This study attempts to determine parental prioritization of goals of care and patterns of change over time for parents of children receiving palliative care.
Key Points
Question
What importance do parents of children receiving palliative care place on 5 common goals of care and how do these goals change over a 2-year period?
Findings
In this cohort study of 603 pediatric palliative care patients, parents most commonly scored seeking quality of life as having the highest importance, followed closely by seeking health or comfort with less importance placed on disease modification or life extension, but individual parents’ scores varied substantially and often changed over time.
Meaning
These findings emphasize the importance of reassessing goals of care with parents to guide appropriate clinical intervention.
Abstract
Importance
While knowing the goals of care (GOCs) for children receiving pediatric palliative care (PPC) are crucial for guiding the care they receive, how parents prioritize these goals and how their priorities may change over time is not known.
Objective
To determine parental prioritization of GOCs and patterns of change over time for parents of children receiving palliative care.
Design, Setting, and Participants
A Pediatric Palliative Care Research Network’s Shared Data and Research cohort study with data collected at 0, 2, 6, 12, 18, and 24 months in hospital, outpatient, or home settings from April 10, 2017, to February 15, 2022, at 7 PPC programs based at children’s hospitals across the US. Participants included parents of patients, birth to 30 years of age, who received PPC services.
Exposures
Analyses were adjusted for demographic characteristics, number of complex chronic conditions, and time enrolled in PPC.
Main Outcomes
Parents’ importance scores, as measured using a discrete choice experiment, of 5 preselected GOCs: seeking quality of life (QOL), health, comfort, disease modification, or life extension. Importance scores for the 5 GOCs summed to 100.
Results
A total of 680 parents of 603 patients reported on GOCs. Median patient age was 4.4 (IQR, 0.8-13.2) years and 320 patients were male (53.1%). At baseline, parents scored QOL as the most important goal (mean score, 31.5 [SD, 8.4]), followed by health (26.3 [SD, 7.5]), comfort (22.4 [SD, 11.7]), disease modification (10.9 [SD, 9.2]), and life extension (8.9 [SD, 9.9]). Importantly, parents varied substantially in their baseline scores for each goal (IQRs more than 9.4), but across patients in different complex chronic conditions categories, the mean scores varied only slightly (means differ 8.7 or less). For each additional study month since PPC initiation, QOL was scored higher by 0.06 (95% CI, 0.04-0.08) and comfort scored higher by 0.3 (95% CI, 0-0.06), while the importance score for life extension decreased by 0.07 (95% CI, 0.04-0.09) and disease modification by 0.02 (95% CI, 0-0.04); health scores did not significantly differ from PPC initiation.
Conclusions and Relevance
Parents of children receiving PPC placed the highest value on QOL, but with considerable individual-level variation and substantial change over time. These findings emphasize the importance of reassessing GOCs with parents to guide appropriate clinical intervention.
Introduction
Both adult and pediatric medicine emphasize the importance of patient and parent goals of care (GOCs) to integrate their beliefs and values into care decisions and guide medical and surgical interventions.1,2,3,4 GOCs are especially important for patients with serious illness confronting difficult decisions in situations where treatment options pose tradeoffs.5 Although clinicians often wait to initiate a GOCs discussion until the patient has experienced a medical crisis or is near the end of life,6,7 general expert consensus holds that GOCs discussions should start early in the treatment of the illness with rediscussions over time.5,8,9
Despite this broad consensus regarding the importance of GOCs, many key aspects of how patients, parents, or other surrogate decision makers prioritize different potential GOCs and how these priorities change over time are unknown. Most of what we do know is based on qualitative analysis of 1-time interviews of parents of children or clinicians who care for children or adults receiving palliative care3,10,11,12,13 and medical record reviews,6,14 with a few exceptions of longitudinal interviews,15 surveys,16 and quantitative assessment of video recordings of clinician-patient interactions while discussing GOCs.17 What is notably missing from the literature is information about how different GOCs relate to each other and how stable these GOCs are over time.
Therefore, we undertook, as part of an encompassing cohort study of children with serious illness who had been receiving pediatric palliative care (PPC) services for different lengths of time, to collect data regarding parents’ importance scores of 5 different GOCs (patient’s quality of life [QOL], health, comfort, disease reduction, and life extension) over the course of 2 years. We limited this study to parents because most of the enrolled patients were too young or too ill or impaired to participate in this rating exercise. Given the paucity of quantitative data regarding pediatric GOCs, our primary analytic objective was descriptive with secondary objectives of determining whether specific GOCs priorities were associated with categories of underlying diseases or conditions, or with length of time receiving palliative care services.
Methods
Study Design, Setting, and Participants
As reported previously,18,19,20 this longitudinal study used parent-reported baseline data from the ongoing Pediatric Palliative Care Research Network’s Shared Data and Research 2-year prospective cohort study being conducted at 7 children’s hospitals across the US. Patients were eligible if they were receiving PPC services and were younger than 30 years. Primary caregivers responsible for the patient (referred to as “parent” in this article) were eligible to participate if they were at least 18 years old or a minor permitted by state law to consent for their own participation and able to speak and understand English or Spanish. Institutional review boards at all participating sites approved the conduct of this study. Parents provided written informed consent. This study followed the Strengthening the Reporting of Observational Studies in Epidemiology (STROBE) reporting guidelines.
