Suicidality in Huntington’s Disease: A Qualitative Study on Coping Styles and Support Strategies

Abstract

Background: Huntington’s disease (HD) mutation carriers are at increased risk of suicidal ideation, suicide attempts, and completed suicide. However, research is lacking on coping strategies and treatment options that can be offered to suicidal HD mutation carriers.

Objective: This study explores how individuals with pre-motor or motor symptomatic HD cope with suicidality, how their partners support them, and their ideas and wishes regarding how relatives and healthcare professionals can help them in coping with suicidality.

Methods: This qualitative study included 11 HD mutation carriers who experienced suicidal ideation or attempted suicide and 3 of their partners. They participated in a focus group discussion or an individual in-depth interview. Two independent researchers fragmented the transcribed interviews, coded these fragments, grouped them under themes, and structured the data.

Results: HD study participants used four main strategies to cope with suicidality, including talking about suicidality, employing self-management activities, using medication, and discussing end-of-life wishes. Partners, relatives, and healthcare professionals can support suicidal HD mutation carriers in each of those four strategies.

Conclusions: Despite the absence of a turnkey solution for suicidality in HD, healthcare professionals can play an important role in supporting suicidal HD mutation carriers by providing an opportunity to talk about suicidality, providing psychoeducation on self-management, prescribing medication, and discussing end-of-life wishes. Future HD-specific intervention studies could investigate the effect of combining these treatment strategies into one holistic approach.

Keywords

Neurodegenerative diseases suicide psychopathology coping behaviour therapy qualitative research

INTRODUCTION

Huntington’s disease (HD) is an autosomal dominant progressive neurodegenerative disease [1]. An expanded CAG repeat on the short arm of chromosome 4 causes a mutant polyglutamin chain in the huntingtin protein [2]. The disease is characterized by motor dysfunction, cognitive decline, and neuropsychiatric symptoms. Although some of these symptoms can be treated, the disease cannot be cured [3].

An important psychiatric sign of HD is the increased risk of suicide, as first described by George Huntington in 1872 [4]. More recent studies showed that HD patients were 2–8 times more likely to die by suicide than individuals from the general population [5 –8], which accounts for up to 11.4% of all deaths in motor symptomatic HD patients [5 –24]. In addition, lifetime suicide attempts have been reported by 3.2–17.7% [6 , 24–31] of the individuals with pre-motor or motor symptomatic HD, compared with only 2.7% in the general population [32]. Furthermore, 8–34% of the individuals with pre-motor or motor symptomatic HD experienced some form of suicidal ideation in the month prior to the interview [26 , 33–36], compared with 0% of the first-degree non-carrier relatives [33]. These numbers emphasize the frequent occurrence of suicidal ideation and suicide attempts (together referred to as ‘suicidality’) in HD. Suicidality is associated with lower quality of life [37, 38] and an increased occurrence of completed suicide [39].

Both suicidal ideation and completed suicide occur most frequently in early-to-middle disease stages [5 , 35]. In addition, depression is reported to be the strongest association of suicidality in HD [28–31 , 40]. Individuals with HD who had other psychiatric symptoms like irritability/aggression [28, 31], obsessive and compulsive symptoms [25], and a previous suicide attempt [28, 40] as well as those who used antidepressants [33] or benzodiazepines [28], were also more likely to experience or develop suicidality.

Despite the high frequency of suicidality in HD, limited information is available about coping with or the treatment of suicidality in HD, comprising only a few case descriptions of suicidal HD patients who were pharmacologically treated [41 –43]. General guidelines on treatment of suicidality are available, but it is unknown whether the treatment of suicidal HD patients should be different, given the in advance known devastating course of the disease.

Therefore, we conducted a qualitative study among HD mutation carriers who had experienced suicidal ideation or attempted suicide and among their partners. The primary aim was to examine how these individuals cope with suicidality and their ideas and wishes regarding how relatives and healthcare professionals can help them cope with suicidality. An additional aim was to explore how the spouses of HD mutation carriers support their partners with regard to suicidality.

