2. Parental Bereavement Following the Death of a Child with a Life-Limiting Condition
There are a number of key differences between parental bereavement processes associated with the death of a child with a prolonged, life-limiting condition, relative to an unexpected death due to an injury or acute illness [4
]. Parents looking after a child with a life-limiting condition commonly experience prolonged suffering in the weeks, months, and years before the death of the child. In some cases, they may have learnt about their child’s diagnosis around the time of their birth. The child’s death may occur at a point when a parent’s coping resources have been tested to the limit across a range of possible areas (e.g., physical, emotional, marital, financial, spiritual, and inter-personal), often for a long period of time. On the one hand, the draining nature of a prolonged period of caring may leave an individual vulnerable to poorer bereavement outcomes. Conversely, however, the stress reduction theory holds that the child’s death coincides with a reduction in stressors that had been associated with the long-term care of the child, thus potentially facilitating post-bereavement adjustment [6
]. It is extremely difficult to compare bereavement outcomes for expected and unexpected child deaths in a methodologically rigorous way, and results have been mixed. One study found lower levels of parental depression following an unexpected death relative to a long-term illnesses [7
], whereas another study found poorer parental bereavement outcomes following a child’s violent death (e.g., resulting from accident, homicide, and suicide) relative to a long-term illness [8
In the context of caring for a child with a life-limiting condition, parental grief processes may have started well before the child’s actual death, with similarities (albeit also some differences) between anticipatory mourning/grief reactions and conventional grief reactions [9
]. Upon learning of a child’s diagnosis and/or prognosis, the parents of a child with a life-limiting condition may start to readjust their cognitive schemas, to accommodate the recognition that their child might not experience a “normal” childhood, nor live to adulthood. However, this recognition does not necessarily equate with preparedness. Indeed, this realization may be associated with such a burden to parents that some authors have described the potential for the draining and debilitating experience of “chronic sorrow” [10
The parents of children with a life-limiting condition may be more likely than the parents of children following an injury or acute illness to be aware if their child’s impending death is imminent. Parents commonly report that the knowledge of their child’s impending death enabled them to make appropriate choices, engage in tasks they deemed most important prior to their child’s death, and to say goodbye to their child [11
]. These actions are likely to help minimize regrets that the parent may feel after their child’s death. Nevertheless, in the context of caring for a child with a life-limiting condition, it is not uncommon for parents to experience several occasions when they believe their child’s death to be imminent, thus making it difficult for them to know when it is really time to say a final goodbye.
It is important to acknowledge differences in parental bereavement processes following the death of a child due to cancer, relative to the death of a child due to a non-cancer, life-limiting illness. In the context of many life-limiting, non-cancer conditions, parents typically learn at the time of diagnosis that, although there may be uncertainty regarding how long their child is likely to live, they as parents are likely to outlive their child. This information is likely to gradually reshape their cognitive schemas. In contrast, parents whose child has been diagnosed with cancer, may recognize the possibility of their child’s death, however, they typically retain hope that their child will make a full recovery and enjoy a full life expectancy. These parents are therefore more inclined to cling to their existing schemas for as long as possible, hoping that the treatments will enable a return to health and normality.
3. The Role of Risk and Resilience Factors in Parental Bereavement
Over the past decade the Integrative Risk Factor Framework of Bereavement [3
] has been valuable in drawing attention to a broad array of risk factors that may adversely impact on parental bereavement. This framework grouped potential risk factors as: (i) loss-oriented and restoration-oriented stressors; (ii) intra-personal factors, which are stable factors that are intrinsic to the bereaved individual; (iii) inter-personal factors, which are stable factors external to the individual (e.g., social support and culture); and (iv) coping and appraisal. These factors, alone and in combination, have the potential to impact on the ability of bereaved parents to resume functioning with necessary and valued activities. However, advances in the resilience literature suggest that intra-personal factors and inter-personal factors should not only be considered as potential risk factors for poorer bereavement outcomes, but that they should also be considered as possible resilience factors [12
]. Although a risk-focussed approach has been useful in some fields of medicine, such as investigating infectious diseases, when investigating more complex conditions with biopsychosocial components, a more comprehensive consideration of both risk and resilience factors is likely to be beneficial [14
Resilience may be regarded as an individual’s ability to respond effectively to challenges or adversity. In some cases, resilience factors may be the opposite end of the spectrum of risk factors, for example, good marital communication may be considered a resilience factor, whereas poor marital communication may be a risk factor. However, risk and resilience factors are not always at opposite ends of the same continuum. For example, substance abuse confers a risk, but it cannot be said that its absence confers any protective value.
