Epilepsy & Behavior 29 (2013) 560–564
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SUDEP: What do parents want to know? Rajesh RamachandranNair a,⁎, Susan M. Jack b, Brandon F. Meaney a, Gabriel M. Ronen a a b
Department of Pediatrics (Neurology), McMaster University, 1280 Main Street West, Hamilton, Ontario L8S 4K1, Canada School of Nursing, McMaster University, 1280 Main Street West, Hamilton, Ontario L8S 4K1, Canada
a r t i c l e
i n f o
Article history: Received 7 June 2013 Revised 24 September 2013 Accepted 29 September 2013 Available online 26 October 2013 Keywords: Sudden unexpected death Epilepsy Qualitative research Counseling
a b s t r a c t Purpose: Our objective was to understand the range of parental views on whether and how to approach the issue of SUDEP with families, to clarify the optimal timing and formulation of the information, and to learn from parents the optimal counseling strategies in order to minimize the inherent emotional burden. Methods: The principles of fundamental qualitative description were used to guide this descriptive and exploratory study of parental experiences and perceptions. Stratified purposeful sampling included parents (i) who lost children to SUDEP, (ii) of children with moderate to severe epilepsy, (iii) of children with mild epilepsy, and (iv) of children with new-onset epilepsy. Focus group and in-depth one-on-one interviews were conducted. The principles of directed content analysis were used to code and categorize the focus group and interview data. Key concepts from the interview guide were used as the first level of coding categories. Codes were subsequently collapsed into broader categories. Results: There was full agreement, across both genders and regardless of seizure severity, that routine counseling about SUDEP should be provided by pediatric neurologists, during the appointment when the diagnosis of epilepsy is shared with parents, and with opportunities for short-term follow-up and discussions with clinical nurses or social workers. Parents described feeling overwhelmed, worried, and increasingly anxious when the risk of SUDEP was explained to them. Parents generally expressed a preference for receiving routine SUDEP counseling at the time of the diagnosis of epilepsy. Across all groups of parents, it was identified that SUDEP counseling should occur in a face-to-face interaction with the neurologist. In learning about SUDEP, parents expressed a need to be informed of the risk of SUDEP. There was group endorsement for receiving written information about SUDEP to reinforce the content shared by the neurologist. There was a consensus that it should be the parents' decision as to whether or not the child should be present at the meeting or when to inform the child about the risk of SUDEP. Conclusion: Participants in this study opined that all parents of children with epilepsy should receive routine SUDEP counseling and have access to ongoing professional support. Based on these findings, it is imperative for pediatric neurologists to refine their communication skills when counseling parents about SUDEP. © 2013 Elsevier Inc. All rights reserved.
1. Introduction There is neither empirical evidence nor consensus on the question of whether to inform patients and parents about the possibility of sudden unexpected death in epilepsy (SUDEP). Advocates of universal SUDEP counseling cite the “right to know,” but others point to the “right not to know” [1]. Potential negative reactions from patients seemed to deter doctors from frequent discussion of SUDEP [2]. Interviews of bereaved relatives of SUDEP fatalities often revealed feelings of guilt or blame, as well as regret that they and their deceased relative had not had the opportunity to make more informed decisions regarding lifestyle and treatment [3]. Although the American Epilepsy Society and the Epilepsy Foundation joint task force on SUDEP recommended discussing SUDEP with patients and their families, their report mentioned that no ⁎ Corresponding author at: Division of Pediatric Neurology, McMaster University, 1280 Main Street West, Hamilton, Ontario L8S 4K1, Canada. Fax: +1 905 521 7914. E-mail addresses: [email protected] (R. RamachandranNair), [email protected] (S.M. Jack), [email protected] (B.F. Meaney), [email protected] (G.M. Ronen). 1525-5050/$ – see front matter © 2013 Elsevier Inc. All rights reserved. http://dx.doi.org/10.1016/j.yebeh.2013.09.040
information was currently available on how SUDEP information could optimally be conveyed [4]. A survey of the views of parents regarding SUDEP disclosure and its impact reported that 91% of the responders wanted the physician to discuss SUDEP, and the majority preferred that the discussion be held at the time of diagnosis [5]. Importantly, the majority of parents (61%) did not want their child to be informed about SUDEP. However, several aspects of SUDEP discussion including the optimal setting for the discussion, who should participate, followup plans after the initial discussion, the content of the discussion, and how to inform the child and siblings still need to be explored. 1.1 We hypothesized that parents are the true experts in deciding on the optimal strategies for counseling about SUDEP in children and youth with epilepsy. Our research questions were as follows: (i) What is the range of perceptions among families of children with epilepsy as to whether or not routine counseling about SUDEP is appropriate?; (ii) If counseling about SUDEP is to be provided, when and how should this be done so that the information is understandable and anxiety is minimized?; and (iii) What is the range of perceptions among families
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of children with epilepsy as to whether or not information on SUDEP should be shared with younger patients?
participants following each session to respond to any emotional distress or questions that arose in response to participating in the interview.
1.2 We believe that qualitative methods are best suited for the exploration of parental preferences regarding SUDEP counseling. This methodology is in line with the NIH/NINDS workshop report on SUDEP that recommended conducting focus group interviews to determine when and how much information should be presented to patients and families [6].
