Abstract
Storytelling is perennial, and central to the human condition. Although illness may shatter identity and one’s role and place in the broader social world, narrative may aid in the process of self-reparation. Despite the merits of the narrative approach, it has been underutilized with children who are living with cystic fibrosis (CF). The role that illness narratives may play in influencing CF youths’ physical activity also remains poorly investigated. This article drew on the qualitative case study methodological tradition to narrate the stories of two children living with CF at a children’s hospital in Canada. The findings beg researchers to consider (a) how children with life-limiting diseases borrow multiple illness narrative types, (b) the role of development in influencing the kinds of stories that children can tell, and (c) the impact of illness narratives on physical activity. By rendering the tales of two CF youth in this study, we respond to Aurthur Frank’s call; taking a multiple narrative turn, we listen to stories of a different kind of suffering.
‘Perhaps the healer and the family, like the historian of human misery, must allow themselves to hear … the wail’ (Kleinman, 1988, p. 284).
Introduction
Narrative and storytelling are perennial, and central to the human condition. For those that live with illnesses, narrative may aid in the process of self-reparation and mend the brokenness of the body (Frank, 2000b). Since people with illnesses often experience a sense of temporal disruption – especially those with chronic and life limiting diseases – narrative may recreate a lost temporal framework, parsing together the shreds of time (Lars-Christer, 1997). Despite the contributions of the narrative approach toward disciplines such as psychology and the social sciences, stories of patients living with cystic fibrosis (CF) – especially children – are notably absent in the literature. CF youths’ storied experiences of physical activity are also limited. In this paper, we respond to Arthur Frank’s (2000a) call to avoid meta-narratives, that is, those grand, totalizing accounts of the illness experience that may eclipse the particularities of suffering. Rather, we extend a study by Moola, Faulkner, and Schneiderman (2011) that described the development, implementation, and evaluation of a physical activity counselling intervention for CF youth known as ‘CF Chatters’. In doing so, we draw on the qualitative case study methodological approach to narrate the health and physical activity stories of two CF children. We illuminate health and illness dilemmas encountered by the CF population that physical activity intervention development approaches may not be sensitive to. Since this may contribute toward a more global understanding of CF, this paper also highlights potentially useful information for future behavioural interventionists. Indeed, Sparkes and Smith (2008) suggest that there is a need to make narratives that do not align with the contours of dominant storylines available, and find those hidden stories that lie beyond conventional plot lines. By chronicling the stories of two CF youth in this paper, we answer this call by attempting to render stories of a different kind of suffering and take a multiple narrative turn (Frank, 2000).
Review of the literature
There is a growing body of qualitative literature that examines how young people living with CF experience their health and illness. In our synthesis of this literature, there are three themes that characterize patients’ experiences: Normalcy, temporal dilemmas, and treatment non-adherence.
A compromised sense of normalcy is a salient issue for people with CF. In a landmark study, Bluebond-Langner (1979) found that parents of youth with CF engage in a variety of tactics to prevent the intrusion of the disease into their everyday lives and preserve a sense of normalcy. Lung transplantation is a serious surgical procedure that may be necessary for some CF patients. Maynard (2006) found that CF patients awaiting double lung transplant surgery seek to maintain a linear, uninterrupted narrative, upholding a vision of normalcy and health for as long as possible until their bodies fail. Admi (1996) also found that young people with CF strive to define themselves as normal and do not dwell on death until negative events force the body into stark relief and threaten normalcy. Huyard (2008) underscored similar findings in adults with CF. If the patient has no symptoms at the time of diagnosis, the disease’s label is rendered irrelevant and normalcy is not compromised. Health deteriorations starkly connect the disease label and symptoms, initiating a critical self-learning process in which patients contemplate the limits of their abilities and question their normalcy. Glasscoe and Smith (2011) also found that the parent of a child with CF strives to attain normalcy in what is otherwise an extra-ordinary situation, seeking to maintain a fine balance between affect and reason, acceptance and denial. However, CF symptoms shatter parents’ fragile sense of normalcy and make them recoil in horror (Bluebond-Langner, 1996).
Temporal disruption is also critical to understanding how health and illness are experienced in CF. The meaning of chronic illness may have a generational impact across time, with family members developing a shared familial compilation of illness stories. For example, in their qualitative study, Seaburn, Lorenz, and Kaplan (1992) found that CF may serve to generationally connect family members across time. Caregiving for ill family members may represent vertical and horizontal commitments of loyalty, and is fuelled by feelings of love, a sense of duty, and guilt. Such loyalty commitments attempt to repair the incomplete caregiver work that was inherited from previous generations (Seaburn et al., 1992). Similarly, Heidegger (1992) suggests that time is the first and necessary precondition for ‘being in the world’ and the apprehension of anything worldly. As humans, we must create a temporal field for meaningful activities to unfold, and strive to maintain a sense of synchronicity between temporal events in the environment and neurochemical processes in the brain (Heidegger, 1992). Life-shortening diseases such as CF illustrate the power of illness to shift the meaning of time and to threaten temporal integrity (Glasscoe & Smith, 2008). Similarly, Glasscoe and Smith (2008) found that rather than a linear series of events, CF parents may experience the telescoping of time in which particular periods of health-related deterioration are magnified. Moola, Faulkner, and Schneiderman (2012) also found that major temporal anxieties and dilemmas are central to the experience of living with CF.
Non-adherence to treatment is a central theme that is relevant to those with CF. Walking a precarious line between sickness, health, and quality of life, researchers have long-since observed that CF patients may push boundaries and compromise time-consuming treatment to preserve quality of life, such as socializing with friends (Admi, 1996; Huyard, 2008; Moola et al., 2012).Wright and Morgan (1990) observed that patients who do not adhere to treatment are considered deviant when they do not uncritically subscribe to the unquestionable authority of medicine. Willis, Miller, and Wyn (2001) examined sociological explanations for the gendered disparity in survival. In relation to boys, CF girls are more likely to die before the age of 20 and compromise their treatment, nutrition, and physical activity. While girls adopt a disengaged role in their health and fail to plan for the future, boys more readily accept CF and demonstrate enthusiasm for the future. Some of the socially-constructed practices of femininity, such as dieting, may be injurious to health in CF, and treatment non-adherence may play out differently in the lives of girls and boys (Willis et al., 2001). Thus, thwarted normalcy, temporal losses, and treatment non- adherence characterize the psycho-social worlds of CF patients.
