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

Open AccessPublished:July 30, 2020DOI:https://doi.org/10.1016/j.jpainsymman.2020.07.024

      Abstract

      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 ([email protected]) families value the most.

      Objectives

      To explore how parents rate and prioritize domains of [email protected] as the first phase of a larger study that developed a parent-reported measure of experiences with [email protected]

      Methods

      Twenty domains of high-value [email protected], 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 [email protected] 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 Caregiver support at 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 [email protected] provides the foundation for further exploration and conversation about priority areas for resource allocation and care improvement efforts.

      Key Words

      Key Message

      This article describes parents' priorities for pediatric palliative and hospice care in the home setting ([email protected]). Parents provided their ratings and rankings of [email protected] domains in a discrete choice experiment. Understanding parents' priorities for [email protected] facilitates further exploration and conversation about priority areas for resource allocation and care improvement efforts.

      Introduction

      Children with life-shortening serious illnesses and medically-complex care needs are increasingly cared for by their families at home, particularly toward the end of life.
      • Feudtner C.
      • Feinstein J.A.
      • Satchell M.
      • Zhao H.
      • Kang T.I.
      Shifting place of death among children with complex chronic conditions in the United States, 1989-2003.
      • Feudtner C.
      • Kang T.I.
      • Hexem K.R.
      • et al.
      Pediatric palliative care patients: a prospective multicenter cohort study.
      • Feudtner C.
      • Silveira M.J.
      • Christakis D.A.
      Where do children with complex chronic conditions die? Patterns in Washington State, 1980-1998.
      • Nageswaran S.
      • Radulovic A.
      • Anania A.
      Transitions to and from the acute inpatient care setting for children with life-threatening illness.
      These children and families are supported in the home setting by palliative and hospice care programs that vary in structure, staffing, funding, and patient census.
      • Feudtner C.
      • Womer J.
      • Augustin R.
      • et al.
      Pediatric palliative care programs in children's hospitals: a cross-sectional national survey.
      • Friebert S.
      • Williams C.
      NHPCO'S facts and figures: Pediatric palliative and hospice care in America.
      • Kaye E.
      • Rubenstein J.
      • Levine D.
      • et al.
      Pediatric palliative care in the community.
      • Boyden J.Y.
      • Curley M.A.Q.
      • Deatrick J.A.
      • Ersek M.
      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.
      • Lindley L.
      • Mark B.
      • Lee S.Y.D.
      Providing hospice care to children and young adults: a descriptive study of end-of-life organizations.
      As a result, many children and their families in the U.S. have variable and, at times, inadequate access to high-quality pediatric palliative and hospice care in the home setting ([email protected]).
      • Kaye E.
      • Rubenstein J.
      • Levine D.
      • et al.
      Pediatric palliative care in the community.
      ,
      • Carroll J.M.
      • Torkildson C.
      • Winsness J.S.
      Issues related to providing quality pediatric palliative care in the community.
      ,
      • Lindley L.C.
      • Kieim-Malpass J.
      Quality of paediatric hospice care for children with and without multiple complex chronic conditions.
      Previous research with primarily inpatient-based samples of parents and providers has identified several factors that improve parents' perceptions of the quality of care, including effective pain and symptom management,
      • Donnelly J.P.
      • Huff S.M.
      • Lindsey M.L.
      • McMahon K.A.
      • Schumacher J.D.
      The needs of children with life-limiting conditions: a healthcare-provider-based model.
      • Contro N.
      • Larson J.
      • Scofield S.
      • Sourkes B.
      • Cohen H.
      Family perspectives on the quality of pediatric palliative care.
      • Mack J.W.
      • Hilden J.M.
      • Watterson J.
      • et al.
      Parent and physician perspectives on quality of care at the end of life in children with cancer.
      • Meyer E.C.
      • Ritholz M.D.
      • Burns J.P.
      • Truog R.D.
      Improving the quality of end-of-life care in the pediatric intensive care unit: parents' priorities and recommendations.
      • Weidner N.J.
      • Cameron M.
      • Lee R.C.
      • et al.
      End-of-life care for the dying child: what matters most to parents.
      child-centered and family-centered care and decision making,
      • Donnelly J.P.
      • Huff S.M.
      • Lindsey M.L.
      • McMahon K.A.
      • Schumacher J.D.
      The needs of children with life-limiting conditions: a healthcare-provider-based model.
      ,
      • Weidner N.J.
      • Cameron M.
      • Lee R.C.
      • et al.
      End-of-life care for the dying child: what matters most to parents.
      inclusion of siblings in care processes,
      • Contro N.
      • Larson J.
      • Scofield S.
      • Sourkes B.
      • Cohen H.
      Family perspectives on the quality of pediatric palliative care.
      ,
      • Andresen E.M.
      • Seecharan G.A.
      • Toce S.S.
      Provider perceptions of child deaths.
      consistent and high-quality communication between family and providers,
      • Contro N.
      • Larson J.
      • Scofield S.
      • Sourkes B.
      • Cohen H.
      Family perspectives on the quality of pediatric palliative care.
      • Mack J.W.
      • Hilden J.M.
      • Watterson J.
      • et al.
      Parent and physician perspectives on quality of care at the end of life in children with cancer.
      • Meyer E.C.
      • Ritholz M.D.
      • Burns J.P.
      • Truog R.D.
      Improving the quality of end-of-life care in the pediatric intensive care unit: parents' priorities and recommendations.
      • Weidner N.J.
      • Cameron M.
      • Lee R.C.
      • et al.
      End-of-life care for the dying child: what matters most to parents.
      • Andresen E.M.
      • Seecharan G.A.
      • Toce S.S.
      Provider perceptions of child deaths.
      family education and preparation for the end of life,
      • Mack J.W.
      • Hilden J.M.
      • Watterson J.
      • et al.
      Parent and physician perspectives on quality of care at the end of life in children with cancer.
      psychosocial and spiritual care,
      • Donnelly J.P.
      • Huff S.M.
      • Lindsey M.L.
      • McMahon K.A.
      • Schumacher J.D.
      The needs of children with life-limiting conditions: a healthcare-provider-based model.
      ,
      • Meyer E.C.
      • Ritholz M.D.
      • Burns J.P.
      • Truog R.D.
      Improving the quality of end-of-life care in the pediatric intensive care unit: parents' priorities and recommendations.
      • Weidner N.J.
      • Cameron M.
      • Lee R.C.
      • et al.
      End-of-life care for the dying child: what matters most to parents.
      • Andresen E.M.
      • Seecharan G.A.
      • Toce S.S.
      Provider perceptions of child deaths.
      a comfortable death,
      • Meyer E.C.
      • Ritholz M.D.
      • Burns J.P.
      • Truog R.D.
      Improving the quality of end-of-life care in the pediatric intensive care unit: parents' priorities and recommendations.
      ,
      • Weidner N.J.
      • Cameron M.
      • Lee R.C.
      • et al.
      End-of-life care for the dying child: what matters most to parents.
      care coordination and management,
      • Donnelly J.P.
      • Huff S.M.
      • Lindsey M.L.
      • McMahon K.A.
      • Schumacher J.D.
      The needs of children with life-limiting conditions: a healthcare-provider-based model.
      ,
      • Weidner N.J.
      • Cameron M.
      • Lee R.C.
      • et al.
      End-of-life care for the dying child: what matters most to parents.
      and bereavement care.
      • Contro N.
      • Larson J.
      • Scofield S.
      • Sourkes B.
      • Cohen H.
      Family perspectives on the quality of pediatric palliative care.
      Evidence suggests that these domains of care are as important in the home setting.
      • Kaye E.
      • Rubenstein J.
      • Levine D.
      • et al.
      Pediatric palliative care in the community.
      ,
      • Carroll J.M.
      • Torkildson C.
      • Winsness J.S.
      Issues related to providing quality pediatric palliative care in the community.
      ,
      • Contro N.
      • Larson J.
      • Scofield S.
      • Sourkes B.
      • Cohen H.
      Family perspectives on the quality of pediatric palliative care.
      ,
      • Malcolm C.
      • Knighting K.
      • Forbat L.
      • Kearney N.
      Prioritisation of future research topics for children's hospice care by its key stakeholders: a Delphi study.
      • Thienprayoon R.
      • Grossoehme D.
      • Humphrey L.
      • et al.
      "There's just no way to help, and they did." Parents name compassionate care as a new domain of quality in pediatric home-based hospice and palliative care.
      • Thienprayoon R.
      • Mark M.S.J.
      • Grossoehme D.
      Provider-prioritized domains of quality in pediatric home-based hospice and palliative care: a study of the Ohio Pediatric Palliative Care and End-of-Life Network.
      • Widger K.A.
      • Wilkins K.
      What are the key components of quality perinatal and pediatric end-of-life care? A literature review.
      National Consensus Project for Quality Palliative Care
      Clinical practice guidelines for quality palliative care.
      Little is known, however, about which of these care domains parents value the most in supporting their child in their home, and how these domains compare in importance to one another. Previous studies prioritized areas for high-quality [email protected] clinical care and research using Delphi methods with [email protected] stakeholders, including providers and parents.
      • Malcolm C.
      • Knighting K.
      • Forbat L.
      • Kearney N.
      Prioritisation of future research topics for children's hospice care by its key stakeholders: a Delphi study.
      ,
      • Thienprayoon R.
      • Mark M.S.J.
      • Grossoehme D.
      Provider-prioritized domains of quality in pediatric home-based hospice and palliative care: a study of the Ohio Pediatric Palliative Care and End-of-Life Network.
      Among top-rated priorities were services, techniques, and resources for pain and symptom relief and psychological support for children and young adults.
      • Malcolm C.
      • Knighting K.
      • Forbat L.
      • Kearney N.
      Prioritisation of future research topics for children's hospice care by its key stakeholders: a Delphi study.
      Given limitations to Delphi-rating methodologies, including the limited ability to differentiate between similar rating scores
      • Malcolm C.
      • Knighting K.
      • Forbat L.
      • Kearney N.
      Prioritisation of future research topics for children's hospice care by its key stakeholders: a Delphi study.
      and concerns about stability of rating scores,
      • Trevelyan E.G.
      • Robinson P.N.
      Delphi methodology in health research: how to do it?.
      we do not know which of these care domains are most important in supporting children at home. Although high-quality [email protected] encompasses the spectrum of care domains,
      • Thienprayoon R.
      • Mark M.S.J.
      • Grossoehme D.
      Provider-prioritized domains of quality in pediatric home-based hospice and palliative care: a study of the Ohio Pediatric Palliative Care and End-of-Life Network.
      ,
      National Consensus Project for Quality Palliative Care
      Clinical practice guidelines for quality palliative care.
      health care resources, particularly in the home and community setting,
      • Kaye E.
      • Rubenstein J.
      • Levine D.
      • et al.
      Pediatric palliative care in the community.
      ,
      • Boyden J.Y.
      • Curley M.A.Q.
      • Deatrick J.A.
      • Ersek M.
      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.
      are finite. Knowing, therefore, what parents value most could help guide allocation of these scarce resources, as well as future clinical and research efforts to measure and improve the quality of [email protected]
      The goal of this analysis was to measure parents' priorities for [email protected] using a quantitative choice-based approach. This analysis represents the first phase of a larger study to develop a parent-reported measure of experiences with [email protected]

