This Article Abstract Full Text (PDF) Purchase Article View Shopping Cart Alert me when this article is cited Alert me if a correction is posted Services Email this article to a colleague Similar articles in this journal Similar articles in PubMed Alert me to new issues of the journal Save to my personal folders Download to citation manager Rights & Permissions Citing Articles Citing Articles via HighWire Citing Articles via Google Scholar Google Scholar Articles by Edwards, K. E. Articles by Wolfe, J. Search for Related Content PubMed PubMed Citation Articles by Edwards, K. E. Articles by Wolfe, J. Social Bookmarking                            What's this?

Understanding of Prognosis and Goals of Care Among Couples Whose Child Died of Cancer

Kelly E. Edwards, Bridget A. Neville, Earl F. Cook, Jr, Sarah H. Aldridge, Veronica Dussel, Joanne Wolfe

From the Harvard Medical School; Center for Outcomes and Policy Research, Dana-Farber Cancer Institute; Department of Medicine, Brigham and Women's Hospital; Department of Epidemiology, Harvard School of Public Health; Department of Medicine, Children's Hospital; Department of Pediatric Oncology, Dana-Farber Cancer Institute, Boston, MA

Corresponding author: Joanne Wolfe, MD, MPH, Dana-Farber Cancer Institute, 44 Binney St, Boston, MA 02115; e-mail: Joanne_wolfe{at}dfci.harvard.edu

	ABSTRACT

TOP ABSTRACT INTRODUCTION METHODS RESULTS DISCUSSION AUTHORS' DISCLOSURES OF... AUTHOR CONTRIBUTIONS REFERENCES

 
Purpose Little is known about how couples care for the terminally ill child with cancer. We assessed both parents’ understanding of prognosis and treatment goals for children with cancer and explored whether sex mediates these views. We also investigated whether discordance within couples regarding treatment goals was related to parental perception of the child's end-of-life (EOL) experience.

Methods We surveyed mothers and fathers of children who died of cancer and were cared for at Children's Hospital (Boston, MA) and the Dana-Farber Cancer Institute (Boston, MA) between 2000 and 2004. Our sample included 38 couples (response rate, 56%).

Results Willingness to participate did not differ by sex. At diagnosis, fathers and mothers held a similar understanding of the child's prognosis, and 58% of couples agreed on the goal of cure. During the EOL period, a majority of fathers and mothers reported lessening suffering as the primary goal. However, within couples there was poor agreement about the primary goal of care ( = 0.07). When parents did not agree on the primary goal of lessening suffering, both parents were more likely to report that the child suffered significantly from cancer-directed treatment (P = .03).

Conclusion Though parent goals are often concurrent at diagnosis, they frequently differ during the EOL period. Parent disagreement about the goal of lessening suffering at the EOL appears to impact how parents describe their child's experience of suffering. Creating opportunities for parents to work through their goals together may lead to improvements in the child's EOL experience.

	INTRODUCTION

TOP ABSTRACT INTRODUCTION METHODS RESULTS DISCUSSION AUTHORS' DISCLOSURES OF... AUTHOR CONTRIBUTIONS REFERENCES

 
Parents of children who died of cancer associate excellent care with receiving clear and compassionate communication.¹ Integrating palliative care for children with advanced cancer begins with communication by ensuring family understanding of the child's prognosis and eliciting goals of care. Studies that have surveyed predominantly mothers of children with cancer have uncovered important areas in need of improvement regarding these communication outcomes. Mack et al² found that among children with newly-diagnosed cancer, 61% of parents were more optimistic than the child's primary oncologist regarding the child's prognosis. Wolfe et al³ reported that parents of children who died of cancer recognized that the child had no realistic chance for cure an average of 3 months later than the primary oncologist. Later recognition of the child's end-of-life (EOL) period was associated with the administration of more aggressive care, later involvement of hospice services, and physician and parent care goals were less likely to be directed at lessening suffering.³

