Patients' beliefs about the causes of systemic lupus erythematosus

Rheumatology Advance Access published December 29, 2009

RHEUMATOLOGY

Original article

doi:10.1093/rheumatology/kep430

Patients’ beliefs about the causes of systemic lupus erythematosus Olivier Taıëb1, Olivier Bricou1,2, Thierry Baubet1, Vale´rie Gaboulaud1, Beátrice Gal1, Luc Mouthon3, Robin Dhote4, Loı¨c Guillevin3 and Marie Rose Moro1,5 Abstract Objective. Beliefs about the causes of SLE have rarely been investigated. The purpose of this study was to explore these beliefs. Methods. Face-to-face interviews were carried out with a total of 33 women with SLE, fulfilling the ACR criteria, with a median age of 40 (range 1565) years. Data were analysed using interpretative phenomenological analysis. Results. Women attributed SLE to many causes, some of them being not congruent with biomedical models. The most frequent beliefs about the causes of SLE related to autoimmunity, psychological and familial causes, heredity, magico-religious causes (especially in first- and second-generation migrants) and infectious causes. Autoimmunity was often seen as a self-destructive process. Conclusions. Being diagnosed with SLE prompted ‘Why me?’ and ‘Why now?’ questioning among these women, who attempted to reconstruct coherence in their life histories. For clinicians, analysis of the beliefs about the causes clarifies what is at stake for the patient. The objective is to allow patients to produce narrative to describe their chronic illness experience in order to facilitate a long-term treatment alliance. Further studies are required to understand relationships between beliefs about causes, psychological distress and SLE morbidity.

Introduction SLE is a systemic autoimmune condition responsible for a wide range of clinical manifestations. The disease course is chronic and unpredictable with the possible occurrence of flares leading to functional disability and psychological 1 Department of Child and Adolescent Psychiatry and General Psychiatry, Avicenne Hospital, AP-HP, Universite´ Paris 13, Bobigny, Inserm U 669, Paris, 2Department of Adolescent Psychiatry, Victor Dupouy Hospital, Argenteuil, 3Universite´ Paris Descartes, Faculte´ de Me´decine Paris Descartes, Cochin Hospital, AP-HP, Paris, 4 Department of Internal Medicine, Avicenne Hospital, AP-HP, Universite´ Paris 13, Bobigny and 5Department of Adolescent Psychiatry, Cochin Hospital, AP-HP, Universite´ Paris Descartes, Faculte´ de Me´decine Paris Descartes, Inserm U 669, Paris, France.

Submitted 3 August 2009; revised version accepted 18 November 2009. Correspondence to: Olivier Taıëb, Department of Child and Adolescent Psychiatry and General Psychiatry, Avicenne Hospital, AP-HP, Universite´ Paris 13, EA 3413, Inserm U 669, 125 rue de Stalingrad, 93000 Bobigny, France. E-mail: [email protected]

CLINICAL SCIENCE

Key words: Systemic lupus erythematosus, Beliefs about the causes, Illness narratives, Migration.

distress [1]. Psychosocial factors, such as stress, play an important role in SLE morbidity and are essential considerations in the management and care of patients [2, 3]. The chronicity of the illness and the disability resulting from this condition raise two fundamental questions for the sick person: Why me? and What can be done? Clinicians need to explore the significance of the answers to these questions among their patients [48]. Understanding of the experiences of illness is important in determining how patients perceive and cope with their situations. People’s beliefs about the causes of their chronic illnesses fit into a comprehensive process of ‘narrative reconstruction’ in ‘an attempt to reconstitute and repair ruptures between body, self and world by linking up and interpreting different aspects of biography in order to realign present, past and self with society’ [9]. Despite the interest in exploring the experience of illness from the point of view of those who are directly affected for the purpose of designing psychosocial

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Olivier Taıëb et al.

interventions, there is little published evidence concerning representations of illness among persons with SLE, and most of the existing studies focused only on the illness experience (see, for example, references [10, 11]) or on one of its dimensions, such as body image [12], parenting [13], with the exception of two studies conducted in the USA [14] and in Australia [15], respectively, which also considered beliefs about causes. The aim of the present study was to examine beliefs about the causes of SLE among SLE patients living in France.

