Introduction
Globally, estimates indicate that 8 million children per year live with some form of life-limiting condition for which there is no hope of cure and from which a child or young person will die (Connor et al. Reference Connor, Downing and Marston2017; Together for Short Lives 2017). Of these, children with severe neurological impairment (SNI) form a distinct and diverse population (Allen et al. Reference Allen, Brenner and Hauer2020). Common features of associated conditions include severe motor and cognitive impairments with co-occurring medical complexity (Cohen et al. Reference Cohen, Kuo and Agrawal2011). Due to medical and technological advances, growing numbers of affected children now survive with lengthier trajectories (Koch and Jones Reference Koch and Jones2018). These children require intensive caregiving and assistance with activities of daily living (Clarke and Quin Reference Clarke and Quin2007). The illness course typically follows a downward trajectory, with periods of relative stability interspersed with episodes of acute ill-health as the condition progresses (Steele Reference Steele2000). Increasingly, it is recognized that good pediatric palliative practice involves caring for parents and assessment of the family situation (Koch and Jones Reference Koch and Jones2018). Recent literature reveals the complex pervasive grief experienced relating to the loss of previously sustaining world assumptions, the wished for child, and inevitable, their child’s death. This type of loss is characteristically disenfranchised (Doka Reference Doka2002), falling outside normative language and customs of death-related grief. Indeed, parents may struggle to express emotions or fully comprehend their loss experience (Neimeyer and Krawchuk Reference Neimeyer, Krawchuk and Harris2020).
However, current theoretical models of grief provide a strong empirical basis on which to approach the care of bereaved individuals (Bonanno et al. Reference Bonanno, Wortman and Lehman2002; Coleman and Neimeyer Reference Coleman and Neimeyer2010; Klass et al. Reference Klass, Silverman and Nickman2014; Stroebe and Schut Reference Stroebe and Schut2010). Notably, newer developments in our understanding of grief incorporate the experience of significant non-death losses and emphasize the significance of meaning reconstruction in the grieving process (Braun and Berg Reference Braun and Berg1994) and have moved away from stage or task model approaches (Parkes Reference Parkes1988; Worden Reference Worden2009). In bereavement, significant losses are shaped by the social environment where interpersonal interactions influence how we grieve. Concepts such as meaning-making, resilience, and identity change have significance for how we relearn the world after loss (Bonanno Reference Bonanno2009; Lichtenthal et al. Reference Lichtenthal, Currier and Neimeyer2010; Neimeyer et al. Reference Neimeyer, Klass and Dennis2014). Studies show that finding meaning in loss helps individuals move forward with life, create new meaning, and build capacity for the future (Lichtenthal et al. Reference Lichtenthal, Currier and Neimeyer2010). In palliative care, the absence of meaning-making care has been shown to predict difficulties with anticipatory grief, and complicated grief symptoms after the death occurs (Burke et al. Reference Burke, Neimeyer and Bottomley2019). In this regard, parents who experience distress associated with rumination about events during the pregnancy or birth may find telling their story to a family member or professional helpful for reaching some consoling explanation for their child’s condition. Over time, communication about important areas of palliative practice, such as goals of care, decision-making, and advance care planning requires professionals to feel comfortable with loss and grief. Studies of pediatric advance care planning have found that avoidance results in later conversations not being beneficial for parents and professionals (Carr et al. Reference Carr, Hasson and McIlfatrick2021).
