“A journey around the world”: Parent narratives of the journey to pediatric resective epilepsy surgery and beyond

Abstract

OBJECTIVE

Although shorter time to pediatric resective epilepsy surgery is strongly associated with greater disease severity, other non-clinical diagnostic and sociodemographic factors also play a role. We aimed to examine parent-reported barriers to timely receipt of pediatric epilepsy surgery.

METHODS

We conducted 37 interviews of parents of children who previously had resective epilepsy surgery at UCLA (2006–2011). Interviews were audio-recorded, transcribed and systematically coded using thematic analysis by two independent coders and subsequently checked for agreement. Clinical data, including `time to surgery' (age of epilepsy onset to surgery) were abstracted from medical records.

RESULTS

The mean time to surgery was 5.3 years (SD=3.8); surgery types included 32% hemispherectomy, 43% lobar/focal, 24% multilobar. At surgery, parents were on average 38.4 years (SD=6.6) and children were 8.2 years (SD=4.7). The more arduous and longer aspect of the journey to surgery was perceived by parents to be experienced prior to presurgical referral. The time from second anti-epileptic drug failure to presurgical referral was ≥1 year in 64% of children. Thematic analysis revealed four themes (with subthemes) along the journey to surgery and beyond: (1) recognition: “something is wrong” (unfamiliarity with epilepsy, identification of medical emergency), (2) searching and finding: “a circuitous journey” (information seeking, finding the right doctors, multiple medications, insurance obstacles, parental stress), (3) surgery is a viable option: “the right spot” (surgery as last resort, surgery as best option, hoping for candidacy), and (4) life now: “we took the steps we needed to” (a new life, giving back).

SIGNIFICANCE

Multi-pronged interventions targeting parent-, provider- and system-based barriers should focus on the critical presurgical referral period; such interventions are needed to remediate delays and improve access to subspecialty care for children with medically refractory epilepsy and potentially eligible for surgery.

INTRODUCTION

In appropriately selected children, resective epilepsy surgery is an efficacious and cost-effective treatment with the potential to eliminate and reduce seizure burden, avert developmental regression and improve quality of life outcomes.^1–5 Despite the accumulating evidence of the benefits of surgery over time and the 2006 International League Against Epilepsy published referral recommendations for epilepsy surgery in children, a substantial proportion of children fail to receive this care in a timely manner.⁶ Epilepsy duration prior to pediatric resective epilepsy surgery is still on average 5–6 years with substantial variability.^7–9 A significant proportion of children in need of epilepsy surgery – particularly those with seizure onset before age two – experience daily seizures and are at risk for epileptic encephalopathy, making early surgical intervention critical.^6,8 In children, shorter times to surgery have been associated with better seizure and developmental outcomes.^2,10–12

While several studies have examined physician and patient-based barriers to adult epilepsy surgery,^13–23 few have examined in-depth the factors that mediate delays in children^7,17,24–26 and none have assessed perspectives of parents of children who have undergone epilepsy surgery. We previously found that although shorter time to pediatric resective epilepsy surgery is most strongly associated with greater disease severity (history of infantile spasms, daily seizures), other non-clinical diagnostic and sociodemographic factors also play a role.⁷ In follow-up, we aimed to identify the nature and range of parent-perceived barriers to timely receipt of pediatric epilepsy surgery.

METHODS

We chose a qualitative methodology to obtain a full range and in-depth assessment of barriers and perceptions in a heterogeneous sample of parents of children who had prior resective epilepsy surgery.²⁷

Sample selection & recruitment

We recruited and interviewed a sample of parents of children who had previously undergone resective epilepsy surgery at UCLA. Inclusion criteria included: (1) parent/guardian of child that had resective epilepsy surgery (<18 years; 2006–2011), (2) English or Spanish speaking and (3) ability to provide verbal informed consent. Parents were recruited purposively oversampling those with longer times to surgery in order to assess barriers to timely epilepsy surgery.

