Parent values and preferences underpinning treatment decision-making in poor-prognosis childhood cancer: A scoping review

doi:10.21203/rs.3.rs-1450408/v1

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Parent values and preferences underpinning treatment decision-making in poor-prognosis childhood cancer: A scoping review

https://doi.org/10.21203/rs.3.rs-1450408/v1

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Background: Parents of children who are diagnosed with a poor-prognosis cancer want to be involved in making treatment-related decisions for their child. They often make repeated decisions depending on their child’s response to treatment and can experience decisional regret as a consequence. Understanding parent values and preferences when making treatment-related decisions may help enhance discussions with healthcare professionals and identify additional ways of providing support to this parent population.

Objectives: To explore parent values and preferences underpinning treatment decision-making for children receiving cancer-directed therapy for a poor prognosis cancer.

Methods: A scoping review of research literature and systematic reviews from qualitative, quantitative, and mixed methods studies was conducted following Joanna Briggs Institute methodology. Articles which included parents of a child who received cancer-directed therapy for a poor-prognosis childhood cancer, under the age of eighteen years were considered. Four electronic databases were searched (CINAHL, Medline, PsychINFO, Web of Science Core Collections). Reference and citation lists of all included full-text articles were also searched. Summative content analysis was used to synthesise findings and develop themes.

Results: Twelve articles were included. Parent decision-making was affected by underpinning factors: hope for a cure, fear of their child dying and uncertainty. Influencing factors: opinions of others, child’s wishes, and faith and religion had the potential to inform decision-making processes. Parents valued having enough time, being a good parent and being involved in decision-making. Preferences within these values varied resulting in the potential for conflict and ‘trade-offs’ in making decisions.

Conclusions: Parent decision-making in poor-prognosis childhood cancer is complex and extends beyond values and preferences. Underpinning factors and values are consistent through the decision-making process with influencing factors and preferences varying between parents. Preferences can conflict when parents want to continue cancer-directed therapy whilst maintaining their child’s quality of life or can change depending on a parents’ cognitive state as they realise cure might be unlikely.

Cancer

Child

Decision-making

Parent

Poor-Prognosis

Approximately 400 000 children and young people aged 0–19 years are diagnosed with cancer each year globally [1]. Of these, in the United Kingdom, approximately 1,645 children are aged 0–14 years when diagnosed [2]. In 2018, approximately 260 children died, accounting for 7% of childhood deaths in the 0–14 population that year [2]. Despite advances in research and treatment, there remains a small cluster of difficult to treat poor-prognosis childhood cancers. These include relapse neuroblastoma, medulloblastoma, hepatocellular carcinoma and peripheral T cell lymphoma.

Over the last decade, there has been an increase in clinical trials and treatment options for children diagnosed with poor-prognosis cancers, with the implementation of targeted inhibitors and immunotherapy treatments [3, 4]. Due to the potential options now available, parents often make multiple repeated treatment decisions as their child’s condition changes and relapses occur. Multiple relapses can often result in a poor-prognosis with an increased likelihood of death [5]. There is a need to understand individual parent values to support decision-making when their child has multiple cancer relapses [6].

Treatment Decision-Making

Parents want to be involved in making treatment-related decisions [7]. Typically, parent involvement in treatment decision-making happens when there is no standard of care treatment protocol, in situations where there is either disease progression, relapse, or failure to respond to treatment [8]. This situation is dependent on the diagnosis and the availability of standard of care protocols at these timepoints. Standard of care treatments, include those that are shown to be the best available for a particular disease, proven through clinical trials [9]. At this timepoint, treatment is guided by healthcare professionals based on the best available standard of care treatment [10]. Parents are not involved in treatment decision-making at this point. When there is no standard of care treatment, parents can be offered various treatment options and become involved in making treatment-related decisions. Decision-making at this point should be informed by parent values and preferences [10].

Values and preferences

Treatment decision-making is based on parents’ individual values and preferences which are underpinned by quality information on the risks and benefits of the treatment options, treatment expectations and cure [7]. Key components of quality information include diagnosis and treatment information, expectations of treatment, potential short and long-term side effects, and their child’s ongoing response to treatment [11]. This information needs to be explicit and provided by healthcare professionals in an open, accurate and clear way [12]. Research has shown the importance of clear prognostic information and the impact this can have on parent decision-making [13]. Understanding parent values and preferences can empower parents, increase their confidence in decision-making [14] and reduce the potential for decisional conflict and regret [15].

Research has acknowledged parents’ pursuit to continue cancer-directed therapies even when the chance of cure is minimal [16, 17]. Parent preferences for cancer-directed therapies in phase I clinical trials, or at the end-of-life, include the need to prolong life, minimise suffering [7], and know they have done everything possible to save their child’s life [16, 18]. Parents are reported to be overly hopeful and optimistic for the likelihood of cure which can result in parents making inadequate or inappropriate treatment decisions [12] which can impact on the child’s quality of life and suffering.

Decisional Regret

Decisional regret can have a lasting impact on parents particularly if their child dies [8.15,17]. One study found most parents regretted not pursuing further cancer-directed therapy or exploring alternative treatment options for their child [15]. Equally some parents regretted not discontinuing cancer-directed therapy earlier when this provided little or no benefit to their child [15]. This suggests confliction in parent treatment decision-making. Regret can result in the potential for prolonged grieving, be more intense and longer lasting, when compared with other types of grief [19].

