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Palliative care in chronic progressive neurological disease
Changing perspectives
Seeber, A.A.
Publication date
2019
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Citation for published version (APA):
Seeber, A. A. (2019). Palliative care in chronic progressive neurological disease: Changing
perspectives.
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eXPerienCes anD reFleCtions oF Patients
With Motor neuron Disease on BreaKing
the neWs in a tWo tiereD aPPointMent.
a Qualitative stuDY
antje a. seeber, a. Jeannette Pols, albert hijdra, hepke F. grupstra, Dick l. Willems, Marianne de visser
aBstraCt
Background
Breaking bad news should be fine-tuned to the individual patient, contain intelligible information, include emotional support and offer a tailor-made treatment plan. to achieve this goal in motor neuron disease (MnD), neurologists of the als centre amsterdam deliver the message on two separate visits within 14 days.
Aim
to evaluate how patients with MnD react to and view disclosure of the diagnosis, in this two-tiered approach.
Methods
non-participating observations and in-depth interviews with patients were conducted in one tertiary als referral centre. Qualitative analysis consisted of open coding and inductive analysis of observation reports and verbatim typed out interviews.
Results
ten two-tiered appointments were observed and 21 Dutch MnD patients interviewed. they experienced the straightforward message to be suffering from a fatal disease as devastating, yet unavoidable. the prospect of a short-term second appointment offered structure for the period immediately following the diagnosis. the time between appointments provided the opportunity for a first reorientation on their changed perspective on their life. the second appointment allowed for detailed discussions about various aspects of MnD and a tailor-made treatment plan.
Conclusion
the two-tiered approach fits well with the way in which Dutch MnD patients process the disclosure of their diagnosis, gather information and handle the changed perspec-tive on their life. it may serve as a model for other life-limiting diseases.
Introduction
Discussing the diagnosis amyotrophic lateral sclerosis (als) or progressive muscular atrophy (PMa) with a patient does not only involve the disclosure of bad news, but also taking the first step in a palliative care trajectory.1-3 Both these motor neuron diseases
(MnD) shorten the patient’s life expectancy significantly. in als, 50% of patients die within 36 months after symptom onset and in PMa within 48 months.4 Death is usually
caused by respiratory failure as a result of progressive muscle weakness or by cachexia.2,5 the only currently available disease-modifying drug, riluzole, prolongs life
by only a few months.6 supportive interventions, such as feeding via a gastrostomy tube
and non-invasive ventilation, do not prevent severe and progressive disability either.7,8
When breaking the news of a life-threatening disease, the medical information discussed should be intelligible and adjusted to the needs and preferences of the patient.9-11 support should be empathic to alleviate the emotional impact of the
diagnosis and the feeling of isolation experienced by patients.1,12,13 Finally, a treatment
plan should be developed based on shared decision making between healthcare professional and patient.14-16 these standards are primarily derived from research
in oncology and oncologists’ expert opinions.9,12,17,18 For the vast majority of cancer
patients, however, disease-modifying therapy is available, and – in contrast to MnD – treatment is not initially labelled ‘palliative’. the effect of a lack of potentially curative treatments on discussing the diagnosis of a life-threatening disease has been investigated in only a few oncology and MnD studies.13,19-27
in the als tertiary referral centre in amsterdam, a patient satisfaction survey concern-ing the diagnostic course revealed that a sconcern-ingle appointment for breakconcern-ing bad news did not fully meet the needs and preferences of MnD patients (unpublished data 2009). Patients stressed that they needed more time at short notice to discuss all their questions and the plans for their treatment. this was also found in other studies on breaking the news in als.13,14,21 the centre therefore decided to plan two consecutive
consultations, 10 to 14 days apart, to break the news and to discuss treatment strate-gies, both with the same MnD specialist. the second appointment would also include an initial consultation with the rehabilitation physician (table 1).
in this report we present an evaluation of this approach by means of non-participating observations of the consultations and semi-structured interviews with patients.
