Parental Distress and Desire for Information about Long-term Implications of Pediatric Cancer Treatment

Abstract

BACKGROUND:

Parents of children with cancer have unmet information needs about future limitations resulting from cancer or its treatment. Prior research shows that clinicians focus on acute effects of therapy rather than long-term limitations in early care discussions, partly due to worries of causing distress. The validity of concerns about distress is unknown. We evaluated parental distress associated with information about future limitations, and the extent to which distress is associated with information preferences.

METHODS:

We surveyed 355 parents of children with cancer within 3 months of diagnosis, and the children’s physicians, at Dana-Farber/Boston Children’s Cancer and Blood Disorders Center and Children’s Hospital of Philadelphia. Our primary outcome was parental distress associated with information about long-term limitations.

RESULTS:

46% of parents found information about future limitations to be extremely or very upsetting. In multivariate analysis, parents were more likely to consider information about future limitations distressing if they also found prognostic information upsetting (OR 5.36, p<0.001), struggled to accept their child’s illness (OR 2.57, p<0.001), or had depression (OR 1.79, p=0.01). However, 92% of parents considered information about potential future limitations to be extremely/very important. Those who found information about future limitations distressing were more likely to consider it important (96% vs 89%, p=0.03), and to desire a precise understanding of their child’s risks (92% vs 80%, p=0.001).

CONCLUSION:

Though information about future limitations caused by cancer treatment is upsetting to many parents, most parents desire this information, and those who are distressed are more likely to value this information.

Precis:

Though information about future impairment as a result of pediatric cancer or its treatment is upsetting to many parents, parents desire this information from the time of diagnosis, and those who are distressed consider this information particularly important.

Background:

The time surrounding a child’s cancer diagnosis can be overwhelming to parents.¹ Beyond the news of the diagnosis, early treatment conversations frequently touch on sensitive topics such as the child’s prognosis and the short- and long-term risks of therapy.^1–3 Parents who are emotionally overwhelmed may struggle to process and incorporate the news of the cancer and the fact that treatment may have life-long consequences.⁴

Prior work suggests that parents value information about late effects from the time of diagnosis,^3,5,6 but parents feel they receive sub-optimal information about future implications of their children’s treatment.⁷ This may be in part because providers offer less information about late effects than acute toxicities during early treatment conversations.⁸ Furthermore, clinicians often describe probabilities of specific late effects of treatment by individual chemotherapeutic agents, yet parents worry about the functional implications of the treatment as a whole⁹ and the potential impact on their children’s lives.¹⁰

The gap between parent communication preferences about future limitations, and the information they receive, may in part be due to provider concerns about discussing potential impairment. Providers often hesitate to discuss late effects throughout the cancer care continuum due to concerns about overwhelming patients with information and causing distress.^11,12 Furthermore, many physicians find it stressful to anticipate and communicate a patient’s likely outcomes,¹³ and fear taking away hope.¹⁴ Even in survivorship clinics, oncologists tend to focus on current symptoms, rather than providing information about potential future health risks.¹⁵

As rates of cure for childhood cancer continue to improve, it is ever more important that we meet parents’ information needs regarding the long-term health implications of cancer and its treatment. Currently, more than 80% of children with cancer will become long-term survivors,¹⁶ and most will experience at least one late effect of cancer treatment.¹⁷ In this study, we sought to evaluate the emotional impact of early conversations about future limitations of cancer treatment. We hypothesized that parents, particularly parents of children with a high likelihood of long-term survival, would consider this information upsetting but important. We also investigated factors associated with parental distress about future impairment, and the extent to which distress is associated with information preferences.

Methods:

We surveyed parents of children with cancer and the children’s pediatric oncologists at Dana-Farber/Boston Children’s Cancer and Blood Disorders Center and the Children’s Hospital of Philadelphia about their medical communication experiences. Questionnaires were administered between November 2008 and April 2014. Parents were approached about participation 1 to 6 weeks after initial diagnosis, and surveys were administered within 12 weeks of the child’s diagnosis. Eligibility criteria included child ≤18 years of age, parent able to read English or Spanish, and permission to approach granted by the child’s primary oncologist. Eligible parents were approached in person during clinic visits or by mail, and given a letter describing participation, the questionnaire, and a postage-paid postcard to decline participation. Parents who did not respond initially were approached a second time. One parent per child completed the survey. After a parent completed a survey, their child’s primary oncologist was approached with the paired physician survey. As a token of appreciation, participating parents received a $10 gift card, and physician participants received a $5 gift card. The institutional review boards of the Dana-Farber Cancer Institute and Children’s Hospital of Philadelphia approved this study.

