‘No one's ever said anything about sleep’: A qualitative investigation into mothers' experiences of sleep in children with epilepsy

Sleep problems in children with epilepsy (CWE) are common. However, little is known about parental experiences and feelings about managing sleep in their CWE. To provide the most appropriate services' provision, it is essential that the lived experience of parents of this patient group and the issues and problems that they face in managing their child's sleep is understood.


| INTRODUCTION
Epilepsy is a common chronic neurological condition, characterized by recurrent seizures; the prevalence in children and adolescents under 18 years of age is estimated to be 0.32%-0.55%. 1,2 Many children with epilepsy (CWE) experience co-existing deficits in behavioural, cognitive, attention, academic and psychosocial domains, [3][4][5] as well as reduced quality of life in comparison to children without epilepsy. 6,7 Appropriate quantity and quality of sleep are crucial to healthy well-being. However, sleep and epilepsy have a complex and bidirectional relationship 8 and have been described as 'unfortunate bedfellows'. 9 In CWE, commonly reported sleep difficulties are problems around initiation (settling and falling asleep), maintenance (experiencing night or early morning wakings), duration, daytime sleepiness as well as sleep anxiety. [10][11][12] Research into sleep deprivation of parents of children with complex needs has found that one of the biggest challenges was the need to be available or vigilant during the night and that the impact of this sleep deprivation is 'relentless' and 'draining'. 13 Over two-thirds (68.6%) of parents in a previous study of parents of CWE reported having concerns about their child having night-time seizures. 14 These worries and concerns are likely to contribute to parental sleep disturbance, which is more common in parents of CWE than in parents of children without epilepsy. 15 In a sample of parents of CWE (with intractable epilepsy), 75% reported that their sleep was affected, and of these, 48% reported the impact on their sleep was stressful. 16 Traditional treatment goals for CWE tend to focus on seizure management, even though the importance of the need to develop child epilepsy care beyond 'seizure control with minimal adverse effects' has been a longstanding recommendation. 17 Yet sleep or problems around sleep are not usually an aspect of standard care that is regularly addressed, even though sleep is a key consideration reported by parents, CWE and healthcare professionals. 18,19 Behavioural and educational approaches have shown signs of being effective treatments for many of the sleep difficulties experienced by these children and their families. 20 Research suggests that there may be special considerations for managing sleep in CWE, which could helpfully be addressed to likely increase families' engagement with and feasibility of intervention. 21 A greater understanding of these considerations could helpfully direct future intervention development 22

| Participants and recruitment
Participants were nine co-habiting mothers of CWE (six boys and three girls who ranged in age from 5 to 15 years, with median = 10 years and mean = 10.3 years, SD = 2.9). See Table 1 for descriptive details about the children of the interviewees, including their epilepsy.
Children suffered from a wide range of sleep problems, which allowed mothers to share their experiences of a broad range of sleeprelated issues, see Table 2 for a summary of the children's maternalreported sleep problems.
Participants were recruited by responding to online advertisements, which were placed on the websites of epilepsy organizations and charities (e.g., Epilepsy Action), the CASTLE study and researchers' university websites. Participants were recruited between March and July 2018, with interviews taking place between March and July 2018. The criteria for participation were being the parent of a child with epilepsy, based in the United Kingdom and having sufficient English skills to partake in the interview and also willing to review a draft online sleep intervention which was part of the larger CASTLE programme of studies reported in a separate paper. 22

| Interviews
A semi-structured interview schedule was developed by the researchers that asked about key topics related to the child's sleep and relevant to the development of an online sleep intervention for a wider study (Supporting Information: 1 for full interview schedule; Cook et al. 21 and Wiggs et al. 22 for development of the sleep intervention).