      Boyden J, Ersek M, Deatrick J, et al. Developing a parent-reported measure of experiences with home-based pediatric palliative and hospice care: a multi-method, multi-stakeholder approach. In development. BMC Palliat Care.

      Methods

      Study Design

      We conducted a cross-sectional assessment of parents' priorities regarding the importance of 20 different [email protected] domains of care. This assessment represents the first phase of a larger project to develop a parent-reported measure of experiences with [email protected]

      Boyden J, Ersek M, Deatrick J, et al. Developing a parent-reported measure of experiences with home-based pediatric palliative and hospice care: a multi-method, multi-stakeholder approach. In development. BMC Palliat Care.

      The Children's Hospital of Philadelphia's Institutional Review Board approved the conduct of this study.

      Conceptual Framework for the Domains

      The framework for the domains used in this study came from the National Consensus Project's (NCP) Clinical Guidelines for Quality Palliative Care (fourth edition),
      National Consensus Project for Quality Palliative Care
      Clinical practice guidelines for quality palliative care.
      which were further adapted using pediatric palliative care (PPC)-specific guidelines and the literature
      • Thienprayoon R.
      • Mark M.S.J.
      • Grossoehme D.
      Provider-prioritized domains of quality in pediatric home-based hospice and palliative care: a study of the Ohio Pediatric Palliative Care and End-of-Life Network.
      ,
      • Feudtner C.
      • Friebert S.
      • Jewell J.
      Pediatric palliative care and hospice care commitments, guidelines, and recommendations.
      • Field M.J.
      • Behrman R.E.
      When children die: Improving palliative and end-of-life care for children and their families.
      National Hospice and Palliative Care Organization (NHPCO)
      Standards of practice for pediatric palliative care.
      • Widger K.
      • Brennenstuhl S.
      • Duc J.
      • Tourangeau A.
      • Rapoport A.
      Factor structure of the Quality of Children's Palliative Care Instrument (QCPCI) when completed by parents of children with cancer.
      and informed by a panel of stakeholders (PPC providers and parent advocates). The adapted framework includes 20 [email protected] domains and subdomains (Table 1).
      Table 1Domains of High-Quality PPHC in the Home
      DomainsDescription
      Communication between family and care teamCare team communicates with the child and family to make sure that the care provided meets the child's and family's values, preferences, goals, and needs
      Relationship between family and care teamRelationship between care team and family is built on trust, respect, and advocacy for the child's and family's needs
      Knowledge and skills of care team providersCare team members have the necessary education and training to provide high-quality palliative care for children and families
      Access to careCare team provides access to palliative and hospice care to the child and family 24 hours a day, seven days a week
      Physical care: CommunicationCare team provides information about treatments for child's pain and other physical symptoms (e.g., nausea, fatigue, constipation)
      Physical care: Symptom managementCare team assesses and manages pain and other physical symptoms and side effects based on the best available medical evidence
      Psychological and emotional aspects of care (child, parents, siblings, and extended social network)
      Separate subdomain for each group.
      Care team assesses and manages psychological and emotional issues (such as anxiety, distress, coping, grief) of the child, family, and family's community based on the best available medical evidence
      Practical aspects of careCare team provides the family with assistance and resources for dealing with financial and insurance-related issues
      Social aspects of care (child, parents)
      Separate subdomain for each group.
      Care team helps with social issues to meet child-family needs, promote child-family goals, and maximize child-family strengths and well-being (examples include helping family maintain and strengthen their support network; help family develop strategies to balance caregiving, work, and family needs)
      Spiritual and religious aspects of careCare team assists with religious and spiritual rituals or practices as desired by the child and family
      Cultural aspects of careCare team respects the child's and family's cultural beliefs and language preferences
      Communication at the end of lifeCare team works with the child and family to develop and implement a care plan to address actual or potential symptoms at the end of life
      Caregiver support at the end of lifeCare team meets the emotional, spiritual, social, and cultural needs of families at the end of life (e.g., preparing parents for the end of life)
      Ethical and legal aspects of careChild's and family's goals, preferences, and choices are respected within the limits of state and federal law, current standards of medical care, and professional standards of practice

      These goals/preferences/choices are documented and shared with all professionals involved in the child's care
      Coordination of careCare team works to make sure that when there are transfers between health care settings and providers, that there is timely and thorough communication of the child's/family's goals, preferences, values, and clinical information to ensure continuity of care and seamless follow-up (e.g., getting needed services, arranging for medical equipment)
      Continuity of careCare team works to make sure that the delivery of palliative and hospice care is seamless across care settings and providers (e.g., the same providers work with family)
      PPHC = pediatric palliative and hospice care.
      Note: These domains are based on the National Consensus Project's Clinical Guidelines for Quality Palliative Care (fourth edition),
      National Consensus Project for Quality Palliative Care
      Clinical practice guidelines for quality palliative care.
      which were further adapted using pediatric palliative care-specific guidelines and the literature
      • Thienprayoon R.
      • Mark M.S.J.
      • Grossoehme D.
      Provider-prioritized domains of quality in pediatric home-based hospice and palliative care: a study of the Ohio Pediatric Palliative Care and End-of-Life Network.
      ,
      • Feudtner C.
      • Friebert S.
      • Jewell J.
      Pediatric palliative care and hospice care commitments, guidelines, and recommendations.
      • Field M.J.
      • Behrman R.E.
      When children die: Improving palliative and end-of-life care for children and their families.
      National Hospice and Palliative Care Organization (NHPCO)
      Standards of practice for pediatric palliative care.
      • Widger K.
      • Brennenstuhl S.
      • Duc J.
      • Tourangeau A.
      • Rapoport A.
      Factor structure of the Quality of Children's Palliative Care Instrument (QCPCI) when completed by parents of children with cancer.
      and informed by a panel of pediatric palliative care stakeholders (providers and parent advocates).
      a Separate subdomain for each group.

      Sample

      Parents were eligible if they were English speaking, older than 18 years, and had a child with a serious illness who was younger than 25 years at the time care was received. We included parents whose children were currently receiving [email protected], as well as bereaved parents whose children had previously received [email protected] Parents were recruited from the Children's Hospital of Philadelphia's (CHOP) Pediatric Advanced Care Team and from the Courageous Parents Network (CPN), which is a virtual community of parents, families, and providers that supports parents with information, skills, tools, and other resources during their child's illness journey.
      Courageous Parents Network.
      For participants recruited from CHOP, the principal investigator (PI; first author [J. Y. B.]) worked closely with Pediatric Advanced Care Team's nurse coordinator and social worker to screen for eligible participants. The PI then contacted participants by phone. Interested parents provided their electronic informed consent and completed the Web-based survey concurrently (in person or by phone) with the PI or independently via a Web link. For participants recruited from CPN, the PI worked closely with staff of CPNs to post recruitment materials to the CPN listserv and social media page. Interested participants contacted the PI via phone or electronic mail and were screened for eligibility. Eligible participants provided their electronic informed consent and completed the survey via a Web link either concurrently with the PI or independently. Participants were compensated with a $30 gift card for their time and effort.