Methods
in this study an inductive content analysis was performed. the article is presented according to the Consolidated Criteria for reporting Qualitative research (CoreQ, table s-1 (supplementary data)).28,29
Table 1. Strategy for MND specialists to disclose the diagnosis ALS/PMA
Derived from the eFns guidelines on the Clinical Management of amyotrophic lateral sclerosis 201214 and regular evaluation of the daily practice at the ‘als Centrum nederland’ (aCn), including
Setting
in the netherlands diagnostic and treatment protocols for MnD patients have been harmonized under the umbrella of the ‘als Centrum nederland’ (aCn), located in amsterdam and utrecht. the consultants at this centre are neurologists and rehabilitation physicians with expertise in MnD. People with suspected MnD can be seen within one week after referral by a general neurologist or general practitioner (gP). as the two-tiered approach for breaking bad news has only been introduced in amsterdam so far, we recruited patients from that location’s outpatient clinic only. Study design
all participants were followed during a six months’ period by means of non-partici-pating observations of all appointments with the specialist in charge. as part of a larger study to explore advance care planning with MnD patients in daily practice, participants were either followed from breaking the bad news onwards, at relatively early stages of disease (group 1; seen by both neurologist and rehabilitation physician during the observation period) or during more advanced stages of disease (group 2; seen by the rehabilitation physician during the observation period). the single in-depth interview with every participant took place after the observation period.
Participants
We purposively sampled adult patients with the two major variants of MnD, amyotro-phic lateral sclerosis (als, including the bulbar-onset form (‘progressive bulbar palsy’) and the limb-onset form), and progressive muscular atrophy (PMa), seeking broad variation concerning the characteristics ‘age’, ‘gender’, ‘site of disease onset’ and ‘disease progression’, measured with the als-Functional rating scale (purposive sampling).30 We considered these to be important and rather easily accessible variables
which influence patients’ experiences and reflections and thus contribute to the hetero- geneity of the sample. Patients needed to know the Dutch language well, but might suffer from speech disorders as long as communication remained possible (e.g. by means of a speech computer). With regard to possible cognitive impairment, the only strict exclusion criterion was cognitive and behavioural decline due to als-FtD (Fronto- temporal dementia). eligible patients were first approached by a member of the aCn outpatient clinic amsterdam, either the neurologist (group 1) or the specialised nurse (group 2), informed about the study and, when interested, asked for verbal consent to be contacted by the researcher (aas). aas would give more detailed information about the study, verbally and in writing, and ask patients for written informed consent to carry out the study and use the gathered – anonymized – data for improvement of the outpatient clinic’s approach to diagnose and support patients with incurable progres-sive diseases. Patients knew that they could withdraw from the study at any moment, without explanation. Furthermore, it was guaranteed that no information obtained during the interview would be shared with anyone of the treating medical team.
Ethical approval
all patients gave informed consent. Dutch law specifies that ethics approval is only needed when ’participants are subject to procedures or are required to follow rules of behaviour’ (http://www.ccmo.nl/en/your-research-does-it-fall-under-the-wmo). as this was not the case, the approval of the local research ethics committee (reC) was not needed, as confirmed in a letter from the aMC reC 14th June 2010. nevertheless, participants were asked to contact the researcher or a member of their als team if participation in the study led to any questions or concerns.
Data collection
all non-participating observations and in-depth interviews were done by the first author (aas). as the interviews took place after an observation period of six months, interviewer and interviewee were acquainted with each other by that time.
During the observations the first author took extensive field notes which she worked out immediately afterwards. the second author read all transcripts and marked and labelled emerging themes, as did the first author. after reaching consensus about important themes, the first author explored and validated them subsequently during the following observations. this process was repeated several times. For the interviews, the authors also used an iterative method, adapting and accentuating the semi-structured interview guide, first developed based on key concepts identified in the literature and during the observations. Furthermore, the first author added questions concerning each interviewed patient’s personal illness trajectory, as observed during the six-month period preceding the interview, to his/her individual topics list. all interviews took place at the patients’ preferred location and time.
Analysis
observations were recorded and anonymized, interviews were audio-recorded, typed out verbatim, and anonymized by only the first author who saved all original data on a extern hard disc. observation reports and transcripts were analysed using MaXqda version 10 software.31 the first author ordered them chronologically. she coded
characteristics of the appointments and of the time in between, such as requests for clarity and information, emotional response of patients and their next of kin, concerns raised by patients, and other positive and negative evaluations of the meetings. all this was done shortly after each interview, so that findings could be iteratively fed into the interview guide and evolving coding tree. starting with the code ‘information’ and subcode ‘clarity of information’, for instance, the first author learned from several of the first interviewees that they understood the information given during the first appointment only after having talked things through a second time. a further focus on this aspect then led to the subcode ‘clearing up misunderstandings’.
