M J Godelief

Gertrudis I J M Mars

Guy A M Kempen

Peter P M Widdershoven

Jacques T M Janssen

Van Eijk

Janssen

Jacques T M Van Eijk

Conceptualizing autonomy in the context of chronic physical illness: relating philosophical theories to social scientifi c perspectives

Keywords:

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Introduction

Chronic physical illness may bring about pervasive changes in people's lives. For example, it may become diffi cult to perform valued activities and social roles, dependency on others may increase and future plans may need adjustment. Under these circumstances, it is a real challenge to preserve self-determination. People with a chronic illness continuously have to negotiate autonomy in their daily activities and in their relationships with family, friends and society [START_REF] Williams | Coming to terms with chronic illness: The negotiation of autonomy in rheumatoid arthritis[END_REF].

Previous research has uncovered various threats to autonomy, such as functional decline, institutionalization, professionals' paternalistic approach, people's negative mindset and lack of clarity about goals, aspirations and purpose in life [START_REF] Collopy | Autonomy in long term care: Some crucial distinctions[END_REF][START_REF] Hickey | Chronic illness and aging: A personalcontextual model of age-related-changes in health-status[END_REF][START_REF] Agich | Autonomy and long-term care[END_REF][START_REF] Becker | The oldest-old: Autonomy in the face of frailty[END_REF][START_REF] Mcwilliam | A new perspective on threatened autonomy in elderly persons: The disempowering process[END_REF]. Opportunities to promote autonomy have been suggested as well, including cognitive behavioral therapy and a patient-centered approach by health professionals [START_REF] Rybarczyk | Applying cognitive-behavioral psychotherapy to the chronically ill elderly: Treatment issues and case illustration[END_REF][START_REF] Williams | Supporting autonomy to motivate patients with diabetes for glucose control[END_REF].

The fact that previous research on autonomy has identifi ed different threats and opportunities might be due to the use of different conceptualizations of autonomy. These may have different implications for research, policy and practice aimed at autonomy support. Consider, for example, the relationship between autonomy and dependence. As [START_REF] Berlin | Four essays on liberty[END_REF] already has pointed out, some scholars equate autonomy with independence and non-interference, which implies that dependence confl icts with autonomy. Yet other scholars conceptualize autonomy as actively shaping life in accordance with one's own values [START_REF] Berlin | Four essays on liberty[END_REF]. Other people may facilitate this, which means that dependence may enable autonomy. Then there are scholars who argue that all people depend on others during the entire life cycle, albeit to various degrees, which means that autonomy can only take shape through this interdependence [START_REF] Tronto | Moral boundaries: A political argument for an ethic of care[END_REF]. Hence, depending on its conceptualization, autonomy may be promoted or impinged on by reducing dependence.

Which of the different conceptualizations of autonomy is 'the right one' is an issue unlikely to be resolved, because who should decide this and on what grounds [START_REF] Seidman | Postmodern social theory as narrative with a moral intent[END_REF]? We might, however, argue on pragmatic grounds why we favor a particular conceptualization in a particular context [START_REF] Seidman | Postmodern social theory as narrative with a moral intent[END_REF]. The implications of different conceptualizations of autonomy have been discussed in the contexts of different disciplines such as gerontology [START_REF] Collopy | Autonomy in long term care: Some crucial distinctions[END_REF], nursing (e.g. [START_REF] Hertz | Conceptualization of perceived enactment of autonomy in the elderly[END_REF][START_REF] Ballou | A concept analysis of autonomy[END_REF][START_REF] Proot | Autonomy in the rehabilitation of stroke patients in nursing homes: A concept analysis[END_REF][START_REF] Keenan | A concept analysis of autonomy[END_REF][START_REF] Aveyard | Is there a concept of autonomy that can usefully inform nursing practice[END_REF] and rehabilitation [START_REF] Proot | Autonomy in the rehabilitation of stroke patients in nursing homes: A concept analysis[END_REF]. In the context of chronic physical illness, however, the implications of different conceptualizations have not been discussed yet. It therefore remains unclear how autonomy might be understood with regard to chronic physical illness.

This article aims to conceptualize autonomy in the context of chronic physical illness. To this end, we compare and contrast a selection of contemporary philosophical theories of autonomy with social scientifi c perspectives on chronic illness. The philosophical theories offer a thorough debate on autonomy, while the social scientifi c perspectives provide a rich source of insights into living with chronic illness. Some of these philosophical theories adopt a view of human beings that has been criticized for its focus on reason and consciousness and its disregard of the social reality people are embedded in [START_REF] Hall | The question of cultural identity[END_REF][START_REF] Tronto | Moral boundaries: A political argument for an ethic of care[END_REF]Mackenzie and Stoljar, 2000a). Although this criticism is deserved, we think the conceptual clarity of these theories may still help to conceptualize autonomy in the context of chronic physical illness, particularly in combination with the other theories that will be discussed.

First, we discuss the difference between negative and positive autonomy, an important classifi cation of contemporary philosophical theories of autonomy. Next, we discuss four philosophical theories of autonomy in greater depth. The fi rst theory [START_REF] Feinberg | Harm to self: The moral limits of the criminal law[END_REF] discusses prerequisites for autonomy, while the three other theories [START_REF] Dworkin | The theory and practice of autonomy[END_REF][START_REF] Dworkin | The concept of autonomy[END_REF][START_REF] Frankfurt | Freedom of the will and the concept of a person[END_REF][START_REF] Agich | Autonomy and long-term care[END_REF]Mackenzie and Stoljar, 2000a) focus primarily on the conceptualization of autonomy. The second [START_REF] Dworkin | The theory and practice of autonomy[END_REF][START_REF] Dworkin | The concept of autonomy[END_REF][START_REF] Frankfurt | Freedom of the will and the concept of a person[END_REF] has an individualistic, psychological focus, while the third [START_REF] Agich | Autonomy and long-term care[END_REF] and fourth (Mackenzie and Stoljar, 2000a) put more emphasis on social conditions and practical circumstances. We relate each theory to social scientifi c perspectives on living with chronic illness, particularly social models of disability and symbolic interactionism. Furthermore, we relate each theory to the case of Anne, which is described in the next section. Finally, we suggest how autonomy might be conceptualized in the context of chronic physical illness and we briefl y discuss some additional critical views on this concept of autonomy.

The case of Anne

Anne is a middle-aged woman who is married and has two sons away at college. Anne worked as a head nurse at the local hospital, when she was diagnosed with emphysema a few years ago. Emphysema is a lung disease that is characterized by shortness of breath and that gets worse gradually. At fi rst, Anne continued her job full time with the support of her colleagues, but then she got to the point where she was so tired when she got home from work that all she could do was eat and go to bed. Her husband and sons took a larger share in housekeeping, but eventually, Anne decided to work shorter hours. It was tough on her, because she had to give up her post as a head nurse. Last year, Anne's company doctor told her that she had to quit her job entirely. Anne felt pushed out and had diffi culty reconciling to disability. She therefore volunteered as a counselor for people with health: 12(3) a terminal illness because she expected that, as a counselor, she could truly be there for patients, which she often had felt she could not when she was head nurse. Today, the positive feedback from patients and colleagues gives Anne much satisfaction and she enjoys counseling.

How might autonomy be understood in Anne's case? Is it impinged on, because she fi rst lost her position as head nurse and then her entire job? Or is this a prime illustration of autonomy, because Anne makes her own decisions, takes initiative and fi nds a solution?

Negative and positive autonomy

An important classifi cation of contemporary philosophical theories of autonomy is the distinction between negative and positive autonomy, which was fi rst made by [START_REF] Berlin | Four essays on liberty[END_REF]. While negative autonomy refers to non-interference, positive autonomy refers to actively shaping one's life and identifying with fundamental values. It is about being in charge of life [START_REF] Berlin | Four essays on liberty[END_REF].

In negative conceptualizations of autonomy, a person is autonomous if no one interferes with his/her actions. In the context of chronic illness, concerned family members who prevent certain activities or, in the case of institutionalization restrictive regulations, may impinge on negative autonomy. In the case of Anne, negative autonomy is not impinged on by working shorter hours, because no one interfered to make this happen. It is however impinged on by the eventual job loss, because the company doctor ordered this.

Since negative autonomy may be promoted by minimizing interference, it is compatible with medical models of disability, which present disability as an individual attribute, which is caused by disease and may be solved by medical intervention [START_REF] Barnes | Exploring disability: A sociological introduction[END_REF]. Furthermore, it is in line with a particular version of social models of disability, the minority group approach. This approach considers people with disabilities as a minority group whose civil rights must be protected by anti-discrimination legislation. Discrimination is an impingement on negative autonomy, because people are hindered in their actions by others [START_REF] Bickenbach | Models of disablement, universalism and the international classifi cation of impairments, disabilities and handicaps[END_REF].

