Research Considerations in Pediatric Palliative Care

Published on 09/04/2015 by admin

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10 Research Considerations in Pediatric Palliative Care

Betty Davies, Kimberley Widger, Rose Steele, Susan Cadell, Harold Siden, Lynn Straatman

No one can whistle a symphony. It takes an orchestra to play it.

— Halford E. Luccock

While the actual number of patients in pediatric palliative care is small relative to the number of adults, the scope of the field is vast. There are many areas and opportunities for research with the children, parents, siblings, extended family members, and peers, as well as the teachers, clinicians, and volunteers who work with the children and their families. There is diversity in the types of diseases, illness trajectories, biopsychosocial and spiritual facets of illness, developmental aspects of childhood, and care at diagnosis, end-of-life, and into bereavement. There also is a need to explore how to best translate completed research into improving care for children and families and a need to better support those who provide the care.

The opportunities, joys, and rewards of conducting research in any of these areas are many, but there also are challenges or pitfalls to be overcome or avoided. While many of the joys and challenges are not unique to pediatric palliative care, some of the distinct aspects of the field bring an additional level of complexity to the associated research. In this chapter, we review priority areas of research in pediatric palliative care. We also identify and explore various methodological and ethical issues inherent in research in this field and suggest methods to address them, including conducting research as a team. Our own research team, Transitions in Pediatric Palliative and End-of-life Care (PEDPALNET), was created following a call for proposals by the Canadian Institutes of Health Research (CIHR) to develop collaborative, multidisciplinary research teams organized around particular areas of study in palliative care. Of the nine teams funded across Canada, our team was the only one with a particular focus on pediatrics. Some of the examples we use in this chapter are from our experiences conducting research as a team over the last five years.

Priority Areas for Research

The Institute of Medicine (IOM) report on improving palliative and end-of-life care for children and their families highlighted the lack of research and systematic data on which to base their recommendations.1 The authors identified a critical need for research in all aspects of pediatric palliative and end-of-life care and recommended focusing research activities on “the effectiveness of: clinical interventions including symptom management; methods for improving communication and decision-making; innovative arrangements for delivering, coordinating, and evaluating care, including interdisciplinary care teams and quality improvement strategies, and different approaches to bereavement care.”1

Others also have worked to identify research priorities. The four top questions identified from a Delphi survey with pediatric palliative care researchers and clinicians in Canada were: What matters most for patients and parents receiving pediatric palliative care? What are the bereavement needs of families in pediatric palliative care? What are the best practice standards in pain and symptom management? What are effective strategies to alleviate suffering at the end of life?2 In pediatric oncology, the areas of palliative and end-of-life care have been identified as research priorities. Recommendations include a focus on: decision making and communication, characteristics of death and profiles of bereaved family members and health professionals, trajectories of dying, a comparison between the care provided and the care that was desired by families, financial cost of death after cancer, and outcomes from symptom management and bereavement support.3,4

The identified priority areas are broad enough to leave opportunities for exploring a wide range of specific research questions. Experience suggests that researchers need to identify the questions that keep them up at night. The questions need to be exciting and interesting enough to researchers so that they will keep going despite the inevitable challenges. Given the current lack of research in the field, it is likely that initial studies are needed to develop appropriate indicators or measures and to more fully understand the experiences and needs of a particular population. Initial studies should be completed before the most burning questions can be answered with designs such as randomized controlled trials (RCT) of interventions.