Potential participants (N=146) were sent an informational invitation letter from the study investigators (CB, GM). A pre-stamped “opt-out” return card was included if they did not wish to be contacted further (only 1 potential participant returned the opt-out card). Those interested in participation could return a pre-stamped card to the study team and the research assistant (RA) then called them to schedule an interview. The RA also called non-responding potential participants two weeks after the mailing to describe the study in more detail and invite them to participate. We recruited a final sample of 37 individual parents (not dyads), at which point data saturation had been achieved.²⁸

Measures and Data Sources

Clinical characteristics of children

Clinical data (date of epilepsy onset and surgery, seizure frequency before surgery, type of surgery, presurgical history of infantile spasms, number of anti-epileptic drugs (AEDs) at surgery and etiology) were obtained by abstraction of medical records. Details regarding the standardized pre-surgical evaluation protocols, surgical procedures and clinical variables have been previously described.^1,2,7 `Time to surgery' was calculated as the interval from date of epilepsy onset to epilepsy surgery.

Parental sociodemographic and baseline information

A trained RA or the PI (CB) first asked parents structured questions regarding parental sociodemographic characteristics (age, gender, race/ethnicity, education level, employment status, marital status, primary language), medical insurance of their child (at surgery), physician specialty type involved in care of child (from seizure onset, diagnosis, and presurgical referral for Video-EEG monitoring), timing of epilepsy care (from seizure onset, diagnosis, two AED failure to presurgical referral), information sources for presurgical referral to UCLA, whether their child had received epilepsy specialty care prior to UCLA and whether their child had experienced any seizure remission periods.

Semi-structured interviews

Based on clinical experience and published research, study investigators constructed a semi-structured interview guide (Supplemental Table 1) to elicit parent perspectives on access and barriers to care and surgery in relationship to the timeline of seizure onset, epilepsy diagnosis, two AED treatment failure, presurgical referral, and surgery. To capture a range of responses, questions were open-ended; directed and non-directed prompts were used to encourage discussion.²⁷

Data Collection

The PI (CB) or a trained bilingual (English/Spanish) RA conducted interviews via telephone. Participants were mailed a $30 gift card. The average duration of the 37 interviews was 29 minutes (range 10–60 minutes).

Analysis

Baseline characteristics and clinical data were analyzed using descriptive statistics. Audiotaped semi-structured interviews were transcribed verbatim, translated (Spanish to English, if applicable) and de-identified. A three-person analysis team (CB, HP, TI) then systematically analyzed the transcripts using thematic analysis.²⁹ Two members of the team (CB, TI) read each interview to familiarize themselves with the data and then independently assigned initial codes to sections of text in each interview. Coding was reviewed and discrepancies were discussed until agreement was reached. All codes were maintained in a codebook (Microsoft Excel version 14.4.2). Once initial coding was completed for the majority of the interviews, the analysis team sorted the initial codes into the most salient themes and subthemes identified as frequent and meaningful across interviews.²⁹ Diagramming was used as a tool to summarize major themes and subthemes.²⁹

Standard Protocol Approvals, Registrations, and Patient Consents

The UCLA Institutional Review Board approved this study. Parents signed research informed consents and Health Insurance Portability and Accountability Act authorizations allowing ongoing participation in the pediatric epilepsy surgery database and recruitment for this study. Parent participants provided verbal consent via phone prior to the interview. This study was not a clinical trial.

RESULTS

Sample characteristics

Sample characteristics (N=37 individual parents) are shown in Table 1. There was high agreement for the `time to surgery' between the medical chart and parent-reports (ICC = 0.96, 95% CI (0.92, 0.98); the mean `time to surgery' by medical chart was 5.4 years (SD=3.8) and by parent-report was 5.1 years (SD=3.7). The majority (70%) of parents did not know that their child was having an epileptic seizure that first time (Table 1). At seizure onset, over half (57%) of children were seen by a primary care or emergency medicine doctor, while 41% were seen by a pediatric neurologist or epileptologist (Table 2). While epilepsy was diagnosed by a pediatric neurologist (or epileptologist) in most children (95%), nearly one-third (30%) were referred for presurgical evaluation by a non-neurologist (n=8) or were self-referred (n=3). The time from seizure onset to epilepsy diagnosis was over 1 month in 40% of children, and the time from the second AED failure to presurgical referral was over 1 year in nearly two-thirds (64%) of children (Table 3).

Table 1.

