Evidence map›Paper›PMID 39609757›Full record

ArticleBMC public health2024

The IMPACT Survey: the humanistic impact of osteogenesis imperfecta in adults.

Taco van Welzenis, Ingunn Westerheim, Tracy Hart, Lena Lande Wekre, Oliver Semler, Frank Rauch, Laetitia Dewavrin, Ruby Dadzie, Samantha Prince, Cathleen Raggio

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In one paragraph

Article in BMC public health, 2024. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 6 papers.

0numbers the graph read from it
0cells of the map it votes in
6citing 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

6 citing papers in PubMed.

  1. Review
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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

10 authors.

Taco van WelzenisOsteogenesis Imperfecta Federation Europe, Heffen, Belgium.
Ingunn WesterheimOsteogenesis Imperfecta Federation Europe, Heffen, Belgium.
Tracy HartOsteogenesis Imperfecta Foundation, Gaithersburg, MD, USA.
Lena Lande WekreTRS National Resource Center for Rare Disorders, Sunnaas Rehabilitation Hospital, Nesodden, Norway.
Oliver SemlerFaculty of Medicine and University Hospital Cologne, Department of Pediatrics, University of Cologne, Cologne, Germany.
Frank RauchMcGill University, Montreal, Canada.
Laetitia DewavrinWickenstones Ltd, Oxford, Oxfordshire, UK. laetitia@wickenstones.com.
Ruby DadzieWickenstones Ltd, Oxford, Oxfordshire, UK.
Samantha PrinceWickenstones Ltd, Oxford, Oxfordshire, UK.
Cathleen RaggioHospital for Special Surgery, New York, USA.

Funding

No grant is acknowledged in the PubMed record.

6 · The paper itself

Abstract

backgroundThe IMPACT Survey explored the humanistic, clinical, and economic burden of osteogenesis imperfecta (OI) on individuals with OI, their families, caregivers, and wider society. Two previous publications report research methodology, initial insights of the survey, and cost of illness of OI. Here, we present data on the impact of OI on the quality of life (QoL) of adults with OI and explore potential drivers of this impact.

methodsThe IMPACT Survey was an international mixed methods online survey in eight languages (fielded July-September 2021), aimed at adults (aged ≥ 18 years) or adolescents (aged 12-17 years) with OI, caregivers (with or without OI) of individuals with OI, and other close relatives. Survey domains included demographics, socioeconomic factors, clinical characteristics, treatment patterns, QoL, and health economics. We conducted a descriptive analysis of the QoL data, as well as exploratory regression analyses to identify drivers of impact of OI on QoL (independent associations between patient characteristics and the impact on QoL).

results1,440 adults with OI participated in the survey. The proportion who reported an impact of OI on their QoL across individual areas in the physical, socioeconomic, and mental well-being domains ranged between 49 and 84%. For instance, 84% of adults reported an impact of OI on the types of leisure activities they could do and 74% on the type of job they could do. More severe self-reported OI and higher fracture frequency were consistently identified as drivers of OI's impact on QoL. The proportion of adults who reported worrying about different aspects of their lives due to their OI, such as mobility loss, future fractures, and ageing, ranged between 31 and 97%.

conclusionIMPACT provides insights into the humanistic burden of OI on adults, revealing that OI has a substantial impact on the QoL of adults. OI severity and fracture frequency were consistently identified as drivers of impact on QoL across all domains. Understanding these drivers may aid in identifying areas for targeted interventions, such as fracture prevention.

Indexed as

Cost of IllnessOsteogenesis ImperfectaQuality of LifeAdolescentAdultAgedCaregiversChildFemaleHumansMaleMiddle AgedSocioeconomic FactorsSurveys and QuestionnairesYoung AdultBurden of diseaseFatigueFracturesHealth worriesHumanistic burdenOsteogenesis imperfectaPainPatient-reported outcomesQuality of lifeSelf-reported disease severitySurvey

Identifiers

PMID39609757
PMCPMC11605868

What OpenQuestion holds

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LicenceCC BY
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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.