Parvaneh Vasli · Maryam Karami · Hanieh AsadiParvar-Masouleh

Abstract Background Providing access to pediatric palliative care (PPC) for children living with a serious illness, such as cancer, is of critical importance, although this specialized intervention, as a novel concept, still seems vague and complicated. The present study analyzed the concept of PPC for children with cancer. Methods Rodgers' evolutionary method was employed for the concept analysis. Articles on PPC, particularly those for children suffering from cancer, published between 2010 and 2021 were searched in valid academic research databases. The inclusion criteria for the full-text articles were based on the characteristics, antecedents, and consequences of PPC for children with cancer. Results In total, 19 relevant articles were selected and then reviewed and analyzed after applying the inclusion criteria and the final sampling. The analysis of the concept of PPC for children affected with cancer revealed four characteristics, including "PPC as holistic and integrated care", "PPC as patient- and family-centered care", "PPC as early-start continuous care", and "PPC as interdisciplinary and team-based care". Some effective factors could also act as antecedents for this concept, i.e., health care providers' training and expertise as well as human resources and financing. Moreover, improved quality of life, symptom reduction, and coordination between patient care and family support were among the PPC consequences for such children. Conclusions The study results demonstrated that delivering PPC to children with cancer demanded a comprehensive view of its various dimensions. Furthermore, numerous factors need to be delineated for its accurate and complete implementation.

Keywords Cancer · Children · Palliative care · Survivor

Introduction

Among vulnerable populations are children living with cancer who have their own complex needs [ 1]. Each year, an estimated 400,000 children and adolescents aged 0–19 years develop cancer [ 2]. According to the SEER (surveillance, epidemiology and end results) Cancer Statistics Review, during 2008–2012, the incidence rate among children and adolescents was further reported to be approximately 22.9 and 21.3 per 100,000 population of boys and girls, respectively [ 3]. The success rate of cancer treatment in childhood is also on an upward curve; however, approximately 20% of pediatric patients lose their lives due to this medical condition [ 4]. Additionally, survival rates among children and adolescents with cancer differ based on the disease type [ 5]. Despite recent advances in cancer treatment together with increased survival rates among such patients, this lifethreatening health problem in children can pose many challenges to their families [ 6]. Empirical research on children with cancer has accordingly revealed significant symptoms and psychosocial suffering [ 7, 8].

Of note, children are more tolerant and require more palliative care (PC) for longer periods than adults [ 9]. Pediatric conditions are also different from the more prevalent terminal illnesses in adults and may demand different plans of care, in particular more aggressive care services up to the end of life. Moreover, the length of illness varies widely in children because of their differential development and resilience, and end-of-life care can extend over many years through several developmental phases [ 10]. PC is thus known as a management approach to optimize quality of life (QoL) in patients (viz. adults and children) and their families, using prevention or relief for life-threatening illnesses [ 11]. In view of that, the early initiation of PC can minimize pain and augment health-related QoL [ 12]. As recommended by the American Academy of Pediatrics (AAP) in 2000, pediatric palliative care (PPC) should start upon the diagnosis of a life-threatening condition and continue throughout the illness, along with life-saving care methods or treatments, even at home. Along these lines, it is necessary to introduce novel patient- and family-centered care services as well as community-based PPC [ 10].

PPC is a family-centered, specialized medical care service for children with a serious illness, such as cancer, that helps in relieving physical, emotional, psychosocial, and spiritual suffering by providing interdisciplinary interventions that mostly focus on elevating QoL [ 7]. PPC was initially introduced as a standard of care in pediatric oncology by Weaver [ 13]. Of note, PPC is a much newer phenomenon compared with that for adults [ 14]. PPC also pursues similar goals for life-threatening conditions as those practiced for adults [ 15]; however, there are multiple differences between PPC and PC for adults, viz. (1) some diagnoses are child-based; (2) care centers are not child specific, and the whole family and parents are typically expected to become caregivers for children with complex needs; and (3) children continue to have physical, emotional, and cognitive growth despite the diagnosis of a life-limiting illness [ 16].

