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Parental experiences before and long-term after their children's hemispherotomy — A population-based qualitative study
Epilepsy & Behavior 60 (2016) 11–16
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Epilepsy & Behavior journal homepage: www.elsevier.com/locate/yebeh
Parental experiences before and long-term after their children's hemispherotomy — A population-based qualitative study Anneli Ozanne a,b,⁎,1, Cecilia Verdinelli c,1, Ingrid Olsson c, Ulla H. Graneheim d,e, Kristina Malmgren b a
Institute of Health and Care Sciences, Sahlgrenska Academy, University of Gothenburg, Box 457, 405 30 Gothenburg, Sweden Department of Clinical Neuroscience and Rehabilitation, Institute of Neuroscience and Physiology, Sahlgrenska Academy, University of Gothenburg, Box 410, 405 30 Gothenburg, Sweden Department of Paediatrics, Institute of Clinical Sciences, Sahlgrenska Academy, University of Gothenburg, Gothenburg, Sweden d Department of Nursing, Umeå University, 901 87 Umeå, Sweden e Department of Health Sciences, University West, Trollhättan, Sweden b c
a r t i c l e
i n f o
Article history: Received 23 December 2015 Revised 16 March 2016 Accepted 10 April 2016 Available online xxxx Keywords: Childhood epilepsy Hemispherotomy Parental experiences Qualitative content analysis
a b s t r a c t Severe childhood epilepsy has an impact on the whole family. For selected children, hemispherotomy is the treatment of choice. The aim of this study was to explore parents' experiences before and after hemispherotomy as reported at a long-term follow-up and their view on received information and support. This was a populationbased qualitative descriptive study, using qualitative content analysis of interviews. Seven to eighteen years after hemispherotomy, parents of twenty-one operated children were interviewed about the family life situation, expectations before surgery, and support and information before and after surgery. Before surgery, the theme ‘Living in a chaotic bubble’ illuminates how parents felt: the family lived in isolation, they felt both dissatisfaction and satisfaction about support and information, and they experienced that surgery was a question about life or death. After surgery, the theme ‘Hovering between success and disaster’ illuminates how parents hovered between happiness if the surgery was successful and sadness about e.g., complications and behavior problems. They experienced both excellent and poor support, in hospital and at rehabilitation. Regardless of all concerns, parents were satisfied that the child had received an operation. The hemispherotomies were successful and generated a better life situation. However, in order to cope, families need support and information throughout the whole process, from the onset of epilepsy and for a long time after surgery. If the child has behavior problems, an assessment should be made before surgery in order for the families to get adequate support. The specialist team needs to be involved as early as possible and follow the families for several years after surgery. Focus should be on the whole family, if needed including grandparents; family-centered care might be relevant for this patient group. © 2016 Elsevier Inc. All rights reserved.
1. Introduction When a child has epilepsy, it affects the whole family life situation in a negative way. Epilepsy is unpredictable, has a potential for injury [1], and may cause both cognitive and behavioral problems [1–3]. Drugresistant epilepsy also causes psychosocial difficulties [4,5], family stress [6,7], and problems at school [8] and with adaptive skills [3]. Healthrelated quality of life (HRQoL) is often reduced in both children [9–11] and parents [11,12].
⁎ Corresponding author at: Institute of Health and Care Sciences, Sahlgrenska Academy, University of Gothenburg, Gothenburg, Sweden. Tel.: +46 766186072. E-mail addresses: [email protected] (A. Ozanne), [email protected] (C. Verdinelli), [email protected] (I. Olsson), [email protected] (U. H. Graneheim), [email protected] (K. Malmgren). 1 Both authors contributed equally.
http://dx.doi.org/10.1016/j.yebeh.2016.04.025 1525-5050/© 2016 Elsevier Inc. All rights reserved.
While hemispherotomy may seem a drastic option, it is an effective treatment for selected children with drug-resistant epilepsy due to widespread pathology in one hemisphere [13–15]. A prerequisite for considering hemispherotomy is a preexisting neurologic deficit with hemiparesis. Two large studies report seizure freedom in about 65% [14,15]. However, seizure recurrences occur over time [14]. In our own population-based study, 55% were seizure-free two years after surgery and 38% at the 5- or 10-year follow-up. Another 41% had ≥75% reduction in seizure frequency [16]. Few studies have examined quality of life (QoL) after hemispherotomy. In a recent study, parents perceived their children's QoL to be good but described problems with their children's self-care, daily activities, and mobility [17]. There is a lack of studies on parental experiences before and after their children's hemispherotomy. Baca et al. interviewed parents of children who had undergone epilepsy surgery (32% hemispherectomy) about their journey to surgery. They experienced both personal and family stresses before surgery; they wanted their child to undergo
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surgery, even though they were frightened [18]. The present study was undertaken as part of a long-term follow-up after hemispherotomy in the Swedish series [16]. The aim of this interview study was to explore parental experiences before and after hemispherotomy as reported at a long-term follow-up and also the parents' views on received information and support. 2. Material and methods
was decided to expand the project by including parents of all patients who underwent a hemispherotomy during the period 1995–2007. The interview guide contained fourteen open-ended questions exploring the experiences about life situation before surgery, the support and information before and after surgery, the expectations and satisfaction with outcome, and the present situation (see supplementary information). Both the recorded interviews and the interviews documented with detailed notes were included (15 telephone interviews, six personal meetings).
