Evidence map›Paper›PMID 32436106›Full record

SynthesisJournal of neurology2021

When does postural instability appear in monogenic parkinsonisms? An individual-patient meta-analysis.

Luca Marsili, Joaquin A Vizcarra, Andrea Sturchio, Alok K Dwivedi, Elizabeth G Keeling, Dhiren Patel, Murli Mishra, Ashar Farooqi, Aristide Merola, Alfonso Fasano and 3 more

Abstract readMeta-AnalysisSystematic Review
PubMed Publisher
In one paragraph

Synthesis in Journal of neurology, 2021. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 12 papers, 1 of them a synthesis that pooled it.

0numbers the graph read from it
0cells of the map it votes in
12citing papers in PubMed, 1 pooled it
1.3field-weighted citation impact, top 15% 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

12 citing papers in PubMed, 1 synthesis or guideline pooled it, 16 citations in OpenAlex.

  1. Pooled it
  2. Pregnancy, fertile life factors, and associated clinical course in PRKN early-onset Parkinson's disease.Neurological sciences : official journal of the Italian Neurological Society and of the Italian Society of Clinical Neurophysiology · 2024
    Article
  3. Article
  4. Article
  5. Review
  6. Article
  7. Review
  8. Device-aided therapies for advanced Parkinson disease: insights from an international survey.Neurological sciences : official journal of the Italian Neurological Society and of the Italian Society of Clinical Neurophysiology · 2021
    Article
  9. Article
  10. Article
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  12. 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

13 authors at 8 institutions in 4 countries.

Luca MarsiliDepartment of Neurology, Gardner Family Center for Parkinson's Disease and Movement Disorders, University 6 of Cincinnati, Cincinnati, OH, USA.
Joaquin A VizcarraDepartment of Neurology, Gardner Family Center for Parkinson's Disease and Movement Disorders, University 6 of Cincinnati, Cincinnati, OH, USA.
Andrea SturchioDepartment of Neurology, Gardner Family Center for Parkinson's Disease and Movement Disorders, University 6 of Cincinnati, Cincinnati, OH, USA.
Alok K DwivediDivision of Biostatistics and Epidemiology, Department of Molecular and Translational Medicine, Texas Tech University Health Sciences Center, El Paso, TX, USA.
Elizabeth G KeelingDepartment of Neurology, Gardner Family Center for Parkinson's Disease and Movement Disorders, University 6 of Cincinnati, Cincinnati, OH, USA.
Dhiren PatelDepartment of Neurology, Gardner Family Center for Parkinson's Disease and Movement Disorders, University 6 of Cincinnati, Cincinnati, OH, USA.
Murli MishraDepartment of Neurology, Gardner Family Center for Parkinson's Disease and Movement Disorders, University 6 of Cincinnati, Cincinnati, OH, USA.
Ashar FarooqiDepartment of Neurology, Gardner Family Center for Parkinson's Disease and Movement Disorders, University 6 of Cincinnati, Cincinnati, OH, USA.
Aristide MerolaWexner Medical Center Department of Neurology, Ohio State University, Columbus, Ohio, USA.
Alfonso FasanoDivision of Neurology, Edmond J. Safra Program in Parkinson's Disease and Morton and Gloria Shulman Movement Disorders Clinic, Toronto Western Hospital, UHN, University of Toronto, Toronto, ON, Canada.
Ignacio F MataLerner Research Institute, Cleveland Clinic, Cleveland, OH, USA.
Marcelo A KauffmanConsultorio Y Laboratorio de Neurogenética, Centro Universitario de Neurología José María Ramos Mejía, Buenos Aires, Argentina.
Alberto J EspayDepartment of Neurology, Gardner Family Center for Parkinson's Disease and Movement Disorders, University 6 of Cincinnati, Cincinnati, OH, USA. alberto.espay@uc.edu.ORCID http://orcid.org/0000-0002-3389-136X
University of Cincinnati · USCleveland Clinic Lerner College of Medicine · USEmory University · USHospital Ramos Mejía · ARSt. George's University · GDTexas Tech University · USThe Ohio State University Wexner Medical Center · USUniversity of Toronto · CA

Funding

No grant is acknowledged in the PubMed record.

6 · The paper itself

Abstract

backgroundPostural instability is a disease milestone signaling advanced disease.

objectivesTo estimate the onset of postural instability in monogenic parkinsonisms.

methodsWe systematically reviewed studies (PubMed 1996-2017) in SNCA, PRKN, PINK1, DJ-1, LRRK2, ATP13A2, FBXO7, VPS35, DNAJC6, or SYNJ1-related monogenic parkinsonisms, with documented postural instability. Genes with ≥ 15 patients were included in an individual-patient meta-analysis and compared with a retrospectively collected sporadic Parkinson's disease cohort from our center. The primary outcome measure was the progression-free survival from postural instability using Kaplan-Meier survival curves. Cox proportional hazards analyses were summarized using hazards ratio (HR).

resultsOf 2085 eligible studies, 124 met full criteria (636 patients) for the systematic review, whereas a total of 871 subjects (270 from sporadic cohort, 601 monogenic parkinsonisms) were included in the individual-patient meta-analysis. Postural instability was reported in 80% of DJ-1, 40% of PRKN, 39% of PINK1, 34% of ATP13A2, 31% of LRRK2, and 29% of SNCA patients. Progression-free survival from postural instability at 10 years after disease onset was longest in ATP13A2 (97%) and shortest in SNCA (50%). Halfway between these two extremes were PRKN (88%), PINK1 (87%), and LRRK2 (81%), similar to sporadic Parkinson's disease (72%). Higher risk of postural instability was observed in SNCA (HR = 3.2, p = 0.007) and DJ-1 (HR = 3.96, p = 0.001) compared to sporadic Parkinson's disease. Young age at onset in PINK1 and female sex in LRRK2 were associated with a decreased risk of postural instability.

conclusionsMonogenic parkinsonisms exhibit differential timelines to postural instability, informing prognostic counseling and interpretation of future genotype-specific treatment trials.

Indexed as

Parkinson DiseaseParkinsonian DisordersCohort StudiesFemaleGenotypeHumansRetrospective StudiesDisease milestonesMeta-analysisMonogenic parkinsonismParkinson’s diseasePostural instability

Identifiers

PMID32436106
OpenAlexW3028189349

What OpenQuestion holds

Textmetadata
Read underepoch 390

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.