Coping strategies in adults with a diagnosis of refractory epilepsy: Development, antecedents, and consequences.

Refractory epilepsy means that epileptic seizures remain uncontrolled despite
adequate treatment with at least two antiepileptic drugs (Kwan et al., 2010).
The unpredictable course of refractory epilepsy is an important factor in
coping with this illness. There are different types of coping strategies, which
means that everyone copes with having epilepsy in a different way. Various
studies have shown that patients with epilepsy often employ an avoidant coping
style, which may have a negative impact on different aspects of psychosocial
functioning. Hence, coping strategies will function as the primary variable of
interest in the present project.

First, we will examine which coping strategies adults with epilepsy commonly
use in dealing with their illness and how these coping strategies develop over
time. Coping is defined as the use of cognitive and behavioral strategies for
dealing with pressures, demands, and emotions involved in stressful situations
(Lazarus & Folkman 1984). Hence, coping is typically viewed as a
multidimensional construct. A particularly important distinction can be made
between engagement coping (aimed at active problem solving and dealing with
stressors and related emotions) and disengagement coping (aimed at avoiding or
withdrawing from problems and escaping feelings of distress) (Carver &
Connor-Smith, 2010). In the current project, core exemplars of these two
strategies (i.e., problem solving and avoidance) will be assessed, in addition
to social support seeking which is generally viewed as part of engagement
coping (Carver & Connor-Smith, 2010; Seiffge-Krenke, Aunola,& Nurmi, 2009).
Previous research has focused mainly on cross-sectional snapshots of coping and
has not examined the development of coping in patients with refractory epilepsy
using multi-wave longitudinal data.

Second, we will examine how these coping strategies are related to, and
potentially influence, quality of life and seizure frequency and severity in
our patients. Although coping strategies are not universally beneficial or
detrimental (Wrosch et al., 2003), prior research in community samples have
typically found that problem solving and social support seeking predict better
physical and psychological health, whereas avoidance predicts poorer outcomes
(Compas et al., 2001). In individuals with epilepsy, the employed coping style
has been shown to be an important predictor of patients* psychological
wellbeing (Kemp, Morley, & Anderson, 1999), quality of life (Westerhuis,
Zijlmans, Fischer, van Andel, & Leijten, 2011), and perceived seizure severity
(Oosterhuis, 1999). By using multi-wave longitudinal data, we can investigate
whether coping, quality of life, and seizure frequency/severity co-develop over
time. Furthermore, we can examine the directionality of effects and identify
reciprocal associations among the different study variables. For instance, one
might hypothesize that the use of avoidant coping strategies may lead to a
higher seizure frequency over time. However, a higher seizure frequency may
also lead to a further increase in avoidant coping, thereby inducing a negative
vicious cycle.

Third, we will examine how antecedent factors (i.e., executive functioning,
illness identity and perceptions, and personality) relate to, and potentially
influence, these coping strategies. With regard to executive functioning, the
neuropsychological literature suggests a connection between coping and
executive functioning. To effectively cope with stressful events (planning), it
is necessary to remember previous confrontations with similar stressors
(working memory), and at the same time to evaluate coping alternatives and
adapt cognitive and behavioral responses to the situation (cognitive
flexibility). A study with patients who have had a stroke shows that deficits
in executive functioning (especially working memory) are associated with an
increase in avoidant coping style (Kegel, Dux, and Macko, 2014). Another study
in adolescents with treatment resistant depression shows that some aspects of
executive functioning (inhibition and cognitive flexibility) affect the
relationship between coping and depressive symptoms (Morris, Evans, Rao, &
Garber, 2015). Decreased active and increased avoidant coping are associated
with a higher number of depressive symptoms. It appears that higher scores on
both inhibition and cognitive flexibility lead to fewer depressive symptoms
through more active and less avoidant coping. In addition, it has been found
that there is a low correlation between subjective executive functioning, as
measured with the BRIEF-A, and objective executive functioning, as measured
with cognitive tasks. Hence, in order to examine executive functioning
thoroughly, both the BRIEF-A and the cognitive tasks are deemed necessary.

Another potentially important factor that could explain why certain patients
experience difficulties, whereas others successfully manage their illness, is
patients* illness identity. The concept of illness identity captures the degree
to which the illness is integrated into patients* identity (Luyckx, Rassart, &
Weets, 2015). This is the first study to date to look at this issue in the
epilepsy population. Recently, four illness identity dimensions have been
proposed in the literature: engulfment (patients completely define themselves
in terms of their illness, which invades all domains of life), rejection (the
illness is viewed as a threat or as being unacceptable to the self), acceptance
(patients accept their illness as part of their identity, besides other social
roles and identity assets), and enrichment (the illness results in positive
life changes, benefits one*s identity, and enables one to grow as a person). In
other chronic illness populations (e.g., patients with type 1 diabetes),
important associations with depressive symptoms, quality of life, and treatment
adherence were uncovered (Oris et al., manuscript in revision). Relatedly,
previous research has demonstrated the importance of patients* illness
perceptions in how they cope with their illness. For instance, a study by
Goldstein, Holland, Soteriou, and Mellers (2005) in adults with epilepsy showed
that patients with negative illness perceptions more often used an avoidant
coping style in dealing with stressors. In contrast, patients feeling more in
control of their illness reported a more active coping style.