Sociodemographic and clinical characteristics (N=37; surgery 2006–2011)

	Mean (SD) or N (%)
Child age at epilepsy onset, years*	2.7 (3.5)
Child age at surgery, years*	8.2 (4.7)
Time to surgery (epilepsy onset to surgery), years*	5.4 (3.8)
Child gender, % male*	17 (46.0)
History of infantile spasms*	15 (40.5)
≥ Daily Seizure frequency*	25 (69.4)
Number of AEDs taking at time of presurgical evaluation*
1 AED	7 (18.9)
2 AEDs	13 (35.1)
≥ 3 AEDs	17 (45.9)
Operation Type*
Hemispherectomy	12 (32.4)
Lobar/Focal	16 (43.2)
Multilobar	9 (24.3)
Etiology*
Cortical Dysplasia	16 (43.2)
Hemimegalencephaly	3 (8.1)
Infarction	8 (21.6)
Tuberous Sclerosis Complex	6 (16.2)
Tumor	4 (10.8)
Parent age at interview, years	41.3 (7.3)
Parent age at child's surgery, years	38.4 (6.6)
Parent, % mothers	31 (83.8)
Parent race/ethnicity
Caucasian	25 (67.6)
Hispanic	11 (29.7)
Pacific Islander	1 (2.7)
Parent primary language English	31 (83.8)
Parent born in United States	28 (75.7)
Parent married (at time of child's surgery)	34 (91.9)
Parent employment (at time of child's surgery)
Full- or part-time	20 (54.1)
Homemaker	15 (40.5)
Unemployed	2 (5.4)
Parent level of education (at time of child's surgery)
≤ High school/GED	7 (18.9)
Vocational School	10 (27.0)
≥ 4 year college	20 (54.1)
Insurance at time of epilepsy surgery
Preferred Provider Organization	21 (56.8)
Health Maintenance Organization	3 (8.1)
Fee for Service	2 (5.4)
Medicaid /California Children's Service	8 (21.6)
Tricare	3 (8.1)
Epilepsy specialty care prior to UCLA referral? % yes	29 (78.4)
How heard about UCLA Pediatric Epilepsy Surgery Program**
Referring physician	26 (70.3)
Friends/family	3 (8.1)
Internet	10 (27.0)
Epilepsy support groups	1 (2.7)
Other	4 (10.8)
Parent knew child was having seizure (that 1st time)	11 (29.7)

^*data from medical chart abstraction;

^**1 missing

Table 2.

Physician Types at Different Pediatric Epilepsy Care Points

Physician Type	Seizure Onset	Epilepsy Diagnosis	Presurgical Referral
Pediatric Epileptologist	8 (21.6)	21 (56.8)	14 (37.8)
Pediatric Neurologist	7 (18.9)	14 (37.8)	10 (27.0)
pediatrician/Family Medicine	13 (35.1)	1 (2.7)	8 (21.6)
Emergency Medicine	8 (21.6)	1 (2.7)	-
Adult Neurologist	-	-	1 (2.7)
Neurosurgeon	-	-	1 (2.7)
Self-referred	-	-	3 (8.1)
Other	1 (2.7)

Table 3.

Timing of Pediatric Epilepsy Care

Time to surgery (epilepsy onset to surgery), years, mean (SD); range*	5.4 (3.8); 0.5–14.7
Time from seizure onset to epilepsy diagnosis#
< 1 week	12 (32.4)
≥ 1 week but < 1 month	10 (27.0)
≥ 1 month but < 1 year	8 (21.6)
≥ 1 year	7 (18.9)
Time from 2nd AED failure to referral to epilepsy center#Φ
< 1 month	6 (16.7)
≥ 1 month but < 1 year	7 (19.4)
≥ 1 year but < 3 years	12 (33.3)
≥ 3 years	11 (30.6)
Any period of extended seizure freedom prior to referral#
No period of remission ≥ 6 months	10 (73.0)
≥ 6 months but < 1 year	2 (5.4)
≥ 1 year but < 2 years	5 (13.5)
≥ 2 years	3 (8.1)

^*data from medical chart abstraction (`time to surgery' parent-report 5.1 years, SD=3.7); high agreement for time to surgery between medical chart and parent report (ICC = 0.96, 95% CI (0.92, 0.98))

^#data from parent-report

^Φ1 missing

Parent Narratives: Themes Along the Journey to Pediatric Epilepsy Surgery and Beyond