Enrollment began in April 2017 and ended in March 2021. Among the 728 eligible parents of the 664 patients who enrolled and consented, 680 of 728 parents (93.4%) representing 603 patients had at least 1 evaluable GOCs survey completed by February 15, 2022.
Data Collection
At study entry and subsequent assessments at 2, 6, 12, 18, and 24 months, parents completed a set of instruments either at the hospital, in outpatient clinic, or at home, with data recorded either via paper or internet-based forms. Parents provided information regarding their child’s and their own age, sex, and self-identified race and ethnicity (to assess for disparities in PPC).21,22,23,24
Time in PPC
At baseline, patients had been receiving PPC for varying amounts of time. Site coordinators conducted medical record review for each patient to identify the date when PPC was initiated.
GOCs Assessment and Scoring
At each assessment, parents completed a discrete choice experiment consisting of 5 predetermined specific GOCs that were selected based on a literature review and the study team’s prior work (eMethods 1 in Supplement 1).9,16,25,26,27,28,29,30 GOCs were stated concisely in terms used commonly in clinical practice: seeking QOL, seeking health, seeking comfort, seeking disease reduction, and seeking longer life. These GOCs were presented to parents 2 goals at a time and parents were asked to pick the more important goal to them (eFigure 2 in Supplement 1). This process repeated across 10 permuted GOCs sets. Based on parents’ choices, relative importance scores for each goal were calculated in comparison with the other goals: a score twice as large indicated that that goal was twice as likely to be picked as more important. The scores were scaled so that the sum of the importance scores of all 5 GOCs equaled 100.
GOCs Differentiation Metric
We conceptualized a spectrum of differentiation of the GOCs from a scenario where all goals had nearly identical importance scores (called equivalence) to scenarios where the 5 goals had scores that increasingly differed (called differentiated.) We operationalized this spectrum by calculating the SD of the importance scores for the 5 goals for each individual parent; an SD of 0 indicated the equivalence end of the spectrum, whereas increases in the SD indicated further differentiation across study assessment time points (eTable 6 and eMethods 2 in Supplement 1).
GOCs Rank
Similarly, at each assessment, GOCs were ranked by importance scores with the highest-scoring GOC ranking as 1 and the lowest as 5. The rank order of goals at each consecutive assessment was then compared with the previous assessment to extract 2 dichotomous outcomes: (1) if any goal was reprioritized in rank order and (2) if there was a change in the top goal (ie, first-ranked goal).
Complex Chronic Condition (CCC) Designation
Enrolled patients’ medical record numbers were used to match to data in the Pediatric Hospital Information System ([PHIS], Children’s Hospital Association). Of the 603 participants, 592 had matching PHIS data. International Statistical Classification of Diseases, Tenth Revision, Clinical Modification codes were used to identify whether participating patients had any of 12 different nonmutually exclusive CCC categories.31 For patients with no PHIS data, CCC categories were identified via medical records review.
Statistical Analysis
Descriptive statistics were calculated to summarize demographic and clinical characteristics of patients receiving PPC, including parents’ GOCs importance scores. Differences in GOCs baseline assessment importance scores by patient characteristics were assessed using bivariable t tests and multivariable linear regression. Missing data over time in this analysis was due to missed data collections, study dropout, and patient death. To account for missing data due to missed data collections and dropout (but not death), all models were run using multiply imputed data.32,33 To account for potential bias due to patient death, parametric joint models (PJM) were run34 to assess the possible joint association between study outcomes and patient death by using a shared random intercept for each parent between the longitudinal model and a survival model. Two separate logistic PJM models were used to assess if there were (1) any rank order changes in goal rankings or (2) a change in the top goal within parents as a function of the previously measured degree of differentiation of the GOCs importance scores. Separate linear PJM models examined change in each goal over time since PPC initiation. Analyses controlled for patient characteristics and standard errors were calculated using robust clustering to account for family-level correlations arising when more than 1 parent per patient participated. All analyses were 2-tailed with an α of .05 and were performed using Stata version 17.0 (StataCorp). See eMethods 1 in Supplement 1 for more detail about study design and methods.
Results
Among the 603 patients receiving PPC for whom 680 parents completed at least 1 GOCs assessment (Table and eTable 1 in Supplement 1), patients had a median age of 4.4 (IQR, 0.8-13.2) years. Just over half were male (320 of 603 [53.1%]) and identified by their parents as White (436 of 603 [72.3%]), Black (50 of 603 [8.3%]), Asian (16 of 603 [2.7%]), Native Population (16 of 603 [2.7%]), or other (parents could respond “other” and then provide an open-ended response)/multiple race (85 of 603 [14.1%]), and 89 were identified as Hispanic (14.8%). In terms of clinical characteristics (eTable 2 in Supplement 1), at the time of study entry, patients had been receiving PPC services for a median of 55 (IQR, 13-413) days. Most patients (505 of 603 [83.7%]) were technology dependent and all patients had at least 1 CCC diagnosis. The most prevalent CCC categories were gastrointestinal (400 of 603 [66.3%]), neurological (346 of 603 [57.4%]), and cardiovascular (331 of 603 [54.9%]). Patients had a median of 5 different CCC categories of diagnoses (IQR, 4-7).
