Abstract
Abstract
Background:
The aim of this study was to explore the professional experience of caring for children and adolescents with lymphedema and to explore the way in which they understand and implement self-management strategies and the influence of their own self-efficacy beliefs on this process.
Methods and Results:
Participants were recruited during an educational camp for children with lymphedema. Three individual semistructured focus groups were undertaken in English, French, and Italian with simultaneous translation. Data were analyzed using interpretative phenomenological analysis (IPA). Analysis of the data produced three superordinate themes: professional concepts of self-management, professional practice, and redefining the cornerstone of lymphedema care. An additional seven subthemes were as follows: readiness to self-management, professional perspectives on self-management, defining success and treatment failure, emotional burden, traditional views on complex decongestive therapy, new ways to practice, and sole practitioner versus multidisciplinary teams.
Conclusions:
The purpose of the study was to explore the challenges professionals face when introducing self-management to children and adolescents with lymphedema and their parents and to explore their own sense of self-efficacy in approaching this. The research allowed in-depth discussion about the ways they conceptualize self-management and faced professional challenges. The research highlighted the need to define what is considered an acceptable outcome within a complex and uncertain condition and the self-management strategies that are needed to support this.
Psychosocial Impact on Families
Research indicates that lymphedema impacts on the lives of children and their families, with greater difficulties occurring during adolescence. 1 However, little is known of the challenges of providing daily treatment and its impact on family life. Even less is known of the professional challenges of managing children and adolescents within the context of different health care systems and service models. The cornerstones of lymphedema management have been traditionally defined as a set of techniques (complex decongestive therapy [CDT]) involving care of the skin, exercise, compression, and manual lymphatic massage, a specialized form of remedial massage. 2 Treatment has been split into two phases. The first involves an intensive treatment, provided by professionals, that is often delivered for several weeks in which the goal of treatment is rapid removal of fluid and stabilization of the skin. The second phase is defined as maintenance in which in an ideal world, parents take on daily responsibility for delivering a modification of these techniques.
Self-Management in Lymphedema
A systematic review of activities or treatments that patients used in lymphedema self-management was undertaken using Orem's self-care deficit theory. 3 The review concluded that self-management does not include professionally delivered treatment and that control of swelling is only one outcome of treatment with symptom control of high importance. Richard and Shea support the importance of managing symptoms and gaining a level of control over the condition. 4
The dichotomous view that professional treatment of lymphedema is separate from patient self-management is difficult to reconcile as both frequently coexist. Traditional techniques, such as compression therapy, are based on professional procedures and are adapted and taught to children and parents. Procedures are largely based on adult recommendations without clear articulation of what should be adapted despite different challenges. Moffatt and colleagues in a study of CDT in adults highlighted the clash between professional training in CDT and the inability to predict which patients would respond to treatment. 5
Aim
The aim of this study was to explore the professional experience of caring for children and adolescents with lymphedema, a rare disease, and to explore the way in which they understand and implement self-management strategies and the influence of their own self-efficacy beliefs on this process. The study was nested within a larger study to explore the enablers and barriers to self-management in children, adolescents, parents, and professionals.
Research Setting
Professionals were recruited during an international educational camp run under the auspices of the International Lymphoedema Framework (ILF) for children with lymphedema in Turin, Italy (2017). The sample included a mix of clinical teams and individual practitioners from Italy, France, and southern Ireland. All professionals recruited were involved in the care of the children and adolescents attending the camp.
Ethics
Approval for this study was given by the University of Nottingham Faculty of Medicine and Health Science Ethics Committee. As it was not an intervention study, no formal ethical approval was required in Italy. The research was performed in accordance with international research standards, including the Helsinki Declaration. 6 All participants gave informed consent and were made fully aware of their right to withdraw from the study if they wished to do so. All study information was translated and back-translated into the different languages to ensure accuracy with English.
Methods
All professionals from each country attending were invited to attend a focus group and could participate in their native language, or a second language if they preferred. Group size was determined by the numbers wishing to attend and therefore the size was not limited.
Two researchers moderated the groups, which were simultaneously translated so that they could be undertaken in their native language (English, French, and Italian). The researchers wore headsets to ensure they understood the translation. The moderators of the focus groups undertook other research roles during the camp.
