family predictors of psychopathology in children with epilepsy

Epilepsia, 47(3):601–614, 2006Blackwell Publishing, Inc.C© 2006 International League Against Epilepsy

Family Predictors of Psychopathology in Children with Epilepsy

∗Roos Rodenburg, ∗Anne Marie Meijer, †Maja Dekovic, and ‡Albert P. Aldenkamp

∗Department of Educational Studies, University of Amsterdam, Amsterdam; †Department of Child and Adolescent Studies, UtrechtUniversity, Utrecht; and ‡Department of Neurology, University Hospital Maastricht & Department of Behavioral Sciences, Epilepsy

Center Kempenhaeghe, Maastricht, The Netherlands

Summary: Purpose: To examine the contribution of epilepsy-related factors, proximal (parent–child relationship quality), dis-tal (parental characteristics), and contextual (quality of otherfamily relationships) family factors to psychopathology (bothbroad-band and narrow-band syndromes) in children withepilepsy and normal intelligence.

Methods: Parents of 91 children (mean, 8.5 years) partici-pated by filling out questionnaires about family factors and childpsychopathology. Regression analyses were used to analyze theunique and combined predictive power of family factors in rela-tion to psychopathology.

Results: In contrast to epilepsy-related factors, family factors,especially those related to the quality of the parent–child rela-

tionship, appeared to be strong predictors of psychopathology.The results supported the mediational model: Distal and contex-tual factors affect child psychopathology by affecting proximalfactors.

Conclusions: In treating children with epilepsy, clini-cians should be aware of the importance of the parent–child relationship quality. Strengthening the relationship qual-ity may prevent or reduce psychopathology. Key Words:Childhood epilepsy—Psychopathology—Parent–child relation-ship quality—Parental characteristics—Family relationships—Mediator.

The literature provides ample evidence that psy-chopathology is more common in children with epilepsythan in children from the general population (1,2). A re-cently conducted meta-analysis confirmed that childrenwith epilepsy have higher levels of psychopathology thando children from the general population (3).

The factors that have been found to contribute topsychopathology in children with epilepsy are multiple(4,5). They consist of neurologic factors (e.g., age atonset, epilepsy duration, epilepsy severity, seizure type,and seizure control) (6,7), medication factors such asside effects (8), and psychosocial factors (e.g., perceivedstigma, attitude toward illness, and family factors) (9–13).While reviewing the behavioral and cognitive correlatesof epilepsy in 1984, Hermann and Whitman (5) pleadedat that time for a more serious examination of family fac-tors as determinants of psychopathology in children withepilepsy. Since then, the examination of distinct familyfactors as contributors to child psychopathology has grad-ually been incorporated in childhood epilepsy research.

Accepted October 8, 2005.Address correspondence and reprint requests to Dr. H.R. Rodenburg

at University of Amsterdam, Faculty of Social and Behavioral Sciences,Department of Educational Studies, Wibautstraat 4, 1090 GEAmsterdam, The Netherlands. E-mail: [email protected]/[email protected]

Family-factor constructs represent different aspects offamily functioning, but to date, an appropriate model todifferentiate between family factors has frequently beenlacking in childhood epilepsy research. It is essential,though, to categorize family factors into distinct types,because each type of factor is considered to play a differ-ent role in relation to child outcome (14–16). Followingsocial interactional and ecologic perspectives, family fac-tors can be ordered, according to the level of proximityto the child’s everyday life, into proximal family factors(the quality of the parent–child relationship and parent-ing), distal family factors (parental characteristics), andcontextual family factors (the quality of other family re-lationships) (14,17–19).

Although it can be deduced from childhood epilepsyliterature that distinct family factors are associated withpsychopathology in children with epilepsy (20), the si-multaneous examination of distinct family factors seldomoccurred. Little is known about the unique and the com-bined predictive power of each of these family factors ascontributors to child psychopathology. The examination ofthe interrelationships among distinct family factors them-selves and the pathways between these distinct family fac-tors and child psychopathology may add new information,which may be helpful for children with epilepsy and theirfamilies.

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602 R. RODENBURG ET AL.

With the current study, we aim to examine the con-tribution of epilepsy-related factors and distinct familyfactors—ordered into proximal, distal, and contextualfactors—to psychopathology in children with epilepsy.Even though it is possible to make clear conceptual dis-tinctions between family factors, it also is clear that fac-tors representing family functioning do not function sepa-rately from each other within families, but are interrelatedconstructs that in joint interaction exert their influenceon the child’s everyday life (21). As a consequence, itmay be that spurious associations arise between distinctfamily factors and child psychopathology. Therefore withthis study, we aimed to examine the independent contri-butions of proximal, distal, and contextual family factorsto child psychopathology and to examine whether certaintypes of family factors predominate in predicting childpsychopathology (18). Both family risk factors and pro-tective factors were selected, because it is considered thatchild outcome is affected by the equilibrium between fam-ily stressors and protective (buffering) factors. That is, ac-cording to the cumulative risk model, the chance that psy-chopathology develops in children increases when familyrisk factors outweigh protective family factors (15,16,22).

Although childhood chronic illness literature has em-phasized the importance of applying a noncategoric ap-proach to examining the effects of chronic conditions onchild development (that is, generic features of chronic con-ditions such as functional status, the limitation of activ-ities, or the burden of daily care on the family) (23,24),disease-specific parameters also have been consistentlydepicted in conceptual models that explain child adjust-ment in chronic conditions (19,25). Among these diseaseparameters are condition severity, which has been foundto be a contributor to child adjustment (26) and condi-tion duration, which received less attention in childhoodchronic illness research (25).