Living with the prospect of losing a child is regarded as one of the most stressful and traumatic experiences for parents (Janoff-Bulman Reference Janoff-Bulman2010; Rando Reference Rando1986). Moreover parents of children with SNI live in a state of constant vigilance for signs of the next crisis, with the care journey described as an emotional rollercoaster (Rallison and Raffin-Bouchal Reference Rallison and Raffin-Bouchal2013). In bereavement literature, intensive caregiving has been found to place parents at greater risk of morbidity, mortality, and prolonged grief disorder after the death occurs (Li Reference Li, Precht and Mortensen2003). Although studies have found that outcomes are influenced by experiences during the illness, the end-of-life phase, and the circumstances of the death (Keesee et al. Reference Keesee, Currier and Neimeyer2008; Kriecberg et al Reference Kreicbergs, Lannen and Onelov2007; Wijngaards-de Meij et al. Reference Wijngaards-de Meij, Stroebe and Schut2008a), gaps remain in our understanding of anticipatory grief. At the same time, a hopeful outlook may present a challenge to professionals seeking to prepare parents for the inevitability of death. To date, interventions developed by healthcare professionals to support parents to deal with loss have tended to focus on bereavement care after the death while pediatric palliative care research has principally focused on alleviating concerns about quality of life during the end-of-life phase (Mack and Wolfe Reference Mack and Wolfe2006; Ribbers et al. Reference Ribbers, Wager and Hartenstein-Pinter2020).
This scoping review aims to synthesize what is known about healthcare professionals’ discussions of loss and grief with parents of children with life-limiting severe neurological impairment (LLSNI) to establish how these issues are considered, conceptualized, and understood within current literature. Identifying the approach, timing, frequency, and setting in which these discussions occur has implications for practitioners.
Study design
Scoping reviews are useful within health and social sciences disciplines when the aim is to seek conceptual clarification by mapping existing research prior to embarking on further research (Khalil et al. Reference Khalil, Peters and Godfrey2016). The methods employed in this review align with methodological developments since the framework was originally established by Arksey and O’Malley (Reference Arksey and O’Malley2005) and included an a priori protocol (Khalil et al. Reference Khalil, Peters and Godfrey2016) which was registered on the Open Science Framework on 8 March 2023 (10.17605/OSF.IO/ZEF5W) and can be accessed at https-//archive.org/details/osf-registrations-zef5w-v1. The review was developed and conducted in accordance with the Preferred Reporting Items for Systematic Reviews and Meta analyses – Scoping Extension (PRISMA-ScR) checklist (Tricco et al. Reference Tricco, Lillie and Zarin2018).
The PCC mnemonic (population, concept, fcontext) guided the construction of the research question, objectives, and eligibility criteria, and informed the search strategy (Khalil et al. Reference Khalil, Peters and Godfrey2016). During the early stages of the review, the definition of medical conditions to be included was reviewed and refined. Ultimately, we chose to use the term children with SNI, as this best describes the population of interest, using Allen et al.’s definition as follows; “Severe neurological impairment describes a group of disorders of the central nervous system which arise in childhood, resulting in motor impairment, cognitive impairment and medical complexity, where much assistance is required with activities of daily living. The impairment is permanent but can be progressive or static” (Allen et al. Reference Allen, Brenner and Hauer2020, p. 85). The population was defined as parents of children with a life-limiting SNI, the concept as professionals’ discussion of loss and grief, while the context refers to the timing, frequency, and settings in which discussions occurred.
Eligibility criteria
The search was exhaustive, using all years of records and countries of publication. Due to resource limitations, the search was confined to articles published in the English language. Children were defined up to the age of 18 in accordance with the UN Convention on the Rights of the Child (UN General Assembly 1989). Table 1 outlines the inclusion and exclusion criteria.
Table 1. Inclusion and exclusion criteria informing the search
Search strategy
Keywords and index terms were initially identified through preliminary searching in the title and abstract of existing reviews that were relevant to the current review across 2 databases (Cooper et al. Reference Cooper, Booth and Varley-Campbell2018). Searching these reference lists identified key articles that were then used to gather synonyms and terminology for the final search string. Boolean operators and truncation were used in each database to efficiently identify all relevant findings. Trial searches were conducted repeatedly, and adjustments made during the months of November and December 2022 to validate the search string by identifying these same key articles in the results of the search. An academic librarian was consulted throughout this process. The scope of the search extended beyond articles specifically focused on professional discussion of loss and grief to include papers that referenced this as part of a wider discussion. For this reason, the term “professional discussion” was not used as a search term and was incorporated in the inclusion criteria instead. The final keywords used included variants across the 4 terms child, parent, life-limiting neurodevelopmental disability (previous descriptor for SNI) and grief, with a sample search string developed for use in PsycINFO provided in supplementary material.
