Living With the Burden of Pseudobulbar Affect: A Qualitative Analysis of the Effects of Education on Patient Experience

Abstract

Pseudobulbar affect (PBA) is associated with several neurological diseases and is underrecognized in clinical practice; however, PBA symptoms are often attributed to psychiatric or mood disorders rather than to neurological etiology. Until recently, there were no US Food and Drug Administration therapies approved for treating this condition, and there are currently few resources to support patients in the recognition and self-management of PBA symptoms. We evaluated the impact of a virtual education symposium on patient knowledge and self-efficacy via qualitative interviews. This evaluation of education impact provides unique insight into the experience of managing PBA symptoms; suggests that there is extensive need for educational resources to support patients with PBA and enable them to engage effectively with their providers; and affirms that online learning is an effective mechanism for delivering education to patients that enables them to more effectively self-manage symptoms in the context of chronic neurological conditions such as PBA.

Introduction

Pseudobulbar affect (PBA) is a condition that is associated with neurological diseases such as Alzheimer’s disease, amyotrophic lateral sclerosis, Parkinson’s disease, multiple sclerosis, stroke, posttraumatic stress disorder, and traumatic brain injury (TBI) (1 –3). Symptoms of PBA may also overlap with psychiatric disorders such as depression or anxiety and PBA is often mistaken as a psychiatric disorder. This debilitating condition affects an estimated 2 to 7 million people in the United States (4-6) and is characterized by unprovoked outbursts of uncontrollable, inappropriate laughter and/or crying that are unrelated, exaggerated, or out of proportion to the emotions felt by the patient or to the social context in which they are expressed (4,7,8). Pseudobulbar affect is underrecognized in clinical practice and PBA symptoms are often attributed to psychiatric or mood disorders rather than to neurological etiology (1,4). Although there is very little research on the burden of symptoms on patients with PBA, existing research indicates that the unpredictable and uncontrollable nature of PBA symptoms has a considerable impact on the social and emotional lives of patients and caregivers, often resulting in embarrassment, social withdrawal or isolation, and reduced work productivity and quality of life (9). Despite this burden, patient voices are seldom heard in relation to symptom self-management and aspects of living with PBA. Until recently, there were no US Food and Drug Administration (FDA) therapies approved for treating this condition (1,4), and there are also few resources to support patients in the recognition and self-management of PBA symptoms. In order to develop and provide such resources, PlatformQ Health Education, LLC, in conjunction with Albert Einstein College of Medicine and Montefiore University Hospital, created a virtual education program. We evaluated the impact of education on patient knowledge and self-efficacy via qualitative interviews.

Methods

Education Program Design and Delivery

Living with the burden of PBA: Education for Patients and Caregivers was designed to enable patients with PBA and their caregivers to adopt strategies for self-care that minimize the burden of PBA and improve quality of life. The learning objectives for the education were to enable patients and caregivers to (1) differentiate PBA episodes from other neuropsychiatric symptoms, (2) identify treatment options for PBA symptoms, and (3) adopt strategies to reduce PBA burden and promote quality of life. Accordingly, program content focused on defining and recognizing PBA, how to manage PBA symptoms, and resources to support self-care. The education program included a video-based streaming lecture presented by 2 PBA experts (one of the authors, E.P., and Maileen Ulep-Reed MSN, APRN) and provided time for participants to ask questions. The online program was broadcast live in a production studio on June 20, 2017, and remained on demand for 1 year, drawing a total of 820 participants. The program was hosted on a dedicated education page on the website of the National Stroke Association (NSA) as well as on the patient education site www.NeuroCarelive.com.

