Thank you for your lovely card: ethical considerations in responding to bereaved parents invited in error to participate in childhood cancer survivorship research
Research exploring the needs of families of childhood cancer survivors is critical to improving the experiences of future families faced by this disease. However, there are numerous challenges in conducting research with this unique population, including a relatively high mortality rate. In recognition that research with cancer survivors is a relational activity, this article presents a series of cases of parents bereaved by childhood cancer who unintentionally received invitations to participate in survivorship research. We explore six ethical considerations, and compare our experiences with that described previously. Our considerations include the sharing of confidential information with external parties to confirm past patients’ vital status and appropriate researcher responses to bereaved parents. The management of researchers’ emotional safety when working with illness populations and the fact that study invitations can elicit grief responses in non-bereaved families are discussed. To conclude, we argue for the benefits of inviting bereaved parents whose children died after treatment completion in survivorship research. Such parents’ early survivorship experiences will otherwise be systemically neglected. We argue that discussing complex cases can assist researchers to protect participants’ and researchers’ emotional well-being. We hope to contribute to the dearth of discussion about research operating procedures to address these issues.
KeywordsBereavement Childhood cancer Parents Qualitative research Research ethics Research methods Adverse events Incidents
Research with ill patients is strongly defensible, and often advocated for, not only by researchers and clinicians, but by patients and families themselves (Dear et al. 2011). Despite the advantages of medical and psychosocial research, there are a number of ethical issues relating to research in illness populations. Many articles have already raised important ethical considerations surrounding research with ill populations, including those affected by childhood disease (Bellavance and Alexander 2012; Bowtell et al. 2013; Roth-Cline et al. 2011). Along with Guillement and Gillam (2009), we argue that sharing research challenges benefits new researchers in the field, as well as the wider research community facing similar dilemmas. The discussion of ethical dilemmas facilitates the management of emotional well-being for both the researchers and their participants (Ridge et al. 1999). We also hope to contribute to the dearth of published discussion available describing appropriate research operating procedures (Bowtell et al. 2013), with a unique focus on childhood cancer survivorship research.
Our research team has a mandate to conduct studies investigating psychological aspects of families’ experiences of childhood cancer. Such research has been beneficial in advancing our psychosocial understanding of childhood cancer survivorship. We conduct research at all points along the cancer trajectory, from diagnosis (Evans et al. 2013), through to survivorship (Wakefield et al. 2010), and bereavement. We also conduct work with different age groups, ranging from young patients (Sansom-Daly and Wakefield 2013), to parents (Wakefield et al. 2012) and even grandparents (Wakefield et al. 2014). In the past 10 years, we have, unsurprisingly, faced ethical and logistical challenges in working with this population, including occasionally contacting past patients in error. Despite careful protocol implementation, the reaction of past patients and families can be highly variable, which highlights the importance of researchers maintaining awareness of any research risks and appropriate responses (Bowtell et al. 2013; Dickson-Swift et al. 2008).
Background: a new study investigating the needs of childhood cancer survivors
In 2011, we commenced work on a new project, investigating possible models of care for long term survivors of childhood cancer across 11 hospitals. Survivors aged over 16 were eligible to participate in the study. Parents of survivors under the age of 16 were also eligible. Potential participants were identified through the electronic databases of participating hospitals. The study invited voluntary participation in a questionnaire and a qualitative telephone interview.
Given that some forms of childhood cancer became ‘curable’ in the 1960s (Mukherjee 2011), we anticipated that eligible survivors may have completed their cancer treatment up to 50 years prior. However, the majority of children who survive cancer develop late side effects such as new cancers, heart disease and abnormal lung function, some of which will be life threatening (Hudson et al. 2013; Oeffinger et al. 2006). Late mortality is also increased among children who have completed cancer treatment, with approximately 14 % of survivors dying within 5 years of completing treatment (Mertens et al. 2008).