It has historically been challenging to obtain funding for pediatric palliative care research, partly because the major government funding agencies have agendas and limited resources that may not be supportive of this type of research. In Canada, the government recognized the importance of creating a health agenda that incorporated palliative care into the spectrum of care provided to its citizens. Therefore, in 2004 Canada took a leadership role in funding palliative care research through the CIHR, which is one of the three major research funding agencies in the country. Palliative care was identified as a priority and a targeted competition was held to create interdisciplinary teams (IDT) to facilitate the development of programs of research. Our own team was funded as a result of this competition and we remain the largest funded team in pediatric palliative care in Canada. However, we work in collaboration with colleagues from across the country who also may have their own more local teams. Subsequent funding for our work was obtained through operating fund competitions, but no further resources have been allocated specifically for palliative care let alone for pediatric palliative care. In the United States, the seminal work of the IOM on Approaching Death, published in 1997, explicated gaps in scientific knowledge. In response, the director of the National Institutes of Health (NIH) designated the National Institute for Nursing Research (NINR) to serve as the lead Institute at NIH for end-of-life research. To coordinate this effort, NINR has established the Office of Research on End-of-Life Science and Palliative Care, Investigator Training, and Education (OEPC) and offered numerous Program Announcements (PA) that solicit research pertaining to end-of-life care. The one PA that specifically addressed pediatrics (Improving Care for Dying Children and Their Families – PA 04-57) is inactive although pediatric proposals can be submitted in response to other PAs.

Methodological Challenges

When considering the development of a study in the area of pediatric palliative care, researchers need to be aware of potential impediments to their success. Some of the methodological challenges include research design, outcome measures, recruitment and sampling, and research teams. While ethical challenges are alluded to in this section, they will be discussed more fully later in the chapter.

Research design

Though the gold standard for research remains the RCT with the highest level of evidence being systematic reviews of multiple RCTs, primarily qualitative designs and descriptive methods may be more appropriate to address the research priorities and knowledge in pediatric palliative care.1 Further, RCTs are difficult to conduct in both adult and pediatric palliative care. The heterogeneity of diseases, illness trajectories, and ages of participants are difficult to accommodate in a design that requires a homogeneous group of participants and testing of simple standardized interventions.5 Bensink et al.6 made two attempts to use an RCT to test videophones as a support for families receiving pediatric palliative care, but all families approached refused to participate. When the research team changed the design to an acceptability study, without randomization or any measures to be completed by the family, they had a participation rate of more than 90%. These researchers suggested that a clinical trial is overwhelming for families when initiated at the transition to palliative care. Kane, Hellsten, and Coldsmith7 discussed the interpersonal relationships at the heart of palliative care and the need for understanding complex interactions among humans in order to identify the social and spiritual interventions needed to address human suffering. It may be that these interactions are not amenable to the quantified measurement and standardization needed for an RCT. Levels of evidence that may be more relevant for palliative care include quasi-experimental studies, qualitative studies, and consensus opinions of palliative care experts.8

Longitudinal studies in pediatric palliative care have been uncommon, but are useful in expanding the evidence base. Researchers who embark on longitudinal work need to design their studies in ways that will minimize attrition. Though families in pediatric palliative care often participate in research as a way of helping others, even when they find it challenging practically or emotionally,9 researchers need to pay attention to keeping families involved in a study over time. A number of effective strategies have been reported in the literature,10–13 and we are using some of them in our longitudinal study with families where a child has a progressive metabolic, neurological, or chromosomal condition and in our parent caregiver study. Development of a solid relationship with families is crucial. An initial face-to-face visit, telephone contact at prescribed intervals, and letters to thank families for their participation are useful for building and maintaining relationships. Continuity of research assistants or other personnel involved in the study is important and will contribute to the trusting relationship that is needed to help participants feel more connected to and interested in the study over time. Efforts should be made to respect families’ time and to recognize the value of their contribution. Researchers should schedule data collection at the family’s convenience and should regularly express verbal and written appreciation for their participation in the study. In addition, providing a monetary or other gift as a token of appreciation along with a letter thanking them for participation may encourage families to remain in a study, because families feel valued when their time and effort is recognized. Strategies that limit attrition will result in the type of high quality data needed to provide solid, evidence-based care.

Parent Participation

Parents in all of the studies we have undertaken as a team have given us feedback about what they see as being the value of their participation. In some cases this feedback was solicited and in others it was offered by the parents without our invitation. Our parent caregiver study has provided several touching examples. Many of the 273 participants took the time to do more than simply answer a survey. They wrote comments on the forms or sent letters to accompany their returning questionnaires. One mother wrote five pages about her story and added questions that she thought should be asked in the research. Another mother wrote a note about her depression, her partner leaving her, and the need to keep moving forward with a smile for her daughter who did not ask to be born as she was. Other parents have called the toll-free number to give us updates on their situations or thank us for the research. Some of our studies include or are solely comprised of qualitative interviews. Parents have commented on their desire to participate in research in order to make the difficult situations that they have experienced less difficult for someone else. Many parents want to participate in research. They also want the opportunity to decide for themselves if a study is right for them.

