Abstract
Background:
This study aims to understand diagnosis communication experiences and preferences of youths with functional seizures and their parents.
Methods:
Semistructured interviews with youths and their parents from a tertiary care children's hospital were conducted separately. We confirmed the diagnosis of functional seizures with the youth's treating providers. All interviews were audio recorded and professionally transcribed. Two coders performed thematic analysis and determined themes.
Results:
Thirteen youths (aged 12 to 18) and 13 parents completed interviews separately. Themes included the following: (1) both parents and youths reported substantial fear about their symptoms, diagnosis, prognosis, and treatment options; (2) good communication skills highlighted included providing reassurance, listening, and allowing time for questions, with different preferences between youths and parents in the mode of communication; (3) poor communication for both youths and parents included use of medical jargon, brevity of communication especially with the youth, minimization of youth's symptoms, lack of apparent compassion, and lack of written materials to supplement discussions; (4) perspectives on the role of mental health in functional seizures were heterogeneous, yet concrete treatment plans were desired including access to behavioral therapy; and (5) youths and parents wanted practical guidance including plans for school and how to respond to functional seizures in all settings.
Conclusions:
Youths and parents provided concrete recommendations for optimal diagnosis communication. Study findings will inform the future development of an educational intervention to improve communication and create a supportive environment for youths with functional seizures.
Keywords: Psychogenic nonepileptic seizures (PNES), Thematic analysis, Adolescents, Parents/caregivers, Diagnosis communication, Perceptions, Therapy
Introduction
Functional seizures (FS), otherwise known as psychogenic nonepileptic seizures, involve transient alterations in behavior, consciousness, and sensation that are associated with acute or chronic biopsychosocial stressors. Although these events may resemble epileptic seizures, they are not caused by epileptiform hypersynchronous neural activity. FS are prevalent in adolescence, representing about 25% of all admissions to the inpatient epilepsy monitoring unit.1,2 The prevalence in adolescents is estimated to be higher than that in adults at 60 in 100,000 people between ages 15 and 19 years compared with 33 per 100,000 among adults. There is a 20% rate of comorbidity between patients with FS and co-occurring epilepsy. About 70% to 80% of patients diagnosed are female.2 Children or adolescents with FS often have frequent emergency visits or hospitalizations when misdiagnosed or when they misunderstand their diagnosis.3,4 Adolescents experience academic and social strain due to frequent FS, including missed school days. Adolescents may also experience associated psychiatric comorbidities, such as depression, anxiety, and somatoform symptoms, including stigmatization on behalf of medical providers such as accusations of faking symptoms.3-6 When compared with children without FS, those with the condition report a poorer quality of life. Additionally, individuals with FS may have been treated unnecessarily with antiseizure medications when cognitive behavioral-informed therapy (CBiT) is currently considered the gold standard of treatment.7
Diagnosis communication and delivery is essential to promoting positive outcomes including remission of FS, particularly by linking patients to treatment resources in behavioral health.2,8,9 Patient acceptance of the diagnosis has been associated with marked reduction in health care utilization, whereas lack of acceptance was associated with increased health care utilization.2 Consequently, effective, credible communication of a diagnosis of FS is paramount to optimize therapeutic outcomes. Although there are many expert opinions on how to deliver the FS diagnosis, there are few tested interventions that currently exist, especially in adolescents. In fact, there is only one evidence-based intervention for how to communicate this diagnosis to adult patients and one for pediatric patients based on a Norwegian qualitative study. This Norwegian study explores the use of a biopsychosocial approach, where the patients were helped to identify their own biological, social, and psychological conditions as precipitating factors for FS.10,11 It is unknown if the adult study is generalizable to children or if the Norwegian study is generalizable to the US context.
In this study, we aimed to understand the diagnosis communication experiences and preferences of adolescents with FS and their parents in the United States, using qualitative methodology. Our objective was to gain insight into what constitutes patient- and family-centered communication about a diagnosis of FS to optimize care including diagnosis acceptance and linkage to appropriate behavioral health treatment.
Materials and Methods
We recruited individuals aged 12 to 18 years with a diagnosis of FS and their parent/guardian. Recruitment was conducted through the neurology outpatient clinics, the epilepsy monitoring unit, inpatient ward service, and behavioral health clinic at a tertiary care center. At our institution, neurologists are typically the first providers to communicate the diagnosis and often refer to behavioral health providers embedded in our department who specialize in FS treatment. Clinicians known to potential participants approached them and asked if they would like to hear about the research study. With permission, the study team contacted them, and if they were interested, they obtained informed consent/assent. Inclusion criteria for youth participants entailed age (12 to 21 years old), diagnosis of FS confirmed by their neurologist, and sufficient proficiency in English to complete a telephone interview without translation services. Inclusion criteria for parent participants entailed being the parent or guardian of a study-eligible youth and sufficient English language proficiency to complete an interview. Youths and their parents each received a $25 gift card following their interviews.
