Journal of Cystic Fibrosis 14 (2015) 540 – 546 www.elsevier.com/locate/jcf
Original Article
Breaking bad news, the diagnosis of cystic fibrosis in childhood Trudy Havermans a,⁎, Jessica Tack b , Anneke Vertommen a , Marijke Proesmans a , Kris de Boeck a a
Cystic Fibrosis Centre, Pediatric Pulmonology, University Hospital Gasthuisberg Leuven, Belgium b Psychology Department, Katholieke Universteit Leuven, Belgium Received 31 July 2014; revised 9 December 2014; accepted 10 December 2014 Available online 3 January 2015
Abstract Background: The day parents are told their child has cystic fibrosis (CF) is imprinted in their memory. Parents often show strong emotions (e.g. shock, anxiety); they need to cope with bad news and restructure their lives taking into account CF. Aims: The aims of this study are (1) to explore how parents recall circumstances of the CF diagnosis and the information they received and (2) to investigate their current coping styles. Methods: Parents (n = 38) of 20 children (diagnosed during the past 5 years) were interviewed using a semi-structured interview. Coping was assessed using the Utrecht Coping List. The association between coping and time since diagnosis/severity of illness was investigated. Results: Fifteen parents first heard the term ‘CF’ from their local pediatrician or GP. All were informed in detail by the CF specialist. All parents recalled specifics about the information, the attitude of the doctor, their thoughts and emotions. Most parents were satisfied with the content and manner in which they had received information. Nineteen appreciated the doctor showing some emotions during the talks. One couple criticized the doctor for not showing emotions. Parents reported higher use (than normative scores) of the active coping style ‘social support seeking’ and the accommodative coping styles ‘palliative reaction pattern’ and ‘comforting cognitions’. Perception of severity of illness was associated with higher scores on palliative coping. Conclusions: This study shows the importance of physicians and CF teams to tailor the way of providing bad news to parents' needs and preferences. It is important to help and encourage parents to use active or accommodative coping strategies. The diagnosis is the starting point of a long-term relationship. ‘Doing things well from the start’ helps families to learn to live with CF and treatment. © 2014 European Cystic Fibrosis Society. Published by Elsevier B.V. All rights reserved. Keywords: Bad news; Cystic fibrosis diagnosis; Parents; Communication; Coping
1. Introduction The diagnosis cystic fibrosis (CF) is, by definition, bad news: “information that changes ones' view of the future in a negative way [1]. The impact of bad news can be observed in a persons' coping behaviors and in the emotional response, both of which may persist for some time after the news is received. The present paper reports on parents hearing the diagnosis CF in their young child. It explores parent's appraisal of the ⁎ Corresponding author at: University Hospital Leuven, 49 Herestraat, B-3000 Leuven, Belgium. Tel.: +32 16 340268; fax: +32 16343817. E-mail address: [email protected] (T. Havermans).
received information, recall of their emotions and thoughts at the time of the diagnosis as well as their current coping styles. 1.1. Bad news A serious childhood disease arouses strong emotions, such as anxiety, shock, grief and feeling out of control [2–4]. The way a diagnosis is given influences these emotions and informing parents with bad news is challenging to the physician [3,5]. In CF, two studies have been conducted on how parents received the CF diagnosis [3,4]. Jedlicka-Köhler [3] reported that parents who were told about the CF diagnosis were often receiving a kind of lecture. This lecture contained more information than parents could
http://dx.doi.org/10.1016/j.jcf.2014.12.005 1569-1993© 2014 European Cystic Fibrosis Society. Published by Elsevier B.V. All rights reserved.
T. Havermans et al. / Journal of Cystic Fibrosis 14 (2015) 540–546
possibly assimilate, especially because of the incompatibility of the emotional distress and optimum learning. As a result, JedlickaKöhler [3] recommended frequent interviews with both parents and the provision of written and audiovisual materials. They also stressed that the initial discussion is only the beginning of an ongoing learning process necessary to enable long-term adjustment to a serious disease. Borowitz et al. (2009) included this recommendation in the guidelines for the management of infants with CF [6] and they are incorporated in the recent ECFS Standards of Care document [7]. 1.2. Coping with chronic illness Coping styles are the behaviors and cognitions a person uses to deal with a stressful situation [8–10]. The perception of the controllability of a stressful situation is important and three distinct coping styles have been described [11,12]: 1. active coping, where a person tries to change the stress (e.g. finding out information, learning new skills); 2. accommodative coping, where a person tries to adapt to the stress by changing his/her emotions (e.g. distraction, meditating, relaxation) or cognitions (e.g. altering goals, humor) and 3. passive coping where a person avoids the stress (e.g. relying on others, helplessness). There is ample research on coping in chronic illness, though limited in CF. An active coping style seems to reduce the overall emotional impact of a chronic illness. It is associated with a better adaptation to illness [13–17] and is positively associated with adherence [18]. Accommodative coping is also related to better adjustment, probably because it helps to adapt to the uncontrollability of a chronic illness [8–17]. Passive coping is related to poorer adjustment to chronic illness, mostly because avoidant and passive behaviors are generally associated with poorer psychological functioning (e.g. depression) [8,17]. An unclear coping strategy is ‘denial’: on one hand denial can be temporarily beneficial, for example when trying to deal with the uncontrolled stress of a chronic illness. However, on the long term denial is not helpful when it leads to non-adherence to treatment [18]. In the present study, parental experiences when hearing the CF diagnosis were further investigated in settings that aim to deliver the CF diagnosis as recommended [6,7]. First we need to note that neonatal screening is not yet available in Belgium and the information parents typically receive minimally includes the diagnosis (also explaining genetics), treatment, expected outcome, prognosis and day-to-day management. The focus of this study was on (1) how parents recall in what way the diagnosis was given, (2) how parents recall their emotions and thoughts about the diagnosis and (3) how they now perceive the sufficiency of specific items of information they received. In addition, current parental coping was examined. Differences in coping styles between mothers and fathers were investigated as well as differences between parents and a normative comparison group. Furthermore, associations between coping styles, time since diagnosis and severity of illness at diagnosis were assessed. Coping styles are relatively stable, though in principle, changeable and they differ between individuals due to learning experiences, personality traits or behavioral styles [10]. Because of the immediate stressful situation parents have to cope with, the hypothesis was
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that nearer the time of diagnosis and/or with more severe illness, parents score higher on passive coping styles and lower on active or accommodative coping styles. The results give insight into parents' experiences around the diagnosis and help to identify families that may need more help and counseling from the start. 2. Method 2.1. Participants and recruitment The study was approved by the Medical Ethical Committee of the University Hospital Leuven. From September 2012 to August 2013, 29 children with CF (diagnosed between 12 and 71 months [or 1 year and 5 years 11 months] prior to interview) were in follow-up at the CF clinic of the University Hospital Leuven. This period was chosen to keep the retrospective timespan manageable and to ensure that parental recall was drawn from a similar developmental phase, namely from infancy through early childhood. All parents were invited to take part in this study. Since two families had two children with CF, twenty-seven families were approached. In total parents of 20 children participated: four families declined because of lack of time, two families were not fluent in Flemish and one family was not approached because of severe familial problems at the time (divorce). When there was more than one child with CF, the first born was included as the reference child. For two families, only mothers participated. In total 20 mothers and 18 fathers participated (Table 1). Parents were invited to participate during outpatient clinics. After consent an appointment was made for the interview, either at the family's home or at the hospital. All but two families preferred the home situation. 3. Measures 3.1. Interview schedule A semi-structured interview was constructed based on the literature concerning bringing bad news and the impact of the diagnosis [3,5,6,19]. The semi-structured interview consists of three parts: 1) Parents' recall of how the diagnosis was communicated and circumstances and characteristics of the diagnostic process. Questions were in open-ended format, multiple choice or rating scales. Examples of questions are ‘How many times did the doctor talk to you about the diagnosis?’ and ‘Who was present at the talks?’. 2) Parents' recall of their emotions and thoughts about the diagnosis. Parents were asked to rate their emotion and thoughts on a five point rating scale (Table 2). Parents were also asked to describe their experiences in their own words. 3) A list of topics of information (Table 3) was provided and parents were asked to rate the sufficiency of the information they received on a five point Likert scale (1 = insufficient– 5 = sufficient). It is important to keep in mind that the focus is on the way parents recall their thoughts and emotions, which makes them valid
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Table 1 Sample characteristics, sources of information, and main symptoms at diagnosis and diagnostic tests (multiple response frequencies). Description
n
Fathers Mothers Male Girls Mean age child at interview Mean days between diagnosis and birth Mean months since diagnosis Recall of sources of initial diagnosis (multiple response) General practitioner/pediatrician CF specialist Nurse Gastroenterologist Geneticist Recall of symptoms leading to diagnosis (multiple response) Airway problems Gastro-intestinal Meconium ileus Failure to thrive Prenatal test Rash Hematoma after immunization Recall of diagnostic tests (multiple response) Sweat test Ultrasound Blood test/genetics Stool tests
18 20 11 9 39 months (15–71) 102 days (range 0–329) 36 months (range 14–71)
15 27 1 1 1
10 37 10 11 8 1 1 33 21 8 4
in their own way. We do not aim to assess the exact thoughts or emotions, but their recall of it at time of the interview. The open-end questions were answered by both parents together; the questions in multiple choice format and rating scales were completed separately. The interview was coded using a Table 2 Number of parents recalling emotional reactions and thoughts at the time of diagnosis. A lot/all the time
A lot/all the time
Feeling
n
Anxious Overwhelmed Relieved (that there was a diagnosis) Out of control
29 ‘How will the future be?’ 28 ‘My child will die too soon.’ 24 ‘The disease is incurable.’