Procedure
Following consent, participants were invited to introduce themselves and explain their roles in relation to the children attending the camp. The Focus groups were semistructured with initial questions focusing on the following: the ways in which they selected children to attend the camp, how they viewed self-management in lymphedema, the professional challenges they faced, and the way in which they addressed these issues. Following this general discussion, questions were responsive and tailored to each individual group. Finally, the researchers summarized the main points raised and asked whether participants' thoughts had been summed up adequately and whether anything had been missed. All focus groups were audiotape recorded and lasted between 1 and 1.5 hours. Immediately following each focus group, both researchers met and completed a reflective diary of the event to capture initial impressions that may have been lost.
Data analysis
Each focus group session was transcribed verbatim and interpretative phenomenological analysis (IPA) used. 7 The first transcript was read several times, the left-hand margin being used to annotate what was interesting and significant about what the respondents said. Once this had been carried out for the whole of the first transcript, attention was returned to the beginning of the transcript with the right-hand margin annotated with emerging themes. These themes were then taken back to the original transcript to validate their existence within the text. Emergent themes were then listed on a sheet of paper and studied for connections between them. All the themes were clustered together to produce a set of superordinate concepts. As the clustering of themes emerged, they were continually checked in the transcript to make sure they were evident in the primary source material.
The next stage was to produce a coherently ordered table of themes to establish which most strongly captured the respondents' issues or concerns. The clustered themes were given names that represented their overall superordinate theme and an identifier was added in each instance to aid the organization of the analysis and facilitate checking back to the original transcript. During this process, themes were dropped if they did not fit well into the emerging structure or were not very rich in evidence. Themes from the first transcript were then used to ordinate the analysis of subsequent transcripts. As such, repeating patterns were established, but the emergence of new issues was also recognized. Data from each country's focus group were then compared across the three transcripts to explore the cultural differences emerging.
To determine reliability, an additional researcher undertook independent thematic analysis of the verbatim transcripts. Both researchers discussed the themes and subthemes they had identified, and agreement was sought when meaning was deemed the same but the language used was different so that informed consensus was achieved.
Results
Participants (N = 14) were asked to discuss in as much detail as possible how they viewed self-management for children with lymphedema and their parents and how they addressed this within their practice. Participants' accounts clustered around three superordinate themes: professional concepts of self-management, professional practice, and redefining the cornerstone of lymphedema care, with an additional seven subthemes: readiness to self-manage, professional perspectives on self-management, defining success and treatment failure, emotional burden, traditional views on CDT, new ways to practice, and sole practitioner versus multidisciplinary teams.
Tables 1–3 outline the superordinate themes and subthemes arising from the data from each country with supporting quotes.
Superordinate Themes (1) and Subthemes (2) with Supporting Quotes (English)
N = 3 participants.
CDT, complex decongestive therapy; P, physician; T, therapist.
Superordinate Themes (1) and Subthemes (2) with Supporting Quotes (French)
N = 6 participants.
P, physicians; Psy, psychologists; T, therapist.
Superordinate Themes (1) and Subthemes (2) with Supporting Quotes (Italian)
N = 5 participants.
P, physician; T, therapist.
Professional Concepts of Self-Management (1)
Parent readiness to self-manage (2)
Professionals varied in the methods they used to appraise whether they considered parents and families were ready to engage with self- management. Appraisal methods varied from completion of a questionnaire to interaction over a prolonged time with the family that included assessment by a psychologist. There was overall concern that introducing self-management strategies too quickly could easily overwhelm families who were already facing anxiety and uncertainty over their child's condition.
Readiness was also influenced by the professional's confidence in dealing with high levels of clinical uncertainty in the control of the child's condition and the possibilities of complications occurring due to inappropriate parental decisions. Sole practitioners were concerned about their isolation and vulnerability in making these decisions without access to a multidisciplinary team. Within teams, the decisions concerning whether families were ready to engage with self-management were made by doctors responsible for the child's care and endorsed by psychologists rather than by a therapist. This differs from the experience of sole practitioners who were often therapists and were therefore facing these decisions alone. Some stressed the importance of parents supporting each other in self-management because of their unique experiences of having a child with a similar condition. There was recognition that this included parental solutions to problems that may differ from the advice given by professionals.