Generally speaking though, negative autonomy is not compatible with social models of disability because these models argue that interference may be enabling as well as disabling. In particular, social models contend that restrictions and opportunities exist side by side, because the material and social organization of the environment varies in the degree in which the needs of people with a chronic illness are taken into account [START_REF] Bickenbach | Models of disablement, universalism and the international classifi cation of impairments, disabilities and handicaps[END_REF]. Hence, a social model might for example reason that when Anne lost her position as head nurse her autonomy could have been preserved if her employer had assigned another head nurse to share the position with Anne part time.

In positive conceptualizations of autonomy, a person is autonomous if she actively shapes her life and identifi es with fundamental values. In Anne's case, positive autonomy is impinged on because Anne does not succeed in holding down her job although she wants to work. Positive autonomy is compatible with social and socio-medical models of disability. The latter relate restrictions in activities and social participation to an interaction between person, disease and environment (e.g. [START_REF] Verbrugge | The Disablement Process[END_REF][START_REF] Who | The international classifi cation of functioning, disability and health -ICF[END_REF]. Hence, in this view, Anne loses her job because she has emphysema and both she and her employer do not adapt to this circumstance adequately.

Positive autonomy may be promoted by increasing people's opportunities to arrange their lives. As mentioned earlier, the minority group version of the social model wants to create equal opportunities by means of anti-discrimination legislation. Another version of the social model calls for universalizing disability policy: rather than focusing on special needs, policy should acknowledge the wide variation between people and commit to 'universal design, not merely for public buildings and transportation, but across the board for housing, workplaces and other human environments' [START_REF] Bickenbach | Models of disablement, universalism and the international classifi cation of impairments, disabilities and handicaps[END_REF](Bickenbach et al., : 1183)).

As mentioned briefl y in the introduction, the difference between negative and positive autonomy is also relevant to the relationship between autonomy and dependence. [START_REF] Gignac | A conceptual model of independence and dependence for adults with chronic physical illness and disability[END_REF] distinguish four types of dependence/independence: dependence (needs and receives assistance); imposed dependence (does not need, but receives assistance); independence (does not need and does not receive assistance); and non-independence (needs, but does not receive assistance). As mentioned earlier, we might also think of people as interdependent, that is, dependent on each other, rather than distinguishing between dependence and independence [START_REF] Tronto | Moral boundaries: A political argument for an ethic of care[END_REF]). Anne's case has elements of dependence (her colleagues and family provided assistance that Anne needed), independence (Anne does not need and does not receive assistance to do her volunteer work), non-independence (Anne did not receive the assistance she needed to keep her paid job in general and her position as head nurse in particular) and interdependence (Anne's family, patients and colleagues depend on her support, care, instructions and so on).

In negative conceptualizations, dependence, imposed dependence and interdependence are impingements on autonomy (interference), while independence and non-independence are not (non-interference). Put differently, in negative conceptualizations of autonomy, it matters whether people receive assistance and not whether they need it. If people do not receive the assistance they need, the environment may not be enabling, but what matters here is that it is not interfering either.

In positive conceptualizations, independence, dependence and interdependence are compatible with autonomy, while non-independence is not. Imposed dependency is compatible if it is self-imposed. [START_REF] Baltes | The many faces of dependency in old age[END_REF] health: 12(3) has called this self-regulated dependence: people accept dependence in certain domains in order to have enough energy for other domains that they value more. In other words, in positive conceptualizations of autonomy, it matters whether people get what they need.

In summary, chronic illness may impinge on both negative and positive autonomy, but the circumstances under which this occurs differ. Negative autonomy is impinged on if someone receives assistance (regardless of whether or not she needs it), while positive autonomy is impinged on if someone does not receive the assistance she needs. Generally, we would argue for a positive conceptualization of autonomy in the context of chronic physical illness, because it takes account of people's actual needs and is compatible with social models of disability and with the assumption that people are interdependent. Despite this compatibility, not all positive philosophical theories of autonomy actually discuss autonomy in a social context, as the discussion of split-level theory below will show. First, we turn to [START_REF] Feinberg | Harm to self: The moral limits of the criminal law[END_REF], who has presented a model of prerequisites for autonomy, which includes both positive and negative elements.

Prerequisites for autonomy

According to [START_REF] Feinberg | Harm to self: The moral limits of the criminal law[END_REF], people are autonomous if they display a certain blend of virtues, including authenticity, identifi cation, initiative and responsibility for self. This condition of autonomy depends on three necessary -but not suffi cient -prerequisites. First, people need the capacity to govern themselves, which is determined by the ability to make rational choices. Second, people need the right to be a sovereign authority. In order to acquire this right, the capacity to govern oneself should be above a certain threshold. Third, people need opportunities, which depend on luck according to Feinberg. It seems that all three prerequisites for autonomy might be affected by chronic illness. First, diseases like Alzheimer's disease or stroke might affect one's capacity. In these cases, someone may not only experience difficulty in refl ecting on what they want, but also in making their wishes known to others (impingement on positive autonomy). Pain or fatigue may also infl uence decision making. Second, the right to be a sovereign authority is at stake if people's legal capacity is questioned or if people have to submit to restrictive regulations in institutions (impingement on negative autonomy). Third, if we extend Feinberg's theory using models of disability, opportunities may be restricted as a result of disease and/or the material and social organization of the environment, which may not adequately take into account the needs of people with a chronic illness (impingement on positive autonomy).

In our example, Anne has both the capacity and the right to be a sovereign authority, but there is a lack of opportunity to hold down her job. This may be explained differently, depending on the model of disability used.

From a medical point of view, Anne might not be fi t enough physically to hold down her job. Social models might focus on the efforts of Anne's employer to meet her special needs. A socio-medical model might consider not only medical and social, but also personal factors like activity accommodation and coping.

Feinberg's model does not reveal clearly in which circumstances Anne might actually be considered autonomous, because Feinberg does not specify the exact composition of characteristics that make up the condition of autonomy. Might Anne be considered autonomous because she takes initiative and responsibility for herself, fi rst by reducing her working hours and then by volunteering as a counselor? Or might she not be considered autonomous because she does not identify with disability? The theories in the following three sections present positive conceptualizations of the condition of autonomy.

Split-level theory

Split-level theory consists of a theory of the person developed by [START_REF] Frankfurt | Freedom of the will and the concept of a person[END_REF] and a theory of autonomy developed by [START_REF] Dworkin | The theory and practice of autonomy[END_REF][START_REF] Dworkin | The concept of autonomy[END_REF]. [START_REF] Frankfurt | Freedom of the will and the concept of a person[END_REF] bases his theory on the capacity of persons to refl ect on what they want to be motivated by. This capacity leads to a hierarchical order of desires. First-order desires express what a person wants. A person can have multiple desires, which may possibly, but not necessarily confl ict. Not all desires result in action. If a fi rst-order desire leads to action, it is called an effective desire. For example, a person with congestive heart failure may have both the desire to take the bus to work as usual, and to travel to work by bicycle, as advised by her doctor to get regular exercise. If the person takes the bus, this is the effective desire. Second-order desires express what a person wants to want, that is, what she wants to be motivated by. They are formed through rational, refl ective self-evaluation. The person in the example might decide that she wants to be motivated by the desire to travel by bike, lifting this desire to the second order. [START_REF] Dworkin | The theory and practice of autonomy[END_REF][START_REF] Dworkin | The concept of autonomy[END_REF] adds to this, stating that second-order desires do not have to be unique or attained uninfl uenced, but they must undergo critical scrutiny in order to become one's own. The desires that people have are theirs, but not automatically their own. For example, health promotion programs may make people feel that they should exercise regularly, adhere to a balanced diet or quit smoking, but in order to make these kinds of lifestyle modifi cations second-order desires, people have to ask themselves whether these modifi cations are something that they really want themselves. It is crucial to autonomy that people possess 'the capacity to raise the question of whether I will identify with or reject the reasons for which I now act' (Dworkin, 1988: 15). This evaluation must occur free from subverting infl uences, such as manipulation or coercive persuasion. Spouses who keep on about regular exercise, and authoritarian medical professionals health: 12(3) who tell people to modify their lifestyle, present obstacles to an evaluation made in freedom.

Dworkin further argues that autonomy is not a mere refl ective notion, but also includes the attempt to make the outcome of evaluation effective. This means that people should try to act for the reasons for which they want to act (e.g. try to travel to work by bike). Dworkin emphasizes that it is the attempt that matters, not whether people actually succeed in acting for the reasons for which they want to act. One of Dworkin's arguments is that people might otherwise realize autonomy via intuitively wrong ways. In particular, Dworkin feels that there is something wrong with scaling down second-order desires in order to become autonomous.

The issue is particularly relevant in the context of chronic illness because scaling down is exactly what may happen in response to chronic illness. [START_REF] Charmaz | The body, identity and self: Adapting to impairment[END_REF] describes how people move up and down an identity hierarchy in response to the course of illness and their social situations. An identity hierarchy consists of identity goals, which represent what or who a person wants to be, strives or hopes for, either explicitly or implicitly. Moving up and down the hierarchy, people weigh different identities against each other and determine which to give up in order to retain others (identity trade-offs). Generally, deterioration in physical functioning will force people to scale down their identity goals, while an improvement in physical functioning will encourage people to scale up their identity goals. Some people are hesitant to scale down identity goals, because they underestimate their decline, fi nd the resulting identity losses too great or have strong feelings of commitment and responsibility. Other people are hesitant to scale up identity goals, because they fear failure or relapse [START_REF] Charmaz | The body, identity and self: Adapting to impairment[END_REF].