Sources of evidence
The electronic databases PsycINFO, CINAHL, and PubMed were chosen for their relevance to psychology, social sciences, nursing, and medical research. All final searches were run in January 2023 and imported to Endnote, with duplicates removed and final records uploaded to Covidence for screening. A pilot title and abstract screening of the first 30 records generated by the PsycINFO database was undertaken by 2 reviewers (EB & SG) to test and refine the eligibility criteria. As a notable number of studies reported on findings from mixed populations, the inclusion criteria were broadened to include articles where findings could be separately extracted. The remaining records were independently screened by title and abstract by the first 2 authors (EB & MC) and disagreements resolved by the third author (SG). Review and selection of full text studies that met the inclusion criteria was undertaken independently by the first author and a researcher (EB & PS), and any disagreements were resolved as above. At this stage, the reason for exclusion was recorded. Finally, reference lists of included full text studies were hand-searched for relevant articles and any records identified subjected to the same screening process.
Data extraction and synthesis
Relevant data from each of the included articles were extracted into a template by the first author (EB), including information on author, year of publication, study aims, methodology, population, key findings, and study limitations. At the same time, quality appraisal of the articles was conducted using the Mixed Methods Appraisal Tool (Hong et al. Reference Hong, Fàbregues and Bartlett2018). The extraction was piloted on 5 studies to ensure the process was effective and all relevant information extracted. Following a review of this sample, data extraction was completed by the first author.
All quantitative data were converted to qualitative data prior to analysis. Qualitative thematic synthesis (Thomas and Harden Reference Thomas and Harden2008) was used to generate descriptive and interpretative themes. Following data familiarization, 2 reviewers (EB & SG) independently assigned open codes to a sample of 5 articles. The reviewers then compared labels assigned to agree a set of codes going forward. All articles were coded in this way to develop a coding framework. Similar codes were then grouped into categories to produce a descriptive analysis of relevant data related to professionals’ discussion of loss and grief. It was noted that none of the papers identified contained explicit discussions of loss and grief between parents and professionals. However, there was clear discussion of aspects of death and dying, and 2 broad themes were developed from the analysis of these discussions: “Loss and grief is part of this context” and “Lack of recognition of loss and grief.”
Results
In total, 35 papers were identified that met the inclusion criteria, with 34 papers included in the final analysis. The search process is represented in Figure 1, while the key details from the 35 papers are presented in Table 2. One study produced 2 papers (Janvier Reference Janvier, Farlow and Barrington2016; Janvier et al. Reference Janvier, Farlow and Barrington2020), 1 produced 3 papers (Bogetz et al. Reference Bogetz, Munjapara and Henderson2022; Bogetz et al. Reference Bogetz, Revette and DeCourcey2021; Bogetz et al. Reference Bogetz, Revette and Rosenberg2020) and a third (Sullivan et al. Reference Sullivan, Gillam and Monagle2015, Reference Sullivan, Gillam and Monagle2019, Reference Sullivan, Gillam and Monagle2020, Reference Sullivan, Monagle and Gillam2014) 4 papers. In 25 papers, the participants were parents (See Table 2), in 7 they were professionals, and the remaining 3 combined both parents’ and professionals’ perspectives. In terms of the assessment of quality, the MMAT was applied to all papers. One paper failed to meet the minimum criteria (based on the screening questions) and was excluded. For the remaining papers, there were no methodological areas of concern in the body of research.
Figure 1. PRISMA flowchart documenting the search process and outcomes.
Table 2. Summary of studies included themes and illustrative findings
ACP = advance care planning, SMA = spinal muscular atrophy, EoL = end of life, SNI = severe neurological impairment, TLD = treatment limitation discussion, NICU = Neonatal Intensive Care Unit, PPC = Pediatric Palliative Care, HMV = Home Mechanical Ventilation, PND = Prenatal Diagnosis, LTLLC = life-threatening or life-limiting condition, NMLLC = nonmalignant life-limiting condition, EoLDM = end-of-life decision-making, PIMD = Profound Intellectual and Multiple Disabilities, QoL = quality of life
* Paper excluded based on MMAT.