      Discrete Choice Experiment

      Participants engaged in a discrete choice experiment (DCE), which is a quantitative choice-based approach to evaluating individuals' stated preferences regarding choices in health care
      • Gomes B.
      • de Brito M.
      • Sarmento V.P.
      • et al.
      Valuing attributes of home palliative care with service users: a pilot discrete choice experiment.
      • Huynh E.
      • Coast J.
      • Rose J.
      • Kinghorn P.
      • Flynn T.
      Values for the ICECAP-Supportive Care Measure (ICECAP-SCM) for use in economic evaluation at end of life.
      • Mangham L.J.
      • Hanson K.
      • McPake B.
      How to do (or not to do) … designing a discrete choice experiment for application in a low-income country.
      • Ryan M.
      • Gerard K.
      • Amaya-Amaya M.
      Using discrete choice experiments to value health and health care.
      • Feudtner C.
      • Walter J.K.
      • Faerber J.A.
      • et al.
      Good-parent beliefs of parents of seriously ill children.
      and other settings.
      Sawtooth Software
      Maximum difference scaling: improved measures of importance and preference for segmentation.
      ,
      • Louviere J.J.
      • Flynn T.N.
      • Carson R.T.
      Discrete choice experiments are not conjoint analysis.
      We used a DCE with maximum difference (MaxDiff) scaling to obtain a quantitative estimate of the relative importance of each domain (i.e., domain importance scores), as rated by parents, and to rank the [email protected] domains by order of the parent-rated importance scores.
      Participants first reviewed a list of all 20 [email protected] domains and associated definitions (as displayed in Table 1); these definitions were available to participants throughout the DCE. Participants were then presented with sets displaying four of the 20 domains. Within each set, participants were instructed to choose, from the four listed domains, the one that they felt was the most important in supporting their child in the home and the domain they felt was least important. This process was repeated for a total of 15 sets, each set being a different combination of the 20 domains, with the same instructions to choose the most and least important among the four domains listed in that set. Across all the sets for each respondent, the design of the DCEs ensured that each domain was shown exactly three times and in a balanced set of combinations and permutations with the other domains. Completing all 15 sets took participants approximately 8–10 minutes.

      Data Analysis

      Based on the choices that participants made across the 15 DCE sets, domain importance scores were calculated using a hierarchical Bayesian application of multinomial logistic regression, which estimated the average (mean) probability, with 95% CIs, of each domain being chosen as the most or least important for individual participants and across the entire sample. The raw logit scores were then transformed to a relative importance score on a 0–100 probability scale, where scores sum to 100 across domains.
      • Chrazan K.
      • Orme B.
      Applied MaxDiff: A practitioner's guide to best-worst scaling.
      ,
      Sawtooth Software
      Aggregate score estimation via logit analysis.
      This transformation facilitates a readily interpretable comparison of domains.
      Sawtooth Software
      The MaxDiff system technical paper.
      More specifically, this transformed score indicates the relative importance of domains on a common scale; for example, a domain that is given a score of 10 is perceived by respondents (as revealed via their choices) as being twice as important as a domain with a score of 5. Domains were rank ordered according to their importance score, where higher scores indicated higher perceived importance. To describe the variation in how respondents rated each domain, we also calculated the interquartile range (IQR) in each domain and reported the median, 25th and 75th percentiles, and any outliers.
      • Agresti A.
      • Finlay B.
      Statistical methods for the social sciences.
      In additional analyses, we explored how domain ratings differed depending on whether there were siblings in the household, whether the parent was bereaved, and whether the parent was a mother or a father. All comparisons were conducted using two-sample independent t-tests. Given the exploratory nature of these subgroup analyses, P-values < 0.1 were considered statistically significant.

      Sample Size Considerations

      DCEs of the design we used converge on stable estimates of the relative scores for items with as few as 20 participants in subgroups.
      • de Bekker-Grob E.W.
      • Donkers B.
      • Jonker M.F.
      • Stolk E.A.
      Sample size requirements for discrete-choice experiments in healthcare: a practical guide.
      ,
      • Lancsar E.
      • Louviere J.
      Conducting discrete choice experiments to inform healthcare decision making.
      There are currently no standard guidelines for defining sample sizes for these DCE studies; therefore, simulation studies, where the effect of different sample sizes are simulated to test the effect on the reliability of estimates, are recommended.
      • Flynn T.N.
      • Louviere J.J.
      • Peters T.J.
      • Coast J.
      Best-worst scaling: what it can do for health care research and how to do it.
      Using data from a previous DCE with a sample of 200 parents of children with serious illnesses,
      • Feudtner C.
      • Walter J.K.
      • Faerber J.A.
      • et al.
      Good-parent beliefs of parents of seriously ill children.
      we used a bootstrap approach to draw overall data set samples of 20, 30, or 50 individual responses to the DCE at a time, doing so with replacement and iterating this process 100 times for each sample size. Across all three sample sizes, all items were consistently ordered from top to bottom. Although the CIs indicated that similarly-rated items may, for some samples, switch ranking, they demonstrated that with a sample size of 30, high-rated items can be clearly differentiated from low-rated items. Thus, we aimed to recruit a minimum of 30 parent participants from 30 unique families.

      Survey and Analytic Software

      We designed the DCE and deployed the survey instruments using Lighthouse Studio (Version 9.6.1; Sawtooth Software, Provo, UT), which includes a cloud-based survey platform. We calculated the domain importance scores based on participants' choices across the DCE sets using Lighthouse Studio and conducted statistical analysis of the importance scores using Stata, version 15.1 (StataCorp, College Station, TX).

      Results

      Forty-seven parents from 45 families participated (Table 2). Most participants were white (89.4%), non-Hispanic (91.5%), and mothers (93.6%) who were married or partnered (87.2%) and had completed college or graduate school (68.1%). Parents' mean age was 42.6 years (SD = 8.5). Fourteen parents (29.8%) were bereaved, and 33 (70.2%) parents were currently caring for their child at home. Sixteen of the 47 parents (34%) received care for their child primarily from CHOP's interprofessional palliative care service.
      Table 2Demographic and Clinical Characteristics of Parents and Children
      n (%)
      Parents' Characteristics (n = 47)
       Parent type
      Mother44 (93.6)
      Father3 (6.4)
       Age
      Mean/SD42.6 (8.5)
       Race
      White42 (89.4)
      Black or African American1 (2.1)
      More than one race/other3 (6.4)
      Prefer not to answer1 (2.1)
       Ethnicity
      Non-Hispanic43 (91.5)
      Hispanic3 (6.4)
      Prefer not to answer1 (2.1)
       Religious preference
      Christian (Protestant, Catholic, Mormon, etc.)31 (66.0)
      Jewish6 (12.8)
      Muslim0
      Buddhist1 (2.1)
      Hindu1 (2.1)
      Atheist3 (6.4)
      Agnostic2 (4.3)
      Prefer not to answer3 (6.4)
       Highest education level completed
      Grade school1 (2.1)
      High school/general educational development2 (4.3)
      Trade/technical/vocational4 (8.5)
      Associates/professional8 (17.0)
      College19 (40.4)
      Graduate school13 (27.7)
       Relationship status
      Married/partnered41 (87.2)
      Separated/divorced/widowed6 (12.8)
       Number of other children
      011 (23.4)
      1–335 (74.5)
      4 or more1 (2.1)
       Employment status
      Full time23 (48.9)
      Part time5 (10.6)
      Not employed outside the home17 (36.2)
      Prefer not to answer2 (4.3)
       Bereavement status
      Bereaved14 (29.8)
      Currently caring for child at home33 (70.2)
       Affiliation
      CHOP16 (34.0)
      CPN31 (66.0)
      Children's Characteristics (n = 45)
       Age (yrs)
      One or less8 (17.8)
      2–49 (20.0)
      5–95 (11.1)
      10–1817 (37.8)
      19–256 (13.3)
       Gender
      Female21 (46.7)
      Male24 (53.3)
       Race
      White37 (82.2)
      Black or African American2 (4.4)
      More than one race/other5 (11.1)
      Prefer not to answer1 (2.2)
       Ethnicity
      Non-Hispanic39 (86.7)
      Hispanic4 (8.9)
      Prefer not to answer2 (4.4)
       Primary complex chronic condition (Note: not mutually exclusive; thus, the percent does not sum to 100%)
      Cardiovascular10 (22.2)
      Gastrointestinal4 (8.9)
      Genetic or congenital22 (48.9)
      Hematologic or immunologic4 (8.9)
      Malignancy5 (11.1)
      Metabolic10 (22.2)
      Neuromuscular, neurologic, or mitochondrial23 (51.1)
      Respiratory6 (13.3)
      Other/unknown1 (2.2)
       Primary care team (hospice v. palliative care)
      Hospice19 (42.2)
      Palliative care24 (53.3)
      Unknown/not sure2 (4.4)
       Length of time receiving home-based palliative or hospice care
      Less than one month5 (11.1)
      One to three months5 (11.1)
      Four to six months7 (15.6)
      Seven to 12 months5 (11.1)
      One to two years8 (17.8)
      More than two years15 (33.3)
      CHOP = Children's Hospital of Philadelphia; CPN = Courageous Parents Network.
      Note: Unless otherwise noted, cell entries are percentages. Percentages are within each demographic or clinical characteristic variable.
      Participants were parents to 45 children who have received [email protected] Approximately half of these children were between birth and nine years, and half were between 10 and 25 years. Most prevalent primary diagnoses included neuromuscular, neurologic, or mitochondrial (51.1%), genetic or congenital (48.9%), cardiovascular (22.2%), and metabolic (22.2%) diseases. More than half of these children received home-based support primarily from a palliative care team (53.3%), and a third of these children received [email protected] for more than two years (33.3%).

