Adjustment in children with intractable epilepsy: importance of seizure duration and family factors Christopher G McCusker* MSc PhD, Consultant Clinical Psychologist, The Royal Belfast Hospital for Sick Children, Belfast, Northern Ireland; Patrick John Kennedy MSc DClinPsych, Clinical Psychologist, The Kolvin Unit, Newcastle General Hospital, Newcastle-Upon-Tyne, England; Jennifer Anderson, Paediatric Neurology Nurse Specialist; Elaine M Hicks MA MB FRCP FRCPCH, Consultant Paediatric Neurologist; Donncha Hanrahan MD MRCP, Consultant Paediatric Neurologist, The Royal Belfast Hospital for Sick Children, Belfast, Northern Ireland. *Correspondence to first author at Department of Clinical Psychology, The Royal Belfast Hospital for Sick Children, 180 Falls Road, Belfast, BT12 6BE. E-mail:
[email protected]
Seventy-five families of children with intractable epilepsy but without a severe learning disability (mean age 7 years 1 month, SD 2 years 6 months; range 2 to 12 years) who attended a regional paediatric neurology service, were surveyed. A postal questionnaire was used which included standardized measures of child and family adjustment; forty-eight families responded (64%; 31 males, 17 females). There was no significant difference between responders and non-responders in terms of age, sex, number of other chronic illnesses and disabilities, age at epilepsy diagnosis, seizure type, nor number of antiepileptic drugs currently prescribed (p>0.05). The importance of including multidimensional measures of outcome was highlighted by the finding that epilepsy, pharmacological, and psychosocial factors were differentially associated with specific adjustment difficulties. Two factors appeared to be most pervasively implicated across a range of adjustment problems: frequency of rectal diazepam administration and family patterns of relating to each other (p0.05) and responders were therefore taken to be representative of the population as a whole. THE QUESTIONNAIRE
Medical notes were used to extract information related to a child’s epilepsy diagnosis (as detailed in Table I) and medication. However, a postal questionnaire was used to quantify all other variables studied. Items in the questionnaire related to three groups of factors: epilepsy-related, pharmacological, and behavioural and psychosocial adjustment. The information listed below was ascertained: Epilepsy-related factors These were: (1) age of child at diagnosis; (2) primary seizure type – this was later collapsed into ‘mixed/generalized’ (absence, tonic–clonic, atonic, myoclonic) or ‘partial’ (complex and simple) and this information was taken from medical records;
(3) seizure frequency – total number of seizures recorded over the previous month; (4) frequency of rectal diazepam administration, i.e number of times rectal diazepam was used over the previous year. Pharmacological factors These were: (1) AEDs currently prescribed (taken from medical records); (2) number of different AEDs currently prescribed (taken from medical records); (3) pharmacotherapy adherence, i.e. number of times in the past month the parents suggested the child had ‘missed’ receiving their medication. Family factors These were listed as: (1) composition – number of parents/ guardians and siblings living with the child; (2) employment status of parents/guardians; (3) family functioning – the Family Relations Index (Moos and Moos 1986) was completed. Comprising 27 items, this is a well-validated method of assessing how family members relate to each other. Three subscales were included: Cohesion, defined as ‘the family’s degree of mutual commitment and the help and support family members provide for one another’; Conflict, defined as ‘the amount of openly expressed anger, aggression, and conflict among family members’; Expressiveness, defined as ‘the extent to which family members are encouraged to act openly and express their feelings directly’(p 2). Child factors and child adjustment These were: (1) age and sex; (2) school attendance (special or mainstream); (3) prevalence and nature of other known chronic illnesses and/or disabilities; (4) adjustment: – behaviour problems. Using the 118 item Child Behaviour Checklist (CBCL; Achenbach and Edelbrock 1983), scores on eight problem subscales were derived. Scales related to problems within the following domains: Attention/Hyperactivity; Withdrawal; Somatic Complaints; Anxiety/Depression; Delinquent Behaviour; Aggression; Thought Disturbance; and Social Problems (relationship difficulties); (5) adjustment: – social competencies. Three subscales of the CBCL were used to measure competencies in the following domains: Activities (e.g. sports, hobbies); Social (e.g. friendships, interpersonal skills); School (e.g. performance, ability, school problems). Results PARTICIPANT PROFILES AND CHILD ADJUSTMENT
The study sample comprised 31 males and 17 females (mean age was 7 years 1 month, SD 2 years 6 months). The sample had early diagnoses of epilepsy (mean age at diagnosis 2 years
Table I: Comparisons between study sample and ‘non-responders’ on key variables Variable Mean (SD) age, y:m M/F, n Those with other chronic illnesses/disabilities, n Mean (SD) age at epilepsy diagnosis, y:m ‘Partial’ seizures, n Mean (SD) number of AEDs prescribed
682
Study sample (n=48)
Non-responders (n=27)
7:1 (2:6) 31/17 33/48 2:8 (2:4) 29/48 1.7 (0.7)
7.11 (2:7) 16/11 16/27 2:6 (2:7) 15/27 1.9 (0.8)
Developmental Medicine & Child Neurology 2002, 44: 681–687
significant range on at least one subscale. In terms of the social competency scales, the figure suggests that between 18 and 21 of the sample were perceived as having significant impairment in their social, school, and recreational functioning.
