“Nothing is carved in stone!”: uncertainty in children with cancer and their families

‘‘Nothing is carved in stone!’’: uncertainty in children with cancer and their families Roberta Lynn Woodgate, Lesley Faith Degner

Although more children are surviving childhood cancer, they and their families still face many new stressors and challenges.Understanding the experiences of childhood cancer in children and families is conditional upon building a sound and comprehensive knowledge base that is grounded in research. Accordingly, a longitudinal interpretive qualitative study was conducted to arrive at an understanding of children’s and families’ perspectives on having to experience the childhood cancer-symptom trajectory. A purposive sample of 39 children with cancer and their families were recruited into the study.Open-ended interviews and participant observation were the primary data collection methods. Data were analyzed by the constant comparative method of grounded theory and analysis of illness narratives. Findings revealed that in addition to the stress and suffering resulting from the children’s multiple symptom experiences, children and their families experienced many other events or ‘rough spots’ that made cancer and life diff|cult to get through. Although the ‘rough spots’ were numerous and varied, they all resulted in children and their families experiencing feelings of uncertainty.This paper focuses on describing the nature of uncertainty experienced by the children with cancer and their families. Recommendations for nursing practice and research are discussed. r 2002 Elsevier Science Ltd. All rights reserved. Keywords: pediatric cancer, uncertainty, families, children, cancer-symptoms

Roberta Lynn Woodgate, RN, MN, PhD, Assistant Professor, Child Health and Illness, Faculty of Nursing, Helen Glass Centre for Nursing, University of Manitoba, Winnipeg, Manitoba, Canada R3T 2N2.Tel.: +1 204 474 8338; Fax: +1 204 474 7682; E-mail: roberta_woodgate @umanitoba.ca Lesley Faith Degner, RN, MA, PhD, Professor (Cancer Nursing), Faculty of Nursing, Helen Glass Centre for Nursing, University of Manitoba, Winnipeg, Manitoba, Canada R3T 2N2. Correspondence and o¡print request for reprints to: Roberta Lynn Woodgate The data in this article are part of the f|rst author’s dissertation study which was completed in 2001. Dr Degner was Dr.Woodgate’s supervisor on the study.

Es gibt kein ehernes Gesetz! Unslcherheit bei krebskranken Klndern und lhren Fammlllen Obwohl heute mehr Kinder eine Krebserkrankung in Kindesalter ˇberieben, werden sle und ihre Familienangeh˛rigen immer noch vielen mit vielen neuen Stresserregern und Herausforderungen konfrontiert. Die Erfahrungen von Kindern und ihren Familien bei Krebserkrankungen im Kindesalter zu verstehen, ist nur dann m˛glich, wenn man eine solide und umfassende Wissensbasis aufbaut, die sich auf die Forschung stˇtzt. Folglich wurde eine interpretative qualitative L,ngsschnitt-Studie unternommen, um einVerst,ndnis des Erlebnisses der Krebs-Symptom-Kurve im Kindesalter aus der Perspektive von Kindern und Familien zu erarbeiten. Eine bewusste Auswahl von 39 krebskranken Kindern und ihren Familienangeh˛rigen wurde fˇr die Studie rekrutiert. Offene Interviews und teilnehmende Beobachtung waren die prim,ren Datenerhebungsmethoden. Die Daten wurden durch die permanente Vergleichs-Methode der grounded theory (empirisch begrˇndeteTheoriebildung) und durch Analysen der Erz,hlungen ˇber die Krankheit analysiert. Die Ergebnisse zeigten, dass die Kinder und ihre Familien zus,tzlich zu der vielf,ltigen symptombedingten Stress- und Leidenserfahrung noch viele andere Ereignisse oder ‘schlimme Zeiten’ erlebten, die die Krebskrankheit und das Leben ˇberhaupt erschwerten. Die ‘schlimmen Zeiten’ waren zwar zahlreich und unterschiedlich, fˇhrten aber alle dazu, dass Kinder und ihre Familien ein Gefˇhl der Unsicherheit erlebten. Diese Untersuchung konzentriert sich auf die Beschreibung der Art dieser von krebskranken Kindern und deren Familien empfundenen Unsicherheit. Empfehlungen fˇr die Pflegepraxis und fˇr die Forschung werden besprochen.

European Journal of Oncology Nursing 6 (4),191^202 # 2002 Elsevier Science Ltd. All rights reserved. doi:10.1054/ejon.2002.0220, available online at http://www.idealibrary.com on

192 European Journal of Oncology Nursing

INTRODUCTION Cancer is the leading cause of disease-related death among children. However, fewer children today are dying from cancer (Faulkner et al.1995, Langeveld et al. 2000). The decrease in mortality rates of children with cancer can be attributed to advances in anti-cancer treatment. However, the aggressiveness of treatment is associated with many adverse effects and consequences not only for the children, but for the entire family unit (Eiser et al. 1995). Increased confusion, decreased energy, caregiver burden, despair, compromised survivorship, and complicated bereavement are just some of the adverse consequences that may ensue (Hinds 1990, Hogan 1997). In reality, despite all the advances, children with cancer and their families still must face many challenges as they embark on their long and arduous journey in trying to ‘beat the cancer.’ The many challenges can make daily living very stressful for families. More than ever, physical and mental suffering become a part of their lives. Compounding the suffering are the multiple symptoms that children experience and that families have difficulty in managing (Ferrell et al. 1994, Rhiner et al. 1994). Accordingly, a longitudinal qualitative study was undertaken to focus on discovering meaningful descriptions and interpretations of children’s and their families’ experiences with the cancer-symptom trajectory (Woodgate 2001). The study reinforced that the symptoms in and of themselves were critically important to the shaping of the overall cancer experience. In the words of one adolescent female with cancer ‘symptoms are the big part of cancer!’ However, there were other difficult events or as identified by families ‘rough spots’ that made cancer and life difficult to get through. Although the ‘rough spots’ varied, all were associated with specific characteristics including that of uncertainty. The purpose of this paper is to increase the reader’s understanding of the uncertainty experienced by the children with cancer and their families. Prior to presenting the findings on the nature of uncertainty in children with cancer and their families, a review of the literature focusing on the construct of uncertainty and uncertainty in children with life-threatening illness is presented to help to provide a context for the findings.