Four interrelated themes with associated subthemes were identified along the journey to surgery and beyond (Table 4; Figure 1): (1) Recognition, (2) Searching and finding, (3) Surgery becomes a viable option, and (4) Life now. At initial onset of seizures, parents were scared and described a sense of urgency to uncover what was happening to their child. Parents depicted a pathway filled with multiple doctors and treatments, insurance battles, and work and life obstacles looking for a cure for their child's seizures, becoming ever more knowledgeable about epilepsy. Once parents found the right doctor and were given the option to pursue epilepsy surgery, the once frightening prospect of brain surgery became a source of hope. Whether this journey took only months or many years, parents expressed that the circuitous journey to surgery felt long due to all the ups and downs and unknowns of the journey. As one parent said, “I wish it was a more direct flight rather than a journey around the world and very circuitous.”

Table 4.

Parent-reported themes along the journey to pediatric resective epilepsy surgery: Themes, subthemes and exemplars (N=37)

Themes	Subthemes	Exemplars
“Something is wrong”: Recognition	Didn't know it was a seizure-unfamiliar with epilepsy	• “My mom's cousin had grand mal seizures, so when he had a grand mal seizure, I knew… I honestly had no idea of all the other little seizures that they can have…”
		• “I mean the normal person knows a seizure as somebody convulsing, the grand mal type seizure, but she doesn't have that type of seizure. When it first happened we thought she was just very very sick from the stomach flu… I was under the assumption that there's only one kind of seizure.”
	Recognition of emergency or medical urgency	• “We were all scared, and we needed to go, and take her to the ER, and get some help, and find out what is going on.”
		• “My first reaction was to call the pediatrician in a state of panic…”
”A circuitous journey”: Searching and finding	Information seeking–learning the language of epilepsy	• “I mean, you've heard of epilepsy, but – so it was pretty much an avalanche of information coming at us. So we didn't in the beginning have a clue… It wasn't that easy to find information….”
		• “There's a whole lot of words that neurologists and a hemispherologist use when – and in the beginning you're like what in the – what are you talking about. So, eventually when you start understanding the terminology and you start understanding the treatments, and you start understanding the medication side effects and all the other stuff and then it becomes a little bit easier”
		• “I firmly believe that the more education you have as a parent…the more educated questions you can ask your doctor”
		• “When you suddenly have a child with a serious disability that comes out of left field that you don't know anything about, there's a lot to learn.”
	Doctors – not enough, too many, finding the right one	• “We'd been bouncing around hospitals for four or five months …Not that care was poor – don't get me wrong – but there's a point where it surpasses the local specialty.”
		• “Every doctor has their own idea about what the deal is”
		• “I don't believe that doctor really knew what she was doing… By having doctors who were experienced, who knew what they were dealing with, and who knew and had a plan of attack in place. I felt that the XXX, when we were there, there was no plan, there was no guidance…”
		• “She again said that he was doing it for attention and that we needed to go to see the behavioral clinic doctor.”
		• “… and so one doctor might see something, but another doctor is gonna see something completely different. It's not black and white. As much as they know, they don't know a lot more.”
	Many and more medications	• “… and all the medications we were giving her were not controlling them and they weren't stopping. We were just upping, taking it down, and putting her on a different one which wasn't controlled.”
		• “When the medications continued to fail and we were on No. 4 and No. 5 and none of it was working, her seizures were getting worse, her behaviors were worse, there were side effects. She would lose short term memory. She wasn't learning. Some days all she did was seize and sleep…It was the medications. We kept trying more medications. Nothing was helping our daughter.”
	Out of network or treatment and testing denied – insurance obstacles	• “I feel that her medical insurance, particularly when we had the HMO, made it very difficult to get in to see doctors within a reasonable amount of time because we had to get an authorization. It wasn't just a phone call, it was four phone calls, continuous follow-ups…”