Given parents want to be involved in making treatment decisions yet they experience decisional regret particularly when their child has a poor-prognosis cancer, this warrants exploration of the concepts of values and preferences which inform their treatment decision-making. Values and preferences are defined as the goals, expectations, predispositions, and beliefs an individual holds when making a decision [20]. Understanding these values and preferences could enhance discussions with healthcare professionals and identify additional ways of providing support to this parent population.

The objective of this scoping review was to explore parent values and preferences underpinning treatment decision-making when their child was receiving cancer-directed therapy for a poor prognosis cancer.

This review allowed for a broad scope of the literature to answer the research questions. It was conducted following the Joanna Briggs Institute (JBI) methodology [21]. For transparency, the objectives, methods, and inclusion criteria were specified in advance. This review was registered on 2nd June 2020 with the Open Science Framework (https://osf.io/n7j9f) and the protocol published in BMJ Open [22].

The review process followed seven different stages: (a) defining the research questions, (b) eligibility criteria, (c) search strategy, (d) evidence screening and selection, (e) data extraction, (f) analysis of the evidence, and (g) presentation of the results [21]. Given the sensitivity of the scoping review topic, the authors included a consultation phase with a parent and carer group who have experienced making treatment decisions for their child with a poor-prognosis cancer. This mirrors the optional consultation phase suggested in the Arksey and O’Malley scoping review framework [22].

Research Questions

The research questions were, 1) what are parent values and preferences when their child is receiving cancer-directed therapy for a poor prognosis cancer?; 2) how do these values and preferences inform treatment decision-making?

Eligibility Criteria

Included was research literature and systematic reviews from qualitative, quantitative, and mixed methods studies. Parents of a child who had received cancer-directed therapy for a poor-prognosis cancer and the child was under the age of 18 years. Cancer-directed therapy was defined as any type of cancer treatment which could be received with or without palliative care or symptom management simultaneously.

Literature could encompass any clinical, medical or homecare setting. Articles were published in English, from any country with the full abstract available from 1996 until the time each database was searched.

Search Strategy

The databases searched included CINAHL, Medline and PsycINFO using the EBSCO platform and Web of Science Core Collections using the Clarivate platform. An initial search was conducted in April 2020 on CINHAL and Medline. Relevant literature was found which supported undertaking a full scoping review. All databases were searched in May 2021. For completeness, the Web of Science was used to screen the reference and citation lists of all included full-text articles.

Keywords were searched for subject headings/Medical Subject Headings (MeSH) for full exploration of academic language associated with each word (Table 1). Phrases were not searched as these were natural language phrases for example “no realistic chance of cure” and “cancer directed-therapy”. Adjacency, truncation, and the wildcard search symbols provided full exploration of words. Boolean operators (AND/OR) were used. For combining keywords, subject headings, phrases, and MeSH terms the Boolean operator OR was used. When combining the main topics (cancer, child, decision-making, parent, poor prognosis, values, and preferences) the Boolean operator AND was used. Full search strategies are shown in Table 2.

Table 1

Keywords and phrases searched
Keywords	Subject Headings/MeSH terms and phrases
Decision Making	“Decision making”
Cancer	Malignancy; Neoplasm; Oncology; “cancer-direct* therap*”
Child	Paediatric; Pediatric
Parent	Family
Poor Prognosis	Advanced Cancer; Deteriorate; Disease Progression; Incurable; Recurrent; Refractory; Relapse; Uncertainty; “advance* cancer”; “no realistic chance of cure”; “disease* progress”; “palliat chemotherap*”; “no reasonable chance of cure”
Value	Attitude; Belief; Choice; Choose; Expectation; Influence; Predisposition; Preference; Perception; “goal* of care”

The University Research Librarian peer-reviewed the search strategy in each database for transparency and robustness. No amendments to search strategies were required.