MATERIALS AND METHODS

Recruitment procedure and participants

This qualitative study was conducted among HD mutation carriers recruited between February and May 2014 and who had previously participated in a Dutch prospective cohort study on behavioral problems and psychiatric disorders in HD [44].

Of all mutation carriers who 1) had participated in the last follow-up measurement of the aforementioned cohort study, 2) had given consent to be contacted for future research, and 3) were not known to have died (n = 90), 40 had reported the presence of thoughts of death, suicidal ideation, or a previous suicide attempt at one of the measurements of the cohort study. Of these 40 HD mutation carriers, 11 consented to participate in the present qualitative study (Fig. 1). If possible, participation in a focus group discussion was the preferred study method [45, 46], as the group processes help participants to explore and clarify their opinions and feelings [46]. Furthermore, as suicidality is a sensitive topic, participants may feel more relaxed to share their feelings about this topic when they see that others have similar feelings [45]. Eight mutation carriers participated in a focus group discussion (divided over 2 groups of 4 participants each) and 3 in an individual in-depth interview.

The focus group discussions were analyzed first, followed by the individual interviews. Data saturation was reached after these focus group discussions, as no new understandings/information on support strategies emerged from the individual interviews [46].

All participants were asked whether they had a partner and, if so, whether he/she knew about their suicidal thoughts. After consent, we contacted their partner for participation in a focus group discussion. Of the 4 partners of participants that we could contact, 3 were willing to participate.

This study was approved by the Medical Ethical Committee of the Leiden University Medical Center (LUMC) and was conducted according to their ethical standards. All participants provided written informed consent. Potential participants who did not have the verbal or cognitive capacities to provide informed consent or to participate in a focus group discussion or individual interview could not participate in the current study.

Focus group/individual interview for HD participants

Before the focus group discussion/individual interview, each HD participant had an individual intake appointment to explain: the purpose of the study, the topic list, and the course of the focus group discussion/individual interview. In addition, the presence and severity of depressed mood and suicidality in the last month were assessed by the items ‘depressed mood’ and ‘suicidal ideation’ of the Problem Behaviors Assessment (PBA) [26]. Also, lifetime suicidality was assessed by the Columbia-Suicide Severity Rating Scale (C-SSRS) [47].

The actual focus group discussions/individual interviews were conducted by a psychiatrist (moderator/interviewer) with longstanding experience in the study and treatment of HD mutation carriers (EvD). This researcher was specifically trained in moderating focus groups for the present study. He was also the treating psychiatrist of one of the participants of the focus group discussion. The first author (AAMH), who is experienced in interviewing HD mutation carriers, conducted the intake appointment and was present as note taker during the focus groups and individual interviews. The focus groups lasted 1.5–2 h (including a short break), and the individual interviews lasted 15–60 min each. During the focus groups and individual interviews, we mainly referred to ‘thoughts of death’, and explained that this term could cover the whole spectrum of suicidal thoughts: ranging from not wanting to live anymore should the disease become more severe, to actual suicide attempts. The moderator/interviewer ensured that the whole range of thoughts of death was discussed. To introduce this sensitive topic, the focus group discussion/individual interview started with an open question about how participants felt about this study actually being conducted, as was specified in the interview guide. Subsequent questions were based on what the participants said themselves. A topic list, also specified in the interview guide, was used as a reminder for the moderator/interviewer and consisted of 3 main items: how the mutation carriers themselves coped with suicidality; how relatives could support suicidal mutation carriers; and how healthcare professionals could help suicidal mutation carriers. If one of these topics was not addressed during the discussion by the participants themselves, some open questions were specified in the interview guide which could be used by the moderator/interviewer to start discussion. For example: ‘If you were a relative (or a healthcare professional/neurologist/psychiatrist/general practitioner) how would you help an individual with HD who expresses suicidal ideation?’

