 Okay, so in an effort to keep us close to on time, I'll get started. I'm gonna present my thesis data, so I'm really excited that uncertainty came up a lot because it's near and dear to me. So my study was on the perceptions of uncertainty among parents of children with undiagnosed medical conditions. Just to give a brief outline, I'll go over what I mean by undiagnosed and a little bit about what's been reported about uncertainty already, and then discuss the design of my study and some of the results I find most interesting. So in undiagnosed, my goal was to recruit parents of children who likely had an underlying syndrome for which the cause remained unknown. So they often had a lot of symptoms identified. They had epilepsy, they had failure to thrive, they had hearing loss, et cetera. But the cause of all of these was still unknown. And so just to give a little background about uncertainty, it's been defined in multiple different ways, and there are multiple different models and ways to look at it and study it. And the definition I like best is the subjective perception of, not illness, actually, but the subjective perception that there's something you don't know. So you have to first know that there's something you don't know to be uncertain about it. And research by fellow alum have shown that uncertainty pervades many aspects of healthcare. It has multiple sources. It leads to multiple different stressors. It leads to struggle coping and adapting to these conditions and it leads to psychosocial emotional distress, anxiety, depression, and helplessness. So my study design was a cross-sectional online survey of multiple scales and open-ended questions and I recruited through support and advocacy groups such as syndromes without a name and you are our hope. And the criteria was that you had a child with a medical condition that had not been diagnosed and involved at least two parts of the body aiming to get at these people who likely had underlying syndromes. I'm sure everyone in the room is familiar with this theoretical framework. It's Lazarus and Fogman. So I won't go into too much detail, transactional model of stress and coping. But my study aimed to measure what I have here in red. So we looked at three key personality traits, resilience, optimism, and tolerance of uncertainty. We looked at uncertainty with a newly developed scale. Coping self-efficacy and coping. But today I'm gonna limit my discussion to the uncertainty and coping efficacy. So we measured perceived uncertainty with this new scale, parental uncertainty about child's health. It's a scale that Barb and Paul Hahn worked really hard to get up and running for this and many other projects. And it looked at seven different domains of how we were conceptualizing uncertainty. And we also used coping self-efficacy scale to measure parents' confidence in their ability to cope successfully. So not that they were actively coping, but that they believed they could cope with it and manage it. So the aims I'm gonna focus on today are the dimensions of uncertainty and their relative importance. The scale allowed us to measure both how uncertain parents were on this domain and how important it was to them to resolve that uncertainty. And also the association between uncertainty and coping efficacy. So just to give a little background on who participated, we had 94 total completed surveys. Not too surprisingly, the vast majority were mothers. They were married, white, fairly well-educated. And for most of the children, they were younger than two when a physician or someone first mentioned that there was concern something was wrong with them. And most of them had only been undiagnosed, if you will, for less than eight years. So uncertainty. So it's weighted both how uncertain are you and then how important is that uncertainty to resolve for you. And what we found is that the majority of people perceive great uncertainty about raising a child with an undiagnosed illness and that the areas in which they are uncertain matter greatly to them. So it's not that they're uncertain and they're okay with being uncertain, but they're actively working to try and resolve that uncertainty. Our scale, as I mentioned, we set out to have seven different domains, but a factor analysis revealed that four domains better captured the uncertainty parents were facing. Medical management, things like having to make treatment decisions and being involved in understanding a prognosis, things like that. Reproductive uncertainty fairly straightforward. You know, the belief that this could happen again, not only to themselves, but to other family members who had queried them about what are my chances given your child is affected. Social uncertainty was the area parents were most uncertain and expressed the greatest desire to resolve that uncertainty. And our survey or the scale discussed things like, I have trouble finding parents who share this situation with me and I don't have an area for support. And anecdotally, Sam and I have just started thoroughly coding the qualitative responses and I think this particular dimension could be expanded further. What we've realized is there are quite a few parents who not only don't know how to talk about it, but they literally lack the words to talk about it. Without a diagnosis, they find they can't, they stumble upon how to begin explaining it. And that that's kind of their first stumbling block that may perhaps I could find a group, but I don't even know how to tell you what's wrong. An existential uncertainty. So the negative value implies that there was uncertainty, but this was an area they were less concerned about resolving this uncertainty. And these questions focused on the purpose of my child's life and what that child means to me. And so naturally it makes sense that they might be questioning that and yet less concerned with trying to discover the purpose of their four-year-old daughter's life in this world. So the third aim, and we explored multiple different relationships between uncertainty and coping efficacy and then our other outcomes of ways of coping, including problem and emotion focus. But what we did find is that lower levels of perceived uncertainty were associated with higher coping efficacy, meaning those parents who perceived less uncertainty felt better able to handle the uncertainty they were facing and fairly significantly. Uncertainty was so in addition, as I said, we looked at personality traits and uncertainty was only predicted by optimism. So all demographic factors fell out of the model, all other time-sense diagnosis, all of those things fell out. And what seemed to matter most in predicting how uncertain someone was was how generally optimistic they