Outcome measures

Science can progress no further than the measurement of its key variables.

— ATTRIBUTED TO J. NUNNALLY, 1994

Unfortunately, in pediatric palliative care the most important variables or outcomes to be measured are not well defined, and there are few valid measures. Outcomes usually involve a change in the health status of the individual, but also can include increased knowledge of health conditions, changes in behavior related to health, or patient and family satisfaction with the care received and its outcomes.14 The expected outcome for the child in pediatric palliative care is death, which cannot be changed. However, one can improve the quality of that death15 and the family’s satisfaction with the care provided. One also may be able to improve health outcomes for the family including reduction in the incidence and severity of depression, anxiety, post traumatic stress, guilt, and complicated grief.

Research in adult palliative care is more advanced than that of pediatrics; however, that field of research is still relatively recent and similar issues about the difficulty of identifying and measuring important outcomes have been reported. There has been some research in adult palliative care asking patients and family members to identify components of quality care and of a good death.16–18 A number of measures have been developed and used with adult palliative care patients and their families (Mularski et al19). However, for multiple reasons, including the developmental level of children, the variables and/or outcomes and measures used in research with adults cannot simply be taken and applied to children. For example, one outcome deemed indicative of quality care for adults is that death occurs at home.20 In children, this outcome may not be as indicative of quality end-of-life care. Dussel, Kreicbergs, Hilden et al.21 found that the opportunity to plan the location of death was more important to parents than where the death actually occurred. Satisfaction is another outcome that has been identified as a component of quality care.14 However, satisfaction has not been well conceptualized in the literature and may be influenced by demographic variables.22 In end-of-life care, there is a concern that families have very low levels of expectation and therefore are easily satisfied with care.23 In pediatric palliative care, there is evidence that parents tend to report high levels of satisfaction even when high levels of distressing symptoms are reported.24 Thus, satisfaction may not be a good indicator of the actual quality of care.

Given that research in adult palliative care is more prevalent than in pediatric palliative care, pediatric researchers can learn from their adult-researching counterparts about possible instruments, methods, and research designs that might be useful. However, this information must be critically examined to see how instruments might need to be altered to fit the pediatric population. Any alterations must be based on clinical expertise and done with an intimate understanding of the population to be studied. This speaks to the need for researchers and clinicians to work together to conduct appropriate research in pediatric palliative care.

Given that there is no single instrument encompassing all of the dimensions of care in pediatric palliative care, researchers must choose among several to measure the specific domains of interest. Those domains will include tools that are specific to areas such as pain and other symptoms, the child’s emotional well-being, family issues, and spiritual support. There is no single instrument in these fields that answers all questions. For example, in studying families one may want to know about marriage and relationship stability, personal growth, family stress and functioning, or the health of individuals.

The Team for Research with Adolescents and Children in Palliation and Grief (TRAC-PG) at Toronto’s Hospital for Sick Children (SickKids) has compiled a list of instruments that some of its members have used with children, adolescents, and adults in pediatric palliative and end-of-life care research. This list is available on TRAC-PG’s website at www.tracpg.ca/index.pho/researchers/instruments_measures_scales, but caution should be used when reviewing these instruments for future research. As previously noted, few measures have been specifically validated for use in pediatric palliative care. Many of the measures have also been used with parents in the months or years after their child’s death.

Recruitment and sampling

There are several challenges in identifying, accessing, and recruiting a sample to take part in a study related to pediatric palliative care. The number of children who die each year is small relative to the number of adults. However, small numbers at any one center may make it difficult to access a large enough sample to fit with particular research designs. As well, many children have rare illnesses where there may be very few in one country or around the world, let alone within a particular institution. Two possible methods to increase the size of the accessible population are to use disease groupings rather than single diseases or to conduct multicenter research.