Evaluation of Knowledge, Competence, and Practice Change

We evaluated the impact of the education on patient knowledge and self-efficacy via qualitative interviews and explored contextual factors that may be important to knowledge uptake and behavior change via qualitative interviews with a sample of education participants. The interview topic guide (Supplementary Figure 1) was designed to explore patient and caregiver understandings about the goals of PBA treatment, strategies to live more comfortably with PBA, and current treatment options. One of the authors (A.H.) conducted confidential, 30- to 45-minute telephone interviews that were scheduled at convenient times for participants. We audio-recorded interviews via cloud-based, web-conferencing software (www.uberconference.com), transcribed interviews verbatim, and imported them into NVivo for Mac 11 (QSR International), a software package designed to support systematic analysis of unstructured data.

Participants, Inclusion/Exclusion Criteria, and Recruitment

Patients and caregivers were recruited to participate in the education via a digital marketing campaign. The program was promoted across Facebook and Twitter, and e-mails were sent to patients and family members of stroke survivors in the NSA’s support group, Careliving. The Stroke Center Network, a network dedicated to developing excellence in acute and rehabilitative stroke care, also advertised the activity to over 100 000 in the affected community via newsletters, e-mail, social media, and support groups. In total, 820 individuals participated in the educational program on NeuroCareLive and Facebook.

We recruited potential interviewees from the larger sample of education program participants; learners were invited to take part in voluntary interviews 6 to 10 weeks following the educational intervention. Participants were eligible to participate in interviews if they had a diagnosis of PBA or cared for a loved one with PBA, currently resided in the United States, and recalled viewing the education program on NeuroCareLive. We determined this eligibility via a screener e-mailed to education program participants. People who did not meet the inclusion criteria were not invited to participate in interviews. When screening for potential interviewees began, 132 participants had completed the education activity and 31 responded to interview invitations. Of these invitation respondents, 16 were eligible per inclusion criteria, and 13 were interviewed (3 did not attend for telephone interview at the designated date and time). A small honorarium was offered for completing an interview. Western Institutional Review Board granted ethical approval for the study and we obtained informed consent from all participants prior to interviews. None of the participants were personally known to the authors.

Qualitative Data Analysis

We used a process of constant comparison to structure analysis of participant responses to questions both across interviews and within interviews (10). One of the authors (A.H.) coded transcript content into descriptive categories that broadly followed the structure and focus of the interview categories concerning the education activity impact on self-reported knowledge and clinical practice. Following descriptive coding, a second round of coding identified themes across the data set until thematic saturation was achieved. These themes were validated by a second author (W.T.) who reviewed tables of text identified as evidence for codes and themes. The themes were further explored for connections and relationships (11).

Results

Interview Participants

We interviewed 13 people with PBA and caregivers for people with PBA (Table 1). All were stroke survivors or caregivers of stroke survivors; one participant also had TBI. Participants were actively seeking information on PBA due to ongoing symptoms and/or an interest in self-care strategies and so responded favorably to e-mail invitations.

Table 1.

Interview Sample Characteristics.

Variable	n	%
Age range (mean = 51)
35-45	3	23
46-55	4	30
56-65	3	23
≥66	2	15
Unknown	1	8
Gender
Male	4	31
Female	9	69
Role
Patient	8	62
Caregiver	5	38
Ethnicity
Non-Hispanic White or Euro-American	10	77
Latino or Hispanic American	1	7
Asian or Asian American (including East, South)	1	7
Unknown	1	7
Region
Northeast	2	15
Southeast	3	23
Midwest	6	46
Pacific	2	15

Low Physician Awareness of PBA

With one exception, all interview participants had a confirmed diagnosis of PBA, and for the most part, participants had initiated conversations about their symptoms with their physician, rather than vice versa. Participants painted a dismal picture of low physician awareness about PBA (Table 2). This was especially evident in primary care but was also a characteristic of patient interactions with neurologists. Allied health professionals were identified as being more aware of PBA symptoms and as having a role in prompting interviewees to talk to their physicians about their symptoms. Other factors that encouraged patients to talk with their physicians included direct-to-consumer (DTC) advertising about the FDA-approved PBA medication dextromethorphan/quinidine (DM/Q). Exposure to such advertising prompted 3 participants to talk to their physicians about persistent symptoms of crying for several years following stroke (mean = 8 years), and PBA symptoms following stroke were the trigger for the remaining participants to initiate discussion with their physicians.

