Memory-making interventions for children and their families receiving pediatric palliative or bereavement care: A systematic review protocol

Background In paediatric palliative and bereavement care, providing comprehensive support that extends beyond medical treatment to address the emotional and psychosocial needs of children and their families is essential. Memory-making interventions play a critical role in capturing cherished moments and fostering emotional resilience. However, widespread consensus on the foundation and scope of memory-making interventions for children and young people remains sparse. This review aims to identify, appraise, and synthesise the evidence on memory-making interventions for children and young people with life-limiting or life-threatening conditions and their family members receiving palliative or bereavement care. Methods This systematic review will follow the Preferred Reporting Items for Systematic Reviews and Meta-Analysis (PRISMA). A systematic search will be undertaken from January 1, 1985, to February 27, 2024, across the following databases: PubMed, EMBASE, CINAHL (EBSCO), PsycINFO (EBSCO), Web of Science, the Cochrane Library, and Scopus. Studies across diverse research designs that examine children (0-19 years) with life-limiting or life-threatening conditions undergoing memory-making interventions with psychosocial or other outcomes will be included. Screening, data extraction, and quality appraisal will be performed by two independent reviewers, with a third reviewer resolving discrepancies. Joanna Briggs Institute guidelines for conducting mixed methods systematic reviews will be used to inform the data analysis and synthesis process. Conclusions This review will provide critical insights into the existing evidence base on memory-making interventions in paediatric palliative and bereavement care, highlighting psychosocial and other impacts, implementation factors, and evidence quality. By identifying best practices and gaps in knowledge, this evidence review may inform future research and intervention design, or adaptation, and contribute to the enhancement of healthcare for children and young people with life-limiting and life-threatening conditions and their families as well as families in bereavement. Registration This review was registered in PROSPERO, the International Prospective Register of Systematic Reviews (CRD42024521388; 18/03/2024).


Introduction
The integration of paediatric palliative and bereavement care within the global health landscape is essential for providing comprehensive care to children facing life-limiting and life-threatening (LLLT) conditions, as well as their families.Despite the challenge of accurately estimating prevalence rates of children with LLLT conditions, the demand for palliative care services is significantly underestimated and is expected to rise (Department of Health and Children (DoHC), 2009;Fraser et al., 2021).Palliative care aimed at improving the quality of life for patients with serious illnesses and their families (Hallenbeck & Hallenbeck, 2022).Children with life-limiting conditions have no reasonable hope of cure and are expected to die prematurely, while life-threatening conditions involve a high probability of premature death but may have feasible curative treatments (Roland et al., 2022).Paediatric palliative and bereavement care services are crucial in circumstances where cure may not be an option, requiring care that extends beyond traditional medical treatments to address holistic care for children's physical, psychological, and spiritual needs, along with supporting families from diagnosis through end-of-life care or ongoing treatment (Mitchell et al., 2020;Together for Short Lives, 2018;World Health Organization, 2020).This aligns with the Sustainable Development Goal 3, which emphasises universal health and well-being, including palliative care as an essential service for achieving global health objectives (United Nations, 2023).
Current estimates suggest around 21 million children globally require palliative care annually, with approximately 8 million needing specialist care (Connor et al., 2017).In England, the prevalence of children with life-limiting conditions rose from 26.7 per 10,000 in 2001/02 to 66.4 per 10,000 in 2017/18, with the highest rates in the under 1-year age group (Fraser et al., 2021).Similarly, in Queensland, Australia, the prevalence increased from 35.2 per 10,000 in 2011 to 43.2 in 2016, especially among children under one year and Aboriginal and Torres Strait Islander children (Bowers et al., 2020).This growing demand highlights the importance of comprehensive palliative and bereavement care with the prevalence of children requiring these services expected to rise due to advancements in medical care and increasing recognition of paediatric palliative needs (Roland et al., 2022).
Globally, the provision of bereavement support tailored to the needs of families of children receiving palliative care is recognised as a critical component of comprehensive care (Applebaum et al., 2023;Hudson et al., 2018;Thornton et al., 2021).This support, crucial for navigating the emotional, psychological, and practical challenges following the loss of a child, emphasises family-centred psychosocial interventions to enhance life quality and well-being (Akard et al., 2018;Benini et al., 2022;Pedraza et al., 2024).Family distress is a concern in paediatric palliative and bereavement care, where emotional burden can be overwhelming for parents (Bronsema et al., 2022;Hasdenteufel & Quintard, 2022).Minimising parents' regret is also essential for healthy bereavement, as unresolved regret can complicate the grieving process.Parents who had minimal involvement in their infant's bedside care expressed feelings of regret and guilt after their loss (Sim et al., 2020).Clarke & Connolly (2022) found that parents appreciated memory-making activities, such as creating personalised memory boxes, hand and footprint jewellery, and moulds, which help document their child's life and maintain a connection after their death, significantly aiding the grieving process.The National Policy on Palliative Care for Children with Life-Limiting Conditions in Ireland (DoHC, 2009)  This review focuses on children and young people aged 0-19 years with LLLT conditions, recognizing that care and support needs vary significantly across different age ranges, developmental stages, and conditions.Younger children may require more family involvement in interventions, such as creating scrapbooks and memory boxes, while adolescents and young adults might engage more independently in activities like hobbies and school.Understanding these differences ensures that interventions are tailored to support infants, children, young people and their families effectively (Coombes et al., 2022).
Memory-making interventions are increasingly recognised within paediatric palliative and bereavement care contexts to help children and families make meaning out of their experiences (Akard et al., 2021a;Akard et al., 2021b;Sisk et al., 2012).Memory-making, also often referred to as creating a legacy (Akard et al., 2021c), is a broad term describing specific processes, such as sense-making and benefit finding, that contribute to adapting to stressful life experiences and is a core element in coming to terms with grief (Kobler et al., 2007;Park, 2013).Memory-making interventions can include tangible activities such as memory boxes, handprint art, or digital storytelling and recording special messages.These interventions serve not only as therapeutic tools to support emotional expression and processing but also as valuable artifacts that families can hold onto, serving as a source of comfort and connection to their loved one (Boles & Jones, 2021).