2.5. Analysis
2. Methods 2.1. Study design The principles of fundamental qualitative description were used to guide this descriptive and exploratory qualitative study of parental experiences and perceptions [7]. 2.2. Participants Stratified purposeful sampling included parents (i) who lost children to SUDEP, (ii) of children with moderate to severe epilepsy, (iii) of children with mild epilepsy, and (iv) of children with new-onset epilepsy. The groups were stratified by parental gender to better understand any gender-specific perceptions [8]. Children and youth with epilepsy aged 17 years and below who had received treatment at the Neurology Clinic at McMaster Children's Hospital (Ontario, Canada) were identified from the hospital epilepsy database. We used the seizure-specific categorization of severity of epilepsy, which is based on the number of seizures in the previous 12 months [9]. For this study, we divided seizure severity into two categories (seizure-free and low frequency as ‘mild’ versus moderate and high frequency as ‘moderate–severe’ epilepsy). Children whose first recognized seizure was within the 12 months prior to the study were classified as having new-onset epilepsy. Children with exclusive absence seizures were not included in the study. We contacted six parents of four children who had SUDEP (three parents were identified through our clinic death registry, and additional three parents were recruited through a lay organization, SudepAware (www. sudepaware.org). This study was approved by the Faculty of Health Sciences–Hamilton Health Sciences Research Ethics Board. 2.3. Sample size As qualitative research is an iterative process of sampling, data collection, and analysis, required sample sizes cannot be calculated a priori. Instead, we estimated that recruiting approximately 50 parents of children and youth with epilepsy would be sufficient to reach data saturation [10]. 2.4. Data collection Parents were contacted by a research nurse to enquire if they would like to know about an interview-based qualitative research study on a rare condition related to epilepsy. The topic of SUDEP was mentioned along with the purpose of the study. Participants were provided, via mail, with the information on the research project and received a clinical pamphlet on SUDEP several days before the sessions. Written informed consent was obtained from all participants. The focus group interviews were conducted in the summer of 2011. Six focus group interviews (separate groups of mothers and fathers) for parents belonging to groups ii, iii, and iv were conducted by an experienced interviewer using a semistructured interview guide (Appendix 1), each lasting 2 h. In-depth one-on-one interviews (with a semistructured interview guide) were conducted with parents from category (i) because of the sensitive nature of the topic. All interviews were digitally recorded and then transcribed verbatim. The Neurology Clinic social worker was available to all
Textual analysis using qualitative software NVivo 8 (QSR International Pty Ltd 2011) occurred immediately following the transcription. The principles of directed content analysis were used to code and categorize the focus group and interview data [11]. Key concepts from the interview guide were used as the first level of coding categories. However, through a process of line-by-line coding, new codes were also established to reflect emerging concepts. Codes were subsequently collapsed into broader categories, and similarities and differences across categories were documented. 3. Results A purposeful sample of 42 parents participated in the study. Thirtysix parents (21 mothers and 15 fathers) of 21 children (new-onset epilepsy: 5, mild epilepsy: 9, and moderate–severe epilepsy: 7) participated in the focus group interviews (Table 1). We contacted six parents of four children who had expired due to SUDEP. Each of them participated in a one-on-one interview. All six parents confirmed that the cause of death in the coroner's report was SUDEP. and they believed that their child had SUDEP. 3.1. Experience before and after meeting the neurologist In all interviews, parents discussed their experiences of having their child diagnosed with epilepsy. Mothers discussed and disclosed, after their child's diagnosis of epilepsy, a significant number of mental health issues: depression, anxiety, fear, worry, guilt, and exhaustion. Fathers expressed feelings of worry, anger (directed toward the health care team), and concern for spouse and family. Both mothers and fathers expressed a high degree of concern or worry that their child would die (either during a seizure or as a result of an injury during a seizure). These fears led them to be highly vigilant regarding their children and wanting to monitor all of their activities. There was a particular fear that the child would have a seizure while sleeping and die at night. The majority of parents disclosed that the child slept in their bed as part of a coping strategy. A small number of parents divulged the adoption of different technologies to take over the nightly monitoring routine (e.g., therapy dog and monitors). 3.2. Parental understanding of SUDEP Among parents of children diagnosed and living with epilepsy (groups ii, iii, and iv), there was a lack of understanding about the exact meaning of the word SUDEP and risk of a child dying from SUDEP. While some parents were able to correctly explain that it is an “unexplained death”, others held the perception that SUDEP meant that the child died as a consequence of having a seizure. Mothers and fathers across all groups, while correctly explaining that it is a rare condition, still overestimated the risk of SUDEP. Parents who lost a child to SUDEP provided a more accurate and in-depth understanding of the criteria for diagnosing SUDEP and a recognition that, as parents, there is very little they could have done to prevent their child's death. 3.3. Parental perceptions about routine counseling for SUDEP 3.3.1 There was full agreement, across both genders and regardless of seizure severity, that routine counseling about SUDEP should be provided by pediatric neurologists, during the appointment when the diagnosis of epilepsy is shared with parents, and with opportunities for short-term follow-up and discussions with clinical nurses or social workers. Parents explained that it is important to have a comprehensive
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Table 1 Semiquantitative response from participants. Question/opinion
Mothers (groups ii, iii, and iv) #21
Fathers (groups ii, iii, and iv) #15
Parents of children who had SUDEP #6
Heard about SUDEP before study Risk of SUDEP overestimated Felt anxious/stressed on hearing about SUDEP SUDEP should be discussed with parents Prefers discussion at first visit SUDEP should be discussed by the pediatric neurologist Method of discussion: prefers face to face Parents to decide whether child should be informed about SUDEP Knowledge of SUDEP will change the interaction with children
16 Around a third Nearly two-thirds All Nearly half All All All None
7 Nearly half Nearly half All Nearly two-thirds All All All None
All None 5 All 3 All All All None
understanding of all of the risks associated with epilepsy. They shared that knowing about SUDEP increases parental awareness of the difficulties associated with the child's condition and allows them to cognitively “prepare for the worst”. Parents identified that with this information, they would be motivated to place monitors or alarms in their child's room. One parent who lost a child to SUDEP explained that by understanding the risks for SUDEP, parents and their children may be more likely to have increased compliance with treatment regimens, including taking medications regularly, minimizing consumption of alcohol and nonprescription drugs, and getting appropriate amounts of sleep. 3.3.2 Across all groups, parents described feeling overwhelmed, worried, and increasingly anxious when the risk of SUDEP was explained to them. Fathers tended to express feeling uneasy, uncertain, and frustrated because of the lack of measures they could implement to prevent SUDEP. The majority of mothers opined that the information had not formally changed their management of their child with the exception that they were now aware of the possibility and the potential need for being mentally prepared for a death in the family. The consensus among fathers was that there was already some level of vigilance and monitoring in the household of the child; this additional information, vital as it is, would not increase that level of monitoring. 3.3.3 Despite these drawbacks, there was agreement among parents that not knowing about the possibility of SUDEP would be worse. Parents indicated that they would feel angry if they were not informed, and if their child subsequently experienced SUDEP, they would feel burdened by guilt that there might have been more they could have done. Across the six focus groups of 36 parents of children living with epilepsy, nearly a third disclosed that the first time they learned of SUDEP was through the invitation to participate in this qualitative study. 3.4. Parental preferences for providing routine SUDEP counseling to parents of children with epilepsy 3.4.1 Participants disclosed personally learning about SUDEP from a broad range of sources including the following: the pediatric neurologist, research on the Internet, connecting with epilepsy support groups, participating in this study, and, for one parent, from the coroner following the postmortem. Based on their experiences, there was agreement in the recommendation from all participants that it is the responsibility of the pediatric neurologist to provide routine SUDEP counseling. Parents were emphatic in explaining that they should not first learn about SUDEP from a pamphlet or an Internet website. 3.4.2 A small number of fathers agreed that this type of counseling should not be the responsibility of a general practitioner or primary care physician. However, it was identified that while it is the responsibility of the pediatric neurologist to conduct the SUDEP counseling, parents expressed a preference for meeting with a clinical nurse who could “translate what the doctors say” or a clinical social worker “to soften