Parent and physician survey instruments utilized items or modified items drawn from previously developed questionnaires.^7,18,19 Limited novel items were developed after literature review, employing general principles of survey design. The 75-item parent survey was pilot tested with parents to assess face and content validity, and took approximately 30–40 minutes to complete. Parent and oncologist surveys were available in paper-and-pencil or electronic format per participant preference; parent surveys were available in English or Spanish. Surveys were initially written in English, translated into Spanish, then back-translated into English to ensure translation accuracy.

Study Outcomes:

The primary outcome for this analysis was parental distress associated with information about future limitations, assessed by a single survey item. Parents were asked how upsetting it was “to know information about how likely it is that cancer or its treatment may affect your child’s life in the future,” with response options of extremely, very, somewhat, a little, and not at all. This item was a modified version of an item previously used to assess distress associated with prognostic information.¹⁸

The secondary outcome was parent information preferences about future limitations, assessed using 2 previously utilized items.²⁰ Parents were asked the importance of knowing “how likely it is that cancer or its treatment may affect your child’s life in the future” and the importance of “having a precise understanding” of the likelihood of future limitations.

Explanatory factors:

We assessed the following factors we hypothesized may be associated with parents finding future limitations information upsetting:

• Parent perception of information quality using a previously developed 5-item scale (α=0.87) with items querying the quality of information received about treatment, cause of cancer, likelihood of cure, likelihood of future limitations, and overall, with response options excellent/good/satisfactory/fair/poor.²¹

• Parent perception of communication quality utilizing a previously developed scale (α=0.76) which asked how often the oncologist gives understandable answers to questions, takes adequate time to answer questions, and conveys information in a sensitive manner, with response options never/sometimes/usually/always.²¹

• Trust in the child’s oncologist using an item from the Trust in Physician scale.^22,23

• Parents and physicians were asked the child’s likelihood of cure, and likelihood of experiencing future limitations in “physical abilities (such as difficulty exercising),” “intelligence (such as difficulty with schoolwork),” and “quality of life (such as difficulty enjoying time with family or friends).”^20,24 Physician-reported prognosis estimations were dichotomized as “favorable” (>75% chance of cure), or “less favorable” (<75% chance of cure) to approximate median survival estimates for all children with cancer. As described in prior analyses, physician-reported likelihood of future limitations in each domain was trichotomized as “high risk” (≥50% chance), “moderate risk” (10–49% chance of future limitation), and “low risk” (≤10% chance).²⁵

• Distress related to prognostic information by asking parents “how upsetting is it to you to know information about your child’s likelihood of cure?” (extremely/ very/somewhat/a little/not at all).¹⁸

• Parental peaceful acceptance and struggle with illness using the validated PEACE scales; the 7-item struggle with illness sub-scale (α=0.81), and the 5-item peaceful acceptance sub-scale (α=0.78).²⁶

• Parental depression and anxiety using the 14-item Hospital Anxiety and Depression Scale (HADS, α=0.92).^27,28

• Parent/child attributes: Parent sex, age, educational level, and race/ethnicity were obtained by questionnaire. Previously validated scales were used to assess parent dispositional optimism (6-item),²⁹ decisional regret (5-item),³⁰ and decision-making preferences and role (2-item).³¹ . Child’s sex, diagnosis, date of diagnosis, and date of birth were obtained by medical record review.

Statistical Methods:

Parental distress associated with information about potential future limitations was dichotomized as highly upsetting (extremely/very) vs. somewhat/a little/not at all. Univariable and multivariable logistic regression were used to evaluate factors associated with parental distress. Backwards selection was used to construct the multivariable model, setting the significance level for removal to 0.05, and including parental education, parent race/ethnicity and child diagnosis category in the model regardless of statistical significance. Variances of parameter estimates were obtained using a sandwich estimator to correct for within-physician clustering. Similar univariable analyses were performed to investigate if information preferences differ by parental distress related to future limitations.

Item non-response was ≤7% for all items. Missing values were imputed by multiple imputation by chained equations, using the -ice-command in Stata. Imputed values were used only for regression analyses, and not for descriptive statistics; moreover, values for the outcome variable (parental distress) were not imputed. Statistical analyses were conducted using Stata version 13.1 (StataCorp, College Station, TX).