Focus group for partners

The focus group discussion for partners lasted±80 min (including a short break). As specified in the interview guide, the session started with an open question regarding how participants felt about this study being conducted and subsequent questions depended on the participants’ responses. The topic list, also specified in the interview guide, was based on results of the mutation carrier focus groups and individual interviews, and covered the following items: discussion of suicidal thoughts with partner; support of suicidal partner; and advice for future partners of suicidal HD mutation carriers. Again, example questions to initiate discussion on one of these topics were provided in the interview guide and used when necessary.

Data analysis

All focus group discussions and individual interviews were recorded on tape and transcribed ad verbatim. The data analysis included fragmenting, open coding, axial coding, and structuring of the data, based on a grounded theory approach according to Strauss and Corbin [48]. Two researchers (AAMH: medical PhD student and AH: physician) independently fragmented the transcripts of the HD mutation carriers in smaller parts consisting of one or multiple sentences which addressed the same topic. Thereafter, open coding of the fragments by the two researchers was conducted. The initial code list included only 3 codes: ‘coping themselves’, ‘support from relatives’, and ‘help from healthcare professionals’, as these were the items on the topic list, but many new codes were created during the open coding process. Most fragments got multiple codes: a general code that described the overall topic of that fragment (like: ‘help from healthcare professionals’) but also codes that described for example the particular strategy used (like: ‘medication use’), or codes that described influencing factors (like: ‘efficacy’ and ‘side effects’). While coding interviews, fragments were compared to other fragments, also from earlier transcripts, to see whether they should receive the same code. After coding of each transcript, the two independent researchers discussed their fragments and codes until consensus was reached. They also grouped codes that were closely related and ordered them under certain themes. For example, the codes ‘listening’ and ‘trust’ were grouped under the theme ‘factors influencing talking’, which was grouped at a higher level under ‘talking about suicidality’. All of these codes and themes emerged from the data and were not pre-specified. Having discussed all the transcripts of the HD participants, themes and codes were displayed in a code tree to get insight in the relationship between the different themes and codes. In the initial code tree, the codes that were related to coping styles and support strategies for suicidality in HD were ordered within the 3 pre-specified general codes derived from the initial code list (‘coping themselves’, ‘support from others’, and ‘help from healthcare professionals’). Several themes, like ‘talking about suicidality’ and ‘end-of-life wishes’ were actually grouped under all three pre-specified general codes. Both researchers independently recognized that all relevant codes and themes could better be grouped differently into four main categories which described all coping and support strategies that were used by the HD participants. Subsequently, both researchers independently read and interpreted all fragments that belonged to these four categories. They explored the experience with and the outcomes of the different coping styles and wanted to explain why some participants had positive experiences with that particular coping style, while others had negative experiences. They analyzed conditions that should be present before a coping style or support strategy was considered helpful, and looked at factors that influenced the outcome of that particular coping style or support strategy. Thereafter, the partner focus group transcripts were analyzed. The transcripts were coded and fragments were grouped according to the four main categories that were identified from the HD participant transcripts. Again, all fragments that belonged to these four categories were closely read and interpreted. We chose not to search for new themes within the transcripts of the partner focus group as our aim was to explore the coping styles and support categories used by the HD participants. We were specifically interested whether the HD mutation carriers’ ideas and on how partners could help were also considered helpful and applied in daily life by their partners. The data was stored and managed in Atlas.ti 7.5 (Scientific Software Development GmbH, Berlin, Germany).

RESULTS

Study population

The disease stage of the participants ranged from self-reported pre-motor symptomatic to participants who had to live in a nursing home. There were three male HD participants and one male partner, the age of the participants ranged from 32–71 years, and the number of CAG repeats ranged from 41–47. Table 1 presents the scores on the ‘depressed mood’ and ‘suicidality’ items of the PBA.