Methods Patients Women with SLE fulfilling the ACR 1997 revised criteria [16] were recruited consecutively from the Department of Internal Medicine of Avicenne Hospital at Bobigny, a Paris suburban area. Women with neurolupus were excluded from the study. Participation in this study was proposed by the internist and study information sheets were provided to all 37 potential participants: 33 patients agreed to participate and four refused. This research received approval from the Ethical Review Committee ‘Comite´ de Protection des Personnes Ile-de-France III’, Paris, France. According to the Declaration of Helsinki, written informed consent was obtained from all the patients included in the study before interview. For minors, parental consent was also obtained. The median (S.D.) age of participants was 40 (12) (range 1565) years. Mean (S.D.) time since diagnosis of SLE was 7.8 (6.8) (range 026) years. Among the 33 women, 8 (24%) were first-generation migrants (i.e. foreign-born) [two of Caribbean origin from the French Antilles, two from North Africa (Algeria), three from sub-Saharan Africa (one from Mali, one from Congo and one from Guinea) and one from Turkey] and eight (24%) second-generation migrants [(i.e. born in France with at least one of their parents born in a foreign country) (three with parents born in Algeria, two with parents born in Morocco, one with her father born in Ivory Coast and her mother in Senegal, one with parents born in Poland, one with parents born in ex-Yougoslavia)]. Firstgeneration migrants from the French Antilles (which are administratively speaking part of France) were included among ‘migrant’ patients for the purpose of this study. Eighteen patients (54%) were living with a partner and among these, 14 were married. Twenty-two (67%) had at least one child. Concerning employment status, 7 (22%) were employed, 12 (37.5%) were not employed because of disability caused by SLE and 7 (22%) were students. Mean (S.D.) educational level was 10 (2.5) (range 817) years.

Interviews The present study used Interpretative Phenomenological Analysis (IPA), an established qualitative methodology used to explore in-depth how individuals perceive particular situations they are facing and how they are making sense of their personal and social world [17, 18].

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In chronic diseases, this method has been used in numerous studies (see for example references [12, 15, 19]). The approach is phenomenological, in that it involves detailed examination of the participant’s life experience and it is concerned with his or her personal perception or account of an object or event. The present study was conducted through a semistructured interview. This form of interviewing is the best way to collect data for IPA [18]: the investigators have a set of questions on an interview schedule, but at the same time, there is an endeavour to enter the psychological and social world of respondents. Respondents consequently share more closely in the direction the interview takes, and they can introduce an issue that investigators have not thought of. Respondents can be perceived as experiential experts on the subject and should be allowed the opportunity to tell their own story. This form of interview facilitates empathy, allows flexibility of coverage and discovery of novel areas, and it tends to produce richer data. Investigators are free to probe interesting areas that arise and they can follow respondents’ interests or concerns. But, of course, this form of interview reduces the control investigators have over the situation and involves longer completion time. The semi-structured interview schedule focused on patients’ beliefs about the causes and meaning of SLE. The explanatory model framework developed by Kleinman [5] helped to construct the schedule, which contained the following questions: ‘1-What do you call your problem? 2-Could you give me a brief history of your illness? 3-What do you think has caused your illness? 4-Why do you think it started when it did? 5-What does your illness do to you? How does it work? 6-How severe is it?’. Interviews took place in the Department of Psychiatry at Avicenne Hospital. All interviews were conducted by psychiatrists (O.T., O.B., T.B., B.G. and M.R.M.) and lasted 23 h. For non-French-speaking patients, an interpreter also participated in the interviews. Notes were taken directly after each interview to facilitate the interpretative process.

