Skip to main content
Download PDF

Promoting an ethic of engagement in pediatric palliative care research

BMC Palliative Care volume 14, Article number: 50 (2015) Cite this article

Abstract

Background

This paper defends the ethical and empirical significance of direct engagement with terminally ill children and adolescents in PPC research on health-related quality of life. Clinical trials and other forms of health research have resulted in tremendous progress for improving clinical outcomes among children and adolescents diagnosed with a life-threatening illness. Less attention has been paid, however, to engaging this patient population directly in studies aimed at optimizing health-related quality of life in PPC. Though not restricted to care at the end of life, PPC—and by extension PPC research—is in part dependent on recognizing the social complexities of death and dying and where health-related quality of life is a fundamental element. To explore these complexities in depth requires partnership with terminally ill children and adolescents, and acknowledgement of their active social and moral agency in research.

Discussion

Principles of pediatric research ethics, theoretical tenets of the “new sociology of the child(hood),” and human rights codified in the United Nations Convention on the Rights of the Child (UNCRC) underpin the position that a more engagement-centered approach is needed in PPC research. The ethics, sociologies and human rights of engagement will each be discussed as they relate to research with terminally ill children and adolescents in PPC. Qualitative method(ologies) presented in this paper, such as deliberative stakeholder consultations and phenomenology of practice can serve as meaningful vehicles for achieving i) participation among terminally ill children and adolescents; ii) evidence-bases for PPC best practices; and iii) fulfillment of research ethics principles.

Conclusion

PPC research based on direct engagement with PPC patients better reflects their unique expertise and social epistemologies of terminal illness. Such an approach to research would strengthen both the ethical and methodological soundness of HRQoL inquiry in PPC.

Peer Review reports

Background

According to the American Academy of Pediatrics, the “goal of pediatric palliative care is to add life to the child’s years, not simply add years to the child’s life” [1]. Promoting a research agenda that makes central the participation of terminally ill children and adolescents in pediatric palliative care (PPC) is needed to ensure the former, and to improve PPC services in line with their lived experiences. While the focus of this paper will be PPC research at the end of life, PPC delivery is not restricted to this point in the care trajectory. Rather, it is well established that PPC should be integrated as early as possible in the care process [2]. Despite global recognition of the need for enhancing the availability and accessibility of PPC, systematic reviews of the literature reveal unmet needs in both developed and developing countries [3-5]. In response, concerted efforts to expand PPC  has enabled i) better identification of eligible patient populations; ii) greater attention to symptom control needs at the end of life,; iii) empirical research investigating health-related quality of life (HRQoL) [6, 7] and iv) evaluation of cost-benefit outcomes [8–10].

Research with terminally ill children and adolescents should therefore also parallel the service expansion of PPC in order to meet demands: “Research involving children and their families occupies a small niche in the world of research in palliative and end-of-life care, which itself is small in comparison to other areas of clinical and health services research” [11]. Considering that healthcare professionals have an ethical duty to palliate when a child is believed to be suffering at the end of life [12, 13], the lacuna of evidence-based care practices derived from PPC research with patients can be both ethically questionable and methodologically unsound [14]. To this end, HRQoL inquiry with terminally ill children and adolescents has been identified as an important, albeit under-researched domain in PPC [6, 15].

HRQoL is a socioculturally complex phenomena that has been used extensively in palliative care research to inform best practices [16, 17]. Yet, “Few reliable, valid, and developmentally appropriate methods are available for measuring the suffering and quality of life of children with life-threatening illness…” [18]. The intensely personal nature of HRQoL necessitates that research investigating its influence on PPC delivery engage participants directly as a rule whenever possible.Footnote 1 Although greater engagement of terminally ill children and adolescents is emerging as a new priority in PPC research [4, 19], limited attention has been paid to their illness experience(s) in PPC for quality of care improvement [20]. Most studies investigating HRQoL among terminally or critically ill children and adolescents overwhelmingly represents the perspectives of parental and/or health professional proxies [21–25]. A recent study of parents’ ability to represent the ‘voice of the child’ in end of life decision-making found, however, that parents often have “difficulty in gaining an insight into their child’s perspective even though they may be intensely involved in his or her care and support during the [end of life] phase” [26]. Where children are engaged in HRQoL research within other pediatric health domains, the level of agreement between self- and proxy-reporting yield mixed results [27–33]. The marked absence of children and adolescents in studies meant to better understand their perspectives on terminal illness [34–36] can be attributed in part to the relative rarity of such illnesses in this population [37, 38]; misconception surrounding the care goals of PPC [39]; a shortage of PPC specialty training [40–42]; and stringent regulations governing the involvement of children in research [43]. Matza et al. nuance the challenges of assessing HRQoL in this population, specifically: “As more pediatric clinical trials have been initiated, however, researchers have encountered a unique set of challenges involved in assessing HRQL among children, such as identifying the age at which children can reliably report various domains of HRQL and determining whether children or their parents are the best respondents” [44].