LITERATURE REVIEW The construct of uncertainty Understanding the construct of uncertainty in chronic illness has been greatly advanced by the theoretical and empirical work of Mishel (1999, 1996, 1988). Since the early 1980’s, Mishel has European Journal of Oncology Nursing 6 (4),191^202

focused on developing and expanding on a middle-range nursing theory of uncertainty in illness that is grounded in literature as well as research. Foundations of this theory are based on Selder’s life transition theory that describes how people restructured their lives and resolved uncertainty after a major experiential transition (Mishel 1999). The life transition theory was expanded on to make it applicable and specific to illness. Mishel (1999) also did not focus on the management of uncertainty in order to eliminate it, but instead focused on the integration of uncertainty into one’s life and one’s life view. Uncertainty as it applies to chronic illness is defined by Mishel (1996) as the inability to determine the meaning of illness-related events and occurs when the individual cannot adequately structure or categorize an event because of lack of sufficient cues. ‘Not knowing’ in relation to many aspects of chronic illness is a critical dimension of uncertainty and includes not knowing when untoward events or tragedy will occur, not knowing what form uncertainty will take, and not knowing what to do with the uncertainty. While uncertainty in chronic illness may be a factor in maintaining hope, it nonetheless undermines the quality of life and is associated with increased stress and decreased levels of life satisfaction (Mishel 1996). There are many types of uncertainty in chronic illness that individuals must manage including symptom uncertainty, medical uncertainty, and daily living uncertainty (Mishel 1996). In managing uncertainty, Mishel (1996) identifies five major approaches: (1) constructing normative frameworks; (2) focusing on the positive; (3) specifying controllable circumstances; (4) carrying out ritualistic behavior; and (5) incorporating uncertainty. A limitation of Mishel’s theory is that its main foundations are based on theoretical and empirical perspectives related to adults experiencing life-threatening illness. However, researchers such as Cohen (1993, 1995) have adapted and extended Mishel’s work to the study of uncertainty specific to children experiencing lifethreatening chronic illness. Cohen (1993) conceptualizes uncertainty as: (1) an universal experience in which varying degrees of certainty can be found in most languages; and (2) a multidimensional phenomenon that can vary in degree of magnitude, intensity, and saliency. Cohen (1993) contends that while the conditions that give rise to uncertainty have changed overtime, the strategies that individuals in primitive societies used to manage uncertainty have remained surprisingly constant and are still relevant today. Critical to dealing with the presence of universal and multiple uncertainties is the assumptive world in which individuals live (Cohen 1993). The assumptive world as defined

Uncertainty in children with cancer and their families

by Cohen consists of a relatively stable cognitive world of knowledge, values, beliefs, and expectations and an action world of predictable events, routines, behaviors and social relationships. Living in an assumptive world provides a sense of continuity and coherence that helps one to mediate the awareness of the presence of universal and multiple uncertainties (Cohen 1993). In childhood cancer, it is assumed that cancer challenges the child’s and family’s assumptive world thereby causing uncertainty and a near total transformation of that world.

Uncertainty in children with chronic, life-threatening childhood illness Research has consistently demonstrated that regardless of the child’s specific diagnosis, families of children with chronic and/or lifethreatening illness including cancer share similar problems, feelings, and experiences including uncertainty (Clarke-Steffen 1993, Cohen 1993, 1995, Comaroff and Maguire 1981, 1986, O’Brien 2001, Sterken 1996). The continual uncertainty experienced by families is related both to the child’s health and survival status and the survival of the family unit (Cohen 1995, O’Brien 2001). For families of children with cancer, the uncertain status and outcome of the illness threat is especially evident and the apparent clinical gains seem to have only heightened remaining uncertainties (Comaroff and Maguire 1981, 1986). In a study that sought to understand what it meant to families to have a child who is technology dependent, experiencing frequent change was the overwhelming response reported by families (O’Brien 2001). The change experienced by families was not only frequent but also unexpected, unpredictable, and limited parental control. No matter how carefully parents planned, something unexpected could, and generally did happen. Families described this experience with an uncertain future as ‘living in a house of cards’ and is comparable to research that describes living with uncertainty as like ‘living with a time bomb’, never knowing when it is going to go off (Cohen 1995). In addition to experiencing unexpected or unpredictable change, research has also demonstrated that common to most families of children with chronic and/or life-threatening illness is that they do attempt to deal with the change and uncertainty (Clarke-Steffen 1993, Cohen 1993, 1995, O’Brien 2001). Strategies used by families to increase stability include the use of vigilance or careful surveillance, advocacy, reframing or evaluating and shifting priorities, living 1 day at a time, and becoming aware or knowledgeable about cancer and its effects on the family (Brett and Davies 1988, Clarke-Steffen 1997, 1993,

193

Cohen 1993, 1995, Comaroff and Maguire 1981, 1986, O’Brien 2001). Challenges reported by families are also similar and include having to make sense out of life, managing daily life and all the additional tasks associated with the child’s condition, dealing with new developmental demands, and perhaps most important, maintaining the family unit (Clarke-Steffen 1997, 1993, Cohen 1993, 1995, O’Brien 2001). The uncertainty and change that families can experience makes it more difficult for them to deal with the challenges. In summary, the research demonstrates that life at times can be overwhelming for families who have a child with a chronic and/or lifethreatening illness regardless of the child’s health problem. Contributing to the stress experienced by families are the constant feelings of uncertainty. Nonetheless, the research also demonstrates that families manage fairly well and are able to adapt to all the change and challenges (Clarke-Steffen 1997, 1993, Cohen 1993, 1995, O’Brien 2001). To suggest that these families are resilient would be an understatement considering the uncertain future that they must face. Developing a comprehensive understanding of this phenomenon in children with cancer and their families is justly warranted considering the magnitude that uncertainty in life-threatening illness may have on individuals.