2.1. Design This was a population-based national study including parents of children with drug-resistant epilepsy who underwent a hemispherotomy. It was a cross-sectional qualitative descriptive study, based on interviews with parents 7–18 years after surgery (median: 13 years). A qualitative content analysis was used for data analyses. 2.2. Participants The study included consecutive patients from all centers in Sweden where hemispherotomies are performed and used data from the Swedish National Epilepsy Surgery Register, which prospectively collects data on all epilepsy surgery procedures in Sweden and at 2-, 5-, 10-, and 15-year follow-ups [19]. Outcome data after hemispherotomy and the long-term patient perspective have been described in a previous paper [16]. Long-term was defined as ≥5 years since surgery. In all, twenty-nine children and adolescents underwent hemispherotomy from 1995–2007 and had at least a 5- or 10-year follow-up. Three young adults responded themselves, but these interviews were not analyzed in this study since the focus was on parents' experiences. One adult could not be contacted. In two cases, parents chose not to participate. Twenty-three parents accepted participation. Two taped interviews could not be analyzed because of technical problems. Thus, the results are based on interviews with parents of 21/25 children (ten mothers, eight fathers, and both parents in three cases). Patient data are presented in Table 1. The study was approved by the Regional Board of Medical Ethics at the University of Gothenburg. 2.3. Data collection Data were collected between July 2012 and March 2014. The interviewer (CV) and the researchers who analyzed the qualitative data (AO and UG) were not part of any epilepsy surgery team. The survey started as a pilot project with four interviews, which were not recorded, but documented in detailed notes. Because of the encouraging pilot data, it
2.4. Analysis A qualitative content analysis was carried out [20]. This method systematically analyzes written or verbal communication [21], by focusing on subject and context and by dealing with similarities and differences between parts of the text [20]. The transcribed interviews were divided into meaning units, i.e., words and sentences containing aspects related to each other, and were grouped by their content and context. In order to shorten the text but still preserve its core, the meaning units were condensed and coded. The codes were sorted and abstracted into nine subthemes, based on threads of meaning running through the codes. Through interpretation, these subthemes were further abstracted into two themes, one concerned the time before surgery and the other concerned the time after surgery. A theme can be explained as a thread of an underlying meaning. Themes are condensed meaning units, codes, or categories at an interpretative level [20]. To ensure trustworthiness of the themes and subthemes, the analytic process involved a back and forth movement between the whole text and its parts. Interviews with only notes were compared with the taperecorded interviews; no significant differences were identified.
3. Results Parents of children who had undergone hemispherotomy experienced that surgery was successful and had given the families better lives. However, they were living in a complex situation both before and after surgery. The theme Living in a chaotic bubble illuminates parents' experiences before surgery (Table 2). The theme Hovering between success and disaster highlights their experiences after surgery (Table 3).
Table 2 Illustration of themes, subthemes, and quotes before the hemispherotomies. Parents' experiences before hemispherotomy
Table 1 Patient data on the 21 children. Sex Median age at epilepsy onset (range) Median duration of epilepsy at surgery (range) Median age at surgery (range) Seizure outcome two years after surgery
Median age at interview (range) Seizure outcome at interview compared to preoperatively
Living condition
Theme
Subtheme
Quotes
Living in a chaotic bubble
Feeling isolated from others
p6: ‘I had to stop working…//…We could not do much. We were locked up in our home. At least I.’ p4: ‘It was I (mother) who had read about the possibilities for surgery. No physician raised the issue with me; at least not before I had raised it myself.’ p11: ‘It (the information) was very good and they included my parents-in-law, my father-in-law, since he found it very difficult to understand that the child was ill. They arranged a meeting at the hospital and when we stepped into the room there were pictures of her brain on the wall…their information was very factual.’ p1: ‘We felt it was a question of life or death. There were no alternatives.’
12 males/9 females 3 m (0 m–7 yrs 7 m) 1 yr 3 m (3 m – 19 yrs 1 m) 4 yrs 10 m (4 m–20 yrs 7 m) Seizure-free ≥75% reduction in seizure frequency b75% reduction in seizure frequency 16 yrs (7 yrs–37 yrs) Seizure-free ≥75% reduction in seizure frequency b75% reduction in seizure frequency Living with parents Living alone with support Sheltered accommodation
Getting too little support and information 12/21 3/21
Being satisfied with support and information
6/21 9/21 6/21 6/21 14a/21 3/21 4/21
Surgery — a question of life or death p: participant.
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Table 3 Illustration of themes, subthemes, and quotes after the hemispherotomies. Parents' experiences after hemispherotomy Theme
Subtheme
Quotes
Hovering between success and disaster
Feeling happy
p18: ‘Now we have learned to take a few days at a time, and we are very glad. Now he has learned to walk! And no one believed that.’ p15: ‘Both she and we as a family are still very much affected by her disease and what has happened. We are not an ordinary family because she is very demanding. The whole family can rarely do things together.’ p17: ‘We got very good support at the hospital where the surgery was performed; we had continuous contact and so on. If we phoned them about something, they always called back.’ p15: ‘We are critical of how the physicians acted during that period. It felt like nobody had time, and everybody carried on stressed as usual.’ p7: ‘One part of me is terrified to describe the child's situation as problematic because it is as it is. My child is my child. He lives and has a good life and we have a good life as a family…//…Because the good thing outweighs everything else…’
Feeling sad Getting excellent support Getting poor support Feeling satisfied despite all concerns p: participant.
