Goals of Care Among Parents of Children Receiving Palliative Care.

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.

Parent Priorities in End-of-Life Care for Children With Cancer.

Importance: Robust quality measures to benchmark end-of-life care for children with cancer do not currently exist; 28 candidate patient-centered quality measures were previously developed. Objective: To prioritize quality measures among parents who lost a child to cancer. Design, Setting, and Participants: This survey study was conducted using an electronic, cross-sectional discrete choice experiment (DCE) with maximum difference scaling from January to June 2021 in the US. In each of 21 questions in the DCE, participants were presented with a set of 4 quality measures and were asked to select the most and least important measures within each set. All 28 quality measures were presented an equal number of times in different permutations. In the volunteer sample, 69 eligible bereaved parents enrolled in the study; 61 parents completed the DCE (participation rate, 88.4%). Main Outcomes and Measures: Using choices participants made, a hierarchical bayesian multinomial logistic regression was fit to derive mean importance scores with 95% credible intervals (95% Crs) for each quality measure, representing the overall probability of a quality measure being selected as most important. Importance scores were rescaled proportionally from 0 to 100, with the sum of scores for all quality measures adding up to 100. This enabled interpretation of scores as the relative importance of quality measures. Results: Participants included 61 bereaved parents (median [range] age, 48 [24-74] years; 55 individuals self-identified as women [90.2%]; 1 American Indian or Alaska Native [1.6%], 1 Asian [1.6%], 2 Black or African American [3.3%], 1 Native Hawaiian or Pacific Islander, and 58 White [91.8%]; 58 not Hispanic or Latinx [95.1%]). Highest-priority quality measures by mean importance score included having a child's symptoms treated well (9.25 [95% Cr, 9.06-9.45]), feeling that a child's needs were heard by the health care team (8.39 [95% Cr, 8.05-8.73]), and having a goal-concordant end-of-life experience (7.45 [95% Cr, 6.84-8.05]). Lowest-priority quality measures included avoiding chemotherapy (0.33 [95% Cr, 0.21-0.45]), provision of psychosocial support for parents (1.01 [95% Cr, 0.57-1.45]), and avoiding the intensive care unit (1.09 [95% Cr, 0.74-1.43]). Rank-ordering measures by mean importance revealed that symptom management was 9 times more important to parents than psychosocial support for themselves. Conclusions and Relevance: This study found that bereaved parents prioritized end-of-life quality measures focused on symptom management and goal-concordant care while characterizing quality measures assessing their own psychosocial support and their child's hospital resource use as substantially less important. These findings suggest that future research should explore innovative strategies to measure care attributes that matter most to families of children with advanced cancer.

The 2023-2026 Hospice and Palliative Nurses Association Research Agenda.

The Hospice and Palliative Nursing Association established the triannual research agenda to ( a ) provide focus for researchers to conduct meaningful scientific and quality improvement initiatives and inform evidence-based practice, ( b ) guide organizational funding, and ( c ) illustrate to other stakeholders the importance of nursing research foci. HPNA Research Agendas are developed to give direction for future research to continue advancing expert care in serious illness and ensure equitable delivery of hospice and palliative care.

Measuring Pediatric Palliative Care Quality: Challenges and Opportunities.

Pediatric palliative care (PPC) programs vary widely in structure, staffing, funding, and patient census, resulting in inconsistency in service provision. Improving the quality of palliative care for children living with serious illness and their families requires measuring care quality, ensuring that quality measurement is embedded into day-to-day clinical practice, and aligning quality measurement with healthcare policy priorities. Yet, numerous challenges exist in measuring PPC quality. This paper provides an overview of PPC quality measurement, including challenges, current initiatives, and future opportunities. While important strides toward addressing quality measurement challenges in PPC have been made, including ongoing quality measurement initiatives like the Cambia Metrics Project, the PPC What Matters Most study, and collaborative learning networks, more work remains. Providing high-quality PPC to all children and families will require a multi-pronged approach. In this paper, we suggest several strategies for advancing high-quality PPC, which includes 1) considering how and by whom success is defined, 2) evaluating, adapting, and developing PPC measures, including those that address care disparities within PPC for historically marginalized and excluded communities, 3) improving the infrastructure with which to routinely and prospectively measure, monitor, and report clinical and administrative quality measures, 4) increasing endorsement of PPC quality measures by prominent quality organizations to facilitate accountability and possible reimbursement, and 5) integrating PPC-specific quality measures into the administrative, funding, and policy landscape of pediatric healthcare.

Hope in the context of life-threatening illness and the end of life.