In the parent participant studies, 13 used qualitative methods. Of these, 2 involved parents, 6 bereaved parents, and the remaining 5 both parents and bereaved parents. Ten papers used a quantitative research design. Four of these included parents who had received a prenatal diagnosis and had opted for an induced termination or where the fetus had not survived. Of the remaining 6 papers, participants in 1 were parents, with 5 papers comprised of both parents and bereaved parents. Both mixed methods papers comprised parents and bereaved parent participants.
In papers reporting professionals’ views, 2 were qualitative and 2 quantitative. The remaining 3 papers were chart reviews. The final paper used mixed methods in conjunction with a retrospective chart review.
All studies analyzed were conducted during a 10-year period from 2012 to 2022 across several countries, including America, Canada, Europe, Australia, and Malaysia. Children’s ages ranged from 1 day to 17 years, with evidence of age clusters associated with the condition. Noting that studies focused on death and dying, the majority of discussions occurred later in the trajectory.
As noted above, it was an unexpected finding that these studies did not include explicit discussions of loss and grief, however analysis of findings relating to discussions of death and dying generated 2 themes relevant to understanding loss and grief in the context of LLSNI.
Theme 1: Loss and grief is part of this context
This theme was developed in light of the unexpected finding that none of the papers identified explicitly reported on discussions between professionals and parents about aspects of loss and grief. Despite this, the analysis of these papers did isolate evidence that loss and grief is part of parents’ experience and professionals’ practice. All papers explicitly reported on discussions of death and dying, and most discussions related to medical interventions during the end-of-life phase. There was evidence of practice being attuned to parents’ anticipatory grief and bereavement needs, with findings in most papers suggesting a broad understanding of the grieving process.
Trust was key to parents’ perceptions and experiences that care received was compassionate and empathic. Several papers reported on types of supportive interactions and care that engendered trust. These included expressions of compassion and empathy, when professionals conveyed they wanted the best for the child, and respected parents’ expertise in their desire to protect and care for their child (Beecham et al. Reference Beecham, Oostendorp and Crocker2017; Lord et al. Reference Lord, Moore and Beatty2020; Zaal-Schuller et al. Reference Zaal-Schuller, Willems and Ewals2016). It was evident that professionals trusted parents knew their child best and would make the right decisions when relationships had developed over a longer illness trajectory. Furthermore, professionals who invested in relationships felt better equipped to understand parents’ perspectives and align with goals of care (Donovan et al. Reference Donovan, Slater and Delaney2022; Hurley et al. Reference Hurley, Kiernan and Price2021; Zaal-Schuller et al. Reference Zaal-Schuller, Willems and Ewals2016). In addition, knowledge of parental dynamics was perceived as helpful for determining parent readiness cues prior to the initiation of advance care planning discussions (Carr et al. Reference Carr, Hasson and McIlfatrick2022a). In this vein, a trusting relationship facilitated ongoing discussions with parents regarding quality-of-life issues as the condition progressed.
However, trust could also be established during a single conversation. Parents described experiences of interactions that supported trust when informed of their child’s diagnosis (Currie et al. Reference Currie, Christian and Hinds2016; Janvier et al. Reference Janvier, Farlow and Barrington2016; Kiernan et al. Reference Kiernan, Hurley and Price2022), specifically the use of empathetic language, when information was imparted in a balanced manner and communicated messages of hope. Parents perceived these interactions as conveying their child’s life was viewed as meaningful and valuable despite their diagnosis. Parents reported feelings of shock, loss, and grief at this time, and understanding the implications of the diagnosis was a complex mental process (Carr et al. Reference Carr, Hasson and McIlfatrick2022a). They reflected that life was unalterably changed, their hopes and dreams for their child had disappeared and they experienced uncertainty about the future (Hurley et al. Reference Hurley, Kiernan and Price2021). While the review uncovered no evidence of emotional support to help process these feelings, it was evident that nursing staff were attuned to the loss of the expected parenting experience at these times. In 2 studies, nurses were perceived to support parent-and-child bonding in challenging circumstances in the neonatal intensive care unit (Currie et al. Reference Currie, Christian and Hinds2016; Guon et al. Reference Guon, Wilfond and Farlow2014). In addition, accounts of professionals’ assisting parents to acquire new parenting skills to meet complex care needs were noted (Courtney et al. Reference Courtney, Kiernan and Guerin2018; Janvier et al. Reference Janvier, Farlow and Barrington2020).