Analysis All interviews were audio-recorded with participants’ permission, they were transcribed verbatim and were analysed using IPA. The first stage of the analysis involved interviewers checking the transcripts for accuracy against tape recordings. Individual transcripts were repeatedly read and then coded to identify initial themes that were annotated in margins. This was close to being a free textual analysis. Each reading had the potential to bring up new insights. Recurrent themes were then identified across transcripts; these recurrent themes reflect a shared understanding of the phenomena in question among participants. This stage involved a more analytical ordering as researchers tried to make sense of the connections between themes. Some of the themes got clustered. This was a dynamic and cyclic process with each transcript leading both to the collection of further data and to their subsequent analysis. The aim was to recognize

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Beliefs about the causes of SLE

ways in which narratives from participants could be similar but also different. Researchers were in fact disciplined to discern returning patterns, but they also integrated new issues emerging so as to take account of convergences and divergences in the data. The last stage was to produce a coherently ordered table of the themes. To enhance validity, a single researcher (O.B.) carried out the main analysis and transcripts were also read by three other researchers (O.T., B.G. and T.B.) to improve consistency and coherence of analysis by ensuring that the themes identified were an accurate reflection of the data and that the analysis was not confined to one perspective. The results were discussed in repeated meetings with this research team. These discussions helped to highlight necessary clarifications or modifications of the themes identified. Another way of enhancing validity was also the care taken to distinguish clearly between what respondents said and researchers’ interpretations or accounts of it [18]. In the results below, extracts of respondents’ verbatim account have been chosen as exemplifying the underlying recurrent themes. In order to protect confidentiality, identifying information has been substituted in the quotes presented. The verbatim account has been freely translated into English, attempting to preserve the essential meaning and tone.

Results The frequencies of SLE causal attributions are reported in Table 1. For each patient, several causal attributions can be associated.

are seen as gradually destroying them and even ‘eating’ them: ‘With the anti-DNA antibodies, I realize now, my body has been destroying itself on its own’ (Respondent 1, aged 23 years); ‘They say that these diseases are illnesses you manufacture yourself. There are antibodies that become active and go places where they are not supposed to go. They choose something they’ve not gobbled up yet, but they can also come back to places they have already been (. . .) I don’t know if it has anything to do with it, but if you don’t like yourself, it can come. For a long time I was in a state where I couldn’t enjoy myself’ (Respondent 13, aged 53 years). One woman associated ANAs with nuclear, atomic and bomb: ‘Nuclear antibodies attack the good antibodies. That makes atomic antibodies, like an atomic bomb’ (Respondent 17, aged 47 years). Two others compared autoimmunity to gradual suicide: ‘It’s a disease you create for yourself, when deep down you want to die. Lupus doesn’t happen by accident, it’s part of a self-destroying process that has been ingrained in me’ (Respondent 3, aged 42 years); ‘It’s a self-suicide of the body, because I didn’t have the courage to do it myself. The body manufactures its own antibodies that destroy it. I’ve realized it’s self-destruction that comes from yourself, so I thought back over my life and everything I have put up with since I was 18’ (Respondent 7, aged 50 years). In all of these patients, autoimmunity appears to raise questions or to strengthen pre-existing questionings about psychological welfare and to contribute to generating psychological causal attributions.

Psychological and familial causes Autoimmunity and self-destruction Many participants (n = 19) reported autoimmunity as the main cause of SLE. Most of them associated autoimmunity with self-destruction. Indeed, the autoimmune process appears to be personified. Their own antibodies

These causes were cited by two-thirds of the patients (n = 23). In several narratives, the women did not differentiate between causes of the disease and factors triggering flares. Certain patients reported family events as being possible causes of their disease, such as the loss of a

TABLE 1 Frequency of SLE causal attributions (n = 33) Category

Frequency

Autoimmunity With notion of self-destruction Psychological causes (stress, emotions, personality factors, worries) Heredity/genes Magico-religious causes Bad fate, destiny, ordeal sent by God Evil eye, witchcraft, transgression A latent disease, an interior disease that ‘was already here’ Familial causes (conflicts, loss and grieving, separations) Infections (virus, etc.) and/or contagion Contagious but not infectious Ideas of guilt: disease as a just punishment Functional causes: humoral imbalance Causes linked to pregnancy Vaccination Weather (exposure to elements, e.g. cold, sun, etc.) Do not know