Two pediatric neurology residents (H.P., R.M.) and one pediatric epileptologist (L.K.) with training in qualitative methodology conducted the interviews with the youth and parent/guardian separately. Interviews lasted between 20 and 60 minutes. During the interviews, we asked questions regarding experiences receiving the diagnosis, including opinions and reactions about how it was communicated and influenced the therapeutic trajectory. We also inquired whether they were asked about mental health and how they perceived questions about mental health during the initial encounter. Additionally, we asked about their recommendations on ideal communication of diagnosis.
We developed an initial codebook reflecting potential categories of participant experiences and recommendations. We performed iterative coding on the transcripts using a thematic analysis approach.12 Using Dedoose software, two coders (R.M., S.B.) coded the deidentified transcripts. The coders used the preliminary codebook deductively and inductively added further codes as needed. The coders convened periodically throughout the study to discuss and reconcile their coding, resolve discrepancies, and create new codes using a consensus coding approach. A senior coinvestigator (L.K.) was available to resolve any discrepancies in the coding process. The coders then used the final codebook to analyze the transcripts, evaluate emerging themes, and select representative quotations. The University of Pittsburgh Institutional Review Board approved this study.
Results
Thirteen youths diagnosed with FS and their parents participated in the interviews. A total of 26 interviews were conducted with 13 youths and 13 parents separately. Youth ages ranged from 12 to 18 years (median age 14 years). Table 1 describes the youth and parent demographics, including age, gender, and age of youth at diagnosis. Several themes emerged related to the experiences and diagnosis communication related to FS, which are summarized in Table 2.
TABLE 1.
Participant and Parent Demographics
| Youth # | Current Youth Age |
Youth Age at Diagnosis |
Patient Race/Ethnicity | Youth Gender | Setting of Diagnosis (Neurology Clinic vs Hospital) |
Treatment—Was Patient Started on Therapy After Diagnosis Was Made? |
|---|---|---|---|---|---|---|
| 1 | 14 | 10 | White | NB | Neurology Clinic | Yes |
| 2 | 13 | 12 | White | W(C) | Hospital | Yes |
| 3 | 13 | 13 | White | W(C) | Hospital | Yes |
| 4 | 15 | 15 | White | W(C) | Hospital | No |
| 5 | 17 | 17 | White | M(T) | Neurology Clinic | Yes |
| 6 | 18 | 15 | White | W(C) | Hospital | No |
| 7 | 13 | 12 | Black/Hispanic | W(C) | Hospital | Yes |
| 8 | 17 | 7 | White | M(C) | Hospital | Yes |
| 9 | 15 | 14 | White | M(C) | Neurology Clinic | No |
| 10 | 14 | 13 | White | W(C) | Hospital | Yes |
| 11 | 12 | 12 | White | M(C) | Hospital | Yes |
| 12 | 16 | 15 | White | W(C) | Neurology Clinic | Yes |
| 13 | 12 | 12 | White | W(C) | Neurology Clinic | Yes |
| Parent # |
Parent Age |
Relationship to Youth |
Parent Race/Ethnicity | Parent Gender | ||
| 1 | 47 | Mother | White | W(C) | ||
| 2 | 38 | Mother | White | W(C) | ||
| 3 | 42 | Father | White | M(C) | ||
| 4 | 48 | Mother | White | W(C) | ||
| 5 | 47 | Parent | White | NB | ||
| 6 | 47 | Mother | White | W(C) | ||
| 7 | 46 | Mother | Black/Hispanic | W(C) | ||
| 8 | 42 | Mother | White | W(C) | ||
| 9 | 41 | Mother | White | W(C) | ||
| 10 | 51 | Mother | White | W(C) | ||
| 11 | 39 | Mother | White | W(C) | ||
| 12 | 51 | Mother | White | W(C) | ||
| 13 | 33 | Mother | White | W(C) |
Abbreviations:
M(C) = Man (cisgender)
M(T) = Man (transgender)
NB = Nonbinary
W(C) = Woman (cisgender)
TABLE 2.
