J. Indian Assoc. Child Adolesc. Ment. Health 2016; 12(3): Award paper Niloufer Award 2015

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1 230 J. Indian Assoc. Child Adolesc. Ment. Health 2016; 12(3): Award paper Niloufer Award 2015 The effectiveness of an intensive, parent mediated, multi-component, early intervention for children with autism Raman Krishnan, Merlin Thanka Jemi Alwin Nesh, Paul Swamidhas Sudhakar Russell, Sushila Russell, Priya Mammen Address for correspondence: Dr Raman Krishnan, Associate Professor, Saveetha Medical College, Chennai. dr_ramkrish@yahoo.com Abstract Aim: Most children with autism do not receive early intervention, unless parents are trained, in countries with low child mental health resources. To maximize the existing resource, we evaluated the feasibility and effectiveness of an intensive, parent mediated, multi-component, early intervention for autism in India. Methods: Data of 77 children with an ICD-10 diagnosis of autism who completed a 12- week, five days a week, intervention program and regular practice at home was collected from the database of a teaching hospital. Intervention components included the standard intervention protocol, the Psycho-Educational Profile intervention (PEP-R) and the Carolina Curriculum for Infants and Toddlers with Special Needs. Pre and postintervention PEP-R rating of parents were used to evaluate the intervention outcome. Appropriate bivariate, multivariate and resampling techniques were used to evaluate the intervention effectiveness. Results: The effect size (ES) for the intervention was moderate to large for the PEP-R developmental age as well as perception, fine motor, gross motor, eye-hand coordination,

2 231 cognitive performance and cognitive verbal domain among children with mild to moderate (ES ranged from 0.70 to 0.88) and severe autism (ES ranged from 0.73 to 0.87). There was no difference in the intervention effect between the groups (ES ranged from to 0.30). The effectiveness of the intervention continued after controlling the confounding effect of baseline developmental quotient, developmental age and adaptive skills. Conclusion: It is feasible to provide an effective parent mediated early intervention for mild to severe autism in countries where parents are the cornerstone in childhood disability care. Key Words: Autism, effectiveness, early intervention, parent-mediated, India. Introduction Prevalence of autism is greater than previously recognised at per [1]. As impairment emerge during the first three years of life [2], early diagnosis followed by early intervention can improve outcomes for individuals with autism, possibly more than in any other developmental disability [3,4]. While there is no cure for autism, an array of interventions like Applied Behavioural Analysis, Communication-focussed Intervention, Developmental Interventions, Integrative Programs, Sensory-motor Interventions have been proposed to improve the symptoms associated with autism. Although reviews of many of the above interventions show either no or modest intervention effects [5], found them complex and resource intensive [6], those treatments that demonstrated intervention gain have not only

3 232 ameliorated autistic symptoms but improved adaptive functioning and parent-child dyadic relationship [7-10]. Interestingly, among the many intervention models available, parent mediated early interventions are known to have the most significant intervention effects which have not been tested in low and middle income countries with paucity of child mental health-care resources [11]. Therefore, the current study was designed to evaluate: (1) if it is feasible to provide a therapist guided, parent mediated, multi-component, early intervention for children with autism in a country with low child mental health resources; (2) if this multi-component intervention program, which is an integration of a standard autism treatment protocol combined with the Psycho-Educational Profile intervention (PEP-R) and The Carolina Curriculum for Infants and Toddlers with Special Needs (CCITSN), is effective for children with autism; (3) if the intervention is more effective for either the mild to moderate or severe autism group. Methods Setting and sample This study was conducted at the Autism Clinic, Child and Adolescent Psychiatry Unit of a tertiary care, teaching hospital in South India. The charts of children and adolescents enrolled for autism training either for a residential or day care program with complete data set were identified from the unit s database for a three-year period. The enrolled toddlers and children had an ICD-10 based clinical diagnosis of Autism Spectrum Disorder (Pervasive Developmental Disorder). Children with a diagnosis of Overactive disorder associated with mental retardation and stereotyped movements (F84.4) were

