Biotteau, M. 1 , Tournay, E. 2 , Baudou, E. 1, 3 , Lelong, S. 3 , Iannuzzi, S. 3 , Faure-Marie, N. 3 , Schweitzer, E. 4 , Rodriguez, D. 5, 6, 7 , Kemlin, I. 5 , Dorison, N. 5 , Rivier, F. 8, 9, 10 , Carneiro, M. 8 , Preclaire, E. 8 , Barbarot, S. 11 , Castelneau, P. 12, 13 , Lauwers-Cancès, V. 2 & Chaix, Y. . 1, 3
1 ToNIC, Toulouse NeuroImaging Center, Université de Toulouse, Inserm, UPS, France
2 Epidemiology Department, Toulouse University Hospital, Toulouse, France
3 Children?s Hospital, Purpan University Hospital, Toulouse, France
4 Neuropediatrics & Disabilities Unit, Gatien de Clocheville Children?s Hospital, Tours University Hospital, Tours, France
5 Pediatric Neurology Department & Neurofibromatosis Referral Center, Armand Trousseau Hospital, East Paris University Hospital, Paris, France
6 University of Paris VI Pierre & Marie Curie, Sorbonne Universities, Paris, France
7 ?Neuroprotection of the Developing Brain? Joint Research Unit (U1141), INSERM, Paris, France
8 Neuropediatric Department & Language Disorders Referral Center, Montpellier Regional University Hospital, Montpellier, France
9 Faculty of Medicine, University of Montpellier, Montpellier, France
10 ?Physiology & Experimental Medicine? Research Unit (U1046 INSERM & UMR 9214 CNRS), University of Montpellier, Montpellier, France
11 Dermatology Clinic, H&ocirc;tel-Dieu University Hospital, Nantes, France
12 ?Brain &amp; Imaging? Joint Research Unit (UMR 930), Bretonneau Hospital, Tours Regional University Hospital, Tours, France
13 University of Tours Fran&ccedil;ois Rabelais, Tours, France
Context: Neurofibromatosis type 1 (NF1) is consider as one of the most common childhood genetic disorders, affecting approximately 1 for 3000 individuals (Evans et al., 2010). Risk for many central nervous system abnormalities are significant, and highlight brain abnormalities (Sabol et al., 2011). A broad spectrum of cognitive deficits occur in 30-70% of cases (Hyman et al., 2005) and negatively affect quality of life. In particular, reading disorders is one of the most concerns of NF1 children: 67% have problems in one or more reading subskills, 75% of these fulfill criteria for phonological dyslexia and 20% meeting criteria for mixed dyslexia (Watts et al, 2008). In NF1 as in others, reading is a multifaceted skill and difficulties in this domain can result from difficulties in other areas. Especially, attention was identified as a possible contributory reading impairment factor in others pathologies and is consider as the main cognitive concern of children with NF1 (Isenberg et al., 2013). We wanted to observe if there is an impact of attention on reading comprehension; what assessment could enable to provide a link with reading comprehension measures; and if there was a difference in such impact between genetic (NF1) or developmental disorders (dyslexia).
Method: A multicenter, cross-sectional study was conducted on two groups of 75 children (72?-78?) with or without NF1 (8-12yo), matched for age, sex, handedness, and reading level thus forming a continuum from good to poors readers in both NF1 and non-NF1 groups (NF1 children with reading impairment were matched with children with dyslexia, whereas NF1 children with reading impairment were matched with typically children). [Excluded: mental retardation, neurological/psychiatric disorder]. Attentional skills were assessed combining (1) a parental questionnaire (CBCL, Achenbach 2001) and (2) performance-based assessment (CPTII, Conners 2000). Reading comprehension (text and sentences) was assessed through a standardized reading comprehension test (ORLEC, Lobrot 1973).
Results: For both groups, the attention performance-based scores were associated to the text and sentences comprehension ability (p=0.0235 and p=0.0164 respectively), while indirect questionnaire scores of attention were associated only to sentences comprehension (p=0.0263). The correlations between questionnaire and performance-based measures were low on NF1 and non-NF1 children.
Discussion: Three important findings are to be considered:
(1) attention capabilities (but not impulsivity and hyperactivity) greatly influence reading comprehension (text and sentence) for NF1 as non-NF1 children. Impact of attention processes in reading skills is the same for both;
(2) predictors of a good reading comprehension are not significantly different between the 2 populations and include 3 factors: attention level, IQ score and Alouette accuracy index;
(3) for both NF1 and non-NF1 children, indirect observer-rated (questionnaires) and direct performance-based measures of attention do not assess the same thing.
Altogether, we show that reading comprehension disorders, that has caused by genetic or developmental disturbance, behaves the same in both cases, greatly influenced by attention. However, no single instrument capture all aspects of attention function and the different attention tools are linked to different reading skills components. Thus, children should benefit from a multimodal assessment of attention skills when reading comprehension is evaluated.