EOL indicates end of life.
eTable. Semistructured Interview Guide for Bereaved Family Caregivers
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Lord S, Moore C, Beatty M, et al. Assessment of Bereaved Caregiver Experiences of Advance Care Planning for Children With Medical Complexity. JAMA Netw Open. 2020;3(7):e2010337. doi:10.1001/jamanetworkopen.2020.10337
How do bereaved family caregivers describe their experiences with advance care planning for children with medical complexity?
In this qualitative study that included 13 parents of children with medical complexity, participants emphasized the importance of involving trusted health care professionals and incorporating parental expertise to guide advance care planning. The relative shock parents experienced regarding the timing of the child’s death, despite recurrent experiences with life-threatening events, and the multiple losses they experienced when the child with a large health care team died were important themes.
The perspectives of bereaved parents in this study revealed important themes that should be considered for future study of advance care planning for children with medical complexity.
Advance care planning (ACP) is the process of discussing values and preferences for care to help inform medical decision-making. Children with medical complexity (CMC) often have a shortened life span with an unpredictable clinical course and timing of death; however, there is a paucity of literature that describes the experience of ACP from the perspective of bereaved family caregivers of CMC.
To explore the experiences of bereaved family caregivers with ACP for CMC.
Design, Setting, and Participants
This qualitative study included 12 interviews with 13 bereaved family caregivers of CMC whose deaths had occurred in the 5 years before study commencement (2013-2018). Participants were recruited at a single tertiary care pediatric center; CMC were treated by the Complex Care or Long-term Ventilation clinic in Toronto, Ontario, Canada. Data were collected from July to October 2018. Thematic analysis with an inductive approach was used.
Qualitative interviews were conducted using purposive sampling of bereaved family caregivers using semistructured interviews that were recorded and transcribed. Interviews were conducted until saturation was reached.
Main Outcomes and Measures
Transcripts were analyzed to create themes that characterized caregiver experiences with ACP.
A total of 13 family caregivers were interviewed in 12 interviews, all of whom were parents (12 [92%] women, 1 [8%] man) of a deceased child (aged 7 months to 12 years). Themes were divided in the 3 following categories, which align with the Donabedian model for health service quality: (1) structure of care, (2) ACP process, and (3) end-of-life experience. Notable subthemes for this population included the importance of accounting for parental expertise in the child’s care, recurrent experiences with life-threatening events, relative shock of the timing of death, and the multiple losses that caregivers experienced.
Conclusions and Relevance
In this study, parental experiences revealed that there are key aspects of the structure of the child’s care, process around ACP, and end-of-life care experiences that provide important reflections on ACP that warrant future study.
Medical and technological advances have resulted in a growing cohort of children with medical complexity (CMC), many of whom would not have survived previously.1,2 These children are often defined as having high family-identified needs, complex chronic disease necessitating specialized care, functional disability, and high health care utilization.3 They are among the most medically fragile pediatric patients and are at high risk of morbidity and mortality during childhood.3,4 Given their baseline medical and technological needs, decision-making regarding medical interventions that optimally balances quantity and quality of life is challenging.
Advance care planning (ACP) is the process of discussing values and preferences to help inform medical decision-making.5 In pediatrics, this often involves shared decision-making among parents and health care professionals to align care with the child’s best interests. ACP discussions are often prompted by the anticipation of impending death, including do-not-resuscitate orders, or when curative therapies are no longer effective and palliative care becomes the focus.6 This approach does not lend itself to optimal care when the course of illness and timing of death are highly unpredictable, as is often the case for CMC. Furthermore, health care professionals’ traditional practice of ACP at the end of life may miss important opportunities to explore families’ goals throughout the disease trajectory for CMC.
Prior studies have demonstrated that parents value ACP because of the potential for optimizing a child’s quality of life and achieving better end-of-life outcomes.5 Given the chronicity, unpredictable disease course, and tendency toward technology dependence for many CMC, we felt it would be worthwhile to investigate ACP experiences in this population specifically. Most studies involving bereaved parents have focused on children with disease categories that tend to have a more predictable trajectory, such as cancer, congenital heart disease, and defined genetic syndromes.5,7,8 While some previous studies have focused on ACP for CMC, they have examined the experiences of parents and health care professionals of living CMC.9-11 The current study was undertaken to examine the experiences of ACP for CMC from the perspective of bereaved parents. Our hypothesis was that the insights from parents who had experienced the full life and death of their CMC would provide valuable additions to the existing literature that helps guide ACP practices for this population.