the blow.” Some opined that a pediatrician with knowledge in the management of epilepsy could also discuss SUDEP. 3.4.3 Parents generally expressed a preference for receiving routine SUDEP counseling at the time of the diagnosis of epilepsy. However, many of the mothers and fathers recognized that an extensive amount of information is shared in that appointment so it can be difficult to process content related to seizure severity, treatments, prognosis as well as the risk of SUDEP. Among mothers of children with moderate to severe epilepsy, many disclosed feeling overwhelmed at the first diagnostic visit and recommended receiving the information in a follow-up appointment scheduled within two to three weeks from the initial visit. A minority of fathers of children with mild epilepsy also concurred that routine SUDEP counseling should occur in a follow-up meeting shortly postdiagnosis. There was a consensus that discussion of SUDEP should not occur in the emergency room or critical care unit. 3.4.4 Across all groups of parents, it was identified that SUDEP counseling should occur in a face-to-face interaction between the neurologist and the parents and not be provided in a phone consultation. Fathers particularly emphasized the need for neurologists to schedule appointments with enough time to share and review SUDEP information. Participants identified that in their interactions with physicians, their wish is not only to be “told” information but also to have opportunities to “discuss” issues related to SUDEP and epilepsy. To facilitate introducing the topic of SUDEP, it was recommended that this conversation occur within the context of a clinician–parent relationship characterized by trust and where rapport had been established. In learning about SUDEP, parents expressed a need to be informed of the risk of SUDEP. However, some parents expressed that it is important for neurologists to stress that SUDEP is rare and to balance the message of risk, with one of hope. Parents who had lost a child to SUDEP were best able to articulate the messages that should be communicated to parents, including discussions of the following: (i) the actual risk of SUDEP; (ii) risk and protective factors associated with SUDEP; (iii) any known prevention strategies; and (iv) an acknowledgement that SUDEP may be beyond the parents' or physicians' ability to prevent or stop. There was group endorsement for receiving written information about SUDEP to reinforce the content shared by the neurologist. This was perceived as an important strategy in supporting parents to manage the extensive amount of information received during their appointments. Moreover, access to written information would allow parents to choose the time to be ready to learn more about SUDEP. Also, parents preferred to have a list of credible websites mentioned in the written pamphlet. In addition, parents wanted an opportunity to schedule a follow-up visit (within a short period of time) in order to have any outstanding questions or concerns addressed. 3.5. Parental perceptions about who should receive routine SUDEP counseling Participants identified that both parents (as appropriate) should be present at the appointment to receive routine SUDEP counseling.
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There was a consensus that it should be the parents' decision as to whether or not the child should be present at the meeting or when to inform the child about the risk of SUDEP. Factors influencing a parent's decision to tell a child about SUDEP included the child's age, level of understanding about epilepsy, and cognitive abilities. In general, parents expressed a willingness to share this information once the child was in early adolescence (e.g., N 12 years), possessed the maturity to process the information, and was not developmentally impaired. Some parents also stressed the importance of sharing information about SUDEP with children once they reached an age when they would be engaging in potentially risky behaviors (e.g., underage consumption of alcohol). A minority of parents expressed that they would not inform their child of SUDEP. There was a lack of agreement among both mothers and fathers about who should explain SUDEP to a child. Some cited that it is the responsibility of the parents, while others felt that the information should come from the pediatric neurologist. However, there was a consensus that it is the parents' right to decide whether children should be informed about SUDEP or not. There was a consensus that older siblings, especially if taking part in the care of the child, should be informed about the risk of SUDEP. 4. Discussion 4.1 The majority of parents who participated in the focus group interviews had heard about SUDEP before being invited to participate in the study. This was higher than the 34% reported in a study based on a questionnaire [5]. It is possible that the parents who attended onsite focus group and one-on-one interviews in our study were more motivated than the parents who participated in a postal questionnaire study. An alternative or additional explanation could be related to the fact that this study was conducted nearly two years after the publication of the UK study. It is reasonable to believe that wider availability of SUDEP information in the cyberspace and more active public discussion have led to increased public awareness about SUDEP during this time. In this study, a majority of parents overestimated the risk of SUDEP. A previous study among parents of children with epilepsy reported that nearly 61% of parents sought health information from the Internet [12]. Families may not be aware that the accuracy of the information provided in cyberspace is often questionable. 4.2 All the participants in the study preferred to have a discussion on SUDEP. This opinion was voiced by parents of children with newonset, mild, and moderate-severe epilepsy as well as by parents who lost a child due to SUDEP. Those who overestimated the risk of SUDEP were relieved to know that risk of SUDEP was relatively low in children. Information on SUDEP increased the overall understanding of the risk associated with epilepsy and motivated some for better seizure control strategies including drug compliance and avoiding seizure triggers like sleep deprivation. In the UK study, 91% of parents wanted to know about SUDEP [5]. Though parents were under stress after the onset of seizures in the child, nearly 50% of the mothers and most of the fathers wanted to hear about SUDEP at the time of diagnosis of epilepsy. Parents who were overwhelmed by the amount of information provided at the first visit preferred to have the discussion on SUDEP on a follow-up visit scheduled within a few weeks. In the UK questionnaire study, 74% of parents expressed similar opinion [5]. Based on these data, physicians should consider postponing the discussion on SUDEP with parents who appear overwhelmed to the next follow-up visit. However, in a busy clinical practice, it might be challenging to schedule a relatively early follow-up appointment. 4.3 Literature is scant on the content of SUDEP counseling. It is a challenging task to provide balanced but accurate information on SUDEP. Our study has provided better clarity regarding what parents would like the physician to discuss. Accurate risk estimation with
