Introduction

Deep brain stimulation (DBS) is utilized to treat movement disorders, including pediatric refractory dystonia under an FDA Humanitarian Device Exemption [1, 2]. The growing use of DBS in children raises several important ethical questions and concerns [3, 4]. The role that hopes and expectations play in decision-making is a key ethical issue. Do caregiver decision makers have realistic expectations? Does hope inspire or cloud good decision-making in this context? Do clinicians’ and caregivers’ views on these matters differ? How ought clinicians respond to high hopes in the clinical setting without quelling them or permitting “false hopes” that might compromise informed decision making? A better understanding of these issues is vital for understanding informed decision-making process about deep brain stimulation for children.

When a patient hopes for a certain outcome, they desire it, believe the desired outcome is possible (probability greater than zero), and tend to engage in certain behaviors such as praying for the outcome, thinking about it or fantasizing about it—even planning for it [5]. Unrealistic optimism, on the other hand, occurs when a patient has a desire for a certain outcome and overestimates the probability of the desired outcome [5,6,7,8,9]. Similarly, denial occurs when a patient has a desire for an outcome and fails to think about or “face” the high probability of the undesired outcome [5, 7, 10, 11]. Self-deception occurs when a patient has a desire for a certain outcome and actively lies to herself or tells herself a narrative about the probability which is in tension with actual evidence but in line with the desired outcome [5, 10, 12]. There is also the phenomenon of “false” hope, which occurs when a patient has a desire for a certain outcome and believes it is possible when in fact it is not possible. Related concepts include “unrealistic hope” and “self-deceived hope” [5]. Although these latter terms include the word “hope,” their defining essence is unrealistic beliefs. One important thing to note about these definitions is an implicit distinction between cases of hope vs. unrealistic optimism/denial/self-deception. In the case of hope, although a person may respond to unfavorable probabilities in very positive ways, she still possesses an accurate understanding of them [5, 10]. In cases of unrealistic optimism/denial/self-deception, on the other hand, there is something awry with the person’s beliefs about the probabilities [5, 13].

Understanding the existence and impact of hope and unrealistic optimism in caregivers is integral to analyzing clinical interactions and enabling shared decision-making. Understanding the ranges of unrealistic hopes as well as their impacts on clinical decision-making can better inform clinician-patient interactions.

Methods

Participants and Procedures

Semi-structured interviews were conducted with clinicians (n = 29) working in DBS programs treating children and adolescents with dystonia as well as caregivers (n = 44) to understand stakeholder perspectives on informed consent and decision making about pediatric DBS. An interview guide was developed based on issues raised in the clinical and ethics literature and through discussions with DBS and movement disorders experts. A draft of the guide was reviewed by an advisory team and piloted with a neurologist specializing in movement disorders, resulting in minor changes for improved clarity. The study was approved by the Institutional Review Board at Baylor College of Medicine. Interviews were conducted via phone or Zoom, lasting an average of 50 min.

Data Analysis

Interviews were audio-recorded, transcribed verbatim, and analyzed using MAXQDA 2018 qualitative data analysis software. Team members (led by KK-Q, a qualitative methods expert and medical anthropologist) developed a codebook to identify thematic patterns in clinicians’ and patients’ responses to questions about hope and unrealistic optimism [14]. Team members (KK-Q, JBB, LS, and YK) all coded 5 transcripts together to ensure inter-coder reliability in application of the codebook and then LS and YK independently coded each interview transcript. With oversight from KK-Q and JBB, LS and YK then iteratively abstracted the coded information and identified thematic patterns across interviews utilizing thematic content analysis. ND calculated final theme and sub-theme frequencies and was responsible for primary data analysis.

Results

A total of 56 clinicians working with pediatric patients with dystonia specializing in pediatrics or having substantial experience with pediatric patients were contacted, and 29 participated (response rate = 52%) (Table 1). A total of 44 caregivers of children receiving DBS for dystonia were contacted (Table 2). We first present the thematic findings from clinician interviews followed by those from caregiver interviews.

Table 1 Clinician characteristics
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Table 2 Caregiver Characteristics
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Clinician Views on Hope and Unrealistic Optimism in Pediatric Deep Brain Stimulation

Clinicians said that because DBS for pediatric dystonia is a relatively new, experimental treatment with variable impacts on dystonia symptoms in pediatric patients, they worry about the potential for unrealistic hopes to impact caregiver decision making, as described below.