| Analysis
Data were thematically analysed according to the six standardized stages outlined by Braun and Clarke. 23 Data familiarization was achieved through the reading and rereading of transcripts before the data were fully coded by a researcher not involved in the interview process (GC). This study employed an inductive analytic strategy driven by the data and participants' own words. Coding was reviewed and discussed amongst the research team, to address any discrepancies and reach agreement. A number of codes were combined following discussions, for example, the codes 'frequent overnight awakenings' and 'night waking issues' contained similar content and were combined.
Other codes that did not relate to mothers' perceptions or experiences around their child's sleep were set aside. During the 'search for themes' stage, one of the researchers (GC) reviewed the codes and clustered them into potential themes and subthemes. Next, coded extracts of raw data were revisited, and the themes/subthemes reviewed across the whole data set to ensure that they comprised an accurate reflection of the transcripts. During the 'defining and naming themes' stage, the research team reviewed the proposed analysis and refined the specific details of themes, as well as agreed names and descriptions. This involved ongoing discussion and iterative amendments until agreement was reached. The final set of themes and subthemes were agreed upon between all authors.

| Procedure
Once parents had expressed an interest (through responding to online adverts), they were provided with an information sheet and given an opportunity to ask any questions. Eligible participants who wished to participate were required to complete a consent form.
Interviews were then completed by two researchers (PG and LW) at a time and in a format convenient for participants (two face-to-face, six by telephone and one via video call). Interviews were audio recorded, transcribed verbatim and thematically analysed.

| RESULTS
Interviews lasted, on average, 61 min. Themes are presented in line with the study's research questions; four represent the nature of maternally reported sleep problems; seven (and associated subthemes) represent mothers' experiences and feelings around managing their child's sleep and any associated problems and one relates to mothers' perceptions of the available help and support to them around sleep. Themes (and subtheme(s) where appropriate) are presented and described below, with detailed illustrative quotations appearing in Table 3.   a C-Current sleep problems refer to problems which are currently present, but it should be noted that the duration of these problems is generally longstanding (since infancy or beyond), with most problems also being also present in the past. P-Some parents also reported past sleep problems, which relate to issues that have now been resolved but which were significant problems at some point.
T A B L E 3 Summary of themes and subthemes supported by detailed quotations Research question 1: Parentally reported sleep problems faced by CWE and their families Theme: Settling issues 'She had trouble falling asleep, so falling asleep sometimes lasted more than an hour to make her fall asleep' (P5). 'He used to just, like, when we put him to bed it would be, like, 2 hours or so afterwards before he'd finally managed to drop off to sleep. Yeah, he really used to struggle to get to sleep and anything like that…' (P4). 'If he can't sleep he's up down, annoying his brother, in and out of his sisters room, up and down the stairs … wandering around the house making a noise' (P6). '…it got to the point, even at that age, where you dreaded bedtime with her. You were, like, oh god it's bedtime. It just came over you like a black cloud, your heart sunk, we both looked at each other, like, it's bedtime' (P7).
Theme: Night-waking issues 'He would go to bed fine and go to sleep fine but it's just in the middle of the night when he wakes up … it would be that bit that would cause the problem for us' (P1). 'So whether they were seizures waking him up or whether he was just not falling to sleep properly, not going into deep sleep and he was just dozing' (P2). 'I'm not sure if they were seizures waking up, because obviously he has quite a lot of seizures.
So whether they were seizures waking him up or whether he was just not falling to sleep properly, not going into deep sleep and he was just dozing' (P2). 'It's just the amount of times he wakes up and gets up in the night and unless you sit and watch him all night I don't know how you would prevent him getting up all night' (P6). 'He'd go to sleep at sort of 8.30/9 and then wake up at between 7 and 8 so he was, but he would always in the night he would wake up and come into us and we'd have to go and put him back' (P1).
Theme: Parasomnias 'Well he's 11 this November probably from the age of 7 he's suffered with night terrors intermittently. So we had a few episodes of that and then he did a bit of sleepwalking and he's never since sort of being the age of 7 years old he's never really fully settled at night time' (P1). 'But she very much still every night still shouting 'no, no, no, no' and shouting out … During her sleep yeah, she has a recurrent dream … when she's having the dream and she was shouting "mum, mum" and I'm like "I'm here"' (P3). 'Sometimes it was very difficult to tell if he was just wandering around because he didn't want to go to sleep because he was anxious or he'd had a seizure or he was literally sleep walking … he was asleep, so he did sleep walk quite a lot' (P2).
Theme: Child anxiety around sleep 'He's always been quite a nervous sleeper you know he's always had to have a light on and things' (P1). 'He was having a lot of difficulty sleeping because he was worried that if he went to sleep, because we had quite a few instances where he went to bed as normal and when he woke up he was in hospital. Because sometime in that night he'd had a tonic clonic and we had to call an ambulance because his tonic clonics tend to be about 10 minutes long and his oxygen levels drop, so we have to call an ambulance. So he did go through a stage of not wanting to go to sleep and I think I wrote in there that he had a massive fear that he was going to die in his sleep. So I think that's, it's not just the seizures it's the emotional side of it as well' (P2).