Caring for terminally ill children and adolescents should be reflective of the complex social dimensions of death and dying patients themselves face if PPC is to be truly patient-centered [6]. Underrepresentation of this crucial stakeholder population (terminally ill children and adolescents) can misguide the development of best practices and fall short of fulfilling the ethical mandates for respect persons and justice, among others. A recent proposal of a ‘charter for the rights of the dying child’ attests to this: “To have access to child-specific palliative-care programmes that avoid futile or excessively burdensome practices and therapeutic abandonment” [45]. Engaging children and adolescents in PPC research enhances their position as a critical stakeholder community for whom effective service delivery should be designed. Pursuant to this goal, the United Nations Convention on the Rights of the Child (UNCRC) and new social theories on the child(hood) can inform the ethical frameworks upon which increased pediatric participation in PPC research can build. Each of these frameworks will be discussed in turn in the subsequent sections.

Social theories and applicability to PPC

Research with terminally ill children and adolescents can accentuate the classical tensions that ethics review committees and researchers consider when involving minors in research. This tension follows that children warrant special protections due to their unique situation(s) of vulnerability, yet should not be categorically excluded from research that could lead to important health(care) advancements for them. Promoting research with—as opposed to on—children is inspired in part by “new sociologies of the child” [46]. The concepts articulated in the new sociology of the child, herein referred to as the new sociology, and those later codified in the UNCRC support the greater inclusion of terminally ill children and adolescents in PPC research.

Of the four guiding principles outlined in the UNCRC, respect for the views of the child under Article 12 lends the strongest support of children’s inclusion and participation in (health) research broadly [47]:

States Parties shall assure to the child who is capable of forming his or her own views the right to express those views freely in all matters affecting the child, the views of the child being given due weight in accordance with the age and maturity of the child.

While Article 12 is one of the most celebrated achievements of the UNCRC, it has also garnered the most controversy [48]. Article 12 explicitly safeguards participatory rights granted to children and adolescents, underscoring the prioritization and legal legitimacy of their voice. It is groundbreaking, “not only for what it says, but because it recognizes the child as a full human being with integrity, personality and the ability to participate freely in society” [49].

Taken together, the UNCRC and the new sociology share many theoretical underpinnings that substantiate a rights-based approach to child participation and protection. Lavalette and Cunningham summarize the central tenets of the new sociology:

  1. 1)

    Childhood is not merely a biological phenomenon, but a social construction, affected and shaped by wider social and cultural elements, within concrete, historical circumstances;

  2. 2)

    Children occupy and conduct themselves in worlds that are full of meaning for them, but about which adults are, at least partially, ignorant. It has led to an emphasis on listening to children’s voices;

  3. 3)

    Politically, children are powerless and disadvantaged. The new sociology is a theory of advocacy, sociology for children rather than sociology of children. This approach has closely tied into children’s rights agenda; and

  4. 4)

    Children are an identifiable social group, with a common [basic] set of needs and rights [50].

Each tenet will be discussed in turn as they relate to existing pediatric research ethics norms and the UNCRC.

An ethical justification of PPC for children, by children

First, societal determinations of ‘childhood’ and ‘adulthood’ are largely the result of physiological and psychological benchmarking. Taking from early child development theory according to Piaget, maturity (and hence autonomous decision-making) surrounding death and dying is presumed to evolve chronologically with age [51]. Maturity is legally recognized to reach full development at the age of majority, 18 in most Western jurisdictions. This accepted linearity of a child’s capacity to make informed decisions is the basis upon which legal standards of a child’s capacity to consent to research (and care) are premised [52]. The primary rationale for an age of majority is then to categorically define the point when a child is capable of making informed decisions, such as the ability to consent to research. Diagnosis of a terminal illness and the pending outcome of death, however, truncates the burgeoning process of autonomy [51, 53]. Placing the same age of majority restrictions on PPC patients’ ability to participate in HRQoL research can be methodologically inappropriate for what is often considered minimal risk, qualitative research. This is particularly the case when recruitment of the research population of interest is critical to addressing knowledge gaps regarding HRQoL for terminally ill children and adolescents receiving PPC. Prevailing assumptions of when a minor reaches full decision-making ability and the level of vulnerability they experience as a result can be (unjustified) barriers to involvement in HRQoL research when taking into account the limited timeframe in which they can participate.Footnote 2

Second, studies investigating child health, including at the end of life, cannot extrapolate findings from adult studies. Just as HRQoL assessment tools have been created specifically for use in pediatric populations, HRQoL research in pediatric palliative care should also be population-specific: “To identify practices that affect the quality of life experienced by a child with a life-threatening medical problem requires measurement tools that can reliably and validly reflect the child’s experiences, particularly when the problem has reached an advanced stage and death is expected or possible in the foreseeable future” [11]. Despite widespread clinical interest, there HRQoL infrequently engages children and adolescents directly in the development of PPC best care practices [54–56].