		• “We had quite a fight with the insurance to get the MEG approved, and actually because of that fight, our insurance changed our protocol concerning MEG and epilepsy.”
		• “It took six months just to get a consultation approved through the insurance… And since XXX Medicaid refused to pay, XXXX would no longer accept the insurance, and I would have to pay out-of pocket in order to have surgery. And I am not a billionaire. I can't pay for brain surgery out of pocket.”
	Being on call 24/7 – parental stress	• “…I was at his side in the hospital for the majority of his life, so my marriage, my home, it was difficult”
		• “… because I don't have a life besides taking her to her doctor's. I could never actually have a job or do anything and take care of my daughter.”
		• “I mean family and friends are very supportive, but in the workplace they're not. I got fired from too many jobs. I lost too many jobs, and I was actually forced to stop working…. There was no balance. It was impossible to keep a job”
		• “You feel alone because you just don't have other friends that have kids who have had brain surgeries necessarily.”
“ The right spot”: Surgery becomes a viable option	Surgery as scary and treatment of last resort only	• “You hear surgery, but it's always considered a last resort option. I think like a lot of families, we kind of just wrote surgery off. We just continued meds.”
		• “I think sometimes with brain surgery, it's scary for a lot of people. The brain is one of the most major organs in the body, and the idea that you as a parent have to say yes or no to them cutting out a portion of your kid's brain, what does that really mean for your child?”
		• “When you say that you're going to cut someone's –half of their brain out – it sounds very extreme, which it is… It sounded like science fiction. I remember thinking there was no way I would ever do that to my child. It seemed insane.”
		• “Well in the beginning when she was diagnosed the neurologist at XXX thought that she would outgrow the seizures, so from age 4 until around 11 or 12 we thought she'd outgrow them. And we weren't about to do a radical surgery with the hopes that she would have been cured by age 12.”
		• “I think part of my son's problem is that he did go two years without a seizure so I think we were hopeful that he had outgrown it or something.”
	Epilepsy is more scary than surgery	• “Because there are people who will tell you scary things about the surgery; I don't have scary things to tell you about the surgery, I have more scary things to tell you about the epilepsy.”
		• “Either she was going to die or we were going to make an effort to do something about it…You need to make an informed decision if you choose not to have surgery. Everything has its risks.”
	Surgery as best option - hoping for surgical candidacy	• “It was more like, just hoping that he was a candidate.”
		• “But until you find a doctor who thinks a little bit more outside the box and you know, explains the fact that surgical, epilepsy surgery is not a last resort, but it is often the best resort, once you hear that, it's kind of very clear that you need to go talk [laughs] to a surgeon.”
“We took the steps we needed to”: Life now	A new life – for child and parent	• “Because her life is, and our life is so much easier without the seizures…”
		• “It's just an amazing – it was, you know, it was an anxiety-ridden process, but it has been the best decision of our entire life for her. She is a changed person.”
	Wanting to give back – importance of shared experience	• “Share your story so that other people can, you know, hopefully be helped.”
		• “Once my child had surgery someone called us and said, `I heard your daughter had the surgery.' So my husband and I actually went and met with those parents and told them our experiences. And then my child met with them as well. …It was just any trial that you have, if somebody's been through it before, it's encouraging to hear their story.”
		• “Find a group of people…reach out to other people so that you have someone who has been there, someone who can understand you, someone that you can call and have that support, because it is different from a family member or a friend that really doesn't get it.”
		• “Now, I'm the biggest proponent for epilepsy surgery. I have a friend in the community who I'm trying to convince to go to XXX right now and just do the surgery.
		• We found some afterward (support groups), but there wasn't really any when we were doing this, when we started going through it.”