34 26 25
23 ‘Why my child?’ ‘There has to be a cure!’ ‘I hope the disease progresses slowly.’
23 23 23
Never/rarely
Thought
n
Never/rarely
Feeling
n
Thought
n
Feeling indifferent Lonely Guilty Desperate Accepting Optimistic In shock Exhausted Angry
33 31 24 23 23 22 20 20 20
‘God will help us’ ‘My child is disabled.’ ‘Why was the diagnosis not made earlier?’ ‘The diagnosis must be wrong!’ ‘It is all my fault.’ ‘I have never heard of such a disease.’
36 34 32 30 25 25
Table 3 Parental ratings of sufficiency of information at diagnosis by CF related topic. Insufficient Neutral Sufficient Not (rating 1–2) (rating 3) (ratings 4–5) Applicable n (%) n (%) n (%) n (%) Nebulizer treatment (n = 38) Airway treatment (n = 38) Treatment digestive tract (n = 38) Physiotherapy (n = 37) CF antibiotics (n = 38) CF symptoms (n = 37) CF treatment (n = 32) CF genetics (n = 38) CF prognosis (n = 37) Infertility (n = 37) CF related liver problems (n = 36) Transplantation (n = 35) CF related diabetes (n = 36) Pseudomonas aeruginosa (n = 38) Osteoporosis (n = 36)
2 (5)
36 (95)
–
5 (13)
–
33 (87)
–
1 (3)
4 (11)
33 (87)
–
(5) (8) (3) (6) (5) (14) 30 (24)
3 (8) 3 (8) 6 (16) – 7 (18) 4 (11) 4 (11) 12 (32)
32 (86) 32 (84) 30 (79) 30 (94) 29 (76) 28 (76) 20 (54) 16 (42)
– – – – – – 2 (5) 1 (3)
8 (23) 15 (41)
11 (31) 7 (19)
15 (43) 14 (38)
1 (3) –
15 (39)
10 (26)
11 (29)
2 (5)
19 (50)
7 (18)
9 (24)
1 (3)
2 3 1 2 2 5 11 9
system which was pre-constructed by the researchers per individual question. For example, the question ‘From whom did you first receive the diagnosis CF?’, was coded into: 1. GP, 2. general pediatrician, 3. CF physician and 4. other. Open-end format questions were categorized based on their respective topics. 3.2. Severity of illness score To assess severity of illness, medical data concerning 1. failure to thrive, 2. stay in hospital, 3. abdominal surgery, 4. intravenous antibiotics and 5. comorbidities, were incorporated. The medical variables were coded in 1 (yes) or 0 (no) and added up (coding was done by a CF physician, taking into account nutritional status, respiratory status, need for surgery etc.). This resulted into a clinical severity score ranging from 1 (not severely ill) to 5 (severely ill). Parental recall of the severity of their child's condition at the moment of CF diagnosis was rated on a five point Likert scale (1 = not at all ill–5 = severely ill). 3.3. UCL: Utrecht Coping List Coping was assessed using the Utrecht Coping List (UCL) which is widely used in the Dutch and Flemish population in health research and is a valid and reliable coping scale [10]. Parents indicate which of the described behaviors, related to strategies of coping, applied to them on a four point rating scale ranging from 1 (rarely) to 4 (very often). The UCL consists of seven conceptual sub-scales (Table 4). Sub-scores describe an individuals' tendency to habit these seven coping strategies. Parent scores were compared to a normative sample of 1200 men and women aged between 23 and 64 years old (mean 43 years), representing all socio-economic classes [10].
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3.4. Statistical analysis Statistical analyses were performed using the program Statistical Package for the Social Science (IBM SPSS Statistics Data Editor 20). The data was described using frequencies and descriptive analysis. Multi-response analyses were used to examine multiple choice questions that allowed more than one response. Mean scores were compared using t-tests and to assess the magnitude of the effect Cohen's d was calculated [20]. An effect size of d ≤ 0.2 is considered a “small effect”, d N 0.2 and ≤ 0.8 a “medium effect” and d ≥ 0.8 is considered a “large effect”. Correlational analyses were used to assess the association between time since diagnosis, severity of illness and coping strategies. P-values ≤ 0.05 were considered statistically significant.
4. Results 4.1. The CF diagnosis Table 1 shows the sources of information during the initial talks and the main symptoms leading to the CF diagnosis. The majority of parents (n = 28) were told the CF diagnosis in a one-to-one talk; 10 parents received the term ‘CF’ by telephone. Most diagnoses were made during hospitalization (16 children), and 5 children were initially at home, but admitted soon after. Sweat test and blood test/genetic tests were recalled as diagnostic tests by most parents. Parents reported a wide variety in the length (from 15 min to one and a half hour) and number of talks about the diagnosis (1–8); all considered the duration and number of talks sufficient. One couple would have preferred more talks, mainly because they were overwhelmed with emotions during the first talk. All parents were pleased with the persons present: typically the doctor and both parents. With one family, the maternal grandparents were present. Multiple response frequencies showed that parents recalled the talks taking place in the child's hospital room (n = 26). Other places for these talks were the doctor's office (n = 17) and a consultation room (n =14). Twenty-two parents expressed that the chosen location was appropriate. Thirteen parents said that the location did not
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matter, because ‘the quality of the information is the most important’. Twenty parents reported that they had never heard of CF prior the diagnosis, four knew someone with CF and 14 parents had heard about CF, e.g. via the media. Most parents recalled searching other sources of information, such as the internet, before and between talks. All parents positively remembered being encouraged to ask questions. Two parents did not ask questions: one mother was too upset and one father prefers to listen and not talk during stressful situations. Sixteen parents said that they did not understand the medical language, but they soon learned. All but one couple had discussed the diagnosis with family and friends. Only 4 parents said they could not answer the questions of friends and family members. Nearly all parents (n = 33) stated that their grief was taken into account by the doctor. One couple described not feeling much grief during the diagnosis talks, since the information gave them a hopeful perspective. The multi-response analyses showed that parents described the doctor mainly as calm (n = 34), competent (n = 27); sensitive (n = 26) and/or precise (n = 23). Fifteen parents wanted the doctor to stay primarily unemotional; 19 appreciated the doctor showing some emotions during the talks. Only one couple criticized the doctor for not showing emotions. All parents mentioned brochures they were given and the physician using drawings to aid their information. All parents were introduced to the members of the CF team (nurse, physiotherapist, dietician, social worker and psychologist), which was valued as “it added to the care of their child”.