Differences in understanding of prognosis and goals between mothers and fathers caring for children with advanced cancer and their potential clinical implications have not been investigated. Qualitative studies have portrayed the father caring for a child with a life-threatening illness as "engaging in an all-consuming battle with illness itself. " ⁴ In particular, fathers have been described as being caught in a struggle against uncertainty with only two options: treat the underlying illness or accept death.^4-6

We conducted a cross-sectional study of pairs of mothers and fathers of a child who died of cancer. We investigated how closely mothers’ and fathers’ understandings of prognosis, treatment goals at diagnosis, and treatment goals during the EOL period were aligned. In addition, we explored whether parental discordance regarding treatment goals is associated with differences in parental perception of the child's experience during the last month of life and parents’ feelings of having fulfilled their goals. By including the voices of both parents, we hoped to generate a more complete picture of the experience of children with advanced cancer and their families.

	METHODS

TOP ABSTRACT INTRODUCTION METHODS RESULTS DISCUSSION AUTHORS' DISCLOSURES OF... AUTHOR CONTRIBUTIONS REFERENCES

 
Study Design
This study was conducted at the Dana-Farber Cancer Institute (DFCI; Boston, MA) and Children's Hospital Boston (CHB; Boston, MA). The methodology was modeled after a study conducted at these sites and has been described previously.^3,7 Between 2004 and 2006, we interviewed mothers and fathers of children who had died of cancer between 2000 and 2004. Parents were eligible if they were English- or Spanish-speaking, they resided in North America, their child died of cancer more than 4 months before enrollment, and their child's primary oncologist permitted contact with the family.

Eligible families were sent a letter addressed to both parents that contained two postage-paid opt-out cards. If the opt-out card was not returned within 2 weeks, we attempted to reach parents by telephone. We conducted separate interviews with each parent that enrolled. Midway through the study, after a population-based study achieved a 75% response rate in parents completing a written survey in EOL care in Sweden,⁸ we introduced the option of a paper-and-pencil survey.

Response Rates
Primary oncologists denied permission to contact the families of 14 children (9%). The most common reason for denial was a perception that the family was angry with medical team members and/or the institution (n = 4). Of 131 eligible families, 78 families were located, 68 were dual-parent families, and among them, 38 pairs of mothers and fathers completed the survey (response rate, 56%). Sixty-three parents completed the survey by phone (median duration, 1.25 hours; range, 0.75 to 2 hours), 13 parents by paper and pencil. Response rate did not differ among mothers or fathers, by mode of participation, and did not change significantly with the written survey option. Parents participated in the study a median of 4.4 years after the child's death. The length of time after the child's death was not significantly different from nonrespondents. There was no significant difference between parent respondents and nonrespondents regarding the child's sex, race/ethnicity, language, or religion (P > .10), nor were there significant differences regarding the child's age, duration of disease, sex, or diagnosis.

Instruments
Survey about Caring for Children with Cancer. The Survey about Caring for Children with Cancer was adapted from a previously validated instrument that measured parental perceptions of EOL care.^3,7 This report focuses on aspects of the questionnaire that addressed parental perspectives regarding prognosis and treatment goals at diagnosis, during the child's EOL period, and in the last month of life using closed-ended questions; some items requested further open-ended elaboration. The EOL period was defined as the time before death when the parent reported s/he realized that her/his child had no realistic chance for cure. Parents were asked about their understanding of the child's likelihood of cure; categoric responses included "very likely," "likely," "unlikely," or "very unlikely." Parents were asked about their primary goal in providing medical care for their child at diagnosis, during the EOL period, and during the last month of life; categoric responses included: "to cure your child's cancer," "for you and/or your child to keep hoping," "to make sure you have done everything," "to extend your child's life as long as possible but without expecting a cure," "to lessen your child's suffering as much as possible," "to help cancer research," or other. Parents were also asked if they felt that they had accomplished their primary goals during the EOL period (using "yes"/"no" response options).

Parents provided sociodemographic information including marital status, whether they considered themselves to be one of the child's primary caregivers, and whether they took time off from work during the EOL period. Additionally, they provided data about the child including date and type of diagnosis, whether the child received cancer-directed therapy or utilized a homecare team during the EOL period, and location of death. Finally, parents described their child's EOL experience. Specifically, they rated their child's suffering from cancer-directed therapy, fatigue, pain, dyspnea, anorexia, sadness, and anxiety. Parents also evaluated the child's quality of life during the EOL period and peacefulness of death.