3.1. Living in a chaotic bubble 3.1.1. Feeling isolated from others Before surgery, the social life of the family was affected by the child's seizures. Parents felt isolated and inactive; they were living in a “bubble”. Often, one parent took the overall responsibility for the ill child. The other one took care of the siblings and his/her professional work. Siblings needed one parent's attention for activities, impossible if the ill child were to participate. Thus, family members did not do many things together. Furthermore, parents who could not work regretted this and felt isolated. It took time to get used to the situation and to adapt expectations. The child's disabilities became evident when compared with other children. Some parents felt discomfort if they were with friends and the child had a seizure. Others were open about the situation, bringing the child to joint activities and inviting friends. 3.1.2. Getting too little support and information Parents described that support and information were inadequate before the epilepsy was recognized to be drug-resistant, especially from the local hospital. Their concerns were not taken seriously. For example, they received information that everything was normal or that they had to leave the hospital despite the fact that their child was improperly treated. Some parents had been referred to several specialists before they ended up with the right specialist. Parents were afraid and worried. The parents questioned if the information about surgery was adequate, especially if the physician said that the child would probably die without surgery. They wondered if they asked the wrong questions and if they received misguided information about brain damage. Parents wanted more information about how psychologically exhausting an operation could be for the child. If the pediatric neurologists revealed their own worries and insecurities regarding epilepsy surgery, this could have negative effects. Parents did not know what to expect or if the child would survive. Sometimes, parents received more information from other parents than from the physician. They were disappointed if they felt that information was withheld, for example, if another hospital had more competence. Parents also felt that it was difficult to manage the grief of the child's grandparents. They wanted grandparents to be offered professional support. 3.1.3. Being satisfied with support and information The support and information went from poor to strong when specialists at the university hospitals were involved. Supporting pediatric neurologists and treatment strategies helped them to manage the situation. Information about the surgery and its possible risks was
burdensome but often good and comprehensive. Being shown images of the child's brain helped parents and grandparents understand the severity of the child's disorder. Trust and safety characterized the relations to the pediatric neurologists and specialist nurses. It strengthened parents' trust to see that the pediatric neurologist fought for the child and that the parents' opinions counted in the decision process. Parents appreciated continuous contact. In contrast to the description in the subtheme above, the pediatric neurologist's worry and sympathy for the family could also strengthen them since they felt invited and could be insecure together. 3.1.4. Surgery — a question of life or death Parents felt that epilepsy surgery was a question of life or death, of chaos or control. It was terrifying to see the child so ill. The epilepsy was uncontrolled, and drugs gave side effects. Parents felt that there was no alternative to surgery. However, it was a difficult decision since they did not know the outcome. The situation at home was unbearable; parents felt that they had no control over their lives. They needed to look after the child day and night; the burden was huge. The lives of the families were described as worthless; everything revolved around the seizures. It was difficult to establish contact with the child, and the parents sometimes felt depressed. They found that the child did not have a normal physical and psychological development, and some described symptoms of behavior disorders. Investigations were often time consuming, which caused worry and frustration. It was demanding not to know if surgery would be possible, and the parents were thankful when the decision was finally made. Information about possible complications related to surgery did not prevent the parents from wishing to proceed with the operation. They preferred to take a risk rather than live in constant fear. 3.2. Hovering between success and disaster 3.2.1. Feeling happy When seizure freedom was achieved or seizure frequency reduced, parents were happy and felt life was like a dream; they never imagined that it could be so good. The children were more alert, even if seizures and drugs sometimes still caused tiredness. It was fantastic to see them in activities they were not expected to manage. They could do more things on their own, and parents did not need to look after them to the same extent. It was like a new beginning. The family also did more things together. Parents were also happy if the child was obstinate and strong-willed, a behavior demonstrating independence and development. It took a long time for parents to believe that the seizures would not return, but realizing that this was actually true, they were happy and felt free. They had less worries and stress and were not so occupied with the epilepsy. Focus was on the future and no longer on medication or the seizure diary.
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Even if life could be unpractical because of the child's disabilities, the positive results of surgery outweighed the negative consequences. Parents thought that the whole family often had a good life. It was important for siblings to be involved but also for them to do things on their own. It became easier to make plans, but on the other hand, the situation was easier if parents could take one day at a time. Grandparents also gave support when they overcame their fear of taking care of the disabled child. Not until parents had control did they focus on taking care of themselves. Hobbies helped them to find strength. But they also wanted to come into contact with other parents whose children had also undergone a hemispherotomy. 3.2.2. Feeling sad If the child did not become seizure-free, or had seizure recurrence, the life situation was burdensome for the whole family. Parents were disappointed, sad, and frightened. Their expectations had not been fulfilled, and they questioned their decision. Even if the seizures were not so frequent and severe, parents worried that they might worsen again. They never became accustomed to their child having seizures. Parents considered that, even if they had not seen any positive results, they could not conclude that the surgery was to blame. The situation might have been worse if the operation had not been performed; maybe the surgeon even saved the child's life. Parents thought that it was important to receive the information that surgery was aimed to treat the epilepsy and not other disabilities, such as behavioral problems, in order to have realistic expectations. They emphasized the importance of being informed about both positive and negative outcomes and risks. Even if surgery was a medical success, the family's life situation was sometimes characterized by sorrow and problems. The deterioration, expected or unexpected, of e.g., motor function, speech, visual field, or attention made life more difficult and burdensome. Even a long time after surgery, some parents needed to keep the child under close observation all the time because of seizures. Everything circled around the child. It was difficult to have a job, making them feel stuck at home; it caused economic problems, and some depended on financial support. If the children had behavioral problems, the parents wondered if they had been caused by surgery, if they were normal teenage problems, or if they were related to neuropsychiatric disorders. Parents were disappointed if it took a long time to get a correct diagnosis. The family situation would have been less complicated if they had been told about the child's diagnosis. Tantrums affected the family most, and to avoid them, the parents sometimes tiptoed around the child. These families could isolate themselves since the child's behavior made social life difficult. However, parents did not want to describe the situation as problematic; the situation was accepted for what it was. Parents worried if their child had learning problems. It was important to find the appropriate school. Parents also worried that the child could be exploited by others. Parental health had been down-prioritized for many years. They had stress symptoms, anxiety, and sleeping problems. They worried about the child's future after their death. Some wanted siblings to take care of the ill child in the future, which was the reason why some had more children. Some children did not feel well. Their energy was affected, and they did not have the strength to do things and thus became isolated. 3.2.3. Getting excellent support Parents often experienced excellent support and information from the team when the children had the operation, helping them to handle their worries that their child would change for the worse. With good support directly after surgery, they felt comfortable to contact the team for a long time after surgery if needed, which was important and