According to Dworkin, people who scale down identity goals are not autonomous. On closer reading, however, Dworkin seems to be particularly concerned that people might scale down second-order desires too easily in order to realize autonomy. Dworkin would have diffi culty accepting that a person who has decided she wants to travel to work by bike realizes autonomy by taking the bus at the fi rst sight of rain and then deciding that traveling by bike is not what she wants after all. However, Dworkin would probably agree that scaling down second-order desires might produce autonomy after critical self-evaluation, as is the case in identity trade-offs. For example, a person may discover that she could really use a shower when she travels to work by bike, but that there are no facilities. After carefully considering whether she really wants to travel to work by bike under these circumstances, she might decide that she would rather travel by bus.

Applying Dworkin's argumentation to the case of Anne, what Anne does is not really all that relevant in the end, as long as she does it after careful consideration. Whether she would have volunteered as counselor or protested against the loss of her job or decided that she wanted to spend her limited energy on, say, social activities, she is considered autonomous if she identifi es with the reasons for her actions.

Actual autonomy

In contrast to [START_REF] Feinberg | Harm to self: The moral limits of the criminal law[END_REF] and [START_REF] Dworkin | The theory and practice of autonomy[END_REF][START_REF] Dworkin | The concept of autonomy[END_REF], [START_REF] Agich | Autonomy and long-term care[END_REF] approaches autonomy as embedded in the practical, social world of everyday life. Rather than making autonomy an abstract ideal, Agich focuses on actual autonomy. He is critical of the idea proposed by split-level theory, that second-order desires are formed through refl ective self-evaluation. According to Agich, higher-order principles (i.e. Dworkin's second-order desires) are not a top-down affair, as claimed by split-level theory, but the product of a two-way process. Higher-level principles are constructed from actual experiences and choices; they guide future actions. These actions in turn infl uence the higher-level principles. Rather than an end-state, autonomy should be conceived as an ongoing process: 'To be an autonomous person in the world is to develop … as an integrative process of accommodating oneself to new circumstances and adapting those circumstances to one's unique structures of meaning' (Agich, 1993: 106).

Certainly, Agich does not deny that people might refl ect on who they are or what guides them. In particular, existential crises, life transitions or sickness may bring people to consciously consider such matters. But this is not how autonomy is displayed in daily life. In daily life, people display autonomy through constant adjustment of their intentions to the opportunities and limitations encountered in their interaction with the world. A sense of self can be derived from people's actual choices, motivations and experiences, which can be expressed in terms of higher-level principles. These principles guide future action and are subject to change. Agich's point is that these principles are the product of real life, rather than critical refl ection. Autonomy is an ongoing process in which people develop, express and confi rm their identity in interaction with the world around them.

Agich's presentation of autonomy including accommodation and identity development shows close parallels to symbolic interactionist work on biographies [START_REF] Bury | Chronic illness as biographical disruption[END_REF][START_REF] Corbin | Unending work and care: Managing chronic illness at home[END_REF]. According to this work, people experience a biographical disruption when they are confronted with chronic illness, meaning that they can no longer rely on assumptions and behaviors that they formerly took for granted [START_REF] Bury | Chronic illness as biographical disruption[END_REF]. Restoring a biography requires the fulfi llment of four tasks, which occur simultaneously and interact with each other. People have to integrate the course of illness into their biography, they must reach a certain degree of understanding and acceptance of the consequences of illness, they must reconstruct their identity, and fi nally, they must give their biography a new direction (Corbin and Strauss, 1988: 68).

health: 12(3)

Both the symbolic interactionists and Agich assume an ongoing process, in which events and experiences in daily life may disturb people and their identities, but may then be integrated and guide future actions. In this view, then, changing higher-order principles is a valid and natural way to establish correspondence between higher-order principles and actions. In fact, from Agich's point of view, it is a necessary course to restore autonomy, since Agich considers accommodating to circumstances as necessary for the process of autonomy.

In Anne's case, autonomy would thus involve an adjustment of her intentions to disability. Anne would have to reconstruct her identity, incorporating disability. She actually does this by volunteering for work as a counselor, which enables her to continue to work with patients despite disability.

Relational autonomy

The developmental nature of autonomy and its embeddedness in the social world, which [START_REF] Agich | Autonomy and long-term care[END_REF] stresses, have also been addressed by feminist scholars (Mackenzie and Stoljar, 2000a). They criticize the inherent individualism and rationalism of prevailing conceptualizations of autonomy. In contrast, they conceptualize autonomy relationally, aiming to express that people are not only rational, but emotional and creative as well, and that they are embedded and socialized in a social and historical context in which they depend on each other. Through social relationships and in networks, people constitute their identity and fi nd recognition of it. Only through the social context, can autonomy take shape [START_REF] Mackenzie | Introduction: Autonomy refi gured[END_REF].

Unlike [START_REF] Agich | Autonomy and long-term care[END_REF] and like split-level theory [START_REF] Dworkin | The theory and practice of autonomy[END_REF][START_REF] Dworkin | The concept of autonomy[END_REF][START_REF] Frankfurt | Freedom of the will and the concept of a person[END_REF], the relational scholars attribute a central place to conscious refl ection in the realization of autonomy. However, unlike split-level theory and like symbolic interactionism [START_REF] Corbin | Unending work and care: Managing chronic illness at home[END_REF][START_REF] Frank | The wounded storyteller: Body, illness and ethics[END_REF], they contend that refl ection does not necessarily have to be rational, but may also involve emotions, creativity or imagination [START_REF] Friedman | Autonomy, social disruption, and women[END_REF][START_REF] Mackenzie | Imagining oneself otherwise[END_REF]. Symbolic interactionism too describes how people make sense of and give meaning to their lives by daydreaming, imagining and constructing stories [START_REF] Corbin | Unending work and care: Managing chronic illness at home[END_REF][START_REF] Frank | The wounded storyteller: Body, illness and ethics[END_REF]. According to the relational scholars, refl ection is not primarily an individual matter, but a process that takes place through interaction and communication. Autonomy thus is based upon dialogical hermeneutic procedures. This entails a view of autonomy as self-development through dialogue [START_REF] Widdershoven | Care, cure and interpersonal understanding[END_REF].

In the case of Anne, relational autonomy is illustrated by the support of family and colleagues who help Anne, fi rst, to continue her job and then to make a tough decision and give up her position as head nurse to reduce work hours. Relational autonomy is impinged on when Anne hears that she will lose her job and feels pushed out. The company doctor does not take into account Anne's circumstances and does not offer any support.

Relational autonomy requires that Anne can take account of her feelings in this diffi cult situation and get support from others to fi nd a solution that fi ts her needs. One might question whether the situation could not have been changed in such a way that Anne would have been able to continue working at the hospital. After this phase, Anne is able to fi nd a new way of living, in interaction with others. The position as a counselor seems an opportunity for Anne to develop herself, and the positive feedback from colleagues and patients seems to reinforce her satisfaction with the position.

Discussion

We have presented a selection of philosophical theories of autonomy in relation to the context of chronic physical illness as described by social scientifi c perspectives. The emphasis was on positive autonomy, although chronic illness might also impinge on negative autonomy. However, a negative conceptualization of autonomy seems unsatisfactory in the context of chronic illness because it focuses on impingements on autonomy due to interference from the environment. In contrast, a positive approach is compatible with interference provided it meets people's needs, although not all positive theories of autonomy take account of the social and practical context of daily life in which people depend on each other, as split-level theory illustrates. [START_REF] Dworkin | The theory and practice of autonomy[END_REF][START_REF] Dworkin | The concept of autonomy[END_REF], [START_REF] Agich | Autonomy and long-term care[END_REF] and the relational scholars (Mackenzie and Stoljar, 2000a) all present positive conceptualizations of autonomy, but Dworkin focuses on an individual, psychological process, while Agich and the relational scholars focus on accommodation to the social, practical world of daily life. According to Dworkin, critical refl ection on desires is the key to autonomy. Hence, as long as chronic illness does not affect the capacity to refl ect, it does not threaten autonomy. Agich argues, that chronic illness may change someone's daily reality in such a way that she can no longer identify with it. Autonomy then requires accommodation to the changed circumstances.

When accommodation is diffi cult, Agich suggests that the circumstances might need to be changed to facilitate identity development. Likewise, social and socio-medical models of disability point out, that a restrictive material and social organization of the environment may restrict opportunities. Hence, reorganization of the environment might increase opportunities and make accommodation superfl uous.