METHODS Study design An interpretive qualitative research design was used to arrive at an understanding of children’s and families’ perspectives. Guided by the philosophy of interpretive interactionism, the qualitative research approach afforded the opportunity to study the human lived experience of children with cancer and their families as it was experienced and grounded in their meanings, interpretations, activities, and interactions (Beck 1990, Bernheimer 1986, Deatrick and Faux 1989, Strauss and Corbin, 1990, van Manen 1990).

Participants and setting This study took place in a major city located in a province of Western Canada. In addition to participants’ homes, the study was conducted in the outpatient unit of the province’s major outpatient clinic and the pediatric oncology inpatient unit of the province’s only pediatric hospital. Both the units provide care for pediatric oncology patients under a universal health system that does not charge user fees. European Journal of Oncology Nursing 6 (4),191^202

194 European Journal of Oncology Nursing

Child and family participants were selected using the grounded theory method of theoretical purposive sampling. This method helps to optimize the probability of describing the full range of the phenomenon (Patton 1990, Field and Morse 1985). A designated nurse intermediary assisted in participant recruitment. A total of 39 children and their families were recruited. In order to capture core experiences that cut across participant and phenomenal variation, children of varying ages with different cancer diagnoses and who were at different stages of their illness were sampled (Patton 1990). Of the 39 families, a subset (n=15) of families were followed throughout the course of the study. These 15 families’ experiences were studied more intensely. Health-care providers including professionals and para-professionals such as physicians, nurses, social workers, and nursing aides were also part of the study’s sample. Their involvement in the study was mainly restricted to observing them when they came in contact and interacted with the children and families during the designated observation periods. They were not formally interviewed. Including these individuals in the study was important because how they responded to the children and families had an effect on children’s and families’ responses. However, the health-care providers’ interactions with families and children are not a focus of this paper.

Procedure and data collection methods Permission to carry out the study was received from the Ethical Review Committee at a University located in Western Canada. Once ethical approval was received for the study, recruitment of participants commenced. Explanation of the study was provided to parents and children. Data collection commenced once parental consent and child assent were obtained. The first author, who was responsible for all data collection, arranged convenient times to meet with each family. Data collection took place between the period of July 1998 and December 2000. Although multiple data collection methods were used to facilitate arriving at a comprehensive understanding of the phenomenon under study, there were two major sources of data collection. The first method involved the use of participant observation. Participant observation affords researchers the opportunity to sustain close contact with participants thereby gaining insight into their viewpoints and practices (Prus 1996). The first author assumed the role of moderate participation in order to maintain a balance between participation and observation, while trying to remain fairly unobtrusive or nondisruptive. The 39 children and their families European Journal of Oncology Nursing 6 (4),191^202

were observed during various periods, at various sites, and at different points in time. A total of 960 observation hours took place in the outpatient unit. In addition, children were observed anywhere from 2 to 5 hours daily or every second day if they were admitted to the inpatient unit. As part of the participant observation experience, informal interviewing took place and involved asking questions that flowed during the immediate context of the observation period. The questions helped to verify what meanings the participants themselves assigned to their situations (Patton 1990). Observations were unstructured in order to permit flexible exploration and were recorded in field notes. A total of 1500 double-spaced typed pages with 1ands16;in margins to all sides, tops, and bottoms of the pages were recorded. The second major source of data collection involved formally interviewing children and their families. An open-ended technique was used in the interview process in order to elicit detailed responses and to focus participants’ responses into areas previously not anticipated (Field and Morse 1985, Morse and Field 1995). Three interview guides with a list of potential probes in the form of questions were developed for the parents, children with cancer, and siblings. Questions were based on key themes identified from the literature and from the investigators’ clinical experience. Even though interview guides were developed, the open-ended interviewing technique assumes that meanings, understandings, and interpretations cannot and should not be standardized (Denzin 1989). The phrasing of the questions and the order in which they were asked varied for each individual. Care was taken to ensure that the interview guides for the children with cancer and their siblings were developmentally appropriate. More than one set of interview sessions were planned in order to validate and follow-up on significant themes. Family members were interviewed individually as well as with other family members, and most interviews took place in the families’ homes. During the course of the study, a total of 230 interviews were conducted with mothers being interviewed 117 times, fathers 46 times, siblings 48 times and children 103 times. The adults’ interviews lasted anywhere from 40 to 180 minutes. The children’s interviews lasted from 20 to 120 minutes with the younger children usually participating in shorter interviews. All interviews were tape-recorded in order to preserve their authenticity and field notes were made during all interviews. Triangulation of data collection methods proved to be of much value to arriving at a more comprehensive understanding of the children’s and families’ experiences. The various methods complemented each other and allowed

Uncertainty in children with cancer and their families

195

the first author to approach emerging themes through multiple perspectives.

and (5) we are certain about some things. Table 1 highlights some of the study’s key findings.

Data analysis

Demographic and cancer-related characteristics

All data collected were transcribed and entered into a computer, and analyzed by the constant comparative method of grounded theory. The goal of this method is to arrive at explanatory, inductively derived theories of basic psychosocial phenomena that exist within or experienced by individuals or groups (Field and Morse 1985, Strauss and Corbin 1990). Analysis occurred concurrently with data collection in order to facilitate the discovery of patterns of the emerging theory. Critical to making comparisons of emerging categories was the ongoing process of coding or giving meaning to all units of information that address the phenomenon under study (Creswell 1998, Morse and Field 1995, Strauss and Corbin 1990). Asking questions and making memos related to code notes and data comparisons were part of the process of coding. Codes were revised and previously coded data were re-coded. When no new information or insights emerged, and when the category development was dense and the relationships between categories were well established and validated, data collection and analysis were terminated (Creswell 1998). The construction of illness narratives helped to confirm the paradigmatic relationships of the theoretical categories as they provided a basis for understanding how illness affects the children’s and families’ biographies by contextualizing illness events and illness symptoms (Hyde´n 1997).