Moreover, the heterogeneity of ‘childhoods’, family contexts and end-of-life beliefs necessitate thoughtful consideration of research methods that can best describe the worlds children occupy. From a methodological standpoint, researcher reflexivity and coherent study design are critical for meeting bioethical mandates as well. As such, lending primacy to children and adolescents’ voices is one avenue for respecting their agency in healthcare settings, including in PPC.Footnote 3 Hart is perhaps best known for his work in this field, describing how participation is stepwise on a ladder that culminates in an ideal empowerment model. Hart loosely defines participation as referring to “the process of sharing decisions which affect one’s life and the life of the community in which one lives…Participation is the fundamental right of citizenship” [57].

Third, in line with the ideas of participation Hart proposes, the new sociology’s emphasis on power can encourage a discussion of social and distributive justice for the inclusion of terminally ill children and adolescents in PPC research. Canada’s Tri Council Policy Statement: Ethical Conduct for Research Involving Humans (TCPS 2) speaks to this point directly:

The principle of Justice holds that particular individuals, groups or communities should neither bear an unfair share of the direct burdens of participating in research, nor should they be unfairly excluded from the potential benefits of research participation…Over-protectionist attitudes or practices of researchers or REBs, whether intentional or inadvertent, can exclude some members of society from participating in research…The inclusion of the young and the elderly in research, for example, ensures that treatments frequently given to these populations are effective and safe [58].

Terms of engagement in research practice

Certainly, a commitment to research engagement with children and adolescents demands methodological consideration. Some scholars have argued the ethics of PPC research compounds the already pressing concerns regarding vulnerability in research with children generally; others deem research in palliative care ipso facto unethical [59]. We posit that neither children’s terminal illnesses nor the extent of their (presumed) vulnerability as a result should categorically prevent research in this area. Rapoport shares this view, urging researchers that, “The imperative to practice evidence-based medicine and strive toward optimal patient care compels us to find ways to overcome obstacles to conducting research on vulnerable patients, not by avoidance, but by confronting them directly” [41].

Engagement can therefore take on a variety of forms and depths in research practice. Ensuring assent is obtained from pediatric participants is a requisite first step. Although there is no consensus on a definition, nor the exact circumstances under which assent should be sought from children or adolescents, it is nevertheless the foundation of ethical engagement [60]. Democratic and non-tokenistic inclusion underlines the notion of participation Hart proposes, but is worth mentioning this norm is more widely accepted in Western contexts. A number of participatory methods have been recommended to overcome the ethical challenges of avoiding tokenistic involvement of children and adolescents in research [61] and other methods for HRQoL research, specifically [62].

Two methodological methods and approaches are proposed to complement the deeply personal nature of HRQoL inquiry with terminally ill children and adolescents. A research team comprised of authors VR, GB, LC and CL, in partnership with the International Childhood Astrocytoma iNtegrated Genomic and Epigenomic (iCHANGE) Consortium is piloting a deliberative stakeholder consultation method to explore facilitators and barriers to HRQoL among children and adolescents with terminal brain tumors (Longo C, Bartlett G, Rahimzadeh V, Crimi L. Deliberative Stakeholder Consultations: Giving Vulnerable Patients a Voice in Genetic Research. Forthcoming. [63]). Contrary to focus groups and other similar qualitative approaches such as citizen juries, the deliberative stakeholder consultation limits researcher mediation [64]. It aims to mitigating power differentials between researchers and participants that have been the source of considerable debate in both the health research [65–67] and bioethics communities. The hallmark of deliberative methods, such as those we use to engage terminally ill children and adolescents in our ongoing study, reflects the idea that “deliberation is more than merely a discussion of the issues. Emphasis is also given to the product that arises from discussion (e.g., a decision or set of recommendations), and the process through which that product comes about” [68].

With equal emphasis placed on understanding through engagement, a phenomenology of practice approach provides further methodological guidance for HRQoL inquiry with PPC patients. van Manen writes the primary aim of a phenomenology of practice-inspired research strives, “…to open up possibilities for creating formative relations between being and acting, between who we are and how we act, between thoughtfulness and tact” [69]. A phenomenology of practice in PPC research would give precedence to the theoretical dimensions of HRQoL as defined by PPC patients themselves.

Conclusion

An ethic of engagement in PPC research defended in this paper activates the rights to free expression and participation of the UNCRC. It advocates for  the involvement of children and adolescents in research meant to better inform PPC delivery; improves care for future patients; promotes accessibility and availability of PPC services that are reflective of children’s experiences at the end of life; expands modes of representation among the PPC population in health research; and achieves direct stakeholder engagement with primary users of PPC services. Drawing on ethical and methodological arguments reinforced by the new sociology of the child(hood) and qualitative research, respectively, the authors propose the engagement of children and adolescents is central to augmenting care capacities in PPC.