We conducted qualitative interviews with bereaved family caregivers of CMC at The Hospital for Sick Children, Toronto, Canada. Institutional research ethics approval was obtained from the Hospital for Sick Children. Written informed consent was obtained by a research coordinator prior to initiation of the interview. Data were collected between July and October 2018 for patients who died between 2013 and 2018. The Consolidated Criteria for Reporting Qualitative Research (COREQ) checklist for reporting qualitative studies was used to guide reporting.23 Parents were eligible for study participation if their child met at least 1 criterion from each of the following conditions, per the Ontario Provincial Council of Maternal and Child Health criteria for CMC12: (1) technology dependent and/or user of high intensity care, (2) fragility, (3) chronicity, and (4) complexity. Furthermore, their children had to have been treated in the Complex Care or Long-term Ventilation (LTV) clinic, parents had to have been the primary caregiver for a CMC and experienced the death of that child between 6 months and 5 years before the recruitment date, and parents had to have had at least 1 ACP discussion (identified by a member of their former Complex Care or LTV team). While definitions of CMC vary, these criteria have been successfully implemented for prospective recruitment to complex care programs across Ontario, Canada’s largest province (population, 14.5 million). It is estimated that as many as 0.67% of children in Ontario meet these criteria.13 Families were excluded if their child’s death occurred within 6 months of the recruitment date to respect their early months of bereavement and avoid any added psychological burden associated with discussing those events in the context of a research study. Parents were recruited through purposive sampling to ensure representation of CMC of various ages, diagnoses, technology supports, and experiences (positive and negative) with ACP. Criteria for ACP discussions were not strictly defined, given that the topics varied depending on the child and context. However, they shared the following characteristics: they pertained to health-related situations that might happen in the future, and they involved identifying goals of care and thinking about how to align future care with those goals.
Eligible families were identified by Complex Care and LTV program physicians using their respective clinical databases. Introductory letters were sent, and follow-up phone calls were made to families who did not opt out. Data collection comprised semistructured interviews. Basic demographic information was obtained from the hospital electronic medical record.
An interview guide was developed by the research team following a review of the relevant literature5-11,14-19 and with input from pediatric complex care, LTV, and palliative care experts. All interviews were conducted in English, although interpreter services were available and offered to any family for whom English was not their preferred language. Following 2 initial interviews, transcripts were reviewed, and revisions were made to the interview guide to improve clarity (eTable in the Supplement). Interviews lasted from 40 to 80 minutes and were conducted at a location of the participant’s choice (eg, home, hospital, by telephone) by a research coordinator (C.M.) who did not have a prior relationship with the participants. Participants were compensated for their time with a nominal gift card.
Semistructured interviews were audio-recorded and transcribed verbatim by a professional transcriptionist. The qualitative analysis was aided by qualitative data management software (NVivo version 12 [QSR]). Four members of the research team (S.L., physician; C.M., research coordinator; M.B., research coordinator, and J.O., physician) followed the Braun and Clarke steps of thematic analysis,20 first by reading and rereading the data, then by the generating initial codes. Codes were organized into themes and subthemes, which were subsequently reviewed to ensure the data supported each theme. Consensus regarding codes and themes was reached through meetings among team members (S.L., C.M., M.B., and J.O.) during which emerging codes, themes, and relationships were discussed. Interviews were completed once thematic saturation was achieved.20-22 Because no statistical tests were performed, not prespecified level of significance was set.
A total of 53 eligible families were identified by the Complex Care and LTV physicians and were sent an introductory letter about the study; none of them opted out of a telephone call. Overall, 29 (55%) were unable to be reached by telephone, and 12 (23%) declined participation; thus, 12 families (23%) agreed to participate. A total of 13 bereaved family caregivers (12 [92%] mothers and 1 [8%] father) whose CMC died between 2013 and 2018 participated in the semistructured interviews. One interview was completed by both the father and the mother, while the rest of the interviews were completed by the mother alone. Demographic information regarding family caregivers and their CMC is summarized in Table 1.
Following analysis and coding of the interviews, the themes were analyzed, then grouped in a way that aligns with Donabedian’s established framework for characterizing health care service quality.24 This model has been used as a foundation for health services research, including conceptual frameworks for communication regarding end-of-life issues, and evaluates aspects of health care according to structure, process, and outcome.25 The overarching themes in our study were organized as follows: (1) structure of care, (2) ACP process, and (3) end-of-life care outcomes, with subthemes grouped accordingly (Figure). Examples of quotations for each theme and subtheme are shown in Table 2.
Parents described how the health care system and team structure during the child’s routine care and acute illnesses provided an important foundation on which ACP was situated (Table 2). Many stated that having trusted health care professionals who knew their child well was an important prerequisite for ACP. The presence of medical supports in their local community, such as physicians, home and school nursing, and the resources in their tertiary care hospital, including subspecialty teams and access to higher intensity care, were important factors that influenced ACP. For example, community and tertiary care resources influenced choices about location of care during times of illness or disease progression. Furthermore, many participants described the involvement of a subspecialty palliative care team as helpful for exploring goals of care.
