Journal of Pediatric Nursing (2014) xx, xxx–xxx
Parental Uncertainty in Illness: Managing Uncertainty Surrounding an ‘Orphan’ Illness1 Anna M. Kerr MA a,⁎, Stephen M. Haas PhD b a
Department of Communication, University of Kentucky, Lexington, KY Department of Communication University of Cincinnati, Cincinnati, OH
b
Key words: Uncertainty management; Parental uncertainty; Healthcare decision-making; Communication
Parents of children with complex chronic illnesses experience substantial uncertainty that is heightened when the condition is an “orphan” illness not belonging to one medical specialty. The current study explores uncertainty experienced by parents of children with “orphan” illnesses requiring multidisciplinary care. Method: Participant-observations over 13 months (n = 200) were combined with questionnaire data (n = 55) to assess parental uncertainty at a multidisciplinary pediatric clinic. Results: Five unique types of uncertainty emerged from a grounded analysis ( Glaser & Strauss, 1967), revealing 11 interrelated uncertainties these parents experience. Findings can help providers understand parents' uncertainty and assist in family-centered decision-making. © 2014 Elsevier Inc. All rights reserved.
WHEN CHILDREN ARE diagnosed with chronic illnesses, parents make a radical shift from a life of takenfor-granted expectations to a life of heightened uncertainty (Cohen, 1993), lack of control, and less optimism (Madeo, O'Brien, Bernhardt, & Biesecker, 2012). One source of uncertainty is a lack of adequate information about the illness (Clarke-Steffen, 1993). For example, parents of children born with vascular anomalies (i.e., birthmarks) experience uncertainty because one consistent terminology does not exist to diagnose the condition (Rieu & Festen, 1996). Consequently, nearly 60% of vascular anomalies are misdiagnosed (Patel & Curry, 2010). Although some vascular anomalies are easily treated, some complex anomalies can be life-threatening if left untreated and can cause blood clots, limb length discrepancies, seizures, and intellectual deficiencies (Patel & Curry, 2010). Many birthmarks also affect other parts of the body, including major organs. Therefore, the complex nature of vascular 1
A previous version of this study was presented at the International Communication Association Annual Convention, Boston, MA: May 26– 30, 2011. ⁎ Corresponding author: Anna M. Kerr, MA. E-mail address: [email protected]. 0882-5963/$ – see front matter © 2014 Elsevier Inc. All rights reserved. http://dx.doi.org/10.1016/j.pedn.2014.01.008
anomalies requires coordinated care by physicians from multiple disciplines (Richter & Friedman, 2012). The need for multiple specialists classifies birthmarks as “orphan” illnesses not belonging to one medical specialty. The multifaceted nature of vascular anomalies provides an ideal context for studying parental uncertainty. The purpose of this study is to explore uncertainty experienced by parents who face unclear diagnosis and treatment options. Uncertainty exists when situations are unpredictable, often due to inconsistent or unavailable information (Brashers, 2001). Therefore, illness uncertainty stems from the struggle to categorize symptoms and information associated with one's health, inhibiting the ability to predict outcomes (Mishel, 1988). Mishel's (1988) theory of uncertainty in illness outlines four types of illness uncertainty: (a) ambiguity concerning the state of the illness, (b) complexity regarding treatment and systems of care, (c) lack of information about the diagnosis and seriousness of the illness, and (d) unpredictability of the course of the disease and prognosis. Additionally, uncertainty can simultaneously be medical, social, and personal–forcing individuals with chronic illnesses to integrate uncertainty into daily life (Brashers et al., 2003; Mishel, 1999).
2 Contrary to previous assumptions that uncertainty is a negative experience reduced by seeking information, recent research suggests more information can actually intensify uncertainty by increasing alternatives or suggesting unwanted outcomes (Brashers et al., 2000; Shannon & Weaver, 1949). Ultimately, uncertainty can only be reduced if information is adequate for decision-making purposes (Clatterbuck, 1978; Sheer & Cline, 1995). However, information that helps answer one question often introduces new questions and uncertainties (Babrow & Dutta-Bergman, 2003). This “chaining of uncertainties” suggests uncertainty cannot be isolated, but exists in relation to other uncertainties (Babrow, 2001). Thus, information seeking might not always be the most helpful strategy for managing uncertainty (Brashers, 2001). Instead, uncertainty management theory (UMT) proposes that individuals communicate in a variety of ways to manage uncertainty depending on their appraisal of uncertainty (Brashers, 2001; Brashers et al., 2000). According to uncertainty management theory (Brashers et al., 2000; Brashers, 2001), uncertainty manifests differently for different people and in different situations. Consequently, individuals may seek or avoid information to eradicate, cultivate, or maintain feelings of uncertainty depending on their appraisal. When uncertainty is appraised as a danger, individuals generally seek relevant information to clarify alternatives (Hogan & Brashers, 2009) or relieve discomfort (Bradac, 2001). If uncertainty is perceived as a way to maintain hope, seeking information can help amplify uncertainty by increasing alternatives or challenging existing beliefs (Brashers et al., 2000; Hogan & Brashers, 2009). Individuals may also avoid information to maintain uncertainty and avoid unwanted outcomes (Barbour, Rintamaki, Ramsey, & Brashers, 2012). Contrary to previous uncertainty theories, UMT posits that more information can often increase uncertainty, so managing information becomes important. Consequently, individuals with chronic illnesses must adapt to chronic uncertainty through continued reappraisals over the trajectory of their illness (Brashers et al., 2000), which results in adequately seeking and avoiding information as necessary. Adapting to chronic uncertainty is vital for parents of children with chronic illnesses. Parents' illness uncertainty stems from parents' desire to envision their child's future (Santacroce, 2001). Uncertainty is typically intensified during the diagnosis phase of the illness when parents experience uncertainty about the severity of the illness and the unpredictability of the future (Clarke-Steffen, 1993). Uncertainty also manifests in fear of the pain the child might endure and the possibility of death (Binger et al., 1969). Not surprisingly, parents with higher levels of uncertainty often report a lack of personal control, less optimism, and higher perceptions of disease severity when their child's medical condition is unknown (Madeo et al., 2012). Therefore, when parents do assign meaning to the illness,
A.M. Kerr, S.M. Haas they often do so negatively because of the perceived threat to the health of the child (Hoff et al., 2005). Therefore, the uncertainty associated with chronic illness can cause high levels of stress (Dodgson et al., 2000; Santacroce, 2003). Parents of children with chronic ‘orphan’ illnesses, such as vascular anomalies, experience uncertainty because of the complexity of care. Vascular anomalies are a challenge for physicians because each occurrence is unique (Rieu & Festen, 1996). Many physicians are willing to treat uncomplicated birthmarks, such as port-wine stains, but less willing to treat, or even diagnose, complex anomalies (Mathes, Haggstrom, Dowd, Hoffman, & Frieden, 2004). These multifaceted vascular anomalies require care from physicians from numerous disciplines to diagnose and to manage (O'Regan et al., 2007). Consequently, patients risk being misdiagnosed or mismanaged if the right physicians are not consulted (Mathes et al., 2004). The review of previous literature suggests parents experience unique uncertainty while caring for a child in the context of multidisciplinary care and in the face of inadequate information. The goal of this study is to examine and extend existing constructions of uncertainty by exploring how parents of children with vascular anomalies experience and manage uncertainty.
Methods Data for this study were collected through triangulating participant-observations, discussions with parents, and an open-ended questionnaire at a multidisciplinary vascular anomalies clinic within a large midwestern children's hospital. The study was approved by the authors' institutional review board, and all participants provided informed consent prior to study involvement. Parents were asked to participate in the study when they arrived for their child's appointment and provided informed consent at that time. One author also visited the clinic weekly over 13 months to observe over 200 physician–parent–child interactions in the exam room as well as interactions among the multi-disciplinary team members. Youth assent was attained if the child (patient) was between the ages of 11 and 17. Although the children (patients) did not complete the survey instrument, the youth assent was required for the observation of the clinic visit during which the child would be present.
Instrument Data were collected through triangulating participantobservation, discussions with parents, and an openended questionnaire developed after several months of participant-observations. Parents completed the questionnaire in the waiting or exam room before seeing
Parental Uncertainty in Illness the physicians. The questionnaire included quantitative measures, several open-ended questions, and demographic items. This study focuses on participant-observations, parent discussions, and the open-ended responses from the questionnaire to explore how parents experience uncertainty. In the initial survey, parents responded to two open-ended questions: (a) “Explain any anxiety, uncertainty, or concerns you have about your child's illness.” and (b) “How knowledgeable do you feel about your child's condition?" In line with emergent qualitative design, after more participant-observations, a third openended question was added that asked participants, (c) “Do you feel capable of explaining the actual condition, treatment options, and future progression of your child's hemangioma or vascular malformation to your family, friends, pediatrician, etc.?” This question stemmed from the recurring observation that many parents were visibly unsure about the fundamentals of their child's health condition. Several parents had trouble even locating the clinic because they were unable to pronounce their child's diagnosis or explain what specialists they were seeing at the hospital. The open-ended responses allowed the researchers to use the parents' own words to explain the experiences of uncertainty salient when managing an orphan illness. In addition to the questionnaire, the first author, who was born with a vascular anomaly, observed approximately 200 parent–child–physician interactions. Parents frequently asked the researcher questions during or after the medical interview about social and health-related issues she had experienced growing up. It was during these discussions that additional experiences of uncertainty emerged beyond those discussed during the physical examination. Throughout the 13-month participant observations, theoretic notes were taken and analyzed in conjunction with questionnaire responses to provide a more holistic analysis of parents' uncertainty.
