Family Management of a Chronic Health Condition

Design and Sample

Data used for this secondary data analysis were a subsample of 60 interviews conducted as a part of a larger study evaluating adaptation in adolescent with SB. An overview of the larger study is included to orient the reader to the sample, recruitment and data collection procedures used in this larger study.

Overview of the Larger Study, Adaptation in Spina Bifida

The overall aims of the larger mixed-methods study were to identify factors associated with adaptation outcomes for adolescents with SB and to understand the experience of living with spina bifida. The study was grounded in the Ecological Framework of Adaptation in Spina Bifida. This framework delineates the relationship of risk factors: (a) characteristics of SB, (b) demographic characteristics, (c) neuropsychological challenges] and protective processes: (a) adolescent resilience factors, (b) family resourcefulness, to adaptation outcomes: (a) physical, social, mental health, and health related quality of life (Sawin, Brei, Buran, & Fastenau, 2002). The original analysis of the qualitative data focused on the experience of adolescents and parents living with spina bifida (Bellin, Sawin, Roux, Buran, & Brei, 2007; Roux, Sawin, Bellin, Buran, & Brei, 2007; Sawin et al., 2003, Sawin, Bellin, Roux, Buran, & Brei, 2009).

After obtaining Institutional Review Board approval, the sample was recruited from a single SB clinic setting in the Midwest. Adolescent and their primary caregiving parent were eligible if the adolescent was 12-21 years of age, with no other major medical conditions, and no major cognitive delays. These dyads were invited to participate either by letter or personal invitation in the clinic. The adolescent and parent participated in separate interviews either in person or by telephone. During the interviews, data were collected from adolescents and parents using quantitative instruments to measure several protective processes and adaptation outcomes as well as qualitative interview that addressed the experience of living with SB. The interview guide started with the general question: “What is it like to be a teen with spina bifida?” Follow-up questions addressed specific life experiences such as everyday stresses, coping strategies, perceptions of self, family relationships, social relationships, self-management activities, responsibility for self-management activities across family members, what self-management adolescents thought they needed to achieve independence and future goals. Both adolescents and their parents were assured that they could end the interview at any time or omit any questions they did not want to answer. All interviews were conducted either in person in the family home or in select cases, over telephone by nurses who were experienced in interacting with adolescents who had a CHC such as SB.

Current Secondary Analysis

This secondary analysis was conducted using 25 of the 60 adolescent transcripts to determine if adolescent exemplars of the FMSF dimensions could be identified in the adolescents’ description of living with SB. Secondary analysis has been shown to be an appropriate strategy for asking unique questions of existing qualitative data (Hinds, Vogel, & Clarke-Steffen, 1997; Santacroce, Deatrick, & Ledlie, 2000; Szabo & Strang, 1997). Although the fact that the original research was conducted for different purposes may pose challenges regarding saturation, data still may contain sufficient richness in descriptions to enable credible results. The purposive subsample of transcripts was chosen to reflect the age range of the sample and the larger sample’s approximate gender distribution. In addition, because of the secondary nature of the analysis, each transcript was subjected to an initial reading and those transcripts with sufficiently thick descriptions identified by the investigator in the original study were included in the subsample. The resulting sample of 25 transcripts included adolescents from 12-21 years of age. Sixty-eight percent were female and the sample reflected all levels of SB lesion: thoracic high lumbar (40%), lumbar (24%), lumbosacral (16%), sacral (16%), other (4%). The number of adolescents who had a shunt (84%) was similar to that of the total sample (85%).

Data Analysis

The transcripts of adolescent interviews were analyzed for the presence of data elements that reflected the components and dimensions of the FMSF (Knafl & Deatrick, 2003). The process began with an open-coding approach and through an iterative process the data units coalesced into clusters under the preliminary coding labels. As this process progressed, the preliminary codes were compared to the labels and definitions within the FMSF for similarities. A decision was made to use Excel spreadsheets labeled with the components and dimensions of the FMSF as the format for the analytic grids. As the most recent version of the FMSF was based solely on data from parents, it was anticipated there would be a lack of congruence between the adolescent data and the existing labels and definitions of the dimensions. In cases where the adolescent data matched the essence of the dimension, but not the label or existing definition, the data element was placed within the appropriate cell in the spreadsheet and a clarifying label was added to make clear that the data were from an adolescent’s point of view. An example of a dimension that did not allow for the adolescent perspective is Child Identity. In this circumstance, the phrase self-identity was placed in parentheses next to the child-identity label in the spreadsheet to indicate the data were coming from the adolescent’s perspective.