Amendments from Version 1
In Version 2 of our study protocol, we made targeted revisions to enhance clarity and depth in some areas.First, we refined the background section to include current and projected estimates of children with life-limiting conditions, emphasizing the growing need for palliative and bereavement care.We deepened the definition of palliative care to highlight its role in improving the quality of life for children and their families.This revision also addresses caregiver distress and regrets and underscores the benefits of memory-making interventions in reducing grief.Additionally, we clarified and standardized the age range as 0-19 years, expanding our discussion to detail the diverse support needs across different developmental stages.The term "children and young people" has been used consistently to maintain clarity throughout the text.For more detailed explanations, please refer to the responses provided to the reviewers' questions.

Any further responses from the reviewers can be found at the end of the article
Four recent review papers have shed light on the diverse approaches to memory-making in healthcare (Boles & Jones, 2021;Keller et al., 2024;MacEachen et al., 2023;Xu et al., 2024).Although these reviews cover a broad spectrum, none specifically focused on memory-making interventions for children with LLLT conditions and their families within the realms of palliative or bereavement care.Boles and Jones (2021) highlighted the significant benefits of legacy interventions, for both adult and paediatric populations, showing significant improvements in well-being and a reduction in depression symptoms among adults.This review demonstrates the utility and benefits of legacy interventions but highlights the scarcity of paediatric focused research and the need for standardized practices in legacy interventions.Keller et al. (2024) took a deep dive into what "legacy" means in the context of paediatric healthcare.They identified that legacy creation in children's healthcare is a collaborative process that leaves something lasting to remember the child by, capturing the unique essence or spirit of the child.They identified that legacy-oriented interventions are provided in most children's hospitals in the United States yet highlighted the absence of a widespread consensus on the foundation or scope of these interventions.This gap emphasises the critical need for evidence-based guidelines and practices in creating and implementing legacy for and with children, suggesting an opportunity to enhance the quality of care for children and their families globally.
In a qualitative thematic synthesis, MacEachen et al. (2023) explored families' experiences of memory-making in both adult and paediatric critical care settings, aiming to understand the impact on bereaved families.The synthesis identified four main themes describing families' experiences: connection, compassion, engagement and creation, and continuation.MacEachen's review illuminates how memory-making facilitates a connection between families and healthcare staff, providing a structure and purpose during the bereavement process, and fostering a continuing bond with the deceased.The review also highlights the lack of research focusing on paediatric critical care settings, marking a vital area for future exploration.Finally, Xu et al.
(2024) conducted a qualitative synthesis examining bereaved parents' perceptions of memory-making.Their findings reveal parents' recognition of memory-making's significance in connecting with their children and navigating grief.Yet, they identified barriers such as a lack of understanding and preparation, along with limited support, which sometimes led parents to miss out on these meaningful opportunities.The review calls for a deeper investigation into how memory-making practices and intervention can be better tailored to meet the diverse needs of families, particularly considering cultural sensitivity and the evolving nature of grief over time.While these previous reviews have revealed important information about memory-making and memory-making interventions in various contexts and with different populations, they also highlight significant gaps in our understanding, particularly concerning paediatric populations, and the need to consider challenges faced by bereaved parents.To date, to our knowledge, no systematic review has been undertaken to specifically examine memory-making interventions for children with LLLT conditions and their family receiving palliative and/or bereavement care.
