Sample
Thirty-three physicians and 22 caregivers participated in four physician and three caregiver focus groups, respectively (Table 1). The children of the 22 caregivers were 55% male, and included infants, children, preadolescents, and adolescents equally.
Evidence
Focus group analysis identified 12 core themes influencing physician decision making around neurotechnological interventions for DRE in pediatric patients and eight for caregivers (Table 2). The final attributes included in both DCEs, reflecting the most significant choice drivers, differed slightly between groups in number and wording. Their derivation is now described.
Table 2 Physician and caregiver themes and attributes Attributes Included in Both DCEs
A number of attributes were common to both DCEs, reflecting the themes identified from both stakeholder focus groups; however, the attributes and levels were worded to fit the intended audience in consultation with physicians and caregivers, respectively. Table 3 includes quotes expressing physician and caregiver perspectives on these attributes.
Table 3 Quotes describing physician and caregiver perspectives on attributes included in both DCEs Chances of Seizure Freedom
Physicians were concerned with the efficacy of the intervention and the chances that their patients would achieve a clinically significant reduction in seizure frequency. The limited evidence on relatively new neurotechnologies was cause for concern, as their goal was to see their patients live a more seizure-free life.
Caregivers reported significant concerns about the quality of life of their children and how it could be improved by intervention. The definition of quality of life in their decision making was driven by whether their child(ren) could actually be free of debilitating seizures and able to live a normal life with a good social network.
Risk (Major and Minor Risk)
Physicians discussed their concerns through a number of themes: risk, safety, mortality, and invasiveness. Physicians described considering new technologies only if they were safe, proven to have very low mortality rates, and would provide some level of health improvement. They also noted that they had to weigh and communicate to caregivers the risk of an intervention based on the invasiveness of the surgery. Additionally, they were aware of both the risk associated with the intervention and the risk of not intervening, namely, ongoing uncontrolled seizures, which they considered to be significant. These themes were considered to be sufficiently captured under the ‘risk’ and ‘chances of seizure freedom’ attributes. The risk theme was split into two attributes describing minor and major risks, to sufficiently capture the complexity of the risk considerations, to provide clarity on the specifics of the risk factors, and for a better understanding of the trade-offs people make between benefits and risks of different severity and the likelihood of occurrence.
For caregivers, balancing risk and benefit was also a salient factor they considered, they recognized the risk of doing nothing and described making choices based on whether the intervention was more or less risky compared to the natural history of uncontrolled epilepsy. The themes of risk described by caregivers were also split into two attributes to replicate the question in the physician DCE, to provide consistency, and to allow for a more direct comparison.
Availability of Evidence
The ‘source of information’ theme captured physician concerns about the source, quality, and trustworthiness of evidence supporting possible interventions. With novel surgical interventions, where rigorous studies such as clinical trials are usually limited, physicians considered anecdotal evidence useful when it came from respected colleagues with significant experience and expertise in well-regarded institutions. This theme was linked with ‘evidence’ and described as ‘availability of evidence’.
Caregivers also considered the available evidence and were interested in knowing the types of studies and the health outcomes of patients who had received the interventions they were considering for their children. Worries about being a test case or the first to have a procedure was also raised by caregivers.
Cost to Families
This attribute described as the ‘financial burden to families’ in the physician DCE was based on our thematic analysis showing that physicians were aware of the possible direct financial cost families may incur in caring for children with DRE, along with the logistical burdens of transport and travel to receive specific interventions in larger centers, often far from home. These issues factored into the physician decisions about whether they would recommend certain interventions.
For caregivers, cost was also a salient concern, given the significant costs associated with these interventions and the complicated nature of insurance coverage, especially in the US focus groups. Caregivers in the USA were especially worried about the implications of a loss of employment on access to high-quality medical insurance to care for their children.
Attributes Included Only in Physician or Caregiver DCEs
A small number of attributes appeared to be only key drivers of decisions for either physicians or caregivers (Table 4).
Table 4 Quotes describing physician and caregiver perspectives on attributes Availability of Intervention at Institution Level (Physicians)
Physicians were very aware of the features of novel neurotechnologies and how to deliver them to patients. However, they noted that the availability of the intervention within their institution would determine their ability to deliver or recommend an intervention, especially because referring patients to other centers could mean increasing the financial burden (because of time lost at work, travel, accommodation) on caregivers.
Access to a Multidisciplinary Care Team (Caregivers)
Caregivers described a need for additional support outside their epilepsy surgery teams in caring for their children with DRE, and often expressed a desire for access to other healthcare professionals, which ranged from counsellors to support their children with the psychological aspects of their disease to social workers for support with other aspects of life.
Invasiveness (Caregivers)
Caregivers frequently discussed concerns about the level of invasiveness of procedures and how novel technologies seemed less invasive and therefore favorable. Conversations from multiple focus groups suggested that preferences for interventions could be influenced by the degree of invasiveness.
Themes that did not Translate to Attributes
Under the ‘burden to family’ theme, physicians expressed concerns about travel costs for caregivers and their ability to complete the treatment process. As this theme appeared to be inextricably linked with cost, this theme contributed to the ‘financial burden on family’ attribute.
‘Pressure to innovate’ was primarily discussed by American physicians, but not by their Canadian counterparts. This theme related closely to the ‘availability of intervention at institution level’ attribute. Similarly, physicians discussed ‘family preferences’ as a theme, describing feelings of being burdened or pressured by the desires of the patients’ family but ultimately they did not believe that this strongly influenced their recommendations or decisions.
We also identified themes such as ‘trust in physicians,’ ‘involving the child (patient) in decision making,’ and ‘appropriateness of treatment’ for an individual. Whilst important, these were either prerequisites to deciding about any type of treatment (‘appropriateness’; ‘trust in physician’), or were inherent to the physician-patient relationship and unlikely to vary between treatments (‘trust in physicians’; ‘involving the child’).
Synthesis/Interpretation
Guided by our core question “does this drive decision making?”, we worked to identify themes that directly affected decision making and varied between options, and then described those key concepts in a minimum set of attributes (Table 5). Overall, physicians prioritized improving the quality of lives of the patients by reducing their seizures, but in a way that would not leave caregivers facing serious financial pressures. Caregivers were similarly focused on long-term outcomes associated with the interventions, and their thought process revolved very much around their child’s quality of life post-intervention, as well as access to comprehensive care.
Table 5 Final attributes and levels included in each DCE