Literature review

Eight relevant articles were uncovered (online supplemental material table 5). Articles included audits,33 59 cohort studies60–64 and one case series study.65 Studies assessed a range of factors in relation to transition, including the status of patient transition needs,33 61 engagement with, and access to adult services,59 satisfaction with a specific transition programme,60 perceived preferences and barriers to various services (eg, transition programmes),60 individual and parent concerns across the lifespan,65 66 adult outcomes62 and individuals’ experiences of healthcare transition.64 65 No Canadian studies were identified.

Environmental scan

From the search across the 13 Canadian children’s hospital websites, several resources related to either intersex or healthcare transitions were uncovered. However, no resources related to both intersex and healthcare transition.

The survey garnered three responses (see online supplemental material table 2) despite over 500 emails being sent. One respondent used transition materials for pediatric-to-adult care. One used discharge summary resources with individuals with intersex.

From the FOI requests sent to each Children’s hospital, two hospitals reported using practice documents (see online supplemental material table 3). Eleven reported no policies, guidelines, or procedures for working with individuals with intersex.

The results of both the literature review and the environmental scan outline the lack of evidence-based and formalised resources, policies, procedures or guidelines used by HCPs when working with individuals with intersex traits in Canada, as well as the lack of resources that are available to these individuals when searching hospital websites for information about their conditions.

Engagement sessions

The following sections examine the three themes dominant across the engagement sessions data, providing excerpts shared verbatim to illustrate impact and highlight metaphors of significance.

Theme 1: transition is a lifespan activity

First, transition was constructed by participants as a lifespan activity encompassing the whole life course, despite transitions in care often being focused on the pediatric-to-adult care experience. Individuals in our study reported facing challenges across their life course, which became part of their changing journey as they aged. Sourcing different providers because of ageing was a common challenge, with the role of a primary care provider becoming central in helping individuals navigate healthcare complexity outside of intersex‘…the intersex or the AIS [androgen insensitivity syndrome] or whatever takes a real background to gerontology, I’m 77,…dealing with sort of end-of-life issues’ (P1).

Changes in care meant that individuals needed to proactively seek out new healthcare partnerships, which for some included letting go of trusted relationships with specialists. When individuals moved geographically, as noted by FG1, there was a stepwise approach or sequence in events that brought about opportunity to make new healthcare relationships:

…like transitioning from like a family doctor who I had since I was a baby to my endocrinologist [in adolescence] when I was in whatever grade to moving here [new city] to then having [to find a new] family doctor so……transitions within those practitioners and the various experiences (FG1).

Geography transitions were also connected with immigration to Canada, which resulted in a new medical team and new diagnosis (P4). A prior misdiagnosis or curiosity about an individual’s diagnosis resulted in transitions in self that were unexpected for P4, while P3 noted neglecting exploring a diagnosis, ‘since I was too busy.’ P2 argued early relationships with specialist providers supported a transition to similar specialists:

[The Specialist] didn’t speak to me like I was a child at any point [but] like we were very much equals…and I feel like very much the same now with the adult endocrinologist, but I don’t know if that would be like a universal thing (P2).

For P2, establishing a satisfactory primary care relationship took a little more effort; ‘(the) nurse practitioner, that I kind of see as my family doctor, set me up with an online service that doesn’t actually have my records.’ For others, there was an expectation that transition also meant a responsibility to have an acceptable level of knowledge about their healthcare needs, as P5 noted,‘I should know these things, being 40.’ Further discussion focused on transitions not only linked to age but also to life events such as motherhood. A request to visit genetic counselling and a requirement to attend prenatal care and connect with new specialist providers who knew less about steroid management resulted in uncertainty:

…gestational diabetes’s twice, nobody told me that [my condition] affects your blood sugars. Not until I got gestational diabetes and it’s basically, oh, by the way this is because of your condition (P5).

Transition then became focused on a reluctance to let go of daily glucose testing, worrying about well-being, and finding courage to seek out community care, as stated by P5,‘you kind of have to seek it out yourself. Nobody refers you out.’ For others, the notion of transition was linked to bodily functionality with age, how a specific condition can influence hormones and limit future fertility,‘down the road,’(P7) or,‘declining hormones,’ resulting in a less visible genital difference (P3). P7 discussed celebrating moments when the body gave signals that there was hope for a future transition to motherhood; ‘at 27 I go months with nothing, or I’ll go months with continuous spotting and then I’ll ovulate like once in a while and I’ll go whoo hoo at least I’m still ovulating’ (P7).

For some, transition related to uncertainty. Despite taking medication as prescribed, attending for blood work, and following medical advice there was a space in which the body ‘transitioned’ between being in harmony with medical care or out of sync, as described by P6:

Almost being 30, I’m really not old yet, but I’m not in my teens anymore…The Dr. is concerned about my bloodwork. In the past year and a half, mainly my steroid dose, we’ve been really trying to get that under control. For some reason, we haven’t been able to get that quite right [even though] we’ve been playing with the dosage (P6).

Transition as a life course event was inherently linked to being relational, with others and self. This changed for individuals over time as they aged, were able to find independence, and confidence in understanding their bodies. There remained a desire for support especially in the transition spaces and phases within adult care with FG2 agreeing that,‘(you’re) on your own’ which is significant since FG1 concisely stated,‘it’s a life of transitions.’

