Advancing a comprehensive cancer care agenda for children and their families: Institute of Medicine Workshop highlights and next steps.

2016

2016 May 4

1542-4863

<p>This article highlights key findings from the "Comprehensive Cancer Care for Children and Their Families" March 2015 joint workshop by the Institute of Medicine (IOM) and the American Cancer Society. This initiative convened more than 100 family members, clinician investigators, advocates, and members of the public to discuss emerging evidence and care models and to determine the next steps for optimizing quality-of-life outcomes and well-being for children and families during pediatric cancer treatment, after treatment completion, and across the life spectrum. Participants affirmed the triple aim of pediatric oncology that strives for every child with cancer to be cured; provides high-quality palliative and psychosocial supportive, restorative, and rehabilitative care to children and families throughout the illness course and survivorship; and assures receipt of high-quality end-of-life care for patients with advancing disease. Workshop outcomes emphasized the need for new pediatric cancer drug development and identified critical opportunities to prioritize palliative care and psychosocial support as an integral part of pediatric cancer research and treatment, including the necessity for adequately resourcing these supportive services to minimize suffering and distress, effectively address quality-of-life needs for children and families at all stages of illness, and mitigate the long-term health risks associated with childhood cancer and its treatment. Next steps include dismantling existing silos and enhancing collaboration between clinical investigators, disease-directed specialists, and supportive care services; expanding the use of patient-reported and parent-reported outcomes; effectively integrating palliative and psychosocial care; and clinical communication skills development. CA Cancer J Clin 2016. © 2016 American Cancer Society.</p>

10.3322/caac.21347

CA Cancer J Clin

27145249

What's missing in missing data? Omissions in survey responses among parents of children with advanced cancer.

2014

953-6

2014 Aug

1557-7740

<p><strong>BACKGROUND: </strong>Missing data is a common phenomenon with survey-based research; patterns of missing data may elucidate why participants decline to answer certain questions.</p>

<p><strong>OBJECTIVE: </strong>To describe patterns of missing data in the Pediatric Quality of Life and Evaluation of Symptoms Technology (PediQUEST) study, and highlight challenges in asking sensitive research questions.</p>

<p><strong>DESIGN: </strong>Cross-sectional, survey-based study embedded within a randomized controlled trial.</p>

<p><strong>SETTING: </strong>Three large children's hospitals: Dana-Farber/Boston Children's Cancer and Blood Disorders Center (DF/BCCDC); Children's Hospital of Philadelphia (CHOP); and Seattle Children's Hospital (SCH).</p>

<p><strong>MEASUREMENTS: </strong>At the time of their child's enrollment, parents completed the Survey about Caring for Children with Cancer (SCCC), including demographics, perceptions of prognosis, treatment goals, quality of life, and psychological distress.</p>

<p><strong>RESULTS: </strong>Eighty-six of 104 parents completed surveys (83% response). The proportion of missing data varied by question type. While 14 parents (16%) left demographic fields blank, over half (n=48; 56%) declined to answer at least one question about their child's prognosis, especially life expectancy. The presence of missing data was unrelated to the child's diagnosis, time from progression, time to death, or parent distress (p&gt;0.3 for each). Written explanations in survey margins suggested that addressing a child's life expectancy is particularly challenging for parents.</p>

<p><strong>CONCLUSIONS AND RELEVANCE: </strong>Parents of children with cancer commonly refrain from answering questions about their child's prognosis, however, they may be more likely to address general cure likelihood than explicit life expectancy. Understanding acceptability of sensitive questions in survey-based research will foster higher quality palliative care research.</p>

10.1089/jpm.2013.0663

J Palliat Med

24865204

Improving the care of children with advanced cancer by using an electronic patient-reported feedback intervention: results from the PediQUEST randomized controlled trial.

2014

1119-26

2014 Apr 10

1527-7755

<p><strong>PURPOSE: </strong>This study aimed to determine whether feeding back patient-reported outcomes (PROs) to providers and families of children with advanced cancer improves symptom distress and health-related quality of life (HRQoL).</p>

<p><strong>PATIENTS AND METHODS: </strong>This study was a parallel, multicentered pilot randomized controlled trial. At most once per week, children age ≥ 2 years old with advanced cancer or their parent completed the computer-based Pediatric Quality of Life and Evaluation of Symptoms Technology (PediQUEST) survey consisting of age- and respondent-adapted versions of the Memorial Symptom Assessment Scale (MSAS), Pediatric Quality of Life Inventory 4.0 Generic Core Scales (PedsQL4.0), and an overall Sickness question. In the intervention group (n = 51), oncologists and families received printed reports summarizing PROs; e-mails were sent to oncologists and subspecialists when predetermined scores were exceeded. No feedback was provided in the control group (n = 53). Primary outcomes included linear trends of MSAS, PedsQL4.0 total and subscale scores, and Sickness scores during 20 weeks of follow-up, along with child, parent, and provider satisfaction with PediQUEST feedback.</p>