19 14 18 16 14 7 7 14 13 12 4 7 3 3 2 2 0


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close person, separation and conflict with spouse, parents or children: ‘It’s my mother’s death that started it all off’ (Respondent 5, aged 43 years); ‘It’s a disease caused by lack of motherly affection and recognition’ (Respondent 15, aged 42 years). The fact that an event is chronologically positioned immediately before the appearance of the symptoms of the disease reinforces its causal links with the disease: ‘My sister had an operation for cancer, and just after that they found I had lupus. It has to be linked’ (Respondent 9, aged 56 years); ‘It remains latent, and then develops following a big upset or a shock. In my case it was my second son’s suicide attempt. It really threw me, I think that is when it started. There must have been something to trigger it. I’m 99% convinced it can appear when you are stressed or have a big fright’ (Respondent 11, aged 65 years). Other patients suggested more general psychological factors that they did not relate to family events, such as ‘stress’, ‘strong emotions’, ‘worries’: ‘At the time, before the diagnosis, I was working in the Paris area, I really had too much to do. I had a period of immense stress at work, I was overworking. Whenever there is a period of great fatigue, it sets everything off, the pain . . .’ (Respondent 19, aged 50 years). For certain patients, the aetiological role of psychological factors over which some control can be exerted can in turn be seen as making the course of the disease controllable: ‘I take care, because I think that getting upset can cause flares. I try to keep calm and worry less’ (Respondent 16, aged 23 years). Conversely, for one patient (Respondent 14, aged 45 years) the disease is ‘uncontrollable’ precisely because it is dependent upon psychological factors.

Transmission and heredity Nearly half of the patients (n = 16) attributed a causal role to genes and heredity: ‘It’s a disease that is in the genes’ (Respondent 11, aged 65 years). Thus, trans-generational transmission of SLE is believed to be possible. The female predominance of the disease may reinforce this hypothesis: ‘If women are more affected by lupus, it must be because it’s genetic’ (Respondent 33, aged 18 years). Certain patients reported that they wondered about the existence of the disease in previous generations: ‘They did not do any research to see if my parents had the disease. I have a sister who doesn’t have it’ (Respondent 13, aged 53 years); ‘One of my cousins on my father’s side has intermittent paralysis in his left foot. I don’t really know’ (Respondent 28, aged 23 years). However, most worry about possible transmission to their children: ‘It comes from the family, I think on my father’s side, because my mother had a blood test and they said she did not have it; but my father died earlier. I hope my boys won’t have it. I’m glad I didn’t have any girls, as it’s more common in girls’ (Respondent 12, aged 35 years); ‘I’m afraid for my girls. I’m afraid of bad news for them, that they have the disease’ (Respondent 22, aged 41 years).

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Contagion and infection SLE was talked about by more than a third of the respondents (n = 12) as a contagious disease and/or classified as an infectious disease. Notions relating to contagious and hereditary diseases are sometimes intermingled: ‘In my opinion, either it’s a hereditary disease or a contagious disease. When they diagnosed me, they couldn’t tell me how you caught it. I didn’t really understand. It’s not really contagious, but I am still worried for my children. We’ll just have to wait and see’ (Respondent 19, aged 50 years). The risk of contagion or transmission can be seen as related to an infectious agent, but rather than talking about an infectious aetiology as such, patients use terms that belong to this register to name the disease and circumscribe it as an identifiable object, separate from the patient: ‘It’s a bug, something foreign that won’t go away’ (Respondent 12, aged 35 years); ‘A horrible virus which ruins your life, psychologically anyway’ (Respondent 16, aged 23 years); ‘It’s a microbe from the cold, in ice; I must have caught it in the cold, working in the fields in Turkey’ (Respondent 22, aged 41 years). Some women wonder about a disease transmissible by blood or sexual intercourse: ‘"Someone told me you shouldn’t give blood. Anyway I never have: my blood is no good’ (Respondent 13, aged 53 years); ‘It’s not a contagious disease and anyway I haven’t been having sex’ (Respondent 21, aged 31 years). The theme of impureness and being sullied is explicitly brought up in some discourse: ‘My blood is impure’ (Respondent 8, aged 35 years); ‘My body isn’t healthy, it isn’t pure’ (Respondent 27, aged 47 years).