created security. Meeting the same professionals also gave a feeling of safety. Long hospitalizations were tiresome for the families. Parents felt that the staff mostly took good care of them. They appreciated early interventions from a rehabilitation team. Rehabilitation was often beneficial, especially at the beginning. Support and assistance helped families in their life situation, and they appreciated e.g., that physiotherapists worked intensely with the child, which made them see him/her improve more than they ever thought possible. 3.2.4. Getting poor support Sometimes, parents experienced poor information and support. They wanted to know more about the future; it was not sufficient to get a telephone number to call if needed. During the long hospitalizations, some parents felt that nobody had time for them. Arrogance of some professionals made them very disappointed. They needed more support in a situation where one parent always had to be in the hospital. Parents had neglected their own needs, and it was difficult to find the time to do things together as a couple. The parents also described disappointment if rehabilitation took longer than expected from the information they had received. The initial rehabilitation was often good, but at the local level, they often felt that it was inadequate. The transfer from pediatric to adult care was sometimes difficult. In some cases, parents decided to reduce rehabilitation, albeit beneficial, because their homes had become a training area, and neither children nor parents were able to rest. It was important that the home could remain an area of rest and preferable if rehabilitation could be given at school or at the day center. The Swedish Social Insurance Agency, technical aids center, school, and municipality were criticized by the parents. Based on their experience, they felt that the bureaucracy had been problematic and had taken a great deal of time. They had to fight for their rights, despite physicians' certificates. Without specific diagnoses of comorbidities, it was even more difficult to get adequate support. They thought that it would be helpful if the authorities understood that parents only asked for help when they had reached their limit, and then, urgent help was necessary. If the seizure frequency was reduced after surgery, support from the municipality was sometimes also reduced, which had a negative effect on the family's life situation, since many problems still remained. Parents who did not feel that they received adequate support sometimes felt that all focus was on the child. They found it obvious that parents should also be offered support. 3.2.5. Feeling satisfied despite all concerns The long-term development after surgery was mostly described in positive terms. Despite seizures, disabilities, and cognitive impairments, children and parents were reported to have a good life situation. According to parents, their children enjoyed the new life situation and felt safe when they had no seizures or reduced seizure frequency. They dared do more than before; they had friends and were active, did sports, listened to music, had daily activities, sheltered jobs, or were at school. They still made progress physically and mentally and had become more independent. Children in need of surveillance by parents due to seizures were reported to be frustrated by this, even though they understood the concern. Parents also recognized that they themselves and professionals sometimes hindered the children's development and social life, for fear the child would not cope or because the child's activities were not prioritized. 4. Discussion The findings of this study illuminate parents' experiences of the family life situation before and long-term after their children's hemispherotomy. Before surgery, they lived in a chaotic bubble and
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felt that surgery was a question of life or death. After surgery, they hovered between success and disaster. They experienced both happiness and sadness, good and poor support. Despite all concerns, the family life situation significantly improved after surgery. There is a lack of long-term studies on parental experiences of family life situation and support before and after hemispherotomy. Baca et al. [18] studied parents' experiences of the journey to epilepsy surgery. They describe parents' desperation to get a diagnosis and proper treatment. Parents talked about surgery as a last resort but also that it was necessary. These findings are in accordance with our results. Also in our study, parents reported that the support and information were limited before the specialist team became involved. We also found that parents experienced varying support and information directly after the hemispherotomy and for a long time afterwards. Further, parents were stressed over the family life situation before and after surgery. These results are in accordance with earlier studies [7,22]. One study on epilepsy management found that families feel that their views on the provision of support are not adhered to [23]. Our study indicates that it is important to involve the families early in the treatment plan for them to feel safe and that the specialist team should also support the families for a long time after surgery. Parents of children with drug-resistant epilepsy have lower QoL compared with population norms [24]. In our study, the finding that parents do not see to their own needs also indicates the importance to support them in getting more time for themselves. Information could also be offered to grandparents who worry but also support the family. Family-centered care might be an alternative to make it possible to meet the family’s needs from their own perspectives [25]. Like other researchers [7,26], we found that the problematic life situation causes isolation, but in accordance with Baca et al. [18], we found that parents wanted more contact with other families in the same situation. Therefore, it might be valuable to have family support groups. We found that the children's behavioral problems cause much concern. They are common in children with epilepsy [27], but in spite of that, comorbidities were underdiagnosed, and parents needed more information before and after surgery. The behavioral problems came as a shock after surgery, and parents wondered if they had always been there. They thought that they would have managed better if they had been informed about the child's additional diagnoses at an earlier phase. That information is a prerequisite for counseling and interventions aiming at improving the psychosocial situation [28].
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Another limitation is that only three patients could be interviewed themselves, which was too low a number for a separate analysis. The majority of patients had intellectual disabilities to a degree that they could not participate in this kind of interview. 5. Conclusion The life situation before hemispherotomy was chaotic, and although the operation caused much concern, its positive effects were appreciated. Parents reported positive effects, not only on seizures but also on the family situation. It led to less worries and stress and to less isolation for the family, including the siblings. However, support and information need to be improved. Specialist teams should be involved as early as possible and follow the family after surgery. Behavioral problems need to be diagnosed before surgery so the families can get adequate support and realistic counseling. Family-centered care with specialist teams in both neurology and rehabilitation might strengthen the possibilities to meet the families' needs. Further qualitative studies on parents' and patients' life situation and experiences of medical care and support are needed to improve the care before and long-term after hemispherotomies. Acknowledgments The authors thank the steering committee of the Swedish National Epilepsy Surgery Register, all of the Swedish epilepsy surgery teams, especially the epilepsy nurses (in Gothenburg, Linköping, Lund, Stockholm, Umeå, and Uppsala), and Sahlgrenska University Hospital, Gothenburg, Sweden. The study was funded by grants from the Swedish Research Council (grant 521-2011-169); the Sahlgrenska Academy, University of Gothenburg through the LUA/ALF agreement (grant ALFGBG137431); the Margarethahem Foundation; and the Research Foundation of Queen Silvia Children's Hospital. Disclosure of conflict of interest None of the authors has any conflict of interest to disclose. We confirm that we have read the Journal's positions on issues involved in ethical publication and affirm that this report is consistent with those guidelines. Appendix A. Supplementary data
4.1. Strengths and limitations One strength of this study is that it is population-based; parents of all patients who had undergone hemispherotomy in Sweden from 1995– 2007 were invited to participate, and only a few declined participation. Another important strength is that the interviewer and the two researchers analyzing and coding data were not part of any epilepsy surgery team and had not been involved in the care of the patients. The fact that interviews were conducted at different time points after surgery might be considered a limitation. An ideal study would have been longitudinal. However, since the shortest time from surgery to interview was seven years, all interviews can be considered as long-term (i.e., ≥5 years). The answers obtained at long-term must be understood as such; not primarily as descriptive testimonies but as memories processed through emotions and colored from further experiences, which were then developed into opinions and personal views. It could be that parents whose children had a good outcome had more positive memories of the information and support than those whose children had less improvement from surgery. However, it seems reasonable that even the shortest time to interview (seven years) is enough for memories to transform into a more general sense of satisfaction or dissatisfaction. Therefore, although the varying times to interviews are suboptimal, they are not considered a serious limitation to this analysis.