Recent research shows the importance of hope in the context of life-threatening illness and the end of life for patients and their families. While some patients and family members continue to hope for a cure or extending life, others may develop more complex hopes related to quality of life and making the most of the time left. Clinicians often worry about taking hope away with bad news about the patient's prognosis, but patients and family members often appreciate honesty without losing hope. Clinicians should recognize that hopes in the context of serious, progressive illness may be complex, contradictory, culturally based, and evolve over time. Recent interventions have been developed to support hope for these patients, caregivers, and parents.

Home-Based Care for Children with Serious Illness: Ecological Framework and Research Implications.

Care for U.S. children living with serious illness and their families at home is a complex and patchwork system. Improving home-based care for children and families requires a comprehensive, multilevel approach that accounts for and examines relationships across home environments, communities, and social contexts in which children and families live and receive care. We propose a multilevel conceptual framework, guided by Bronfenbrenner's ecological model, that conceptualizes the complex system of home-based care into five levels. Levels 1 and 2 contain patient and family characteristics. Level 3 contains factors that influence family health, well-being, and experience with care in the home. Level 4 includes the community, including community groups, schools, and providers. Level 5 includes the broader regional system of care that impacts the care of children and families across communities. Finally, care coordination and care disparities transcend levels, impacting care at each level. A multilevel ecological framework of home-based care for children with serious illness and families can be used in future multilevel research to describe and test hypotheses about aspects of this system of care, as well as to inform interventions across levels to improve patient and family outcomes.

The Design of a Data Management System for a Multicenter Palliative Care Cohort Study.

CONTEXT: Prospective cohort studies of individuals with serious illness and their family members, such as children receiving palliative care and their parents, pose challenges regarding data management. OBJECTIVE: To describe the design and lessons learned regarding the data management system for the Pediatric Palliative Care Research Network's Shared Data and Research (SHARE) project, a multicenter prospective cohort study of children receiving pediatric palliative care (PPC) and their parents, and to describe important attributes of this system, with specific considerations for the design of future studies. METHODS: The SHARE study consists of 643 PPC patients and up to two of their parents who enrolled from April 2017 to December 2020 at seven children's hospitals across the United States. Data regarding demographics, patient symptoms, goals of care, and other characteristics were collected directly from parents or patients at 6 timepoints over a 24-month follow-up period and stored electronically in a centralized location. Using medical record numbers, primary collected data was linked to administrative hospitalization data containing diagnostic and procedure codes and other data elements. Important attributes of the data infrastructure include linkage of primary and administrative data; centralized availability of multilingual questionnaires; electronic data collection and storage system; time-stamping of instrument completion; and a separate but connected study administrative database used to track enrollment. CONCLUSIONS: Investigators planning future multicenter prospective cohort studies can consider attributes of the data infrastructure we describe when designing their data management system.

Pediatric Palliative Care Parents' Distress, Financial Difficulty, and Child Symptoms.

CONTEXT: Parents of patients with a serious illness experience psychological distress, which impacts parents' wellbeing and, potentially, their ability to care for their children. Parent psychological distress may be influenced by children's symptom burden and by families' financial difficulty. OBJECTIVES: This study examined the associations among parent psychological distress, parent-reported patient symptoms, and financial difficulty, seeking to determine the relative association of financial difficulty and of patient symptoms to parent psychological distress. METHODS: Cross-sectional study of baseline data for 601 parents of 532 pediatric palliative care patients enrolled in a prospective cohort study conducted at seven US children's hospitals. Data included self-reported parent psychological distress and parent report of child's symptoms and family financial difficulty. We used ordinary least squares multiple regressions to examine the association between psychological distress and symptom score, between psychological distress and financial difficulty, and whether the degree of financial difficulty modified the relationship between psychological distress and symptom score. RESULTS: The majority of parents were moderately to severely distressed (69%) or severely distressed (17%) and experienced some degree of financial difficulty (65%). While children's symptom scores and family financial difficulty together explained more of the variance in parental psychological distress than either variable alone, parental distress was associated more strongly, and to a larger degree, with financial difficulty than with symptom scores alone. CONCLUSIONS: Parent psychological distress was associated with parent-reported patient symptoms and financial difficulty. Future work should examine these relationships longitudinally, and whether interventions to improve symptom management and ameliorate financial difficulties improve parental outcomes.

Association Between Children With Life-Threatening Conditions and Their Parents' and Siblings' Mental and Physical Health.

