Pediatric end-of-life care in rural America: A systematic review

Abstract

Background:

Families increasingly desire to bring their children home from the acute care setting at end of life. This transition includes home to rural or remote areas. Little is known about the end-of-life care for children who reside in rural areas.

Objective:

The purpose of this study was to comprehensively review and summarize the evidence regarding end-of-life care for children living in rural areas, identify key findings and gaps in the literature, and make recommendations for future research.

Methods:

A systematic review was conducted from 2011 to 2021 using MEDLINE and CINAHL databases.

Results:

Nine studies met inclusion criteria. Key themes from the literature included: barriers, facilitators, and needs. Three articles identified barriers to end-of-life care for children in rural communities, which included access to end-of-life care and clinicians trained to provide pediatric care. Three studies identified and evaluated the facilitators of end-of-life care for rural children. The articles identified technology and additional training as facilitators. Four studies reported on the needs of rural children for end-of-life care with serious illness.

Conclusions:

We found major barriers and unmet needs in the delivery of rural pediatric end-of-life care. A few facilitators in delivery of this type of care were explored. Overall research in this area was sparse. Future studies should focus on understanding the complexities associated with delivery of pediatric end-of-life care in rural areas.

Children with serious illness are a growing population within the 40,000 children who die in the United States (US) each year.^1–3 Many of these children have multisystem diseases, technology dependence, and complex medication regimens that become more profound as they come to the end of their lives. Although most children spend their last days in an acute care setting, families increasingly desire to bring their children home at end of life.^4,5 The arrival home is typically recounted as a celebration and one which provides time with the child in the context of their family and friends. These experience bring comfort and a sense of fulfillment, and parents find the experience positive and meaningful.⁶ This transition includes home to rural or remote areas. The end-of-life care available for rural children can range from palliative care, concurrent hospice care, to traditional hospice care. These services offer expertise in pain and symptom management, availability for urgent needs and escalating end-of-life symptoms, and an array of family-centered services ranging from spiritual care to bereavement.

The World Health Organization and the American Academy of Pediatrics recognize the importance of end-of-life care extend to children living with chronic and life-limiting, terminal conditions.^7,8 However, there are challenges to accessing end-of-life care for children, especially for those residing in rural areas. We know that children and their families encounter a range issues related to the difficulty of prognosis of their pediatric serious illnesses, insufficient policy and resources, lack of health professionals with training in pediatric end-of-life care, lack of pediatric advance directives, and cultural/social beliefs related to pediatric death and dying.^9–13 We also know that many of these challenges are intensified when children reside in rural areas. For example, in areas of rural Appalachia many families hold the belief that hospice care hastens death.¹⁴ Consequently, a third of the 40,000 of children in the United States who die with a life-limiting illnesses access pediatric hospice care, and among rural children it is estimated that approximately 3,000 children/annually utilize hospice care.^15–17

Although there is emerging evidence about pediatric end-of-life care in rural areas, we lack a systematic review of the literature and information about what is known to guide future research and clinical practice. To that end, there is a gap in our knowledge about the current state of the science for rural children at the end of life. Therefore, the purpose of this article was to review the literature pertaining to end-of-life care for children who reside in rural areas. Specifically, we sought to comprehensively review and summarize the evidence regarding end-of-life care for children living in rural areas, identify key findings and gaps in the literature, and make recommendations for future research. The findings from this review contribute to existing knowledge on end-of-life care for children.

Methods

Search Strategy

Using available health care literature, we conducted a systematic review in accordance with the Preferred Reporting Items for Systematic Reviews and Meta-Analyses Statement (PRISMA) (Figure 1). MEDLINE and CINAHL were searched for peer-reviewed original research. The search strategy included a combination of Medical Subject Heading (i.e., MESH) terms and key words. The query included the terms: rural, pediatrics, children, adolescents, hospice, hospice care, palliative care, palliative, end-of-life care, end of life, and concurrent care. The search was limited to articles published between January 1, 2011 to January 31, 2021. We also conducted a hand search of reference lists to identity any articles not captured through the database searches and solicited additional citations through discussion with topic experts.

FIGURE 1:

PRISMA flowchart

Inclusion and Exclusion Criteria

Inclusion and exclusion criteria were used to refine the initial literature search. Studies were included if they were conducted in the US and pertained to pediatrics and rural care. We included qualitative and quantitative articles. Exclusion criteria were publication prior to 2011 and those that were not peer-reviewed. Studies written in a language other than English were excluded. Editorials, position papers, case studies, commentaries, expert reports, abstract, reviews, and dissertations were excluded. After duplicates were eliminated and inclusion and exclusion criteria applied, two reviewers independently screened the titles and abstracts to identify relevant articles.