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Contents lists available at ScienceDirect
Epilepsy & Behavior journal homepage: www.elsevier.com/locate/yebeh
Parental experiences before and long-term after their children's hemispherotomy — A population-based qualitative study Anneli Ozanne a,b,⁎,1, Cecilia Verdinelli c,1, Ingrid Olsson c, Ulla H. Graneheim d,e, Kristina Malmgren b a
Institute of Health and Care Sciences, Sahlgrenska Academy, University of Gothenburg, Box 457, 405 30 Gothenburg, Sweden Department of Clinical Neuroscience and Rehabilitation, Institute of Neuroscience and Physiology, Sahlgrenska Academy, University of Gothenburg, Box 410, 405 30 Gothenburg, Sweden Department of Paediatrics, Institute of Clinical Sciences, Sahlgrenska Academy, University of Gothenburg, Gothenburg, Sweden d Department of Nursing, Umeå University, 901 87 Umeå, Sweden e Department of Health Sciences, University West, Trollhättan, Sweden b c
a r t i c l e
i n f o
Article history: Received 23 December 2015 Revised 16 March 2016 Accepted 10 April 2016 Available online xxxx Keywords: Childhood epilepsy Hemispherotomy Parental experiences Qualitative content analysis
a b s t r a c t Severe childhood epilepsy has an impact on the whole family. For selected children, hemispherotomy is the treatment of choice. The aim of this study was to explore parents' experiences before and after hemispherotomy as reported at a long-term follow-up and their view on received information and support. This was a populationbased qualitative descriptive study, using qualitative content analysis of interviews. Seven to eighteen years after hemispherotomy, parents of twenty-one operated children were interviewed about the family life situation, expectations before surgery, and support and information before and after surgery. Before surgery, the theme ‘Living in a chaotic bubble’ illuminates how parents felt: the family lived in isolation, they felt both dissatisfaction and satisfaction about support and information, and they experienced that surgery was a question about life or death. After surgery, the theme ‘Hovering between success and disaster’ illuminates how parents hovered between happiness if the surgery was successful and sadness about e.g., complications and behavior problems. They experienced both excellent and poor support, in hospital and at rehabilitation. Regardless of all concerns, parents were satisfied that the child had received an operation. The hemispherotomies were successful and generated a better life situation. However, in order to cope, families need support and information throughout the whole process, from the onset of epilepsy and for a long time after surgery. If the child has behavior problems, an assessment should be made before surgery in order for the families to get adequate support. The specialist team needs to be involved as early as possible and follow the families for several years after surgery. Focus should be on the whole family, if needed including grandparents; family-centered care might be relevant for this patient group. © 2016 Elsevier Inc. All rights reserved.
1. Introduction When a child has epilepsy, it affects the whole family life situation in a negative way. Epilepsy is unpredictable, has a potential for injury [1], and may cause both cognitive and behavioral problems [1–3]. Drugresistant epilepsy also causes psychosocial difficulties [4,5], family stress [6,7], and problems at school [8] and with adaptive skills [3]. Healthrelated quality of life (HRQoL) is often reduced in both children [9–11] and parents [11,12].
⁎ Corresponding author at: Institute of Health and Care Sciences, Sahlgrenska Academy, University of Gothenburg, Gothenburg, Sweden. Tel.: +46 766186072. E-mail addresses: [email protected] (A. Ozanne), [email protected] (C. Verdinelli), [email protected] (I. Olsson), [email protected] (U. H. Graneheim), [email protected] (K. Malmgren). 1 Both authors contributed equally.
http://dx.doi.org/10.1016/j.yebeh.2016.04.025 1525-5050/© 2016 Elsevier Inc. All rights reserved.
While hemispherotomy may seem a drastic option, it is an effective treatment for selected children with drug-resistant epilepsy due to widespread pathology in one hemisphere [13–15]. A prerequisite for considering hemispherotomy is a preexisting neurologic deficit with hemiparesis. Two large studies report seizure freedom in about 65% [14,15]. However, seizure recurrences occur over time [14]. In our own population-based study, 55% were seizure-free two years after surgery and 38% at the 5- or 10-year follow-up. Another 41% had ≥75% reduction in seizure frequency [16]. Few studies have examined quality of life (QoL) after hemispherotomy. In a recent study, parents perceived their children's QoL to be good but described problems with their children's self-care, daily activities, and mobility [17]. There is a lack of studies on parental experiences before and after their children's hemispherotomy. Baca et al. interviewed parents of children who had undergone epilepsy surgery (32% hemispherectomy) about their journey to surgery. They experienced both personal and family stresses before surgery; they wanted their child to undergo
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surgery, even though they were frightened [18]. The present study was undertaken as part of a long-term follow-up after hemispherotomy in the Swedish series [16]. The aim of this interview study was to explore parental experiences before and after hemispherotomy as reported at a long-term follow-up and also the parents' views on received information and support. 2. Material and methods
was decided to expand the project by including parents of all patients who underwent a hemispherotomy during the period 1995–2007. The interview guide contained fourteen open-ended questions exploring the experiences about life situation before surgery, the support and information before and after surgery, the expectations and satisfaction with outcome, and the present situation (see supplementary information). Both the recorded interviews and the interviews documented with detailed notes were included (15 telephone interviews, six personal meetings).