RESULTS This section first describes the primary demographic and cancer-related characteristics of the child and family participants. A description of the nature of uncertainty in childhood cancer then follows. Five themes related to uncertainty emerged: (1) uncertainty and everything else that comes with the ‘rough spots’; (2) uncertainty, more than living with the unknown; (3) knowing what to expect; (4) uncertainty is sometimes OK!;

Of the 39 child participants, 21 were female (54%) and 18 were male (46%). Ninety-five percent of the children were white. Children ranged in age from 5 to 18 years with 10 years as the mean age. Most of the 39 children had two parents (87%) and siblings (87%). Twenty-six families (70%) resided in the city, 13 families (30%) in the rural areas. The majority of the children were diagnosed with either leukemia or lymphoma (72%). All the children received chemotherapy either alone (56% ) or in combination with surgery (18%), radiation (5%), radiation and bone marrow transplant (BMT) (8%), radiation and surgery (10%), and surgery, radiation, and BMT (3%). During the course of the study, five children (13%) had a history of relapse. At completion of the study, most of the 39 children remained in remission (90%) and four children had died (10%).

Uncertainty and everything else that comes with the ‘rough spots’ To describe childhood cancer as an ‘up and down’ or ‘roller coaster’ experience, as many of the families did, would not be an exaggeration. Contributing to the ‘roller coaster’ nature of cancer were the ‘rough spots’ or experiences that made getting through the cancer difficult for most families. The ‘rough spots’ that children and families experienced included such events as being informed of the diagnosis and getting past the diagnosis, repeated invasive procedures, missed work and school, death, changes in social relationships, hospitalization, and so on. There were just so many ‘rough spots’ that at times cancer was a very overwhelming phenomenon to experience as one 16-year-old female with cancer reinforced, ‘I can’t go to parties with my friends. And then I’m sixteen and I can’t drive. Well I can drive, I can get my licence I just (pause) I can’t, I don’t really want to bother right now? I have too much on my plate, you know!’

Table 1 Key findings of uncertainty in children with cancer and their families Uncertainty is present throughout the cancer trajectory and even after the child has successfully completed the cancertreatment course KIn addition to ‘not knowing’, uncertainty involves dimensions of indecisiveness, self-doubt, and a lack of security KExperiencing the ‘what ifs’ is common to the experience of uncertainty KCertainty is not always a desired state KBoth uncertainty and certainty can cause a lot of distress KComing to know what to expect and taking1day at a time are two strategies that help to reduce a sense of uncertainty in children and families and help them to cope KMany di¡icult events or experiences especially the children’s cancer-symptoms contribute to the feelings of uncertainty K

European Journal of Oncology Nursing 6 (4),191^202

196 European Journal of Oncology Nursing

Although the children’s cancer was associated with many ‘rough spots’ and varied among families, all the ‘rough spots’ that families experienced, shared the common characteristic of uncertainty. From the moment parents, children, and siblings suspected that the child’s illness was more serious than originally thought (i.e., prior to the cancer diagnosis), feelings of uncertainty pervaded their worlds. The characteristic of uncertainty was associated with feelings of living with the ‘unknown’ and involved children and their families ‘not knowing’ what to expect. This included ‘not knowing’ what was going to happen to them in treatment, whether the child would be able to go to school, and whether they as a family unit would be able to deal with the cancer and survive as a family unit. There was always some type of ‘not knowing’ for families to deal with as is evident by the following comment made by a mother whose son had completed all his chemotherapy and radiation treatment and was about to undergo a BMT, ‘I feel like it is the beginning of the end and that another unknown is now out there...there will be another unknown to deal with.’ There was no real distinct beginning or end to the children’s and families’ not knowing, but instead it was like the beginning and the end melded into one. Regardless of the time frame, it was like one mother said, ‘you just never know what to prepare for and that’s the difficulty’. While the families and children used a variety of strategies to help them cope under such situations, learning to take 1 day at a time proved to be especially valuable. It was important for them to focus on the present. Children and families experienced much angst and uneasiness because of their ‘not knowing.’ Not having any sense of the future was difficult for families. One mother emphasized this by stating ‘If you could have a vision of this far, you could get through the end. We had no vision and no, there is no way you can give anybody that because they don’t know, there is no guarantees with anything. But how far, you can’t say this is the way it is going to because it is the unknowing, the unknown that is the complete, that destroys you.’ The uncertainty of ‘not knowing’ made getting through the cancer experience so much more difficult for children and their families. However, through the course of the study it soon became evident in the children’s and families’ narratives that there was a whole lot more that contributed to the difficult nature of cancer and its ‘rough spots.’ Moreover, families did not always feel that uncertainty was the worst part of experiencing cancer and all its ‘rough spots.’ In addition to the uncertainty, the ‘rough spots’ experienced by children and their families were also associated with: (1) a multitude of intense feelings, including prevailing feelings of sadness; (2) restrictions on life; (3) extra physical, mental, European Journal of Oncology Nursing 6 (4),191^202

and emotional work; and (4) a sense of loss. The degree of intensity of each characteristic including uncertainty varied depending on the context of the situation and therefore, affected how each ‘rough spot’ were perceived and experienced by the children and families. All the characteristics were consequences of, as well as contributors to the ‘rough spots.’ Likewise, the degree of uncertainty felt by children and their families was influenced by as well as influenced their experiences with intense feelings, restrictions, extra work, and loss.

Uncertainty, more than living with the unknown Through the course of the study it also became evident that there was more to the children’s and families’ experiences with uncertainty than living in the unknown. The uncertainty experienced could range from falling short of uncertainty to an almost complete lack of confidence or knowledge about something, especially in relation to outcomes. Indeed, even when children and families knew what possibly was going to occur and knew what to expect, they still could experience a lack of a certainty. In addition to feelings of ‘not knowing,’ indecisiveness and selfdoubt were experienced by children and families. For example, even when one mother was provided with the information needed to make the decision related to scheduling certain treatment for her daughter, she made the following comment, ‘I do not know, I, I have thought about it and thought about it and there is just no good answer.’ Phrases or words such as ‘I guess,’ ‘sort of,’ ‘probably,’ and ‘kind of’ were frequently used by children and parents in response to many of the interview questions reinforcing their selfdoubt. The indecisiveness that families at times experienced could be related to their feelings of mistrust as well as apprehension of the information provided by health-care providers and was demonstrated by them by varying degrees of skepticism and suspicion of what ‘really’ may happen. Even when children and families knew what to expect, there was always that feeling of ‘what if...’ as all expressed. So in addition to ‘not knowing’ and the indecisiveness, the ‘what ifs’ contributed to the uncertainty. Experiencing the ‘what ifs’ resulted in families feeling at times like they were living in a state of limbo, being unsure of the future as well as not knowing how to respond even though they knew what to expect. For example, one parent who received a lot of strength from the belief that God would ‘set things straight,’ nonetheless still expressed some uncertainty in her faith that her son would survive his cancer, ‘God has helped us before, he’ll probably help us again.’ Simply put, uncertainty