Magico-religious causes Almost half of the patients (n = 14) referred to magicoreligious causes. Among these respondents, the majority were first- (n = 4) and second-generation (n = 5) migrants. Illness and distress can take on the meaning of an ordeal sent by God that has to be overcome: ‘It’s God testing you, you have to pray’ (Respondent 1, aged 23 years, parents born in ex-Yugoslavia); ‘It’s a message from God, but I don’t deserve it’ (Respondent 2, aged 41 years, parents born in France). Other causes were also quoted, such as witchcraft or the ‘evil eye’. These theories relate patients to other members of their families or other members of their cultural groups, in particular where certain alliances are involved: ‘I am someone who shows when I am well, so you see . . . . I always show it when I’m happy or pleased. I live in a place where you mustn’t tell lies to yourself, there’s jealousy everywhere. I’ve got nothing more than the others, but people think I have. Maybe someone sent me the evil eye. The evil eye exists for us, it’s no good pretending it doesn’t. After that, the family was broken up, my parents separated’ (Respondent 16, aged 23 years, parents born in Morocco); ‘Witchcraft from my previous mother-in-law, my former husband’s family . . . . My husband gave back the jewellery from my dowry eight years after the separation. That confirms it for me. They gave it back because he didn’t need it any longer for the spells, they had been cast’



(Respondent 26, aged 32 years, parents born in Algeria); ‘It could be an attack by a witch from the mistress of the father of my children. In a dream I had a fight with that woman’ (Respondent 27, aged 33 years, born in Congo). Among these patients, almost half (n = 8) also reported having resorted to traditional medicine in France or in their original home country: ‘I saw several healers, one of them, an African, said I didn’t have very long to live . . . I’ll maybe go and see a marabout’ (Respondent 4, aged 39 years, born in Guadeloupe); ‘I saw a fortune-teller, just once. I wanted to see one after my first lupus flare. I made the big mistake of telling my husband, and he immediately told me I was crazy. I should have gone, I would have found out something’ (Respondent 8, aged 35 years, parents born in Algeria).

A latent disease, ‘already there’ For around half of the patients (n = 14), SLE was ‘there all along, lying quiet’, before the diagnosis was made. It is a latent internal disease ‘waiting for its hour’ to come out into the open: ‘It’s like a lot of things you have inside you, there comes a time when it develops, it’s not because my life changed, but because the thing ripened, like something foreign, something I have extra to other people’ (Respondent 13, aged 53 years); ‘It’s a disease that wakes up when it wants’ (Respondent 25, aged 27 years). Among these patients, some compare the causes of SLE to other more frequent and more wellknown conditions, emphasizing the similarities or the differences. For instance, cancer can be brought up: ‘It’s a skulking disease you can’t predict. It all happens inside, the cells go mad, like in cancer. ( . . . ) We all have cancer in us, it may develop or it may not, in fact it’s rather like lupus’ (Respondent 8, aged 35 years). Chronic illnesses are also mentioned, present either in the patient herself or someone close, like diabetes; ‘Lupus is a disease you can’t understand compared to diabetes. I saw my mother lose her eyesight because of diabetes, nobody was taking care of her. It’s humiliating not to be able to do things for your children any more. I’d rather die if it happened to me’ (Respondent 8, aged 35 years); or asthma: ‘I keep strictly to the treatment, so that the disease doesn’t come back. I’ve stopped taking my treatment for asthma, I could see it was not a good thing’ (Respondent 25, aged 27 years).