The ACP process was divided into 2 subthemes: (1) the family and patient context and (2) the ACP discussions. The family and patient context (Table 2) included an understanding of the child’s existing medical and technological needs, given that these often informed ACP decisions (eg, whether to add new technology, feasible locations of care in instances of worsening illness or for end-of-life care). Parents also described the degree of prognostic uncertainty, which was often high, as another aspect of their child’s unique situation that needed to be taken into account. Parents’ perceptions of their child’s quality of life and their specific goals for their children (both short-term and long-term) were key contributors to ACP. Examples included goals for being at home together as a family as much as possible or having typical family outings. Parents appreciated when their own expertise in their child’s care was acknowledged and valued, given that their knowledge was important for contributing to medical care decisions. Finally, CMC’s recurrent experiences with severe illness and life-threatening deteriorations were common and important influencers of ACP. Medical decisions regarding care escalation during an acute deterioration were influenced by the child’s past experiences with escalations in care under similar clinical circumstances. For example, parents spoke about prior experiences with escalations and what interventions were successful in stabilizing their child; this guided decisions about whether to embark on similar interventions in the future.
Parents’ reflections on their experiences with ACP discussions (Table 2) revealed that their preferences regarding pace and timing varied. Many felt that ACP discussions should occur early and continue regularly. Others expressed that they should be the ones to indicate readiness to engage in these conversations or felt that the conversations were too frequent. Having a comfortable setting, such as a quiet room with adequate seating, was important. Another theme was having the appropriate people present, including health care professionals who knew the patient and family well and key family caregiver(s) (eg, ensuring both parents were present). From the perspective of communication, the importance of health care professionals expressing compassion above all else was a common theme.
Parents shared a number of reflections related to their experiences with end-of-life care (Table 2). None of the parents in our study had a child die at home. They felt more comfortable with their child dying in the hospital or hospice and reflected that ACP did help them consider different options for the location of end-of-life care. Parents noted that although their child experienced recurrent life-threatening events and they knew their child would likely have a shortened life span, the timing and circumstances around death often came as a shock. In addition to this, many parents described feeling not only the loss of their child but also the loss of the relationships they had built with individuals in their child’s large care team. Finally, several parents highlighted the need for improved grief and bereavement support following their child’s death, specifically in terms of having counselors and family support groups who understand the unique experience of caring for and losing a CMC.
This is a novel study of bereaved family caregivers’ experiences with ACP in that it focuses on CMC. The study adds to a growing body of literature in the area of pediatric ACP that no longer focuses only on typically developing adolescents who are capable of participating in their own ACP14,15 but also considers younger or noncommunicative children whose caregivers are faced with making decisions on their children’s behalf. In the case of CMC, caregivers often have to make those decisions in the context of a chronic disease course without a predictable trajectory. Perspectives from parents who have experienced the full life and death of their child are crucial for improving our understanding of optimal ACP for CMC.
The results of our study align with previous findings that a supportive relationship with health care professionals is key for effective ACP. A study by Mitchell et al16 that examined parental experiences with end-of-life decision-making for children in the intensive care setting also revealed the importance of having trusted health care professionals involved in ACP. In terms of the ACP process itself, the study by Mitchell et al16 also demonstrated the importance of parental knowledge as a key factor in ACP. Furthermore, demonstrating compassion in the discussion was identified in our and previous studies17 as being of utmost importance. As part of that compassionate approach, involving both parents and guardians in the discussion was preferred. In keeping with other centers,18 our study revealed that a high proportion of CMC whose families engaged in ACP had a pediatric palliative care team involvement.
Another theme that arose in our study was the optimal timing of ACP. The parent perspectives gathered in our study reflected that timing is important and that an earlier, step-wise conversation is appreciated by some, but there is no single approach that is preferred by all. In a similar study including health care professionals and parents of current CMC at our center,9 a flexible approach that was adaptable to parent needs was preferred. In another study of parental perceptions of ACP for children on long-term assisted ventilation,19 the issue of deciding when to start ACP discussions was challenging, and many parents preferred not to discuss advance directives during periods of stability. Others have found that parents prefer an individualized and gradual approach to ACP and a health care professional who has worked with the family consistently to facilitate those discussions.5,7 A dynamic and family-centered approach is likely needed, with the ability to initiate ACP outside of a time of crisis and then reflect with parents on the best way to discuss goals of care moving forward.
