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Autism

Outcome of young children with autism : Does the amount of intervention influence developmental trajectories?
Cline Darrou, Ren Pry, Eric Pernon, Ccile Michelon, Charles Aussilloux and Amaria Baghdadli Autism 2010 14: 663 DOI: 10.1177/1362361310374156 The online version of this article can be found at: http://aut.sagepub.com/content/14/6/663

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Outcome of young children with autism


Does the amount of intervention inuence developmental trajectories?
CLINE DARROU
University, France Montpellier I University and Montpellier III

autism 2010 SAGE Publications and The National Autistic Society Vol 14(6) 663677; 374156 1362-3613(2010)

R E N P RY
University, France

Montpellier I University and Montpellier III University, France Montpellier I University and Montpellier III Montpellier I University, France Montpellier I University and Montpellier I University and

ERIC PERNON

CCILE MICHELON
Montpellier III University, France

C H A R L E S AU S S I L L O U X A M A R I A B AG H DA D L I
Montpellier III University, France

A B S T R AC T

The study aims were to identify developmental trajectories of young children with autism and investigate their prognostic factors. The participants were 208 children, assessed first at the age of 5 years, followed longitudinally, and reassessed 3 years later. The childrens clinical characteristics and the interventions received were recorded. The results indicated two distinct outcome groups with more stability than change. When changes did occur, they pertained to symptom severity (which decreased) and speech level and adaptive behavior (which improved). A logistic regression analysis pointed out two main risk factors (symptom severity and speech level) and two main protection factors (communication skills and person-related cognition). Surprisingly, the amount of intervention (in terms of number of hours) was not related to outcome.

K E Y WO R D S

autism; children; intervention; outcome; risk factors

ADDRESS Correspondence should be addressed to: A M A R I A B AG H DA D L I , Child and Adolescent Psychiatry Department, Centre de Ressources Autisme, Clinique PeyrePlantade, Centre Hospitalier Universitaire de Montpellier, 291 avenue du Doyen Giraud, 34295 Montpellier, Cedex 5, France. e-mail: cent-ress-autisme@chu-montpellier.fr

Copyright The Author(s), 2010. Reprints and permissions: http://www.sagepub.co.uk/journalspermissions.nav DOI: 10.1177/1362361310374156
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14(6) Autism is a developmental disorder with an early-childhood onset that is characterized by signicant impairment in socialization and communication, as well as repetitive patterns of behavior (American Psychiatric Association, 1994; WHO, 1993). The question of outcome and prognosis has been raised since the very rst studies on autism (Kanner, 1971). Most studies describe outcome on the basis of social adaptation, intelligence quotient (IQ), and speech, but the results are difcult to compare because different samples, models, and variables were used. Longitudinal studies show a wide range of outcomes in adaptive behavior domains (Bolte and Poustka, 2002), degree of autism (Jonsdottir et al., 2006; Szatmari et al., 2003), speech (Eaves and Ho, 1996; Turner et al., 2006), and IQ (Eaves and Ho, 1996; Szatmari et al., 2003). There is a consensus regarding the importance, as prognostic factors, of IQ and speech level measured in childhood. The absence of speech at age 5 and an IQ under 50 predict the worst outcomes (Lord et al., 1989; Pry et al., 2005). Most studies on the outcome of autistic children (Charman et al., 2004) describe the stability or change of one or more of the childs characteristics. The variables most often studied are social adaptation and cognitive factors. Social adaptation was described by Lotter (1974) in terms of four outcome levels (good, satisfactory, poor, and very poor). Pursuing this classication, Fombonne et al. (1989) described the heterogeneity of social adaptation, although the majority of the outcomes they examined were qualied as poor in Lotters (1974) sense of the term. McGovern and Sigman (2005) added that symptom severity can change even if the diagnosis remains the same. On the issue of IQ stability, the literature is inconsistent. Lord and Schopler (1989) and Szatmari et al. (2000) showed that IQ changed little across development.Yet studies by Ballaban-Gil et al. (1996) pointed out IQ heterogeneity when individual outcomes were considered separately. Currently, there is a consensus regarding the predictive power of both speech and cognitive level (Lord and Schopler, 1989). While the importance of IQ and speech level is not questioned, the impact of intervention is poorly understood. Many studies suggest that intervention can affect outcome. Lovaas (1987) reported that an intensive behavioral program dramatically improved IQ. The literature as a whole suggests that a behavioral intervention program is more effective if it is intensive (Eldevik et al., 2006; McEachin et al., 1993; Smith et al., 1997). Several studies suggest that intensive, eclectic intervention approaches are not as effective as intensive behavioral approaches (Gabriels et al., 2001; Jonsdottir et al., 2006). The sheer amount of intervention, then, is not the only factor related to outcome the type of intervention is also an important component of prognosis. The effect of intervention also depends on the childs clinical characteristics, with the best outcomes being achieved
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by children with the highest cognitive level (Howard et al., 2005; Smith et al., 1997). In France, intervention programs for children with pervasive developmental disorders (PDD) are very eclectic and vary considerably in terms of theories, number of hours, and types. Usually, behavioral programs are not used, but rather a mixture of special education, rehabilitation, and psychotherapy. Furthermore, descriptive data on intervention programs are lacking and there are no controlled studies about any of the approaches administered in France to children with PDD. The present article is a follow-up to Baghdadli et al. (2007). After demonstrating the importance of speech and cognitive level, these authors looked into the effect of educational and therapeutic intervention on the outcome of their PDD children. The current article is based on the same sample and attempts to predict outcome, but using a different approach. Unlike the paper published in 2007, which focused on measuring the difference in the childrens scores on adaptive skills between Time 1 and Time 2 and then explored prognostic factors using a multivariate analysis, the present paper uses latent class analysis and logistic regression in order to identify subgroups of children at Time 1, examine their outcome at Time 2, and thus investigate their risk and protection factors (including intervention variables). The purpose of this study was to determine the predictive factors, including intervention, linked to the developmental pathways of children with autism between the ages of 5 and 8.