Importance: Despite concerns regarding the potential deleterious physical and mental health outcomes among family members of a child with a life-threatening condition (LTC), few studies have examined empirical measures of health outcomes among these family members. Objectives: To examine whether mothers, fathers, sisters, and brothers of children with 1 of 4 types of pediatric LTCs have higher rates of health care encounters, diagnoses, and prescriptions compared with families of children without these conditions. Design, Setting, and Participants: This retrospective cohort study included US families with commercial insurance coverage from a single carrier. Children who had 1 of 4 LTCs (substantial prematurity, critical congenital heart disease, cancer, or a condition resulting in severe neurologic impairment) were identified by a diagnosis in their insurance claim data between July 1, 2015, and June 30, 2016. Each case child and their family was matched with up to 4 control children and their families based on the age of the case and control children. Data were analyzed between August 2020 and March 2021. Exposures: Having a child or sibling with substantial prematurity, critical congenital heart disease, cancer, or a condition resulting in severe and progressive neurologic impairment. Main Outcomes: Rates of occurrence of health care encounters, physical and mental health diagnoses, and physical and mental health medication prescriptions, identified from insurance claims data, were compared between case and control families using a multivariable negative binomial regression model. The statistical analysis adjusted for observed differences between case and control families and accounted for clustering at the family level. Results: The study included 25 528 children (6909 case children [27.1%] and 18 619 control children [72.9%]; median age, 6.0 years [IQR, 1-13 years]; 13 294 [52.1%] male), 43 357 parents (11 586 case parents [26.7%] and 31 771 control parents [73.3%]; mean [SD] age, 40.4 [8.1] years; 22 318 [51.5%] female), and 25 706 siblings (7664 case siblings [29.8%] and 18 042 control siblings [70.2%]; mean [SD] age, 12.1 [6.5] years; 13 114 [51.0%] male). Overall, case mothers had higher rates of the composite outcome of health care encounters, diagnoses, and prescriptions compared with control mothers (incident rate ratio [IRR], 1.61; 95% CI, 1.54-1.68), as did case fathers compared with control fathers (IRR, 1.55; 95% CI, 1.46-1.64). Sisters of children with LTCs had higher rates of the composite outcome compared with sisters of children without LTCs (IRR, 1.68; 95% CI, 1.55-1.82), as did brothers of children with LTCs compared with brothers of children without LTCs (IRR, 1.70; 95% CI, 1.56-1.85). Conclusions and Relevance: In this cohort study, mothers, fathers, sisters, and brothers who had a child or sibling with 1 of 4 types of LTCs had higher rates of health care encounters, diagnoses, and medication prescriptions compared with families who did not have a child with that condition. The findings suggest that family members of children with LTCs may experience poorer mental and physical health outcomes. Interventions for parents and siblings of children with LTCs that aim to safeguard their mental and physical well-being appear to be warranted.

Developing a family-reported measure of experiences with home-based pediatric palliative and hospice care: a multi-method, multi-stakeholder approach.

BACKGROUND: Many children with serious illnesses are receiving palliative and end-of-life care from pediatric palliative and hospice care teams at home (PPHC@Home). Despite the growth in PPHC@Home, no standardized measures exist to evaluate whether PPHC@Home provided in the U.S. meets the needs and priorities of children and their families. METHODS: We developed and conducted a preliminary evaluation of a family-reported measure of PPHC@Home experiences using a multi-method, multi-stakeholder approach. Our instrument development process consisted of four phases. Item identification and development (Phase 1) involved a comprehensive literature search of existing instruments, guidelines, standards of practice, and PPHC@Home outcome studies, as well as guidance from a PPHC stakeholder panel. Phase 2 involved the initial item prioiritization and reduction using a discrete choice experiment (DCE) with PPHC professionals and parent advocates. Phase 3 involved a second DCE with bereaved parents and parents currently receiving care for their child to further prioritize and winnow the items to a set of the most highly-valued items. Finally, we conducted cognitive interviews with parents to provide information about the content validity and clarity of the newly-developed instrument (Phase 4). RESULTS: Items were compiled predominantly from three existing instruments. Phase 2 participants included 34 PPHC providers, researchers, and parent advocates; Phase 3 participants included 47 parents; and Phase 4 participants included 11 parents. At the completion of Phase 4, the Experiences of Palliative and Hospice Care for Children and Caregivers at Home (EXPERIENCE@Home) Measure contains 22 of the most highly-valued items for evaluating PPHC@Home. These items include "The care team treats my child's physical symptoms so that my child has as good a quality of life as possible", "I have regular access to on-call services from our care team", and "The nurses have the knowledge, skills, and experience to support my child's palliative or hospice care at home." CONCLUSIONS: The EXPERIENCE@Home Measure is the first known to specifically measure family-reported experiences with PPHC@Home in the U.S. Future work will include formal psychometric evaluation with a larger sample of parents, as well as evaluation of the clinical utility of the instrument with PPHC@Home teams.