Table 2

Full database search strategies
Database & Records Retrieved	Search Strategy
Medline Search Strategy (N = 218)	S1 parent* S2 mother* S3 father* S4 famil* S5 S1 OR S2 OR S3 OR S4 S6 value* S7 (MH "Value of Life") OR (MH "Social Values") S8 preference* S9 "goal* of care" S10 choice* S11 belief* S12 (MH "Social Norms") S13 attitude* S14 expect* S15 (MH "Motivation") S16 predisposition* S17 influenc* S18 experienc* S19 (MH "Clinical Decision-Making") S20 choose* S21 percept* S22 S6 OR S7 OR S8 OR S9 OR S10 OR S11 OR S12 OR S13 OR S14 OR S15 OR S16 OR S17 OR S18 OR S19 OR S20 OR S21 S23 "decision making" S24 (MH "Decision Making, Shared") S25 decision N2 making S26 decision* S27 decide* S28 S23 OR S24 OR S25 OR S26 OR S27 S29 S5 AND S22 AND S28 S30 "poor prognosis" S31 relaps* S32 (MH "Recurrence") S33 recurrenc* S34 refractory S35 incurable S36 (MH "Terminally Ill") S37 "advanc* cancer" S38 "no realistic chance of cure" S39 uncertain* S40 deteriorat* S41 (MH "Clinical Deterioration") S42 "disease* progress" S43 "palliat chemotherap" S44 "no reasonable chance of cure" S45 S30 OR S31 OR S32 OR S33 OR S34 OR S35 OR S36 OR S37 OR S38 OR S39 OR S40 OR S41 OR S42 OR S43 OR S44 S46 child S47 p#ediatric* S48 S46 OR S47 S49 cancer S50 neoplasm* S51 (MH "Neoplasm Recurrence, Local") S52 malignan* S53 (MH "Neoplasms") S54 "cancer direct* therap" S55 oncolog S56 S49 OR S50 OR S51 OR S52 OR S53 OR S54 OR S55 S57 S45 AND S48 AND S56 S58 S29 AND S57
CINAHL Search Strategy (N = 115)	S1 parent* S2 mother* S3 father* S4 famil* S5 S1 OR S2 OR S3 OR S4 S6 value* S7 (MH "Social Values") OR (MH "Values Clarification") S8 preference* S9 "goal* of care" S10 choice* S11 belief* S12 (MH "Health Beliefs") OR (MH "Attitude to Illness") S13 attitude* S14 expect* S15 predisposition* S16 influenc* S17 experienc* S18 choose* S19 percept* S20 S6 OR S7 OR S8 OR S9 OR S10 OR S11 OR S12 OR S13 OR S14 OR S15 OR S16 OR S17 OR S18 OR S19 S21 "decision making" S22 (MH "Decision Making, Shared") OR (MH "Decision Making, Clinical") S23 decision N2 making S24 decision* S25 decide* S26 S21 OR S22 OR S23 OR S24 OR S25 S27 S5 AND S20 AND S26 S28 "poor prognosis" S29 relaps* S30 (MH "Recurrence") S31 recurrenc* S32 refractory S33 incurable S34 "advanc* cancer" S35 "no realistic chance of cure" S36 uncertain* S37 deteriorat* S38 (MH "Clinical Deterioration") S39 "disease* progress" S40 "palliat chemotherap" S41 "no reasonable chance of cure" S42 S28 OR S29 OR S31 OR S32 OR S33 OR S34 OR S35 OR S36 OR S37 OR S38 OR S39 OR S40 OR S41 S43 child S44 p#ediatric S45 S43 OR S44 S46 cancer S47 neoplasm* S48 malignan* S49 "cancer direct* therap" S50 oncolog S51 S46 OR S47 OR S48 OR S49 OR S50 S52 S42 AND S45 AND S51 S53 S27 AND S52
APA PsycInfo Search Strategy (N = 91)	S1 parent* S2 mother* S3 father* S4 famil* S5 S1 OR S2 OR S3 OR S4 S6 value* S7 DE "Social Values" OR DE "Personal Values" S8 preference* S9 "goal* of care" S10 choice* S11 DE "Uncertainty" S12 belief* S13 DE "Attitudes" S14 attitude* S15 DE "Parental Attitudes" OR DE "Parental Role" S16 expect* S17 predisposition* S18 influenc* S19 DE "Personal Values" OR DE "Social Influences" S20 experienc* S21 choose* S22 percept* S23 S6 OR S7 OR S8 OR S9 OR S10 OR S11 OR S12 OR S13 OR S14 OR S15 OR S16 OR S17 OR S18 OR S19 OR S20 OR S21 OR S22 S24 "decision making" S25 DE "Choice Behavior" S26 decision N2 making S27 decision* S28 decide* S29 S24 OR S25 OR S26 OR S27 OR S28 S30 S5 AND S23 AND S29 S31 "poor prognosis" S32 DE "Disease Progression" S33 relaps* S34 recurrenc* S35 refractory S36 incurable S37 "advanc* cancer" S38 DE "Terminal Cancer" S39 "no realistic chance of cure" S40 uncertain* S41 deteriorat* S42 "disease* progress" S43 "palliat chemotherap" S44 "no reasonable chance of cure" S45 S31 OR S32 OR S33 OR S34 OR S35 OR S36 OR S37 OR S38 OR S39 OR S40 OR S41 OR S42 OR S43 OR S44 S46 child S47 p#ediatric* S48 S46 OR S47 S49 cancer S50 DE "Neoplasms" S51 neoplasm* S52 DE "Oncology" S53 malignan* S54 DE "Neoplasms" OR DE "Oncology" S55 "cancer direct* therap" S56 oncolog S57 S49 OR S50 OR S51 OR S52 OR S53 OR S54 OR S55 OR S56 S58 S45 AND S48 AND S57 S59 S30 AND S58
Web of Science Core Collections Search Strategy (N = 149)	# 1 TOPIC: (parent* OR mother* OR father* OR famil) # 2 TOPIC: (value OR preference* OR "goal* of care" OR choice* OR belief* OR attitude* OR expect* OR predisposition* OR influenc* OR experienc* OR choose* OR percept) # 3 TOPIC: ("decision making" OR decision NEAR/2 making OR decision OR decide) #4 TOPIC: #3 AND #2 AND #1 # 5 TOPIC: ("poor prognosis" OR relaps OR recurrenc* OR refractory OR incurable OR "advanc* cancer" OR "no realistic chance of cure" OR uncertain* OR deteriorat* OR "disease* progress" OR "palliat chemotherap" OR "no reasonable chance of cure") # 6 TOPIC: (child OR p?ediatric) # 7 TOPIC: (cancer OR neoplasm OR malignan* OR "cancer direct* therap" OR oncolog) #8 TOPIC: #7 AND #6 AND #5 # 9 TOPIC: (#8 AND #4)

Patient Public Involvement Consultation Phase

Within the European paediatric oncology community, there is a move to include patient public involvement in all aspects of patient care [24] and involvement in the peer review publication process more widely [25]. Within the parent and carer group, one member (CK) was given the extracted data from each included article on two specific questions: ‘what parent values and preferences were identified’ and ‘how did these values and preferences inform treatment decision-making’. This parent looked for recurring patterns in the data, considered the associated meaning and how these related to parent values and preferences in decision-making. The review findings were co-constructed between the lead author and parent (HP and CK), and this same parent reviewed the scoping review manuscript with input into the data analysis, results, discussion, and limitation sections. The whole parent and carer group reviewed the study results to see how representative these values and preferences were to parent experiences. They provided insight and feedback which is addressed in the patient public involvement consultation discussion section.

