Evidence map›Paper›PMID 40364572›Full record

ArticleMovement disorders : official journal of the Movement Disorder Society2025

Development and Preliminary Validation of a Parkinsonism-Dystonia Scale for Infants and Young Children.

Roser Pons, Toni S Pearson, Belen Perez-Dueñas, Angels Garcia-Cazorla, Manju A Kurian, Zoi Dalivigka, Vasiliki Zouvelou, Chrysa Outsika, Eleftheria Kokkinou, Maria Sigatullina-Bondarenko and 7 more

Abstract readValidation Study
In one paragraph

Article in Movement disorders : official journal of the Movement Disorder Society, 2025. 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
–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

3 citing papers in PubMed.

  1. Review
  2. Observational
  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

17 authors.

Roser PonsFirst Department of Pediatrics, Agia Sofia Children's Hospital, National and Kapodistrian University of Athens, Athens, Greece.ORCID https://orcid.org/0000-0001-6749-1906
Toni S PearsonDivision of Neurology, Nationwide Children's Hospital, The Ohio State University College of Medicine, Columbus, Ohio, USA.
Belen Perez-DueñasPediatric Neurology Research Group, Vall d'Hebron Research Institute, Autonomous University of Barcelona, CIBERER, Spanish National Network for Research on Rare Diseases, Barcelona, Spain.ORCID https://orcid.org/0000-0002-4979-2788
Angels Garcia-CazorlaNeurometabolic Unit, Neurology Department, Institut de Recerca, MetabERN and CIBERER, Hospital Sant Joan de Déu, Barcelona, Spain.ORCID https://orcid.org/0000-0001-9853-688X
Manju A KurianMolecular Neurosciences, Developmental Neurosciences, Zayed Centre for Research into Rare Disease in Children, UCL Great Ormond Street Institute of Child Health, London, United Kingdom.ORCID https://orcid.org/0000-0003-3529-5075
Zoi DalivigkaPediatric Rehabilitation Department, Pan & Aglaias Kyriakou Children's Hospital, Kallithea, Greece.
Vasiliki ZouvelouFirst Department of Pediatrics, Agia Sofia Children's Hospital, National and Kapodistrian University of Athens, Athens, Greece.
Chrysa OutsikaFirst Department of Pediatrics, Agia Sofia Children's Hospital, National and Kapodistrian University of Athens, Athens, Greece.ORCID https://orcid.org/0000-0003-2384-3468
Eleftheria KokkinouFirst Department of Pediatrics, Agia Sofia Children's Hospital, National and Kapodistrian University of Athens, Athens, Greece.
Maria Sigatullina-BondarenkoNeurometabolic Unit, Neurology Department, Institut de Recerca, MetabERN and CIBERER, Hospital Sant Joan de Déu, Barcelona, Spain.
Alejandra DarlingNeurometabolic Unit, Neurology Department, Institut de Recerca, MetabERN and CIBERER, Hospital Sant Joan de Déu, Barcelona, Spain.
Maria Del Mar O'CallaghanNeurometabolic Unit, Neurology Department, Institut de Recerca, MetabERN and CIBERER, Hospital Sant Joan de Déu, Barcelona, Spain.
Robert SpaullMolecular Neurosciences, Developmental Neurosciences, Zayed Centre for Research into Rare Disease in Children, UCL Great Ormond Street Institute of Child Health, London, United Kingdom.ORCID https://orcid.org/0000-0003-4096-6945
Dora B D SteelMolecular Neurosciences, Developmental Neurosciences, Zayed Centre for Research into Rare Disease in Children, UCL Great Ormond Street Institute of Child Health, London, United Kingdom.
Evdokia SalamouFirst Department of Pediatrics, Agia Sofia Children's Hospital, National and Kapodistrian University of Athens, Athens, Greece.
Maria João ForjazRed de Investigación en Cronicidad, Atención Primaria y Promoción de la Salud, Madrid, Spain.
Carmen Rodriguez-BlazquezCentro Nacional de Epidemiología y Centro de Investigaciones Biomédicas en Red de Enfermedades Neurodegenerativas, Instituto de Salud Carlos III, Madrid, Spain.ORCID https://orcid.org/0000-0003-3829-0675

Funding

AADC Research TrustInstituto de Salud Carlos III FORT23/00034Instituto de Salud Carlos III PI21/00248
6 · The paper itself

Abstract

backgroundParkinsonism in infancy is rare and is highly correlated with the presence of dystonia. Advances in treating and characterizing developmental and infantile degenerative parkinsonism have highlighted the need for a specialized assessment scale.

objectiveThe aim of this study was to design and validate a scale that effectively assesses parkinsonism-dystonia in early life.

methodsThe Infantile Parkinsonism-Dystonia Rating Scale (IPDRS) was designed to capture the key clinical features of parkinsonism-dystonia in early life. It consists of 28 items across three subscales: Non-motor symptoms, Motor symptoms, and Dyskinesias. Thirty-two patients with hypokinetic movement disorder were scored following a standardized protocol. Filmed motor examinations were analyzed independently by three pediatric movement disorders specialists to evaluate interrater reliability. Twenty additional patients with primary neurotransmitter disorders were scored, and nine of them were evaluated at baseline and after treatment. Psychometric validation was conducted.

resultsA total of 52 patients were scored using the IPDRS. Mean age was 3.1 years (standard deviation [SD]: 2.0), and the mean IPDRS score was 40.8 (SD: 13.17). Internal consistency analysis demonstrated a Cronbach's α of 0.21 for Non-motor symptoms subscale, 0.84 for Motor symptoms subscale, and 0.95 for Dyskinesia subscale. Kappa indexes exceeded 0.70 in seven items. Correlation coefficients for dystonia items with the Barry-Albright-Dystonia Scale ranged from 0.46 to 0.64. After treatment, all IPDRS scores changed significantly, with an effect size of 2.42.

conclusionsThe IPDRS appears to be a reliable and valid tool for assessing parkinsonism in early life. Further validation studies with a larger sample size are needed to confirm these findings and complete the validation process. © 2025 The Author(s). Movement Disorders published by Wiley Periodicals LLC on behalf of International Parkinson and Movement Disorder Society.

Indexed as

DystoniaDystonic DisordersParkinsonian DisordersSeverity of Illness IndexChildChild, PreschoolFemaleHumansInfantMalePsychometricsReproducibility of Resultsbradykinesiasdopaminedystoniahypokinesiaparkinsonism

Identifiers

PMID40364572
PMCPMC12371672

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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.