There is yet so much to learn from children of the death and dying process in order to better care for future children during the death and dying process. The continued underrepresentation of terminally ill children and adolescents in PPC research, however, fails to ground modalities of care in the unique and evolving clinical realities they face as PPC patients. Because ensuring an optimal HRQoL is the goal of PPC, it is an ethical and methodological imperative that research in this field is inclusive of primary stakeholder populations, namely PPC patients. The deliberative stakeholder consultation and phenomenology of practice approach are two methodological decisions that can help PPC researchers achieve such inclusion. While the former has been used to engage stakeholders in other domains of health research [70], assessment of its methodological utility for exploring HRQoL in the PPC research context is currently underway.

How research engagement with PPC patients will be affected by shifts in conceptions of the child(hood) remain to be seen. But, “Ultimately the children’s rights project is not just about making a better world for children, it is about making a better world for all of us” [71]. Part of granting greater legitimacy to children’s moral and social agency, particularly in the face of terminal illness, is rendering their continued absence in PPC an antiquated protectionist argument of the past. Doing so can make way for more creative and inclusive methods in PPC research that indeed “add life to a child’s years.” The spirit of engagement promulgated by the deliberative stakeholder consultation method could also be transferable to improving understanding of HRQoL for adults in similar situations of vulnerability, namely elderly patients living with dementia [72].

The year 2014 marked the 25th anniversary of the UNCRC’s adoption, and was coined the “Year of Innovation for Equity.” In addition to encouraging novel approaches to equity, it gives us pause to reflect on the victories of the UNCRC, and spotlight areas for improvement in furthering the rights and welfare of children and adolescents worldwide. Healthcare is an ideal setting in which to strive towards realizing the ideals of participation, protection and provision the UNCRC espouses as it invokes the most basic human right afforded to all: the right to health. The emergence of international initiatives in PPC research, and identification of its pressing need in the pediatric community is one of many victories.

Notes

  1. Some circumstances complicate children’s ability to participate in research, to be sure. This can be particularly true for research using language-based methods (such as in qualitative research) with infants, children who are otherwise nonverbal, or those who suffer from severe cognitive impairment.

  2. The ethical complexities concerning mature minors and clinical decision-making typify this case scenario. A number of studies have sought to gauge the level of decision-making ability and maturity that minors demonstrate in the face of critical illness (see for example Miller et al. [73]) where findings indicate some adolescents exhibit abilities that match, or surpass, those of consenting adults. On this point, there is some consensus in the adolescent psychology literature that suggest minors appeal to morality and logic in informing their decisions by middle adolescence [74].

  3. It is worth noting that some scholars argue listening to children’s voices alone is not sufficient to promote due representation [71] or can be tokenistic if not followed up with measurable action [57, 75].

Abbreviations

PPC:

Pediatric palliative care

HRQoL:

Health-related quality of life

UNCRC:

United Nations Convention on the Rights of the Child

TCPS 2:

Tri Council Policy Statement: Ethical Conduct for Research Involving Humans, 2nd edition

References

  1. American Academy of Pediatrics Committee on Bioethics and Committee on Hospital Care. Palliative care for children. Pediatrics. 2000;106:351–7.

    Article  Google Scholar 

  2. Liben S. Palliative care for children: Is it really needed? Am J Hosp Palliat Med. 2000;17:294–5.

    Article  CAS  Google Scholar 

  3. Knapp C, Woodworth L, Wright M, Downing J, Drake R, Fowler-Kerry S, et al. Pediatric palliative care provision around the world: a systematic review. Pediatr Blood Cancer. 2011;57:361–8.

    Article  PubMed  Google Scholar 

  4. Downing J, Knapp C, Muckaden MA, Fowler-Kerry S, Marston J. Priorities for global research into children’s palliative care: results of an International Delphi Study. BMC Palliat Care. 2015;14:36.

    Article  PubMed  PubMed Central  Google Scholar 

  5. Linton JM, Feudtner C. What accounts for differences or disparities in pediatric palliative and end-of-life care? A systematic review focusing on possible multilevel mechanisms. Pediatrics. 2008;122:574–82.

    Article  PubMed  Google Scholar 

  6. Feeny D, Furlong W, Mulhern RK, Barr RD, Hudson M. A framework for assessing health-related quality of life among children with cancer. Int J Cancer Suppl. 1999;12:2–9.

    Article  CAS  PubMed  Google Scholar 

  7. Wolfe J, Orellana L, Cook EF, Ullrich C, Kang T, Geyer JR, et al. Improving the care of children with advanced cancer by using an electronic patient-reported feedback intervention: results from the PediQUEST randomized controlled trial. J Clin Oncol. 2014;32:1119–26.