Table. Patient Demographic Characteristics and Goals of Care Importance Scores at Baseline Assessmenta.
| Characteristic | No. (%) | Mean (SD) | ||||
|---|---|---|---|---|---|---|
| Quality of life | Health | Comfort | Disease reduction | Life extension | ||
| Full sample | 603 (100.0) | 31.5 (8.4) | 26.3 (7.5) | 22.4 (11.7) | 10.9 (9.2) | 8.9 (9.9) |
| Age | ||||||
| <1 mo | 18 (3.0) | 32.1 (7.9) | 25.5 (9.3) | 23.0 (12.1) | 6.4 (7.0) | 12.9 (10.4) |
| 1-11 mo | 149 (24.7) | 30.4 (8.8) | 27.5 (7.0) | 20.4 (11.3) | 11.4 (9.7) | 10.3 (11.1) |
| 1-9 y | 223 (37.0) | 31.5 (8.7) | 26.3 (7.5) | 22.6 (11.6) | 11.3 (8.9) | 8.4 (9.2) |
| 10-17 y | 159 (26.4) | 32.1 (7.9) | 25.6 (7.5) | 22.9 (12.1) | 11.4 (9.7) | 8.0 (9.3) |
| 18-28 y | 54 (9.0) | 33.3 (7.4) | 25.0 (7.9) | 25.3 (11.4) | 7.6 (7.4) | 8.7 (10.2) |
| Sex | ||||||
| Female | 283 (46.9) | 31.8 (8.3) | 26.8 (7.1) | 22.2 (11.9) | 11.1 (9.4) | 8.1 (9.7) |
| Male | 320 (53.1) | 31.4 (8.5) | 25.8 (7.8) | 22.5 (11.6) | 10.7 (9.0) | 9.7 (10.0) |
| Raceb | ||||||
| Asian | 16 (2.7) | 26.5 (9.0) | 30.0 (7.4) | 14.4 (12.6) | 17.1 (11.4) | 12.0 (10.1) |
| Black | 50 (8.3) | 28.3 (9.3) | 28.0 (7.4) | 19.1 (9.9) | 12.8 (9.7) | 11.9 (10.1) |
| Native Hawaiian/Pacific Islander | 16 (2.7) | 30.3 (10.1) | 30.4 (7.1) | 19.1 (12.3) | 11.6 (7.9) | 8.7 (9.6) |
| White | 436 (72.3) | 32.2 (8.1) | 26.0 (7.4) | 23.2 (11.7) | 10.1 (8.9) | 8.5 (10.0) |
| Otherc or more than 1 race | 85 (14.1) | 31.4 (8.2) | 25.3 (7.5) | 22.0 (11.8) | 12.4 (10.0) | 8.9 (8.9) |
| Ethnicityb | ||||||
| Hispanic | 89 (14.8) | 29.9 (8.5) | 27.7 (7.9) | 22.1 (12.0) | 10.9 (9.4) | 9.3 (9.2) |
| Non-Hispanic | 495 (82.1) | 31.9 (8.3) | 26.0 (7.4) | 22.5 (11.7) | 10.7 (9.1) | 8.9 (9.9) |
| Prefer not to answer | 17 (2.8) | 29.5 (9.1) | 26.1 (6.5) | 20.0 (10.4) | 15.1 (11.2) | 9.4 (11.4) |
| Missing | 2 (0.3) | NA | NA | NA | NA | NA |
Abbreviation: NA, not available.
Demographic characteristics numbers and percents represent the full sample of patients. Goals of care scores only represent 667 parents of 592 patients who completed the goals of care instrument at baseline. For a version of this table with statistical comparisons of the various strata of demographic characteristics, please see eTable 2 in Supplement 1.
Race and ethnicity were identified by patients’ parents.
Parents could respond “other” and then provide an open-ended response.
The 680 parents (eTable 3 in Supplement 1) included in the study had a mean age of 37.7 (SD, 9.5) years, were mostly female (543 of 680 [79.9%]), most self-identified as White (524 of 680 [77.1%]), and 12.6% (86 of 680) self-identified as Hispanic. Almost a quarter (156 of 680 [23.0%]) reported quite a bit or a great deal of financial difficulties, 51.5% (344 of 680) reported being moderately distressed, and 17.4% (116 of 680) severely distressed.
