Michel Lecendreux1,2, Sophie Lavault2,3, Régis Lopez2,4, Clara Odilia Inocente5, Eric Konofal1,2,3, Samuele Cortese6,7, Patricia Franco2,5,8, Isabelle Arnulf2,3, Yves Dauvilliers2,4. 1. AP-HP, Pediatric Sleep Center, CHU Robert-Debré, Paris, France. 2. National Reference Centre for Orphan Diseases, Narcolepsy, Idiopathic hypersomnia and Kleine-Levin Syndrome, France. 3. AP-HP, Groupe Hospitalier Pitié-Salpétrière, Service des Pathologies du Sommeil & Université Pierre et Marie Curie - Paris 6, Centre de Recherche de l'Institut du Cerveau et de la Moelle épinière, Paris, France. 4. Sleep Disorders Center, Department of Neurology, Gui-de-Chauliac Hospital, CHU Montpellier, Inserm U1061, Montpellier, France. 5. Integrative Physiology of Brain Arousal System, CRNL, University Lyon 1, Lyon, France. 6. Department of Psychology, Developmental Brain-Behaviour Laboratory, University of Southampton, Southampton, UK. 7. New York University Child Study Center, New York, NY. 8. Pediatric Sleep Unit, Hôpital Femme-Mère Enfant, University Lyon 1, Lyon, France.
Abstract
STUDY OBJECTIVES: To evaluate the frequency, severity, and associations of symptoms of attention-deficit/hyperactivity disorder (ADHD) in children with narcolepsy with and without cataplexy. DESIGN: Cross-sectional survey. SETTINGS: Four French national reference centers for narcolepsy. PATIENTS: One hundred eight consecutively referred children aged younger than 18 y with narcolepsy, with (NwC, n = 86) or without cataplexy (NwoC, n = 22), and 67 healthy controls. INTERVENTIONS: The participants, their families, and sleep specialists completed a structured interview and questionnaires about sleep, daytime sleepiness, fatigue, and ADHD symptoms (ADHD-rating scale based upon Diagnostic and Statistical Manual of Mental Disorders, Fourth Edition, Text Revision [DSM-IV-TR] symptoms), and use of psychostimulants for the treatment of narcolepsy (administered in 68.2%). Polysomnographic measures were collected. MEASUREMENTS AND RESULTS: Clinically significant levels of ADHD symptoms were found in 4.8% of controls compared with 35.3% in patients with NwoC (P < 0.001) and 19.7% in patients with NwC (P < 0.01). Total ADHD scores were 6.4 (95% confidence interval [CI]: 4.5, 9.0) in controls compared with 14.2 (95% CI: 10.6, 18.9; P < 0.001), in patients with NwoC and 12.2 (95% CI: 9.8, 15.3; P < 0.01) in patients with NwC; subscores of inattention and hyperactivity/impulsivity were also significantly higher in both narcolepsy groups compared with controls. No difference was found between the NwC and NwoC groups for any ADHD measure. ADHD symptom severity was associated with increased levels of sleepiness, fatigue, and insomnia. Compared with the 34 untreated patients, the 73 patients treated with psychostimulants (modafinil in 91%) showed a trend toward lower narcolepsy symptoms but not lower ADHD symptoms. CONCLUSIONS: Pediatric patients with narcolepsy have high levels of treatment-resistant attention-deficit/hyperactivity disorder (ADHD) symptoms. The optimal treatment for ADHD symptoms in these patients warrants further evaluation in longitudinal intervention studies.
STUDY OBJECTIVES: To evaluate the frequency, severity, and associations of symptoms of attention-deficit/hyperactivity disorder (ADHD) in children with narcolepsy with and without cataplexy. DESIGN: Cross-sectional survey. SETTINGS: Four French national reference centers for narcolepsy. PATIENTS: One hundred eight consecutively referred children aged younger than 18 y with narcolepsy, with (NwC, n = 86) or without cataplexy (NwoC, n = 22), and 67 healthy controls. INTERVENTIONS: The participants, their families, and sleep specialists completed a structured interview and questionnaires about sleep, daytime sleepiness, fatigue, and ADHD symptoms (ADHD-rating scale based upon Diagnostic and Statistical Manual of Mental Disorders, Fourth Edition, Text Revision [DSM-IV-TR] symptoms), and use of psychostimulants for the treatment of narcolepsy (administered in 68.2%). Polysomnographic measures were collected. MEASUREMENTS AND RESULTS: Clinically significant levels of ADHD symptoms were found in 4.8% of controls compared with 35.3% in patients with NwoC (P < 0.001) and 19.7% in patients with NwC (P < 0.01). Total ADHD scores were 6.4 (95% confidence interval [CI]: 4.5, 9.0) in controls compared with 14.2 (95% CI: 10.6, 18.9; P < 0.001), in patients with NwoC and 12.2 (95% CI: 9.8, 15.3; P < 0.01) in patients with NwC; subscores of inattention and hyperactivity/impulsivity were also significantly higher in both narcolepsy groups compared with controls. No difference was found between the NwC and NwoC groups for any ADHD measure. ADHD symptom severity was associated with increased levels of sleepiness, fatigue, and insomnia. Compared with the 34 untreated patients, the 73 patients treated with psychostimulants (modafinil in 91%) showed a trend toward lower narcolepsy symptoms but not lower ADHD symptoms. CONCLUSIONS: Pediatric patients with narcolepsy have high levels of treatment-resistant attention-deficit/hyperactivity disorder (ADHD) symptoms. The optimal treatment for ADHD symptoms in these patients warrants further evaluation in longitudinal intervention studies.
Authors: Christopher Drake; Chelsea Nickel; Eleni Burduvali; Thomas Roth; Catherine Jefferson; Badia Pietro Journal: Sleep Date: 2003-06-15 Impact factor: 5.849
Authors: Arthur S Walters; Rosalia Silvestri; Marco Zucconi; Ranju Chandrashekariah; Eric Konofal Journal: J Clin Sleep Med Date: 2008-12-15 Impact factor: 4.062