The related literature has thus far confirmed the necessity of research developments about PPC [ 17]. Although PPC studies are ongoing, they have focused on parental decisionmaking parameters, epidemiology, care models, ethical and legal considerations, symptom management, and complete family-based care methods [ 18]. Among the aspects required for this purpose, an explanation of the PPC concept is of utmost importance [ 19]. The clarity in the PPC definition also affects how it is described in all fields [ 20]. Moreover, this concept is newer than PC for adults [ 10]. Since its introduction in 1975, the term PC has undergone several semantic fl uctuations. For almost two decades, PC has been practiced to replace hospice and end-of-life or terminal care [ 21]. This specialized medical care is thus considered a concept with a wide variety of definitions in the literature [ 22]. Hence, providing a proper definition and placing some limitations on the nature and scope of PC are vital for integrating it into ongoing care, identifying human, financial, and physical resources to meet global needs, and filling the gaps of unfair access. More clarity in the concept of PC in turn shapes the scope of action, therapeutic objectives, and evaluation of results. However, conceptual ambiguity may hinder efforts for PC implementation and, as a result, create barriers to accessing global health care, particularly in low- and middleincome countries [ 23]. Research must thus focus much on the explicit use of such concepts [ 24].

To the best of the authors' knowledge, no study has previously analyzed the concept of PPC for children living with cancer. There are only some articles on the analysis of the concept of PC, e.g., "Concept analysis of palliative care using Rodgers' evolutionary method" [ 25], "Concept analysis of palliative care in nursing: introducing a hybrid model" [ 22], "A concept analysis of neonatal palliative care in nursing: introducing a dimensional analysis" [ 26], "A concept analysis of palliative care in the United States" [ 27], and "Pediatric palliative care: a conceptual analysis for pediatric nursing practice" [ 28].

PPC for children living with cancer needs to be explained since this concept has been vague and multifaceted, and there have been even overlaps among pediatric oncology, terminal care, hospice care, and PC, making PPC conceptualization much more difficult [ 19, 29, 30]. The terms PC and hospice care are often used incorrectly in place of one another [ 30]. In addition, similar definitions have been provided for them. The AAP suggests that PPC is a treatment to address the needs of children with serious life-threatening or inevitably life-shortening conditions and their families [ 31]. This statement is similar to the definition of hospice care in pediatrics that is provided in life-limiting conditions [ 32]. Contrary to this resemblance, some literatures have noted that hospice care is exclusively intended for people with a prognosis of 6 months or less, while PPC is not limited to the end of life [ 33]. In addition, end-of-life care has sometimes been employed interchangeably with PPC [ 34, 35].

Since one of the major barriers to implementing PPC in pediatric oncology patients is the definitions and the scope misconceptions about this type of intervention [ 30], the present study aimed to analyze the concept of PPC for children suffering from cancer using Rodgers' evolutionary method (REM) for concept analysis, as it could develop the classification or description of this phenomenon and provide the possibility of assessing its strengths and weaknesses [ 24].

Methods

In the present study, REM was applied to analyze the concept of PPC for children living with cancer. This could provide an inductive, dynamic, and fl exible approach to detecting PPC evolution and definition over time. Moreover, it could allow for the current consensus over the concept to be identified using an accurate analysis. The most important outcome was identifying a strong conceptual basis for the development of future knowledge and providing a basis for continuous communication and understanding [ 36].

In this study, REM was implemented in six steps: (1) identifying the concept of interest and its associated expressions (including surrogate terms); (2) recognizing and selecting the appropriate realm for data collection; (3) collecting the relevant data to identify the attributes and contextual bases of the concept; (4) analyzing the data to ascertain the concept characteristics; and (5) developing a model case (viz. an exemplary); and (6) finding the implications and hypotheses for the further development of the concept [ 36].

Identifying the concept of interest and its associated expressions (including surrogate terms)

Reviewing the related literature, consulting with the research team, and browsing the Medical Subject Headings (MeSH) database ( https:// www. ncbi. nlm. nih. gov/ mesh ), all the keywords and phrases associated with the concept of PPC for children with cancer, i.e., "oncology, cancer, children, pediatric, and palliative care", using AND and OR in both English and Persian, were identified during the first step (Supplementary Table 1).

Recognizing and selecting the appropriate realm for data collection

In the present study, the fields of nursing, medicine, psychology, and social work were the areas of data collection. The search was also carried out in several phases. Accordingly, English articles published from 2010 to 2021 were first searched in the Scopus, ProQuest, Elsevier, Cochrane, PubMed, Google Scholar, and Ovid databases. The inclusion criteria for the full-text articles were also based on the characteristics, antecedents, and consequences of PPC for children with cancer and being published in English. It is notable that Persian articles were searched in the Scientific Information Database and Magiran.

Collecting the relevant data to identify the attributes and contextual bases of the concept

In the initial search, a total of 325 articles were retrieved. The number of duplicates also reached 205 upon observing the inclusion and exclusion criteria, which were mostly related to the research topic. After a complete review at the final stage, the number of articles published in the mentioned period reached 48. The preliminarily selected articles were then analyzed to answer questions such as "Is there any information about the characteristics of the concept of PPC in children with cancer?" and "Is there any information on the consequences of PPC in children with cancer?". If the answer was positive, that article was reviewed as one in the study-related topic. In total, 19 articles were included in the conceptual analysis after removing irrelevant articles (Fig. 1).

