Limits...
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

Handwriting fluency and DGM-P test box plot.Box plot of (A) handwriting fluency test and (B) DGM-P test with z-scores in the four subgroups.
© Copyright Policy
Related In: Results  -  Collection

License
getmorefigures.php?uid=PMC4488862&req=5

pone.0130883.g003: Handwriting fluency and DGM-P test box plot.Box plot of (A) handwriting fluency test and (B) DGM-P test with z-scores in the four subgroups.

Mentions: Analysis of results of the “UNO” and “LE” tests in the four subgroups revealed the z-score distribution shown in the box plot (Fig 3A); children with absence epilepsy/dysgraphia performed more slowly than controls with dysgraphia, although a statistically significant difference was detected only for the “LE” test (t = 2.2247, P = 0.037).


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)

Handwriting fluency and DGM-P test box plot.Box plot of (A) handwriting fluency test and (B) DGM-P test with z-scores in the four subgroups.
© Copyright Policy
Related In: Results  -  Collection

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

pone.0130883.g003: Handwriting fluency and DGM-P test box plot.Box plot of (A) handwriting fluency test and (B) DGM-P test with z-scores in the four subgroups.
Mentions: Analysis of results of the “UNO” and “LE” tests in the four subgroups revealed the z-score distribution shown in the box plot (Fig 3A); children with absence epilepsy/dysgraphia performed more slowly than controls with dysgraphia, although a statistically significant difference was detected only for the “LE” test (t = 2.2247, P = 0.037).

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