Physical activity and cystic fibrosis
Despite the well documented benefits associated with physical activity for people with CF, the majority of these patients are inactive (Nixon, Orenstein, & Kelsey, 2001). While physical activity is promoted for the CF population, exercise training interventions characterize current approaches. Such exercise training programmes may not be relevant to patients’ interests or local environments, nor display sensitivity to the lived experience of CF – exemplified by the themes of normalcy, temporal dilemmas, and treatment non-adherence described above (Moola et al., 2011). Thus far, only two studies have utilized a qualitative approach to explore the physical activity experiences of CF patients. This research suggests that disease barriers characterize their physical activity. For example, Moola et al. (2011) and Swisher and Erickson (2008) found that the burden of disease and treatment is the most noxious barrier toward physical activity, leading to activity avoidance and a sense of compromised normalcy. While active CF youth are characterized by parental support for physical activity, mastery experiences that confirm success, an inherent sense of pleasure during activity, and hopeful illness narratives, inactive youth ascribe low value to physical activity, lack parental engagement, and narrate feelings of helplessness and futility as a result of CF. According to supporting literature that documents CF patients’ temporal concerns (Glasscoe & Smith, 2008), youth negotiate physical activity within the context of reduced time (Moola et al., 2012).
To address these population-specific barriers to physical activity, we developed a theoretically informed, parent-mediated, behavioural counselling intervention for CF youth known as ‘CF Chatters’ (Moola et al., 2011). The programme was pilot-tested with six CF patients and their caregivers to assess its feasibility and acceptability at the CF Centre of a large children’s hospital in a Canadian metropolis. Participants demonstrated self-reported increases in physical activity and quality of life. While further development and more robust objective measures are needed to extend this investigative pilot work, our findings suggest that behavioural counselling might be an effective modality for enhancing physical activity and quality of life in this life-limited group of youth (Moola et al., 2011).
However, in conducting this research, we were also conscious of the ways in which such an exercise intervention approach fails to capture patients’ health and illness narratives. In describing the development and evaluation of the CF Chatters intervention, we were not able to share the participants’ health and illness stories, and the ways in which one’s perception toward their disease strongly influences physical activity experiences. Thus, in this paper, we attempt to extend Moola et al’s (2011) work by illuminating some of the health and illness dilemmas that may not be captured by such a structured intervention and evaluation approach.
Furthermore, studies that utilize the case study approach to understand how children living with CF narrate their health and physical activity experiences are absent from the literature. The case study approach has proved critical to understanding the health and physical activity experiences of those whose lives are circumscribed by different kinds of illnesses. For instance, in chronicling the sporting life of elite cyclist Lance Armstrong, who was diagnosed with an aggressive form of testicular cancer at the pinnacle of his career, Sparkes (2004) suggested that the body remains absent and only enters the registers of meaning as a liability when it demonstrates signs of failure. Sparkes (2004) illuminated the multiple body–self relationships that Armstrong adopted during the transition from world class athlete, to cancer patient, to cancer survivor, and his affinity for different narrative types. Rooted in a tough, masculinist working-class culture, Armstrong’s pre-cancerous self is one that is demarcated by staunch individualism and the corporeal manipulation necessary to achieve a disciplined and regimented sporting body that is a closed, monadic entity unto itself. When stricken by cancer, illness becomes yet another challenge to defeat. In the post-cancer period, Armstrong contemplates the ways in which illness has changed him and broadens his purview beyond the narrow world of cycling to include helping others with cancer and considering his role as a husband and father (Sparkes, 2004).
Similarly, in narrating Jaime’s case story, that is, a young athlete who suffered a severe spinal cord injury that terminated his rugby career, Smith and Sparkes (2008) assert that desirable stories of suffering assume a particular structure that emphasizes transcendence and minimizes despair. Because physical activity and illness narratives like Jaime’s are so despairing – told on the edges of a wound (Smith & Sparkes, 2008) – they are unlikely to be heard. Because such stories illuminate an empty present, a psychologically battered self, and a desolate future post-injury, they push the limits of tellability and are too frightening to hear. Similarly, mental illness is associated with notions of deficit. For those who live with mental illness, narrative processes play a critical role in aiding meaningful re-storying and facilitating the process of self-reparation (Carless, 2008; Carless & Douglas, 2008).
Thus, qualitative researchers in psychology and the social sciences have done much to better our understanding of how CF patients experience their illness, and the ways in which thwarted normalcy, temporal disruption, disease non-adherence, and physical activity barriers compromise health and physical activity. However, the case study is underutilized as a methodological approach for the CF population. As demonstrated by Smith and Sparkes (2008) and Carless (2008), narrative is important within the context of illness for both those that suffer with illnesses, and those that care for and treat them. By distancing people from the threat of illness, the narrative process may (a) facilitate self-reparation and bodily integrity; (b) illustrate body–society relationships and cultural ways of understanding disease; and (c) bridge the chasm between health providers and patients by providing shared means for understanding illness. Furthermore, the illness narrative approach may (d) provide us with an insider emic and ideographic perspective on what it means to be ill, one that cannot be captured by nomoethic studies; and (e) teach us about physical activity–illness relationships.
Since the narrative approach has rarely been utilized with CF youth, we considered it important to build on the work of Smith (2004) and Carless (2008) to include this specific clinical population. Adopting the case study methodological tradition to understand how two children living with CF narrate their experience of illness and physical activity, we ask: 1. How do children living with CF understand and narrate their illness? 2. What impact, if any, do illness narratives have on outlook toward physical activity? 3. What theoretical advancements can be made from children with CF toward the concept of illness narratives? We employ a hermeneutic device to narrate the children’s words through our own interpretive lens. In doing so, we refract their voices through ours, and uphold the virtues of storytelling as a mechanism to better understand how CF children negotiate health, illness, and physical activity.