Participants Table 1 presents an overview of the survey participant demographics. The sample consisted of 55, Englishspeaking, parents of children with vascular anomalies (46 females, 7 males) (M = 32.9 years of age, SD = 6.9, range = 19–50 years). The children (patients) included 39 females and 14 males (M = 3.9 years of age, SD = 5.3, range = newborn–17 years). This proportion is consistent with the research that indicates that the most prevalent vascular anomalies (hemangiomas) occur most frequently in White females, premature infants, and twins (Burrows, Laor, Paltiel, & Robertson, 1998; Fishman & Mulliken, 1993). Approximately 34.5% of the parents were visiting the clinic for the first time, while 60% of parents had visited at least once before (see Table 1). Similar demographics were observed in the parent–child–physician interactions.
3 Table 1 Summary of Survey Participant Demographic Variables (n = 55). Variable Gender Male Female Age Education Some high school Completed high school Some college Completed college Some graduate school Completed graduate school Income $15,000 or below $16,000–$30,000 $31,000–$50,000 $51,000–$75,000 $76,000–$100,000 $100,000 or above Clinic visit First visit Return visit 2–5 visits 6–10 visits 10 or more visits
Parents (%, n)
Children (%, n)
12.7%, (7) 83.6%, (46) M = 32.9 years
25.5% (14) 70.9% (39) M = 3.9 years
12.7% 14.5%
(7) (8)
14.5% 29.1% 5.5% 21.8%
(8) (16) (3) (12)
14.5% 10.9% 20.0% 18.2% 14.5% 20.0%
(8) (6) (11) (19) (8) (11)
34.5% 60.0% 32.7% 16.4% 10.9%
(19) (33) (18) (9) (6)
Data Analysis The researchers analyzed field notes and open-ended responses for themes through a process consistent with constant comparison analysis (Glaser & Strauss, 1967; Strauss & Corbin, 1990). Open coding was used to identify categories and sub-categories within the data. Through a process of axial coding, the categories were organized into themes that emerged inductively and in line with the authors' knowledge of previous research. The authors met regularly to organize emergent themes into sub-categories to form an accurate representation of the findings (Strauss & Corbin, 1990). Sub-categories were combined into larger super-ordinate categories based on their conceptual uncertainty-related connections until saturation occurred (Glaser & Strauss, 1967). The themes emerged from analysis across the data sources, and parents expressed similar types of uncertainty within participant-observations, discussions, and the open-ended responses providing a participant validity check for the presence of the themes across the sources of data. In addition, three scholars familiar with uncertainty, health, and family communication reviewed the emergent themes and exemplar quotes for validity. There was unanimous agreement that the themes reflected parents'
4
A.M. Kerr, S.M. Haas
descriptions of their experiences and uncertainty management.
Results The analysis of the open-ended questionnaire data and participant-observations revealed five themes capturing parents' uncertainty experiences and management: (a) normalization uncertainty, (b) information uncertainty, (c) orphan-illness uncertainty, (d) parental proxy uncertainty, and (e) social stigma uncertainty.
Normalization Uncertainty Parents expressed uncertainty about the future and were concerned about how their children's birthmarks would influence their ability to be “normal.” For example, a mother of a 4-year-old girl wrote, “I am most concerned about how my child's illness will affect her life.” The mother of a 12-year-old girl expressed: [I] Just want to be able to help my child and someone to tell me all what's going on and is this something she will have to live with rest of her life, getting her pain controlled, and getting her back to being the run of the mill child.
One mother also had concerns about having more children stating, “I am most concerned about how my child's illness will affect her life. I am also anxious about knowing exactly what her diagnosis is so I can be aware when I have more children.” Normalization uncertainty was also common during participant observations. Parents often asked about the child's ability to engage in activities at school. For example, one mother was concerned about her teenage daughter's ability to play sports at school because her birthmark that was located on the bottom of her foot. While the mother also was concerned about pain, the overall source of her concern was whether her daughter could participate in “normal” activities.
Information Uncertainty Parents expressed concerns about inadequate information regarding whether surgery was an option, how difficult surgery would be, and whether additional surgeries were needed. Because treatment options for birthmarks are not consistent, it is not always possible for physicians to give parents confident answers when asked about treatment. One mother of an 11-week old daughter expressed concern about the physicians' uncertainties about surgery. She stated, “On the first visit, it seemed that the derm[atology] resident and the surgeon were discussing the treatment option in front of me as ‘experimental’ and I wasn't sure they really felt it would work.”
A mother of a 17-week old daughter also expressed concern about surgery after receiving multiple conflicting opinions: We were [originally] referred to a pediatric surgeon. His advice was to remove [the birthmark], we opted to observe for another month, but the size continued to increase and became very painful. I was concerned it was infected, so the surgeon offered to perform surgery that next Tuesday….The radiologist strongly recommended we find a ‘clinic’ who specializes in hemangiomas/ vascular [malformations] for a second opinion. He did not agree with the surgeon to remove it the next day. The surgeon wanted to go through with the surgery, but we went on the Internet & found this clinic. That night before the scheduled surgery I was very anxious & tearful. I couldn't put my finger on it, but going ahead with surgery with a general pediatric surgeon did not ‘feel’ right. I felt we needed a second opinion from someone who specializes in this area and does this all the time....I am still anxious a bit, b/c I don't know what the [treatment] will entail, but I do feel I am at the correct place, with the right expertise to get the best care for our precious baby girl.
This mother's story illustrates the inadequate and conflicting information that parents receive in treating vascular anomalies and how that information can elicit more uncertainty. Many parents also had uncertainty about the basic risks of performing surgery. One mother of an 8-year-old son expressed, “Since I am unsure about the future, I want things taken care of now, but I am afraid of the risk of surgery,” illustrating the complex dilemma stemming from incomplete and inconsistent information about treatment and the future.
Orphan-Illness Uncertainty Some parents indicated concern about the influence of their child's birthmark on other parts of the body. For example, one mother stated, “My main concern is that she will lose her vision if the [birthmark] grows. I worry that I could let it go too long before I bring her back.” Another mother, whose daughter had a birthmark on her breast, expressed concern that the nipple would be damaged during surgery. Uncertainty about complex health risks was also prevalent during participant observations. One mother of a newborn child seeing the oncologist (one of the many physicians from the multidisciplinary team), waited until the physician had nearly left the room before blurting out “So it's not cancer, right?” Despite the conversation she had just had with an oncologist about the benign vascular anomaly, she was concerned that the birthmark had other health risks – especially cancer. The mother might not have had previous concerns about the birthmark being cancerous, but because
Parental Uncertainty in Illness she was seeing a pediatric oncologist, who traditionally cares for children with cancer, she became preoccupied with the confusing health risks of an orphan condition.
Parental Proxy Uncertainty Assessing pain and deciding when to seek care was a particularly difficult experience for parents in this study. This is common for proxy decision makers who frequently have different perceptions of the intensity of pain and the ability to control pain than the patient for which they are making medical decisions (Ferrell, Grant, Borneman, Juarez, & ter Veer, 1999). For example, a mother of a 2-year-old boy expressed how hard it is for a parent to determine when and how much pain their child is suffering: My son's lump has grown in size and I worry how big it's going to get, and I pray it doesn't hurt him. I know he doesn't like it to be touched but I don't know what it feels like every time he sits down.
The father of a 6-month-old girl was also uncertain about pain: We are concerned about her lip possibly getting worse, but we understand with [birthmarks] that many times they get bigger then subside and go away. We just want to be sure that she does not suffer any pain with ulcerations to her lip.
Parents also frequently asked the first author about the amount of pain she experienced and how her parents decided to pursue surgery. They wanted to find out from someone who had been through similar experiences if the pain was tolerable and how other parents have made ‘proxy’ decisions about surgeries for their child.
Social Stigma Uncertainty Parents frequently expressed concern about their child experiencing social stigma by being teased in school or asked about their birthmark by strangers. One mother expressed concern about the birthmark being in a prominent place on her son's body. She explained, “[His] Birthmark is in a visible place on [his] neck and [I'm] afraid that kids will make fun of him now and later on.” Another mother of a 7year old girl, who has been to the clinic more than ten times, explained how she tries to prevent potentially harmful psychological issues, but is still uncertain about stigma her child may face. She explained, “I think the not knowing what will happen with her illness is the hard part. I have tried to raise my daughter to be confident, but the questions and the teasing from other kids makes it hard.” Another mother referenced the concerns her 8-year-old son already has about his birthmark on his leg and the compression hose prescribed to him to control pain and swelling. She explained that her uncertainty came from “[t]he concerns of how his peers react
5 as he gets older to his compression hose. He doesn't want people thinking he wears ‘panty hose.’” The uncertainty about social stigma was also present during participant observations. Over the course of the 13months, several parents were visibly upset at the physicians' cautions about treatment or advice to “wait and see.” These parents frequently requested treatments or surgery to manage their child's often-concealable birthmark, against medical advice to wait, because they feared social stigma being expressed toward their child.
