Evidence map›Paper›PMID 36996759›Full record

ArticlePediatric neurology2023

Motor Function and Physiology in Youth With Neurofibromatosis Type 1.

Alexander C Doherty, David A Huddleston, Paul S Horn, Nancy Ratner, Brittany N Simpson, Elizabeth K Schorry, Lindsey Aschbacher-Smith, Carlos E Prada, Donald L Gilbert

2 registry-linked trialsOpen access · greenAbstract read
In one paragraph

Article in Pediatric neurology, 2023. The graph could read no effect estimate from its abstract, so it casts no vote on the map. It is linked to 2 registered trials, which are not on this map. Cited by 3 papers.

0numbers the graph read from it
0cells of the map it votes in
3citing papers in PubMed
0.9field-weighted citation impact, top 27% 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.

NCT04421248 phase4recruitingnot on this map

Anomalous Motor System Physiology in Attention Deficit Hyperactivity Disorder: Biomarker Validation and Modeling Domains of Function

TypeinterventionalSponsorDonald Gilbert, MD, MS, FAAN, FAAPRan2020 to 2026Enrolled214ConditionsAttention Deficit Hyperactivity Disorder CombinedArmsMethylphenidate, Placebo
NCT04481048 phase2completednot on this map

Antioxidant Therapy With N-acetylcysteine for Motor Behavior and/or Learning in Children With Neurofibromatosis Type 1

TypeinterventionalSponsorChildren's Hospital Medical Center, CincinnatiRan2021 to 2024Enrolled25ConditionsNeurofibromatosis 1ArmsN-Acetyl cysteine, Placebo
3 · Its place in the literature

Who cites it

3 citing papers in PubMed, 7 citations in OpenAlex.

  1. Review
  2. NF1Clinical and translational science · 2024
    Article
  3. 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

9 authors at 2 institutions in 1 country.

Alexander C DohertyUniversity of Cincinnati College of Medicine, Cincinnati, Ohio; Division of Neurology, Cincinnati Children's Hospital Medical Center, Cincinnati, Ohio. Electronic address: dohertar@mail.uc.edu.
David A HuddlestonDivision of Neurology, Cincinnati Children's Hospital Medical Center, Cincinnati, Ohio.
Paul S HornDivision of Neurology, Cincinnati Children's Hospital Medical Center, Cincinnati, Ohio; Department of Pediatrics, University of Cincinnati College of Medicine, Cincinnati, Ohio.
Nancy RatnerDepartment of Pediatrics, University of Cincinnati College of Medicine, Cincinnati, Ohio; Division of Experimental Hematology and Cancer Biology - Rasopathy Program, Cincinnati Children's Hospital Medical Center, Cincinnati, Ohio.
Brittany N SimpsonDepartment of Pediatrics, University of Cincinnati College of Medicine, Cincinnati, Ohio; Division of Human Genetics, Cincinnati Children's Hospital Medical Center, Cincinnati, Ohio.
Elizabeth K SchorryDepartment of Pediatrics, University of Cincinnati College of Medicine, Cincinnati, Ohio; Division of Human Genetics, Cincinnati Children's Hospital Medical Center, Cincinnati, Ohio.
Lindsey Aschbacher-SmithDivision of Human Genetics, Cincinnati Children's Hospital Medical Center, Cincinnati, Ohio.
Carlos E PradaDivision of Genetics, Ann & Robert Lurie Children's Hospital of Chicago, Chicago, Illinois; Department of Pediatrics, Northwestern University Feinberg School of Medicine, Chicago, Illinois.
Donald L GilbertDivision of Neurology, Cincinnati Children's Hospital Medical Center, Cincinnati, Ohio; Department of Pediatrics, University of Cincinnati College of Medicine, Cincinnati, Ohio.
Cincinnati Children's Hospital Medical Center · USNorthwestern University · US

Funding

Anomalous Motor Physiology In ADHDR01MH078160 · NIMH · HUGO W. MOSER RES INST KENNEDY KRIEGER · PI MOSTOFSKY, STEWART H · 2006 to 2024
$5.8M
2/2-Anomalous Motor Physiology in ADHDR01MH095014 · NIMH · CINCINNATI CHILDRENS HOSP MED CTR · PI GILBERT, DONALD L · 2012 to 2024
$3.3M
GABAergic Sensorimotor Dysfunction in Tourette SyndromeR01NS096207 · NINDS · HUGO W. MOSER RES INST KENNEDY KRIEGER · PI GILBERT, DONALD L, MOSTOFSKY, STEWART H · 2016 to 2020
$2.9M
Short Term Medical Student Training GrantT35DK060444 · NIDDK · UNIVERSITY OF CINCINNATI · PI JASON T BLACKARD · 2002 to 2026
$2.3M
NIDDK NIH HHS T35 DK060444NIMH NIH HHS R01 MH078160NIMH NIH HHS R01 MH095014NINDS NIH HHS R01 NS096207
6 · The paper itself

Abstract

backgroundNeurofibromatosis type 1 (NF1) is a genetic neurocutaneous disorder commonly associated with motor and cognitive symptoms that greatly impact quality of life. Transcranial magnetic stimulation (TMS) can quantify motor cortex physiology, reflecting the basis for impaired motor function as well as, possibly, clues for mechanisms of effective treatment. We hypothesized that children with NF1 have impaired motor function and altered motor cortex physiology compared to typically developing (TD) control children and children with attention-deficit/hyperactivity disorder (ADHD).

methodsChildren aged 8-17 years with NF1 (n = 21) were compared to children aged 8-12 years with ADHD (n = 59) and TD controls (n = 88). Motor development was assessed using the Physical and Neurological Examination for Subtle Signs (PANESS) scale. The balance of inhibition and excitation in motor cortex was assessed using the TMS measures short-interval cortical inhibition (SICI) and intracortical facilitation (ICF). Measures were compared by diagnosis and tested using bivariate correlations and regression for association with clinical characteristics.

resultsIn NF1, ADHD severity scores were intermediate between the ADHD and TD cohorts, but total PANESS scores were markedly elevated (worse) compared to both (P < 0.001). Motor cortex ICF (excitatory) was significantly lower in NF1 than in TD and ADHD (P < 0.001), but SICI (inhibitory) did not differ. However, in NF1, better PANESS scores correlated with lower SICI ratios (more inhibition; ρ = 0.62, P = 0.003) and lower ICF ratios (less excitation; ρ = 0.38, P = 0.06).

conclusionsTMS-evoked SICI and ICF may reflect processes underlying abnormal motor function in children with NF1.

Indexed as

Neural InhibitionNeurofibromatosis 1AdolescentChildElectromyographyEvoked Potentials, MotorHumansQuality of LifeTranscranial Magnetic StimulationBiomarkerMotor developmentNeurofibromatosisPediatric neurologyTranscranial magnetic stimulation

Identifiers

PMID36996759
PMCPMC10228140
OpenAlexW4322761594

What OpenQuestion holds

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LicenceTDM
Read underepoch 390

Registered trials

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.