4.2. Emotional reactions and thoughts No significant differences were found between fathers and mothers recalling emotions and thoughts at diagnosis. The only exception was the difference on ‘feeling indifferent’, with fathers scoring higher than mothers (1.78 (sd 1.11) versus 1.05 (sd 0.22); t = 2.862, p b 0.05). Table 2 shows the number of parents recalling or not recalling emotional reactions and thoughts. Eight parents reported negative events they were still upset about. For example, one couple was unhappy about the crude way a non CF-specialist had communicated the CF diagnosis; one parent said that the diagnosis was made too late
Table 4 Description of the Utrecht Coping List (UCL) subscales by coping style. Subscales
Description
Item example
Active coping style Active problem focusing Expression of emotions Social support seeking
Trying to solve the problem or analyzing the situation. Express anger, frustration, venting stress. Seek company, comfort and understanding, ask for help.
“Working purposeful to try and solve a problem.” “Venting tension.” “Discussing the problem with friends or relatives.”
Distract attention away from the problem, relax. Sooth oneself with the thought that thing will get better or that others face difficulties as well.
“Trying to relax.” “Thinking: Every cloud has a silver lining.”
Do not intervene, avoid the situation. Ruminate, isolate, withdraw into oneself.
“Setting your worries aside by taking a break.” “Ruminating about the past.”
Accommodative coping style Palliative reaction pattern Comforting cognitions Passive coping style Avoidance Passive reaction pattern
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Table 5 Parental coping: comparison of mean subscale scores between parents and the normative sample, Cohen's effect size °, correlation with times since diagnosis and severity of illness. Coping strategy
Mean parents (SD)
Mean normative sample (SD)
t
Cohen's d°
Active coping Active problem focusing Expression of emotions Social support seeking
19.38 (3.84) 6.65 (3.77) 14.27 (3.35)
18.4 (3.6) 6.25 (1.7) 11.1 (3.0)
1.63 1.34 6.31 ⁎
Accommodative coping Palliative reaction Comforting cognitions
16.62 (4.04) 13.00 (2.72)
15.3 (3.6) 11.5 (2.6)
− 4.44 ⁎ 3.45 ⁎
Passive coping Passive reaction pattern Avoidance
11.16 (1.93) 14.05 (2.60)
10.6 (2.0) 14.7 (3.3)
1.17 − 1.19
Time since diagnosis r
Severity of illness r
0.26 0.13 0.99
0.08 0.26 − 0.20
0.11 − 0.04 0.10
0.34 0.56
0.08 0.07
− 0.28 − 0.22
− 0.09 − 0.30
0.34 ⁎ 0.01 0.15 0.13
° d ≤ 0.2 = “small effect”; 2 N d b 0.8 = “medium effect”; d ≥ 0.8 “strong effect”. ⁎ p b 0.05.
and this was still upsetting and two parents thought the talks gave too much hope. 4.3. Recall of sufficiency of the information Most parents were satisfied with the information they received (Table 3). A significant number of parents (more than 30%) rated the information about the possibility of CFRD (Cystic Fibrosis Related Diabetes), infertility, CF related osteoporosis and/or Pseudomonas aeruginosa as insufficient. 4.4. Severity of illness Parents rated their perception of the severity of illness at time of diagnosis (mean = 3.18, SD = 1.73). The clinical severity of illness score (mean 2.79, SD = 1.38) and parental rating of illness severity were significantly correlated (r = 0.35, p b 0.05). 4.5. Coping One of the parents did not complete the UCL questionnaire. ‘Social support seeking’ is the only coping strategy on which mothers and fathers differ, with mothers using this strategy more than fathers (M = 16.26 (SD = 3.62) and 12.17 (SD = 3.40) respectively; t = − 3.54, p b 0.05, Cohen's d = 1.16). Table 5 shows the comparison between parents and the normative sample. Parents reported significantly higher on ‘social support seeking’, ‘palliative reaction pattern’ and ‘comforting cognitions’ than the comparison group. Time since diagnosis was not significantly associated with coping strategy. Severity of illness was associated with higher scores on the palliative coping scale (r = 0.34, p b 0.05). 5. Discussion Jurkovich [21] concisely described the essentials of bringing bad news as follows: “The attitude of the news-giver, combined with clarity of the message and the time, privacy, and knowledge to
answer questions are the most important aspects of giving bad news.” The present study examined early parental experiences of the CF diagnosis. Similar to previous research [3,4], parents vividly recalled the way the diagnosis was given, with details on location, time and persons present. Visual information (e.g. drawings) used by the physician were remembered and considered helpful. Nearly all parents searched the internet for information. Information people find on the Internet may affect their health care decisions, health status, and mental status. For example, parents who use the internet following a positive newborn screening for CF ask more questions and feel more confident in actively participating in treatment decisions [22]. In our clinic parents are advised to be cautious when searching the internet and we encourage them to ask about the information they find. Parents recalled varying emotions and thoughts. Most common were ‘anxiety’ and ‘how will the future be?’. Few parents recalled to feel indifferent and very few recalled thinking ‘God will help us’ or ‘my child will be disabled’. The diversity in parental emotions and thoughts shows the importance of encouraging parents to regularly talk about their feelings and cognitions. It is important to gain insight into parents' perception to provide help and support tailored to their needs. Parents recalled most information sufficient, which is contrary to earlier reports were parents reported an ‘information overload’ [23]. More than 30% considered the information on CFRD, osteoporosis and Pseudomonas aeruginosa insufficient. One explanation is that the doctor paid little attention to these elements or that the doctor considered this information not essential at the time [24,25]. Another possibility is that these subjects were discussed, but parents did not recall them, since there was a lot of information to take in. During all the interviews parents stressed the importance of how the diagnosis was given. Despite large variety in timing, location and number of talks, the vast majority of parents were generally content with their own experience. The parental reports especially point to the importance of the attitude of the news-giver, and most preferred a ‘professional attitude with a personal touch’. This description is similar to views of parents
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in a pediatric intensive care unit, who want the physician to be accessible, to provide honest and complete information, with a caring affect [26]. The present results confirm the importance of customization of the information to each family and the realization of the impact of emotions on the retention of information whilst providing the information [3,6,26]. The challenge seems to be finding the right balance between ‘attitude–amount–content’ when providing information. The quality of the ‘bad-news’ interview will predominantly depend on the physician's ability to estimate preferences of the parent. Oshea [19] concluded that informing parents about their child's serious illness is one of the most unpleasant tasks for pediatricians and Ptacek [1] found that the stress to the physician after giving bad news could last up to 3 days. Physicians are at times uncertain whether families want to know everything or just part of the news and no two families will react to bad news in a similar way [24,25]. The present data confirm that there is no ‘best way’ and doctors need training in providing optimal information whilst adapting to parents' preferences and needs. Mothers and father use of coping styles was similar, though mothers used social support more often. Compared to a normative sample the parents used more social support, more distraction and more comforting thoughts. These are adequate strategies to cope with chronic illness [12,17,27]. It was hypothesized that nearer the diagnosis and/or with higher perceived illness severity parents would use more passive coping styles and fewer active or accommodative coping styles. The hypothesis was partly confirmed. No association with time since diagnosis or severity of illness was found with parents ratings of passive coping strategies, which is a promising finding, since these strategies are related to poorer adjustment to chronic illness [12,17]. But, parents who perceived their child more severely ill at the time of the diagnosis currently reported to use more accommodative strategies. These strategies probably help them to take their mind off from worrying. Parents were not asked about their current perception of the severity of their child's illness. Further research may look into fluctuations over time of parent's perception of the severity of their child's illness in association with their coping styles. Several limitations have to be reported. The number of parents participating in this study is small and three children received the diagnosis in a different, be it also a Belgian clinic. The sample cannot be seen as homogenous, also because of the range in time since diagnosis. The semi-structured interview was based on similar previous studies, but not piloted in a target group. Although no obvious problems were noted, the validity of the questions was not assessed. When interpreting the results these limitations should be taken into account. Finally, the study had to be retrospective, because asking parents about their experiences immediately after the diagnosis is difficult and unethical. But, retrospective interviewing has the disadvantage of inaccuracies [28]. It is known that memory for emotional events is typically more vivid and accurate than memory for neutral ones [29]. Similar to Jedlicka-Köhler [3] it was therefore assumed that the impression of the ‘bad news talks’ was well memorized, since the diagnosis of CF is such an emotionally salient event. The time limit of less than 5 years 11 months since diagnosis was chosen to keep the retrospective timespan manageable.