to ensure that the data analysis was accurate, the second author validated the whole analytic process by reading all transcripts and taking an active part in coding as described above. the first author also discussed the codes with research colleagues from multiple backgrounds involved in other qualitative studies (investigator triangu-lation).32 the iterative coding and recoding operations ended when saturation was
reached, i.e. when no new categories or variations were needed to address the main research questions. the lists of transcripts under each code served further analysis following a qualitative approach to study ethical issues as they take place in clinical practice (empirical ethics).33
Results
ten patients were observed during their two-tiered appointment with the MnD specialists, and six of them were interviewed six months later. the other four patients were too ill to communicate (3) or had passed away (1) at the time that the interview could be scheduled. Fifteen additional MnD patients who had not been observed during their two-tiered appointment were interviewed about their experiences with this approach as well. one patient declined participation (table s-2 (supplementary data)). all twenty-one interviewed patients (table 2) were born and raised in the netherlands. sixteen interviews were held in the presence of the patients’ partners or other relatives who actively took part in the conversation. all interviews took place at the patients’ home and lasted between 45 and 120 minutes. no evidently new information regarding our main research question emerged after sixteen interviews and after an additional five interviews thematic saturation was reached. no patient or relative voiced any concern about the study during or after participation.
The first appointment
sixteen patients stated that the definite diagnosis ‘als’ or ‘PMa’ was disclosed in the als centre, the other nine patients came for the confirmation of the diagnosis. Most patients reported to have consulted several physicians before they finally got the diagnosis MnD. all of them explained that the unambiguous message to be suffering from a deadly disease was shocking, regardless of whether this was entirely unex-pected or the confirmation of a serious suspicion.
You’ve known it for a long time. it’s been getting worse and worse. But you hope they’re wrong and that the muscle specialist will come up with another explanation. Deep down you know it, but it’s still devastating news. (Patient 22)
all patients stated that they had wanted to hear the truth about their condition. nearly half of them explicitly mentioned that they felt relieved when the diagnosis was given or confirmed after a long period of uncertainty.
oddly enough, at first i was actually just glad to be told something – to know that there really was something going on. (P3)
in the other hospital, they’d been beating about the bush, but in the referral letter i read ‘suspected MnD’. When you google that term you are scared to death. […] But i wanted clarity… You need to know, otherwise you can’t go on and you’re continuously stuck between denial and despair. (P16)
reactions of patients observed during the first appointment varied. some of them wanted to know as much as possible about the disease, while others were more reserved. regardless of individual coping strategies, however, all patients asked the MnD expert how much time they had left and what could be done to stop or slow down disease progression. During the interviews many patients said that there had been little left to talk about with the specialist at the very moment they heard that their life expectancy was considerably limited.
When you’re told during your first appointment, ‘Madam, you’ve got als. there is no cure, and these are the consequences’,… at that stage, you don’t really need any more information than that. (P22)
the prospect of a short-term follow-up visit with the same MnD expert prior to meeting the rehabilitation physician was experienced as supportive. as one patient said, having a follow-up appointment at short notice gave him ‘something to hold on to’ Table 2. Characteristics of interviewees
als = amyotrophic lateral sclerosis; PMa = Progressive Muscular atrophy; als-Frs = als functional rating scale (maximum score: 48; a higher score represents better function retention); * moderate or severe dysarthria
in a situation where he ‘had just lost all track’.
in the 1990s, i’d visited the hiv outpatient clinic where i got the diagnosis, and a prescription. the doctor told me to come back in six months. Your world is falling apart, and you’re told that you can come back in six months! i was afraid the same would happen again, but they immediately drew up a whole treatment plan for me. i still remember how relieved i felt. (P11)
Patients did not reflect negatively on the sudden and full disclosure of their diagnosis and on all the information about the consequences of the diagnosis.
it was horrible to be told. Yes, it certainly was! But of course, she [MnD specialist] couldn’t make it sound any better than it was. i mean, what could she have told me, other than the truth? (P3)
she was quite straightforward, but she immediately took good care of us, too. ‘We’ll be doing this together,’ she said… (P21)
any deviations from the strategy of the aCn for disclosing the diagnosis MnD (table 1), as could occur when patients were told the diagnosis elsewhere, made patients perceive the bad news message as even more stressful.
