A five-year longitudinal study reveals progressive cortical thinning in narcolepsy and faster cortical thinning in relation to early-onset.

Narcolepsy with cataplexy is characterized by excessive daytime sleepiness, cataplexy, and other REM sleep phenomena. Previous MRI studies were cross-sectional in design and could not adequately address if disease progression leads the brain structural abnormalities in narcolepsy. Our analysis in pa...

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Published in:Brain Imaging & Behavior Vol. 14; no. 1; pp. 200 - 213
Main Authors: Jeon, Seun, Cho, Jae Wook, Kim, Hosung, Evans, Alan C., Hong, Seung Bong, Joo, Eun Yeon
Format: Journal Article
Published: Springer Nature Feb2020
Online Access:View this record in EBSCOhost
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      pub: Springer Nature
      place: New York, New York
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        atl: A five-year longitudinal study reveals progressive cortical thinning in narcolepsy and faster cortical thinning in relation to early-onset.
      aug:
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          Jeon, Seun
          Cho, Jae Wook
          Kim, Hosung
          Evans, Alan C.
          Hong, Seung Bong
          Joo, Eun Yeon
        affil: McGill Centre for Integrative Neuroscience, Montreal Neurological Institute, McGill University, Montreal, Quebec, Canada
      sug:
        subj:
          Narcolepsy Physiopathology
          Narcolepsy
          Frontal Lobe Physiopathology
          Severity of Illness Indices
          Cerebral Cortex Physiopathology
          Female
          Prospective Studies
          Adult
          Age of Onset
          Magnetic Resonance Imaging
          Cross Sectional Studies
          Limbic System Physiopathology
          Male
          Brain Physiopathology
          Frontal Lobe
          Disease Progression
          Middle Age
          Clinical Assessment Tools
          Adult: 19-44 years
          Middle Aged: 45-64 years
          Female
          Male
      ab: Narcolepsy with cataplexy is characterized by excessive daytime sleepiness, cataplexy, and other REM sleep phenomena. Previous MRI studies were cross-sectional in design and could not adequately address if disease progression leads the brain structural abnormalities in narcolepsy. Our analysis in patients using longitudinally collected brain MRIs (n = 17; 2 scans per patient; scan interval: 4.7 ± 1.9 years) revealed widespread progressive cortical thinning in bilateral dorsolateral frontal and fusiform cortices, right anterior cingulate (corrected p < 0.05). Cross-sectional analyses showed faster progressive cortical thinning in patients than controls (n = 83, one scan per subject available), which we confirmed significant in the analysis of a small-set of longitudinal control data (n = 10). The pattern of progressive thinning in patients was overlapped well with those found in structural and functional studies of narcolepsy. We also found a faster progression of cortical thinning and worse disease severity (decreased sleep efficiency, increased sleep latency and arousal index) over time in a subgroup of patients with earlier disease onset (n = 9, onset age: 15.9 ± 2.5 years old) compared to later disease onset (n = 8, 25.3 ± 4.9). The faster progressive cortical thinning and worse disease severity over time in the patients with early-onset suggest compelling evidence of disease progression existing in this phenotype of narcolepsy syndrome. Our result based on a small dataset, however, demands a more careful investigation of the underlying mechanism.
      pubtype: Academic Journal
      doctype: Journal Article
      ougenre: Article
    language: English
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