It is a key principle of mental health policy that service users’ views are at the heart of mental health services (A Vision for Change, 2006). Similarly, enabling the participation of young people to engage with issues that concern them is a central tenet of contemporary Irish child and youth policy (Department of Children and Youth Affairs 2014; 2015) and indeed of international human rights policy (United Nations Convention of the Rights of the Child, Article 12, 1989). However, although young people with the genetic disorder 22q11.2 Deletion Syndrome are at known elevated risk of psychiatric disorders (Schneider 2014), their voice has not yet informed research on their lived experience of mental health nor the development of appropriate mental health services.

This research project sought to address this gap by offering a vehicle through which these young people could communicate their lived experiences and service recommendations to parents, educators, researchers, service providers and policy makers. It also aimed to support the development of mental health resilience by providing spaces for young adults to share their stories of mental health challenges and recovery. Resilience was defined simply to the young participants by the researcher as ‘the ability to bounce back after times when you felt upset by your emotions, thoughts or relationships’.

Methodology

Participatory Action Research (PAR) was chosen as a methodology as it aims to empower participants by validating lived experience as a ‘legitimate form of knowledge that can influence practice’ (Baum, F., MacDougall, C., and Smith, D., 2006, :854). Acknowledged as a new paradigm science (Wadsworth, 1998) differing from the old paradigm of positivist science, PAR is located within a transformative research framework (Mertens, 2009), which is particularly appropriate where research aims to change oppressive and outdated social practises, such as the exclusion of marginalised populations. 

Research process

Phase one involved a collaborative grant application and subsequent project planning by a researcher, a family support organisation and a consultant psychiatrist. Ethical approval was received from UCD Human Research Ethics Committee following a full ethical review. Six female participants, aged 18-35 years and diagnosed with 22q11.2DS, were recruited to a ‘Youth Expert by Experience Panel’ (YEEP) through 22q11 Ireland, the national family support organisation. Written informed consent was sought from the young adult participants, and from their parents due to their mild learning disability. Additional written consent was sought regarding the use of participants’ images in photographs, videos and artwork. Participant choice and ability to withdraw consent or to disguise identity was emphasised at the beginning and end of each research session. Participants emphasised that they wished their images and words to be publically shared, therefore several images have been included in this article.

Phase two involved four groups facilitated by the researcher through the use of creative arts, to support peer group cohesion and to identify emergent key themes regarding mental health. Specific Participatory Action Research (PAR) methods included:

– Somatic methods to decrease anxiety 
– Narrative, arts based methods to develop group trust and creative expression 
– Photo elicitation methods to facilitate voice and resistance to invisibility and silencing 
– Digital storytelling methods to disseminate key messages targeted at parents, clinicians and policy makers

One of the six key themes that emerged from these creative participatory sessions was ‘mental resilience’, which the group defined through discussion and consensus as the ‘ability to keep going even when times get tough’. Two videorecorded focus groups were conducted to explore the six themes in more depth, co-facilitated by the researcher and the child psychiatrist. Phase three consisted of creating a digital story of participants’ discussion regarding mental health and their experience of participating in the action research process; this was collaboratively edited by group members and supported by the researcher. Participants were facilitated to present this digital story at 22q11 Ireland’s national conference in November 2016 to an audience of over a hundred parents and professionals. The focus groups and reflective discussions were transcribed and analysed by the researcher. Key findings regarding the impact of participation in the participatory action research process on participants’ mental health resilience are outlined below. 

Key findings

Finding 1: Participation increased the protective factor of mental health awareness and literacy

Participation in a group that discussed mental health problems, treatment and mental resilience normalised talking about these issues for some participants. One young woman expressed her realisation that she was not alone in her struggle with anxiety and stress: 

We all experience anxiety and feeling stress and down from time to time. (E) 

Another young woman commented: 

Being in a group helped me talk about how I feel. For example, we talked about thoughts and feelings and mental health, which usually we don’t talk about every day. We need to talk about mental health more. — Ai

Finding 2: Participation decreased risk factors of social isolation and stigma and increased protective factors of belonging and confidence

Most participants commented on their past experience of social isolation and stigma and credited meeting others with the same genetic condition as increasing their sense of being connected and of being accepted: 

Growing up I always thought I was the only one who had this syndrome, I thought wrong. I used to feel lonely and afraid. But then I met these lovely ladies who now I call friends for life. (A) 