Progression of the Cohort Over Time
At baseline, 667 parents (98.1%) reported on their GOCs (with 13 parents completing GOCs questionnaires only at later assessments) regarding 592 patients (98.2%). At various time points after the baseline data collection, 195 patients died (32.3%) and 38 parents withdrew or disengaged from the study (5.7%). This attrition, combined with some parents not completing each assessment of data collection, affected follow-up data collection. GOCs questionnaire completion (eFigure 1 in Supplement 1) decreased over time as follows: 436 parents at 2 months (72.3%), 355 parents at 6 months (58.9%), 275 parents at 12 months (40.4%), 211 parents at 18 months (31.0%), and 180 parents at 24 months (26.5%).
GOCs Importance Scores at Baseline
Among the 5 GOCs, QOL was given the highest mean (SD) score at baseline (31.5 [SD, 8.4]), followed in descending order by pursuit of health (26.3 [SD, 7.5]), pursuit of comfort (22.4 [SD, 11.7]), disease reduction (10.9 [SD, 9.2]), and life extension (8.9 [SD, 9.9]) (Figure 1). Notably, compared with parents of White patients, parents of Black patients scored life extension (mean, 11.9 [SD, 10.1] to 8.5 [SD, 10.0]; P = .03) and QOL (32.2 [SD, 8.1] to 28.3 [SD, 9.3]; P = .003) significantly higher and comfort (19.1 [SD, 9.9] to 23.2 [SD, 11.7]; P = .02) significantly lower with parents of Asian patients showing similar patterns to White patients for health, comfort, disease modification, and QOL (eTable 1 in Supplement 1). Additionally, the importance scores for each GOC varied considerably across participants with an IQR span of 9.4 or more (Figure 1). The sort order of the importance scores was consistent across 10 of the 11 CCC categories (Figure 2) with the exception of parents of children with neonatal CCC, who scored life extension as more important than disease modification.
Figure 1. Distribution of Importance Scores for Each Goal of Care at Study Entry.
For each goal, the box represents data between the 25th to 75th percentile; the break within the box represents the median value, the horizontal capped bars extending from each box represent 1.5 IQR limits, and the circles outside the capped bars represent values beyond 1.5 IQR from the median.
Figure 2. Goals of Care Importance Scores at Baseline by Types of Complex Chronic Condition (CCC).
All goals of care were stated as seeking each specific goal. The dots are located at the mean score at baseline for each goal. QOL indicates quality of life.
Trajectories of GOCs Importance Scores Over Time
The study team assembled trajectories of the GOCs importance scores over time (Figure 3), accounting for the various amounts of time between when patients initiated PPC and when they entered the study. In models adjusted for patient demographics and number of CCCs (eTable 4 in Supplement 1), at PPC initiation, QOL had the highest importance score (32.2; 95% CI, 30.8-33.6), followed by health (28.8; 95% CI, 27.6-30.1), comfort (20.6; 95% CI, 18.1-23.1), disease modification (11.7; 95% CI, 9.8-13.6), and life extension (6.5; 95% CI, 4.6-8.5 ). Over time, each additional study month enrolled in PPC was associated with a score increase of 0.06 (95% CI, 0.04-0.08) for QOL and 0.03 (95% CI, 0-0.06) for comfort.
Figure 3. Goals of Care Importance Scores From Initiation of Pediatric Palliative Care (PPC) Services Forward Over Time.
Goals of care importance scores overtime smoothed with a lowest fit. Note that the horizontal time scale is log (base 10) transformed.
Individual-Level Change in Rank Ordering of GOCs Over Time
While the trajectories described above present a pattern marked more by continuity than change, this was at the group and not the individual level. Focusing on individual participants in the sample (Figure 4), the likelihood over time that a given participant’s GOCs would change in rank ordering was essentially 80% over the 5 separate time intervals where change could have been detected in the study. More important, the percentage of participants who changed their top goal during any 1 of these intervals hovered in the 35% to 40% range throughout the study.
Figure 4. Occurrence of Change in Rank Ordering of Goals of Care During 5 Successive Time Intervals.
Goal Differentiation and Subsequent Change of GOCs Rank Ordering
Parents with greater differentiation of the GOCs at 1 point in time (that is, their importance scores among the 5 GOCs differed to a greater degree compared with other parents) were less likely to change the ranking of any goal at the subsequent assessment (adjusted odds ratio, 0.78; 95% CI, 0.69-0.88) or change the top scored goal (adjusted odds ratio, 0.88; 95% CI, 0.81-0.95) after adjustment for patient and parent demographic and clinical characteristics (eTable 5 in Supplement 1).
Discussion
In this large sample of parents of children and young adults receiving PPC services, we found that the GOC with the highest average importance score was the pursuit of QOL for their child. This was true at the point of study entry and across the range of time that patients had been receiving PPC services. The pursuit of health had the next highest average importance score at study entry, followed closely by the pursuit of comfort. Importantly, GOCs scores varied substantially across parents (at all time points, some parents scored the importance of each of the 5 GOCs as the most important) but much less variation in scores was observed regarding patients across different categories of CCCs. Over the 2 years of the study, parents often changed the relative importance of their GOCs, with the top-scored goal changing about 40% of the time from 1 study visit to the next. If the parent had differentiated the importance of the different GOCs to a greater degree, they were less likely to subsequently change the prioritization of their GOCs.