were. And so since it's a personality trait, it's a measure of their state and not relative to the experience they're going through. And coping efficacy was predicted by uncertainty, as I said, and then interestingly, both optimism and resilience, which I think also makes sense in the context of people who are more optimistic and resilient are more likely to believe they can make it through a difficult situation and that they have the resources and strength to get through that. So it's just a short review of my thesis because there's a lot more, but the conclusions we can draw from this is that for all of these parents, the uncertainty is inevitable. It exists and it's important for, they view it as important to resolve and in particular, the social uncertainty and that the uncertainty they experience influences parental coping efficacy. But, oh, sorry. So the limitations of the study are that, of course, it's a small sample size and the cross-sectional nature makes it difficult to make inferences about the relationships in particular, except for the fact that we can base it in a theoretical model. And the recruitment sources, what I've always been interested in is when people reach a point where they no longer need to search. So when that uncertainty, they've kind of gotten to a point where it naturally resolves or they've accepted that that's what it will be like and I don't need to actively search for a diagnosis and a reason. And recruiting through advocacy groups and support groups, we're naturally going to capture the people who are still searching and looking and very involved in that particular community. So we are missing the portion of people who may have uncertainty, but don't view it as important for whatever reason. And I think the clinical implications kind of go to what we've spent a lot of time talking about today that often there aren't tests to offer and what we do in a session, we have to change the way we do that and help with these more persistent long-term problems such as this uncertainty and how our family's going to manage that on a longer-term basis. And I think one way we can help is obviously help identify resources such as SWAN and the other support groups that exist for them, but also help work to reduce these feelings of isolation and help them to find words around which they can talk about it or what's your major focus of your child and which areas do you think you need more support in? Is it the hearing loss or the epilepsy? What's the hardest part about it for you? Where do you feel most alone in it? And I think additionally we can work to increase coping efficacy certainly by helping parents identify where they've succeeded in the past. You've made it this far. What have you done to get this far and get to the office today? And to reframe appraisals about why they perhaps feel unable to cope with this uncertainty or what part of it is most difficult for them? And is there anything we can do to help them with that? So the future directions, I'd absolutely like to continue this research. I'm very interested also in how uncertainty changes when a diagnosis is present, but the diagnosis doesn't necessarily give you a strict course. Luckily, Megan, another student in the program is gonna take this on with Holly a little bit in the muscular dystrophy environment and see how those parents are experiencing uncertainty. I think after going through the qualitative data with Sam there's a lot more about uncertainty that we could explore further and help tease apart which areas are most salient and whether, in addition with longitudinal studies, when those areas become most salient and what changes along the course of a diagnostic odyssey. And then, of course, explore more personality traits and see if we can tease apart what types of people might be most receptive to certain interventions or where our intervention is going to work best and does that have to do with the type of person they are or the situation they're in. So, of course, thank you to the participants in my Barband Thesis Committee. Questions for Ellen? So, I am so interested in this finding of the social isolation and just want to make sure that I'm understanding correctly. So, you recruited from groups or people with kids that are undiagnosed and they still feel very isolated. Yes. I mean, that's just, that's really interesting. So, it's meeting some needs, but not all of their needs. It's absolutely interesting. For community, right? Yes. For community. Mm-hmm. Mm-hmm. Why are they competing? Right. And they feel that way, even though they're a part of a group. So, they have theoretically social organization. Mm-hmm. But isn't that kind of expected? Yeah, no. I'm not, I don't think it's terribly... I don't think it's terribly... There's someone who wants that and values that, but what they want is a matched experience. Right. That can't exist. That's what I think is interesting about this, is that they're obviously, it's not that surprising to me, I guess, is that they are seeking to have to identify with someone that these support groups are the best that's available to them, that is still not meeting their needs. There's huge unmet needs, even though they're parts of these formalized support structures. Right, I would agree that to an extent, these support groups aren't meeting the types of social support they were looking for. And I've had that experience recommending it to parents of kids in UDP who look at me like I'm crazy because there's a group, but everybody is different, so how would that be helpful to me? So what are your thoughts working in UDP about how perceptions of uncertainty might change over time for people who don't have a diagnosis? So assuming they don't get a diagnosis, do you think that the, and specifically related to how important it is to them versus the sort of what's uncertain? Yeah, I think they absolutely change over time and while I'd like to capture it while they're here, I think I'd probably be capturing their heightened need for a diagnosis if I talk to them and report about that while they're here for a week at NIH. But there are a few patients we see more longitudinally, and there's a patient here on a clinical trial who started in the Undiagnosed Diseases Program and we've diagnosed her enough to know that it's mitochondrial, but she still feels very much undiagnosed. And so I think it's very interesting, I think it does change over time and as certain issues become more salient, so patients for whom all of a sudden there is another medical emergency and other crisis, having that diagnosis at that point became more salient because it's hard to go into the ER and have to describe 16 pages of a medical record before you can be treated.