Disease Groupings

The Association for Children with Life Threatening or Terminal Conditions and their Families (ACT) and the Royal College of Paediatrics and Child Health identified four distinct groups, or quadrants, of conditions (Table 10-1) seen in children who may not live until adulthood and who require palliative care.25 Though there is some fluidity regarding which quadrant a condition fits into, conditions in a quadrant are similar to each other in terms of the expected trajectory and time course of the illness. Therefore, when planning a study, rather than only including children with a particular neurodegenerative condition, it may be possible, depending on the type of research questions being asked, to increase the available population by including children with any condition that fits into Quadrant 3.

TABLE 10-1 Association for Children with Life-Threatening or Terminal Conditions and Their Families and the Royal College of Paediatrics and Child Health Quadrants of Conditions That May Require Palliative Care

Quadrant 1 Quadrant 2
Conditions that can be cured but have a possibility of death (e.g., cancer) Conditions requiring intensive medical therapy but are ultimately terminal (e.g., cystic fibrosis, HIV/AIDS)
Quadrant 3 Quadrant 4
Conditions that have no cure but whose symptoms can be managed (e.g., neurodegenerative, metabolic disease) Severe neurological impairments where complications may lead to early death (e.g., anoxic brain injury)

Multicenter Research

Recruiting participants through a number of centers is another way to increase the size of the research population and to broaden the racial, ethnic, and geographic diversity of the sample.26 However, multicenter research may raise other challenges. The need for ethical approval at each site may add an additional six months or more to the time required to start a study.27 Additional associated costs must be factored in to grant proposals. A great deal of negotiation may be needed to ensure that recruitment strategies at each site will meet the requirements of local ethics boards, but are similar enough to each other to not threaten the validity of the study findings.

Participatory action research (PAR) is one method that may help to overcome some recruitment challenges. PAR involves extensive collaboration between the researchers and those affected by the issue that is being studied.28,29 In pediatric palliative care research, PAR has been used to some extent with both clinicians and families. For example, one study involved collaboration between a group of researchers and a group of clinicians who were providing hospice care. This collaboration included design and implementation of a project to evaluate the care provided by the clinical team. Trust between the two groups and active involvement of the clinicians in all aspects of the research facilitated recruitment of families as participants, as well as increased the uptake of study findings by the clinician group.30 In a similar project designed to evaluate a hospice program, the research team used PAR with the families. This collaboration ensured project objectives and methods used for data collection were sensitive to family needs and issues and were meaningful to participants.31

The positive outcomes from such studies suggest that using PAR with clinicians and families in pediatric palliative care may assist with some of the challenges to recruitment. Even if all of the principles of PAR are not followed, some evidence that a research protocol has been developed with direct input of families may be helpful in receiving ethics approval as well as funding for pediatric palliative care studies. We have found that including families’ input early as we design our studies, for example, asking families about the suitability of the potential instruments or piloting the accrual process with families, has contributed to an increased success rate for our funding proposals. It has also smoothed the process of conducting research, from receiving funding to ethical approval to recruitment.

Accessing Families

Once approval for a study has been obtained from the appropriate ethics boards, researchers generally need to rely on clinicians for assistance in recruiting participants. Clinicians who are not part of the research team or who have little experience with palliative or end-of-life care research may add to recruitment challenges if they act as gatekeepers to potential participants, especially if they are reluctant to identify potential participants.32 Yet, parents often want to make the decision themselves about whether they participate, rather than have clinicians make that decision for them.32 Tomlinson and colleagues highlighted the important impact that clinicians can have on recruitment.33 Hinds34 found that up to 8.6% of potential parent participants for a study that took place months to years after a child’s death were placed in a “do not approach” category by clinicians. The proportion deemed not approachable by clinicians rose to 26.8% in studies where the child was still alive, but where an end-of-life decision had been made. Hinds also reported that 54.9% of potentially eligible families in an end-of-life decision-making study were not referred to the study team, often because of the rush to transfer the child home once this type of decision was made. The urgency of the situation meant that clinicians sometimes forgot to make the referral or believed that the timing was inappropriate. As noted above, the use of PAR with clinicians may help to address their concerns about approaching families and may facilitate recruitment. As well, sharing existing evidence about the benefits of research for families and the ability of families to make their own choices may help to allay concerns. (This type of evidence is discussed later in this chapter under ethics.) Researchers should track the numbers of eligible participants who are not approached to participate in a study because the final sample may not be representative of the population under study. The findings of a study must be interpreted with this possibility in mind.

