C International Psychogeriatric Association 2014 International Psychogeriatrics: page 1 of 16 doi:10.1017/S1041610214000969
Preferences regarding disclosure of a diagnosis of dementia: a systematic review ...........................................................................................................................................................................................................................................................................................................................................................................................................................................................................................................................................................................................................................................
Pim van den Dungen,1 Lisa van Kuijk,1 Harm van Marwijk,1 Johannes van der Wouden,1 Eric Moll van Charante,2 Henriette van der Horst1 and Hein van Hout1 1
Department of General Practice and Elderly Care Medicine, EMGO Institute for Health and Care Research, VU University Medical Center Amsterdam, Amsterdam, the Netherlands 2 Department of General Practice, Academic Medical Center, University of Amsterdam, Amsterdam, the Netherlands
Abstract
Background: Studies in memory clinics suggest that the majority of patients would like to know of a diagnosis of dementia. It is less clear what preferences are in the community. Our objective was to review the literature on preferences regarding disclosure of a diagnosis of dementia and to assess key arguments in favor of and against disclosure. Methods: Systematic search of empirical studies was performed in Pubmed, Embase, and Psycinfo. We extracted preferences of individuals without cognitive impairment (general population; relatives of dementia patients; and physicians) and preferences of individuals referred to a memory clinic or already diagnosed with dementia. A meta-analysis was done using a random effects model. Our main conclusions are based on studies with a response rate ࣙ75%. Results: We included 23 articles (9.065 respondents). In studies with individuals without cognitive impairment, the pooled percentage in favor of disclosure was 90.7% (95%CI: 83.8%–97.5%). In studies with patients who were referred to a memory clinic or already diagnosed with dementia, the pooled percentage that considered disclosure favorable was 84.8% (95%CI: 75.6%–94.0%). The central arguments in favor of disclosure pertained to autonomy and the possibility to plan one’s future. Arguments against disclosure were fear of getting upset and that knowing has no use. Conclusions: The vast majority of individuals without and with cognitive impairment prefers to be informed about a diagnosis of dementia for reasons pertaining to autonomy. Key words attitude of health personnel, dementia, diagnosis, disclosure, early diagnosis, informed consent, patient preference, primary health care
Introduction National and international dementia guidelines recommend straightforward diagnosis of dementia and provision of information about, among other things, the course, prognosis, treatment options, services, and legal and financial issues (Patterson et al., 1999; National Collaborating Centre for Mental Health (UK), 2007; Albert Weale et al., 2009; Segal-Gidan et al., 2011; Galvin and Sadowsky, 2012; Sorbi et al., 2012). This should be done in a manner consistent with the patient’s
Correspondence should be addressed to: Pim van den Dungen, Department of General Practice and Elderly Care Medicine, EMGO Institute for Health and Care Research, VU University Medical Center Amsterdam, Van der Boechorststraat 7, 1081 BT Amsterdam, the Netherlands. Phone: +31-204448199, Fax +31-20-4448361. Email:
[email protected]. Received 6 Feb 2014; revision requested 21 Mar 2014; revised version received 30 Apr 2014; accepted 3 May 2014.
preferences, culture, educational level, and abilities. Still, dementia is regularly not diagnosed and when it is, physicians may not disclose. A 2012 review showed that 14%–33% of mild dementia cases and 28%–61% of moderate to severe dementia cases were diagnosed in the primary care setting (van den Dungen et al., 2012). Whether disclosure occurs seems to depend much on the individual physician encountered. Of family physicians, 39%– 70% reported to disclose regularly (Bamford et al., 2004; Carpenter and Dave, 2004; van Hout et al., 2006). For old-age specialists, this was 38%–96% (Bamford et al., 2004; Carpenter and Dave, 2004; van Hout et al., 2006). Many physicians fear to harm the relationship with their patients, or to upset them unnecessarily, by disclosing a diagnosis of dementia (Bradford et al., 2009). Underlying this may be the awareness of a progressive neurodegenerative disorder for
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which a curative treatment is not available (Bradford et al., 2009; Hort et al., 2010; Sorbi et al., 2012). However, deciding for, or “protecting” the patient, may not be in the patient’s best interest. When asked, individuals with dementia experience and express the same emotions as persons without this condition and value being involved in planning their own future (Kutzleben, von, et al., 2012). A 2004 review focused on (post hoc) appreciation of disclosure of individuals who were already diagnosed with dementia, which is a very specific group (Bamford et al., 2004). However, from a clinical perspective, it is more valuable to know whether individuals not yet diagnosed with dementia would want to be told the diagnosis. Our objective was to review the preferences of individuals, without cognitive impairment, regarding disclosure of a hypothetical future diagnosis of dementia. In addition, we assessed rates in favor of disclosure of individuals referred to a memory clinic or already diagnosed with dementia. To provide context, reasons for and against disclosure were assessed. Based on the results, we make recommendations for practice.