Data Extraction

We used a modified tool developed for systematic reviews of end-of-life literature to extract key information from all included studies in a uniform way.¹⁸ Each member of our team independently extracted data from the articles and recorded the information on the data extraction tool. This information included: design, sample size, target population, purpose, location, definitions, study questions, interventions, measures, and results. The information extracted was reviewed collectively by all members of the team to validate accuracy, resolve disagreements, and build consensus.

Data Analysis

Information extracted from the selected articles was reviewed and analyzed for common themes and representative conclusions. An evidence assessment tool, Johns Hopkins Nursing Evidence-Based Practice Research Evidence Appraisal Tool, was used to rate the quality of the included articles within this review.¹⁹ This tool evaluated the level of evidence and quality of each research study. A quality rating was assigned based on whether the study was a randomized control trial (level I), quasi-experimental (level II), or non-experimental (level III) design. Additional qualifiers of high, good, or low quality was assigned based upon the study’s methodology. The quality assessments of each article were conducted by two team members. Any articles containing discrepancies were evaluated by a third member.

Results

Characteristics of Studies

The initial search strategy resulted in 269 citations from electronic databases and hand searches of the reference lists (Figure 1). One hundred eighty-nine citations were removed because they were duplicates. Based on screening the titles and abstracts, 54 citations were removed because they did not meet one or more of the screening criteria, leaving 26 for full-text screening. Following the full-text screening process, 17 were removed because they did not meet one or more exclusion criteria. The search for literature resulted in a final set of 9 articles for the review.

Included studies and their characteristics are listed in Table 1. The 9 included rural studies were published between 2012 and 2021. Four were published in the last 5 years.^10,20,21,23 Three studies were qualitative^14,20,21 and 6 were quantitative^10,22–26 designs. Rural study samples included hospice organizations,^{10,21–23,26} registered nurses,^10,20,21 pediatric patients,^20,22–25 and families/caregivers.^10,14,20 Data were collected via surveys,^10,20,26 and interviews,^14,21 along with administrative data.^22–25 The location of the rural studies varied from Tennessee,^14,22,24 Nebraska,^10,20,21 California,^{23, 25} and North Carolina.²⁶ For all articles in the review, the quality of the studies was assessed at a Level III - quality grade of B according to the Johns Hopkins Quality Assessment tool.¹⁹

TABLE 1:

Study characteristics of review publications (n = 9)

Authors	Purpose	Methods	Sample	Conclusions
Lindley and Edwards16	Describe the geographic distribution of pediatric hospice need and supply in rural and urban areas	Quantitative Design	119 pediatric decedents with cancer in Tennessee from 2009 to 2011 and 188 hospice providers	Rural counties surrounding Knoxville, Tennessee generally lacked pediatric hospice providers even though there was a need for care
Lindley and Edwards17	Describe the geographic distribution of pediatric hospice care need versus supply in rural and urban areas	Quantitative Design	2036 children and adolescent decedents and 136 pediatric hospice providers in California (2007–2010)	Distinctive geographic patterns of potential and realized need High density of potential and realized need in Los Angeles and high density of realized need in the San Diego area Supply of pediatric hospice care generally matched the needs of children and adolescents
Lindley and Oyana18	Investigate geographic variation by race in mortality among children and adolescents diagnosed with cancer in rural and urban areas	Quantitative Design	192 children and adolescents in Tennessee	African American children and adolescents with cancer in Tennessee were more likely than their counterparts of other races to reside in rural areas with close proximity to mortality clusters
Lindley and Shaw19	Examine the characteristics of children who use hospice care in rural and urban areas	Quantitative Design	76 pediatric decedents in California	California has 58 counties, and children who utilized hospice care resided in 21 of the counties A majority of children (96%) in the study resided in a county with a pediatric hospice provider 16 counties (28%), which were generally in major metropolitan areas, had multiple hospices that provided care for children
Mixer et al.11	Explore the culturally congruent end-of-life care needs of rural Appalachians	Qualitative Design	Six key informants and nine general informants in Tennessee	Lack of pediatric hospice services in rural Appalachia Faith is fundamental to rural Appalachians and their transition through end of life Family care at end of life is essential for culturally congruent care Integrating generic and professional care is essential for culturally congruent end-of-life care
Varela et al.20	Describe the status of pediatric hospice care and availability of services in rural and urban counties	Quantitative Design	Representatives of 61 hospice agencies in North Carolina	Families want to continue curative therapies while receiving hospice care services Lack of pediatric trained staff Lack of pediatrician consultation and referrals to end-of-life care Lack of pediatric pharmacy Inconsistent plan of care between pediatrician and hospice
Weaver et al.14	Determine acceptability of telehealth pediatric palliative care in rural settings	Qualitative Design	15 patients, 15 caregivers, and 15 nurses in Nebraska	Telehealth acceptability improved over time Acceptability was higher among hospice nurses All children “liked” telehealth Pediatric palliative telehealth visits partnered with in-person hospice nurse offer acceptable access to services, while extending support
Weaver et al.15	Understand the experiences of rural hospice nurses caring for children at the end of life using telehealth modalities to inform palliative communication	Qualitative Design	9 rural hospice agencies and 15 registered nurses in Nebraska	Telehealth may assist palliative care teams in honoring the communication needs of patients and families while striving to improve rural care access
Weaver et al.7	Develop a pediatric palliative care curriculum for inpatient nurses and adult-trained hospice teams caring for children in a rural region	Quantitative Design	180 respondents to needs assessment, 18 participants in pilot phase, and 31 participants in curricular expansion in Nebraska	Rural hospices previously unwilling to accept children enrolled pediatric patients in the 4 months following the training