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In Belgium, newborn screening is currently under evaluation. Advantages of newborn screening are: better survival, improved growth, better nutritional status, less burden of care and avoidance of stress related to delayed diagnosis. Disadvantages of newborn screening are: anxiety in parents of infants with a false positive screening result and unwarranted detection of some asymptomatic CF carriers [30]. When newborn screening is introduced in Belgium, parental experience of the diagnosis will be different, because the diagnostic process starts at a different point in time and most children will not show serious CF symptoms (yet). Parents will be emotional and the situation will be stressful, but in the absence of symptoms parents most likely perceive their situation different. After the introduction of neonatal screening further study, including the impact of false positive screening will be necessary [22]. In conclusion, this study shows that parents recall the period around the diagnosis as full of emotions and difficult thoughts. When working with parents it is important to pay attention to these early emotions and thoughts. This study also shows that parents can be content with the course of the diagnostic and information process, despite differences in experiences and/or preferences. The attitude of the physician is important, especially his/her ability to adapt to parent's needs [23]. Finally, not long after diagnosis parents use coping strategies that are adaptive to their situation, which is a positive sign of resilience in young families. The results are preliminary and warrant further investigation, but they are an indication that ‘Doing things well from the start’ is important and may prevent long-term problems in coping with CF. Acknowledgments The authors wish to thank the parents participating in this study and the Belgian Patient Association (BVSM, CP2013/ UZLEUVEN) for funding the project. References [1] Ptacek J, Eberhardt T. The patient–physician relationship: breaking bad news—a review of the literature. JAMA 1996;276:496–502. [2] Fallowfield L, Jenkins V. Communicating sad, bad and difficult news in medicine. Lancet 2004;363:312–9. [3] Jedlicka-Köhler I, Götz M, Eichler I. Parents' recollection of the initial communication of the diagnosis of cystic fibrosis. Pediatrics 1996;97:204–9. [4] De Monestrol I, Brucefors AB, Sjöberg B, Hjelte L. Parental experiences of a clinical cystic fibrosis diagnosis and impact on siblings and relatives. http://www.dissertations.se; 2011. [5] Mack JW, Grier HE. The day one talk. J Clin Oncol 2004;22(3):563–6. [6] Borrowitz D, Robinson KA, Rosenfield M, Davis SD, Sabadosa KA, Spear SL, et al. Cystic Fibrosis Foundation evidence-based guidelines for management of infants with cystic fibrosis. J Pediatr 2009;155:S73–93. http://dx.doi.org/10.1016/j.jpeds.2009.09.001. [7] Smyth AR, Bell SC, Bojcin S, Bryon M, Duff A, Flume P, et al. European Cystic Fibrosis Society Standards of Care: best practice guidelines. J Cyst Fibros May 2014;13(Suppl. 1):S23–42. http://dx.doi.org/10.1016/ j.jcf.2014. 03.010. [8] Compas BE, Connor-Smith JK, Saltzman H, Thomsen AH, Wadsworth M. Coping with stress during childhood and adolescence: progress, problems and potential. Psychol Bull 2001;127:87–127. [9] Abbott J. Coping with cystic fibrosis. J R Soc Med 2003;96:42–50. [10] Schreurs PJG, van de Willige G, Tellegen B, Brosschot JF. De Utrechtse Coping Lijst: UCL-handleiding. Lisse Swets & Zeitlinger; 1988.
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[11] Weiten W, Lloyd MA. Psychology applied to modern life. 9th ed. Wadsworth Cengage Learning; 2008. [12] Carver CS, Connor-Smith J. Personality and coping. Annu Rev Psychol 2010;61:679–704. http://dx.doi.org/10.1146/ annurev.psych.093008.100352 (Review). [13] Marin TJ, Chen E, Munch T, Miller G. Double exposure to acute stress and chronic family stress is associated with immune changes in children with asthma. Psychosom Med 2009;71:378–84. http://dx.doi.org/10.1097/ PSY.0b013e318199dbc3. [14] Wong MG, Heriot SA. Parents of children with CF: how they hope, cope and despair. Child Care Health 2008;34(4):344–54. http://dx.doi.org/10. 1111/j.1365-2214.2007.00804.x. [15] Sheehan J, Hisock H, Massie J, Jaffe A, Hay M. Caregiver coping, mental health and child problem behavior in cystic fibrosis: a cross-sectional study. Int Soc Behav Med 2014;21(2):211–20. http://dx.doi.org/10.1007/s12529-0139289-y. [16] Hodkova P, Abbott J, Mala I, Chladova H. Coping with cystic fibrosis—CF adults and parents of a child with CF in Czech Republic. J Cyst Fibros 2008;7(2):110. [17] Compas BE, Jaser SS, Dunn MJ, Rodriguez EM. Coping with chronic illness in childhood and adolescence. Annu Rev Clin Psychol 2012;8: 455–80. http://dx.doi.org/10.1146/annurev-clinpsy-032511-143108. [18] Abbott J, Dodd M, Gee L, Webb K. Ways of coping with cystic fibrosis: implications for treatment adherence. Disabil Rehabil May 2001;23(8):315–24. [19] Oshea J, Smith O, O'Marcaigh A, McMahon C, Geoghegan R, Cotter M. Breaking bad news: parents' experience of learning that their child has leukemia. Ir Med J 2007;100(9):588–90. [20] Cohen J. Statistical power analysis for the behavioral sciences. 2nd ed. Hillsdale NJ: Lawrence Earlbaum Associates; 1988. [21] Jurkovich GJ, Pierce B, Pananen L, Rivara FP. Giving bad news: the family perspective. J Trauma Inj Infect Crit Care May 2000;48(5):865–73.
[22] Price Dillard J, Shen Lijiang, Robinson Jeffrey D, Farrell Phillip M. Parental information seeking following a positive newborn screening for cystic fibrosis. J Health Commun Int Perspect 2010;15(8):880–94. [23] Hummelinck A, Pollock K. Parents' information needs about the treatment of their chronically ill child: a qualitative study. Patient Educ Couns 2006; 62:228–34. [24] Rosenbaum ME, Ferguson KJ, Lobas JG. Teaching medical students and residents skills for delivering bad news: a review of strategies. Acad Med 2004;79(2):107–17. [25] Ptacek JT, McIntosh EGJ. Physician challenges in communicating bad news. Behav Med 2009;32(4):380–7. http://dx.doi.org/10.1007/s10865009-9213-8. [26] Meert KL, Eggly S, Pollack M, Anand KJ, Zimmerman J, Carcillo J, et al. Parents' perspectives on physician–parent communication near the time of a child's death in the pediatric intensive care unit. Pediatr Crit Care Med 2004;9(1):2–7. [27] Tluczek A, Koscik RL, Modaff P, Pfeil D, Rock MJ, Farrell PM, et al. Newborn screening for cystic fibrosis: parents' preferences regarding counseling at the time of infants' sweat test. J Genet Couns 2006;15(4):277–91. [28] Henry B, Moffitt TE, Caspi A, Langley J, Silva PA. On the “remembrance of things past”: a longitudinal evaluation of the retrospective method. Psychol Assess 1994;6:92–101. [29] Todd RM, Talmi D, Schmitz TW, Susskind J, Anderson AK. Psychophysical and neural evidence for emotion-enhanced perceptual vividness. J Neurosci 2012;32:11201–12. http://dx.doi.org/10.1523/ JNEUROSCI. 0155-12.2012. [30] Castellani C, Southern KW, Brownlee K, Dankert Roelse J, Duff A, Farrell M, et al. European best practice guidelines for cystic fibrosis neonatal screening. J Cyst Fibros 2009;8(3):153–73. http://dx.doi.org/10. 1016/j.jcf.2009.01.004.