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strategies for possible prevention of SUDEP should be the focus of discussion. Parents preferred to have the discussion on SUDEP with a physician who is an expert in epilepsy and who could answer questions on SUDEP. In their opinion, a pediatric neurologist was the physician with these attributes. In the UK questionnaire study, 74% of the pediatric neurologists provided SUDEP information only to a selected group of patients [5]. Involvement of nurses or social workers in the SUDEP discussion has not been studied well. A survey conducted in the UK examined what, when, and how information about SUDEP was disseminated to patients by clinical nurse specialists in epilepsy (CNSEs). Clinical nurse specialists in epilepsy discussed SUDEP with most patients (50%). Ninety-five percent responded that both CNSEs and neurologists should discuss SUDEP with the patients [13]. All the participants in our study had children treated by a pediatric neurologist at some point. This experience might have biased their opinions. 4.4 It is a common practice in North America to involve an older child in discussions with health care providers regarding their health. The Health Care Consent Act, 1996, states that every person in Ontario, regardless of age, is presumed to be capable of consenting to or refusing medical treatment unless he or she is found incapable with respect to a specific treatment or plan of treatment [14]. Opinions expressed by the parents that it was up to them to decide whether the child should be informed about the risk of SUDEP makes it challenging for the neurologist to discuss SUDEP with the child. There is practical difficulty in sending the child outside the clinic room when the neurologist discusses SUDEP with parents. Forty-six percent of the pediatric neurologists in the UK study discussed SUDEP with both parents and children [5]. Several studies have reported negative reactions by the patients as perceived by the physicians [2,15,16]. We believe that a major reason for physicians not discussing SUDEP is their fear of causing negative emotions and anxiety in their patients. After the focus group interview, none of the parents contacted our social worker though that option was provided to them. Though parents described feeling overwhelmed, worried, and increasingly anxious when the risk of SUDEP was explained to them, knowing the actual low risk of SUDEP was relieving, especially to the parents who overestimated the risk. Knowledge about SUDEP was unlikely to result in the way they interacted with the child except for enhanced monitoring and perhaps continued cosleeping. 4.5 It is possible that parents who participated in the interview were information seekers as opposed to information avoiders. We contacted 70 parents, and only three refused to participate. Twenty-five parents could not participate in the focus group interviews on a Saturday during the summer because of scheduled vacations, work, or lack of child care support. In the UK, 100 parents were contacted for a questionnaire study, but only 67 parents completed and returned the first survey, and 47 parents completed and returned the second survey (39 completed and returned both surveys) [5]. Though we have not formally recorded the ethnicity of the participants, more than 90% were Caucasians. It is unclear whether the results of this study are transferable to all cultural and ethnic backgrounds. Overall, credibility of the study was enhanced by data type (focus group and one-on-one interviews) and data source (stratified sampling) triangulation [17]. Saturation of data was inferred by the emergence of the same themes across different focus groups and personal interviews. We suggest that the stability of the results of this study be retested after a few years. We used qualitative methodology to capture the full complexity and dimensionality of parents' perspectives and beliefs on how or when to be informed about SUDEP. The aim was to generate nonnumerical preliminary data to help guide clinicians and policy makers. However, for research that aims to directly impact on policy and practice, the findings of qualitative research can be limited by the small sample size that is necessary for in-depth exploratory work with the consequent lack of generalizability. A complementary mix of qualitative and
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quantitative methodologies can overcome weaknesses inherent to either method alone [18]. Sequential development of a survey instrument built upon the findings from the qualitative approach and illuminated by the parents' own phrases would be a sound methodology to achieve generalizability. 5. Conclusion This descriptive qualitative study has investigated parental opinions concerning the provision of counseling about the risk of SUDEP in their children. The results indicate very clearly that parents of children with epilepsy do want to be informed about SUDEP. The optimal timing and setting of SUDEP counseling should certainly be determined on a case-by-case basis, taking into account the degree of emotional and mental saturation that might currently exist in parents of newly diagnosed children. However, as a general rule, parents indicated a strong preference for the counseling to occur early on, preferably at the first or second clinical visit. There should be sufficient time allotted to allow for a two-way discussion regarding SUDEP. Preferably, printed material should be available for the parents to take away with them, and there should be the possibility of a short-term follow-up appointment for additional discussion and clarification, either with the neurologist or with a nurse or a social worker. The content of the counseling regarding SUDEP should include a realistic appraisal of the risk, with an emphasis on the actual prevalence of the condition. Conflict of interest statement None of the authors has any conflict of interest to disclose. Acknowledgments This study was supported by the innovation fund provided by the Ministry of Health and Long Term Care, Ontario through the Hamilton Academic Health Sciences Organization. We thank Tamzin Teffs, MSc, Executive Director, SUDEP Aware for helping with the participant recruitment. Appendix 1. Semistructured interview guide for the focus group interviews 1. To allow us to understand you and your family a bit better, we would appreciate it if you could each briefly introduce yourself, and if you are comfortable doing so, tell us briefly how old your child is currently and how old he/she was when diagnosed with epilepsy. 2. Please take a minute to think back to when you were formally told that your child had epilepsy. Can you each describe this experience to us? 3. When you were first learning about epilepsy, what were your main concerns or worries? 4. Are you familiar with the term: sudden unexplained death in epilepsy — SUDEP? a) What is your understanding of what SUDEP is? b) When did you first hear about it? c) Where, or from whom, did you learn about SUDEP?
5. When you first learned about SUDEP, what was your initial reaction? 6. What is your opinion on informing parents and other caregivers about SUDEP? 7. Who do you, as a parent, want to hear about SUDEP from? 8. If you feel you want to be told about SUDEP, when would be the best time to be given this information? 9. Who from your family should participate in the conversation about SUDEP? Why? 10. What details should be told to families in the first discussion about SUDEP? 11. Who (or what) do you see as a credible source of information about SUDEP? 12. Has anything changed for you or how you manage your child's epilepsy since learning about SUDEP? 13. Do you have any additional advice on how or if to inform families about SUDEP?
References [1] Beran RG, Weber S, Sungaran R, Venn N, Hung A. Review of the legal obligations of the doctor to discuss sudden unexplained death in epilepsy (SUDEP)—a cohort controlled comparative cross-matched study in an outpatient epilepsy clinic. Seizure 2004;13:523–8. [2] Morton B, Richardson A, Duncan S. Sudden unexpected death in epilepsy (SUDEP): don't ask, don't tell? J Neurol Neurosurg Psychiatry 2006;77:199–202. [3] Nashef L, Garner S, Sander JW, Fish DR, Shorvon SD. Circumstances of death in sudden death in epilepsy: interviews of bereaved relatives. J Neurol Neurosurg Psychiatry 1998;64:349–52. [4] So EL, Bainbridge J, Buchhalter JR, Donalty J, Donner EJ, Finucane A, et al. Report of the American Epilepsy Society and the Epilepsy Foundation Joint Task Force on Sudden Unexplained Death in Epilepsy. Epilepsia 2008. [5] Gayatri NA, Morrall MC, Jain V, Kashyape P, Pysden K, Ferrie C. Parental and physician beliefs regarding the provision and content of written sudden unexpected death in epilepsy (SUDEP) information. Epilepsia 2010;51:777–82. [6] Hirsch LJ, Donner EJ, So EL, Jacobs M, Nashef L, Noebels JL, et al. Abbreviated report of the NIH/NINDS workshop on sudden unexpected death in epilepsy. Neurology 2011;76:1932–8. [7] Sandelowski M. Whatever happened to qualitative description? Res Nurs Health 2000;23:334–40. [8] Shore C, Austin J, Musick B, Dunn D, McBride A, Creasy K. Psychosocial care needs of parents of children with new-onset seizures. 3. J Neurosci Nurs 1998;30:169–74. [9] Devinsky O, Vickrey BG, Cramer J, Perrine K, Hermann B, Meador K, et al. Development of the quality of life in epilepsy inventory. Epilepsia 1995;36:1089–104. [10] Krueger RA. Focus groups: a practical guide for applied research. 2nd ed. Thousand Oaks: Sage Publications Inc.; 1994. [11] Hsieh HF, Shannon SE. Three approaches to qualitative content analysis. Qual Health Res 2005;15:1277–88. [12] Lu C, Wirrell E, Blackman M. Where do families of children with epilepsy obtain their information? J Child Neurol 2005;20:905–10. [13] Lewis S, Higgins S, Goodwin M. Informing patients about sudden unexpected death in epilepsy: a survey of specialist nurses. Br J Neurosci Nurs 2008;4:30–4. [14] Geist R, Opler SE. A guide for health care practitioners in the assessment of young people's capacity to consent to treatment. Clin Pediatr (Phila) 2010;49:834–9. [15] Friedman D, Donner E, Stephens D, Wright C, Devinsky O. Sudden unexpected death in epilepsy: knowledge and experience among us and Canadian neurologists, abstract 2.324. American Epilepsy Society, 66th Annual Meeting. San Diego; 2012. [16] Vegni E, Leone D, Canevini MP, Tinuper P, Moja EA. Sudden unexpected death in epilepsy (SUDEP): a pilot study on truth telling among Italian epileptologists. Neurol Sci 2011;32:331–5. [17] Krefting L. Rigor in qualitative research: the assessment of trustworthiness. Am J Occup Ther 1991;45:214–22. [18] Tariq S, Woodman J. Using mixed methods in health research. JRSM short rep, 4; 2013 [2042533313479197].