Table 2.

Examples of Physician Awareness of PBA and DM/Q.

My GP had never heard of PBA before, and so I had to tell him what it was. The main thing has been the American Stroke Association. Physicians just basically said go there. Google PBA. I didn’t really get a ton of information. I probably got more information from my OT than I did from the physicians.

She [neurologist] basically didn’t really make a big deal about it. She said, “They’ve got some medicine out there that they think can help to control it somewhat. It’s really expensive, but if you want to try it, go ahead.”

She [PCP] was confused about my mom’s whole situation. She wanted to give her—she didn’t recognize the PBA either, which is really weird for a doctor. And that’s why I’m saying that people don’t know enough about PBA, including individuals in the medical field.

Not at all. Just to be mindful it may or may not be how she’s feeling. I don’t think I was given enough information about it. And I pretty much just had to go researching on like MayoClinic.com, just looking up different things.

Not much at all. She [PA] admitted to me that she didn’t know much about PBA, and she suggested that I go see a psychiatrist about it. And I said, “It’s my understanding that this is not a mental thing. This is like a neurological thing,” and she says, “Well, if you want to go see a psychiatrist, we can send you.” And so, I think there’s not as much understanding in the medical community as there should be about this condition.

I told the physicians I know they have [a new drug for] PBA, [and asked] if they are using it? The doctor said “we are already doing everything for your father, and we will give your father good care. So we’ll just do our best.”

I don’t know that he told me anything about what to expect. He just said I could take this medicine, and it would improve my condition.

[My HCP explained:] That the trauma to the brain, as a result of the stroke, affected my ability to produce serotonin as it had been able to before and that the medication would help to stabilize.

She explained that it was a condition that happens sometimes with people who have had some sort of a brain injury and that they can laugh and cry inappropriately. And it really has nothing to do with how they’re feeling at that moment. They just do it because it’s like a neurological response.

I asked my new doctor about it and she was a little more informed and maybe more aware, but she’s also an internal medicine specialist, and she said that she thought that I was suffering from PBA symptoms.

Abbreviations: DM/Q, dextromethorphan/quinidine; GP, general physician; HCP, healthcare provider; OT, Occupational Therapist; PBA, pseudobulbar affect; PCP, primary care physician.

A “New Normal”

The PBA symptoms represented a “new normal” for participants, which was characterized by “lack of control” in not only functionality but also social status. Participants described getting “funny looks” or embarrassed laughter from family members, and their experience of others’ responses was characterized by a general lack of understanding and even bullying (Table 3). Moreover, PBA symptoms disrupted established patterns of communication between parents and children, husbands and wives, and engendered considerable frustration for caregivers around deciphering the needs of their loved one.

Table 3.

Participant Experience of How Others Respond to PBA Episodes.

Early on, I would get some kind of funny looks from family members when I’d start to laugh and cry at the same time, and their eyebrows would go down and they’d look at me kind of funny. But I really don’t think anybody really notices it anymore. My wife does occasionally, but I think they’ve: (a) gotten used to it somewhat and (b) as I said it’s gotten better.

I’ve gone through bullying because of it because people don’t understand. They assume that you’re being—have emotional problems when you don’t. They don’t realize that it’s a neurological—it’s a physical condition, not an emotional condition.

Most people when I tell them, either one of two things happen. Number one, they laugh at me and say that it’s a commercial with Danny Glover where you just laugh a little bit, and a lot of people think that it’s not a real thing. Or, two, they look at me like I’m crazy and say, “Pseudobulbar Affect? What does that mean?”

People don’t understand it

My family, you know, “You’ve got to get a hold of yourself. You’re acting like you’ve got Alzheimer’s.