Support strategies for suicidality in HD

First of all, participants indicated that there is no patent solution for suicidality in HD. They mentioned several ways of support that can be grouped in the following categories: 1) talking about suicidality, 2) self-management, 3) medication use, and 4) discussing end-of-life wishes.

1. Talking about suicidality

All participants stressed that they had a great need to talk about their suicidality, even though this was considered difficult and the threshold to do so was high. This is illustrated by one of the participants who said: “Someone who thinks about death already has difficulty expressing this... and has feelings of self-disapproval because of this thought [ ... .] But my experience is that it really helps when you eventually do talk about everything - with all your sorrows and misery. This always gives relief.”

Table 2 shows factors that, according to the participants, could facilitate or complicate talking about suicidality. Listening, taking the thoughts seriously and not trivializing these thoughts were the most important factors in helping them to express their feelings to a relative or healthcare professional. The preferred person to talk to varied per participant and included their partner, a family member, friend, care manager, psychologist, or hypnotherapist. Some participants stressed that this person should not be too close, and they preferred someone who is “a relative outsider”, because they did not want to hurt their partner or children by telling them about their suicidal thoughts. However, the spouses mentioned that they did want their partner to tell them about suicidal ideation, as was stated by a female spouse: “I don’t want him to think about that all on his own - I think that’s far too lonely”, although the spouses admitted that their partner’s expression of suicidal ideation was also a burden on their own lives. When the person to talk to was less close (either a professional or a non-professional), a trustful relationship needed to be established before the participants felt ‘close’ enough to talk about suicidal ideation, although the extent of this relationship differed per participant. Furthermore, some participants found it difficult when family members who experienced the same disease process did not want to talk about the disease at all, as illustrated by one participant: “You’d think you’d have a good relationship with someone [ ... ] somebody who lives the same sort of life as you, but who’s really very different.”

Participants stated that positive experiences with talking to someone about suicidal thoughts resulted in a decrease of suicidal ideation, and in relief, improved self-esteem, and increased ability to see the positive things in life. A few participants who read up on the disease since the moment they became aware of their genetic status, recognized that they had now learned to think more positively when they felt depressed for a longer period, or when someone pointed out the good things in life. However, most other participants felt that the ‘helping person’ ignored their suicidal thoughts when they simply responded by pointing out the positive things in life. This difficulty experienced by persons trying to give support is illustrated by one of the partners who said: “If you even make a small reference to something positive [ ... ] this works in a counterproductive way [ ... .] what you’re really trying to do is cheer the other person up” and sometimes it is better “not go into it” but “just be there [for your partner]”. Also, the HD participants themselves mentioned that non-verbal signs of support, like someone putting an arm around you, can be very helpful. Negative experiences with talking about suicidal ideation resulted, for example, in withdrawing from the contact “which made me feel lonely and yes, that made everything worse.” Others mentioned that they had a negative experience with too muchtalking about suicidality to too many different healthcare professionals. One participant attributed this to the fact that she had not yet learned “to let go”.

Given the importance of talking about suicidality, the moderator specifically asked participants whether others should actively address the presence of suicidal thoughts. They pointed out that it is best to ask about their mood when others see that they are sad or doing less well; also, when an individual with HD indicates a depressed mood, questions about suicidality could be asked (Table 2). Partners acknowledged that it is difficult to find the right moment to talk, e.g. due to a busy daily life, and because they did not want to introduce this painful topic when their partner was feeling well. Given the emotional burden for both HD mutation carriers and partners, one of the partners advised other partners to try and talk about the suicidality during multiple conversations, as aperson can only handle a certain amount each time. One participant could talk with her partner about suicidality very well because he “allowed me to talk about it and he took the time to discuss it.” Talking about suicidality was also difficult for the partners themselves, particularly when they had to decide on the amount of control they wished to exercise in order to prevent suicide. Partners themselves also needed to talk about suicidality with others, which one partner did by joining a peer support group, whereas another partner had negative experiences with such a group because of all the unpleasant stories she heard about what was still to come.