Several findings emerged from our study, including the themes related to the patient and family context, that help frame options for care moving forward. Parents consistently raised the child’s existing medical and technology supports as well as the degree of prognostic uncertainty as important factors of ACP. The family’s experiences with their child’s prior life-threatening events emerged repeatedly as a theme that played a key role in informing ACP. These aspects of the CMC patient and family experience may differ from other patients engaging in ACP, whose illness trajectory and end of life follow a more predictable trajectory. As such, they are important factors that clinicians should seek to understand when embarking on ACP with CMC.
Having lived through the entire disease trajectory and end of life of their child, family caregivers were able to reflect on the end-of-life care experience and how this was informed by ACP. Many parents described a feeling of relative shock when their child’s death actually happened, even though they had experienced multiple life-threatening events in the past. Despite knowing their child would likely die young and from similar causes, the final illness was often similar to clinical deteriorations in the past. We posit that there may be a degree of desensitization that occurs over time for parents who experience the up-and-down clinical course that is common for children with chronic medical fragility. Once they have faced a life-threatening situation several times, parents may adjust to their child surviving against the odds each time, resulting in a feeling of shock when the child ultimately does not survive.
When examining the bereavement experience, the feeling of experiencing multiple losses after the child’s death was shared strongly among participants. Parents described that this added sense of loss from the sudden absence of multiple health care professionals and community professionals who surrounded the child in their day-to-day care. Grief and bereavement support was desired not only to cope with the loss of the child but also to deal with this abrupt change and loss of relationships with the professionals who cared for their child and family.
This study has limitations. First, it took place at a single center. In the future, interviewing parents of CMC from other centers would be beneficial to gain broader perspectives. Although we reached thematic saturation, the available participant pool was small, given that many families’ contact information was not available. Although sampling began as purposive, all eligible families were ultimately contacted. Data on those we were unable to contact or who declined participation were not available. Our sampling only included the Complex Care and LTV clinics although there are some patients who meet criteria for CMC who were not treated in these clinics. It is possible that access to the multidisciplinary professionals in these clinics informed ACP. Interestingly, all CMC in our study experienced an in-hospital or hospice death, in contrast with the wider pediatric population in the center, in which a significant proportion of deaths occur at home. This in itself warrants further study. Additionally, we had participation almost exclusively from mothers, so a future study of fathers’ insights would be helpful.
In this study, bereaved families’ experiences shed light on important aspects of ACP for CMC that warrant further study. CMC experience an unpredictable disease trajectory, and effective ACP discussions reflected this uncertainty. Parents appreciated a compassionate approach that involved trusted health care professionals who followed the pace and timing that were best for that particular family. Their experiences suggest that discussions should be informed by the child’s unique context and parental expertise regarding the child’s care. Understanding the common experiences with recurrent life-threatening events may be useful for providing anticipatory guidance. The added sense of loss that may be experienced by the family after the child’s death, when they no longer have the large circle of health care professionals providing support, is an important consideration for those providing grief support. The key concepts captured in this study can help inform future research and development of ACP processes for CMC.
Accepted for Publication: May 2, 2020.
Published: July 28, 2020. doi:10.1001/jamanetworkopen.2020.10337
Open Access: This is an open access article distributed under the terms of the CC-BY License. © 2020 Lord S et al. JAMA Network Open.
Corresponding Author: Sarah Lord, MD, MSc, Division of Pediatric Medicine, The Hospital for Sick Children, 555 University Ave, Toronto, ON M5G 1X8, Canada (email@example.com).
Author Contributions: Drs Lord and Orkin had full access to all of the data in the study and take responsibility for the integrity of the data and the accuracy of the data analysis.
Concept and design: Lord, Cohen, Rapoport, Hellmann, Amin, Orkin.
Acquisition, analysis, or interpretation of data: Lord, Moore, Beatty, Rapoport, Netten, Amin, Orkin.
Drafting of the manuscript: Lord, Orkin.
Critical revision of the manuscript for important intellectual content: All authors.
Statistical analysis: Moore.
Obtained funding: Lord, Amin, Orkin.
Administrative, technical, or material support: Beatty, Netten, Amin.
Supervision: Rapoport, Hellmann, Amin, Orkin.
Conflict of Interest Disclosures: None reported.
Funding/Support: This study was funded by the Norman Saunders Complex Care Initiative at the Hospital for Sick Children.
Role of the Funder/Sponsor: The funder had no role in the design and conduct of the study; collection, management, analysis, and interpretation of the data; preparation, review, or approval of the manuscript; and decision to submit the manuscript for publication.
Additional Contributions: We gratefully acknowledge the time and openness of the parents who were willing to share their stories and insights with us.
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