Figure 1.

Parent narratives of the journey to pediatric epilepsy surgery and beyond

The more arduous and “longer” aspect of the journey to surgery was largely perceived to be experienced prior to the presurgical evaluation, or prior to referral to the epilepsy center. “It was a long time only because nobody referred us. But once we found an epilepsy specialist that would refer us, it was very quick,” stated a parent. Once referred, parents felt that the path was more direct and less arduous. As one parent recounted, “We met Dr. X (pediatric neurologist). She said, `I'm going to address the seizures as best I can while I'm getting you evaluated for pediatric neurosurgery.' And it was a straight line from there.”

THEME 1. RECOGNITION: “Something is wrong”

The initial onset of seizures was often a frightening experience, with many parents describing a feeling of desperation to get a diagnosis and proper treatment.

Unfamiliarity with Epilepsy

At the time of initial seizure onset the majority of parents reported that they were unfamiliar with seizures and how they could manifest. Many parents mistook their child's initial seizure for something else – including, for example, “daydreaming”, “the flu”, “hiccups”, or “food poisoning because it looked like she was choking.” While some parents reported some prior familiarity with seizures, most parents' conception of a seizure was that of a motor convulsion exclusively.

Recognition of Medical Emergency or Urgency

Despite not recognizing their child's initial epileptic seizure as such, parents knew that something was wrong and that they needed to seek medical attention urgently. As one mother expressed, “I didn't know what was going on and I had no idea. I just knew it was an emergency. So I called 911.”

THEME 2. SEARCHING AND FINDING: “A journey around the world and very circuitous”

With the recognition that “something is wrong” parents sought information from multiple sources in an attempt to understand seizures and epilepsy and become better advocates for their child. Through this journey, parents learned to navigate a complicated medical system filled with doctors of different specialties, multiple hospitals, insurance approvals, and numerous treatment options. Parents traversed all of this within the context of their daily lives, having to juggle life responsibilities such as other children and work, in addition to caring for a sick child.

Information Seeking

Parents reported difficulties as they sought information about seizures, epilepsy, medications, and surgery. Parents also reported little initial familiarity with epilepsy and that they felt overwhelmed trying to learn a new language of epilepsy because of the “avalanche of information coming at us.” Parents sought information from a variety of disparate sources including physicians, the Internet, books and family/friends. As they acquired more epilepsy knowledge, however, many parents felt more empowered and able to ask questions and participate more effectively in the care of their child. A mother who stated, “I just think knowledge is power and it also brings some comfort to making a good decision”, exemplified the sense of empowerment associated with gaining fluency in the language of epilepsy.

Finding the Right Doctor

Parents expressed that their children were seen, evaluated and cared for by a variety of doctors of different specialties throughout their journey. Parents frequently described difficulty finding the “right” doctor(s). Difficulty was associated with a lack of pediatric neurologists in their local area and with different doctors having different recommendations. Several parents experienced feeling doubted by a doctor; some were told that nothing was wrong with their child initially. As one mother revealed, “Initially the first thing she [doctor] thought that I was crazy. Well, she thought I was a little hypochondriac or something. I'm thinking, well, four kids, I'm not here that often. I don't think that's the case.” The journey to finding the right doctor was further complicated by parental perceptions that doctors sometimes lacked knowledge or familiarity of epilepsy. Many parents went to numerous doctors searching for the “right” doctor, or one who had a strong epilepsy knowledge base, could effectively identify the problem, and then make a clear plan of action.

Many Medications

The majority of parents reported that their children tried many AEDs. AEDs were often associated with no improvement in seizure control and numerous adverse effects. Parents frequently described that medications trials continued often with little to no mention of surgery. One mother told of her frustrations with multiple medications, “It was, `Let's work on all the different medical aspects or medicines before going to surgery or even talking about surgery.”

Insurance Obstacles

Parents reported insurance related barriers that impacted their journey to surgery, and several parents stated that they purposively changed insurance in order to make care for their child with epilepsy easier. Parents noted that insurance policies impacted their ability to see certain physicians and centers and to have specific diagnostic tests in a timely and stress-free manner. Even if approval was eventually granted, authorizations were needed and parents had to spend significant time and energy navigating how to get specific visits, procedures or tests approved. Parents had to learn to navigate insurance policies, a learning process that was frustrating and time consuming. One mother explained her insurance frustrations, stating, “I firmly believe that if the insurance companies would have been more responsive, it would have changed the outlook of the whole thing.”

Parental Stress

Parents reported personal and family stress throughout the journey. Caring for children with a chronic, severe and paroxysmal neurological condition meant being “on call” all the time; they had to be prepared to respond to an emergency at all times. For many parents, such caretaking roles meant they had less time and energy for other persons and activities in their life – including their spouse, other children, and work. A mother reflected, “It was a miracle I didn't drink or do drugs. I mean that is the honest truth…I always had to be on call…I can't believe my marriage is still intact as well – it was so stressful.” Many parents felt very alone in their roles as caretakers of and advocates for their children and in the decisions that they had to make for their children's care.

THEME 3. SURGERY AS A VIABLE OPTION: “The right spot”

After an arduous journey, parents reached the right doctor and center; epilepsy surgery, although previously seen as a last ditch option, became a viable option and a source of hope.

Surgery as Scary and Last Resort

Many parents initially perceived epilepsy brain surgery to be “pretty horrific” and a treatment of “last resort.” The prospect of needing to make a decision about surgery for their child was stressful. Although knowing about the possibility of surgery, some parents continued to hold on to a notion that their child might “outgrow” their seizures; such a notion often came from a treating doctor saying that this would happen or from a child's prior experience with a period of remission.