<p><strong>RESULTS: </strong>Feedback did not significantly affect average MSAS, PedsQL4.0, or Sickness score trends. Post hoc subgroup analyses among children age ≥ 8 years who survived 20 weeks showed that feedback improved PedsQL4.0 emotional (+8.1; 95% CI, 1.8 to 14.4) and Sickness (-8.2; 95% CI, -14.2 to -2.2) scores. PediQUEST reports were valued by children, parents, and providers and contributed at least sometimes to physician initiation of a psychosocial consult (56%).</p>

<p><strong>CONCLUSION: </strong>Although routine feedback of PROs did not significantly affect the child's symptoms or HRQoL, changes were in expected directions and improvements observed in emotional HRQoL through exploratory analyses were encouraging. Importantly, children, parents, and providers value PRO feedback.</p>

10.1200/JCO.2013.51.5981

J. Clin. Oncol.

24616307

Economic impact of advanced pediatric cancer on families.

2014

594-603

2014 Mar

1873-6513

<p><strong>CONTEXT: </strong>Despite emerging evidence of substantial financial distress in families of children with complex illness, little is known about economic hardship in families of children with advanced cancer.</p>

<p><strong>OBJECTIVES: </strong>To describe perceived financial hardship, work disruptions, income losses, and associated economic impact in families of children with advanced cancer stratified by federal poverty level (FPL).</p>

<p><strong>METHODS: </strong>Cross-sectional survey of 86 parents of children with progressive, recurrent, or nonresponsive cancer at three children's hospitals. Seventy-one families with complete income data (82%) are included in this analysis.</p>

<p><strong>RESULTS: </strong>Parental work disruptions were prevalent across all income levels, with 67 (94%) families reporting some disruption. At least one parent quit a job because of the child's illness in 29 (42%) families. Nineteen (27%) families described their child's illness as a great economic hardship. Income losses because of work disruptions were substantial for all families; families at or below 200% FPL, however, were disproportionately affected. Six (50%) of the poorest families lost more than 40% of their annual income as compared with two (5%) of the wealthiest families (P&nbsp;=&nbsp;0.006). As a result of income losses, nine (15%) previously nonpoor families fell from above to below 200% FPL.</p>

<p><strong>CONCLUSION: </strong>The economic impact of pediatric advanced cancer on families is significant at all income levels, although poorer families suffer disproportionate losses. Development of ameliorative intervention strategies is warranted.</p>

10.1016/j.jpainsymman.2013.04.003

J Pain Symptom Manage

23870843

Psychological distress in parents of children with advanced cancer.

2013

537-43

2013 Jun

2168-6211

<p><strong>IMPORTANCE: </strong>Parent psychological distress can impact the well-being of childhood cancer patients and other children in the home. Recognizing and alleviating factors of parent distress may improve overall family survivorship experiences following childhood cancer.</p>

<p><strong>OBJECTIVES: </strong>To describe the prevalence and factors of psychological distress (PD) among parents of children with advanced cancer.</p>

<p><strong>DESIGN: </strong>Cohort study embedded within a randomized clinical trial (Pediatric Quality of Life and Evaluation of Symptoms Technology [PediQUEST] study).</p>

<p><strong>SETTING: </strong>Multicenter study conducted at 3 children's hospitals (Boston Children's Hospital, Children's Hospital of Philadelphia, and Seattle Children's Hospital).</p>

<p><strong>PARTICIPANTS: </strong>Parents of children with advanced (progressive, recurrent, or refractory) cancer.</p>

<p><strong>MAIN OUTCOME MEASURE: </strong>Parental PD, as measured by the Kessler-6 Psychological Distress Scale.</p>

<p><strong>RESULTS: </strong>Eighty-six of 104 parents completed the Survey About Caring for Children With Cancer (83% participation); 81 parents had complete Kessler-6 Psychological Distress Scale data. More than 50% of parents reported high PD and 16% met criteria for serious PD (compared with US prevalence of 2%-3%). Parent perceptions of prognosis, goals of therapy, child symptoms/suffering, and financial hardship were associated with PD. In multivariate analyses, average parent Kessler-6 Psychological Distress Scale scores were higher among parents who believed their child was suffering highly and who reported great economic hardship. Conversely, PD was significantly lower among parents whose prognostic understanding was aligned with concrete goals of care.</p>

<p><strong>CONCLUSIONS AND RELEVANCE: </strong>Parenting a child with advanced cancer is strongly associated with high to severe levels of PD. Interventions aimed at aligning prognostic understanding with concrete care goals and easing child suffering and financial hardship may mitigate parental PD.</p>

10.1001/jamapediatrics.2013.628

JAMA Pediatr

23545569

Hospital Use in the Last Year of Life for Children With Life-Threatening Complex Chronic Conditions.