Method
Setting This study was collaborative, longitudinal, and prospective (Aussilloux et al., 2001). All participants were contacted between December 1997 and December 1999. They were being treated at 51 centers that service children with psychiatric disorders (in France, Switzerland, and Luxemburg). Most of the centers use institutional psychotherapy approaches, but with differences in practices and forms of service provision. Usually, intervention is administered via comprehensive, multidisciplinary approaches that can include psychotherapy, educational sessions, speech therapy, and psychomotor rehabilitation. Intervention approaches are eclectic and vary considerably across individuals. Participants The eligibility criteria for the study were diagnosis of PDD on the basis of the ICD-10 checklist, age under 7, and written parental consent. In all, 280
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Description of clinical characteristics of the sample at inclusion n %

Table 1

Gender Male Female PDD diagnosis category Infantile autism Atypical autism Others Presence of medical condition Genetic or malformation syndrome Epilepsy Perinatal antecedents Intervention hours per week

166 42 175 28 5 7 16 10 Median 30.5

79.8 20.2 84.1 13.4 2.5 3.3 7.7 4.8 IntQa 2438

a. IntQ (interquartile) = Q25-Q75 (rst and third quartile).

children were included in the cohort and rst assessed at a median age of 5 years. Three years later, there were only 222 children (74%) from the original cohort: 16 children could not be found, 43 had been dropped from the study because their family or center had decided not to continue participation, and 2 children had died. The characteristics of the sample at the two testing times are given in Table 2.
Table 2 Description of clinical variables at times T1 and T2 T1 Median Total CARS score Intervention hours per week Person-related cognition (months) Communication skills (months) Speech level Sentences Words No speech Developmental quotient (DQ) DQ > 70 DQ 5069 DQ < 49
a. IntQ = Q25-Q75.