Professional perspectives on self-management (2)
Many professionals defined self-management in relation to the ability of the family to be independent from continuous professional care and for the child to have a normal life. However, there was a deep concern that families had a direct access to professional advice when they needed this and that they remained connected to the services rather than being discharged.
Therapists discussed self-management in relation to the techniques such as exercise and compression that are part of CDT, and the challenges of parents taking on these roles. Adherence to these procedures was often described in a causal relationship to treatment success or failure and was attributed to the level of parental engagement. Attitudes to the strictness that parents should adopt in self-management varied considerably and suggested this was linked to the professional ability to tolerate uncertainty. Some reported that continuous adherence to a set of techniques was directly linked to the control of swelling, while others were more relaxed in their assessment of this.
The integration of self-management into providing professional care was seen to be time-consuming and required continuous engagement with families. There was a strong belief in the need for a therapeutic relationship based on trust that could not always be rapidly or easily established. The challenges of having adequate time for families were discussed by professionals from all disciplines and countries.
Traditional methods of training about the treatment of lymphedema left therapists feeling ill-equipped to address psychosocial issues. This was exacerbated by the challenge of integrating this aspect within clinical visits, which were focused on providing clinical techniques, often leaving little time to address the wider issues. Physicians also discussed the difficulties of making decisions about the role parents could play and the creative solutions they had to take. This included admission of the child to hospital if parents were not coping.
The concepts of self-management were understood differently and ranged from paternalistic attitudes to following professionally prescribed treatment regimens to flexible approaches that sought solutions with the families. Self-management was often conceptualized as a set of techniques or procedures that parents would have to undertake rather than by a broader definition. Professionals from all countries recognized the importance of providing correct information for parents who were facing an array of information that was complex, rapidly evolving, and could be incorrect or misleading, leading to escalating parental anxiety.
Some approached self-management in a formal way and provided training, while others viewed this more fluidly. The camp was viewed as a multifaced intervention that would enable parents to cope better with their child's situation as well as breaking their sense of isolation. The professional views on self-efficacy and the ability of parents to succeed with self-management were complex and integrally linked to the judgments they made on successful or failed outcomes of care.
Professional Practice (1)
Defining success and failure of treatment (2)
The findings from this study indicate that stability of swelling measured by limb volume remains the dominant outcome of treatment across all services and countries. However, the different professional responses to interpreting these measurements suggest that they are seeking a “good enough” outcome. This is more nebulous to define and influenced by many clinical and psychosocial issues within the patient and family and within professional belief systems.
Poor control of swelling in children who are clinically stable without frequent complications and who appear to be developing normally is viewed with less concern than those who are not. Experienced clinicians working within teams appear to show greater tolerance to this than sole practitioners. There is evidence that professionals struggle with families in whom there is a clash in the goals of treatment between the child and parent, particularly if these expectations appear unrealistic. The importance of the child or adolescent leading a normal life was of critical importance but appeared difficult to quantify. Attitudes to adherence to treatment indicate that some professionals define poor outcome with reference to poor adherence.
Emotional burden (2)
Professionals describe a high emotional burden. During all focus groups they became distressed and expressed their surprise at this. The emotional burden was a result of a complex array of issues and was present in those working within teams and by sole practitioners. The level of uncertainty concerning many of the children's future was difficult for many to manage.
Professionals felt overwhelmed by parents in whom there was an expectation of cure or stability and in those who expressed frustration with the limitations of the current knowledge and treatment options. Relationships with children and parents were often deep and led to some being concerned of an overattachment and an inability to manage when treatment outcomes were not as they wished or when parents or children rejected help and sought other solutions.
Professionals as well as children and parents appeared to be seeking a “cure” and were therefore facing their own limitations and feelings of impotence. Teams appear to share the dilemmas together describing this as a “psychodrama,” whereas individual practitioners speak of isolation and being emotionally overwhelmed and ill-equipped to cope. The ability to tolerate ambiguity and uncertainty is a dominant factor underpinning the emotional responses.