Methods Search strategy A literature search was performed in Pubmed (Medline), Embase, and Psycinfo from inception up to May 2012. The search strategy was developed after exploring keywords in relevant articles and reviews. The final search combined variations on the keywords “dementia,” “diagnosis,” and “disclosure.” The full search strategy is provided as an appendix (Appendix 1). In addition, reference lists of articles retrieved and of previous reviews on this subject were checked. Inclusion and exclusion criteria We only included original empirical studies on preferences regarding disclosure of a diagnosis of dementia. We imposed no restrictions on geography, setting, or studied populations. Articles in languages other than English or Dutch were excluded. Two authors (LK and PD, HM, HvH, respectively) independently assessed the relevance and quality of retrieved studies. Disagreement was resolved through discussion. As studies ranged from predominantly qualitative to more quantitative and because we only used a very specific part of their results, we used a brief, self-developed instrument to assess the quality. Studies were scored on their internal validity (e.g., clear description of respondent characteristics; clear in- and exclusion
criteria) and external validity (e.g., sampling strategy; response rate). The full instrument is provided as an appendix (Appendix 2). Data extraction From included articles, two authors independently extracted study and respondent characteristics, quantitative data on response rates, and preferences regarding disclosure. Again, disagreement was resolved through discussion. In addition, central arguments in favor of and against disclosure were extracted from articles presenting these. An argument was considered to be a “central argument” if it was encountered frequently across different studies. In addition, we extracted striking or clinically relevant arguments (e.g., “to plan a suicide”). Data synthesis We divided the included studies into four subgroups. Three subgroups without cognitive impairment: general population or individuals not selected on cognitive status; relatives of individuals with dementia; and physicians; and one subgroup with cognitive impairment: patients with dementia or patients referred to a memory clinic. Arguments were ordered based on how frequently they occurred. Statistical analysis Several studies had a low response rate or did not report the response rate. As selection bias may be present in studies with a low response rate, we performed an overall meta-analysis and a meta-analysis in which only studies with a response rate ࣙ75% were included. The results of both are provided. Q’s and I2 ’s were calculated to assess statistical heterogeneity. Because there was statistical and methodological heterogeneity between studies and because preferences may differ between study populations and subgroups, a random effects model was chosen for the metaanalysis (DerSimonian and Laird, 1986). We used a Microsoft Excel spreadsheet developed by Neyeloff et al. for the meta-analysis and plots (Neyeloff et al., 2012). To assess whether rates in favor of disclosure were lower in studies older than 2003 (> 10 years old), we performed a post hoc analyses using a t-test.
Results Included studies Figure 1 provides a flow chart of the selection process. Of the 23 included articles, most provided
Dementia—preferences regarding disclosure
3
Figure 1. Flowchart of selection process. ∗ Boxes at the bottom add up to more than 23 because three articles describe preferences of two subgroups.
semi-quantitative data; two papers concerned qualitative interviews (Byszewski et al., 2007; Pratt and Wilkinson, 2001). Some studies appear more than once in the results (Table 1) because they examined two subgroups of respondents. The average quality score of the included studies was 6.0 (SD 1.9) on a scale ranging from 1 to 10. The majority of (sub-)studies (17/25) used clear
in- and exclusion criteria and (19/25) described respondent subgroups clearly. Most studies (22/25) used a questionnaire or fixed interview schedule, but only 10 studies explicitly described the question about preference regarding disclosure. Most studies (17/25) reported an adequate sampling strategy. Seventeen studies included >100 participants and 13 studies reported a response rate ࣙ75%. The