Discussion The current findings confirm existing literature on illness uncertainty, and highlight unique dilemmas experienced with orphan conditions. Patients with birthmarks are at a higher risk of misdiagnosis, require the care of multiple specialists, and often receive inconsistent information. These challenges illustrate the potential for more information to increase parental uncertainty (Brashers et al., 2000). Parents reported experiencing: (a) normalization uncertainty, (b) information uncertainty, (c) orphan-illness uncertainty, (d) parental proxy uncertainty, and (e) social stigma uncertainty. These interwoven uncertainties illustrate that chronic-illness uncertainty is frequently a combination of medical, social, and personal uncertainties and is multilayered and complex (Brashers et al., 2003). In Table 2, we summarize eleven explanatory principles that emerged across the five uncertainty areas. Normalization uncertainty exemplifies the conceptualization of uncertainty as unpredictability (Mishel, 1988). Parents struggled with the inability to predict a “normal” future for their child and assessing the valence of the situation. As a result, parents overwhelmingly appeared to be preoccupied with thoughts of potentially negative outcomes (danger appraisals), leading them to seek information from multiple health care providers to manage their uncertainty. These results suggest that parents are highly unlikely to appraise uncertainty about their child's future as an opportunity or believe it is acceptable to avoid information about their child's medical condition. However, as the results suggest, seeking and increasing information can lead parents to increased uncertainty (Brashers et al., 2000). The area of information uncertainty highlights three of the four established types of illness uncertainty: (a) complexity regarding treatment, (b) lack of information, and (c) unpredictability of the illness (Mishel, 1988). Parents' tendency to seek information from health care providers illustrates the role physicians play in illness uncertainty management (Brashers et al., 2000). However, physicians are not always able to provide complete information regarding the diagnosis and seriousness of the illness because consistent information about treatment for vascular anomalies has not been established. This lack, or
6
A.M. Kerr, S.M. Haas Table 2
Core Principles of Parental Uncertainty Management.
Uncertainty
Core principles
Normalization uncertainty
1. Parents experience uncertainty about the impact the condition may have on their child’s ability to be “normal” in the future. 2. Parents' thoughts of potentially negative circumstances that are unknown about the future lead to information-seeking behaviors. 1. Increased and/or inconsistent information can complicate parents' understanding of treatment options and predicting outcomes. 2. Parents face ‘ambivalent uncertainty dilemmas’ from treatment-related danger versus recovery-related opportunity appraisals. 3. Parents rely on emotional (‘gut feeling’) appraisals to manage uncertainty resulting from increased or inconsistent information. 1. Concerns about additional health risks related to multiple parts of the body complicate parents' uncertainty regarding the complex condition. 2. Multidisciplinary uncertainties create interrelated uncertainty chains. 1. Parents' uncertainty can stem from making decisions on their child's behalf. 2. Parents face uncertainty when deciding to pursue or withhold treatment due to lack of direct physical sensation and pain cues, heightened when the child is too young to communicate pain levels. 1. Parents have uncertainty from the fear of social stigma put on their child. 2. Parents' emotional appraisal of social stigma uncertainty can push parents to pursue treatments in haste or against medical advice.
Information uncertainty
Orphan-illness uncertainty
Parental proxy uncertainty
Social stigma uncertainty
inconsistency, of information does not provide parents with the cues needed to predict outcomes (Mishel, 1990). Information uncertainty from multiple medical opinions can also create a dilemma for parents. Parents want to treat their child's condition as early as possible to ensure a healthy future for their child and avoid future obstacles, but the potentially dangerous (and unknown) outcomes of treatment complicate the decision to undergo surgery. This illustrates a paradoxical and ambivalent uncertainty dilemma between treatment-related danger and recovery-related hope that influences decision-making. This dilemma confirms that more information can increase uncertainty and compromises parents' decision-making abilities, forcing them to rely on emotional ‘gut feelings’ to manage uncertainty (Hogan & Brashers, 2009). The findings supporting orphan-illness uncertainty highlight the uncertainty associated with caring for one condition, but worrying about how the treatment influences other aspects of the child's health. The involvement of other organs complicates decision-making and prevents parents from predicting treatment outcomes and evaluating the valence of those outcomes (Brashers et al., 2003). The chain of uncertainties stemming from information uncertainty and orphan-illness uncertainty underscores the inability to isolate parental uncertainty and the presence of multiple, interrelated uncertainties (Babrow, 2001). Additionally, unlike making medical decisions about one's own healthcare, it can be more difficult for a parent to decide to pursue or withhold treatment for their child because the pain and risk factors are not their own. While the original four types of uncertainty capture how patients experience uncertainty, the topic of pain management that emerged in our findings suggests parents
experience a fourth form of uncertainty–parental proxy uncertainty (i.e., uncertainty by one who acts on behalf of their child). Parental proxy uncertainty is caused by the lack of direct illness experiences and the inability to judge pain levels to guide decision-making. This resembles the literature on couples coping with a serious illness that suggests partners of someone who is ill experience additional uncertainty because they do not know how their partner is feeling and must trust that their partner is reporting and managing pain appropriately (Goldsmith, 2009). Parental proxy uncertainty is heightened when children are too young to communicate levels of pain. As the proxy decision-maker, parents may feel compelled to control all aspects of their child's health and rely more heavily on ‘gut-feeling decisions’ than rational, educated decisions. The parental proxy uncertainty intensifies the treatment-related danger vs. recovery-related opportunity dilemma and is complicated when the fear of social stigma is involved. The stigma surrounding these visible malformations is also a concern for parents and is a prominent source of uncertainty. Social stigma uncertainty experienced by parents is a useful addition expanding the original uncertainty in illness typology (Mishel, 1988). Goffman (1963) conceptualized stigma as a mark that identifies someone as lacking social acceptance. Parents' perceptions of stigma may trigger danger appraisals of uncertainty leading them to seek as much information about treatment as possible, or even hastily decide on surgery that may be in opposition to medical advice that suggests waiting. In recent studies exploring HIV/AIDS or mental illness, stigma associated with chronic illness has become more recognized, however, the potential relationship of stigma to uncertainty has yet to be fully explored (Weiss, Ramakrishna, & Somma, 2006).
Parental Uncertainty in Illness The findings of this study confirm the original tenets of the model of illness uncertainty (Mishel, 1988), but revealed two additional areas of uncertainty that parents experience: (e) parental proxy uncertainty and (f) social stigma uncertainty. Additionally, the study confirmed that more information frequently leads to more uncertainty by increasing the number of choices and complicating expectations (Brashers, 2001). Therefore, the findings of this study provide further evidence that UMT provides a more comprehensive framework for understanding uncertainty management of parents of children with orphan illnesses than an uncertainty reduction perspective alone, which is frequently adopted in the medical environment to suggest that information-seeking will reduce unwanted uncertainty.
Practice Implications The results of our study indicate that parents frequently report wanting multiple types of information from their health care providers as a result of their uncertainty. For example, parents experiencing normalization uncertainty wanted to know whether their child could participate in activities at school, and parents experiencing parental proxy uncertainty desired information about the level of pain their child may be experiencing. However, parents' information-seeking behaviors in response to their negative appraisal of uncertainty recurrently led to more uncertainty. These results suggest that parents could be educated to expand their conception of uncertainty management and to understand that danger appraisals may be leading to premature or ineffective decisions. UMT proposes that information avoidance can be functional at times in managing chronic uncertainty (Brashers et al., 2000), however, parents in this study did not mention information avoidance. It may be that parents feel they do not have the choice to manage their uncertainty by avoiding information about their child's condition because of the social implications of appearing to be an irresponsible parent. Avoiding information could lead to a stigmatized identity as a “bad parent” for choosing to avoid information because of their own uncertainty. Thus, it is important for future research to further examine the implications of avoiding information by parents as part of the uncertainty management process. The current findings also have the potential to enlighten parents and medical practitioners about the types of uncertainty commonly experienced by parents coping with orphan illnesses and their potential impact on communication and medical decision-making.
Limitations As with every study, there are limitations to the current investigation. First, the small sample size may limit the transferability of the findings to other parents of children with complex chronic illnesses. However, the insight gained
7 by developing findings grounded in participants' lived experiences provides a deeper understanding of parents' uncertainty experiences. Additionally, the parents in this study were primarily mothers. Although mothers and fathers tend to agree on many aspects of their child's chronic health condition (Perrin, Lewkowicz, & Young, 2000), their perceptions of uncertainty and ability to cope with their child's illness may differ (Kupst & Schulman, 1988). The predominantly female patient sample in the current study may also be considered a limitation. It is common for parents of female children born with physical anomalies to express more concern about the physical appearance than parents of male children (Broder, Smith, & Strauss, 1992; Johansson & Ringsberg, 2004). Additionally, although the sample represented a wide range of both income and education levels, the parent sample on average was relatively more educated and the middle class socioeconomic status representation was the greatest (see Table 1). It is possible that education and income influence uncertainty experiences if parents have more access to diverse sources of information, but previous findings on the moderating effects of education and income on uncertainty are not consistent (Mishel, 1997). Finally, the use of self-report measures did not allow for further probing into emergent areas of uncertainty, and participant observation and note-taking are inherently biased by the researchers' perceptions. There may be parental uncertainty experiences that were missed in the data collection. Future studies should not only further investigate these areas of parental illness uncertainty using in-depth interviews to elaborate on parental uncertainty management processes, but also strive to incorporate more fathers and male patients to provide a broader understanding of uncertainty experiences.
Conclusion The results of this study suggest parents of children with chronic “orphan” illnesses experience unique uncertainty not captured by existing uncertainty theories. The themes identified in the data help further our understanding of parents' appraisals of uncertainty as a danger and how this influences their communication behaviors. The results also suggest parents rely on strategies such as emotion management (e.g., reacting to a ‘gut feeling’ of fear to make a decision), not solely information management, to cope with uncertainty (Hogan & Brashers, 2009). In the future, examining parental uncertainty and emotional management could reveal processes used by parents dealing with childrelated illness uncertainty. Additionally, the current findings should be investigated in other types of complex, multidisciplinary “orphan” illnesses, such as autoimmune diseases. Exploring these findings across multiple conditions may shed additional light on the unique uncertainty experiences of parental caregivers and improve decisionmaking with health care providers and quality of care.