If this is so, then Dworkin is right to express his concern that people might scale down second-order desires too easily. Not because people might scale down desires without careful consideration, but rather because people might accommodate to opportunity ranges that are unnecessarily restrictive. In the case of Anne, allowing part-timers to share the position of head nurse might have enabled Anne to keep her position as head nurse while she worked at the hospital.

health: 12(3)

The importance of opportunities is recognized by [START_REF] Feinberg | Harm to self: The moral limits of the criminal law[END_REF], who presents opportunities as a necessary prerequisite of autonomy. Feinberg's model offers conceptual clarity, putting the condition of autonomy at the center, with capacity, right and opportunities as prerequisites. However, it also has three important shortcomings. First, it ignores the social context people are embedded in, because it presents opportunities simply as the result of bad luck. As already indicated above, this might be 'fi xed' in the context of physical chronic illness by extending Feinberg's model with a model of disability. We would suggest a socio-medical model, because it considers the interplay between person, disease and environment instead of focusing on one aspect. Second, Feinberg's model narrows the capacity to direct one's life down to the ability to make rational choices, which feminist scholars, among others, have long agitated against (Mackenzie and Stoljar, 2000a). And third, it does not specify the exact composition of characteristics that make up the condition of autonomy.

The second and third shortcoming of Feinberg's model might be fi xed by combining it with the theories of the condition of autonomy (split-level theory, actual autonomy and relational autonomy). In all these three theories, achieving correspondence between higher-order principles and actions appears to be the 'ultimate goal' of autonomy. However, split-level theory still has in common with Feinberg's model an ignorance of the social context and a focus on rational refl ection. Also, its conceptualization of autonomy implies that autonomy requires that people know their desires. Yet this assumption has been criticized from different perspectives. Psychoanalysts, for instance, argue that persons do not know all their desires, because unconscious psychological processes may be at work. Marxist structuralist scholars argue that persons can never truly know what they want, because they are always shaped by circumstances formed by former generations. And poststructuralist scholars, for example, add to this that the meaning of an identity can never be completely fi xed, as meanings in general never are, because they are expressed in language, which is a social system in which meanings arise and vary in relation to others [START_REF] Hall | The question of cultural identity[END_REF].

These points of critique have led scholars to reject the idea of autonomous persons altogether. Alternatively, the relational scholars and Agich present conceptualizations of autonomy that meet those critiques to some extent. Relational and actual autonomy can only take shape through the social, historical environment. Furthermore, the relational scholars broaden the capacity to direct one's life from rational refl ection to involve emotions, creativity and dialogue as well, while Agich argues that conscious refl ection is possible but not the usual way in which principles are developed and modifi ed. Higher-order principles are rather the product of choices and experiences in real life.

We therefore suggest conceptualizing autonomy in the context of physical chronic illness as correspondence between what people want their lives to be like and what their lives are actually like. To achieve this correspondence, people need to develop principles expressing what they want their lives to be like. Furthermore, they need the opportunities to arrange their lives. People generally develop these principles in daily life, as they interact, make choices and gather experiences. Refl ection -involving ratio, emotions, creativity, imagination or dialogue -can, however, also play a role in the process of developing and modifying principles. If chronic illness has disturbed the correspondence between what people want their lives to be like and what their lives are actually like, the correspondence may be restored either by increasing the opportunities to arrange life or by adjusting how one wants life to be arranged. The grounds for the second approach might be questioned, fi rst, if people have not carefully changed what they want, and second, if reorganization of the material and social environment would have made it unnecessary to adjust principles about the arrangement of life.

several studies focusing on disease management in primary care, of elderly patients with diabetes type 2, asthma, chronic obstructive pulmonary disease and other diseases. Currently, he is also involved in observational studies on the contextual determinants of health, the role of depression in physical decline and decline in social participation.

Author biographies

godelief m.j. mars is a PhD candidate at the Care and Public Health Research Institute of Maastricht University, the Netherlands, with a background in sociology. The aim of her PhD project is to develop measurement instruments for autonomy and social participation of older adults with a chronic illness, particularly diabetes mellitus type 2 and chronic obstructive pulmonary disease. g.i.j.m. (ruud) kempen is Professor of Social Gerontology at Maastricht University, the Netherlands. Originally trained as a medical sociologist, he became interested in research into psychosocial and environmental determinants of health problems, social participation, independency and quality of life among older persons, and the development and evaluation of related innovative health care programs to slow down processes of disablement in older persons. He is also interested in measurement issues in the area of health and quality of life among older persons. guy a.m. widdershoven is Professor of Ethics of Health Care at Maastricht University, the Netherlands. His main theoretical interest is to contribute to the development of practice-oriented approaches to bioethics (hermeneutic ethics, narrative ethics, ethics of care). He supervises research projects on ethics of chronic care (care for the elderly, psychiatry, care for people with a mental disability). He also supervises projects on ethical aspects of care around the end of life, particularly palliative care and euthanasia. peter p.m. janssen (deceased) was Associate Professor of Work and Health at the Department of Health Organization, Policy and Economics at Maastricht University, the Netherlands. With a background in psychology, he started his academic career with a dissertation concerning the issue of relative deprivation among highly educated men in mid-career. After that, his research focused on issues such as stress model refi nement, burnout, workload and work rehabilitation. He coordinated several PhD projects and contract research projects on work and health issues. jacques t.m. van eijk is Professor of Medical Sociology at Maastricht University, the Netherlands. His current research interests are 'coping with chronic disease', the role of depression in the course of chronic disease, disablement, self-management and social participation of patients with chronic disease. He was and is engaged in


Richard Hallam

Paul Ashton

Katerina Sherbourne

Lorraine Gailey

Persons with acquired profound hearing loss (APHL): how do they and their families adapt to the challenge?

Keywords: adjustment, coping, deafened, intimate relationships, psychosocial impact

come

Introduction

It would be surprising if acquired profound hearing loss (APHL) did not present a severe challenge to the maintenance of close relationships with partners and close family members. People affected by APHL have formerly been able to communicate normally; but, later in life, their hearing deteriorates, gradually or suddenly, to the point that it is no longer usable for spoken communication. The condition is relatively rare -perhaps between 1 in 250 and 1 in 500 of the population [START_REF] Davis | Hearing in adults[END_REF]) -but prevalence is diffi cult to estimate because functional hearing is not solely dependent on auditory thresholds. Mild to severe acquired hearing loss (AHL) is far more prevalent and its associated disabilities and handicaps have been more thoroughly investigated. People with AHL usually strive to preserve their 'normally hearing' identity and to function at parity with the hearing majority [START_REF] Hétu | Qualitative analysis of the handicap associated with occupational hearing loss[END_REF][START_REF] Hallberg | Hearing impairment, coping, and consequences on family life[END_REF]. This is usually possible with the help of technical aids, although there is often a cost associated with the effort to keep up. People with APHL may get some benefi t from hearing aids but their ability to portray themselves as normal communicators is severely compromised. Effi cient communication may be achieved in one-to-one situations with the help of lip-reading, signing or a cochlear implant but the effects of the hearing impairment are rarely invisible. For this reason, research on AHL is only partially applicable to the situation of people with APHL who have for some time been recognized as a distinct group (e.g. [START_REF] Luey | Between worlds: The problems of deafened adults[END_REF][START_REF] Hogan | Hearing rehabilitation for deafened adults: A psychosocial approach[END_REF].

Although there have been no systematic studies of the impact of APHL on close relationships, the evidence that exists paints a negative picture. [START_REF] Hogan | Hearing rehabilitation for deafened adults: A psychosocial approach[END_REF] reported that there was a break-up of a signifi cant relationship in 18 of his sample of 38. [START_REF] Schlau | I did not die, I just can't hear': A grounded theory study of acquired deafness[END_REF] collected qualitative data via e-mail from 24 late-deafened adults and reported that the vast majority of comments about the response of family members were negative.

There have been some surveys of people with moderate to severe hearing loss, using questionnaire and interview methods. [START_REF] Kerr | Acquired deafness: A multidimensional experience[END_REF] found that one in 10 of their respondents felt that deafness had almost destroyed their lives, although the impact on close relationships was not addressed directly. [START_REF] Thomas | Acquired hearing loss: Psychological and social implications[END_REF] compared a mixed severity hard-ofhearing group (N = 211) with a group whose loss exceeded 60 dB HL (N = 88). The rate of separation or divorce was similar to population norms in the former (7/211, 3.3%) but far greater for the latter (13/88, 14.7%). A subgroup with severe hearing loss in the mixed sample (> 70 dB HL, n = 23) were also more likely to be separated or divorced (13% compared to 2% in the remainder of the sample). Fifty-three per cent of the severe group reported that hearing impairment had affected their marriage as against only 24 per cent in the remainder. There was evidence of considerable strain in a sample of 27 families who were interviewed, and several respondents acknowledged that they coped only with family support.

Research on the impact of AHL on close relationships has been conducted mainly on outpatients [START_REF] Stephens | Effects of hearing impairment on the patient's family and friends[END_REF] or on people with progressive losses due to occupational noise exposure [START_REF] Hétu | The impact of acquired hearing loss on intimate relationships: Implications for rehabilitation[END_REF][START_REF] Hallberg | Coping with noise-induced hearing loss: Experience from the perspective of male victims[END_REF] or ageing [START_REF] Donaldson | Older people with hearing impairment: A literature review of the spouse's perspective[END_REF]. In general, this research has underlined the need for professionals to focus on the whole family to help them to adjust [START_REF] Hétu | The impact of acquired hearing loss on intimate relationships: Implications for rehabilitation[END_REF]. The stresses experienced by the impaired and unimpaired members of a close relationship are somewhat different and there are considerable opportunities for misunderstanding, blame and confl ict. [START_REF] Jones | Words apart: Losing your hearing as an adult[END_REF] found that in the 29 families they studied, 40 per cent felt that the relationship had changed negatively as a result of hearing loss. In only 4 per cent had it changed for the better.