T2 IntQa 3141 2438 926.5 1119 % 20.8 24.2 55 4.3 23.1 72.6 Median 32.5 30.5 26.5 19 n 96 35 76 15 41 152 IntQa 2639 2438 938.5 1443 % 47 16 37 7.2 19.1 73.7

36.5 30.5 20.5 15 n 43 50 115 9 48 151

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Procedure
Enrollment phase All patients in the 51 centers were contacted and screened for a diagnosis of autism. Before the enrollment phase, staff training sessions were organized during which diagnoses of children with autism and other developmental disorders were rated blindly. Assignment of each child to the infantile autism, atypical autism, or Aspergers syndrome cohort was done by the centers in accordance with ICD-10 ratings, assessments, and the clinical judgments of an experienced, multidisciplinary team. To qualify for a diagnosis of atypical autism, a child had to have at least two areas of impairment according to ICD-10 criteria for infantile autism. All diagnoses were validated by two independent, experienced psychiatrists (from the research staff) on the basis of medical records and videotaped observations made during enrollment. Regular group meetings were also organized during the enrollment phase to improve clinical agreement between the centers collaborating in the study. The overall diagnosis agreement rate between the two clinicians resulted in a kappa value of 0.76. Evaluation Assessment at Time 1 (T1) included retrospective data collection, psychological testing, autism rating, and a measure of speech. The evaluations were performed by experienced, trained professionals. Three years later, at follow-up time (T2), the children included in the cohort were reassessed using the same procedures. To ensure impartiality, the follow-up assessors were different from the initial ones. Data collection was validated by a research staff member. Measures
Degree of autism Autism was rated using the Childhood Autism Rating Scale (CARS; Schopler et al., 1980). The total CARS score was used as the measure of autistic symptom severity. Scoring was based on a 20-minute videotape of the child interacting with an adult. Each child was scored twice by two trained raters. Cases subject to disagreement were discussed in order to obtain the best nal estimate. Inter-rater agreement for the total CARS score calculated using an intraclass correlation coefcient (ICC) was 0.73 (95% condence interval: CI = [0.67; 0.78]). Speech The childrens speech was assessed by experienced clinicians using observation data and parental interviews based on an item adapted from the Autism Diagnostic Interview (ADI-R; Lord et al., 1994). Speech 667

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14(6) level was rated good = sentences for spontaneous functional speech with sentences made up of three or more words, intermediate = words for no three-word sentences but the use of at least ve different words, and poor = no speech for the use of fewer than ve words.
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Adaptive behavior and developmental level The Vineland Adaptive Behavior Scale or VABS (Sparrow et al., 1984) was used to assess adaptive behavior in communication, daily living skills and socialization. The American norms for this scale have proven valid for the French population. VABS scoring was based on a semi-structured interview with the parents conducted by a trained professional. Given that the measures could be expressed either as standard scores or in developmental age, we chose developmental months so as to improve comparability across tests. In order to specify the extent of mental retardation, we used the daily living skills score as an index of nonverbal development age, and a developmental quotient [DQ = (developmental age/chronological age) / 100] was calculated for this domain. This approach alleviates problems related to using a range of tests to assess discrepancies in childrens developmental levels, because IQ correlates with adaptive behaviors and daily living skills are relatively independent of language (Stone et al., 1999). Person-related cognition Considering the limitations of testing large samples containing low-functioning subjects, and the problematic use of global psychological measures in autism, we assessed psychological development in terms of person-related cognition. Person-related cognition was measured using Seibert and Hogans scale (Seibert and Hogan, 1982). The French version (ECSP) has already been standardized and validated. The ECSP denes four levels (simple/complex: 0 to 6 months; conventional gesture: 7 to 16 months; conventional speech: 17 to 24 months; symbolic play: 25 to 30 months). Three subscales were used (social interaction, joint attention, and behavioral adjustment) and coded based on behavioral observation during a session in which the child was interacting with an investigator. Intervention Data about the type and amount of intervention per week delivered in special education and regular classrooms were gathered from a detailed report. Only periods that lasted more than three consecutive months and interventions administered by licensed professionals were considered. Independent raters calculated the total number of hours of intervention provided per week. A mean per-child measure of intervention intensity was computed. 668