Redefining the Cornerstone of Lymphedema Practice (1)
Traditional views of CDT (2)
The perspectives held by professionals about the role of CDT in children and adolescents were complex. There was some evidence that this was influenced by the underpinning professional background. Examples of this were seen in physiotherapists who talked of the importance of maintaining mobility. Nurses and physiotherapists were deeply embedded in the methods of CDT training they had received. Physicians also discussed CDT and the need to adapt the protocols they used in “a trial and error basis.” They appeared more able to step back from CDT and consider the wider issues but expressed the burden of overall responsibility for the child. The burden of finding an effective adaption that children and parents could manage was evident in all focus groups irrespective of the country. Teams were prepared to share how these dilemmas impacted on the team and could result in tension that was not usually discussed. Access to a psychologist was discussed as being a vital component to assist both the patient and teams to understand the complex issues they were facing.
New ways to practice (2)
The experience of the camp offered new ways for professionals to consider delivering care and support for families. The opportunity to connect with families during social events further developed the relationships they held with them in a nonhierarchical way.
Sharing the difficulties as well as learning together appeared to be a cathartic experience for sole practitioners as well as teams, and was not readily available in clinical practice. While parents sought the advice of professionals during the camp, there was also evidence of how professionals valued the opportunity to discuss difficult challenges together, as well as developing new approaches to activities such as aqua aerobic sessions.
Therapists working alone explored the difficulties they faced in influencing physicians who did not listen to their recommendations or prescribed incorrect treatment approaches. They faced the dilemma of parents who had been wrongly advised on sole treatments such as manual lymphatic massage use without compression. They discussed how much value there would be in providing ways to support themselves through networks and clinical supervision.
Professionals were challenged to explore how problems such as mosquito bites and the fear of infection were of much greater clinical significance than they appreciated. Hearing the stories from parents about the control of their child's swelling challenged their assumptions about how they defined “normality” and the significance of the problem to the family.
An underpinning ethos behind the camp is the ability to break the cycle of isolation felt by families and an ability to empower them with the strategies to manage more effectively. The research with professionals indicates a mirror image of the need for support for professionals who are facing complex issues and prolonged uncertainty of outcome.
Sole practitioner versus multidisciplinary team (2)
The research indicates that professionals face many challenges whether they are sole practitioners or working in large teams. Large multidisciplinary teams were able to share the burden between them but also recognized they faced challenges of working effectively together. Psychology support for patients and professionals emerged as an important issue throughout the camp.
Sole practitioners were frequently therapists who expressed the vulnerability they faced of managing the complex patients in their care when they had no access to specialist advice. Despite this, the challenges of managing these patients were evident for all professionals irrespective of where they worked and the country they were based. Professional boundaries of practice vary between countries and the levels of autonomy these roles require. The research would indicate an urgent need to define what is required in specialist practice and service models and define how children and families can best be supported given the national and international variations in health care that will continue to exist.
Discussion
This study explored the professional experience of caring for children and adolescents with lymphedema, and the ways in which they understood and implemented self-management strategies. Analysis of the data produced three superordinate themes: professional concepts of self-management, professional practice, and redefining the cornerstone of lymphedema care. From these an additional seven subthemes were identified: professional readiness to self-manage, professional perspectives on self-management, defining success and treatment failure, emotional burden, traditional views on CDT, new ways to practice, and sole practitioner versus multidisciplinary teams.
The research showed the difficulties professionals have in conceptualizing self-management and the clash that exists between the traditional views of CDT and self-management strategies. Research has shown that patients with lymphedema navigate a complex path that makes adherence to treatment difficult. The intrusiveness on daily life may contribute to the low levels of adherence reported in adult studies. 8 This research found that professionals differed in their views on adherence and whether this contributed to unstable swelling and complications.
Multiple studies in lymphedema show that while the traditional primary focus is control of swelling, this is not the only troubling symptom with altered sensations, psychological distress, changes in body image, fatigue, and functional limitations leading to reduced activity.9–13 The benefits of adherence to treatment can be expected to therefore influence beyond control of swelling to other aspects of life and are therefore of critical importance for professionals to understand and integrate into clinical practice.