Uncertainty in children with cancer and their families

resulted in families never feeling as secure as they once felt prior to the cancer diagnosis. This was even the case for parents of children who had completed treatment and remained in remission. Most parents expressed that they ‘were always waiting for the other shoe to drop.’ The parents’ sense of security was especially shaken with respect to how they responded to their child’s symptom experiences. At times their feelings of the uncertainty related to the overall cancer experience paralleled their feelings related to the symptom trajectory. Prior to embarking on the cancer journey, parents felt fairly secure in their ability to figure out what particular symptoms meant in their child. But with the very start of the cancer-symptom trajectory, that is, in the pre-diagnosis stage of cancer, parents’ faith in their own judgement was shaken. They knew something was not right, but could not really figure it out. Moreover, when they found out that the symptoms resulted from cancer, their confidence was completely shaken. They could no longer be totally confident in their judgement. Parents’ sense of security was especially challenged when they perceived certain symptoms to be less of a concern but in fact were much more serious than originally judged. In such instances parents doubted themselves and their ability to parent as one mother noted, ‘You start doubting your own parenting skills!’ The children’s confidence in their parents’ ability to help them feel better had also been fragmented to some degree because they now experienced symptoms that could not be easily fixed by their parents. Years after completing treatment, children and families always expressed some degree of selfdoubt with respect to symptoms resulting from minor illnesses (e.g., flu). Although most felt relatively confident that symptoms such as a headache or stomachache were minor inconveniences, they nonetheless expressed some selfdoubt. Another dimension of the uncertainty experienced by families was the ‘waiting.’ Families found that in general cancer meant ‘having to wait a lot’ such as having to wait for the next cycle of treatment to commence or to end. A good deal of the waiting involved ‘not knowing’ what in fact would take place. The waiting required of families compounded the degree of uncertainty experienced by families.

Knowing what to expect Although each family approached the uncertainty in their own unique way, it was important for all families to arrive at some sense of knowing what to expect. Coming to know what to expect helped families and children to cope. Knowing what to expect involved children and families seeking out information from many

197

sources such as health-care providers, other families experiencing childhood cancer, and from the literature on childhood cancer. Knowing what to expect in relation to the children’s cancer was important as such knowledge helped families to contain their feelings of uncertainty. To some extent it helped them to prepare ‘for things to come!’ But even when families knew what to expect, many parents voiced ‘you can never be completely prepared!’ As always, there were the little and sometimes big surprises waiting for them; some degree of self-doubt was always present. While it was important to be informed of all aspects of the child’s care and prognosis, both children and parents felt that timing of accessing or receiving information was critical. Depending on the situation, they wanted to wait for the information. One mother expressed ‘In a way I don’t want all the answers right away. Sometimes I don’t want to know everything right away!’ Considering the symptom experiences were so much interrelated to the degree of uncertainty felt by families, children and families especially needed to find out everything they could about the symptom experiences as it helped children and families to be prepared in responding to the cancer symptoms. However, even after having gained knowledge from other families, physicians, and nurses, and becoming increasingly became attuned to what to expect with a particular medication and resulting symptoms, the feelings of uncertainty would surface again. This was especially the case when the child would be put on a new medication as it involved more learning about what to expect in relation to a whole new set of symptoms. As one father noted ‘The first three months of chemotherapy was the worse...but her body adjusted to chemotherapy, and things got better....but then she would get another new set of chemotherapy that resulted in us adjusting to a whole new set of symptoms!’ Parents could never be totally sure or feel secure with respect to the children’s symptoms because each parent saw their child as an unique individual who could respond differently to specific treatment in comparison to other children with cancer. In the voices of many parents ‘nothing is carved in stone!’ Things could turn out differently for their child despite all the information that they processed. Parents as well as children reinforced that it was ‘different for all children and their families.’ It is perhaps for this reason that many families were reluctant to give advice to other families about their experiences. Often they were at loss for words when asked what other families experiencing cancer should know in order to get through the cancer in general and how to manage the cancer symptoms. European Journal of Oncology Nursing 6 (4),191^202

198

European Journal of Oncology Nursing

Uncertainty is sometimes OK! Although uncertainty brought on feelings of uneasiness and made life harder to live, it was nonetheless sometimes accepted or desired. Parents emphasized that having some degree of uncertainty was inevitable and that it was important to accept the inevitability of uncertainty. Families reinforced it was important to ‘expect the unexpected.’ In fact, uncertainty was viewed sometimes as a positive and comfortable state to be in. For example, the children sometimes felt that knowing what to expect with treatment was difficult and caused a lot of worrying and stress. Accordingly, remaining uncertain about specific aspects about their treatment at times was a good feeling. One adolescent male with cancer expressed ‘Um yeah, sometimes it was good (not knowing) cause you wouldn’t go from Friday to Friday thinking ‘oh my God this gonna happen to me next time and you spend a whole week worrying.’ It’s kinda like...Do you almost feel worse, like, than when you had your treatments with your cancer?’ For another adolescent having to go for another surgery was especially stressful for her because she knew what to expect the second time around. Her father felt it was more difficult because ‘Lynn now has experiences and memories, so it (second surgery) was more tearful for her because she knew what to expect. And while the surgery was half in terms of duration, aahh it was in some sense more difficult. Certainly more difficult for Lynn. She knew what to expect now...’ In effect a sense of uncertainty at times was sometimes welcomed and depended on, as one child noted, ‘how differently they or things work out.’ Things could turn out to be more positive than what was expected. Parents and children would in ceratin instances tried to retain some degree of uncertainty even when they had the opportunity to do otherwise. For example, even when parents or children had some sense that certain symptoms were not ‘normal’ or ‘not a good sign,’ they nonetheless kept silent at times. That is, they would not always immediately talk to the physicians or nurses about what the symptoms could possibly be indicative of in relation to the child’s prognosis. Despite their suspicions, many days and sometimes weeks would pass before ‘seasoned’ parents and their children would directly seek confirmation from members of the health-care team in relation to what the symptoms meant. At time this was further compounded by children being silent to their parents about certain symptoms that indicated to the children that they were ‘getting sick’ again. Being silent and not asking question resulted in both children and parents remaining in a state of uncertainty even if it was artificial at European Journal of Oncology Nursing 6 (4),191^202