In childhood epilepsy, disease parameters could be de-scribed as pathogenetic contributors of psychopathology,that is, central nervous system (CNS)-related or epilepsy-related factors (27). When epilepsy-related factors wereexamined with parenting factors, it was found that par-enting factors predicted internalizing and externalizingbehavior problems better than the effects of epilepsy-related factors (12,13). However, significant relations alsohave been found between seizure severity and child psy-chopathology (28), including internalizing, externalizing,attention, and thought problems (7,29). Hoare and Mann(30) found an association between longer epilepsy dura-tion and child psychopathology, including depression andanxiety (31).

Proximal family factors comprise the quality of theparent–child relationship and parenting (18,32). Parent–child relationship quality is defined as a constellation ofparental attitudes that has been built up in the long historybetween parent and child, in which the parent’s behaviors

are expressed (18,32). Good quality in terms of the parent–child relationship has generally been associated with lowerlevels of child psychopathology in samples of both healthychildren and children with epilepsy (9,13,33–35).

In contrast, low parent-child relationship quality, suchas parental rejection, has been found to exert negative ef-fects on child development and is thus considered to bea risk factor for the development of child psychopathol-ogy (36–38). One study that measured expressed emotion(EE) in mothers of children with epilepsy showed that highmaternal criticism, an indicator of a low parent–child re-lationship quality, was related to higher levels of childpsychiatric disturbances and antisocial behavior (33).

Distal family factors are dispositional parental charac-teristics that are assumed to influence the child indirectly,by disrupting the quality of parenting (18). Maternal de-pression is a distal family factor that is of current interestin child developmental and family research, and also inchildhood epilepsy literature. Child developmental litera-ture consistently showed both direct associations betweenmaternal depression and child psychopathology (39,40)and indirect associations between maternal depression andchild psychopathology by disrupting the quality of par-enting (18,41). Childhood epilepsy literature showed that∼30% of the mothers of children with epilepsy are atincreased risk for depression and showed associations be-tween maternal depression and higher levels of child psy-chopathology (33,42,43).

Counter to the risk-factor depression, we selected an-other dispositional characteristic that protects againstpsychopathology, parental competence in parenting. Al-though this characteristic has scarcely been researched inrelation to child psychopathology in the epilepsy literature(9), in the child developmental literature, it has frequentlybeen linked to adequate parenting (44,45), which in turn isrelated to lower levels of child psychopathology (46,47).

Contextual family factors are often understood as in-dicative of psychopathology in individual family mem-bers, reflecting overall family dysfunction (48). In light ofchildhood chronic conditions, family adaptation is com-monly seen as a key factor for healthy child adjustment(49,50). Childhood chronic conditions are considered toaffect the whole family, requiring new modes of organiza-tion and structure for the family (51). The family failure toadapt adequately to the demands of childhood chronic ill-ness could, therefore, be considered a risk factor for devel-opment of psychopathology. Studies with samples of bothhealthy children and children with a chronic illness, in-cluding children with epilepsy, showed a relation betweenproblems with family adaptation and the development ormaintenance of child psychopathology (9,52–54).

Marital satisfaction also has frequently been linked tochild psychopathology. Unsatisfactory marital relation-ships have been shown to function as a risk factor forthe development of child psychopathology by interfering

Epilepsia, Vol. 47, No. 3, 2006

FAMILY FACTORS IN PEDIATRIC EPILEPSY 603

with parent–child relationship quality (55–57). In contrast,good marital relationship quality is positively associatedwith adaptive child outcome (58). To date, marital rela-tionship quality has been scarcely researched in samplesof children with epilepsy. One study, carried out by Austin,Risinger, and Beckett (59), found that marital strain wascorrelated with higher levels of child behavior problems.Another earlier study (60) found that a disrupted parentalmarriage contributed to internalizing and externalizing be-havior problems in children with epilepsy.

The relation between family factors and psychopathol-ogy in children with epilepsy has been examined mainlyon the level of Internalizing (e.g., Anxiety/Depressionand Withdrawal) and Externalizing (e.g., Aggression andDelinquency) broad-band behavior problems, measuredwith the Child Behavior Checklist (61,62). The relationbetween the syndrome scales (Anxiety/Depression, With-drawal, Somatic complaints, Delinquency, Aggression,Social problems, Attention problems, and Thought prob-lems) and family factors has been less frequently assessedin children with epilepsy (20). To overcome this gap and,in particular, to study thought problems and attention prob-lems that were considered relatively specific to epilepsy(3), the current study includes all the syndrome scales.

In sum, we aimed to examine simultaneously the rela-tion between epilepsy-related factors, distinct family fac-tors, and distinct types of psychopathology in childrenwith epilepsy by examining the independent contributionof each cluster of epilepsy and family factors to child psy-chopathology. More specifically, concerning the relationbetween distinct family factors and child psychopathol-ogy, two mechanisms may play a role. First, it may bethat each family factor has an independent effect on childpsychopathology. Although we expect that each of thesefactors would be related to child psychopathology, we ex-pect that the proximal factors would exert the greatest in-fluence on child psychopathology (16). Second, it may bethat family factors are interdependent in their influence onchild psychopathology. On basis of the social interactionaland ecologic model, it is subsequently assumed that theeffects of distal (parental depression and parental compe-tence) and contextual factors (problems with family adap-tation and marital satisfaction) are mediated by proximalfamily factors. In other words, the distal and contextualfamily factors are expected to affect the parent–child re-lationship (proximal factors), which, in turn, would affectchild psychopathology.