8
References Babrow, A. S. (2001). Uncertainty, value, communication, and problematic integration. Journal of Communication, 51, 553–573, http://dx.doi.org/ 10.1111/j.1460-2466.2001.tb02896.x. Babrow, A. S., & Dutta-Bergman, M. J. (2003). Constructing the uncertainties of bioterror: A study of u.S. News reporting on the anthrax attack of fall, 2001. In C. B. Grant (Ed.), Rethinking communicative interaction: New interdisciplinary horizons (pp. 297–317). Amsterdam: John Benjamins. Barbour, J. B., Rintamaki, L. S., Ramsey, J. A., & Brashers, D. E. (2012). Avoiding health information. Journal of Health Communication: International Perspectives, 17, 212–229, http://dx.doi.org/10.1080/10810730.2011.585691. Binger, C. M., Albin, A., Fuerstien, R., Kushner, J., Zoger, S., & Mikkelsen, C. (1969). Childhood leukemia: Emotional impact on the family. The New England Journal of Medicine, 280, 414–418, http: //dx.doi.org/10.1056/NEJM196902202800804. Bradac, J. J. (2001). Theory comparison: Uncertainty reduction, problematic integration, uncertainty management, and other curious constructs. Journal of Communication, 51, 456–476, http://dx.doi.org/10.1111/ j.1460-2466.2001.tb02891.x. Brashers, D. E. (2001). Communication and uncertainty management. Journal of Communication, 51, 477–497, http://dx.doi.org/10.1111/j.14602466.2001.tb02892.x. Brashers, D. E., Neidig, J. L., Haas, S. M., Dobbs, L. K., Cardillo, L. W., & Russell, J. A. (2000). Communication in the management of uncertainty: The case of persons living with hiv or aids. Communication Monographs, 67, 63–84, http://dx.doi.org/10.1080/03637750009376495. Brashers, D. E., Neidig, J. L., Russell, J. A., Cardillo, L. W., Haas, S. M., Dobbs, L. K., & Nemeth, S. (2003). The medical, personal, and social causes of uncertainty in hiv illness. Issues in Mental Health Nursing, 24, 497–522, http://dx.doi.org/10.1080/01612840305292. Broder, H. L., Smith, F. B., & Strauss, R. P. (1992). Habilitation of patients with clefts: Parent and child ratings of satisfaction with appearance and speech. The Cleft Palate-Craniofacial Journal, 29, 262–267, http: //dx.doi.org/10.1597/1545-1569(1992)029b0262: HOPWCPN2.3.CO;2. Burrows, P. E., Laor, T., Paltiel, H., & Robertson, R. L. (1998). Diagnostic imaging in the evaluation of vascular birthmarks. Dermatologic Clinics, 16, 455–488, http://dx.doi.org/10.1016/S0733-8635(05)70246-1. Clarke-Steffen, L. (1993). Waiting and not knowing: The diagnosis of cancer in a child. Journal of Pediatric Oncology Nursing, 10, 146–153, http://dx.doi.org/10.1177/104345429301000405. Clatterbuck, C. W. (1978). Attributional confidence and uncertainty in initial interaction. Human Communication Research, 5, 147–157, http: //dx.doi.org/10.1111/j.1468-2958.1979.tb00630.x. Cohen, M. H. (1993). The unknown and the unknowable: Managing sustained uncertainty. Western Journal of Nursing Research, 15, 77–96, http://dx.doi.org/10.1177/019394599301500106. Dodgson, J. E., Garwick, A., Blozis, S. A., Patterson, J. M., Bennett, F. C., & Blum, R. W. (2000). Uncertainty in childhood chronic conditions and family distress in families of young children. Journal of Family Nursing, 6, 252–266. Ferrell, B. R., Grant, M., Borneman, T., Juarez, G., & ter Veer, A. (1999). Family caregiving in cancer pain management. Journal of Palliative Medicine, 2, 185–195, http://dx.doi.org/10.1089/jpm.1999.2.185. Fishman, S. J., & Mulliken, J. B. (1993). Hemangiomas and vascular malformations of infancy and childhood. Pediatric Clinics of North America, 40, 1177–1200. Glaser, B. G., & Strauss, A. L. (1967). The discovery of grounded theory: Strategies for qualitative research. Chicago: Aldine. Goffman, E. (1963). Stigma: Notes on the management of spoiled identify. Englewood Cliffs, NJ: Prentice Hall. Goldsmith, D. J. (2009). Uncertainty and communication in couples coping with serious illness. In T. D. Afifi, & W. A. Afifi (Eds.), Uncertainty, information management, and disclosure decisions (pp. 203–225). New York: Routledge.
A.M. Kerr, S.M. Haas Hoff, A. L., Mullins, L. L., Gillaspy, S. R., Page, M. C., Van Pelt, J. C., & Chaney, J. M. (2005). An intervention to decrease uncertainty and distress among parents of children newly diagnosed with diabetes: A pilot study. Families, Systems, and Health, 23, 329–342, http://dx.doi.org/10.1037/10917527.23.3.329. Hogan, T. P., & Brashers, D. E. (2009). The theory of communication and uncertainty management: Implications from the wider realm of information behavior. In T. D. Afifi, & W. A. Afifi (Eds.), Uncertainty, information management, and disclosure decisions (pp. 45–66). New York: Routledge. Johansson, B., & Ringsberg, K. C. (2004). Parents' experiences of having a child with cleft lip and palate. Journal of Advanced Nursing, 47, 165–170, http://dx.doi.org/10.1111/j.1365-2648.2004.03075.x. Kupst, M. J., & Schulman, J. L. (1988). Long-term coping with pediatric leukemia: A six-year follow-up study. Journal of Pediatric Psychology, 13, 7–22, http://dx.doi.org/10.1093/jpepsy/13.1.7. Madeo, A. C., O'Brien, K. E., Bernhardt, B. A., & Biesecker, B. B. (2012). Factors associated with perceived uncertainty among parnets of children with undiagnosed medical conditions. American Journal of Medical Genetics, 158A, 1877–1884, http://dx.doi.org/10.1002/ajmg.a.35425. Mathes, E. F. D., Haggstrom, A. N., Dowd, C., Hoffman, W. Y., & Frieden, I. (2004). Clinical characteristics and management of vascular anomalies: Findings of a multidisciplinary clinic. Archives of Dermatology, 140, 979–983, http://dx.doi.org/10.1001/archderm.140.8.979. Mishel, M. H. (1988). Uncertainty in illness. Journal of Nursing Scholarship, 20, 225–232, http://dx.doi.org/10.1111/j.1547-5069.1988.tb00082.x. Mishel, M. H. (1990). Reconceptualization of the uncertainty in illness theory.Journal of Nursing Scholarship, 22, 256–262, http://dx.doi.org/10.1111/j.15475069.1990.tb00225.x. Mishel, M. H. (1997). Uncertainty in actue illness. Annual Review of Nursing Research, 15, 57–80. Mishel, M. H. (1999). Uncertainty in chronic illness. Annual Review of Nursing Research, 17, 269–294. O'Regan, G. M., Watson, R., Orr, D., O'Donovan, D., Russell, J., Phelan, E., & Irvine, A. (2007). Management of vascular birthmarks: Review of a multidisciplinary clinic. Irish Medical Journal, 100, 425–427. Patel, R. V., & Curry, J. I. (2010). Vascular malformations. In C. K. Sinha, & M. Davenport (Eds.), Handbook of pediatric surgery (pp. 213–227). London: Springer. Perrin, E. C., Lewkowicz, C., & Young, M. H. (2000). Shared vision: Concordance among fathers, mothers, and pediatricians about unmet needs of children with chronic health conditions. Pediatrics, 105, 277–285. Richter, G. T., & Friedman, A. B. (2012). Hemangiomas and vascular malformations: Current theory and management. International Journal of Pediatrics, 2012, 1–10, http://dx.doi.org/10.1155/2012/645678. Rieu, P. N. M. A., & Festen, C. (1996). Hemangioma and vascular malformations. Pediatric Surgery International, 11, 289, http: //dx.doi.org/10.1007/BF00497794. Santacroce, S. J. (2001). Measuring parental uncertainty during the diagnosis phase of serious illness in a child. Journal of Pediatric Nursing, 16, 3–12, http://dx.doi.org/10.1053/jpdn.2001.20547. Santacroce, S. J. (2003). Parental uncertainty and posttraumatic stress in serious childhood illness. Journal of Nursing Scholarship, 35, 45–51, http://dx.doi.org/10.1111/j.1547-5069.2003.00045.x. Shannon, C. E., & Weaver, W. (1949). The mathematical theory of communication. Urbana, IL: University of Illinois Press. Sheer, V. C., & Cline, R. J. (1995). Testing a model of perceived information adequacy and uncertainty reduction in physician-patient interactions. Journal of Applied Communication Research, 23, 44–59, http://dx.doi.org/10.1080/00909889509365413. Strauss, A., & Corbin, J. M. (1990). Basics of qualitative research: Grounded theory procedures and techniques. Thousand Oaks, CA: Sage. Weiss, M. G., Ramakrishna, J., & Somma, D. (2006). Health-related stigma: Rethinking concepts and interventions. Psychology, Health, and Medicine, 11, 277–287, http://dx.doi.org/10.1080/13548500600595053.