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Epilepsy & Behavior journal homepage: www.elsevier.com/locate/yebeh
SUDEP: What do parents want to know? Rajesh RamachandranNair a,⁎, Susan M. Jack b, Brandon F. Meaney a, Gabriel M. Ronen a a b
Department of Pediatrics (Neurology), McMaster University, 1280 Main Street West, Hamilton, Ontario L8S 4K1, Canada School of Nursing, McMaster University, 1280 Main Street West, Hamilton, Ontario L8S 4K1, Canada
a r t i c l e
i n f o
Article history: Received 7 June 2013 Revised 24 September 2013 Accepted 29 September 2013 Available online 26 October 2013 Keywords: Sudden unexpected death Epilepsy Qualitative research Counseling
a b s t r a c t Purpose: Our objective was to understand the range of parental views on whether and how to approach the issue of SUDEP with families, to clarify the optimal timing and formulation of the information, and to learn from parents the optimal counseling strategies in order to minimize the inherent emotional burden. Methods: The principles of fundamental qualitative description were used to guide this descriptive and exploratory study of parental experiences and perceptions. Stratified purposeful sampling included parents (i) who lost children to SUDEP, (ii) of children with moderate to severe epilepsy, (iii) of children with mild epilepsy, and (iv) of children with new-onset epilepsy. Focus group and in-depth one-on-one interviews were conducted. The principles of directed content analysis were used to code and categorize the focus group and interview data. Key concepts from the interview guide were used as the first level of coding categories. Codes were subsequently collapsed into broader categories. Results: There was full agreement, across both genders and regardless of seizure severity, that routine counseling about SUDEP should be provided by pediatric neurologists, during the appointment when the diagnosis of epilepsy is shared with parents, and with opportunities for short-term follow-up and discussions with clinical nurses or social workers. Parents described feeling overwhelmed, worried, and increasingly anxious when the risk of SUDEP was explained to them. Parents generally expressed a preference for receiving routine SUDEP counseling at the time of the diagnosis of epilepsy. Across all groups of parents, it was identified that SUDEP counseling should occur in a face-to-face interaction with the neurologist. In learning about SUDEP, parents expressed a need to be informed of the risk of SUDEP. There was group endorsement for receiving written information about SUDEP to reinforce the content shared by the neurologist. There was a consensus that it should be the parents' decision as to whether or not the child should be present at the meeting or when to inform the child about the risk of SUDEP. Conclusion: Participants in this study opined that all parents of children with epilepsy should receive routine SUDEP counseling and have access to ongoing professional support. Based on these findings, it is imperative for pediatric neurologists to refine their communication skills when counseling parents about SUDEP. © 2013 Elsevier Inc. All rights reserved.
1. Introduction There is neither empirical evidence nor consensus on the question of whether to inform patients and parents about the possibility of sudden unexpected death in epilepsy (SUDEP). Advocates of universal SUDEP counseling cite the “right to know,” but others point to the “right not to know” [1]. Potential negative reactions from patients seemed to deter doctors from frequent discussion of SUDEP [2]. Interviews of bereaved relatives of SUDEP fatalities often revealed feelings of guilt or blame, as well as regret that they and their deceased relative had not had the opportunity to make more informed decisions regarding lifestyle and treatment [3]. Although the American Epilepsy Society and the Epilepsy Foundation joint task force on SUDEP recommended discussing SUDEP with patients and their families, their report mentioned that no ⁎ Corresponding author at: Division of Pediatric Neurology, McMaster University, 1280 Main Street West, Hamilton, Ontario L8S 4K1, Canada. Fax: +1 905 521 7914. E-mail addresses: [email protected] (R. RamachandranNair), [email protected] (S.M. Jack), [email protected] (B.F. Meaney), [email protected] (G.M. Ronen). 1525-5050/$ – see front matter © 2013 Elsevier Inc. All rights reserved. http://dx.doi.org/10.1016/j.yebeh.2013.09.040
information was currently available on how SUDEP information could optimally be conveyed [4]. A survey of the views of parents regarding SUDEP disclosure and its impact reported that 91% of the responders wanted the physician to discuss SUDEP, and the majority preferred that the discussion be held at the time of diagnosis [5]. Importantly, the majority of parents (61%) did not want their child to be informed about SUDEP. However, several aspects of SUDEP discussion including the optimal setting for the discussion, who should participate, followup plans after the initial discussion, the content of the discussion, and how to inform the child and siblings still need to be explored. 1.1 We hypothesized that parents are the true experts in deciding on the optimal strategies for counseling about SUDEP in children and youth with epilepsy. Our research questions were as follows: (i) What is the range of perceptions among families of children with epilepsy as to whether or not routine counseling about SUDEP is appropriate?; (ii) If counseling about SUDEP is to be provided, when and how should this be done so that the information is understandable and anxiety is minimized?; and (iii) What is the range of perceptions among families
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of children with epilepsy as to whether or not information on SUDEP should be shared with younger patients?
participants following each session to respond to any emotional distress or questions that arose in response to participating in the interview.