METHODS

SampleParents of children referred for epilepsy to the out-

patient clinic of the tertiary epilepsy center Kempen-haeghe (The Netherlands) filled out questionnaires con-cerning child psychopathology, epilepsy-related aspects,

and family factors, that is, parent–child relationship qual-ity, parental characteristics, and family relationships. Thestudy was approved by the scientific review committee ofthe Kempenhaeghe Institute, and informed, written con-sent was obtained from every participating parent. Thecriteria for inclusion of children in the study were (a) anIQ >70 points, (b) aged between 4 and 18 years, and (c)no psychiatric illnesses, such as autism or obsessive–compulsive disorders. An exception to this criterion wasthe presence of Attention Deficit and Hyperactivity Dis-order (ADHD), which is very common for childrenwith epilepsy (1,63). In addition, child psychopathologyaccording to conceptualizations of the Child BehaviorChecklist was not defined as psychiatric illness.

In total, 91 children met the inclusion criteria. The num-ber of potential family inclusions was 135. About 33% ofthe families did not participate in the study because ofnot fulfilling inclusion criteria (i.e., low IQ/severe men-tal retardation: five children); refusal to participate, andnondelivered questionnaires. No information about non-participating families was available, unfortunately. Boyswere slightly overrepresented, 58% (n = 53). The childrenhad a mean age of 8 years, 5 months (SD, 2.42). Most chil-dren (86.8%) were younger than 12 years. Mean child IQwas 90 points (SD, 13.18). The majority of the children(60.4%) were in primary school, and 20.9% received spe-cial educational services.

Epilepsy-related factors (i.e., seizure type, epilepsysyndrome, frequency of seizures, the presence ofmono/polytherapy, epilepsy onset) were specified basedon an inspection of the children’s medical files. The def-inition of the type of epilepsy was based on the Interna-tional League Against Epilepsy criteria (64). The majorityof children in the sample had complex partial seizures(44%) and absences (39.6%). The commonest formwas generalized idiopathic epilepsy (40.7%), followedby localization-related idiopathic epilepsy (29.7%) andlocalization-related symptomatic epilepsy (27.5%). Meanepilepsy onset was at 4 years and 1 month (SD, 2.12).Children experienced seizures on a daily (22%), weekly(25.3%), monthly (26.4%), or yearly basis (24.2%). Thisis a normal representation of the epilepsy center Kem-penhaeghe. Because Kempenhaeghe is a tertiary epilepsycenter, a slight bias may exist regarding the more severeepilepsies. Moreover, most referred children were in thefirst phase of diagnosis before the onset of therapy or dur-ing the initial phase of treatment, and consequently, theseizures are not very well controlled yet in a large groupof children. A number of children had no antiepilepticdrug (AED) prescription (19%), the majority of childrenreceived monotherapy (59%), a minority had more thanone AED prescribed (8%), and for 14% of the children,no medication use information was available.

Eighty-one mothers and 10 fathers completed the ques-tionnaires. Mothers (mean age, 38.8 years; SD, 5.1) had



mainly completed lower secondary vocational education(23.1%), secondary education (18.7%), or senior sec-ondary vocational education (27.5%). Seventy-one per-cent of the mothers had a paid job for several hours aweek, and 24% did not work and ran the housekeeping (1%unemployed, and for 4% no information was available).Educational training for fathers (mean age, 42.0 years;SD, 6.4 years) was mainly lower secondary vocational ed-ucation (27.5%), senior secondary vocational education(20%), or higher professional education (20%). Ninety-two percent of the fathers had a paid job. Parents were over-represented concerning the lower and senior secondaryeducational levels and underrepresented concerning thehigher educational levels, whereas the percentage of moth-ers and fathers with paid jobs was relatively high whencompared with the general population (65). Most fami-lies were intact families with two biologic parents (92%),which is considerably higher than the percentage of two-parent families (∼75%) in the general population (66).One child lived in a stepfamily, two children lived insingle-parent families with coparenting, and one childlived in a single-parent family without coparenting. Allchildren were of Dutch origin. Only 12% of the childrenwere single children, 48% had a brother or sister, and 34%of the children had two or more siblings (for 5.5%, no in-formation was available).

Measures

Child psychopathologyPsychopathology was measured with the Child Behav-

ior Checklist (CBCL), which is well known for its reliabil-ity and validity (61,62). The CBCL assesses psychopathol-ogy on both broad-band and narrow-band syndromelevels. Broad-band psychopathology includes Internaliz-ing behavior problems (e.g., depression) and Externalizingbehavior problems (e.g., aggression). Narrow-band syn-drome scales consist of Somatic complaints, Withdrawal,Anxiety/Depression, Aggressive behavior, Delinquent be-havior, Social problems, Attention problems, and Thoughtproblems. At the item level, parents indicate whether de-scriptions of behavior are (0) not at all true, (1) somewhattrue, or (2) very true for their child.

The CBCL uses cutoff scores that indicate whether achild has psychopathology in the borderline or in the clin-ical range. Normally, raw scores of the CBCL are used instatistical analysis for both males and females in the ageranges younger than 12 years and older than 12 years. Sucha division in boys and girls in age ranges younger than 12years and older than 12 years would divide the currentsample into relatively small subsamples. Therefore withthe present study, CBCL T-scores were used, which arescores that correct for age and gender. Regarding broad-band behavior problems, children scoring T ≥60 are inthe borderline range (scores 60–62), and children scoringT ≥63 are in the clinical range. For the syndrome scales,

children scoring T ≥67 are in the borderline range, andchildren scoring T ≥70 are in the clinical range. Meannormative CBCL scores are T = 50.

