Development of a core outcome set for evaluative research into paediatric cerebral visual impairment (CVI), in the UK and Eire

Abstract

Objectives: Cerebral visual impairment (CVI) comprises a heterogeneous group of brain-related vision problems. A core outcome set (COS) represents the most important condition-specific outcomes according to patients, carers, professionals and researchers. We aimed to produce a COS for studies evaluating interventions for children with CVI, to increase the relevance of research for families and professionals and thereby to improve outcomes for affected children.

Design: We used methods recommended by the Core Outcome Measures in Effectiveness Trials Initiative. These included a proportionate literature review of outcomes used in previous studies; qualitative interviews with children and families; a two-round Delphi survey involving parents, children and professionals and a consensus meeting to ratify the most important outcomes.

Setting: Telephone interviews and online Delphi surveys of participants who all lived in UK or Eire.

Participants: Eighteen parents and six young people were interviewed. Delphi participants (n=80 did both rounds) included professionals working with children who have CVI (teachers, orthoptists, ophthalmologists, optometrists, qualified teachers for visually impaired, family members (parents and siblings) and affected children.

Results: The literature review included 13 studies yielding 37 outcomes. Qualitative interviews provided 22 outcomes. After combining and refining similar items, the first round contained 23 outcomes and the second 46. At the consensus meeting, 5 attendees recommended 27 outcomes for inclusion in the CVI COS, of which 15 were ratified as most important, including 4 related to vision; 1 to family well-being; 1 to adults around the child being informed about CVI and the rest to the child's abilities to engage with people and surroundings.

Conclusions: Good engagement from participants led to the development of a COS. Future research will be useful to identify the best ways to measure COS items and potentially to update this COS as more interventions for CVI are developed.

Trial registration number: ISRCTN13762177.

Keywords: epidemiology; paediatric neurology; paediatric ophthalmology; qualitative research.