Identification of QoL, clinical outcomes & research priorities in OI

  • Research type

    Research Study

  • Full title

    Evaluation of the content validity of the OIQoL questionnaire within an international cohort and identification of patient and family priorities and practices for research and clinical outcomes in children with Osteogenesis Imperfecta (OI).

  • IRAS ID

    262258

  • Contact name

    Claire Hill

  • Contact email

    claire.wagstaff@sch.nhs.uk

  • Sponsor organisation

    Sheffield Children's NHS Foundation Trust

  • Duration of Study in the UK

    0 years, 11 months, 31 days

  • Research summary

    We wish to validate the OIQoL, a quality of life questionnaire for children with Brittle Bone Disease, in populations outside the United Kingdom. We want to ensure that when the OIQoL questionnaire is used either clinically, for research or for service evaluation and development, that the results are meaningful for the children or young people in whom it is undertaken. The OIQoL was developed in the UK, which has a particular set of environmental, social and economic circumstances. Those circumstances could well impact on the content of the questionnaire. Clearly, those circumstances will vary from country to country, and we want to be sure that the OIQoL is a valid and robust tool wherever it is used. The proposed multicentre, multi-country validation will determine the extent to which the tool can be used reliably across Europe and more globally to help drive improvements for patients with brittle bones.

    As an additional piece of work (secondary study), we will use the convened focus groups to provide patient views on priorities for research and clinical outcomes in brittle bone disease, as well as their views on what participation in research could reasonably involve and which clinical outcomes are most important to them to be assessed in prospective intervention studies.

  • REC name

    London - Stanmore Research Ethics Committee

  • REC reference

    19/LO/1249

  • Date of REC Opinion

    29 Jul 2019

  • REC opinion

    Further Information Favourable Opinion