1.2 We believe that qualitative methods are best suited for the exploration of parental preferences regarding SUDEP counseling. This methodology is in line with the NIH/NINDS workshop report on SUDEP that recommended conducting focus group interviews to determine when and how much information should be presented to patients and families [6].
2.5. Analysis
2. Methods 2.1. Study design The principles of fundamental qualitative description were used to guide this descriptive and exploratory qualitative study of parental experiences and perceptions [7]. 2.2. Participants Stratified purposeful sampling included parents (i) who lost children to SUDEP, (ii) of children with moderate to severe epilepsy, (iii) of children with mild epilepsy, and (iv) of children with new-onset epilepsy. The groups were stratified by parental gender to better understand any gender-specific perceptions [8]. Children and youth with epilepsy aged 17 years and below who had received treatment at the Neurology Clinic at McMaster Children's Hospital (Ontario, Canada) were identified from the hospital epilepsy database. We used the seizure-specific categorization of severity of epilepsy, which is based on the number of seizures in the previous 12 months [9]. For this study, we divided seizure severity into two categories (seizure-free and low frequency as ‘mild’ versus moderate and high frequency as ‘moderate–severe’ epilepsy). Children whose first recognized seizure was within the 12 months prior to the study were classified as having new-onset epilepsy. Children with exclusive absence seizures were not included in the study. We contacted six parents of four children who had SUDEP (three parents were identified through our clinic death registry, and additional three parents were recruited through a lay organization, SudepAware (www. sudepaware.org). This study was approved by the Faculty of Health Sciences–Hamilton Health Sciences Research Ethics Board. 2.3. Sample size As qualitative research is an iterative process of sampling, data collection, and analysis, required sample sizes cannot be calculated a priori. Instead, we estimated that recruiting approximately 50 parents of children and youth with epilepsy would be sufficient to reach data saturation [10]. 2.4. Data collection Parents were contacted by a research nurse to enquire if they would like to know about an interview-based qualitative research study on a rare condition related to epilepsy. The topic of SUDEP was mentioned along with the purpose of the study. Participants were provided, via mail, with the information on the research project and received a clinical pamphlet on SUDEP several days before the sessions. Written informed consent was obtained from all participants. The focus group interviews were conducted in the summer of 2011. Six focus group interviews (separate groups of mothers and fathers) for parents belonging to groups ii, iii, and iv were conducted by an experienced interviewer using a semistructured interview guide (Appendix 1), each lasting 2 h. In-depth one-on-one interviews (with a semistructured interview guide) were conducted with parents from category (i) because of the sensitive nature of the topic. All interviews were digitally recorded and then transcribed verbatim. The Neurology Clinic social worker was available to all
Textual analysis using qualitative software NVivo 8 (QSR International Pty Ltd 2011) occurred immediately following the transcription. The principles of directed content analysis were used to code and categorize the focus group and interview data [11]. Key concepts from the interview guide were used as the first level of coding categories. However, through a process of line-by-line coding, new codes were also established to reflect emerging concepts. Codes were subsequently collapsed into broader categories, and similarities and differences across categories were documented. 3. Results A purposeful sample of 42 parents participated in the study. Thirtysix parents (21 mothers and 15 fathers) of 21 children (new-onset epilepsy: 5, mild epilepsy: 9, and moderate–severe epilepsy: 7) participated in the focus group interviews (Table 1). We contacted six parents of four children who had expired due to SUDEP. Each of them participated in a one-on-one interview. All six parents confirmed that the cause of death in the coroner's report was SUDEP. and they believed that their child had SUDEP. 3.1. Experience before and after meeting the neurologist In all interviews, parents discussed their experiences of having their child diagnosed with epilepsy. Mothers discussed and disclosed, after their child's diagnosis of epilepsy, a significant number of mental health issues: depression, anxiety, fear, worry, guilt, and exhaustion. Fathers expressed feelings of worry, anger (directed toward the health care team), and concern for spouse and family. Both mothers and fathers expressed a high degree of concern or worry that their child would die (either during a seizure or as a result of an injury during a seizure). These fears led them to be highly vigilant regarding their children and wanting to monitor all of their activities. There was a particular fear that the child would have a seizure while sleeping and die at night. The majority of parents disclosed that the child slept in their bed as part of a coping strategy. A small number of parents divulged the adoption of different technologies to take over the nightly monitoring routine (e.g., therapy dog and monitors). 3.2. Parental understanding of SUDEP Among parents of children diagnosed and living with epilepsy (groups ii, iii, and iv), there was a lack of understanding about the exact meaning of the word SUDEP and risk of a child dying from SUDEP. While some parents were able to correctly explain that it is an “unexplained death”, others held the perception that SUDEP meant that the child died as a consequence of having a seizure. Mothers and fathers across all groups, while correctly explaining that it is a rare condition, still overestimated the risk of SUDEP. Parents who lost a child to SUDEP provided a more accurate and in-depth understanding of the criteria for diagnosing SUDEP and a recognition that, as parents, there is very little they could have done to prevent their child's death. 3.3. Parental perceptions about routine counseling for SUDEP 3.3.1 There was full agreement, across both genders and regardless of seizure severity, that routine counseling about SUDEP should be provided by pediatric neurologists, during the appointment when the diagnosis of epilepsy is shared with parents, and with opportunities for short-term follow-up and discussions with clinical nurses or social workers. Parents explained that it is important to have a comprehensive
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Table 1 Semiquantitative response from participants. Question/opinion
Mothers (groups ii, iii, and iv) #21
Fathers (groups ii, iii, and iv) #15
Parents of children who had SUDEP #6
Heard about SUDEP before study Risk of SUDEP overestimated Felt anxious/stressed on hearing about SUDEP SUDEP should be discussed with parents Prefers discussion at first visit SUDEP should be discussed by the pediatric neurologist Method of discussion: prefers face to face Parents to decide whether child should be informed about SUDEP Knowledge of SUDEP will change the interaction with children
16 Around a third Nearly two-thirds All Nearly half All All All None
7 Nearly half Nearly half All Nearly two-thirds All All All None
All None 5 All 3 All All All None
