The experiences of adolescents with osteosarcoma during the one-year of treatment in Taiwan

Abstract

Osteosarcoma (OS) typically occurs during puberty. The one-year treatment for OS can be very challenging. However, little empirical research has explored the experiences of adolescents with OS. This research explored the experiences of Taiwanese adolescents with OS, utilizing a qualitative inductive content analysis with in-depth interviews and field notes that captured interviewer observations. In total, 20 participants—aged between 10 and 19 years and being treated for OS—were recruited. Ten categories were extracted: disbelief, hope for recovery and maintaining body integrity, experience with physical symptoms, inappropriate sleep hygiene, experience of psychosocial distress, use of spirituality to recover from misfortune, information acquisition, preparation to return to school, interdependence, and dedication to hope of recovery. This is the first empirical qualitative study to explore the experiences of Taiwanese adolescents with OS during treatment since being diagnosed. Close surveillance of the compliance in rehabilitation and physical function is needed. Providing age-appropriate and culture-oriented support systems would be helpful for addressing their psychosocial difficulties. Social networking sites closely moderated by health professionals may be a feasible way enhancing psychosocial well-being. Conducting a descriptive exploratory qualitative study for further development of psychosocial supportive care interventions is recommended.

Keywords

Osteosarcoma adolescent qualitative research

Introduction

Osteosarcoma (OS) typically occurs during puberty (Ward et al., 2014) in areas where the bone is growing rapidly, such as the distal femur and proximal tibia, suggesting a relationship between rapid bone growth and malignancy development (Ottaviani and Jaffe, 2009). Adolescents with OS not only risk death from OS but also risk disability due to the less-than-perfect surgical options (limb salvaging or amputation) (Anderson et al., 2013), and the standard chemotherapeutic agents for OS generate many adverse short-term and long-term health impacts (Ottaviani et al., 2013). Thus, treatment can be very challenging. There is a paucity of published empirical research into the experiences of adolescents with OS in Taiwan, perhaps contributing to their needs for services not being appropriately met during their cancer trajectory.

Literature review

Among all bone cancers in Taiwan, OS is the most commonly reported (Hung et al., 2014). The peak incidence of OS occurs in adolescents. In Taiwan, the five-year survival rate between 2004 and 2011 reached 90.4% for adolescents with OS without metastasis at diagnosis, and for those with metastasis, the five-year survival rate dropped to 25% (Hung et al., 2015), a consequence of limited access to early diagnosis and appropriate treatment (Lasebikan et al., 2014). A common early symptom of OS is sporadic pain that may worsen during the night, with progressions to local swelling (Ward et al., 2014). This symptom can be easily overlooked by adolescents or clinicians as sports injury, thus leading to a delayed diagnosis (ACS, 2015). Once an OS diagnosis has been confirmed, the standard one-year treatment of limb salvage, including limb surgery combined with neoadjuvant and adjuvant chemotherapy, is initiated (Hsieh et al., 2009).

Adolescents with cancer who are treated with chemotherapy and who have the increased cluster of fatigue and sleep disturbances subsequently display more depressive symptoms and behavioral changes (Hockenberry et al., 2010) as well as worsening daily functioning (Verberne et al., 2012). Additionally, the limb salvage procedure can directly impact mobility and physical functioning. One study demonstrated that adolescents with a primary malignant bone tumor had significantly lower scores in the domains of physical well-being and social functioning compared to healthy peers (van Riel et al., 2014). These results underline the fact that adolescents with OS may not be able to participate in the regular physical activity that they were accustomed to prior to their condition and may encounter related difficulties adjusting socially.

Adolescents of childhood cancer survivors are at risk for psychosocial difficulties compared to their siblings (Stuber et al., 2010), indicating the needs for psychosocial services. Studies have demonstrated that the psychosocial needs of adolescents with cancer are neither adequately nor appropriately served (Kahalley et al., 2013; Li et al., 2012; Zebrack et al., 2014), an outcome perhaps resulting from a lack of age-appropriate (Richter et al., 2015) and culture-oriented psychosocial services (Yeh, 2001). Unmet psychosocial needs may not only affect adolescents’ abilities to successfully achieve psychosocial transitions from childhood to adulthood but also influence their health-related quality of life (Smith et al., 2013). Some excellent works have been undertaken to provide psychosocial services for adolescents with cancer in England (Grimer et al., 2010a, 2010b; National Institute for Health and Clinical Excellence (NICE), 2005; Pearce, 2009). The NICE (2005) develops the guidance for improving outcome in children and young people with cancer, and the guidance focuses on enhancing the age-appropriate care. Pearce (2009) further outlines the ongoing developments for the provision of cancer care for teenagers and young adults, specifically highlighting the centrality of the place of teenage and young adults cancer care, effective multidisciplinary teamwork, improvement of clinical outcomes and psychosocial care of cancer care for teenager and their families.

