An Investigation of Eye Movement Dysfunctions in Children with Neurofibromatosis type 1

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An Investigation of Eye Movement Dysfunctions in Children with Neurofibromatosis type 1

Stéphanie Ducrot, Marianne Jover, Jérémy Danna, Stéphanie Maziero, Marie Vernet, Mélanie Jucla, Frédérique Audic, Yves Chaix

To cite this version:

Stéphanie Ducrot, Marianne Jover, Jérémy Danna, Stéphanie Maziero, Marie Vernet, et al.. An Investigation of Eye Movement Dysfunctions in Children with Neurofibromatosis type 1. Join Global Fibromatosis Conference, Nov 2018, Paris, France. �hal-02479462�

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An Investigation of Eye Movement Dysfunctions

in Children with Neurofibromatosis type 1

S Ducrot

, M Jover

, J Danna

, S Maziero

, M Vernet

, M Jucla

, F Audic

, Y Chaix

Today’s estimates indicate that 30-60% of Neurofibromatosis type 1 (NF1) children suffer from learning disorder, including reading

disabilities, secondary to language and/or visuospatial deficits (Chaix et al., 2017) with a profound impact on their academic achievement (Krab et al., 2008). Some studies suggest a delay in the maturation of low-level vision processes in children with NF1 (e.g. saccadic system, Lasker et al., 2003; magnocellular processing, Ribeiro et al., 2012). The findings from typically developing readers suggest a strong

relationship between reading ability and visual processing (Leibnitz, et al., 2017). The main goal of this study was to investigate the

occurrence of perceptual, visuo-attentional and oculomotor deficits in NF1 children and their potential to explain reading behavior and reading problems in this population.

2018 Joint Global Neurofibromatosis Conference Paris, november 2-6 2018

Background

Results

Conclusions

DEM test : digit reading tasks

contact : stephanie.ducrot@univ-amu.fr

1 Aix Marseille Univ, CNRS, LPL, Marseille, France 2 Aix Marseille Univ, PSYCLE, Aix-en-Provence, France

3 Aix Marseille Univ, CNRS, LNC, Aix-en-Provence, France 4 ToNIC, Toulouse NeuroImaging Center, Université de Toulouse, Inserm, UPS, France 5 URI Octogone-Lordat, Université de Toulouse, France 6 Service de Neurologie Pédiatrique, CHU Timone-Enfants, Marseille, France

7 Centre Hospitalier Universitaire de Toulouse-Purpan, Toulouse, France

Forty-two children with NF1 (9,8 ± 1,5 years, 26 girls) and forty-two control children (TD, 10 ± 1,1 years, 20 girls) participated in the study. Parents and children gave their informed consent prior to the experiment, approved by the local ethics committee. Reading and visual processing skills were respectively evaluated with the Alouette test (Lefavrais, 2005) and the DEM test (Garzia et al., 1990). We also recorded eye movements while a subgroup of 17 NF1 children (9;8 ± 1;4 years, 10 girls) and 21 control children (9;4 ± 0;9 years, 10 girls) performed an oculomotor lateralized bisection task on words, strings of hash marks and solid lines.

Method

DEM test (especially the HT) can be used clinically to distinguish NF1 with RD from NF1 without RD. Analysis of eye movement patterns represents a potential way to identify differences in the cognitive processing and visuo-attentional mechanisms underlying reading in NF1 children before the occurrence of school failure. Both language and visual aspects of reading should be targeted in intervention programs.

Chaix, Y., Lauwers-Cancesc, V.,Faure Marie, N. et al. (2017). Deficit in phonological processes: A characteristic of the neuropsychological profile of children with NF1. Child Neuropsychology, 24, 1-17. Garzia, R. P., Richman, J. E., Nicholson, S. B., & Gaines, C. S. (1990). A new visual-verbal saccade test: the development eye movement test (DEM). J Am Optom Assoc, 61, 124–35. Krab, L. C., de Goede-Bolder, A., Aarsen, F. K., Pluijm, S. M., Bouman, M. J., van der Geest, J. N., et al. (2008). Effect of simvastatin on cognitive functioning in children with neurofibromatosis type 1: a randomized controlled trial. JAMA, 300, 287–294. Lasker, A. G., Denckla, M. B., Zee, D. S. (2003). Ocular motor behavior of children with neurofibromatosis 1. Journal of Child Neurology, 18, 348–55. Lefavrais, P. (2005). Test de l'Alouette, version révisée ECPA, Paris. Leibnitz, L., Grainger, J., Muneaux, M., & Ducrot, S. (2017). Visuo-attentional processes and reading: Assessment and prospects. Topics in Cognitive Psychology, 116, 1-26. Ribeiro, M., Violante, I., Bernardina, I., Ramos, F., Saraiva, J., Reviriego, P….CasteloBranco, M. (2012). Abnormal achromatic and chromatic contrast sensitivity in neurofibromatosis.

Investigative Opthalmology and Visual Science, 53, 287-293.

No significant correlations were found between DEM parameters and the density of landing positions (r=-0.368 and r=-0.011 , for VT/density in P2-P3 and HT/density in P2-P3)

Children with NF1 presented lower reading skills than TD children (**p<.001).

-> 18 NF1 presented a Reading Delay (NF1-RD) and 24 didn’t (NF1-nonRD)

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- NF1-RD children

presented more atypical oculomotor behavior

than NF1-nonRD (p<.001).

- groups also differed according to the DEM z-scores for HT, VT/Ht

proportion (rapport) and error (**=p<.001)

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A link between reading age and performance in both tasks : DEM test (r=.55, p<.05) and ocular bisection task (r=.59, p<.05) was evidenced in NF1 children.

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- Preferred Viewing Location (PVL) for TD and NF1-nonRD between the beginning and the middle of the word

This work has benefited from support from the French Government, managed by the French National Agency for Research (ANR), under the project title DYSTAC-MAP (ANR-13-APPR-0010) and from the Association Neurofibromatoses & Recklinghausen in France.

Mobile infrared head mounted eye tracker (EYELINK II, SR Research Ltd).

NF1-RD displayed poor results in visual attention and generation of saccadic processing (e.g., less

accurate, more variability, no sensibility to the discreteness of the stimuli).

- No PVL for NF1-RD who were less accurate, and

more variable in saccadic

programming

- More flattened curve for NF1-nonRD -> TD children locate their 1st fixation mostly around P2 whereas the landing positions cover a more expanded zone, P2-P3 with NF1-nonRD

Oculomotor bisection task : participants were

asked to move their eyes as quickly as

possible to a position they thought to be the middle of the stimulus and to validate this position by pressing a button

DEM test : type of oculomotor