What Do Parents Value Regarding Pediatric Palliative and Hospice Care in the Home Setting?

CONTEXT: Children with life-shortening serious illnesses and medically-complex care needs are often cared for by their families at home. Little, however, is known about what aspects of pediatric palliative and hospice care in the home setting (PPHC@Home) families value the most. OBJECTIVES: To explore how parents rate and prioritize domains of PPHC@Home as the first phase of a larger study that developed a parent-reported measure of experiences with PPHC@Home. METHODS: Twenty domains of high-value PPHC@Home, derived from the National Consensus Project's Guidelines for Quality Palliative Care, the literature, and a stakeholder panel, were evaluated. Using a discrete choice experiment, parents provided their ratings of the most and least valued PPHC@Home domains. We also explored potential differences in how subgroups of parents rated the domains. RESULTS: Forty-seven parents participated. Overall, highest-rated domains included Physical aspects of care: Symptom management, Psychological/emotional aspects of care for the child, and Care coordination. Lowest-rated domains included Spiritual and religious aspects of care and Cultural aspects of care. In exploratory analyses, parents who had other children rated the Psychological/emotional aspects of care for the sibling(s) domain significantly higher than parents who did not have other children (P = 0.02). Furthermore, bereaved parents rated the Caregiversupportat the end of life domain significantly higher than parents who were currently caring for their child (P = 0.04). No other significant differences in domain ratings were observed. CONCLUSION: Knowing what parents value most about PPHC@Home provides the foundation for further exploration and conversation about priority areas for resource allocation and care improvement efforts.

The Association of Perceived Social Support with Anxiety over Time in Parents of Children with Serious Illnesses.

Background: Parenting a child with a serious life-threatening illness (SLTI) may impact parents' mental health. The protective association of social support with anxiety over time following an acute medical event has not been empirically tested in a sample of parents of children with oncologic and nononcologic serious illnesses. Objective: To test the potential association of perceived social support with anxiety in parents of children with SLTIs over time. Design: Prospective cohort study. Setting/Subjects: Two hundred parents of 158 children in the Decision Making in Serious Pediatric Illness study, conducted at the Children's Hospital of Philadelphia. Measurements: Parental anxiety and perceived social support were assessed using the Hospital Anxiety and Depression Scale (HADS) and the Social Provisions Scale (SPS). We performed bivariate linear regressions to test cross-sectional and longitudinal associations between the SPS and anxiety scores at baseline, 12 months, and 24 months. Results: The average SPS total and subscale scores decreased significantly from baseline to 12 months, and increased from 12 to 24 months. The average HADS-Anxiety scores decreased significantly from baseline to 12 months, and remained stable at 24 months. Cross-sectionally, total SPS scores were negatively associated with anxiety scores at each time point. Longitudinally, SPS scores were associated with anxiety scores, although this association weakened in adjusted modeling. Conclusions: Over a two-year period, higher levels of perceived social support were associated with lower levels of anxiety in parents of seriously ill children. Clinicians and researchers should work to optimize social support for families to improve parental mental health outcomes.

Factors Associated With the Use of U.S. Community-Based Palliative Care for Children With Life-Limiting or Life-Threatening Illnesses and Their Families: An Integrative Review.

CONTEXT: As children with life-limiting illnesses (LLIs) and life-threatening illnesses (LTIs) live longer, challenges to meeting their complex health care needs arise in homes and communities, as well as in hospitals. Integrated knowledge regarding community-based pediatric palliative care (CBPPC) is needed to strategically plan for a seamless continuum of care for children and their families. OBJECTIVES: The purpose of this integrative review article is to explore factors that are associated with the use of CBPPC for U.S. children with LLIs and LTIs and their families. METHODS: A literature search of PubMed, CINAHL, Scopus, Google Scholar, and an ancestry search was performed to identify empirical studies and program evaluations published between 2000 and 2016. The methodological protocol included an evaluation of empirical quality and explicit data collection of synthesis procedures. RESULTS: Forty peer-reviewed quantitative and qualitative methodological interdisciplinary articles were included in the final sample. Patient characteristics such as older age and a solid tumor cancer diagnosis and interpersonal factors such as family support were associated with higher CBPPC use. Organizational features were the most frequently discussed factors that increased CBPPC, including the importance of interprofessional hospice services and interorganizational care coordination for supporting the child and family at home. Finally, geography, concurrent care and hospice eligibility regulations, and funding and reimbursement mechanisms were associated with CBPPC use on a community and systemic level. CONCLUSION: Multilevel factors are associated with increased CBPPC use for children with LLIs or LTIs and their families in the U.S.