Aim

The aim of this research was to serve as a pioneer study exploring the experiences of Taiwanese adolescents with OS during treatment since being diagnosed.

Methods

Design

This study used a qualitative research design with one-on-one, in-depth interviews of adolescents with OS and field notes that recorded the interviewer’s observations that enabled teenagers to recall memories during the one-year treatment.

The study was conducted at one pediatric oncology ward in a medical center attached to a university in Taiwan. This 2980-bed medical center has the most well-regarded pediatric orthopedic medical team in the country, receiving almost 40% of all adolescents with OS from across Taiwan (Hung et al., 2015).

Interviews were conducted from October 2012 through June 2014. Participants were asked to answer open-ended semi-structured questions about the changes to their lives since diagnosis, their responses to cancer treatment, what their needs were and the changes they had experienced in their social lives. Participants had to be: (a) 10–19 years of age, (b) completed neoadjuvant chemotherapy and limb surgery, and (c) hospitalized and undergoing adjuvant chemotherapy.

Approval to conduct this research was obtained from the institutional review board of the medical center.

Procedures

Purposive sampling was used to ensure that the sample included diverse backgrounds and perspectives. Potential participants were referred by the head nurse of the ward to a research assistant who introduced the research purpose. The adolescents who agreed to participate met with the principal investigator (PI) at the medical center in either a conference room or a quiet corner at the patient’s bedside, whichever location offered the most privacy. All participants were asked the same series of questions. All interviews were digitally recorded and transcribed verbatim and lasted one to two hours. Each transcription was checked by the appropriate participant to further ensure reflecting their experiences faithfully. The recruitment process ended when no new content emerged, at which point a total of 20 participants had been enrolled.

Data analysis

Qualitative inductive content analysis was used to derive meaning from the data of this study. This included processes of open coding, creating categories, and abstraction (Elo and Kyngas, 2008). All transcriptions were checked against the original recordings and compared to the interviewer’s field notes for consistency. All transcriptions were imported to NVivo 10.0 for analysis and independently coded by the PI and another researcher, and the research team repeatedly met to redefine the main categories in an iterative, continuous process until consensus had been reached.

Trustworthiness was established using Lincoln and Guba’s (1985) criteria (Lincoln and Guba, 1985). Credibility was ensured of by the data each transcription being confirmed by the participant (via member check) before data analysis. Dependability was ensured through feedback by peer debriefing in which the interview transcripts. To establish confirmability, the interview recordings, transcripts, and written documents were retained for a confirmability audit. To provide a sturdy database for transferability, participants were requested to provide the widest possible range of descriptions.

Findings

Nine categories were extracted. The following section describes these categories.

Disbelief

An early symptom of OS is a painless lump or bone aches, which can be easily overlooked by adolescents themselves or by primary care physicians due to lack of awareness since adolescents often participate in strenuous physical activities that easily contribute to bumps and bruises. The participants reported experiencing disbelief when they learned that their bone aches were symptoms of OS.

I was suffering from leg aches for a while since the beginning of this summer. I thought that should be fine because I ran on the school track team and practiced running daily. I thought it should be muscle injury or something like that … I could not believe it when the doctor told me it was osteosarcoma.

Hope for recovery and maintaining body integrity

Before transferring to the medical center where this study was located, many participants were told by their local health-care providers that they would require a limb amputation. Through the Internet research, friends’ suggestions, or physicians’ referral, they eventually found or transferred to this medical center’s well-regarded team of pediatric orthopedic surgeons. They regarded this team as their hope to save their lives and preserve their limbs.

I was told by the previous physician that my leg needed to be amputated in order to save my life. I kept on surfing the Internet and finally found Dr. ****, the sophisticated physician …. As expected, Dr **** did keep my leg and save my life.

Experience with physical symptoms

Participants had been experiencing many physical symptoms while receiving chemotherapy, including fatigue, sleep disturbances, limited mobility and movement, pain, and neutropenia. Of these symptoms, fatigue and sleep disturbances were the most reported during chemotherapy treatment.

During those days chemotherapy was going on, I felt tired but couldn’t sleep well because I wasn’t feeling good, like vomiting and nausea. Sometimes my sleep was interrupted by some treatment-related activities of mine or other patients in the same room.