In the cases where the adolescent data reflected the essence of the definition of the dimension (e.g., Parental Mutuality) but not a perfect fit for the parent-focused definition, the authors identified preliminary definition revisions from the adolescents’ perspective. For the majority of the dimensions, the revision consisted only of substituting the word “adolescent” for “parent.” However, for four dimensions: Child (Self) Identity, Parental Mutuality (mutuality of family members), Management Mindset, and Future Expectations, slight wording changes of the definitions were needed to accommodate the adolescent as reporter. When this process was completed, there were data within each of the cells of the spreadsheets that corresponded to a dimension within the FMSF. The labels and definitions of the dimensions that needed to be revised to accommodate the adolescent perspective had been developed. Finally, a summary table was created for side-by-side comparisons of the original definitions and the proposed expansion of the definitions. The data presented in the results section are organized using the proposed revisions (see Table 1).

Further, the original work on the FMSF in the 1990s focused on families with children who had a variety of illnesses such as diabetes. Within the FMSF definitions, the words illness and Illness View were used. The social movement that expanded the definition of illness, disabilities, and function (World Health Organization, 2001) focuses on a broader classification of these conditions. To broaden the definitions of the FMSF dimensions to encompass adolescents with conditions such as SB which are not considered as an illness, the authors of this article chose to use the term CHC rather than illness or Illness View throughout the description of the data.

Throughout the process, the primary author consulted with the second coauthor who is an experienced qualitative researcher and the third coauthor who is a researcher experienced with the population of adolescents with SB and their parents. The team met frequently to review and affirm the analysis and discussed the implications of the results for the further refinement of the FMSF.

Component I: Definition of the Situation

In the FMSF, four dimensions: Child Identity, CHC Views, Management Mindset, and Parental Mutuality, contribute to the family’s definition of the situation. The adolescents in this study had a clear sense of identity, the first of these dimensions, expressing both feelings of normalcy and feelings of vulnerability about themselves. Examples from the adolescent interviews revealed positive perceptions such as not feeling “limited” by their condition, as being someone who is “friendly, patient, nice,” and in one case, that having SB had a “pretty positive influence” on the adolescent’s self-perception. Another said: “I’m a normal person. I’m just in a wheelchair.” One participant shared his thinking about self-identity in this positive way:

I don’t let my condition limit me to what I can do. I never have . . . In some aspects I wish I wasn’t like I was, but then I think maybe there was a reason why I’m like that . . . I really feel that I was blessed with the way that I am.

However, difficulty with mobility, and other people’s perception of people in wheelchairs seemed to cause some of the adolescents (n = 11) to think of themselves in a vulnerable or negative way. As one participant said:

I like the fact that I’m good at archery, but I don’t really like the fact that I’m in the chair. I mean cause a lot of times kids look at the chair, not me, so I get judged by it.

The adolescents described parental behaviors that reflected their parents’ emphasis on normalcy and capability or vulnerability as presented in the Knafl and Deatrick (2003) definition. Adolescents in this study talked about how their parents interacted with them, encouraged them, or set limits in an effort to promote normalcy. One adolescent responded:

My parents try to keep me normal. They don’t point out all the differences . . . They encourage me to do things that people with spina bifida normally don’t do . . . I mean they try to just get me out in the world so I'm not enclosed in my own little world.

These examples from the adolescent data support the definition of Child Identity presented by Knafl and Deatrick (2003). Yet it is apparent that these adolescents also have a clear perception of self-identity. The recognition of the adolescent’s ability to articulate self-identity in the context of a CHC may prove useful in the expansion of the definition of the Identity dimension of the FMSF.