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results of the quality assessment are provided as an appendix (Appendix 2). Pooled preferences of individuals without cognitive impairment Percentages in favor of disclosure ranged from 57.7% to 98.0% in studies among individuals without cognitive impairment. Pooling these studies (n = 18), we found that 87.3% (95%CI: 81.0%– 93.0%) of individuals without cognitive impairment would want to know if diagnosed with dementia. In the subgroup of studies with a response rate ࣙ75% (n = 7), 90.7% (95%CI: 83.8%–97.5%) would want to know. Pooled preferences of individuals with cognitive impairment Percentages in favor of disclosure ranged from 70.0% to 100% in studies among individuals referred to a memory clinic or diagnosed with dementia. Pooling these studies (n = 8), we found that 84.1% (95%CI: 78.1%–90.2%) of this group considered full disclosure of a diagnosis of dementia preferable. In the subgroup of studies with a response rate ࣙ75% (n = 6), 84.8% (95%CI: 75.6%–94.0%) considered disclosure preferable. Figure 2 provides a forest plot of percentages in favor of disclosure per study; the pooled percentage of studies in individuals without cognitive impairment and the pooled percentage of studies in individuals with dementia or referred to a memory clinic. Table 1 provides an overview of the main characteristics of the included studies. Preferences in subgroups The studies concerning the preferences of individuals without known cognitive impairment (n = 8) showed 76% to 98% in favor of disclosure if they were to develop dementia with a pooled percentage of 90% in favor of disclosure (Erde et al., 1988; Holroyd et al., 1996; Sullivan and O’Conor, 2001; Turnbull et al., 2003; Ouimet et al., 2004; Umegaki et al., 2007; Shimizu et al., 2008; TNS for the Harvard School of Public Health and Alzheimer Europe, 2011; ). Only two articles reported less than 90% in favor of disclosure (Holroyd et al., 1996; Umegaki et al., 2007). In these studies, 4% and 11% of population studied, respectively, was against disclosure, with the latter study being of an earlier date (Holroyd et al., 1996; Umegaki et al., 2007). The two studies on the preference of physicians (UK 2000; Brazil 2008) showed that 72% and 77%, respectively, would favor disclosure if they were to develop this condition (Johnson et al., 2000; Raicher et al., 2008). In these studies, 9% and 13%,
respectively, did not want to know and the rest of physicians were unsure. The studies investigating the preferences of relatives of dementia patients if they were to develop dementia (n = 8) showed that 58% to 98% favored disclosure (Maguire et al., 1996; Heal and Husband, 1998; Mimura, 2003; Pinner and Bouman, 2003; Lin et al., 2005; Shimizu et al., 2008; Mormont et al., 2012; ). The pooled percentage in favor was 85%. In six of the eight studies, percentages in favor of disclosure were higher than 90%. The two studies with lower percentages (71% and 58% in favor) were older (1996 and 1998, respectively) (Maguire et al., 1996; Heal and Husband, 1998). In the study with 58% in favor, 21% did not want to know and 21% was unsure. The studies concerning the preferences of individuals referred to a memory clinic or already diagnosed with dementia (n = 8) showed 69% to 100% in favor of disclosure, with a pooled percentage in favor of disclosure of 84% (Marzanski, 2000; Jha et al., 2001; Pratt and Wilkinson, 2001; Dautzenberg et al., 2003; Pinner and Bouman, 2003; Elson, 2006; Byszewski et al., 2007; Mormont et al., 2012). In the two qualitative studies included in this review, none of the patients believed that the diagnosis should be withheld from the patient, except for two individuals who were unsure.
Not wanting to know or being unsure Not all studies reported on “not wanting to know.” In the studies that explicitly reported on this (n = 12; one study in two subgroups), the pooled percentage of individuals explicitly stating that they would not want to be informed if they would have dementia was 4.8% (95%CI: 3.3%–6.3%). In studies in individuals without cognitive impairment (n = 8), the pooled percentage against disclosure was 4.8% (95%CI: 3.2%–6.4%) as well. In studies in individuals referred to a memory clinic or diagnosed with dementia (n = 5), it was 4.0% (95%CI: 0.0%–8.6%). Nine studies reported on being “unsure” about disclosure and in one study no respondents were unsure. The pooled percentage that was unsure was 9.2% (95%CI: 5.0%–13.40%). In studies in individuals without cognitive impairment that reported on this (n = 7), the pooled percentage that was unsure was 11.8% (95%CI: 3.5%–20.0%). In two studies in individuals referred to a memory clinic 7% and 8% of participants were unsure about whether they would want to be informed if diagnosed with dementia. In one study among individuals with dementia, no respondents were
Table 1. Characteristics of included studies and main outcomes PREFERENCE REGARDING
N STUDY
COUNTRY
METHODS
PARTICIPANTS
/
RESPONSE
AGE
(%)
(YEARS)
DISCLOSURE OF
MAIN REASONS
A DIAGNOSIS
IN FAVOR OF OR
OF DEMENTIA
(%)
AGAINST DISCLOSURE
.........................................................................................................................................................................................................................................................................................................................................................................................................................................................