Given this was a high-risk population, we considered the project’s potential ethical and logistical challenges prior to commencement. We faced substantial difficulties contacting survivors, who may not have had contact with their hospital for several decades. Some of the factors considered were their mortality rate, rates of movement of survivors from their parents’ homes and rates of survivors changing their names (e.g. for marriage). Younger Australians are also highly mobile, with up to 90 % of those aged under 35 without children having moved house in the last 5 years (Australian Bureau of Statistics 2010).
Standard operating procedures
Standard operating procedures for identifying participants for research
Standard operating procedure
Team member responsible
Step 1: Medical records at treating hospital searched, according to study criteria. Patients identified as deceased in hospital records are excluded
Clinical research associate
Step 2: List is manually reviewed by clinical nurse consultant responsible for long term follow up care of survivors at treating hospital. Patients known to be deceased or ineligible are excluded
Clinical nurse consultant
Step 3: Remaining participant list is matched against the Australian electronic ‘White Pages’*. This step ensures hospital records match the current address. All non-matches are removed from the list (i.e. only those with confirmed addresses are approached)
Electronic ‘White Pages’*
Step 4: Remaining participant list is reviewed by the Australian Births Deaths and Marriages Registry (BDM), for a small fee. All patients identified as deceased are excluded
Step 5: The patient’s original treating clinician/oncologist is asked to manually review the remaining list of their past patients and identify any they know to be deceased or ineligible based on study criteria. Where the treating clinician is no longer available, a second clinical representative from the treating hospital (for example, the Head of Department or Head of Oncology) is asked to review the list
Step 6: If a bereaved parent/relative is contacted (despite following steps 1–5), all relevant staff/organizations are notified, including their oncologist (or Head of Department), the clinical nurse consultant responsible for long term follow up care at the treating hospital, any relevant data managers and the institutional review board
Oncologist, clinical nurse consultant, data manager
Step 7: If a bereaved parent/relative is contacted (despite following steps 1–5), a personalized card is sent to the family, after receiving permission from treating hospital staff**
Clinician at treating hospital
Despite the rigor of the procedures there was still no guarantee that the families of deceased patients would not be contacted (Guillemin and Gillam 2004; Rossman and Rallis 2010). The Registry of Births, Deaths and Marriages acknowledged that it did not hold data on all Australian deaths and provided the following reasons for missing deaths: (a) Errors in death notifications submitted by funeral directors or the coroner (e.g. inaccurate date of birth of the deceased); (b) Name changes of the deceased which were not registered prior to the death; (c) Overseas deaths or funerals; and (d) Deaths without a formal funeral, in which case, it is the next of kin’s responsibility to notify the Registry.
Cases of bereaved families inappropriately invited to participate in survivorship research
Available child details
Research team response, and outcomes
Case 1: Phone call from father to study coordinator
Female, brain stem glioma, 6 years old at diagnosis
Manner was kind, but upset. He reported that “it felt strange receiving mail addressed to a daughter who had been gone so long.”
Hand written card was mailed to father. Treating hospital was notified of the death. No further contact with family
Case 2: Mother wrote a note and included it in the study package, returned using provided reply paid envelope
Female, osteogenic sarcoma, 15 years old at diagnosis, died 2008, aged 48 years
Mother’s note was brief, but understanding and kind
Hand written card was mailed to mother. Mother responded by sending a card to the research team (see quotes from card within manuscript). Treating hospital was notified of the death
Case 3: Mother wrote a letter and included it in the study package, returned using provided reply paid envelope
Male, died 1999. Further details unknown
Quote: “Thank you but our son [son’s name] passed away May 1st, 1999.” [Mother’s name]. Love heart drawn
Hand written card was mailed to mother
Treating hospital was notified of the death. No further contact with the family
Case 4: Mother wrote an email to the principal investigator
Female, died 2013, aged 30, from a fifth cancer
Quote: “Last year, you sent to my daughter [name], papers regarding a study. Unfortunately, and with great sadness, [her] time with us, ran out on [date of death]…. We would like it if you could inform Dr. [name] and Dr. [name].” The email then described her daughter’s cancer battle and her achievements, in spite of her multiple cancers (see manuscript text)
Principal investigator replied by email, to which the mother replied “There is no distress, so do not worry about the fact that the questionnaire arrived. We just wanted to let you know as well as letting Dr. [name] know, as he was her doctor for many years.” Treating doctors, and nurse, were notified, each of whom contacted the mother to express their condolences, as well as expressing to the study team their distress at not having been aware of the death, and relief at the opportunity to contact the family. Hand written card was sent to the mother from the study team
As outlined in our procedure, in each case we sent a hand-written card to the family, expressing our sympathy, apologizing for contacting the parent and explaining that a checking procedure was in place (see Table 2 for the wording of the card). To date, these cards appear to have been well received by parents, although the numbers have been small. One card elicited a return card from a bereaved mother (Case 2) and contained heartfelt thanks. “Hello [name of study coordinator], Just to say thank you for your lovely card—it was a lovely thought.”