The study found that there is considerable emotional burden from providing care. Central to this is the uncertainty of outcome and difficulties in being able to define the expected pathway for children and the consequent impact this placed on practitioners. The importance of uncertainty in managing chronic illness has been known for many decades. Uncertainty is the inability to determine the meaning of illness-related events and occurs in situations where the decision maker is unable to assign definite values to objects and events and is unable to accurately predict outcomes due to a lack of information. 14 Because uncertainty indicates the situation is vague and ill defined, there is potential for many diverse evaluations and conclusions about the uncertainty. While the focus is frequently on the outcomes of care for patients, nevertheless, the professional response to this uncertainty has been shown in adult patients with lymphedema to cause professional anxiety and an inability to define a set of reasonable outcomes in a complex patient population. 5
A central concept in self-management is self-efficacy, which has been defined as the confidence to carry out behaviors that are necessary to reach a desired goal. Self-efficacy is enhanced when patients succeed in solving patient-identified problems. People's belief that they can take control over their condition is of special relevance to self-management. 15 Research has shown that motivational, cognitive, affective, and physiological processes affect self-efficacy beliefs and alter health outcomes such as symptoms, physical and emotional well-being, and enhanced social activities. 16 The perception people have of their self-efficacy also affects their thought patterns and ability to manage stress and depression. 17 People's belief systems have been shown to affect both acute and chronic illness outcomes, including the degree of benefit they perceive from a health intervention. 17
Lymphedema management (CDT) is typical of many conditions developed in an era when an acute illness model still predominated, and concepts and structures continue to influence the way in which care is approached.18,19 Lymphedema may be defined as a chronic disease, however, treatment with CDT is based more on an acute model of care with interventions delivered during acute exacerbations. This may influence the ineffectiveness of health care systems designed to treat acute disease rather than chronic disease.
Sound management of lymphedema requires participation of patients and families at most levels of health care, from understanding the disease process through to applying self-management strategies, and requires a functioning participation between the patient, family, and the professionals. This study found that professionals struggled to make decisions about when and how to ask families to engage with self-management, with concern for the parental ability to manage additional burdens of care. The study indicated that professionals from all disciplines struggled with their own self-efficacy beliefs in the face of children and adolescents in whom the future was uncertain. This resulted in a tension between preventing deterioration through ensuring self-management techniques were used to prevent deterioration and an awareness of the burden placed on families.
The educational camp was seen to play a complex role in addressing the issues of engaging parents in self-management and highlighted the diversity of challenges faced by all professionals. Central to future research is the description of self-management practices that can be evaluated for their efficacy and impact on the wide group of stakeholders involved in successful support of children, adolescents, and parents who face this important but neglected condition.
Study limitations
This study has several limitations. It only represents the views of the professionals who agreed to take part in the focus groups during an international educational camp. As such, they may not reflect the views of others who do not access such camps. It is well acknowledged that professional education in lymphedema practice is highly varied with a lack of evidence in many aspects of treatment. The views of the participants will be influenced by many factors, including their professional background and the way in which they have been trained to asses and treat patients with lymphedema. There are many different service models operating internationally with a variation in roles and responsibilities. Caution should be given to the generalizability of the research, which must be extended to a wider participation. Despite this caveat, the study shows that many of the issues raised were common across the groups, indicating that self-management in this is an important professional issue.
Conclusion
The purpose of investigating the experience of professionals working with children and families with lymphedema was to give a greater understanding of the challenges they face and their own sense of self-efficacy. The focus groups provided a forum that allowed in-depth discussion about the ways they conceptualize self-management and faced professional challenges. The research highlighted the need to define what is considered an acceptable outcome within a complex and uncertain condition and the self-management strategies that are needed to support this.
Implications for Clinical Practice
The research has shown an urgent need to address the following issues:
Develop multidisciplinary services for children in all countries. Provide clarity on self-management strategies in lymphedema. Define a range of outcome parameters that extend beyond control of swelling to psychosocial health. Educate health professionals about the reality of managing a child or adolescent with lymphedema. Develop and evaluate low-intrusion self-management programs that incorporate self-efficacy assessment.
Footnotes
Acknowledgments
We thank and acknowledge all the help provided by the parents and children during this camp, with specific help from Delphine Lanchais for translating the documents in this research and the professional translators. The following professional and patient organizations provided technical and financial assistance: (International Lymphoedema Framework, Partenariat Francais du Lymphoedeme, and Association Vivre Mieux le Lymphoedeme.) Additional financial support was provided through unrestricted research grants from Cizeta and Thuasne. This research is dedicated to all the wonderful children, adolescents, and young adults who suffer from lymphedema and participated in this camp.
Author Disclosure Statement
No competing financial interests exist.