Title: Parents and children’s communication about inherited genetic conditions and the effects on family functioning
Funder: Department of Health (England)

Abstract: WORK ELEMENTS Element 1: Consolidating the programme research team (PRT) (Months 1-12) The PRT will meet monthly to oversee the PDG work, take part in activity related to their research expertise and further design the programme in detail. Additional personnel will be recruited based on emergent gaps in expertiseincluding a health economist, family behaviourist and a trialist. The work delivered in the PDG will determine whether the PRT can establish strong working relationships across research sites. The PRT will set inclusion and exclusion criteria and decide the IGCs to be included, which will be representative of the main genetic inheritance patterns and associated mortality and morbidity. Element 2: Recruitment of genetics centres (Months 1-4) Three genetic centres, London, Manchester and Cardiff, are collaborating on this programme. Depending on the IGCs chosen for inclusion in the programme grant, additional genetic centres may be required as research sites, to assist with recruitment for some work packages due to the relative rarity of specific IGCs. PRT members will meet researchers and clinicians from other centres who have indicate an interest in participating and enlist collaboration in developing the interventions, informing outcome measures and recruitment to future programme grant studies. Element 3: Design the MFDG intervention (Months 2-6) We are proposing using a MFDG intervention to assist families affected by IGC, however it is important that that it meets the needs of families and is implementable. Therefore families affected by IGCs are invited to take part in focus groups to inform the design of the MFDG sessions. Two essential aspects where the research users (families and HPs) input is required are; i) agreeing on the composition, set up, and timings and delivery schedule of the MFDG and the acceptability of the intervention to families and HPs, fitting with their schedules and availability, and ii) agreeing the importance of outcomes to be measured and included in future studies. High quality focus groups will be undertaken with families and HPs over two sessions62,63. Participants: The families will be divided into three separate groups; i) parents, ii) children (aged up to 12 years) and iii) young people (13-18 years). Two HPs will join each group. Focus groups will be carried out with up to ten participants in each in order to gain a range of views55. Recruitment will be adjusted to take into account ethnicity, social class and educational backgrounds56. Focus groups will be conducted in lay language. Researchers experienced with working with children and young people will conduct these focus groups using developmentally appropriate language and child-centred methods. The venue will be suitable for implementation methodologies appropriate to the participants’ age group and abilities to allow activities eg drawing, colouring, role play and mental mapping. This will assist building a rapport between participants and with researchers57 and assist with setting up discussions. Risk assessments will be carried out for all venues and researchers will have CRB checks. Focus group 1: The interview schedule will focus on the design of the MFDG sessions, for example what they should include, who should be included, how should they be organised, their frequency and venue. Focus group 2: Response rates and completion rates are higher where service users users are involved in choosing the outcome measures for research studies19. Therefore the second focus groups will explore participants’ views of the acceptability and suitability of the validated outcome measures for the research programme studies, in relation to family communication and well-being, previously identified in systematic literature reviews (See pCase for Support Part 3). Participants will receive materials to review 2-3 weeks before arriving at their focus groups. A facilitator will conduct each focus group using a non-directive style with open and neutral questions. A moderator will assist with managing the session, guiding the facilitator in exploring the issues raised, observing participants’ behaviours and recording field notes55. The focus group interviewer and moderator will be sensitive to the morbidities and mortality associated with different IGCs. Recruitment: Representative families affected by IGCs that meet inclusion criteria agreed by the PRT (See Element 1) will be invited to participate. Families will be invited from charities through Genetic Alliance UK via an invitational letter. Data Analysis: In addition to compiling field notes and observations, all focus groups will be audio-recorded, transcribed and managed using Atlas T.i 6.2. Thematic analysis will be used for data analyses, since the issues posed to the focus groups are well defined and the data obtained will lend themselves well to descriptive categorisation51,58,59 Outcome: Identification of parents’, children’s, young people’s and HPs views of MFDG set up to support discussion of IGC with families, and refinement of the outcome measures for use in the research programme. Element 4: Family surveys to assess feasibility and acceptability of studies in programme grant (Months 6-12) With little empirical work on a large scale that explores psycho-social research, an essential aspect of the PDG will be to