Pediatrician Perspectives on Brief Resolved Unexplained Events

We conducted a qualitative study using semistructured interviews with providers who care for infants <12 months of age in a pediatric ED setting. The investigators are pediatric emergency medicine physicians with an interest in quality improvement who practice at one of the participating university-affiliated institutions. One of the investigators has a specific interest in BRUE-related research. The local institutional review board approved this study, and all participants provided verbal consent before participation.

A convenience sample was recruited via e-mail. Within our state, study information was distributed to pediatric emergency medicine fellowship program directors via publicly available e-mail addresses. There are 4 institutions within the state with pediatric emergency medicine fellowship programs. The institutions include both level 1 and level II trauma centers, evaluating 20 000 to 85 000 patients annually, in 16 to 52 beds at both tertiary-care and affiliated community sites. The institutions have up to 70 faculty, including physicians with pediatric, emergency medicine, and pediatric emergency medicine training.⁹  Fellowship directors were asked to electronically forward the study information to their institutional pediatric ED colleagues. A follow-up reminder e-mail was sent to the fellowship directors 3 weeks after the initial e-mail, but compliance with the request to forward the study information to their colleagues was not confirmed. Study information contained the contact details for the principal investigator, and participants self-selected and contacted the principal investigator directly to arrange a date and time for an interview.

After completion of a literature review, a semistructured interview guide was developed by the study authors in consultation with local and national experts (Supplemental Information). The guide was reviewed by pediatric emergency medicine providers for feedback on content and question clarity and then piloted by the principal investigator with the first 6 respondents to the recruitment e-mail. During the pilot phase there were no changes to the content of the questions, but minor adjustments were made to the interview guide to include additional open-ended questions intended to allow providers an opportunity to distinguish between caregiver and physician perceptions of BRUEs.

A total of 25 participants responded to the recruitment e-mail and all were interviewed by the principal investigator. Participants completed a semistructured audio-recorded interview at their convenience by telephone or in person between October 2019 and January 2020. The first 6 interviews were used as a pilot, whereas the others were included in the analysis. Demographic information including age group, sex, residency and/or fellowship training, years in practice, and practice setting was collected verbally at the start of the interview. Audio-recordings were transcribed by a medical transcription service.

The 6-phase approach to reflexive thematic analysis, developed by Braun and Clark, was used to conduct the qualitative analysis.¹⁰  Using an inductive approach to thematic analysis, the investigators identified patterns of meaning within the data set by following a stepwise yet fluid process of data familiarization, code generation, theme identification, theme refinement, and thematic mapping.^10,11  Before conducting the analysis, transcripts from the first 6 pilot interviews were reviewed, and after data familiarization, these transcripts were used to develop the initial codes. All 3 investigators completed this process independently and then collaborative group discussion followed wherein the transcripts and codes were reviewed until consensus regarding the initial codes was achieved (Supplemental Table 4). The analysis was then conducted in batches of 4 to 6 transcripts, and with each batch, a similar process of data familiarization was used and the transcripts were coded by using the initial codes. Because this approach is flexible by nature, additional codes were generated as needed. As with the pilot transcripts, all 3 investigators reviewed the transcripts independently, and then collaborative group discussion was used to finalize the coding of each transcript. With each batch of transcripts, the investigators collaboratively identified and modified themes based on the codes. There was notable overlap between the themes identified in the first and second batches of transcripts. Therefore, the final batch of transcripts was used to confirm and finalize the identified themes, resulting in a final sample size of 19 physicians. Identified themes were noted to be interconnected, largely reflecting the patterns and complexities of clinical decision-making; thus, the themes were reviewed with attention to the transitions and connectors noted in the audio-recorded interviews and transcripts to facilitate thematic mapping and delineate the primary themes and underlying topics.¹²  Because multiple investigators were involved in the data analysis, investigator triangulation was used to enhance the trustworthiness of the data.¹³  Descriptive statistics including frequency and percentage were calculated for the demographic variables.

Nineteen physicians, practicing in 4 nonaffiliated pediatric EDs across our state, participated in the study. The interviews ranged in length from 4 minutes 15 seconds to 13 minutes 9 seconds, with a mean length of 7 minutes 32 seconds. As shown in Table 1, the majority of participants work in a university-affiliated setting and 68% report an annual ED vol ≥80 000 patient visits. Only 4 (21%) participants reported that the BRUE CPG had been implemented in their ED. The primary themes related to providing care for patients with a BRUE identified in our analysis were (1) reassurance, (2) caregiver or provider concern, and (3) CPG availability and interpretation. Closely related underlying topics informing BRUE patient management were also noted: (1) ambiguity in the BRUE diagnosis and its management; (2) a need for shared decision-making between the caregiver and the provider; and (3) concern over the increased time spent with families during an ED visit for a diagnosis of BRUE (reference Supplemental Table 4). In Table 2 and Table 3, we provide illustrative quotes from participating physicians that are representative of the identified thematic relationships. Ultimately, these complex relationships were found to influence patient evaluation and disposition (Fig 1).