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Statistical analysis
Data collection was centralized at the Centre de Ressources-Autism in Montpellier, France. The clinical characteristics of the children were described globally with medians and with interquartiles (75th and 25th percentiles) for the numerical variables. The normality of the distribution was checked using the Shapiro-Wilk test, and in terms of category frequencies. Latent Class Analysis (LCA) using Mplus software (version 3.0) was conducted on four variables: chronological age, developmental age, degree of autism (CARS), and speech level (sentences, words, no speech) at the two testing times, in order to identify subgroups of individuals. Next, the developmental trajectory of each child was dened according to his/her group of membership at the two testing times. A high-level group (HL) and a low-level group (LL) were identied using LCA, and then a univariate analysis was performed using the chisquare test, or Fishers exact test. The explained variable was belonging to Group LL at Time 2 and the explanatory variables were gender, medical condition, speech level, DQ, hours of intervention, communication, socialization, person-related cognition and the parents socioeconomic status at Time 1. A logistic regression was computed to nd out which of the clinical and interventional variables found to be signicantly linked in the univariate analysis (at a cutoff of 0.2) could be considered to predict a negative outcome. A stepwise selection procedure was used with an input cutoff at 0.2 and an output cutoff at 0.1. The t of the model was assessed using the Hosmer-Lemeshow Test.

Results
Developmental trajectories At the two testing times, the children in the high-level group (HL) had a lower degree of autism, a higher speech level, and a higher developmental age than the children in the low level group (LL) (see Table 3). The majority of the children in the sample (72%) had a stable outcome prole: 44 children belonged to Group HL at both testing times, and 105 children belonged to Group LL at both testing times. For 149 children, then, the prole remained stable over the past three years. Stability in this case means that the children continued to progress, but at a slower pace than typical children, thereby causing a lag with respect to the norm. Changes occurred for the other 28% of the children in the sample: 55 children (27%) shifted from Group LL to Group HL and 3 children (1%) shifted from Group HL to Group LL. For the children who belonged to
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T1 Group HL N = 47 Median 68 37 30 32.5 30 n 40 4 3 20 1 1 3 46 112 % n % 1 22 54 2732 38 3442 26.535.5 9 921 20.535 12 1016 3144.5 19 1624 48 43.5 38.5 25.5 n 90 7 3 6078 55 4470 95 IntQa Median Median IntQa IntQa 85107 3861 2960 32.556.5 2230 % 43 3 1 n 6 28 74 Group LL N = 161 Group HL N = 100 T2 Group LL N = 108 Median 102 25 14 9 39.5 IntQa 85112 1925 1216 921 3442 % 2 13 38

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Table 3

Description of the groups at the two testing times

Chronological age (months)

Developmental age (months)

Communication (months)

Person related-cognition (months)

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Degree of autism

Speech

Sentences Words No speech

a. IntQ = Q25-Q75.

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Group HL at both testing times, autism severity decreased signicantly (t(43) = 6.38, p = .00) and developmental age increased (t(43) = 8.34, p = .00). The children who moved up from Group LL to Group HL also beneted from a signicant improvement in autism severity (t(55) = 10.3, p = .00) and developmental age (t(55) = 13.5, p = .00). As for the children who remained in Group LL, only their developmental age rose signicantly (t(104) = 10.18, p = .00). Most of the children in Group HL exhibited speech at time T1 (20%), and all of these children still did so at time T2. For Group LL, most of the children did not exhibit speech at either testing time. Finally, among the children with a positively-evolving prole who did not speak at T1, 43% did speak at T2.