Results:

Sixty-eight percent (382/565) of eligible parents completed the survey within 12 weeks of their child’s diagnosis (mean 6 weeks post-diagnosis), and 95 oncologists completed paired surveys for 361/382 (95%). Six surveys were excluded as the parent did not respond to the primary outcome of the present analysis, for a final cohort of 355 parent-physician pairs. Parents of children with hematologic malignancies, extra-cranial solid tumors, and brain tumors were equally likely to participate, although there was a borderline association toward decreased participation of parents of children with brain tumors (p=0.07). The majority of parent respondents were female, married and highly educated (Table 1). Oncologists considered 22% of children at high risk of physical limitations, 9% high risk for impaired intelligence, and 6% high risk for limitations in quality of life.

Table 1.

Characteristics of participating parents and their children (N=355)

	N (%)
Parent age*
<30	38(11)
30-39	137(39)
40-49	134(39)
50+	39(11)
Parent gender*
Female	285(81)
Male	67(19)
Parent race/ethnicity*
White	276(78)
Black	23(7)
Hispanic	28(8)
Other	25(7)
Parent education*
<College graduate	123(35)
College graduate	137(39)
Graduate/professional school	88(25)
Parent marital status*
Married/living as married	293(84)
Other	56(16)
Child age at diagnosis (years)
0-2	96(27)
3-6	71(20)
7-12	79(22)
13-18	109(31)
Child gender*
Male	156(44)
Female	198(56)
Diagnosis
Hematologic malignancies	174(49)
Solid tumor	138(39)
Brain tumor	43(12)
Physician-rated prognosis
Favorable	216(61)
Unfavorable	139(39)
Site
Boston	263(74)
Philadelphia	92(26)
Language of Survey Completion
English	341(96)
Spanish	14(4)

^*Missing data include child gender (N=1); parental age (N=7); parental gender (N=3); parental race/ethnicity (N=3); parental education (N=7); and parental marital status (N=6).

Nearly half (46%, 165/355) of parents found information about potential future limitations due to cancer or its treatment to be extremely or very upsetting. In bivariable analyses, parental distress related to future limitations was not significantly associated with physician estimates of the child’s prognosis or the child’s risk of future limitations (Table 2). However, parents who found prognostic information upsetting (35%, 121/348) also tended to find information about future limitations upsetting (OR 5.64; 95% CI 3.78–8.42). Distress about future limitations was also more likely among parents who struggled to accept their child’s illness (OR 2.95; 95% CI 1.9–4.56), and those with depression (OR 2.52; 95% CI 1.65–3.85), and anxiety (OR 1.87, 95% CI 1.17–2.98). In contrast, parents reported less distress if they held a passive role in decision-making relative to the oncologist (OR 0.57; 95% CI 0.36–0.89), and if they reported peaceful acceptance of the child’s illness (OR 0.43; 95% CI 0.3–0.63).

Table 2.

Bivariable analysis of factors associated with parents finding it “extremely” or “very” upsetting to know information about how likely it is that cancer or its treatment may affect their child’s life in the future.

	Number of patients*	Unadjusted % extremely/very upset	Unadjusted Odds Ratio	95% CI	P Value
Prognosis and Prognostic Communication
Physician-Estimated Prognosis
Favorable (>75% chance of cure)	216	44	1.00
Unfavorable	139	50	1.23	0.86-1.76	0.25
Distress related to prognostic information
Somewhat-Not at all	227	32	1.00
Extremely/very upset	121	72	5.64	3.78-8.42	<0.001
Risk of Future Limitations: Physician-rated likelihood of impairment in
Intelligence
Low risk	121	40	1.00
Moderate risk	155	52	1.54	0.96-2.48	0.19
High risk	76	45	1.14	0.70-1.87
Physical function
Low risk	175	43	1.00
Moderate risk	145	50	1.31	0.81-2.12	0.47
High risk	32	44	1.00	0.51-1.96
Quality of life
Low risk	167	43	1.00
Moderate risk	163	52	1.40	0.85-2.31	0.22
High risk	22	36	0.80	0.28-2.26
Information, Communication and Decision-Making Experiences
Quality of information received about future limitations
Satisfactory-Poor	126	44	1.00
Excellent-good	220	47	1.17	0.77-1.80	0.46
Overall information quality
Low	143	50	1.00
High	194	44	0.83	0.55-1.24	0.36
Communication quality
Low	170	42	1.00
High	178	49	1.35	0.85-2.14	0.20
Trust in physician
Not completely	107	44	1.00
Completely	246	47	1.13	0.65-1.95	0.67
Held ideal role in decision-making
No	130	45	1.00
Yes	218	47	1.08	0.69-1.70	0.73
Held a passive role in decision-making
No	252	50	1.00
Yes	97	36	0.57	0.36-0.89	0.01
Decisional Regret
Low	289	45	1.00
High	66	55	1.49	0.91-2.42	0.11
Parent and Child Characteristics
Child diagnosis
Hematologic malignancy	174	45	1.00
Extra-cranial solid tumor	138	46	1.01	0.63-1.62	0.62
Brain tumor	43	53	1.38	0.74-2.57
Child age at diagnosis
0-2	96	51	1.00
3-6	71	48	0.88	0.52-1.49	0.68
7-12	79	44	0.76	0.42-1.39
13-18	109	43	0.73	0.45-1.17
Child gender
Female	156	49	1.00
Male	198	44	0.80	0.50-1.26	0.34
Parent education
<College graduate	123	53	1.00
College graduate	137	47	0.80	0.53-1.20	0.13
Graduate/professional school	88	39	0.56	0.32-0.98
Parent race/ethnicity*
White	276	46	1.00
Black	23	48	1.07	0.49-2.30	0.49
Hispanic	28	61	1.71	0.75-3.90
Other	25	40	0.75	0.30-1.90
Parent Psychological Factors
Dispositional optimism
Low	166	49	1.00
High	189	44	0.80	0.55-1.16	0.25
Peaceful acceptance of illness
Low	167	58	1.00
High	172	38	0.43	0.30-0.63	<0.001
Struggle with illness
Low	149	32	1.00
High	179	58	2.95	1.90-4.56	<0.001
Parental depression
HADS score not suggestive	241	40	1.00
Suggestive	103	62	2.52	1.65-3.85	<0.001
Parental anxiety
HADS score not suggestive	156	37	1.00
Suggestive	177	53	1.87	1.17-2.98	0.009