Original Article
Breaking bad news, the diagnosis of cystic fibrosis in childhood Trudy Havermans a,⁎, Jessica Tack b , Anneke Vertommen a , Marijke Proesmans a , Kris de Boeck a a
Cystic Fibrosis Centre, Pediatric Pulmonology, University Hospital Gasthuisberg Leuven, Belgium b Psychology Department, Katholieke Universteit Leuven, Belgium Received 31 July 2014; revised 9 December 2014; accepted 10 December 2014 Available online 3 January 2015
Abstract Background: The day parents are told their child has cystic fibrosis (CF) is imprinted in their memory. Parents often show strong emotions (e.g. shock, anxiety); they need to cope with bad news and restructure their lives taking into account CF. Aims: The aims of this study are (1) to explore how parents recall circumstances of the CF diagnosis and the information they received and (2) to investigate their current coping styles. Methods: Parents (n = 38) of 20 children (diagnosed during the past 5 years) were interviewed using a semi-structured interview. Coping was assessed using the Utrecht Coping List. The association between coping and time since diagnosis/severity of illness was investigated. Results: Fifteen parents first heard the term ‘CF’ from their local pediatrician or GP. All were informed in detail by the CF specialist. All parents recalled specifics about the information, the attitude of the doctor, their thoughts and emotions. Most parents were satisfied with the content and manner in which they had received information. Nineteen appreciated the doctor showing some emotions during the talks. One couple criticized the doctor for not showing emotions. Parents reported higher use (than normative scores) of the active coping style ‘social support seeking’ and the accommodative coping styles ‘palliative reaction pattern’ and ‘comforting cognitions’. Perception of severity of illness was associated with higher scores on palliative coping. Conclusions: This study shows the importance of physicians and CF teams to tailor the way of providing bad news to parents' needs and preferences. It is important to help and encourage parents to use active or accommodative coping strategies. The diagnosis is the starting point of a long-term relationship. ‘Doing things well from the start’ helps families to learn to live with CF and treatment. © 2014 European Cystic Fibrosis Society. Published by Elsevier B.V. All rights reserved. Keywords: Bad news; Cystic fibrosis diagnosis; Parents; Communication; Coping
1. Introduction The diagnosis cystic fibrosis (CF) is, by definition, bad news: “information that changes ones' view of the future in a negative way [1]. The impact of bad news can be observed in a persons' coping behaviors and in the emotional response, both of which may persist for some time after the news is received. The present paper reports on parents hearing the diagnosis CF in their young child. It explores parent's appraisal of the ⁎ Corresponding author at: University Hospital Leuven, 49 Herestraat, B-3000 Leuven, Belgium. Tel.: +32 16 340268; fax: +32 16343817. E-mail address: [email protected] (T. Havermans).
received information, recall of their emotions and thoughts at the time of the diagnosis as well as their current coping styles. 1.1. Bad news A serious childhood disease arouses strong emotions, such as anxiety, shock, grief and feeling out of control [2–4]. The way a diagnosis is given influences these emotions and informing parents with bad news is challenging to the physician [3,5]. In CF, two studies have been conducted on how parents received the CF diagnosis [3,4]. Jedlicka-Köhler [3] reported that parents who were told about the CF diagnosis were often receiving a kind of lecture. This lecture contained more information than parents could
http://dx.doi.org/10.1016/j.jcf.2014.12.005 1569-1993© 2014 European Cystic Fibrosis Society. Published by Elsevier B.V. All rights reserved.
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possibly assimilate, especially because of the incompatibility of the emotional distress and optimum learning. As a result, JedlickaKöhler [3] recommended frequent interviews with both parents and the provision of written and audiovisual materials. They also stressed that the initial discussion is only the beginning of an ongoing learning process necessary to enable long-term adjustment to a serious disease. Borowitz et al. (2009) included this recommendation in the guidelines for the management of infants with CF [6] and they are incorporated in the recent ECFS Standards of Care document [7]. 1.2. Coping with chronic illness Coping styles are the behaviors and cognitions a person uses to deal with a stressful situation [8–10]. The perception of the controllability of a stressful situation is important and three distinct coping styles have been described [11,12]: 1. active coping, where a person tries to change the stress (e.g. finding out information, learning new skills); 2. accommodative coping, where a person tries to adapt to the stress by changing his/her emotions (e.g. distraction, meditating, relaxation) or cognitions (e.g. altering goals, humor) and 3. passive coping where a person avoids the stress (e.g. relying on others, helplessness). There is ample research on coping in chronic illness, though limited in CF. An active coping style seems to reduce the overall emotional impact of a chronic illness. It is associated with a better adaptation to illness [13–17] and is positively associated with adherence [18]. Accommodative coping is also related to better adjustment, probably because it helps to adapt to the uncontrollability of a chronic illness [8–17]. Passive coping is related to poorer adjustment to chronic illness, mostly because avoidant and passive behaviors are generally associated with poorer psychological functioning (e.g. depression) [8,17]. An unclear coping strategy is ‘denial’: on one hand denial can be temporarily beneficial, for example when trying to deal with the uncontrolled stress of a chronic illness. However, on the long term denial is not helpful when it leads to non-adherence to treatment [18]. In the present study, parental experiences when hearing the CF diagnosis were further investigated in settings that aim to deliver the CF diagnosis as recommended [6,7]. First we need to note that neonatal screening is not yet available in Belgium and the information parents typically receive minimally includes the diagnosis (also explaining genetics), treatment, expected outcome, prognosis and day-to-day management. The focus of this study was on (1) how parents recall in what way the diagnosis was given, (2) how parents recall their emotions and thoughts about the diagnosis and (3) how they now perceive the sufficiency of specific items of information they received. In addition, current parental coping was examined. Differences in coping styles between mothers and fathers were investigated as well as differences between parents and a normative comparison group. Furthermore, associations between coping styles, time since diagnosis and severity of illness at diagnosis were assessed. Coping styles are relatively stable, though in principle, changeable and they differ between individuals due to learning experiences, personality traits or behavioral styles [10]. Because of the immediate stressful situation parents have to cope with, the hypothesis was
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that nearer the time of diagnosis and/or with more severe illness, parents score higher on passive coping styles and lower on active or accommodative coping styles. The results give insight into parents' experiences around the diagnosis and help to identify families that may need more help and counseling from the start. 2. Method 2.1. Participants and recruitment The study was approved by the Medical Ethical Committee of the University Hospital Leuven. From September 2012 to August 2013, 29 children with CF (diagnosed between 12 and 71 months [or 1 year and 5 years 11 months] prior to interview) were in follow-up at the CF clinic of the University Hospital Leuven. This period was chosen to keep the retrospective timespan manageable and to ensure that parental recall was drawn from a similar developmental phase, namely from infancy through early childhood. All parents were invited to take part in this study. Since two families had two children with CF, twenty-seven families were approached. In total parents of 20 children participated: four families declined because of lack of time, two families were not fluent in Flemish and one family was not approached because of severe familial problems at the time (divorce). When there was more than one child with CF, the first born was included as the reference child. For two families, only mothers participated. In total 20 mothers and 18 fathers participated (Table 1). Parents were invited to participate during outpatient clinics. After consent an appointment was made for the interview, either at the family's home or at the hospital. All but two families preferred the home situation. 3. Measures 3.1. Interview schedule A semi-structured interview was constructed based on the literature concerning bringing bad news and the impact of the diagnosis [3,5,6,19]. The semi-structured interview consists of three parts: 1) Parents' recall of how the diagnosis was communicated and circumstances and characteristics of the diagnostic process. Questions were in open-ended format, multiple choice or rating scales. Examples of questions are ‘How many times did the doctor talk to you about the diagnosis?’ and ‘Who was present at the talks?’. 2) Parents' recall of their emotions and thoughts about the diagnosis. Parents were asked to rate their emotion and thoughts on a five point rating scale (Table 2). Parents were also asked to describe their experiences in their own words. 3) A list of topics of information (Table 3) was provided and parents were asked to rate the sufficiency of the information they received on a five point Likert scale (1 = insufficient– 5 = sufficient). It is important to keep in mind that the focus is on the way parents recall their thoughts and emotions, which makes them valid
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Table 1 Sample characteristics, sources of information, and main symptoms at diagnosis and diagnostic tests (multiple response frequencies). Description
n
Fathers Mothers Male Girls Mean age child at interview Mean days between diagnosis and birth Mean months since diagnosis Recall of sources of initial diagnosis (multiple response) General practitioner/pediatrician CF specialist Nurse Gastroenterologist Geneticist Recall of symptoms leading to diagnosis (multiple response) Airway problems Gastro-intestinal Meconium ileus Failure to thrive Prenatal test Rash Hematoma after immunization Recall of diagnostic tests (multiple response) Sweat test Ultrasound Blood test/genetics Stool tests
18 20 11 9 39 months (15–71) 102 days (range 0–329) 36 months (range 14–71)
15 27 1 1 1
10 37 10 11 8 1 1 33 21 8 4
in their own way. We do not aim to assess the exact thoughts or emotions, but their recall of it at time of the interview. The open-end questions were answered by both parents together; the questions in multiple choice format and rating scales were completed separately. The interview was coded using a Table 2 Number of parents recalling emotional reactions and thoughts at the time of diagnosis. A lot/all the time
A lot/all the time
Feeling
n
Anxious Overwhelmed Relieved (that there was a diagnosis) Out of control
29 ‘How will the future be?’ 28 ‘My child will die too soon.’ 24 ‘The disease is incurable.’