      Average Domain Importance Scores and Ranking

      Among the 20 domains of [email protected] (Fig. 1), parents ranked the Physical aspects of care: Symptom management domain the highest (mean score = 9.68; 95% CI = 8.71, 10.64), followed by Psychological/emotional aspects of care for the child (mean score = 8.97; 95% CI = 7.74, 10.21) and Care coordination (mean score = 8.92; 95% CI = 7.92, 9.92). Among the lowest-ranked domains were Emotional aspects of care for the extended social network (mean score = 0.90; 95% CI = 0.36, 1.45), Spiritual and religious aspects of care (mean score = 0.22; 95% CI = 0.12, 0.32), and Cultural aspects of care (mean score = 0.12; 95% CI = 0.07, 0.17).
      Figure thumbnail gr1
      Fig. 1Average importance scores across domains. Note: This figure contains the point estimates of the mean importance score for each domain. The 95% CI around each point estimate represents our level of confidence that the interval captures the true point estimate. EOL = end of life.
      In terms of relative importance, parents rated the Physical aspects of care: Symptom management domain as being twice as important as the Practical aspects of care domain (mean score = 9.68 vs. 4.75) and nearly 10 times as important as the Emotional aspects of care for the family's extended social network domain (mean score = 9.68 vs. 0.90).

      Individual-Level Variation in Domain Importance Scores and Ranking

      Domains differed not only regarding their mean importance score, but also in the degree to which the participants, as a group, varied in their scores for given domains. For example, the Physical aspects of care: Symptom management domain had a median score of 11.18 and an IQR range of 5.25, whereas the Psychological/emotional aspects of care for the sibling(s) domain had a median score of 6.35, yet a much larger IQR of 9.91, representing greater variation in how respondents rated this domain (Fig. 2).
      Figure thumbnail gr2
      Fig. 2Individual-level variation across domain importance scores. Note: This graph presents the interquartile range of domains, which represents the spread of the middle half of the scores in each domain. The line within each box represents the median, the left edge of the box represents the 25th percentile, and the right edge represents the 75th percentile of scores. The whiskers extending out of the box represents minimum and maximum scores, except for outliers (defined as more than 1.5 interquartile range beyond the 25th and 75th quartiles), which are represented by the dots.
      • Agresti A.
      • Finlay B.
      Statistical methods for the social sciences.
      EOL = end of life.

      Additional Analysis: Subgroup Comparisons in Domain Importance Scores

      We examined three potential notions of how parents might differ in their importance ratings of domains. First, we assessed how parents with other children rated the sibling-specific domain (Psychological/emotional aspects of care for the sibling) compared with parents who did not have other children. The mean score for parents with at least one other child (n = 38) was 7.55 (95% CI = 6.01, 9.10), whereas for parents without other children (n = 9), the mean score was 3.27 (95% CI = −0.48, 7.02) (t = −2.44; P = 0.02). Second, we compared parents currently caring for their child (n = 33) and bereaved parents (n = 14) across all 20 domains; only the Caregiver support at the end of life domain had significantly different scores at the P < 0.1 level (t = −2.07; P = 0.04), where bereaved parents rated this domain 1.5 times as important as parents currently caring for their child. Third, mothers' (n = 44) and fathers' (n = 3) domain importance scores did not display any statistically significant differences (Table 3).
      Table 3Subgroup Domain Score Comparisons: Two-Sample Independent t-Tests
      DomainSubgroupMean95% CIP
      Single domain: Parents without other children (n = 9) vs. parents with other children (n = 38)
       Psychological and emotional aspects of care for the sibling(s)
      No siblings3.27−0.48, 7.020.02
      Significant at P < 0.1 level.
      One or more siblings7.556.01, 9.10
      Across all domains: parents currently caring for child (n = 33) vs. bereaved parents (n = 14)
       Communication between family and care team
      Currently caring for child6.084.99, 7.160.98
      Bereaved6.054.00, 8.10
       Relationship between family and care team
      Currently caring for child5.534.24, 6.820.45
      Bereaved4.612.21, 7.00
       Knowledge and skills of care team providers
      Currently caring for child6.635.12, 8.130.81
      Bereaved6.944.93, 8.96
       Access to care
      Currently caring for child6.555.32, 7.790.29
      Bereaved7.775.56, 9.97
       Physical care: Communication
      Currently caring for child5.394.46, 6.310.62
      Bereaved4.963.31, 6.62
       Physical care: Symptom management
      Currently caring for child9.338.05, 10.600.28
      Bereaved10.508.92, 12.07
       Psychological and emotional aspects of care: Parent
      Currently caring for child5.704.42, 6.990.97
      Bereaved5.753.98, 7.53
       Psychological and emotional aspects of care: Child
      Currently caring for child8.787.23, 10.320.64
      Bereaved9.436.92, 11.95
       Psychological and emotional aspects of care: Sibling(s)
      Currently caring for child6.844.99, 8.690.83
      Bereaved6.493.86, 9.13
       Psychological and emotional aspects of care: Extended social network
      Currently caring for child1.040.30, 1.790.45
      Bereaved0.57−0.18, 1.33
       Practical aspects of care
      Currently caring for child5.213.76, 6.660.24
      Bereaved3.651.36, 5.95
       Social aspects of care: Parents
      Currently caring for child3.182.02, 4.350.31
      Bereaved2.160.60, 3.73
       Social aspects of care: Child
      Currently caring for child2.020.96, 3.080.47
      Bereaved1.400.56, 2.24
       Spiritual and religious aspects of care
      Currently caring for child0.200.09, 0.310.48
      Bereaved0.280.01, 0.56
       Cultural aspects of care
      Currently caring for child0.140.07, 0.220.19
      Bereaved0.070.03, 0.11
       Communication at the end of life
      Currently caring for child5.804.43, 7.170.51
      Bereaved6.565.08, 8.04
       Caregiver support at the end of life
      Currently caring for child4.723.34, 6.100.04
      Significant at P < 0.1 level.
      Bereaved7.105.47, 8.72
       Ethical and legal aspects of care
      Currently caring for child2.041.09, 3.000.49
      Bereaved2.650.99, 4.31
       Coordination of care
      Currently caring for child8.997.75, 10.230.84
      Bereaved8.766.67, 10.85
       Continuity of care
      Currently caring for child5.824.43, 7.220.20
      Bereaved4.282.56, 5.99
      Across all domains: Mothers (n = 44) vs. fathers (n = 3)
       Communication between family and care team
      Mothers6.105.13, 7.070.77
      Fathers5.55−3.12, 14.21
       Relationship between family and care team
      Mothers5.133.97, 6.290.40
      Fathers7.07−1.87, 16.02
       Knowledge and skills of care team providers
      Mothers6.725.51, 7.930.99
      Fathers6.73−6.81, 20.28
       Access to care
      Mothers6.995.90, 8.080.60
      Fathers5.84−3.88, 15.56
       Physical care: Communication
      Mothers5.104.30, 5.910.12
      Fathers7.571.95, 13.19
       Physical care: Symptom management
      Mothers9.658.62, 10.680.28
      Fathers10.08−0.11, 20.26
       Psychological and emotional aspects of care: Parent
      Mothers5.744.67, 6.800.89
      Fathers5.44−1.23, 12.11
       Psychological and emotional aspects of care: Child
      Mothers8.757.42, 10.080.17
      Fathers12.2910.14, 14.44
       Psychological and emotional aspects of care: Sibling(s)
      Mothers6.805.29, 8.310.72
      Fathers5.73−9.79, 21.25
       Psychological and emotional aspects of care: Extended social network
      Mothers0.910.32, 1.500.97
      Fathers0.86−2.29, 4.01
       Practical aspects of care
      Mothers4.823.57, 6.060.64
      Fathers3.67−7.66, 15.01
       Social aspects of care: Parents
      Mothers3.012.04, 3.990.27
      Fathers0.92−0.11, 1.94
       Social aspects of care: Child
      Mothers1.790.98, 2.590.65
      Fathers2.52−4.44, 9.48
       Spiritual and religious aspects of care
      Mothers0.240.12, 0.350.43
      Fathers0.06−0.01, 0.14
       Cultural aspects of care
      Mothers0.130.07, 0.180.40
      Fathers0.04−0.01, 0.08
       Communication at the end of life
      Mothers6.095.01, 7.180.63
      Fathers5.06−2.87, 13.00
       Caregiver support at the end of life
      Mothers5.504.36, 6.640.60
      Fathers4.33−5.20, 13.87
       Ethical and legal aspects of care
      Mothers2.341.50, 3.190.26
      Fathers0.49−0.88, 1.85
       Coordination of care
      Mothers8.897.81, 9.960.78
      Fathers9.481.41, 17.55
       Continuity of care
      Mothers5.304.20, 6.410.67
      Fathers6.27−7.75, 20.29
      a Significant at P < 0.1 level.