Some definitions of resilience highlight an individual’s sustainability of purpose in the face of stress [13
]. In recent years, the concept of resilience has been applied to the health psychology literature to identify why some individuals adjust to chronic health stressors more readily than others [15
]. Resilience has been identified as being of importance in the bereavement literature, with the potential to help account for why some individuals are able to resume functioning more readily than others, despite experiencing painful and life-changing losses [17
]. From a theoretical perspective, resilience factors may operate in a number of ways. Firstly, they may buffer or serve to compensate or minimize the effect of the stressor or loss in some way. For example, having other children in the family may prevent a parent from facing a childless existence, thus enabling, indeed requiring, them to maintain their role as parent. Secondly, resilience factors may facilitate the individual’s process of recovery. In this case, the stressor or loss may be experienced just as acutely, but the resilience mechanisms may facilitate coping and accelerate the process of recovery [19
]. For example, good marital communication may enable the bereaved parents to assist each other with more effective problem-solving.
The current paper draws from the multi-faceted risk framework of bereavement outlined by Stroebe et al. [3
] and integrates this framework with a more comprehensive consideration of resilience factors. Thus, we have proposed a new model, namely the Risk and Resilience Model of Parental Bereavement (see Figure 1
). Like the Stroebe et al. [3
] framework of risk factors, this model groups loss-oriented factors, intra-personal factors, inter-personal factors, and appraisal and coping, separately. However, each of these classes of factors is considered in terms of potential risk and resilience influences on parental bereavement outcomes. This more comprehensive and holistic framework for considering how multiple risk and resilience factors interact is paramount to an improved understanding of parental bereavement outcomes, promoting theoretically-driven research, and guiding evidence-based clinical practice. The specific factors outlined in Figure 1
have been included based on available empirical or theoretical justification. Following a brief discussion of the varied nature of parental bereavement outcomes, the literature on each of the four classes of bereavement risk and resilience factors will be reviewed, where possible with a particular focus on parental bereavement associated with the death of a child following a life-limiting condition.
4. Parental Bereavement Outcomes
Whilst there is consensus that grief is a normal experience following a major loss, it is difficult to define the process of normal grief. It is generally recognized that the grief response is dynamic, pervasive and highly individualized [21
]. The process of grief is not linear and does not fit neatly into predetermined categories. The death of a child commonly results in detrimental effects on the psychological and physical well-being of parents [22
]. Psychological responses to parental bereavement may include heightened anxiety, depression, suicidal ideation, and reduced quality of life [22
]. Increased risk of psychiatric hospitalization has also been reported, especially in mothers [27
]. Detrimental physical outcomes that have been reported in response to parental bereavement include a greater risk of cardiovascular problems [28
], cancer [26
], and higher rates of mortality due to natural and unnatural causes [26
]. A wide range of detrimental social [29
], marital [30
], occupational and financial consequences [31
] have also been reported amongst bereaved parents.
The adverse outcomes listed above are certainly not experienced by all individuals, with there being considerable variability in symptoms experienced. Moreover, there is also much variability in the duration of intense grief reactions [22
]. Most bereaved individuals return to relatively normal functioning, as judged by external standards, within a relatively short time-frame [1
], even if their experience of life is now different.
Over the years, various terms and classifications have been used to describe intense and debilitating grief reactions, including persistent complex bereavement disorder, prolonged grief disorder, bereavement-related major depression, complicated grief, pathological grief. Although there remain differences in opinion as to how best to classify these individuals [32
], it is generally recognized that 5–10% of bereaved individuals experience significant and prolonged impairment of functioning [34
]. It is not clear what these figures are for parental bereavement following the loss of a child due to a life-limiting condition, however it has been found that the context of an expected death poses lower risk than unexpected deaths [35
]. Where data are available, the current paper will consider risk and resilience factors associated with such intense and debilitating grief reactions; however, the paper will primarily encompass discussion of risk and resilience factors associated with the full spectrum of grief reactions.