Statistical Analysis
Analyses were conducted with the SAS 9.1 statistical software package for Windows (SAS Institute, Cary, NC). No attempt was made to impute missing data. Categoric variables were dichotomized as specified in the text and tables. We conducted a paired analysis restricted to families in which both mothers and fathers completed surveys. We used the statistic to assess agreement within couples regarding prognosis and treatment goals at diagnosis, during the EOL period, and during the last month of life. When there was discordance, we used the McNemar statistic to determine significant directionality.⁹ To assess whether parental agreement about treatment goals was clinically meaningful, we conducted univariate analyses with the dependent variable being "both parents agreed that their primary treatment goal was to lessen suffering during the EOL period." To analyze categoric variables, we used ² or Fisher's exact tests, and for continuous variables, t test or Wilcoxon rank sum test, depending on the distribution of the independent variable.⁹ A P value less than .05 was considered significant.

This study was approved by the DFCI institutional review board.

	RESULTS

TOP ABSTRACT INTRODUCTION METHODS RESULTS DISCUSSION AUTHORS' DISCLOSURES OF... AUTHOR CONTRIBUTIONS REFERENCES

 
Characteristics of the 38 pairs of parents are presented in Table 1. Most parents had postsecondary education, were white, Christian, and described themselves as either somewhat or very religious. Only three couples were not married. Almost all mothers and half of fathers identified themselves as one of the child's primary caregivers, and more than 60% of both mothers and fathers reported taking off time from work during their child's illness.

Primary Goal of Care: Diagnosis, EOL Period, Last Month of Life. Fathers’ and mothers’ primary goals at diagnosis and at EOL are displayed in Figure 1. When goals were dichotomized into cure versus others, fathers were significantly more likely than mothers to report cure as their primary goal when the child was initially diagnosed (P = .01). During the EOL period, however, a majority of both fathers and mothers (56%) reported lessening suffering as their primary treatment goal. Similarly, during the child's last month of life, 59% of mothers and 56% of fathers chose lessening suffering as their primary goal.

View larger version (28K): [in this window] [in a new window] [PowerPoint Slide for Teaching]   Fig 1. Primary treatment goals, according to parents, at the time of the child's diagnosis with cancer and during the child's end-of-life period are displayed (n = 38 mothers and 38 fathers).

View larger version (14K): [in this window] [in a new window] [PowerPoint Slide for Teaching]   Fig 2. Within couple agreement throughout the course of the child's illness regarding prognosis and treatment goals (n = 38 couples). *Prognosis at diagnosis ("cure very or somewhat likely" v other). Primary treatment goal at diagnosis (to cure v other), during the end of life and during the last month of life (to lessen suffering v other).

Accomplishment of EOL Goals
Seventy-four percent of parents reported that their primary treatment goal during the child's EOL period was accomplished (Table 3). Eighty-eight percent of parents with the goal of lessening suffering were able to achieve that goal. An equal percentage of fathers and mothers felt that their goals had been accomplished. Within couples, when at least one parent focused on lessening suffering, both parents were more likely to accomplish their goals (P = .04).

	DISCUSSION

TOP ABSTRACT INTRODUCTION METHODS RESULTS DISCUSSION AUTHORS' DISCLOSURES OF... AUTHOR CONTRIBUTIONS REFERENCES

Gender
Our study did not uncover consistent differences between mothers and fathers with regard to understanding of prognosis or primary treatment goals. The only difference found was at diagnosis; although fathers were not more optimistic than mothers regarding their child's prognosis at diagnosis, fathers were more likely than mothers to choose cure as their primary goal. Prior qualitative research suggests that fathers, particularly heavily involved fathers, may conceptualize treating their child's illness as a battle that is won or lost. As one father in our study stated, "The only goal from the hour of diagnosis was cure... we failed." However, we found that many fathers moved from focus on cure to lessening suffering. Thus, while a father's initial approach may be more directed at cure, this should not imply that fathers are less likely than mothers to transition in their views as the illness progresses.

