Neurol Sci (2011) 32:331–335 DOI 10.1007/s10072-010-0365-7
BRIEF COMMUNICATION
Sudden unexpected death in epilepsy (SUDEP): a pilot study on truth telling among Italian epileptologists Elena Vegni • Daniela Leone • Maria Paola Canevini Paolo Tinuper • Egidio Aldo Moja
•
Received: 27 April 2009 / Accepted: 22 June 2010 / Published online: 6 July 2010 Springer-Verlag 2010
Abstract Sudden unexpected death in epilepsy (SUDEP) is a syndrome where a person with epilepsy dies suddenly and no other cause of death is found. The question of informing patients and their families about SUDEP remains a problematic issue. The aim of this study is to explore whether Italian physicians interested in epilepsy believe that they should discuss SUDEP with patients and/ or their families. A total of 315 questionnaire were distributed, of which 195 (61.9%) were returned. Seventeen respondents (8.76%) discussed SUDEP with all of their patients, 38 (19.59%) with the majority of patients, 120 (61.85%) with very few of their patients and 15 (7.73%) with none of their patients. No statistical differences among groups were found for gender, professional age (B10 years; C11 yearsB20 years; C21 yearsB30 years; C31 years) and medical specialty (neurologists vs. others). Open questions offered insights into the physicians’ problem of managing the negative emotions of E. Vegni E. A. Moja School of Medicine, University of Milan, Milan, Italy D. Leone Centro CURA, University of Milan, Milan, Italy M. P. Canevini Epilepsy Centre, School of Medicine, San Paolo University Hospital, University of Milan, Milan, Italy P. Tinuper Department of Neurological Sciences, University of Bologna, Bologna, Italy E. Vegni (&) E. A. Moja Department of Medicine Surgery and Dentistry, San Paolo University Hospital, Via di Rudinı` 8, 20142 Milan, Italy e-mail:
[email protected]
patients/family and why the physicians decided to give information. Keywords Epilepsy SUDEP Truth telling Questionnaires
Introduction People with epilepsy have a two- to threefold higher mortality rate than the general population, because of the epilepsy itself (epilepsy-related death) or the underlying cause of epilepsy [1]. Sudden unexpected death in epilepsy (SUDEP) is a syndrome where a person with epilepsy dies suddenly and no other cause of death is found [2]. It is the commonest cause of epilepsy-related death in refractory epilepsies [3] The incidence of SUDEP can vary from 1 in 11,400 people-years of follow-up in people with newly diagnosed epilepsy to 1 in 200 for adults with refractory epilepsy attending a tertiary referral center [4, 5]. Reports of people dying alone in their sleep seem to be frequent. Death is thought to occur following a seizure, but since the deaths are often unwitnessed this is only an assumption. The most important risk factor for SUDEP appears to be poor seizure control [6]. The question of informing patients and their families about the risk of SUDEP, and whether and when SUDEP should be discussed remain a problematic issue [7]. Reasons in favor of informing are based on the evidence that awareness of the increased risk may enhance patients’ and parents’ cooperation with therapy. Compliance with treatment and frequent nightly observations of patients at high risk may help to minimize the incidence of SUDEP. Patients and their families should be made aware of SUDEP [8], and the education of patients, relatives and
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caregivers should be a core medical goal to reduce its incidence [8]. Last, but not least, most relatives of patients who died from SUDEP claimed that they would have preferred to have known that epilepsy could be fatal [9]. Nonetheless, reasons for not discussing SUDEP may also be advocated. Information on SUDEP may not alter the outcome, and, conversely, disclosure that is unsolicited by the patient may adversely affect his/her quality of life [10]. Only one study, among UK neurologists, has explored the habit of disclosing the risk of SUDEP [11]. The results showed that only 4.7% of respondents discussed SUDEP with all of their patients, while 7.5% did not discuss the issue with any of their patients [11]. The findings are interesting because they emerge from an Anglo-Saxon culture where the task of fully informing patients is a leading principle and a necessary requirement for medical practice. The cultural attitude of Anglo-Saxon countries about the truth-telling practices seems to be largely different from the Italian culture [12]. In Italy medical ethics and legislation on patients’ self-determination have evolved dramatically in the past 10 years. Yet, the practice of partial or non-disclosure is still common, and the general level of patients’ awareness of diagnosis and prognosis is still limited [13]. Two reports on truth telling in Italy suggest the importance of the ongoing debate. Only 34% of Italian respondents in a survey of different European intensive care units believed that full information should always be given to patients. Moreover, a discrepancy between what physicians state they believe and what they actually do in their own practice is discussed in the paper [14]. The second study estimated the proportion of Italians dying of cancer who had received information on their diagnosis and prognosis. Results showed that only 37% of people had received information on diagnosis and 13% on the poor prognosis [15]. Despite the possible differences in the intensive care and oncology context from that of epilepsy, it is interesting to note that the two studies seem to witness an attitude of Italian doctors to protect their patients from bad news, in particular when the news is about the patients’ death. The aim of this preliminary study is to explore whether Italian physicians interested in epilepsy believe that they should discuss SUDEP with patients and/or their families or not.
