Grief reactions in dementia carers: a systematic review

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1 REVIEW ARTICLE Grief reactions in dementia carers: a systematic review Diana Chan 1, Gill Livingston 2, Louise Jones 1 Elizabeth L Sampson 1 1 Marie Curie Palliative Care Research Unit, UCL, London, UK 2 Department of Mental Health Sciences, Archway campus, Highgate Hill, London, N19 5NL, UK Correspondence to: D. Chan, diana.chan.09@ucl.ac.uk Objective: Supporting dementia carers is an identified target of the UK government, yet we know little about such family carers grief before after the death of the person with dementia for whom they care. We systematically review the existing literature on characteristics, prevalence, predictors associations of grief in dementia carers before after death. Methods: We searched electronic databases found 31 publications meeting predetermined criteria. Results: Grief in dementia carers, which may be normal or complicated, is a complex reaction to losses occurring before after death. Carers experience anticipatory grief as multiple losses for themselves (companionship, personal freedom control) the person with dementia. Anticipation ambiguity about the future, anger, frustration guilt are core features. Anticipatory grief is greatest in moderate to severe stage dementia spouse carers, especially when the person with dementia is institutionalised. There was poor quality evidence about the prevalence of grief; studies reported anticipatory grief between 47% 71%, complicated grief after death is estimated around 20%. Carer depression increases with anticipatory grief. Being a spouse carer being depressed are the strongest predictors of complicated normal grief after death. Conclusion: Grief in dementia carers can be expected; however, those at risk of distressing anticipatory complicated grief may be identified targeted for intervention when necessary. Higher quality research from a wider range of samples countries is needed to explore this complex emergent topic. Copyright # 2012 John Wiley & Sons, Ltd. Key words: grief; dementia; carers; bereavement History: Received 8 August 2011; Accepted 3 February 2012; Published online in Wiley Online Library (wileyonlinelibrary.com) DOI: /gps.3795 Introduction Grief is a normal complex psychological emotional reaction occurring in response to a significant loss. One definition is the reaction to the perception of loss with symptoms including yearning, sadness, anger, guilt, regret, anxiety, loneliness, fatigue, shock, numbness, positive feelings a variety of physical symptoms unique to the individual (Ro, 2000). Complicated grief is recognised in clinical practice but ill defined as a syndrome; yet diagnostic criteria proposed for the fifth Diagnostic Statistical Manual of Mental Disorders of the American Psychiatric Association the 11th International Classification of Diseases suggest the possible diagnosis in someone who experiences cognitive, behavioural emotional symptoms or separation distress to a disabling degree, causing significant impairment in social or occupational functioning for more than 6 months after bereavement (Prigerson et al., 2009). Psychiatric co-morbidity (post traumatic stress disorder, depression, anxiety (Simon et al., 2007), suicidal thoughts (Prigerson et al., 1997a)) poor physical health (Prigerson et al., 1997a) associated with complicated grief suggest that early interventions from health professionals for these carers might be helpful.

2 D. Chan et al. Anticipatory grief occurs before the death of a family member (Ro, 1986). This may be especially relevant in dementia as the median life span after diagnosis is 5 8years (Xie et al., 2008). Cognitive decline, loss of personal autonomy, lack of insight coping with difficult behaviours makes it difficult for carers the person with dementia to grieve together, therefore the tasks of normal grieving (Worden, 1991) may be incomplete (Sachs et al., 2004). If anticipatory grief is processed openly adaptively, grief after death may be less intense distressing for carers; however, the evidence for this is inconclusive (Ro, 2000). As the worldwide population of people with dementia grows (Ferri et al., 2005), supporting dementia carers becomes more important. For example, in Engl, providing better services for carers is a target of the National Dementia Strategy (Department of Health, 2009). However, little is known about how to identify support dementia carers who grieve before after death. support intervention, we systematically reviewed the literature to answer the following questions: In carers of people with dementia, (i) what are the prevalence, characteristics, predictors associations of grief reactions? (ii) What is the relationship between anticipatory grief reactions post-death grief? Methods Search strategy We searched electronic databases Medline (1950 ), Embase (1980 ) Cinahl Plus (1987 ) up to July 2010, combining the MeSH terms keywords grief bereavement with dementia. This search was repeated with these terms as free text. We also h searched references of included papers contacted experts in the field (Figure 1). Objective In order to facilitate early identification of carers before after death who may require further Inclusion exclusion criteria We included qualitative quantitative primary research on the following: Figure 1 Results of literature search.