      Discussion

      We conducted a DCE to prioritize 20 [email protected] domains based on quantitative parent-rated domain importance scores and identified, through exploratory analysis, potential differences in how subgroups of parents rated these domains. Several of our findings warrant discussion.
      First, parents ranked Physical aspects of care: Symptom management and Psychological/emotional aspects of care for the child as the most important domains. This prioritization is unsurprising, given the proportion of children with serious illness who experience pain and other distressing physical symptoms, such as fatigue, reduced mobility, and constipation,
      • Feudtner C.
      • Kang T.I.
      • Hexem K.R.
      • et al.
      Pediatric palliative care patients: a prospective multicenter cohort study.
      ,
      • Jalmsell L.
      • Kreicbergs U.
      • Onelöv E.
      • Steineck G.
      • Henter J.-I.
      Symptoms affecting children with malignancies during the last month of life: a nationwide follow-up.
      • Rosenberg A.R.
      • Orellana L.
      • Ullrich C.
      • et al.
      Quality of life in children with advanced cancer: a report from the PediQUEST study.
      • Schmidt P.
      • Otto M.
      • Hechler T.
      • et al.
      Did increased availability of pediatric palliative care lead to improved palliative care outcomes in children with cancer?.
      • Wolfe J.
      • Grier H.E.
      • Klar N.
      Symptoms and suffering at the end of life in children with cancer.
      and psychological symptoms, such as sadness, worry, and anxiety.
      • Feudtner C.
      • Kang T.I.
      • Hexem K.R.
      • et al.
      Pediatric palliative care patients: a prospective multicenter cohort study.
      ,
      • Rosenberg A.R.
      • Orellana L.
      • Ullrich C.
      • et al.
      Quality of life in children with advanced cancer: a report from the PediQUEST study.
      ,
      • Kersun L.S.
      • Shemesh E.
      Depression and anxiety in children at the end of life.
      ,
      • Theunissen J.M.
      • Hoogerbrugge P.M.
      • Achterberg T.V.
      • et al.
      Symptoms in the palliative phase of children with cancer.
      The relief of symptoms is often described by pediatric and adult patients and their family caregivers as a top concern and priority for quality palliative and end-of-life care;
      • Donnelly J.P.
      • Huff S.M.
      • Lindsey M.L.
      • McMahon K.A.
      • Schumacher J.D.
      The needs of children with life-limiting conditions: a healthcare-provider-based model.
      ,
      • Meyer E.C.
      • Ritholz M.D.
      • Burns J.P.
      • Truog R.D.
      Improving the quality of end-of-life care in the pediatric intensive care unit: parents' priorities and recommendations.
      ,
      • Heyland D.K.
      • Dodek P.
      • Rocker G.
      • et al.
      What matters most in end-of-life care: perceptions of seriously ill patients and their family members.
      • Namisango E.
      • Bristowe K.
      • Allsop M.J.
      • et al.
      Symptoms and concerns among children and young people with life-limiting and life-threatening conditions: a systematic review highlighting meaningful health outcomes.
      • Stevenson M.
      • Achille M.
      • Lugasi T.
      Pediatric palliative care in Canada and the United States: a qualitative metasummary of the needs of patients and families.
      • Vohra J.U.
      • Brazil K.
      • Hanna S.
      • Abelson J.
      Family perceptions of end-of-life care in long-term care facilities.
      yet, the assessment and management of pain and other symptoms has room for improvement for patients across ages, diagnoses, and settings.
      • Contro N.
      • Larson J.
      • Scofield S.
      • Sourkes B.
      • Cohen H.
      Family perspectives on the quality of pediatric palliative care.
      ,
      • Meyer E.C.
      • Ritholz M.D.
      • Burns J.P.
      • Truog R.D.
      Improving the quality of end-of-life care in the pediatric intensive care unit: parents' priorities and recommendations.
      ,
      • Theunissen J.M.
      • Hoogerbrugge P.M.
      • Achterberg T.V.
      • et al.
      Symptoms in the palliative phase of children with cancer.
      ,
      • Vohra J.U.
      • Brazil K.
      • Hanna S.
      • Abelson J.
      Family perceptions of end-of-life care in long-term care facilities.
      • Meyer E.C.
      • Burns J.P.
      • Griffith J.L.
      • Truog R.D.
      Parental perspectives on end-of-life care in the pediatric intensive care unit.
      • Singer P.
      • Martin D.
      • Kelner M.
      Quality end-of-life care: patients' perspectives.
      • Thienprayoon R.
      • Marks E.
      • Funes M.
      • et al.
      Perceptions of the pediatric hospice experience among English- and Spanish-speaking families.
      • Zheng N.T.
      • Li Q.
      • Hanson L.C.
      • et al.
      Nationwide quality of hospice care: findings from the Centers for Medicare & Medicaid Services hospice quality reporting program.
      Inadequate pain and symptom management has been associated with child and family outcomes, such as lower child health-related quality of life,
      • Rosenberg A.R.
      • Orellana L.
      • Ullrich C.
      • et al.
      Quality of life in children with advanced cancer: a report from the PediQUEST study.
      ,
      • Meeske K.A.
      • Patel S.K.
      • Palmer S.N.
      • Nelson M.B.
      • Parow A.M.
      Factors associated with health-related quality of life in pediatric cancer survivors.
      long-term parental grief and distress,
      • Kreicbergs U.
      • Valdimarsdottir U.
      • Onelov E.
      • et al.
      Care-related distress: a nationwide study of parents who lost their child to cancer.
      ,
      • van der Geest I.M.M.
      • Darlington A.
      • Streng I.C.
      • et al.
      Parents' experiences of pediatric palliative care and the impact on long-term parental grief.
      and parents' more negative perceptions of care quality.
      • Donnelly J.P.
      • Huff S.M.
      • Lindsey M.L.
      • McMahon K.A.
      • Schumacher J.D.
      The needs of children with life-limiting conditions: a healthcare-provider-based model.
      ,
      • Contro N.
      • Larson J.
      • Scofield S.
      • Sourkes B.
      • Cohen H.
      Family perspectives on the quality of pediatric palliative care.
      ,
      • Homer C.J.
      • Marino B.
      • Cleary P.D.
      • et al.
      Quality of care at a children's hospital: the parent's perspective.
      Parents also ranked Care coordination among the most important domains. Seriously-ill and medically-complex children often have significant care needs, necessitating care from a complex network of providers across numerous settings and institutions.
      • Cohen E.
      • Lacombe-Duncan A.
      • Spalding K.
      • et al.
      Integrated complex care coordination for children with medical complexity: a mixed-methods evaluation of tertiary care-community collaboration.
      As a result, parents, providers, and health care leaders have identified care coordination as a significant need
      • Field M.J.
      • Behrman R.E.
      When children die: Improving palliative and end-of-life care for children and their families.
      ,
      • Stevenson M.
      • Achille M.
      • Lugasi T.
      Pediatric palliative care in Canada and the United States: a qualitative metasummary of the needs of patients and families.
      ,
      • Cady R.G.
      • Belew J.L.J.C.
      Parent perspective on care coordination services for their child with medical complexity.
      and a priority for clinical practice, policy, and research.
      • Feudtner C.
      • Friebert S.
      • Jewell J.
      Pediatric palliative care and hospice care commitments, guidelines, and recommendations.
      ,
      • Field M.J.
      • Behrman R.E.
      When children die: Improving palliative and end-of-life care for children and their families.
      ,
      • Baker J.N.
      • Levine D.R.
      • Hinds P.S.
      • et al.
      Research priorities in pediatric palliative care.
      ,
      • Lipkin P.
      • Alexander J.
      • Cartwright J.
      Care coordination in the medical home: integrating health and related systems of care for children with special health care needs.
      Notably, care coordination was added as a key theme in each domain in the most recent edition of the NCP Guidelines,
      National Consensus Project for Quality Palliative Care
      Clinical practice guidelines for quality palliative care.
      and with good reason: care coordination is associated with improved care quality and child and family outcomes (e.g., quality of life, symptom management, death in preferred location) in the home and community settings.
      • Boyden J.Y.
      • Curley M.A.Q.
      • Deatrick J.A.
      • Ersek M.
      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.
      ,
      • Thienprayoon R.
      • Grossoehme D.
      • Humphrey L.
      • et al.
      "There's just no way to help, and they did." Parents name compassionate care as a new domain of quality in pediatric home-based hospice and palliative care.
      ,
      • Thienprayoon R.
      • Mark M.S.J.
      • Grossoehme D.
      Provider-prioritized domains of quality in pediatric home-based hospice and palliative care: a study of the Ohio Pediatric Palliative Care and End-of-Life Network.
      ,
      • Cohen E.
      • Lacombe-Duncan A.
      • Spalding K.
      • et al.
      Integrated complex care coordination for children with medical complexity: a mixed-methods evaluation of tertiary care-community collaboration.
      ,
      • Friedrichsdorf S.J.
      • Postier A.
      • Dreyfus J.
      • et al.
      Improved quality of life at end of life related to home-based palliative care in children with cancer.
      • Bona K.
      • Bates J.
      • Wolfe J.
      Massachusetts' Pediatric Palliative Care Network: successful implementation of a novel state-funded pediatric palliative care program.
      • Knapp C.A.
      • Madden V.L.
      • Curtis C.M.
      • et al.
      Partners in care: together for kids: Florida's model of pediatric palliative care.
      However, coordinating care by health care teams, a time-consuming endeavor, is often not reimbursed.
      • Ronis S.
      • Grossberg R.
      • Allen R.
      • Hertz A.
      • Kleinman L.
      Estimated nonreimbursed costs for care coordination for children with medical complexity.
      ,
      • Coller R.
      • Ehlenbach M.
      Making time to coordinate care for children with medical complexity.
      Future research should examine the effectiveness and efficiency of different models of care coordination, followed by advocacy to align reimbursement with high-value service models.
      The lowest-ranked domains in this study were Spiritual and religious aspects of care and Cultural aspects of care. Although we know that these care domains are critical components of effective PPHC,
      • Hexem K.R.
      • Mollen C.J.
      • Carroll K.W.
      • Lanctot D.A.
      • Feudtner C.
      How parents of children receiving pediatric palliative care use religion, spirituality, or life philosophy in tough times.
      • Wiener L.
      • McConnell D.G.
      • Latella L.
      • Ludi E.
      Cultural and religious considerations in pediatric palliative care.
      • Davies B.
      • Brenner P.
      • Orloff S.