Methods
Theoretical framework
Informed by the sociology of health and illness, this study was theoretically sensitized by the concept of illness narratives. Although historically the field of medicine has accorded an ambivalent status toward what patients have to say, more attention is being devoted toward the merits of listening to stories of illness (Lars-Christer, 1997). Illness narratives have particular functions and effects. Illness may ‘rupture our sense of temporal continuity, and, if the rupture is not mended, the fabric of our lives may be ripped to shreads’ (Lars-Christer, 1997, p. 53). For this reason, the telling of illness narratives not only provides a frame of reference to understand the world, but brings form and meaning to suffering. Where illness is associated with identity threat, telling stories may aid in the recuperation and reparation of the self and offer an explanatory model for patient’s experiences. Without a storied structure to articulate the experience of suffering, the relations between patients and their health-providers may remain plagued by misunderstanding. In helping to mend the yawning chasm between patients and providers, narratives can liberate the health-provider from often narrow and oppressive accounts of why patients act in the ways that they do. Narrative may help providers to break free of such a reductive ‘iron cage’, helping them to listen and better know the Other (Frank, 1998). Narrative provides a way to write across the lines of fault that demarcate the lived experience of illness from the academic analysis of what it means to be ill. Until patients and providers can see glimpses of one another’s world, the clinic will remain rife with tension (Frank, 2000b).
There are particular theoretical tenets associated with the use of the illness narrative. Although narratives appear to be personal stories, they do not emanate from the caverns of our minds. Rather, personal illness narratives are profoundly cultural, and, in the telling of individual stories, we draw on culturally available and dominant story lines (Smith, 2010). Often, we can look to personal stories of illness to teach us about the broader culture. Bodily symptoms or indicators of distress, such as a loss of perceived time for young CF patients, can be read as embodied metaphors for social experience and distress in the inter-personal world, such as the emphasis on attaining milestones on time in modern capitalist cultures. Thus, although illness narratives are realized as local worlds of experience, they are patterned by culture (Ware & Kleinman, 1992). And, because narratives are cultural, they can help to collectivize an illness experience (Frank, 2000a).
In what is described as an increasingly fragmented and polymorphous postmodern world, illness narrative scholars remind us of the importance of avoiding teleological, grand meta-narratives of illness (Lars-Christer, 1997). Totalizing accounts may foreclose alternative possibilities and fail to expand the available cultural narrative repertoire. Rather, as we seek to do here in rendering the health and physical activity stories of Chase and Layla, there is a need to consider tales that do not accord with the contours of dominant plot lines (Sparkes, 2004). Finally, Frank (2000a) reminds us that stories are not matter to be analyzed, but rather relationships to be entered. In entering into a narrative relationship with the patient, the narrator must respect and preserve the other’s alterity. Following Baktim, who states that ‘two voices are the minimum for life’, (Frank, 2000, p. 135) storytellers must willingly enter into a dialogical relationship characterized by shared listening, hearing, and understanding (Frank, 2000a). In doing so, it may be possible to engage in a far more ethical narrative relationship.
Although Frank (2000) encourages us to avoid grand meta-narratives of illness, it is important to outline the three dominant narrative tropes that account for most illness experiences. As the culturally preferred narrative, the restitution story emphasizes triumph in the face of adversity and the recuperation of the self after illness. As the classic tale of overcoming hardship, restitution narratives are pleasing to hear. Ultimately, the ill body with an affinity toward the restitution narrative restores the pre-illness body–self relationship and rebuilds and remoralizes the body (Smith & Sparkes, 2008; Sparkes, 2004). The chaos narrative stands in direct contrast to restitution and narrates a tale of being swept away by life’s fundamental contingency. The body in chaos is broken; the present is empty and the future is desolate and experiences of terror tend to characterize these plotless stories. Since the body in chaos is never restored, these tales push the upper threshold of tellability – what can be told and what can be heard. Finally, the quest narrative type is characterized by fundamentally altered body–self relationships (Smith & Sparkes, 2008). The quest narrative emphasizes the acquisition of insight and new understandings, in which body, self, values, and beliefs are fundamentally changed through illness as a journey. Theoretical tenets from the illness narrative perspective provided a useful way for thinking about and analyzing our findings.
The case study approach
Informed by the concept of illness narratives, the qualitative case study research tradition provided the methodological underpinning for this study. The case study employs multiple methods to explore the particularities of individual lives (Flyvbjerg, 2004; Stake, 2000). While the case study has typically been grounded in a post-positivist epistemology that assumes certain truths and fixed relationships between language, meaning, and experience, it is increasingly employed by social constructivists in ways that take account of the socially constructed nature of social life. The first epistemic tenet that underpins the case study is that much information can be derived from the study of the unique and the particular, and that there is inherent value to studying the specific. Secondly, case studies value comparison and contrast as critical to the production of knowledge, and carefully tread the line between the general and the specific, or the generic and the particular (Stake, 2000). As suggested by Woods in Faulkner and Sparkes (1999), case studies ‘emphasize the context bound uniqueness of participants’ stories while common themes reveal the junctures in their shared experiences’ (p. 96). Through case comparison, then, it can thus act as a useful forum to confirm existing understandings about CF and physical activity, and generate novel insights which capture the particularities of participants’ lives. As in the instance of this study, case research is an excellent forum to understand how participants appraise behavioural interventions (Flyvbjerg, 2004; Stake, 2000).
There are many case study types and guidelines regarding how to select appropriate cases. To maximize the analytic potential of comparison, researchers are advised to conduct multiple case studies (Stake, 2000). Although a total of six participants were enrolled in the CF Chatters programme (Moola et al., 2011), for the purposes of this study and due to space limitations, we tell the stories of the two adolescent participants – Chase and Layla. We assigned these pseudonyms to the participants to protect their confidentiality. The case stories of the other child and adult participants have been compiled as narratives, and are available upon request from the first author (FJM).