3 Systematic review of grief reactions in carers of people with dementia Grief family carers of older people with dementia in community settings including people romised to the control arm of interventional trials. We excluded studies that were as follows: Non-English language, reviews opinion articles. Non-grief or dementia carer specific. Validating grief-rating scales. Using depression scales not validated in bereaved people; participants may score above the threshold without necessarily being depressed. Exploring young onset or HIV dementia; dementia in these populations has a different disease trajectory to that of dementia in older people. Data extraction We extracted data on the characteristics, associations predictors of grief before after death. Prevalence rates were obtained from studies where stated. Associations predictive factors were identified from quantitative studies that used regression analysis. We extracted key themes identified by authors in each qualitative study combined these where possible to summarise qualitative results. Quality assessment (Table 1 for Quality Assessment Tools) We assessed the quality of the studies using modified stardised assessment tools: National Institute for Health Clinical Excellence (NICE) guidance for qualitative studies (NICE, 2009) Boyle s criteria for quantitative studies (Boyle, 1998). Mixed method studies were rated for both qualitative quantitative methodology. Each study was scored out of eight points independently rated by two authors (D. C., E. S. or G. L.). In the event of discrepancies, another independent rater (L. J.) was involved. Table 1 Quality assessment tools of qualitative quantitative studies Qualitative (NICE, 2009) Quantitative (Boyle, 1998) 1. Did the study have clear aims use the qualitative approach appropriately? Is aim stated should it be explored qualitatively? 2. Did the study use describe rigorous sampling methodology? Did the method of sampling cover all appropriate groups, was this justified? Is the sampling strategy reproducible? 3. Was the data collection robust, were methods clearly described? Was there a topic guide for interviews or focus groups? 4. Has the role of the researcher been clearly described? Was data collected face to face conversation facilitated? 5. Were characteristics of the participants the setting clearly described? 6. Did the study use more than one method to collect the data? Was more than one perspective used, that is different people interviewing or groups with different characteristics being interviewed? 7. Was the content diversity of perspective of the participants explored in depth (i.e. did the researchers achieve saturation of data)? Did they state that they achieved saturation? 8. Was the data analysis sufficiently reliable (i.e. at least two raters, participant feedback on transcripts, discrepant results resolved)? 1. Was the target population defined clearly by inclusion exclusion criteria? (Must define precisely what is meant by a carer what population they come from. Is it replicable in principle?) 2. Was probability sampling used to identify potential respondents or whole sample approached? (Rom or fully inclusive sample from known pool) 3. Did characteristics of respondents match the target population (i.e. response rate >80% or appropriate analysis including responders non-responders) 4. Were data collection methods stardised? Same methods must be used each time (i.e. replicable in another study; could be a validated measure or a measure devised for the study) 5. Was the grief instrument reliable? Some evidence of reliability testing its use in this population or psychometric properties published evidence of reliability in similar populations. Score one if in similar half if in dissimilar populations 6. Was the grief instrument valid? Some evidence of reliability testing its use in this population or psychometric properties published evidence of reliability in similar populations. Score one if in similar half if in dissimilar populations 7. Were special features of the sampling design accounted for in the analysis, through proper weighting of the data or the whole population approached? If not applicable, award full score (one) 8. Were confidence intervals included in the results?

4 D. Chan et al. Results We identified 31 total studies: 17 quantitative, 11 qualitative three mixed methods; 20 anticipatory grief 11 post-death grief. Characteristics, quality findings of these studies are shown in Tables 2 3; the studies are listed in order of quality, highest first. All studies were conducted from 1992 to 2009; all but four were from the. Three studies were set in care homes or long-term dementia units one in a hospice. Two studies sampled advanced dementia carers, one sampled carers at dementia diagnosis. In reporting results, we gave precedence to higher quality studies. The qualitative quantitative quality scores ranged from 1.5 to 6 (median 3) 1 to 8 (median 3), respectively. The commonest grief measurement (six studies) was the Meuser Marwit Caregiver Grief Inventory (Marwit Meuser, 2002). Four studies used the Grief Experience Inventory (Sers et al., 1985). Complicated grief was measured using the Inventory of Complicated Grief (Prigerson et al., 1995) Prolonged Grief Disorder-12 (Prigerson et al., 2008). Studies describing the characteristics of anticipatory grief Grief losses A high quality study examined ambiguous loss, when a loved one is physically present but psychologically absent (Boss, 2004). Three sequential phases of loss were identified: anticipatory (ambiguity about the future), progressive (helplessness dealing with gradual loss) acknowledged (avoidance acceptance as coping strategies) (Dupuis, 2002). Other high quality studies also described anticipatory grief using themes of ambiguity (Sers Corley, 2003; Adams Sers, 2004) anticipation (Meuser Marwit, 2001; Moyle et al., 2002). As dementia progressed, the most frequently reported losses were of intimacy/companionship (Meuser Marwit, 2001; Moyle et al., 2002; Sers Corley, 2003; Adams Sers, 2004; Sers et al., 2008), personal freedom (Loos Bowd, 1997; Meuser Marwit, 2001), control (Loos Bowd, 1997; Sers et al., 2002; Sers Corley, 2003), wellbeing (Loos Bowd, 1997) loss of personhood for the care recipient (CR) (Moyle et al., 2002). Carers also experienced sadness (Dupuis, 2002; Moyle et al., 2002; Adams Sers, 2004), desperateness (Dupuis, 2002; Sers et al., 2002; Adams Sers, 2004), anger (Meuser Marwit, 2001; Sers Corley, 2003), frustration (Loos Bowd, 1997; Marwit Meuser, 2002; Adams Sers, 2004) guilt (Loos Bowd, 1997; Meuser Marwit, 2001; Sers et al., 2008). Relationship between carer care recipient Meuser s high quality study found different grieving patterns in adult children compared with spouse carers. Adult children carers of people with mild dementia were in denial; spouses accepted the illness, engaging in open discussion about the future. At moderate dementia, adult children felt anger, frustration, sadness, but guilt, wishing the CR would die; spouses felt deep sadness empathy focussed on maintaining their relationship. In severe dementia, adult children accepted institutionalisation with sadness relief; spouses felt relieved from physical burden of care (Meuser Marwit, 2001). Two lower quality quantitative analyses found no significant difference with dementia severity grief between spouse adult children carers (Lindgren et al., 1999; Adams Sers, 2004). In a lower quality sub-study, some male carers described feelings of self-awareness insight; others described isolation, desperation, thoughts of harm to self the CR. Some men did not associate losses with grief (Sers et al., 2002). A comparison of sons with daughters found that fewer sons acknowledged themselves as grieving before death (Diwan et al., 2009). Summary. Carers experience grief as serial multiple losses for themselves (companionship, personal freedom, control) the CR. Anticipation ambiguity about the future, anger, frustration guilt are core features. Adult children spouse carers may experience grief in different ways, both have changing emotions as dementia progresses. Some male carers may be less willing to acknowledge grief experience thoughts of harm to self the CR. Stages of dementia High quality studies showed significantly more grief symptoms in severe stages of dementia compared with middle early stages (Meuser Marwit, 2001; Adams Sers, 2004; Ott et al., 2007). An intermediate quality study found that moderate to severe