34 26 25
23 ‘Why my child?’ ‘There has to be a cure!’ ‘I hope the disease progresses slowly.’
23 23 23
Never/rarely
Thought
n
Never/rarely
Feeling
n
Thought
n
Feeling indifferent Lonely Guilty Desperate Accepting Optimistic In shock Exhausted Angry
33 31 24 23 23 22 20 20 20
‘God will help us’ ‘My child is disabled.’ ‘Why was the diagnosis not made earlier?’ ‘The diagnosis must be wrong!’ ‘It is all my fault.’ ‘I have never heard of such a disease.’
36 34 32 30 25 25
Table 3 Parental ratings of sufficiency of information at diagnosis by CF related topic. Insufficient Neutral Sufficient Not (rating 1–2) (rating 3) (ratings 4–5) Applicable n (%) n (%) n (%) n (%) Nebulizer treatment (n = 38) Airway treatment (n = 38) Treatment digestive tract (n = 38) Physiotherapy (n = 37) CF antibiotics (n = 38) CF symptoms (n = 37) CF treatment (n = 32) CF genetics (n = 38) CF prognosis (n = 37) Infertility (n = 37) CF related liver problems (n = 36) Transplantation (n = 35) CF related diabetes (n = 36) Pseudomonas aeruginosa (n = 38) Osteoporosis (n = 36)
2 (5)
36 (95)
–
5 (13)
–
33 (87)
–
1 (3)
4 (11)
33 (87)
–
(5) (8) (3) (6) (5) (14) 30 (24)
3 (8) 3 (8) 6 (16) – 7 (18) 4 (11) 4 (11) 12 (32)
32 (86) 32 (84) 30 (79) 30 (94) 29 (76) 28 (76) 20 (54) 16 (42)
– – – – – – 2 (5) 1 (3)
8 (23) 15 (41)
11 (31) 7 (19)
15 (43) 14 (38)
1 (3) –
15 (39)
10 (26)
11 (29)
2 (5)
19 (50)
7 (18)
9 (24)
1 (3)
2 3 1 2 2 5 11 9
system which was pre-constructed by the researchers per individual question. For example, the question ‘From whom did you first receive the diagnosis CF?’, was coded into: 1. GP, 2. general pediatrician, 3. CF physician and 4. other. Open-end format questions were categorized based on their respective topics. 3.2. Severity of illness score To assess severity of illness, medical data concerning 1. failure to thrive, 2. stay in hospital, 3. abdominal surgery, 4. intravenous antibiotics and 5. comorbidities, were incorporated. The medical variables were coded in 1 (yes) or 0 (no) and added up (coding was done by a CF physician, taking into account nutritional status, respiratory status, need for surgery etc.). This resulted into a clinical severity score ranging from 1 (not severely ill) to 5 (severely ill). Parental recall of the severity of their child's condition at the moment of CF diagnosis was rated on a five point Likert scale (1 = not at all ill–5 = severely ill). 3.3. UCL: Utrecht Coping List Coping was assessed using the Utrecht Coping List (UCL) which is widely used in the Dutch and Flemish population in health research and is a valid and reliable coping scale [10]. Parents indicate which of the described behaviors, related to strategies of coping, applied to them on a four point rating scale ranging from 1 (rarely) to 4 (very often). The UCL consists of seven conceptual sub-scales (Table 4). Sub-scores describe an individuals' tendency to habit these seven coping strategies. Parent scores were compared to a normative sample of 1200 men and women aged between 23 and 64 years old (mean 43 years), representing all socio-economic classes [10].
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3.4. Statistical analysis Statistical analyses were performed using the program Statistical Package for the Social Science (IBM SPSS Statistics Data Editor 20). The data was described using frequencies and descriptive analysis. Multi-response analyses were used to examine multiple choice questions that allowed more than one response. Mean scores were compared using t-tests and to assess the magnitude of the effect Cohen's d was calculated [20]. An effect size of d ≤ 0.2 is considered a “small effect”, d N 0.2 and ≤ 0.8 a “medium effect” and d ≥ 0.8 is considered a “large effect”. Correlational analyses were used to assess the association between time since diagnosis, severity of illness and coping strategies. P-values ≤ 0.05 were considered statistically significant.
4. Results 4.1. The CF diagnosis Table 1 shows the sources of information during the initial talks and the main symptoms leading to the CF diagnosis. The majority of parents (n = 28) were told the CF diagnosis in a one-to-one talk; 10 parents received the term ‘CF’ by telephone. Most diagnoses were made during hospitalization (16 children), and 5 children were initially at home, but admitted soon after. Sweat test and blood test/genetic tests were recalled as diagnostic tests by most parents. Parents reported a wide variety in the length (from 15 min to one and a half hour) and number of talks about the diagnosis (1–8); all considered the duration and number of talks sufficient. One couple would have preferred more talks, mainly because they were overwhelmed with emotions during the first talk. All parents were pleased with the persons present: typically the doctor and both parents. With one family, the maternal grandparents were present. Multiple response frequencies showed that parents recalled the talks taking place in the child's hospital room (n = 26). Other places for these talks were the doctor's office (n = 17) and a consultation room (n =14). Twenty-two parents expressed that the chosen location was appropriate. Thirteen parents said that the location did not
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matter, because ‘the quality of the information is the most important’. Twenty parents reported that they had never heard of CF prior the diagnosis, four knew someone with CF and 14 parents had heard about CF, e.g. via the media. Most parents recalled searching other sources of information, such as the internet, before and between talks. All parents positively remembered being encouraged to ask questions. Two parents did not ask questions: one mother was too upset and one father prefers to listen and not talk during stressful situations. Sixteen parents said that they did not understand the medical language, but they soon learned. All but one couple had discussed the diagnosis with family and friends. Only 4 parents said they could not answer the questions of friends and family members. Nearly all parents (n = 33) stated that their grief was taken into account by the doctor. One couple described not feeling much grief during the diagnosis talks, since the information gave them a hopeful perspective. The multi-response analyses showed that parents described the doctor mainly as calm (n = 34), competent (n = 27); sensitive (n = 26) and/or precise (n = 23). Fifteen parents wanted the doctor to stay primarily unemotional; 19 appreciated the doctor showing some emotions during the talks. Only one couple criticized the doctor for not showing emotions. All parents mentioned brochures they were given and the physician using drawings to aid their information. All parents were introduced to the members of the CF team (nurse, physiotherapist, dietician, social worker and psychologist), which was valued as “it added to the care of their child”.