Methodological tools
Semi structured interviews were employed to explore participants’ health and illness experiences and thoughts toward physical activity (Mason, 2002). The participants engaged in multiple, lengthy interviews over the course of the six-week programme, and we developed a sustained relationship and rapport with the participants. Although a semi-structured interview guide was developed by the researchers to understand their experience of the intervention, the participants’ responses drove further lines of inquiry and the interview proceeded in terms of a recursive conversation. These interviews were digitally taped and transcribed verbatim (Kvale, 1996; Mason, 2002). A detailed field note journal was used throughout the study to record, describe, and characterize participants’ responses to the CF Chatters programme, institutional observations, personal reflections, and novel programme insights. A few hundred pages of field work data was collected over the course of the CF Chatters programme, and observations were recorded before, during, and after each session. The field work data further corroborated the interviews and provided an excellent forum to understand the unique facets of participants’ lives and their response to the programme (Faulkner & Sparkes, 1999).
Case study analysis
There are many analytical frameworks that researchers can employ to analyze the findings from case research. Researchers can analyze a case study chronologically by describing the events of a story over time. Alternatively, researchers can analyze a case by way of theoretical proposition in which a particular theoretical postulate provides the interpretive framework for the case (Stake, 2000). In this study, we adopted a variety of different approaches to aid the analysis of our cases.
To analyze the case studies, first, a thematic analysis of the transcribed qualitative interview data was generated (Braun & Clarke, 2006). These interview question responses, such as thoughts and feelings toward CF, were coded, named, and grouped into broader themes. Novel responses that were not a component of the interview guide, such as the psychological benefits of the programme, were also named, coded, and grouped into themes. This thematic analysis of the transcribed data allowed us to understand participants’ overall perceptions toward the intervention (Braun & Clarke, 2006).
Secondly, multiple analytical techniques – such as analysis by way of description and theoretical postulate – were employed, and novel information was garnered by contrasting the different CF cases (Stake, 2000). For example, the case studies were analyzed within illness narrative frameworks, and the first author created ‘story profiles’ of each participant. Then, the field work and transcribed data were combined as multiple data sources to compile case stories, and provided the broader framework for how the cases were organized. For example, fieldwork notes and the transcribed interview data were drawn on to compile a description of the participants’ history, personal story living with CF, and response to the CF Chatters programme.
The findings, including extracts from the interviews and field notes, are provided below. By ‘invite[ing] the reader into a vicarious experience (however brief) of the life or lives being described’ (Sparkes, 2000, p. 33), case studies provide a provocative story and rich description of a few lives in context and are a useful way of understanding an individual’s experience of an intervention. Below, we tell Chase and Layla’s stories.
Findings
Chase: ‘I want to see the world through another person’s perspective’
Context
Chase is an intelligent and thoughtful 17-year-old boy with moderate CF. Although Chase was experiencing a respiratory infection at the time of the study, he has the outward appearance of a healthy, normal weight boy. Chase is from a two-parent middle class family and resides in a comfortable urban home. His parents are educated working professionals. Chase’s younger brother does not have CF.
Chase’s story
During the programme, Chase explained that his perspective toward his illness had recently undergone a radical change. He recounted negative stories of himself as a child and spoke of obtaining a new lease on life. With a grimace, Chase conjured up an unpleasant image of himself as a child in his mind’s eye – a child who was uninformed, apathetic, lacked awareness into health matters, and passively followed the rules outlined by his parents without question. Chase explained that as a child, he did not know what CF ‘really was’ and simply ‘did what his parents told him to’ without understanding the rationale for – or consequences of – treatment. With a near-visible disdain and moral repugnance for the boy that he once was, Chase recounted memories of a child who lacked a sense of passion and initiative; during our sessions, he appeared to psychically distance himself from this abject former self. The ‘former Chase’ was one who enjoyed physical activity, but who only undertook it at specialized centres for children with disabilities. The former Chase did not want to go away to college or university for fear of health problems, and instead envisioned himself living at home with his parents where he did not have to expose himself to unnecessary risks. The former Chase had a small social network and was largely isolated from his peers. The former Chase expressed disdain for the distressing bodily changes that CF medications caused.
Chase attributed his personal metamorphosis to a 60-day River Trip that he undertook at a wilderness residential summer camp when he was 15. On the River Trip, Chase’s social network expanded to include socially conscious and interesting new friends who saw past CF and accepted him as he was. The physically demanding nature of the trip, and the tremendous endurance required to paddle the river on a daily basis, allowed Chase to develop a new-found respect for his body, and a desire to take better care of himself through nutrition and physical activity. Camp provided Chase with the opportunity to meet other youth with disabilities, such as autism. Although Chase has a contradictory relationship with his own disease – simultaneously constructing himself as normal/ abnormal and healthy/sick – meeting others with disabilities at camp has led him to be fascinated by various illnesses and fostered an overwhelming desire to help others who are ill. Today, Chase has a rich and ever-expanding social network that includes friends and a romantic relationship. He challenges himself intellectually and pursues studies outside of the confines of the high school curriculum, ‘which really does not teach you anything useful’. He is an avid reader of Euro-Western philosophy, especially the history of time. Chase envisions pursuing a career in Kinesiology in the future and volunteering his time to help others who are afflicted by illness. He is also interested in travel and is planning to volunteer in Europe this summer.
A pro-health orientation
The changed boy who participated in the CF Chatters intervention was insightful and reflective and possessed a strong health orientation. In the following quote, Chase explains that he has changed the approach that he adopts toward managing his health. Now, he values health, has a new-found sense of responsibility to independently monitor his treatment, and expresses reluctance to rely on his parents to assist with treatments:
I understand that I have a lung disease and it needs monitoring. When I was a kid, I had no idea what was going on. Mom was like ‘take this pill’ and I took it, but that was coming from the doctor. I know now – I am aware. Things are clear to me now versus when I was younger. I understand. I do not need the parent to tell me to do this.
I am glad that you are doing as well as you are.
It is my own personal health. I like being healthy. It makes me feel good and I did not know before that I would feel this way about being healthy. Once I did, I felt good, I felt great, and once you do that, you do not want to go back. So all you need to do is just try. I think that most people should stick with it.