Guilt and punishment For less than a quarter of the patients (n = 7), SLE leads them to search their past for behaviour that was inappropriate or wrong according to their individual moral standards, behaviour they were ‘guilty’ of and for which they were punished by the onset of the disease. Thus, some patients associate wrongful behaviour with their disease and therefore view it as deserved and just; others are more ambivalent and are against this punishment model: ‘It’s not possible, just when I was beginning to enjoy life and have new experiences. I don’t understand. Maybe I got overtired with going out too much, but there’s no way this disease could be a punishment,


even if at times I have felt guilty about thinking only of myself and leading a rather frivolous life’ (Respondent 7, aged 50 years). Patient 29 (aged 52 years) wondered about HIV diagnosis when the first lupus symptoms appeared after a period of ‘living it up’, including in particular an abortion. Even though she now knows she does not have AIDS, guilt pervades her discourse on the disease: ‘But I remember, after the abortion, I had a whole lot of health problems. So may be it was after the abortion that it was triggered ( . . . ) AIDS worries me a lot. Because, well, for a while I was really living it up. I was going out a lot. I used to go to night clubs when I left my husband. I had experiences I hadn’t had before because we were twenty when we got married. So I made the most of it . . . I was never home. As soon as I had an hour to spare I had to go and hang about in the streets. I loved it, seeing friends. I loved dancing, I used to dance. I’d go out in the evenings around ten, and I’d come back at four in the morning ( . . . ) Nowadays whenever a fellow knows you’re ill, he’ll ditch you, or he’ll say: she’s sick, she’s got something more serious and she doesn’t want to tell me the truth.’ In contrast, for one patient the arrival of the illness is all the more unfair because one of her activities is voluntary work with disabled people: ‘I did so many lovely things before, I helped sick and disabled people. I think that should have protected me, I’ve never done anything wrong. I never thought I would end up being ill myself’ (Respondent 16, aged 23 years).

Multiple causes and meanings Many patients suggested more than one cause for their SLE. The narrative transcribed below demonstrates how one patient (Respondent 14, aged 45 years) attributes her SLE to a combination of causes including autoimmunity, heredity, stress and psychology, fate and an ordeal sent by God. Her genetic theory was associated with her relationship with her maternal grandmother, which she described as being a happy one: ‘So let me explain, for me lupus is like AIDS, except that it is something I produce myself, while AIDS is something that comes from outside, that’s my explanation. Anyway, even AIDS one day—I say one day mind you—will be cured. But I’m sure the lupus won’t get better because it’s a psychological factor. It seems to me the more frustrated you get about it the worse it gets, and the more that thing inside you grows and nibbles and gnaws away at you. ( . . . ) I produce bad antibodies, but a whole lot of them. And they eat me up left, right and centre. It’s pretty clear to me . . . I think it comes from way back in the past, because there are symptoms my grandmother had. It’s just crazy! I get her pains, her diarrhea, her sickness. She couldn’t walk any more. Yes, I can see my grandmother in me, and I gather my great grandmother was the same ( . . . ) There is part of it that’s genetic, and the other part the doctors don’t know ( . . . ) There it is, it’s bad luck. It’ll just be as God wills it, I always say’.

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Discussion Patients’ beliefs about the causes of SLE have not often been reported [14, 15]. This is the first study in France. Most of the patients interviewed cited numerous causes that might explain their illness, a finding that differs from that reported by Goodman et al. [15], where 6 of the 36 patients interviewed stated that they did not know what caused their SLE and half made a single causal attribution. In contrast, the content of the themes noted in the present study is fairly similar to the Goodman et al. study [15]. In the Goodman study, the main causal attributions were heredity, stress, viral infections, family relationships and pregnancy. In our study, certain patients do not always differentiate the causes of the disease per se from the causes of flares, as was also noted in the study by Baker and Wiginton [14] on 38 women. Thus SLE leads sufferers to look for causes, similar to the quest for causes noted in RA [20]. Any illness or disease tends to generate questioning about the causes and even more so about the meaning of the illness: Why me? Why here? Why now? The answers provided by sufferers to these questions reach beyond a search for cause and become a search for meaning [8, 21]. Lay perceptions of disease are neither an impoverishment nor a distortion of medical conceptions. Nor do they exist in isolation; they can integrate numerous elements of medical knowledge, as can be seen from patient discourse. Since SLE is a relatively little-known disease, many patients in the present study compared it to other more common chronic diseases, which are in line with the observations in the study by Goodman et al. [15], where the majority of participants had no previous knowledge of SLE before diagnosis. The causes quoted are variously subscribed to by the different patients in our study; many of them were uncertain and in some cases suggested contradictory elements. It is well known that narrative concerning illness can be seen as a medium for discussing possible explanations for the illness and also for finding ways of relating to the illness [6, 22]. Some of the causes quoted are congruent with the SLE biomedical model, while others are not; for instance, references to possible genetic or infectious transmission of the disease. It is also interesting to see how the patients appropriate medical knowledge concerning autoimmunity and the role of stress in the pathophysiology of the condition. Autoimmunity is not an easy process to understand. The process is subjective for a number of patients. It can then come to be seen as a form of self-destruction. For two patients (Respondents 3 and 7), it had even become a sort of suicide equivalent: their bodies were destroying themselves because they had not managed to commit suicide. For these two patients, psychiatric disorders clearly pre-existed the diagnosis of SLE, but for most patients it is probably the very notion of autoimmunity and the related semantic network that generates representations of self-destruction [23]. SLE in patient discourse is a disease of the unloved. It appears ‘when you don’t like yourself’ (Respondent 13) or when one has lacked love or affection (Respondent 15).