4.2. Emotional reactions and thoughts No significant differences were found between fathers and mothers recalling emotions and thoughts at diagnosis. The only exception was the difference on ‘feeling indifferent’, with fathers scoring higher than mothers (1.78 (sd 1.11) versus 1.05 (sd 0.22); t = 2.862, p b 0.05). Table 2 shows the number of parents recalling or not recalling emotional reactions and thoughts. Eight parents reported negative events they were still upset about. For example, one couple was unhappy about the crude way a non CF-specialist had communicated the CF diagnosis; one parent said that the diagnosis was made too late
Table 4 Description of the Utrecht Coping List (UCL) subscales by coping style. Subscales
Description
Item example
Active coping style Active problem focusing Expression of emotions Social support seeking
Trying to solve the problem or analyzing the situation. Express anger, frustration, venting stress. Seek company, comfort and understanding, ask for help.
“Working purposeful to try and solve a problem.” “Venting tension.” “Discussing the problem with friends or relatives.”
Distract attention away from the problem, relax. Sooth oneself with the thought that thing will get better or that others face difficulties as well.
“Trying to relax.” “Thinking: Every cloud has a silver lining.”
Do not intervene, avoid the situation. Ruminate, isolate, withdraw into oneself.
“Setting your worries aside by taking a break.” “Ruminating about the past.”
Accommodative coping style Palliative reaction pattern Comforting cognitions Passive coping style Avoidance Passive reaction pattern
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Table 5 Parental coping: comparison of mean subscale scores between parents and the normative sample, Cohen's effect size °, correlation with times since diagnosis and severity of illness. Coping strategy
Mean parents (SD)
Mean normative sample (SD)
t
Cohen's d°
Active coping Active problem focusing Expression of emotions Social support seeking
19.38 (3.84) 6.65 (3.77) 14.27 (3.35)
18.4 (3.6) 6.25 (1.7) 11.1 (3.0)
1.63 1.34 6.31 ⁎
Accommodative coping Palliative reaction Comforting cognitions
16.62 (4.04) 13.00 (2.72)
15.3 (3.6) 11.5 (2.6)
− 4.44 ⁎ 3.45 ⁎
Passive coping Passive reaction pattern Avoidance
11.16 (1.93) 14.05 (2.60)
10.6 (2.0) 14.7 (3.3)
1.17 − 1.19
Time since diagnosis r
Severity of illness r
0.26 0.13 0.99
0.08 0.26 − 0.20
0.11 − 0.04 0.10
0.34 0.56
0.08 0.07
− 0.28 − 0.22
− 0.09 − 0.30
0.34 ⁎ 0.01 0.15 0.13
° d ≤ 0.2 = “small effect”; 2 N d b 0.8 = “medium effect”; d ≥ 0.8 “strong effect”. ⁎ p b 0.05.
and this was still upsetting and two parents thought the talks gave too much hope. 4.3. Recall of sufficiency of the information Most parents were satisfied with the information they received (Table 3). A significant number of parents (more than 30%) rated the information about the possibility of CFRD (Cystic Fibrosis Related Diabetes), infertility, CF related osteoporosis and/or Pseudomonas aeruginosa as insufficient. 4.4. Severity of illness Parents rated their perception of the severity of illness at time of diagnosis (mean = 3.18, SD = 1.73). The clinical severity of illness score (mean 2.79, SD = 1.38) and parental rating of illness severity were significantly correlated (r = 0.35, p b 0.05). 4.5. Coping One of the parents did not complete the UCL questionnaire. ‘Social support seeking’ is the only coping strategy on which mothers and fathers differ, with mothers using this strategy more than fathers (M = 16.26 (SD = 3.62) and 12.17 (SD = 3.40) respectively; t = − 3.54, p b 0.05, Cohen's d = 1.16). Table 5 shows the comparison between parents and the normative sample. Parents reported significantly higher on ‘social support seeking’, ‘palliative reaction pattern’ and ‘comforting cognitions’ than the comparison group. Time since diagnosis was not significantly associated with coping strategy. Severity of illness was associated with higher scores on the palliative coping scale (r = 0.34, p b 0.05). 5. Discussion Jurkovich [21] concisely described the essentials of bringing bad news as follows: “The attitude of the news-giver, combined with clarity of the message and the time, privacy, and knowledge to
answer questions are the most important aspects of giving bad news.” The present study examined early parental experiences of the CF diagnosis. Similar to previous research [3,4], parents vividly recalled the way the diagnosis was given, with details on location, time and persons present. Visual information (e.g. drawings) used by the physician were remembered and considered helpful. Nearly all parents searched the internet for information. Information people find on the Internet may affect their health care decisions, health status, and mental status. For example, parents who use the internet following a positive newborn screening for CF ask more questions and feel more confident in actively participating in treatment decisions [22]. In our clinic parents are advised to be cautious when searching the internet and we encourage them to ask about the information they find. Parents recalled varying emotions and thoughts. Most common were ‘anxiety’ and ‘how will the future be?’. Few parents recalled to feel indifferent and very few recalled thinking ‘God will help us’ or ‘my child will be disabled’. The diversity in parental emotions and thoughts shows the importance of encouraging parents to regularly talk about their feelings and cognitions. It is important to gain insight into parents' perception to provide help and support tailored to their needs. Parents recalled most information sufficient, which is contrary to earlier reports were parents reported an ‘information overload’ [23]. More than 30% considered the information on CFRD, osteoporosis and Pseudomonas aeruginosa insufficient. One explanation is that the doctor paid little attention to these elements or that the doctor considered this information not essential at the time [24,25]. Another possibility is that these subjects were discussed, but parents did not recall them, since there was a lot of information to take in. During all the interviews parents stressed the importance of how the diagnosis was given. Despite large variety in timing, location and number of talks, the vast majority of parents were generally content with their own experience. The parental reports especially point to the importance of the attitude of the news-giver, and most preferred a ‘professional attitude with a personal touch’. This description is similar to views of parents
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in a pediatric intensive care unit, who want the physician to be accessible, to provide honest and complete information, with a caring affect [26]. The present results confirm the importance of customization of the information to each family and the realization of the impact of emotions on the retention of information whilst providing the information [3,6,26]. The challenge seems to be finding the right balance between ‘attitude–amount–content’ when providing information. The quality of the ‘bad-news’ interview will predominantly depend on the physician's ability to estimate preferences of the parent. Oshea [19] concluded that informing parents about their child's serious illness is one of the most unpleasant tasks for pediatricians and Ptacek [1] found that the stress to the physician after giving bad news could last up to 3 days. Physicians are at times uncertain whether families want to know everything or just part of the news and no two families will react to bad news in a similar way [24,25]. The present data confirm that there is no ‘best way’ and doctors need training in providing optimal information whilst adapting to parents' preferences and needs. Mothers and father use of coping styles was similar, though mothers used social support more often. Compared to a normative sample the parents used more social support, more distraction and more comforting thoughts. These are adequate strategies to cope with chronic illness [12,17,27]. It was hypothesized that nearer the diagnosis and/or with higher perceived illness severity parents would use more passive coping styles and fewer active or accommodative coping styles. The hypothesis was partly confirmed. No association with time since diagnosis or severity of illness was found with parents ratings of passive coping strategies, which is a promising finding, since these strategies are related to poorer adjustment to chronic illness [12,17]. But, parents who perceived their child more severely ill at the time of the diagnosis currently reported to use more accommodative strategies. These strategies probably help them to take their mind off from worrying. Parents were not asked about their current perception of the severity of their child's illness. Further research may look into fluctuations over time of parent's perception of the severity of their child's illness in association with their coping styles. Several limitations have to be reported. The number of parents participating in this study is small and three children received the diagnosis in a different, be it also a Belgian clinic. The sample cannot be seen as homogenous, also because of the range in time since diagnosis. The semi-structured interview was based on similar previous studies, but not piloted in a target group. Although no obvious problems were noted, the validity of the questions was not assessed. When interpreting the results these limitations should be taken into account. Finally, the study had to be retrospective, because asking parents about their experiences immediately after the diagnosis is difficult and unethical. But, retrospective interviewing has the disadvantage of inaccuracies [28]. It is known that memory for emotional events is typically more vivid and accurate than memory for neutral ones [29]. Similar to Jedlicka-Köhler [3] it was therefore assumed that the impression of the ‘bad news talks’ was well memorized, since the diagnosis of CF is such an emotionally salient event. The time limit of less than 5 years 11 months since diagnosis was chosen to keep the retrospective timespan manageable.