It is worth noting that once a researcher gains access to families, response rates are relatively good so long as it is easy for them to take part.34 Our own experience in a recent study about parental caregiving (CIHR – MOP89984; S. Cadell, PI) when a child has a life-limiting illness illustrates the high response rate. Of the 367 potential participants who called our toll-free number, only 4 declined to participate after hearing about the study. After eliminating ineligible parents and a few who could not be contacted, a total of 339 survey packages were sent out. The return rate was 82.6% (n = 280 packages), but seven packages were unusable so the final sample size was 273. In another study exploring fathers’ experiences, only two of the 70 fathers who agreed to participate in the study were unavailable for a second interview. Families who choose to participate in pediatric palliative care research tend to be committed to following through on their participation.

Involving Children

Another issue in sampling is the inability of children to participate in research because of developmental and cognitive limitations associated with their young age or as a result of their illness. However, researchers must make special efforts to listen to children themselves whenever possible so that their points of view are also taken into account in the development of knowledge in pediatric palliative care.35 Creative ways of collecting data, such as video diaries, may be a useful complement to traditional ways of obtaining information.36

Gender and Diversity Challenges

Much of the research in pediatric palliative care in North America is based on English-speaking mothers who are Caucasian. In a recent review of 45 papers reporting on research about parents’ perspectives on pediatric palliative care, only 25% of the total sample was made up of fathers.37 Yet, there is some evidence of gender differences that may impact the types of interventions that should be offered to parents.38 An NIH-funded study is currently examining fathers’ experiences in pediatric palliative care (1R01 NR009430-01A1 [B. Davies, PI]). Similarly, a review of the prevalence of different languages and cultures in North American pediatric palliative care research found very few studies with greater than 20% non-Caucasian participants and only two that offered interviews or instruments in languages other than English.39 Work is needed to translate validated tools into languages other than English. Careful planning and use of novel study designs, methods of recruitment, and data collection may be needed to ensure that the voices of children, fathers, and families from various cultural backgrounds are included in research in pediatric palliative care.37,40,41

Research teams

Embedded in valuing the various perspectives that different disciplines bring to care, pediatric palliative care is usually delivered by an IDT. Similarly the conduct of research can be enhanced by working with an interdisciplinary research team. Though interdisciplinary research teams predominate in pediatric palliative care, one-discipline teams also have contributed to the field. For example, there has been work by physicians in symptom control, such as Joanne Wolfe24 and her team in the United States and John Collins with his team in Australia.

Given that children who receive palliative and end-of-life care can be found in many areas of the hospital and community, a team of researchers who come from several care areas may facilitate recruitment. Conducting research as a team can assist in overcoming some recruitment challenges, but it also can create others. A multidisciplinary research approach and multicenter studies may bring together a team of researchers with a different set of philosophies, which can be challenging to accommodate. Yet finding ways to learn from one another can lead to uncovering new and better ways of conducting research and contributing to quality care for children and families.

Research Team Functioning

We knew from the literature and our clinical experiences that we needed to study the trajectories of children requiring palliative care. So we met face to face to lay the groundwork for developing a proposal. Everyone had really good ideas, but our discussion went in circles for a time. We were getting frustrated because we seemed to be at cross-purposes and we could not zero in on the central issue. We stopped to reflect on the process, and then it hit us—we were talking about the same topic, but from very different perspectives. A couple of us were focused on children’s symptoms in orphaned diseases; another was imagining a study about the psychosocial aspects of children with cancer and other conditions; and others wanted to focus on the needs of families with children of varying diagnoses. Once we understood where we were all coming from, we could clearly identify and appreciate our varying points of view. It then became easy to name the paths of discovery that could be incorporated into our proposed study and to see how they fit with one another. Given our commitment to a non-hierarchical approach and to having a consensus in decisions, no single point of view took priority. Rather we combined all perspectives, including our various clinical and research backgrounds, and this approach resulted in a proposal for a much more comprehensive study than any one of us alone imagined or would have written. The synergy produced a longitudinal study, which was funded by CIHR and is in progress, that is innovative and opportune. It focuses on a population that is under-researched and not well-understood and will provide information about the symptoms of 300 children across Canada. These children have progressive metabolic, neurological, or chromosomal conditions and the study will include their family’s associated emotional, social, physical, and spiritual experiences. None of us separately would have seen the whole picture that was developed by the team.