Common Themes
| Theme | Representative Quotations |
|---|---|
| 1. Both parents and youth reported substantial fear about the youth's symptoms, diagnosis, prognosis, and treatment option of CBiT. | |
| a. Fear of presenting symptoms as being seizures |
[Parent 2]:
“Originally, it started as her falling over, shaking all over and it appeared like an epileptic seizure, but then it turned into her hallucinating and seeing evil things. She was running out of the house, searching for her father who wasn't involved in her life anymore” [Parent 3]: “She got hurt at school a couple times. It was, sort of, like they grabbed her and knocked her to the ground. It was very concerning, watching videos of what was going on when she was having them at school.” [Parent 4]: “We thought she was having a stroke or seizures.” [Youth 1]: “I was horrified I was dying… I didn't know what was happening. I was scared, and I didn't really know what was going on.” |
| b. Fear of not knowing what to do in the situation and feeling helpless |
[Youth 5]:
“I had a seizure where I couldn't move for 36 hours; it was awful and very scary… a lot of them would leave me incapacitated, and not being able to like control my body.” [Parent 6] “I think it was scary for her because she had gotten hurt a couple of times, and being told that there wasn't anything we could do immediately to help stop it was scary for her.” |
| c. Frightened that there is no medication or treatment for the condition, equated to the perception that patient may have this condition permanently |
[Parent 1]:
“Oh, gosh. Is she going to go on medication? Like-how are we going to get this under control? How are we going to keep this from happening” [Parent 2]: “How is she going to go to school?” Is there any way to cure this? [Youth 5]: “There is no medicine for it, like its going to be around forever, I think that would be kind of the scariest thing to hear.” |
| d. Counterpoint: many felt relief of having a diagnosis and a small subset reporting relief that diagnosis was not epilepsy |
[Parent 7]:
“I was also kind of relieved because I had a name to it now and it wasn't just an unknown condition.” [Youth 6]: “I was happy it wasn't epileptic because I've helped my sister get through her seizures.” [Parent 8]: “We do realize that it's not brain-based, necessarily. His brain is not affected in the same way that epilepsy would.” [Youth 8]: “I feel like they [parents] were relieved that it wasn't as serious [as epilepsy].” [Parent 2]: “We were thrilled. I felt like, I could calm down. We were told it wasn't affecting her brain.” [Youth 6]: “I was pretty happy to have a diagnosis.” |
| 2. Good communication skills highlighted during the interview included providing reassurance, listening, and allowing time for questions, with different preferences between youth and parents in the mode of communication. | |
| a. Good communication skills liked by both youth and parents |
[Youth 1]:
“I think everyone was polite, nice and open about the subject.” “…the bedside manner was very good. Overall, I've always had a very good experience…everyone was very sweet and had my best interests in mind.” [Parent 2]: “The neurologist was very calm. She was easy to talk to. She just put me at east with saying it wasn't epilepsy.” [Parent 8]: “The doctor was very good at listening and seeing him [the patient] as the expert, rather than just being like you are just the kid.” [Parent 3]: “They [medical team] were very clear, empathetic and straightforward.” [Parent 4]: “To have it come from a medical professional really made me accept that this is what it is and this is what I need to do to help my daughter through this.” [Youth 4]: “The doctors [at the hospital] actually listened to me. They took time and tried to figure out what was going on.” [Youth 1]: “They made me feel really safe.” [Youth 2]: “They did give me a printout of some typical PNES seizures.” |
| b. Differences between skills liked by parents and youth |
[Parent 6]:
“The best example was one that she gave me, explaining that some people get stomachaches and headaches when they're stressed out. So [my child's] body responds differently. That helped me understand it better.” [Parent 2]: “She just broke it down more. She gave me (parent) an example related to a computer and hardware and how it works, related that to the brain and how the brain is sending incorrect signals to the rest of his body.” [Youth 1]: “Having written materials or pictures can be helpful to understand how nervous systems function and is impacted by stressors.” |
| 3. Poor communication for both youth and parents included medical jargon, minimization of youth's symptoms, lack of apparent compassion, brevity of communication, and lack of written materials to supplement discussions. | |
|
[Youth 5]:
