Coping with the uncertainty of uncontrolled epilepsy

Seizure 1993; 2:167-178

Coping with the uncertainty of uncontrolled epilepsy J E N N Y MURRAY

University of Melbourne, Parkville, Victoria, Australia Correspondence to J. Murray, Epilepsy Foundation of Victoria, 818 Burke Road, Camberwell,Victoria 3124, Australia

Parents of children with uncontrolled epilepsy experience chronic uncertainty in relation to their child's condition. In this study the author sought to explore the levels and dimensions of uncertainty experienced by parents of children with Lennox-Gastaut syndrome; the relationship between parental uncertainty and stress; and the range of cognitive and behavioural coping responses employed by parents. The population is drawn from a self-help group for parents of children with L.G.S. coordinated by the author. Data was collected through questionnaires as well as in-depth interviews with parents. The findings revealed five key dimensions of uncertainty: in respect to (1) diagnosis, (2) aetiology, (3) seizure activity, (4) treatment, and (5) prognosis. The study highlights the importance of information, contact with other parents of children with uncontrolled epilepsy and diary writing as aids to reducing uncertainty and enhancing coping for parents. The paper concludes by calling for a re-evaluation of the role of parents of children with epilepsy which moves away from the notion of parents as a source of pathology for their children to parents as part of a family unit attempting to cope as best it can with a chronic illness.

Key words: epilepsy; family; parents; stress; uncertainty; coping.

INTRODUCTION

'It's the variability t h a t really makes it so stressful. You never know when it is going to be chaos again and you'll have one. J u s t because this morning is terrific doesn't m e a n tonight is going to be terrific, either behaviour-wise, medication-wise, or any other-wise. So it is the unpredictability of it t h a t is really nerve-racking to live with.' The impetus for this study came from the author's involvement with a support group for parents of children with Lennox-Gastaut syndrome (L.G.S.). L.G.S. is a severe epileptic syndrome clinically manifested by intractible seizures of several types, usually some degree of intellectual disability and a typical electroencephalographic pattern, with the onset generally in the pre-school years 1. Over several years of coordinating the group, the author became increasingly aware of the apparent isolation and uncertainty facing parents of children with this syndrome and a need to document the knowledge they had acquired through years of experience as well as their strategies for coping. 1059-1311/93/030167+ 12 $08.00/0

The major areas of inquiry were as follows: --levels and dimensions of uncertainty experienced by parents of children with L.G.S.; - - t h e relationship between parental uncert a i n t y surrounding L.G.S. and stress; --coping strategies employed by parents to reduce uncertainty and regain a sense of control. The concept of uncertainty is recognized as a key factor in the experience and management of chronic illness. Strauss and Glaser 2 assert t h a t illnesses which have uncertain prognoses are associated with great hardship for the individual and family concerned. Yet the seemingly striking degree of uncertainty associated with the experience of epilepsy is not wellunderstood or recognized. Epilepsy, by its very nature, implies unpredictability with respect to the occurrence of seizures. In addition, people with epilepsy and their families often report having great difficulty in accessing adequate information about the condition, in finding clear and definitive answers to fundam e n t a l questions, such as those surrounding aetiology, diagnosis, t r e a t m e n t and prognosis.