^*Numbers may not sum to 355 due to missing values. Imputed values were used for logistic regression.

In a multivariable logistic regression model (Table 3), parents were more likely to find information about future limitations upsetting if they were upset by prognostic information (OR 5.36; 95% CI 3.34–8.6), if they struggled to accept their child’s illness (OR 2.57; 95% CI 1.53–4.31), and if they had HADS scores suggestive of depression (OR 1.79; 95% CI 1.18–2.72). Parents who felt they held a passive role in decision making were less likely to be upset by future limitations information (OR 0.52; 95% CI 0.3–0.94).

Table 3.

Factors associated with parents finding information about future limitations extremely/very upsetting in multivariable logistic regression. The model was determined using a backwards selection procedure where the significance level for removal from the model was set at 0.05; parental education, parent race/ethnicity and diagnosis category were forced into the model regardless of statistical significance. Odds ratios >1 represent a higher likelihood of finding information about future limitations to be extremely/very upsetting.

	Odds Ratio	95% CI	P Value
Distress related to prognostic information
Somewhat-Not at all	1.00
Extremely-very upset	5.36	3.34-8.60	<0.001
Struggle with illness
Low	1.00
High	2.57	1.53-4.31	0.001
Parental depression
HADS score not suggestive	1.00
Suggestive	1.79	1.18-2.72	0.01
Held a passive role in decision-making
No	1.00
Yes	0.52	0.30-0.94	0.03
Parent education
<College graduate	1.00
College graduate	0.63	0.38-1.03
Graduate/professional school	0.69	0.38-1.23	0.13
Parent race/ethnicity*
White	1.00
Black	1.30	0.50-3.40
Hispanic	0.97	0.38-2.48
Other	0.59	0.26-1.32	0.56
Child diagnosis
Hematologic malignancies	1.00
Solid tumor	0.96	0.57-1.63
Brain tumor	0.86	0.41-1.80	0.92

Despite the prevalence of distress, 92% of parents thought information about long-term limitations was extremely or very important, and 86% desired a precise understanding of possible future limitations. Parents who found information about future limitations to be extremely or very upsetting were more likely to consider this information important (OR 2.83; 95% CI 1.12–7.15) and more likely to value a precise understanding of the risk of future limitations (OR 2.90; 95% CI 1.58–5.29) relative to parents who were less upset by this information (Table 4).

Table 4.

Association between parents finding information about future limitations extremely or very upsetting and parent information preferences. N=355.

	Number of patients	Importance of information about possible future limitations			Importance of having a precise understanding of possible future limitations
		Unadjusted % “extremely or very important”	Unadjusted OR (95% CI)	P value	Unadjusted % “extremely or very important”	Unadjusted OR (95% CI)	P value
Parental distress related to future limitations information
Somewhat-not at all	190	89	1.00		80	1.00
Extremely/very	165	96	2.83 (1.12-7.15)	0.03	92	2.90 (1.58-5.29)	0.001

Discussion:

Nearly half of the parents in our study considered information about future limitations resulting from cancer or its treatment to be upsetting. However, those who were distressed by this information expressed greater desire for detailed information about long-term outcomes. Many oncologists worry that providing information about potential future limitations may upset parents,^11,12 and while this work supports this as a legitimate concern, our study suggests that parental distress should not be considered a reason to avoid conversations. Instead, parents who are distressed may need more information, or an opportunity to have continued discussions over time.