General Population or Individuals Not Selected on Cognitive Status USA
Self-completion questionnaire after description of a patient with dementia
Harvard 2011
Germany France Spain Poland
Telephone survey
Holroyd et al., 1996
USA
Ouimet et al., 2004
Canada
Patients waiting to see their physician (family practice or internal medicine practice) > 21 years Nationally representative random samples of adults > 18 years
224 / Not measured Age: < 50: 43.6% 50–64: 37.8% >65: 18.5%
92% wanted to know
Main reasons for disclosure: “advanced planning for finances or personal care” and “obtaining a second opinion”
2678 / Not reported Age: Not reported
Not reported
Self-completion questionnaire after description of a patient with AD
Community dwelling older persons > 65 years
156 / 237 (66%) Mean age: 79.7 (SD 6.9)
97% (France), 98% (Germany), 96% (Poland), 94% (Spain) wanted to know 3% (France), 2% (Germany), 2% (Poland) 5% (Spain) would not want to know The rest was not sure 80% wanted to know 11 % would not want to know 10% was not sure
Structured interview after description of progressive AD
Elderly subjects (>65) attending medical and surgical outpatient clinics
204 / 236 (86%) Age: 65–79: 91.7% >80: 8.3%
98% wanted to know When the possibility of treatment with medication was mentioned, this increased to 99%
Main reasons for disclosure: “to do advance planning,” “Financial planning,” and to “settle family matters.” 3.2% (n = 4) answered: “to consider suicide while able. Main reason against disclosure: “no cure anyway” and “would be to upset” Main reasons for disclosure: “to understand what happens, to try to help themselves,” “to prepare, to plan,” and “always want to know diagnosis”
Dementia—preferences regarding disclosure
Erde et al., 1988
5
6
PREFERENCE REGARDING
N STUDY
COUNTRY
METHODS
PARTICIPANTS
/
RESPONSE
AGE
(%)
(YEARS)
DISCLOSURE OF
MAIN REASONS
A DIAGNOSIS
IN FAVOR OF OR
OF DEMENTIA
(%)
AGAINST DISCLOSURE
.........................................................................................................................................................................................................................................................................................................................................................................................................................................................
Shimizu et al., 2008
Brazil
Structured interview before and after receiving information on AD
A control group without a family member with AD
100 / Not measured Mean age: 56.3
98% wanted to know
Sullivan and O’Conor 2001
Australia
Questionnaire before and after exposure to AD education
First year undergraduate psychology students
100 / Not reported Mean age: 26.8 (SD 10.2)
93% wanted to know
Turnbull et al., 2003
USA
Structured interview after description of a patient with AD
Subjects > 65 years at a medical outpatient clinic. Patients with known dementia were excluded
200 / 206 (97%) Mean age: 73.0
92% wanted to know 4% did not want to know 5% was not sure.
Main reasons for disclosing: “right to know” and “to receive treatment and more information” Reasons against disclosure: “would get depressed or shocked” Main reasons for disclosure: “right to know,” “plan ahead for future,” and “explore treatment options.” 9% (n = 6) answered: “to plan my suicide” Main reasons against disclosure: “don’t want to know of any illness,” “no benefit in knowing,” and “diagnosis would depress or agitate.” 50% (n = 2) answered “fear of committing suicide” Main reasons for disclosure: “advance/Financial planning” and “to get a second opinion.” 2% (n = 3) answered: “to commit suicide” Main reason against disclosure: “would be too upset” and “no cure anyway”
P. van den Dungen et al.
Table 1. Continued
Table 1. Continued PREFERENCE REGARDING
N STUDY
COUNTRY
METHODS
PARTICIPANTS
/
RESPONSE
AGE
(%)
(YEARS)
DISCLOSURE OF
MAIN REASONS
A DIAGNOSIS
IN FAVOR OF OR
OF DEMENTIA
(%)
AGAINST DISCLOSURE
.........................................................................................................................................................................................................................................................................................................................................................................................................................................................
Umegaki et al., 2007
Japan
Postal questionnaire
Random sample of residents > 40 years in Nagoya
4042 / 7000 (58%) Age: 40–65: 23% > 65: 77%
80% of younger and 76% of older respondents wanted to know 3% of younger and 5% of older respondents would not want to know. The rest was not sure
Main reasons for disclosure: “to consult or convey wills about the future,” “to receive treatment,” “to accept and cope with it” Main reasons against disclosure: “I may be depressed or lose hope” “because you cannot help having dementia when you grow old” and “no advantage of knowing”
Johnson et al., 2000
UK
Postal questionnaire
40 / 55 (73%) Age: Not reported
72% wanted to know 12.5 would not want to know 15% was not sure
Not reported
Raicher et al., 2008
Brazil
Questionnaire sent by email
Doctors working in old-age psychiatry and geriatric medicine Geriatricians, neurologists and psychiatrists
181 / 970 (19%) Mean age: 45 (SD: 11)
77% wanted to know 9% would not want to know 14% was not sure
Not reported
Belgium
Self-completion questionnaire
Family members of people with Alzheimer’s disease
100 / 100 (100%) Age: Not reported
90% wanted to know the diagnosis of AD
Not reported
UK
Postal questionnaire
Caregivers of people with dementia
71 / 155 (46%) Mean age: 61.3 (SD: 13.5)
58% wanted to know 21% would not want to know 21% was not sure
Not reported
Physicians
Bouckaert and Van den Bosch 2005 Heal et al., 1998
Dementia—preferences regarding disclosure
Relatives
7
8
PREFERENCE REGARDING
N STUDY
COUNTRY
METHODS
PARTICIPANTS
/
RESPONSE
AGE
(%)
(YEARS)
DISCLOSURE OF
MAIN REASONS
A DIAGNOSIS
IN FAVOR OF OR
OF DEMENTIA
(%)
AGAINST DISCLOSURE
.........................................................................................................................................................................................................................................................................................................................................................................................................................................................
