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.
Anna Pease, Trudy Goodenough, Cath Borwick, Rose Watanabe, Christopher Morris, Cathy Williams
Disease Category: Eyes & vision
Disease Name: Cerebral Visual Impairment (CVI)
Age Range: 6 - 18
Sex: Either
Nature of Intervention: Any
- Charities
- Clinical experts
- Consumers (caregivers)
- Consumers (patients)
- Epidemiologists
- Families
- Patient/ support group representatives
- Researchers
- Service providers
- Service users
- COS for clinical trials or clinical research
- COS for practice
- Consensus meeting
- Delphi process
- Interview
- Literature review
We will first conduct a systematic review of the existing literature on outcomes used in evaluations of services or interventions for children with CVI (Aim 1)
We will then collect new data in interviews (Aim 2) to “fill in the gaps” revealed by the systematic review and capture the language and perspectives of children and their families.
Participants
Participants will be children and young people aged 6 - 18 years with a diagnosis of CVI, using any definition, and/or parents or carers of children with this diagnosis.
Sampling method
To include a range of ages and physical capabilities, maximum variation purposive sample will be used to recruit families to a matrix including younger (6-11 years old) and older (12-18 years old) children, and those with and without a diagnosis of cerebral palsy.
Recruitment
We will recruit children and young people from three sources initially and will use a snowballing approach to recruit more if needed. Firstly, we will ask the local Specialist teachers for Vision Impairment to post a notice with information and contact details on their website. They will also notify individuals they think might be interested. We will visit a local school for children with visual impairment (The WESC Foundation, Specialist Centre for Visual Impairment, Exeter) and distribute notices and information. Thirdly we will give the same information to a national parent support group for families of children with CVI (www.cvisociety.org.uk).
Data collection – interviews
Interviews will be carried out in person, at school or at home as preferred or as convenient. Children will be interviewed on their own, after their parent has been interviewed. All interviews will be audio-recorded with consent. A topic guide based on the systematic review and comments from the advisory groups and based on approaches already reported and successful with similar-aged children will be used (for example an activity discussing “a day in the life of a child with CVI”). Interviews will be expected to take 30-45 minutes for adults and 10-30 minutes for children. When interviewing children a range of creative activities will help us to elicit views of young people about what matters most to them. We will use drawings, stories and a tablet to support these approaches to follow the topic guide.
Interviews will continue with families until the point of diminishing returns, when little or no new perspectives are forthcoming and no new outcome domains are being identified. We expect to conduct interviews with approximately 30 families in total.
Analysis
A framework analysis consisting of five steps will be used. First familiarisation with the data will allow for submersion in the topic area. Secondly an initial framework will be applied by coding key issues in the transcripts. Third, the codes will be indexed into categories, fourth, thematic charts will be produced and compared by independent coders (members of the research team) and finally the charts will be interpreted into themes using key ideas and quotes from the original data.