Epilepsy is Scarier than Surgery

On their journey, many parents transformed their view of surgery from a scary, last resort treatment to a necessary and hopeful option. One mother explained, “By the time she got it – it got so serious I was looking toward that as being as answer to our prayers rather than just something that I couldn't even think about.” This transformation occurred as parents became increasingly aware of the severity of their child's epilepsy and its impact on their child's and their future life. While surgery remained scary, epilepsy was or became scarier than surgery. As some parents feared that their child would die from epilepsy, surgery was no longer perceived to be an “elective” treatment.

Hoping for Surgical Candidacy

As parents learned that surgery was a potential viable option for their child, they “hoped” for candidacy. This hoping required persistence and sometimes disregarding doctors who initially felt surgery was not an option. Looking back, one father voiced happiness that he listened to his parental instincts rather than the opinion of one doctor. He (doctor) said, “ `I don't believe that she is a surgical candidate.' As a parent, I listened and I wanted to think otherwise… In hindsight, I'm glad I didn't listen to him.”

THEME 4. LIFE NOW: “We took the steps we needed to”

Though the purpose of the interviews was to examine parental perspectives along the journey to surgery, parents spontaneously spoke of their lives post-surgery.

A New Life – Child and Parent

Many parents reported that the journey to surgery was a life changing process for their child and family. For some, life before surgery felt like “another life” and sometimes one that they would prefer not think about. Post-surgery, one family came to refer to the seizures as “things you don't like to talk about because that's been taken care of.”

Giving Back

Post-surgery, parents often wanted to give back to others by sharing their story about their journey to surgery, supporting other parents now going through a similar journey and advocating for brain surgery. Many parents recommended support groups and felt a need to give back to others, particularly since some parents didn't make “parental connections” until late in the journey.

DISCUSSION

Parents that travelled the journey to pediatric resective epilepsy surgery identified four main themes. The journey started with recognition that “something is wrong” with their child; unfamiliar with seizures and epilepsy, however, parents still identified their child's medical urgency. Following this, parents were searching and finding along a “journey around the world” that was “circuitous”. This journey encompassed information seeking, multiple medication trials, insurance obstacles and parental stress until they found the right doctors. Although initially considered to be a treatment of last resort, parents perceived surgery to be a viable option and hoped for candidacy as epilepsy reached a threshold of being scarier than surgery. Parents spontaneously reflected on life post-surgery and were convinced that they made the right decision in pursuing surgery fueled with a desire to give back to other families. Along this path, parents perceived the more arduous and longer aspect of the journey to surgery to be experienced prior to presurgical referral.

These data provide evidence for a spectrum of parent-, provider- and health-system based barriers that influence the ease of getting to and the timeliness of receipt of pediatric resective epilepsy surgery. Parents perceived these barriers to be most pronounced early in the journey, prior to the referral for presurgical evaluation. While prior literature has reported parental difficulty with recognition of initial epileptic seizures in children that may result in delays in seeking medical care,^30,31 we found that although the majority of parents did not know that their child was having a seizure that very first time, most parents, nonetheless, sought urgent or emergent medical attention. Further, the time from second AED failure to presurgical referral was ≥1 year in nearly two-thirds of children perhaps highlighting that the interval from two AED failure until access at a pediatric epilepsy center for presurgical evaluation is a critical window for interventions targeting barriers.

Parent-based perceived barriers along the journey to epilepsy surgery included knowledge of epilepsy, parental stress and the perception of surgery as risky and a last resort treatment. Parents reported little baseline knowledge related to seizures and epilepsy, prompting them to “learn the language” of epilepsy, highlighting the need to develop strategies to mitigate stigma³² and to improve public awareness of epilepsy for families with epilepsy.³³ Our observation that parents experienced significant stress along the path to surgery³⁴ further underscores the need for family-centered care and community resources for parental support, including for example, peer-to-peer support groups.³⁵ Consistent with prior work showing a high proportion of adults perceived epilepsy surgery to be a very dangerous treatment of last resort^{13,14,19,22,23} we found that many parents shared similar sentiments. In one study of pediatric epilepsy surgery, Italian parents of young children had a more favorable attitude toward surgery compared to parents of adolescents; however, subjects were parents of children with predominantly well-controlled epilepsy who had not undergone surgery.²⁴ Future research is needed to determine the extent to which such parental attitudes toward surgery mediate the timing of presurgical referral and surgery, in addition to how physician-parent communication about surgery impacts perceptions and decision-making.