times. Sustaining a semi-authentic state of uncertainty helped them to get through some of the more difficult times of the cancer course. It was better for them in such instances to delay being told the possibility of ‘bad news,’ even though they realized they would inevitably have to know one way or another. Another example of the need to remain uncertain was provided by a mother who felt more comfortable with her son taking a chance with a BMT than knowing for sure whether or not her child would live or die. She had commented that ‘well once the doctor told me he only had two months to live (pause), I talked to the other kids and Chad (ill child) and my parents and Chad wants to fight...Chad had wants to go out fighting. He wants to give up the option that he has to live with two months of certainly dying and instead take a chance, we are taking a chance.’ This mother, however, also at times felt it would have helped if she had been made more aware of all potentially bad news and was certain about the child’s future, ‘I did not even know if we were going to have him for Christmas, I was not sure what was going on. It was frustrating in that I want to know, it gets frustrating with God, like what, what, you know...because I would get on with dealing with it. But I do not know what I am dealing with right now.’ In this instance knowing what to expect for this mother was both a negative and positive experience. Additionally, while children and their parents in the end wanted to know everything about the cancer and its treatment regimen including potential symptoms, knowing what to expect did not always ease their discomfort. That is, experiencing ‘certainty’ was not without some discomfort as made evident by a mother whose adolescent child knew from past experience how she would feel after receiving a particular chemotherapy agent. The mother expressed ‘She (the adolescent child) was saying that she was really sick after that medication...she did not eat well and she was quite nauseated. So she is not looking forward to this day cause today is the beginning of that cycle in which she gets that medication again...I think she gets it 5 times every ten days and the dose keeps increasing unless her liver functions go off and then if they are off, she will not get it at all. So she is really hoping that her liver functions go off.’ In experiencing the ‘rough spots’ both certainty and uncertainty could be experienced simultaneously. Families found at times life harder to live regardless if they were experiencing certainty or uncertainty.

We are certain about some things While uncertainty became a way of life for children and families, this did not mean that they experienced a complete absence of certainty in

Uncertainty in children with cancer and their families

their lives in relation to the cancer. In fact, because of the cancer children and families developed definite beliefs about particular aspects of their lives. Most of them expressed that they were ‘certain that their lives would forever be different’ because of the cancer diagnosis. Although initially they did not what the difference would entail, they nonetheless knew they were now embarking on a new journey. A journey different from the one in which they had originally planned and hoped for. Another certainty acknowledged by all of the children and their families was that their lives would now be filled with more suffering and anguish. They were certain that as a family they would experience both mental and physical suffering. In fact, many families felt that the added suffering was necessary in order to get through the cancer trajectory. Perhaps even more revealing and troubling was that some parents were certain that in the end, their child’s cancer would eventually be responsible to ending their child’s life. Be it in the immediate future during the course of their child’s initial cancer treatment or in the remote future, many years after the child had successfully completed his/her cancer treatment. Parents always had the nagging feeling that one way or another, the cancer would succumb their child. Some parents were certain that at the end of the day, the cancer would eventually lead to their children dying even if did not happen till years later or until the children became adults, ‘Regardless how well he does, I know something bad will happen and that he will eventually die from the cancer.’ In contrast, children (children with cancer and their siblings) more often held the attitude that they were certain the cancer could be beaten and even more importantly would never return. Feeling and believing that the cancer would not get the better of them helped them to get through the cancer trajectory.

DISCUSSION The literature related to the impact of childhood cancer on children and families describes many difficult aspects of cancer that can result in confusion, despair, worry, caregiver burden, and most notably the presence of uncertainty (Clarke-Steffen 1997, Comaroff and Maguire 1981, 1986, Hinds 1990, Neville 1998). However, although research has indicated that uncertainty is the most striking feature of childhood cancer for children and families (Comaroff and Maguire 1981, 1986), this study revealed that what families found to be most difficult about cancer varied and depended on the context of the situation. In addition to the uncertainty, the

199

families found the characteristics of restrictions, extra work, multitude of feelings, and loss very difficult at times to deal with and were closely associated with the families’ sense of uncertainty. The variability in the intensity of each characteristic explains why children and families could not definitely pinpoint what was the most difficult part of having to get through the cancer experience. All the characteristics identified in this study including uncertainty contributed to the arduous cancer journey. Throughout the cancer trajectory, families experienced varying degrees of uncertainty depending on what families were experiencing in relation to the ‘rough spots.’ However, even when children and families experienced fewer difficulties, the families nonetheless experienced uncertainty. This was also the norm once treatment was completed and the children remained in remission. While the cancer experience no longer dominated the life of those families in which children remained cancer-free and while certain details of the cancer experiences faded over time for them, there was always the uncertainty of the cancer possibly returning. Even when the child remains in remission and is symptom-free, cancer remains problematic for families due to the uncertain nature of the cancer; there is the ever-present threat of fatal relapse, a threat that does not diminish with time (Comaroff and Maguire 1981, 1986). The uncertainty experienced by families who have children with chronic, life-threatening illness is best referred to as sustained uncertainty (Cohen 1993, 1995). Acknowledging that families always experience uncertainty in relation to cancer has implications for nurses and other health-care providers caring for these individuals. As most families expressed in this study ‘it (cancer) is never over with,’ points to the need for those caring for survivors of childhood cancer to always assess and be aware of increased stress resulting from feelings of uncertainty. The findings reinforce the complex nature of uncertainty. For the families in this study, it was much more than ‘not knowing.’ The multidimensional nature and variability of uncertainty warrant further study in order verify and expand on the different dimensions of uncertainty. Scale development assessing the type, frequency, and severity of uncertainty in relation to the different ‘rough spots’ would help nurses and others in designing interventions that are more familyfocused and sensitive. Perceived quality of life could also be assessed in relation to the overall frequency and severity of the uncertainty. One of the most significant findings that emerged from this study was that the experience of uncertainty was not always perceived as something totally negative. That sometimes waiting was not worse than knowing. Likewise, European Journal of Oncology Nursing 6 (4),191^202