© 1993 Bailliere Tindall

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Uncertainty theory Uncertainty can be defined as a cognitive state created when an event cannot be adequately structured or categorized because of insufficient cues 3. Uncertainty occurs when the person is unable to assign definite values to events and/or is unable to accurately predict outcomes 4. Lazarus and Folkman 5 make a clear distinction between the concepts of ambiguity and uncertainty. Ambiguity is defined as a lack of situational clarity whereas uncertainty is defined as the person's confusion about the meaning of the environmental configuration. In other words, ambiguity has a cause external to the person whereas uncertainty is internally linked to the person's appraisal of the situation. Ambiguity arises when the family cannot get clear facts about the event. However, in some situations the facts are clear but the family continues to deny or ignore them. Information from the environment can be unambiguous and yet the person still experiences uncertainty. On the other hand, even when there is ambiguity in the environment, a person may feel confident about what to do. Uncertainty reduction theory was first put forward by Berger and Calabrese 6 as an explanation for interpersonal communication behaviours displayed during initial interactions. The inability to predict and explain the action of others was suggested as the central motivating force guiding behaviour in first encounters with other people. Of relevance to this study is one of the most widely believed principles of uncertainty reduction theory, axiom 3, which states that there is a positive relationship between uncertainty and information seeking. 'This axiom seems to make a great deal of intuitive sense: if one is uncertain (i.e. does not know) then seek information (i.e. ask); if one is certain (i.e. knows) then do not seek information (i.e. do not ask)7. ' Despite widespread belief in the validity of this principle, empirical tests of it are few in number and inconsistent in their findings. It seems the equation is too simplistic. Berger s has since made an important addition to the theory by distinguishing between a person's level of uncertainty and their concern for its reduction. For instance, some people may have higher levels of tolerance for uncertainty than others. Furthermore, uncertainty reduction

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m a y have either positive or negative consequences. Possible examples of the negative impact of reducing uncertainty are where hope is extinguished or there is an unreasonable lowering of expectations. Numerous studies have suggested a relationship between uncertainty and stress. The idea that if coping does not lessen uncertainty then the environment is perceived as stressful has been widely lauded 9. Nowhere is the role played by uncertainty in generating stress more widely noted than in cases of physical illness and disability. Moos and Tsu 1° and Cohen and Lazarus 11 in reviewing studies of how people cope with physical illness cite dealing with uncertainty as a major adaptive task, A major reason why uncertainty can be stressful is that it has an immobilizing effect on anticipatory coping processes 12.

Coping theory This study adopted a process-centred approach to coping as developed by Lazarus and Folkman (1984). Coping is defined as: 'Constantly changing cognitive and behavioural efforts to manage external and/or internal demands that are appraised as taxing or exceeding the resources of the personl3. ' This definition is process-orientated rather than trait-orientated, defining coping in terms of what the person actually thinks or does rather than what the person usually does. Also important is the use of the word 'manage' which may include minimizing, avoiding, tolerating and accepting stressful situations as well as the more widely noted attempts to master the environment. Hence coping is not equated with mastery here as elsewhere in the literature. The process approach to coping has a number of other significant features. It is concerned with understanding coping in a specific context. Coping is defined independently of outcome which means that no one strategy is considered inherently better than any other. And finally the coping process is characterized by change in thoughts and behaviour over time. These changes are a function of continuous reappraisals of the shifting person-environment relationship.

Coping with epilepsy

Epilepsy and the family As with other chronic conditions, the attention of researchers and clinicians in the epilepsy field has tended to focus on the 'patient' in isolation. Investigation of the impact of epilepsy on family functioning and social networks in general has lagged far behind. This is an incongruous and lamentable state of affairs given the profound impact epilepsy can have on families, particularly where seizures cannot be controlled by medication 14-1s. In general the literature portrays parents of children with epilepsy in a negative light, with a great deal of explicit and implicit blaming directed their way. Green and Hartlage 19 suggest t h a t over-protective attitudes of parents as well as their acceptance of lower levels of performance was related to underachievement of children with epilepsy at school. Ferrari 2° suggests t h a t poor selfconcept in a child with epilepsy is related to low parental expectations. Parental attitudes have also been linked with a range of behavioural disorders in children, increased psychopathology and even an increase in seizure occurrence 21. As a result, parents of children with epilepsy have been depicted in the literature in a narrow and predominantly negative way: as a source of pathology for their children. Yet it is increasingly recognized elsewhere in the health literature t h a t families are a key resource in the care and rehabilitation of people with chronic illnesses 22' 23. This study sought to explore family strengths as well as vulnerabilities, to assess how families do in fact cope in the face of a severe chronic stressor like uncontrolled epilepsy. This comparatively optimistic approach is consistent with family stress research since the 1970s and 1980s, where the emphasis has shifted from crisis to coping. Researchers into the effects of stress have become less concerned with why families fail and more concerned with why families succeed 24'25. Epilepsy is what stress researcher Pauline Boss would describe as an ambiguous, chronic stressor in t h a t it generally runs a long course and is fundamentally unpredictable in nature 26.