    Article  PubMed  PubMed Central  Google Scholar 

  8. Pascuet E, Cowin L, Vaillancourt R, Splinter W, Vadeboncoeur C, Grandmaison Dumond L, et al. A comparative cost-minimization analysis of providing paediatric palliative respite care before and after the opening of services at a paediatric hospice. Healthc Manag Forum. 2010;23:63–6.

    Article  Google Scholar 

  9. Ward-smith P, Korphage RM. Where health care dollars are spent when pediatric palliative care is provided. Nurs Econ. 2008;26:175–9.

    PubMed  Google Scholar 

  10. Gans D, Kominski GF, Roby DH, Diamant AL, Chen X, Lin W, et al. Better outcomes, lower costs: palliative care program reduces stress, costs of care for children with life-threatening conditions. Policy Brief UCLA Cent Health Policy Res 2012;(PB2012-3):1-8.

  11. Field M, Behrman R. Committee on Palliative and End-of-Life Care for Children and Their Families: When Children Die: Improving Palliative and End-of-Life Care for Children and Their Families. Washington: National Academies Press; 2003.

    Google Scholar 

  12. Norton CK, Joos OH. Caring for Catherine: a cry to support ethical activism. J Pediatr Oncol Nurs. 2005;22:119–20.

    Article  PubMed  Google Scholar 

  13. Wolfe J. Suffering in children at the end of life: recognizing an ethical duty to palliate. J Clin Ethics. 2000;11:157–63.

    CAS  PubMed  Google Scholar 

  14. Theunissen N, Vogels T, Koopman H, Verrips G, Zwinderman K, Verloove-Vanhorick S, et al. The proxy problem: child report versus parent report in health-related quality of life research. Qual Life Res. 2014;7:387–97.

    Article  Google Scholar 

  15. Friedman DL, Hilden JM, Powaski K. Issues and challenges in palliative care for children with cancer. Curr Pain Headache Rep. 2005;9:249–55.

    Article  PubMed  Google Scholar 

  16. Detmar SB, Muller MJ, Schornagel JH, Wever LDV, Aaronson NK. Role of health-related quality of life in palliative chemotherapy treatment decisions. J Clin Oncol. 2002;20:1056–62.

    Article  CAS  PubMed  Google Scholar 

  17. Sprangers MA, Schwartz CE. Integrating response shift into health-related quality of life research: a theoretical model. Soc Sci Med. 1999;48:1507–15.

    Article  CAS  PubMed  Google Scholar 

  18. Himelstein BP, Hilden JM, Boldt AM, Weissman D. Pediatric palliative care. N Engl J Med. 2004;350:1752–62.

    Article  CAS  PubMed  Google Scholar 

  19. Ullrich C, Morrison RS. Pediatric palliative care research comes of age: what we stand to learn from children with life-threatening illness. J Palliat Med. 2013;16:334–6.

    Article  PubMed  PubMed Central  Google Scholar 

  20. Margherita G, Martino ML, Recano F, Camera F. Invented fairy tales in groups with onco-haematological children. Child Care Health Dev. 2014;40:426–34.

    Article  CAS  PubMed  Google Scholar 

  21. Blume ED, Balkin EM, Aiyagari R, Ziniel S, Beke DM, Thiagarajan R, et al. Parental perspectives on suffering and quality of life at end-of-life in children with advanced heart disease: an exploratory study. Pediatr Crit Care Med. 2014;15:336–42.

    Article  PubMed  Google Scholar 

  22. Tomlinson D, Bartels U, Gammon J, Hinds PS, Volpe J, Bouffet E, et al. Chemotherapy versus supportive care alone in pediatric palliative care for cancer: comparing the preferences of parents and health care professionals. CMAJ. 2011;183:E1252–8.

    Article  PubMed  PubMed Central  Google Scholar 

  23. Knapp C, Madden V, Revicki D, Feeny D, Wang H, Curtis C, et al. Health status and health-related quality of life in a pediatric palliative care program. J Palliat Med. 2012;15:790–7.

    Article  PubMed  Google Scholar 

  24. Sheetz MJ, Bowman M-AS. Parents’ perceptions of a pediatric palliative program. Am J Hosp Palliat Care. 2013;30:291–6.

    Article  PubMed  Google Scholar 

  25. Kassam A, Skiadaresis J, Habib S, Alexander S, Wolfe J. Moving Toward Quality Palliative Cancer Care: Parent and Clinician Perspectives on Gaps Between What Matters and What Is Accessible. J Clin Oncol. 2012;31:910–5.

    Article  PubMed  Google Scholar 

  26. Kars MC, Grypdonck MHF, de Bock LC, van Delden JJM. The parents’ ability to attend to the “voice of their child” with incurable cancer during the palliative phase. Health Psychol. 2015;34:446–52.