Therefore, the aim of this review is to identify, appraise, and synthesise the current evidence on memory-making interventions for children with LLLT conditions and their family members who are receiving palliative or bereavement care.Our objectives include synthesizing evidence on the (1) range of memory-making interventions used for children with LLLT conditions and their families receiving palliative or bereavement care, (2) effectiveness of memory-making interventions by synthesizing outcomes reported in the literature and mapping the measures used to assess psychosocial outcomes like resilience, emotional coping, family communication, and any other relevant outcomes, (3) experiences of families who participate in memory-making interventions, focusing on their perceptions, the perceived impact on their emotional and psychosocial well-being, and the contribution to family cohesion, and (4) barriers and enablers affecting the successful implementation of memory-making interventions.

Methods
This protocol was developed in accordance with the guidelines for the Preferred Reporting Items for Systematic Reviews and Meta-Analysis Protocols Checklist (PRISMA-P) (Moher et al., 2015).This review protocol has been registered prospectively on the PROSPERO database for systematic reviews (registration number CRD42024521388; 18/03/2024).This systematic review will be reported in accordance with the Preferred Reporting Items for Systematic Reviews and Meta-Analysis (PRISMA) statement (Page et al., 2021).

Eligibility criteria
The eligibility criteria for inclusion in this review will be outlined using the Population, Intervention, Comparison, Outcome (PICO) framework.

Population
This review targets children and young people aged 0-19 years with LLLT conditions and their families.A life-limiting condition is defined as an illness or disorder with no reasonable expectation of cure, leading to premature death and requiring comprehensive care that addresses the child's physical, psychological, social, and spiritual well-being.A life-threatening condition, conversely, is characterized by a significant risk of death but may be amenable to curative treatment or prolonged management.This review extends to family members involved in or benefiting from memory-making interventions in the context of palliative or bereavement care.Family members include, but are not limited to, parents, siblings, and other relatives, as identified by the studies included in this review.

Interventions
Memory-making interventions are identified as therapeutic activities designed to aid children with LLLT conditions, and their families, in crafting lasting memories.These interventions encompass a broad range of activities, including digital storytelling, the creation of physical mementos, and participatory art projects.The interventions focus on enhancing emotional well-being, offering comfort, and bolstering coping and resilience in the face of adversity.This review considers interventions that involve the child alone, child-parent dyads, child-other family member dyads, child-parent-other family member triads, the entire family unit, and interventions for bereaved family members.The scope includes interventions directed by, with, or about children and their families, aiming to support them during palliative care or the bereavement process.

Comparators/control
This review will compare memory-making interventions against usual care practices or other active interventions as defined by included studies.In qualitative or exploratory studies without direct comparators, no specific control will be applied.

Main outcome(s)
The primary focus of this systematic review is on psychosocial outcomes, which are broadly conceptualized to cover various aspects of psychosocial outcomes for children with LLLT conditions and their families.These outcomes include, but are not limited to: • Quality of life, • Mental health indicators such as stress, anxiety, and depression, • Aspects of communication within the family and strength of family communication.