Themes

The analysis of evidence revealed three themes: barriers, facilitators, and needs (Table 2). Among the studies included in this review studies, three articles^{14,22, 24,26} identified barriers to end-of-life care for children in rural communities. The most common barrier was a lack of pediatric providers.^14,22 Mixer and colleagues found that hospices in rural Appalachia had organizational policies prohibiting the admission of children,¹⁴ while Lindley and Edwards identified rural counties in Tennessee using geographic information systems (GIS) methodology that had no pediatric hospice providers.²² Other noted barriers were insufficient pediatric trained hospice staff and pediatric pharmacy resources.²⁶ Nineteen percent of hospices perceived the lack of pediatric pharmacy and consultation about pediatric dosaging as “very important” and “important” reason that hospices are unable to provide pediatric care.²⁶ Other noted barriers to rural care included racial disparities and underrepresented ages such as adolescents.^24,26 Thus, the hospices presented the major barriers to rural pediatric hospice care.

TABLE 2:

Major Themes

Major themes	Representative conclusions
Barriers	Families focus on curative therapies only Lack of knowledge about palliative care resources Lack of referrals (upstream clinician knowledge needed) Lack of access (no pediatric hospices available in rural settings) Racial disparities persist in the rural context Some ages (i.e., adolescents) underrepresented
Facilitators	Alternative approaches to end-of-life care delivery (i.e., telehealth) can foster improved symptom management and inclusive supportive care Locally developed, stakeholder-centric education can enable trust
Needs	Enhanced workforce to care for diverse children (pediatric-trained professionals, Spanish speaking professionals) Identification and matching of populations missing from comprehensive end-of-life care with optimal supportive care services Align care with policies and regulatory practices (i.e., advanced care planning)

Three studies^10,20,21 identified and evaluated the facilitators of end-of-life care for rural children. Two studies discussed telehealth as strategy to improve care for rural children.^20,21 Pediatric palliative telehealth care was found acceptable to children, hospice nurses, and caregivers,²⁰ and assisted palliative care teams in improving communication with rural children and their families.²¹ In addition, Weaver and colleagues found that clinical training for inpatient nurses and adult-trained hospice teams focused specifically on caring for children at end of life improved enrollment in pediatric hospice care after training.¹⁰ Technology and education were found to be critical facilitators of hospices providing rural pediatric hospice care.

Among the studies in the review, four studies reported on the needs of rural children for end-of-life care.^22–25 Using GIS methodology, two studies identified the geographic need for end-of-life care among children and adolescents, who died with advanced cancer in rural areas of Tennessee.^22,24 These studies examined the spatial distribution of children who died with a cancer diagnosis and identified rural hotspots were children would benefit from pediatric hospice care. The need for rural pediatric end-of-life care was identified by city- and county-level in California among pediatric decedents with a serious, life-limiting diagnosis.^23,25 These studies suggested that there was a consistent need for rural hospice care across the United States.

Discussion

We provided a review of the published literature on rural end-of-life care for children and found that there was sparse evidence with only nine studies specifically focused on this population. The lack of evidence for rural children at the end of life is consistent with other reviews of pediatric end-of-life literature (e.g., concurrent hospice care).²⁷ Although the studies conducted over the last decade have included a wide range of participants such as hospice, nurses, patients, and families/caregivers, they were limited in their geographic scope with a majority of studies conducted in the South. Additionally, the evidence produced from the studies in the review was of moderate quality and did not include any high-level evidence such as randomized control trials.