Several papers identified that prolonging the child’s life was of major concern to parents, but this had to be balanced with the overarching priority to maintain their child’s comfort (Bogetz et al. Reference Bogetz, Revette and Rosenberg2020, Reference Bogetz, Munjapara and Henderson2022, Reference Bogetz, Revette and DeCourcey2021; Zaal-Schuller et al. Reference Zaal-Schuller, Willems and Ewals2018a). Parents held on to this liminal space by accepting their situation, normalizing the experience, and suppressing grief-related emotions (Bogetz et al. Reference Bogetz, Revette and Rosenberg2020; Carr et al. Reference Carr, Hasson and McIlfatrick2022a; Verberne Reference Verberne, Kars and Schouten-van Meeteren2019). Professional support for their goals of care alleviated some of the pressure experienced by these competing demands. Since disease progression provoked anxiety and fear, the creation and accomplishment of goals appeared to help parents feel they had some control over certain aspects of the outcome (Verberne et al. Reference Verberne, Kars and Schouten-van Meeteren2019). In this vein, communication practices that were open, honest, and understandable helped their engagement in discussions and decisions about future care needs. While accepting the inevitable deterioration in their child’s condition required continual adaption, parents felt supported when professionals acknowledged the sacrifices they made.
Findings, however, suggested that parents experienced advance care planning discussions as simultaneously supportive and anxiety provoking. In one paper, the introduction of palliative care was perceived as a clear sign of disease progression and the next step in the child’s journey (Verberne et al. Reference Verberne, Kars and Schouten-van Meeteren2019). While papers identified differences in professional practices regarding the timing of these discussions, it was evident that the involvement of palliative care teams facilitated discussions about the child’s impending death, particularly when parents perceived the regular medical team to be uncomfortable discussing death (Carr et al. Reference Carr, Hasson and McIlfatrick2022a). It encouraged the contemplation of goals and wishes, memory-making, and greater attention to symptom management and quality of life issues (Donovan Reference Donovan, Slater and Delaney2022; Lord et al. Reference Lord, Moore and Beatty2020). Legacy-making activities enabled parents to mentally prepare for their child’s end-of-life, acknowledged their parenting role, and enhanced their sense of control by affirming parents’ decision-making at the end-of-life (Beecham et al. Reference Beecham, Oostendorp and Crocker2017). While not reflecting a discussion with parents about loss, in one study professionals stated the involvement of parents in end-of-life decision-making facilitated the grieving process, showing an understanding of this issue while not explicitly addressing it with the parent (Zaal-Schuller et al. Reference Zaal-Schuller, Willems and Ewals2016).
Post-loss, caring for the child’s body was identified as something parents valued along with being able to spend time with their child to say goodbye (Lövgren et al. Reference Lövgren, Sejersen and Kreicbergs2016). Integrating bereavement supports with end-of-life care by sign-posting sources of support was linked to increased satisfaction with care (Walker et al. Reference Walker, Miller and Dalton2008), although some parents advocated that bereavement care should ideally be delivered by the treating team or counsellors familiar with complex needs (Lord et al. Reference Lord, Moore and Beatty2020).
Theme 2: Lack of recognition of loss and grief
While parents recognized that palliative care was challenging work, they felt the onus was on professionals to understand what they were going through, to express empathy and compassion, and adjust their practice accordingly. This contributed to the development of this theme.