Most participants agreed that healthcare professionals could (or even should) ask about suicidal thoughts, even though they might feel “taken by surprise”. This topic should be introduced delicately by asking about sad feelings and, if present, follow-up questions should be asked (Table 2). In case of suicidal thoughts, healthcare professionals should guarantee further possibilities to talk about suicidality, e.g. by referral to a psychologist. Both HD participants and partner participants emphasized that healthcare professionals should have adequate knowledge on HD and “be familiar with the whole picture”. A combination of individual therapy for the individual with HD and therapy sessions that partners could also attend was preferred by the participating spouses.

2. Self-management

Participants mentioned many different activities that they used to cope with suicidality and with HD itself. These activities could result in increased self-esteem and decreased stress and thoughts of death. Most of these activities could be grouped as either 1) actively facing suicidality/HD (e.g. talking about suicidal ideation/HD, gaining information on HD, realizing that you are not the only one, contacting an HD support center, making a crisis plan, setting future goals, healthy living, and hypnotherapy), or 2) taking their mind off it, mainly by exercise and relaxation (e.g. yoga, meditation, going outside, cycling, walking, distraction, focusing on daily life, social contacts, family life, taking rest, and creative therapy). “The art” of “balancing” these elements was considered important, since activities on both sides of this spectrum could become too extreme (Fig. 2) and then worsen their feelings. Reducing suicidal thoughts and getting back in balance after too much confrontation with HD or thoughts of death (e.g. in family, by rumination, listening to sad music or watching films or TV shows about death) could be achieved by taking medication, calling someone, taking rest, letting go, taking distance, focusing on yourself, or having someone who can take you out. At the same time, for some participants, negative experiences were induced by too much avoidance and not wanting to face the disease which resulted in destructive behavior like taking drugs and alcohol, aggression, or isolating oneself. At that point, participants said they need people who can “pull you out of the mud, so to speak”. One of the participants gave the following advice: “Don’t stay at home worrying ... It is a cliché, but it actually does help: go out for a walk with someone, or go cycling! Or ... I don’t know ... if you stay at home in front of the TV all day, or behind the computer ... if you isolate yourself, in my experience, it’ll only get worse and worse.”

It was advised to employ such self-management strategies and create an environment of people you can talk to before the disease starts, as it is more difficult to do this when the disease becomes more severe or when someone is depressed. Coping styles witnessed in their own family also influenced how participants dealt with the disease themselves. Several participants criticized the coping styles of their family members, e.g. when they refused to talk about it and/or avoided everything that had to do with HD.

3. Medication use

Almost all participants who used medication (mostly antidepressants) were positive about its effects on their mood and suicidal thoughts. Only one woman reported that she stopped using antidepressants because she always felt unwell after taking these pills. In her opinion the pills suppressed her feelings, and she emphasized that the “the instruction leaflet already states [ ... ] suicidal tendencies.”

The reluctance of some participants to start taking medication was recognized by the participants who were currently using medication from the period before they started. Reasons for this reluctance were: potential side-effects and being unable to separate these from disease symptoms, fear of emotional suppression, and “wanting to do it on your own strength”. Some participants needed time and discussion with relatives to decide about starting medication. Participants who used medication for a longer period of time mentioned that they did not want to quit, because of its effectiveness.

Many participants who used medication combined this with talking to someone: the importance of this was stressed by one participant who described an appointment with a neurologist as follows: “when I mention I feel depressed, [the neurologist says]: ‘Well –here’s the medication’, whereas in fact he could say something like: ‘Take this medication, and try and see a psychologist in a couple of days.’ People might not always want to do this, but offering this, yes that can help some people [ ... .] I think it often helps to literally say these things out loud to a psychologist or psychiatrist.” Partners recognized the positive effects of medication for various HD symptoms, and one partner stated “without all of that [HD medication] I couldn’t have coped for so long.”

