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Título

Social/Cognitive and Responsibility domains are the most affected functioning areas in children and adolescents with epilepsy.

Resumo

1. Introduction
Children and adolescents with epilepsy (CAWE) may have emotional and social difficulties, behavioral and psychiatric disorders. Age of onset of the disease and neurobehavioral problems are associated with limitations in child's and caregivers' daily activities. Functioning covers body functions and structures, activity and participation, while disability consists of impairments, activity limitation or restrictions in participation. Functional skills can be defined as the person's ability to take care of himself and to adapt to different contexts. Epileptic seizures, regardless of frequency, are associated with lower functioning levels. Relevant factors for assessing epilepsy impact on functioning in CAWE are number of antiseizure medicines (ASM), onset age, illness duration and presence of tonic-clonic seizures. The relationship between functioning and epilepsy is complex, not explained only by disease severity. The aim of this study is to evaluate functioning through analysis of capacity and performance in daily activities and factors related to them in CAWE without moderate/severe disability.
2. Methods
After approval by local Ethics Committee patients were recruited (March 2019- March 2020) at the outpatient unit of a tertiary care hospital in São Paulo, Brazil. Inclusion criteria were: age 4-18 years, epilepsy with regular treatment and seizures in last year, and exclusion criteria, severe clinical, physical, intellectual and neuropsychiatric comorbidities. We used ILAE definitions (2017) for classification of epileptic seizures, epilepsy and epileptic syndromes. Pediatric Evaluation of Disability Inventory Computer Adaptive Test (PEDI-CAT) (validation Portuguese-Brazil) assessed functional abilities in domains: Daily activities; Mobility; Social/cognitive; Responsibility. Its balanced content consists of 30 items per domain, according to age, including 276 functional activities acquired during childhood, adolescence, and young adult life. Aimed to people aged 1-21 years, PEDI-CAT seeks to identify functional deficits and evaluate treatment/rehabilitation. The Wechsler Abbreviated Scale of Intelligence (WASI) was used for IQ and SON-R 2½-7 [a] was applied in children aged 4-5 years old. Burden Interview (BI) scale (validation Portuguese-Brazil) was used to assess caregiver burden. Caregivers and patients were recruited by the attending neurologist. Two psychologists applied PEDI-CAT Content-balanced version and BI to caregivers and intelligence tests to patients. Clinical and demographic data were collected in medical records and clinical interview. Associations between PEDI-CAT, IQ and seizure-related variables were analyzed (ANOVA). Spearman’s correlation was used for continuous variables. Multiple Linear Regression method established multivariate correlation between BI and PEDI-CAT. We regarded significant values p≤0.05 .
3. Results
Forty-two children and adolescents and caregivers were evaluated (23 boys, 54.8%) with mean age of 10.4 years (±3.72). Mean epilepsy onset age was 4.5 years (±3.43) and epilepsy duration, 5.9 years (±3.94). Mean IQ were: VIQ 75.5 (±16.78); PIQ 76.5 (±17.00); and FIQ 73.4 (±16.93).
Mean results of PEDI-CAT scale, according to the normative table (z-score), were: Daily Activities [-1.34]; Mobility [-1.07]; Social/Cognitive [-2.20]; Responsibility [-2.07]. All domains of PEDI-CAT showed no significant differences when analyzed with clinical variables such as seizure onset age, focal or generalized epilepsy, etiology and seizure frequency. ASM number showed no differences regarding Daily Activities, Mobility and Responsibility. Two or more ASM were associated with lower scores (p=0.045) in Social/Cognitive domain. Diagnosis of genetically determined generalized electroclinic syndrome was associated with higher values in Daily Activities (p=0.045), Mobility (p=0.042) and Social/Cognitive (p=0.059). The same was observed with history of absence and myoclonic seizures for Mobility (p=0.042) and a trend for Social/Cognitive (p=0.059) and Daily Activities (p=0.097).
Daily Activities (r=-0.366;p=0.017) and Mobility (r=-0.450;p=0.003) showed negative correlation with epilepsy duration. Negative correlations were also found in Social/Cognitive with age (r=-0.476;p=0.001) and epilepsy duration (r=-0.498;p=0.001). Finally, in Responsibility, negative correlation was observed with epilepsy duration (r=-0.430;p=0.005).
There was positive correlation between PEDI-CAT and VIQ: Daily Activities (r=0.635;p<0.001); Mobility (r=0.437;p=0.004); Social/Cognitive (r=0.644;p<0.001); and Responsibility (r=0.575;p<0.001). There were also positive correlations with PIQ: Daily Activities (r=0.533;p<0.001); Mobility (r=0.353;p=0.022); Social/Cognitive (r=0.434;p<0.004); and Responsibility (r=0.391;p<0.010), and positive correlation with FIQ: Daily Activities (r=0.631;p<0.001); Mobility (r= 0.439;p=0.004); Social/Cognitive (r=0.585;p<0.001); and Responsibility (r=0.492;p=0.001).
There was negative correlation between PEDI-CAT and BI: Daily Activities (r=-0.355;p=0.021); Mobility (r=-0.424;p=0.005); Social/Cognitive (r=-0.480;p=0.001); Responsibility (r=-0.347;p=0.025).
Multiple Linear Regression explained 25.5% of data variability, in domains: Daily Activities 14.7% (p=0.622); Mobility 24.6% (p=0.373); Social/Cognitive 57% (p=0.064); Responsibility 3.8% (p=0.901).
4. Discussion