On the basis of several qualitative studies of people with AHL, [START_REF] Hallberg | Hearing impairment, coping, and consequences on family life[END_REF] concluded that participants coped either by controlling the social scene or by avoiding it. Hearing people were frequently neither understanding of, nor sympathetic to, any diffi culties arising. Males tended to deny or minimize their disabilities in order to protect their self-image and avoid being defi ned as deviant. Some partners backed up this strategy; others mediated on behalf of their spouse, while another group distanced themselves from the relationship. One might expect to fi nd similar coping patterns in people with profound hearing loss; however, their situation is more challenging and the options of ignoring and minimizing are not available to them in most situations.

The aim of the present study was to explore the ways people with APHL, and a close family member whom they had nominated, adapted to the challenge that profound hearing impairment had imposed on their relationship. The participants were interviewed separately, face-to-face, in their own homes. The interview was loosely structured but followed a pre-defi ned set of topics, of which a principal one was the effect on relationships. Other topics covered in the interview do not form part of this analysis. Data of a general nature were also collected by questionnaire.

Methodology

Participants

The sample consisted of 27 deafened people, 13 male and 14 female, of whom 17 had, at some time, attended a one-week residential rehabilitation course run by the LINK Centre for Deafened People, eight were recruited from respondents to a Web-based survey on APD (Hallam et al., submitted) and two were recruited through contacts at LINK. The rehabilitation programme at LINK (described in [START_REF] Sherbourne | Intensive rehabilitation programmes for deafened men and women: An evaluation study[END_REF], contains a large health: 12(3) educational component and, while addressing the impact on family members, is not a family therapy intervention. Participants for the study volunteered after receiving full information about its nature. Two respondents were excluded -one who failed to nominate a family member and another who nominated a neighbour. In 19 cases the second informant was a spouse/ partner, in six cases a parent, sibling or daughter. The latter six informants were, in nearly all cases, the closest family member of an unmarried or widowed participant. Demographic and other details of the participants with APHL are shown in Table 1.

The defi nition of APHL used by LINK is an inability to follow speech by using sound alone (aided or unaided). The participants recruited through the Web survey (see Hallam et al., 2003) either could not hear sounds at all or endorsed three out of four items indicating the most severe level of hearing loss, as used in a United Kingdom government survey of disabilities [START_REF] Martin | The prevalence of disability amongst adults: OPCS surveys of disability in Great Britain Report 1. London: Her Majesty's Stationery Offi ce[END_REF]. It was not our purpose to obtain a representative sample but to include participants who could provide a wide range of experience and perspectives. However, the extensive LINK database enabled us to select participants of all ages; we also ensured that gender, ethnicity, socio-economic status and urban and rural location approximated population norms. Their demographic, medical and audiological characteristics closely mirrored the results of our Web-based survey of 95 people with APHL.

It seemed likely that participants' individual circumstances and characteristics would infl uence the impact of APHL but our focus was not on families taken separately. It aimed to explore typical stresses, modes of adjustment and patterns of interaction. One relevant individual difference variable is suddenness of onset -13 participants had become deafened in a matter of weeks. Other variables are age, gender, employment status, the pre-existing personality of the hearing impaired person and family member,

Research methodology

The phenomena of interest were patterns of interaction and interpersonal perceptions in real settings, and so we adopted a critical realist approach for which grounded theory seemed most suited [START_REF] Strauss | Basics of qualitative research: Grounded theory procedures and techniques[END_REF][START_REF] Dey | Grounded theory[END_REF]. We were concerned with observable processes rather than a linguistic or discursive interpretation of the way our participants construed their relationships. Our approach to coding followed grounded theory guidelines and worked upwards from open (preliminary, descriptive) codes, to axial (conceptually connecting) codes and fi nally to selective codes (core categories that relate to a broader conceptual framework).

As the focus of our study was on family relationships, we did not view the adjustments as 'belonging' exclusively to the individual with hearing loss. The pairs of participants spoke about themselves and each other. We were interested in generic processes of adjustment rather than treating each dyad as a case study in adjustment. Strategies of adjustment with positive and negative effects were identifi ed but, beyond a simple classifi cation of the overall quality of the relationship (see later), we did not wish to characterize each relationship as an example of good or poor adjustment. For these reasons, we use quotations from either the person with APHL (D) or family member (F) to illustrate the categories we develop. Where there is a clear difference of perspective, based on being hearing impaired or normally hearing, we attempt to integrate the relevant categories in our general framework. We also saw no sound reason for separate analyses of married couples versus other kinds of close family relationship. All participants nominated their family member and presumably entrusted them to divulge confi dential details of their relationship.

Interview process

Participants were interviewed separately at home for 60-90 minutes each. We consider it to be important that the interviewer (PA) has a long-standing moderate to severe acquired hearing loss and many years' experience of the kinds of problems that a person with AHL may encounter. We assume that his fi rst-hand knowledge regarding the problems of communication, his association with the LINK charity, and to a large degree, his shared experience has enhanced his credibility, facilitated empathy and eased the exploration of relevant issues. If anything, his sympathies would probably have been more with the hearing impaired person than the family member, although we had no evidence of a bias.

The interview guide listed a number of open-ended questions, beginning with a history of the development of hearing loss, help sought and the health: 12(3) impact it had had on social and family life. The questions were developed in consultation with staff at the LINK centre who have had long experience of working with adults with APHL. Some questions were directed to the relationship but the interview was kept conversational in tone to allow participants to enlarge freely on their feelings. Questions were available in written form but this was rarely needed. The interviews were recorded on a digital sound recorder and the fi les e-mailed to a transcriber who produced a typed transcript that was then e-mailed back to the two main researchers. Transcripts could be compared to the original sound recording where necessary.

Participants were at different stages in adapting to their changed circumstances. Some participants referred back to an earlier critical period, after which there was a gradual coming to terms with their deafness. This was especially true of participants who had received a cochlear implant, the effects of which were invariably benefi cial. Other participants were in the midst of a critical phase of adjustment and, in one case, in the process of a marital separation.

Process of analysis

The transcripts were entered as documents into the NVivo software package [START_REF] Richards | Using NVivo in qualitative research[END_REF]. Coding was discussed in meetings between the fi rst author and the interviewer in order to achieve a consensual interpretation of interview material. As the open codes were developed, on an interview by interview basis, all of them were retained for potential application to the next transcript. Aspects of the relationship emerged as one of the most prominent themes. As the analysis was ongoing, the number of codes expanded as the analysis progressed. Each line of the transcribed data was examined; open coding avoided conceptual interpretation and aimed to remain close in meaning to the words used by participants. Very few new codes were added towards the end of the analysis and so an acceptable degree of saturation seemed to have been achieved. When all transcripts had been coded, the open codes were compared, contrasted, refi ned and reduced in number through a process of re-reading of transcripts and discussion within the wider research team.

In order to make the analysis more manageable, the open codes relating to aspects of the relationship were separated from other themes such as events surrounding onset of APD, professional help received, response of friends and the general public, communication techniques and change in perception of self, habits or lifestyle. These other themes were subsequently analysed separately.

Conceptual coding

There were initially over 120 open codes for aspects of the relationship but when all transcripts had been coded, these were reduced in number. In a conceptual level of analysis, the relationship between the open codes was subjected to axial coding, informed by criteria of internal consistency, completeness and a theoretical interpretation that began to emerge from the analysis. A core category was also proposed (see below). Our general conceptual model will now be presented (see Figure 1) in order to prepare the reader for a more detailed presentation of the fi ndings.

We were aware that concepts of stress and coping had been applied to similar data (e.g. [START_REF] Jones | Words apart: Losing your hearing as an adult[END_REF]. These authors conceptualized adjustment from the point of view of the hearing impaired individual, that is, as changes in belief, behaviour or lifestyle designed to preserve the perception of control over normal functioning. We focused more on the family system as seen by pairs of family members. Adjustments were therefore seen as mutual, though not necessarily as complementary. The open codes were interpreted as refl ecting attempts to stabilize the relationship or as adjustments that became a source of further stress. It is clearly extremely diffi cult to attribute causality to specifi c elements in a family system. It is our conviction that problems of communication (and their social consequences) occasioned by APHL made a signifi cant contribution to changes in family relationships but these problems must be viewed as being in dynamic causal interaction with other factors. In our model, we have emphasized the role of social processes that establish the ground rules and motivation for individuals to maintain their commitment to each other and strive to make adjustments to an imposed diffi culty. There was insuffi cient evidence to describe the unique aspects of interactions within a family as this had been an incidental focus among others in the interview. However, when the analysis was complete, the transcripts of pairs of married participants were read in conjunction in order to make an informed judgement as to whether the relationship was 'stable' or showing signs of signifi cant confl ict or interpersonal stress. This enabled us to make some approximate comparisons with previously published quantitative data.