4. Discussing end-of-life wishes

Some participants had a wish and/or advance directive for euthanasia in case the disease became very severe: in the Netherlands, under strictly controlled conditions, this is legal. The prospect of such a way out could help in coping with suicidal thoughts, as explained by one of the participants: “I kind of made my own crisis plan: what to do if I think I want to jump in front of a train. Because ... I don’t want to end up that way. So I made arrangements about this [euthanasia directive] and that helps.” Most participants had seen examples of HD in their family and clearly knew what they did not want to experience (such as admission to a nursing home), but some also mentioned that their views might change. Also, some participants in an early stage of the disease did not know whether or not they would want euthanasia, or had not yet thought about this. Some found it difficult to decide at what point you need to think about end-of-life wishes: this was illustrated by one participant who was recently clinically diagnosed and still fully occupied with accepting the functional decline he experienced: “When should I start thinking about that [euthanasia directive]? [ ... .]I don’t want to face it yet, because I feel reasonably OK [ ... ] but there’s a point when you’ll go further downhill, and then ... then you can’t properly think about it.” These participants did not want the doctor to actively address euthanasia, or they considered it a topic that should first be discussed with their partner. However, another participant, who had thought about euthanasia since her genetic diagnosis, would like the doctor to address this. Also, one partner thought it was normal that the doctor had asked about euthanasia when his wife was admitted to a nursing home, since they had discussed this together since the beginning of the disease.

Those who expressed a wish for euthanasia emphasized the importance of this being their own choice. One participant described both her positive and negative experiences with healthcare professionals as follows: “I was always used to the fact that healthcare professionals [ ... ] are focused on making sure you won’t end your life [ ... ], but at the moment it does help me that I can just be very honest about it and yes ... that people understand. That maybe I don’t want to continue living at some point in time ... and, yes that this is my own choice and not ... not someone else’s.” The fact that the doctor might not want to carry out their request at the time they have reached this point, which some participants had witnessed in their close surroundings, worried them as “you have no guarantee”. Also, one partner expressed such doubts, but acknowledged she had no knowledge about the legal requirements for euthanasia. Once a participant had the confidence that his/her wish would be carried out, this provided some relief. Positive euthanasia experiences in the family strengthened this confidence, and further thoughts about end-of-life wishes were also influenced by family experiences. One participant realized that in an advanced disease stage he might not understand his situation and he wondered “So who is euthanasia most important for? For me?... Or for my wife and children?”

DISCUSSION

This qualitative study shows that HD mutation carriers use various strategies to cope with suicidality including talking about suicidality, employing self-management activities, using medication, and discussing end-of-life wishes. Relatives and healthcare professionals can support suicidal HD mutation carriers in each of those four strategies. Finding a delicate balance between different coping styles was an important central concept in all of these four strategies.

Coping styles and support strategies

Our study participants emphasized the urgent need to talk about their suicidality, either with a healthcare professional or a relative. However, professionals might be less likely to consider treatment and avoid further conversations about suicidality in HD when they feel they cannot help or consider suicidal ideation more normal or rational in a particular group of patients, as was previously shown for older non-HD populations with a death wish or suicidal ideation [49 –51]. Several participants mentioned the importance of involving specialist HD expertise as was previously also considered very important in coping with HD in general [52, 53]. With regard to timing, another important central concept in most of the mentioned strategies, participants stressed that questions about suicidality should be asked in case of a depressed mood, which is the most important association and predictor of suicidality in HD [28–31 , 40]. Similar to non-HD populations, the majority of patients do not easily disclose their suicidal thoughts to healthcare professionals [54, 55] and general practitioners only asked questions about suicide in 36% of patients with depressive symptoms [56]. Furthermore, both general practitioners and neurologists thought that less than 50% of the patients with suicidal ideation needed psychiatric assessment [57]. It was previously suggested that suicidal persons communicate their thoughts more often to relatives than to healthcare professionals [58]. The participating spouses stressed they wanted their partners to tell them about suicidal thoughts and wanted to be involved in the treatment, although participants themselves were sometimes afraid to hurt their partners’ feelings. Educating and involving caregivers in treatment for non-HD suicidal patients was shown to result in improved caring ability [59] and a greater improvement on suicidality, depression, and hopelessness measures [60]. Within HD, involving partners in treatment of suicidal HD patients might help to determine the best moments and ways to discuss suicidality for this individual and his/her partner and to deal with the ambivalence in ‘protecting’ the other.