The card went on to describe her child’s life in detail. “She accomplished so much, in spite of everything.” The card described her daughter’s subsequent health challenges, “then of all things, she was faced with a brain tumor—just unbelievable” and her passing “she had an ongoing battle with that until her death—was so brave the whole time—never complained.” The card concluded by describing the mother’s favorite photograph of her daughter, which was connected to a very personal memory, still held tight by her mother. The card concluded with a final thank you and was signed with the mother’s name, followed by an ‘x’ (symbolizing a ‘kiss’).
Resulting considerations from the described incidents
This experience led our team to ponder a number of ethical considerations related specifically to our study and more generally to all research with vulnerable families. Our experiences echoed those reported by Bowtell et al. (2013), whose team experienced two unexpected deaths of young research participants. The researchers considered the deaths as ‘ethically important moments’, and after consultation with their institutional review board, also chose to send a card to the family (Bowtell et al. 2013).
Should research teams share the names of past patients with ‘external’ parties such as the registry of Births, Deaths and Marriages (BDM) and the electronic ‘White Pages’?
Should researchers contact parents who respond that their child has died, and if so, how?
If a bereaved parent contacts the study team, a second dilemma emerges, namely, should the team instigate additional contact and risk distressing the family further? An apology from the team may provide some acknowledgement of the parents’ experience, as well as help the team to consider their own response to the situation. There is however, little evidence to guide ‘how’ this should be done, for example, through a personal telephone call or a letter/card.
After consultation with an experienced bereavement counselor and a review of the literature (Macdonald et al. 2005; Macnab et al. 2003; Serwint and Nellis 2005), we decided a card was appropriate. Parents whose children have died in the ICU of a pediatric hospital greatly appreciated when hospital staff sent their sympathies in a form of a card (Macdonald et al. 2005). This approach was also condoned by the review board involved in the Bowtell et al. (2013) study.
Can study invitations elicit grief responses in non-bereaved parents?
Negative reactions were not limited to bereaved parents but also included non-bereaved parents who received invitations. Some parents of surviving children expressed sadness and grief relating to their cancer experience, which was intensified when they received the study invitation. Careful consideration of how best to manage these feelings is important when developing study protocols.
Specific cases in this study included a mother and father who requested a joint teleconference with the study team because the final diagnosis of their son’s suspected malignancy was not cancer.
Another case included a mother who felt angry that her son might have been reminded of his cancer experience immediately before the birth of his own child. The mother had opened her son’s invitation and chosen not to share it with him. Other cases involved managing non-study related distress in parents, such as a parent whose own father had died recently, and several survivors who were dissatisfied with the medical care they had received since completing treatment. A final case involved an expression of grief in a survivor who had learned that her treating oncologist had died in the years after her treatment.
Should bereaved families be excluded from survivorship research?
Many bereaved families experience a period of ‘survivorship’ between the time their child completes cancer treatment and their death from disease recurrence or other factors. Case 2 provides the strongest evidence of this, as the patient experienced more than 30 years of survivorship before her death at age 48. Her family’s 30 year survivorship experience was not captured or acknowledged when they were excluded from this study. It remains unknown whether their experiences were similar to other families, or whether their experience was unique. Given this, it might be appropriate to consider the inclusion of bereaved families in survivorship research, with appropriate safeguards.