Fig. 1 Flow diagram of the study. PPC pediatric palliative care

Analyzing the data to ascertain the concept characteristics

After data collection, data analysis was carried out to extract the true characteristics of PPC for children living with cancer with no bias and premature data saturation from the dataset. Upon selecting the relevant documents, they were adjusted according to the year of publication to determine the changes in the definition of PPC for children with cancer. Then, each article was read in its entirety to find the characteristics, antecedents, and consequences. Here, the characteristics referred to the attributes that constituted the definition of PPC for children with cancer. The identification of the antecedents could also help define the attributes or events emerging prior to the concept embodiment. Additionally, the consequences denoted the incidents or events that emerged and were related to the concept [ 37].

This study was an attempt to address the following questions: "What conditions are needed to implement PPC for children with cancer?" (antecedents), "What are the PPC characteristics of children with cancer?" (features or attributes), and "What are the consequences of implementing PPC for children with cancer?" (consequences). Next, suitable semantic units were identified to answer these questions and record them in three different files, viz. characteristics, antecedents, and consequences. The existing data in each file were also analyzed separately by Graneheim and Lundman's qualitative content analysis. In this line, there were attempts to discover the patterns in the data and allow the main themes to emerge from them. In this regard, the selected articles were read several times to extract the codes. Then, the semantic units were identified, and the initial codes were established. Afterward, considering the similarities and differences, the codes were categorized into several subcategories, which then, with the increase in codes, the subcategories formed categories based on the similarities and differences [ 38].

Results

The study results are provided in the form of some current definitions of PPC (Supplementary Table 2), along with the characteristics, antecedents, and consequences of this specialized intervention for children suffering from cancer (Table 1) and PC-related terms. A summary of the characteristics, antecedents, and consequences of PPC for children with cancer is given in Supplementary Table 3 and Fig. 2.

Fig. 2 Antecedents, characteristics and consequences of pediatric palliative care for children with cancer. PPC pediatric palliative care, ICU intensive care unit

Current definitions of pediatric palliative care

PPC provides a great opportunity to care not only for physical symptoms but also for the impact of illnesses on children's psychological, developmental, and spiritual health in the social context of their family and community [ 8]. There is no specific definition of PPC for children with cancer in the literature; however, several definitions of PPC have been offered in general, some of which are listed in Supplementary Table 2.

Characteristics of pediatric palliative care for children with cancer

Based on the concept analysis results, PPC for children living with cancer was summarized into four general characteristics: (1) PPC as holistic and integrated care; (2) PPC as patient- and family-centered care; (3) PPC as early-start and continuous care; and (4) PPC as interdisciplinary and team-based care, discussed below (Table 1).

Pediatric palliative care as holistic and integrated care

Based on the definition released by the World Health Organization (WHO), PPC refers to the comprehensive care for the mind, body, and soul in children [ 30], incorporating the physical, emotional, and spiritual aspects of care [ 48]. Various aspects of this type of care are symptom surveying and management, pain control and complex symptom management, mental health care, psychological and social support, care orientation, spiritual considerations, and family support [ 8, 51, 52]. The early integration of PPC for children with life-threatening disease can thus enhance holistic care provision, address psychological, social, spiritual, and physical concerns, and even prevent treatment with no cure goals [ 46].

Pediatric palliative care as patient- and family-centered care

One of the peculiar aspects of PPC, particularly for children with cancer, is its simultaneous attention to children and their families [ 7, 26]. PPC can thus be defined as a specialized care service and an approach to the care of a pediatric oncology patient, which encompasses child and family care [ 8]. This type of care is unique since it takes into account the legal issues of adults, including factors such as the involvement of children in medical decision-making and the ethical and legal considerations when children are affected with a serious illness. In other words, children must be respected with regard to ethical and legal considerations as adults in medical decisionmaking when they become seriously ill [ 33]. PPC for children and adolescents with cancer also means supporting all individuals and families, regardless of the path to disease prognosis. In this type of care, compassionate and honest communication with patients and their families along with much attention to their preferences is one of the principles [ 8]. Therefore, the siblings of sick children must be considered [ 52].

Pediatric palliative care as early-start and continuous care

PPC for children living with cancer is a continuous process that must start immediately after diagnosis, regardless of specific treatments [ 30, 48, 54], and then sustained to outpatient care [ 46]. PPC may also be provided regularly in mobile clinics, outpatient centers, community health centers, and intensive care units (ICUs) [ 22]. The early integration of PPC into oncology care has thus been shown to reduce symptoms and improve outcomes by providing patient- and family-centered care experiences [ 1].