Concerns about Unrealistic Expectations and False Hopes

The most common unrealistic hope that clinicians reported seeing in caregivers was that DBS will be a “cure,” with a third of all clinicians interviewed explicitly mentioning that many caregivers hold this unrealistic hope. One clinician explained that some caregivers believe that DBS will completely heal their child and enable them to do things that they were unable to previously. Some parents go so far as to say that they believe their child will be “reborn” through DBS (see Tables 3 and 4).

Table 3 Clinician views on hope and unrealistic optimism in DBS
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Table 4 Caregiver views on hope and unrealistic optimism in DBS
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A majority (86%, 25/29) of clinicians expressed concern that caregivers may have false hopes that may lead caregivers to overestimate the potential benefits and therefore inadequately assess the risk/benefit ratio. One clinician explained that realistic expectations are important for meaningful engagement in the informed consent process. However, some (21%, 6/29) clinicians noted that the rise in access to videos and testimonials on social media websites depicting significant improvements for other dystonia pediatric patients has increased false hopes among some caregivers.Footnote 1 One clinician explained that caregivers see videos that depict patients with significant improvements, which leads them to believe that DBS will solve all the patient’s problems (see Tables 3 and 4). Another clinician said caregivers can also be swayed by results from DBS for adults with Parkinson’s, explaining that the caregivers who view these videos assume that DBS will have the same outcomes for dystonia patients.

Another unrealistic expectation that clinicians cited is that caregivers believed DBS will be a simple procedure rather than acknowledging the complexity of the treatment process. Multiple clinicians expressed that many caregivers believe that DBS is a single operation procedure, and neglect to factor in the fact that DBS will require multiple programming appointments. If caregivers are under the impression that their child will only need one surgery or be miraculously cured by DBS, they may overlook the potential complications and number of surgeries their child will require. Further, one clinician explained that caregivers “need to be prepared that there can be complications and if it gets infected, which is the most common complication, then the device is going to come out… they need to be mindful that this is going to indicate multiple surgeries over their lifetime” (C_Dy_029).

Concerns about Last Resort Options and Desperation

Many (68.9%, 20/29) clinicians pointed out that most pediatric candidates for DBS have refractory conditions and their caregivers and patients are considering DBS as a last resort. Clinicians note that most patients who enter the DBS program have exhausted all other known clinical options. As one clinician explained, dystonia patients who are eligible for DBS are very sick, so caregivers view DBS as a “Hail Mary” (see Tables 3 and 4). The clinician then explained that this can lead to unrealistic optimism, explaining that it is difficult to dissuade caregivers from holding this view.

Patients and families may also arrive frustrated with the lack of progress in their clinical care to date. One clinician explained that the frustration that caregivers feel can lead to desperation and limit adaptive decision-making on the part of the caregivers. Desperate parents can beg clinicians for DBS and can get angry if the clinician believes that DBS would not be helpful (see Tables 3 and 4). In these scenarios, clinicians feel that caregivers are irrational because their evaluations of DBS are colored by emotion over reason.

In deciding treatment course, clinicians try to evaluate how desperate caregivers are (see Tables 3 and 4). Clinicians said that in some cases this desperation can lead to a deterioration of clinician-parent trust and communication. One clinician recounted a situation wherein the caregivers overestimated the potential benefits of DBS and began to believe that the physicians were not providing good care for their child.

Another concern about this being a last resort option is that caregivers and patients may be disappointed if their unrealistic hopes remain unmet. One clinician explained that some caregivers are so desperate that there is a significant psychological and emotional impact to poor DBS outcomes. Another clinician stated that this psychological impact may be even worse for patients than for caregivers (see Tables 3 and 4).

The Practice of Setting Low Expectations to Combat Unrealistic Optimism

To combat unrealistic optimism and decrease unrealistic expectations, 68.9% (20/29) of clinicians expressed that they intentionally try to lower parent expectations about DBS outcomes (the practice of “laying crepe” as we expand on in the discussion). Clinicians emphasized the importance of managing expectations. One clinician stated that clinicians have to be very clear that DBS results are mixed and uncertain (see Tables 3 and 4).

In order to help clinicians set good expectations, one clinician expressed that they would significantly underestimate the chances of success so that clinicians would engage in good decision-making (see Tables 3 and 4). Another clinician agreed that most DBS clinicians are harsh in their estimation of potential outcomes. The clinician explained that when the family is engaged in the decision-making process, the clinician sets low goals for treatment outcomes (see Tables 3 and 4). Clinicians also expressed that downplaying potential outcomes was important because there is little clinical information about how patients will do post-DBS. One clinician explained that because there is a lack of information about potential treatment outcomes, caregivers should not be setting high or low expectations.