Methods The present study partly replicated the UK study previously described [11]. In particular, on the basis of the UK questionnaire, an Italian questionnaire was developed asking respondents to give general and professional data (age, sex, degree, specialization, years of professional
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experience); a closed question (‘‘I believe that SUDEP should be discussed…’’) with one choice among four answers (A. with all of my patients; B. with the majority of my patients; C. with very few of my patients; D. with none of my patients) and two open questions were presented. The questionnaire used in our study is presented in Table 1. The questionnaire was distributed to all the participants at the National Congress organized by the Italian Association Against Epilepsy (LICE). The LICE is the main Italian Scientific Association whose participants are medical doctors from different disciplines, particularly interested in the field of epilepsy. Descriptive statistics were used for general and professional data and for closed questions. Data were stratified for the following variables: gender, professional experience (B10 years; C11 yearsB20 years; C21 yearsB30 years; C31 years), and medical specialization (i.e., neurologists vs. others). A v2 test was used for statistical analysis of differences among groups. Open questions were analyzed by a content analysis [16] that included three stages. In the first stage, to identify the main issues in the participants’ contributions, two coders independently discussed and listed all the possible themes arising from the answers. In a second stage, the themes were attributed to one or more content categories independently by the two coders (in case of different ratings, the coders discussed them until a consensus was reached). Finally, in the third stage, the frequency of the participants’ answers for each content category identified was calculated.
Results As much as 315 questionnaires were distributed, of which 195 (61.9%) were returned. One questionnaire was excluded because it was filled by a biologist. Among respondents, 98 (50.5%) were women and 96 (49.5%) were men. Their mean age was 45.4 years (range 25–64), and their mean professional experience was 21.4 years (range 1–42). Seven (3.6%) had no specialization; 9 (4.6%) were residents and 179 (91.8%) had one or more specializations (125 in neurology; 53 in child neuropsychiatry; 28 in neurophysiopathology; 11 in pediatrics; 4 in psychiatry; 2 in neurosurgery; 1 in clinical pharmacology; 1 in physiatry). The distribution of the participants’ answers to the closed question is presented in Table 2. The v2 analysis of differences was not statistically significant (for the gender: v2 = 0.330, df 3, n.s.; for the professional experience: v2 = 0.041; df 3, n.s.; for the medical specialization: v2 = 0.197, df 3, n.s.). For the first open question (‘‘When I happened to discuss SUDEP with my patients, their reaction was’’), 110
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Table 1 The questionnaire distributed to all the participants at the National Congress organized by the Italian Association Against Epilepsy (LICE)
Age ……… Sex …… If not graduated in medicine, specify qualifications ……………………………….. Years after graduation ……………………………….. Specialization ………………………………………… I believe that the possibility of a risk of SUDEP should be discussed (mark one): With all of my patients With the majority of my patients With very few of my patients With none of my patients When I happened to discuss SUDEP with my patients, their reaction was (answer only if you discussed SUDEP with any patients): …………………………………………………………………………………………………… …………………………………………………………………………………………………… …………………………………………………………………………………………………… …………………………………………………………………………………………………… Why did you discussed the risk of SUDEP (if you did) with your patients? …………………………………………………………………………………………………… …………………………………………………………………………………………………… …………………………………………………………………………………………………… …………………………………………………………………………………………………….
