Evidence map›Paper›PMID 28947125›Full record

ReviewHandbook of clinical neurology2017

Statistical modeling of Huntington disease onset.

Tanya P Garcia, Karen Marder, Yuanjia Wang

Abstract readReview
In one paragraph

Review in Handbook of clinical neurology, 2017. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 4 papers.

0numbers the graph read from it
0cells of the map it votes in
4citing papers in PubMed
–field-weighted citation impact
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

4 citing papers in PubMed.

  1. Article
  2. Article
  3. Article
  4. Article
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

3 authors.

Tanya P GarciaDepartment of Epidemiology and Biostatistics, Texas A&M Health Science Center, College Station, TX, United States. Electronic address: tpgarcia@sph.tamhsc.edu.
Karen MarderDepartments of Neurology and Psychiatry, Sergievsky Center and Taub Institute, Columbia University Medical Center, New York, NY, United States.
Yuanjia WangDepartment of Biostatistics, Mailman School of Public Health, Columbia University, New York, NY, United States.

Funding

CTSA INFRASTRUCTURE FOR PEDIATRIC RESEARCHUL1RR024156 · NCRR · COLUMBIA UNIVERSITY HEALTH SCIENCES · PI GINSBERG, HENRY N · 2006 to 2011
$53.0M
NEUROBIOLOGICAL PREDICTORS OF HUNTINGTON'S DISEASER01NS040068 · NINDS · UNIVERSITY OF IOWA · PI PAULSEN, JANE S · 2001 to 2013
$48.5M
Statistical Methods for Integrating Mixed-type Biomarkers and Phenotypes in Neurodegenerative Disease ModelingR01NS073671 · NINDS · COLUMBIA UNIVERSITY HEALTH SCIENCES · PI WANG, YUANJIA · 2011 to 2025
$3.9M
Identifying Huntington's disease markers by modern statistical learning methods.U01NS082062 · NINDS · COLUMBIA UNIVERSITY HEALTH SCIENCES · PI WANG, YUANJIA · 2014 to 2016
$1.1M
Innovative Statistical Models for Development of First Huntington's Disease Progression Risk Assessment ToolK01NS099343 · NINDS · UNIV OF NORTH CAROLINA CHAPEL HILL · PI GARCIA, TANYA PAMELA · 2017 to 2019
$739k
NCRR NIH HHS UL1 RR024156NINDS NIH HHS K01 NS099343NINDS NIH HHS R01 NS040068NINDS NIH HHS R01 NS073671NINDS NIH HHS U01 NS082062
6 · The paper itself

Abstract

Huntington disease (HD) is caused by a CAG trinucleotide expansion in the huntingtin gene. We now have the power to predict age-at-onset from subject-specific features like motor and neuroimaging measures. In clinical trials, properly modeling onset age is important, because it improves power calculations and directs clinicians to recruit subjects with certain features. The history of modeling onset, from simple linear and logistic regression to advanced survival models, is discussed. We highlight their advantages and disadvantages, emphasizing the methodological challenges when genetic mutation status is unavailable. We also discuss the potential bias and higher variability incurred from the uncertainty associated with subjective definitions for onset. Methods to adjust for the uncertainty in survival models are still in their infancy, but would be beneficial for HD and neurodegenerative diseases with long prodromal periods like Alzheimer's and Parkinson's disease.

Indexed as

Age of OnsetHumansHuntingtin ProteinHuntington DiseaseModels, NeurologicalMutationTrinucleotide RepeatsHuntingtin ProteinAge-at-onsetDiagnosis definitionsKin-cohort studyMeasurement errorPenetrancePrediction

Identifiers

PMID28947125
PMCPMC8934048

What OpenQuestion holds

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Registered trials

None linked

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.