Research question 2: Parents' experiences and feelings around managing their child's sleep and any associated problems
Theme: Longstanding challenging nature of child sleep issues 'I said to him yesterday how's your sleep and he said to be honest I don't think I've slept through the night for the last 6 months' (P2). 'He used to just be saying I can't sleep mum I can't sleep all the time and you, it's just awful when they want to sleep but they can't get to sleep' (P4). 'It's so draining for her and us really, you know. Because when she's awake she's like Bagpuss the whole house is awake. So she's got a little sister so then she's awake because [child's] awake … It affects everyone' (P7).
Theme: Management strategies for child sleep 'He's never slept and we've gone through lots of different things, of early to bed, late to bed, exercise before bed, everything' (P2).
Subtheme: Co-sleeping 'Practical wise it was easier for him to be in with us because he is there all night. Rather than sort of lying there uncomfortably with my arm up in the air trying to hold his hand and then creeping out of the room half an hour later' (P2).
'Ever since he had his first seizure he co-sleeps with his dad because we don't feel comfortable to let him sleep on his own … we'd just be up all night checking on him … it's us more than him' (P1). 'But the main thing was we all slept together … I think I was probably 50/50 us wanting to sleep with him as well as him wanting to sleep with us' (P2). 'Well he [child] slept in my bed and my husband slept on the floor in our room bless him, on and off for about 3 years' (P2). '…we have to deal what we're living with at the minute and just get on with it and as long as we all get sleep … however we feel, we just feel a lot calmer and more settled if one of us is with him' (P1).
Subtheme: Sleep environment adaptations 'It's petrifying isn't it if you wake up and it's pitch black and you're not sure, so he did have various night lights throughout the years, until he was a bit older and now he's got a sort of electric one he can flick on when he wants to by the side of the bed' (P2). 'We had one of those safety pillows and I think that's a massive help and it's a massive reassurance for [child] and for parents that you've got one of these safety pillows. That if they do have a tonic clonic face down … they can breathe through the pillow, yes I think that's a massive thing' (P2). 'The early nights and everything that we're doing, we've got we've bought black out curtains, we've bought well 2 sets of black out blinds and curtains, we have a fan going, we have all these kind of little interventions that we've put in place just anecdotal' (P3). 'Now whether or not that [blackout blinds] cancels out the noise of the rest of the just the general household as well. Because then her brother will be going to bed an hour later. You know there's just those things that I'm very conscious of might wake her up again' (P3).