The review indicated that children in rural areas lack access to end-of-life care. Our findings were not consistent with adult hospice studies of access to hospice care. Carlson and colleagues examined access to hospice care for adults nationwide and found that as of 2008, 88% of the population lived in communities within 30 minutes and 98% lived in communities within 60 minutes of a hospice.²⁸ They reported that adults living in rural areas generally had access to hospice care. With the overall increase in the number of hospices and palliative care services across the United States during the past 10 years,^29,30 it was surprising to find that children did not have access to care, especially if they had a medical need for end-of-life care.^22–25 The literature in the review provided an explanation that organizational policies in the South sometimes did not allow hospices to admit pediatric patients.¹⁴ An alternative explanation was that the lack of pediatric trained clinical and pharmacy staff in the South limited hospice providers from admitting children.²⁶ These findings suggest that administrative policies and practices present in the organization may act as a barrier to end-of-life care and that interventions aimed at reducing these barriers should be focused on hospices in the southern region of the US.

The findings from our review on telehealth were interesting. Two recent studies in the review examined the use of telehealth in the context of care for rural children^20,21 The findings from these studies suggested that it was feasible to extend hospice care into rural regions and to enable end-of-life care to occur at home.¹⁰ Telehealth was found to be effective at identifying and treating physical symptom burden and especially psycho-logical symptoms experienced by children. It also enables hospice clinicians to extend support for rural family caregiver well-being.¹⁰ Telehealth combined with in-person visits by hospice nurses, offered access to services, while extending support to the child and family.³¹ The use of telehealth in rural pediatric hospice care may be an important factor in meeting the needs of rural children to accessible end-of-life care in the home. In addition, it may assist hospice and palliative care teams better honor the communication needs of patients and families while striving to improve care access.³²

These findings have implications for pediatric end-of-life research. First, given the dearth of existing studies, more research is needed to expand on the existing knowledge of barriers, facilitators, and needs in rural communities. Expansion of research should also extend the geographic scope of focus to different rural communities beyond North Carolina, Tennessee, Nebraska, and California. Research examining the national landscape are warranted. Second, more comparative studies will be needed to determine similarities and differences across rural regions. A better foundation of comparative studies might pave the way for development of policy solutions addressing rural communities’ needs. Finally, there is a need to expand the knowledge bases regarding facilitators aimed at pediatric end-of-life care delivery in rural areas. Information about the communication infrastructure available in the homes of rural children is limited. For example, we know that many parts of the US lack access to the internet, which is critical for the delivery of telehospice and palliative care. Innovations aimed at mitigating or resolving this infrastructure problem have not been developed or tested in the pediatric population. There are opportunities to expand our knowledge on facilitators of rural end-of-life care.

A clear understanding of rural end-of-life care for children has implications for clinical practice. Clinicians caring for children at end of life in rural communities may need additional training and resources to ensure quality care. Many educational resources focused specifically on children at end of life such as End of Life Nursing Education Consortium (ELNEC) are available in several locations across the US. There are also educational opportunities devoted to pediatric end-of-life care at hospice/palliative care professional meetings (e.g., American Academy of Hospice and Palliative Medicine/Hospice and Palliative Nurses Association Annual Assembly). In addition, clinicians might benefit from gaining a better understanding of technology and how it can be utilized for rural pediatric patients. Examples from countries such as Scotland where the nation’s pediatric hospice provider went entirely online at the onset of COVID-19 might provide examples and ideas for US clinicians.³³

Limitations

This review has several limitations. First, there is a possibility that some studies did not make the initial or final pool of articles for the evaluation due to the use of different search terms or the presence of other exclusion criteria. Second, personal bias in identification of major themes and interpretation of study conclusions are possible. In order to minimize the limitations, all authors met to discuss key words used within the search strategy as well as to brainstorm additional avenues through which relevant research could be identified. The authors also discussed resulting themes emerging from the literature and major conclusions to minimize potential bias. The study used a standardized tool to extract data from studies meeting the inclusion criteria. All authors met to discuss and come to consensus on the use of the tool and its results. In addition to analyzing data gathered from the study for major themes, the authors also rated the quality of studies found in this review by applying the Johns Hopkins Nursing Evidence-Based Practice Research Evidence Appraisal Tool. Finally, the study focused on rural pediatric end-of-life care and the findings cannot be generalized to children in urban areas. Although the scope of our study did not allow for us to examine access to urban care for children, future research might investigate the differences and similarities between rural and urban children with respect to barriers, facilitators, and need for end-of-life care.

Conclusion

This review found that there are major barriers and unmet needs in the delivery of rural pediatric end-of-life care in the US. Although a few facilitators in delivery of this type of care were explored, overall research in this area was sparse. Future studies should focus on understanding the complexities associated with the delivery of pediatric end-of-life care in rural areas as well as exploring the feasibility of interventions to overcome those challenges. Expanding this area of research will provide a foundation for the development of effective policy solutions that may address the needs of rural children and their families at end of life.