ascertain whether families affected by IGC are likely to participate in studies, and if so, what type. Process: Supported through the relevant country's local research network, participating genetics centres will conduct an audit of attending families, numbers, family size and children’s age groups and the IGC for which they attended. Families attending the genetic clinics in the previous two years will be contacted via a letter with brief questionnaire, designed by the PRT (an electronic questionnaire version will also be provided). The 15minute questionnaire will briefly describe the research activities planned in the research programme. Parents will be asked which studies they would consider their family participating in. The questionnaire will collate baseline demographic information. Descriptive statistics will indicate potential recruitment rates for different research programme work packages and multiple regression models will be used to predict variables affecting families’ participation. The questionnaire will ask whether families wish to be approached for research studies if the future programme is funded and a database of prospective participants developed. Element 5: Testing the acceptability of an MFDG intervention (Months 3-15) Research question: Is a MFDG psycho-educational intervention to facilitate family communication about IGC acceptable to parents, children and young people? Background: Positively developed family communication skills result in resilient families that cope with the stress and strains upon it through strong parent-child relationships28. With the stresses of living with an IGC and its risks, our previous work suggests highly developed communication skills are needed by parents and children to support better family communication and functioning 4,8,19. The most successful interventions for facilitating communication and improving family functioning are MFDG, psycho-educational and interactive, involving parents and children43. We propose drawing on these interventions to develop a MFDG for families affected by IGCs that can be delivered by Genetic Counsellors (GCs). Intervention: The psycho-educational intervention will use a MFDG format38,41,43. based on model designed and agreed following the focus groups (Element 3). This will ensure the intervention is relevant to the needs of families and fits with service provision. Three GCs from Guy’s and St Thomas NHS Trust (GSTT) Genetics Centre will be trained in the intervention’s use by highly experienced family therapists. The GCs will be supported by family therapists with systemic family intervention skills and assessed for their fidelity to the intervention. A training manual will be developed for GCs around delivering the MFDG for families with IGCs. Once designed the intervention’s acceptability for use will be tested through a small exploratory RCT Participants: All families with at least one child Recruitment: Families will be recruited from the GSTT genetics centre for this exploratory trial Method: An exploratory RCT will ascertain the acceptability of the MFDG psycho-educational intervention to parents and their families. Families will be randomised to receive the intervention or information only (control arm). A waiting list control may be considered in the future programme depending on findings from elements four and five Sample size: Twenty families will be randomised to either receive the intervention or the information only. Data collection: Questionnaires to both groups, administered by a researcher at baseline and six months post the intervention group’s completion of the last MFDG session, and prior to the control group commencing their sessions. Questionnaires will contain assessments of all the relevant outcome measures listed below. Primary outcome measure: The primary outcome will be the acceptability of the MFDG intervention to parents, children and young people. Secondary outcome measures: Outcome assessment will take place according to findings from focus groups (Element 3) but will include measures known to affect families communication; demographic information, family functioning44, parental anxiety and depression (GHQ-12)60, children and young people’s well-being (Kidscreen-52)61. Other assessments will include the feasibility and acceptability of a definitive trial. Inclusion criteria: Families who are wishing to discuss the IGC with their children, where the children are 15 years, children and young people will provide assent. Exclusion criteria: Families unwilling to discuss IGC with their children. Parents who are unable to speak English, which will limit the delivery of the intervention. Data analysis : The effect of the intervention on family functioning, anxiety and depression of parents and well-being of children for the six month period will be analysed using repeated analysis of variance. Associations between outcome measures for parents and children will be statistically analysed using similar methods. All analyses will take into account clustering effects within families and will be on an “an intent to treat” basis. Data will be collated on dropout rates and interviews will take place to ascertain reasons. No previous RCTs of psycho-educational interventions for families affected by an IGC have been carried out. Therefore potential influences that may affect the success of a definitive trial will need to be determined.


Research activity codes: 7.1 INDIVIDUAL CARE NEEDS
Health categories: CONGENITAL DISORDERS