Lin et al., 2005
Taiwan
Self-completion questionnaire
Family members of neurologic patients
143 / 150 (95%) Mean age: 55 (SD: 14.3)
97% wanted to know Of subjects with AD family members 93% wanted to know the diagnosis
Maguire et al., 1996
Ireland
Structured interview
Family members of people with dementia
100 / 100 (100%) Age: Not reported
71% wanted to know
Mimura 2003 Mormont et al., 2012 Pinner and Bouman 2003
Japan
Self-completion questionnaire Structured interview
Caregivers of people with dementia Caregivers of patients with dementia Caregivers of patients with mild dementia
46 / 46 (100%) Age: Not reported 107 / 108 (99%) Mean age: 65 (SD: 14.7) 50 / Not measured Age: Not reported
93% wanted to know
Main reasons for disclosure: “right to know” and “accepting treatment and activity training” Main reasons against disclosure: “causing emotional disturbance,” “would worsen disease.” 20% (n = 1) answered “diagnosis could cause suicidal ideation” Main reason for disclosure: “right to know” and “future planning”. 8% (n = 6) answered: “commit suicide” Main reasons against disclosure: “it would depress me.” 7% (n = 2) answered: “commit suicide” Not reported
93% wanted to know
Not reported
98% wanted to know
Main reasons for disclosure: “to know the truth” and “future planning” Reason against disclosure: “it would upset me”
Belgium
UK
Semi-structured interview
P. van den Dungen et al.
Table 1. Continued
Table 1. Continued PREFERENCE REGARDING
N STUDY
COUNTRY
METHODS
PARTICIPANTS
/
RESPONSE
AGE
(%)
(YEARS)
DISCLOSURE OF
MAIN REASONS
A DIAGNOSIS
IN FAVOR OF OR
OF DEMENTIA
(%)
AGAINST DISCLOSURE
.........................................................................................................................................................................................................................................................................................................................................................................................................................................................
Shimizu et al., 2008
Brazil
Semi-structured interview before and after receiving information on AD
Caregivers of AD patients
100 / Not measured Mean age: 60 (SD not reported)
90% wanted to know
Main reasons for disclosure: “right to know” and “ to receive treatment and information” Main reason against disclosure: “would get depressed or shocked”
30 / 38 (79%) Age: 65–74: 10% 75–84: 17% >85: 33 % 46 / 50 (92%) Mean age: 79 (61–90) 36 / 95 (38%) Mean age: 75.9 (SD 5.8)
83% wanted to know No patients said they would not want to know 7% was not sure
Main reasons for disclosure: “want to know the truth,” “to prepare for future” and “to obtain more information” Not reported
Individuals with Dementia or Referred to a Memory Clinic Canada
Qualitative interview
New patients referred to the Geriatric Day hospital
Dautzenberg et al., 2003
Nether-lands
Self-completion questionnaire
Elson, 2006
UK
Individual interviews
Out-patients referred to a memory clinic Patients > 65 years referred for investigation of memory complaints
96% wanted to know.
86% wanted to know 6% did not want to know 8% was not sure. After receiving information about AD: 69% wanted to know 17% did not want to know 11% was not sure.
Main reasons for disclosure: “to enable advance planning,” “general desire to be well informed,” and “to consider treatment.” Main reason against disclosure: “diagnosis would cause anxiety/distress”
Dementia—preferences regarding disclosure
Byszewski et al., 2007
9
10
PREFERENCE REGARDING
N STUDY
COUNTRY
METHODS
PARTICIPANTS
/
RESPONSE
AGE
(%)
(YEARS)
DISCLOSURE OF
MAIN REASONS
A DIAGNOSIS
IN FAVOR OF OR
OF DEMENTIA
(%)
AGAINST DISCLOSURE
.........................................................................................................................................................................................................................................................................................................................................................................................................................................................
