Resilience Processes in Children with Leukemia: Developing a New Resilience Model

Background: To clarify the details of resilience processes in children with leukemia who repeatedly undergo examination and treatment, with the aim of providing a basis for the prevention of posttraumatic stress disorder in such children. Methods: A qualitative study based on the hybrid model of concept development. To demonstrate that resilience processes have been exclusively examined in theoretical studies in the theoretical phase, and clarify the details of such processes in the fieldwork phase, semi-structured interviews regarding examination and treatment were conducted with 7 children (aged 9 to 15 years) with leukemia. In the final analytical phase, the findings obtained in the theoretical and field phases were integrated to confirm the details of resilience processes in children with leukemia based on evidence and develop a new resilience model. Results: Through interviews, 273 codes, 45 labels, 15 sub-categories, and 6 categories were extracted. In children with leukemia, resilience comprised processes by which protective factors control the senses of disgust and fear associated with stressors and vulnerability factors, and induced resilient responses, such as self-denial, proactive preparedness, coping, and emotional adjustments (in this order), leading to adaptation at the time of the study. Among these responses, self-denial and proactive preparedness were promoted by protective factors, while emotional adjustments were made through coping, consequently controlling the senses of disgust and fear and developing an accepting attitude toward unwillingness. This had helped the children achieve adaptation in the absence of post-traumatic stress disorder by the time of the study. Conclusion: This study clarified the details of resilience processes in children with leukemia, including their resilient responses promoted by protective factors, and confirmed the usefulness of a new resilience model for such children. Resilience Processes in Children with Leukemia: Developing a New Resilience Model Publication History: Received: December 19, 2014 Accepted: February 19, 2015 Published: February 21, 2015


Introduction
Children with leukemia suffer from long-term distress due to repeated examinations and treatments, such as bone marrow aspirations, lumbar punctures, and intraspinal injections. Stuber el al. [1][2][3] suggested that it may be possible to examine socio-psychological issues in children who have undergone bone marrow transplantation within the framework of post-traumatic stress disorder (PTSD), and that facing a severe disease and undergoing invasive treatment may be a traumatic experience for children. In line with this, repeated examinations and treatments are also likely to cause severe distress corresponding to type II trauma in children with leukemia, involving the senses of disgust and fear, and possibly leading to PTSD. On the other hand, in the studies on PTSD in pediatric cancer patients conducted by Bulter et al. [4] and Kazak et al. [5], the proportion of those completely meeting the PTSD criteria was limited, even when some symptoms of post-traumatic stress disorder were self-reported.
Resilience is being increasingly focused on as a basis for understanding such children. It is defined as the ability to appropriately adapt despite a threatening situation and the outcome of such adaptation, and also as an important concept for positive health. Furthermore, enabling researchers to develop effective intervention methods addressing stress responses by examining the characteristics of resilient individuals and related factors, it is also drawing increased attention as a useful concept in nursing.
As the phase of resilience (successful adaptation process, ability, and outcome) focused on varies among researchers, literature reviews, including conceptual classification, are needed when conducting resilience studies involving children with leukemia. Therefore, with the aim of developing effective support techniques to prevent trauma from leading to PTSD in such children, the hybrid model of concept development (HMCD) (Schwartz-Barcott & Kim, 2000) [6], developed to sophisticate concepts, was used to confirm the details of resilience processes in children with leukemia based on evidence, and to develop a new resilience model.

Methods
The concept analysis of resilience was performed according to the hybrid model of concept development (HMCD), which was developed by Schwartz-Barcott & Kim in 2000 to sophisticate concepts [6]. This model combines theoretical and empirical techniques, with a view to promoting the development of concepts, and consists of 3 phases integrating inductive and deductive/analytical approaches: theoretical, fieldwork, and final analytical ( Figure 1). In the present study, a literature review on resilience of children with leukemia was conducted in the theoretical phase. In the fieldwork phase, semi-structured interviews regarding examination and treatment IJNCP Only two articles were found in CHINAL [7] and ICHUSHI [8].
Because it was difficult to find articles directly related to the purpose of this study, a manual search of the reference lists was conducted to identify the articles that included the following words: resilience and leukemia or pediatric or child.