understanding of all of the risks associated with epilepsy. They shared that knowing about SUDEP increases parental awareness of the difficulties associated with the child's condition and allows them to cognitively “prepare for the worst”. Parents identified that with this information, they would be motivated to place monitors or alarms in their child's room. One parent who lost a child to SUDEP explained that by understanding the risks for SUDEP, parents and their children may be more likely to have increased compliance with treatment regimens, including taking medications regularly, minimizing consumption of alcohol and nonprescription drugs, and getting appropriate amounts of sleep. 3.3.2 Across all groups, parents described feeling overwhelmed, worried, and increasingly anxious when the risk of SUDEP was explained to them. Fathers tended to express feeling uneasy, uncertain, and frustrated because of the lack of measures they could implement to prevent SUDEP. The majority of mothers opined that the information had not formally changed their management of their child with the exception that they were now aware of the possibility and the potential need for being mentally prepared for a death in the family. The consensus among fathers was that there was already some level of vigilance and monitoring in the household of the child; this additional information, vital as it is, would not increase that level of monitoring. 3.3.3 Despite these drawbacks, there was agreement among parents that not knowing about the possibility of SUDEP would be worse. Parents indicated that they would feel angry if they were not informed, and if their child subsequently experienced SUDEP, they would feel burdened by guilt that there might have been more they could have done. Across the six focus groups of 36 parents of children living with epilepsy, nearly a third disclosed that the first time they learned of SUDEP was through the invitation to participate in this qualitative study. 3.4. Parental preferences for providing routine SUDEP counseling to parents of children with epilepsy 3.4.1 Participants disclosed personally learning about SUDEP from a broad range of sources including the following: the pediatric neurologist, research on the Internet, connecting with epilepsy support groups, participating in this study, and, for one parent, from the coroner following the postmortem. Based on their experiences, there was agreement in the recommendation from all participants that it is the responsibility of the pediatric neurologist to provide routine SUDEP counseling. Parents were emphatic in explaining that they should not first learn about SUDEP from a pamphlet or an Internet website. 3.4.2 A small number of fathers agreed that this type of counseling should not be the responsibility of a general practitioner or primary care physician. However, it was identified that while it is the responsibility of the pediatric neurologist to conduct the SUDEP counseling, parents expressed a preference for meeting with a clinical nurse who could “translate what the doctors say” or a clinical social worker “to soften
the blow.” Some opined that a pediatrician with knowledge in the management of epilepsy could also discuss SUDEP. 3.4.3 Parents generally expressed a preference for receiving routine SUDEP counseling at the time of the diagnosis of epilepsy. However, many of the mothers and fathers recognized that an extensive amount of information is shared in that appointment so it can be difficult to process content related to seizure severity, treatments, prognosis as well as the risk of SUDEP. Among mothers of children with moderate to severe epilepsy, many disclosed feeling overwhelmed at the first diagnostic visit and recommended receiving the information in a follow-up appointment scheduled within two to three weeks from the initial visit. A minority of fathers of children with mild epilepsy also concurred that routine SUDEP counseling should occur in a follow-up meeting shortly postdiagnosis. There was a consensus that discussion of SUDEP should not occur in the emergency room or critical care unit. 3.4.4 Across all groups of parents, it was identified that SUDEP counseling should occur in a face-to-face interaction between the neurologist and the parents and not be provided in a phone consultation. Fathers particularly emphasized the need for neurologists to schedule appointments with enough time to share and review SUDEP information. Participants identified that in their interactions with physicians, their wish is not only to be “told” information but also to have opportunities to “discuss” issues related to SUDEP and epilepsy. To facilitate introducing the topic of SUDEP, it was recommended that this conversation occur within the context of a clinician–parent relationship characterized by trust and where rapport had been established. In learning about SUDEP, parents expressed a need to be informed of the risk of SUDEP. However, some parents expressed that it is important for neurologists to stress that SUDEP is rare and to balance the message of risk, with one of hope. Parents who had lost a child to SUDEP were best able to articulate the messages that should be communicated to parents, including discussions of the following: (i) the actual risk of SUDEP; (ii) risk and protective factors associated with SUDEP; (iii) any known prevention strategies; and (iv) an acknowledgement that SUDEP may be beyond the parents' or physicians' ability to prevent or stop. There was group endorsement for receiving written information about SUDEP to reinforce the content shared by the neurologist. This was perceived as an important strategy in supporting parents to manage the extensive amount of information received during their appointments. Moreover, access to written information would allow parents to choose the time to be ready to learn more about SUDEP. Also, parents preferred to have a list of credible websites mentioned in the written pamphlet. In addition, parents wanted an opportunity to schedule a follow-up visit (within a short period of time) in order to have any outstanding questions or concerns addressed. 3.5. Parental perceptions about who should receive routine SUDEP counseling Participants identified that both parents (as appropriate) should be present at the appointment to receive routine SUDEP counseling.
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There was a consensus that it should be the parents' decision as to whether or not the child should be present at the meeting or when to inform the child about the risk of SUDEP. Factors influencing a parent's decision to tell a child about SUDEP included the child's age, level of understanding about epilepsy, and cognitive abilities. In general, parents expressed a willingness to share this information once the child was in early adolescence (e.g., N 12 years), possessed the maturity to process the information, and was not developmentally impaired. Some parents also stressed the importance of sharing information about SUDEP with children once they reached an age when they would be engaging in potentially risky behaviors (e.g., underage consumption of alcohol). A minority of parents expressed that they would not inform their child of SUDEP. There was a lack of agreement among both mothers and fathers about who should explain SUDEP to a child. Some cited that it is the responsibility of the parents, while others felt that the information should come from the pediatric neurologist. However, there was a consensus that it is the parents' right to decide whether children should be informed about SUDEP or not. There was a consensus that older siblings, especially if taking part in the care of the child, should be informed about the risk of SUDEP. 4. Discussion 4.1 The majority of parents who participated in the focus group interviews had heard about SUDEP before being invited to participate in the study. This was higher than the 34% reported in a study based on a questionnaire [5]. It is possible that the parents who attended onsite focus group and one-on-one interviews in our study were more motivated than the parents who participated in a postal questionnaire study. An alternative or additional explanation could be related to the fact that this study was conducted nearly two years after the publication of the UK study. It is reasonable to believe that wider availability of SUDEP information in the cyberspace and more active public discussion have led to increased public awareness about SUDEP during this time. In this study, a majority of parents overestimated the risk of SUDEP. A previous study among parents of children with epilepsy reported that nearly 61% of parents sought health information from the Internet [12]. Families may not be aware that the accuracy of the information provided in cyberspace is often questionable. 4.2 All the participants in the study preferred to have a discussion on SUDEP. This opinion was voiced by parents of children with newonset, mild, and moderate-severe epilepsy as well as by parents who lost a child due to SUDEP. Those who overestimated the risk of SUDEP were relieved to know that risk of SUDEP was relatively low in children. Information on SUDEP increased the overall understanding of the risk associated with epilepsy and motivated some for better seizure control strategies including drug compliance and avoiding seizure triggers like sleep deprivation. In the UK study, 91% of parents wanted to know about SUDEP [5]. Though parents were under stress after the onset of seizures in the child, nearly 50% of the mothers and most of the fathers wanted to hear about SUDEP at the time of diagnosis of epilepsy. Parents who were overwhelmed by the amount of information provided at the first visit preferred to have the discussion on SUDEP on a follow-up visit scheduled within a few weeks. In the UK questionnaire study, 74% of parents expressed similar opinion [5]. Based on these data, physicians should consider postponing the discussion on SUDEP with parents who appear overwhelmed to the next follow-up visit. However, in a busy clinical practice, it might be challenging to schedule a relatively early follow-up appointment. 4.3 Literature is scant on the content of SUDEP counseling. It is a challenging task to provide balanced but accurate information on SUDEP. Our study has provided better clarity regarding what parents would like the physician to discuss. Accurate risk estimation with