Adolescents’ view of the CHC, the second dimension under Definition of the Situation included beliefs about the seriousness, predictability, and to a lesser extent the course of the CHC. Adolescents in this study who were born with SB did not discuss the cause of the condition, which is part of the original FMSF dimension. Questions regarding cause were not a part of the interview guide. However, lack of reference to the cause of SB in the transcripts may also be influenced by the congenital nature of SB and the lack of clarity about causative processes.

The adolescents’ outlook on having this condition was primarily a result of attempts to manage when they were “feeling tired” or “in pain” or “never having time off from having to take care of myself.” Adolescents with SB seemed to view the seriousness of their condition in the context of the ways in which having the condition has limited mobility and what they are able to do. That is, they did not focus on the CHC itself but rather their ability to function with their CHC. There were missed opportunities to “stay overnight with friends”; reports of being unable to “do things I’d like to do like playing basketball or doing other activities” and the impact of limited mobility on their independence. For some adolescents, the daily activities such as self-catheterization became routine and for others, these activities were constant reminders of the seriousness of the condition. For the adolescents in this sample, the social consequences, such as interference with school attendance, were central to their definition of the seriousness of having SB. One adolescent described this perception of seriousness in the following way: “I was having problems at school because I was sick so much and in the hospital, then I would get behind on my work and I’d have trouble with the teachers.”

A few of the adolescents (n = 4) spoke about the unpredictability of the condition. Specifically, they reported pain, problems such as urinary tract infections, unanticipated shunt revisions, or other surgeries. Individuals with SB are living well into adulthood and there is much uncertainty about future health problems for those aging with SB as reflected by the participant who shared: “I’m kind of worried about good health because I don’t know how good it will be. I know I’ve got it and everything, but I’m not sure if it will get worse, or if it will just stay the same like it is now.”

The course or trajectory of SB varies substantially across individuals. For many, there are numerous orthopedic surgeries that occur most often in early childhood. Urological surgeries are more typical in childhood and adolescents and neurosurgical procedures are most often for shunt revisions and occur when the shunt is not functioning. Although there is not a typical course, the number of surgeries was often cited as important in the adolescents’ view of their CHC. One participant indicated: “I don’t want to have another surgery. I had my 15th surgery in March.”

The third dimension of Definition of the Situation is Management Mindset, which includes the perceptions of adolescents with SB about the ease or difficulty of implementing the treatment regime and their ability to manage effectively. A major focus of these adolescents’ Management Mindset was perceptions of the ease or difficulty of carrying out their bladder- and bowel-management programs, which were common activities in the treatment regime. The most common way to manage the neurogenic bladder is with clean intermittent catheterization 4-6 times a day and there are a wide variety of bowel programs depending on the individual’s response. With appropriate programs, social continence can be achieved. A few adolescents (n = 4) were “very happy with the bladder program and the bowel program.” One adolescent was neutral on the subject stating that the way to increase satisfaction would be “just quit having accidents altogether.” Another reported “What would make me satisfied is if I didn’t have to cath.” The omnipresent nature of these responsibilities for self-care may be viewed as a part of the difficulty of managing this condition. One adolescent described the additional stress the individual may feel when given responsibility for the routine management of this CHC: “Sometimes it gets real emotional; I went through a couple of years I just wanted to die. I didn’t want to do anything. It’s a lot of responsibility for a 10 year old to have.” Similarly another adolescent stated “I never have time off from having to take care of myself.”

The adolescents described how they and their parents communicate and make decisions, or manage the physical care required to maintain health in someone who has SB. They described the skills and techniques, such as bladder catheterization, placement of braces, or use of the wheelchair that either they themselves or their parents had to learn to manage their condition. Two adolescents with SB described the difficulty in managing the condition not as the treatment regimen itself so as much as the mobility challenges presented by the condition.

Being a teenager with spina bifida, it’s rough. You know there are kids that have got it worse, but it’s rough. Getting relationships that’s not working cause you’re in your wheelchair, but you deal with it. I usually go into my room and turn my music on real loud and cry for about half an hour.