Chase comments on a friend who lives with another chronic disease, childhood onset diabetes. He critiques her apathy and over-reliance on her parents to do her treatment for her and liaise with doctors on her behalf:
Yes, an example is one friend who comes here [hospital] with diabetes. My diabetes friend is someone with very low motivation. She hates exercise and physical activity. She just lets her mom speak. And she knows that what her mom is saying is wrong, but she does not care about her health. I am just like ‘you should correct your mom, or you are not going to get the right stuff!’
Psycho-social challenges
Despite his strong pro-health orientation, Chase expressed frustration with CF-related disease symptoms and limitations. Respiratory infections, and dealing with the numerous physical limitations associated with CF, are particularly arduous burdens for Chase:
Infection is such a big thing with CF. When the doctors tell me that I have an infection, it is like ‘ohhh, this is bad,’ and I know what comes with it. And I do not like thinking about it. I think that no CF patient likes to think about it … partly, it is the disease – that is a big factor. It comes with so many limitations. You think that ‘I cannot do this, so I am not going to do this.’
However, despite the burden of disease and treatment, Chase underscores the importance of talking about disease barriers. He encourages himself to never give up and to persevere in the face of illness related adversity:
CF has to be discussed. It cannot be ignored. It has to be talked about, but when it is talked about is a different issue … I tell myself ‘that is not true. You can do it – it is going to be harder, but you can still do it.’
The numerous social restrictions that prevent youth with CF from interacting with one another given the risk of cross-contamination is another CF barrier for Chase that negatively influences his psycho-social health. Interestingly, Chase expressed an almost insatiable desire to know more about the lives of other children with CF and was interested in details, such as how ill other children are relative to him. Whenever any mention was made of other patients during the study, Chase was immediately captivated and hungered to know more. The medical restrictions that prevent children with CF from socially interacting with one another are burdensome for Chase because they deprive him of critical learning and social opportunities.
I learned a little about other kids with CF through the programme. I wish that I could see more of them and to relate my CF to theirs … I want to know their fitness plans and their lifestyles. I do not know them. Maybe they have something that I do not know about. And I have no way to learn about that. You will learn about all that stuff but I cannot know that stuff … Because there is no one that I can really relate to. When I am having a conversation, there is no one that I can talk to unless it is asthma or another disease … But, I don’t know, it would be nice to talk with someone that has CF as well.
He skeptically questioned best clinical practice, and, by discussing the social costs of clinical restrictions, he illuminated a complicated bioethical dilemma:
I wish that we could all be in the same room at the same time and just talk in some sort of crazy configuration. And just talk. Kind of like the sports games and hockey games. I do not know why they let us do that, but not other things.
Programme response toward physical activity
Chase was already interested in leading an active lifestyle prior to the programme. However, as a result of participation, he improved his physical activity pre- to post-intervention, increasing his treadmill running from 0 to 15 minutes a day. He also reported increases in quality of life:
My quality of life altered a little. With the running – wanting to run more and then actually changing to run more – that is only going to help. So, my quality of life did raise a little.
What part of your quality of life did it help?
It helped the physical part of quality of life with endurance and being able to run more. Endurance comes up all of the time. It does not have to be running. It could be who knows? Whatever kind of activity. Endurance and stamina are important in CF … I can avoid situations that bring me down because I am not able to do something. I will be able to avoid those things with better endurance and have a better quality of life.
Chase enjoyed the counselling-based nature of the programme, describing it as distinctly different from medical appointments. He also reported enhanced knowledge about the self, and gained insight into issues such as personal coping and resiliency:
Yes, I learned things about myself by doing things like this – ‘the life wheel’ activity [a workbook activity focussed on life values]. I saw things that I had structured but had not realized … I have always known about my barriers but I have never thought about them as much as I have in the programme. I have always known that CF is a barrier, but, until we talked about it on this page, I did not know just how much it was … I have someone to talk to and talk with you. To have someone here with me to talk about this … You definitely need a counsellor for this.
The CF Chatters programme supported and enhanced Chase’s already positive appraisal toward activity.
Summary
Chase is an exemplar of a young person who is living well with CF. Chase has largely overcome his early struggles to have an active role in his health care, and the avoidant approach that he once adopted to deal with the burden of CF. Chase is a motivated, intelligent, inquisitive, and socially conscious boy who is devoted to attaining optimal personal health with CF. Chase desires to take care of himself despite knowledge of the harsh reality of CF. Although infections frustrate him, he shows no indication of helplessness or futility, and engaging in treatment, physical activity, and good nutrition appear to be worthwhile time investments that enhance perceptions of autonomy over his health. While he foresees that he may need ‘treatment reminders’, Chase expressed no fear of transition to the adult CF centre in the future. Chase also displayed concern for the broader social world around him and its complexities – especially to help those that suffer – and a desire to see the world from others’ perspectives.
Layla’s story: ‘I never tell others what I really think’
Context and background
Layla is a 16-year-old girl with moderate CF. During the study, Layla was also experiencing excessive fatigue of unknown etiology. Layla is thin, pale, and fragile in appearance and her countenance can generally be described as one of sadness. She has great difficulty making eye-contact with staff and her voice lacks confidence. Layla was the only participant in the study who is a visible minority. Layla is from a two-parent, low socio-economic status suburban family. She resides in a housing complex with her many brothers and sisters and her parents, who immigrated to Canada from the Middle East a few years ago. Layla’s parents are unemployed and she ‘has no idea how they make their money’. With the exception of her youngest brother who also has CF, Layla’s siblings are healthy. Layla’s family adheres to the principles of fundamentalist religion. Although Layla expressed disagreement with the strict religious sanctions in her home, her relationship to her religious faith is contradictory. She enjoys attending the temple and other religious gatherings with her family. For Layla, religion is a ‘guide which keeps me on track and explains things’.