Evidence screening and selection

All articles were collated and uploaded into EndNote X9.2. Duplications were removed. The first level screen of title and abstract was completed independently by two reviewers (HP and ASD). Five articles had limited information provided within the abstracts. The third reviewer (FG) decided to include these articles for full-text review to avoid excluding potential research literature.

The second level screen involved full-text review. This was completed by the same two reviewers independently (HP and ASD). There were no discrepancies. The reference and citation list of included full-text articles were reviewed via the Web of Science by HP and resulted in additional articles for review. First and second level screening of these were undertaken by HP and ASD independently with no discrepancies.

A total of 573 articles were identified via electronic databases (CINAHL = 115, MEDLINE = 218, PyscInfo = 91 Web of Science Core Collections = 149). Of these, 242 were duplicates leaving a potential 331 articles. Based on title, 305 articles were excluded, resulting in 26 articles for first level screening. Upon completion, 16 articles remained for full-text review.

A further six articles were excluded at this stage. One article was a mixed-method systematic review incorporating the facilitators and barriers to decision-making in non-curative childhood cancer [13]. This review consisted of eighteen articles, twelve of which were found in database searches. Five articles were excluded at first level screening, and seven articles were included in this review. For cross checking purposes, the remaining six articles were first level screened and deemed not relevant to the scoping review objectives. A total of ten articles were included from database searches.

The reference and citation list of these ten articles were searched. This yielded an additional seven articles. Two articles were excluded at first level screening. At full-text review, three articles were excluded and two were included. These articles were not identified through database searches due to missing keywords in the article title or abstract.

Twelve articles were included in this scoping review (ten from database searches and two from reference/citation lists). The PRISMA flow diagram (Fig. 1) details study selection and inclusion process.

Data extraction

Data were extracted into a table developed during the protocol phase, and amended at this stage. Data extraction was completed independently by two reviewers (HP and GB), who met virtually to discuss. A third reviewer (FG) randomly selected 25% of the articles and completed data extraction table as a quality check. The review team (HP, GB, FG) met virtually to discuss. There were no discrepancies.

Analysis of the evidence

Summative content analysis identified repetitive words and phrases across the data extracted which was grouped to provide meaning of context [26]. Nvivo supported extraction of these repetitive words and phrases to establish frequency. These were then looked at in relation to the context meaning and grouped according to values and preferences, underpinning, and influencing factors in decision-making.

Description of the studies

The citation details of included articled can be found in Table 3. Most research was generated from the United States of America (US), represented by the first author for eight of the articles [8, 17, 27, 28, 29, 30, 21, 32]. Research methods were stated for all studies with interviews being the primary source of data collection. Research design was not explicitly stated for each article. Eight of the studies were qualitative [8, 27, 28, 29, 30, 32, 33, 34] and four were quantitative [16, 17, 31, 35]. One study was described as retrospective [8]. Other studies included bereaved parents but did not state a retrospective design. Three studies were described as prospective [17, 30, 33]. Studies had a significantly higher percentage of mothers participating than fathers, with one study having an equal number participating [16].