Table 2 Descriptive analysis of the answers to the closed question ‘‘I believe that the possibility of a risk of SUDEP should be discussed’’
N
%
With all of my patients
17
8.76
With the majority of my patients
38
19.59
120
61.85
15
7.73
4
2.06
With very few of my patients With none of my patients Missing dataa Total no. of respondents a
194
answers were collected. Two main themes were identified with four sub-themes each. For the second open question (‘‘Why did you discuss the risk of a SUDEP (if you did) with your patients?’’), 100 answers were collected. Five main themes were identified. The themes, the frequency and percentage that resulted from the content analysis for the first and second open questions are reported in Table 3. Discussion
100
Three respondents gave an open answer, while one gave no answer
Our pilot study aimed to verify attitudes on disclosing information on SUDEP among Italian physicians involved
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Table 3 The themes, frequency and percentage of the result of content analysis of the first and second open questions Open question 1: ‘‘When I happened to discuss SUDEP with my patients, their reaction was…’’
N 110
100%
1. A patient’s emotional response
89
80.2
1.1. An emotional response characterized by anguish and fear
45
40.5
1.2. A response characterized by different degrees of emotional and cognitive disturbance (e.g., disbelief, refusal, relative awareness)
34
30.6
1.3 An emotional response characterized by anger
3
2.7
1.4 A positive emotional response (e.g., satisfaction for the involvement)
7
6.3
21
19.8
15
13.5
2. A patient’s concrete response 2.1 Patient’s request for information, practical suggestions and reassurance 2.2 Patient’s intention to take care of him/herself and his/her own health
4
3.6
2.3 A change in the doctor/medical center in charge
2
1.8
2.4 A decrease in the quality of life
1
0.9
N 100
100%
Open question 2: ‘‘Why did you discuss the risk of SUDEP (if you did) with your patients?’’ 1. Because of the peculiar status of the disease/treatment
47
47
2. Because he/she asked me
16
16
3. To strengthen the therapeutic relationship and to favor the patient’s choice
15
15
4. Because it is a medical duty
14
14
8
8
5. For reasons concerning myself as a person
in the care of epilepsy patients. Of the respondents, 28.35% claimed that they deemed SUDEP should be discussed with all or almost all their patients. In a similar UK study, 30.30% of neurologists claimed that they discussed SUDEP with all or almost all their patients. At least in the field of epilepsy, and particularly for SUDEP, Italian doctors seemed to have the same cultural attitude of informing patients as the British sample, even with a higher percentage of answers ‘to all my patients’ (8.76%) in the Italian sample than among British doctors (4.7%). It is possible that this percentage could be an overestimation: in fact, about 40% of physicians involved did not respond to the questionnaire and probably this group included a high percentage of physicians who were not much aware of SUDEP aspects or did not care a lot about information on SUDEP. So, though 30% of doctors deemed that SUDEP should be discussed with all or almost all of their patients, they are to be considered as constituting the upper bound of a particularly sensitive sub-group of physicians. Moreover, the study did not yield information on actual practice, and it is possible that Italians (but also UK) physicians may act differently from what they claim. The lack of a significant relationship between hypothesized predictors (gender, professional experience, medical specialization) and propensity to disclose SUDEP information may have been due to the small sample and suggests the need for further development of the research. The insights obtained from open questions may be more interesting. The question about patients’ reactions emphasizes the enormous problem of managing a response that is mostly (73.8%)
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emotional and negative, such as anguish (40.5%), refusal (30.6%) or anger (2.7%). As in other fields such that of the oncology, therapeutic privilege is one of the main issues involved in breaking news [17], in particular when managing communication with patients affected by severe illness, and is linked to the problem of dealing with the patient’s emotional reaction [18]. Lack of experience and training may be an important deterrent that daunts physicians in talking about the risks caused by epilepsy. The second question may disclose why the physicians decide to give information. The answers seem to reflect a disease-centered approach, mostly recognized in the pertinent literature [19]: the most common answer (47%) suggests that the decision is due to specific clinical situations or therapeutic choice. However, about 50% of the answers seem to reflect a changing culture where the decision to talk may also be driven by the patient or by nonclinical reasons, such as ethical or deontological issues. This may constitute a new perspective where it is crucial to enhance patient empowerment, or even a very subjective view of the doctor as a person saying (excerpts are from the questionnaires) ‘if I were him/her, I would want to know’, ‘because I’ve lost a patient from SUDEP’, ‘I would be worried about the consequences for my own job of not speaking out’. Our study suffers from many limitations due in particular to the small and nonrepresentative sample, recruited from among participants of a National Congress on epilepsy. This method could have engendered selection bias. Moreover, we had no chance to identify participants
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particularly interested in epilepsy: we compared groups based on speciality, but not specifically on their clinical interest and practice. Our results are descriptive and, as such, the study needs further research.
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