You don’t want to be at a meeting or something or at church or whatever, and then you all of a sudden start crying, or having dinner with someone and you’re crying. They start looking at you like, “What in the hell is going on?”

Abbreviation: PBA, Pseudobulbar affect.

It’s just so tiring, not being able to control your emotions. I just don’t think, at least my doctors, understand to the full extent of what it does. [Participant 7, patient]

Gender and PBA Symptoms

Some participants felt that crying posed a particular barrier to physician engagement with patients who have PBA, on the grounds that crying is often culturally viewed as a weakness. One participant, who was reluctant to take any medication to manage his symptoms, nonetheless noted that his physician had not offered him any treatment for PBA symptoms. He felt that this was because crying among men is viewed as culturally problematic.

None of the physicians I’ve been to have ever offered anything for PBA. I think it’s new to everyone, even though it obviously affects a lot of stroke victims. It’s just not—especially with men, you don’t want to deal with a bunch of crying men, maybe more with women. [Participant 5, patient]

Another participant, a caregiver, also noted the social and cultural challenge for men associated with excessive crying.

We’ve experienced that people are even afraid to touch a person that’s going through this. They can’t hold their hand. They can’t put their hand under their arm to make sure that they’re okay. If they choke during an episode of the crying or the laughing during a meal, people can’t get up and touch them. Women are more likely to help and to intervene. I don’t know if it’s because men are just the way they’re made, or they just don’t know what to do, or if they think they’ll embarrass their friend if they point it out. [Participant 8, caregiver]

Education Content as a Validation of PBA

Participants emphasized that exposure to education content validated their PBA diagnosis as a neurological versus a psychiatric condition and provided both a sense of hope and reassurance that participants were not alone in their experience of PBA. Depression was a common initial diagnosis for many participants, although they or their caregivers did not feel they were actually depressed. Yet depression, as one participant put it, “got in the way of an earlier diagnosis.” Even participants who acknowledged their depression following stroke felt they were able to distinguish between their symptoms of depression and PBA and were frustrated that their physicians could not make this distinction. The validation that the education content gave to the distinction between PBA as a neurological condition versus depression and other psychiatric diagnoses was vital to participants who were keen to dissociate themselves from psychological/psychiatric labels or associations (Table 4).

Table 4.

How Education Content Validated PBA: Distinguishing Between Neurological Versus Psychiatric Conditions.

I think having the knowledge to know that it wasn’t depression because that got in the way of the earlier diagnosis.

The main thing is it’s not psychological, so they think something’s wrong, you have a mental illness, and that’s what we have to get rid of right there because I think that carries a really bad stigma and that’s what you want to avoid.

I think that that video is something very important for anyone who has had brain trauma to view and caregivers to view to understand that it’s okay. It’s common, and there’s help for it.

After watching the program, I really like the section about the difference between depression and PBA because I don’t think psychiatrists totally get it either. Was I depressed after having the stroke? Yes. But the difference between depression lasting for a long period of time and PBA just instantaneously happening, I like that because I think more people in the psychiatric counseling—just doctors in general need to be educated more about this.

First of all, that I’m not crazy. And that may sound silly but it’s very true. I thought at first I was the only person in the world and I was, that was going through this. And it was a problem unique only to me.

Just to manage the stress, that it can be mistaken for depression most times when they’re totally separate. I definitely understand that because I understand depression. I’ve been there before. That is strictly neurological, which I get that.

The idea that it’s a result of an injury or something to the brain. It’s more specific. And, plus, when they talked about what areas of the brain and stuff, it pertains to my stroke because where my stroke was it affected, what, the frontal something? Frontal lobe, or wherever the emotions are housed. And so, it makes total sense now, and I wish somebody would have told me, “Since you had this kind of stroke, it’s possible you’re going to have this kind of thing going on. And if you do, this is how we fix it or treat it or whatever.”