2.1. Design This was a population-based national study including parents of children with drug-resistant epilepsy who underwent a hemispherotomy. It was a cross-sectional qualitative descriptive study, based on interviews with parents 7–18 years after surgery (median: 13 years). A qualitative content analysis was used for data analyses. 2.2. Participants The study included consecutive patients from all centers in Sweden where hemispherotomies are performed and used data from the Swedish National Epilepsy Surgery Register, which prospectively collects data on all epilepsy surgery procedures in Sweden and at 2-, 5-, 10-, and 15-year follow-ups [19]. Outcome data after hemispherotomy and the long-term patient perspective have been described in a previous paper [16]. Long-term was defined as ≥5 years since surgery. In all, twenty-nine children and adolescents underwent hemispherotomy from 1995–2007 and had at least a 5- or 10-year follow-up. Three young adults responded themselves, but these interviews were not analyzed in this study since the focus was on parents' experiences. One adult could not be contacted. In two cases, parents chose not to participate. Twenty-three parents accepted participation. Two taped interviews could not be analyzed because of technical problems. Thus, the results are based on interviews with parents of 21/25 children (ten mothers, eight fathers, and both parents in three cases). Patient data are presented in Table 1. The study was approved by the Regional Board of Medical Ethics at the University of Gothenburg. 2.3. Data collection Data were collected between July 2012 and March 2014. The interviewer (CV) and the researchers who analyzed the qualitative data (AO and UG) were not part of any epilepsy surgery team. The survey started as a pilot project with four interviews, which were not recorded, but documented in detailed notes. Because of the encouraging pilot data, it
2.4. Analysis A qualitative content analysis was carried out [20]. This method systematically analyzes written or verbal communication [21], by focusing on subject and context and by dealing with similarities and differences between parts of the text [20]. The transcribed interviews were divided into meaning units, i.e., words and sentences containing aspects related to each other, and were grouped by their content and context. In order to shorten the text but still preserve its core, the meaning units were condensed and coded. The codes were sorted and abstracted into nine subthemes, based on threads of meaning running through the codes. Through interpretation, these subthemes were further abstracted into two themes, one concerned the time before surgery and the other concerned the time after surgery. A theme can be explained as a thread of an underlying meaning. Themes are condensed meaning units, codes, or categories at an interpretative level [20]. To ensure trustworthiness of the themes and subthemes, the analytic process involved a back and forth movement between the whole text and its parts. Interviews with only notes were compared with the taperecorded interviews; no significant differences were identified.
3. Results Parents of children who had undergone hemispherotomy experienced that surgery was successful and had given the families better lives. However, they were living in a complex situation both before and after surgery. The theme Living in a chaotic bubble illuminates parents' experiences before surgery (Table 2). The theme Hovering between success and disaster highlights their experiences after surgery (Table 3).
Table 2 Illustration of themes, subthemes, and quotes before the hemispherotomies. Parents' experiences before hemispherotomy
Table 1 Patient data on the 21 children. Sex Median age at epilepsy onset (range) Median duration of epilepsy at surgery (range) Median age at surgery (range) Seizure outcome two years after surgery
Median age at interview (range) Seizure outcome at interview compared to preoperatively
Living condition
Theme
Subtheme
Quotes
Living in a chaotic bubble
Feeling isolated from others
p6: ‘I had to stop working…//…We could not do much. We were locked up in our home. At least I.’ p4: ‘It was I (mother) who had read about the possibilities for surgery. No physician raised the issue with me; at least not before I had raised it myself.’ p11: ‘It (the information) was very good and they included my parents-in-law, my father-in-law, since he found it very difficult to understand that the child was ill. They arranged a meeting at the hospital and when we stepped into the room there were pictures of her brain on the wall…their information was very factual.’ p1: ‘We felt it was a question of life or death. There were no alternatives.’
12 males/9 females 3 m (0 m–7 yrs 7 m) 1 yr 3 m (3 m – 19 yrs 1 m) 4 yrs 10 m (4 m–20 yrs 7 m) Seizure-free ≥75% reduction in seizure frequency b75% reduction in seizure frequency 16 yrs (7 yrs–37 yrs) Seizure-free ≥75% reduction in seizure frequency b75% reduction in seizure frequency Living with parents Living alone with support Sheltered accommodation
Getting too little support and information 12/21 3/21
Being satisfied with support and information
6/21 9/21 6/21 6/21 14a/21 3/21 4/21
Surgery — a question of life or death p: participant.
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Table 3 Illustration of themes, subthemes, and quotes after the hemispherotomies. Parents' experiences after hemispherotomy Theme
Subtheme
Quotes
Hovering between success and disaster
Feeling happy
p18: ‘Now we have learned to take a few days at a time, and we are very glad. Now he has learned to walk! And no one believed that.’ p15: ‘Both she and we as a family are still very much affected by her disease and what has happened. We are not an ordinary family because she is very demanding. The whole family can rarely do things together.’ p17: ‘We got very good support at the hospital where the surgery was performed; we had continuous contact and so on. If we phoned them about something, they always called back.’ p15: ‘We are critical of how the physicians acted during that period. It felt like nobody had time, and everybody carried on stressed as usual.’ p7: ‘One part of me is terrified to describe the child's situation as problematic because it is as it is. My child is my child. He lives and has a good life and we have a good life as a family…//…Because the good thing outweighs everything else…’
Feeling sad Getting excellent support Getting poor support Feeling satisfied despite all concerns p: participant.