Theoretical phase
Up to the present, resilience has mostly been examined in theoretical studies, and empirical studies focusing on it have been limited to measurement of the personality traits of resilient individuals and related factors.
Masten et al. defined resilience as the ability to appropriately adapt and the outcome of such adaptation, and described it as overcoming difficulties, abilities maintained in the presence of stress, and recovery from trauma [9]. Resilience allows two different focuses: abilities and successful outcomes as a resilience-related trait; and the process of adaptation.
Resilience is promoted through dynamic interactions between abilities and environmental factors that have been regarded as resilience-related traits, considering that both are helpful for overcoming difficult situations and risks. Protective factors are traits promoting resilience and functioning as learning experiences. In diverse and difficult situations due to poverty, abuse, disease, or other causes during two developmental stages-infancy and adulthood-the ability to recognize, address, and redefine stress is a personality trait functioning as a protective factor, and it is associated with self-control, esteem, and efficacy. Family traits are related to strong family bonds, while social traits are related to supporters other than family members, supportive relationships, and support itself [10][11][12][13][14][15][16][17][18]. Protective factors play an important role in helping individuals overcome risks and direct themselves toward positive perspectives as media, and integrate four functions: reducing the impact of risks; avoiding negative chain reactions (vicious circles); enhancing and maintaining senses of selfesteem and efficacy; and creating opportunities to grow [19].  Rutter regarded resilience as a process [19,20], and Luthar et al. defined it as a dynamic process to achieve positive adaptation despite severely adverse contexts [21]. In short, resilience is a dynamic process, and adaptation is under the influence of interactions between protective and risk factors in each situation [19]; however, up to the present, the influences of protective factors on stressors and vulnerability factors, as well as adaptation, in resilience processes have been examined only in theoretical studies. In a resilience model for adolescent cancer patients developed by Woodgate in 1999, resilience is regarded as a set of processes to adapt, and Rutter's and Garmezy's theories are adopted as theoretical pillars. It consists of stressors, protective and vulnerability factors, processes, and outcomes (maladaptation or adaptation) [22,23] (Figure 2). Stressors are specific events or situations (such as losses, events requiring social adaptation, and physical injuries) inducing such patients' emotional responses [24]. Vulnerability factors promote their negative responses or vulnerability to stressors, leading to maladaptation [19]. In children with cancer, stressors and vulnerability factors increase risks and the incidence of dysfunction. In contrast, protective factors control their negative responses to stressors and vulnerability factors, and guide them toward improvement or a shift from maladaptation to adaptation [20]. As all of these components are interrelated, Woodgate's resilience model comprises a continuum between adaptation and maladaptation [25]. Although it regards resilience as a set of processes, their details are limited to the contents suggested by Rutter.
To the authors' knowledge, resilience processes were previously examined exclusively in theoretical studies, without fully clarifying their details. In order to prevent PTSD in children with leukemia, it may be essential to further develop resilience theories. It may also be necessary to examine resilient responses and coping supported by protective factors as processes to achieve resilient personality traits or outcomes (adaptation).

Objective
To clarify resilience processes in children with leukemia who repeatedly undergo examination and treatment, with the aim of providing a basis for the prevention of PTSD in such children.  [22].

Subjects
Seven children with leukemia (5 males and 2 females aged 11.7±1.80 years), who had been treated on Ward B of Hospital A between September and October 2006, were studied (Table 1). Among those referred by the doctor in charge in consideration of their confidence in his explanations, expectations for healing, and the absence of lifethreatening conditions, the following pediatric patients were included: those currently undergoing maintenance therapy in the remission phase or under observation after the termination of treatment, and who had been provided with explanations regarding the disease or pathological condition; those aged 9 or over; and those in whom type II trauma-specific symptoms (such as escapism or self-mutilation due to painful treatment) were observed. Based on Piaget's theory of cognitive development, children aged 9 or over are considered able to develop theoretical thoughts, to view the disease using their knowledge, and to verbally express their emotions and feelings [26]. In Cases B, C, E, F, and G, escapism, such as making a detour on the passageway toward the treatment room to avoid painful examination or treatment during hospitalization, or taking 30 minutes or more to enter the treatment room, was observed. In Cases D and F, selfmutilation, such as repetitive picking of the skin of the fingertips or heels to an extent where bleeding was caused, was observed when waiting for examination or treatment after a notification.
Semi-structured interviews were conducted in a closed, single room to protect the privacy of the children, who were accompanied by their parents. The duration of each session was 30 to 60 minutes.