METHOD Given the dearth of research on the subject, an exploratory study was undertaken using both quantitative and qualitative methods. Several

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methods of data collection were combined to highight different dimensions of the same phenomena. These were a questionnaire, indepth interviews and the insights of the practitioner-researcher.

Design Questionnaire The data collection process was initiated with a questionnaire sent to all members of a support group for parents of children with LennoxGastaut syndrome run through the Epilepsy Foundation of Victoria. The questionnaire consisted of 44 questions, the majority of which were of a multiple-choice format, as well as a small number of open-ended questions. Respondents had the option of anonymity with the questionnaire.

In-depth interviews Subsequent to receipt of the questionnaires, indepth interviews were conducted by the researcher with a sub-group of 10 parents. Survey respondents living in Victoria were asked to indicate whether or not they were willing to be interviewed. As m a n y as 83% (25) of Victorian respondents agreed to be interviewed but time constraints permitted only 10 interviews. Incoming data from the surveys suggested t h a t the age of the child with L.G.S. was a critical factor in parents' experience of the syndrome. A stratified sampling procedure was used which involved applying a simple random sampling procedure based on children's ages. All of the interviews were carried out in the homes of parents and were taped to increase the accuracy of data collection and to allow the researcher to be more attentive to the interviewee. The interview style was a semi-structured focused interviewing format, using an interview guide. To some extent the questions evolved and changed from one interview to the next in response to emerging conceptual insights, in a m a n n e r typical of qualitative research of this nature 27. The selection of indepth interviews as a method of qualitative research was in accordance with the study's aim of understanding the significance of the experience of L.G.S. from the parents' perspective.

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Practitioner-researcher approach A key feature of this study was the integration of practitioner and researcher roles. The author has worked as a social worker at the Epilepsy Foundation of Victoria for the past 5 years, two of which involved the coordination of the L.G.S. support group. Involvement with parents in the group has ranged from lengthy telephone consultations, written correspondence and facilitating monthly group meetings. The dual practitioner-researcher approach has the advantage that the person conducting the research has some understanding and sensitivity to the issues from the outset. It has been argued by Scott 2s that lengthy exposure to similar situations in social work practice contributes significantly to the cognitive schema of the practitioner. This is sometimes referred to as 'practice wisdom'. However, it must also be acknowledged that 'practice wisdom is not always wise '29. It may be incorrect or distorted by non-representative cases and stubbornly resistant to change.

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- - A g e of respondents ranged from 29-70, with an average age of 44 years. --Thirty-eight (93%) respondents were female and three were male. --Respondents' children with L.G.S. ranged in age from 3 to 34 years with an average age of 13 years and 71% were of school-age. --Respondents reported that 56% of children had a moderate to severe intellectual disability, 32% of children had a mild intellectual disability, 7% had learning difficulties, and 2% had normal learning abilities.

Parents interviewed The interview sample was a sub-set of 10 of the survey sample and comprised a representative number of parents of children in each of the following age-groups: 1 - 5 years (one parent), 6 - 1 2 years (three parents), 13-17 years (three parents), and 18+ years (three parents). The 10 parents interviewed were all mothers of children with L.G.S. They were aged between 35 and 55 years and all lived with their husbands and children except one whose children were all adults and no longer living at home.

Consultations Key stake holders in the L.G.S. program were identified and consulted at various stages in the research process. These were L.G.S. group members, staff at the Epilepsy Foundation of Victoria and two Neurologists with a particular interest in epilepsy.

Data analysis Survey data was compiled on computer using a spreadsheet software package called 'Excel'. Interview data was transcribed and coded by the researcher for the purposes of a content analysis. Common themes linking critical issues were extrapolated from respondents' statements of beliefs, feelings and situations.