3.1. Living in a chaotic bubble 3.1.1. Feeling isolated from others Before surgery, the social life of the family was affected by the child's seizures. Parents felt isolated and inactive; they were living in a “bubble”. Often, one parent took the overall responsibility for the ill child. The other one took care of the siblings and his/her professional work. Siblings needed one parent's attention for activities, impossible if the ill child were to participate. Thus, family members did not do many things together. Furthermore, parents who could not work regretted this and felt isolated. It took time to get used to the situation and to adapt expectations. The child's disabilities became evident when compared with other children. Some parents felt discomfort if they were with friends and the child had a seizure. Others were open about the situation, bringing the child to joint activities and inviting friends. 3.1.2. Getting too little support and information Parents described that support and information were inadequate before the epilepsy was recognized to be drug-resistant, especially from the local hospital. Their concerns were not taken seriously. For example, they received information that everything was normal or that they had to leave the hospital despite the fact that their child was improperly treated. Some parents had been referred to several specialists before they ended up with the right specialist. Parents were afraid and worried. The parents questioned if the information about surgery was adequate, especially if the physician said that the child would probably die without surgery. They wondered if they asked the wrong questions and if they received misguided information about brain damage. Parents wanted more information about how psychologically exhausting an operation could be for the child. If the pediatric neurologists revealed their own worries and insecurities regarding epilepsy surgery, this could have negative effects. Parents did not know what to expect or if the child would survive. Sometimes, parents received more information from other parents than from the physician. They were disappointed if they felt that information was withheld, for example, if another hospital had more competence. Parents also felt that it was difficult to manage the grief of the child's grandparents. They wanted grandparents to be offered professional support. 3.1.3. Being satisfied with support and information The support and information went from poor to strong when specialists at the university hospitals were involved. Supporting pediatric neurologists and treatment strategies helped them to manage the situation. Information about the surgery and its possible risks was
burdensome but often good and comprehensive. Being shown images of the child's brain helped parents and grandparents understand the severity of the child's disorder. Trust and safety characterized the relations to the pediatric neurologists and specialist nurses. It strengthened parents' trust to see that the pediatric neurologist fought for the child and that the parents' opinions counted in the decision process. Parents appreciated continuous contact. In contrast to the description in the subtheme above, the pediatric neurologist's worry and sympathy for the family could also strengthen them since they felt invited and could be insecure together. 3.1.4. Surgery — a question of life or death Parents felt that epilepsy surgery was a question of life or death, of chaos or control. It was terrifying to see the child so ill. The epilepsy was uncontrolled, and drugs gave side effects. Parents felt that there was no alternative to surgery. However, it was a difficult decision since they did not know the outcome. The situation at home was unbearable; parents felt that they had no control over their lives. They needed to look after the child day and night; the burden was huge. The lives of the families were described as worthless; everything revolved around the seizures. It was difficult to establish contact with the child, and the parents sometimes felt depressed. They found that the child did not have a normal physical and psychological development, and some described symptoms of behavior disorders. Investigations were often time consuming, which caused worry and frustration. It was demanding not to know if surgery would be possible, and the parents were thankful when the decision was finally made. Information about possible complications related to surgery did not prevent the parents from wishing to proceed with the operation. They preferred to take a risk rather than live in constant fear. 3.2. Hovering between success and disaster 3.2.1. Feeling happy When seizure freedom was achieved or seizure frequency reduced, parents were happy and felt life was like a dream; they never imagined that it could be so good. The children were more alert, even if seizures and drugs sometimes still caused tiredness. It was fantastic to see them in activities they were not expected to manage. They could do more things on their own, and parents did not need to look after them to the same extent. It was like a new beginning. The family also did more things together. Parents were also happy if the child was obstinate and strong-willed, a behavior demonstrating independence and development. It took a long time for parents to believe that the seizures would not return, but realizing that this was actually true, they were happy and felt free. They had less worries and stress and were not so occupied with the epilepsy. Focus was on the future and no longer on medication or the seizure diary.
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Even if life could be unpractical because of the child's disabilities, the positive results of surgery outweighed the negative consequences. Parents thought that the whole family often had a good life. It was important for siblings to be involved but also for them to do things on their own. It became easier to make plans, but on the other hand, the situation was easier if parents could take one day at a time. Grandparents also gave support when they overcame their fear of taking care of the disabled child. Not until parents had control did they focus on taking care of themselves. Hobbies helped them to find strength. But they also wanted to come into contact with other parents whose children had also undergone a hemispherotomy. 3.2.2. Feeling sad If the child did not become seizure-free, or had seizure recurrence, the life situation was burdensome for the whole family. Parents were disappointed, sad, and frightened. Their expectations had not been fulfilled, and they questioned their decision. Even if the seizures were not so frequent and severe, parents worried that they might worsen again. They never became accustomed to their child having seizures. Parents considered that, even if they had not seen any positive results, they could not conclude that the surgery was to blame. The situation might have been worse if the operation had not been performed; maybe the surgeon even saved the child's life. Parents thought that it was important to receive the information that surgery was aimed to treat the epilepsy and not other disabilities, such as behavioral problems, in order to have realistic expectations. They emphasized the importance of being informed about both positive and negative outcomes and risks. Even if surgery was a medical success, the family's life situation was sometimes characterized by sorrow and problems. The deterioration, expected or unexpected, of e.g., motor function, speech, visual field, or attention made life more difficult and burdensome. Even a long time after surgery, some parents needed to keep the child under close observation all the time because of seizures. Everything circled around the child. It was difficult to have a job, making them feel stuck at home; it caused economic problems, and some depended on financial support. If the children had behavioral problems, the parents wondered if they had been caused by surgery, if they were normal teenage problems, or if they were related to neuropsychiatric disorders. Parents were disappointed if it took a long time to get a correct diagnosis. The family situation would have been less complicated if they had been told about the child's diagnosis. Tantrums affected the family most, and to avoid them, the parents sometimes tiptoed around the child. These families could isolate themselves since the child's behavior made social life difficult. However, parents did not want to describe the situation as problematic; the situation was accepted for what it was. Parents worried if their child had learning problems. It was important to find the appropriate school. Parents also worried that the child could be exploited by others. Parental health had been down-prioritized for many years. They had stress symptoms, anxiety, and sleeping problems. They worried about the child's future after their death. Some wanted siblings to take care of the ill child in the future, which was the reason why some had more children. Some children did not feel well. Their energy was affected, and they did not have the strength to do things and thus became isolated. 3.2.3. Getting excellent support Parents often experienced excellent support and information from the team when the children had the operation, helping them to handle their worries that their child would change for the worse. With good support directly after surgery, they felt comfortable to contact the team for a long time after surgery if needed, which was important and