Poor communication practices at the diagnostic stage were perceived to have implications for parental acceptance of the diagnosis and prognosis and set a tone for future encounters with professionals. Parents reported that the shock of the diagnosis impacted on their ability to process and understand what they were being told (Carr et al. Reference Carr, Hasson and McIlfatrick2022a). Parents reported feeling like “their world had fallen apart” (p. 29, Hurley et al. Reference Hurley, Kiernan and Price2021) but received no support from healthcare professionals to process their emotions. Parents described being informed of their child’s diagnosis by professionals who used insensitive or derogatory language, as well as unfamiliar medical terms (Janvier et al. Reference Janvier, Farlow and Barrington2016, Reference Janvier, Farlow and Barrington2020). Years later, parents were able to recall the exact words used during such encounters. Other factors that contributed to perceptions that professionals lacked awareness of parents’ emotional distress included diagnostic information imparted in inappropriate settings, alone, and without specific information on the condition. Moreover, remarks overhead regarding potential survival or future abilities were perceived as a judgment on the value of their child’s life (Janvier et al. Reference Janvier, Farlow and Barrington2016). These troubling experiences were etched in parents’ memories and became part of their story.
Prognostic uncertainty gave rise to tensions within parent-professionals’ relationships when it was perceived that care delivered was not individualized to family needs (Bogetz et al. Reference Bogetz, Munjapara and Henderson2022; Heckford and Beringer Reference Heckford and Beringer2014). Papers indicated that while parents preferred information to be delivered in a straightforward way, differences in personality and coping styles influenced preferences regarding the detail, timing, and nature of information imparted (Beecham et al. Reference Beecham, Oostendorp and Crocker2017). For example, some parents perceived there had been missed opportunities to plan for the end-of-life and were given false hope when positives were emphasized in treatment discussions, whereas others coped by prioritizing the “here and now” to normalize life and tended to avoid prognostic discussions (Bogetz et al. Reference Bogetz, Revette and DeCourcey2021). In addition, parents highlighted improvements in communication across various agencies involved in their child’s care would enhance the care received (Kiernan et al. Reference Kiernan, Hurley and Price2022).
In total, 2 papers reported that bereaved parents viewed their involvement in end-of-life decisions improved bereavement outcomes (Carr et al. Reference Carr, Hasson and McIlfatrick2022a; Zaal-Schuller et al. Reference Zaal-Schuller, Willems and Ewals2018a). However, preparing for the end-of-life was also a time when unsettling disagreements could occur. In particular, discussions about withdrawal of treatment were perceived to be acrimonious when goals of care and values differed (Bogetz et al. Reference Bogetz, Revette and DeCourcey2021). In 2 studies, parents perceived that professionals’ candid descriptions of the child’s likely physical deterioration, as well as their repeated reminders of the child’s poor prognosis were used to influence their treatment decision-making (Beecham et al. Reference Beecham, Oostendorp and Crocker2017; Hurley et al. Reference Hurley, Kiernan and Price2021). Notably, these studies also reported occasions when overwhelming emotions hampered parents’ ability to make important decisions that could affect treatment outcomes. Parents wanted professionals to take control at such times.
It was evident that the uncertain trajectory had an influence on the timing of advance care planning discussions. These discussions typically occurred in either hospital or hospice settings. Bereaved parents felt discussions should ideally have occurred earlier and regularly, whereas many proceeded a hospital admission following a deterioration in the child’s condition. Evidence of inconsistent access to advance care planning before the child’s death were identified in 3 studies (Carr et al. Reference Carr, Hasson and McIlfatrick2022a; Guerin et al. Reference Guerin, Kiernan and Courtney2020; Heckford and Beringer Reference Heckford and Beringer2014). Findings from one study indicated that where curative management was ongoing, professionals were unlikely to discuss the likelihood the child would die until close to death (Heckford and Beringer Reference Heckford and Beringer2014). In this vein, professionals’ discomfort caused frustration to parents who sought to know what to expect when their child reached the end-of-life. While palliative care was perceived to give permission for discussions about end-of-life plans (Sullivan et al. Reference Sullivan, Gillam and Monagle2019), in one study, palliative professionals expressed a view that the majority of medical teams failed to recognize the palliative care needs of children with SNI (Guerin et al. Reference Guerin, Kiernan and Courtney2020).