4 233 excluded because of its uncertain nosological status [12]. All clinical diagnoses were made by consultant psychiatrists and endorsed by the multidisciplinary team with a mean (SD) clinical experience of 12.74(8.21) years. Measures The Psycho-Educational Profile-Revised (PEP-R) [13] was developed to assess children with autism and formulate their Individualized Education Programs (IEP). The Developmental Scale of PEP-R assesses the overall functioning of the child, the developmental age, based on the imitation, perception, fine motor, gross motor, eye-hand coordination, cognitive performance and cognitive verbal subscales. The PEP-R also includes a Behavioral Scale, which is used to identify the degree of behavioral abnormality The Developmental Scale of PEP-R has been validated for children with autism in India [14]. The pre-intervention and post-intervention PEP-R scores rated, in terms of improvement in months, was used as the intervention outcome measure in this study. The Childhood Autism Rating Scale (CARS) [15], is a 15-item behavior-rating scale, designed to detect and quantify symptoms of autism. Children with scores of 30.5 to 37 are rated as mildly moderately autistic, and 37.5 to 60 as severely autistic by CARS. This measure has been validated for India [16]. Gesell s Developmental Schedule (GDS) [17] gives the developmental skills in four areas in months: motor behavior, adaptive behavior, language and personal as well as social behavior.

5 234 Vineland Social Maturity Scale (VSMS) [18] measures the adaptive skills of the child in eight areas in weeks: self help general, self help dressing, self help eating, socialization, self direction, communication, locomotion and occupation. As the baseline GDS developmental quotient and VSMS adaptive skills can confound the intervention outcome independent of the severity of autism both had to be controlled in this study [19, 20]. The study was reviewed and approved by the local institutional review board. Intervention Within the first week of starting the intervention and at end of 12 th week the parent assisted by the therapists assessed the child using the PEP-R and CCITSN assessment schedules. This clinic based multi-component early intervention package had three components. (1) the standard clinical intervention being used for the past 11 years in the clinic that included training in self-care skills, social skills and control of problem behaviour using special education and behavioural techniques. Throughout the 12-week program, the parents, using interactive group psycho education technique, were taught about various aspects of autism and developmental disabilities. This psycho education module is proven to improve the knowledge and attitude of the parents towards developmental disability [21].; (2) The PEP-R intervention [22] included teaching activities from Individualized Assessment and Treatments for Autistic and Developmentally Disabled Children,

6 235 Teaching Strategies for Parents and Professionals and Teaching Activities for Autistic Children and (3) the CCITSN module [23] addressed the intellectual disability component with focus mainly on cognition, communication, social adaptation, fine motor, and gross motor of the children using a developmental approach. Each session of the therapist guided, parent mediated intervention process would start with a 10-minute briefing about the goals for the day which are set on a weekly basis. The child was engaged in play routines and social stories and each parent child dyad received applied behaviour analysis aimed at improving the behavioural control and interactive skills of parents using principles of rewarding and guided practice. The whole intervention was in the form of closed group sessions conducted five times a week by two therapists and each session lasted for 4 hours. The parents were encouraged to continue the intervention, at home using adaptations to suit the home environment. Participation from other significant family members was encouraged. Data analysis Non-parametric analyses were used because of the relatively small and skewed sample. The baseline differences between the groups were analyzed using Fisher s Exact Test and Mann-Whitney U test for the categorical and continuous variables respectively. To compare the change in PEP-R scores within the mild to moderate and severe autism group, before and after intervention, Wilcoxon matched pair rank test was used. Based on the median of the post and pre intervention PEP-R scores, within groups and between groups, the effect size (ES) for both groups was calculated using the formula r=z/ n. For

7 236 the comparison of difference (post-pre intervention) between the groups, Mann-Whitney U test was used. Also, 95% confidence intervals were calculated by resampling with replacement, using boot strapping (1000 times), for all the effect sizes. In addition, for the outcomes, multiple regression analysis was used to take into account the possible confounding effect of the developmental age, adaptive skills and baseline PEP-R developmental age imbalance autism groups. P<0.05 (2-tailed test) was considered significant. Statistical analyses were performed using the SPSS (version 19) and R (Version ). Results Participant and baseline characteristics The mean (SD) chronological age of the children was 3.66(1.64) years. There were more boys (81.8%) than girls (18.2%) in the sample. The chronological age (mild vs. severe = 3.74 (1.86) vs (1.59), z=-0.20; P=0.8) in the mild to moderate and severe autism group were comparable at the baseline. Similarly, other socio-demographic and clinical variables were not different between the groups (Table 1).