Finally, personality has been proven to be an important factor in how patients
cope with their illness (Rassart, Luyckx, Klimstra, Moons, & Weets, 2014). In
the current study, we focus on the Big five personality traits: extraversion
(engaging in social behaviors and experiencing positive emotions),
agreeableness (showing empathy and engaging in prosocial behavior),
conscientiousness (organizational and motivational aspects of behavior and
taking responsibility), emotional stability (the tendency to be emotionally
stable and managing negative emotions), and openness to experience (the way
people seek and deal with new information) (Caspi, Roberts, and Shiner, 2005).
Previous research, for instance, has shown that patients low in extraversion
and emotional stability are more prone to depression after epilepsy surgery
(Wilson, Wrench, McIntosh, Bladin, and Berkovic, 2010). However, research
looking at the role of personality in this specific population is scarce.

In sum, we want to gain more insight into the coping strategies of adults with
epilepsy, their associations with quality of life and seizure
severity/frequency, and potential antecedent factors (i.e., executive
functioning, illness identity and perceptions, and personality functioning).

Primary Objectives: To formulate answers to the following questions:

Three main study objectives will be examined in the present project.
1. What are the employed coping strategies in patients with refractory epilepsy
and how do these develop over time?
2. How do these coping strategies relate to, and potentially predict, quality
of life and seizure frequency/severity in these patients (consequences of
coping)? Can coping predict these outcomes over time after controlling for the
effects of demographical and clinical variables (e.g., gender, age, type of
epilepsy, medication use)?
3. How do executive functioning, illness identity and perceptions, and
personality functioning relate to, and potentially predict, these coping
strategies over time (antecedents of coping)? Can these factors predict coping
strategies after controlling for the effects of specific life-events?

Our study is an observational longitudinal research with three time points
(once a year for the duration of two years, that is, at baseline, baseline +
one year, and baseline + two years) in which patients between the ages of 18-40
with refractory epilepsy complete the following questionnaires:
- Coping strategy: Shortened Coping Strategy Indicator (CSI)
- Illness identity: Illness Identity Resolution Scale (IIRS)
- Personality: Big Five Inventory (BFI-25)
- Subjective executive functioning: Behavior Rating Inventory of Executive
Function (BRIEF-A)
- Quality of life: Shortened Quality of Life in Epilepsy (QOLIE-31)
- Illness perceptions: Brief Illness Perception Questionnaire (BIPQ)
- Severity and frequency of the seizures: Liverpool Seizure Severity Scale
(LSSS)
- Life-events: one open and one impact question

The following demographic and clinical variables will also be measured:
- Gender
- Age (date of birth)
- Level of education
- Employment status
- Ethnic background
- Marital status
- Age of onset of the epilepsy
- Type of epilepsy
- Medication use
- Epilepsy surgery (yes/no)
- Psychogenic non-epileptic seizures (PNES) (yes/no)

An at random selected subgroup will be invited at three different time points
(once a year for the duration of two years) to measure objective executive
functioning. The executive tasks are:
- Cognitive flexibility and inhibition: D-Kefs Color Word Interference Test
- Working memory: WAIS-IV-NL Number sequences
- Planning: D-Kefs Tower Test
- Attention and information processing speed: WAIS-IV-NL Digit Symbol
Substitution Test

The time required for completing the questionnaires per time point (3, once a
year) is expected to be 60 minutes. A subgroup will be invited at three
different time points (once a year) to measure objective executive functioning.
The time expected to complete the executive tasks is 45 minutes. These
executive tasks will be conducted at the Hans Berger Clinic, which means that
some patients will need to travel. Patients could experience participation and
the time required to participate as a burden. In addition, certain questions,
for example about depressive symptoms, may cause some discomfort to some
participants. Although our past experience with such questionnaires indicates
that feelings of discomfort are very limited. The opportunity to contact the
principal investigators (dr. Ruth Mark and drs. Ludo Verdyck) and independent
expert (prof. dr. Marrie Bekker) is given in case certain questions evoke
negative emotions or if the person has trouble handling them.

There are a number of steps taken to protect the subjects. The informed consent
will be provided to the participants along with the questionnaires and the
documents must be returned to the researchers in a sealed envelope. We will
explicitly ask the participants for permission to view their electronic medical
record and use certain data from it (e.g. type of epilepsy). Confidentiality of
the data is ensured in several ways:

(1) All analyses will be carried out on a group level and not on an individual
level.
(2) All documents (questionnaires and informed consents) will be provided with
meaningless numbers that allow us to link the questionnaires over time and to
link the informed consents to the questionnaires. These numbers will be used
for entering and organizing the data. The numbers and the associated list of
participants are kept separately in a secure document to which only the
involved researchers from the University of Tilburg, Kempenhaeghe-HBK and
KULeuven have access.
(3) The informed consents and contact details of the participants will be saved
seperately from the dataset and questionnaires.
(4) Individual data obtained from the aforementioned questionnaires will not be
passed to doctors and clinicians. This study, therefore, is independent from
the possible clinical follow-up for patients, which means that patients are not
in a dependent position in relation to the researchers of this project.
(5) Contact details of the principal investigator (dr. Ruth Mark and drs. Ludo
Verdyck) and independent expert (prof. dr. Marrie Bekker) are given for any
questions or remarks.