For the broad-band scales, excellent alphas were foundfor Internalizing problems (α = 0.87) and Externalizingproblems (α = 0.93). The narrow-band Internalizing syn-drome scales also had satisfactory reliability, with α =0.74 for both Withdrawal and Somatic complaints and α =0.84 for Anxiety/Depression. Alphas for the narrow-bandExternalizing syndrome scales were 0.71 (Delinquent be-havior) and 0.93 (Aggression). Attention problems (α =0.78) had a relatively high alpha, whereas Thought prob-lems (α = 0.64) and Social problems (α = 0.58) had ratherlow alphas. Social problems were not included in the anal-yses because of unsatisfactory reliability.

Epilepsy-related factors

Epilepsy durationThe estimation of epilepsy duration also was based on

an inspection of the child’s medical files. Mean epilepsyduration was 4 years, 6 months (SD, 2.71).

Epilepsy severitySimilar to the approach of Austin et al. (67), epilepsy

severity—a composite variable—was computed by as-signing scores from 0 to 3, based on seizure type, seizurefrequency, and the presence of mono/polytherapy. Seizuretype was scored 3 in case of generalized tonic–clonicseizures, 2 in case of partial seizures, and 1 in case ofabsences. A score of 3 was given if the child had weeklyor daily seizures, 2 if seizures happened monthly, and 1 ifthe child had seizures once a year. Absence of a medica-tion regimen was scored 1, the presence of monotherapywas scored 2, and in case of polytherapy, a score of 3 wasassigned. Scores for seizure type, seizure frequency, andthe presence of mono- or polytherapy were then summed.Children scoring between 1 and 5 were considered to havelow epilepsy severity, and children with scores ≥6 wereconsidered to have high epilepsy severity. Mean epilepsyseverity score was 5.60 (SD, 1.55). Forty-five percent ofthe children had low epilepsy severity, and 55% had highepilepsy severity.

Proximal Family Factors

RejectionRejection refers to the degree to which the child does

not fulfill parental expectations regarding physical, intel-lectual, and emotional characteristics and was measuredwith the rejection scale of the Nijmegen Parental StressIndex (PSI) (68,69). The scale consists of 12 items (e.g.,“It is not always easy to accept my child the way he/sheis”). Parents rated the items on a 6-point Likert scale, rang-ing from “completely disagree” (1) to “completely agree”(6). Alpha was 0.81. The higher the score, the higher thelevel of rejection the parent felt toward the child.



Positive parent–child relationship qualityPositive parent–child relationship quality, a composite

measure existing of the subscales parental authority andthe resolution of conflict between parent and child, andparental acceptance, was measured with the Parent–ChildInteraction Questionnaire Revised (PACIQ-R) (70), whichincludes 21 items (e.g., “I take my time to listen to mychild”) to be rated on a 5-point Likert scale ranging from“does not apply to me at all” (1) to “applies to me exactly”(5), and from “never” (1) to “always” (5). Alpha was 0.75.The higher the score, the more positively the parent feltabout the quality of the parent–child relationship.

Distal family factors

Parental competenceParental competence was measured with the compe-

tence scale of the Nijmegen PSI (68,69), that is, the de-gree to which the parent feels confident about dealing withthe child. The initial scale consists of 15 items (e.g., “Ican take decisions without help”). For the current study,a shortened version, consisting of five items, was used.Parents rated each item on a 6-point Likert scale, rangingfrom “completely disagree” (1) to “completely agree” (6).Alpha was 0.71. A higher score reflects more confidence.

Parental depressionParental feelings of depression were measured with the

Self-Rating Depression Scale (SDS) (71). This instrumenthas 20 items (e.g., “Life is worthless for me”). Parentshad to rate items on a 4-point Likert scale, ranging from“seldom or never” (1) to “almost always or always” (4).Alpha was 0.82.

Contextual family factors

Family adaptation problemsThe Dutch Version of the Family Adaptability and Co-

hesion Evaluation Scales (FACES) (72,73) was used toassess family adaptation. This scale refers to the degreeto which the family system adapts the power structure,role definitions, and relationship rules to changing inter-nal and external circumstances. It includes 13 items (e.g.,“In our family, it is unclear which rules are the standard,because they always change”). Although the definition ofthe scale is positively formulated, the scale is negative: ahigher score reflects more problems with family adapta-tion. Parents had to rate items on a 4-point Likert scale,ranging from “never true” (1) to “always true” (4). Alphawas 0.70.

Marital satisfactionThe satisfaction scale of the Interactional Problem Solv-

ing Questionnaire (IPOV) (74) was used to measure thedegree of marital satisfaction (e.g., “How satisfied are youabout the love and affection that you receive from yourpartner?”). A higher score reflects more marital satisfac-

tion. A 5-point Likert scale was used, ranging from “un-happy” (1) to “happy” (5). Alpha was 0.84.

Statistical analysisTo replace missing data, we carried out expectation–

maximization analysis (75,76). In this procedure, maximallikelihood estimates are computed, which subsequently“treat the missing data as random variables to be removedfrom the likelihood function as if they were never sam-pled” (75, p. 148). In our sample, missing data were dueto parents (a) simply forgetting to fill out items (itemsthat were not related to any other particular items), or (b)not filling out a part of the questionnaire because the spe-cific part was accidentally not included among the set ofquestionnaires (n = 21 for positive relationship quality,and n = 11 for adaptation problems). As a consequence,we were able to specify that our missing data were miss-ing completely at random, which is required to conductmissing-data analysis (75).