Some Caregivers are Too Pessimistic, Overweigh Risks, and do not Have Enough Hope

A smaller percentage of clinicians (34.5%, 10/29) expressed the opposite concern that some caregivers may be too pessimistic about DBS and over-estimate potential risks, which may dissuade patients and families from proceeding. Clinicians expressed that caregivers have misconceptions about DBS and perceive it to be overly dangerous and risky without significant benefits (see Tables 3 and 4).

Many clinicians noted that there were significant consequences of undue pessimism—it could prevent patients from accessing treatment. One clinician gave the example of a case wherein a patient was a perfect candidate for DBS but never got DBS as the caregivers perceived surgery to be too dangerous. Another clinician expressed frustration, stating that there are some cases wherein the child is a perfect candidate for DBS, but the caregivers are too scared to consent to brain surgery for a young patient (see Tables 3 and 4). The clinician elaborated that the patient will sometimes suffer for years before caregivers will consent to surgery. These families are so focused on risks, and neglect to consider the potential benefits of DBS.

Caregivers’ Views on Hope and Unrealistic Optimism in Pediatric Deep Brain Stimulation

Realistic Hopes and Expectations for DBS

A majority of caregivers (73%, 32/44) reported that contrary to clinician concerns they had realistic hopes about DBS. The most common hopes expressed by caregivers included a better quality of life for the child (45%n 20/44), for the child to have [some of] their “life back” (32%, 14/44), for the child to regain independent skills (32%, 14/44), and for the child to gain the ability to walk or run (34%, 13/44).

While caregivers acknowledged that they had high hopes for the potential outcomes of DBS, they explained that their expectations were realistic. One post-DBS caregiver explained that they had set realistic expectations while maintaining high hopes (see Table 5). Many caregivers (60%, 26/44) had hopes for small improvements: these included hopes for a decrease in dystonic storms, controlling chorea, improved sleep, increased confidence, and for DBS to make feeding the child easier. Achieving minor improvements in quality of life for both patients and caregivers was the central hope for most caregivers. One caregiver explained that even small improvements to patient functioning were important (see Table 5).

Table 5 Patient demographics
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Another parent explained that they simply hoped that their child would experience some decrease in pain (see Table 5). Another caregiver noted that they were hoping for a miracle cure, but also accepted that DBS would likely only help with symptom management (see Table 5). High (even “unrealistic”) hopes could coexist with realistic expectations for outcomes.

DBS Simply had to be Done—Choice (and Hope) was Irrelevant

Caregivers (77%, 34/44) expressed that DBS essentially had to be done because it was their last option. Some caregivers expressed that the patients wanted to get DBS due to the significant impact on quality of life and ability to engage in normal activities (see Table 5).

Some caregivers reported that their child’s condition had progressed so far that they felt they had no other choice. One caregiver expressed that they were desperate to get DBS in order to increase patient independence. Another caregiver expressed that DBS progressed so quickly that the caregivers felt they had no other option (see Table 5). One caregiver stated that clinicians had stated that DBS was the last option for the patient, which led the caregiver to ignore potential risks. In their view, hope and choice did not play much of a role.

Expectation Setting for Self/Child can be Difficult

Caregivers (54.3%) expressed that they struggled setting outcome expectations due to the uncertainty of DBS. One caregiver explained that variability and complexity of the disease made expectation setting difficult. Caregivers also cited the lack of information about childhood dystonia as another issue making expectation setting difficult. The fac that DBS was an experimental treatment with uncertain outcomes complicated decision-making and made expectation-setting impossible. Because even clinicians could not provide estimates for potential outcomes with high levels of confidence, caregivers felt ill-equipped to set expectations (see Table 5).

One caregiver explained that they wished the clinic had given more information for what to expect during DBS surgery and programming in order to help caregivers set better expectations (see Table 5). Another caregiver explained that because DBS is situated as a miracle cure, it can be difficult to set realistic expectations about DBS outcomes (see Table 5). This highlights the need for realistic expectations from clinicians to help facilitate goal setting for caregivers.

Unmet Expectations can Result in Disappointment

There was a large number (43%, 19/44) of caregivers who expressed negative feelings post-DBS treatment due to unmet expectations. caregivers expressed that difficulty in setting expectations was one factor leading to disappointment. One caregiver explained that the family expected the child would begin walking and running. When those expectations were unrealized, family members were unhappy (see Table 5). Multiple caregivers expressed that they had high hopes for how DBS would impact patients, and they felt disappointed post-intervention when those expectations were unmet (see Table 5).

