Evidence map›Paper›PMID 38532733›Full record

ArticleJournal of child neurology2024

Characterization of Fine Motor and Visual Motor Skills in Aicardi-Goutières Syndrome.

Stacy V Cusack, Francesco Gavazzi, Isabella Peixoto de Barcelos, Nicholson B Modesti, Sarah Woidill, Brielle Formanowski, Sara B DeMauro, Scott Lorch, Ariel Vincent, Abbas F Jawad and 4 more

Open access · greenAbstract read
In one paragraph

Article in Journal of child neurology, 2024. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 3 papers.

0numbers the graph read from it
0cells of the map it votes in
3citing papers in PubMed
0.2field-weighted citation impact, top 46% 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

3 citing papers in PubMed, 1 citations in OpenAlex.

  1. Article
  2. Rigorous genetic diagnosis review in natural history studies.Orphanet journal of rare diseases · 2026
    Article
  3. 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

14 authors at 1 institution in 1 country.

Stacy V CusackDepartment of Occupational Therapy, Children's Hospital of Philadelphia, Philadelphia, PA, USA.
Francesco GavazziDivision of Neurology, Children's Hospital of Philadelphia, Philadelphia, PA, USA.ORCID 0000-0002-4908-8395
Isabella Peixoto de BarcelosDivision of Neurology, Children's Hospital of Philadelphia, Philadelphia, PA, USA.
Nicholson B ModestiDivision of Neurology, Children's Hospital of Philadelphia, Philadelphia, PA, USA.
Sarah WoidillDivision of Neurology, Children's Hospital of Philadelphia, Philadelphia, PA, USA.
Brielle FormanowskiDepartment of Neonatology, Children's Hospital of Philadelphia, Philadelphia, PA, USA.ORCID 0009-0009-3916-847X
Sara B DeMauroDepartment of Neonatology, Children's Hospital of Philadelphia, Philadelphia, PA, USA.
Scott LorchDepartment of Neonatology, Children's Hospital of Philadelphia, Philadelphia, PA, USA.
Ariel VincentDivision of Neurology, Children's Hospital of Philadelphia, Philadelphia, PA, USA.
Abbas F JawadDivision of General Peds, Children's Hospital of Philadelphia, Philadelphia, PA, USA.
Timothy EstilowDepartment of Occupational Therapy, Children's Hospital of Philadelphia, Philadelphia, PA, USA.
Allan M GlanzmanDepartment of Physical Therapy, Children's Hospital of Philadelphia, Philadelphia, PA, USA.
Adeline VanderverDivision of Neurology, Children's Hospital of Philadelphia, Philadelphia, PA, USA.
Laura A AdangDivision of Neurology, Children's Hospital of Philadelphia, Philadelphia, PA, USA.
Children's Hospital of Philadelphia · US

Funding

Validation of the GMFC-MLDU54NS115052 · NINDS · CHILDREN'S HOSP OF PHILADELPHIA · PI Adeline Lucie Vanderver · 2019 to 2026
$14.3M
Clinical Outcomes in Aicardi Goutières SyndromeU01NS106845 · NINDS · CHILDREN'S HOSP OF PHILADELPHIA · PI VANDERVER, ADELINE LUCIE · 2019 to 2023
$7.5M
Clinical measures in Aicardi Goutieres SyndromeK23NS114113 · NINDS · CHILDREN'S HOSP OF PHILADELPHIA · PI ADANG, LAURA ANN · 2019 to 2023
$947k
Optimizing Trial Readiness for AdrenomyeloneuropathyU54TR002823 · NCATS · CHILDREN'S HOSP OF PHILADELPHIA · PI VANDERVER, ADELINE LUCIE · 2019 to 2019
–
NCATS NIH HHS U54 TR002823NINDS NIH HHS K23 NS114113NINDS NIH HHS U01 NS106845NINDS NIH HHS U54 NS115052
6 · The paper itself

Abstract

Aicardi-Goutières syndrome is a genetic inflammatory disorder resulting in dispersed neurologic dysfunction. Despite a recognition of overall motor impairment, fine and visual motor skills are undercharacterized. We hypothesize that there is a spectrum of fine and visual motor skills in the Aicardi-Goutières syndrome population as captured by a standard outcome measure, the Peabody Developmental Motor Scales (PDMS-2), which will be proportional to overall disease severity.In a cohort of 74 subjects, the Peabody Developmental Motor Scales-2 grasping and visual-motor integration subtests were administered concurrently with the Aicardi-Goutières syndrome Severity Scale (severe [range 0-3], moderate [range 4-8], and attenuated [range 9-11]). The cohort was also compared by genotype and performance as defined by raw scores. The distribution of Peabody Developmental Motor Scales-2 scores within a genotype was assessed by interquartile ranges (IQRs).Peabody Developmental Motor Scales-2 grasping and visual-motor integration performance was the least variable in the

Indexed as

Autoimmune Diseases of the Nervous SystemMotor SkillsNervous System MalformationsAdolescentChildChild, PreschoolCohort StudiesFemaleHumansInfantMalePsychomotor PerformanceSeverity of Illness Indexdevelopmental delayleukodystrophyoutcomepediatric

Identifiers

PMID38532733
PMCPMC11098691
OpenAlexW4393232269

What OpenQuestion holds

Textmetadata
LicenceTDM
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.