Pediatric palliative care as interdisciplinary and team-based care

PPC, as an interdisciplinary and evidence-based service, addresses the needs of infants, children, and adolescents living with life-threatening or life-limiting conditions, along with those of both families and caregivers. The treatments here are to alleviate physical, psychosocial, spiritual, and practical suffering. Interdisciplinary teams accordingly focus on the role of families in improving children's QoL to cope with diseases and grief and consequently facilitate informed decision-making and more coordination [ 45]. PPC can thus expedite the achievement of outcomes, such as boosting QoL to alleviate physical, emotional, and mental suffering in children with serious illnesses by providing interdisciplinary services [ 7].

Palliative care-related terms in cancer

Several synonyms or equivalents of PC are often used during cancer treatment, including supportive, end-oflife, and hospice care. Of note, PC was originally called "end-of-life care"; however, it is now regarded as a treatment option for relieving emotional and physical suffering, which may occur at any stage of the disease [ 56]. Here, supportive care is defined as "the provision of the necessary services for those living with or affected by cancer to meet their informational, emotional, spiritual, social or physical needs during their diagnostic treatment or followup phases, encompassing the issues of health promotion, prevention, survivorship, palliation, and bereavement" [ 11]. In view of this, PC is the subset of supportive care and is further described by the WHO as"an approach that improves the quality of life of patients (adults and children) and their families who are facing problems associated with life-threatening illness. It prevents and relieves suffering through the early identification, correct assessment and treatment of pain and other problems, whether physical, psychosocial or spiritual" [ 57].

In many cases, PC is utilized synonymously with end-oflife or terminal and hospice care, which can result in misconceptions [ 58]. For instance, the National Hospice and Palliative Care Organization also utilized the term PPC in addition to hospice care in its definition of PPC as follows: "Pediatric palliative and/or hospice care is a philosophy and an organized method for delivering individualized care to children with life-threatening conditions. This care focuses on enhancing quality of life for the child and family, preventing and/or minimizing suffering, optimizing function, and providing opportunities for personal and spiritual growth. As such, it can be provided along with concurrent life-prolonging or curative care or as the focus of care …" [ 43]. In some documents, the definition of hospice care as a subset of supportive care is as follows: "Hospice care is a form of community-based palliative care predominantly serving patients and their loved ones at the end of life" [ 11].

Antecedents of pediatric palliative care for children with cancer

The study results indicated that the antecedents, or in fact, the PPC prerequisites for children with cancer, included a variety of care models, viz. holistic and integrated, patient- and family-centered, interdisciplinary and team-based, and early-start and continuous. Some antecedents were clarity in PPC philosophy [ 30], codification and implementation of models for PPC approval [ 46], codification of an interdisciplinary educational curriculum [ 1], PPC training to health care providers (HCPs) [ 1, 46, 51, 53], effective use of resources to achieve child and family care standards [ 8], adequate knowledge about PPC [ 48], and care provision by an individual or a team of qualified and trained individuals [ 46]. Additionally, sufficient funding, PPC programs, and human resources at the health system level, PC integration into existing child oncology caregiver models at the organizational level, ample knowledge and skills about PPC, cultural competence in HCPs [ 33], confirmation and acceptance of the child's disease by parents, and nurses' awareness of services [ 30] were of utmost importance. The existence of PPC teams, no intersection in the description of the duties of this team and oncologists, families' readiness for accepting an incurable disease, sufficient time, agreement between HCPs and families about PPC objectives, and PC delivery in the form of teamwork [ 7] were among other antecedents. Moreover, adequate financial resources, PPC training, awareness of politics and policymaking, acceptable psychological and professional supervision of the staff, manpower shortages, satisfactory hospital infrastructure, a harmony of real and emotional knowledge between parents and HCPs, attention to cultural aspects [ 53], and integration of scheduled PPC in oncology clinics into home PC [ 55] were highlighted.

Consequences of pediatric palliative care for children with cancer

The present study suggested that PPC for children living with cancer generally had many positive effects, including improved QoL in children with cancer and their family members [ 8, 13, 30, 33, 51, 52, 54, 55] using the management of physical symptoms [ 33, 45, 54, 55], disease perceptions, satisfaction with care and overall survival [ 50], reduction of emotional distress and suffering, better coordination and communication between families and HCPs [ 14], lower parental stress [ 45], and an increment in care coordination [ 13, 33, 54]. Additionally, patient and family support [ 13, 30, 33], higher life expectancy, prolonged life, pain surveying and relief, lower ICU death rate [ 33, 54], more comfort and meaning through bereavement care [ 7], treatment cost reduction, lower hospitalization and emergency visits [ 1], and less utilization of health care, chemotherapy, and intensive care resources at different stages of problem-solving and decision-making [ 13] are important.

