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

Guerrini R, Melani F, Brancati C, Ferrari AR, Brovedani P, Biggeri A, Grisotto L, Pellacani S - PLoS ONE (2015)

Bottom Line: Absence epilepsy (AE) is etiologically heterogeneous and has at times been associated with idiopathic dystonia.We compared the blink reflex recovery cycle in children belonging to all four subgroups.In children with AE, dysgraphia is highly prevalent and has a homogeneous, distinctive pathophysiological substrate consistent with idiopathic dystonia.

View Article: PubMed Central - PubMed

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.

ABSTRACT

Background: Absence epilepsy (AE) is etiologically heterogeneous and has at times been associated with idiopathic dystonia.

Objectives: 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.

No MeSH data available.


Related in: MedlinePlus

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pone.0130883.g001: Diagram of the study.

Mentions: Using the above criteria, we selected 82 patients (mean age 9.7; SD 1.78; 53.7% females) (Fig 1). We labelled as “poor writers” 38 /82 patients (mean age 9.67; SD 1.73; 57.9% females) based on assessment of two pediatric neurologists who examined recent and remote, school and homework handwriting samples. Whenever seriously poor legibility or exceedingly slow writing speed, or both, were apparent, a child neuropsychologist formally tested children using a handwriting test battery [18–19]. We subsequently divided “poor writers” with absence epilepsy into two subgroups: children with absence epilepsy/dysgraphia and children with absence epilepsy without dysgraphia.


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

Guerrini R, Melani F, Brancati C, Ferrari AR, Brovedani P, Biggeri A, Grisotto L, Pellacani S - PLoS ONE (2015)

Diagram of the study.
© Copyright Policy
Related In: Results  -  Collection

License
Show All Figures
getmorefigures.php?uid=PMC4488862&req=5

pone.0130883.g001: Diagram of the study.
Mentions: Using the above criteria, we selected 82 patients (mean age 9.7; SD 1.78; 53.7% females) (Fig 1). We labelled as “poor writers” 38 /82 patients (mean age 9.67; SD 1.73; 57.9% females) based on assessment of two pediatric neurologists who examined recent and remote, school and homework handwriting samples. Whenever seriously poor legibility or exceedingly slow writing speed, or both, were apparent, a child neuropsychologist formally tested children using a handwriting test battery [18–19]. We subsequently divided “poor writers” with absence epilepsy into two subgroups: children with absence epilepsy/dysgraphia and children with absence epilepsy without dysgraphia.

Bottom Line: Absence epilepsy (AE) is etiologically heterogeneous and has at times been associated with idiopathic dystonia.We compared the blink reflex recovery cycle in children belonging to all four subgroups.In children with AE, dysgraphia is highly prevalent and has a homogeneous, distinctive pathophysiological substrate consistent with idiopathic dystonia.

View Article: PubMed Central - PubMed

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.

ABSTRACT

Background: Absence epilepsy (AE) is etiologically heterogeneous and has at times been associated with idiopathic dystonia.

Objectives: 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.

No MeSH data available.


Related in: MedlinePlus