Subtheme: Use of monitoring devices 'He had a couple of tonic clonics in the middle of the night, which we did pick up on because I had this video monitor and you know I had like an alarm and everything … I have that on my phone pointed at a video monitor. So that if he moves on the video monitor even if I don't hear it, it'll pick up the movement as well and sound an alarm, so we did beef up on the seizures' (P4). 'Once I got all the monitors setup and everything, I was kind of happy that I wasn't going to miss any [seizures], if he did have any' (P4). 'We have monitors as well and I think which have given her some security. I think they help her to go to sleep knowing that she has a monitor … She feels less anxious because she's got a bed monitor and she's got a room like a video monitor on' (P3). 'Obviously it's movement [that is monitored by the device] and he's a bit of a rough sleeper so that goes off quite often … I just check on the video monitor and if I can see he's just turning over or doing a normal movement then I know he's fine. If I can't see him properly on the monitor then I'll just pop in and make sure he's alright' (P4). 'My problem with the seizure detection monitor is that I've not seen it work, it's not proved itself to me because we've only had it 2 weeks and she hasn't had that kind of seizure' (P3).
Theme: Challenges related to managing sleep over time 'It's very hard if she's resistant to want to go bed … because of your seizures. You don't want to bring it into that just before you go to bed. But for an older child they've got to take ownership of it a little bit' (P4). 'We have had relaxation DVDs in the past, before she was diagnosed with epilepsy … They were kind of like a story that went into relaxation … We did use those for a good while … but then she outgrew them' (P3).
Theme: Link between sleep and seizures '…a vicious circle because the seizures were caused by lack of sleep and … they're getting stressed because they're not getting enough' (P4). 'I know for a fact that if we put him back in his own bed he would be waking up in the night and then obviously we're told that maybe one of the causes of the seizure is lack of sleep and tiredness so we don't want him to, we're scared if he goes back in his own bed and he's waking up all the time is it going to bring on a seizure' (P1). 'I was kind of neurotic with trying to make sure that he was getting enough sleep. Because I knew that that was a major trigger for his seizures, because it was when he was tired, he always had more' (P4).
Theme: Negative impact of poor sleep on daytime functioning 'Sleep obviously has a massive effect on the way he functions during the day' (P2). 'If he's been up then he really does suffer the next day at school, whether that's a knock on effect that he's tired so he has more seizures and its just a constant knock on effect' (P2). 'He doesn't do well without sleep and I think that's the other big thing, he does need a lot of sleep and again I don't know whether that's because he wakes numerous times or whether he's a teenager' (P2).
sleep which was problematic, such as, '2 hours or so afterwards before he'd finally managed to drop off to sleep' (P4).
In some families, the CWE settling issues negatively influenced the wider family, 'annoying his brother, in and out of his sister's room…' (P6).
For some, the extent of settling issues influenced their feelings and experiences around putting their child to bed, '…you dreaded bedtime with her…' (P7). Settling issues were problematic for mothers, families and CWE but were one of the most commonly reported sleep problems.