Another young woman said 

It was a nice feeling being in the same room as other people who have the same condition as you, and not being under pressure to have to fit in. — E

Several participants spoke of their increased confidence and sense of being understood due to participating in the YEEP:

It has made me feel more confident with having 22q as I didn’t really like to talk about it with anyone else. But when you’re in a group with people that have it, it makes it so much easier and it takes the stress off you because they know how you understand and feel about it. (A) 

Another young woman shared: 

I feel a bit more confident since being part of the group because I am surrounded by people who are going through the same things as me and it helps that I have someone to turn to. — E

Finding 3: Participation increased acceptance of having 22q11.2DS

Initially, several participants commented on being uncomfortable disclosing their genetic condition to others and one young woman denied even having the condition. However, over the six months study period, there was a growing appreciation of the uniqueness of having 22q11.2DS and the development of a shared solidarity. As one young woman commented: 

I learned that we are the only ones that understand about us like nobody else. — N.

Increased acceptance of having the genetic condition was commented on by all, including the young woman who initially denied having 22q11.2DS: 

I found the group very worthwhile, it helped me accept my condition more. — E.

Each participant agreed to speak about their condition and their mental health onstage at 22q11 Ireland’s national conference where they encouraged other young people and their parents to be open and accepting of their condition:

Just tell everyone about 22q, it just makes life easier. You get so much help and support once you trust friends. — S

Finding 4: Participation facilitated the identification of a need for a multidisciplinary care centre to support complex health needs

The young women spoke of their frustration of attending multiple medical appointments due to the complexity of their condition and their difficulty in not being able to understand what doctors were saying due to their learning difficulties. Great emphasis was placed on the value of doctors speaking in ways that the young women could understand:

I think the clinic should be colourful and friendly and the doctors should listen to us and explain stuff in a way that we can understand. — N

The need for doctors to consult with both the patient and with the patient’s caregiver was emphasised: 

I want the doctors to read the files before we come in so we don’t have to explain ourselves and they should educate the new doctors who come in. Doctors should talk to the 22q patient first and then to the parent or carer or partner. — Ai. 

The young women stated that they need more support with managing mental health challenges such as social anxiety and psychotic symptoms: 

I think there should be doctors in the clinic to talk about anxiety as it is common for everyone with 22q and the doctors should know how to help me to cope with anxiety and big groups. Sometimes I hear sounds and voices like ghosts and I think doctors should be able to help. — K

Finally, there was consensus among all the young women about the need for a multidisciplinary care clinic to support the health, including mental health, of children and young people with this rare genetic disorder. They explained it is simply too overwhelming for them to coordinate appointments due to their complex medical needs and learning difficulties and there was deep concern and anxiety about their future ability to manage their care when their parents are no longer alive to support them. They strongly advocated that a care coordinator should be a central support at such an integrated care clinic:

I think the clinic should have a care coordinator to help and be there at appointments. — E

Research dissemination 

The YEEP, co-facilitated by the research team, presented at two major Irish conferences in 2016, to parents at the National 22q11 Ireland conference and to researchers in a ‘research dialogue’ at the national Children’s Research Network conference (http://www.childrensresearchne...). Their digital story and recommendations to establish a multidisciplinary clinic were also presented to the Minister for Disability and to the HSE National Director for Clinical Programmes in December 2016.

Conclusion

This project illustrates the benefit to seldom heard participants of engaging in an empowering participatory action research process about their mental health experience and needs. The participatory process strengthened mental health resilience by decreasing risk factors such as social isolation and stigma and increased protective factors of mental health awareness, social connectedness, confidence and acceptance of the genetic disorder. Participation also empowered participants to use their digital story to encourage greater understanding of children and young people’s mental health needs among parents and to explain the need for an integrated care clinic to Irish policy makers in their own words. It is imperative that Irish mental health services not only hear the voices of these young service users but act on their recommendations to support mental health resilience through prevention and early intervention initiatives and mental health support services which are appropriate to the needs of children and young people with rare chromosomal disorders such as 22q11.2DS.

Acknowledgements:

This participatory action research was kindly funded by the Irish Research Council New Foundations Scheme. Warmest thanks to the22q Ireland Youth Expert by Experience Panel for sharing their experiences, thoughts, feelings and Recommendations.