Several of the self-management strategies mentioned by the participants (e.g. relaxation and exercise) have been shown effective in reducing suicidal ideation [61 –63] and/or depressive symptoms [64 –68] in non-HD populations, including patients with a chronic illness [66]. Within HD, positive effects of relaxation [69, 70] and exercise [71 –74] on several clinical outcomes have been reported, including on depression [74].

Case reports on treatment for suicidality in HD have only focused on pharmacotherapy [41 –43]. Positive effects were highlighted by our study participants who used medication, although others were hesitant to start and should be given time to think about this and room to discuss this with relatives. In non-HD populations, antidepressants (most frequently mentioned by our participants) have been shown effective in reducing suicidal ideation in patients with major depressive disorder [75]. Within HD, one case report showed that mirtazapine could reduce suicidal ideation [41] and a HD case series demonstrated the effectiveness of lithium in reducing suicidality [42]. Lithium was not specifically mentioned by our study participants, but in both non-HD unipolar and bipolar patients treatment with lithium resulted in a reduced suicide risk [76]. Pharmacotherapy for depression is recommended in guidelines for non-HD suicidal patients [55, 77], whereas in HD undertreatment of depression is likely [78] and there is insufficient evidence to guide treatment choice [79].

Finally, discussing end-of-life wishes helped some participants to cope with suicidality; however, this is not one of the treatment strategies recommended in general non-HD guidelines on suicidality and, moreover, is illegal in many countries. It has been estimated that 7% of the affected HD patients in the Netherlands die by euthanasia or physician-assisted suicide [80]. In non-HD populations, euthanasia is often considered to be inconceivable for psychiatric patients with a death wish [81]. It was previously recommended that HD physicians address end-of-life wishes [82], even though they often do not initiate this conversation [83]. In the present study, not all study participants wanted their physician to actively address euthanasia. Timing is crucial, as HD participants do have thoughts about their future [82 , 85] which are mainly based on experiences in their family [84] and are usually discussed first with the family [82]. It was beyond the scope of the present study to disentangle the best moment to address these thoughts. Although not specifically mentioned by participants in our study, a letter of intent [83], repeated conversations [82, 86], a trusting relationship [82] and informing patients about legal criteria/requirements [82] might result in more peace of mind for some of our participants with a wish for euthanasia.

Finding the balance between contradicting coping styles was an important central concept in all four different coping styles and support strategies for suicidality in this study. Previous studies showed that active coping, planning, seeking instrumental and emotional support, acceptance [87 –89] but also denial [88] are frequently used coping styles in HD. Too much active coping can prevent chronically ill people from experiencing life as ‘normal’. In this case, denial can become a positive coping strategy, but will have negative consequences when HD mutation carriers or partners fail to take action when necessary [89, 90]. The search for a balance between different coping styles was evident with regard to the self-management strategies, but also noteworthy with regard to the other strategies. For talking about suicidality, some participants had negative experiences with too much talking about HD/suicidality, which resulted in too much confrontation without being able to let go. They had to find a balance between being busy with suicidality/HD and focusing on other important things in life. Concerning medication use, multiple participants weighted wanting ‘to do it on their own strength’ versus the efficacy of medication on disease symptoms. With regard to discussing end-of-life wishes, there were opposing opinions between different participants. Some had active coping styles and had an advance directive for euthanasia as they wanted to have arranged everything, while others used more avoidant coping styles and did not talk or think about end-of-life wishes as they did not want to face the future yet. For some in this latter group it was also considered difficult to decide at what point they needed to start thinking about end-of-life wishes. These examples all stress the importance of a delicate balance within each support strategy with regard to the contradicting active versus avoidant coping styles to experience life as ‘normal’.