Categories were developed to describe different kinds of adjustment outcome, positive or negative. These outcomes were solutions to shared problems and do not refer to overall adjustment of the individual or family. The core category informing our concept of adjustment was the social construction of a committed relationship, that is, the generally accepted expectations of the duties, obligations and benefi ts to the partners of such a relationship, and the implicit norms governing behaviour within it. This core category enabled us to integrate the fi ndings but, as a theoretical construct, evidence for it was indirect. A sense of commitment to a relationship is, of course, compatible with open disagreement and less obvious forms of dissatisfaction. A relationship may be committed without necessarily illustrating effective mutual adjustments. We have interpreted many of the interactions between our informants as attempts to preserve aspects of a committed relationship or to compensate for factors that threaten it. In a few cases, the person with APHL and their close family seem to have formed a new commitment on the basis of a changed lifestyle and altered values.

Mutual positive adjustment was generally refl ected in shared coping strategies such as an agreement to avoid socializing in certain group situations. Strategies of this kind may have had short-term positive benefi ts but less fortunate consequences in the longer term. The benefi ts of avoidance depended on the nature of the relationship, and any signifi cant degree of social avoidance could have produced a negative outcome for certain couples. Individual outcomes appear to have resulted from a mismatch or unfortunate interaction between the adjustments that each person made. Sources of stress also arose as an indirect consequence of the hearing impaired person's auditory limitations, especially restrictions on leisure activities, loss of social status or unemployment.

Illustration of the model

In what follows, pairs of participants are assigned the same number and identifi ed as D (deafened) or F (family member) e.g. 10D or 10F. A series of dots indicates that part of an excerpt has been omitted. Sentences in italics are spoken by the interviewer.

Auditory impairments

The analysis of these impairments was based on references to aural perception and they are seen as prompting the adjustments that participants employed. They are familiar from previous literature [START_REF] Thomas | Acquired hearing loss: Psychological and social implications[END_REF][START_REF] Jones | Words apart: Losing your hearing as an adult[END_REF][START_REF] Hallberg | A qualitative study of situations turning a hearing disability into a handicap[END_REF][START_REF] Morgan-Jones | Hearing differently: The impact of hearing impairment on family life[END_REF]) and will not be illustrated in detail. They are: (1) Quality of one-to-one communication is reduced as a result of a loss of speed and accuracy of verbal exchanges, in turn leading to a loss of spontaneity. (2) Ditto in group situations, impinging especially on family gatherings and social/leisure activities. (3) Poor voice modulation, especially in public places, leading to embarrassment, or giving the impression of an inappropriate emotional tone. (4) Failure to respond to everyday auditory signals, with consequences such as increased vulnerability to danger, substitution of a signal in a different sensory modality or reliance on others. (5) Intolerance of background sounds which are experienced as unpleasant or as interfering with spoken communication. ( 6) Inability to hear audio-visual equipment (e.g. TV) at normal volumes, with consequences for shared viewing and use of captioned material. ( 7) Altered experience of sound in general (e.g. distortion) and a negative effect of silence (absence of sound). ( 8) Finally, the effort of spoken communication, miscommunication and reduced social participation was interpreted as giving rise to negative emotional states such as irritability, tiredness, frustration or depression. The following quotations illustrate some of the impairments (it should be noted that we have to infer impairments from quotations that are, naturally enough, mainly about their psychosocial effects):

.… it is receding at speed, the whole quality of our interactions were being stripped away and I was fearful that I couldn't communicate with him …(3F) … I was fi nding groups of friends very hard by that point, I was tending very, very much to want to talk to people one at a time and meet people one at a time. (7D) … she has an assertive voice and quite ordinary things are said which, by the tone of them, makes them sound offensive. (6F) … so the loud telly, I found very, very stressful … (7F)

Pragmatic adjustments designed to deal with impairments

These were coded as adjustments to (1) spoken communication and (2) domestic, social and leisure activities. They can be regarded as practical manoeuvres designed to facilitate the business of daily life, to speed up spoken communication or reduce the need for it. The impact of these adjustments on relationships was not necessarily either good or bad; they have the potential to be perceived positively, as signals of support, or negatively, as a sign of personal inadequacy or negative social evaluation.

health: 12(3)

Adjustments to communication A common adjustment is simply for D to exclude him/herself or be excluded from conversations that would otherwise be effortful. A modifi ed form of this is for F to give D edited messages that would take too long to repeat fully. This adjustment impacts especially on initiating conversation, sharing gossip or partaking in social gatherings: 'So once you have spoken to someone for half an hour, you don't really repeat everything, so he probably gets frustrated that we just give him a little summary …' (24F).

F can compensate for the tendency for D to be excluded by making sure that D is included in a conversation or informed of its content, although this is not always successful:

Sometimes, depending on the situation, if it is important then I will sort of stop the person and say, 'oh, I am not sure if he has understood, just check with him' and sometimes he just feels too polite to just ask. (24F) So then I try and explain, by which time the moment has gone, and that makes him feel even worse. But you don't want him to feel left out so that is quite diffi cult. (23F) D can compensate for missing out on spoken communication by exerting greater control over a conversation or by generally taking charge. The following quotation illustrates how normal turn-taking is superseded: 'I don't think I talked about it to R. Although I would tell him about it!' (2D). In this extract, D explains why she is regarded as bossy:

Bossy towards who? Everybody, I have to be in control of things just so I know what is going on. (14D) Instead of communicating directly, D may ask F or others to mediate in order to make a statement, convey a message, use the telephone, listen out for a signal or repeat what has been said. While this can be a helpful adjustment in certain circumstances, it diminishes D's sense of agency and, in some cases, privacy: '… and the lost privacy of going to a doctor and having to take me with him' (2F). It also has the potential for miscommunication: '… I had to take messages and this was another point of friction' (6F).

D sometimes relied on children as mediators and felt this was wrong: 'I probably did say to him "can you keep an eye on the baby" "can you tell me if the baby is crying", you know, he was eight then and he's too young really' (1D). If D fails to hear, F may direct D's attention by means of touch. Often this is innocuous but it can be experienced as intrusive: 'I try not to shout and I try not to prod … but occasionally … it does turn into a prod and I hate it when I do that …' (20F).

Adjustments to domestic, social and leisure activities For routine tasks that require conversation, such as shopping, F may take them over as a matter of practical convenience. D may later fi nd that they would prefer to resume these tasks: 'Shopping, my husband is mostly doing … he would volunteer to do it on a Saturday, although he never used to. Although I'm trying to wrestle it back now …' (1D).

Social and leisure activities may be curtailed because they are no longer pleasurable: 'We stopped going out and I get quite worked up about the idea, if they were to invite us I don't want to go … it is not pleasure' (3F). F may lose out, too, on joint activities that have been given up: '… we used to like going to the cinema, a concert, R loves music' (6D). D and F may modify their activities without giving them up altogether: '… what I prefer now is, well, four people maximum … we used to go for a meal, eight of us, and then I said I wasn't going to go because I just couldn't cope with it' (11D).

Managing the adjustments without negative consequences for the relationship

As noted earlier, adjustments need not become a source of contention. For example, F may give support, as required, as part of the commitment to the relationship: 'I have a disability myself and I rely on him or other people to do things for me …we sort of do things for each other, sort of thing …' (24F). Partners may be receptive to learning how best to adapt: It's not so much the communication, it's more the strategies she's taught me like when we sit in the kitchen, I sit so that I'm facing the light so she can see my face and I just know to do that, but then she is a good teacher. (11F)

If diffi culties arise, these are just treated as a matter of course, in an atmosphere of mutual acceptance: 'J sometimes gets a bit frustrated with me but she does not mean it, it is just a normal thing' (13D). F may want to compensate but is careful not to overstep the mark and provide unwanted support: 'I think he expects the support. I let it go as far as I can without intervening or interrupting …' (23F).

F may be careful to preserve D's sense of autonomy: '… she is the kind of person who says she knows what she can and she can't hear, and if she can't hear, she is quite happy to pass it over to someone who can' (17F). F has to exercise judgement in not being over-supportive and may have to 'push' D to be more autonomous: 'I have made him go out and do that … But you have got to know when to do it and when the time is' (10F). F may strive to maintain D's autonomy and feel frustrated if this is not possible: '… in a way we have taken over, he does try, he does want to stay in control and manage but he can't. That is frustrating' (18F). The adjustments that F has to make often require extra effort but D acknowledges this as a potential burden on F: 'I just did not think it was very fair to keep asking him what they were saying. I couldn't keep up with it so I just stopped going' (20D).

health: 12(3)

F may acknowledge their own defi ciencies but this does not seem to imply self-condemnation or resentment of D (an interpretation supported by the surrounding text): I don't know. I would be a long way from being perfect and being the most sympathetic or empathetic that I think I could have been. (1F) I am probably the world's worst. I will just treat her as though she can hear me … (17F) F makes allowances for D when failures of communication occur: 'Yes, well this is where the breakdown of communication comes, isn't it? Through no fault of hers' (6F). F may have to sacrifi ce some of their own enjoyments but this seems to be done as part of the give and take of a committed relationship. For example, F reduced his sporting activities: 'So I cut them down because it would mean leaving her too often and apart from that I don't want to play that much now' (8F). F may 'take charge' of a situation but there is an implication that this is exceptional and temporary: I said to him not long ago that I felt more like his mother than his wife for a while … I stayed as strong as possible because I thought somebody has got to be strong in the partnership. (10F) I think sometimes Mum probably prefers me to stand up, rather than her have to say 'I'm deaf, I can't hear you.' Especially, over and over again to close family members, I will have a go at them instead. (21F)

In some instances, the adjustments that F has to make give them greater self-confi dence: '… and it has made me a much stronger person, much stronger, I didn't know I had this much strength in me' (10F).