Two bereaved parents did express interest in study participation despite their loss. In the study protocol, we had excluded bereaved families to minimize any emotional burden on this vulnerable group. Nevertheless, our reluctance to include bereaved parents may not have been empirically justified. Bereaved relatives have expressed a strong desire to remain involved in research, citing this as a positive opportunity to give altruistically and to process their loss (Koffman et al. 2012). Bereaved parents have also described their exclusion from research and other involvement with their child’s treating center as adding to their grief and sense of abandonment (D’Agostino et al. 2008).
Clearly, Case 2’s mother felt a keen desire to share her memories and grief with the study team. Appropriate invitations to participate in research may, in fact, allow bereaved parents to remain engaged with their hospital and allow staff an appropriate forum through which to maintain contact. This possibility was realized in Case 4: the child’s treating doctors and nurse were grateful to be notified of the death and subsequently contacted the family. Case 4’s mother also demonstrated a strong desire to share her daughter’s experiences “She ended up suffering from [disease name]”.
Again, [name] coped with more chemotherapy, but was unable this time to beat the odds.” She was also eager to share her achievements “[Name] did, however, reach the age of 30 years and achieved a degree, and was also admitted full membership of [professional body]. She was employed full time.”
Who is the appropriate person to contact a bereaved parent?
How do we ensure the emotional safety of the research team in this process?
There has been a shift in focus within the literature to address not only participant wellbeing, but also that of the researchers (Bowtell et al. 2013). A number of emotional safety issues are apparent when engaging in research of this kind. Researchers need to be aware of the possibility of contacting a participant who has died and would benefit from being prepared with strategies to cope with emotions prior to such events (Dickson-Swift et al. 2008). Bowtell et al. (2013) clearly described a common juxtaposition in research, in which researchers may be encouraged to remain emotionally distant from their participants, yet also experience a valuable, human connection with them. In order to address this, prior training on boundary management strategies and regular debriefing with either the chief supervisor or a trusted research confidant are two main recommendations suggested in the literature (Dickson-Swift et al. 2008; Johnson and Clarke 2003; Wray et al. 2007).
One emotion described by several members of the research team was a feeling of nervousness surrounding answering the study telephone line. This uneasiness surrounded the possibility of receiving a call from a distressed participant/relative and their uncertainty regarding how best to respond. Similar feelings of uneasiness have been mentioned in previous research (Duncan et al. 2009; Johnson et al. 2003), and can be attributed to the unpredictable nature of conducting research (Duncan et al. 2009). However, providing appropriate training with a set of professional competencies may increase researcher confidence to adequately maneuver through conversations they did not foresee (Dickson-Swift et al. 2008). Bowtell et al. (2013) provided a useful list of ‘Ten emotional safety recommendations for researchers’, which is a framework that is applicable to this study as well.
Though survivors call for more cancer research to support their medical and psychosocial needs, this may come at the emotional expense of bereaved or distressed families who are inadvertently contacted. Awareness of this possibility, self-care and professional boundary management typify prepared researchers. Nevertheless, comprehensive referral networks and active collaboration with the staff that cared for the invited families are critical. With support and open communication, participants contacted in error have expressed hope that more is being done to help children like theirs have a chance not only to survive, but to thrive, after completing cancer treatment, as exemplified by one of our described cases (Case 3). “We hope your survey helps children/adults who still need follow-up.”
We acknowledge the contribution of Alison Young (Researcher; Sydney Children’s Hospital). This study was funded by the Kids Cancer Alliance, which is supported by a translational research grant from the Cancer Institute of NSW. Claire Wakefield is supported by a Career Development Fellowship from the National Health and Medical Research Council of Australia (APP1067501) and an Early Career Development fellowship from the Cancer Institute of NSW (ID: 11/ECF/3-43). The Behavioural Sciences Unit is supported by the Kids with Cancer Foundation.
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