“Explaining a lot of words to younger kids, so that way they understand it more.” [Youth 7]: “The first doctor told me that I was faking it, basically.” “[second doctor] weren't like dismissive the way the first doctor was.” [Parent 8]: “Seeing him more than one time to understand where he is and who he is.” [Parent 2]: “I think she handed me a pamphlet on therapy but did not really explain what NEE was… I feel like there wasn't a lot of guidance in what to do moving forward.” [Parent 5]: “Sometimes you'll get a doctor that comes in and is extremely intelligent and they talk above everybody. I think that is a big turnoff, feeling like they're not necessarily communicating to a person but they're talking about them, not with them.” [Youth 3]: “I think they dealt with my parents really well, but I feel like it would have been a lot nicer for someone to sit down with me and talk about it too.” [Youth 4]: “I liked how she went into depth of explaining it and my fifth-grade self didn't appreciate that enough. But now, my eight-grade self understands better.” [Youth 9]: “Nothing really stressful was happening at that time. If I did have something super stressful happening, I would've felt uncomfortable revealing what it was, especially in front of parents… I think she could have told my family to step outside to talk to me.” [Youth 10]: “My doctor was straight to the point… Iwas honestly scared of her… be nice and understanding of how kids must be feeling at the time of diagnosis.” [Parent 9]: “I think having a sheet or overview of diagnostic criteria and treatment would have been nice.” |
|
| 4. Parents' and youths' perspective on the role of mental health in FS was very heterogeneous, yet they wanted concrete treatment plans including access to behavioral therapy, although reported several barriers. | |
| a. Parents’/youths’ discussion of mental health |
[Parent 8]:
“That [asking about mental health] made me feel good. It was more personalized, it made me seem like she really cared about her.” [Parent 2]: “It helps communication go a lot better…clue them into what stressors are going on in people's lives.” |
| b. Both parents/youth wanting concrete next steps after diagnosis |
[Parent 9]:
“Seeing him more than one time to understand where he is and who he is.” [Parent 5]: “Give a little more information about how it may or may not affect your daily life, what you should do if you start to going into an episode.” [Parent 6]: “…maybe suggesting there's a support group or this is a place that has a lot of information that could be helpful for families. Just so I know where to start to help figure it out.” [Youth 10]: “Just if there was a website that I could join or a group of people that would help each other out.” |
| c. Barriers to treatment (behavioral therapy) referrals |
[Parent 3]:
“The one thing I didn't like is there was a long wait for her get into cognitive behavioral therapy.” [Parent 10]: “They said, “she needed therapy, but then nobody wanted to take her…” [Parent 9]: “Giving appropriate list of referrals for therapists. There wasn't a lot of guidance in how to manage it… but when I started digging in, I found different resources and then we started working on therapy.” |
| 5. Youths and parents wanted practical guidance including plans for school and how to respond to FS in all settings. |
[Parent 1]:
“Having some written materials can be helpful because a lot of things can be talked about very quickly and can be difficult to remember afterwards.” [Youth 1]: “…probably having some type of resource, pamphlet, on what is a non-epileptic event or something basic…” [Parent 5]: “I think they should've definitely focused on how he felt about it, what he needed to do and what he would need for school to help him navigate this way through this.” [Parent 7]: “I think having something that might be an overview of diagnostic criteria, what this means for treatment and next steps, would have been nice.” [Parent 6]: “I don't think there's enough information out there for our schools… I guess having more resources for what the kids are experiencing.” “What should I do specifically or tell the school to do… we talked about to make sure he is in a safe place.” [Parent 4]: “Notifying her school staff members and her teachers, so we can setup a plan to when she has an episode.” [Parent 2]: “What we needed to do about her being in school. Can she lead a normal life? Can she drive? Can she still do sports?” [Youth 2]: “Make sure they know if driving is going to be affected by it because that was one of my biggest concerns that I wasn't gonna be able to drive.” |
Abbreviation:
CBiT = Cognitive behavioral-informed therapy
Theme 1
Both parents and youths reported substantial fear about symptoms, diagnosis, prognosis, and treatment option of CBiT, with a minority reporting relief at having a diagnosis.
One theme was fear related to the presenting symptoms, diagnosis, treatment, and prognosis. Parents and youths both expressed panic and alarm with the onset of FS. For example, Parent 2 described what her daughter's episode was like. She stated “it all started as her falling over, shaking all over and it appeared like an epileptic seizure, but then it turned into her hallucinating and seeing evil things…she was running out of the house, searching for her father who wasn't involved in her life anymore.” Parent 3 expressed worry about physical harm, specifically mentioning “It was, sort of, like they grabbed her and knocked her to the ground… it was very concerning, watching videos of what was going on when she was having them at school.”