We found that parents of Black patients placed greater importance on health and life extension and less importance on QOL compared with White parents. This finding is consistent with prior research showing that non-White and Hispanic patients spend more time in the hospital,35 are less likely to receive hospice care,36 and that these differences in care may be concordant with parent goals.37
These findings accord with much of what has been learned about parental GOCs, although given the novelty of the questions posed in this study and of the quantitative approach to providing the answers, comparison with other studies is not direct. Prior research has focused on whether clinicians and parents have similar GOCs,3,16 whether parents and adolescents/young adults agree on GOCs,38 whether GOCs are discussed toward the end of life or earlier,6,7,34 and whether parent understanding of prognosis predicts GOCs.11,15,16 Many of these studies conceptualize the GOCs over time in a simplified binary manner with a shift from curative goals at initial diagnosis to the goal of lessening suffering at the end of life, especially once parents understand the child is likely to die.11,12,15,16 No prior studies, to our knowledge, used a quantitative approach to examine how parents prioritize multiple GOCs over time across multiple life-threatening conditions and across disease trajectories.
Strengths and Limitations
Several features of this study strengthen the generalizability of the findings, including the large, multicenter, geographically diverse sample, the representation of fathers as well as mothers, the multilingual cohort, the use of quantitative methodology, and the 2-year cohort study design. This study has 4 main limitations: the GOCs discrete choice experiment method is a closed-form exercise that does not allow for a discursive GOCs conversation, the choices that lead to the importance scores may not map to the importance that parents place on a goal in a specific clinical situation, attrition of the study cohort over time may have introduced potential biases over time (which the multiple imputation and joint modeling methods sought to minimize), and the predominately White participants limit generalizability.
With these strengths and limitations in mind, 4 points warrant discussion. First, we found considerable variation among what individual parents considered most important for their child. Prior studies have also observed a range of different goals among parents of children receiving palliative care.3,13,16,23 Second, on average, parents placed greater importance on QOL over comfort. Over time, parents tended to give higher importance ratings for QOL and comfort while importance scores for other goals remained the same or decreased. Notably, we did not define the goals for parents in this study, including QOL and comfort, as these concepts are often referred to but not typically defined in clinical practice. Prior work, however, has shown regarding these 2 concepts that parents and adult patients generally think of comfort as being free from unpleasant physical symptoms, such as pain or nausea, whereas QOL includes broader issues like being able to participate in life in a meaningful and enjoyable way.39,40,41,42 Parental focus on QOL aligns with earlier studies, with the exception of parents of children with cancer who perceived their child’s illness to be curable.11,12,15,16,29,43 Third, across months and years of received palliative care, maintaining the child’s health continued to be important to parents. The few studies that have examined parents’ goals at different times have been retrospective or 1-time cross-sectional studies with qualitative data (eg, in 1 study visit, parents were asked to recall and report on goals at multiple time points in the past).43,44,45,46 Lastly, we found that parents who differentiated the GOCs by importance scores had more stable goal priorities over time, an issue which, to out knowledge, has not been examined before.
These findings suggest important implications for clinical practice. First, while the overall pattern of goal priorities of parents of patients receiving PPC may be similar over time, individual parents may change their priorities and some significantly. Pediatric clinicians should therefore reassess parents’ GOCs regularly for potential changes throughout the course of a child’s illness and not solely when there is a significant change in clinical status. Second, while QOL and comfort may be most important for these parents over time, their desire to maintain their child’s health and prolong their child’s life never goes away. Third, parents who indicate that all GOCs are equally important may need to be reassessed more often as their GOCs may shift more substantially over time while they are in the process of determining what goals are most important for their child.
Conclusions
Parents of children receiving PPC placed the highest value on QOL, but with considerable individual-level variation. Furthermore, while these parents overall show somewhat stable patterns in their GOCs priorities, individual parents may often change their GOCs over time, particularly those who initially reported that all GOCs are equally important.