Univariate analysis The relationship between outcome (Group HL / Group LL at T2) and initial clinical characteristics (i.e., degree of autism, communication, socialization, person-related cognition, hours of intervention, presence of a medical condition and speech level) was examined. The results indicated signicant differences between Group HL and Group LL at T2 on the following initial clinical characteristics: degree of autism at T1 (HLmedian = 31.7; LLmed = 40; p < .001), communication at T1 (HLmed = 18; LLmed = 12; p < .001), socialization at T1 (HLmed = 19.5; LLmed = 10.5; p < .001), person-related cognition at T1 (HLmed = 26.5; LLmed = 9; p < .001), hours of intervention (HLmed = 33.6; LLmed = 29.7; p = .015), presence of a medical condition at T1 (HL% = 29; LL% = 39; p = .02), and speech level at T1 (HLsentences(%)= 20; LLsentences(%) = 0.4; p < .001). These results show that the children with the best outcomes were those without a medical condition and those exhibiting at least one of the following characteristics at T1: more hours of intervention, lower degree of autism, and better levels of communication and socialization. By contrast, no link was detected with the parents socioeconomic status. Risk and protection factors The variables signicantly linked to outcome in the unvariate analysis were entered into the logistic regression model. The results pointed out two risk factors and two protection factors (Table 4) of a negative outcome (i.e., belonging to Group LL at Time 2). The number of hours of intervention, however, had no effect on outcome. The two risk factors identied were autism severity (CARS score) and speech level. At T1, a difference of 5 CARS points multiplied the likelihood of belonging to Group LL at T2 by 1.75 (odds ratio OR = 1.75, CI = [1.082.91]). For those children who had no speech at T1, the risk of
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Table 4 Logistic regression: Risk and protection factors of a poor outcome (Group LL) Unit Degree of autism (CARS score) Presence of speech Person related-cognition (months) Communication (months) 5 1 10 10 OR 1.75 23.92 0.20 0.136 95% 1.08 1.80 0.08 0.02 CI 2.91 820 0.44 0.50 p-wald 0.0001 0.04 0.0001 0.0006

Note. Hosmer and Lemeshow Goodness-of-Fit 0.99; percent concordant 94.5. OR = odds ratio; CI = condence interval.

belonging to Group LL at T2 was 23.9 times higher than for children who exhibited speech (OR = 23.92, CI = [1.80820]). The protection factors observed at T1 were the childs person-related cognition level and communication skills. For an increase of 10 months in person cognition, the likelihood of belonging to Group LL at T2 was divided by ve (OR = 0.20, CI = [0.080.44]). For example, a child who had a person-cognition age of 30 months was ve times more likely to belong to Group LL than a child with 40 months on this variable. Likewise, a communication skill increase of 10 months divided the risk of belonging to Group LL at T2 by 7.3 (OR = 0.13, CI = [0.020.50]).

Discussion
Our results show that, for the sample as a whole, there were signicant changes during the follow-up period in each domain considered. Firstly, as shown in previous studies, there was a decrease in the degree of autism as measured by the CARS (Jonsdottir et al., 2006). Secondly, there was an improvement in speech level, with 46% of the children producing sentences at the follow-up assessment whereas only 20% did so initially. This frequency is lower than the 70% reported by the only two studies that have used these same parameters to examine speech (Chakrabarti and Fombonne, 2001; Jonsdottir et al., 2006). The discrepancy is probably due to the different ages of the samples (60 months for our children vs. 41 months for theirs). Thirdly, although the initial 72% of the children with a DQ under 50 did not move up into the higher group, developmental gains were observed in the areas of communication and person related cognition. In our cohort, four outcome proles were noted. The rst (27% of the sample) was characterized by a positive outcome with improved speech, developmental gains (mean rise of 21 months), and decreased autism severity (mean drop of 6 points), which is consistent with past results. In spite of these important developmental gains (in months), the median 672

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developmental quotient at T2 was 50 (IntQ: 4061), which means that even in the highest functioning group, developmental age did not go up as quickly as chronological age, as it does in typical children. The second and third proles (21% and 50%, respectively) were characterized by stability. Note that stability here does not imply a lack of improvement on individual factors, but that the child belonged to the same group (highlevel or low-level) at both testing times. Despite their stability, these two proles involved slight changes consistent with those observed by Gabriels et al. (2001). Most of the children in our high-level group improved in developmental age and in degree of autism. All of these children exhibited speech. On the other hand, the low-level group improved less in developmental age, had an unchanged autism severity level, and for most of them, no speech. The fourth prole (2%) was characterized by developmental regression. In other studies where a regression prole has been observed (Davidovitch et al., 2000), the prevalence was much higher (between 20% and 40%) than in our sample. The low frequency of regression in our sample may be due to the higher mean chronological age (59 months) as compared to the younger populations studied by Davidovitch et al. (2000). The different outcome proles in our sample were related to degree of autism, speech level, communication skills, and person-related cognition. Autism severity and speech level are widely acknowledged as very robust prognostic factors no matter what methodology is used (Baghdadli et al., 2007; Sutera et al., 2007). The presence or absence of speech is an important variable that has long been considered a predictive factor of outcome. Our results showed that the absence of speech was a risk factor of a negative outcome. The children with an intermediate speech level at Time 1 improved their speech by 14%. Being able to express oneself even with only a few words and a still-fragile syntax appears to correspond to a critical moment in development and seems to be an important protective factor. A higher degree of autism is also related to a negative outcome. These ndings, which point to symptom severity as a risk factor, support Lotters (1974) study showing that children with a high degree of autism will become severely handicapped. Thus, communication and person related cognition appear to be protecting factors against a negative outcome. These abilities can be regarded as prerequisites for the acquisition of speech, to the extent that using language requires the child to interrelate the physical and social environments. Moreover, Charman et al. (2004) contended that a childs initial communication level is a predictor of changes in daily living skills. The outcome proles obtained here were not linked to gender, socioeconomic status, medical condition, or surprisingly, intervention. While there is a current consensus on the positive impact of early intensive 673