8. Coping and Appraisal
Coping may broadly be considered as the process by which an individual appraises the personal significance of a situation or event and the options that they have for responding to that situation or event [3
]. Over the years, individuals typically develop a tendency to utilize certain coping styles. However, when faced with specific challenges, the individual must appraise the situation and their capacity to respond to it, and apply particular coping strategies accordingly.
Coping is known to be associated with, and likely to mediate, the relationship between interpersonal and intrapersonal variables with adjustment [3
]. Within the context of bereavement, the coping process is of particular importance because it offers possible targets for effective intervention, given that these constructs may be amenable to change [3
]. In an early paper addressing intervention strategies for coping with transitions, a useful list of coping competencies was outlined [117
]. These coping competencies included skills for assessing, developing and utilizing internal and external resources, skills for managing emotional and physiological distress, and skills associated with planning and implementing change. Despite these transition-based coping competencies being articulated more than 35 years ago, more research is needed to investigate the role of coping interventions in the context of parental bereavement.
The majority of the literature on coping pertains to situations that have occurred and present a current challenge or threat to the individual. However, a small body of literature addresses what has been referred to as proactive coping, pertaining to anticipated threats or stressors [118
]. The literature regarding bereavement following the death of a child due to a life-limiting condition would benefit from a consideration of both types of coping, given that parents face many ongoing stressors whilst caring for a child with a life-limiting condition, whilst also being mindful that they will one day need to face their child’s death. In other contexts, proactive coping has been found to be beneficial, and indeed teachable, heightening an individual’s awareness of their personal and social resources, so that they are better placed to make effective coping decisions [119
]. However, the concept of proactive coping has, to date, not been well applied to parental bereavement following a child’s death due to a life-limiting condition, and warrants further research.
It is generally recognized that specific coping strategies are not uniformly effective (or ineffective) across all contexts and situations. Instead, an individual’s coping efficacy is dependent on an efficient process of self-regulation, which requires an awareness of contextual demands, availability of a range of coping skills to select from, and responsivity to internal and external feedback [80
]. A small-scale study investigating the coping of bereaved parents found that the coping strategies used by those bereaved for 18 months or less differed considerably from non-bereaved normative samples, whereas those bereaved for more than 18 months engaged in coping strategies similar to normative samples [120
]. The efficacy of the coping strategies, however, was not examined.
Within the bereavement literature, Stroebe and Schut [95
] made a distinction between loss-oriented coping (namely a focus on appraising and processing some aspect of the loss experience) and restoration-oriented coping (namely a focus on reorienting oneself in a changed world without the deceased person). The dual process theory of bereavement holds that oscillation between these different types of cognitive processing is essential for adaptive bereavement outcomes and that over time more focus is placed on a restoration orientation and less on a loss orientation [121
]. In a study of 219 couples following the death of their child, Wijngaards-de Meij et al. investigated parental use of restoration-coping and loss-oriented coping [123
]. Although utilizing quite a limited set of items to assess coping, Wijngaards-de Meij et al. found that a greater focus on future-oriented, restoration-coping, irrespective of the amount of loss-oriented coping that was used, was associated with more beneficial outcomes [123
]. Moreover, when women made greater use of restoration coping, their husbands also benefited, showing lower levels of depression.
Although psychological flexibility is sometimes regarded as a personality dimension, it may also be considered as a more malleable, cognitive coping process. Within the Acceptance and Commitment Therapy (ACT) framework, psychological flexibility has been taken to refer to an ability to be present-focussed, acting in a manner consistent with one’s values, even in the presence of interfering thoughts and emotions [93
]. ACT interventions with the parents of children with a life-limiting condition have successfully increased aspects of parental psychological flexibility [124
]. However, to our knowledge, studies have not investigated parental psychological flexibility following the death of a child following a life-limiting condition.