5 Systematic review of grief reactions in carers of people with dementia Table 2 Qualitative mixed methods studies describing the characteristics prevalence of anticipatory post-death grief Paper country Type of study Sample setting dementia Main results/themes Anticipatory grief Dupuis (2002), Active interviews 61 adult children carers in long-term care facility; purposive sample Meuser Marwit (2001), Aminzadeh et al. (2007), Canada Sers et al. (2008), Sers Corley (2003), Sers et al. (2002), Moyle et al. (2002), Australia Focus groups 16 (eight spouses eight adult children) in mild, moderate, severe dementia group (M) Focus groups, interviews, field observations Semi-structured interviews (M) Cross-sectional survey open question asking whether carers were grieving whilst PWD was still alive, response rate not stated Cross-sectional survey open question asking whether carers were grieving whilst PWD was still alive, response rate not stated Focus groups 11 4 in each group 87 carers (42 spouses, 45 adult children) recruited through chapter of AA, memory ageing project 33 dyads for interview, sub-group 12 carers for focus group, people referred for assessment of cognitive function to day hospital 201 self-selected carers recruited through support groups via AA, memory loss clinics 253 carers from chapter of AA sent postal survey 71 male carers who returned survey recruited from chapter of AA sent postal survey 15 next of kin of residents on long-term Not stated Anticipatory loss; progressive loss; acknowledged loss 20 mild Adult children: mild denial, losses self focussed; moderate 22 moderate 21 severe, using CDR Early diagnosis Moderate to severe, using FAST guilt, grief focussed on self; severe placement, relief, sadness. Spouses: mild acceptance, sadness; moderate sadness, grief focussed on other; severe uncertainty about future, relief burden Stages of emotional reaction to diagnosis (denial, grief reaction, positive coping responses); experience of loss stigma; uncertainty insecurity High grief yearning for the past (n = 31); regret guilt (n = 26); isolation (n = 20); restricted freedom (n = 19); life stresses conflict (n = 19); systemic illness (n = 11); coping strategies (n = 15) Not stated 68% grief No grief themes: carer relief, religiosity, spirituality, past relationship conflict. Grief reaction themes: ambiguity; loss of intimacy; feelings of desperateness loss of control; self-awareness of grief 11 mild 68 % grief moderate 30 severe Grief reaction themes grief turned into crisis (68%); selfawareness insight into losses grief journey (67%). no grief themes: emotional dissociation from care recipient (57%); life could be worse Not stated Relief versus burden of loss; loss from observation of the cognitive decline; loss of companionship; (Continues)