Effective teams are built on respect, flexibility, and shared goals, and behaviors such as communication, continuous assessment of progress on goals, and taking time to have fun together. It also needs to have efficient and effective support from staff members who also are valued team members. Each member needs to work on understanding the views of others and must strive to respect and accept the differences that can arise when people come from various disciplines and philosophical and/or theoretical perspectives. It is not uncommon for members of two disciplines to use the same term, but to hold it to different meanings. Communication is essential so that misunderstandings and differing interpretations are recognized, discussed, and resolved to the greatest extent possible. Even something as simple as an acronym can cause confusion. For example, a physician or social worker may not understand a nurse who introduces herself as a CNS, because to them that term means central nervous system. The nurse, however, is a Clinical Nurse Specialist.

For a research team to be effective and productive, it is essential to lay a solid foundation where team functioning is detailed and formalized at the beginning, such as through a charter. The charter may include a process for decision making, inter-researcher relations, researcher-staff relations, researcher-collaborator relations and management roles, frequency of communication, roles and responsibilities of team members, guidelines for authorship, and so on (see Box 10-1 for a section of the PEDPALNET Charter on decision-making). Time spent in getting to know each other, and laying out expectations, including time commitment, and goals for the team at the start will enhance team functioning help get the team off to a good start. If team members are separated geographically, both face-to-face and frequent virtual meetings are essential to keep the team connected and allow for ongoing development. We recommend regularly scheduled teleconferences, as well as a web-based system to share files and develop and refine proposals and manuscripts, as ways of maintaining needed contact amongst members.

BOX 10-1 PEDPALNET Process for Decision Making

The PEDPALNET will use a modified “shades of agreement” approach where there are classes of agreement. Result is one of five classes: unconditional agreement, agreement with minor modifications, agreement with major modifications, agreement with strong reservations, such as “disagree, but will not stop the decision,” and full veto. In the case of full veto, discussion continues until one of the other four levels is reached.

A research team may be from a single institution or from multiple institutions. The team may come together for a single study or it may work multiple studies around a particular theme. Other collaborators may be involved on specific projects as needed. Teams that come together for a single study can certainly be effective; however, a team that comes together for multiple studies will have more opportunities to develop truly collaborative relationships within the team.

One of the challenges of conducting team research is the varied interests of each member. It may be difficult to include all members on every project. We have found it helpful to rotate the person responsible for each project, so it fits well with the leader’s interests and experience. In terms of authorship for the various publications that result from each project, we have found the International Committee of Medical Journal Editors (ICMJE) guidelines most useful.42 It also is best to discuss potential authors and the author order prior to writing. Finally, though collaborative research is valued in many areas, academic institutions may favor individual authorship. It is incumbent on researchers to familiarize their academic settings with the importance of collaborative research in an interdisciplinary field. Researchers also need them to be aware that a study from a multidisciplinary team is more likely to be applied to practice and thus to meet the knowledge translation requirements of major funding agencies.

Ethics

Even with the best research team assembled and a population available to meet the requirements of the research design, there may still be challenges to starting a study. These challenges may begin with an institutional ethics review board, which may include members reluctant to approve studies related to palliative, end-of-life, or bereavement care. Challenges also may come from the clinicians involved with families, who are reluctant to identify eligible participants even if the study has been ethically approved. Clinicians not familiar with pediatric palliative care research may have concerns about overburdening families during a difficult time, and they may try to protect families from these perceived burdens.