Parental Uncertainty in Illness: Managing Uncertainty Surrounding an ‘Orphan’ Illness1 Anna M. Kerr MA a,⁎, Stephen M. Haas PhD b a
Department of Communication, University of Kentucky, Lexington, KY Department of Communication University of Cincinnati, Cincinnati, OH
b
Key words: Uncertainty management; Parental uncertainty; Healthcare decision-making; Communication
Parents of children with complex chronic illnesses experience substantial uncertainty that is heightened when the condition is an “orphan” illness not belonging to one medical specialty. The current study explores uncertainty experienced by parents of children with “orphan” illnesses requiring multidisciplinary care. Method: Participant-observations over 13 months (n = 200) were combined with questionnaire data (n = 55) to assess parental uncertainty at a multidisciplinary pediatric clinic. Results: Five unique types of uncertainty emerged from a grounded analysis ( Glaser & Strauss, 1967), revealing 11 interrelated uncertainties these parents experience. Findings can help providers understand parents' uncertainty and assist in family-centered decision-making. © 2014 Elsevier Inc. All rights reserved.
WHEN CHILDREN ARE diagnosed with chronic illnesses, parents make a radical shift from a life of takenfor-granted expectations to a life of heightened uncertainty (Cohen, 1993), lack of control, and less optimism (Madeo, O'Brien, Bernhardt, & Biesecker, 2012). One source of uncertainty is a lack of adequate information about the illness (Clarke-Steffen, 1993). For example, parents of children born with vascular anomalies (i.e., birthmarks) experience uncertainty because one consistent terminology does not exist to diagnose the condition (Rieu & Festen, 1996). Consequently, nearly 60% of vascular anomalies are misdiagnosed (Patel & Curry, 2010). Although some vascular anomalies are easily treated, some complex anomalies can be life-threatening if left untreated and can cause blood clots, limb length discrepancies, seizures, and intellectual deficiencies (Patel & Curry, 2010). Many birthmarks also affect other parts of the body, including major organs. Therefore, the complex nature of vascular 1
A previous version of this study was presented at the International Communication Association Annual Convention, Boston, MA: May 26– 30, 2011. ⁎ Corresponding author: Anna M. Kerr, MA. E-mail address: [email protected]. 0882-5963/$ – see front matter © 2014 Elsevier Inc. All rights reserved. http://dx.doi.org/10.1016/j.pedn.2014.01.008
anomalies requires coordinated care by physicians from multiple disciplines (Richter & Friedman, 2012). The need for multiple specialists classifies birthmarks as “orphan” illnesses not belonging to one medical specialty. The multifaceted nature of vascular anomalies provides an ideal context for studying parental uncertainty. The purpose of this study is to explore uncertainty experienced by parents who face unclear diagnosis and treatment options. Uncertainty exists when situations are unpredictable, often due to inconsistent or unavailable information (Brashers, 2001). Therefore, illness uncertainty stems from the struggle to categorize symptoms and information associated with one's health, inhibiting the ability to predict outcomes (Mishel, 1988). Mishel's (1988) theory of uncertainty in illness outlines four types of illness uncertainty: (a) ambiguity concerning the state of the illness, (b) complexity regarding treatment and systems of care, (c) lack of information about the diagnosis and seriousness of the illness, and (d) unpredictability of the course of the disease and prognosis. Additionally, uncertainty can simultaneously be medical, social, and personal–forcing individuals with chronic illnesses to integrate uncertainty into daily life (Brashers et al., 2003; Mishel, 1999).
2 Contrary to previous assumptions that uncertainty is a negative experience reduced by seeking information, recent research suggests more information can actually intensify uncertainty by increasing alternatives or suggesting unwanted outcomes (Brashers et al., 2000; Shannon & Weaver, 1949). Ultimately, uncertainty can only be reduced if information is adequate for decision-making purposes (Clatterbuck, 1978; Sheer & Cline, 1995). However, information that helps answer one question often introduces new questions and uncertainties (Babrow & Dutta-Bergman, 2003). This “chaining of uncertainties” suggests uncertainty cannot be isolated, but exists in relation to other uncertainties (Babrow, 2001). Thus, information seeking might not always be the most helpful strategy for managing uncertainty (Brashers, 2001). Instead, uncertainty management theory (UMT) proposes that individuals communicate in a variety of ways to manage uncertainty depending on their appraisal of uncertainty (Brashers, 2001; Brashers et al., 2000). According to uncertainty management theory (Brashers et al., 2000; Brashers, 2001), uncertainty manifests differently for different people and in different situations. Consequently, individuals may seek or avoid information to eradicate, cultivate, or maintain feelings of uncertainty depending on their appraisal. When uncertainty is appraised as a danger, individuals generally seek relevant information to clarify alternatives (Hogan & Brashers, 2009) or relieve discomfort (Bradac, 2001). If uncertainty is perceived as a way to maintain hope, seeking information can help amplify uncertainty by increasing alternatives or challenging existing beliefs (Brashers et al., 2000; Hogan & Brashers, 2009). Individuals may also avoid information to maintain uncertainty and avoid unwanted outcomes (Barbour, Rintamaki, Ramsey, & Brashers, 2012). Contrary to previous uncertainty theories, UMT posits that more information can often increase uncertainty, so managing information becomes important. Consequently, individuals with chronic illnesses must adapt to chronic uncertainty through continued reappraisals over the trajectory of their illness (Brashers et al., 2000), which results in adequately seeking and avoiding information as necessary. Adapting to chronic uncertainty is vital for parents of children with chronic illnesses. Parents' illness uncertainty stems from parents' desire to envision their child's future (Santacroce, 2001). Uncertainty is typically intensified during the diagnosis phase of the illness when parents experience uncertainty about the severity of the illness and the unpredictability of the future (Clarke-Steffen, 1993). Uncertainty also manifests in fear of the pain the child might endure and the possibility of death (Binger et al., 1969). Not surprisingly, parents with higher levels of uncertainty often report a lack of personal control, less optimism, and higher perceptions of disease severity when their child's medical condition is unknown (Madeo et al., 2012). Therefore, when parents do assign meaning to the illness,
A.M. Kerr, S.M. Haas they often do so negatively because of the perceived threat to the health of the child (Hoff et al., 2005). Therefore, the uncertainty associated with chronic illness can cause high levels of stress (Dodgson et al., 2000; Santacroce, 2003). Parents of children with chronic ‘orphan’ illnesses, such as vascular anomalies, experience uncertainty because of the complexity of care. Vascular anomalies are a challenge for physicians because each occurrence is unique (Rieu & Festen, 1996). Many physicians are willing to treat uncomplicated birthmarks, such as port-wine stains, but less willing to treat, or even diagnose, complex anomalies (Mathes, Haggstrom, Dowd, Hoffman, & Frieden, 2004). These multifaceted vascular anomalies require care from physicians from numerous disciplines to diagnose and to manage (O'Regan et al., 2007). Consequently, patients risk being misdiagnosed or mismanaged if the right physicians are not consulted (Mathes et al., 2004). The review of previous literature suggests parents experience unique uncertainty while caring for a child in the context of multidisciplinary care and in the face of inadequate information. The goal of this study is to examine and extend existing constructions of uncertainty by exploring how parents of children with vascular anomalies experience and manage uncertainty.
Methods Data for this study were collected through triangulating participant-observations, discussions with parents, and an open-ended questionnaire at a multidisciplinary vascular anomalies clinic within a large midwestern children's hospital. The study was approved by the authors' institutional review board, and all participants provided informed consent prior to study involvement. Parents were asked to participate in the study when they arrived for their child's appointment and provided informed consent at that time. One author also visited the clinic weekly over 13 months to observe over 200 physician–parent–child interactions in the exam room as well as interactions among the multi-disciplinary team members. Youth assent was attained if the child (patient) was between the ages of 11 and 17. Although the children (patients) did not complete the survey instrument, the youth assent was required for the observation of the clinic visit during which the child would be present.
Instrument Data were collected through triangulating participantobservation, discussions with parents, and an openended questionnaire developed after several months of participant-observations. Parents completed the questionnaire in the waiting or exam room before seeing
Parental Uncertainty in Illness the physicians. The questionnaire included quantitative measures, several open-ended questions, and demographic items. This study focuses on participant-observations, parent discussions, and the open-ended responses from the questionnaire to explore how parents experience uncertainty. In the initial survey, parents responded to two open-ended questions: (a) “Explain any anxiety, uncertainty, or concerns you have about your child's illness.” and (b) “How knowledgeable do you feel about your child's condition?" In line with emergent qualitative design, after more participant-observations, a third openended question was added that asked participants, (c) “Do you feel capable of explaining the actual condition, treatment options, and future progression of your child's hemangioma or vascular malformation to your family, friends, pediatrician, etc.?” This question stemmed from the recurring observation that many parents were visibly unsure about the fundamentals of their child's health condition. Several parents had trouble even locating the clinic because they were unable to pronounce their child's diagnosis or explain what specialists they were seeing at the hospital. The open-ended responses allowed the researchers to use the parents' own words to explain the experiences of uncertainty salient when managing an orphan illness. In addition to the questionnaire, the first author, who was born with a vascular anomaly, observed approximately 200 parent–child–physician interactions. Parents frequently asked the researcher questions during or after the medical interview about social and health-related issues she had experienced growing up. It was during these discussions that additional experiences of uncertainty emerged beyond those discussed during the physical examination. Throughout the 13-month participant observations, theoretic notes were taken and analyzed in conjunction with questionnaire responses to provide a more holistic analysis of parents' uncertainty.