6 D. Chan et al. Table 2. (Continued) Paper country Type of study Sample setting dementia unit, recruited through open invitation Diwan et al. (2009), Structured telephone interview after death, response rate not stated 135 bereaved carers from primary care geriatric clinics Adams Sers (2004), Cross-sectional survey open questions, response rate not stated (M) 99 carers romly selected from community database of AA Loos Bowd (1997), Canada Cross-sectional survey open question asking what carers had given up during caring role 68 carers recruited through local, regional social service health professionals Almberg et al. (2000), Sweden Serial interviews with open-ended questions, part of a larger longitudinal study Post-death grief 30 carers whose elderly relative died in Swedish community Collins et al. (1993), Cross-sectional survey three open questions about predeath post-death 82 bereaved primary carers recruited through mailing list by local chapters of ADRDA dementia Main results/themes loss creating fears frustration; loss of personhood; anticipation of death Not stated 71% grief other than when death Initial diagnosis (n = 7); decline in cognition, loss of person (n = 13); decline in function (n = 1); declining health (n = 5); nearing death (n = 13); behavioural symptoms (n = 2); interpersonal issues or conflict (n = 3); nursing home placement (n = 2); continuously (n = 10) 25% early 59% grieving % middle No grief themes: early missing 34% late activities, clear communication 81% Alzheimer s disease (52%). Middle personal changes within caregivers (time, energy) (40%). Late interpersonal relationship (36%) Loss of social recreational interaction; loss of control over life events; loss of well-being; loss of occupation Diagnosed using DSM- 3 Pre-death grief positive memories (n = 21); burden before death (n = 12); saying goodbye (n = 11); mixed feelings after death (loneliness, relief, guilty); importance of social support: negative support; social isolation; formal support. Link between prepost-death experience social support: availability of support; positive memories; great burden; possibility of saying goodbye Not stated Grief before death (47%) Loss of familiarity intimacy; loss of hope (46%); expectancy of death (28%); post-death relief (42%); post-death reflections (Continues)

7 Systematic review of grief reactions in carers of people with dementia Table 2. (Continued) dementia Main results/themes Paper country Type of study Sample setting experiences, response rate not stated Not stated Intense grief during caring; readiness to let go; the death was a relief; ready to get on with life; bereavement conceptualisations; process of detachment; complex relief (sorrowful, guilty, grateful) 30 carers purposive sampling recruited through AD clinics carer support groups Serial interviews over 3.5 years, before after death Jones Martinson (1992), Not stated Carer role versus personal life Patient-related challenges caused by AD Personal grief loss 353 rom sample of carers, then convenience sample for Alzheimer s carers at AA (4.5% carers where PWD had died) Frank (2007), Cross-sectional survey open questions about barriers to caring M, mixed methods study; AA, Alzheimer s Association; AD, Alzheimer s disease; DSM, Diagnostic Statistical Manual of Mental Disorders; PWD, person with dementia; ADRDA, Alzheimer s Disease Related Disorders Association; CDR, Clinical Dementia Rating Scale; FAST, Functional Assessment Staging. 1 Prevalence rate of anticipatory grief. dementia carers were more functionally impaired experienced high (severe) grief reactions, identifying carer burden stress, coping strategies life stressors as qualitative themes (Sers et al., 2008). Other studies highlighted how grief might occur at diagnosis (Lindgren et al., 1999; Aminzadeh et al., 2007; Diwan et al., 2009). Institutionalisation Two qualitative studies conducted in long-term dementia units found that grief persisted created new challenges; adult children carers faced ambivalence around visiting (Dupuis, 2002), carers experienced loss of role a necessity to build new relationships to avoid staff conflict (Moyle et al., 2002). Other studies described deeper feelings of guilt failure related to institutionalisation (Loos Bowd, 1997; Diwan et al., 2009). This was worse for spouse carers (Rudd et al., 1999; Meuser Marwit, 2001; Ott et al., 2007). Summary. Anticipatory grief in dementia carers may occur at any stage from diagnosis to severe dementia. Severe grief reactions mostly occur in the moderate severe stages. Carers who suffer with severe grief are likely to have functional impairment feel stressed burdened. Placing a person with dementia in a care home is likely to precipitate or exacerbate feelings of guilt failure in spouse carers. Studies reporting the prevalence of anticipatory grief Two intermediate quality studies reported the prevalence of anticipatory grief as 47% (Collins et al., 1993) 71% (Diwan et al., 2009). Dichotomous survey responses found that 68% of responders (Sers Corley, 2003) 68% of this male subset (Sers et al., 2002) reported themselves as grieving, even though the CR was still alive. The same question asked to another sample reported 59% as grieving (Adams Sers, 2004). One study found that 10% of carers were at risk of serious grief reactions needing further support (Sers Adams, 2005). Another compared the prevalence of grief in dementia carers with that in carers of people with cardiac disease. Grief occurred almost twice as commonly compared with cardiac carers, but we do not know about the severity of illnesses (Ross Dagley, 2009).