Ethical considerations
With the permission of the Nursing Department of the study hospital, explanations outlining the study were provided to the doctor in charge and chief nurse on Ward B to obtain their approval. After obtaining the parents' informed consent, and in their presence, the children were provided with oral and written explanations regarding the study objective, methods, data use limited to research purposes, voluntary participation, unconditional refusal and withdrawal, maintenance of anonymity, personal information protection, recording during interview sessions, and appropriate data destruction. Written informed assent and consent forms were received from the children and their parents, respectively.

Analysis
In each case, recorded narratives were repeatedly read and classified for encoding, without changing their semantic contents. Similar contents were labeled, related to corresponding events, and classified into sub-categories. Subsequently, based on the similarity among the 7 cases, sub-categories and categories were created.

Final analytical phase
The final step was to integrate the findings of the previous studies in the theoretical phase, and the consequences of the interviews in the fieldwork phase. A new resilience model was constructed by including the process of resilience.

Characteristics of narratives and associations among categories (1)[Stressors], [vulnerability factors], and [stress responses]
In all cases, the {sense of disgust} and {sense of fear} intensified as [stress responses] to repeated examinations and treatments. As     In fact, in Case A, such an attitude toward the unwillingness to undergo examination and treatment was observed when the child stated, "It is inevitable. I know it..." In this case, {proactive preparedness}, such as making efforts to achieve the goal of curing the disease, was developed through {self-denial} and expressed in the statement that she knew that it was inevitable for her to overcome anxiety. She also stated, "I had to control my mind. That was all I could do. I needed to avoid thinking about bone marrow aspirations.
I just thought about my favorite things and kept watching my favorite TV programs...," indicating that she made {emotional adjustments} and developed an accepting attitude toward unwillingness by such {coping}.
In Case B, this attitude was observed in the statement, "I didn't want to enter the treatment room, but they forced me to do it. It was inevitable. " As he also stated, "I don't want to, but I have to undergo them to cure my disease and move on, " he was aware of the necessity of undergoing examination and treatment, showing {proactive preparedness}. His statement, "When they notified me of treatment, I used to play games, trying to take my mind off it. Games were helpful to change my feelings" indicated that he controlled the {sense of disgust} and {sense of fear} by {coping} and making {emotional adjustments}, and developed an accepting attitude toward unwillingness.

(4) [Adaptation observed in the absence of PTSD at the time of the study]
Considering that its symptoms are not necessarily or continuously manifested immediately after a traumatic event, and that it may influence children throughout their lives, PTSD was regarded as absent at the time of the study. As {no re-experience} was extracted, the children were considered to have appropriately adapted by that time.