Table 3

Scoping review articles included in this review
First Author, Publication year	Title of Study	Journal	Country of Origin	Study Aim	Research Methods	Study Design	No of Participants	No of Mothers	No of Fathers
Hinds, P.S. et al (1996)	Coming to Terms: Parents’ Response to a First Cancer Recurrence in their child	Nursing Research	USA	Identify and describe the coping processes (meaning the behaviours and thoughts) used by parents of pediatric oncology patients to deal with stress of a first cancer recurrence in their child	Semi-structured interviews, observations	Qualitative	33	27	5
Hinds, P.S. et al (1997)	Decision Making by Parents and Healthcare Professionals when Considering Continued Care for Pediatric Patients with Cancer	Oncology Nursing Forum	USA	To better define the treatment related decisions considered most difficult by parents of paediatric patients with cancer and the factors that influence their final decisions	Semi-structured interviews, questionnaires	Retrospective-descriptive design	39	Not stated	Not stated
Hinds, P.S. et al (2000)	An International Feasibility Study of Parental Decision Making in Pediatric Oncology	Oncology Nursing Forum	USA	To describe parental decision making about treatment options for children with cancer and determine the feasibility of a similar but larger international study	Open-ended interviews	Qualitative Exploratory descriptive cross-sectional design	43	38	5
Bluebond-Langner, M. et al (2007)	Understanding Parents’ Approaches to Care and Treatment of Children with Cancer when Standard Therapy has Failed	Journal of Clinical Oncology	USA	To examine US and UK parents’ approaches to care and treatment when standard therapy has failed and consider implications for clinical practice	Semi-structured interviews, observation	Qualitative Prospective Ethnographic study	34	Not stated	Not stated
De Graves (2008)	Lving with Hope and Fear – The Uncertainty of Childhood Cancer after Relapse	Cancer Nursing	Australia	To develop an understanding of human actions and emotions that shape the experience of relapse, to question what influences the care provided at relapse, and to challenge current practice	In-depth interviews and field notes (case studies)	Qualitative Prospective Critical Ethnography	17	12	5
Mack et al (2008)	Parents’ Views of Cancer-Directed Therapy for Children with No Realistic Chance of Cure	Journal of Clinical Oncology	USA	Assess parents’ experiences with treatment for their child with cancer and no realistic chance of cure	Close-ended questionnaires, vignettes	Quantitative	141	117	24
Hinds et al (2009)	“Trying to be a Good Parent” As defined by Interviews with Parents who made Phase I, Terminal Care and Resuscitation Decisions for their Child	Journal of Clinical Oncology	USA	To define what it means to be a good parent to a child with incurable cancer	Interviews	Qualitative Descriptive study	62	91.4%	44.8%
Maurer et al (2010)	Decision Making by Parents of children with Incurable Cancer who opt for enrolment on a Phase I trial compared with Choosing a Do Not Resuscitate/Terminal Care option	Journal of Clinical Oncology	USA	To compare the self-reported rationale, good parent definition, and desired clinical staff behaviours of parents who recently decided for phase 1 (P1) chemotherapy with parents who chose do not resuscitate (DNR) or terminal care (TC) option	Interviews	Qualitative (secondary analysis)	62	53	9
Tomlinson et al (2011a)	Concordance between couples Reporting their Child’s Quality of Life and their Decision Making in Pediatric Oncology Palliative Care	Journal of Pediatric Oncology Nursing	Canada	To (1) describe concordance between fathers' and mothers' evaluation of QoL and (2) determine correlation between mother and father for how factors such as hope, anticipated QoL, and prolonged survival time influence the decision between supportive care alone versus aggressive chemotherapy	Hypothetical scenarios interview, format	Quantitative Cross-sectional study	26	13	13
Tomlinson et al (2011b)	Chemotherapy versus supportive Care alone in pediatric palliative care for cancer: comparing the preferences of parents and health care professionals	Canadian Medical Association Journal	Canada	The goal of this study was to compare the strength of preference between parents and healthcare professionals for supportive care alone versus palliative chemotherapy for children whose cancer has no reasonable chance of being cured, and to determine how specific factors affect these preferences	Hypothetical interviews (prepared scripts, visual aids)	Quantitative	77	60	17
Matsuoka et al (2012)	Parents’ thoughts and perceptions on hearing that their child has incurable cancer	Journal of Palliative Medicine	Japan	To describe parental thoughts and perceptions when they hear that their child has incurable cancer	Semi-structured open-ended interviews	Qualitative	23	16	7
Mack et al (2019)	Unrealistic Parental Expectations for Cure in Poor-Prognosis Childhood Cancer	Cancer	USA	The goal of the current study was to identify communication and care experiences that drive parents' care goals and decisions	Questionnaires	Quantitative Prospective cohort study	95	81%	19%
Abbreviations: No = number; USA = United States of America

What informs treatment decision-making?

Underpinning parent drive to continue with cancer-directed therapy were three factors: hope for a cure, fear of their child dying and uncertainty. These factors were constant through parent treatment decision-making, underpinning decisions which were made. Aspects which influenced parent decision-making included the opinion of others, child’s wishes and faith and religion. Parents valued having enough time, being a good parent and being involved in the decision-making process. Within these values were varying preferences which at times were conflicted and involved ‘trade-offs’ in order to pursue and continue with cancer-directed therapy. Figure 2 maps how underpinning factors, influencing factors and values and preferences informed parent treatment decision-making.

Underpinning factors

Hope for a cure

Hope was fundamental in the pursuit for cancer-directed therapies, highlighted 13 times in the literature. When informed of a new relapse parents underwent a process of “coming to terms”, adapting to what was happening to their child [27]. This adaptation involved parents’ managing their emotional reactions to make rational decisions in the hope for cure with the conflicting lingering possibility that their child might die [27]. Hope for a cure encouraged parents to believe this was possible and parents searched for this in conversations with healthcare professionals [33]. For parents, maintaining hope was the driving force in pursuing treatment and in some cases outweighed their child’s potential harm, suffering [33] and quality of life [16]. Continuing cancer-directed therapy in some cases was a parent obligation which corresponded with being a good parent and represented not giving up [34]. A chance for survival provided hope [28] and hope was ranked highest in the need to continue cancer-directed therapy [35]. One study found disagreement between mothers and fathers relating to quality of life and hope resulting in gender differences to continuing cancer-directed therapy [16].

Hope could be associated with leaving “no stone unturned” [30]. Despite parents recognising a cure was unlikely, parents continued to hope for a cure and therefore continued with cancer-directed therapy [31]. Treatment options included exploring alternative therapies [33] and seeking treatment opinions outside of what their child’s healthcare team offered [30]. The obligation to continue treatment, despite the knowledge that cure was unlikely resulted in parents pursuing treatments to ensure they had done everything possible to save their child’s life [28], in effect leaving “no stone unturned” [30]. This related with being a good parent, to keep all options open and not allow their child to die despite knowledge that this was a potential.