eTable 1. Patient demographic characteristics and goals of care importance scores at baseline assessment
eTable 2. Patient clinical characteristics and goals of care importance scores at baseline assessment
eTable 3. Parent characteristics and goals of care importance scores at baseline assessment
eTable 4. Parametric joint models of goals of care importance scores since PPC initiation
eTable 5. Patient demographic and clinical characteristics associated with changes in rankings of goals of care based on importance scores over time
eFigure 1. CONSORT diagram of study’s longitudinal cohort
eMethods 1. Elaboration on study design, methods, and findings
eMethods 2. Supplemental results
eReferences
eTable 6. Example of differentiation metric calculations
eFigure 2. Goals of care measure
Group information
Data sharing statement
References
- 1.Myers J, Cosby R, Gzik D, et al. Provider tools for advance care planning and goals of care discussions: a systematic review. Am J Hosp Palliat Care. 2018;35(8):1123-1132. doi: 10.1177/1049909118760303 [DOI] [PubMed] [Google Scholar]
- 2.Bernacki RE, Block SD; American College of Physicians High Value Care Task Force . Communication about serious illness care goals: a review and synthesis of best practices. JAMA Intern Med. 2014;174(12):1994-2003. doi: 10.1001/jamainternmed.2014.5271 [DOI] [PubMed] [Google Scholar]
- 3.Hoang K, Halpern-Felsher B, Brooks M, Blankenburg R. Addressing goals of parents of hospitalized children: a qualitative analysis of parents’ and physicians’ perspectives. Acad Pediatr. 2021;21(2):244-251. [DOI] [PubMed] [Google Scholar]
- 4.Secunda K, Wirpsa MJ, Neely KJ, et al. Use and meaning of “goals of care” in the healthcare literature: a systematic review and qualitative discourse analysis. J Gen Intern Med. 2020;35(5):1559-1566. doi: 10.1007/s11606-019-05446-0 [DOI] [PMC free article] [PubMed] [Google Scholar]
- 5.Feudtner C, Schall T, Hill D. Parental personal sense of duty as a foundation of pediatric medical decision-making. Pediatrics. 2018;142(suppl 3):S133-S141. doi: 10.1542/peds.2018-0516C [DOI] [PubMed] [Google Scholar]
- 6.Hui D, Con A, Christie G, Hawley PH. Goals of care and end-of-life decision making for hospitalized patients at a Canadian tertiary care cancer center. J Pain Symptom Manage. 2009;38(6):871-881. doi: 10.1016/j.jpainsymman.2009.05.017 [DOI] [PubMed] [Google Scholar]
- 7.Verberne LM, Fahner JC, Sondaal SFV, et al. Anticipating the future of the child and family in pediatric palliative care: a qualitative study into the perspectives of parents and healthcare professionals. Eur J Pediatr. 2021;180(3):949-957. doi: 10.1007/s00431-020-03824-z [DOI] [PMC free article] [PubMed] [Google Scholar]
- 8.Thomas RL, Zubair MY, Hayes B, Ashby MA. Goals of care: a clinical framework for limitation of medical treatment. Med J Aust. 2014;201(8):452-455. doi: 10.5694/mja14.00623 [DOI] [PubMed] [Google Scholar]
- 9.Walter JK, Nestor K, Feudtner C. Parental perspectives on goals of care discussions with the healthcare team for their child with cancer. Psychooncology. 2016;25(8):990-993. doi: 10.1002/pon.3985 [DOI] [PubMed] [Google Scholar]
- 10.Hinds PS, Oakes LL, Hicks J, et al. “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. 2009;27(35):5979-5985. doi: 10.1200/JCO.2008.20.0204 [DOI] [PMC free article] [PubMed] [Google Scholar]
- 11.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. 2000;284(19):2469-2475. doi: 10.1001/jama.284.19.2469 [DOI] [PubMed] [Google Scholar]
- 12.Edwards KE, Neville BA, Cook EF Jr, Aldridge SH, Dussel V, Wolfe J. Understanding of prognosis and goals of care among couples whose child died of cancer. J Clin Oncol. 2008;26(8):1310-1315. doi: 10.1200/JCO.2007.13.4056 [DOI] [PubMed] [Google Scholar]
- 13.Tamburro RF, Shaffer ML, Hahnlen NC, Felker P, Ceneviva GD. Care goals and decisions for children referred to a pediatric palliative care program. J Palliat Med. 2011;14(5):607-613. doi: 10.1089/jpm.2010.0450 [DOI] [PubMed] [Google Scholar]
- 14.Niswander LM, Cromwell P, Chirico J, Gupton A, Korones DN. End-of-life care for children enrolled in a community-based pediatric palliative care program. J Palliat Med. 2014;17(5):589-591. doi: 10.1089/jpm.2013.0576 [DOI] [PubMed] [Google Scholar]
- 15.Mack JW, Cronin AM, Uno H, et al. Unrealistic parental expectations for cure in poor-prognosis childhood cancer. Cancer. 2020;126(2):416-424. doi: 10.1002/cncr.32553 [DOI] [PMC free article] [PubMed] [Google Scholar]