I think to explain to him, I guess to explain there is one disease and it’s called PBA and maybe it’s not depression.

Abbreviation: PBA, pseudobulbar affect.

Dextromethorphan/Quinidine

Prior to education exposure, 6 participants were aware of DM/Q via DTC advertising. This awareness prompted them to seek further information about treatment via the Internet and likely primed them to be receptive to the invitational e-mail for this study. The participants with no reported prior awareness of DM/Q were able to recall education content about DM/Q. Overall, participants were open to information about treatment options and, following participation in the education activity, were aware that a medication specific to PBA could be of benefit for themselves or the loved ones they cared for. Despite seeing this potential benefit, most participants were reluctant to consider DM/Q as PBA symptom therapy. Polypharmacy was the most common barrier to considering DM/Q, as participants did not wish to add another drug to their already complex regimens. Some were also concerned about drug–drug interactions, and one participant felt that her current treatment (venlofaxine) already adequately reduced her symptoms. Cost was a barrier for remaining participants.

I want to explore more with the different types of meds that they talk about. Now, I am concerned about cost because my retirement is not great, I mean financially wise. And so I’ve got to come up with a regimen plan that I can afford. [Participant 6, patient]

Self-Care Strategies: Acceptance, Hustling Through, and Distraction

Prior to education exposure, participants managed their PBA symptoms by “accepting” symptoms as part of their stroke experience or intuitively adopting strategies such as changing position or distraction. Half the participants talked about trying to accept or adapt to PBA symptoms as part of the “new normal” and described themselves as “hustling through,” “hanging in there,” or just “getting through” episodes of laughing or crying.

We’re trying to get through it. We try to make it light because we try to get through it at that moment. [Participant 2, caregiver]

Participants described avoiding public situations or social occasions because, as one participant observed, she felt that her symptoms would “ruin” the experience of others who were present in the same space.

A lot of times I wind up having my episodes in front of people, which I would prefer not to. I’d prefer to do it in private. A lot of times I avoid situations where I know that a PBA may come in or on. I have avoided funerals, weddings, anything—movies, any situation where I think it’s going to be quiet or if I feel like one may come on, then I just avoid it altogether. [Participant 7, patient]

The remaining participants felt that movement or distraction strategies were effective in helping them defray the intensity of PBA symptoms and redirecting the course of symptoms. These strategies were intuitive for participants and rooted in their personal experience. Simply, they worked. Education content also reinforced the value of what participants were already doing and validated their self-care behaviors.

It validated my thinking of having him take a deep breath and relax and move. I didn’t realize that was really something you were really supposed to do, but it was just the nurse in me, trying to get somebody to calm down and do whatever, that kind of seemed to help him. And it just kind of validated my thinking. [Participant 11, caregiver]

Following education exposure, all participants were able to identify a range of new distraction or relaxation strategies that they would be willing to try as ways to manage their PBA symptoms (Supplementary Table 1).

Discussion

Participants’ knowledge and skill concerning self-management strategies increased following program participation and were maintained at follow-up (Supplementary Table 2). This study upheld observations from other studies that PBA is typically underrecognized in clinical practice, and symptoms are often attributed to psychiatric or mood disorders rather than to neurological etiology (1,4). While some studies suggest that patients with PBA tend not to report their symptoms, participants in our study described scenarios in which they were typically driving conversations with their health-care providers about PBA. Indeed, at times, participants felt that they knew more about the topic than their providers, and it seems likely that DTC advertising enabled participants to initiate these conversations. In contrast, studies on clinicians knowledge affirm the varied terminology that is used in clinical practice to describe PBA symptoms, which might reinforce confusion around diagnostic criteria for PBA and undermine clinician confidence in asking patients about PBA symptoms in the setting of associated neurological conditions (1,12).