8 As expected, the mean (SD) CARS score [mild vs. severe = (1.58) vs (2.88), z= -5.95; P=0.001] was statistically different between the groups. Also the GDS developmental quotient [mild vs. severe = (19.27) vs (14.11); z=-2.76; P=0.006] and VSMS adaptive functioning [mild vs. severe = 2.71 (1.03) vs (0.73); z=-2.48; P=0.01] were statistically significantly different between the groups. The mean (SD) outcome measure, total PEP-R score (developmental age), at the baseline was statistically significantly different between the groups [mild vs. severe = 25.43(11.13) vs (7.18), z=-2.72; P=0.007]. Similarly, the domains of imitation, 237

9 238 perception, fine-motor, gross motor, eye hand integration, cognitive performance and cognitive verbal were statistically significantly different between the mild and severe autism groups. Feasibility of parent mediating intervention The attrition of parents from this intensive multi-component early intervention was only 2% over three months of training and there was no difference between the groups. Clinical observations demonstrated that parents were able to implement different aspect of the intervention effectively. The ability to continue the training at home, for about 3 to 4 hours a day (high level) was observed in 74% of the parents. There was no difference between the groups in participation by other family members in the training. Effectiveness of the intervention within group The intervention was effective in children with mild to moderate and severe autism in the bivariate analysis (Table 2). The mean (SD) gain in the total PEP-R score (developmental age) was 9.88 (3.47) months and 7.50 (1.97) months among those with mild to moderate and severe autism respectively. The effect size (ES) for the intervention, as indicated by the PEP-R developmental age, was 0.87 for both groups indicating a large effect size. The effect size for the various PEP-R subscale scores also varied between 0.70 to 0.88 for the mild to moderate autism group and 0.73 to 0.87 for the severe autism group, indicating moderate to large effect size for the intervention. The confidence intervals for the effect sizes, indicated that the intervention effects would fall within the mentioned range 95% of the time and remain significant even if the study were to be repeated as many as thousand times (Table 2).

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11 240 Effectiveness of the intervention between groups In the bivariate analysis, when the post-intervention differences in the total and subscale PEP-R scores was compared, there was no statistically significant difference except in the cognitive verbal subscale (Table 3). The mean (SD) gain in the developmental age was 2.37 (1.16) months between autism groups. The effect size for the intervention was 0.21 between the mild and severe autism group. The intervention effect for the PEP-R subscales of cognitive verbal and cognitive perceptual was 0.20 and 0.30 respectively suggesting a small intervention effect. The effect size for all the other PEP-R subscales varied between and 0.18 indicating no difference in the intervention effect between the groups. The confidence intervals for the effect sizes, indicated that the intervention effects would fall within the mentioned range 95% of the time and remain insignificant even if the study were to be repeated as many as thousand times (Table 3).

12 In the multivariate analyses, despite controlling for the possible confounding effect of the developmental age, adaptive functioning and the baseline PEP-R differences, neither the post-intervention total PEP-R-score nor any other subscale demonstrated a statistically significant difference between the groups (Table 4). 241

13 Discussion In summary, the findings of this study are that: (1) it is feasible to provide a parent mediated multi-component, early interventions for children with autism in a low child mental health resource country; (2) this intervention is effective for enhancing the PEP-R developmental age and its domains for children with autism (both mild to moderate and severe group). It is proven that it is critical to include parents in the early intervention for 242