Layla’s story
Within the clinic, Layla is the quintessential ‘high risk’ or ‘difficult’ patient. Known as a trickster, clinical staff suspect that Layla fails to adhere to her life-saving treatment protocol and regularly procure medical evidence to indicate her propensity to tell falsehoods. She rarely tells the truth about how she is feeling or whether she is taking her medication and the staff suggest that she ‘lies to your face’. Layla has also mastered the art of the silent treatment and often does not answer questions, or rather answers with one- or two-syllable responses. Layla lacks initiative in matters related to her health and suspects that her health and treatment adherence will be worse upon transition to the adult clinic in two years. Rather than contacting the clinic when she is feeling unwell, she waits until she is very ill and relies on parents and staff to intervene. Thus, Layla has encountered largely preventable health deteriorations associated with the poor monitoring of CF. Layla’s poor adherence to her clinical treatment practices extend to multiple health behaviours and she is physically inactive and underweight.
In addition to poor personal health, Layla has a complex social world. With little command of the English language, and very strict parenting sanctions such as not permitting their children to attend social gatherings or mixed gender activities, Layla’s relationship with her parents is characterized by conflict. Although her parents did not participate in the intervention, Layla appeared to be imprisoned in a complicated cultural clash between two opposing world views, and her parents’ strict sanctions around social activities saddened her. Given that several of her siblings are very young, she has few points for identification with them. Layla has good relationships with two older sisters and two same-aged girl friends at school. Thus, Layla’s social network is small, and, interestingly, she does not derive social support from adults.
Layla spends between five and six hours doing homework in the evening and often goes to sleep between 2:00 and 3:00 am. However, her ability to concentrate at school for prolonged periods is compromised and her academic performance is mediocre. I was quite astounded by the sight of Layla’s weekly schedule. With little time for play, her scholastic efforts do not allow her to attain desired academic outcomes. I felt particularly dismayed when, on top of this gruelling homework schedule, Layla grudgingly admitted that CF often leaves her stricken with fatigue and bed-ridden at home. Layla’s scholastic aptitude for the future and career ambitions are poorly conceptualized. Although she stated that she wants a ‘job and a home one day’, she is worried that CF will thwart her attainment of these goals and she lacks passion. Layla unenthusiastically stated that she ‘may apply to college’. Although I repeatedly encouraged her to complete the necessary applications and tried to foster broader discussions about the benefits of education for women, I am not convinced that Layla will attend college.
Anti-health orientation
When Layla was a little girl, she generally adhered to the CF treatments that her parents administered without complaint but without any real understanding of why she was required to receive such treatment. Occasionally, she asked her parents why her older and younger sisters did not have to take medication like she did. Layla learned more about CF at the age of 13. In particular, learning about the fatal and genetic nature of the disease – that treatment did not lead to a cure and had to be performed for life – was particularly dismaying for Layla, and marked her descent into sadness and despondency:
When I was little, I would always have to do treatment and I did not know why. I would always look at my sisters and ask my parents ‘how come Ayesha does not do this? How come I only have to do it?’ But that is when you are little and you do not know why you have to do treatment. When I started to grow up – in grade eight and nine – when you start to learn more about CF. When I learned that it is actually inherited, you cannot get rid of it – there is no cure. That is when I started to slack off with treatment and to not care as much.
Unlike Chase, who simultaneously constructed himself as normal and not normal, the sense that she is not normal – an abhorrent and deviant body that is distinctly different from her healthy sisters – leads to a sense of distress. Perceptions of not being normal threaten her sense of self, and contribute toward an anti-health orientation. Like Jaime, the young man who sustained a severe, sporting-career-ending spinal cord injury (Smith & Sparkes, 2008), when she discusses her sense of non-normativity, Layla refers to her body in the third person. Indeed, she describes her body as an object in mechanistic terms:
Sometimes, you want to fit in with everyone else and to not be different. Not having to do things that other kids do not have to do. To live – to be healthy, and stuff. Sometimes, you desire to fit in.
How does CF make you not fit in?
Because you have to take all of these enzymes and stuff and sometimes you get sick more. It changes the way that your life is … and it makes it difficult to think of yourself as normal. I do not think of myself as normal. . .When I look at my sisters, I think that they are normal. They are healthy and they have no sicknesses and stuff. It makes you feel not so good.
Knowledge about the fatal nature of CF and perceptions of non-normativity have a cumulative effect, resulting in Layla’s disregard for treatment and her willingness to lie to health professionals about treatment adherence. The knowledge that there are no immediate negative consequences for skipping treatment, and that people with CF are required to do treatment for life irrespective of poor disease outcomes, constitute Layla’s rationale for treatment non-adherence:
If there was something that would get rid of CF, I would do it all the time [laughing]! It is not like that. With antibiotics too, it is something that will get rid of your infection or whatever, so I want to take it all and to finish it all. And you remember to take it on time and you care about taking it. But the thing is with this CF – it is ‘like whatever, I have to do it for the rest of my life anyways.’ After a while, it gets very irritating. It is like ‘I have to do this for the rest of my life? Screw it! Who cares! I am not going to do it anymore.’ You know that you have to do it all the time. You think of it and you say, ‘it is OK if I miss it once or twice. Because I will have to do it for the rest of my life anyways.’
However, Layla is cognizant that her treatment ‘lies’ to parents and staff members also contribute toward personal guilt and distress and is ultimately in no one’s best interest:
It does make you feel bad when you do tell the doctor that you do take the treatment. Sometimes, I will tell them that I have missed it once or twice. But, like, afterwards, you know how important it is and you know that they know too. When you do not do it, you feel bad.
CF chatters programme response
Despite her generally negative world view and resistance to talking with adults about her health, Layla tolerated the programme well and derived benefits. Although it required a great deal of encouragement, Layla attended all her appointments in the programme, attained all of her weekly physical activity goals, mastered the programme content, and demonstrated the ability to continue with the programme after getting sick. For instance, since Layla was completely inactive prior to the programme, she set physical activity goals that would facilitate reduced sedentary time, prevent movement losses, and improve participation in activities of daily living. She also displayed improvements in physical quality of life and reported enhanced knowledge about the self. The programme helped Layla to contemplate the potential benefits of taking better care of her physical health, including eating well, doing treatment, and staying active:
I learned that you have to stay on schedule with treatment and stuff. It is not good to leave things until they get worse. It is good to stay on top or things … I learned that exercise is really important for a lot of different things. Like physically and mentally too … yes, like the questions in the book make you think about things and the kind of person that you are. It gives you a reason to care about yourself. It makes you think that taking care of your health is way more important.