It’s really hard being a teenager with spina bifida, like getting around and in certain places with certain people looking at you and saying “Well she’s not right because she’s in that wheelchair.”

Thus, even if the adolescent was able to manage their condition effectively, the cost or difficulty of doing so was high.

The final dimension in this component is Parental Mutuality. As the focus of this analysis was on the adolescent’s perspective, data that addressed adolescents’ perception of Parental Mutuality and/or family mutuality were included. Adolescents were able to describe characteristics of their parents, and their own views of the parent to parent relationship, and/or parent to adolescent relationship which may add insight into the shared or discrepant views present in their family relationships.

In most families, adolescents reported mutuality with shared views of the CHC, the roles of family members, and approach to the management of the CHC. Participants reported parent involvement in monitoring but also making joints decisions about optimal health and everyday life as one adolescent reported “If I forget about [cathing] she reminds me: “When was the last time you cathed yourself?” or:

I usually know when things run out and stuff, so I tell them that I need something. Then they get on the phone and order it, and they usually go pick it up at the drug store or it comes in by mail. (Sawin et al., 2009)

The adolescents also provided insight into the issue of privacy in the family and the struggle between parent and adolescent. In some situations, the adolescent perceived that parents overstepped the adolescent’s boundaries, which can have implications for the possibility of conflict in the family’s management attempts. One adolescent described tension existing in the relationship with her parents due to their discrepant views:

My dad is easiest to be around. I feel sometimes that mom thinks she knows more than I do about myself. I feel sometimes that she doesn't respect me, what I say. One thing that really annoys me about her is, she’s always got to get into my catheters . . . I’ll come home and they'll be laying out, I’m a private person. I don’t like my personal stuff laid out. (Sawin et al., 2009)

One adolescent described his observation of the discrepant views his parents have related to him:

My mom and dad think I’m completely different. Mom, she’s protective of me. She tried to tell me she doesn’t want to be a big influence on me. She wants me to make my own decision, but she also wants to tell me her experiences so I can work with that. Dad’s pretty much like you’re going to do it, do it, experience it, get over it, make your mistakes, let me ground you, no big deal.

Another adolescent described the triadic relationship present in families when talking about the discrepant views his parents have of him as well as how his view of himself differs from that of his mother:

I’m a good swimmer. I love to swim and mom’s like well be careful, you might drop (sibling). I don’t think you can hold him. There’s certain things she tells me that drives me insane. Like my dad tells me that this summer he wants me to walk, cause I’m getting pretty good. Mom doesn’t let me try anything. It’s too hard. I’m 18, it’s not too hard.

Component II: Management Behaviors

This component of the FMSF has two dimensions, Philosophy and Management Approach. This component was not the focus of the larger study of adolescents with SB and adolescents were not particularly articulate about a philosophy that guided their overall approach to self-management. For some, self-management was not described as an explicit goal, although there was a consistent emphasis on treatment of the adolescent as “normal.” Although a clear philosophy was not always articulated, the behaviors demonstrated by parents in working with the adolescent to manage the condition do reflect the underlying philosophy and includes the adolescents as a participant in managing the condition. There is evidence in the interviews with adolescents that they were able to identify parental goals or priorities with which they agreed. As was noted by one participant:

My mom wants me to have friends and be liked and everything so she pushes me like makes sure that I keep the lower part of me clean cause it's kind of hard because I leak . . . she pushes me to do that.

The adolescents also shared their unique goals priorities and values. For example one adolescent reported:

I set my own limits as far as what I can do and what I can’t do. If I don’t think that I can do something well I elect not to. If I think that it’s going to be a challenge then I’ll go ahead and try it I’d tell that you do what you think you can and if you don’t think you can do something, that’s your choice to make.

Another participant clearly stated her personal goals:

I told mom my goals right now, cause they told me I would never be able to walk without crutches, and I probably would not be able to drive. Those are my goals right now that I'm shooting for.