this man [referring neurologist] was apparently struggling with the news himself. he didn’t know how to tell me […] Weirdly enough, i felt sad for him that he had to tell me! (P14) i was sitting there alone. half an hour later, i was back at the tube station, holding a red information folder with my death sentence, and, well, i was simply desperate. i should never have gone there on my own. they [the neurologists] should have told me that. (P18)
the neurologist asked whether i wanted another appointment with him. But he had just told me that he couldn’t do anything for me. i couldn’t see any reason to go back… (P2)
Patients generally did not forget the date of diagnosis. receiving the diagnosis was hard but unavoidable, and necessary in order to be able to start making plans for the future. Consequently, the prospect of support by a specialized team gave them some grip on their situation.
Time between appointments
Many patients remembered that they felt as if they had received a ‘sledgehammer blow’ or their ‘death sentence’ after the first appointment.
During the appointment, you just try to keep up appearances. But as soon as we were back in the car, it was terrible. i didn’t know which way to turn anymore, and my boyfriend lost his head as well… (P11)
Patients spent the days following the disclosure of the bad news differently, but they all reported positively on the ‘breathing space’ between the two appointments with
the MnD neurologist. as one patient said, the most important questions could not be answered by the medical team anyway.
My head was in total chaos. no one could help me at that point – not my wife, not my friends, and certainly no als team. You have questions that only you yourself can hopefully find the answers to – and that takes time. (P3)
the only thing i immediately knew for sure was that i would do anything to see my son grow up for as long as possible. (P13)
according to the vast majority of patients, sharing the news with close family members and friends was of great importance. their reactions, whether experienced as supportive or distressing, were said to be essential for their plans for the future with this incurable disease.
We came home and my friends immediately said, ‘of course you will stay at home. We’ll take care of you!’ (P3)
… My wife said, ‘But we won’t sell the boat. We’ve been sailing all our lives and that’s exactly what we’ll do again this year.’ (P19)
Patients often reported a strong desire to learn more about their disease. they used the time between appointments to obtain information at their own pace. While some said that they were satisfied with the information folders of the als Centre, others tried to increase their knowledge by surfing the net. Partners, children and friends often provided patients with articles, documentaries, and links to als websites via (social) media.
During the first few days i didn’t check anything on the internet. i was far too emotional for that. it took me about a week before i started digging deeper. (P14)
i immediately hid the information folder from the hospital in a closet: i didn’t want to have it lying on the table. My partner, on the other hand, immediately started googling… (P11)
all patients consulted their general practitioner (gP) between the two appointments with the specialist. in most cases the gP took the initiative for a consultation after having been informed by the MnD expert – usually within 24 hours of the bad news message (table 1). this consultation served to discuss wishes and options for support during the course of the illness and to talk about end-of-life decisions. thirteen of the 21 patients reported to have asked their gP directly to support their wish for euthanasia when life would become unbearable.
i made arrangements immediately, together with my children and our family doctor. i signed a statement for euthanasia straightaway. i didn’t know how much time i’d have. those last few weeks of life – i can do without them, if i’m not going to get better. (P6)
Patients differed in their assessment of the duration of the interval between appoint-ments. While most of them were satisfied with the two-week interval, some patients felt that several days would have been enough. others felt that several weeks would have been more appropriate to come to terms with their altered situation. Patients were unanimous about the intrinsic value of the ‘breathing space’. it gave them time to accommodate to their dramatically changed future in their own way, before involving a medical care team.
The second appointment
all observed and interviewed MnD patients confirmed that the second appointment had been with the same neurologist they had spoken to during the first appointment. upon meeting each other for the second time, one very often got down to the heart of the matter immediately.
the neurologist starts the conversation by asking how the patient has been doing since the diagnosis. ‘it’s intense when you are completely preoccupied with it, but otherwise it’s been oK.’ ‘so you’re able to get it out of your mind from time to time?’ ‘sure.’ ‘i can see you’re all very well-prepared.’ the patient and his daughter hold notes in their hands, his wife and son have notepads. the neurologist looks around encouragingly. ‘should we start with your questions [to the patient]?’ ‘Yes. i have a few questions, just out of curiosity. how will i die? that’s what actually keeps me occupied.’ (observation with neurologist 1, Patient 2)
Patients mentioned various reasons why they felt the second appointment with the MnD neurologist was helpful. one important reason was the opportunity to discuss facts about the disease and its course with a specialist, who is obviously better informed than a gP.