Parents also expressed how upsetting it was to see their child go through an episode with one parent (Parent 4) commenting on their child's episode looking like “a stroke or seizure.” Many parents felt helpless, not knowing what to do or how to help when their child had an episode. In addition, the youths were very frightened by the episodes, especially when they lasted for “30-45 minutes or hours in duration” and left them “incapacitated” (Youth 1). As Youth 5 pointed out, “I had a seizure where I couldn't move for 36 hours; it was awful and very scary.” Specifically, they felt scared when they were at school in anticipation of episodes occurring in public.
Many parents and youths expressed fear, including whether the FS might be lifelong and how FS will impact the youth's quality of life. Several parents wondered “how is she going to go to school? Is there a way to cure this?” (Parents 1 and 2). Some parents and youths were also upset at the fact that there is not a pill to treat the condition immediately or were afraid about pursuing CBiT for treatment. Despite the recommendation for CBiT, several parents felt like there were no treatments and were concerned: “There wasn't a clear thing that what I was supposed to do with her… there was no answer [sic]” (Parents 4 and 5). For others, the absence of medication was particularly upsetting: “There is no medicine for it, like it's going to be around forever, I think that would be kind of the scariest thing to hear” (Youth 3).
Despite most youths reporting prevailing experiences of fear, some expressed relief that they finally had a diagnosis after months or years of trying to find an answer. Similarly, a minority of parents were relieved to discover that their child's episodes were not epileptic seizures. As Parent 2 related after discovering the diagnosis of FS, “We were thrilled. I felt like I could calm down. We were told it wasn't affecting her brain.”
Theme 2
Good communication skills highlighted included providing reassurance, active listening, and allowing time for questions, with different preferences between youths and parents in the mode of communication.
There were several aspects of diagnosis communication that parents recognized and appreciated from neurologists. Some included talking in simple language that both parents and youths could understand, having a gentle bedside manner, taking time to listen to the young person and family's experiences, and allowing time for questions. Another theme was that parents and youths appreciated when the health care provider (HCPs) reassured them that FS were not related to epileptic seizures and would not result in serious damage to the brain. Additionally, Parent 8 emphasized how “the doctor was good at listening, to seeing him [the youth] as the expert” rather than being dismissive of their concerns.
Parents and youths highlighted different communication skills as being helpful. Some parents appreciated analogies. For example, Parent 6 appreciated how the neurologist provided an analogy of their child's symptoms to having a “stomachache or headache” and FS as being the physical manifestation of the stress the child was experiencing. Parent 2 appreciated the analogy of “computer and hardware and how it works, related that to the brain and how the brain is sending incorrect signals to the rest of his body.” A few parents liked and understood the concept of FS better with analogies, whereas one parent felt that such comparisons could be dismissive of the underlying condition (Parent 3). He said, “The doctor's like, ‘my stress comes out I have to go poop. Your daughter's stress comes out, her right side goes numb.’ I'm like, that's a pretty big difference in my mind. But, hey, you're the doctor.”
There were no youths who specifically mentioned that the analogies were helpful. However, several wanted concrete neurobiological explanations for why FS occur. For example, Youth 1 suggested using a diagram to “show how the nervous system is connected and how it causes the episodes.”
Theme 3
Poor communication for both youths and parents included use of medical jargon, brevity of communication, especially with the youth, minimization of youth's symptoms, lack of apparent compassion, and lack of written materials to supplement discussions.
Most parents and youths expressed wanting to talk in simple terms without the use of excessive medical jargon. As Youth 5 noted, they wanted their HCP to “explain a lot of [medical] words to younger kids, so that way they understand it more.” Some youths received explanations with terminology that any child at their age would not be able to understand. For example, as Youth 4 mentioned, “I liked how she went into depth of explaining it, but my fifth-grade self didn't appreciate it at that time.” Youth 7 wished that their doctor would “speak in words that I would know” and not use “big words.”
Parents and youths felt that their encounters with HCPs were very short and inadequate to contribute to their understanding of the diagnosis. Most youths mentioned feeling confused or left the hospital without a clear understanding of their diagnosis. Youths reported that HCPs spent the most time trying to explain the new diagnosis to the parents but did not take time to directly talk with them. Nevertheless, many parents also reported feeling confused about the diagnosis. Most parents and youths reported not understanding the diagnosis enough to ask questions during the initial encounter.
A common subtheme was parents and youths being concerned about the symptoms being minimized or dismissed. Parents wanted to hear that the FS are “real” and that their children are not “faking it.” Parent 7 brought up that they sought a second opinion from another neurologist because they felt that their original neurologist “dismissed” their concerns and thought that their child was “faking it.”
