title: Dysgraphia as a Mild Expression of Dystonia in Children with Absence Epilepsy.
abstract: BACKGROUND
  Absence epilepsy (AE) is etiologically heterogeneous and has at times been associated with idiopathic dystonia.
  
  
  OBJECTIVE
  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.
  
  
  METHODS
  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.
  
  
  RESULTS
  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).
  
  
  CONCLUSIONS
  In children with AE, dysgraphia is highly prevalent and has a homogeneous, distinctive pathophysiological substrate consistent with idiopathic dystonia.
authors.affiliation: Pediatric Neurology and Neurogenetics Unit and Laboratories, Neuroscience Department, A. Meyer Children's Hospital, University of Florence, 50139, Florence, Italy; IRCCS Stella Maris Foundation, 56128, Pisa, Italy.
authors.forename: Renzo
authors.initials: R
authors.lastname: Guerrini
authors.affiliation: Pediatric Neurology and Neurogenetics Unit and Laboratories, Neuroscience Department, A. Meyer Children's Hospital, University of Florence, 50139, Florence, Italy.
authors.forename: Federico
authors.initials: F
authors.lastname: Melani
authors.affiliation: Pediatric Neurology and Neurogenetics Unit and Laboratories, Neuroscience Department, A. Meyer Children's Hospital, University of Florence, 50139, Florence, Italy.
authors.forename: Claudia
authors.initials: C
authors.lastname: Brancati
authors.affiliation: IRCCS Stella Maris Foundation, 56128, Pisa, Italy.
authors.forename: Anna Rita
authors.initials: AR
authors.lastname: Ferrari
authors.affiliation: IRCCS Stella Maris Foundation, 56128, Pisa, Italy.
authors.forename: Paola
authors.initials: P
authors.lastname: Brovedani
authors.affiliation: Department of Statistics, Informatics, Applications "G. Parenti", University of Florence, Florence, Italy.
authors.forename: Annibale
authors.initials: A
authors.lastname: Biggeri
authors.affiliation: Department of Statistics, Informatics, Applications "G. Parenti", University of Florence, Florence, Italy.
authors.forename: Laura
authors.initials: L
authors.lastname: Grisotto
authors.affiliation: Pediatric Neurology and Neurogenetics Unit and Laboratories, Neuroscience Department, A. Meyer Children's Hospital, University of Florence, 50139, Florence, Italy; IRCCS Stella Maris Foundation, 56128, Pisa, Italy.
authors.forename: Simona
authors.initials: S
authors.lastname: Pellacani
mesh_terms: D000293:Adolescent; D000381:Agraphia / Q000175:diagnosis* / Q000209:etiology; D016022:Case-Control Studies; D002648:Child; D004421:Dystonia / Q000175:diagnosis* / Q000209:etiology; D004832:Epilepsy, Absence / Q000150:complications* / Q000175:diagnosis; D005260:Female; D006236:Handwriting; D006801:Humans; D008297:Male