Adjustments leading to negative interactions or vicious circles

Signs of manifest interpersonal stress were evident in more than half the transcripts. The manner in which participants had adjusted to impaired communication seems to have led to increasing strain and, in one couple, breakdown of the relationship. An uneasy compromise prevailed in some cases although this may have existed prior to D losing their hearing. In line with our core category, we interpret manifest stress as originating in:

(1) A perception by D or F that the other 'ought to do better' because he/she has departed from their (perhaps implicit) model of expected behaviour. This perception may create a sense of inequity in the relationship overall.

(2) A loss of relationship quality, experienced as frustrating or unfair.

(3) Negative public perceptions of D that in turn impact negatively on F.

Departure from expected behaviour or attitude F may feel that D is either simply not trying hard enough to compensate for their disability or ought just to accept it: 'Uhm … you shouldn't dwell on these things, you should just get on with living and you do the things you can do and feel that maybe that you are better off than a lot of other people' (6F). F may perceive that D expects too much of them and sets limits on how much they are willing to adjust: 'I intended to carry on leading what had become a normal life. Although it wasn't going to be normal any more, there was no reason why you should suddenly have your whole world collapse about you …' (4F).

F may feel they are giving support and it is not being appreciated: 'Yes, she does not look for support but she certainly needs support and I give her support. Now, she might argue with that and say I don't support her, I don't know what she would say about that' (8F). F may try to be accommodating but then feel that they are falling short of D's expectations: Whenever I came off the phone invariably she would say 'oh, did you ask them so and so' and you know, 'shit', I have gone through all this and that is the one question she wanted to ask and I did not ask them and that seemed to happen repeatedly. (6F)

Conversely, D may think that F should be more supportive or may feel that F has a grudging attitude. The implication from the context of the fi rst quotation is that her husband should have been more involved in helping her to adjust: … my husband, I felt that it didn't affect him very much. He wasn't unkind or unhelpful, he just carried on as normal. (20D).

… having to wait for him to make the phone call. And not that I couldn't do it myself or that he had to do it, but his reluctance to do it, you know, 'oh, do I have to?' (2D) D may feel that F should be more understanding about being asked to make specifi c adjustments (in this instance, reported by F): '… I think J found that annoying because he was fi nding it so diffi cult to control his volume, so he found it quite annoying to be asked' (7F). D may criticize F's hearing tactics with the implication that he/she should try harder:

I was aware of it causing tension, but not of my point of view. It was they that weren't doing the communicating (laughs) … I felt very much it was me having to do all the work. Giving them deaf awareness all the time. (2D) F may feel that D is not making an effort to communicate: … as soon as I get in, M is able to unload the story of her day, what she has been doing whereas I am trapped in as much as I couldn't go into detail and tell M about my day. It is sometimes just a few sentences so, whereas she is able to communicate to the relationship, I am not. (14F)

Even when pragmatic adjustments have been made jointly, D may still feel disadvantaged and appear to implicate F in their disappointment: 'I defi nitely feel as if I have lost my independence because you always seem to need somebody with you and doing things for you' (6D).

health: 12(3) D's hearing loss may bring about changes to the relationship that lead to a reassessment of its reciprocal benefi ts and/or distribution of power. This may be experienced as an inequality that is resented: … I think she probably resents me a little bit because I am still able to do things that we have always done and she can't and she sees me now as the one that is in control. (8F)

It is very hard to live with the fact that I did not have time on my own. (10F).

F may feel that D has somehow emerged with greater benefi t from the new situation:

… it seems really strange to say it but it seems that everything positive has happened to B and all the negative comes down on me but that is the way you view life I suppose, and what happens to you. (14F) F's sense of being the person on whom D depends may be experienced as a burden: 'I could never leave him because he is so dependent. He doesn't recognize it, he would never say or even think it, but I think he is … very dependent on our relationship …' (9F).

Loss of quality in the relationship

One cause of a loss of quality is a failure to repair a breakdown of conversation in a satisfactory manner: I can't make him understand, well I say, it doesn't matter, forget it. He says, it does matter, so I sort of have to try and understand what I mean, otherwise he gets all …, and his heart starts beating faster and I don't want to get his heart up. (13F) D or F may also miss the spontaneity and depth of one-to-one conversation and companionship generally:

I have pretty well missed regular normal conversations with them and it has been rather tedious both from their point of view and from mine. (3D) It is very stressful, I cannot, a part of our relationship in a way has died in …, and I might weep here (obviously upset and tape paused). (3F) One function of a close relationship is to talk about stresses that originate outside it (i.e. a mutual 'offl oading of emotion'). Understandably, D or F might feel a need for emotional release but the usual give-and-take exchange seems to break down: … this problem never seemed to gravitate outside the front door and it was all directed at me. (6F) She doesn't like being excluded and sometimes that can be upsetting for her and for me. I mean I get upset by it but then I get the double whammy because she gets upset with me as well. (11F) D or F may go outside the relationship to unburden themselves: '… she has got a couple of friends, one of them is my cousin who she talks to and really tells her feelings to whereas she knows it would probably upset me' (14F). The opportunity for shared intimacy in social situations is also affected: 'Often left the evening with feeling annoyed, angry even, rather than having said "oh that was a good evening", just because she couldn't follow' (11F).

Public perceptions of the deafened person D and/or F may feel upset when D is perceived differently because of their deafness:

Annoyance when we are out socializing and I am stood at the side of my husband and people look past me to him, and don't include me. Even if they are asking me something, they ask J as a third party to ask me and it is like you become a different person. (8D).

S may perceive others' response to D as personally humiliating: 'And to be faced across the table with two people who are in hysterics over W confi rming what he thinks he has heard, I couldn't handle it' (3F).

F may begin to perceive D differently as a result of their changed social persona:

He hasn't been able to show his sense of humour, and within the group. It's probably affected us in ways that I'm not really aware of because I don't know how it would be different otherwise, but I'm sure that has had an effect on the way I perceive him socially. (7F) D may realize that they are embarrassing F. In the following situation, in a restaurant, D had complained about the behaviour of some other diners: 'C said I positively shouted at her and we could all have hid under the table. It doesn't sound like shouting to me but they went which was good' (11D).

Perceived threat to the relationship

Several participants intimated that their relationship had been placed in jeopardy by the challenge of deafness. One couple was in the process of separating and problems caused by deafness appeared to be important contributing factors. Another volunteer for the research, who later withdrew, said that her own hearing diffi culties had led to the ending of her marriage. The following quotations illustrate participants' worries:

He always looks shocked when I talk about the threat to our marriage, it is more fundamental and I told him it is a threat to our relationship and I was fearful … (3F) … it was quite distressing and it has led to some enormous strains on the relationship which I think we have patched up … I hope we have patched up anyway. (6F)

Discussion

The decision to interview family members as well as the person with APHL seems to have resulted in an even-handed representation of the diffi culties health: 12(3) that couples and families were facing. We did not encounter much blame or acrimony -and this was true also of the couple who were in the process of separating. Family members provided a considerable amount of information about the impact on the person with the hearing impairment, as refl ected in the number of quotations they provide. For many, it was their fi rst opportunity to voice their point of view as none of the participants had ever received any counselling together. (It should be noted that the LINK course provided an opportunity for this but not all family members had attended and many of the sessions were in a group format.) The decision to interview participants separately meant that feelings were expressed that could not have been expressed in a joint interview. However, in view of the fact that some participants reported humiliating and other negative experiences in their dealings with professionals or the general public, it seems likely that some views were held back.

The results indicate that APHL can affect a family in complex and subtle ways. Evidence of interpersonal stress was found in at least half of the married participants, a proportion close to the 53 per cent of people with a severe hearing loss who reported that deafness had affected their marriage (Thomas, 1984: 95). Although not emphasized here, the contribution of additional medical problems (especially tinnitus and imbalance) and the stress associated with obtaining diagnostic information and appropriate medical and rehabilitation support should not be underestimated.