Physicians recognized the important role they played in providing families with comfort and reassurance that their child was not in imminent danger after a BRUE. Physicians were aware that parents expected an explanation for this frightening event and wanted specific guidelines for its prevention: “They’re scared and they’re willing to pretty much do what the doctor wants to do” (participant 8). As a result, many physicians relied on the guidelines put forth by the AAP to explain the diagnosis of BRUE to caregivers. By “using that terminology and trying to be transparent to the family to provide some reassurance on the basis of what information [has] been provided to us from the American Academy of Pediatrics,” physicians felt equipped to explain to caregivers that a lower-risk BRUE can “be safely followed up with their primary care physician” without additional evaluation or observation (participant 1). Physicians perceived, however, that although research supports limited testing and observation for a lower-risk BRUE, caregivers remained uncomfortable with this approach. Thus, providing reassurance is difficult and often results in the need for a longer conversation: “I think [discussions with parents regarding diagnosis] go pretty well but parents do have a lot of questions so those are times when you need to spend a lot of time with families” (participant 15).

Although the criterion of life-threatening has been removed by the AAP from the definition of BRUE, physicians noted continued caregiver anxiety surrounding the event that led to ED evaluation. Acknowledging how frightening an apneic event can be for the caregiver of a young infant, physicians expressed that it can be difficult to label the event as benign. Consequently, providers reported having a low threshold for admitting patients for observation, particularly if the caretaker appears uncomfortable: “… if I think the family has a high degree of anxiety, stress over this whole event, I tend to admit them for observation” (participant 1). They will often have an open discussion with the family explaining the diagnosis and its differential and determine the caregivers’ comfort with discharge from the hospital. As a result, the decision of discharge versus admission for observation is a shared decision between provider and caregiver: “If they appear hesitant then I will open up the conversation of a possible observation period and if they are agreeable to that then I will admit them for observation… I really leave this decision of going home more to families and their degree of comfort with the particular event” (participant 11).

Physicians also voiced concern centered on the fact that BRUE is a clinical diagnosis lacking a confirmatory diagnostic test. Physicians expressed that it can be difficult to elicit the necessary history to determine if a patient is lower-risk. This leads to doubt and frustration in the diagnostic process related to the fact that they “don’t have an obvious diagnosis to explain this random apneic episode” (participant 6). Ambiguity in the BRUE diagnosis, the limited approach to testing for a lower-risk BRUE recommended by the AAP, and a paucity of data to “predict whether this would happen again” ultimately influenced ED management and final disposition (participant 15).

Although only a minority of participants reported guideline implementation within their ED, physicians indicated that they referenced the CPG to inform their clinical decision-making. Physicians also referred to the guideline in discussions with caregivers, finding that “the framework of BRUE itself can be helpful… knowing that this is a studied population on a national scale that’s understood by the American Academy of Pediatrics” (participant 1). The unexplained nature of the event, however, was difficult for providers to accept. Some providers voiced that “all these kids have a diagnosis” and thus actively avoided diagnosing a patient with a BRUE, erring instead toward an assumed or confirmed, but more familiar, etiology of the event (eg gastroesophageal reflux). In contrast, although some providers committed to the diagnosis of BRUE, they struggled with its obscure nature, desiring a more comprehensive explanation for the event. Oftentimes, the explanation for the event was thought to be more useful to caregivers because this enabled them to anticipate potential future events. In either case, physicians felt more comfortable explaining to caregivers what a BRUE is not rather than vaguely discussing what it is because “labeling [the patient] with something that’s a little bit obscure can confuse the family” (participant 2).

Using qualitative methodology, we are the first study to explore the perspectives of health care providers in the interpretation and application of the recommendations for the care of infants with BRUE. Our analysis reveals that providers rely on the CPG to engage in shared decision-making with families, but offering reassurance is time-consuming and challenging because the ambiguous and unexplained nature of the event leaves room for doubt among providers and caregivers.

Our results extend the previous literature examining caregiver perceptions of BRUE.^7,8  Caregivers of hospitalized patients with a higher-risk BRUE expressed that diagnostic testing and hospitalization offered some reassurance but the unexplained nature of the event led to continued uncertainty that contributed to a degree of ambivalence regarding discharge from the hospital.⁷  Caregivers interviewed after bringing their child to the ED after a BRUE and being discharged from the hospital noted 3 similar key themes: “apprehension about their child’s well-being, ambivalence about the best course of action after the initial evaluation in the ED, and a need for reassurance that their infant was now fine, that the cause of the event was benign, and that they did not have to worry about a recurrence.”⁸  Thus, our results reveal that the provider–caregiver dyad share a similar perspective, because both parties need to balance continued concern with the need for pursuing additional diagnostic testing, which, particularly in the case of a lower-risk BRUE, is not needed but may serve to provide reassurance.