In this sample of CAWE without moderate/severe disability PEDI-CAT identified most impaired functional skills. Although results in Daily Activities and Mobility were considered within expected age range, it was possible to identify impairment in Social/Cognitive and Responsibility fields. PEDI-CAT results were negatively correlated with disease duration and caregiver burden, while positively correlated with IQ. Higher results in Daily Activities, Mobility and Social/Cognitive were associated with diagnosis of absence and myoclonic seizures and genetically determined generalized electroclinic syndromes. Epilepsy duration was the main clinical variable related to domains, in accordance with others. However, this was different in a study that evaluated children with epilepsy and adaptive behavior with Behavior Assessment System for Children scale.
History of absence/myoclonic seizures and genetically determined generalized electroclinic syndromes showed association with higher results in Daily Activities, Mobility and Social/Cognitive. We did not identify studies with similar findings. Due to exclusion criteria, these diagnosed electroclinic syndromes had less impact on functional abilities compared to other with greater impact on development, such as epileptic and developmental encephalopathies. Nevertheless, it highlights that epilepsy without moderate/severe disability can affect functioning. One study identified that children with epilepsy who had one or more epileptic seizures in the past year had higher impairment compared to those with controlled seizures. They analyzed frequency and found no significant association with functional skills development, suggesting that children with controlled seizures, are also at risk of functional deficits. Another showed that there was no significant difference in adaptive skills among children with focal and generalized seizures and deficits were found in adaptive behavior of both.
Daily Activities scored below normative range in approximately 40% in our study. Disease duration directly impacted patient’s independence development. Lower scores in activities related to daily living skills were found in other surveys. In a longitudinal study with children with epilepsy in first years of life, it was identified significant statistical decline of daily living skills over time, especially in children with symptomatic etiology, encephalopathies and drug resistance. Mobility domain presented the most developed skills in our study and disease duration influenced activities performance that involved gross motor skills.
In Social/Cognitive domain participants’ performance was far below normative values. Epilepsy duration and participants’ age were identified as risk factors for impairments in these areas. Some studies have found negative association between disease severity and parental behaviors characterized by intensified care, expanded monitoring of activities and resistance to encourage autonomy. Epilepsy clinical variables and family/school context seem to be predictive of impairment in this area. Learning problems contribute to social deficits and underdevelopment of communication and stigma affects social bonds formation. High level of parental anxiety can influence child's involvement in social situations and emotional regulation necessary for group functioning.
Regarding Responsibility, patients had lower performance and greater deficit compared to other domains. This ability reflects management of life tasks that are important in transition to adulthood and independent living, such as preparing meals, planning and following schedules, managing health, safety and using public transport. This aims to assess patient performance, considering degree of responsibility assumed by family and children in routine functions. In this study, epilepsy duration showed negative correlation with Responsibility that involves participation in life situations–implying difficulties in typical cultural activities and tendency to increased deficit according to illness span. The longer disease duration, the greater number of seizures throughout life, and consequently longer continuous abnormal brain functioning, which can affect adaptive behavior development and contribute to family restrictions.
PEDI-CAT domains were also positively correlated with IQ, indicating that better cognitive skills influence daily life activities. In children with focal or generalized seizures, some studies showed that adaptive behavior can be affected by IQ, learning difficulties and epilepsy severity. Nevertheless, another study indicated that functioning in children with epilepsy with IQ between 50-90 and learning difficulties were similar and independent of IQ. Even in children with middle range IQ, adaptive functioning influenced more academic performance.
BI results in this study showed that caregiver's burden was negatively correlated with patient's functional abilities. In contrast to other studies, epilepsy clinical variables did not show significant correlation. This can be due to the sample composed by children with active epilepsy without severe comorbidities or ASM polytherapy. One study with BI on 106 caregivers of children with epilepsy identified mild/moderate overload and high levels of anxiety/depression. In addition, behavioral difficulties and development delays also impacted well-being of these patients and family dynamics. Another study showed that onset age and neurobehavioral problems were associated with limitations in child's and caregivers' daily activities.
5. Conclusion
Patients with epilepsy without moderate/severe disability had deficits in Social/Cognitive and Responsibility domains of PEDI-CAT scale. Longer epilepsy duration in children and adolescents was associated with lower functioning, reinforcing the importance of better seizure control to alleviate physical and psychosocial difficulties. IQ and caregiver burden were correlated with functioning in CAWE which should be supported in clinical practice.
Acknowledgments: CNPq (Brazil).

Área

EPILEPSIA NA INFÂNCIA