Participants Table 1 presents an overview of the survey participant demographics. The sample consisted of 55, Englishspeaking, parents of children with vascular anomalies (46 females, 7 males) (M = 32.9 years of age, SD = 6.9, range = 19–50 years). The children (patients) included 39 females and 14 males (M = 3.9 years of age, SD = 5.3, range = newborn–17 years). This proportion is consistent with the research that indicates that the most prevalent vascular anomalies (hemangiomas) occur most frequently in White females, premature infants, and twins (Burrows, Laor, Paltiel, & Robertson, 1998; Fishman & Mulliken, 1993). Approximately 34.5% of the parents were visiting the clinic for the first time, while 60% of parents had visited at least once before (see Table 1). Similar demographics were observed in the parent–child–physician interactions.
3 Table 1 Summary of Survey Participant Demographic Variables (n = 55). Variable Gender Male Female Age Education Some high school Completed high school Some college Completed college Some graduate school Completed graduate school Income $15,000 or below $16,000–$30,000 $31,000–$50,000 $51,000–$75,000 $76,000–$100,000 $100,000 or above Clinic visit First visit Return visit 2–5 visits 6–10 visits 10 or more visits
Parents (%, n)
Children (%, n)
12.7%, (7) 83.6%, (46) M = 32.9 years
25.5% (14) 70.9% (39) M = 3.9 years
12.7% 14.5%
(7) (8)
14.5% 29.1% 5.5% 21.8%
(8) (16) (3) (12)
14.5% 10.9% 20.0% 18.2% 14.5% 20.0%
(8) (6) (11) (19) (8) (11)
34.5% 60.0% 32.7% 16.4% 10.9%
(19) (33) (18) (9) (6)
Data Analysis The researchers analyzed field notes and open-ended responses for themes through a process consistent with constant comparison analysis (Glaser & Strauss, 1967; Strauss & Corbin, 1990). Open coding was used to identify categories and sub-categories within the data. Through a process of axial coding, the categories were organized into themes that emerged inductively and in line with the authors' knowledge of previous research. The authors met regularly to organize emergent themes into sub-categories to form an accurate representation of the findings (Strauss & Corbin, 1990). Sub-categories were combined into larger super-ordinate categories based on their conceptual uncertainty-related connections until saturation occurred (Glaser & Strauss, 1967). The themes emerged from analysis across the data sources, and parents expressed similar types of uncertainty within participant-observations, discussions, and the open-ended responses providing a participant validity check for the presence of the themes across the sources of data. In addition, three scholars familiar with uncertainty, health, and family communication reviewed the emergent themes and exemplar quotes for validity. There was unanimous agreement that the themes reflected parents'
4
A.M. Kerr, S.M. Haas
descriptions of their experiences and uncertainty management.
Results The analysis of the open-ended questionnaire data and participant-observations revealed five themes capturing parents' uncertainty experiences and management: (a) normalization uncertainty, (b) information uncertainty, (c) orphan-illness uncertainty, (d) parental proxy uncertainty, and (e) social stigma uncertainty.
Normalization Uncertainty Parents expressed uncertainty about the future and were concerned about how their children's birthmarks would influence their ability to be “normal.” For example, a mother of a 4-year-old girl wrote, “I am most concerned about how my child's illness will affect her life.” The mother of a 12-year-old girl expressed: [I] Just want to be able to help my child and someone to tell me all what's going on and is this something she will have to live with rest of her life, getting her pain controlled, and getting her back to being the run of the mill child.
One mother also had concerns about having more children stating, “I am most concerned about how my child's illness will affect her life. I am also anxious about knowing exactly what her diagnosis is so I can be aware when I have more children.” Normalization uncertainty was also common during participant observations. Parents often asked about the child's ability to engage in activities at school. For example, one mother was concerned about her teenage daughter's ability to play sports at school because her birthmark that was located on the bottom of her foot. While the mother also was concerned about pain, the overall source of her concern was whether her daughter could participate in “normal” activities.
Information Uncertainty Parents expressed concerns about inadequate information regarding whether surgery was an option, how difficult surgery would be, and whether additional surgeries were needed. Because treatment options for birthmarks are not consistent, it is not always possible for physicians to give parents confident answers when asked about treatment. One mother of an 11-week old daughter expressed concern about the physicians' uncertainties about surgery. She stated, “On the first visit, it seemed that the derm[atology] resident and the surgeon were discussing the treatment option in front of me as ‘experimental’ and I wasn't sure they really felt it would work.”
A mother of a 17-week old daughter also expressed concern about surgery after receiving multiple conflicting opinions: We were [originally] referred to a pediatric surgeon. His advice was to remove [the birthmark], we opted to observe for another month, but the size continued to increase and became very painful. I was concerned it was infected, so the surgeon offered to perform surgery that next Tuesday….The radiologist strongly recommended we find a ‘clinic’ who specializes in hemangiomas/ vascular [malformations] for a second opinion. He did not agree with the surgeon to remove it the next day. The surgeon wanted to go through with the surgery, but we went on the Internet & found this clinic. That night before the scheduled surgery I was very anxious & tearful. I couldn't put my finger on it, but going ahead with surgery with a general pediatric surgeon did not ‘feel’ right. I felt we needed a second opinion from someone who specializes in this area and does this all the time....I am still anxious a bit, b/c I don't know what the [treatment] will entail, but I do feel I am at the correct place, with the right expertise to get the best care for our precious baby girl.
This mother's story illustrates the inadequate and conflicting information that parents receive in treating vascular anomalies and how that information can elicit more uncertainty. Many parents also had uncertainty about the basic risks of performing surgery. One mother of an 8-year-old son expressed, “Since I am unsure about the future, I want things taken care of now, but I am afraid of the risk of surgery,” illustrating the complex dilemma stemming from incomplete and inconsistent information about treatment and the future.
Orphan-Illness Uncertainty Some parents indicated concern about the influence of their child's birthmark on other parts of the body. For example, one mother stated, “My main concern is that she will lose her vision if the [birthmark] grows. I worry that I could let it go too long before I bring her back.” Another mother, whose daughter had a birthmark on her breast, expressed concern that the nipple would be damaged during surgery. Uncertainty about complex health risks was also prevalent during participant observations. One mother of a newborn child seeing the oncologist (one of the many physicians from the multidisciplinary team), waited until the physician had nearly left the room before blurting out “So it's not cancer, right?” Despite the conversation she had just had with an oncologist about the benign vascular anomaly, she was concerned that the birthmark had other health risks – especially cancer. The mother might not have had previous concerns about the birthmark being cancerous, but because
Parental Uncertainty in Illness she was seeing a pediatric oncologist, who traditionally cares for children with cancer, she became preoccupied with the confusing health risks of an orphan condition.
Parental Proxy Uncertainty Assessing pain and deciding when to seek care was a particularly difficult experience for parents in this study. This is common for proxy decision makers who frequently have different perceptions of the intensity of pain and the ability to control pain than the patient for which they are making medical decisions (Ferrell, Grant, Borneman, Juarez, & ter Veer, 1999). For example, a mother of a 2-year-old boy expressed how hard it is for a parent to determine when and how much pain their child is suffering: My son's lump has grown in size and I worry how big it's going to get, and I pray it doesn't hurt him. I know he doesn't like it to be touched but I don't know what it feels like every time he sits down.
The father of a 6-month-old girl was also uncertain about pain: We are concerned about her lip possibly getting worse, but we understand with [birthmarks] that many times they get bigger then subside and go away. We just want to be sure that she does not suffer any pain with ulcerations to her lip.
Parents also frequently asked the first author about the amount of pain she experienced and how her parents decided to pursue surgery. They wanted to find out from someone who had been through similar experiences if the pain was tolerable and how other parents have made ‘proxy’ decisions about surgeries for their child.
Social Stigma Uncertainty Parents frequently expressed concern about their child experiencing social stigma by being teased in school or asked about their birthmark by strangers. One mother expressed concern about the birthmark being in a prominent place on her son's body. She explained, “[His] Birthmark is in a visible place on [his] neck and [I'm] afraid that kids will make fun of him now and later on.” Another mother of a 7year old girl, who has been to the clinic more than ten times, explained how she tries to prevent potentially harmful psychological issues, but is still uncertain about stigma her child may face. She explained, “I think the not knowing what will happen with her illness is the hard part. I have tried to raise my daughter to be confident, but the questions and the teasing from other kids makes it hard.” Another mother referenced the concerns her 8-year-old son already has about his birthmark on his leg and the compression hose prescribed to him to control pain and swelling. She explained that her uncertainty came from “[t]he concerns of how his peers react
5 as he gets older to his compression hose. He doesn't want people thinking he wears ‘panty hose.’” The uncertainty about social stigma was also present during participant observations. Over the course of the 13months, several parents were visibly upset at the physicians' cautions about treatment or advice to “wait and see.” These parents frequently requested treatments or surgery to manage their child's often-concealable birthmark, against medical advice to wait, because they feared social stigma being expressed toward their child.
