Abstract
BackgroundAbsence epilepsy (AE) is etiologically heterogeneous and has at times been associated with idiopathic dystonia.ObjectivesBased on the clinical observation that children with AE often exhibit, interictally, a disorder resembling writer’s cramp but fully definable as dysgraphia, we tested the hypothesis that in this particular population dysgraphia would represent a subtle expression of dystonia.MethodsWe ascertained the prevalence of dysgraphia in 82 children with AE (mean age 9.7) and average intelligence and compared them with 89 age-, gender- and class-matched healthy children (mean age 10.57) using tests for handwriting fluency and quality, based on which we divided patients and controls into four subgroups: AE/dysgraphia, AE without dysgraphia, controls with dysgraphia and healthy controls. We compared the blink reflex recovery cycle in children belonging to all four subgroups.ResultsWe identified dysgraphia in 17/82 children with AE and in 7/89 controls (20.7 vs 7.8%; P = 0.016) with the former having a 3.4-times higher risk of dysgraphia regardless of age and gender (odd ratio: 3.49; 95% CI 1.2, 8.8%). The AE/dysgraphia subgroup performed worse than controls with dysgraphia in one test of handwriting fluency (P = 0.037) and in most trials testing handwriting quality (P< 0.02). In children with AE/dysgraphia the blink reflex showed no suppression at short interstimulus intervals, with a difference for each value emerging when comparing the study group with the three remaining subgroups (P<0.001).ConclusionsIn children with AE, dysgraphia is highly prevalent and has a homogeneous, distinctive pathophysiological substrate consistent with idiopathic dystonia.
Highlights
We identified dysgraphia in 17/82 children with Absence epilepsy (AE) and in 7/89 controls (20.7 vs 7.8%; P = 0.016) with the former having a 3.4-times higher risk of dysgraphia regardless of age and gender
In children with AE/dysgraphia the blink reflex showed no suppression at short interstimulus intervals, with a difference for each value emerging when comparing the study group with the three remaining subgroups (P
As children with absence epilepsy/dysgraphia manifested abnormal force and posturing in holding the pen, reminiscent of writer’s cramp [20,21], we investigated them with polymyographic recordings at rest and during handwriting and using the blink reflex recovery cycle
Summary
In turn, underlies variable pathophysiological backgrounds, as suggested by the association of absence epilepsy with paroxysmal dyskinesia, cognitive impairment, or other seizure types in later life. Co-occurrence of absence epilepsy and paroxysmal dyskinesia, in particular [7], suggests a common dysfunction of cortico-subcortical networks. We noticed that a substantial number of children in our cohort of typical absence epilepsy of childhood (CAE) exhibited a distinct handwriting disorder resembling writer’s cramp but fully definable as dysgraphia, a disorder of written expression featuring writing skills substantially below those expected considering age and intelligence [8]. Absence epilepsy (AE) is etiologically heterogeneous and has at times been associated with idiopathic dystonia
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