Jha et al., 2001
UK
Postal questionnaire
Marzanski et al., 2000
UK
Structured interview
Patient seen by Old age psychiatry Services diagnosed with dementia and depression (as control group) Patients with dementia
Mormont et al., 2012
Belgium
Structured interview
Patients with dementia
Pinner and Bouman 200332
England
Semi-structured interview
Patients with mild dementia
Pratt and Wilkinson 2001
UK
Qualitative interview
People with dementia
100 / 113 (88%) 53 dementia; 47 depression Mean age: 77 (SD 7)
After reading an unedited copy of their full clinical assessment 75% of people with dementia said it was a good idea to read the diagnosis
Not reported
30 / 30 (100%) Mean age: 81 (63–92)
70% wanted to know what was wrong with them or get more information if already knew. 33% wanted to know their diagnosis 30% did not want to know what was wrong with them or receive information 95% wanted to know 5% would not want to know.
Not reported
107 / 108 (99%) Mean age: 76.6 (SD 5.2) 50 / Not measured Mean age: 78.5 (SD: 6.6)
24 / 24 (100%) Age: < 65: 46% ࣙ 65: 54%
92% would want to know
All participants who knew their diagnosis felt that people with dementia should be told their diagnosis
Not reported
Main reasons for disclosure: “to know the truth” and “right to know Main reason against disclosure: “it would upset me” Reasons for disclosure: “like any other illness I had to know,” “I had to know,” “there’s nothing worse than other people know something about you and try to protect you”
P. van den Dungen et al.
Table 1. Continued
Dementia—preferences regarding disclosure
11
Figure 2. Meta-analysis of percentages in favor of disclosure. ∗ Articles occur twice because they describe preferences of (two) subgroups. All pooled percentages are based on meta-analysis using a random effects model (DerSimonian & Laird 1986).
unsure, the other four studies in individuals with dementia did not report on this. Preferences and publication date A post hoc analysis revealed that overall, the mean percentage in favor of disclosure in studies published less than 10 years ago (2003 or later) was significantly higher compared to studies older than 10 years (91.1% vs. 78.9%, p = 0.03). The same was found in studies with individuals without cognitive impairment (all studies: 91.5% vs. 77.6%, p = 0.01; only studies with a response rate ࣙ75%: 93.5% vs. 71.0, p = 0.001). In studies in individuals with cognitive impairment, the trend was similar but not statistically significant (all studies: 90.4% vs. 81.7%, p = 0.45; only studies with a response rate ࣙ75%: 91.3% vs. 81.7%, p = 0.40). Central arguments in favor of and against disclosure In 13 articles, respondents were asked for their arguments in favor of or against disclosure. The most frequently encountered arguments are presented here. The possibility to compare arguments between groups was limited due to
the different methods used: open questions versus prescribed answer categories and one answer required versus the option to choose more than one answer. The most frequently expressed arguments by individuals from all subgroups who would want to be told about a diagnosis of dementia were: “to plan the future” (reported in 10 studies), “it’s my right to know” (7 studies), “to look for treatment options” (6 studies), and “to obtain more information” (4 studies) or “a second opinion” (2 studies). Notably, specifically individuals with dementia expressed that they wanted “to know the truth” (four studies; three among individuals with dementia). Another argument we encountered was “to plan a suicide.” This weighty argument for wanting to be told was expressed by 2% to 9% of respondents in favor of disclosure in three studies; one among relatives and two among the general population (Maguire et al., 1996; Sullivan and O’Conor, 2001; Turnbull et al., 2003). This argument was never mentioned by individuals referred for evaluation or diagnosed with dementia. The main arguments against disclosure were: “it would upset or depress me” (10 studies) and “there are no benefits in knowing” (4 studies). A
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P. van den Dungen et al.
noteworthy argument against disclosure expressed by some respondents in studies performed in Taiwan and Japan was the believe that knowing the diagnosis might exacerbate the symptoms of dementia (Lin et al., 2005; Umegaki et al., 2007). In contrast to favoring disclosure to plan a suicide, “fear of committing suicide” was mentioned by 11% to 50% of individuals who would not want to know, in three studies: one in the general population and two in relatives (Maguire et al., 1996; Sullivan and O’Conor, 2001; Lin et al., 2005).