Theme 2: the building of personal agency

Second, participants emphasised the value in building personal agency over the life course. Personal agency focused on the ways in which individuals had the capacity to act, both towards self-care and knowledge with confidence, throughout transition phases. For some participants, awareness of being intersex started in childhood which continued until present day as adults or older adults. For others, there was low-agency, that is, little power or control over their feelings or actions since they had knowledge gaps about their body from childhood,‘I have no idea what the purpose of this [genital examination] was,’ only later connecting this with being, ‘big enough ‘down there’ for intercourse,’ (P5). When the diagnosis of intersex came during adolescence, there was a sense of drowning in information, needing to process it quickly but being unable to do so effectively and with self-compassion. For example, participants said, ‘You’re still downloading a lot of information about yourself and realising that you’re different and what is that difference and how does that resonate with you and everything?’ (FG1); and ‘[t]here was a lot of things I didn’t understand about my condition and my understanding was so limited that I didn’t even know what questions to ask’ (P8). A diagnosis in young adulthood was also hard, with P1 saying over 50 years ago, ‘I was sort of hoping it wasn’t true’ and:

I’m sort of sugar coating because I sound as though I had it all together and I must admit that I thought I had it all together when I was 25. I thought well, this is it, this is what’s gone down and then it didn’t hit me until I was 29 and … I was just unhappy (P1).

Constructing a healthy level of agency, or even countering perceived over-agency from others, that is, those with good intentions or overly caring approaches, was difficult for adolescents growing up aware they were intersex. As P5 reflected, ‘not sharing everything with mom and dad…or the doctor’ was a way to learn about holding ‘privacy’ and managing anger. Emotions were reflective of individuals’ states of mind at various stages in their lives described as ‘emotional health,’ (FG1) which influenced relationships with self and others. While there was no specific time, transition experience, or event that triggered emotional awareness or growth, it was talked about by most participants as being in their mid-to-late-twenties and early-thirties. Recognising, reflecting, and attending to emotions was also linked to personal agency, time, resources and supports:

…people who have intersex problems, they need therapy when they’re ready…it has to be when you’re actually in a position to be able to deal with it and then it has to be somehow available and somehow recognized (P1).

The focus groups allowed for a general discussion of the harm misinformation had. For example, some individuals believed they ‘couldn’t have sex at all,’ (FG2) which generated dialogue around the idea of information literacy and sex education in schools. One participant reflected on how their loss of confidence was linked to school education being ‘very heteronormative and not very informative’ (FG2).

Developing agency was also possible because of peer-to-peer networking and support groups. Those with CAH did not connect with others with CAH while the remaining participants accessed support groups, conferences, or online forums. Such spaces allowed individuals to use language to describe themselves, ‘saying [at the] conference, they have AIS, DSD but [not] using it outside of that context,’ (FG2). Overwhelmingly, once an individual decided to access peer support there were positive outcomes after initial hesitations; ‘I wasn’t sure whether I was even in the right place [support group meeting], whether… I had this feeling, am I intersex enough?’ (FG2). Building personal agency took time and emotional investment which in combination had the potential to support self-enquiry; ‘I feel like now as an adult a great deal of curiosity about it’ (FG1), which in turn developed well-being.

Theme 3: promotion of well-being as personal health literacy

Finally, the concept of well-being was framed indirectly within day-to-day life, living well and appreciating self and others. Well-being was informed by personal health literacy, that is, being able to find, understand and use information to guide health-related decisions and actions. A critical aspect of being well was linked to sourcing health information that was ‘trustworthy,’ and at an ‘understandable level’ (FG1). This included information about daily activities such as diet, exercise, and sleep. The need for rest to nurture wellness was reported in a physical sense;’ I’ll sleep for hours and hours and I still don’t feel refreshed in the morning,’ (P7). Wellness was also, for several participants, linked to a history of difficulties with mental health or the combination of medication and physiology as described by P8; ‘(I was) struggling with my blood pressure……I saw a cardiologist [but it was] the anti-depressants,’ that impacted sleep. Participants were keen to understand how to be healthy, recognising they had knowledge gaps which confounded their ability to determine which aspects of public health messaging applied to them. This was important as individuals transitioned across the life span, as they developed new conditions such as diabetes, hypertension, depression, anxiety, osteoporosis, coagulation conditions, obesity and other chronic conditions. A key aspect of being well was linked to education:

…education is the most important thing and there’s going to be, unfortunately many people who are not going to be educated, you know, I want to sort of acknowledge my privilege and so to say, you know, well people should be able to educate themselves (P1).

Being able to seek, access, and critique information is also dependent on the ways in which it is presented; its complexity and inference should not be ‘offensive’ (P1). Having the capacity to check information was also part of staying well because, ‘there’s room for error too when you learn things yourself, right. Like you might not be getting the right information’ (P9). When there were uncertainties that is, how medication influences physical wellbeing, health became a ‘mystery’ (P9). P3 discussed recognising that the health landscape changes at a rapid pace, with new generations of doctors acknowledging both complexity and uncertainty with, ‘new information being added to the systems every single day.’ Yet for many, there remained frustrations when participants wanted to be educated by others, not be ‘the educators’ themselves.

I don’t want to have to like re-teach somebody and I think that’s where a lot of the frustration or like upset comes from is when you have to re-educate somebody who’s supposed to be educating you. So I’m supposed to, to take care of me?…I need to find someone who has the answers…so finding my community it’s definitely been helpful…to like guide you through what you need to do (P2).

Relationships with providers were an important part of well-being. Some providers had greater capability than others; ‘I’ve also had a lot of really positive experience with healthcare practitioners,’ (FG1). Those who were responsive were noted as helpful; ‘(…) it was really great to have these questions (…) and say [to my community] I asked this and got this back’ (P2). Others lacked education and connection,‘gave terrible information’ (P3) or left participants ‘in the dark about my condition’ (P8). Interactions with providers thus could either discuss (un)wellness in ways that supported individuals to secure a sense of well-being or result in frustration and uncertainty.