    Article  PubMed  Google Scholar 

  27. Bray P, Bundy AC, Ryan MM, North KN, Everett A. Health-related quality of life in boys with Duchenne muscular dystrophy: agreement between parents and their sons. J Child Neurol. 2010;25:1188–94.

    Article  PubMed  Google Scholar 

  28. Penn A, Shortman RI, Lowis SP, Hons BA, Stevens MCG, Hunt LP, et al. Child-Related Determinants of Health-Related Quality of Life in Children With Brain Tumours 1 Year After Diagnosis. Pediatr Blood Cancer. 2010;55(7):1377–85.

    Article  PubMed  Google Scholar 

  29. Noyes J. Comparison of ventilator-dependent child reports of health-related quality of life with parent reports and normative populations. J Adv Nurs. 2007;58:1–10.

    Article  PubMed  Google Scholar 

  30. Havermans T, Vreys M, Proesmans M, De Boeck C. Assessment of agreement between parents and children on health-related quality of life in children with cystic fibrosis. Child Care Health Dev. 2006;32:1–7.

    Article  CAS  PubMed  Google Scholar 

  31. Varni JW, Burwinkle TM, Sherman SA, Hanna K, Berrin SJ, Malcarne VL, et al. Health-related quality of life of children and adolescents with cerebral palsy: hearing the voices of the children. Dev Med Child Neurol. 2005;47:592–7.

    Article  PubMed  Google Scholar 

  32. Cremeens J, Eiser C, Blades M. Factors influencing agreement between child self-report and parent proxy-reports on the Pediatric Quality of Life Inventory 4.0 (PedsQL) generic core scales. Health Qual Life Outcomes. 2014;4:58.

    Article  Google Scholar 

  33. Eiser C, Morse R. Can parents rate their child’s health-related quality of life? Results of a systematic review. Qual Life Res. 2001;10:347–57.

    Article  CAS  PubMed  Google Scholar 

  34. Akard TF, Gilmer MJ, Friedman DL, Given B, Hendricks-Ferguson VL, Hinds PS. From qualitative work to intervention development in pediatric oncology palliative care research. J Pediatr Oncol Nurs. 2013;30:153–60.

    Article  PubMed  PubMed Central  Google Scholar 

  35. Carroll JM, Santucci G, Kang TI, Feudtner C. Partners in pediatric palliative care: a program to enhance collaboration between hospital and community palliative care services. Am J Hosp Palliat Care. 2007;24:191–5.

    Article  PubMed  Google Scholar 

  36. Bates AT, Kearney JA. Understanding death with limited experience in life. Curr Opin Support Palliat Care. 2015;9:40–5.

    Article  PubMed  Google Scholar 

  37. Ward E, DeSantis C, Robbins A, Kohler B, Jemal A. Childhood and adolescent cancer statistics, 2014. CA Cancer J Clin. 2014;64:83–103.

    Article  PubMed  Google Scholar 

  38. Ellison LF, Pogany L, Mery LS. Childhood and adolescent cancer survival: a period analysis of data from the Canadian Cancer Registry. Eur J Cancer. 2007;43:1967–75.

    Article  PubMed  Google Scholar 

  39. Waldman E, Wolfe J. Palliative care for children with cancer. Nat Rev Clin Oncol. 2013;10:100–7.

    Article  PubMed  Google Scholar 

  40. Hughes MT, Smith TJ. The growth of palliative care in the United States. Annu Rev Public Health. 2014;35:459–75.

    Article  PubMed  Google Scholar 

  41. Rapoport A, Obwanga C, Sirianni G, Librach SL, Husain A. Not just little adults: palliative care physician attitudes toward pediatric patients. J Palliat Med. 2013;16:675–9.

    Article  PubMed  Google Scholar 

  42. Schaefer K, Chittenden E. Raising the Bar for the Care of Seriously Ill Patients: Results of a National Survey to Define Essential Palliative Care Competencies for Medical Students and. Acad Med 2014;89:1024–1031.

  43. Diekema DS. Conducting ethical research in pediatrics: a brief historical overview and review of pediatric regulations. J Pediatr. 2006;149(1 Suppl):S3–S11.

    Article  PubMed  Google Scholar 

  44. Matza LS, Swensen AR, Flood EM, Secnik K, Leidy NK. Assessment of health-related quality of life in children: a review of conceptual, methodological, and regulatory issues. Value Health. 2004;7:79–92.

    Article  PubMed  Google Scholar 

  45. Benini F, Vecchi R, Orzalesi M. A charter for the rights of the dying child. Lancet. 2014;383:1547–8.

    Article  PubMed  Google Scholar 

  46. James A, Prout A. Constructing and Reconstructing Childhood: Contemporary Issues in the Sociological Study of Childhood. 2nd ed. London: Falmer Press; 1997.