Children are under-represented in many areas of health research. They are considered to be inherently vulnerable due to their cognitive development, inability to understand the risks and benefits of taking part in research, and power differentials between the researcher and the child.1,43 When the child is also dying, a second layer of vulnerability is added. Dying persons are considered vulnerable because of: perceived mental and physical frailty, the risks of imposing additional psychological distress, the burden of spending time in research rather than with loved ones or enjoying activities, the concern that patients may feel an obligation to participate in research conducted by their clinicians.44

Proxy reports by parents about their child’s experience have frequently been used because of the ethical and methodological difficulties of including children in palliative and end-of-life research. Researchers comparing adult patients’ and their family members’ assessments of care quality found that the greatest congruence was between patients and the family members they lived with or saw every day.45 In pediatric care, parents generally spend a great deal of time with the ill child and so it is likely that they are the best proxy for the child. Given that children may be non-verbal due to age, their illness, or too weak to participate in research at end-of-life, it is likely that parent reports will continue to be an important component of pediatric palliative care research. However, a skilled clinician or researcher can work directly with children as young as six years.46 Based on the ethical principle of justice,47 it is important that children be given more opportunities to share in both the burdens and benefits of the research that has the potential for the greatest impact on meeting their needs and improving the quality of their lives and care.

Another ethical concern about conducting research with dying persons is that the principle of beneficence is difficult to meet—the patient generally does not receive direct benefit from taking part in the research.47 This concern holds particularly true in pediatric palliative care research, where most studies are qualitative or descriptive and are primarily designed to gain knowledge that will improve the care provided in the future.48 However, adult patients facing death who participate in research have identified benefits of research participation, such as giving the opportunity for social interaction, making a contribution to society, and discussing their experiences.49 Similarly, there is some evidence that adolescents with cancer nearing death take part in research for altruistic reasons, hoping to help others and bring some meaning to their illness.50 Bereaved parents who have participated in research identify personal benefits including being able to express feelings; revisiting, reflecting on, and making sense of their experiences; and bringing some meaning to the child’s life and death.51 From our experiences, many families in pediatric palliative care identify benefits of research participation and want to decide for themselves about participating in research. The majority of study participants are willing to be contacted about future research. For example, in our recent caregiving study previously discussed more than 90% of the 273 parents indicated their interest in future research and wanted us to contact them if we developed a suitable study.

Family-centered care is the norm in pediatrics and is based on the recognition that there are, in effect, two patients – the child and the family.52 In addition to acting as proxies for the child, parents are an important group to be included in research to better understand their experiences and need for support. Similarly, research with siblings is important. Ethical concerns about involving siblings in research are similar to those outlined previously because the sibling is usually a child and, therefore, considered inherently vulnerable. Yet, it could be considered paternalistic if siblings are not asked about their experiences. Further, who better to report on their experiences than the siblings themselves?

There is some concern from health professionals and ethics boards that interviewing bereaved families may cause them further pain. However, some studies have highlighted parents’ eagerness to share the story of their child’s death and to provide input to help other families53–55 and a few have included a component to systematically examine the emotional impact of participating in research. In the majority of studies, there were no parent reports of negative impact or distress caused by participation.34,51,56,57 Parents in other studies have reported some degree of negative impact,58 emotional difficulty,59 or stress60 as a result of taking part. However, in one of these studies 99% of parents reported that they thought the research was valuable.58 Parents also have reported that even though they became tearful or very emotional while talking about their child, it did not mean they wanted or needed to end the interview.56,61,62

Hinds et al.34 provided details from a number of studies related to end-of-life care in pediatric oncology and discussed the challenges faced in obtaining study approval from ethics review boards. Their research also offered methods for overcoming the challenges. It is important to include evidence about the benefits of pediatric palliative care research in grant and ethics proposals. As well, it may be useful to include a component within new studies to assess the participants’ perspectives of the benefits and burdens of taking part in the study. We have added a few questions about impact of participation to a longitudinal study we recently started (CIHR – MOP79526, R. Steele & H. Siden, Co-PIs). Parents of children with life-limiting conditions who complete surveys every six months for 18 to 48 months, depending on when they are accrued, will provide information about impact after one year and at the end of participation. Most studies to date have assessed the impact on bereaved parents of taking part in cross-sectional studies that use interviews or written surveys. Little is known about the burdens on parents associated with focus groups, longitudinal studies, and prospective designs, or the burdens of research that may be placed on the ill child or siblings. However, our experiences are that perceived burden by clinicians and ethics board members may not be the same as actual burden.