They also experienced different levels of functional impairment due to the various locations and stages of their OS. Some were no longer allowed to participate in strenuous activities due to adverse consequences of the cancer itself (pathologic fracture) or the cancer treatment (functional impairment resulting from surgery).

Playing basketball is my favorite exercise, but I’m not allowed to play it anymore because it’s too strenuous for me.

In fact, adolescents with OS were recommended to perform adequate rehabilitation after surgery, such as range of motion (ROM) exercises; however, sometimes they lacked the perseverance or had forgotten to do exercises.

Inappropriate sleep hygiene

In addition, participants, especially the males, liked to play online games with friends during the late evening when their physical condition was relatively positive. They slept late and got up late, resulting in sleep disturbances.

I go play online games with friends during late night while returning back home waiting for the next course of chemotherapy. Because I’m in sick leave now, I don’t have to go to school. I usually don’t get up until 11 a.m.

Experience of psychosocial distress

Friendship was important to participants. However, due to their extended medical leave from school, they experienced changes in their friendships with their peers, leaving them to feel lonely and misunderstood. They tried their best to stay connected with peers via social networking sites or by telephone. They also sought acknowledgment by disclosing their terrible experiences to close friends. However, school happenings gradually became unclear to participants, while hospital happenings seemed not to be understood by their friends. These conditions created an emotional gulf between participants and their closest friends and peers.

You cannot imagine how lonely I was. Sometimes I messaged good friends how painful I felt, but they responded I had to be braver. They couldn’t understand me and I don’t know what their life in school was like. Also, My girlfriend left me. Everything I had in my life is gone!

Additionally, the altered appearance made them feel embarrassed in front of their peers. Over time, they came to want only superficial conversations with close friends in order to relieve loneliness. To maintain privacy, participants wanted to avoid deeper peer conversations, especially those regarding their health condition and new appearance. They described how ‘life sucks’ because they lost everything important to their lives.

Use of spirituality to recover from misfortune

Participants were physically carried by their parents to perform religious practices, such as temple worshipping or the lighting of blessing lamps, indicating their desire for protection from the gods. It was believed that these actions could diminish the negative effects of bad karma that had accumulated from the teenagers’ current or previous lives or to remove the evil spirits believed to be attached to the patients. Participants would love to cooperate with parents’ demands because they would rather believe it to be truth than not.

My mother took me to the temple. We believe religion helps. We worshiped in temple, lighting up blessing lamp, and making donations to charity. Then we met the good medical team.

Information acquisition

Participants reported that they seldom read the health education booklets. They preferred to acquire OS-related information from the Internet due to its convenience and efficiency, although they admitted that they could not distinguish between accurate or wrong information they had found online. They also reported that they would rather see tables, figures, or pictures than read many words.

In order to learn more about OS, I had browsed all the related web pages that I could. To tell the truth, I didn’t read the booklets that the nurses gave me. Too many words!

Preparation to return to school

In Taiwanese culture, the most important social value for students is to achieve good academic performance. Participants believed that treatment eventually would come to an end and then return to school. During hospitalization, they did their best to catch up with school’s schedules while their health condition was relatively stable.

I have largely lagged behind my classmates. I have to catch up on the schedule as much as I can.

Interdependence

Parents regarded protecting adolescent children as their responsibility, helping participants to feel a sense of security and satisfaction with their parents’ unconditional love.

Experiencing those terrible adverse effects of chemotherapy is too hard for me to stand. Parents always encouraged me, supporting me unconditionally. Suddenly I realize I need my Mom so much!

Dedication to hope of recovery

Due to the complexity of the OS trajectory, participants felt uncertainty about their future. They loved hearing about successful outcomes to learn positive attitudes of life and healthy behaviors from other adolescents with OS.

I would love to learn from other adolescents’ experiences about how they overcame the hardship. These experiences remind me of being positive and teaching me how to fight cancer.

Discussion

The adolescents with OS were found to have overlooked the severity of the early symptoms of OS, and the onset of treatment may therefore be delayed because of low suspicion of cancer on the parts of both patients and health-care providers. This finding is in line with a finding which indicates that bone tumors in young people have long symptom interval, the time from first symptom/sign to diagnosis and initiation of treatment, which are significantly affected by age and local health-care support systems (Goyal et al., 2004). This reflects that health professionals and society often perceive ‘young people do not get cancer’ (Goyal et al., 2004). Efforts need to be geared toward public education for healthy adolescents and society and the continuing education for health professionals to ensure early diagnosis of OS, thereby reducing morbidity and mortality from the primary bone tumor (Lasebikan et al., 2014).