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14(6) intervention (Howlin, 2000; Webster et al., 2003), our results showed that the number of hours of eclectic intervention was not linked to outcome prole (Eaves and Ho, 2004). As shown by the univariate analysis, the childrens initial clinical characteristics (speech level, adaptive behavior, person-related cognition, autism degree, presence of medical antecedents, and the childs treatment) inuence their developmental trajectories. Those children with the highest cognitive, adaptive, and speech levels, the least severe autism, and with no medical condition had the best outcomes. The two groups differed signicantly in terms of hours of intervention (regular school and special education). The children that had more intervention were the ones with a lesser degree of autism, a better language level, and a higher developmental age. Regarding the lack of a link between intervention and outcome, this nding is in line with the results obtained by Gabriels et al. (2001) and Eaves and Ho (2004), and suggests that the impact of the amount of intervention may not be as critical to outcome as the type of intervention. It is known that children with autism need routines and regularity in their environment. Thus, eclectic intervention approaches could be a source of excessive change and transition between activities and could thereby cause problematic behavior or anxiety. The present results may imply that the effectiveness of intervention is more dependent upon its nature than its intensity keeping in mind, however, that our interventions were eclectic in nature. The failure of intervention intensity and diversity to have an impact in our study raises the question of whether the specicities of an intervention method are a source of gain and improvement for the child (Eldevik et al., 2006; Howard et al., 2005).
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Limitations The ndings of this study should be interpreted in the light of a number of considerations. First, inclusion in our cohort, which took place between 1997 and 1999, was decided by centers during a period where conceptions of PDD were more restrictive than they are today (Allen, 1988). Children with the severest clinical pictures were proposed more often for inclusion, whereas very few children with an Asperger syndrome or a nonspecic PDD were included. Secondly, the intervention approaches were highly diverse, so it was not possible to compare them. For this reason, we focused rst on the impact of the amount of intervention, and then later analyzed the effect of the type of intervention. Thirdly, the fact that we did not use the conventional psychometric batteries to assess DQ constitutes a limitation on comparison with previous studies where IQ was the main variable used to assess outcome. The ratio674

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nale for our strategy was, rstly, that the heterogeneity of chronological age and developmental age in our sample limited the systematic use of the same instrument for the whole sample; secondly, that conventional psychometric batteries are based on typical development and their items are not appropriate for heterochronic development; and thirdly, that verbal-item relevance is limited for very young children with PDD because of their behavioral and speech disorders (Coplan and Jawad, 2005).

Conclusion
First, it seems here that when eclectic interventions are used, their intensity (in terms of number of hours) does not have a signicant effect on the outcome of children with autism, which raises questions about intervention content and approach. Second, the early clinical characteristics of children with autism appear to play an important role in their developmental trajectories, which argues against a universal model of intervention. It is essential to individualize intervention and to adapt it to each childs specic needs.

Acknowledgments This research beneted from the nancial support of the Hospital Program for Clinical Research (PHRC), France Telecom, and the AVA Foundation. We are very grateful to the families who took part in the study and to the staff of the participating teams. References
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