The risk of inflicting harm on participants in research about pediatric palliative care is certainly a real one. However, this risk does not mean that research should not be done. Families have clearly indicated that research is valuable even when it may be emotionally difficult for them.56,58 There is growing evidence that children and parents can have their autonomy preserved in being given choices about if, when, and how to take part in research and that they may experience direct benefit from participation even when the study design is primarily meant to benefit future families. Researchers are encouraged to use and expand on this body of evidence, but also to be cognizant of the risks. A good research proposal will identify the areas for potential harm or risk and have plans to prevent, minimize, or address it should it occur. These plans should include careful training and ongoing support for those involved in recruiting or collecting data from participants, lists of easily accessible support services, and availability of emergency supports if a participant becomes severely upset during the research.26

Dissemination and Knowledge Exchange

Though still limited, the knowledge base in pediatric palliative care is growing. The new challenge is to translate this knowledge into improved care for children and their families. Traditional dissemination methods, such as publications in peer-reviewed journals, can be difficult because the field remains relatively small, so an editor may not be willing to report results that will not be used by the majority of the journal’s readers. Further, trying to publish descriptive and qualitative research in high-impact journals may pose a challenge. There is no specific pediatric palliative care journal, so reaching the right audience can be difficult. In addition, while pediatric and palliative care journals are useful vehicles for publication, there is a tendency to target specific audiences though the knowledge needs to be shared among the professionals who provide interdisciplinary, pediatric palliative care. Yet, given the nature of the field, the development of a specific journal faces its own challenges, such as finding qualified reviewers, attracting sufficient contributors, and obtaining funds to support a journal. In the meantime, potential authors are encouraged to target the mainstream journals and to find other ways of sharing their work.

Most funding agencies now require a knowledge translation plan that goes beyond the traditional venues of conference presentations and publications. In Canada, the CIHR encourages interaction between researchers and the public by funding Café Scientifiques. These CIHR Cafés are designed for the general public and are advertised through various media outlets. Everyone is welcome to attend and there is no admission fee. Our team has held two successful Cafés in coffee shops where we gave short presentations related to pediatric palliative care and then held a question and answer period. The public was very enthusiastic about the format and strongly supported more of these events. They also enjoyed the refreshments that were provided as an integral part of the Café. CIHR also funds some end-of-grant dissemination workshops for researchers, clinicians, and policy makers to meet and facilitate uptake of research into practice. Funding for such innovative dissemination strategies is crucial and researchers need to lobby funding agencies so that support in this burgeoning field of pediatric palliative care is forthcoming.

Researchers in pediatric palliative care need to build capacity in order to address the research priorities. Clinicians who want to conduct their own studies may need to seek additional training and mentorship. Beginning researchers also may need to work with more established researchers or teams to gain experience before leading large projects. For clinicians in particular, there may be opportunities to be involved as a site collaborator in multicenter projects conducted by experienced researchers or to take a lead role in a small part of a much larger study. These experiences may act as stepping stones to developing their own studies. There also is a role for clinicians to translate research that has been done by others into best practices in their own institutions.

Summary

The rewards and joys of conducting research that can improve the life and death of a child and the experience of family members and caregivers are well worth the care and effort required. This effort includes gaining research knowledge, skills, and experience; selecting important research questions; designing rigorous but sensitive studies; and overcoming the inevitable challenges that all researchers face but that may be particularly difficult in pediatric palliative care.

Acknowledgments

This work was supported by Canadian Institutes of Health Research (CIHR) grant, New Emerging Team (NET) Transitions in Pediatric Palliative Care (PET-69769). Kimberley Widger received a PhD Research Trainee Award related to this grant, a TD Meloche Monnex Scholarship from the Canadian Nurses Foundation, and a University of Toronto Fellowship.

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