Discussion This review shows that nine of ten individuals without cognitive impairment would prefer to be informed if diagnosed with dementia in the future. A smaller proportion, but still the vast majority, of individuals referred to a memory clinic or already diagnosed with dementia preferred full disclosure. Only few individuals explicitly stated not wanting to know. Remarkably, in the two studies among physicians a relatively large number preferred not to be informed. The main arguments in favor of disclosure pertained to autonomy: knowing what is going on, being involved in the planning of future care, looking for treatment options, and getting information were important for many respondents. The main arguments against disclosure were the assumption that knowing the diagnosis would not have any benefit and the fear of getting upset or depressed. A striking argument, both used in favor of and against disclosure, was the possibility to plan suicide, or the fear of committing suicide. The latter was not uncommon as an argument against disclosure. However, in the studies among referred or diagnosed patients, suicide was not mentioned at all. This may reflect that a selected group was referred or diagnosed (see limitations section), or that such notions change over time, possibly related to the progression of cognitive impairment or adaptation to disease. One argument against disclosure that was reported in studies from Taiwan and Japan was the fear that disclosure could worsen the disease (Lin et al., 2005; Umegaki et al., 2007). Other studies did report fear of getting upset or depressed by disclosure but did not link disclosure to disease progression. Whether this reflects cultural differences in disease perception, or whether this is due to language or to methodological differences between studies, is not clear. We did not find studies specifically designed to assess cultural influences on preferences regarding disclosure. So far, no study has shown an association between demographic
characteristics and wanting or not wanting to know (Erde et al., 1988; Holroyd et al., 1996; Lin et al., 2005). Thus, although some cohort effect is likely also present, our finding that more respondents preferred to know in more recent studies is probably mostly explained by the trend toward much greater diagnostic openness over the last decades (VernooijDassen et al., 2005). Physician behavior and attitude A discrepancy between patient preferences and physician behavior emerges. Where we found that the majority of individuals wanted to be informed and thus empowered, a review published in 2004 concluded that around 50% of clinicians routinely withheld a dementia diagnosis (Carpenter and Dave, 2004). The two studies on physician behavior included in this review that also studied disclosure behavior and other recent studies on this topic report similar results (Johnson et al., 2000; van Hout et al., 2006; Raicher et al., 2008; Segers, 2009). It was shown that physicians may encounter a “double taboo” when communicating a diagnosis of dementia: their own barriers to disclosure and the patient’s resistance to accept or receive the diagnosis (Kaduszkiewicz et al., 2008). Physician barriers to diagnosis include their attitudes toward dementia but also educational needs and time constraints (Bradford et al., 2009). Physician barriers to disclosure include fear to harm the physician–patient relationship; the assumption that the patient would not understand the diagnosis; fear to harm the patient and diagnostic and therapeutic nihilism. In this context, it is striking that a relatively large part of physicians would not want to be informed if diagnosed with dementia (Johnson et al., 2000; Raicher et al., 2008). This may reflect a negatively biased experience with the natural course of dementia resulting from overexposure to the dreadful course that dementia can follow (Phillips et al., 2012; Vroomen et al., 2013). Physicians may not perceive that dementia patients’ self-reported quality of life is, if they have a good social network, comparable to that of older individuals without cognitive impairment (Cooper et al., 2011). Strength and limitations This systematic review provides insight into preferences regarding disclosure of unselected groups of individuals without cognitive impairment, and of more selected groups of individuals already known with cognitive impairment. The strength of this review lies in the comprehensive search, the thorough methodology used, and in the combination of quantitative data about preferences
Dementia—preferences regarding disclosure
with qualitative information about arguments for these preferences. A limitation of this review is that we aggregated data from studies with different goals and data collection mechanisms. However, all the included studies reported high percentages in favor of disclosure, supporting the validity of our main conclusions. A limitation present in studies concerning a hypothetical future diagnosis of dementia is that the answers may not represent respondents’ actual reaction if they would in fact develop dementia. We did not find any longitudinal studies measuring preferences before and after disclosure or before and after development of dementia. Therefore, we cannot say whether the lower rate in favor of disclosure among individuals with cognitive impairment is mostly due to the conversion of preference over time or rather to differences between studies (e.g., studied population or methodology). A potential limitation of some of the studies in individuals with dementia is that in the case of more advanced dementia, disease insight and awareness may be limited (Zanetti et al., 1999). As a result, the validity of a preference regarding disclosure becomes questionable. Moreover, individuals referred for evaluation or already diagnosed with dementia are a selected group that may be more receptive to disclosure. Individuals with dementia who do not seek or avoid diagnostic evaluation may also be more reluctant to disclosure. Finally, notions of and knowledge about dementia may vary considerably among respondents. Some studies tried to deal with this by providing information about dementia to respondents (Erde et al., 1988; Holroyd et al., 1996; Turnbull et al., 2003; Ouimet et al., 2004; Shimizu et al., 2008). In these studies the content is clearer. However, such studies may not reflect clinical practice where patients and families generally lack such standardized information. Implications for clinical practice Based on our findings, we think that physicians need to be willing and able to disclose a diagnosis of dementia. In comparison with other common conditions, patients with dementia are more dependent on their physicians (or family) to initiate the process of diagnosis and disclosure. Apart from the emotional barriers, dementia patients’ help seeking behavior may be hampered by limited disease awareness or impaired cognitive function (Bradford et al., 2009). Willingness to diagnose and disclose requires that physicians appreciate the value of diagnosis for the patient (this review) and relatives, even when diagnosis may not have immediate practical or therapeutic consequences. Being able to diagnose requires that physicians are