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When this occurs there is a risk of doing oneself harm by manufacturing antibodies against oneself. The metaphors of wars and battles to wage, to be won or to be lost are frequent as with patient 17, who associated the ANAs with nuclear war. The role of stress is also familiar to patients. Numerous patients identified unhappy events in their past history before the diagnosis, around which they constructed the ‘plot’ of the narrative of their illness. SLE can thus sometimes be seen as a pathology of loss or grieving. It is again probable that the stress model favours identification by the patients of these events as happenings that change the course of events and turn good fortune into bad, thus offering anchorage points for narrative [22, 24]. These accounts need to be read as attempts to reconstruct a sense of order from the biographical disruption and fragmentation produced by SLE, as in other chronic illnesses such as RA [9, 25, 26] or OA [19]. As noted by Williams and Wood [25], the object of interpretation is not only the disease itself but also the disruption in the course of life. Closely allied to the psychological origin of the disease is the question of the control of stress to slow the progress of the disease and avoid flares. Several patients reported that they were perplexed as to the possibility of controlling the disease, which would give them an active rather than a passive role, with the corollary of genuine responsibility in relation to the disease. The appropriation of biomedical theories is thus not straightforward for patients and it does involve risk [27]. It is clear that the impact of these theories and the way in which they are interpreted depend on patients’ individual and family life history, on their personalities, on their therapeutic itineraries and on their socio-cultural environments. The search for meaning undertaken by these patients can also tend towards magico-religious explanations. These explanations are more frequent among migrant women, both first- and second generation, than among non-migrants. The disease can be seen as an ordeal sent by God to test them, a challenge to be met or as the result of something evil, the evil eye, witchcraft or something sent out intentionally by persons close to harm them or punish them for wrongdoing on their own part or by members of their families. It would be interesting to determine whether these theories are shared by other family members by recording narrative from other persons close to the subject [28, 29]. It would also be interesting to assess the other cultural affiliations of the migrant patients so as to determine whether the onset of the disease alters their acculturation strategies in a more global manner. Finally, in our study, migrants had emigrated primarily from French ex-colonies, mostly from either North or sub-Saharan Africa, as expected. Their origins are likely to have played a role in shaping magico-religious explanations, but only a larger sample size would enable us to compare the different influences of their North or sub-Saharan African vs European background. In addition to this, the existence of these theories, as in the case of theories about infectious and genetic causes,