14 243 autism and without parental participation the intervention gains are unlikely to be maintained [24]. The observation based on this real world study can pave way to primary-care intervention models involving parents. The overall intervention gain in this study was an increase in the developmental age of about 9 and 7.6 months during the 3 month training among children with mild to moderate and severe autism respectively. The within group intervention effect size (ES) of 0.87 in our study was comparable with the effect size of 0.88, 0.82 and 0.80 reported in the literature [25-27] but lesser than that documented by others [28-30]. Our intervention effect size was far better than the reported by Salt et al [31]. The intervention was equally effective in the mild to moderate and severe autism group except for the cognitive verbal domain. This difference in the cognitive verbal domain did not persist when the confounders were controlled. Similar intervention effectiveness across varying severity of autism has also been documented in the literature although effect sizes are not available [32]. We speculate that our intervention was greatly effective, firstly, because of the integration of the multiple components as it also addressed the adaptive behaviour and behavioural problems. Studies documenting the efficaciousness of various multicomponent interventions have consistently revealed that they improve the developmental age significantly (ES = 0.92 to 1.35), develops play behaviours (ES = 0.84 to 1.18) [33], decreases symptoms of autism (ES = 2.27) [34], improves other abilities like perceptual fine motor, cognition, language, social/emotional (ES = 0.66 to 1.04) and

15 244 receptive/expressive language skills (p =0.001, ES = 1.64) had significantly improved with multi-component intervention [34], which is much similar to our finding. Secondly, the parent involvement possibly has been an enhancing factor in the outcome measure in this study which is reported in literature as well [35-38]. When parents were used as co-therapists in the training of their children, pre-academic skills (ES=0.59), problem behaviours (ES= 1.07 to 1.38) (36), face gaze (ES=1.04) and socialisation (ES=1.27), autism symptom clusters (ES = 0.86) [39] and language skills (ES=0.86) had significantly improved. It is also known that parents can be trained to effectively implement early interventions for children as young as 3 years of age [40]. Thirdly, the early intervention could have played a role in the intervention success. The mean age of the children in our study was 3.66(1.64) years, possibly the earliest age that can be enrolled for clinical intervention in a country with poor awareness about autism [41]. The role of early intervention in improving outcome has been documented [42]. Fourthly, the intervention program was intensive in its approach and perhaps enabled children to learn skills. Our intervention was for 4 hours a day for 5 days a week for 12 weeks. Intensive approaches have been known to bring about significant therapeutic effects in areas of intelligence, self-help, pre-academic skills, communication (ES=0.57 to 2.14) [43,33] and autistic symptoms (ES = 2.58) in a previous study [45]. Finally, closely related to the intensity is the treatment duration. Although the clinic based treatment was only for 20 hours a week, the parents seemingly followed training at home, which couldn t be quantified. A rough estimate is that the parents would have achieved about 20 to 40 hours of total training a week. Although the recent studies show

16 245 that longer treatments, of more than 40 hours, yielded positive outcome (ES = 0.86 to 2.11) [46] considering home setting, researchers suggest at least 30 hours of training [30]. Limitations would include its retrospective design. The generalisability is compromised as the study children have greater familial resources and less heterogeneity than those served in community settings [47]. But these children in clinical setting present more challenges [48] and hence this study which has participants closer to real world improves the external validity of our finding. In conclusion, we document that it is feasible to provide an intensive, therapist guided, parent mediated, multi-component, early intervention program in an Indian setting. In future, the best age for intervention, optimal intensity, duration of treatment and parental involvement as predictive factors need to be quantified, and thus prospective pragmatic trials with factorial design are warranted. References 1. Baird G, Simonoff E, Pickles A, et al: Prevalence of disorders of the autism spectrum in a population cohort of children in South Thames: the Special Needs and Autism Project (SNAP). Lancet 2006, 368(9531): Bethea TC, Sikich L: Early pharmacological treatment of autism: a rationale for developmental treatment. Biol Psychiatry 2007,61(4): Olley JG: Curriculum and classroom structure. In: Volkmar FR, Paul R, Klin A, Cohen D, eds. Handbook of autism and pervasive developmental disorders. 3 rd edn. New Jersey: J. Wiley & Sons; 2005:

17 Lipkin PH, Schertz M: Early intervention and its efficacy. In: Accardo PJ, Paul H. Brookes, eds. Capute and Accardo's Neurodevelopmental Disabilities in Infancy and Childhood: Neurodevelopmental diagnosis and treatment. Vol I. 3 rd edn.. Baltimore MD: 2008; Ospina MB, Krebs Seida J, et al: Behavioural and developmental interventions for autism spectrum disorder: a clinical systematic review. PLoS One 2008, 3(11): e Motiwala SS, Gupta S, Lilly MB, Ungar WJ, Coyte PC: The cost-effectiveness of expanding intensive behavioural intervention to all autistic children in Ontario. Healthcare Policy 2006, 1(2): Aldred C, Green J, Adams C: A new social communication intervention for children with autism: pilot randomised controlled treatment study suggesting effectiveness. J Child Psychol Psychiatry 2004 Nov; 45(8): Tonge B, Brereton A, Kiomall M, Mackinnon A, King N, Rinehart N: Effects on parental mental health of an education and skills training program for parents of young children with autism: a randomized controlled trial. J Am Acad Child Adolesc Psychiatry 2006 May; 45(5): Ben Itzchak E, Lahat E, Burgin R, Zachor AD: Cognitive, behavior and intervention outcome in young children with autism. Res Dev Disabil Sep-Oct; 29(5): Dawson G, Rogers S, Munson J, et al: Randomized controlled trial of an intervention for toddlers with autism: the Early Start Denver Model. Pediatrics 2010 Jan; 125(1): e17-23.

18 McConachie H, Diggle T: Parent implemented early intervention for young children with autism spectrum disorder: a systematic review. J Eval Clin Pract Feb; 13(1): Sponheim E: Changing criteria of autistic disorders: a comparison of the ICD-10 research criteria and DSM-IV with DSM-III-R, CARS, and ABC. J Autism Dev Disord Oct; 26(5): Schopler E, Reichler RJ, Bashford A, Lansing MD, Marcus LM: Individual assessment and treatment for autistic and developmental disabled children. Psychoeducational Profile Revised (PEP-R), Vol I. Pro-Ed. Austin TX: Alwin Nesh MTJ, Joseph RBJ, Daniel A, Abel JS, Shankar SR, Mammen P, Russell S, Russell PS: Psychometrics and utility of Psycho Educational Profile-Revised (PEP- R) as a Developmental quotient measure among children with the dual disability of intellectual disability and autism. Journal of Intellectual Disabilities (in press). 15. Schopler E, Reichler RJ, Renner BR: Childhood Autism Rating Scale. Los Angeles: Western Psychological Services; Russell PS, Daniel A, Russell S, Mammen P, Abel JS, Raj LE, et al: Diagnostic accuracy, reliability and validity of Childhood Autism Rating Scale in India. World J Pediatr May; 6(2): Gesell A: The first five years of life: The preschool years. New York: Harper and Brothers publishers; Doll EA: A Genetic Scale of Social Maturity. American Journal of Orthopsychiatry, 1935; 5:

19 Ben Itzchak E, Zachor DA: The effects of intellectual functioning and autism severity on outcome of early behavioral intervention for children with autism. Res Dev Disabil May-Jun; 28(3): Gordon K, Pasco G, McElduff F, Wade A, Howlin P, Charman TA: Communicationbased intervention for nonverbal children with autism: what changes? Who benefits? J Consult Clin Psychol Aug; 79(4): Russell PS, John JK, Lakshmanan JL: Family intervention for intellectually disabled children. Randomised controlled trial. Br J Psychiatry 1999 Mar; 174: Schopler E, Reichler RJ, Lansing MD: Teaching Strategies for Parents and Professionals: Individualized Assessment and Treatment for Autistic and Developmentally Disabled Children. Vol II. Pro-ed. Austin, TX: Johnson-Martin NM, Jens GK, Attermeier SM, Hacker BJ: The Carolina Curriculum for Infant and Toddlers with special Needs, 2 nd edn. Baltimore, Maryland: Paul H. Brookes Publishing Co; Whalen C, Schreibman L, Ingersoll B: The collateral effects of joint attention training on social initiations, positive affect, imitation, and spontaneous speech for young children with autism. J Autism Dev Disord Jul; 36(5): McEachin JJ, Smith T, Lovaas OI: Long-term outcome for children with autism who received early intensive behavioral treatment. Am J Ment Retard Jan; 97(4): Shallows, Graupner TD:Replicating Lovaas' treatment and findings: Preliminary results. PEACH Putting Research into Practice Conference, London: 1999.