Sample

Survey respondents

RESULTS

Of the 60 surveys sent to all members of the L.G.S. network, 41 were returned, a response rate of 68%, which seemed reasonable and viable. A major factor serving to reduce the number of questionnaires received was the timing of distribution which coincided with the Christmas holiday period. Characteristics of the survey respondents can be summarised as follows. - - T h i r t y lived in Victoria, seven interstate and four overseas. - - A l l spoke English as the language normally spoken at home, and 66% were born in Australia.

The findings revealed that parents of children with L.G.S. experience a pervasive sense of uncertainty about many aspects of their child's condition. This uncertainty is particularly acute around the time of onset of frequent seizure activity in early childhood. As one parent said: 'Something is happening so drastically and there were no answers anyone could give you.' However, parents also reported that periods of uncertainty and the associated stress recur in an episodic way which perhaps reflects the changing and episodic nature of the syndrome itself. Five key dimensions of uncertainty were

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Table 1 : Seizures

Range Average

Age at time of 1st seizure

Age when L.G.S. diagnosed

Time between 1st seizure and diagnosis

Birth to 8 years 2.7 years

2 months to 26 years 7.4 years

Birth to 25 years 4,7 years

identified: these were in relation to: (1) diagnosis; (2) aetiology; (3) seizure activity; (4) treatment; and (5) prognosis. Each of these will be discussed in t u r n with consideration given to the emotional impact on parents, factors t h a t m a y have contributed to uncertainty and strategies adopted by parents (if any) to reduce uncertainty and enhance a sense of control.

Uncertainty Diagnosis The period of uncertainty preceding the exact diagnosis of their child's condition was a lengthy one for m a n y parents. Parents completing the survey reported t h a t their children had their first seizure somewhere between birth and 8 years of age, with the average age being 2.7 years. However, the time between the first seizure occurring and parents learning of the L.G.S. diagnosis varied widely from virtually no time to 25 years, with as m a n y as 10 parents (24%) reporting a waiting period of 8 years or more. During this time parents knew only t h a t their child had epilepsy. Information acquired on 'epilepsy' through books or talking with others with personal experience of epilepsy was of limited value because of the incongruence with their own unusually severe experience of epilepsy. A wide range of reasons were given by parents for perceived delays in learning of the diagnosis. Common responses were that there had been a change in seizure pattern eventually evolving into L.G.S. and the time needed to conduct various diagnostic tests such as electroencephelograms and CT scans. Several parents of older children pointed out t h a t the term L.G.S. was not widely used in Australia when their children were young. Some parents wrote of difficulties in convincing doctors t h a t there was a significant problem. Others wrote t h a t doctors knew the diagnosis but had not communicated it to them and they later found out by incidental means. As one parent stated: 'From the time my child had his first seizure, his neurologist always told me he had a "complex form of epilepsy"--2 years ago

I happened to see the school psychologist's report and the words "Lennox-Gastaut syndrome" jumped out at me.' The perceived benefits of having the more precise diagnosis varied from parent to parent. However, for m a n y it brought a sense of relief to finally have some explanation for what was occurring. The label helped to diminish uncert a i n t y and enabled parents to communicate with greater clarity to others about their child's condition. And m a n y parents reported t h a t the diagnosis of L.G.S. enabled t h e m to obtain more relevant information and needed support services. A typical response was: 'I immediately researched L.G.S. as well as I was able and even though the prognosis isn't fantastic, there is some comfort in having a degree of knowledge about my son's condition instead of being completely in the dark.'