2015

938-46

2015 Nov

1098-4275

<p><strong>BACKGROUND AND OBJECTIVES: </strong>Although many adults experience resource-intensive and costly health care in the last year of life, less is known about these health care experiences in children with life-threatening complex chronic conditions (LT-CCCs). We assessed hospital resource use in children by type and number of LT-CCCs.</p>

<p><strong>METHODS: </strong>A retrospective analysis of 1252 children with LT-CCCs, ages 1 to 18 years, who died in 2012 within 40 US children's hospitals of the Pediatric Health Information System database. LT-CCCs were identified with International Classification of Diseases, 9th Revision, Clinical Modification codes. Using generalized linear models, we assessed hospital admissions, days, costs, and interventions (mechanical ventilation and surgeries) in the last year of life by type and number of LT-CCCs.</p>

<p><strong>RESULTS: </strong>In the last year of life, children with LT-CCCs experienced a median of 2 admissions (interquartile range [IQR] 1-5), 27 hospital days (IQR 7-84), and $142 562 (IQR $45 270-$410 087) in hospital costs. During the terminal admission, 76% (n = 946) were mechanically ventilated; 36% (n = 453) underwent surgery. Hospital use was greatest (P &lt; .001) among children with hematologic/immunologic conditions (99 hospital days [IQR 51-146]; cost = $504 145 [IQR $250 147-$879 331]) and children with ≥3 LT-CCCs (75 hospital days [IQR 28-132]; cost = $341 222 [IQR $146 698-$686 585]).</p>

<p><strong>CONCLUSIONS: </strong>Hospital use for children with LT-CCCs in the last year of life varies significantly across the type and number of conditions. Children with hematologic/immunologic or multiple conditions have the greatest hospital use. This information may be useful for clinicians striving to improve care for children with LT-CCCs nearing the end of life.</p>

10.1542/peds.2015-0260

Pediatrics

26438707

Pediatric palliative care programs in children's hospitals: a cross-sectional national survey.

2013

1063-70

12/2013

1098-4275

<p><strong>BACKGROUND: </strong>Pediatric palliative care (PPC) programs facilitate the provision of comprehensive care to seriously ill children. Over the past 10 years many such programs have been initiated by children's hospitals, but little is known about their number, staff composition, services offered, sources of support, or national distribution.</p>

<p><strong>METHODS: </strong>In the summer of 2012, we surveyed 226 hospitals as identified by the National Association of Children's Hospitals and Related Institutions. The survey instrument gathered data about whether their institution had a PPC program, and for hospitals with programs, it asked for a wide range of information including staffing, patient age range, services provided, and financial support.</p>

<p><strong>RESULTS: </strong>Of the 162 hospitals that provided data (71.7% response rate), 69% reported having a PPC program. The rate of new program creation peaked in 2008, with 12 new programs created that year, and 10 new programs in 2011. Most programs offer only inpatient services, and most only during the work week. The number of consults per year varied substantially across programs, and was positively associated with hospital bed size and number of funded staff members. PPC programs report a high level of dependence on hospital funding.</p>

<p><strong>CONCLUSIONS: </strong>PPC programs are becoming common in children's hospitals throughout the United States yet with marked variation in how these programs are staffed, the level of funding for staff effort to provide PPC, and the number of consultations performed annually. Guidelines for PPC team composition, funding, and consultation standards may be warranted to ensure the highest quality of PPC.</p>

10.1542/peds.2013-1286

Pediatrics

24190689

Pediatric palliative care patients: a prospective multicenter cohort study.

2011

1094-101

06/2011

1098-4275

<p><strong>OBJECTIVE: </strong>To describe demographic and clinical characteristics and outcomes of patients who received hospital-based pediatric palliative care (PPC) consultations.</p>

<p><strong>DESIGN, SETTING, AND PATIENTS: </strong>Prospective observational cohort study of all patients served by 6 hospital-based PPC teams in the United States and Canada from January to March 2008.</p>