Uncertainty with BRUE is implicit in the diagnosis. Although diagnostic uncertainty is an understood and accepted part of clinical practice,¹⁴  the uncertainty with BRUE is not necessarily with the diagnosis itself but with the underlying pathophysiological explanation or etiology of the event. Thus, although the literature related to diagnostic uncertainty suggests that caregivers are more receptive to broad differential diagnoses in the face of uncertainty,¹⁵  this approach is not applicable in the case of BRUE, in which the diagnosis may be certain but the provider is “not sure” why the event occurred. Uncertainty is further exacerbated by the fact that guidelines exist for patients stratified as lower risk yet the majority of patients with BRUE meet higher-risk criteria for whom evidence-based recommendations published as a CPG do not exist.^1,5,16  Because caregivers prefer differential diagnoses when uncertainty exists,¹⁵  providers can frame their discussion around the diagnoses considered for patients who sought care for a BRUE when an etiology was not found.¹⁶  By explaining why these varied diagnoses are not the case when the BRUE diagnosis is being made, providers may help to alleviate some of the uncertainty voiced by caregivers and minimize the ambiguity noted by providers.

Providers can also use shared decision-making to help address uncertainty during a health care encounter.¹⁷  Notably, our analyses reflect several important and previously reported themes related to shared decision-making in the ED setting: time constraints are a significant barrier to shared decision-making,^18,19  and providers are generally “more comfortable ordering more tests and admitting patients rather than risking a bad outcome or a missed diagnosis.”¹⁹ 

Furthermore, shared decision-making requires a certain degree of risk tolerance on the part of the provider and the caregiver.^17,19  In the case of BRUE, risk tolerance will vary substantially because caregivers are not uniform in their response to uncertainty regarding their child.⁸  To facilitate shared decision-making, providers should consider use of a clinical decision aid because these have been shown to increase patient knowledge and involvement in decision-making.²⁰  In the case of minor head trauma, for example, caregivers who were randomly assigned to use a decision aid with their provider had “greater knowledge, less decisional conflict, greater physician trust, and greater involvement in…decision-making.”²¹  A decision aid does not address the time constraint voiced by participants in our study, but as demonstrated for other pediatric ED presentations,²¹  a decision aid is a caregiver-centered approach that may help to provide a structured framework for addressing ongoing concerns while also providing reassurance about what is known regarding patient outcomes after a BRUE.

Caregiver engagement, along with leadership, team building, culture, and context, are the “softer skills” required for successful quality improvement initiatives.²²  In light of our results, institutions might address some of these softer skills by ensuring that caregivers are part of a multidisciplinary quality improvement team that includes stakeholders from the inpatient and outpatient setting with endorsement from institutional leaders.²³  These softer skills, however, must be combined with multifaceted change concepts to ultimately lead to improvement.^23,24  Given our identified themes and the importance of managing variation, organizations might consider implementation of (1) an institutional version of the BRUE CPG, and (2) in the absence of a CPG for higher-risk BRUE, an institutional approach to patients with higher-risk BRUE based on the published framework.^1,5,24  Standardized care might also include scripted discharge instructions for caregivers and a routine expedited outpatient appointment with the primary care provider facilitated at the time of discharge. Finally, in addition to the aforementioned decision aid, the change concept of the producer/consumer interface can also be addressed by providing training in cardiopulmonary resuscitation, which has previously been noted to increase caregiver comfort.^7,24 

There are several limitations to this study. First, the recruitment methods used in this study lend to a selection bias given that participants were pediatric providers from a single state, and the majority practiced in a large university-affiliated center. As a result, their patient population, previous clinical experience, and available community resources may differ from other practice settings, particularly adult EDs, where the majority of pediatric patients receive care.²⁵  In addition, participants volunteered to participate and therefore may differ from health care providers who did not volunteer their participation. Finally, the principal investigator has a collegial relationship with some of the participants, which may have influenced the responses obtained during the interviews. Second, a small number of providers participated in this study. With a narrow topic such as BRUE, thematic saturation may have been achieved with a small number of participants. However, in future studies, researchers might consider using base size, run length, and new information threshold as a reliable approach to determining an adequate sample size.²⁶  Third, researcher biases may have influenced the qualitative analyses, but investigator triangulation enhances the reliability of the interpretation of the data, and some of the inferences made align with previous studies of caregivers’ perception of BRUE.^7,13  Finally, the researchers do not have expertise in qualitative methodology. This lack of expertise may have affected the richness and depth of the answers provided by the participants and, as a result, impacted data transferability. In eliciting a more reflective data set, different themes may also have been identified, which could have led to alternative conclusions. Researchers in future qualitative studies might focus on effective approaches to shared decision-making by including focus groups with parents and providers from both the outpatient and inpatient settings.

Quality improvement initiatives focused on care delivery for patients after a BRUE should include multifaceted interventions that address caregiver and provider concerns regarding the diagnosis of BRUE and incorporate decision aids for clinicians and families that align with recommendations from the CPG and support shared decision-making. Further research is needed to evaluate whether integration of these quality improvement interventions translates to safe, effective, and patient-centered care for infants and families after a BRUE.