Cause Approximately two-thirds of parents responding to the survey reported being uncertain about the cause of their child's L.G.S. (34% very uncertain, 34% quite uncertain). In the face of no medical explanation for the cause of the syndrome, parents attempted to construct their own meaning for why it had occurred. The process of searching for an answer to the question of why often involved perceptions of guilt and self-blame. As one mother said to me: 'I t h i n k you can probably accept it easier if you can s a y - - " o h well, t h a t is what did it" ... if you're not looking for the cause, looking at yourself for the cause. You wonder what you have done. There's t h a t whole horrible, what i f . . . ' However, some parents seemed to have accepted the likelihood t h a t they would never have an answer and have long since given up asking why. For some the question was simply not relevant as they focused on the here-andnow.

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J. Murray 14 12 10 Z

8 6 4 2 0

Very certain

Quite certain

Quite uncertain

Very uncertain

Level of uncertainty Fig. 1 : Perceived level of uncertainty regarding cause of L.G.S.

Seizures As expected the seizure picture described by parents was one of frequent seizures and multiple types of seizures. Parents reported experiencing both 'event uncertainty', that is uncertainty about what is and is not a seizure; and 'temporal uncertainty', that is uncertainty about w h e n seizures will occur and the duration of 'good' and 'bad' phases. During interview several parents reported having difficulty in describing the number and types of seizures their children were having because (1) some seizures did not fit neatly into the commonly-defined categories and (2) some seizures were reportedly difficult to differentiate from particular behavioural disturbances or sideeffects of medication. The often subtle and confusing nature of seizures is clearly conveyed by one parent in the following statement: 'His absences have never been "pure", they have never been the kind of ones where you have a start and a finish but you will suddenly notice a change where he doesn't listen or can't, where he is struggling to articulate what he wants to say or be totally disorientated.' The fact that such seizure activity is unclear and open to interpretation can be stressful for parents and lead to conflict both within the family and between the family and professionals who may impose differing interpretations on the situation. For example, two mothers spoke of conflict with teachers who had reportedly misconstrued seizure activity and side-effects of medication to be naughty behaviour. Such uncertainty also led to selfdoubt for some parents who reported feeling unprepared and unsure of what was happening to their child, particularly in the early days.

The findings demonstrated that parents perceived their children's seizures to be essentially unpredictable in that they occurred without warning. The unpredictability of seizures with the potential to cause serious injury was found to be very stressful for parents. The timing and duration of 'good' and 'bad' phases of the condition was also widely reported to be unpredictable. An unexpected finding was that even the 'good' phases could be as stressful or more stressful for the family because of temporal uncertainty. These periods of relatively few seizures were characterized by ambiguous and contradictory appraisals by parents who were attempting to reconcile feelings of hope for sustained improvement with dread of yet another relapse. As one parent said: 'I think it is soul destroying actually. It doesn't matter how many times you tell yourself this isn't going to last when it is a good patch, you still hope that this medication is the right one or some maturation has happened and so on; so its still devastating when that's followed by a bad patch.'

Medications All 41 parents reported that their children were taking anticonvulsant medications on a daily basis. Nine (22%) children were on monotherapy (one drug) but the vast majority (78%) were taking two, three or four different medications. S u r v e y data shows that 76% parents felt some degree of uncertainty about the ongoing effectiveness of medications (17% very uncertain, 59% quite uncertain). Many of the parents interviewed also expressed grave doubts about the value of available medi-

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cations for their children, particularly parents of older 'Children' who had experienced many years of unsuccessful treatment.

Prognos~ Uncertainty about their child's future was the area of greatest uncertainty and anxiety for parents. It is also the area where least information is available. Unfortunately, very few longitudinal studies of L.G.S. have been conducted to date. As many as 85% of parents responding to the survey reported feeling some degree of uncertainty about their child's prognosis and future (46% very uncertain, 39% quite uncertain). Higher levels of stress seemed to be more apparent where the child's condition was appraised as fluctuating dramatically from very bad phases characterized by frequent seizures and/or behavioural disturbances to periods of relatively high functioning which provided parents with tantalizing glimpses of the child's potential. As one parent said: 'It's an awful conflict. I often think he would be better off if he was a nice little vegetable and didn't know anything about it. It wouldn't cause the immense conflict that goes on with it. He knows that he should be able to do things but he can't do things and why can't he do things?'

verse was true: these parents needed to plan ahead, to consider future options even if highly improbable. There seemed to be a need to at least make an effort to reduce perceived uncertainty about the future for the sake of the child and other members of the family.