Additional outcome(s)
Beyond the primary focus, this review also seeks to explore the wider impacts of memory-making interventions on families and children with LLLT conditions.These additional outcomes include, but are not limited to: • Coping and resilience mechanisms developed because of the interventions, • Satisfaction with and perceived value of the intervention by participants, • The role of these interventions in the creation and preservation of memories.

Study type
This review will include studies that provide qualitative, quantitative, or mixed-method data, including but not limited to randomized controlled trials, cohort studies, case-control studies, cross-sectional studies, qualitative studies, and mixed-methods studies.This approach allows for a broad synthesis of evidence across diverse research designs, offering a comprehensive understanding of memory-making interventions within paediatric palliative and bereavement care.

Information sources and search strategy
To ensure a comprehensive literature review, we will conduct searches across multiple databases for peer-reviewed publications from January 1, 1985, to February 27, 2024.These databases include PubMed, EMBASE, CINAHL (EBSCO), PsycINFO (EBSCO), Web of Science, Cochrane Library, and Scopus.Additionally, we will examine the reference lists of related reviews to identify further studies that meet the review eligibility criteria.
The search strategy of this review has been systematically developed according to the PICO framework, focusing on the Population (P) and Intervention (I) components.This involved the utilization of the MeSH database in PubMed and Emtree via Embase and was informed by previous systematic reviews.A sample search strategy developed with a librarian is presented in Table 1.

Screening and selection of studies
Search outputs will be imported into Covidence where a twopart screening process will be undertaken once duplicates are removed.Part one screening will involve two reviewers (RS, VL) independently screening titles and abstracts against the review eligibility criteria.Any discrepancies between reviewers will be resolved through consensus or discussion with a third reviewer (EC).For part two screening, two reviewers (RS, VL) will independently assess full texts against the eligibility criteria before a final decision regarding inclusion or exclusion is confirmed.Any discrepancies will be resolved through consensus or discussion with a third reviewer (EC) acting as arbitrator as required.We will record reasons for excluding studies at the full-text stage.An adapted PRISMA flow chart will be used to report the screening and selection process at each stage of the review.

Data extraction
Data extraction will be conducted by one reviewer (RS) using designed data extraction forms tailored to each study type (e.g., quantitative, qualitative, mixed methods), which will be refined based on pilot data extraction.Across the included studies, where relevant, data will be extracted on authors, title, year of publication, country, aim, study design, participant demographics, interventions and comparators, outcome measures, and key findings.Where we encounter missing, unclear, or incomplete data, we will contact the study authors to obtain additional information.Extracted data will be cross-checked by a second reviewer (VL) for accuracy and completeness.Any discrepancies will be resolved through consensus, or, if necessary, through consultation with a third reviewer (GK).

Quality appraisal
Utilizing the Mixed Methods Appraisal Tool (MMAT), two independent reviewers (RS, YC) will appraise the methodological quality of the included studies.This appraisal will focus on the robustness of the study design, data collection, and analysis.The MMAT's versatility allows for a detailed appraisal across different study types (i.e., qualitative, quantitative, mixed methods etc.), ensuring a thorough examination of bias and methodological integrity (Hong et al., 2018).In instances of divergent assessments, consensus will be sought through discussion or consultation with a third reviewer (VL).

Data analysis and synthesis
Joanna Briggs Institute (JBI) guidelines for conducting mixed methods systematic reviews will be used to inform the data analysis and synthesis process.By adhering to the JBI Manual for Evidence Synthesis, we plan to integrate quantitative and qualitative findings effectively (Peters et al., 2020).
Through narrative synthesis, we will assess the effectiveness, implementation, and quality of memory-making interventions in palliative care, identifying common patterns, themes, and points of divergence across studies.Where feasible, quantitative data may be subjected to meta-analysis to examine intervention outcomes, provided that the data demonstrate sufficient homogeneity.Qualitative synthesis, on the other hand, will focus on identifying barriers, facilitators, and the contextual factors influencing the efficacy and implementation of interventions.This process will be informed by the Template for Intervention Description and Replication (TIDieR) checklist and JBI's convergence framework for mixed methods studies, ensuring clarity in intervention description and a nuanced understanding of the combined evidence (Hoffmann et al., 2014).This review's goal is to provide a comprehensive synthesis that reveals the complex dimensions and impacts of interventions, thereby informing practice and guiding future research on targeted intervention strategies.