Similarly, within the body of literature on mindfulness, the concept of acceptance has emerged as being of importance in the face of life stressors [125
]. Within the trauma literature, acceptance has been found to be associated with greater psychological adjustment following exposure to trauma (for review, see [125
]). Within the context of bereavement, the concept of acceptance has been integral to clinical mindfulness interventions that have been developed, such as based on the ATTEND (attunement, trust, touch, egalitarianism, nuance, and death education) framework [81
]. However, evaluations of these interventions have thus far been limited [126
Meaning-making is a term that has emerged and proliferated in the literature over the past decade, referring to the restoration of meaning following a highly stressful situation [127
]. Meaning-making requires the integration of the meaning given to a stressful event with one’s global orienting system or cognitive framework [127
]. Highly stressful situations have the potential to challenge one’s global cognitive systems. The extent to which the meaning that an individual attributes to a stressful event is discrepant with their global cognitive system is likely to determine the extent to which they experience distress. It has been found that the degree to which parents have made sense or meaning out of their child’s unexpected death was inversely related to their degree of distress [128
], albeit one study found this relationship was only significant in the first year of bereavement [129
The nature of a child’s death has been found to impact on the ability of parents to find meaning in the situation. Parents whose child died a violent death (i.e., accident, homicide or suicide) found it more challenging to make-meaning of the situation relative to parents whose child died a non-violent death (perinatal, natural anticipated, or natural sudden) [130
]. There has been little work investigating the process of meaning-making specifically among parents of children who died from a life-limiting disorder.
Positive emotions have been suggested to serve a restorative role in bereavement, and as a catalyst for meaning finding and benefit finding [92
]. Moskowitz, Folkman and Acree [79
] noted a positive association between positive affect and bereavement outcomes, which they attributed to an increased likelihood to engage in positive reappraisal. Moreover, a positive affect renders individuals more able to seek social support. However, the overlapping nature of measures of positive emotions and of bereavement outcomes makes it difficult to disentangle these constructs or to consider issues of causality.
Rumination is a coping style characterized by recurrent, self-focused negative thinking. It is widely regarded as a normal part of grieving. However, more extreme rumination is likely to be problematic and a predictor of poorer bereavement outcomes [131
]. In a study with 55 bereaved individuals (following the death of a first degree relative within the previous three years), greater rumination was associated with symptoms of psychopathology over a 12-month period [132
]. It has been suggested that the repeated focus of attention on negative emotions associated with rumination interferes with problem solving capabilities, and impedes instrumental behaviour and the utilization of social support [131
]. Females and individuals with lower social support have been found to engage in more ruminating behaviour following the death of a loved one [116
Rumination used to be considered a confrontational strategy, requiring individuals to confront and experience distress and negative emotions. More recently, though, rumination has been appraised as an avoidance strategy [133
], whereby an individual focuses disproportionately on loss-oriented coping with little attention to restoration-oriented coping. According to this view, individuals engage in ruminative thinking, dwelling on negative aspects of their personal loss, and in so doing avoid confronting the new realities of their life and fail to restructure their cognitive schemata accordingly.
The nature of the cognitive appraisal that an individual engages in may be influenced by their cultural beliefs. For example, individuals holding traditional Chinese beliefs are more likely than individuals holding Western beliefs to adopt an external locus of control and attribute the cause of a death to predestined rules or higher powers such as sick qi
(negative energy that could pass to the unlucky) or evil spirits [134
]. This remains an under-researched area.
9. Challenges to Carrying Out Bereavement Research
Studying the bereavement process and factors associated with parental bereavement outcomes is fraught with challenges [135
]. Key areas of difficulty relate to participant recruitment, inter-relatedness of variables, and the selection and use of multiple outcome measures. Each of these areas of difficulty will be briefly described.
Research participation is voluntary; hence self-selection is likely to influence who participates in bereavement research [136
]. Individuals may decline to participate due to: depressed mood, feeling they are too upset to answer questions about their bereavement, fear that participation may increase their grief, or greater use of avoidant coping strategies. It is therefore possible that individuals who are most disabled by grief, perhaps meeting the Diagnostic and Statistical Manual of Mental Disorders—5th Edition (DSM-5) criteria of persistent complex bereavement disorder, may commonly not participate in bereavement research. Individuals who agree to participate in bereavement research may be more willing to talk about their experience, and therefore may already be engaging in more adaptive coping strategies. Notably, given that women are more likely to seek social support and talk about their feelings and experiences [46
], they may be more likely to engage in bereavement research. This issue is likely to be magnified if recruitment occurs through bereavement support services, which may predominantly be utilized by individuals who are willing to talk about their experiences. Alternatively, it is conceivable that individuals who are more distressed may be more likely to take up the opportunity to talk with someone about their feelings [136
]. Moreover, there is evidence of a selective invitation bias in paediatric palliative care research, whereby not all eligible families are invited to participate due to non-random factors [137
Factors that potentially render parents at greater or lower risk of poor bereavement outcomes are often difficult to disentangle from complex, inter-related circumstances and attitudes. For example, decisions regarding the preferred location of the child’s death may be related to the family’s coping efficacy, their perceptions of how well their child’s symptoms are able to be managed outside of hospital, the supports and services available to the family, and considerations regarding the presence of other siblings. Randomization is rarely appropriate to study such factors in a methodologically rigorous way. A multivariate statistical approach would also be useful in minimizing the reporting of spurious associations, but requires an adequate sample size.