Coping strategies Parents reported a complex and wide range of strategies for coping with L.G.S. Three of the most widely reported behavioural responses were to seek information, make contact with other parents through the L.G.S. support group and to keep a diary. H a l f the respondents reported difficulties in obtaining relevant information on L.G.S. The most common contributing factors were both of a medical nature, namely: 'unsatisfactory communication with your doctor' and 'delay in diagnosing L.G.S.' Parents generally sought a frank and full disclosure of information, particularly from their child's specialist. A typical response was: 'If you could be given hard and fast facts, then maybe you could accept it or maybe even if you were prepared for the worst maybe that wouldn't be as bad but they don't tell you anything.'

In the face of a profoundly uncertain future, parents reported responding in quite different ways. Some felt unable to plan for the future and lived from day to day. For others, the con-

The majority of parents surveyed (63%) felt that the information they had managed to find on L.GoS. was 'generally relevant and useful', but with some important qualifications. Many believed the information to be 'too focused on medical aspects, ignoring other issues relating

25

20

15 d Z 10

0~

Very certain

Quite certain

Quite uncertain

Very uncertain

Level of u n c e r t a i n t y Fig. 2: Perceived level of uncertainty regarding ongoing effectiveness of medications.

Not answered

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20 18 16 14 ¢/

Z

12' 10 8 6 4 2 0

Very certain

Quite certain

Quiteuncertain

Very uncertain

Level of uncertainty Fig. 3: Perceived level of uncertainty regarding prognosis.

to the daily mangement of L.G.S. and there was 'not enough information from families themselves recounting personal experiences'. One way of gaining information from other parents in a similar situation was through membership of the L.G.S. support group. The most commonly reported benefits of the group for parents were enhanced knowledge and the feeling of not being alone in coping with a severe epilepsy syndrome. The support gained from others going through a similarly stressful experience cannot be over-estimated. The following response was typical:

Table 2:

In general, how suitable h a s the written information you have received on L.G.S. been to your particular needs? No. % Generally relevant and useful Too focused on medical aspects ignoring other issues related to the daily m a n a g e m e n t of L.G.S. Not enough information from families themselves recounting personal experiences Too technical and difficult to understand Other Too simple and general

26 19

63 46

17

41

7 2 0

17 5 0

Table 3: Gains from group

'To be part of a group which speaks one language is a comfort, as the outside world can only be outwardly sympathetic but actually glad t h a t they're not in our situation. I feel t h a t applies to the medical profession also. No-one can possibly understand how disabled the whole family becomes with an L.G.S. child except another family.' Almost all of the parents interviewed reported having kept a diary of their child's symptoms in the past, and m a n y of them were still doing so. The reasons for doing so were mixed and included: to enhance accurate reporting to doctors and to identify seizure precipitants, patterns and responses to different medications. Not only did the diary seem to enhance parents' perceptions of control, but occasionally seizure precipitants or medication side-effects were discovered in this way. As one parent said: 'It makes you feel like you are doing something useful. I'm not sure if it is useful at all but it feels like it is.' On the other hand note-taking served to exacerbate parental stress in some cases.

Overall, what have you gained from your contact with the L.G.S. network? No. Increased knowledge Sense of not being alone Information for own purposes Comfort of knowing it is there Increased confidence Reduced sense of isolation Information to give to others Sense t h a t we are all in this together/worldwide Provides hope Not much No answer

13 10 4 3 3 1 1 1 1 1 6

When seizures are very frequent, perhaps as m a n y as 100 per day, it becomes extremely tedious to document them all. This is compounded by difficulties in interpreting some behaviours. One parent reported giving up keeping a diary because it only served to confirm a pattern of deterioration which was distressing. DISCUSSION

The study yielded data t h a t was diverse and complex. The respondents were a homogeneous group in terms of the medical diagnosis of their