20 Lovaas OI: Behavioral treatment and normal educational and intellectual functioning in young autistic children. Journal of Consulting and Clinical Psychology, 1987; 55: Eikeseth S, Jahr E, Eldevik E: Intensive school-based behavioral treatment for four to seven year old children with autism: A one-year follow-up. PEACH Putting Research into Practice Conference, London: Smith T: Outcome of early intervention for children with autism. Clinical Psychology: Science and Practice 1999; 6: Smith T, Groen AD, Wynn JW: Randomized trial of intensive early intervention for children with pervasive developmental disorder. Am J Ment Retard. 2000; 105: Salt J, Shemilt J, Sellars V, Boyd S, Coulson T, McCool S: The Scottish Centre for autism preschool treatment programme. II: The results of a controlled treatment outcome study. Autism 2002 Mar; 6(1): Rogers SJ, Vismara LA: Evidence-based comprehensive treatments for early autism. J Clin Child Adolesc Psychol Jan; 37(1): Rogers SJ, Lewis H: An effective day treatment model for young children with pervasive developmental disorders. J Am Acad Child Adolesc Psychiatry 1989 Mar; 28(2):

21 Rogers SJ, DiLalla D: A comprehensive study of a developmentally based preschool curriculum on young children with autism and young children with other disorders of behavior and development. Topics in Early Childhood Special Education 1991; 11: Dawson G, Osterling J: Early intervention in autism. In: Guralnick MJ, ed. The Effectiveness of Early Intervention. Baltimore MD: Paul H. Brookes Publishing Co., 1997; Robbins FR, Dunlap G: Effects of task difficulty on parent teaching skills and behavior problems of young children with autism. Am J Ment Retard May; 96(6): Ozonoff S, Cathcart K: Effectiveness of a home program intervention for young children with autism. J Autism Dev Disord Feb; 28(1): Jocelyn LJ, Casiro OG, Beattie D, Bow J, Kneisz J: Treatment of children with autism: a randomized controlled trial to evaluate a caregiver-based intervention program in community day-care centers. J Dev Behav Pediatr Oct; 19(5): Zappela M: Young autistic children treated with ethologically oriented family therapy. Family Systems Medicine 1990; 8 (1): Moes DR, Frea WD: Contextualized behavioral support in early intervention for children with autism and their families. J Autism Dev Disord Dec; 32(6):

22 Daley TC, Sigman MD: Diagnostic conceptualization of autism among Indian psychiatrists, psychologists, and pediatricians. J Autism Dev Disord Feb; 32(1): National Research Council. Educating children with autism. Committee on Educational Interventions for Children with Autism. Division of Behavioral and Social Sciences and Education. Washington, DC: National Academy Press, Anderson SR, Avery DL, DiPietro EK, et al: Intensive home-based early intervention with autistic children. Education & Treatment of Children. Special Issue: New developments in the treatment of persons exhibiting autism and severe behavior disorders 1987; 10(4): Sheinkopf SJ, Siegel B: Home-based behavioral treatment of young children with autism. J Autism Dev Disord 1998 Feb; 28(1): Weiss MJ: Differential rates of skill acquisition and outcomes of early intensive behavioral intervention for autism. Behavioral Interventions 1999; 14(1): Schleien SJ, Mustonen T, Rynders JE: Participation of children with autism and nondisabled peers in a cooperatively structured community art program. J Autism Dev Disord Aug; 25(4): Lord C, Wagner A, Rogers S, Szatmari P, Aman M, Charman T, et al: Challenges in evaluating psychosocial interventions for Autistic Spectrum Disorders. J Autism Dev Disord Dec; 35(6):

23 Cochran-Smith M, Lytle S: Relationships of knowledge and practice: Teacher learning in communities. In: Iran-Nejar A, Pearson PD, eds. Review of research in education. Washington, DC: American Educational Research Associates, Raman Krishnan, Associate Professor, Saveetha Medical College, Chennai, Merlin Thanka Jemi Alwin Nesh Psychologist, Christian Medical College, Vellore,Paul Swamidhas Sudhakar Russell- Professor and Head, Sushila Russell,Assistant Professor, Clinical Psychology, Priya Mammen, Professor, Child and Adolescent Psychiatry, Christian Medical College, Vellore.