<p><strong>RESULTS: </strong>There were 515 new (35.7%) or established (64.3%) patients who received care from the 6 programs during the 3-month enrollment interval. Of these, 54.0% were male, and 69.5% were identified as white and 8.1% as Hispanic. Patient age ranged from less than one month (4.7%) to 19 years or older (15.5%). Of the patients, 60.4% lived with both parents, and 72.6% had siblings. The predominant primary clinical conditions were genetic/congenital (40.8%), neuromuscular (39.2%), cancer (19.8%), respiratory (12.8%), and gastrointestinal (10.7%). Most patients had chronic use of some form of medical technology, with gastrostomy tubes (48.5%) being the most common. At the time of consultation, 47.2% of the patients had cognitive impairment; 30.9% of the cohort experienced pain. Patients were receiving many medications (mean: 9.1). During the 12-month follow-up, 30.3% of the cohort died; the median time from consult to death was 107 days. Patients who died within 30 days of cohort entry were more likely to be infants and have cancer or cardiovascular conditions.</p>

<p><strong>CONCLUSIONS: </strong>PPC teams currently serve a diverse cohort of children and young adults with life-threatening conditions. In contrast to the reported experience of adult-oriented palliative care teams, most PPC patients are alive for more than a year after initiating PPC.</p>

10.1542/peds.2010-3225

Pediatrics

21555495

Research Priorities in Pediatric Palliative Care.

2015

467-70.e3

08/2015

1097-6833

<p><strong>OBJECTIVE: </strong>To synthesize the perspectives of a broad range of pediatric palliative care (PPC) clinicians and parents, to formulate a consensus on prioritization of the PPC research agenda.</p>

<p><strong>STUDY DESIGN: </strong>A 4-round modified Delphi online survey was administered to PPC experts and to parents of children who had received PPC. In round 1, research priorities were generated spontaneously. Rounds 2 and 3 then served as convergence rounds to synthesize priorities. In round 4, participants were asked to rank the research priorities that had reached at least 80% consensus.</p>

<p><strong>RESULTS: </strong>A total of 3093 concepts were spontaneously generated by 170 experts and 72 parents in round 1 (65.8% response rate [RR]). These concepts were thematically organized into 78 priorities and recirculated for round 2 ratings (n = 130; 53.7% RR). Round 3 achieved response stability, with 31 consensus priorities oscillating within 10% of the mode (n = 98; 75.4% RR). Round 4 resulted in consensus recognition of 20 research priorities, which were thematically grouped as decision making, care coordination, symptom management, quality improvement, and education.</p>

<p><strong>CONCLUSIONS: </strong>This modified Delphi survey used professional and parental consensus to identify preeminent PPC research priorities. Attentiveness to these priorities may help direct resources and efforts toward building a formative evidence base. Investigating PPC implementation approaches and outcomes can help improve the quality of care services for children and families.</p>

10.1016/j.jpeds.2015.05.002

J. Pediatr.

26028284

Symptoms and Distress in Children With Advanced Cancer: Prospective Patient-Reported Outcomes From the PediQUEST Study.

2015

1928-35

06/2015

1527-7755

<p><strong>PURPOSE: </strong>Thousands of children are living with advanced cancer; yet patient-reported outcomes (PROs) have rarely been used to describe their experiences. We aimed to describe symptom distress in 104 children age 2 years or older with advanced cancer enrolled onto the Pediatric Quality of Life and Evaluation of Symptoms Technology (PediQUEST) Study (multisite clinical trial evaluating an electronic PRO system).</p>

<p><strong>METHODS: </strong>Symptom data were collected using age- and respondent-adapted versions of the PediQUEST Memorial Symptom Assessment Scale (PQ-MSAS) at most once per week. Clinical and treatment data were obtained from medical records. Individual symptom scores were dichotomized into high/low distress. Determinants of PQ-MSAS scores were explored using linear mixed-effects models.</p>

<p><strong>RESULTS: </strong>During 9 months of follow-up, PQ-MSAS was administered 920 times: 459 times in teens (99% self-report), 249 times in children ages 7 to 12 years (96% child/parent report), and 212 times in those ages 2 to 6 years (parent reports). Common symptoms included pain (48%), fatigue (46%), drowsiness (39%), and irritability (37%); most scores indicated high distress. Among the 73 PQ-MSAS surveys administered in the last 12 weeks of life, pain was highly prevalent (62%; 58% with high distress). Being female, having a brain tumor, experiencing recent disease progression, and receiving moderate- or high-intensity cancer-directed therapy in the prior 10 days were associated with worse PQ-MSAS scores. In the final 12 weeks of life, receiving mild cancer-directed therapy was associated with improved psychological PQ-MSAS scores.</p>

<p><strong>CONCLUSION: </strong>Children with advanced cancer experience high symptom distress. Strategies to promote intensive symptom management are indicated, especially with disease progression or administration of intensive treatments.</p>

10.1200/JCO.2014.59.1222

J. Clin. Oncol.

25918277