200 European Journal of Oncology Nursing

feelings of certainty did not always bring peace and ease of mind to the individual, but instead like uncertainty led to more unrest and stress. Even when families wanted to be certain about the future and know everything that there was in relation to specific events, they still could experience increased stress. This study showed that perceptions of living in a world with more or less uncertainty and certainty varied depending on the context of the situation. There is tendency, however, to assume that uncertainty is the greatest psychosocial stressor confronting families and that any reduction of improves coping with stress (Cohen and Martinson 1988, Comaroff and Maguire 1981, 1986, Humphrey et al. 1996). However, Cohen (1993) noted that if individuals were fully aware of all the uncertainties of human life, that awareness would be incapacitating. Mishel (1988) reinforced that uncertainty is neither an inherently dreaded or desired state, but instead its assigned value is based on the individual’s determination of what the potential implications of the uncertainty may mean to that individual. We speculate that living in a world in which one is always certain about the future can be negative for two reasons. First, families’ sense of hope may be diminished if they always know what to expect in relation to future negative events. They may become paralyzed in responding and moving on during the cancer trajectory. Secondly, always knowing what to expect in relation to all future positive events could take away the surprise and joy of the good times that they may experience in the future. Always being certain also does not allow for a change of plans. Depending on the context, we suggest that uncertainty like certainty may at times help families to cope. It is important that nurses and other health-care providers realize that increased stress can be associated with feelings of certainty as well as uncertainty. This study supports the finding that families of children with cancer want to become informed in order to be able to better deal with the cancer (Brett and Davies 1988, Clarke-Steffen 1997, 1993, Cohen 1993, 1995, Comaroff and Maguire 1981, 1986). Nurses and other health-care providers need to inform families of all aspects of the child’s illness condition. However, healthcare providers need to do so in a manner that accounts for the unique needs of each family. They need to be cognizant of the families’ readiness and desire to find out more about the children’s cancer. In addition to the type of information, the timing of information-giving needs to be considered. Practicing anticipatory guidance on the part of nurse is also vital in helping to prepare families to deal with all the ‘rough spots’ of cancer and helps to overcome some of their uncertainty (Cohen 1995). Nurses European Journal of Oncology Nursing 6 (4),191^202

need to acknowledge the uniqueness of each family’s experience, yet also support families to share their knowledge and wealth of experience related to the uncertain nature of cancer with other families experiencing similar cancer-related events. Poorly controlled symptoms contributed greatly to the sense of distress experienced by children and family. Research has shown that changing symptom states and body variability impacts greatly on how families manage lifethreatening illness and contributes to feelings of uncertainty (Cohen 1995). This reinforces the importance of controlling symptoms in childhood cancer (Schmidt et al. 1994). It is important to note that parents and children were sometimes silent about the symptom experiences. They relied on the process of mutual pretence to help get them through the uncertainty of the cancer-symptom trajectory. Bluebond-Langner (1978) noted that practicing mutual pretense enabled children with cancer and others to fulfill their role expectations and preserve social membership when continued membership was threatened. It allowed them to prevent relationships from breaking as well as maintaining some sense of normalcy. Ways that will promote ongoing and open-ended communication within the family unit and between families and health-care providers need to be encouraged. Although the findings cannot be generalized, nurses and other health-care providers when deemed appropriate within a particular context may use the findings to help guide them in developing relevant plans of care. Nurses need to focus on the experiences that contribute to the uncertain nature of the cancer journey. This involves seeking an understanding of children’s and families’ perspectives about specific cancer events and the meanings that families assign to the events and degree of uncertainty. Assessing how much information families desire about the cancer experience must be ongoing with recognition that each family experiences cancer in their own unique way. Most important is acknowledging that clinical knowledge cannot be either evaluated or transformed in any simple, decontextualized manner (Comaroff and Maguire 1981, 1986). Ongoing research that involves larger sample sizes of children with cancer will add to our understanding about uncertainty in the cancer trajectory. Recognizing that the majority of children and their families were Caucasian, research attention needs to be directed at how children from other ethnic and culture groups experience uncertainty. To date, there is minimal understanding with respect to the role culture plays in childhood cancer and how uncertainty is experienced.

Uncertainty in children with cancer and their families

CONCLUSION To conclude, findings from this study add to our understanding about the experience of uncertainty in children with cancer and their families. Children and families reinforced that there are many difficult events that one must go through because of childhood cancer, and that the events can lead to increased uncertainty. The uncertainty is experienced as a dynamic condition that can vary with each ‘rough spot’ as well as within each family and from family to family. To suggest that the uncertainty associated with childhood cancer is complex and multi-dimensional in nature would be an understatement. Further development of research and education programs specific to uncertainty in childhood cancer will assist nurses and other health-care providers to provide care to children with cancer and their families that is both sensitive and comprehensive. Cohen and Martinson (1988) suggested that the focus should be on helping families cope with uncertainty rather than focusing on trying to remove the uncertainty. They believed that the latter is an unlikely goal to achieve, considering the fact that uncertainty becomes an embedded dimension in the lives of families experiencing childhood cancer. Ultimately, future investigations and interventions need to focus on the long-term goal of enhancing the quality of life in children with cancer and their families. Anything that eases the anguish of the childhood cancer that results from the feelings of uncertainty is critical. Helping families feel and know that some things are indeed carved in stone would be of benefit and may give families a greater sense of control and certainty that they may prevent the shoe from dropping. As one mother noted ‘I think the shoe has attempted to drop but it was not there...’ would be more desirable than feeling for certain that the other shoe would always end up dropping.