    Google Scholar 

  47. Understanding the CRC | Convention on the Rights of the Child | UNICEF. 2014.

  48. Smolin DM. Overcoming Religious Objections To the Convention on the Rights of the Child. Emory Int Law Rev. 2006;20:81–110.

    Google Scholar 

  49. Freeman MDA. The Moral Status of Children: Essays on the Rights of the Children. The Hague: Martinus Nijhoff Publishers; 1997.

    Google Scholar 

  50. Lavalette M, Cunningham S. The New Sociology of Childhood. In: Goldson B, Lavalette M, McKechnie J, editors. Children, Welfare and the State. London: SAGE; 2002.

    Google Scholar 

  51. Stillion JM, Papadatou D. Suffer the Children: An Examination of Psychosocial Issues in Children and Adolescents with Terminal Illness. Am Behav Sci. 2002;46:299–315.

    Article  Google Scholar 

  52. Alderson P. In the genes or in the stars? Children’s competence to consent. J Med Ethics. 1992;18:119–24.

    Article  CAS  PubMed  PubMed Central  Google Scholar 

  53. Hinds PS, Drew D, Oakes LL, Fouladi M, Spunt SL, Church C, et al. End-of-life care preferences of pediatric patients with cancer. J Clin Oncol. 2005;23:9146–54.

    Article  PubMed  Google Scholar 

  54. Liben S, Papadatou D, Wolfe J. Paediatric palliative care: challenges and emerging ideas. Lancet. 2008;371:852–64.

    Article  PubMed  Google Scholar 

  55. Balaguer A, Martín-Ancel A, Ortigoza-Escobar D, Escribano J, Argemi J. The model of Palliative Care in the perinatal setting: a review of the literature. BMC Pediatr. 2012;12:25.

    Article  PubMed  PubMed Central  Google Scholar 

  56. Stevenson M, Achille M, Lugasi T. Pediatric palliative care in Canada and the United States: a qualitative metasummary of the needs of patients and families. J Palliat Med. 2013;16:566–77.

    Article  PubMed  Google Scholar 

  57. Hart RA. Children’s Participation: From Tokenism to Citizenship. Florence: UNICEF ICDC; 1992.

    Google Scholar 

  58. Canadian Institutes of Health Research Natural Sciences and Engineering Research Council of Canada and Social Sciences and Humanities Research Council of Canada: Tri-Council Policy Statement: Ethical Conduct for Research Involving Humans. 2014.

  59. De Raeve L. Ethical issues in palliative care research. Palliat Med. 1994;8:298–305.

    Article  PubMed  Google Scholar 

  60. Giesbertz NAA, Bredenoord AL, van Delden JJM. Clarifying assent in pediatric research. Eur J Hum Genet. 2014;22:266–9.

    Article  PubMed  Google Scholar 

  61. Thomas N, O’Kane C. The ethics of participatory research with children. Child Soc. 1998;12:336–48.

    Article  Google Scholar 

  62. Drotar D. Measuring Health-Related Quality of Life in Children and Adolescents: Implications for Research and Practice. Mahwah: Lawrence Erlbaum Associates, Publishers; 1998.

    Google Scholar 

  63. Bartlett G, Longo C, Rahimzadeh V, Crimi L. iChange. Ethics and Methods for Engagement in Genomic Research with Vulnerable Populations. Preliminary GE3LS Report Submitted to the Research Oversight Committee for the Genome Canada Project: Study of Biomarkers for Pediatric Glioblastoma through Genomi. Volume 28; 2014(November 2013).

  64. Doherty KO, Burgess MM. Defining Deliberative Outputs in a Public Engagement about Biobanks. Health Law Rev. 2008;16:4–6.

  65. Schelbe L, Chanmugam A, Moses T, Saltzburg S, Williams LR, Letendre J. Youth participation in qualitative research: Challenges and possibilities. Qual Soc Work 2014:1-18. doi: 10.1177/1473325014556792.

  66. Greig AD, Taylor J, MacKay T. Doing Research with Children: A Practical Guide. 16th ed. London: SAGE Publications; 2012.

  67. Christensen P, Prout A. Working with Ethical Symmetry in Social Research with Children. Childhood. 2002;9:477–97.

    Article  Google Scholar 

  68. Abelson J, Forest P-G, Eyles J, Smith P, Martin E, Gauvin F-P. Deliberations about deliberative methods: issues in the design and evaluation of public participation processes. Soc Sci Med. 2003;57:239–51.

    Article  PubMed  Google Scholar 

  69. Van Manen M. Phenomenology of Practice. Phenomenol Pract. 2007;1:11–30.

  70. O’Doherty KC, Burgess MM, Edwards K, Gallagher RP, Hawkins AK, Kaye J, et al. From consent to institutions: Designing adaptive governance for genomic biobanks. Soc Sci Med. 2011;73:367–74.