We examined the severity of the different types of psy-chopathology by comparing children with epilepsy withchildren from the normative population (CBCL norms).Differences between children with epilepsy and childrenfrom the normative population were calculated with ttests. Subsequently, Pearson correlations were used to ex-plore associations among epilepsy-related factors; proxi-mal, distal, and contextual family factors; and child psy-chopathology. Next, hierarchic regression analyses wereconducted to examine the predictability of child psy-chopathology from epilepsy-related factors, and the prox-imal, distal, and contextual family predictors. Two typesof hierarchic regression analyses were performed. First,direct effects of epilepsy-related factors and family pre-dictors were examined by entering each type of predictoron the first step of the regression. Putting in a cluster offactors first has the advantage that it shows the indepen-dent contribution of the factor to the dependent variable(i.e., psychopathology), compared with the other clustersof factors. Then the remaining significant predictors wereentered on the last step of the regression, thereby con-trolling for the other significant predictors. Entering eachcluster of predictors on the last step allows determinationof which factor has the most influential effect on childpsychopathology. Thus this approach allows the relativeinfluence of each predictor on child psychopathology tobe examined (18,77).

Second, we tested the mediational model that proposesthat contextual and distal family factors generate their ef-fects on child psychopathology through proximal familyfactors. To perform a test of mediation, the three follow-ing criteria must be met. First, distal and contextual fam-ily predictors have to be associated with proximal familymediators. Second, distal and contextual family factorshave to be related to child psychopathology outcome; andthird, proximal family factors have to be related to child



psychopathology (78). Evidence for mediation is achievedif the relation between distal and contextual family fac-tors and psychopathology is reduced when the effects ofproximal family factors are controlled for. However, be-fore concluding that proximal family factors mediate theeffects of distal and contextual family factors on child psy-chopathology, mediation effects should be examined at thelevel of the beta coefficients themselves (78). Therefore aseries of Sobel tests was conducted to test whether prox-imal family factors mediated, to a significant extent, theinfluence of distal and contextual family factors on the dif-ferent types of psychopathology (79,80). Holmbeck (81)wrote an excellent article in which he explained the neces-sity of conducting Sobel tests when examining mediatingeffects, because failure to do so often leads to conclusionsthat are false positive or false negative. Sobel tests werecomputed with a SPSS macro developed by Preacher andHayes (82), because SPSS does not provide the possi-bility of testing indirect effects with the Sobel test. Thismacro can be downloaded from the following website:http://www.comm.ohio-state.edu/ahayes/sobel.htm (82).

RESULTS

Psychopathology in children with epilepsyThe children in the current study scored far above the

normative T-score of 50 (SD, 10) on all syndromes: com-pared with children in the normative population, thesechildren with epilepsy had significantly higher levels ofpsychopathology (Table 1). The percentage of childrenscoring above the clinical cutoff was especially high on In-ternalizing behavior problems (57%) and Attention prob-lems (48%).

Parental depressionThe mean score on parental depression was 42.03 (SD,

9.15). Twelve percent of the parents scored above the cut-off of 50 for mild depression, and 6% scored above the

TABLE 1. Means, standard deviations and percentages of children scoring within the borderline range and above the clinical cutoffof the CBCL

Children with epilepsy Comparison with normative population Cutoff

Psychopathology M SD t Valuea Borderline Clinical

Broad-band syndromesInternalizing problems 63.55 10.46 12.09 7% 57%Externalizing problems 58.55 12.14 6.53 9% 37%

Narrow-band syndromesWithdrawal 63.09 9.59 12.66 9% 27%Somatic complaints 62.23 10.32 10.99 17% 18%Anxiety/depression 61.94 10.04 11.03 14% 18%Aggressive behavior 61.02 11.03 9.27 6% 20%Delinquent behavior 57.33 8.44 8.05 4% 13%Thought problems 65.08 10.18 13.74 6% 38%Attention problems 70.18 10.82 17.30 15% 48%

ap < 0.001.

cutoff of 60 for moderate depression. None of the par-ents scored above the cutoff of 70 for severe depression(71).

Associations among epilepsy, proximal, distal, andcontextual family factors, and child psychopathology

Epilepsy-related factors were not related to either dis-tinct family factors or child psychopathology (Table 2).Significant associations were found between almost allfamily factors and Internalizing and Externalizing be-havior problems. Parental rejection was correlated withhigher levels of Internalizing and Externalizing problems,whereas a positive parent–child relationship was associ-ated with lower levels of Internalizing and Externalizingbehavior problems. Parental confidence in parenting wasrelated to Externalizing behavior problems only. Parentaldepression was connected with both higher levels of Inter-nalizing and Externalizing behavior problems. Problemswith family adaptation were related to higher levels of In-ternalizing and Externalizing behavior problems. Finally,marital satisfaction was related to lower levels of Exter-nalizing behavior problems.

Family factors appeared to be interrelated. Especiallystrong association was found between two proximal fac-tors: parental rejection and parent–child relationship qual-ity. The magnitude of associations among other familyfactors varied from 0.24 to 0.48, indicating that, althoughrelated, each of these factors offers unique informationabout the family.

Table 3 presents the association between the CBCLsyndrome scales and epilepsy and family factors. Again,epilepsy-related factors were not associated with any par-ticular type of psychopathology. With the exception of So-matic problems that were only marginally linked to familyfactors, the pattern of the correlations between the syn-drome scales and family factors was largely similar to thecorrelations obtained with the broad-band scales.



