Evidence map›Paper›PMID 39138478›Full record

ArticleOrphanet journal of rare diseases2024

A standard set of outcome measures for the comprehensive assessment of oral health and occlusion in individuals with osteogenesis imperfecta.

L Blokland, H Arponen, A Ahmad, S Colijn, H Gjørup, R John, M Li, D Mekking, S Parekh, J M Retrouvey and 3 more

Abstract read
PubMed Full text at PMC Publisher
In one paragraph

Article in Orphanet journal of rare diseases, 2024. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 2 papers, 1 of them a synthesis that pooled it.

0numbers the graph read from it
0cells of the map it votes in
2citing papers in PubMed, 1 pooled it
–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

2 citing papers in PubMed, 1 synthesis or guideline pooled it.

  1. Pooled it
  2. 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.

L BloklandVogellanden, Center of Rehabilitation Medicine and Special Care in Dentistry, Zwolle, The Netherlands. l.blokland@vogellanden.nl.ORCID 0009-0003-7215-6709
H ArponenDepartment of Oral and Maxillofacial Diseases, University of Helsinki, Helsinki, Finland.
A AhmadWhittington Health NHS Trust Dental Services, London, UK.
S ColijnCare4BrittleBones Foundation, Wassenaar, The Netherlands.
H GjørupCenter for Oral Health in Rare Diseases, Department of Dental and Maxillofacial Surgery, Aarhus University Hospital, Aarhus, Denmark.
R JohnDepartment of Paediatric Dentistry, University of Bristol, Bristol, UK.
M LiDepartment of Stomatology, The University of Hong Kong-Shenzhen Hospital, Shenzhen, China.
D MekkingCare4BrittleBones Foundation, Wassenaar, The Netherlands.
S ParekhDepartment of Paediatric Dentistry, UCL Eastman Dental Institute, London, UK.
J M RetrouveyDepartment of Molecular Genetics, Baylor School of Medicine, Houston, TX, USA.
T Stutz SteigerProRaris, Vuarrens, Switzerland.
L ZhouDepartment of Stomatology, The University of Hong Kong-Shenzhen Hospital, Shenzhen, China.
K AnderssonMun-H-Center, National Orofacial Resource Centre for Rare Diseases and Clinic of Pedodontics, Public Dental Service, Region Västra Götaland, Göteborg, Sweden.

Funding

No grant is acknowledged in the PubMed record.

6 · The paper itself

Abstract

backgroundOsteogenesis imperfecta (OI) is a group of inherited connective tissue disorders of varying severity characterized by bone fragility. The primary objective of this international multidisciplinary collaboration initiative was to reach a consensus for a standardized set of clinician and patient-reported outcome measures, as well as associated measuring instruments for dental care of individuals with OI, based on the aspects considered important by both experts and patients. This project is a subsequent to the Key4OI project initiated by the Care4BrittleBones foundation which aims to develop a standard set of outcome measures covering a large domain of factors affecting quality of life for people with OI. An international team of experts comprising orthodontists, pediatric dentists, oral and maxillofacial surgeons, and prosthetic dentists used a modified Delphi consensus process to select clinician-reported outcome measures (CROMs) and patient-reported outcome measures (PROMs) to evaluate oral health in individuals with OI. Important domains were identified through a literature review and by professional expertise (both CROMs and PROMs). In three focus groups of individuals with OI, important and relevant issues regarding dental health were identified. The input from the focus groups was used as the basis for the final set of outcome measures: the selected issues were attributed to relevant CROMs and, when appropriate, matched with validated questionnaires to establish the final PROMs which represented best the specific oral health-related concerns of individuals with OI.

resultsConsensus was reached on selected CROMs and PROMs for a standard set of outcome measures and measuring instruments of oral health in individuals with OI.

conclusionsOur project resulted in consensus statements for standardization oral health PROMs and CROMs in individuals with OI. This outcome set can improve the standard of care by incorporating recommendations of professionals involved in dental care of individuals with OI. Further, it can facilitate research and international research co-operation. In addition, the significant contribution of the focus groups highlights the relevance of dental and oral health-related problems of individuals with OI.

Indexed as

Oral HealthOsteogenesis ImperfectaFemaleHumansMaleOutcome Assessment, Health CarePatient Reported Outcome MeasuresQuality of LifeDentinogenesis imperfectaMalocclusionMeasuring instrumentsOsteogenesis imperfectaOutcome measures

Identifiers

PMID39138478
PMCPMC11320983

What OpenQuestion holds

Textmetadata
LicenceCC BY
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.