| Night-waking issues
Mothers also commonly reported issues around night wakings, '…it's just Parasomnias could be longstanding and challenging for parents and children to manage.

| Child anxiety around sleep
A difficulty faced by many mothers was their child's sleep-related worries, concerns or anxieties, 'He's always been quite a nervous sleeper' (P1). Some mothers highlighted that the child's epilepsy and Theme: Antiseizure medication 'I mean she still wakes up but since we, I've noticed improvements since we started medicating, that's also the truth' (P5). 'We finally found a medicine that stopped the tonic clonics and him just having the partial focals that then I think that that was the turning point that it seemed to be much easier to be much easier to cope with that we knew he wasn't in his bedroom having tonic clonics on his own' (P2) 'Because we go to clinic and they say she needs to sleep because you know the better the sleep the less seizures but then they go but she won't sleep because of the condition and the medicine. Side effect is it won't, she won't sleep' (P7).
Theme: Maternal concerns about child sleep 'I think it's just that now because at the point of diagnosis they said this is you know the one thing you can do. It feels more pressured' (P3).

Research question 3: Parents' perception of available help and support when parenting a CWE around sleep
Theme: Lack of information, help and support available 'I think even in all the pamphlets and leaflets and whatever we've been given over the years, nobody's ever mentioned sleep' (P2). 'Sleep is the one thing that we can do, help but then not really much assistance comes along with that at that point of diagnosis' (P3). 'His consultant gives you all sorts of stupid advice and you're like really, "don't let him drink coffee" and I'm like he's 9 I don't let him drink coffee anyway' (P6). '…we are given nothing in hospital, you know or when we've been to A&E, there's nothing that you are told, as I say all we were told at that point is you must get good sleep and so it would be great if somebody then said and here have a look at this … and I would have come straight home and gone straight onto it' (P3).
COOK ET AL.
| 699 the possibility of sleep-related seizures was a source of anxiety for their child, '…he had a massive fear that he was going to die in his sleep' (P2).
For some mothers, the challenge was managing a calm and relaxing bedtime when child anxiety was most commonly expressed or shared at this time, 'That's usually when it comes out [child's anxieties or worries] … just when we're doing the bedtime' (P3).
A key factor in dealing with sleep in CWE was managing the child's worries that could surface around sleep or the anxieties the child had about sleep due to epilepsy. In some cases, it was not just the mothers, but also the children, that acknowledged their disturbed sleep 'He said to be honest I don't think I've slept through the night for the last 6 months' (P2). For many mothers, the sleep problems experienced by their CWE negatively impacted the whole family, 'It's so draining for her and us … It affects everyone' (P7). Overall, the evidence in this theme suggested that sleep problems were common and longstanding, and their influence was negative for the CWE, their parents and the wider family unit.

| Management strategies for child sleep
Many mothers reported adopting certain approaches or practices to facilitate what they perceived to be safe sleep for their children.
These were broad and varied in nature and encapsulated within three subthemes.

Co-sleeping
Some mothers found advantages of co-sleeping (bed and/or room sharing), 'Practical wise it was easier for him to be in with us because he is there all night' (P2). For some parents, a key motivation for cosleeping was so that they could monitor their child and their safety overnight, which allayed some parental concerns, '…we don't feel comfortable to let him sleep on his own' (P1).
In other cases, co-sleeping was beneficial as having parents nearby helped to alleviate some of their child's fears or worries, 'I There was also a range of issues and concerns about the use of these devices. These were around them going off unnecessarily, Some mothers had reservations about the use of these methods as they did not feel these methods were acceptable for their child, '…kind of feels like an invasion of privacy' (P6). Another concern for some was the reliability and efficacy of monitoring devices, 'It's not proved itself to me' (P3).
Monitoring devices were relatively widely used and appeared to offer highly sought-after reassurance to both parent and child that seizures would not be missed. However, these systems had limitations for some families.

| Challenges related to managing sleep over time
The majority of mothers reported that many of the challenges in managing their child's sleep changed over time.
As children became older, they were offered more independence, 'I can't honestly say whether he was a lot better at sleeping or he was more independent at sorting himself out' (P2). Mothers also acknowledged that as their child aged and developed, the child needed to take additional responsibility for their epilepsy, '…for an older child they've got to take ownership of it a little bit' (P4).
The type, success and suitability of parental management techniques also evolved over time, 'We are very conscious that as  Mothers' experiences or concerns around false alarms are also reiterated in the literature in the context of challenges around sensitivity and specificity around these devices. 29 There is currently no individual seizure-monitoring device, which is effective for detecting all types of seizures in adults or children. 30 In addition, a topic of key concern to parents is Sudden Unexpected Death in Epilepsy (SUDEP), 31 yet no devices have been demonstrated to prevent SUDEP. 32  The current findings need to be considered in the context of some limitations. The sample size (n = 9) was smaller than intended the data collection (e.g., following up with questioning on specific sleep issues or problems) and in the analysis of the data (e.g., identifying codes informed by our existing knowledge). However, every attempt was made to foreground parents' words and experiences in the data collection and analysis.

| CONCLUSION
To provide the most appropriate services and support for sleep issues in this patient group, it is essential that the lived experience of parents and the issues and problems that they face in managing their

AUTHOR CONTRIBUTIONS
All authors contributed to the conception and design, acquisition of data, analysis and interpretation of data; and drafting the article or revising it critically for important intellectual content.