A Hospice Rotation for Military Medical Residents: A Mixed Methods, Multi-Perspective Program Evaluation.

BACKGROUND: An estimated 6,000 to 18,000 additional hospice and palliative medicine (HPM) physicians are needed in the United States. A source could be the military graduate medical education system where 15% of U.S. medical residents are trained. A community-based hospice and palliative care organization created a one-week rotation for military residents including participation in interdisciplinary group visits at patients' homes, facilities, and an inpatient hospice unit. OBJECTIVE: Our goal was to evaluate the effectiveness of a one-week community HPM rotation for military medical residents. METHODS: A mixed-methods, multi-stakeholder perspective program evaluation model was used for program years 2011 to 2013. Data were managed and analyzed using Microsoft Excel and Atlas.ti. Participants in the rotation were residents training at two local military hospitals. Program evaluation data were collected from residents, military program liaisons, and hospice clinical preceptors. Quantitative data included pre- and post-tests based on Accreditation Council for Graduate Medical Education competencies completed by residents. Qualitative data included resident essays and semi-structured interviews with hospice preceptors and military program liaisons. RESULTS: Quantitative and qualitative data suggested that the rotation increased military residents' knowledge, attitudes, and comfort level with HPM. Quantitative analysis of test scores indicated improvements from pre- to post-tests in each of five areas of learning. Qualitative data indicated the rotation created a greater appreciation for the overall importance of HPM and increased understanding of eligibility and methods for pain and symptom management. CONCLUSIONS: A one-week community hospice rotation for medical military residents impacts participant's knowledge of and attitudes toward HPM.

Implementation challenges in end-of-life research with adults with intellectual and developmental disabilities.

Although the 4 million+people in the U.S. with an intellectual or developmental disability (I/DD) experience the same life expectancy as those in the general population, end-of-life research including these individuals is lacking and can be difficult to implement. As will be described in this paper, it is possible to overcome barriers to successfully include people with I/DD in end-of-life research. In this paper, the implementation challenges, feasibility, and implications for successful end-of-life research with individuals with I/DD using focus groups are described. Individuals with I/DD were able to discuss their experiences and views about end-of-life care. However, while people with I/DD made valuable contributions to the focus groups, there were several modifications needed in order to execute this study. In order to gain a complete picture of end-of-life care for people with I/DD, it is imperative to include them in research to the best of their ability. By anticipating issues related to recruitment, the consent process, setting, and support needs of participants, focus groups can be successfully implemented.

Nebulized medications for the treatment of dyspnea: a literature review.

BACKGROUND: Dyspnea significantly impacts quality of life and is one of the most common symptoms in advanced illness. Systemically-administered opioids and benzodiazepines have been the most studied and utilized pharmacologic treatments for refractory dyspnea. Less attention has been given to the use of these medications and others when nebulized. This article presents a review of the literature on the use of nebulized medications for the treatment of dyspnea related to cancer, chronic obstructive pulmonary disease, cystic fibrosis, interstitial lung disease, or experimentally-induced dyspnea. METHODS: A systematic review of peer-reviewed literature was conducted using Medline/PubMed, CINAHL, Cochrane, and Google Scholar. RESULTS: Thirty-nine publications were included in this review, including 17 high-quality clinical research studies, as defined by the GRADE system. The evidence for nebulized morphine remains mixed, whereas a potential benefit was suggested for nebulized furosemide, hydromorphone, and fentanyl. No conclusions could be drawn as to which disease population derived greatest benefit from nebulized medications, or whether jet or ultrasonic nebulizers were more effective for the delivery of these medications. CONCLUSIONS: More research is needed to assess the characteristics of specific diseases and the combination of different nebulizers and medications that may yield the greatest benefit, and to assess the safety and efficacy of the chronic use of nebulized opioids and furosemide. Until larger, longer-term studies are completed, the use of nebulized medications to treat dyspnea should be assessed on a case-by-case basis and may be considered if the hoped-for benefits outweigh potential harm.

Experiences of african american parents following perinatal or pediatric death: a literature review.

A child's death is one of life's most difficult experiences. Little is known about the unique factors that influence the grief experience for bereaved African American parents. Through an integrative review of 10 publications, the authors describe the grief responses, outcomes, and implications for African American parents who experience the death of a child. Four themes emerged: (a) emotional response to loss; (b) factors that added to the burden of loss; (c) coping strategies; and (d) health consequences of grief. Healthcare providers, administrators, and policymakers should be sensitive to the unique needs of African American parents following a child's death.