Perceptions of doctors minimizing or dismissing symptoms were at times linked to confusion around the relationship of FS to stress and psychiatric diagnoses. Many parents and youths particularly reported confusion about being told that the condition was “stress-related” —and they were especially confused if the youth was not apparently experiencing acute stressors during their FS. Parents and youths perceived this message as a minimization or poor fit for their circumstances, especially in youths who did not have any prior mental health conditions or youths who did not report feeling acutely stressed or anxious.
Additionally, both parents and youths voiced the need for a “written resource” or “pamphlet” on FS to be provided during the initial diagnosis encounter as a reference. Parent 2 mentioned receiving a “printout” on therapy but still did not fully understand the diagnosis of FS.
Theme 4
Perspectives on the role of mental health in FS were heterogeneous, yet concrete treatment plans were desired including access to behavioral therapy.
Although youths and parents at times did not appreciate HCPs connecting FS to stress, they were largely accepting of HCPs taking a psychiatric history, including asking about any prior diagnoses, symptoms, or stressors. When asked about their experience on being questioned about their child's mental health, most parents thought such questions were appropriate. Most youths also agreed that mental health should be discussed in the initial diagnostic communication, with some caveats. For example, Youth 6 reported feeling stigmatized by being asked about their current mental health. Youth 9 reported that they would have been more comfortable sharing their stressors, if the “doctor had asked parents to step outside the room” and they were questioned in private. Youth 8 brought up that they might have preferred answering questions about depression and anxiety via a written questionnaire rather than verbally, similar to screening tools used at their pediatrician's office. Mostly, youths and parents felt that the providers “genuinely cared” by asking questions related to the mental health.
Despite parent and youth concerns about providers connecting FS to stress and mental health, they were also largely open to learning about CBiT as a treatment option, although (as stated in Theme 1) they also had apprehensions about behavioral health treatment. Parents were very serious about wanting concrete, actionable plans with next steps for connecting their children to behavioral therapy. However, some youths voiced frustration about needing therapy while understanding its importance.
Parents also reported barriers to connecting their child to CBiT. In some cases, parents reported long wait times for behavioral health appointments and a lack of behavioral health providers in certain geographic areas. In other cases, parents reported that the diagnosing clinician did not provide referrals at all. Others felt that they were given broad referrals without a clear sense of where to start and how to approach therapy. Still others reported that the referred behavioral health professional subsequently informed them that they were not able to treat FS.
Theme 5
Youths and parents wanted practical guidance including plans for school and how to respond to FS in all settings.
Parents specifically asked questions about practical guidance on how to manage an acute event at home and school and generally reported not receiving enough information at the time of diagnosis. As Parent 7 suggested, the providers should have “given a little more information about how [functional seizures] may affect their child's daily life, what you should do if they have an episode.” Parent 4 mentioned it would have been helpful to have resources and guidance on how to “notify school staff members and teachers about a plan for when she has an episode.” Most parents felt teachers and school staff members lack knowledge about FS and did not know how to handle episodes. Youths often reported feelings of “shame” or “embarrassment” during school when they had episodes. Youths disliked being treated differently than their peers and wished for normalization of their condition.
Recommendations
A summary of concrete recommendations for diagnosis communication and clinical care extrapolated from study findings are detailed in Table 3.
TABLE 3.