8 D. Chan et al. Table 3 Quantitative mixed methods studies describing the characteristics, prevalence, predictors associations of anticipatory post-death grief Paper country Type of study Sample setting dementia Measure of grief Main results Contribution to synthesis (C, P, A, Pr) Factors controlled for in analysis Kiely et al. (2008), Ott et al. (2007), Sers Adams survey, part of larger prospective cohort study survey survey, 41% 315 HCP of NH residents with advanced dementia. Convenience sample of NH 201 carers recruited from support groups, memory loss clinics. 90 spouses, 11 adult children. 96 home carers, 105 non-home carers 122 carers recruited from chapter of AA Advanced dementia GDS 7 AD 27% mild to moderate, 47% moderate, 26% severe Prolonged grief disorder 12 MM-CGI- SF Anticipatory grief HCP variables associated with grief: non-english primary language PE = 6.66 (10.53, 2.79)**; living with resident before institutionalisation PE = 1.48 (0.36, 2.60) **; depressive symptoms PE = 0.7 (0.53, 0.87)**; satisfaction with care PE = 0.13 ( 0.22, 0.08)**; younger resident age PE = 0.15 ( 0.22, 0.88) Spouse non-home carers experienced more grief F = 7*; more grief in moderate, severe disease than mild F = 7.66** Factors associated with increased grief for non-home carers: planning Β = 0.184*; self-blame Β = 0.287**; depression Β = 0.382** Factors associated with increased grief for home carers: venting Β = 0.204*; positive states of mind Β = 0.345**; depression Β = 0.251* Not stated MM-CGI 10% at risk of serious grief TSNS A Carer: gender, ethnicity, education, employment, hours visiting, physical health, directives with MD; PWD: age, gender, ethnicity, length of stay, health status C Carer: kinship, hours of care, support, religion, acceptance A PWD: severity of illness P No (Continues)

9 Systematic review of grief reactions in carers of people with dementia Table 3. (Continued) Paper country Type of study (2005), response rate Walker Pomeroy (1996), survey Sers et al. (2008), survey (M) Adams Sers (2004), survey (M) Meuser Marwit (2001), Postal survey (M) Ross Dagley (2009), survey of cases controls, 30% response rate Holley Mast survey Sample setting 100 carers, using volunteers from chapters of ADRDA 201 selfselected carers, recruited through support groups via AA, memory loss clinics 99 carers romly selected from community database of AA. 35 spouses, 49 adult children 98 selfvolunteers recruited through chapter of AA, memory ageing project 176 carers 138 dementia 38 cardiac volunteers 80 carers recruited from adverts, support dementia Measure of grief Main results Symptom severity mean score 66.65, using MBPC 48% of variance is grief in depression scores R 2 = 0.48** GEI 63% variance is grief in depression scores, TSNS Moderate severe FAST MM-CGI- SF Carers experiencing high grief group more functionally impaired compared with moderate/low grief t = 2.62** 25% early MM-CGI Carers of late stage 42% middle disease scored higher 34% late on grief than early or middle stages TSNS** Mild, moderate severe, CDR rated by carers AGS Negative correlation MFG all subjects between MFG score years since diagnosis r = 0.45** Not stated MM-CGI- SF Mild, moderate, Carers of people with dementia experience almost two times more grief than carers of people with cardiac disease, difference in mean score F = 102.7* MM-CGI Grief positively correlated with carer burden AGS Contribution to synthesis (C, P, A, Pr) Factors controlled for in analysis A Carer: gender, kinship, support group, knowledge AD, duration caring. PWD: symptom severity C N/A C N/A A No P No A No (Continues)

10 D. Chan et al. Table 3. (Continued) Paper country Type of study (2009), Walker Pomeroy (1997), survey Rudd et al. (1999), survey Lindgren et al (1999), survey Ponder Pomeroy (1996), survey Sample setting groups, nursing homes, community physicians 100 carers recruited from chapters of ADRDA 60 spouse carers (30 wives, 30 husbs; equal numbers of home NH carers) 33 carers (22 spouse, 11 children carers) contacted through home health care agencies, support groups 100 carers recruited from chapters of ADRDA dementia severe, using FAST Symptom severity, using MBPC CDR rated by carers Not stated Not stated Measure of grief Main results Despair scale of non-death GEI, SGI Hostility in Scale (Sadness), Hostility Out Scale (Anger), Guilt Anxiety Scale (Guilt) using two burden scales: MM-CGI r = 0.71**; r = 0.73**; AGS 0.68**; r = 0.72** Intensity of grief (anger guilt) associated with: subjective distress r = 0.56**; health problems r = 0.5**; expectation to institutionalise r = 0.31**; life satisfaction r = 0.35** Female home carers experienced more grief than male home carers TSNS; nursing home carers experienced higher levels of guilt than home carers TSNS GEI Length of time caring inversely related to guilt r = 0.44**; less grief related to higher affection before illness r = 0.39*; satisfaction with marriage r = 0.45* Despair scale of non-death GEI, SGI, PEAF Carers in earlier later periods of caring show greatest amount of despair TSNS Symptoms of dementia intensity of grief correlated r = 0.35** Contribution to synthesis (C, P, A, Pr) Factors controlled for in analysis A Carer: gender, kinship, support group, knowledge AD, duration caring PWD: symptom severity C N/A A No C No A (Continues)