      • Sumner L.
      • Worden W.
      Addressing spirituality in pediatric hospice and palliative care.
      • Davies B.
      • Contro N.
      • Larson J.
      • Widger K.
      Culturally-sensitive information-sharing in pediatric palliative care.
      they have been found to be more important for some families than others.
      • Meyer E.C.
      • Burns J.P.
      • Griffith J.L.
      • Truog R.D.
      Parental perspectives on end-of-life care in the pediatric intensive care unit.
      ,
      • Hexem K.R.
      • Mollen C.J.
      • Carroll K.W.
      • Lanctot D.A.
      • Feudtner C.
      How parents of children receiving pediatric palliative care use religion, spirituality, or life philosophy in tough times.
      In particular, these two domains may be ranked lower by parents in our sample, who were largely white, non-Hispanic, and Christian; a more diverse sample of parents may have prioritized these domains higher. Another possible explanation is that when parents are forced to choose between domains, as in this discrete choice exercise, parents may prioritize the management of their child's symptoms and the effective coordination of their child's care over spiritual or cultural aspects of care. A previous DCE study identified parents' beliefs about what they need to do to be a good parent to their seriously-ill child. This study, conducted with 200 parents of children with serious illness, similarly found that beliefs such as Making my child feel loved and Focusing on my child's comfort were ranked higher by parents than the belief Focusing on my child's spiritual well-being.
      • Feudtner C.
      • Walter J.K.
      • Faerber J.A.
      • et al.
      Good-parent beliefs of parents of seriously ill children.
      Finally, some children and families may receive support for their spiritual, religious, and cultural needs from community-based organizations or other social support networks and may simply not expect or require this type of support from their [email protected] team.
      We did observe potential differences in how subgroups of parents ranked the domains. In particular, we found that parents who had other children ranked the Psychological/emotional aspects of care for sibling(s) domain more than twice as important as parents who did not have other children. This finding is unsurprising in light of the documented needs of siblings of seriously-ill children
      • Stevenson M.
      • Achille M.
      • Lugasi T.
      Pediatric palliative care in Canada and the United States: a qualitative metasummary of the needs of patients and families.
      ,
      • Thienprayoon R.
      • Marks E.
      • Funes M.
      • et al.
      Perceptions of the pediatric hospice experience among English- and Spanish-speaking families.
      ,
      • Gaab E.
      • Owens G.
      • MacLeod R.
      Siblings caring for and about pediatric palliative care patients.
      ,
      • Eaton Russell C.
      • Widger K.
      • Beaune L.
      • et al.
      Siblings' voices: a prospective investigation of experiences with a dying child.
      and the significant stress parents face when balancing the care of their seriously-ill child with care of the child's siblings.
      • Verberne L.M.
      • Kars M.C.
      • Schouten-van Meeteren A.Y.
      • et al.
      Aims and tasks in parental caregiving for children receiving palliative care at home: a qualitative study.
      ,
      • Mooney-Doyle K.
      • Deatrick J.A.
      Parenting in the face of childhood life-threatening conditions: the ordinary in the context of the extraordinary.
      Given the proximity of siblings to their ill sibling's care in the home setting,
      • Eaton Russell C.
      • Widger K.
      • Beaune L.
      • et al.
      Siblings' voices: a prospective investigation of experiences with a dying child.
      further intervention-based research is needed to understand how this support may best be provided to siblings in the home setting in particular. We also observed differences in how bereaved parents and parents currently caring for their child rated the Caregiver support at the end of life domain. This is likely because parents who are actively caring for their child who is not yet at the end of life may not see bereavement care as important when compared with other domains representing more immediate needs or concerns. Alternatively, parents may be unwilling or unable to consider a time when bereavement care is needed for their family.
      Finally, we did not observe significant differences in how mothers and fathers ranked the domains, although these rankings represent the views of only three fathers and, thus, precludes any conclusions to be drawn. Although further exploration with larger samples of fathers is greatly needed in this area, previous research with larger samples of fathers have found that the major areas of problems, hopes, and goals related to a child's care were not significantly different between mothers and fathers,
      • Hill D.L.
      • Miller V.A.
      • Hexem K.R.
      • et al.
      Problems and hopes perceived by mothers, fathers and physicians of children receiving palliative care.
      and that the type of support parents need to care for their child with serious illness at home may be the same, regardless of gender.
      • Verberne L.M.
      • Kars M.C.
      • Schouten-van Meeteren A.Y.
      • et al.
      Aims and tasks in parental caregiving for children receiving palliative care at home: a qualitative study.
      This study has four limitations that warrant mention. First, the gender and sociodemographic characteristics of our sample were relatively homogenous. Although this homogeneity is not surprising given issues with gender
      • Hinds P.S.
      • Burghen E.A.
      • Pritchard M.
      Conducting end-of-life studies in pediatric oncology.
      ,
      • Macdonald M.E.
      • Chilibeck G.
      • Affleck W.
      • Cadell S.
      Gender imbalance in pediatric palliative care research samples.
      and minority
      • Knapp C.A.
      • Madden V.L.
      • Curtis C.
      • Sloyer P.J.
      • Shenkman E.A.
      Assessing non-response bias in pediatric palliative care research.
      imbalance in PPC studies, as well as disparities in access to home-based hospice care in general by minority patients,
      • Cohen L.L.
      Racial/ethnic disparities in hospice care: a systematic review.
      additional research with larger and more sociodemographically diverse samples is critically needed to more fully understand parental priorities, particularly among underrepresented groups (e.g., fathers, racial and ethnic minorities, single parents, other cultural and religious groups, families from lower socioeconomic groups).
      Second, although families in our study received care from various geographic regions of the U.S. (e.g., East Coast, Mid-Atlantic, Mid-West, South), we did not collect data about the [email protected] programs that served the families in our sample. As a result, parents may have evaluated a domain of care as less important if services addressing that domain (e.g., spiritual care, support for the sibling) were ineffective or unavailable or if families chose not to use particular services. These variations in experiences may have influenced parents' domain ratings. We do note that, anecdotally, as part of this overall larger study, several bereaved parents recounted receiving less-than-effective aspects of their [email protected] experiences that they felt were very important to improve on for future children and families. For example, several parents discussed a lack of adequate access to appropriately-trained (e.g., pediatric and palliative/hospice care trained) nursing support in the home; yet, all of these parents acknowledged the critical role nursing care plays in good [email protected] Future research is needed to evaluate differences in care experiences with larger samples of families who receive different models of care (e.g., adult hospice programs that also care for children, pediatric-specific hospice programs, hospital-based PPC programs that also support families at home) from different geographic regions of the U.S.
      Third, this study may have omitted important domains of [email protected] We believe, however, that this is unlikely because the domains were based on the NCP Guidelines for Quality Palliative Care
      National Consensus Project for Quality Palliative Care
      Clinical practice guidelines for quality palliative care.
      and informed by PPC-specific guidelines and literature
      • Thienprayoon R.
      • Mark M.S.J.
      • Grossoehme D.
      Provider-prioritized domains of quality in pediatric home-based hospice and palliative care: a study of the Ohio Pediatric Palliative Care and End-of-Life Network.
      ,
      • Feudtner C.
      • Friebert S.
      • Jewell J.
      Pediatric palliative care and hospice care commitments, guidelines, and recommendations.
      • Field M.J.
      • Behrman R.E.
      When children die: Improving palliative and end-of-life care for children and their families.
      National Hospice and Palliative Care Organization (NHPCO)
      Standards of practice for pediatric palliative care.
      • Widger K.
      • Brennenstuhl S.
      • Duc J.
      • Tourangeau A.
      • Rapoport A.
      Factor structure of the Quality of Children's Palliative Care Instrument (QCPCI) when completed by parents of children with cancer.
      and by a panel of PPHC stakeholders.
      Fourth, because of the cross-sectional design of this study, we do not know how much individual parental ratings of importance change over time. Future longitudinal studies will be important to examine trends in parents' priorities over the course of their child's illness. Our findings, however, build on and extend beyond findings from previous studies, providing quantitative evidence for the overall importance of excellent pain and symptom management, psychological and emotional support for seriously-ill children, and integrated, coordinated care for children with serious illness and their families in the home setting.
      • Thienprayoon R.
      • Grossoehme D.
      • Humphrey L.
      • et al.
      "There's just no way to help, and they did." Parents name compassionate care as a new domain of quality in pediatric home-based hospice and palliative care.
      ,
      • Friedrichsdorf S.J.
      • Postier A.
      • Dreyfus J.
      • et al.
      Improved quality of life at end of life related to home-based palliative care in children with cancer.
      • Bona K.
      • Bates J.
      • Wolfe J.
      Massachusetts' Pediatric Palliative Care Network: successful implementation of a novel state-funded pediatric palliative care program.
      • Knapp C.A.
      • Madden V.L.
      • Curtis C.M.
      • et al.
      Partners in care: together for kids: Florida's model of pediatric palliative care.
      ,
      • Gaab E.
      • Owens G.
      • MacLeod R.
      Siblings caring for and about pediatric palliative care patients.
      ,
      • Gans D.
      • Hadler M.W.
      • Chen X.
      • et al.
      Impact of a pediatric palliative care program on the caregiver experience.
      ,
      • Vollenbroich R.
      • Duroux A.
      • Grasser M.
      • et al.
      Effectiveness of a pediatric palliative home care team as experienced by parents and health care professionals.