- 16.Rosenberg AR, Orellana L, Kang TI, et al. Differences in parent-provider concordance regarding prognosis and goals of care among children with advanced cancer. J Clin Oncol. 2014;32(27):3005-3011. doi: 10.1200/JCO.2014.55.4659 [DOI] [PMC free article] [PubMed] [Google Scholar]
- 17.Land V, Parry R, Pino M, Jenkins L, Feathers L, Faull C. Addressing possible problems with patients’ expectations, plans and decisions for the future: one strategy used by experienced clinicians in advance care planning conversations. Patient Educ Couns. 2019;102(4):670-679. doi: 10.1016/j.pec.2018.11.008 [DOI] [PubMed] [Google Scholar]
- 18.Feudtner C, Nye R, Hill DL, et al. ; Pediatric Palliative Care Research Network Shared Data and Research (PPCRN SHARE) Project Group . Polysymptomatology in pediatric patients receiving palliative care based on parent-reported data. JAMA Netw Open. 2021;4(8):e2119730. doi: 10.1001/jamanetworkopen.2021.19730 [DOI] [PMC free article] [PubMed] [Google Scholar]
- 19.Boyden JY, Hill DL, Nye RT, et al. Pediatric palliative care parents’ distress, financial difficulty, and child symptoms. J Pain Symptom Manage. 2022;(2):271-282. [DOI] [PMC free article] [PubMed] [Google Scholar]
- 20.Nye RT, Hill DL, Carroll KW, et al. The design of a data management system for a multicenter palliative care cohort study. J Pain Symptom Manage. 2022;64(1):e53-e60. doi: 10.1016/j.jpainsymman.2022.03.006 [DOI] [PMC free article] [PubMed] [Google Scholar]
- 21.Johnston EE, Bogetz J, Saynina O, Chamberlain LJ, Bhatia S, Sanders L. Disparities in inpatient intensity of end-of-life care for complex chronic conditions. Pediatrics. 2019;143(5):e20182228. doi: 10.1542/peds.2018-2228 [DOI] [PubMed] [Google Scholar]
- 22.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(3):574-582. doi: 10.1542/peds.2007-3042 [DOI] [PubMed] [Google Scholar]
- 23.Mack JW, Uno H, Twist CJ, et al. Racial and ethnic differences in communication and care for children with advanced cancer. J Pain Symptom Manage. 2020;60(4):782-789. doi: 10.1016/j.jpainsymman.2020.04.020 [DOI] [PMC free article] [PubMed] [Google Scholar]
- 24.Trowbridge A, Walter JK, McConathey E, Morrison W, Feudtner C. Modes of death within a children’s hospital. Pediatrics. 2018;142(4):e20174182. doi: 10.1542/peds.2017-4182 [DOI] [PubMed] [Google Scholar]
- 25.Feudtner C, Walter JK, Faerber JA, et al. Good-parent beliefs of parents of seriously ill children. JAMA Pediatr. 2015;169(1):39-47. doi: 10.1001/jamapediatrics.2014.2341 [DOI] [PMC free article] [PubMed] [Google Scholar]
- 26.Mooney-Doyle K, Deatrick JA, Ulrich CM, Meghani SH, Feudtner C. Parenting in childhood life-threatening illness: a mixed-methods study. J Palliat Med. 2018;21(2):208-215. doi: 10.1089/jpm.2017.0054 [DOI] [PMC free article] [PubMed] [Google Scholar]
- 27.Tubbs-Cooley HL, Santucci G, Kang TI, Feinstein JA, Hexem KR, Feudtner C. Pediatric nurses’ individual and group assessments of palliative, end-of-life, and bereavement care. J Palliat Med. 2011;14(5):631-637. doi: 10.1089/jpm.2010.0409 [DOI] [PMC free article] [PubMed] [Google Scholar]
- 28.Mack JW, Feudtner C, Hinds PS. Communication and decision support for children with advanced cancer and their families. Am Soc Clin Oncol Educ Book. 2012:637-643. doi: 10.14694/EdBook_AM.2012.32.164 [DOI] [PubMed] [Google Scholar]
- 29.Rosenberg AR, Dussel V, Kang T, et al. Psychological distress in parents of children with advanced cancer. JAMA Pediatr. 2013;167(6):537-543. doi: 10.1001/jamapediatrics.2013.628 [DOI] [PMC free article] [PubMed] [Google Scholar]
- 30.Carroll KW, Mollen CJ, Aldridge S, Hexem KR, Feudtner C. Influences on decision making identified by parents of children receiving pediatric palliative care. AJOB Prim Res. 2012;3(1):1-7. doi: 10.1080/21507716.2011.638019 [DOI] [PMC free article] [PubMed] [Google Scholar]
- 31.Feudtner C, Feinstein JA, Zhong W, Hall M, Dai D. Pediatric complex chronic conditions classification system version 2: updated for ICD-10 and complex medical technology dependence and transplantation. BMC Pediatr. 2014;14:199. doi: 10.1186/1471-2431-14-199 [DOI] [PMC free article] [PubMed] [Google Scholar]
- 32.Sterne JA, White IR, Carlin JB, et al. Multiple imputation for missing data in epidemiological and clinical research: potential and pitfalls. BMJ. 2009;338:b2393. doi: 10.1136/bmj.b2393 [DOI] [PMC free article] [PubMed] [Google Scholar]
- 33.Harel O, Mitchell EM, Perkins NJ, et al. Multiple imputation for incomplete data in epidemiologic studies. Am J Epidemiol. 2018;187(3):576-584. doi: 10.1093/aje/kwx349 [DOI] [PMC free article] [PubMed] [Google Scholar]