Caregivers also expressed that the patients experienced disappointment with outcomes as well. One caregiver stated that their child believed DBS would be a complete cure, but the patient did not internalize this and was disappointed that they had undergone a complicated procedure for imperfect results (see Table 5). While caregivers tried to set realistic expectations with patients, they were not always successful, which could lead to disappointment and other negative outcomes among patient populations.

Another factor leading to disappointment was the length of time that side effects from DBS lasted. One caregiver explained that post-DBS, the child would display delays in processing and replying to questions, which was a surprising outcome (see Table 5). Lack of expectation setting and management regarding outcomes and side effects of DBS leads caregivers to feel disappointed with outcomes that they otherwise would be happy with.

The finding that 54.3% of caregivers expressed that they struggled setting outcome expectations and 43% of caregivers expressed disappointment with outcomes post-DBS highlights the need for clinicians to set and manage expectations throughout the treatment process.

The Benefits of Hope

While physicians viewed caregivers’ unrealistic hopes and expectations as potentially having negative consequences, caregivers described that maintaining high hopes – even if potentially unrealistic – could still be beneficial for them. For example, caregivers said that hoping for the dramatic improvements in dystonia symptoms portrayed in certain of the educational videos allowed caregivers to overcome their fear of potential risks. One parent explained that seeing a child who received DBS and who was now able to run marathons gave her “the hope I needed to move forward” with DBS treatment (see Table 5). Another caregiver expressed that viewing videos of patients who had significant improvements post-DBS was necessary in order to consent to DBS.

Experiences with Clinicians Setting Low Expectations

Many caregivers (43%, 19/44) had experiences with clinicians who seemed to intentionally try to set low expectations about the potential benefits of DBS (described earlier as “laying crepe”). Many caregivers expressed that the clinician warned that they were unable to guarantee any specific positive outcomes. One caregiver explained that the clinician emphasized DBS may only slightly improve patient symptoms (see Table 5). Multiple caregivers explained that clinicians explained that DBS had uncertain outcomes and clinicians could not guarantee significant improvements. Caregivers expressed that clinicians seemed to intentionally lower their expectations from the outset to decrease instances of disappointment.

Some caregivers felt that the clinician laying crepe helped them make a more informed decision. One caregiver explained that it was better to know potential realistic outcomes than be ignorant (see Table 5). However, laying crepe sometimes led to caregivers unnecessarily delaying DBS. One caregiver expressed that the clinician laying crepe made them nervous about deciding to consent to DBS, explaining that the over-emphasis on risks of DBS was scary and led them to put DBS off (see Table 5). The overly-negative expression of potential risks and benefits scared some caregivers from utilizing the intervention earlier in the disease course.

Discussion

There has been previous research on suggested and clinician-utilized attempts to manage caregiver hope and optimism within DBS which has found that clinicians attempt to showcase the uncertainty about potential outcomes for patients. [15, 18] Our findings build off of these data: we found that clinicians worried that caregivers developed unrealistic expectations for DBS outcomes in a way that could compromise informed consent, and thus attempted to temper caregiver hopes intentionally setting low expectations and/or emphasizing the uncertainties involved (a practice called “laying crepe”). The phrase “laying crepe” or “hanging crepe” is used in medical literature (often surgery) to refer to the practice of providing pessimistic and dismal portrayal of outcomes, often to avoid patient disappointment. The origin of the term comes from funeral history—undertaker assistants would hang black cloth (crepe) over the windows of homes of the deceased and over their casket—these assistants were called “crepe hangers” [19]. The clinicians that we interviewed reported that they believed the practice of laying crepe would help caregivers develop more realistic expectations and thereby aid in good decision-making. However, many caregivers considered themselves to indeed have high hopes for DBS for their child but counterbalanced those hopes with realistic expectations. What are we to make of this practice of clinicians laying crepe to counter potential unrealistic expectations or false hopes by families during decision making?

One issue with laying crepe is that, instead of enabling quality decision-making, it can be a barrier to caregivers engaging in an accurate risk-benefit analysis. Clinicians have an obligation to facilitate informed consent and help patients make informed medical decisions. Understanding the risk-benefit profile of medical interventions is critical for informed consent (and assent in children to the extent appropriate) as well as establishing realistic expectations of treatment response. While clinicians engaged in laying crepe with the intention of improving decision-making, this practice can undermine the process of informed consent.

