It is acknowledged that children should collaborate in research about their health conditions, and DSD research has been criticised for promoting views of parents and health professionals. However parents are concerned about professionals talking to children about sensitive subjects including fertility and sexual activity. Children may have little experience of talking about their sex development. This makes direct research involving children with DSD particularly challenging. As part of a wider qualitative interview study concerning family responses to management of girls with CAH, we asked 27 parents about the possibility of involving their children in research. We had gained ethical approval to speak to children 515 years, enabling us to prompt parents to consider this possibility. We proposed talking to girls with CAH about common childhood leisure activities as a non-threatening method for engaging children in research discussions. For those parents of younger children the question was more theoretical; allowing them to think about possible future research options. Parents concerns in relation to their daughters research participation are summarised in the following themes.
Necessity: Although parents wanted their daughters to be able to have their questions about CAH answered, they feared taking part in research would evoke concerns prematurely.
Ending ignorance: Research participation might initiate their daughters awareness of potential differences and cause distress.
Promoting talking: Having considered CAH for the purpose of research, their daughter might then discuss further with friends without parental guidance.
Implications: These barriers to childrens participation in DSD research may also counter understanding of the diagnosis throughout childhood a recommended aim of multi-professional clinical care. Some parents were enthusiastic about childrens involvement in research and had helpful methodological ideas including: whether the researcher should be known, research activity location; the use of standardised questionnaires; the minimum age for participation, and question design. However parents advice was varied and contradictory, highlighting the need for careful patient and parent involvement in study design. We conclude that childrens research participation needs to be flexible and involve a range of modalities. We piloted the leisure activity questions with four girls aged 1214 years who had had variable surgical management for atypical genitalia. Only one participant acknowledged any awareness of or concern regarding genital difference. The indirect approach was most appropriate for the youngest child. We conclude that it is difficult to engage participation in DSD research when prior awareness of the genital difference is low.
27 - 29 Sep 2018
European Society for Paediatric Endocrinology