Parents also reported notable physician-based barriers including variability in perceived knowledge of epilepsy, criteria for presurgical referral and appropriateness of epilepsy surgery. Our finding that 30% of children were referred by a non-neurologist suggests that parents had to find alternate referral routes and that some child neurologists may not be aware of or follow pediatric epilepsy surgery referral recommendations⁶ as has previously been found in adult patients.^17,18,36 To date, studies that have found variability in physician knowledge of AED resistance, presurgical referral criteria and attitudes toward surgery have primarily focused on adults.^17,18,20 Two non-U.S. surveys of child neurologists demonstrated variable views on the definition of drug resistant epilepsy and surgery eligibility, in addition to non-adherence to referral recommendations.^17,25 The extent to which these findings are representative of U.S. provider attitudes, knowledge and referral practices for pediatric epilepsy surgery and their potential impact on the timing of presurgical referral and surgical treatment is not known, although they do suggest that physician-targeted interventions are needed. While a neurologist utilized web-based intervention was developed in Canada to identify potential appropriate adolescent and adult epilepsy surgery candidates, further research is needed to determine its impact on timing of presurgical referral and patient outcomes.³⁷

Parents reported numerous health-system based barriers including health insurance related obstacles. While studies have examined insurance disparities in utilization^15,21,26,38 or timing⁷ of surgery in adult and pediatric epilepsy surgery, and delays in access to pediatric specialty care,³⁹ this study reports parents' perceptions of the nature of insurance related barriers along the path to epilepsy surgery. Parents reported challenges associated with the navigation of complex insurance procedures related to approval of diagnostic testing or care with physicians out of network; they had to learn to navigate insurance policies and the medical system. The extent to which health insurance policies and authorization procedures are concordant with presurgical referral recommendations⁶ at the time of two AED failure, or medical intractability,⁴⁰ however, is unknown.

There are limitations to our study. Interviews provided retrospective parental perspectives only. Such retrospective accounts, however, provided perspectives of the journey through the epilepsy continuum – from seizure onset, epilepsy diagnosis, initial treatment, to presurgical referral and surgery; parents additionally spontaneously discussed life beyond surgery. While the `time to surgery' reported by parents was in agreement with that documented in the medical chart, the timing of other parent-reported epilepsy care was not reliably recorded in medical records, thereby limiting potential comparisons between parent-reported and medical chart data. Because the goal of our study was to ensure a heterogeneous sample from which to identify the full range of parent-reported perspectives along the journey to surgery, the extent to which they occur in the population of children with surgically remediable epilepsy would have to be quantified in a study with a different design.²⁷ We focused on parents, as they typically are the proxy-decision makers and health care navigators for their children. Because we only interviewed parents of children who had surgery, parents of children who were never referred for surgery or who underwent a presurgical evaluation for surgery and were not deemed candidates or those who were offered surgery but later declined it are not included. The extent to which children with medically refractory epilepsy are never referred to a comprehensive epilepsy center for evaluation including potential presurgical evaluation is unknown, but of significant concern given our observation that the journey to surgery was more arduous for many parents prior to the presurgical evaluation. While our findings represent the parental perceptions of physician knowledge and attitudes, they do suggest that physician factors may – at least in part – mediate – delays in timing presurgical referral and surgery.

Along the journey to pediatric epilepsy surgery, parents perceive a broad array of modifiable parent-, provider- and health-system based barriers. Multi-pronged interventions are needed to remediate delays and improve access to subspecialty care for children with medically refractory epilepsy in need of surgery given the multitude of diverse barriers identified.³⁰ The recent Institute of Medicine report, “Epilepsy Across the Spectrum: Promoting Health and Understanding” noted, “early identification of patients with persistent seizures that would lead to earlier referral” is needed.³³ Given the large burden of barriers and arduous journey of parents prior to presurgical evaluation, the interval from establishment of drug resistant epilepsy until access at a pediatric epilepsy center for presurgical evaluation is a critical window for interventions targeting these barriers.