When carrying out research with bereaved individuals it is difficult to find the right balance between assessing multiple outcome domains of possible interest and not wanting to over-burden bereaved parents. Many studies have considered only a single measure of bereavement outcome, failing to acknowledge the complex and varied ways in which different individuals respond to the death of a child. For example, it is increasingly recognized that mothers and fathers experience the loss of a child differently [108
]. Some outcome measures may be more sensitive to identifying the responses of women rather than men, or vice versa.
10. Future Directions for Research and Clinical Practice
It is important to achieve better alignment between quality research in the area of parental bereavement, particularly in the context of a death following life-limiting condition, and clinical practice. Although the natural trajectory of bereavement has been documented in the context of bereaved older spouses [138
], at present the natural history of bereavement in parents following the death of a child has not been well studied. Prospective studies in this area are needed, though challenging due to the relatively small numbers of children known to be approaching death and the difficulty of engaging parents at this time. Multi-centre collaboration would be useful to achieve sufficient sample sizes. Alternatively, the use of large-scale, longitudinal databases (i.e., Big Data) would not only provide useful longitudinal data, but, importantly, also avoid many self-selection and recruitment biases common in this area of research, given that participation is not specific to their bereavement status.
The evidence for clinical interventions with bereaved parents is currently poor [139
]. In a systematic review by Endo et al. [139
], nine articles were retrieved, describing eight randomized controlled trials of clinical interventions with bereaved parents or siblings following a child’s death. The interventions were varied, and included support groups, counselling, psychotherapy and crisis intervention. However, the authors of the systematic review concluded that there was limited evidence of sufficient quality to support the intervention techniques used. Similarly, the literature in other areas of bereavement suggests that most individuals regain their pre-loss levels of functioning after a transitory period of distress (e.g., 6–12 months) irrespective of whether they receive any intervention [140
]. The authors of an earlier meta-analysis evaluating the efficacy of psychotherapeutic interventions for bereavement concluded that more favourable outcomes were obtained for programs that specifically targeted bereaved individuals experiencing most marked difficulties [141
Within the context of the Risk and Resilience Model of Parental Bereavement proposed in this paper, the current review has identified various inter-personal and intra-personal factors that may positively or detrimentally impact parental bereavement outcomes. These risk and resilience factors may be identified in the weeks, months, or even years prior to a child’s death within the supportive context of a relationship with a palliative healthcare provider. Many risk factors, such as low social support, previous losses, predisposing personal vulnerabilities (such as psychiatric history or history of substance abuse), are likely to be identifiable through clinical interview. Other factors, such as attachment style, trait mindfulness and psychological flexibility, may warrant the use of brief, validated questionnaires. A clearer identification of which parents are at greatest risk of adverse bereavement outcomes will help pave the way for the development and evaluation of targeted interventions. Consideration should be given to the possibility of enhancing the resilience of parents, arguably even prior to their child’s death, such as by enhancing mindfulness, acceptance and psychological flexibility. Importantly, the Risk and Resilience Model of Parental Bereavement highlights the importance of considering both risk and resilience factors, and how these may, in combination, impact on bereavement outcomes.
A number of key issues warrant further research in order to better inform the development of evidence-based clinical interventions. If factors such as psychological flexibility and mindfulness are indeed associated with more favourable parental bereavement outcomes, how can these coping styles be taught? At what point should they be taught—before or after a child’s death? Would all parents facing bereavement benefit from these approaches, or is there a subset of parents who would receive most benefit? Notably, Bonanno [17
] cautioned against assuming that there is a single resilience pathway. It may be that individuals with certain risk factors receive particular benefit from specific resilience factors that serve to compensate for the risk. More research is needed to address these questions.