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In Belgium, newborn screening is currently under evaluation. Advantages of newborn screening are: better survival, improved growth, better nutritional status, less burden of care and avoidance of stress related to delayed diagnosis. Disadvantages of newborn screening are: anxiety in parents of infants with a false positive screening result and unwarranted detection of some asymptomatic CF carriers [30]. When newborn screening is introduced in Belgium, parental experience of the diagnosis will be different, because the diagnostic process starts at a different point in time and most children will not show serious CF symptoms (yet). Parents will be emotional and the situation will be stressful, but in the absence of symptoms parents most likely perceive their situation different. After the introduction of neonatal screening further study, including the impact of false positive screening will be necessary [22]. In conclusion, this study shows that parents recall the period around the diagnosis as full of emotions and difficult thoughts. When working with parents it is important to pay attention to these early emotions and thoughts. This study also shows that parents can be content with the course of the diagnostic and information process, despite differences in experiences and/or preferences. The attitude of the physician is important, especially his/her ability to adapt to parent's needs [23]. Finally, not long after diagnosis parents use coping strategies that are adaptive to their situation, which is a positive sign of resilience in young families. The results are preliminary and warrant further investigation, but they are an indication that ‘Doing things well from the start’ is important and may prevent long-term problems in coping with CF. Acknowledgments The authors wish to thank the parents participating in this study and the Belgian Patient Association (BVSM, CP2013/ UZLEUVEN) for funding the project. References [1] Ptacek J, Eberhardt T. The patient–physician relationship: breaking bad news—a review of the literature. JAMA 1996;276:496–502. [2] Fallowfield L, Jenkins V. Communicating sad, bad and difficult news in medicine. Lancet 2004;363:312–9. [3] Jedlicka-Köhler I, Götz M, Eichler I. Parents' recollection of the initial communication of the diagnosis of cystic fibrosis. Pediatrics 1996;97:204–9. [4] De Monestrol I, Brucefors AB, Sjöberg B, Hjelte L. Parental experiences of a clinical cystic fibrosis diagnosis and impact on siblings and relatives. http://www.dissertations.se; 2011. [5] Mack JW, Grier HE. The day one talk. J Clin Oncol 2004;22(3):563–6. [6] Borrowitz D, Robinson KA, Rosenfield M, Davis SD, Sabadosa KA, Spear SL, et al. Cystic Fibrosis Foundation evidence-based guidelines for management of infants with cystic fibrosis. J Pediatr 2009;155:S73–93. http://dx.doi.org/10.1016/j.jpeds.2009.09.001. [7] Smyth AR, Bell SC, Bojcin S, Bryon M, Duff A, Flume P, et al. European Cystic Fibrosis Society Standards of Care: best practice guidelines. J Cyst Fibros May 2014;13(Suppl. 1):S23–42. http://dx.doi.org/10.1016/ j.jcf.2014. 03.010. [8] Compas BE, Connor-Smith JK, Saltzman H, Thomsen AH, Wadsworth M. Coping with stress during childhood and adolescence: progress, problems and potential. Psychol Bull 2001;127:87–127. [9] Abbott J. Coping with cystic fibrosis. J R Soc Med 2003;96:42–50. [10] Schreurs PJG, van de Willige G, Tellegen B, Brosschot JF. De Utrechtse Coping Lijst: UCL-handleiding. Lisse Swets & Zeitlinger; 1988.
546
T. Havermans et al. / Journal of Cystic Fibrosis 14 (2015) 540–546
[11] Weiten W, Lloyd MA. Psychology applied to modern life. 9th ed. Wadsworth Cengage Learning; 2008. [12] Carver CS, Connor-Smith J. Personality and coping. Annu Rev Psychol 2010;61:679–704. http://dx.doi.org/10.1146/ annurev.psych.093008.100352 (Review). [13] Marin TJ, Chen E, Munch T, Miller G. Double exposure to acute stress and chronic family stress is associated with immune changes in children with asthma. Psychosom Med 2009;71:378–84. http://dx.doi.org/10.1097/ PSY.0b013e318199dbc3. [14] Wong MG, Heriot SA. Parents of children with CF: how they hope, cope and despair. Child Care Health 2008;34(4):344–54. http://dx.doi.org/10. 1111/j.1365-2214.2007.00804.x. [15] Sheehan J, Hisock H, Massie J, Jaffe A, Hay M. Caregiver coping, mental health and child problem behavior in cystic fibrosis: a cross-sectional study. Int Soc Behav Med 2014;21(2):211–20. http://dx.doi.org/10.1007/s12529-0139289-y. [16] Hodkova P, Abbott J, Mala I, Chladova H. Coping with cystic fibrosis—CF adults and parents of a child with CF in Czech Republic. J Cyst Fibros 2008;7(2):110. [17] Compas BE, Jaser SS, Dunn MJ, Rodriguez EM. Coping with chronic illness in childhood and adolescence. Annu Rev Clin Psychol 2012;8: 455–80. http://dx.doi.org/10.1146/annurev-clinpsy-032511-143108. [18] Abbott J, Dodd M, Gee L, Webb K. Ways of coping with cystic fibrosis: implications for treatment adherence. Disabil Rehabil May 2001;23(8):315–24. [19] Oshea J, Smith O, O'Marcaigh A, McMahon C, Geoghegan R, Cotter M. Breaking bad news: parents' experience of learning that their child has leukemia. Ir Med J 2007;100(9):588–90. [20] Cohen J. Statistical power analysis for the behavioral sciences. 2nd ed. Hillsdale NJ: Lawrence Earlbaum Associates; 1988. [21] Jurkovich GJ, Pierce B, Pananen L, Rivara FP. Giving bad news: the family perspective. J Trauma Inj Infect Crit Care May 2000;48(5):865–73.
[22] Price Dillard J, Shen Lijiang, Robinson Jeffrey D, Farrell Phillip M. Parental information seeking following a positive newborn screening for cystic fibrosis. J Health Commun Int Perspect 2010;15(8):880–94. [23] Hummelinck A, Pollock K. Parents' information needs about the treatment of their chronically ill child: a qualitative study. Patient Educ Couns 2006; 62:228–34. [24] Rosenbaum ME, Ferguson KJ, Lobas JG. Teaching medical students and residents skills for delivering bad news: a review of strategies. Acad Med 2004;79(2):107–17. [25] Ptacek JT, McIntosh EGJ. Physician challenges in communicating bad news. Behav Med 2009;32(4):380–7. http://dx.doi.org/10.1007/s10865009-9213-8. [26] Meert KL, Eggly S, Pollack M, Anand KJ, Zimmerman J, Carcillo J, et al. Parents' perspectives on physician–parent communication near the time of a child's death in the pediatric intensive care unit. Pediatr Crit Care Med 2004;9(1):2–7. [27] Tluczek A, Koscik RL, Modaff P, Pfeil D, Rock MJ, Farrell PM, et al. Newborn screening for cystic fibrosis: parents' preferences regarding counseling at the time of infants' sweat test. J Genet Couns 2006;15(4):277–91. [28] Henry B, Moffitt TE, Caspi A, Langley J, Silva PA. On the “remembrance of things past”: a longitudinal evaluation of the retrospective method. Psychol Assess 1994;6:92–101. [29] Todd RM, Talmi D, Schmitz TW, Susskind J, Anderson AK. Psychophysical and neural evidence for emotion-enhanced perceptual vividness. J Neurosci 2012;32:11201–12. http://dx.doi.org/10.1523/ JNEUROSCI. 0155-12.2012. [30] Castellani C, Southern KW, Brownlee K, Dankert Roelse J, Duff A, Farrell M, et al. European best practice guidelines for cystic fibrosis neonatal screening. J Cyst Fibros 2009;8(3):153–73. http://dx.doi.org/10. 1016/j.jcf.2009.01.004.