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skilled and confident in the process of diagnosing dementia. Establishing a firm diagnosis of dementia requires more time and effort from physicians than many other conditions. One important but difficult aspect of the diagnostic process is distinguishing dementia from mild cognitive impairment (or mild neurocognitive disorder). The latter condition is prognostically heterogeneous and whether diagnosing this condition is overall beneficial to the patient is subject of debate. Physicians should try to make this distinction early in the diagnostic process by assessing whether cognitive deficits cause a functional decline and thus interfere with independence. Where physicians need time to gather all the information required for a firm diagnosis, the patient and relatives may also appreciate some time to become receptive to this diagnosis. It was shown that “hints dropped along the way” helped patients to deal with the diagnosis (Byszewski et al., 2007). To be able to provide tailored information and to assess the risk of a possible catastrophic reaction (e.g., wanting to commit suicide), physicians should explore the patient’s ideas about the cause of their impairment, the patient’s knowledge of dementia, and what a diagnosis of dementia would mean to the patient. Although current evidence refutes that disclosure of a diagnosis of dementia is likely to cause long-term harm, it is clear that the period after disclosure is often very stressful for patients and their close ones (Aminzadeh et al., 2007; Lecouturier et al., 2008; Haw et al., 2009; Robinson et al., 2011; Seyfried et al., 2011). Thus, disclosure should always be an ongoing process of information provision and support. Previous studies suggest that numbers of physicians in favor of early diagnosis are increasing and that reluctance to disclose is decreasing (Johnson et al., 2000; Gely-Nargeot et al., 2003; Cahill et al., 2006; Raicher et al., 2008; Segers, 2009; Tarek et al., 2009). However, prerequisites for establishing a firm diagnosis of dementia may be missing in daily practice. Family physicians, who play an important role in the initial diagnosis of dementia, were shown to lack knowledge and confidence to diagnose dementia (van Hout et al., 2000; Koch and Iliffe, 2010). In addition, comorbid conditions, likely to be present in elderly patients, may compete for attention and time (Iliffe et al., 2009). Similar barriers are likely present in the hospital setting. To improve the process of diagnosis and disclosure, physicians need to be aware of their own barriers to discuss cognitive impairment and need to learn how to initiate diagnostic assessment and disclosure of a dementia diagnosis. In addition, physicians may benefit from education to learn
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to communicate about the diagnosis of dementia in a patient (and family)-centered manner and to provide patients with the information and support they need. Some physicians may also benefit from freshening of their knowledge and skills to diagnose dementia (Foy et al., 2007a; Lecouturier et al., 2008). In case patients appear reluctant to face disclosure of a dementia diagnosis, we recommend to cautiously explore their arguments against disclosure. Such exploration may help to identify fear of or misunderstandings about dementia. Often, some form of disclosure can hardly be avoided. If someone with dementia is, for example, still driving, this person may be potentially hazardous to himself or other people. Implications for future research Although the majority prefers disclosure, empirical evidence on the impact of pro-active diagnosis and disclosure on the wellbeing of patients and relatives is not yet available. In addition, we need to develop and test methods to learn physicians to raise the topic of cognitive impairment, to disclose a diagnosis of dementia, and offer education in a patient-centered manner (Byszewski et al., 2007; Lecouturier et al., 2008; Eccles et al., 2009; Foy et al. 2007b). As part of this, we need to gain more insight into understanding of dementia and preferences regarding disclosure of a diagnosis of dementia of populations from different cultural backgrounds.
Conclusion The vast majority of individuals with and without cognitive impairment both in the community and in specialist settings wants to be told about this diagnosis if they would develop dementia. The main arguments for this preference pertain to autonomy. Patients may benefit most from physicians who inquire openly about what the patient would like to know or fears to face, and who are aware of their own barriers in diagnosing and disclosing dementia. Conflict of interest None. Description of authors’ roles P. van den Dungen, L. van Kuijk, and H. van Hout designed the study. H. van Hout, H. van Marwijk, and P. van den Dungen supervised the data collection. J. van der Wouden, H. van Marwijk, and P. van den Dungen were responsible for the statistical design of the study and P. van den Dungen carried out the statistical analysis. E. Moll
van Charante and H. van der Horst assisted with the statistical design and with writing the article.
Acknowledgements We thank René Otten, medical information specialist at the VU Amsterdam University Library for his support when designing the search strategy. This study was sponsored by grants from the Stoffels-Hornstra foundation, a Dutch non-profit organization supporting research in cancer, brain diseases, cardiovascular disease, and eye diseases, and the National Programme for Improving Care for Older Persons (ZonMw no. 313080201).
Supplementary material To view supplementary material for this article, please visit http://dx.doi.org/10.1017/ S1041610214000969
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