      Conclusion

      The priorities for [email protected] among parents in this study represent a foundation for further exploration and conversation about how to prioritize finite health care resources and where to focus care improvement efforts and future research, particularly in the home-based setting, for seriously-ill children and their families.

      Disclosures and Acknowledgments

      The authors acknowledge the Justin Michael Ingerman Center for Palliative Care at the Children's Hospital of Philadelphia and the Courageous Parents Network, who facilitated the design and data collection phases of this study. In particular, they thank Karen Crew and Dana Dombrowski for their guidance and effort on this project, as well as Russell Nye for his review of this manuscript. They also acknowledge all the clinicians, researchers, and parent advocates who shared their time and expertise with them. And finally, they thank the parents who so generously gave their time and energy to participate in this project—they are truly grateful for parents' willingness to share their perspectives with them.
      This work was supported by the National Institute of Nursing Research of the National Institutes of Health under award number F31NR017554. The content is solely the responsibility of the authors and does not necessarily represent the official views of the National Institutes of Health. This work was additionally supported by the University of Pennsylvania School of Nursing Office of Nursing Research's Student Research grant, the Independence Blue Cross Foundation's Nurses for Tomorrow Scholars Program, and the Sawtooth Software Academic Grant.
      The authors declare no conflicts of interest.

      References

        • Feudtner C.
        • Feinstein J.A.
        • Satchell M.
        • Zhao H.
        • Kang T.I.
        Shifting place of death among children with complex chronic conditions in the United States, 1989-2003.
        JAMA. 2007; 297: 2725-2732
        • Feudtner C.
        • Kang T.I.
        • Hexem K.R.
        • et al.
        Pediatric palliative care patients: a prospective multicenter cohort study.
        Pediatrics. 2011; 127: 1094-1101
        • Feudtner C.
        • Silveira M.J.
        • Christakis D.A.
        Where do children with complex chronic conditions die? Patterns in Washington State, 1980-1998.
        Pediatrics. 2002; 109: 656-660
        • Nageswaran S.
        • Radulovic A.
        • Anania A.
        Transitions to and from the acute inpatient care setting for children with life-threatening illness.
        Pediatr Clin North Am. 2014; 61: 761-783
        • Feudtner C.
        • Womer J.
        • Augustin R.
        • et al.
        Pediatric palliative care programs in children's hospitals: a cross-sectional national survey.
        Pediatrics. 2013; 132: 1063-1070
        • Friebert S.
        • Williams C.
        NHPCO'S facts and figures: Pediatric palliative and hospice care in America.
        NHPCO, Alexandria, VA2015
        • Kaye E.
        • Rubenstein J.
        • Levine D.
        • et al.
        Pediatric palliative care in the community.
        CA Cancer J Clin. 2015; 65: 315-333
        • Boyden J.Y.
        • Curley M.A.Q.
        • Deatrick J.A.
        • Ersek M.
        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.
        J Pain Symptom Manage. 2018; 55: 117-131
        • Lindley L.
        • Mark B.
        • Lee S.Y.D.
        Providing hospice care to children and young adults: a descriptive study of end-of-life organizations.
        J Hosp Palliat Nurs. 2009; 11: 315-323
        • Carroll J.M.
        • Torkildson C.
        • Winsness J.S.
        Issues related to providing quality pediatric palliative care in the community.
        Pediatr Clin North Am. 2007; 54 (xiii): 813-827
        • Lindley L.C.
        • Kieim-Malpass J.
        Quality of paediatric hospice care for children with and without multiple complex chronic conditions.
        Int J Palliat Nurs. 2017; 23: 230-237
        • Donnelly J.P.
        • Huff S.M.
        • Lindsey M.L.
        • McMahon K.A.
        • Schumacher J.D.
        The needs of children with life-limiting conditions: a healthcare-provider-based model.
        Am J Hosp Palliat Med. 2005; 22: 259-267
        • Contro N.
        • Larson J.
        • Scofield S.
        • Sourkes B.
        • Cohen H.
        Family perspectives on the quality of pediatric palliative care.
        Arch Pediatr Adolesc Med. 2002; 156: 14-19
        • Mack J.W.
        • Hilden J.M.
        • Watterson J.
        • et al.
        Parent and physician perspectives on quality of care at the end of life in children with cancer.
        J Clin Oncol. 2005; 23: 9155-9161
        • Meyer E.C.
        • Ritholz M.D.
        • Burns J.P.
        • Truog R.D.
        Improving the quality of end-of-life care in the pediatric intensive care unit: parents' priorities and recommendations.
        Pediatrics. 2006; 117: 649-657
        • Weidner N.J.
        • Cameron M.
        • Lee R.C.
        • et al.
        End-of-life care for the dying child: what matters most to parents.
        J Palliat Care. 2011; 27: 279-286
        • Andresen E.M.
        • Seecharan G.A.
        • Toce S.S.
        Provider perceptions of child deaths.
        Arch Pediatr Adolesc Med. 2004; 158: 430-435
        • Malcolm C.
        • Knighting K.
        • Forbat L.
        • Kearney N.
        Prioritisation of future research topics for children's hospice care by its key stakeholders: a Delphi study.
        Palliat Med. 2009; 23: 398-405
        • Thienprayoon R.
        • Grossoehme D.
        • Humphrey L.
        • et al.
        "There's just no way to help, and they did." Parents name compassionate care as a new domain of quality in pediatric home-based hospice and palliative care.
        J Palliat Med. 2020; 23: 767-776
        • Thienprayoon R.
        • Mark M.S.J.
        • Grossoehme D.
        Provider-prioritized domains of quality in pediatric home-based hospice and palliative care: a study of the Ohio Pediatric Palliative Care and End-of-Life Network.
        J Palliat Med. 2018; 31: 290-296
        • Widger K.A.
        • Wilkins K.
        What are the key components of quality perinatal and pediatric end-of-life care? A literature review.
        J Palliat Care. 2004; 20: 105-112
        • National Consensus Project for Quality Palliative Care
        Clinical practice guidelines for quality palliative care.
        4th ed. National Coalition for Hospice and Palliative Care, Richmond, VA2018
        • Trevelyan E.G.
        • Robinson P.N.
        Delphi methodology in health research: how to do it?.
        Euro J Integr Med. 2015; 7: 423-428
      1. Boyden J, Ersek M, Deatrick J, et al. Developing a parent-reported measure of experiences with home-based pediatric palliative and hospice care: a multi-method, multi-stakeholder approach. In development. BMC Palliat Care.