Several papers identified the loss of long-standing medical relationships after death as a further significant loss (e.g., Lövgren et al. Reference Lövgren, Sejersen and Kreicbergs2016). Parents expressed a view that since these professionals were now part of their child’s legacy, maintaining contact, post-loss, was important for their grieving process.
Discussion
It was evident that parents were confronted with loss from the moment the devastating news of their child’s diagnosis was first imparted. The reality of being separated from their child at some point in the future provoked anxiety and fear, with parents engaging in a complex mental process to comprehend their situation and to cope with challenging emotions over time. Uncertainty in prognosis and trajectory were perceived to have both redemptive and unsettling consequences. On the one hand, it enabled parents to advocate to ensure their child’s needs were prioritized, to be a “good parent” in the time they had, and to live life to the full (Donovan et al. Reference Donovan, Slater and Delaney2022). In this respect, parents perceived it as important that their child was treated as normally as possible. At the same time, reminders of their child’s shortened lifespan, whether subtle or obvious, prompted parents to reevaluate goals, and to engage in a deeply emotional process to adjust to their loss (Carr et al. Reference Carr, Hasson and McIlfatrick2022a). While parents demonstrated personal resilience in normalizing the profound challenges that confronted them (Verberne et al. Reference Verberne, Kars and Schouten-van Meeteren2019), there was evidence that professionals who showed empathy and compassion (Hurley et al. Reference Hurley, Kiernan and Price2021) took time to understand the whole family’s needs (Beecham et al. Reference Beecham, Oostendorp and Crocker2017), and acknowledging their parenting role aided them to move forward (Lövgren et al. Reference Lövgren, Sejersen and Kreicbergs2016). Findings suggested that parents turned to known and trusted professionals, who had intimate knowledge of what the family had been through to make sense of loss (Kiernan et al. Reference Kiernan, Hurley and Price2022). Finally, involving parents in end-of-life planning, as well as encouraging their participation in legacy-making activities showed that professionals were attuned to what might be helpful to grieving parents after the child died (Donovan et al. Reference Donovan, Slater and Delaney2022). However, in only a small number of papers were these practices explicitly linked to improved bereavement outcomes.
The importance of interactions with healthcare professionals, as well as symptom-management at the end-of-life has been shown to influence long-term parental grief (van der Geest et al. Reference van der Geest, Darlington and Streng2014). In the current review, parents of children with SNI reported mixed experiences of professional interactions, with particularly troubling experiences described during the diagnostic period. Parental distress resulting from these interactions appeared to linger and added to their loss (Lövgren et al. Reference Lövgren, Sejersen and Kreicbergs2016). While reflecting on these events after the death, it is conceivable that parents may have experienced regret and unfinished business. While the literature on regret within pediatric populations is limited, regret in adult populations has been shown to be linked with reduced quality of life, problems with grief symptomatology, and depression (Holland et al. Reference Holland, Plant and Klingspon2020). In this vein, the review highlighted certain practices that were seemingly undertaken to facilitate positive reflections regarding the care received after the death. Professionals who provided emotional and instrumental support in the home were uniquely placed to observe parents’ workload and the tremendous strain on parents to extenuate periods when the child was well. Parents’ attempts to make sense of disease progression by clarifying medical information or reviewing the child’s quality of life with known and trusted professionals appeared to have a buffering effect, and aid parental coping over time (Kiernan et al. Reference Kiernan, Hurley and Price2022; Zaal-Schuller et al. Reference Zaal-Schuller, Willems and Ewals2018b). In bereavement, sense-making has been found to account for 15 times the intensity of parent’s distress than other variables such as gender, type of death, or length of time bereaved (Keesee et al. Reference Keesee, Currier and Neimeyer2008).