11 Systematic review of grief reactions in carers of people with dementia Table 3. (Continued) Paper country Type of study Holl et al. (2009), Prospective multi-site study 1 Hebert et al. (2007), Prospective multi-site study 1 Hebert et al. (2006), Prospective multi-site study 1 Schulz et al. (2006), Prospective multi-site study 1 Sample setting dementia 224 bereaved Not carers 2 stated 1229 carers 2 AD average MMSE bereaved AD carers 2 average MMSE bereaved AD carers 2 average MMSE 10.9 Measure of grief Main results Contribution to synthesis (C, P, A, Pr) Factors controlled for in analysis Post-death grief ICG Predictors of CG, carer factors: depression PE = 0.35**; African American, PE = 4.82*; spouse PE = 6.99**; younger age PE = 0.22**; less education PE = 0.64*; fewer days since death PE = 0.01* TRIG Predictors of uncg, carer factors: depression PE = 0.07**; spouse PE = 3.66**; less education PE = 0.84*; fewer days since death PE = 0.01** ICG Frequent religious attendance associated with CG Β = 1.41 ( 2.30 to 0.533)** ICG Carers not prepared for death had more CG OR 2.88 (1.28, 3.20)* Pr Carer: gender, ethnicity (Hispanic), post REACH counselling A Carer: social integration, depression, physical health, negative interactions P/A Carer: prebereavement depression ICG 20% CG P N/A Carer characteristics C N/A of CG: less education z = 2.29*; lower income z = 2.14*; preloss depression z = 5.37**; post-loss depression z = 7.16**; pre-loss anxiety z = 5.59**; post-loss anxiety z = 6.15**; anxiolytic use preloss Χ 2 = 5.99**; antidepressant use (Continues)

12 D. Chan et al. Table 3. (Continued) Paper country Type of study Boerner et al. (2004), Prospective multi-site study 1 Burton et al. (2008), Longitudinal study, part of larger study Owen et al. (2001), Structured interview post death, part of Sample setting 217 bereaved carers 2 50 spouses of hospice patients (21 dementia, 29 end-stage lung cancer) 63 bereaved carers recruited from memory disorders clinic. dementia Measure of grief Main results post-loss Χ 2 = 4.56; Burden z = 3.27**; + aspects of caring z = 2.18*. younger age of care recipient z = 2.52** Pre-loss predictors of CG: depression OR 1.13 (1.04, 1.23)**; burden OR 1.06 (1.01, 1.11)*; + aspects of caring OR 1.22 (1.11, 1.34)**; MMSE OR 0.89 (0.81, 0.98)** Post-loss predictors of CG: depression OR 1.16 (1.08, 1.25)**; time since death OR 1.08 (1.03, 1.13)** AD average MMSE 13 TRIG Associations with uncg: caring benefit r = 0.39**; pre-loss depression r = 0.37*; carer health r = 0.19** Predictors of uncg, carer factors: spouse b = 0.31**; pre-loss depression b = 0.44**; ADL burden b = 0.15*; caring benefit b = 0.47** Advanced TRIG Grief associated with fewer months caring r = 0.35* 3 ; less carer ADL/IADL impairment r = 0.31* 3 AD or progressive dementia TRIG African American carers: less likely to grieve before death t = 1.83**; experience Contribution to synthesis (C, P, A, Pr) Factors controlled for in analysis Pr Carer: age, sex, education, income, race, kinship PWD: age, sex, education Pre-loss: antidepressant use, anxiolytic use, ADL/IADL impairment, MMSE Post-loss: antidepressant use, anxiety, anxiolytic use A No Pr Carer: gender, age, health, (R) MBPC burden, antidepressant use post loss, time since death A No C No (Continues)

13 Systematic review of grief reactions in carers of people with dementia Table 3. (Continued) Factors controlled for in analysis Contribution to synthesis (C, P, A, Pr) Main results Measure of grief dementia Sample setting Type of study Paper country death as a relief t = 2.52**; accept death t = Death perceived as greater loss t = 2.02** 47 white, 16 African American longitudinal study M, mixed methods study; C, characteristics; P, prevalence; Pr, predictors; A, associations; PE, parameter estimate; TSNS, test statistic not stated; HCP, healthcare proxy; NH, nursing home; GDS, Global Deterioration Scale; MD, Medical Doctor; AD, Alzheimer s disease; AA, Alzheimer s association; ADRDA, Alzheimer s Disease Related Disorders Association; PWD, person with dementia; CDR, Clinical Dementia Rating Scale; FAST, Functional Assessment Staging; ADL, activities of daily living; IADL, instrumental activities of daily living; MMSE, Mini Mental State Examination; (R)MBPC, (Revised) Memory Behaviour Problems Checklist; MM-CGI-(SF), Meuser Marwit Caregiver Grief Inventory (Short Form); GEI, Grief Experience Inventory; AGS, Anticipatory Grief Scale; MFG, Many Faces of Grief; SGI, Stages of Grief Inventory; PEAF, Parental Experience Assessment Form; CG, complicated grief; uncg, uncomplicated grief; ICG, Inventory of Complicated Grief; TRIG, Texas Revised Inventory of Grief; OR, odds ratio. 1 Part of Resources for Enhancing Alzheimer s Caregiver Health (REACH) project, a romised interventional study. 2 Participants recruited from six sites through media, memory clinics, primary care clinics social services. Carers followed for 18 months assessed at baseline, 6, months. 3 Results of non-dementia carers combined with dementia carers. Results of dementia carers only not stated. *p 0.05 (95% CI). **p 0.01 (95% CI). Summary. There were no high quality prevalence studies of anticipatory grief. Survey responses from five intermediate/low quality studies found a prevalence of anticipatory grief between 47% 71%. Around 10% of carers may be at risk of serious grief reactions requiring support. Studies describing the predictors associations of anticipatory grief Carer emotional mental health Several high quality studies found that carer depression increased with anticipatory grief (Sers Adams, 2005; Ott et al., 2007; Kiely et al., 2008). An excellent quality study found that grief in advanced dementia carers accounted for 32% of the common variance; depressive symptoms accounted for 12% (Kiely et al., 2008). Grief accounted for 48% (Sers Adams, 2005) 63% (Walker Pomeroy, 1996) of the variance in two studies using different depression scales, suggesting that what appears to be clinical depression may actually be a grief reaction; the full Beck Depression Inventory also includes items that are features of bereavement (Walker Pomeroy, 1996). Ott found several correlations between grief coping strategies in carers looking after the CR within or outside the carer s home. When caring for the CR at home, as carers positive state of mind increased, grief decreased; as ventilation of feelings increased, grief increased. Within a care home, as carer planning self-blame increased, grief also increased (Ott et al., 2007). Other studies showed that increasing burden decreased emotional heath were associated with increasing grief (Lindgren et al., 1999; Holley Mast, 2009), higher affection pre-morbid marriage satisfaction decreased grief (Lindgren et al., 1999). Carer distress, health problems expectation to institutionalise were all positively correlated with intensity of grief, whilst life satisfaction was negatively correlated (Walker Pomeroy, 1997). Other carer care recipient factors In an excellent quality study of people with advanced dementia living in a US care home, family carers whose primary language was not English carers who were less satisfied with care had lived with the CR before institutionalisation experienced greater grief symptoms