On the other hand, clinicians engage in laying crepe in order to prevent disappointment. The concern that caregivers will be disappointed is warranted, as 50% (14/28) caregivers expressed disappointment with outcomes post-DBS. However, feelings of disappointment with outcome are not exclusive or incongruent with caregiver’s making good, informed decisions.

Given this information, it is clear that there are both benefits and drawbacks of clinicians laying crepe. Among caregivers that have high hopes and unrealistic expectations for DBS outcomes, laying crepe may be a necessary precaution for clinicians to take in order to ensure good decision-making on the part of caregivers. If caregivers believe that DBS will be a miraculous cure, they are unlikely to be engaging in an accurate and informative risk–benefit analysis. Clinicians could be right to engage in laying crepe in situations like this to help facilitate good decision-making and prevent potential decisional-regret post-intervention. However, it appears that among most caregivers pursuing DBS, laying crepe may be an unnecessary and even ethically dubious practice, as the practice may pose a barrier to effective decision-making. If caregiver ability to accurately evaluate the risks and benefits of DBS is disrupted by laying crepe, clinicians should change their risk communication methods accordingly. Furthermore, if clinicians engage in the practice of laying crepe among caregivers who already have unrealistically pessimistic expectations for DBS outcomes, clinicians can unintentionally disincentivize caregivers from pursuing a helpful intervention.

Importantly, laying crepe is not always a strictly negative endeavor. For example, with some forms of dystonia, DBS is significantly less effective. In these cases, clinicians have an obligation to help patients and caregivers understand that the benefits of DBS may be limited. In these cases, engaging in the practice of laying crepe can better prepare caregivers for realistic outcomes. However, given that DBS is only offered for patients with refractory dystonia and is a last-resort treatment, many caregivers already have low expectations for outcomes. This was reflected in our results showing that most caregivers self-reported realistic expectations for DBS outcomes.

Limitations

Empirically evaluating the effects of unrealistic hope and optimism in the case of DBS for pediatric refractory dystonia is complicated and multi-faceted. One limitation of our study was that it is difficult to parse whether caregiver self-reports about levels of realistic hope and optimism. While 74% of caregivers viewed themselves as having high hopes but reasonable expectations, this self-report may be inaccurate. Firstly, caregivers’ self-perceptions of accuracy of expectations is subjective. This self-perception can be influenced by biases, such as hindsight bias, making such self-reports inaccurate. Even caregivers who had overwhelmingly realistic expectations still voiced a mix of some unrealistic expectations as well. This included caregivers who stated that they had hopes for a miraculous cure while simultaneously hoping for very minor patient improvements, such as the patient sleeping better.

Another major challenge in differentiating unrealistic optimism from realistic hope is that some patients and caregivers may outwardly present themselves as overly optimistic in communication with physicians. This could be due to concerns that others may prefer or expect them to remain positive, or due to fears of ‘jinxing’ themselves (Blumenthal-Barby and Ubel). Indeed, this may be what was happening with our 74% of caregivers who viewed themselves as having high hopes but reasonable (even low) expectations for DBS when the clinicians (85%) worried they had false hopes. As a result, clinicians may end up laying crepe with caregivers who actually hold realistic expectations for DBS. If laying crepe negatively skews previously realistic expectations, then the practice may compromise the informed decision making of caregivers about DBS for their children.

Finally, our research was focused on the perspectives of the legal decision-makers (i.e., clinicians and caregivers) on realistic versus unrealistic hope and optimism. One area for future research is to include the perspectives of pediatric patients. Given that patients have insight into their own disorder, it would be beneficial to elicit their perspectives.

Conclusion

Understanding caregiver and clinician perspectives on hope and optimism in decision-making is vital for implementing novel neurotechnologies in clinical settings [20, 21]. Many of the concerns that clinicians expressed about false hope and unrealistic optimism distorting caregivers informed decision making about DBS for their child were minimized by caregivers. While clinicians expressed concerns that caregivers would have unrealistic hopes about DBS outcomes, 74.3% of caregivers reported that they believed their hopes were realistic and that they often hoped for small changes. Clinicians expressed concerns that unrealistic hopes might be generated by social media, and conversely, caregivers reported that social media improved their support network and enabled them to make difficult healthcare decisions. Caregivers felt that high hopes paired with realistic expectations enabled them to make difficult healthcare decisions. Collectively, these data highlight the need for ongoing and thorough discussion between clinicians and stakeholders to set realistic expectations for child health outcomes.