TABLE 2. Means, standard deviations, and correlations among psychopathology (broad-band syndromes), epilepsy, proximal,distal, and contextual family factors

Variable M SD 1 2 3 4 5 6 7 8 9

1. Internalizing problems 63.55 10.46 −2. Externalizing problems 58.55 12.14 0.54a −

Epilepsy-related factors3. Duration 4.60 2.71 0.06 0.05 −4. Severity 5.61 1.50 −0.11 0.02 0.03 −

Proximal factors5. Rejection 2.54 0.90 0.41a 0.59a 0.16 0.156. Positive quality 4.16 0.32 −0.35a −0.64a −0.08 −0.07 −0.60a −

Distal factors

7. Parental competence 4.77 0.76 −0.05 −0.23b 0.15 −0.02 −0.42a 0.41a −8. Depression 1.68 0.35 0.33c 0.32c 0.06 0.11 0.47a −0.41a −0.43a −

Contextual factors

9. Adaptation problems 1.54 0.24 0.24b 0.28c 0.01 −0.04 0.43a −0.30c −0.37a 0.40a −10. Marital satisfaction 4.23 0.82 −0.14 −0.31c −0.01 −0.12 −0.47a 0.30c 0.24b −0.48a −0.43a

ap < 0.001.bp < 0.05.cp < 0.01.

Predicting psychopathology in children with epilepsy:direct effects

Direct effects of epilepsy-related factors and family fac-tors were investigated by entering each group of factors inthe first step of the regression. Subsequently, the remain-ing significant predictors were entered in the last step ofthe regression. The results of these regression analysesare presented in Table 4. When entered first, epilepsy fac-tors were nonsignificant contributors of both broad-bandand narrow-band syndromes. The factor most closely re-lated to the child’s everyday life, proximal family factors,accounted for most variance in all types of problem be-havior. The only exception was Somatic complaints, forwhich none of the family factors appeared to be a sig-nificant predictor. Distal factors were significant predic-tors of broad-band syndromes (Internalizing and Exter-nalizing problems). These factors also accounted for asignificant percentage of the variance in Withdrawal, Ag-

TABLE 3. Correlations between psychopathology (narrow-band syndromes) and epilepsy, proximal, distal, and contextual familyfactors

Somatic Thought AttentionVariable Withdrawal Depression problems Aggression Delinquency problems problems

Epilepsy-related factorsDuration 0.04 0.13 −0.05 0.04 0.09 0.15 0.03Severity −0.03 −0.10 −0.12 0.00 0.00 −0.14 −0.08

Proximal factorsRejection 0.42a 0.40a 0.11 0.58a 0.45a 0.47a 0.48a

Positive quality −0.36a −0.35a −0.12 −0.61a −0.54a −0.35a −0.33b

Distal factorsParental competence −0.06 −0.06 0.01 −0.18 −0.07 −0.13 −0.22c

Depression 0.36a 0.22c 0.22c 0.33b 0.29b 0.26c 0.34b

Contextual factors

Adaptation problems 0.28b 0.26c 0.13 0.29b 0.16 0.28b 0.28b

Marital satisfaction −0.21c −0.06 −0.09 −0.31b −0.27c −0.03 −0.15

ap < 0.001.bp < 0.01.cp < 0.05.

gressive and Delinquent behavior, and Thought and At-tention problems. Finally, contextual factors significantlypredicted Externalizing problems and five narrow-bandsyndromes: Withdrawal, Anxiety/Depression, Aggressivebehavior, Thought problems, and Attention problems.

At the level of individual predictors (Table 5), of theproximal factors, rejection and positive quality were bothsignificant predictors in almost all regression analyses.Predictive power of distal factors was, however, mostlydue to the parental depression. Beta coefficients for com-petence did not reach significance in any of the analyses.Two indicators of contextual factors were about equal re-garding their predictive power, although marital satisfac-tion appeared to be a stronger predictor of the externalizingtypes of problems (broad-band syndromes Externalizingproblems, Aggressive behavior, and Delinquent behavior),whereas adaptation problems were a stronger predictor ofother types of problems.



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In the next step of the analyses, only the contributorsthat had significant effects in the first step were enteredin the last step (Tables 4 and 5). Given the nonsignificantcontribution of each type of family factors to Somatic com-plaints, this narrow-band syndrome was excluded from thesubsequent analyses. The same results were obtained forall types of problems. Proximal family factors remainedsignificant contributors, even after the distal and contex-tual factors were controlled for. The significant effects ofdistal and contextual factors, however, disappeared afterthe other factors were controlled for.

Test of the mediational modelIn the next series of analyses, we tested the hypoth-

esis that distal and contextual factors exert their effectson child psychopathology by affecting the proximal fac-tors. In other words, the proximal factors are expectedto mediate the relation between distal/contextual factorsand psychopathology. Epilepsy-related factors were notentered in these analyses, because these variables did notmeet the criteria formulated by Baron and Kenny (78):epilepsy duration and severity were not correlated withproximal family factors, were not correlated with childpsychopathology, and were not related to the other pre-dictors.

For each test of mediation, only those predictors wereentered that had proved to be significant in the first step ofthe regression analyses for testing direct effects (see Ta-ble 5). Table 5 also shows which type of proximal familyfactor was a significant predictor of child psychopathol-ogy. This particular type (i.e., rejection or positive rela-tionship quality) was entered in each of the analyses fortesting mediation. On the level of the distal family factors,only depression was a significant contributor, and there-fore whether the effects of parental competence were me-diated by proximal family factors was not tested. In thecase of Internalizing problems, only effects of depressioncould be tested for, as both indicators of contextual factorsappeared to be nonsignificant predictors. In each analysis,proximal factors were entered in the first step, followedby distal and contextual factors. Table 6 shows the betacoefficients of individual predictors after controlling forthe proximal factors and shows the results of the Sobeltests.