Fear of their child dying

Exploring second opinions and alternative therapies were all strategies in managing fear of their child dying, to reduce the potential of that becoming a reality [33]. This was what a good parent would do and in doing so this maintained hope in finding a cure.

Uncertainty

Uncertainty related to the unknown of how their child would tolerate treatment and the treatment outcome. Given the impact of uncertainty on parents, the word only appeared 9 times in the literature. Parents considered the effects of treatment on their child [8], and if treatment provided a positive outcome, then parents were satisfied with the decisions which they had made [28].

Adjustments within the family unit and parent role caused uncertainty. Parents were concerned how treatment would impact their employment, finances, and family life whilst simultaneously fearing their child would die with losing a sense of normality [27].

The uncertainty of their child’s outcome provided a “cognitive shift” between cure and death resulting in “fighting for cure” and “preparing for loss” [27]. The need to pursue all treatment options available, being a good parent maintaining hope and enable time with their child. This ensured parents had done everything possible whilst acknowledging treatment might not work, and death was a possibility. Not all parents were open or willing to consider this cognitive shift and did not recognise death as a potential reality [34].

Influencing factors

Opinions of others

The opinions of family members, other parents and healthcare professionals influenced parent decisions for cancer-directed therapy [16]. Searching outcomes from families that had gone through treatment and whose child had died helped parents in their adjustments [27].

The actions, information, communication, and support from healthcare professionals were major factors in parents feeling they were being accepted in pursuing cancer-directed therapies. Open communication on their child’s clinical condition, treatment response [8, 28, 29] and knowing everything possible was being done provided a sense of trust and reassurance [8, 29, 32]. A positive relationship where healthcare professionals knew the family and child provided a sense of belonging [29, 32].

Child’s Wishes

The child’s wishes were acknowledged in some studies [8, 27, 28]. This related to children expressing the extent of their symptoms and their preferences around the continuation of treatment.

Faith and Religion

Faith and religion were important factors from studies particularly in the US [27, 28, 35]. This related to being guided by religion and beliefs that their child’s outcome was out of their control.

Values and preferences:

Having enough time

Parents valued the need for time and was a component as to why parents choose to continue with cancer-directed therapies. Time was mentioned 18 times by parents across the literature. Time related to more time with their child, “prolonging life”, to give time for treatments to work or time for treatments to become available, time to realise and adjust to the situation and time to make informed decisions [16, 17, 28, 30].

Specific to buying time was parents wanting to start new treatments earlier or giving them longer time to work which enabled more time with their child [30]. Informed decisions having explored all possible treatment options enabled parents to plan for future possibilities for their child [28]. Parents valued time and the preferences of what time could give them.

Being a Good Parent

A good parent to a sick child pursued cancer-directed therapies, cared, protected, and advocated for their child within the healthcare professional team [30]. The definition of a “good parent” to a child with incurable cancer [29] was defined as unconditional love, provided basic provisions such as a home, clothing and food, prevented suffering, provided protection, promoted health, was a life teacher and made unselfish informed decisions in the best interests of their child [29, 32]. The words protect, advocate, “good parent” was mentioned 27 times in the literature. A good parent continued cancer-directed therapy to cure [17, 34], keep all treatment options open [30], and did not allow their child to die despite realisations that this could be the outcome [34].

Being involved in Decision-Making

Parents valued being involved in the decision-making process. Decision-making included parents exploring treatment options and navigating the options available to them. Decisions, making decisions and decision-making was mentioned 53 times in the literature. In some circumstances, parents felt they did not have a choice if they wanted their child to live with the need to “fight for life” [28]. Parents made decisions which were evidence-based [28, 32], yet experienced turmoil between fighting for a cure and having to decide [27]. There was strong emphasis on the need to continue cancer-directed therapies [30, 32, 34], yet this need in some circumstances was weighed against their child’s quality of life, level of suffering, others’ opinions [16, 34, 35], child’s wishes and religious beliefs [28]. Parents tried to balance saving their child with cancer-directed therapies and protecting from suffering and harm [33]. In the process of deciding, some parents were influenced by more than one preference [28] which had the potential to cause conflict within their decision-making. There was a sense of needing to “do the right thing” [28, 32] however a definition of what this meant was lacking.

In some circumstances, parents made repeated decisions relating to whether to continue cancer-directed therapy [8]. This was dependent on their child’s response to treatment and toxicities experienced. During the process of “coming to terms”, parents began to realise their child’s ability to tolerate and respond to treatments, the suffering this was causing and therefore started to consider the limitations of continuing treatment [27]. Despite literature which suggests parents may reach this realisation, this does not necessarily mean parents will discontinue cancer-directed therapy. Parents continued cancer-directed therapy for goals other than cure such as prolonging life and to decrease suffering [30, 31]. Awareness that their child was deteriorating with the potential for their child to struggle to tolerate further treatments and subsequently die was acknowledged [27, 34]. Parents recognised there could be an endpoint to cancer-directed therapy, and they might have to participate in making that decision [27]. For some parents, the need to repeatedly make decisions lead to the conclusion that their child would not get better resulting in the final decision to terminate cancer-directed therapy [8].