The negative impact of APHL on many of the families replicates the fi ndings of previous research. It would be inappropriate to designate partners as 'carers' but APHL imposes a considerable burden of extra effort. Old habits of communication have to be changed and family members often admitted their own defi ciencies in this respect. Domestic, social and leisure activities may have to be modifi ed or curtailed. All this presupposes a willingness on the part of the family to engage with the problem. Family members, including children, may have to take over domestic duties or act as intermediaries in a variety of situations. As reported by [START_REF] Jones | Words apart: Losing your hearing as an adult[END_REF], the presence of problems in communication may subvert prevailing power relationships as the person with APHL becomes more assertive or more dependent.

Our sample was not designed to be representative of all people with APHL although we assume that our model describes the generic pathways that lead to maintenance or breakdown of relationships. We have not taken account of stage of adjustment to hearing loss, stage in the life cycle, effect on employment or the benefi ts of cochlear implantation, all of which are likely to be important moderating infl uences that deserve further study. However, from the limited information we obtained in interviews, none of these factors would be appear to be critical to the relationship, acting independently of other infl uences. We are also unclear why a large number of potential volunteers declined to participate. We suspect that many families did not want their relationships put under the microscope. In one case, a volunteer withdrew after her relationship broke down. It seems likely that a random sample of people with APHL would evidence more signs of stress or dissatisfaction. Many of our participants had responded favourably to help offered earlier by LINK and were willing to devote time and effort to our research.

The adjustments made by our participants are similar to ones described in research on AHL (e.g. Thomas, 1984: 134-45;[START_REF] Hallberg | Hearing impairment, coping, and consequences on family life[END_REF]. The person with hearing impairment tended either to be excluded from conversation or, less frequently, to take control of it. For many families in our study, the adjustments were made in a spirit of give-and-take and unquestioned commitment. In managing the necessary changes, many families negotiated with respect and consideration. This involved a mutual acknowledgement of the burden, each party accepting and forgiving an inevitable falling short of expectations and the need to make allowances, a respect for the dignity, autonomy and capabilities of the person who needs support and an obligation on the latter to make reasonable efforts. Participants in the study were well aware of the potential to do too much, to neglect a person's need for independence or to fall prey to a loss of respect.

However, for some families, APHL sets in train a negative pattern of interaction. Whether or not this happens may depend on the prior state of the relationship and the personalities involved. Participants certainly expressed expectations of each other that, in some cases, remained unfulfi lled, leading to a sense of loss, frustration or resentment. It cannot help matters that problems of communication are likely to obstruct the means by which any problem usually gets solved, that is, through negotiation. Profound hearing loss hinders the spontaneous expression of mutual regard and reduces the opportunity for positive shared experiences. Some participants also mentioned their frustration at not being able to 'offl oad' about the normal stresses of the day.

The strain on the family includes a public response to APHL that is often deeply prejudiced or, at best, uncomprehending. An analysis of this aspect of the data is currently in progress but participants spoke of family members or longstanding friends who could not really be bothered to alter their usual mode of communication. The person with APHL was sometimes regarded as stupid or was simply bypassed in conversation. If they were not able to deal effectively with this treatment, family members often felt obliged to intervene. In these circumstances, the family was precipitated into a joint defensive posture.

In terms of our core category -the prevailing social construction of a committed relationship and its rights, obligations and benefi ts -we infer that a substantial proportion of participants felt short-changed in their opportunities for intimacy and shared social enjoyments. Participants expressed a variety of mutual criticisms such as a lack of support or understanding, and poor hearing tactics. These complaints must be viewed in a social context of signifi cant disadvantages for people with a profound health: 12(3) hearing loss in the job market (if not loss of employment), and little public recognition of the needs of people with APHL (LINK Centre, 2005).

Authors such as [START_REF] Schlau | I did not die, I just can't hear': A grounded theory study of acquired deafness[END_REF] have rightly argued that there are stages of adjustment to APHL but her view that it is desirable for the person concerned to accept a new identity as a 'deafened person' who goes through a defi ning moment of acceptance, learns to sign, fi nds deaf friends and accepts deaf ways, is unlikely to be generally accepted. In fact, only one-third of her respondents had managed to achieve this and the remainder were described as 'struggling' or 'resigned'. Our participants did not seem to place such an emphasis on identity, which might refl ect differences between a US and UK cultural context. Our participants depended primarily on lip-reading and only one signed regularly. Adopting the identity of person who is deaf is not a preferred way forward in the UK where the deaf community tends to exclude most people with APHL.

A focus on individual adjustment also neglects to consider how the family of the affected person fi ts into this developmental picture. As Hétu et al. (1993: 375) have rightly stressed, professionals must attend to the interactive dimension of the diffi culties and of the coping process. The impact on family members often seemed greater than on the person with hearing loss, him or herself. Given the variety of individual circumstances, it would be diffi cult to defi ne an ideal outcome of adjustment applicable to all. In a similar vein, Jones et al. (1987: 215) paint a complex and non-prescriptive picture of family adjustment in the hard-of-hearing community.

As noted earlier, we were unable to do suffi cient justice to variables such as age, gender, the prior quality of the relationship, personality characteristics and having to cope with other medical conditions or sensory impairments. We are currently analysing our data to investigate the infl uence of gender, which has been found to be an important variable [START_REF] Stephens | Effects of hearing impairment on the patient's family and friends[END_REF][START_REF] Wallhagen | Impact of self-assessed hearing loss on a spouse: A longitudinal analysis of couples[END_REF]. Like other qualitative researchers in this fi eld, we have found it necessary to locate these variables within higher order constructs. [START_REF] Hallberg | Coping with post-lingual severe profound hearing impairment: A grounded theory study[END_REF] derived a core concept of 'fi nding fl ow', with the implication that individuals enter a 'positive circle' if they do or a 'negative circle' if they do not. This formulation hints at a dynamic system but in fact their concepts refer to individual dispositions related to [START_REF] Antonowsky | Unravelling the mystery of health: How people manage stress and stay well[END_REF] concept of sense of coherence. These authors were investigating coping with everyday life situations, such as work, and the dispositions they describe may or may not be suited to maintaining fl ow in family relationships.

Our model and the fi ndings on which it is based are entirely consistent with the views of [START_REF] Hétu | The impact of acquired hearing loss on intimate relationships: Implications for rehabilitation[END_REF] working with older people with AHL. These authors stress that counselling should focus equally on the needs of the hearing impaired person and their partner. The aim is to facilitate communication about confl icting views, roles, needs and costs, so that misunderstandings and disagreements can be resolved by negotiation. Hétu and colleagues recommend that this can be done in groups. Our research suggests that taken-for-granted assumptions about the grounds of commitment to a relationship can be put into question by the consequences of APHL. When a relationship is seriously threatened, couples counselling is indicated. In the acute stage of onset of hearing loss, especially if this is sudden, counselling may have to focus on managing a crisis and extreme emotional responses. The most appropriate intervention would depend on the individual case and the stage of adjustment.

Table 1 Characteristics of sample with APHL (N = 25)

 1

	Characteristics
	Gender (male:female)	11:14
	Mean age: yrs (SD and range)	53.4 (13.0, 25-77)
	Married: single or widowed	20:04
	Separated	01
	Employed	11
	Cannot hear sounds at all (unaided)	16
	Wears a hearing aid	12
	Wears a cochlear implant	10
	Use of lip-reading, very often or always	20
	Recency of onset of profound hearing loss a
	< 1 year	05
	1-5 years	10
	> 5 years	09
	a 1 missing datum

Figure 1 Conceptual model of the relationship consequences of aural impairments

	Aural impairments interacting with processes
	serving to maintain a commitment to the
	success of the relationship
	Pragmatic adjustments
	Communication	Domestic, social and
		leisure activities
	Negotiated with mutual	Incomplete negotiation with
	respect/willingness to engage	unfulfi lled expectations/
	with the problem/tolerance	diffi culty accepting losses/
	of mutual failings	perceived imbalance of
		power or benefi ts
	Stable or strengthened	Potential for chronic
	relationship	dissatisfaction or relationship
		breakdown

health:

12(3)

We gratefully thank the Big Lottery Fund Research Branch for a grant to pursue this research. We also appreciate the assistance of Maria Priestly and comments on the manuscript from Roslyn Corney.

Author biographies

richard hallam is a researcher and clinical psychologist. He worked at the Royal National Throat Nose and Ear Hospital, London, for eight years and has published widely on psychological aspects of audiological medicine. He is currently Visiting Professor of Clinical Psychology at the University of Greenwich and also works in the UK National Health Service.

paul ashton trained as an economist and for 14 years conducted academic research, mainly at Liverpool University's Macroeconomic Research Group. As someone who has a hearing loss, he has experienced at fi rst hand some of the social and psychological effects investigated in this study.

katerina sherbourne trained as a medical doctor and has an MSc in social research. She has worked at the LINK Centre for Deafened People for 11 years and has published a formal evaluation of the effi cacy of its rehabilitation programmes. lorraine gailey completed a PhD in the psychology of speechreading in 1981 and qualifi ed as a speech and language therapist with a special interest in the rehabilitation of adults with acquired deafness. Since 1991, she has been Chief Executive of the Link Centre for Deafened People in Eastbourne, East Sussex.