The results were consistent across all types of prob-lems: after controlling for the proximal factors, the effectsof previously significant predictors, within the distal andcontextual groups of factors, were reduced to nonsignif-icance. Although this reduction to nonsignificance indi-cates evidence for indirect effects, the significance of in-direct effects was formally confirmed with the Sobel tests.The 95% confidence intervals for the size of the indirecteffects also were reported (82). The effects of parental de-pression were mainly mediated by parental rejection (onthe internalizing types of psychopathology, thought and


FAMILY FACTORS IN PEDIATRIC EPILEPSY 609T

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attention problems), whereas positive relationship qual-ity also mediated the effects of parental depression onexternalizing problems and aggression. In addition, pos-itive relationship quality mediated the effects of depres-sion on delinquent behavior problems. Family adaptationproblems were mediated by parental rejection for their in-fluence on withdrawal, anxiety/depression, thought prob-lems, and attention problems. The effects of marital sat-isfaction were mediated both by rejection and positiverelationship quality on externalizing behavior problemsand aggression, whereas positive relationship quality onlymediated the effects of marital satisfaction on delinquentbehavior problems.

DISCUSSION

The goal of this study was to explore the relative influ-ence of epilepsy-related factors and distinct family factorson child psychopathology. Based on the social interactionand ecologic model, we hypothesized that the most prox-imal interactions between parent and child (parent–childrelationship quality) would exert the greatest influence onchild psychopathology. In addition, it was hypothesizedthat proximal family factors would mediate the effects ofdistal and contextual family factors on child psychopathol-ogy.

First, however, the severity of child psychopathologyin our sample was compared with the norm group. Thecomparison showed that children with epilepsy had sig-nificantly higher levels of psychopathology. This holdsparticularly for the presence of Internalizing problems,Externalizing problems, and Attention problems, therebyconfirming findings from the existing childhood epilepsyliterature that indicated that children with epilepsy areat higher risk for the development of psychopathology(1,2,83).

Considering the relative influences of epilepsy-relatedfactors and distinct family factors on both broad-band andnarrow-band behavior problems, it appears that epilepsy-related factors were nonsignificant contributors, irrespec-tive of the type of psychopathology, whereas each typeof family factor significantly influenced almost each typeof child psychopathology (an exception appeared for So-matic complaints, distal factors as predictors of Anxi-ety/Depression, and contextual factors as predictors of In-ternalizing behavior problems). However, when each typeof family factor that appeared to be significant in the firststep of the regression analyses was entered in the last step(i.e., when the other significant family factors were con-trolled for), only proximal family factors exerted influenceon child psychopathology, whereas former significant ef-fects of distal and contextual factors disappeared. Thesefindings support the social interaction and ecologic modelin that the effects of the most proximal family factorson child psychopathology are stronger than the effects of



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the more distal and contextual family factors (15–17). In-ternalizing behavior problems (including Depression andWithdrawal) and Thought and Attention problems weresolely predicted by three family risk factors: rejection, de-pression, and family adaptation problems. In contrast, abetter equilibrium appears to exist between stressors andcompensatory factors in case of Externalizing behaviorproblems (at least as many protective factors as risk fac-tors are found).

Besides that the parent–child relationship quality wasrevealed to be the most important contributor to child psy-chopathology, even when the other family factors werecontrolled for, the results also confirm our hypothesisthat proximal family factors would mediate the effectsof distal and contextual family factors. First, parental de-pression and marital satisfaction influence Externalizingbehavior problems and Aggression through parent–childrelationship quality (both rejection and positive parent–child relationship quality). Second, the effects of parentaldepression and marital satisfaction on Delinquency aremediated by the positive parent–child relationship quality.Third, parental depression and problems of family adap-tation (except for Internalizing behavior problems) exerttheir influence on Internalizing behavior problems, Anx-iety/Depression, Withdrawal, and Thought and Attentionproblems through parental rejection. Especially in the lat-ter case, the adverse effects of distal and contextual familyrisk factors seem to spill over into poor parent–child re-lationship quality, which in turn, negatively affects childpsychopathology.

It should be pointed out that, because of the cross-sectional nature of this study, we are not able to drawany conclusions regarding causality. Although we assumethat family factors contribute to the development of psy-chopathology in children with epilepsy, it is just as pos-sible that problems within the family are reactions toalready existing pathology and thus emerge as contrib-utors to the maintenance of psychopathology. Besides, itmay also be true that child psychopathology contributesto higher levels of maternal depression, family adaptationproblems, or marital conflict, which in turn, contribute tothe maintenance of child psychopathology. Such pathwaysmay be addressed in future research that examines pedi-atric epilepsy from a child-developmental psychopathol-ogy perspective (84).

Regardless of the direction of the effects, the resultsclearly show that family factors are more strongly relatedto psychopathology in children with epilepsy than areepilepsy-related factors. This may indicate that it is es-pecially the presence of a chronic condition (i.e., genericfactors) that places demands on the family, rather than theeffects of specific disease parameters (e.g., severity andduration). It may be that the burden of epilepsy interactswith family processes and that these factors form togethera complex web of contributors to child psychopathology



(19,51). Therefore it may be of importance for future child-hood epilepsy research to use generic effects of epilepsy(for instance, the degree to which epilepsy affects thechild’s functional status, visibility and stability of the con-dition, the need for extra medical care, and the impair-ments of daily activities) (19,85). In addition, it might beimportant to study parental beliefs and child beliefs aboutthe child’s illness and treatment (10,11,13).