14 D. Chan et al. (Kiely et al., 2008). This another high quality study found no significant relationship between grief hours of care (Ott et al., 2007; Kiely et al., 2008). Ott also found no significant relationship between carer social support grief (Ott et al., 2007). Summary. The following carer factors may be positively associated with increasing anticipatory grief: (i) depression poor emotional health, (ii) burden, (iii) non-english primary language in an English-speaking country, (iv) cohabitation with the CR prior to institutionalisation (v) less satisfaction with care. One quantitative study found no association between social support anticipatory grief. Studies describing the characteristics of postdeath grief Post-death grief themes were identified from three intermediate quality qualitative studies: losses (Collins et al., 1993; Almberg et al., 2000; Frank, 2007), carer burden (Almberg et al., 2000; Frank, 2007), regret guilt related to care home placement (Collins et al., 1993). Carers who expected death (said goodbye were present at time of death) coped better with grieving. Carers also needed to psychologically detach themselves from the CR before moving on after death (Collins et al., 1993; Almberg et al., 2000). Death signalled relief for many carers, also ending suffering for the CR (Collins et al., 1993; Almberg et al., 2000). Carers felt more relieved when they had looked after the CR at home until death (Jones Martinson, 1992), acknowledged losses before death but only in the context of high level support from family friends (Collins et al., 1993), reflected on positive experiences experienced less burden during the caring period (Almberg et al., 2000). Three types of complex relief were described: (i) sorrowful (grief not resolved quickly), (ii) guilty (institutionalisation) (iii) grateful (grief resolved quickly) (Jones Martinson, 1992). African Americans were less likely to grieve before death, accept death or experience it as relief. They also perceived death as a greater loss compared with white carers (Owen et al., 2001). Carers highlighted the importance of social, religious, practical emotional support from family friends (Collins et al., 1993; Almberg et al., 2000). Carers who had support before after death from the same person were more likely feel relieved after death (Almberg et al., 2000). Summary. Carers continue to reflect upon predeath experiences when grieving after death. Expectation preparation for death may be important to process grief. Carer relief after death is common related to positive experiences, less burden acknowledged grief in the caring period, as well as consistent support before after death. Studies reporting the prevalence of post-death grief A high quality study of carers (84% male, 66% white, equal numbers of spouse non-spouses) measured the prevalence of complicated grief, interviewing them once or twice (6 12 months) after death. Grief increased during the first 15 weeks after death decreased in subsequent months. Nevertheless, at approximately 12 months after death, 20% of carers were still grieving had complicated grief (Schulz et al., 2006). Summary. The prevalence of complicated grief in carers 12 months after death is 20%, but this has not been replicated. Studies describing the predictors associations of post-death grief Several associations predictors of complicated uncomplicated grief were identified using secondary analysis of a large high quality longitudinal interventional study. Strongest predictors of complicated grief were being a spouse carer, African American ethnicity higher levels of pre-death depression (Schulz et al., 2006; Holl et al., 2009). Other carer predictive factors include younger age, less education (Holl et al., 2009), carer burden, increased positive aspects of caring higher levels of post-death depression (Schulz et al., 2006). CR cognitive impairment also predicted complicated grief (Schulz et al., 2006). Complicated grief in carers also correlated positively with less frequent religious attendance before death (Hebert et al., 2007) carers who were unprepared for death (Hebert et al., 2006). Studies looking at uncomplicated (normal) postdeath grief found that the strongest predictive factors