Conclusion
This systematic review will culminate in a comprehensive synthesis of evidence on memory-making interventions in paediatric palliative and bereavement care, highlighting psychosocial and other impacts, implementation factors, and evidence quality.By identifying best practices and gaps in knowledge, this evidence review may inform future research and intervention design, or adaptation, and contribute to the enhancement of future healthcare for children and young people with life-limiting and life-threatening conditions and their families, as well as those in bereavement.

Alison Rodriguez
University of Leeds, Leeds, England, UK Thank you for the invitation to review this article.The manuscript details a well-structured review protocol that aims to identify, appraise, and synthesise the evidence base that reports on memory making activities for children and young people (CYP) (0-19yrs) with life-limiting and lifethreatening conditions (LLLTC) and their families within both palliative and bereavement pediatric care contexts.These activities are undertaken in practice to provide some level of emotional and psychosocial support to CYP and families.A review of this nature is welcomed.The authors draw on relevant review guidance and propose methods to safeguard the reliability/trustworthiness of the review procedures and findings.There are just a few points that the authors may want to further consider: The abstract of the protocol summarises the protocol well but there could be further acknowledgement of bereavement care in the conclusion section given the focus is on memory making activities in palliative and bereavement care for CYP and their families. 1.
In the introduction section of the protocol, it is stated that there are difficulties with accurately estimating prevalence rates for the population under study.This is true but it would be useful for the readership to know if there are any current and projected estimates proposed?(To further consider how impactful these types of interventions can/could be).

2.
It is unclear what age range this work is focused upon until we get to the methods section of the protocol.Given the age range is 0-19 there could be some earlier acknowledgement of this in the introduction and some consideration of how care and support may be different for CYP of different age ranges (within this 0-19 range), and how support for families and their involvement in care maybe different for different age ranges, developmental stage/ability, and condition type.The authors are considering a patient group that includes many conditions with a multitude of impacts in terms of life 3.
expectation, related disease trajectories, emotional and psycho-social needs, and that may engage different models of care.Also, where some CYP will be able to engage in interventions independently, others will not.The review undoubtedly will unveil issues relevant to these points, but I think an acknowledgement of the variance in support need and levels of CYP participation in these types of activities may be worthwhile.Use of the terms 'children and young people' is generally preferred over just 'children' given the proposed age range includes adolescents and young adults.

4.
Within the method section there is narrative around how appraisal of any included study will acknowledge standard care practices.It will be interesting to see if studies have done this in exploring these types of interventions within paediatric care because there is no standard of care with respect to emotional and psycho-social support for these CYP.

5.
If the review will be limited to paediatric settings, there may be potential to exclude studies reporting on memory making interventions that could be accessed by CYP transitioning to adult care services and intervention studies that have included people above 18 years of age in their inclusion criteria.If the authors remain happy to exclude these studies, then perhaps a stronger case to only include studies that have explored memory making within paediatric care settings could be afforded.It would be useful for the authors to at least note how models of care can be different and this may mean some CYP and families access memory making interventions outside of paediatric services.

6.
In terms of considering outcome measures it is good that the authors have considered to 'include but not be limited to' criteria as it is expected that a wide array of outcomes could be reported (and not necessarily measured).

