PLoS ONE (Jan 2015)

Dysgraphia as a Mild Expression of Dystonia in Children with Absence Epilepsy.

  • Renzo Guerrini,
  • Federico Melani,
  • Claudia Brancati,
  • Anna Rita Ferrari,
  • Paola Brovedani,
  • Annibale Biggeri,
  • Laura Grisotto,
  • Simona Pellacani

DOI
https://doi.org/10.1371/journal.pone.0130883
Journal volume & issue
Vol. 10, no. 7
p. e0130883

Abstract

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Absence epilepsy (AE) is etiologically heterogeneous and has at times been associated with idiopathic dystonia.Based 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.We 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.We 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).In children with AE, dysgraphia is highly prevalent and has a homogeneous, distinctive pathophysiological substrate consistent with idiopathic dystonia.