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strategies for possible prevention of SUDEP should be the focus of discussion. Parents preferred to have the discussion on SUDEP with a physician who is an expert in epilepsy and who could answer questions on SUDEP. In their opinion, a pediatric neurologist was the physician with these attributes. In the UK questionnaire study, 74% of the pediatric neurologists provided SUDEP information only to a selected group of patients [5]. Involvement of nurses or social workers in the SUDEP discussion has not been studied well. A survey conducted in the UK examined what, when, and how information about SUDEP was disseminated to patients by clinical nurse specialists in epilepsy (CNSEs). Clinical nurse specialists in epilepsy discussed SUDEP with most patients (50%). Ninety-five percent responded that both CNSEs and neurologists should discuss SUDEP with the patients [13]. All the participants in our study had children treated by a pediatric neurologist at some point. This experience might have biased their opinions. 4.4 It is a common practice in North America to involve an older child in discussions with health care providers regarding their health. The Health Care Consent Act, 1996, states that every person in Ontario, regardless of age, is presumed to be capable of consenting to or refusing medical treatment unless he or she is found incapable with respect to a specific treatment or plan of treatment [14]. Opinions expressed by the parents that it was up to them to decide whether the child should be informed about the risk of SUDEP makes it challenging for the neurologist to discuss SUDEP with the child. There is practical difficulty in sending the child outside the clinic room when the neurologist discusses SUDEP with parents. Forty-six percent of the pediatric neurologists in the UK study discussed SUDEP with both parents and children [5]. Several studies have reported negative reactions by the patients as perceived by the physicians [2,15,16]. We believe that a major reason for physicians not discussing SUDEP is their fear of causing negative emotions and anxiety in their patients. After the focus group interview, none of the parents contacted our social worker though that option was provided to them. Though parents described feeling overwhelmed, worried, and increasingly anxious when the risk of SUDEP was explained to them, knowing the actual low risk of SUDEP was relieving, especially to the parents who overestimated the risk. Knowledge about SUDEP was unlikely to result in the way they interacted with the child except for enhanced monitoring and perhaps continued cosleeping. 4.5 It is possible that parents who participated in the interview were information seekers as opposed to information avoiders. We contacted 70 parents, and only three refused to participate. Twenty-five parents could not participate in the focus group interviews on a Saturday during the summer because of scheduled vacations, work, or lack of child care support. In the UK, 100 parents were contacted for a questionnaire study, but only 67 parents completed and returned the first survey, and 47 parents completed and returned the second survey (39 completed and returned both surveys) [5]. Though we have not formally recorded the ethnicity of the participants, more than 90% were Caucasians. It is unclear whether the results of this study are transferable to all cultural and ethnic backgrounds. Overall, credibility of the study was enhanced by data type (focus group and one-on-one interviews) and data source (stratified sampling) triangulation [17]. Saturation of data was inferred by the emergence of the same themes across different focus groups and personal interviews. We suggest that the stability of the results of this study be retested after a few years. We used qualitative methodology to capture the full complexity and dimensionality of parents' perspectives and beliefs on how or when to be informed about SUDEP. The aim was to generate nonnumerical preliminary data to help guide clinicians and policy makers. However, for research that aims to directly impact on policy and practice, the findings of qualitative research can be limited by the small sample size that is necessary for in-depth exploratory work with the consequent lack of generalizability. A complementary mix of qualitative and
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quantitative methodologies can overcome weaknesses inherent to either method alone [18]. Sequential development of a survey instrument built upon the findings from the qualitative approach and illuminated by the parents' own phrases would be a sound methodology to achieve generalizability. 5. Conclusion This descriptive qualitative study has investigated parental opinions concerning the provision of counseling about the risk of SUDEP in their children. The results indicate very clearly that parents of children with epilepsy do want to be informed about SUDEP. The optimal timing and setting of SUDEP counseling should certainly be determined on a case-by-case basis, taking into account the degree of emotional and mental saturation that might currently exist in parents of newly diagnosed children. However, as a general rule, parents indicated a strong preference for the counseling to occur early on, preferably at the first or second clinical visit. There should be sufficient time allotted to allow for a two-way discussion regarding SUDEP. Preferably, printed material should be available for the parents to take away with them, and there should be the possibility of a short-term follow-up appointment for additional discussion and clarification, either with the neurologist or with a nurse or a social worker. The content of the counseling regarding SUDEP should include a realistic appraisal of the risk, with an emphasis on the actual prevalence of the condition. Conflict of interest statement None of the authors has any conflict of interest to disclose. Acknowledgments This study was supported by the innovation fund provided by the Ministry of Health and Long Term Care, Ontario through the Hamilton Academic Health Sciences Organization. We thank Tamzin Teffs, MSc, Executive Director, SUDEP Aware for helping with the participant recruitment. Appendix 1. Semistructured interview guide for the focus group interviews 1. To allow us to understand you and your family a bit better, we would appreciate it if you could each briefly introduce yourself, and if you are comfortable doing so, tell us briefly how old your child is currently and how old he/she was when diagnosed with epilepsy. 2. Please take a minute to think back to when you were formally told that your child had epilepsy. Can you each describe this experience to us? 3. When you were first learning about epilepsy, what were your main concerns or worries? 4. Are you familiar with the term: sudden unexplained death in epilepsy — SUDEP? a) What is your understanding of what SUDEP is? b) When did you first hear about it? c) Where, or from whom, did you learn about SUDEP?
5. When you first learned about SUDEP, what was your initial reaction? 6. What is your opinion on informing parents and other caregivers about SUDEP? 7. Who do you, as a parent, want to hear about SUDEP from? 8. If you feel you want to be told about SUDEP, when would be the best time to be given this information? 9. Who from your family should participate in the conversation about SUDEP? Why? 10. What details should be told to families in the first discussion about SUDEP? 11. Who (or what) do you see as a credible source of information about SUDEP? 12. Has anything changed for you or how you manage your child's epilepsy since learning about SUDEP? 13. Do you have any additional advice on how or if to inform families about SUDEP?
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