Discussion The current findings confirm existing literature on illness uncertainty, and highlight unique dilemmas experienced with orphan conditions. Patients with birthmarks are at a higher risk of misdiagnosis, require the care of multiple specialists, and often receive inconsistent information. These challenges illustrate the potential for more information to increase parental uncertainty (Brashers et al., 2000). Parents reported experiencing: (a) normalization uncertainty, (b) information uncertainty, (c) orphan-illness uncertainty, (d) parental proxy uncertainty, and (e) social stigma uncertainty. These interwoven uncertainties illustrate that chronic-illness uncertainty is frequently a combination of medical, social, and personal uncertainties and is multilayered and complex (Brashers et al., 2003). In Table 2, we summarize eleven explanatory principles that emerged across the five uncertainty areas. Normalization uncertainty exemplifies the conceptualization of uncertainty as unpredictability (Mishel, 1988). Parents struggled with the inability to predict a “normal” future for their child and assessing the valence of the situation. As a result, parents overwhelmingly appeared to be preoccupied with thoughts of potentially negative outcomes (danger appraisals), leading them to seek information from multiple health care providers to manage their uncertainty. These results suggest that parents are highly unlikely to appraise uncertainty about their child's future as an opportunity or believe it is acceptable to avoid information about their child's medical condition. However, as the results suggest, seeking and increasing information can lead parents to increased uncertainty (Brashers et al., 2000). The area of information uncertainty highlights three of the four established types of illness uncertainty: (a) complexity regarding treatment, (b) lack of information, and (c) unpredictability of the illness (Mishel, 1988). Parents' tendency to seek information from health care providers illustrates the role physicians play in illness uncertainty management (Brashers et al., 2000). However, physicians are not always able to provide complete information regarding the diagnosis and seriousness of the illness because consistent information about treatment for vascular anomalies has not been established. This lack, or
6
A.M. Kerr, S.M. Haas Table 2
Core Principles of Parental Uncertainty Management.
Uncertainty
Core principles
Normalization uncertainty
1. Parents experience uncertainty about the impact the condition may have on their child’s ability to be “normal” in the future. 2. Parents' thoughts of potentially negative circumstances that are unknown about the future lead to information-seeking behaviors. 1. Increased and/or inconsistent information can complicate parents' understanding of treatment options and predicting outcomes. 2. Parents face ‘ambivalent uncertainty dilemmas’ from treatment-related danger versus recovery-related opportunity appraisals. 3. Parents rely on emotional (‘gut feeling’) appraisals to manage uncertainty resulting from increased or inconsistent information. 1. Concerns about additional health risks related to multiple parts of the body complicate parents' uncertainty regarding the complex condition. 2. Multidisciplinary uncertainties create interrelated uncertainty chains. 1. Parents' uncertainty can stem from making decisions on their child's behalf. 2. Parents face uncertainty when deciding to pursue or withhold treatment due to lack of direct physical sensation and pain cues, heightened when the child is too young to communicate pain levels. 1. Parents have uncertainty from the fear of social stigma put on their child. 2. Parents' emotional appraisal of social stigma uncertainty can push parents to pursue treatments in haste or against medical advice.
Information uncertainty
Orphan-illness uncertainty
Parental proxy uncertainty
Social stigma uncertainty
inconsistency, of information does not provide parents with the cues needed to predict outcomes (Mishel, 1990). Information uncertainty from multiple medical opinions can also create a dilemma for parents. Parents want to treat their child's condition as early as possible to ensure a healthy future for their child and avoid future obstacles, but the potentially dangerous (and unknown) outcomes of treatment complicate the decision to undergo surgery. This illustrates a paradoxical and ambivalent uncertainty dilemma between treatment-related danger and recovery-related hope that influences decision-making. This dilemma confirms that more information can increase uncertainty and compromises parents' decision-making abilities, forcing them to rely on emotional ‘gut feelings’ to manage uncertainty (Hogan & Brashers, 2009). The findings supporting orphan-illness uncertainty highlight the uncertainty associated with caring for one condition, but worrying about how the treatment influences other aspects of the child's health. The involvement of other organs complicates decision-making and prevents parents from predicting treatment outcomes and evaluating the valence of those outcomes (Brashers et al., 2003). The chain of uncertainties stemming from information uncertainty and orphan-illness uncertainty underscores the inability to isolate parental uncertainty and the presence of multiple, interrelated uncertainties (Babrow, 2001). Additionally, unlike making medical decisions about one's own healthcare, it can be more difficult for a parent to decide to pursue or withhold treatment for their child because the pain and risk factors are not their own. While the original four types of uncertainty capture how patients experience uncertainty, the topic of pain management that emerged in our findings suggests parents
experience a fourth form of uncertainty–parental proxy uncertainty (i.e., uncertainty by one who acts on behalf of their child). Parental proxy uncertainty is caused by the lack of direct illness experiences and the inability to judge pain levels to guide decision-making. This resembles the literature on couples coping with a serious illness that suggests partners of someone who is ill experience additional uncertainty because they do not know how their partner is feeling and must trust that their partner is reporting and managing pain appropriately (Goldsmith, 2009). Parental proxy uncertainty is heightened when children are too young to communicate levels of pain. As the proxy decision-maker, parents may feel compelled to control all aspects of their child's health and rely more heavily on ‘gut-feeling decisions’ than rational, educated decisions. The parental proxy uncertainty intensifies the treatment-related danger vs. recovery-related opportunity dilemma and is complicated when the fear of social stigma is involved. The stigma surrounding these visible malformations is also a concern for parents and is a prominent source of uncertainty. Social stigma uncertainty experienced by parents is a useful addition expanding the original uncertainty in illness typology (Mishel, 1988). Goffman (1963) conceptualized stigma as a mark that identifies someone as lacking social acceptance. Parents' perceptions of stigma may trigger danger appraisals of uncertainty leading them to seek as much information about treatment as possible, or even hastily decide on surgery that may be in opposition to medical advice that suggests waiting. In recent studies exploring HIV/AIDS or mental illness, stigma associated with chronic illness has become more recognized, however, the potential relationship of stigma to uncertainty has yet to be fully explored (Weiss, Ramakrishna, & Somma, 2006).
Parental Uncertainty in Illness The findings of this study confirm the original tenets of the model of illness uncertainty (Mishel, 1988), but revealed two additional areas of uncertainty that parents experience: (e) parental proxy uncertainty and (f) social stigma uncertainty. Additionally, the study confirmed that more information frequently leads to more uncertainty by increasing the number of choices and complicating expectations (Brashers, 2001). Therefore, the findings of this study provide further evidence that UMT provides a more comprehensive framework for understanding uncertainty management of parents of children with orphan illnesses than an uncertainty reduction perspective alone, which is frequently adopted in the medical environment to suggest that information-seeking will reduce unwanted uncertainty.
Practice Implications The results of our study indicate that parents frequently report wanting multiple types of information from their health care providers as a result of their uncertainty. For example, parents experiencing normalization uncertainty wanted to know whether their child could participate in activities at school, and parents experiencing parental proxy uncertainty desired information about the level of pain their child may be experiencing. However, parents' information-seeking behaviors in response to their negative appraisal of uncertainty recurrently led to more uncertainty. These results suggest that parents could be educated to expand their conception of uncertainty management and to understand that danger appraisals may be leading to premature or ineffective decisions. UMT proposes that information avoidance can be functional at times in managing chronic uncertainty (Brashers et al., 2000), however, parents in this study did not mention information avoidance. It may be that parents feel they do not have the choice to manage their uncertainty by avoiding information about their child's condition because of the social implications of appearing to be an irresponsible parent. Avoiding information could lead to a stigmatized identity as a “bad parent” for choosing to avoid information because of their own uncertainty. Thus, it is important for future research to further examine the implications of avoiding information by parents as part of the uncertainty management process. The current findings also have the potential to enlighten parents and medical practitioners about the types of uncertainty commonly experienced by parents coping with orphan illnesses and their potential impact on communication and medical decision-making.
Limitations As with every study, there are limitations to the current investigation. First, the small sample size may limit the transferability of the findings to other parents of children with complex chronic illnesses. However, the insight gained
7 by developing findings grounded in participants' lived experiences provides a deeper understanding of parents' uncertainty experiences. Additionally, the parents in this study were primarily mothers. Although mothers and fathers tend to agree on many aspects of their child's chronic health condition (Perrin, Lewkowicz, & Young, 2000), their perceptions of uncertainty and ability to cope with their child's illness may differ (Kupst & Schulman, 1988). The predominantly female patient sample in the current study may also be considered a limitation. It is common for parents of female children born with physical anomalies to express more concern about the physical appearance than parents of male children (Broder, Smith, & Strauss, 1992; Johansson & Ringsberg, 2004). Additionally, although the sample represented a wide range of both income and education levels, the parent sample on average was relatively more educated and the middle class socioeconomic status representation was the greatest (see Table 1). It is possible that education and income influence uncertainty experiences if parents have more access to diverse sources of information, but previous findings on the moderating effects of education and income on uncertainty are not consistent (Mishel, 1997). Finally, the use of self-report measures did not allow for further probing into emergent areas of uncertainty, and participant observation and note-taking are inherently biased by the researchers' perceptions. There may be parental uncertainty experiences that were missed in the data collection. Future studies should not only further investigate these areas of parental illness uncertainty using in-depth interviews to elaborate on parental uncertainty management processes, but also strive to incorporate more fathers and male patients to provide a broader understanding of uncertainty experiences.
Conclusion The results of this study suggest parents of children with chronic “orphan” illnesses experience unique uncertainty not captured by existing uncertainty theories. The themes identified in the data help further our understanding of parents' appraisals of uncertainty as a danger and how this influences their communication behaviors. The results also suggest parents rely on strategies such as emotion management (e.g., reacting to a ‘gut feeling’ of fear to make a decision), not solely information management, to cope with uncertainty (Hogan & Brashers, 2009). In the future, examining parental uncertainty and emotional management could reveal processes used by parents dealing with childrelated illness uncertainty. Additionally, the current findings should be investigated in other types of complex, multidisciplinary “orphan” illnesses, such as autoimmune diseases. Exploring these findings across multiple conditions may shed additional light on the unique uncertainty experiences of parental caregivers and improve decisionmaking with health care providers and quality of care.