Within Couples
At diagnosis, there was moderate agreement within couples regarding the child's prognosis. Further, the majority of couples agreed on the primary goal of the child's care. However, most couples did not agree on the primary treatment goal during the child's EOL period. Although both parents recognized that the child had no realistic chance for cure, only one third of couples shared the primary goal of lessening suffering. During the EOL period, parents may bring different perspectives to the goal-setting process further underscoring the need for oncologists to communicate directly with both parents and elicit their priorities for care.

Agreement within couples did not appear to be a critical factor in allowing both parents to feel they had accomplished their goals for the child. However, both parents were more likely to achieve their goals when at least one parent focused on lessening suffering. Furthermore, parents who focused on more realistic goals were better able to achieve their goals.

More importantly, however, our data suggest that when both parents focused on lessening suffering during the EOL period, they were more likely to report that the child suffered less, especially from cancer-directed therapy. Families often face the decision of whether to continue cancer-directed therapy at the EOL and how intensive that therapy should be. Suffering from cancer-directed therapy may reflect the burden of care imposed on the child and family by the necessity to make trips to the clinic, stay in the hospital, or occurrence of physical symptoms such as anorexia and fatigue. When parents agree that the primary goal is lessening suffering, though they may choose to pursue further cancer-directed therapy for their child,¹² they may be more likely to consider potential toxicities in selecting the treatment. This in turn may impact the child's experience of suffering.³

Fatigue is the most common symptom experienced by children with cancer at the EOL,^7,13,14 accounting for the greatest degree of suffering. When families share the primary purpose of relief of suffering, they may be able to practice a multifaceted approach that aids in the prevention and treatment of fatigue.¹⁵ Although our sample size limited our ability to detect significant differences in distress related to other common EOL symptoms, reported suffering from other symptoms was consistently lower among children whose parents agreed on the goal of lessening suffering. Parents who focused on lessening suffering also reported that their children had a better quality of life and a more peaceful death. These data suggest the potential benefit of interventions that facilitate parent agreement about lessening child suffering as a high priority during the EOL period.

Our data is drawn from a small sample. However, it is important to recognize that mothers and fathers of children who died between 2000 and 2004 continue to report suffering at the EOL. In particular, many families report that their child's quality of life was fair/poor and describe their child's death as not "very peaceful." These findings highlight the need to systematically develop and evaluate interventions aimed at improving the experience of children at the EOL.

Limitations
First, parents were interviewed an average of 4 years after their child's death; their reports may not accurately reflect their understanding of their child's prognosis or goals for care during their child's illness. However, the events of their child's diagnosis and the progression of illness through the EOL period are so significant that parents are likely to recall them accurately. Selection bias may also have influenced the study findings. Primary oncologists denied permission to contact 9% of families, and both fathers and mothers participated among only 56% of couples. Interestingly, permission was most commonly denied for couples who were perceived by the oncologist to be angry. A higher degree of perceived anger may be correlated with increased discord in the dyad, and agreement about goals at the EOL may be even lower than found in our sample. Larger studies of couples are warranted. Reassuringly, respondents did not differ significantly from nonrespondents with respect to relevant child characteristics. Nonetheless, this study was conducted at a single institution, and though pediatric oncology patients are often referred to a specialized center due to the need for subspecialty training,¹⁶ it is important to acknowledge that minorities are not adequately represented in our sample. Previous research has identified the importance, for example, of the availability of interpreters to Hispanic families during the EOL period.¹⁷ It is critical to make a conscious effort to include a diverse patient population in future studies that seek to describe the family's experience caring for a child with advanced illness.

It is also important to acknowledge that this study is based entirely on parent report. Interestingly, a recent article confirmed that parent and child assessment of health-related quality of life showed strong agreement and consistency across multiple domains of experience including physical and emotional symptoms.¹⁸ The authors concluded that it is important to utilize parents’ perspectives particularly when children are coping with advanced illness.¹⁸ Nonetheless, future prospective studies should be aimed at including the child's voice.