Coping with epilepsy

children (as recommended by Mishel, 1984 for future research into uncertainty and illness) but were in many ways a heterogeneous population in respect to their responses and adaptation to the chronic condition in question. The finding t h a t m a n y parents did not learn of the diagnosis of L.G.S. for m a n y years after the onset of seizures is probably due to a multiplicity of factors. It must be emphasized t h a t in m a n y of these cases the more precise diagnosis could not have been made sooner. It is recognized t h a t the diagnostic process for L.G.S. is a complex one, generally requiring the expertise of a paediatric neurologist and, furthermore, the children of m a n y respondents began having seizures when the diagnostic term, Lennox-Gastaut syndrome was not commonly used. However the number of parents who reported learning of the L.G.S. diagnosis by incidental means is of concern because it undermines their role as equal partners in m a n a g i n g a severe chronic condition and seems to afford little respect and sensitivity for the stressful situation with which they are confronted. It also suggests t h a t there may be m a n y families in our community who are u n a w a r e t h a t their child has L.G.S., either because doctors are yet to diagnose it or the diagnosis has yet to be communicated to parents. The high degree of uncertainty about cause perceived by parents is in accordance with medical research on the subject--in m a n y cases doctors cannot ascertain a clear cause for the syndrome 3°. As with some other aspects of L.G.S. one would expect this to be primarily a case of externally caused ambiguity. Facts on aetiology in individual cases are often simply not available. The phenomenon of parental guilt has been noted elsewhere in the literature on childhood illness. For example, researchers in the leuk a e m i a field, Comaroff and Maguire offer the following explanation for parental feelings of guilt in situations of childhood illness: 'The identity of children is generally regarded as a function of t h a t of their parents, who feel practical and moral responsibility for their well-being and their suffering. Threatening illness is frequently seen as an assault on child rearing capacities. Hence the quest for cause and meaning in such illness is closely tied to the attempt to allocate responsibility for its occurrence31. '

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The level of stress associated with an uncertain cause varied. Some regarded the question in a pragmatic and accepting way, as one parent said: 'what's done is done'. Others found it to be a source of recurring feelings of guilt and stress. It is the researcher's contention t h a t the event uncertainty reported by parents in this study is compounded by the fact t h a t much of the information and literature available to families focuses on the more common, clearly defined seizure types. Many people's seizures do not fit neatly into well-defined categories but rather are subtle, complex and idiosyncratic in nature. This problem is particularly apparent in difficult-to-control epilepsies associated with other disabilities. Parents would benefit from more information on these subtle and confusing aspects of L.G.S. Assessments from neuropsychologists can also assist in distinguishing between seizures, behaviour problems and medication effects, leading to more appropriate intervention strategies. The findings show t h a t parents perceived the occurrence of seizures in their children to be essentially unpredictable and that this was a source of enormous stress. The concept of predictability is a major theme in stress research. A large body of evidence suggests t h a t predictable shock is less aversive t h a n unpredictable shock 32. This m a y be because signalled events allow for anticipatory coping to occur, t h a t is people can prepare in some way for the stressor event. An alternative explanation, which may also be applicable here, is t h a t the knowledge t h a t one will get a warning is useful because it allows periods of 'safety', during which one can relax 33. Conversely, where warnings are known not to occur as with most seizures, there may be no 'safe' time when total relaxation is possible except perhaps when the child with epilepsy is in the care of someone else. Weinberg and Levine 34 assert t h a t control and predictability are closely inter-related. It is difficult to control a situation t h a t is not predictable. This study found evidence of parents going to enormous lengths to try and establish some degree of predictability and control over seizures. For example, a wide range of 'alternative treatments' were tried by parents (sometimes at great expense) which, unfortunately, proved futile in most cases. This sort of activity perhaps reflected a family belief system of mastery over problems, a determination to find a solution. Many parents expressed concern about the