The complexity of parent treatment decision-making resulted in three aspects: underpinning factors, influencing factors and values and preferences. Underpinning factors were constant through the decision-making process. These could be interrupted as parent psychological and emotional aspects within decision-making. Influencing factors were those parents may consider when making treatment decisions but appeared to hold different weighting to parents across the data. Parents valued having enough time, being a good parent and being involved in decision-making. Each of these values contained preferences, aspects which were important to parents yet ones they might be willing to compromise on in order to continue with cancer-directed therapy. The literature defines these as ‘trade-offs’, the risks and benefits associated with each treatment option [36] and what parents were willing to accept in relation to their values.

Parents acknowledged their child may die, yet this did not result in a linear process of moving from cancer-directed therapy to symptom management/palliative care alone. The preferences of continuing cancer-directed therapy could alter at this timepoint with the overriding need to maintain hope, have time with their child and leave “no stone unturned” [30, 31]. This timepoint could be defined as a ‘tipping point’ in identifying a change of goals from continuing cancer-directed therapy in the hope of a cure to the realisation that their child will not survive and wanting to increase time with their child.

Parents experienced emotional adjustment when their child’s cancer relapsed managing the grief, shock, and anguish [27]. Parent emotions may fluctuate throughout the decision-making process depending on their child’s clinical condition, the decisions to be made and their own adjustment to the situation. Emotions have the potential to influence rational decision-making processes [27]. There is a body of decision-making literature on how people make decisions which is typically divided into rational [37, 38], descriptive/psychological [39, 40] and emotional [41] decision-making. Rationally, parents want to make informed decisions opting for the best treatment that is underpinned by science and statistics. Descriptive/psychological decision-making acknowledges how parents may obtain and process information, and how their previous experiences and intuition can influence decision-making. Emotion in decision-making focuses on parent feelings and emotions of the situation which can be informed by previous experiences, their current situation and anticipated future emotions [41]. The combination of these three decision-making components, (rational, emotional, and descriptive) may produce conflict in the pursuit of a decision that is underpinned by parent values and preferences and in the best interests of their child.

The “coming to terms” between cure and death was not seen in all parents which suggests hope as a central underpinning factor to these parents whose focus was solely on cure [27]. Parents may subconsciously acknowledge what is happening but doing so consciously means confronting or speaking about this fear which some parents may not be able or willing to do. The value of being a good parent and the preferences associated with this differed between parents. Preferences appeared conflicted wanting to prevent suffering, protect their child and act in their child’s best interests whilst continuing with cancer-directed therapies [29, 32] which could induce suffering and not be in their child’s best interests. Parents emphasised quality of life and to reduce suffering and harm [35]. However, only one study explicitly stated symptom management was given in conjunction with cancer-directed therapy [30]. It cannot be assumed that parents did not engage with symptom management, more that studies did not clearly identify this. A good parent continued cancer-directed therapy to cure even when parents realised their child may die. The need to continue cancer-directed therapy could be underpinned by hope. Hope for a cure enabled parents to pursue cancer-directed therapy even at the expense of the child’s suffering.

The involvement of the child’s wishes is likely to be dependent on age and cognitive ability. Protection was a core component of being a good parent [29] and in some instances parents my protect their child from the full extent of the seriousness of the situation.

In the early 2000s the internet and use of social media platforms became more established. Nowadays many childhood cancer diagnoses have parent-led social media pages on platforms such as Facebook, Instagram and Twitter with a community that share personal knowledge and experience. Furthermore, parents can research treatment options and clinical trials internationally empowering them to explore treatment options beyond what their child’s clinician offers which was seen in some studies [30, 33].

Parents are faced with making complex treatment decisions when their child has a poor-prognosis childhood cancer. Their values can contain conflicting preferences in opting for cancer-directed therapy. Research has shown regret is less evident in parents who trust their child’s oncologist, have concise prognostic information, and are involved in the decision-making process [7]. This reinforces the need to provide adequate support in the decision-making process. Treatment decision-making becomes complex when there is no standard of care treatment protocol resulting in the potential for parents to make multiple repeated treatment decisions as their child’s condition changes. Understanding these parent values and preferences and how they inform treatment decision-making provides the basis for developing support tools such as decision aids to support parent treatment decision-making and enhance discussions between parents and healthcare professionals.

Patient Public Involvement Consultation Discussion

The definition of values and preferences resonated with parents, but the individual meaning was seen as subjective to each parent. For example, what one parent may define as suffering another parent may not. The need for parents to define their meaning of a preference may support decision-making discussions with healthcare professionals in clinical practice. The group saw preferences as the trade-offs parents made to prioritise what they valued most. For example, one parent spoke of prioritising prolonging life but in doing so this may increase suffering resulting in conflicting preferences.

The complexity of decision-making was hard to define. For example, the emotion of decision-making was lacking, and the literature was ‘sanitised’ as a definitive way of how parents made decisions but, it is not linear and difficult to articulate. One study alluded to the emotional component of making treatment decisions [27]. Parent emotion is huge in decision-making and one that the group felt was not fully addressed within the literature.

The values presented were an accurate perception of decision-making in poor-prognosis childhood cancer, but the richness and depth of the complexities of this decision-making was not captured possibly due to the topic sensitivity. The group acknowledged limitations for example journal word restrictions, or the data collection methods used which could inhibit the understanding of these complexities. For example, the use of questionnaires for a sensitive topic felt inappropriate and data produced during interviews is dependent on the trust built between parent and researcher particularly if there is no prior relationship.