        • Feudtner C.
        • Friebert S.
        • Jewell J.
        Pediatric palliative care and hospice care commitments, guidelines, and recommendations.
        Pediatrics. 2013; 132: 966-972
        • Field M.J.
        • Behrman R.E.
        When children die: Improving palliative and end-of-life care for children and their families.
        The National Academies Press, Washington, DC2003: 419-420
        • National Hospice and Palliative Care Organization (NHPCO)
        Standards of practice for pediatric palliative care.
        NHPCO, Alexandria, VA2019
        • Widger K.
        • Brennenstuhl S.
        • Duc J.
        • Tourangeau A.
        • Rapoport A.
        Factor structure of the Quality of Children's Palliative Care Instrument (QCPCI) when completed by parents of children with cancer.
        BMC Palliat Care. 2019; 18
      2. Courageous Parents Network.
        (Available from)
        https://courageousparentsnetwork.org/
        Date accessed: April 23, 2020
        • Gomes B.
        • de Brito M.
        • Sarmento V.P.
        • et al.
        Valuing attributes of home palliative care with service users: a pilot discrete choice experiment.
        J Pain Symptom Manage. 2017; 54: 973-985
        • Huynh E.
        • Coast J.
        • Rose J.
        • Kinghorn P.
        • Flynn T.
        Values for the ICECAP-Supportive Care Measure (ICECAP-SCM) for use in economic evaluation at end of life.
        Soc Sci Med. 2017; 189: 114-128
        • Mangham L.J.
        • Hanson K.
        • McPake B.
        How to do (or not to do) … designing a discrete choice experiment for application in a low-income country.
        Health Policy Plan. 2009; 24: 151-158
        • Ryan M.
        • Gerard K.
        • Amaya-Amaya M.
        Using discrete choice experiments to value health and health care.
        Springer, Dordrecht, The Netherlands2008
        • Feudtner C.
        • Walter J.K.
        • Faerber J.A.
        • et al.
        Good-parent beliefs of parents of seriously ill children.
        JAMA Pediatr. 2015; 169: 39-47
        • Sawtooth Software
        Maximum difference scaling: improved measures of importance and preference for segmentation.
        (Available from)
        • Louviere J.J.
        • Flynn T.N.
        • Carson R.T.
        Discrete choice experiments are not conjoint analysis.
        J Choice Model. 2010; 3: 57-72
        • Chrazan K.
        • Orme B.
        Applied MaxDiff: A practitioner's guide to best-worst scaling.
        Sawtooth Software, Inc., Provo, UT2019
        • Sawtooth Software
        Aggregate score estimation via logit analysis.
        (Available from)
        • Sawtooth Software
        The MaxDiff system technical paper.
        (Available from)
        • Agresti A.
        • Finlay B.
        Statistical methods for the social sciences.
        4th ed. Pearson Prentice Hall, Upper Saddle River, NJ2009
        • de Bekker-Grob E.W.
        • Donkers B.
        • Jonker M.F.
        • Stolk E.A.
        Sample size requirements for discrete-choice experiments in healthcare: a practical guide.
        Patient. 2015; 8: 373-384
        • Lancsar E.
        • Louviere J.
        Conducting discrete choice experiments to inform healthcare decision making.
        Pharmacoeconomics. 2008; 26: 661-677
        • Flynn T.N.
        • Louviere J.J.
        • Peters T.J.
        • Coast J.
        Best-worst scaling: what it can do for health care research and how to do it.
        J Health Econ. 2007; 26: 171-189
        • Jalmsell L.
        • Kreicbergs U.
        • Onelöv E.
        • Steineck G.
        • Henter J.-I.
        Symptoms affecting children with malignancies during the last month of life: a nationwide follow-up.
        Pediatrics. 2006; 117: 1314-1320
        • Rosenberg A.R.
        • Orellana L.
        • Ullrich C.
        • et al.
        Quality of life in children with advanced cancer: a report from the PediQUEST study.
        J Pain Symptom Manage. 2016; 52: 243-253
        • Schmidt P.
        • Otto M.
        • Hechler T.
        • et al.
        Did increased availability of pediatric palliative care lead to improved palliative care outcomes in children with cancer?.
        J Palliat Med. 2013; 16: 1034-1039
        • Wolfe J.
        • Grier H.E.
        • Klar N.
        Symptoms and suffering at the end of life in children with cancer.
        N Engl J Med. 2000; 342: 326-333
        • Kersun L.S.
        • Shemesh E.
        Depression and anxiety in children at the end of life.
        Pediatr Clin North Am. 2007; 54 (xi): 691-708
        • Theunissen J.M.
        • Hoogerbrugge P.M.
        • Achterberg T.V.
        • et al.
        Symptoms in the palliative phase of children with cancer.
        Pediatr Blood Cancer. 2007; 49: 160-165
        • Heyland D.K.
        • Dodek P.
        • Rocker G.
        • et al.
        What matters most in end-of-life care: perceptions of seriously ill patients and their family members.
        CMAJ. 2006; 174: 627-633
        • Namisango E.
        • Bristowe K.
        • Allsop M.J.
        • et al.
        Symptoms and concerns among children and young people with life-limiting and life-threatening conditions: a systematic review highlighting meaningful health outcomes.
        Patient. 2019; 12: 15-55
        • Stevenson M.
        • Achille M.
        • Lugasi T.
        Pediatric palliative care in Canada and the United States: a qualitative metasummary of the needs of patients and families.
        J Palliat Med. 2013; 16: 566-577
        • Vohra J.U.
        • Brazil K.
        • Hanna S.
        • Abelson J.
        Family perceptions of end-of-life care in long-term care facilities.
        J Palliat Care. 2004; 20: 297-302
        • Meyer E.C.
        • Burns J.P.
        • Griffith J.L.
        • Truog R.D.
        Parental perspectives on end-of-life care in the pediatric intensive care unit.
        Crit Care Med. 2002; 30: 226-231
        • Singer P.
        • Martin D.
        • Kelner M.
        Quality end-of-life care: patients' perspectives.
        JAMA. 1999; 281: 163-168
        • Thienprayoon R.
        • Marks E.
        • Funes M.
        • et al.
        Perceptions of the pediatric hospice experience among English- and Spanish-speaking families.
        J Palliat Med. 2016; 19: 30-41
        • Zheng N.T.
        • Li Q.
        • Hanson L.C.
        • et al.
        Nationwide quality of hospice care: findings from the Centers for Medicare & Medicaid Services hospice quality reporting program.
        J Pain Symptom Manage. 2018; 55: 427-432
        • Meeske K.A.
        • Patel S.K.
        • Palmer S.N.
        • Nelson M.B.
        • Parow A.M.
        Factors associated with health-related quality of life in pediatric cancer survivors.
        Pediatr Blood Cancer. 2007; 49: 298-305
        • Kreicbergs U.
        • Valdimarsdottir U.
        • Onelov E.
        • et al.
        Care-related distress: a nationwide study of parents who lost their child to cancer.
        J Clin Oncol. 2005; 23: 9162-9171
        • van der Geest I.M.M.
        • Darlington A.
        • Streng I.C.
        • et al.
        Parents' experiences of pediatric palliative care and the impact on long-term parental grief.
        J Pain Symptom Manage. 2014; 47: 1043-1053
        • Homer C.J.
        • Marino B.
        • Cleary P.D.
        • et al.
        Quality of care at a children's hospital: the parent's perspective.
        Arch Pediatr Adolesc Med. 1999; 153: 1123-1129
        • Cohen E.
        • Lacombe-Duncan A.
        • Spalding K.
        • et al.
        Integrated complex care coordination for children with medical complexity: a mixed-methods evaluation of tertiary care-community collaboration.
        BMC Health Serv Res. 2012; 12: 366
        • Cady R.G.
        • Belew J.L.J.C.
        Parent perspective on care coordination services for their child with medical complexity.
        Children (Basel). 2017; 4: 45
        • Baker J.N.
        • Levine D.R.
        • Hinds P.S.
        • et al.
        Research priorities in pediatric palliative care.
        J Pediatr. 2015; 167: 467-470
        • Lipkin P.
        • Alexander J.
        • Cartwright J.
        Care coordination in the medical home: integrating health and related systems of care for children with special health care needs.
        Pediatrics. 2005; 116: 1238-1244
        • Friedrichsdorf S.J.
        • Postier A.
        • Dreyfus J.
        • et al.
        Improved quality of life at end of life related to home-based palliative care in children with cancer.
        J Palliat Med. 2015; 18: 143-150
        • Bona K.
        • Bates J.
        • Wolfe J.
        Massachusetts' Pediatric Palliative Care Network: successful implementation of a novel state-funded pediatric palliative care program.
        J Palliat Med. 2011; 14: 1217-1223
        • Knapp C.A.
        • Madden V.L.
        • Curtis C.M.
        • et al.
        Partners in care: together for kids: Florida's model of pediatric palliative care.
        J Palliat Med. 2008; 11: 1212-1220
        • Ronis S.
        • Grossberg R.
        • Allen R.
        • Hertz A.
        • Kleinman L.
        Estimated nonreimbursed costs for care coordination for children with medical complexity.
        Pediatrics. 2019; 143: e20173562
        • Coller R.
        • Ehlenbach M.
        Making time to coordinate care for children with medical complexity.
        Pediatrics. 2019; 143: e20182958
        • Hexem K.R.
        • Mollen C.J.
        • Carroll K.W.
        • Lanctot D.A.
        • Feudtner C.
        How parents of children receiving pediatric palliative care use religion, spirituality, or life philosophy in tough times.
        J Palliat Med. 2011; 14: 39-44
        • Wiener L.
        • McConnell D.G.
        • Latella L.
        • Ludi E.
        Cultural and religious considerations in pediatric palliative care.
        Palliat Support Care. 2013; 11: 47-67
        • Davies B.
        • Brenner P.
        • Orloff S.
        • Sumner L.
        • Worden W.
        Addressing spirituality in pediatric hospice and palliative care.
        J Palliat Care. 2002; 18: 59-67
        • Davies B.
        • Contro N.
        • Larson J.
        • Widger K.
        Culturally-sensitive information-sharing in pediatric palliative care.
        Pediatrics. 2010; 125: e859-e865
        • Gaab E.
        • Owens G.
        • MacLeod R.
        Siblings caring for and about pediatric palliative care patients.
        J Palliat Med. 2014; 17: 62-67
        • Eaton Russell C.
        • Widger K.
        • Beaune L.
        • et al.
        Siblings' voices: a prospective investigation of experiences with a dying child.
        Death Stud. 2018; 42: 184-194
        • Verberne L.M.
        • Kars M.C.
        • Schouten-van Meeteren A.Y.
        • et al.
        Aims and tasks in parental caregiving for children receiving palliative care at home: a qualitative study.
        Eur J Pediatr. 2017; 176: 343-354
        • Mooney-Doyle K.
        • Deatrick J.A.
        Parenting in the face of childhood life-threatening conditions: the ordinary in the context of the extraordinary.
        Palliat Support Care. 2016; 14: 187-198
        • Hill D.L.
        • Miller V.A.
        • Hexem K.R.
        • et al.
        Problems and hopes perceived by mothers, fathers and physicians of children receiving palliative care.
        Health Expect. 2015; 18: 1052-1065
        • Hinds P.S.
        • Burghen E.A.
        • Pritchard M.
        Conducting end-of-life studies in pediatric oncology.
        West J Nurs Res. 2007; 29: 448-465
        • Macdonald M.E.
        • Chilibeck G.
        • Affleck W.
        • Cadell S.
        Gender imbalance in pediatric palliative care research samples.
        Palliat Med. 2010; 24: 435-444
        • Knapp C.A.
        • Madden V.L.
        • Curtis C.
        • Sloyer P.J.
        • Shenkman E.A.
        Assessing non-response bias in pediatric palliative care research.
        Palliat Med. 2010; 24: 340-347
        • Cohen L.L.
        Racial/ethnic disparities in hospice care: a systematic review.
        J Palliat Med. 2008; 11: 763-768
        • Gans D.
        • Hadler M.W.
        • Chen X.
        • et al.
        Impact of a pediatric palliative care program on the caregiver experience.
        J Hosp Palliat Nurs. 2015; 17: 559-565
        • Vollenbroich R.
        • Duroux A.
        • Grasser M.
        • et al.
        Effectiveness of a pediatric palliative home care team as experienced by parents and health care professionals.
        J Palliat Med. 2012; 15: 294-300