Final analytical phase
The children's {sense of disgust} and {sense of fear} as stress responses associated with stressors and vulnerability factors correspond to a DSM-IV-TR criterion for PTSD -exposure to traumatic events [27]; however, as none of them showed its symptoms, such as reexperience of traumatic events, continuous avoidance of traumarelated stimuli, numbing of general responsiveness, or persistent hyper-arousal, PTSD was regarded as absent. The findings obtained in the theoretical phase and the results of comprehensive analysis in the fieldwork phase confirmed the usefulness of the developed resilience model comprising stressors, vulnerability and protective factors, and processes.
As novel findings, it was demonstrated that: protective factors convert stress responses into resilient responses; the latter responses comprise 4 processes: {self-denial}, {proactive preparedness}, {coping}, and {emotional adjustments} (in this order); and that the children had achieved adaptation by the time of the study. For example, as observed in the statement, "I want to finish my treatment and be discharged as soon as possible. So, I have to hold on to this, although it disgusts me, " <future-oriented thoughts> as a protective factor may be regarded as a consequence of a shift from the sense of disgust to self-denial. The children were unwilling to undergo examination and treatment, but were aware of the necessity or benefit of undergoing them to cure the disease; in short, {self-denial} may have contributed to the development of {proactive preparedness}. Furthermore, stated as "I tried hard, expecting such a reward, " {coping} and {emotional adjustments} supported {self-denial} and {proactive preparedness}, and resilient responses promoted resilience. Consequently, during the interview, one of the children stated, "It seems that the experience of overcoming distress in the hospital has guided me in the right direction. Now I can say that it was a good experience", indicating his stressrelated growth.
As important psychological traits commonly observed in resilience, 'future-oriented positive thoughts' , 'emotional adjustments' , and 'diverse interests' have been reported [28][29][30]. The {proactive preparedness} observed in the present study is similar to 'futureoriented positive thoughts' , and {coping} and {emotional adjustments} may correspond to 'emotional adjustments' [31]. As these responses are psychological traits achieved through the process of adaptation in a stressful situation, they naturally correspond to psychological traits common among resilient individuals. Based on this, {self-denial} may be a novel response, considering that it has not been reported in any previous study, to the authors' knowledge.
In the 'Diary of Rinaldo' , Goethe noted that self-denial is an unavoidable choice for humans [32], while Okazaki defined it as abnegating lusts or concentrating and making efforts to achieve goals, rather than passively or quietly relinquishing desires disturbing such achievement [33]. Self-denial is not an ideological product, but is wisdom achieved through coping with distress [34]. The statement, "I don't want to, but I have to undergo it to cure my disease and move on," observed in the present study, suggested that the children may have defined their senses of disgust and fear as factors disturbing their goal achievement-making efforts to appropriately undergo examination and treatment and cure the disease. Similarly, the statements "Nobody but myself can help me" and "I will do my best" may be regarded as responses leading to the self-denial of unwillingness and more active attitudes; in short, {self-denial} may be a resilient response contributing to the development of {proactive preparedness}.
Although {self-denial} was nurtured by {personality traits} in the present study, it may be essential to examine it with the other protective factors, using exploratory methods, as it may be a response playing an important role in the initial stages toward resilience.

Clinical application
Protective factors revealed the details of preparation that help pediatric patients, and were shown to enhance their ability to cope with difficult situations and acquire strength to lead their lives. For such preparation, the children regarded the explanation of examination or treatment on the previous day as important, but stated, "Detailed explanations scare me even more, " "Repeated encouragement is not needed, " or "I find ordinary topics more comfortable." These protective factors supported the process of resilience from {self-denial} to {emotional adjustments}. For children with leukemia repeatedly undergoing examination and treatment, it was crucial to avoid thinking about examination or treatment as part of the preparation for the avoidance of fear or disgust. Games were an indispensable instrument for them to control their senses of disgust and fear and maintain {self-denial} and {proactive preparedness}, by changing their feelings and avoiding thinking about treatment. Weeks & Kagan reported that such a strategy -avoiding thinking about treatmentincreases the strength of children with cancer, who face uncertainty during treatment [35].
On the other hand, repeated encouragement by other family members or medical professionals and detailed explanations were shown to be vulnerability factors intensifying the senses of disgust and fear due to examination and treatment. In some cases, the side effects of anesthesia also intensified such senses.
Based on these findings, nurses should improve their skills to differentiate protective and risk factors and assess the preparedness levels of children with leukemia. It may also be necessary to improve the quality of care, with a view to promoting resilience in pediatric patients by adopting preventive measures against risk factors and effectively using protective factors.

Study limitations and future perspectives
Up to the present, resilience has been exclusively examined in theoretical studies even in Western countries leading in this area, and the majority of empirical studies focused on its protective factors and the characteristics of resilient individuals, generating findings that are insufficient to examine and develop resilience processes. Considering this situation, in the present study, a new resilience model was developed and its usefulness was examined, based on the hybrid model of concept development.
The study has the following limitations: the number of subjects was limited to 7; the absence of PTSD was not confirmed by doctors; the nurse who cared for the children also conducted interviews; and there was no other researcher to conduct observation as a third party. As preparation markedly influences resilience in pediatric patients who repeatedly undergo examination and treatment, it may be necessary to conduct further studies, adopting appropriate approaches to obtain reliable data, such as setting stricter inclusion criteria and expanding the contents of interviews in consideration of the developmental stage.

Conclusion
To confirm the usefulness of a resilience model for children with leukemia, semi-structured interviews were conducted with 7 children