clearly shows that there can be a marked discrepancy between patient narratives on their illness and the professionals’ explanatory models. It has been shown (for other disorders and in different settings) that discordance of this type does not necessarily lead to poor compliance (see, for example, Weiss et al. [30]). The objective should not be agreement between patient and physician explanatory models, but rather the co-construction of a therapeutic ‘plot’ that places the therapeutic action within a wider third narrative [31, 32]. This is particularly important for first- and second-generation migrants for whom bridges between their two worlds of reference need to be built by constructing a shared meaning to what has happened. The cultural dimension of their distress needs to find modes of expression and must be taken into account to reduce the gulf between the two worlds, rather than setting one off against the other. Various transcultural consultation systems have been developed to reduce the cultural misunderstandings that can lead to failed treatment alliances [29, 33]. Across discourses overall, ideas of guilt were also expressed by the patients, especially through narratives in which the disease is considered as a punishment for past wrongdoing or mistakes. It is a form of reverse legal proceeding: after the diagnosis of the disease, the offence, whether committed by the subject or another person, then needs to be identified [21]. Several authors have underlined how narratives carry moral considerations connecting family background, the coincidence of symptoms and biographical events and notions of shame or blame [6, 27]. In this perspective, in the narratives, the presence of theories relating to transmission, contagion, impurity or sullying could be related to gender [34]. The issue of the cause of the disease is closely associated for some patients with trajectory, consequences being very dependent upon cause: when patients wonder ‘Where did the disease come from?’ they also wonder ‘Now that I have it, where is it going?’ Is it transmissible genetically, by sexual intercourse, by blood? Is it contagious? The answers that the patients provide are sometimes surprising and show, alongside a quest for meaning, considerable fear of the disease being handed on. One important limitation of the study is that it is solely cross-sectional and provides no information on the stability or changes in beliefs about causes over time. Explanations for illness are most often dynamic entities and this has been found for illnesses other than SLE, for example by Hunt et al. [35] or Williams and Healy [36]. In their study, Goodman et al. [15] reported that representations of SLE changed over time following diagnosis, these changes occurring not in a progressive fashion, but at specific points, especially during or after recovery from a flare. In addition to this, relationships between meanings attributed to the disease and the psychological impact of the disease need further exploration, on account of the potential clinical implications and effects on coping strategies, since effective treatment relies on a sustained therapeutic alliance in chronic illnesses. Can theories that


provide meaning, that are well-organized, integrating the illness into the subject’s history and that are able to change according to the progress of the disease, reduce the mental suffering attendant upon the condition? Can these theories improve quality of life? In this perspective, in their study on patients with RA, Affleck et al. [20] found that patients who searched more actively for causes of their illness and who continued to ask ‘Why me?’ reported greater psychological distress. Longitudinal studies on narratives articulated with psychological and quality-of-life outcomes in SLE are required to explore this hypothesis. The question of our sample size must be addressed. Case-by-case analysis of transcripts takes a long time and the aim of the IPA studies is to say something in detail about the perceptions and understandings of the chosen group. IPA is an idiographic mode of inquiry as opposed to a nomothetic approach [18]. Of course, further studies can be conducted with other groups, gradually enabling more general claims to be made. But the power of an IPA study is judged by the light it sheds within its broader context. The question of the sample size depends partly on two factors: the degree of commitment to the case study level of analysis and the wealth of content of the individual cases. Some IPA studies have been published with very small samples, less than 10 cases. In our study, a larger sample size was needed because of the marked cultural heterogeneity of our patients, but we did not seek ‘data saturation’, as this is not a prominent feature of IPA. This is why some caution must be applied in considering the transferability and generalization of our results in other contexts and settings. In conclusion, to accurately diagnose and treat patients with SLE, it is essential to consider that psycho-social adaptation to illness is the result of the combination of personal, interpersonal, social, political and cultural contexts. For clinicians, the analysis of the experience of illness and beliefs about its causes sheds light on what is at stake for patients and on how patients attempt to give coherence to long-term distress. In order to address patients’ needs and expectations and to facilitate the long-term treatment alliance required for effective care, it is imperative that clinicians attend not only to questions of diagnosis according to professional concepts, but also to the subjective experience of their patients and the meanings they attribute to their problems. Through exploration of these issues, health care providers can find themselves in a better position to promote collaboration and to improve clinical outcomes. Rheumatology key messages Patients attribute SLE to many causes: autoimmune, psychological, familial, hereditary, magicoreligious and infectious causes. . Autoimmunity is often broadly conceived as a self-destructive process. . By their questioning, patients attempt to reconstruct coherence in their life histories. .

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Acknowledgements The authors would like to address their sincerest thanks to the participants. Disclosure statement: The authors have declared no conflicts of interest.

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