From meta-analytic results, it was recently deducedthat family factors were less strongly involved in Thoughtand Attention problems, as these were found to be rela-tively specific to epilepsy (3). This study showed, however,that distinct family factors also are strongly involved withThought and Attention problems. Therefore the contribu-tion of family factors to Thought and Attention problemsmay be attributed to reactions of the family to alreadyexisting pathogenetic causes of psychopathology.

Moreover, as many pathways explain psychopathologyin children with epilepsy (16,86), it may be that the in-fluence of pathogenetic contributors is easier to detectin other childhood epilepsy populations. For example, itmight be that symptomatic epilepsy syndromes, which in-volve central nervous system (CNS) lesions, are, amongother factors, strong contributors to child psychopathology(27). Therefore future research should focus on longitu-dinal studies with large samples, which would enable usto examine transactional pathways of social ecologic con-tributors and pathogenetic epilepsy contributors to childpsychopathology (19,87).

Some other limitations also should be addressed. First,psychopathology was assessed with the CBCL, a screen-ing instrument that examines the risk at psychopathology.Although it has consistently been demonstrated that theempirically based CBCL scales converge with the Diag-nostic and Statistical Manual of Mental Disorders (DSM-IV) diagnostic categories (88,89) use of the CBCL in clin-ical samples should be considered with some caution. Forinstance, Perrin et al. (85) stated that the interpretation ofSomatic complaints in clinical populations should be donecautiously, because it is difficult to discriminate betweensymptoms that may reflect psychopathology and symp-toms that reflect the effects of chronic conditions or reflectside effects of medical treatment itself. Besides, Perrin etal. also stated that Somatic complaints could be reflec-tions of psychopathology but nevertheless are interpretedas consequences of a chronic condition. Moreover, it hasbeen suggested that certain behavioral items on the CBCLchecklist reflect seizure features rather than behavior prob-lems (90). However, if parents of children with epilepsywere asked to exclude behaviors that they considered tobe seizure specific, parents still rated their children higheron levels of psychopathology than did parents of childrenfrom the general population (6).

Second, psychopathology and distinct family factorswere measured by means of parental questionnaires only.

Therefore the findings of the present study should be as-cribed to parent’s reflections on their own behaviors, be-liefs, and attitudes as well as to reflections on their chil-dren’s behaviors and should be interpreted with caution.In particular, solely using parental report of child psy-chopathology may lead to shared method variance. Thatis, parental depression may distort parental ratings of childpsychopathology, leading to artificial stronger relations.For instance, maternal reports of internalizing behaviorproblems, but not externalizing behavior problems, weresignificantly biased by maternal psychopathology (91).With regard to the current study, shared method varianceshould then be ascribed to the more internalizing types ofchild psychopathology rather than to the more externaliz-ing and unambiguous types of psychopathology such asattention problems and thought problems. It also has beendemonstrated that, contrary to former research, depressedmothers are accurate reporters of their children’s behav-iors (92,93), indicating that parental depression does notdistort parental ratings of their children’s behavior.

In addition, although inconsistent findings exist aboutthe convergence between parental reports of parenting andother informants of parenting (e.g., 94,95), it has beenfound that mothers are not inclined to rate their parent-ing behaviors more positively (95). Because parental sub-jective experiences are of importance for child behaviorand development, it can then be considered that, in par-ticular, in the context of parenting, parents seem to beoptimal informants of rating family factors and child be-havior. Therefore we concur with the optimal informantstrategy (96) that assumes that the person most engagedin a particular context is also the person in best posi-tion to rate the constructs belonging to that context. Thisstrategy, however, may eliminate additional, unique infor-mation of other informants (e.g., child report). Thereforemultisource (by means of multimethod) reporting of psy-chopathology and social ecologic factors may elucidatehow parental, child, and environmental influences affectpsychopathology in children with epilepsy, which, as aconsequence, should be more consistently applied in fu-ture research (96).

Third, as children with idiopathic epilepsy syndromesconstituted the majority of children in this study, the re-search findings may not be representative for the wholepopulation of children with epilepsy. Seizure-conditionseverity is generally assumed to contribute to adjustmentin childhood epilepsy, but one of the contributors to con-dition severity—epileptic syndrome severity—is seldomincluded in research. Dunn et al. (97) recently pleadedfor the inclusion of epilepsy-syndrome severity in themeasurement of condition severity, and they developeda syndrome-severity rating. Therefore future childhoodepilepsy research should examine whether certain sam-ples of children (children with symptomatic epilepsy)are at higher risk for psychopathology and whether the



pathways that explain psychopathology are different forthese particular groups of children.

This study showed that when epilepsy-related factorsand distinct family factors are examined within a socialinteraction and ecologic framework, it is the most prox-imal family factors that exert the greatest influence onpsychopathology in children with epilepsy. In particular,parental rejection appeared to be a risk factor that con-tributes to Internalizing behavior problems, Withdrawal,Depression, Thought, and Attention problems. Moreover,with respect to the same types of psychopathology, rejec-tion appeared to be a mediator of two other family riskfactors: depression and problems with family adaptation.Thus clinicians should be aware of the detrimental ef-fects of negative parent–child relationship quality on childpsychopathology and the risk factors that are associatedwith parent–child relationship quality. In contrast, posi-tive parent–child relationship quality may compensate foradverse effects of family risk factors in case of External-izing behavior problems. Therefore in treating childrenwith epilepsy, attention should be paid to the quality ofthe parent–child relationship and to the use of strategiesthat ameliorate and strengthen it.

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