In conclusion, to our knowledge, this study is one of the first to include the perspectives of couples whose child died of cancer. We have shown that a majority of couples disagree on treatment goals during the child's EOL period, and disagreement was not explained by sex differences. The first step for clinicians may be to recognize that frequently, mothers and fathers do not hold the same primary goals for their child's care. When couples disagree, parents perceive their children to suffer more, particularly from cancer-directed therapy. It is therefore critical that clinicians work with both parents to heighten awareness of their priorities and facilitate effective decision making at the EOL.

	AUTHORS’ DISCLOSURES OF POTENTIAL CONFLICTS OF INTEREST

TOP ABSTRACT INTRODUCTION METHODS RESULTS DISCUSSION AUTHORS' DISCLOSURES OF... AUTHOR CONTRIBUTIONS REFERENCES

 
The author(s) indicated no potential conflicts of interest.

	AUTHOR CONTRIBUTIONS

TOP ABSTRACT INTRODUCTION METHODS RESULTS DISCUSSION AUTHORS' DISCLOSURES OF... AUTHOR CONTRIBUTIONS REFERENCES

 
Conception and design: Kelly E. Edwards, Bridget A. Neville, Earl F. Cook Jr, Veronica Dussel, Joanne Wolfe

Financial support: Kelly E. Edwards, Veronica Dussel, Joanne Wolfe

Administrative support: Sarah H. Aldridge

Collection and assembly of data: Kelly E. Edwards, Sarah H. Aldridge, Veronica Dussel, Joanne Wolfe

Data analysis and interpretation: Kelly E. Edwards, Bridget A. Neville, Earl F. Cook Jr, Sarah H. Aldridge, Veronica Dussel, Joanne Wolfe

Manuscript writing: Kelly E. Edwards, Bridget A. Neville, Veronica Dussel, Joanne Wolfe

Final approval of manuscript: Kelly E. Edwards, Bridget A. Neville, Earl F. Cook Jr, Sarah H. Aldridge, Veronica Dussel, Joanne Wolfe

	NOTES

 
Supported in part by the Alexandra Miliotis Fellowship in Pediatric Oncology; the Neil Samuel Ghiso Foundation, the Office of Enrichments Programs, Harvard Medical School; the Agency for Health Research and Quality Grant No. T32HP10018; from the National Cancer Institute Grant No. NCI 5 K07 CA096746; and a Child Health Research Grant from the Charles H. Hood Foundation Inc.

Authors’ disclosures of potential conflicts of interest and author contributions are found at the end of this article.

	REFERENCES

TOP ABSTRACT INTRODUCTION METHODS RESULTS DISCUSSION AUTHORS' DISCLOSURES OF... AUTHOR CONTRIBUTIONS REFERENCES

 
1. Mack JW, Hilden JM, Watterson J, et al: Parent and physician perspectives on quality of care at the end of life in children with cancer. J Clin Oncol 23:9155-9161, 2005[Abstract/Free Full Text]

2. Mack JWM, Cook FE, Wolfe J, et al: Understanding of prognosis among parents of children with cancer: parental optimism and the parent-physician interaction. J Clin Oncol 25:1357-1362, 2007.[Abstract/Free Full Text]

3. Wolfe J, Klar N, Grier HE, et al: Understanding of prognosis among parents of children who died of cancer: Impact on treatment goals and integration of palliative care. JAMA 284:2469-2475, 2000[Abstract/Free Full Text]

4. Davies B, Gudmundsdottir M, Worden B, et al: "Living in the Dragon's Shadow:" Fathers’ experiences of a child's life-limiting illness. Death Studies 28:111-135, 2004[CrossRef][Medline]

5. Jones JB, Neil-Urban S: Father to father: Focus groups of fathers of children with cancer. Social Work in Health Care 37:41-61, 2003[CrossRef][Medline]

6. Neil-Urban S, Jones JB: Father-to-father support: Fathers of children with cancer share their experience. J Pediatr Oncol Nurs 19:97-103, 2002[Abstract/Free Full Text]