176 high dosages of medications prescribed for their children. Uncertainty about possible damage from side-effects was compounded by real doubts in many cases about the effectiveness of the drugs in reducing seizure activity. The high incidence of polytherapy reported by parents is a legitimate concern for them given that polypharmacy has been found to contribute to psychosocial disturbance. There is mounting evidence that a reduction in polypharmacy can lead to cognitive improvement across a range of mental and motor abilities35,36, and has resulted in improved seizure control for a substantial proportion of people with epilepsy37. Behavioural correlates of polytherapy have received less attention, although there is some evidence that a reduction in the number of anticonvulsants may diminish problematic behaviour 3s. For these reasons, Espie and colleagues 39 call for the 'active limitation of the use of polytherapy in mental handicap' and highlight the need for further research into the social effects of drug treatments, especially where more than one drug is prescribed. The study clearly demonstrates that the experience of living with L.G.S. from the parents' perspective was one of deep and chronic uncertainty. It is difficult to assess the relative significance of internal uncertainty and external ambiguity in relation to parents' perceptions of their child's condition. However, the consistently high levels of uncertainty found here m a y indicate that causes of uncertainty external to the individuals concerned were of critical importance. Theirs is an ambiguous environment of limited information, uncontrolled seizures and unclear prognosis. They must live with the reality of limitations in the efficacy of medical treatment in this area of epileptology: L.G.S. remains, in most cases, stubbornly resistant to treatment despite major technological advances in recent years.

Implications for health professionals The goal of health professionals should be to reduce uncertainty and enhance perceptions of control. Parents have a strong need to know all they can about their child's condition. At the same time, the communication of such information needs to be done in a sensitive way, taking into account the potential for negative consequences arising from uncertainty reduction as identified by Berger 4°. For example,

J. Murray

some parents spoke of not wanting to know of worse-case scenarios concerning the future course of L.G.S. In other words, enhanced knowledge m a y serve to unduly increase anxiety and stress in a way that diminishes the parents' ability to cope. The diagnosis of a severe chronic condition like Lennox-Gastaut syndrome is best conceived of by health professionals as a family diagnosis. As one parent said in this study: 'the impact of L.G.S. on the whole family over the years has been so disruptive that the whole family has become disabled in a social sense'. Epilepsy cannot generally be cured but must be managed by families for long periods of time. The tasks of managing a chronic condition have as much to do with psychosocial considerations as medical management of the condition itself. Hence there is a need to broaden the focus of intervention from the needs of the child to the provision of information and support services for the entire family. The role of information broker is a critical one: families need information that is targeted to their particular needs and made accessible both in geographical and cultural terms. This finding was in accordance with previous research underlining the value of information in coping with chronic illness 41-43. In particular there is a need for early diagnosis, improved communication between doctors and parents and greater availability of written information specifically designed for parents. The study also endorses the value of support groups for parents of children with uncontrolled epilepsy in reducing uncertainty and stress. Such groups m a y not radically alter the way in which parents manage epilepsy, but can allow parents to reappraise their situation in the light of enhanced knowledge and social comparison in a way that facilitates coping. In view of the small sample, generalizations must remain tentative until tested on a larger scale. Nevertheless, the study was enlightening and should be of value to its major stakeholder: parents of children with uncontrolled epilepsy. It highlights the value of experiential knowledge, the relatively untapped resource of knowledge contained within families which deserves further attention from researchers. Most importantly it challenges the notion that parents of children with epilepsy have a predominantly negative influence on their children's lives. To the contrary the amount of caring and energy that these parents provide for

Coping with epilepsy

their children is extraordinary. As one parent said to me" 'The amount of energy that goes into facilitating things for your child and to ensure that other people do understand and respect and offer your child support is just enormous, and it never stops, and probably never will stop.'

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Medicine.

15. 16.

17.

18.

ACKNOWLEDGEMENTS 19.

The author would like to thank the Epilepsy Foundation of Victoria for its support of this research project. My thanks also to Dorothy Scott for her supervision and guidance and to the parents who participated in the study.

20.

21.

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