8 months, SD 2 years 4 months). Forty-one of the children lived in two-parent families and at least one parent was in employment in 38 families. Almost all children had at least one sibling (n=47) and 29 had two or more. The nature of seizures experienced was collapsed into two types as described above. Nineteen children fell into the ‘mixed/generalized’ category and 29 were recorded as having ‘simple/complex partial’ seizures. Thirty-three children had at least one other additional chronic illness or disability which mainly involved some developmental delay (n=23) and asthma (n=12). Table II summarizes the epilepsy and pharmacological profile at the time of the study.
Table II: Epilepsy-related and pharmacological outcomes for study sample Epilepsy/pharmacological variables
Means (SD) for total sample and/or number of sample who met variable criterion
CHILD ADJUSTMENT
The CBCL (Achenbach and Edelbrock 1983) was used to measure child adjustment. Poor adjustment was defined as clinically significant high scores on the behaviour problem subscales, and clinically significant low scores on the social competency subscales of the checklist. These scales yield ‘T’ scores with a normal population mean of 50 (SD 10). T scores of 67+ represent the clinically significant range, and the number of children in the study sample that fell within this range on each individual subscale is summarized in Figure 1. Figure 1 shows that the greatest behavioural problems experienced by these children are related to attentional and hyperactivity disturbances. In contrast, relatively few of the sample displayed delinquency or conduct disorders, or anxiety or depression. However, an important minority of children were displaying other behavioural disturbances such as relationship difficulties, thought disturbance, and withdrawn behaviour. A proportion of the children crossed into the clinically significant range on more than one subscale and a concerning number (n=38) fell within the clinically
Total seizures over previous month None 1–19 20+
23.3 (47.9) 12/48 24/48 12/48
Rectal diazepam administration over the past year
3.4 (8.4)
Number of AEDs prescribed
1.7 (0.7)
Number prescribed each drug Valproate Lamotrigine Carbamazepine Vigabatrin Gabapentin Clonazepam Phenytoin Ethosuximide Topiramate Primodone Clobazam Phenobarbitone
23 22 21 9 6 4 2 2 2 1 1 1
Total reported adherence to medication regimen
43/48
Behaviour BehaviourProblems Problems Delinquent DelinquentBehaviour Behaviour
1
Anxiety/Depression Anxiety/Depression
9 9
Aggressive AggressiveBehaviour Behaviour
12 12
Withdrawn Behaviour Withdrawn Behaviour
16 16
Thought Disturbance Thought Disturbance
17 17
Somatic SomaticProblems Complaints
19 19
Social SocialDifficulties Difficulties
19 19
Attention/Hyperactivity Attention/Hyperactivity
33 33
Social Competencies Social Competencies Activities (recreational) Recreational Activities
18 18
School Functioning School Functioning
21 21
Social Functioning Social Functioning
20 20 00
44
88
12 12
16 16
20 20
24 24
28 28
32 32
36 36
40 40
44 44
48 48
Nr inof study Number Totalsample Sample
Figure 1: Number of children in study sample who scored in clinically significant range (T score=67+) on problem and competency subscales of Child Behaviour Checklist (Achenbach and Edelbrock 1983).
Adjustment in Children with Intractable Epilepsy Christopher McCusker et al.
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FACTORS ASSOCIATED WITH POOR ADJUSTMENT
Table III summarizes the epilepsy-related, pharmacological, and family variables that were statistically associated with any of the dimensions of adjustment (i.e. behaviour problems and/or social competencies). Where associated variables were continuous in nature, Pearson’s product–moment correlations were performed. Where the associated factor was categorical (e.g. sex or the use or not of a given AED), independent t-tests were conducted. Multiple r2s were derived from subsequent regression analyses and summarize the percentage of the adjustment variance on each dimension explained by the significant predictor variables. It can be seen that some factors had very specific associations with particular adjustment difficulties, while others were more pervasively implicated across multiple dimensions of adjustment. Results suggested that frequency of rectal diazepam administration, degree of familial cohesion and conflict, and seizure frequency were the three variables most strongly implicated in negative outcomes across multiple areas of adjustment. The extent of rectal diazepam administration in the previous year, which is likely to be indicative of longer duration seizures or, indeed, status epilepticus, was strongly implicated in negative outcomes. This variable correlated significantly with six of eight behaviour problem scales and one of the three measures of social competency. In other words, more frequent use of rectal diazepam in the past year was associated with higher scores on the behaviour problem scales of Attention/Hyperactivity (p