In this study, these adolescents were treated with limb salvage procedures and experienced functional impairment for the treated limb. This was consistent with prior studies that indicated adolescents with a primary malignant bone tumor experienced worse physical well-being (van Riel et al., 2014) and muscular integrity (Carty et al., 2009). The physical immobility negatively impacted on these adolescents, perhaps contributing toward increased dependence to parents. This may also explain why adolescents with bone tumors have poorer autonomy compared to their healthy peers (van Riel et al., 2014).

Adequate ROM exercise prescribed by physicians is an important component for adolescents with sarcoma after limb-sparing surgery (Marchese et al., 2006). However, this study’s findings indicated that participants sometimes did not perform ROM exercises as many times as prescribed. The cause of this might be related to insufficient knowledge designed specifically for this population regarding the severity of surgical complications. Participants reported that they seldom read the provided health educational materials because the content was too wordy and boring. Instead, they relied on the Internet as a source of information about cancer, but some online information could have been outdated and inaccurate.

In this study, fatigue and sleep disturbance were the most frequently mentioned symptoms. The same finding was identified previously (Wu et al., 2015). Evidence indicates that adolescents treated for cancer report decreased total sleep time, increased waking after onset of sleep, and decreased sleep efficiency (Orsey et al., 2013). However, sleep disturbance during the night is not the only cause of daytime sleepiness. A circadian rhythm sleep disorder—delayed sleep phase disorder—is a possible cause of daytime sleepiness, and it is commonly seen in healthy adolescents as well (Bartlett et al., 2013). Disease and treatment are also possible factors in sleep disruption. Evidences demonstrate that an adequately supervised physical activity program can reduce fatigue and improve physical function (Huang and Ness, 2011) as well as diminish sleep disturbance (Orsey et al., 2013).

This study’s participants experienced psychosocial difficulties, a result which was in line with a prior study whose adolescents with malignant bone tumors experienced psychosocial maladjustment (van Riel et al., 2014). The conclusion of a systematic review article strengthens the need of age-appropriate interventions for adolescents and young adults with cancer (Richter et al., 2015). People in Asia may not actively express negative thoughts due to emotional restraint (Davidhizar et al., 1999). Actively detecting psychosocial difficulties and implementing culture-oriented and age-appropriate interventions is needed. However, the psychosocial intervention in Taiwan is seldom conducted in clinical practices because its residents stigmatized and discriminated against associates who psychosocial service for mental illness (Lauber and Rossler, 2007). Other alternative approaches, such as supernatural and religious approaches, are common instead. Traditional religions (Buddhism and Daoism) strengthen inner strength by interpreting the cause of misfortune from the perspective of causality, thereby improving teenagers’ coping mechanisms and serving as an alternative source of hope than that from Western medicine (Yeh, 2001). In addition to religion, peer support groups can enhance psychosocial difficulties. A prior study indicates that the online cancer support community can promote mental/emotional health for adolescents with cancer (Love et al., 2013). Social networking sites having high popularity among adolescents may provide as platforms of online support that need to be carefully moderated by health professionals for adolescents with OS.

This research was conducted in a single Taiwan medical center, limiting transferability. Future studies should recruit adolescents with OS from a variety of hospitals to aid understanding of their experiences. It would be useful to compare the experiences of other adolescent malignant diseases.

Implications for nursing practice and nursing research

Close surveillance of the compliance in rehabilitation and physical function is needed. Providing age-appropriate and culture-oriented support systems would be helpful for addressing their psychosocial difficulties. Social networking sites closely moderated by health professionals, possessing with functions of distributing information and online support, may be a feasible way enhancing health-related knowledge and psychosocial well-being.

Conclusion

The 10 experiential categories that emerged from the data contribute to an initial understanding of how adolescents with OS experience their challenges and changes, which could be used to support research and intervention development in caring for this population.

Footnotes

Acknowledgments

The authors thank all participants and staff of the 93 ward in the Taipei Veteran General Hospital for assistance with this study.

Declaration of conflicting interests

The author(s) declared no potential conflicts of interest with respect to the research, authorship, and/or publication of this article.

Funding

The author(s) disclosed receipt of the following financial support for the research, authorship, and/or publication of this article: This study was supported by the Ministry of Science and Technology, Taiwan (grant no: NSC 101-2511-S-227-003-MY2).

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