created security. Meeting the same professionals also gave a feeling of safety. Long hospitalizations were tiresome for the families. Parents felt that the staff mostly took good care of them. They appreciated early interventions from a rehabilitation team. Rehabilitation was often beneficial, especially at the beginning. Support and assistance helped families in their life situation, and they appreciated e.g., that physiotherapists worked intensely with the child, which made them see him/her improve more than they ever thought possible. 3.2.4. Getting poor support Sometimes, parents experienced poor information and support. They wanted to know more about the future; it was not sufficient to get a telephone number to call if needed. During the long hospitalizations, some parents felt that nobody had time for them. Arrogance of some professionals made them very disappointed. They needed more support in a situation where one parent always had to be in the hospital. Parents had neglected their own needs, and it was difficult to find the time to do things together as a couple. The parents also described disappointment if rehabilitation took longer than expected from the information they had received. The initial rehabilitation was often good, but at the local level, they often felt that it was inadequate. The transfer from pediatric to adult care was sometimes difficult. In some cases, parents decided to reduce rehabilitation, albeit beneficial, because their homes had become a training area, and neither children nor parents were able to rest. It was important that the home could remain an area of rest and preferable if rehabilitation could be given at school or at the day center. The Swedish Social Insurance Agency, technical aids center, school, and municipality were criticized by the parents. Based on their experience, they felt that the bureaucracy had been problematic and had taken a great deal of time. They had to fight for their rights, despite physicians' certificates. Without specific diagnoses of comorbidities, it was even more difficult to get adequate support. They thought that it would be helpful if the authorities understood that parents only asked for help when they had reached their limit, and then, urgent help was necessary. If the seizure frequency was reduced after surgery, support from the municipality was sometimes also reduced, which had a negative effect on the family's life situation, since many problems still remained. Parents who did not feel that they received adequate support sometimes felt that all focus was on the child. They found it obvious that parents should also be offered support. 3.2.5. Feeling satisfied despite all concerns The long-term development after surgery was mostly described in positive terms. Despite seizures, disabilities, and cognitive impairments, children and parents were reported to have a good life situation. According to parents, their children enjoyed the new life situation and felt safe when they had no seizures or reduced seizure frequency. They dared do more than before; they had friends and were active, did sports, listened to music, had daily activities, sheltered jobs, or were at school. They still made progress physically and mentally and had become more independent. Children in need of surveillance by parents due to seizures were reported to be frustrated by this, even though they understood the concern. Parents also recognized that they themselves and professionals sometimes hindered the children's development and social life, for fear the child would not cope or because the child's activities were not prioritized. 4. Discussion The findings of this study illuminate parents' experiences of the family life situation before and long-term after their children's hemispherotomy. Before surgery, they lived in a chaotic bubble and
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felt that surgery was a question of life or death. After surgery, they hovered between success and disaster. They experienced both happiness and sadness, good and poor support. Despite all concerns, the family life situation significantly improved after surgery. There is a lack of long-term studies on parental experiences of family life situation and support before and after hemispherotomy. Baca et al. [18] studied parents' experiences of the journey to epilepsy surgery. They describe parents' desperation to get a diagnosis and proper treatment. Parents talked about surgery as a last resort but also that it was necessary. These findings are in accordance with our results. Also in our study, parents reported that the support and information were limited before the specialist team became involved. We also found that parents experienced varying support and information directly after the hemispherotomy and for a long time afterwards. Further, parents were stressed over the family life situation before and after surgery. These results are in accordance with earlier studies [7,22]. One study on epilepsy management found that families feel that their views on the provision of support are not adhered to [23]. Our study indicates that it is important to involve the families early in the treatment plan for them to feel safe and that the specialist team should also support the families for a long time after surgery. Parents of children with drug-resistant epilepsy have lower QoL compared with population norms [24]. In our study, the finding that parents do not see to their own needs also indicates the importance to support them in getting more time for themselves. Information could also be offered to grandparents who worry but also support the family. Family-centered care might be an alternative to make it possible to meet the family’s needs from their own perspectives [25]. Like other researchers [7,26], we found that the problematic life situation causes isolation, but in accordance with Baca et al. [18], we found that parents wanted more contact with other families in the same situation. Therefore, it might be valuable to have family support groups. We found that the children's behavioral problems cause much concern. They are common in children with epilepsy [27], but in spite of that, comorbidities were underdiagnosed, and parents needed more information before and after surgery. The behavioral problems came as a shock after surgery, and parents wondered if they had always been there. They thought that they would have managed better if they had been informed about the child's additional diagnoses at an earlier phase. That information is a prerequisite for counseling and interventions aiming at improving the psychosocial situation [28].
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Another limitation is that only three patients could be interviewed themselves, which was too low a number for a separate analysis. The majority of patients had intellectual disabilities to a degree that they could not participate in this kind of interview. 5. Conclusion The life situation before hemispherotomy was chaotic, and although the operation caused much concern, its positive effects were appreciated. Parents reported positive effects, not only on seizures but also on the family situation. It led to less worries and stress and to less isolation for the family, including the siblings. However, support and information need to be improved. Specialist teams should be involved as early as possible and follow the family after surgery. Behavioral problems need to be diagnosed before surgery so the families can get adequate support and realistic counseling. Family-centered care with specialist teams in both neurology and rehabilitation might strengthen the possibilities to meet the families' needs. Further qualitative studies on parents' and patients' life situation and experiences of medical care and support are needed to improve the care before and long-term after hemispherotomies. Acknowledgments The authors thank the steering committee of the Swedish National Epilepsy Surgery Register, all of the Swedish epilepsy surgery teams, especially the epilepsy nurses (in Gothenburg, Linköping, Lund, Stockholm, Umeå, and Uppsala), and Sahlgrenska University Hospital, Gothenburg, Sweden. The study was funded by grants from the Swedish Research Council (grant 521-2011-169); the Sahlgrenska Academy, University of Gothenburg through the LUA/ALF agreement (grant ALFGBG137431); the Margarethahem Foundation; and the Research Foundation of Queen Silvia Children's Hospital. Disclosure of conflict of interest None of the authors has any conflict of interest to disclose. We confirm that we have read the Journal's positions on issues involved in ethical publication and affirm that this report is consistent with those guidelines. Appendix A. Supplementary data
4.1. Strengths and limitations One strength of this study is that it is population-based; parents of all patients who had undergone hemispherotomy in Sweden from 1995– 2007 were invited to participate, and only a few declined participation. Another important strength is that the interviewer and the two researchers analyzing and coding data were not part of any epilepsy surgery team and had not been involved in the care of the patients. The fact that interviews were conducted at different time points after surgery might be considered a limitation. An ideal study would have been longitudinal. However, since the shortest time from surgery to interview was seven years, all interviews can be considered as long-term (i.e., ≥5 years). The answers obtained at long-term must be understood as such; not primarily as descriptive testimonies but as memories processed through emotions and colored from further experiences, which were then developed into opinions and personal views. It could be that parents whose children had a good outcome had more positive memories of the information and support than those whose children had less improvement from surgery. However, it seems reasonable that even the shortest time to interview (seven years) is enough for memories to transform into a more general sense of satisfaction or dissatisfaction. Therefore, although the varying times to interviews are suboptimal, they are not considered a serious limitation to this analysis.
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