While previous research has shown that preparation for end-of-life is helpful for dealing with anticipatory grief and facilitates the grieving process (D’Agostino et al. Reference D’Agostino, Berlin-Romalis and Jovcevska2008; Kreicbergs et al. Reference Kreicbergs, Lannen and Onelov2007), the review highlighted the tendency for discussions to proceed following a deterioration in the child’s condition (Carr et al. Reference Carr, Hasson and McIlfatrick2022b). Nevertheless, there was consistent evidence that the introduction of palliative care was associated with a greater focus on family-centered care (Donovan et al. Reference Donovan, Slater and Delaney2022). Findings however, suggested that not all children had timely access to palliative services, if they received them at all (Guerin et al. Reference Guerin, Kiernan and Courtney2020). A study by Price et al. found that different trajectories lead to inequitable provision, with services for nonmalignant conditions less well-developed compared to those for pediatric cancer patients (Price et al. Reference Price, Jordan and Prior2012). These findings suggest the uncertain trajectory for children with SNI may contribute to missed opportunities for referrals to palliative services. The review highlighted that offering choice about location of care and death facilitated parents to maintain their parenting role to the end, through their involvement in decisions and the physical care of their child. Findings also evidenced professional engagement in legacy-making tasks. In bereavement, legacy activities play an important role in maintaining a relationship with the deceased post-loss (Klass et al. Reference Klass, Silverman and Nickman2014), however such activities were not explicitly linked to bereavement outcomes in the papers reviewed. Therefore, it was unclear whether professionals, in actuality, understood the theoretical basis underpinning these practices. Finally, being facilitated to spend time with the dying child in private to say goodbye and attend to the body after death was valued by parents. Previously, Wijngaards-de Meij et al. (Reference Wijngaards-de Meij, Stroebe and Stroebe2008b) found that the opportunity to say goodbye was associated with lower levels of grief in parents. However, it seemed only those parents who accepted their child would eventually die were open to these types of interventions.
With regard to the quality of the research, the MMAT findings would suggest that the studies meet the minimum criteria. However across the research there are some consistent limitations including a reliance on convenience or purposive sampling, a concern about the generalizability of the samples based on medical and other criteria, a lack of access to consistent information where audits are conducted and a possible over reliance on mothers’ reports. While these are not fatal methodological flaws, they do limit the representativeness of the research. In terms of the quality of the review, the work done to adhere to international guidelines for scoping studies and the involvement of multiple researchers in screening and interpretation give confidence. However it is notable that the review was not able to answer the main research question as originally intended.
Overall, these findings point to major gaps in the evidence base and our understanding of professionals’ practice in this area, although it is possible that discussions on aspects of death and dying may have been richer and more nuanced than was reported. Nevertheless, the significant impact that professionals’ have in the lives of these parents is clear from the papers reviewed, together with first-hand knowledge of the losses faced during their child’s trajectory. Going forward, it is recommended that research focus on further exploration of professional views to elicit whether more can be done to enhance bereavement outcomes given the trajectory of children with life-limiting SNI. To this end, it would be important to elucidate the evidence for practice that aligns with or is at variance with current bereavement research. In addition, the views of parents regarding their support needs were not adequately captured in the findings reported and this needs further investigation given the implications for evidence-based practice. Finally, since this area causes enormous anxiety for professionals, identifying training and support needs would benefit practitioners.
Conclusion
The review revealed major gaps in our understanding of professionals’ discussions of loss and grief within this population. Notably, most papers did not consider bereavement outcomes when reporting findings. Despite this, all papers discussed aspects of death and dying, and findings suggest that professionals implicitly provided support for grief in this context. The focus on the end-of-life phase provided limited scope for evidence of discussions earlier in the trajectory. Findings point to the possibility that prognostic uncertainty may contribute to less well-developed palliative services for children with SNI. Initial ideas to advance evidence-based knowledge have been highlighted.
Supplementary material
The supplementary material for this article can be found at https://doi.org/10.1017/S1478951524001743.
Acknowledgments
The authors would like to thank Ms Philie Sheehan for her support with aspects of the systematic review. We would also like to thank LauraLynn Ireland’s Children’s Hospice for their support of this research through the Mc Kenna Scholarship.
Funding
This work was supported by LauraLynn, Ireland’s Children’s Hospice, through the McKenna Scholarship awarded to Elaine Brennan.
Competing interests
The author(s) declare none
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