Evidence map›Paper›PMID 26224419›Full record

ArticleAnnals of neurology2015

Sleep deficits but no metabolic deficits in premanifest Huntington's disease.

Alpar S Lazar, Francesca Panin, Anna O G Goodman, Stanley E Lazic, Zsolt I Lazar, Sarah L Mason, Lorraine Rogers, Peter R Murgatroyd, Laura P E Watson, Priya Singh and 3 more

Open access · hybridAbstract read
In one paragraph

Article in Annals of neurology, 2015. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 55 papers, 2 of them syntheses that pooled it.

0numbers the graph read from it
0cells of the map it votes in
55citing papers in PubMed, 2 pooled it
5.5field-weighted citation impact, top 3% of its field
1 · What the graph read from it

What it found

Each row is one number read from the abstract, on the scale the paper reported it, with its interval. Left of the dashed line favours the treatment, right favours the comparator. Under each row is the sentence it came from. New to these charts? A ten-minute tutorial.

The abstract states no effect estimate the extractor could read, or names no intervention and outcome on the map, so this paper lights no cell and moves no belief. It is still indexed, cited and linked below.

2 · The registry

The trial behind it

Trials whose registry record cites this paper, or whose number appears in the abstract. A trial that started after this paper was published is citing it as background, not reporting it.

Neither the registry nor the abstract names a trial number. If this is a trial report, that itself is worth knowing.

3 · Its place in the literature

Who cites it

55 citing papers in PubMed, 2 syntheses or guidelines pooled it, 118 citations in OpenAlex.

  1. Pooled it
  2. Pooled it
  3. Review
  4. Article
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  6. Article
  7. Review
  8. Assessment of Fitbit Charge 4 for sleep stage and heart rate monitoring against polysomnography and during home monitoring in Huntington's disease.Journal of clinical sleep medicine : JCSM : official publication of the American Academy of Sleep Medicine · 2024
    Article
  9. Article
  10. The multidimensional sleep health of individuals with multiple sclerosis and Huntington's disease and healthy controls.Journal of clinical sleep medicine : JCSM : official publication of the American Academy of Sleep Medicine · 2024
    Article
  11. Article
  12. Review
  13. Article
  14. Article
  15. Review
  16. Review
  17. Sleep Disorders in Patients with Choreic Syndromes.Current neurology and neuroscience reports · 2023
    Review
  18. Review
  19. Article
  20. Review
4 · The record

Corrections and comments

PubMed lists nothing against this paper. Absence here is not a guarantee, only a check that was made.

5 · Who and what money

Authors and funding

13 authors at 9 institutions in 4 countries.

Alpar S LazarJohn van Geest Centre for Brain Repair, Department of Clinical Neurosciences, University of Cambridge, Cambridge, United Kingdom.
Francesca PaninJohn van Geest Centre for Brain Repair, Department of Clinical Neurosciences, University of Cambridge, Cambridge, United Kingdom.
Anna O G GoodmanJohn van Geest Centre for Brain Repair, Department of Clinical Neurosciences, University of Cambridge, Cambridge, United Kingdom.
Stanley E LazicIn Silico Lead Discovery, Novartis Institutes for Biomedical Research, Basel, Switzerland.
Zsolt I LazarDepartment of Physics, Babes-Bolyai University, Cluj-Napoca, Romania.
Sarah L MasonJohn van Geest Centre for Brain Repair, Department of Clinical Neurosciences, University of Cambridge, Cambridge, United Kingdom.
Lorraine RogersRespiratory Support and Sleep Centre, Papworth Hospital, Cambridge, United Kingdom.
Peter R MurgatroydNational Institute for Health Research/Wellcome Trust Clinical Research Facility, Addenbrooke's Hospital, Cambridge, United Kingdom.
Laura P E WatsonNational Institute for Health Research/Wellcome Trust Clinical Research Facility, Addenbrooke's Hospital, Cambridge, United Kingdom.
Priya SinghMedical Research Council Human Nutrition Research, Elsie Widdowson Laboratory, Cambridge, United Kingdom.
Beth BorowskyCHDI Management/CHDI Foundation, Princeton, NJ, United States of America.
John M ShneersonRespiratory Support and Sleep Centre, Papworth Hospital, Cambridge, United Kingdom.
Roger A BarkerJohn van Geest Centre for Brain Repair, Department of Clinical Neurosciences, University of Cambridge, Cambridge, United Kingdom.
University of Cambridge · GBPapworth Hospital · GBAddenbrooke's Hospital · GBAnglia Ruskin University · GBBabeș-Bolyai University · ROCHDI Foundation · USMedical Research Council · GBNIHR Wellcome Trust Southampton Clinical Research Facility · GBNovartis (Switzerland) · CH

Funding

Medical Research Council MC_U105960384Medical Research Council MC_UP_A090_1005Wellcome Trust
6 · The paper itself

Abstract

objectiveHuntington disease (HD) is a fatal autosomal dominant, neurodegenerative condition characterized by progressively worsening motor and nonmotor problems including cognitive and neuropsychiatric disturbances, along with sleep abnormalities and weight loss. However, it is not known whether sleep disturbances and metabolic abnormalities underlying the weight loss are present at a premanifest stage.

methodsWe performed a comprehensive sleep and metabolic study in 38 premanifest gene carrier individuals and 36 age- and sex-matched controls. The study consisted of 2 weeks of actigraphy at home, 2 nights of polysomnography and multiple sleep latency tests in the laboratory, and body composition assessment using dual energy x-ray absorptiometry scanning with energy expenditure measured over 10 days at home by doubly labeled water and for 36 hours in the laboratory by indirect calorimetry along with detailed cognitive and clinical assessments. We performed a principal component analyses across all measures within each studied domain.

resultsCompared to controls, premanifest gene carriers had more disrupted sleep, which was best characterized by a fragmented sleep profile. These abnormalities, as well as a theta power (4-7Hz) decrease in rapid eye movement sleep, were associated with disease burden score. Objectively measured sleep problems coincided with the development of cognitive, affective, and subtle motor deficits and were not associated with any metabolic alterations.

interpretationThe results show that among the earliest abnormalities in premanifest HD is sleep disturbances. This raises questions as to where the pathology in HD begins and also whether it could drive some of the early features and even possibly the pathology.

Indexed as

Asymptomatic DiseasesAdultFemaleHumansHuntington DiseaseMaleMiddle AgedSleep Wake Disorders

Identifiers

PMID26224419
PMCPMC4832311
OpenAlexW2138162732

What OpenQuestion holds

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LicenceCC BY
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Registered trials

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Read under generation 80e0d062 · epoch 390. Bibliography from PubMed, PubMed Central and OpenAlex; grants from NIH RePORTER; trial links from ClinicalTrials.gov; estimates, votes and beliefs from the OpenQuestion graph.