Strengths and limitations

This is the first study to investigate attitudes towards, and coping and support strategies for suicidality in HD, which included HD participants in various disease stages. Also, the use of focus group discussions facilitated the observation of differences of opinion between participants about the coping and support strategies that were proposed.

Some limitations need to be addressed. First, we included a small and selected group which limits the generalizability of the findings. Some participants mentioned that their family members had more avoidant ways of dealing with the disease, but did not want to participate in research in general. Also, males were underrepresented in the sample, while in general they might apply different coping styles than women [91]. Furthermore, individuals who had not expressed suicidal ideations in a previous study setting were not included in this study, while their coping styles are of interest as they might differ from those who do express suicidal ideation. Second, only three partners participated and their data were used only to determine whether they had similar or contrasting ideas about treatment strategies as the HD participants themselves. Data saturation on partner ideas about any of the themes could not be reached with only three partner participants. Third, the actual data analysis of the HD participant transcripts was done after all HD participant data was collected, while analysis and subsequent revision of the topic list during the study can support data saturation. Fourth, the moderator and researchers who analyzed the interviews had received medical education, which might have unintentionally focused their attention on strategies that are similar to those used in current clinical practice. Fifth, generalizability of the results to other countries might be limited, especially since (under specific circumstances) euthanasia is permissible in the Netherlands, whereas this is illegal in many other countries.

Clinical recommendations

Despite the absence of a patent solution for suicidality in HD, healthcare professionals can play an important role in all four strategies, which are largely in line with general non-HD suicide guidelines. All healthcare professionals who treat HD patients should ask about suicidal ideation, especially when symptoms of depression are present, and refer them to a (preferably HD-related) psychologist or psychiatrist for regular sessions to talk about suicidality. Active and non-judgmental listening, and taking their thoughts seriously, will strongly facilitate talking about suicidality. The application of the different self-management strategies might be enhanced with psychoeducation, which should also address the frequent occurrence of suicidality in HD so that patients realize that they are ‘not the only one’. If the physician considers prescribing medication, this should always be combined with talking about suicidality. Furthermore, both the effectiveness and side-effects have to be addressed, and time should be allowed for the patient to decide on starting medication. Significant others should also be involved in the treatment and (often) need to receive psychoeducation from healthcare professionals [55, 92], which must at least contain guidance on talking about suicidality and suggestions for support strategies for this person him or herself. If a patient has a wish for euthanasia, and euthanasia is permissible in their country, the healthcare professional should ensure repeated conversations about this topic and show a commitment of best intentions [82, 83].

Recommendations for further study

Future studies on the treatment of suicidality in HD should ideally investigate a combination of the proposed support strategies; this was shown to be effective in elderly populations [93, 94] in whom a program of psychoeducation, access to a care manager, behavioral activation, and pharmacotherapy or psychotherapy resulted in lower rates of suicidal ideation and improved quality of life compared with the treatment-as-usual group [94, 95]. Also, the perspective of and necessary support for partners should be explored in future studies, which should preferably also include partners of individuals with HD who use more avoidant coping styles. Furthermore, it should be investigated in what way involvement of a significant other in the experimental treatment can result in a greater decrease in suicidality for HD mutation carriers and less stress for caregivers. It is also interesting for further studies to explore whether ideas and wishes about coping styles and support strategies differ between patients with various diseasestages.

Conclusion

This study indicates that the best practice for suicidality in HD is talking about suicidality, self-management strategies, using medication, and discussing end-of-life wishes. Future HD-specific intervention studies should investigate a combined approach of these treatment strategies to establish clinical evidence and improve guidance for the treatment of suicidality in HD.

CONFLICT OF INTEREST

The authors have no conflict of interest to report.

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