7. Wolfe J, Grier HE, Klar N, et a: Symptoms and Suffering at the end of life in children with cancer. N Engl J Med 342: 326-333, 2000[Abstract/Free Full Text]

8. Kreicbergs U, Valdimarsdottir UI, Steineck G, et al: A population-based nationwide study of parents’ perceptions of a questionnaire on their child's death due to cancer. Lancet 364:787-789, 2004[CrossRef][Medline]

9. Rosner B: Fundamentals of biostatistics. Belmont, CA, Duxbury Press, 1995

10. Garfield CF, Isaaco A: Fathers and the well-child visit. Pediatrics 117:e637-645, 2006[Abstract/Free Full Text]

11. Kreicbergs U, Lannen P, Onelov E, et al: Parental grief after losing a child to cancer: Impact of professional and social support on long-term grief outcomes. J Clin Oncol 25:3307-3312, 2007[Abstract/Free Full Text]

12. Bluebond-Langner M, Belasco JB, Goldman A, et al: Understanding parents’ approaches to care and treatment of children with cancer when standard therapy has failed. J Clin Oncol 25:2414-2419, 2007[Abstract/Free Full Text]

13. Jalmsell L, Kreicbergs U, Onelöv E, et al: Symptoms affecting children with malignancies during the last month of life: A nationwide follow-up. Pediatrics 117:1314-1320, 2006[Abstract/Free Full Text]

14. Goldman A, Hewitt M, Collins GS, et al: Symptoms in children/young people with progressive malignant disease: United Kingdom Children's Center Study Group/Pediatric Nurses Forum survey. Pediatrics 117:e1179-1186, 2006[Abstract/Free Full Text]

15. National Health Service: The NHS Cancer Plan. Investing in the future: Research and genetics, in The NHS Plan 2003-2006. 2002, London, UK, Department of Health

16. Guidelines for the pediatric cancer center and role of such centers in diagnosis and treatment. Pediatrics 99:139-141, 1997[Abstract/Free Full Text]

17. Contro N, Larson J, Scofield S, et al: Family perspectives on the quality of pediatric palliative care. Archives of Paediatric and Adolescent Medicine 156:14-19, 2002

18. Russell KM, Hudson M, Long A, et al: Assessment of health-related quality of life in children with cancer: Consistency and agreement between parent and child reports. Cancer 106:2267-2274, 2006[CrossRef][Medline]

Submitted July 6, 2007; accepted November 27, 2007.

CiteULike    Complore    Connotea    Del.icio.us    Digg    Facebook    Reddit    Technorati    Twitter    What's this?

This article has been cited by other articles:

				P. S. Hinds, L. L. Oakes, J. Hicks, B. Powell, D. K. Srivastava, S. L. Spunt, J. Harper, J. N. Baker, N. K. West, and W. L. Furman "Trying to Be a Good Parent" As Defined By Interviews With Parents Who Made Phase I, Terminal Care, and Resuscitation Decisions for Their Children J. Clin. Oncol., December 10, 2009; 27(35): 5979 - 5985. [Abstract] [Full Text] [PDF]

				L. C. Kaldjian, A. E. Curtis, L. A. Shinkunas, and K. T. Cannon Review Article: Goals of Care Toward the End of Life: A Structured Literature Review American Journal of Hospice and Palliative Medicine, January 1, 2009; 25(6): 501 - 511. [Abstract] [PDF]

This Article Abstract Full Text (PDF) Purchase Article View Shopping Cart Alert me when this article is cited Alert me if a correction is posted Services Email this article to a colleague Similar articles in this journal Similar articles in PubMed Alert me to new issues of the journal Save to my personal folders Download to citation manager Rights & Permissions Citing Articles Citing Articles via HighWire Citing Articles via Google Scholar Google Scholar Articles by Edwards, K. E. Articles by Wolfe, J. Search for Related Content PubMed PubMed Citation Articles by Edwards, K. E. Articles by Wolfe, J. Social Bookmarking                            What's this?