they [the neurologists] can’t do anything about it, but you want to discuss your questions about the disease and about research with the most specialised doctor… (P16)
People have so many questions, and they look everything up on the internet. and information you can find on the internet is not always easy to understand… (P14)
another important element of the second appointment was the correction of misunderstandings and misconceptions that had emerged in the time between appointments, e.g. about the course of the disease or the involvement of organs like the heart or bladder.
i thought, ‘all muscles will eventually fail. suppose i have a heart attack – that might spare me a lot of misery.’ But when we talked things over with the neurologist, it turned out that the heart muscle was not involved in the disease. My husband was greatly relieved, but i – i don’t know… anyhow, it was clear… (P22)
suddenly realised that my biggest fear, to wake up one morning with completely paralysed legs, was unfounded. (P16)
several patients, especially the ones who had been treated for chronic conditions before, felt relieved to be listened to when they expressed their anxieties about a future with progressive disability. Being able to express their own wishes and expectations about future care was regularly reported as ‘making you feel less unsettled’.
the neurologist immediately said that the team would arrange for me to stay at home if that was my wish. he had no preconceived plan of his own, and he did not think my questions were ridiculous… (P12)
the neurologist said that he found it difficult at this stage [during the second appointment] to write down that i didn’t want any respiratory support. ‘these are quite com-plex decisions that we have to keep on discussing…’ (P25)
For most patients it was also important to be informed about the latest research news by the MnD neurologist. the option to participate in a clinical trial was considered a valuable – altruistic – contribution, and there were a lot of questions concerning alternative treatments, such as neural stem cell therapy which is highly recommended on the internet. some patients were disappointed or even angry about the fact that participation in clinical trials had not been offered directly after the diagnosis as they felt they had lost precious time in which they might have benefitted from new treatments ‘before it was too late’.
the most horrible thing is to do nothing and just wait for it to get worse. Maybe the situation can at least be kept stable. i always say ‘it doesn’t hurt to try’. (P1)
i wanted to know what he [neurologist] thought about stem cell therapy. it seemed a specta-cular option to be cured, but he had not mentioned that option during our first appointment. Why? (P20)
Patients reported that the direct referral to the multidisciplinary als team was helpful. even patients who expressed having difficulties adapting to the sudden reality of a severe illness said that discussing the consequences of the disease and outlining a care plan with their own multidisciplinary team had reduced their anxiety of being left alone. two patients preferred to postpone the support by the multidisciplinary team, although they did not experience the consultation with the rehabilitation physician as unnecessary or unsettling.
the other neurologist [from another hospital] had told me that there was nothing to be done about it, but then our general practitioner arranged an appointment in the als centre… You know what’s important? that they don’t let you down, even though they can’t cure you… (P6)
i didn’t want to have anything to do with the team. i thought, ‘i’ll see what happens…’ But i did appreciate these first appointments, just to know whom you could contact if you needed help. (P15)
all patients stated that the second appointment, followed by the introduction to the multidisciplinary team, led to fruitful discussions about their future medical care. MnD patients suffering from other chronic diseases and particularly MnD patients who did not want too much professional intervention made it clear that they experienced the fact that care was offered, but not forced upon them, as ‘being granted the kind of dignity befitting their values and personality’.
Discussion
the straightforward and full disclosure to be suffering from an incurable and fatal disease was considered unavoidable and justified by the patients in this study. the prospect of a short-term second appointment offered structure. the time between appointments provided the opportunity for a first reorientation. in line with that the second appointment could be used for detailed discussions of various aspects of the diagnosis and its consequences and to develop a plan for the supportive treatment trajectory.
the appropriateness of the direct disclosure to be suffering from a life-limiting disease has been described before for both patients with MnD and patients with cancer.1,9,10,14
our data confirm that a reduction in the delay between symptom onset and diagnosis is key for patients with MnD.24 Despite the introduction of a fast-track diagnostic
process in tertiary referral centres, which has shortened the time between referral and diagnosis, the overall delay still amounts to 10 to 12 months.34,35 in this context, the
patients’ feeling of relief when they are finally informed about the cause of their progressive disability is understandable.