The adolescents described how they and their parents communicate and make decisions, or manage the physical care required to maintain health for someone who has SB. In some instances, the adolescent described how they negotiated the management routine with their parents over time and shared their perception of how the family developed strategies for managing the routine. However, one young woman responded that she does not think she and mom deliberately decided how to manage her needs in her daily routine: “I think it's just happened.” In contrast, some of the adolescents clearly described the strategies and routine the family used to manage the special challenges a person with SB may have. As one reported,

Things I can do myself are brushing teeth, washing, combing hair, bathing; dressing independent on bed or floor, I need Mom’s help (to get) in chair. I self cath; if I’m in a hurry mom does it. She manually extracts stool.

In addition, planning ahead to manage an unexpected situation or accident occurs in some families:

If I’m going to be gone for a long time, I usually have supplies with me.

In case I have a bowel accident or my catheter would leak, I carry pads with me . . . I carry wipes and everything . . . If it’s something where I’ll be gone most of the day, I take an extra pair of pants and underwear.

Unlike parents in previous studies of the FMSF, adolescents did not seem to focus on strategies to incorporate condition management into everyday family life. The adolescents’ focus is on their own everyday life, not the overall family. Developing strategies for incorporating the condition into family life may be a primarily parent responsibility or may not be evident in this secondary data as it was not a specific question in the interview guide.

Component III: Perceived Consequences

The third major component of the FMSF, Perceived Consequences, includes two dimensions, Family Focus and Future Expectations. Adolescents in this study were able to provide personal perspectives of CHC management to enhance this aspect of the framework. Some of these adolescents (n = 5) perceived that the family has not really been affected by having a member with SB and see their family as very adaptable: “I think my family has learned to take my having spina bifida pretty well and just go with whatever happens.” Another participant said, “I don’t think my family has been affected by my spina bifida,” or if they have “they don’t tell me.” In contrast, another participant recalled the changes the family made after a birthday party, “I had a difficult experience at my birthday party, groups of people ignored me . . . they were leaving me out and I wanted to be in the group. We manage this by inviting only one person now.”

Another adolescent had a clear understanding of the balance between CHC management and other aspects of family. He was aware of the sacrifices made by his parents and shared the difficulty he experienced in learning that his family struggled financially due to his condition. His mother had to make a major change in her life to maintain the family balance:

My having spina bifida, it's changed a lot of stuff which I’ve found out recently. That was one of the reasons my mom quit her bakery business and took a job at the factory, cause she had to pay the bills and take care of my doctor bills. It kind of makes me feel kind of bad, mom really enjoyed it. I know it’s not my fault, but it makes me feel kind of bad cause she loved it.

The adolescents related not only how they see their future but how their individual circumstance may affect the future of their family of origin. A few of these adolescents (n = 3) focused on the more immediate “future” as evidenced by statements such as “I’m going to start a job when I turn 15” and “The job I would like to get is bagging at a grocery store.” Others shared their thoughts about how they envision their more distant future life. Their hope for the future includes living independently, further education, engaging in a career, getting married and having children. In some of the responses, there was concern for the future and others sounded eager to experience it. One female participant shared her thoughts for the future:

If I have a baby I won’t worry about whether it will be normal or not . . . For the baby’s sake I hope it’s normal. That’s why I want to marry someone who’s not handicapped. If I end up marrying someone who’s handicapped, I’m not going to have any kids.

Other worries were about the ability to be successful at self-management to live outside the family of origin as exemplified by this adolescent statement:

I don't know if I’ll ever live on my own. I would need to learn to call my own doctors, call for my own diapers, try to have bowel movements on my own, do my own bath, put on my own clothes, I think that’s it. I don’t know what happens after graduation.

These statements indicate that the adolescents had expectations concerning their own future, consistent with the FMSF.

From the adolescent’s point of view how the family experiences the future will likely center around the adolescent’s ability to “live away from home.” This could have implications for the family of origin, especially if the members envision the adolescent will be out of the house in a few years. Three of the adolescents statements were: “I’ll live here”; “I'm not sure if I can live away from home” and “I don't think I'll ever live by myself.” One adolescent was insightful and realized that his parents are aging and impact it might have on their helping in management of the CHC: “I’ve always got my parents until they get older. They won’t be able to help me as much, but I don’t worry about it much.”