We asked participants if there were elements of the education that were especially effective. In addition to describing faculty as “approachable,” “friendly,” or “knowledgeable,” they most often identified specific areas of content as validating, reassuring, and knowledge boosting. For instance, following education exposure, participants were able to describe a range of questions that people should ask their physicians when they are diagnosed with PBA as well as practical self-care strategies and tips to share with PBA patients and their caregivers to help them manage symptoms (Supplementary Tables 3 and 4). Interview responses suggest that exposure to information about distraction and relaxation strategies in particular boosted their confidence in self-care. Both patients and caregivers also highlighted the need for additional education directed toward caregivers who are overwhelmed by the needs of caring for family members with stroke and neuropsychiatric sequelae, such as PBA. Finally, in the absence of physician engagement, participants turned to friends and family for support. However, participants felt that it was often incumbent on them to educate their own networks about PBA. For this reason, participants felt that the education further raised their own awareness about PBA and that they could share this acquired information with others (ie, patients/caregivers, family, friends, and physicians).

Participants also emphasized that educational content confirmed their experience of the social and emotional burden of PBA symptoms and had the effect of normalizing this stigmatized condition for them by emphasizing its neurological versus psychological etiology. Participants were relieved to find that there was a biological, and therefore, “legitimate,” basis for their symptoms. Education also raised awareness of the therapeutic value of treatment and reinforced the potential for treatment in the future. Following education exposure, participants were more aware of the advantages of DM/Q in reducing the frequency of symptoms. Although the additional burden of taking another medication on an already heavily loaded pharmacy regimen represented a barrier to treatment, participants said that learning about newer treatment options would equip them to initiate a conversation about medication with their providers in the future.

Limitations

Participants self-selected to seek further information about PBA through this education activity and described themselves as aggressive researchers on this topic. Therefore, interview participants may be atypical of patients with conditions associated with PBA. The qualitative sample size was small, as is conventional in qualitative research. However, qualitative analysis does not typically require large sample sizes to yield meaningful insights across a range of perspectives (10).

Conclusion

Analysis of interview data showed that learners emphatically endorsed the relevance of the education as a resource to help them improve their own symptom management. This unique qualitative insight into the experience of managing PBA symptoms suggests that there is extensive need for educational resources to support patients with PBA and enable them to engage effectively with their providers. Self-management support, such as patient education, is associated with small but significant health outcomes improvements in conditions such as diabetes, asthma, coronary heart disease, and depression (13). This evaluation affirms that online learning is an effective mechanism for delivering education to patients and providing health-related information and resources that enable them to more effectively self-manage symptoms in the context of chronic neurological conditions such as PBA.

Author Biographies

Wendy Turell, began her work in health care as a public health researcher and administrator for New York City public and private hospitals, and has focused on CME/CE outcomes evaluation and research since 2003. Dr. Turell holds a doctorate from the Columbia University Mailman School of Public Health and an MS in Psychology from University of Pennsylvania.

Anne Roc, has focused her career on the development and management of medical education for health care providers and patients, mainly in neurology, psychiatry, oncology and primary care. She completed a combined BS/MS program in neuroscience and biology from Brandeis University in Waltham, Massachusetts, and earned a doctoral degree in neuroscience from the University of Pennsylvania's School of Medicine in Philadelphia.

Erik Pioro is Director of the Section of Amyotrophic Lateral Sclerosis and Related Disorders at Cleveland Clinic, Cleveland, Ohio, where he specializes in the care of patients with ALS and other complex neurodegenerative motor neuron disorders and runs clinical trials to find effective treatments for these diseases. He is also professor of Neurology at Cleveland Clinic Lerner College of Medicine of Case Western Reserve University, and Barry Winovich Endowed Chair in ALS Research at the Lerner Research Institute.

Alexandra Howson, trained in qualitative methodologies at the University of Edinburgh. She has conducted qualitative research concerning a range of clinical challenges in oncology, cardiology, neurology, endocrinology, and HIV as well as evaluation of continuing education programs.