15 Systematic review of grief reactions in carers of people with dementia were pre-death depression, spouse carer (Boerner et al., 2004; Holl et al., 2009), caring benefit (Boerner et al., 2004) less education (Holl et al., 2009). Fewer days since the death (Holl et al., 2009) less carer burden were weak predictors of uncomplicated grief. There were positive correlations between increasing grief pre-death depression caring benefit ; the latter accounted for 20%ofthevarianceofgrief(Boerneret al., 2004). A lower quality hospice study examined post-death grief in spouse carers of advanced dementia lung cancer CRs. No significant associations were found between measures of well-being, positive aspects of caring, social measures or demographics (age, race, gender, education, ethnicity); however, there were no comparative results for the two illness groups (Burton et al., 2008). Summary. The strongest predictors of complicated uncomplicated grief are being a spouse carer depression before death. African American ethnicity carer burden may also predict complicated grief. Preparation for death frequent religious attendance before death is associated with less complicated grief. Caring benefit less education may predict uncomplicated grief. Discussion Grief reactions in dementia carers can be described as reactions to serial losses occurring before death (anticipatory grief) persisting when the CR dies. The prevalence of anticipatory grief ranged between 47% 71%, although these studies were of poor quality. Carers commonly experience the CR s death as a relief, although only one study considered the prevalence of complicated grief after death found it in 20% of carers. The most consistent predictive factors of complicated (severe) uncomplicated (normal) grief after death are being a spouse carer experiencing depression. Depression is also strongly associated with increasing anticipatory grief. We found two studies suggesting that grieving in anopenadaptivemannerbeforedeathmaybebeneficial for carers after death. Our finding that spouse carers carer burden may predict complicated grief supports the resource depletion theory in the caring bereavement literature; that is fewer resources resulting from prolonged exposure to caring stressors consequently increase the surviving spouses vulnerability to negative outcomes during widowhood (Schulz et al., 1997). Prigerson suggests that carers who find positive aspects of caring tend to do worse after the person with dementia dies, which may reflect excess dependency or unstable attachment between carer CR (Prigerson et al., 1997b). Although the wider literature in this area is limited, evidence suggests that caring for a person with dementia may well be different from caring for persons with other long-term illnesses (Sachs et al., 2004). We identified one study where dementia carers experienced twice as much anticipatory grief as cardiac disease carers (Ross Dagley, 2009). Reported elsewhere, there is over 50% increased need for carer bereavement services after the death of people with Alzheimer s, lung cancer renal failure compared with other cancers chronic airways disease (Jones, 2010). Loss of carer recognition communication by the CR occurring well before the terminal phase later development of behavioural problems are factors unique to dementia that may impact on grief (Sachs et al., 2004; Jones, 2010). Limitations We excluded non-english studies may have missed useful evidence. We found it difficult to score studies where the methodology was not clearly stated although implied. However, two independent raters agreed on the scores. We found several limitations of the literature. Studies used small samples were mostly from the, so the results may not be generalisable; several used grief measures with poor psychometric properties (reflected by low median quality scores) cross-sectional design (including prevalence studies) with non-probability sampling; we found few longitudinal studies before after death. Grief reactions may not be directly comparable where grief is not rigorously defined, as the results may be associated with other features of caring such as depression anxiety. However, this is the first review using systematic methodology to synthesise this complex area, it highlights how we may move this subject area forward. Clinical implications Our review highlights factors that could be used to identify dementia carers who may need support during the grieving period to prevent the development of subsequent complicated grief related comorbidity. Early identification of modifiable risk factors

16 D. Chan et al. (i.e. decision to institutionalise, carer depression carer burden), grieving in an open adaptive manner before death support provided by the same person before after death may be helpful. Implications for future research Future studies should use more rigorous methodology design. In quantitative studies, grief should be measured using validated scales for dementia carers (e.g. Meuser Marwit Caregiver Inventory) (Marwit Meuser, 2002), thus clearly differentiating this from depression, anxiety the emotional distress of caring (Walker Pomeroy, 1996; Sers Adams, 2005; Kiely et al., 2008). Longitudinal studies exploring the relationship between anticipatory post-death grief, sampling from carer populations outside the would also further knowledge in this field. Conclusions We have identified several factors that may increase grief reactions in dementia carers. For anticipatory grief, the best evidence is for carer depression, moderate to severe dementia institutionalisation. For complicated uncomplicated grief after death, the strongest predictive factors are spouse carers carer depression. Grief in dementia carers can be viewed as a normal reaction; however, a substantial proportion may require further support intervention. Conflict of interest No funding received, no disclosures to report. Key points Grief in dementia carers is a complex reaction to losses occurring before (anticipatory grief) after death of the person with dementia. Anticipatory grief is greatest in moderate to severe stage dementia spouse carers, especially when the person with dementia is institutionalised. Carer depression increases with anticipatory grief. Being a spouse carer being depressed are the strongest predictors of complicated normal grief after death. 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