Like Chase, Layla appreciated the counselling-based nature of the programme and willingness of the interventionist to suspend judgment about treatment non adherence:
It felt good to talk.
How?
Just, I do not have to hide things. You can admit that that is how you are. I usually keep secrets because I do not want people to judge me for the way that I am. I am worried about what they will think or say … adults sometimes they just look at – they focus on the bad thing that you are doing and do not focus on the good things. The programme felt different. It was good! It was a good programme.
Summary
As a wounded storyteller (Frank, 2005), Layla is an intelligent but profoundly sad girl who rejects health and begs researchers to further study the experiences of those who do not adhere to treatment. Characterized by a prolonged sense of sadness and resignation, Layla does not always ‘see the point of taking care of herself’ if CF’s end result is always death. Unlike Chase, who had embarked on a process of deconstructing normalcy, Layla’s conceptualization of herself as ‘sick and abnormal’ as a result of CF is static. Supporting the literature (Admi, 1996), Layla ascribed extremely negative value judgments to herself for her non-normalcy and was willing to compromise her health and treatment.
Despite this, Layla made significant cognitive and behavioural gains in the area of physical activity, quality of life, and self-knowledge that exceeded those of the other patients in the programme. However, we believe that Layla’s important gains were temporary and attributed to the high level of intervention assistance and emotional support. Unless Layla acquires a strong social support network and learns to cognitively and behaviourally re-appraise CF such that she adheres to treatment, physical activity, and nutrition, she may continue to engage in risky behaviours that compromise her health.
Discussion
Illness narrative scholars have encouraged researchers to study stories of a different kind of suffering, and to make available those stories that do not fit the contours of conventional narratives (Sparkes & Smith, 2008). This study contributes toward the illness narrative literature in five ways. This study (a) illustrates how children with chronic and progressive diseases such as CF draw on multiple illness narrative types – in a sort of narrative bricolage – to articulate their experiences. This study (b) also draws our attention to the potential role of development in influencing the kinds of stories that sick children can tell. Drawing on the work of Carless and Sparkes (2008), this study (c) also begs consideration of the influence that illness narratives may have on physical activity experiences. Beyond the contributions of this study to the illness narrative literature, the findings (d) contribute toward scholarly discussions on treatment non-adherence in CF, and (e) the role of social position in influencing storytelling.
The findings extend the existing literature by illustrating how CF youth borrow on multiple illness narrative types to tell their stories. Rather than conforming to one of Frank’s (2005) three illness narrative types, Chase and Layla’s stories appeared to blend multiple narrative types in a narrative bricolage of sorts that encompassed restitution, quest, and chaos, as well as evolving forms. Despite similar ages, the realization of illness played out differently for Chase and Layla. As a child, Chase did not describe a period of normalcy and always knew that he was sick. He passively gave way to illness in a blunted ‘childhood chaos’, marked by parental dependence, lack of goal direction, passivity, and lack of awareness about his disease. Chase’s River Trip accorded to certain elements of the restitution narrative, marking his own personal overcoming story. Although previous body–self relationships were not restored (Sparkes & Smith, 2004) – indeed, Chase expressed disdain for his formal self – the River Trip aided in Chase’s self-realization for the first time. Chase today values health, knows what he wants, and expresses a robust self-esteem and vision for the future. Chase does not display the fundamentally changed values that are characteristic of the quest narrative. From a developmental perspective, the quest narrative may not be available to Chase, perhaps emerging as a story structure later when he has lived longer with CF. However, Chase is aware of the ever-present nature of his disease and the ways in which it limits his potential and saps his energy; he is thus cognizant that health is fragile. The development of a communicative and ethical body–self relationship – his desire to not look inward, and rather, to give to others that are ill and to listen to the stories of other CF children – were elements of the quest narrative that Chase displayed (Sparkes & Smith, 2004). In a more communicative body–self relationship (Sparkes & Smith, 2004), Chase desires to be closer to the CF community. Despite the fact that CF is a fatal disease, Chase borrowed from all three of the narrative types, displaying childhood chaos and adolescent restitution with glimmers of quest.
Unlike Chase, who did not describe a period of childhood normalcy, Layla recounted a period of time when she was confused by the fact that she had to do treatment unlike her siblings, but did not know that she was sick. This accords with some of the literature in which, despite affixing a disease label, one does not feel ill in the absence of other bodily indicators (Admi, 1996). Learning about the incurable nature of the disease, and that there is no respite even if one does treatment, has marked Layla’s rapid descent into an ‘adolescent chaos’ of sorts. While researchers need to explore the form that chaos takes in adolescence – and how it differs from adults – Layla did recount an empty present and a desolate future (Frank, 2000a; Smith & Sparkes, 2008). While she attends academic and religious classes and goes to the mall with her siblings, she expresses profound sadness and futility and the future appears desolate. In addition to raising the question of whether the restitution narrative holds weight for those with chronic and life-limiting illnesses in which a return to a prior self is impossible, the findings extend the existing narrative literature by illustrating how CF youth draw on many storied structures to articulate their experiences.
Our findings also support Glasscoe and Smith’s (2008) assertions by illuminating how development may influence storytelling. For instance, in their qualitatative study, Glasscoe and Smith (2008) recount the tale of a young boy with CF by the name of Daniel. Daniel displayed the fantastical thinking associated with the pre-operational phase of early childhood, believing that although CF is a foe, he could conquer it by cheating fate; this accords with his pre-operational developmental phase (Glasscoe & Smith, 2008). In contrast, the youth in our study dialogued in abstract terms and were able to draw judgments and conclusions without concrete evidence. However, unlike the child in Glasscoe and Smith’s study (2008), our participants were less able to modify thoughts with fantasy and to take on a vision of the world that accords with an egosyntonic view of what they want. Since development is an invariably complex and heterogeneous process unfolding differently for different children (Moola & Norman, 2011), the notion of distinct temporal phases is subject to critique. However, it is important that future researchers study the impact of development on health, illness, and physical activity storytelling, and not assume that children narrate illness in the same manner as adults.