- 34.Dil E, Karasoy D. gsem: A Stata command for parametric joint modelling of longitudinal and accelerated failure time models. Comput Methods Programs Biomed. 2020;196:105612. doi: 10.1016/j.cmpb.2020.105612 [DOI] [PubMed] [Google Scholar]
- 35.DeGroote NP, Allen KE, Falk EE, et al. Relationship of race and ethnicity on access, timing, and disparities in pediatric palliative care for children with cancer. Support Care Cancer. 2022;30(1):923-930. doi: 10.1007/s00520-021-06500-6 [DOI] [PubMed] [Google Scholar]
- 36.Thienprayoon R, Lee SC, Leonard D, Winick N. Racial and ethnic differences in hospice enrollment among children with cancer. Pediatr Blood Cancer. 2013;60(10):1662-1666. doi: 10.1002/pbc.24590 [DOI] [PubMed] [Google Scholar]
- 37.Umaretiya PJ, Li A, McGovern A, Ma C, Wolfe J, Bona K. Race, ethnicity, and goal-concordance of end-of-life palliative care in pediatric oncology. Cancer. 2021;127(20):3893-3900. doi: 10.1002/cncr.33768 [DOI] [PubMed] [Google Scholar]
- 38.Friebert S, Grossoehme DH, Baker JN, et al. Congruence gaps between adolescents with cancer and their families regarding values, goals, and beliefs about end-of-life care. JAMA Netw Open. 2020;3(5):e205424. doi: 10.1001/jamanetworkopen.2020.5424 [DOI] [PMC free article] [PubMed] [Google Scholar]
- 39.Hill DL, Miller VA, Hexem KR, et al. Problems and hopes perceived by mothers, fathers and physicians of children receiving palliative care. Health Expect. 2015;18(5):1052-1065. [DOI] [PMC free article] [PubMed] [Google Scholar]
- 40.Hill DL, Nathanson PG, Fenderson RM, Carroll KW, Feudtner C. Parental concordance regarding problems and hopes for seriously ill children: a two-year cohort study. J Pain Symptom Manage. 2017;53(5):911-918. doi: 10.1016/j.jpainsymman.2016.11.007 [DOI] [PMC free article] [PubMed] [Google Scholar]
- 41.Pinto S, Caldeira S, Martins JC. A systematic literature review toward the characterization of comfort. Holist Nurs Pract. 2016;30(1):14-24. doi: 10.1097/HNP.0000000000000126 [DOI] [PubMed] [Google Scholar]
- 42.Pinto S, Fumincelli L, Mazzo A, Caldeira S, Martins JC. Comfort, well-being and quality of life: discussion of the differences and similarities among the concepts. Porto Biomed J. 2017;2(1):6-12. doi: 10.1016/j.pbj.2016.11.003 [DOI] [PMC free article] [PubMed] [Google Scholar]
- 43.Levine DR, Johnson LM, Snyder A, et al. Integrating palliative care in pediatric oncology: evidence for an evolving paradigm for comprehensive cancer care. J Natl Compr Canc Netw. 2016;14(6):741-748. doi: 10.6004/jnccn.2016.0076 [DOI] [PMC free article] [PubMed] [Google Scholar]
- 44.Fratantoni K, Livingston J, Schellinger SE, Aoun SM, Lyon ME. Family-centered advance care planning: what matters most for parents of children with rare diseases. Children (Basel). 2022;9(3):445. doi: 10.3390/children9030445 [DOI] [PMC free article] [PubMed] [Google Scholar]
- 45.Levine DR, Van Noy K, Talleur AC, Snyder A, Kaye EC, Baker JN. Thoughts from the threshold: patient and family hopes, fears, values, and goals at the onset of pediatric hematopoietic cell transplantation. Bone Marrow Transplant. 2020;55(6):1103-1113. doi: 10.1038/s41409-020-0794-8 [DOI] [PMC free article] [PubMed] [Google Scholar]
- 46.Mack JW, Fisher L, Kushi L, et al. Patient, family, and clinician perspectives on end-of-life care quality domains and candidate indicators for adolescents and young adults with cancer. JAMA Netw Open. 2021;4(8):e2121888. doi: 10.1001/jamanetworkopen.2021.21888 [DOI] [PMC free article] [PubMed] [Google Scholar]
Associated Data
This section collects any data citations, data availability statements, or supplementary materials included in this article.
Supplementary Materials
eTable 1. Patient demographic characteristics and goals of care importance scores at baseline assessment
eTable 2. Patient clinical characteristics and goals of care importance scores at baseline assessment
eTable 3. Parent characteristics and goals of care importance scores at baseline assessment
eTable 4. Parametric joint models of goals of care importance scores since PPC initiation
eTable 5. Patient demographic and clinical characteristics associated with changes in rankings of goals of care based on importance scores over time
eFigure 1. CONSORT diagram of study’s longitudinal cohort
eMethods 1. Elaboration on study design, methods, and findings
eMethods 2. Supplemental results
eReferences
eTable 6. Example of differentiation metric calculations
eFigure 2. Goals of care measure
Group information
Data sharing statement