients and the 50 controls to define the optimal cut-off values using receiver operating characteristic curves. Their sensitivity was then assessed in the other 60 patients with DOA. The SWS fragmentation index and the mixed, slow, slow/mixed arousal indexes and ratios were higher in patients with DOA than controls. The highest area under the curve (AUC) values were obtained for the SWS fragmentation and slow/mixed arousal indexes (AUC = 0.88 and 0.90, respectively). The SWS frag...
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To assess the diagnostic value of extended sleep duration on a controlled 32-hour bed-rest protocol in idiopathic hypersomnia (IH). 116 patients with high suspicion of IH (37 clear-cut-IH according to multiple sleep latency test criteria... more
To assess the diagnostic value of extended sleep duration on a controlled 32-hour bed-rest protocol in idiopathic hypersomnia (IH). 116 patients with high suspicion of IH (37 clear-cut-IH according to multiple sleep latency test criteria and 79 probable-IH), 32 with hypersomnolence associated with a comorbid disorder (non-IH) and 21 controls underwent a polysomnography, modified-sleep latency tests and a 32-h bed-rest protocol. Receiver operating characteristic curves were used to find optimal total sleep time (TST) cut-off values on various periods that discriminate patients to controls. TST was longer in patients with clear-cut-IH than other groups (probable-IH, non-IH and controls) and in patients with probable-IH than non-IH and controls. TST cut-off best discriminating clear-cut-IH and controls was 19h for the 32-h recording (sensitivity 91.9%, specificity 85.7%) and 12h (100%, 85.7%) for the first 24h. The 19-h cut-off displayed a specificity and sensitivity of 91.9% and 81.2%...
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Objectif L’implication d’un processus auto-immun dans la perte selective des neurones a orexine est fortement suspectee dans la narcolepsie de type 1 (NT1). Notre etude a pour but de rapporter les comorbidites personnelles et les... more
Objectif L’implication d’un processus auto-immun dans la perte selective des neurones a orexine est fortement suspectee dans la narcolepsie de type 1 (NT1). Notre etude a pour but de rapporter les comorbidites personnelles et les antecedents (ATCD) familiaux de maladies auto-immunes (MAI), inflammatoires (MI), et allergiques (AL) chez des patients NT1, en comparaison a des patients avec une narcolepsie de type 2 (NT2), une hypersomnie idiopathique (HI), et des temoins. Methodes Au total, 488 patients consecutifs (392 adultes – 206 NT1, 106 NT2, 80 HI – et 96 enfants – 82 NT1, 10 NT2, 4HI) de la cohorte Narcobank avec un diagnostic etabli selon les criteres ICSD-3, ont ete compares a 751 temoins (700 adultes et 51 enfants). Une evaluation structuree recueillait les caracteristiques sociodemographiques, cliniques, polysomnographiques (PSG) et les ATCD personnels et familiaux de MAI, MI et AL. Resultats Dix adultes (4,9 %) et 6 enfants NT1 (7,3 %) presentaient une MAI (i.e. alopecia aerata, diabete de type 1, lupus, thyroidite d’Hashimoto, sclerose en plaque) contre 3,4 % des adultes et aucun enfant chez les temoins, sans difference entre les groupes. Les caracteristiques cliniques et PSG n’etaient pas differentes entre les adultes NT1 avec ou sans MAI. Les adultes NT1 presentaient plus d’ATCD familiaux de MAI que les temoins (15,5 % vs 9 %, p Conclusion Cette etude objective une faible prevalence de MAI dans une population d’adultes et d’enfants NT1, sans difference avec les autres hypersomnies centrales, ni les temoins.
Research Interests: Medicine and Gynecology
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Study Objectives Sleep inertia is a frequent and disabling symptom in idiopathic hypersomnia (IH), but poorly defined and without objective measures. The study objective was to determine whether the psychomotor vigilance task (PVT) can... more
Study Objectives Sleep inertia is a frequent and disabling symptom in idiopathic hypersomnia (IH), but poorly defined and without objective measures. The study objective was to determine whether the psychomotor vigilance task (PVT) can reliably measure sleep inertia in patients with IH or other sleep disorders (non-IH). Methods A total of 62 (51 women, mean age: 27.7 ± 9.2) patients with IH and 140 (71 women, age: 33.3 ± 12.1) with non-IH (narcolepsy = 29, non-specified hypersomnolence [NSH] = 47, obstructive sleep apnea = 39, insomnia = 25) were included. Sleep inertia and sleep drunkenness in the last month (M-sleep inertia) and on PVT day (D-sleep inertia) were assessed with three items of the Idiopathic Hypersomnia Severity Scale (IHSS), in drug-free conditions. The PVT was performed four times (07:00 pm, 07:00 am, 07:30 am, and 11:00 am) and three metrics were used: lapses, mean 1/reaction time (RT), and slowest 10% 1/RT. Results Sleep inertia was more frequent in patients with...