We found that patients appreciated the short-term follow-up visit with the same specialist. several interviewees even said that the plain truth they had been told during the first appointment was bearable only with the prospect of a second appointment at short notice. as shown in another empirical study on communicating the diagnosis MnD in a single appointment, the directness in breaking the news, even when wished for, can be experienced negatively.24 During both the observations and the interviews
patients in our study expressed that they considered the short-term follow-up visit with their specialist as an indication that they were not abandoned. the experiences of several of them prior to their referral to the als centre underline this; they felt isolated when the first consultant they had met elsewhere had told them that he could offer no treatment, or they felt confused when the decision whether to have a follow-up appointment with the specialist was left to themselves.
research in oncology has demonstrated that directly after receiving the diagnosis of a life-threatening disease, most patients are not capable of taking in much more information – let alone of making decisions about future treatment options.11,36-39 our
empirical study shows, as experienced and postulated by MnD experts for quite some time, that this phenomenon holds true for MnD patients as well.14,40 our interviewees
stated that they could hardly recall any concrete content of the first appointment after the diagnosis had been given. in line with that they uttered their appreciation for the time between the two appointments as an opportunity to catch their breath, to start adapting to the dramatically changed life prospects and to seek information at their own pace. of course, one to two weeks are not sufficient to fully adjust to a dramatic life-changing event.36,41 however, our study shows that patients had had time to come
to terms with the situation between appointments and had drawn up their own agendas for the second appointment with the MnD expert.
it is known that the opportunity to make concrete plans for the (near) future and to further substantiate these with the medical expert reduces anxiety and uncertainty in patients with MnD and other life-threatening diseases.9,26 the short-term second
appointment also proves to be important for recapitulating information about the disease and invalidating misunderstandings and misconceptions, such as the fear of sudden respiratory arrest during the night or involvement of the heart muscle. By the time of the second appointment, the vast majority of patients had searched the internet or had been informed by friends and relatives, for example about new life-prolonging or even life-saving therapeutic options (usually unsubstantiated complementary treatments).10 Checking the reliability of this information in a discussion with the expert
appeared to be reassuring for patients. the fact that patients make more confident treatment choices when they have been informed in more detail about treatment options and when their preferences have been evaluated further stresses the added value of a short-term second appointment.42
Strengths and limitations
our findings may be considered valid, since the experiences and opinions of partici-pants of different ages, sex, and with different illness trajectories were very similar. as only one of the patients declined participation in the study, a significant selection bias is unlikely. our data is rich, given the number of interviewed patients and the complementary data from non-participating observations. the findings are in agreement with other studies on MnD patients, cancer or other chronic progressive diseases which demonstrate the appreciation of the full disclosure of bad news, even if it involves information about a reduced life expectancy.43-45 these studies also show
that patients who suffer from incurable progressive diseases value participation in the decision making about treatment and care.10,21,26,46-49
the facts that interviewer and interviewees were acquainted with each other by the time the interview took place and that patients’ partners or other next of kin often attended the interview may have encouraged patients to openly and critically talk about their experiences.
nevertheless, our study also has limitations. it took place in a single tertiary referral centre located in the netherlands, where end-of-life considerations, including eutha-nasia, are openly discussed, and it included only patients who had been born and raised in this country. Furthermore, acquaintance with the interviewer and presence of patients’ partners or other next of kin during the interview might have elicited socially desirable answers or led to concealment of important information.
as far as we know, there are no empirical data on a two-tiered approach to break bad news as described and evaluated in this study, neither for MnD patients nor for patients with other life-threatening diseases. our study provides concrete results about the added value of this approach. and it has led to an even firmer compliance with the outpatient clinic’s protocol (table 1) as our evaluation made clear that people suspected from an MnD must be encouraged to come to the consultation with a significant other and that the second appointment with the same MnD specialist must not be presented as an informal offer, but as a necessary follow-up.
Conclusion
our study examined how Dutch MnD patients experience a two-tiered appointment approach for breaking bad news. the results show that this approach fits well with the way in which patients process the bad news message, gather information and handle their changed life perspective. in turn, the treating specialists are recommended to respond to the patients’ ability to cope with the devastating news by following a clear protocol and by offering a second consultation at short notice.
\\
Acknowledgements
the authors would like to thank the participating patients for their time and frankness in the interviews.
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