In-field assessment of sodium oxybate effect in pediatric type 1 narcolepsy: an actigraphic study.

Study Objectives: Sodium oxybate (SXB) is a GABAergic agent widely used as off-label treatment in pediatric type 1 narcolepsy (NT1). Here, we aimed at analyzing by wrist actigraphy the sleep/wake profile of NT1 children and adolescents in drug-naïve condition and after 1 year of SXB treatment. As secondary aim, we investigated changes on sleepiness, cataplexy, and children's anthropometric profile after 1 year of SXB treatment. Methods: Twenty-four drug-naïve NT1 children underwent 7 days of actigraphy during the school week. Information on sleepiness, narcolepsy symptoms, and anthropometric features were collected during the same week with questionnaires and semistructured clinical interview. Children started SXB treatment and underwent a second evaluation encompassing actigraphy, clinical interview, questionnaires, and anthropometric assessment after 1 year of stable treatment. Results: Actigraphy effectively documented an improvement of nocturnal sleep quality and duration coupled with a reduction of diurnal total sleep time, nap frequency, and duration at 1 year follow-up. Reduction of sleepiness, cataplexy frequency and severity, and weight loss, mainly in obese and overweight NT1 children, were also observed at the 1 year follow-up. Conclusions: Actigraphy objectively documented changes in nocturnal sleep quality and diurnal napping behavior after 1 year of SXB treatment, thus representing a valid approach to ecologically assess SXB treatment effect on NT1 children's sleep/wake profile. NT1 symptoms severity and children's anthropometric features also changed as expected. Actigraphy offers the possibility to longitudinally follow up children and has potential to become a key tool to tailor treatment in pediatric patients.

Actigraphic assessment of sleep/wake behavior in central disorders of hypersomnolence.

OBJECTIVE: To evaluate the reliability of actigraphy to distinguish the features of estimated daytime and nighttime sleep between patients with central disorders of hypersomnolence and healthy controls. METHODS: Thirty-nine drug-naïve patients with Narcolepsy Type 1, twenty-four drug-naïve patients with Idiopathic Hypersomnia, and thirty age- and sex- matched healthy controls underwent seven days of actigraphic and self-report monitoring of sleep/wake behavior. The following variables were examined: estimated time in bed (eTIB), estimated total sleep time, estimated sleep latency (eSOL), estimated sleep efficiency, estimated wake after sleep onset, number of estimated awakenings (eAwk), number of estimated awakenings longer than 5 minutes, estimated sleep motor activity (eSMA), number of estimated naps, mean duration of the longest estimated nap (eNapD), and daytime motor activity. RESULTS: All actigraphic parameters significantly differentiated the three groups, except eTIB and eSOL. A discriminant score computed combining actigraphic parameters from nighttime (eSMA, eAwk) and daytime (eNapD) periods showed a wide area under the curve (0.935) and a good balance between positive (95%) and negative predictive (87%) values in Narcolepsy Type 1 cases. CONCLUSION: Actigraphy provided a reliable objective measurement of sleep quality and daytime napping behavior able to distinguish central disorders of hypersomnolence and in particular Narcolepsy Type 1. The nycthemeral profile, combined with a careful clinical evaluation, may be an ecological information, useful to track disease course.