Thirdly, our findings extend research undertaken by Sparkes and Smith (2004) and Carless and Douglas (2008), who assert that illness narratives may influence one’s orientation toward physical activity. For instance, in elite athletics, Sparkes (2004) found that one’s pre-illness sporting affinity largely provided the blueprint for later illness narrations. Similarly, Smith and Sparkes (2008) explored how disability is the antithesis of athleticism, and thus the failed disabled body is all the more distressing for those who once defined themselves primarily through their physicality. Extending a small but burgeoning area of research by Sparkes and Smith (2008) and Carless (2008), future physical activity interventionists must be aware of how the broader health and illness context – and the manner in which children narrate their experience of CF – profoundly influences the orientation adopted toward physical activity. For example, it appears that CF youth who display hopelessness are less likely to see the ‘point of being active’ and experience greater difficulty initiating and maintaining physical activity even though they might have the most to gain in terms of physical health. Thus, the delivery of physical activity recommendations by interventionists must be sensitive to the kind of health and illness stories that patients narrate.
Beyond the contributions of this study to the illness narrative literature, the findings facilitate a better understanding of treatment non-adherence in CF. In our study, case participant Layla frequently skipped treatment, stating that ‘there is no point’. In Layla’s explanatory model of disease, she either (a) does not understand or (b) value as important the notion of using treatment to slow the progression of a disease that is ultimately fatal. Layla vehemently asserts that if treatment led to a cure, she would adhere to it religiously. Layla feels cheated by the fact that CF treatment does not equate to remission or cure, and is thus readily willing to compromise treatment.
Non-adherence to treatment reported by Layla in this study accords with pre-existing literature that has examined similar behaviours in other CF patients (Wright & Morgan, 1990). For instance, in a qualitative study, Wright and Morgan (1990) recount the story of a CF patient by the name of Nancy. In her local explanatory model of disease, Nancy believed that ‘excessive water’ – rather than other conventional indicators of disease such as respiratory distress – was responsible for her bodily complaints. Believing that she was ‘drowning in water’, she rejected conventional treatments and doctors’ orders. Instead, she requested more diuretics, which she logically concluded – from her explanatory framework – would alleviate her sensation of drowning (Wright & Morgan, 1990). Her illogical treatment choices led physicians to construct Nancy as a problem patient.
Attending to patients’ particular explanatory models of disease, however seemingly nonsensical, may help physicians to support such problematic, non-adherent patients like Layla and others, whose sedimented system of beliefs guide treatment behaviours and choices that accord with their local explanatory models of disease, while rejecting conventional medical explanations. While treatment skipping is ultimately dangerous and compromises long term health in CF, future researchers could also consider what function and meaning treatment non-adherence in CF serves, helping patients to identify more positive and functional ways of communicating distress.
Finally, this study begs researchers to consider how social position and location, like race, gender, and class, influence storytelling. Chase and Layla are only 13 months apart in biological age, and, with an FEV1 (Forced expiratory volume in one second) of 85%, they have an identical ‘objective’ marker of disease severity. However, they are vastly different in terms of their social position. Born into a white, English speaking family, Chase enjoys many of the privileges associated with a middle class urban lifestyle, such as opportunities to attend residential summer camps, trips to Europe, and a great degree of social freedom. Further, his upwardly mobile parents are well-educated about CF, as well as research advancements in the field. As a result, they neither overemphasize the negative aspects associated with CF, nor downplay the importance of treatment. In contrast, Layla can be located at the intersection of socio-economic deprivation and cultural and religious barriers to optimal health care. With little disposable income, Layla explained that it would not ever be possible for her parents to enroll her in physical activity classes of her choice. Her home is cramped and overcrowded; indeed, Layla stated that the only pay-off associated with being sick is that her parents allow her to have her own room unlike her siblings. Layla’s parents do not permit her to engage in any mixed-gender social gatherings and impose – by Western standards – extremely strict standards for behavioural conduct, such as not being allowed to go out after school or on the weekends with friends. While Layla’s parents did not participate in the study, they appear to adhere to a non-Western model of disease that emphasizes fatalism and belief in divine causes of illness. Thus, certainly, Chase and Layla’s stories must also be studied by future researchers along the lines of gender, race, religion, culture, and social class. Whether a hostile social milieu has furthered Layla’s unrelenting chaos demands further study (Wilkinson & Marmot, 2003).
Conclusion
‘And we require, it seems, in our moments of courage or shame, at least one witness’ (Carol Shields, 1993, p. 334).
Qualitative research in psychology and the social sciences has contributed much toward our understanding of how people living with CF interpret their health and illness. Treading the line between the general and the specific, and exposing us to the minutiae of the teller’s life, case studies with CF children that seek to examine both their health and physical activity experiences are underutilized as a forum for learning. Informed by narrative inquiry, this study built on the work of Moola et al. (2011) to illuminate pertinent health and illness dilemmas that typical structured intervention approaches may not be sensitive to. This study sought to work toward this aim by asking how two children living with CF – Chase and Layla – understand their health, illness, and physical activity. Chase told a story of early childhood chaos, and later adolescent restitution and quest, and displayed a positive orientation toward physical activity pre- and post-intervention. In stark contrast, Layla enjoyed a period of relative normalcy before descending into adolescent chaos. Her pre-intervention physical activity attitude was negative. The findings extend the existing narrative literature by forcing us to consider (a) the relevance of conventional illness narrative structures for those that live with life limiting disease, (b) the role of development in shaping the stories that children can tell, and (c) the ways in which these stories may influence the adoption and maintenance of physical activity. As co-narrators, it is our hope that Chase and Layla’s narrations have illuminated health and physical activity stories of a different kind, those that lie outside of conventional emplotments (Sparkes & Smith, 2008).
Footnotes
Funding
This research received no specific grant from any funding agency in the public, commercial or not-for-profit sectors.