Discussion

Implementing REM, this study aimed to analyze the concept of PPC for children living with cancer. The study results indicated that the given concept had four main characteristics, viz. "PPC as holistic and integrated care", "PPC as patient- and family-centered care", "PPC as early-start and continuous care", and "PPC as interdisciplinary and teambased care".

Regarding the first characteristic, Meghani obtained similar results in a conceptual analysis of PC in the United States, wherein individualized patient care, family support, and open communication had been identified [ 27]. In Stayer’s study, PPC was analyzed using the approach developed by Walker and Avant, and care comprehensiveness was reported as one of its characteristics [ 28]. In a qualitative study on PPC providers' perceptions in Switzerland, the participants noted the comprehensiveness of this type of specialized medical care [ 21]. In the analysis of the concept of PC in nursing, exploiting a hybrid method, in Iran, holistic care had been similarly refl ected as one of its major characteristics [ 22].

Another characteristic of PPC highlighted in the present study was patient- and family-centeredness, as reported in Beasley et al.’s study, refl ecting on the evolutionary and conceptual foundations of nonhospice PC [ 59]. Moreover, Meghani delineated three characteristics of PC in the United States, viz. individualized patient care, family support, and open communication are elements of patient- and family-centered care [ 27]. However, the term patient- and family-centered care had not been directly raised as one of the characteristics of the PPC concept [ 28], but the need for open and effective communication in addition to individualized care had been identified as two of its characteristics, which could be considered equivalent to patient- and family-centered care.

The third characteristic of PPC was early-start and continuous care. In the vein of the aforementioned findings, there were two characteristics of nonhospice PC, viz. early intervention at any stage in illness in Beasley et al.’s study [ 59], which might be equivalent to early continuous care. Furthermore, Labudde et al. found in their action research that identifying a soothing opportunity for the early start of PPC for children affected with leukemia and lymphoma could reduce their suffering [ 60]. Accordingly, academic associations working in the field of pediatric oncology have emphasized that children with cancer must receive PC as soon as possible in a timely manner [ 61]. The early initiation of PPC could thus help in decision-making and identification of care objectives, which meet the needs of families, and consequently, children can even be treated during the course of the disease [ 20].

The fourth characteristic of PPC was interdisciplinary and team-based care. In Meghani’s study, one of the characteristics of PC in the United States was reported as interdisciplinary teamwork [ 27]. According to the AAP, "all general and subspecialty pediatricians, family physicians, pain specialists, and pediatric surgeons need to become familiar and comfortable with the provision of PC to children" [ 56]. PPC also provides a program in which members from various specialties cooperate with the patient's family [ 62]. Such care is recognized as a distinct specialty that includes multidisciplinary teams working in a variety of organizations, such as hospitals, community health centers, and children's hostels [ 62, 63].

In conclusion, the present study aimed to explain PPC for children living with cancer using REM, suggesting that the aforementioned concept had four main characteristics, i.e., PPC as holistic and integrated care, PPC as patient- and family-centered care, PPC as early-start and continuous care, and PPC as interdisciplinary and teambased care. Furthermore, some antecedents and consequences were identified. HCPs and administrators in PPC centers for children with cancer can thus make use of the study results for improving this type of care. Having a comprehensive view of PPC for children living with cancer, family involvement, early start and continuation during treatment, interdisciplinary practice and teamwork, codification of training curricula for specialist training, funding and manpower, and codification and implementation of PPC models for children with cancer are some of the main measures that can have impacts on the optimal implementation of PPC for such children.

Supplementary InformationThe online version contains supplementary material available at https:// doi. org/ 10. 1007/ s12519- 022- 00600-x.

AcknowledgementsWe would like to thank Kazem Najafifor cooperating in the research process.

Author contributionsVP contributed to conceptualization, analysis and interpretation of data, project administration, and final approval of the manuscript. KM contributed to conceptualization, analysis and interpretation of data, and writing of the original draft. AMH contributed to final approval of the manuscript.

FundingNone.

Data availabilityThe datasets generated during and/or analyzed during the current study are available from the corresponding author on reasonable request.

Declarations

Ethical approvalNot applicable.

Conflict of interestNo financial or non-financial benefits have been received or will be received from any party related directly or indirectly to the subject of this article. The authors have no confl ict of interest to declare.