Recommendations for Diagnosis Communication of FS to Youth and Families
| Phase | Recommendation |
|---|---|
| Initial diagnosis disclosure | • Allot significant time to explain and discuss the diagnosis • Listen to youth and family experiences • Express compassion and use a gentle bedside manner • Understand that youth and parents are experiencing fear and stress • Inform them that the FS are “real” and not “faked” • Communicate the diagnosis in clear, simple language to both the family and youth as well as the youth individually/separately • Understand that family and youth have different developmental communication needs (i.e., analogies may work for family but not youth, who may need more concrete explanations) • Reassure them that cognitive behavioral-informed therapy is effective • Reassure them that the prognosis is good • Allow time for questions |
| Discussion of mental health and cognitive behavioral-informed therapy | • Normalize discussion of mental health as part of routine medical care • Inquire about the youth's mental health and stressors, but consider doing so privately with the youth alone and asking if they prefer to speak verbally or complete written questionnaires like the PHQ-2 or GAD-2 • Convey that FS can be linked to stressors, although also state that up to one third of the time there is no identifiable stressor or mental health condition • Maintain a list of appropriate referrals of behavioral health providers willing to treat FS to share with youth and families in your catchment area |
| Written resources | • Provide written resources to family and youth at a developmentally appropriate level • Provide a written explanation of the diagnosis and action plan for use at home and in schools • Go over the written resources during initial diagnosis communication • Consider including a concrete diagram of the nervous system and its connection to FS • Provide vetted referrals for behavioral health treatment as described above |
| Follow-up appointment | • Schedule a follow-up appointment with pediatric neurology for verbal reinforcement of the diagnosis and opportunity for further questions |
Abbreviations:
FS = Functional seizures
GAD-2 = Generalized anxiety disorder 2-item
PHQ-2 = Patient health questionnaire 2
Discussion
In this qualitative study of diagnosis communication of FS, youths and parents reported wanting simple, straightforward explanations and concrete plan for management, including both appropriate connections to therapy and guidance for treatment of the condition in daily life. Youths specifically desired inclusion in the conversation and additional time from providers to explain the diagnosis to them, perhaps using a more concrete neurobiological approach referring to the interaction of the brain, body, and mind. Parents endorsed analogies as an effective means of communication, although it is not clear that these strategies are appreciated by youths. Most youths and parents appreciated reassurance that FS do not have the same complications and effect on the brain as epilepsy. Many of the poor experiences highlighted minimization of the diagnosis as “just stress related” without any further explanation.
Qualitative studies in adult literature describe ambivalent emotional responses to receiving an FS diagnosis including relief, confusion, anger, and despair.13,14 In contrast, youths and parents in our study highlighted fear as a more prevailing emotion. There is evidence to suggest that adults with FS may prefer somatic to psychological explanations, interpreting the diagnosis as a problem in their brain's ability to manage conscious or subconscious stress or trauma.9,13-15 Based on our study findings, it is not clear that this explanation resonates with children and families, who may prefer explanations that are less tied to conceptions of stress or trauma.
There are few tested interventions for diagnosis communication of FS. For example, there is one prospective multicenter study in adults. This study looked at the acceptability and effectiveness of a communication intervention consisting of a patient information leaflet. This study interviewed 50 adult patients diagnosed with FS who were informed of the diagnosis by 10 different neurologists using a communication procedure that included an information leaflet. About 94% of the patients found the leaflet easy to understand and 94% found their questions answered by the doctor.11 The information leaflet was created by expert opinion only (without input from patients or other key stakeholders), and although effective when tested in adults, one could question whether it might have been further optimized if it had been developed with input from patients.11,14 The participants of our study provided key insight on specific information that they would like to be included in a written resource.
A practical implication, as implied by the participants of our study, would be for neurology clinics to develop individualized pamphlets with local resources as well as links to helpful web sites. However, another adult qualitative study performed thematic analysis of written accounts of experiences in living with FS and demonstrated that simply providing a pamphlet or web site is not enough to bridge the gap of communication between the clinician and family.6 Consequently, written resources must be paired with evidence-based, high-quality verbal counseling about the diagnosis at both the time of diagnosis and ideally also at an outpatient follow-up visit. The potential need for an outpatient follow-up visit to reinforce diagnosis communication is suggested by our study finding that most parents and youths were confused about the diagnosis after the initial encounter.
There is only one qualitative study concerning diagnosis disclosure to pediatric patients, which explored the impact of using a biopsychosocial approach to explain the diagnosis to pediatric patients based in Norway. When comparing our study to this study, there were key similarities and differences in themes. The Norwegian study identified three key themes of threatened selfimage with perception of diagnosis through fear, participants being suspected by their HCPs of staging their seizures, and continued doubts about their diagnosis after getting an adequate explanation.10 Common themes in both studies were related to the parents’ and youths’ fear of diagnosis and perception of minimization of diagnosis by HCPs. The key difference was that some participants in the Norwegian study identified connections between their personal histories and their seizures and became seizure free. In our study, most youths did not endorse having a solid explanation from which they could relieve their doubts and identify a link between their seizures and past stressful experiences. In addition, a key distinction is that the results of this Norwegian study may not be transferrable to the United States.