We identified risk factors for finding information about future limitations upsetting. For example, parents who experience depression, those who struggle with their child’s diagnosis, and those who are upset by prognostic information may benefit from added emotional support overall, and particular care in presenting information about future limitations. Additionally, we found that parents who held a more passive role in decision-making were less likely to be upset by late effects information. Research suggests that individuals feel greater regret for decisions for which they feel more responsibility,³² a phenomenon that may be at work here. Parents who felt less engaged in decisions may have felt less responsibility for potential outcomes, and subsequently less distressed. Notably, communication and information quality were not associated with distress.

Although we identified an association between distress and desire for more detailed information about long-term outcomes, we do not know the direction of this association and cannot speak to causation. Prior work suggests some potential mechanisms for the association. First, it is possible that inadequate information contributes to distress. Though we did not find an association between parent perception of information quality and distress, we do not have detailed information on what was said. Prior work in prognostic communication has identified greater upset among parents who report lack of prognostic disclosure, and those who are dissatisfied with the quantity of information received.¹⁸ Parents who feel they do not have enough information about the potential long-term implication of therapy may presume the worst and worry accordingly. Alternatively, distress may impair a parent’s ability to process or remember the information presented. We previously found that parents who were upset by information about future limitations were less likely to understand their child’s risk of impairment, relative to parents who were less upset by such information,²⁵ suggesting that parental distress may have implications beyond the immediate emotionality of the information.

We also found that parents who found information about long-term limitations distressing were more likely to find prognostic information distressing, reinforcing the parallels between these types of information. Studies of prognostic disclosure have shown that a subset of parents find prognostic information quite upsetting, however parents value prognostic information, and receiving information about prognosis does not diminish hope.^18,21,33 We hypothesized that parents of children with good prognoses might be most worried about future functional limitations, but prognosis was not significantly associated with concerns about future limitations. We also found that a higher risk of functional impairment was not significantly associated with increased parental distress. Therefore, distress may reflect the emotional experience of the parent more than the medical reality for the child.

Parents expressed a strong desire for information they found distressing, but our study did not investigate how this information could be shared in a less upsetting way. Further research should explore how best to convey information about potential functional impairment in a way that accurately conveys risks while meeting parents’ emotional needs. Studies audio- or video-recording early conversations about long-term effects of treatment could be utilized to help identify best practices and explore the impact of spoken communication on distress. In the meantime, clinicians should consider asking parents how much information they desire, and with what level of detail, and tailor their information delivery accordingly.

Our findings should be interpreted in the context of potential limitations. We surveyed parents of children with many different cancer diagnoses with different risks of late effects, and some long-term limitations may be more distressing than others. We asked about general categories of future limitations, as opposed to exploring distress associated with specific late effects, in part to address the varied late effects that patients might face, and as pilot testing revealed these functional outcomes to be more important to parents. Our study surveyed largely Caucasian, highly-educated parents at two large pediatric cancer centers, raising concerns about generalizability. Future research should explore the unique information needs and preferences of more vulnerable populations. While we did not capture the detailed content of the initial discussions between parents and providers, the majority of children with cancer are enrolled on clinical trials with standardized informed consent documents.³⁴ These documents are often utilized during discussions of potential long-term effects of therapy, suggesting some uniformity of information provided across centers. We have limited information about parents who declined participation, so we do not know if they differed from our study sample in meaningful ways. Lastly, this study did not explore patient perspectives. Survivors of childhood cancer with unmet information needs about long-term outcomes are more likely to experience depression, anxiety and inferior health-related quality of life.^35–37 We focused on parents in this analysis as they are the primary decision-makers for their children, but adolescent and young adult patient perspectives on information delivery and distress associated with future limitations should be explored in future work.

Though emotional to learn about, parents desire early information about long-term functional implications of cancer and its treatment. Risks of future impairment have typically been addressed in depth after treatment completion and during survivorship, yet parents wish to hear this information in early treatment discussions as they consider it germane to decision-making.⁹ Information about potential functional limitations is also crucial to preparing families for their children’s future health needs.²⁰ While many parents in our study found information about future limitations upsetting, limited discussions of the long-term implications of cancer treatment do not meet parent preferences for early, detailed information. Best practices for these conversations must be investigated and tested so that we can impart difficult information honestly while also supporting its emotional impact.