8
References Babrow, A. S. (2001). Uncertainty, value, communication, and problematic integration. Journal of Communication, 51, 553–573, http://dx.doi.org/ 10.1111/j.1460-2466.2001.tb02896.x. Babrow, A. S., & Dutta-Bergman, M. J. (2003). Constructing the uncertainties of bioterror: A study of u.S. News reporting on the anthrax attack of fall, 2001. In C. B. Grant (Ed.), Rethinking communicative interaction: New interdisciplinary horizons (pp. 297–317). Amsterdam: John Benjamins. Barbour, J. B., Rintamaki, L. S., Ramsey, J. A., & Brashers, D. E. (2012). Avoiding health information. Journal of Health Communication: International Perspectives, 17, 212–229, http://dx.doi.org/10.1080/10810730.2011.585691. Binger, C. M., Albin, A., Fuerstien, R., Kushner, J., Zoger, S., & Mikkelsen, C. (1969). Childhood leukemia: Emotional impact on the family. The New England Journal of Medicine, 280, 414–418, http: //dx.doi.org/10.1056/NEJM196902202800804. Bradac, J. J. (2001). Theory comparison: Uncertainty reduction, problematic integration, uncertainty management, and other curious constructs. Journal of Communication, 51, 456–476, http://dx.doi.org/10.1111/ j.1460-2466.2001.tb02891.x. Brashers, D. E. (2001). Communication and uncertainty management. Journal of Communication, 51, 477–497, http://dx.doi.org/10.1111/j.14602466.2001.tb02892.x. Brashers, D. E., Neidig, J. L., Haas, S. M., Dobbs, L. K., Cardillo, L. W., & Russell, J. A. (2000). Communication in the management of uncertainty: The case of persons living with hiv or aids. Communication Monographs, 67, 63–84, http://dx.doi.org/10.1080/03637750009376495. Brashers, D. E., Neidig, J. L., Russell, J. A., Cardillo, L. W., Haas, S. M., Dobbs, L. K., & Nemeth, S. (2003). The medical, personal, and social causes of uncertainty in hiv illness. Issues in Mental Health Nursing, 24, 497–522, http://dx.doi.org/10.1080/01612840305292. Broder, H. L., Smith, F. B., & Strauss, R. P. (1992). Habilitation of patients with clefts: Parent and child ratings of satisfaction with appearance and speech. The Cleft Palate-Craniofacial Journal, 29, 262–267, http: //dx.doi.org/10.1597/1545-1569(1992)029b0262: HOPWCPN2.3.CO;2. Burrows, P. E., Laor, T., Paltiel, H., & Robertson, R. L. (1998). Diagnostic imaging in the evaluation of vascular birthmarks. Dermatologic Clinics, 16, 455–488, http://dx.doi.org/10.1016/S0733-8635(05)70246-1. Clarke-Steffen, L. (1993). Waiting and not knowing: The diagnosis of cancer in a child. Journal of Pediatric Oncology Nursing, 10, 146–153, http://dx.doi.org/10.1177/104345429301000405. Clatterbuck, C. W. (1978). Attributional confidence and uncertainty in initial interaction. Human Communication Research, 5, 147–157, http: //dx.doi.org/10.1111/j.1468-2958.1979.tb00630.x. Cohen, M. H. (1993). The unknown and the unknowable: Managing sustained uncertainty. Western Journal of Nursing Research, 15, 77–96, http://dx.doi.org/10.1177/019394599301500106. Dodgson, J. E., Garwick, A., Blozis, S. A., Patterson, J. M., Bennett, F. C., & Blum, R. W. (2000). Uncertainty in childhood chronic conditions and family distress in families of young children. Journal of Family Nursing, 6, 252–266. Ferrell, B. R., Grant, M., Borneman, T., Juarez, G., & ter Veer, A. (1999). Family caregiving in cancer pain management. Journal of Palliative Medicine, 2, 185–195, http://dx.doi.org/10.1089/jpm.1999.2.185. Fishman, S. J., & Mulliken, J. B. (1993). Hemangiomas and vascular malformations of infancy and childhood. Pediatric Clinics of North America, 40, 1177–1200. Glaser, B. G., & Strauss, A. L. (1967). The discovery of grounded theory: Strategies for qualitative research. Chicago: Aldine. Goffman, E. (1963). Stigma: Notes on the management of spoiled identify. Englewood Cliffs, NJ: Prentice Hall. Goldsmith, D. J. (2009). Uncertainty and communication in couples coping with serious illness. In T. D. Afifi, & W. A. Afifi (Eds.), Uncertainty, information management, and disclosure decisions (pp. 203–225). New York: Routledge.
A.M. Kerr, S.M. Haas Hoff, A. L., Mullins, L. L., Gillaspy, S. R., Page, M. C., Van Pelt, J. C., & Chaney, J. M. (2005). An intervention to decrease uncertainty and distress among parents of children newly diagnosed with diabetes: A pilot study. Families, Systems, and Health, 23, 329–342, http://dx.doi.org/10.1037/10917527.23.3.329. Hogan, T. P., & Brashers, D. E. (2009). The theory of communication and uncertainty management: Implications from the wider realm of information behavior. In T. D. Afifi, & W. A. Afifi (Eds.), Uncertainty, information management, and disclosure decisions (pp. 45–66). New York: Routledge. Johansson, B., & Ringsberg, K. C. (2004). Parents' experiences of having a child with cleft lip and palate. Journal of Advanced Nursing, 47, 165–170, http://dx.doi.org/10.1111/j.1365-2648.2004.03075.x. Kupst, M. J., & Schulman, J. L. (1988). Long-term coping with pediatric leukemia: A six-year follow-up study. Journal of Pediatric Psychology, 13, 7–22, http://dx.doi.org/10.1093/jpepsy/13.1.7. Madeo, A. C., O'Brien, K. E., Bernhardt, B. A., & Biesecker, B. B. (2012). Factors associated with perceived uncertainty among parnets of children with undiagnosed medical conditions. American Journal of Medical Genetics, 158A, 1877–1884, http://dx.doi.org/10.1002/ajmg.a.35425. Mathes, E. F. D., Haggstrom, A. N., Dowd, C., Hoffman, W. Y., & Frieden, I. (2004). Clinical characteristics and management of vascular anomalies: Findings of a multidisciplinary clinic. Archives of Dermatology, 140, 979–983, http://dx.doi.org/10.1001/archderm.140.8.979. Mishel, M. H. (1988). Uncertainty in illness. Journal of Nursing Scholarship, 20, 225–232, http://dx.doi.org/10.1111/j.1547-5069.1988.tb00082.x. Mishel, M. H. (1990). Reconceptualization of the uncertainty in illness theory.Journal of Nursing Scholarship, 22, 256–262, http://dx.doi.org/10.1111/j.15475069.1990.tb00225.x. Mishel, M. H. (1997). Uncertainty in actue illness. Annual Review of Nursing Research, 15, 57–80. Mishel, M. H. (1999). Uncertainty in chronic illness. Annual Review of Nursing Research, 17, 269–294. O'Regan, G. M., Watson, R., Orr, D., O'Donovan, D., Russell, J., Phelan, E., & Irvine, A. (2007). Management of vascular birthmarks: Review of a multidisciplinary clinic. Irish Medical Journal, 100, 425–427. Patel, R. V., & Curry, J. I. (2010). Vascular malformations. In C. K. Sinha, & M. Davenport (Eds.), Handbook of pediatric surgery (pp. 213–227). London: Springer. Perrin, E. C., Lewkowicz, C., & Young, M. H. (2000). Shared vision: Concordance among fathers, mothers, and pediatricians about unmet needs of children with chronic health conditions. Pediatrics, 105, 277–285. Richter, G. T., & Friedman, A. B. (2012). Hemangiomas and vascular malformations: Current theory and management. International Journal of Pediatrics, 2012, 1–10, http://dx.doi.org/10.1155/2012/645678. Rieu, P. N. M. A., & Festen, C. (1996). Hemangioma and vascular malformations. Pediatric Surgery International, 11, 289, http: //dx.doi.org/10.1007/BF00497794. Santacroce, S. J. (2001). Measuring parental uncertainty during the diagnosis phase of serious illness in a child. Journal of Pediatric Nursing, 16, 3–12, http://dx.doi.org/10.1053/jpdn.2001.20547. Santacroce, S. J. (2003). Parental uncertainty and posttraumatic stress in serious childhood illness. Journal of Nursing Scholarship, 35, 45–51, http://dx.doi.org/10.1111/j.1547-5069.2003.00045.x. Shannon, C. E., & Weaver, W. (1949). The mathematical theory of communication. Urbana, IL: University of Illinois Press. Sheer, V. C., & Cline, R. J. (1995). Testing a model of perceived information adequacy and uncertainty reduction in physician-patient interactions. Journal of Applied Communication Research, 23, 44–59, http://dx.doi.org/10.1080/00909889509365413. Strauss, A., & Corbin, J. M. (1990). Basics of qualitative research: Grounded theory procedures and techniques. Thousand Oaks, CA: Sage. Weiss, M. G., Ramakrishna, J., & Somma, D. (2006). Health-related stigma: Rethinking concepts and interventions. Psychology, Health, and Medicine, 11, 277–287, http://dx.doi.org/10.1080/13548500600595053.