The group spoke of a constant shift in parent values and preferences relevant to the situation. How a parent defines these values and preferences is not constant through time but changes as the situation develops depending on their child’s clinical condition and treatment decisions to be made. This was acknowledged in a shared decision-making framework where values and preferences are not seen as stable, but change based on parent capacity and reflection [42]. The group represented this shift as a weighing scale, the options parents have at each decision point with the compromises (preferences) required to make that decision. Parents spoke of this shift being underpinned by a constant feeling of pressure, needing to do the right thing for their child, not wanting to let their child down and the uncertainty and fear that their child may or is dying. This related to the underpinning factors of hope for a cure, fear of their child dying and uncertainty.

The language used within the literature caused concern. Words such as “fighting” were seen in a negative context like a parent going to war. The group had experienced parents using words like fight, battle, winning and losing in clinical practice. This language does not support parents in their “cognitive shift” and adjustment to the situation.

Parents would regret their decisions if their child had died as an outcome, was the consensus of decisional regret. As a result, retrospective study designs could produce biased findings and not be a true representation of parent decision-making at the time of making those decisions. A more accurate representation would involve parents at or just after the time of making a decision whilst the decision outcome is unknown. The group felt parents would value prospective study designs which provide opportunity to talk through their decision-making processes in real time. They did not feel strongly that this approach would add burden to parents.

Limitations

One study included parents whose child was between 1–24 years old at the time of death [8]. This study did not provide details on how old the child was at the time of receiving treatment nor were the results broken down by age groups. Although all relevant data were extracted it is not known whether this was relevant to those under the age of 18 years.

There was a lack of father involvement across the studies. There is a need to increase the participation of fathers in research such as this to further explore whether there are gender differences in the values and preferences which underpin treatment decision-making. One research study suggested there were differences [16].

The literature on decision-making acknowledges rational, descriptive, and emotional processes in decision-making. The parent and carer group provided consensus that parent emotion in treatment decision-making had not been addressed fully in the literature. Emotion can be difficult to articulate which might be the reason why there is limited acknowledgement of this in the literature.

The complexity of treatment decision-making in poor-prognosis childhood cancer considers more than parent values and preferences. There are constant underpinning factors to these decisions for parents, hope for a cure, fear of their child dying and uncertainty throughout the decision-making process. Influencing factors can support or refute the treatment options parents are considering yet these are not consistent factors for every parent. Values appeared consistent but the preferences within these can be conflicting resulting in complex decision-making which is in their child’s best interests. Preferences conflict when parents want to continue cancer-directed therapy whilst maintaining their child’s quality of life or can change depending on a parents’ cognitive state as they realise cure might be unlikely. Complexities of decision-making in this situation is difficult to articulate and the literature lacked the emotional component involved in making these decisions.

Implications of the findings for research

The exploration of emotion in this type of decision-making is warranted. Preferences in treatment decision-making can change depending on several aspects. Research to explore how these values and preferences may change over time with parents who make multiple repeated treatment decisions could provide more effective ways of supporting parents in clinical practice. Studies included in this review stated the need to develop decision support tools, guidelines and care models which can be integrated into clinical practice. Development of such tools and models are required to support parents in clinical practice in this complex decision-making.

CINAHL: Cumulative Index of Nursing and Allied Health Research; JBI: Joanna Briggs Institute; MeSH: Medical Subject Headings; PRISMA: Preferred Reporting Items for Systematic Reviews and Meta-Analysis’ US: United States of America

Ethics approval and consent to participate

Not applicable

Consent for publication

Not applicable

Availability of data and materials

Data extracted using the data extraction tool are available from the corresponding author on reasonable request. As this is not empirical data owned by the authors the data extracted is available within the public domain.

Competing interests

The authors declare that they have no completing interests

Funding

Helen Pearson is supported by National Institute Health Research (NIHR) Clinical Doctoral Research Fellowship NIHR300548.

Author Contributions

HP conceptualised the review questions. HP and CK contributed to the writing of the manuscript with review from GB, FG and ASD. HP, ASD and FG conducted screening and full text reviewing, whilst data extraction was undertaken by HP and GB. HP and CK conducted the analysis of the results. The final manuscript was approved by all authors.

Acknowledgements

With thanks to Vicky Fenerty, Research Engagement Librarian at The University of Southampton for assistance in developing the search strategy and peer-reviewing the database searches.

With thanks to Carol Bell, Karl Cox, Leona Knox and Nick Bird, parent and carers who reviewed this scoping review report and had input into the interpretation of the results.

This research was supported in part by the NIHR Great Ormond Street Hospital Biomedical Research Centre.

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No competing interests reported.

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Parent values and preferences underpinning treatment decision-making in poor-prognosis childhood cancer: A scoping review

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Abstract

Figures

Background

Treatment Decision-Making

Values and preferences

Decisional Regret

Methods

Research Questions

Eligibility Criteria

Search Strategy

Patient Public Involvement Consultation Phase

Evidence screening and selection

Data extraction

Analysis of the evidence

Presentation Of The Results

Description of the studies

What informs treatment decision-making?

Underpinning factors

Hope for a cure

Fear of their child dying

Uncertainty

Influencing factors

Opinions of others

Child’s Wishes

Faith and Religion

Values and preferences:

Having enough time

Being a Good Parent

Being involved in Decision-Making

Discussion

Conclusions

Abbreviations

Declarations

References

Additional Declarations

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