ACKNOWLEDGEMENTS The first author was supported by fellowships from the National Health and Research Development Program Doctoral Student (NHRDP: Canada), the Pat Scorer Fellowship (Winnipeg Children’s Hospital Foundation of Manitoba Inc.), the Maurice Legault Clinical Cancer Nursing Fellowship (Canada), and the Toronto Sick Children’s Hospital Fellowship: Canadian Nurses Doctoral Student Study Award. The second author is supported by a Chair of the Canadian Institutes of Health Research and Canadian Health Services Research Foundation.

REFERENCES Beck C (1990) Qualitative research: methodologies and use in pediatric nursing. Issues in Comprehensive Pediatric Nursing 13: 193–201

201

Bernheimer L (1986) The use of qualitative methodology in child health research. Children’s Health Care 14(4): 224–232 Bluebond-Langner M (1978) The private worlds of dying Children. Princeton University Press, Princeton, NJ Brett K, Davies E (1988) ‘What does it mean?’ Sibling and parental appraisals of childhood leukemia. Cancer Nursing 11(6): 329–338 Clarke-Steffen L (1993) A model of the family transition to living with childhood cancer. Cancer Practice 1(4): 285–292 Clarke-Steffen L (1997) Reconstructing reality: family strategies for managing childhood cancer. Journal of Pediatric Nursing 12(5): 278–287 Cohen M (1993) The unknown and the unknowableFmanaging sustained uncertainty. Western Journal of Nursing Research 15(1): 77–96 Cohen M (1995) The triggers of heightened parental uncertainty in chronic, life-threatening childhood illness. Qualitative Health Research 5(1): 63–77 Cohen M, Martinson I (1988) Chronic uncertainty: its effect on parental appraisal of a child’s health. Journal of Pediatric Nursing 3(2): 89–96 Comaroff J, Maguire P (1981) Ambiguity and the search for meaning: childhood leukemia in the modern clinical context. Social Science and Medicine 15B: 115–123 Comaroff J, Maguire P (1986) Ambiguity and the search for meaning: childhood leukaemia in modern clinical context. In: Conrad P, Kern R (Eds.) The sociology of health and illness: critical perspectives, (2nd edn.). St. Martin’s Press, New York Creswell J (1998) Qualitative inquiry and research design: choosing among five traditions. Sage, Thousand Oaks, CA Deatrick J, Faux S (1989) Conducting qualitative studies with children and adolescents. In: Morse J (Ed) Qualitative nursing research: a contemporary dialogue. Aspen, Rockville, MD Denzin N (1989) Interpretive Interactionism. Sage, Newbury Park, CA Eiser C, Havermans T, Eiser J (1995) Parents’ attributions about childhood cancer: implications for relationships with medical staff. Child: Care, Health and Development 21(1): 31–42. Faulkner A, Peace G, O’Keefe C (1995) When a child has cancer. Chapman & Hall, London, Great Britain Ferrell B, Rhiner M, Shapiro B, Dierkes M (1994) The experience of pediatric cancer pain, Part I: impact of pain on the family. Journal of Pediatric Nursing 9(6): 368–379 Field P, Morse J (1985). Nursing research: The application of qualitative approaches. Aspen, Rockville, MD.0 Hinds P (1990) Quality of life in children and adolescents with cancer. Seminars in Oncology Nursing 6(4): 285–291 Hogan C (1997) Cancer nursing: the art of symptom management. Oncology Nursing Forum 24(8): 1335–1341 Humphrey G, Skeel J, Boon C (1996) Uncertainty: a conceptual model applied to parental stress associated with prognostic uncertainty in pediatric oncology. International Journal of Pediatric Oncology 3: 47–51 Hyde´n L (1997) Illness and narrative. Sociology of Health and Illness 19(1): 48–69 Langeveld N, Ubbink M, Smets E (2000) ‘I don’t have any energy’: the experience of fatigue in young adult survivors of childhood cancer. European Journal of Oncology Nursing 4(1): 20–28 Mishel M (1988) Uncertainty in illness. IMAGE: Journal of Nursing Scholarship 20(40): 225–232 European Journal of Oncology Nursing 6 (4),191^202

202 European Journal of Oncology Nursing Mishel M (1996) Living with chronic illness: Living with uncertainty. In: Funk S, Tornquist E, Champagne M, Wiese R (Eds) Key aspects of caring for the chronically ill, hospital and home. Springer Publishing Company, New York Mishel M (1999) Uncertainty in chronic illness. In: Fitzpatrick J (Ed) Annual review of nursing research Vol. 17. Springer Publishing Company, New York Morse J, Field P (1995) Qualitative research methods for health professionals, 2nd Edn. Sage, Thousand Oaks, CA Neville K (1998) The relationship among uncertainty, social support, and psychological distress in adolescents with cancer. Journal of Pediatric Oncology Nursing 15(1): 37–46 O’Brien M (2001) Living in a house of cards: family experiences with long-term childhood technology dependence. Journal of Pediatric Nursing 16(1): 13–22 Patton M (1990) Qualitative evaluation and research methods, 2nd Edn. Sage, Newbury Park, CA Prus R (1996) Symbolic interaction and ethnographic research. State University of New York Press, Albany, New York

European Journal of Oncology Nursing 6 (4),191^202

Rhiner M, Ferrell B, Shapiro B, Dierkes M (1994) The experience of pediatric cancer pain, part II: management of pain. Journal of Pediatric Nursing 9(6): 380– 387 Schmidt K, Eland J, Weiler K (1994) Pediatric cancer pain management: a survey of nurses’ knowledge. Journal of Pediatric Oncology Nursing 11(1): 4–12 Sterken D (1996) Uncertainty and coping in fathers of children with cancer. Journal of Pediatric Oncology Nursing 13(2): 81–88 Strauss A, Corbin J (1990) Basics of qualitative research: grounded theory procedures and techniques. Sage, Newbury Park, CA van Manen M(1990) Researching lived experience: human science for an action sensitive pedagogy. Althouse, London, Ontario Woodgate, R (2001). Symptom experiences in the illness trajectory of children with cancer and their families. Dissertation Study, University of Manitoba, Winnipeg, Manitoba, Canada