  71. Roche J. Children: Rights, Participation and Citizenship. Childhood. 1999;6:475–93.

    Google Scholar 

  72. Bowling A, Rowe G, Adams S, Sands P, Samsi K, Crane M, et al. Quality of life in dementia: a systematically conducted narrative review of dementia-specific measurement scales. Aging Ment Health. 2014;19:13–30.

    Article  PubMed  Google Scholar 

  73. Miller VA, Baker JN, Leek AC, Hizlan S, Rheingold SR, Yamokoski AD, et al. Adolescent perspectives on phase I cancer research. Pediatr Blood Cancer. 2013;60:873–8.

    Article  PubMed  Google Scholar 

  74. Mann L, Harmoni R, Power C. Adolescent decision-making: the development of competence. J Adolesc. 1989;12:265–78.

    Article  CAS  PubMed  Google Scholar 

  75. Balen R. Involving Children in Health and Social Research: “Human becomings” or “active beings”? Childhood. 2006;13:29–48.

    Article  Google Scholar 

Download references

Acknowledgements

The authors would like to acknowledge the following for their funding support: the David McCutcheon Pediatric Palliative Care Fellowship (Montreal Children’s Hospital) in collaboration with the I-CHANGE (International Childhood Astrocytoma Integrated Genomics and Epigenomics) Consortium, Genome Canada, Genome Québec, the Institute for Cancer Research of the Canadian Institutes for Health Research (CIHR), as well as Dr. Elaine Davis (McGill Faculty of Medicine) for her contributions.

Author information

Authors and Affiliations

  1. Department of Family Medicine, Centre of Genomics and Policy, McGill University, 5858 Côte-des-Neiges, Suite 300, Montréal, QC, H3S 1Z1, Canada

    Vasiliki Rahimzadeh

  2. Department of Family Medicine, McGill University, 5858 Côte-des-Neiges, Suite 300, Montréal, QC, H3S 1Z1, Canada

    Gillian Bartlett, Cristina Longo & Laura Crimi

  3. Division of Oral Health and Society, Pediatric Palliative Care Research, Montreal Children’s Hospital, McGill University, #530-2001 McGill College Avenue, Montreal, QC, H3A 1G1, Canada

    Mary Ellen Macdonald

  4. Department of Pediatrics, Montreal Children’s Hospital Research Institute, McGill University Health Center, 1001 Décarie Boulevard, Montreal, H4A 3J1, QC, Canada

    Nada Jabado

  5. Biomedical Ethics Unit, McGill University, 3647 Peel St, Montreal, QC, H3A 1X1, Canada

    Carolyn Ells

Authors
  1. Vasiliki Rahimzadeh
    View author publications

    You can also search for this author in PubMed Google Scholar

  2. Gillian Bartlett
    View author publications

    You can also search for this author in PubMed Google Scholar

  3. Cristina Longo
    View author publications

    You can also search for this author in PubMed Google Scholar

  4. Laura Crimi
    View author publications

    You can also search for this author in PubMed Google Scholar

  5. Mary Ellen Macdonald
    View author publications

    You can also search for this author in PubMed Google Scholar

  6. Nada Jabado
    View author publications

    You can also search for this author in PubMed Google Scholar

  7. Carolyn Ells
    View author publications

    You can also search for this author in PubMed Google Scholar

Corresponding author

Correspondence to Vasiliki Rahimzadeh.

Additional information

Competing interests

The authors declare they have no competing interests.

Authors’ contributions

This paper is inspired from an MSc thesis completed by the first author VR, under the supervision of authors GB and CE. Authors CL, LC and MM contributed editorial comments in earlier versions, while author NJ contributed clinical expertise relevant to therapeutic trajectories in pediatric palliative care and oncology. All authors read and approved the final manuscript.

Rights and permissions

Open Access This article is distributed under the terms of the Creative Commons Attribution 4.0 International License (http://creativecommons.org/licenses/by/4.0/), which permits unrestricted use, distribution, and reproduction in any medium, provided you give appropriate credit to the original author(s) and the source, provide a link to the Creative Commons license, and indicate if changes were made. The Creative Commons Public Domain Dedication waiver (http://creativecommons.org/publicdomain/zero/1.0/) applies to the data made available in this article, unless otherwise stated.

Reprints and permissions

About this article

Check for updates. Verify currency and authenticity via CrossMark

Cite this article

Rahimzadeh, V., Bartlett, G., Longo, C. et al. Promoting an ethic of engagement in pediatric palliative care research. BMC Palliat Care 14, 50 (2015). https://doi.org/10.1186/s12904-015-0048-5

Download citation

  • Received:

  • Accepted:

  • Published:

  • DOI: https://doi.org/10.1186/s12904-015-0048-5

Keywords