7.
Is the rationale for, and objectives of, the study clearly described?Yes

Is the study design appropriate for the research question? Yes
Are sufficient details of the methods provided to allow replication by others?Yes Are the datasets clearly presented in a useable and accessible format?Yes Competing Interests: No competing interests were disclosed.
Reviewer Expertise: Health psychology -Children and young people with life limiting and life threatening conditions, lived experience, life review and meaning making, phenomenology, long term conditions, and self management.
I confirm that I have read this submission and believe that I have an appropriate level of expertise to confirm that it is of an acceptable scientific standard.
Author Response 03 Jul 2024

Razieh Safarifard
Dear Reviewer, Thank you for your thoughtful and constructive feedback on our manuscript.We appreciate your positive comments and have carefully considered your suggestions.Below, we address each of your points and outline the revisions made in the second version of our protocol: 1. Bereavement Care in the Conclusion Section of the Abstract: The conclusion has been revised to explicitly include bereavement care, enhancing the clarity and scope of the abstract.
2. Prevalence Rates in the Introduction: We added current and projected estimates of children with life-limiting conditions to the introduction, highlighting the growing demand for palliative and bereavement care.
3. Acknowledgement of Age Range and Support Needs: The age range (0-19 years) has been clarified, and the background section of the protocol has been expanded to detail the diverse support needs across different ages and developmental stages.
4. Terminology -Children and Young People (CYP): The manuscript was reviewed to ensure the term "children and young people" is used consistently and appropriately throughout the text.
5. Standard Care Practices in Methods Section: The term "standard care" has been reworded to "usual care" to better capture studies that report any standard regarding the intervention, clarifying our evaluation criteria.
6. Focus on Paediatric Settings: Thank you for your comment and suggestion.We are not just focused on paediatric palliative care but are also interested in other relevant settings to provide a comprehensive review.
7. Outcome Measures: We are grateful for your acknowledgment of the inclusiveness of our approach to outcome measures.We concur that a wide array of outcomes, both reported and measurable, is essential for a comprehensive systematic review.
We value your insights, which have significantly enhanced our manuscript.We look forward to continuing this productive dialogue.

Sincerely,
Competing Interests: No competing interests were disclosed.

Myra Glajchen
Albert Einstein College of Medicine, Bronx, New York, USA Memory-making is an important intervention in oncology, palliative care and hospice.According to this review, such interventions confer benefits for children with life-limiting conditions.This timely systematic review aims to fill an important gap in the literature given that this area has been under-studied.Unfortunately, the review is not scientifically sound, literature is lacking in lacking critical areas including palliative care, caregiver distress, minimizing caregiver regret as part of healthy bereavement, and understanding meaning-making from a developmental perspective.In addition, it is impossible to assess the quality of the data given that the authors do not share their findings.The utility of this paper lies in its list of references and description of its methodology.
Is the rationale for, and objectives of, the study clearly described?Yes Is the study design appropriate for the research question?Yes

Are sufficient details of the methods provided to allow replication by others? No
Are the datasets clearly presented in a useable and accessible format?Partly Competing Interests: No competing interests were disclosed.
Reviewer Expertise: Psychosocial oncology, palliative care, oncology care, social work, interprofessional training, caregiver burden./I confirm that I have read this submission and believe that I have an appropriate level of expertise to state that I do not consider it to be of an acceptable scientific standard, for reasons outlined above.
Author Response 03 Jul 2024

Razieh Safarifard
Dear Reviewer, Thank you for your thoughtful review of our systematic review protocol paper.We appreciate your feedback and have made several revisions to address your concerns and enhance our work.
We have expanded the literature to define palliative care more clearly, emphasizing its role in improving the quality of life for both the child/young person and their families.We acknowledge the emotional burden on caregivers in paediatric palliative care and the importance of minimizing caregiver regret for healthy bereavement.Additionally, we have highlighted that memory-making interventions help families create meaningful memories, reducing feelings of regret and supporting a healthier grieving process.We refined our background section to emphasize the critical areas you mentioned.Our protocol now reflects these concerns more clearly.
Regarding your comment on the impossibility of assessing the quality of the data due to the lack of shared findings, we acknowledge the challenge.As this is a protocol for a systematic review, we appreciate the difficulty in assessing data quality at this stage.We will report more explicitly on this aspect in the findings once the systematic review is completed.
We appreciate your valuable insights, which have significantly helped us improve our manuscript.

Sincerely,
Competing Interests: No competing interests were disclosed.
and studies such as Clarke & Connolly (2022) highlight the significance of interventions like memory-making interventions, which allow families to create lasting memories and maintain a continuing bond with their child.

Table 1 . Sample search strategy.
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