An actionable clinical implication of this study's findings was the need for a structured resource to be utilized in different settings such as school. Another qualitative study evaluated adolescents' experience of attending school with FS with an overwhelming negative response from the participants. The main themes identified were negative experiences emerging from stress, bullying, accusations of “faking” seizures, feeling left out because of the condition, and school management of FS.16 Similarly, our study identified themes of adolescents and youths feeling more afraid and stressed about school after discovery of diagnosis and feeling apprehensive about how to manage FS in school without clear and appropriate guidance from health care professionals. In addition, the results of this study inform the need for a structured resource, written or verbal, that will provide guidance to the teachers, school staff, and peers on actionable steps to mitigate the youths' suffering from FS.
Parents and youths with FS provided valuable insight into the positive and negative aspects of diagnosis communication from their experiences. Their experiences may inform future pediatric neurologists on how to communicate this diagnosis with reassurance and provide appropriate therapy referrals. Next steps include developing and implementing an educational intervention, such as a simulation for neurologists to practice using simple and effective communication skills for diagnosis delivery. Other future interventions include the creation of a written resource for parents and young individuals to supplement diagnosis communication as well as formation of action plans for episodes and a tool for communicating those plans to schools.
Limitations of this study include that qualitative research aims for in-depth understanding of a phenomenon in a limited sample with elicitation of a heterogeneity of perspectives. Further surveys with a larger sample size across various institutions are needed to clarify differences in preferences, which can be stratified by age of the youth. This research may be limited by sampling bias, although it is unclear if participants who are the most satisfied or dissatisfied may be more likely to participate. Another study gathering data on perspectives of the diagnosis nomenclature would provide more insight in improving communication. Additionally, we recognize that our study only included youths and parents who were able to speak the English language proficiently, and therefore, findings may not be transferrable across different cultures and linguistic groups. The main demographic population in our study was white and female gender, hence the lack of minority group perspectives may limit the generalizability of the study. Last, the time from diagnosis to their interview for this study varied significantly among the youth, which may impact recollection.
Conclusion
Youths with FS and their parents provided recommendations including positive and negative examples of diagnosis communication techniques. Most parents desired a clear explanation with concrete next steps for treatment. Many youths wanted more inclusion in the initial diagnosis conversation and desired explanations through varying methods such as through pamphlets or diagrams. With variable nuances in the preferred techniques of diagnosis communication, a common aspect was the need for collaboration among the youths, their parent(s), HCPs, and other key members, such as school staff or teachers, to create a supportive environment for treatment of FS. This study's findings will inform the future development of an educational intervention to improve pediatric neurologists' communication of a diagnosis of FS to adolescent youths, parents, and caregivers.
Funding:
This work was funded by Department of Pediatrics, University of Pittsburgh. Dr. Kerr's research time is funded, in part, by the American Academy of Neurology, American Epilepsy Society, Epilepsy Foundation, and American Brain Foundation. Dr. Harris's research time is funded by the National Palliative Care Research Center. Dr. Kirkpatrick was partially supported by a grant from the Child Neurologist Career Development Program (grant # 5K12NS098482-07).
Footnotes
CRediT authorship contribution statement
Himadri Patel: Writing – review & editing, Writing – original draft, Validation, Methodology, Investigation, Formal analysis, Data curation, Conceptualization. Ryan Malave: Writing – review & editing, Writing – original draft, Supervision, Methodology, Investigation, Formal analysis, Data curation. Sydney Bitting: Writing – review & editing, Methodology, Investigation, Formal analysis, Data curation. Kaycee Weir: Writing – review & editing, Resources, Data curation. Taylor Naus: Writing – review & editing, Resources, Data curation. Levi Shelton: Writing – review & editing, Validation, Supervision, Conceptualization. Kelly W. Harris: Writing – review & editing. Wesley T. Kerr: Writing – review & editing, Visualization, Validation, Supervision, Resources, Conceptualization. Anne C. Van Cott: Writing – review & editing, Visualization, Validation, Supervision. Laura Kirkpatrick: Writing – review & editing, Writing – original draft, Visualization, Validation, Supervision, Resources, Methodology, Investigation, Funding acquisition, Formal analysis, Data curation, Conceptualization.
Declaration of Competing Interest
Dr. Kerr writes paid review articles for Medlink Neurology on topic; is a paid consultant for SK Lifesciences, Biohaven Pharmaceuticals, Cerebral Therapeutics, UCB, EpiTel, and the Epilepsy Study Consortium; and has collaborative or data use agreements with Eisai, Janssen, Radius Health, GlaxoSmithKline (GSK), Johnson & Johnson, and Jazz Pharmaceuticals. The remaining authors have no conflicts of interest to declare. We confirm that we have read the Journal's position on issues involved in ethical publication and affirm that this report is consistent with those guidelines.
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