Richard Solomon MD, Principle Inves#gator

The PLAY Project Home Consulta6on Interven6on for Young Children with Au6sm Spectrum Disorders: A Randomized Controlled Trial Na#onal Ins#tutes of Mental Health (NIMH) Richard Solomon MD, Principle Inves#gator Ann Arbor Center for Developmental and Behavioral Pediatrics Laurie A. Van Egeren PhD Michigan State University, E. Lansing Gerald Mahoney PhD Case Western Reserve University, Cleveland Melissa S. Quon Huber PhD Michigan State University Perri Zimmerman, Ann Arbor Center for Developmental and Behavioral Pediatrics.

Research Collabora#on NIMH SBIR (clinicaltrials.gov) Easter Seals Na#onal office: Ellen Harrington- Kane OTR, VP Regional affiliate agencies Illinois: Peoria/Bloomington Michigan: Detroit/Oakland County Montana: Missoula Indiana: Evansville

Julian & Mom

Introduc#on The PLAY Project was born out of despera#on. Pennsylvania and University of Michigan Pilot pre/post study published in the journal Au6sm 2007 (see references) Phase 1 Small Business Innova#ons Research (SBIR) Feasibility Study (2005-07) Phase 2 SBIR RCT 2009-2012 Published: Journal of Developmental and Behavioral Pediatrics J Dev Beh Pediatr. 2014; 35(8): 475-485.

Overview Background and Review of Literature Need Parent- mediated, Developmental Rela#onship- based PLAY Project Model: From Prac#ce to Research to Prac#ce (With video examples) Research Design Hypotheses Methods Measures/Outcomes Research Results Discussion

BACKGROUND & REVIEW OF THE LITERATURE

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The Need The Na#onal Research Council (Lord et al., 2001) recommends intensive intervencon. 15-25 hours per WEEK of Engaging interven#on with a High adult to child ra#o (1:1 to start) Strategic and comprehensive Thus the many children with ASD need more intensive intervencon

The Need States are struggling to address the needs of young children with ASDs A majority of states (>30) require ABA/Behavioral methods (operant condi#oning) by law But ABA is hard to administer Not enough trained personnel Very high costs: $40-70K/child/year Difficult to implement with fidelity in community seings Recent reviews of state services reveal that too many young children with ASC are not receiving services* *Croen et al, Ruble et al, www.childhealthdata.org

The Need A major priority for health and educacon systems has emerged for: Evidence- based and effec#ve Less expensive and More easily disseminated forms of intensive interven#on for children with ASD Recent DSM V: Emphasis on Social Impairment Behavioral research on ASD largely focused on language and cogni#on, not social impairment. Parent- mediated, Developmental Rela#onship- based Interven#ons may have a role to play in ASD if shown to be effec#ve.

Parent- mediated Models Parent- mediated models olen focus on social reciprocity and improvements in development. Much less costly & more easily implemented because parents, once trained, do the interven#on PMM recognized as evidence based by the Na#onal Professional Development Center (Wong/Odom) Recent review* of PMM however showed good short term gains in interac#on but less effect on language, IQ, and au#sm. Only 3 studies lasted a year and none showed changes in ASD symptomatology. Conclusion: RCTs needed. Monitor parent stress *See Oono et al

Developmental, Rela#onship- based Interven#on Emerging research literature DRI (see refs). Do NOT use operant condi#oning such as Discrete Trial Training or other ABA type interven#ons. Focus is on following the child s intent in a co- regulated, dyadic way to promote func#onal (social interac#onal) development leading to improved language and cogni#on. DSM V focus on improving social impairment. More engagement, ini#a#on, reciprocal exchanges Values spontaneous interac#on that follows the child s lead and is playful and fun. Child loves being with people!

PLAY Project RCT The PLAY Project ConsultaCon Model Trains professionals to coach parents of young children with ASD to be child s best play partner Parent- mediated, developmental, rela#onship- based Opera#onalizes Greenspan and Wieder DIR theory Affect Diathesis Hypothesis Adult learning framework: Coaching, Modeling, Video and wriren feedback. Monthly 3- hour home visits and video feedback mid month. Consistent with NRC intensity Occurs in a community context One of the largest ASD RCT done in the U.S.

PLAY Project RCT Hypotheses 1. Compared to controls, PLAY Parents would improve in their interac#on skills 2. Parents would then facilitate improvement in children s interac#on, language, development, and ASD symptoms 3. PLAY parents would experience more stress b/c of demands to provide intensive interven#on 4. PLAY Consultants would show fidelity

METHODS

Methods Families recruited in two cohorts (2010-2012) from 5 ci#es in U.S. through MDs. 12 per site x 2 yrs = 120 See Figure 1. Power analysis completed. Easter Seals, a na#onal, private, non- profit, disability service agency. Ages 3-5 (actual 2-8 to 5-11) DSM IV clinical diagnosis confirmed by ADOS and SCQ Exclusion: AS, gene#c, severe medical, parent with mental/ cog impairment, English not primary

Figure 1: Timeline Pre- Eval I Post- Eval I Pre- Eval II Post- Eval II Start up 6 months 1st cohort 60 children 2nd cohort 60 children Wind down 6 months Grant start: September 2009 4/10 1 year 4/11 1 year Completed Sept. 2012

Methods Families recruited in two cohorts (2010-2012) from 5 ci#es in U.S. through MDs. 12 per site x 2 yrs = 120 See Figure 1. Power analysis completed. Easter Seals, a na#onal, private, non- profit, disability service agency recruited the children. Ages 3-5 (actual 2-8 to 5-11) DSM IV clinical diagnosis confirmed by ADOS and SCQ Exclusion: AS, gene#c, severe medical, parent with mental/ cog impairment, English not primary

Figure 2: Par#cipant Enrollment 148 Screened for Eligibility Analysis was Intent to Treat i.e. n = 128 20 Excluded 16 did not meet inclusion criteria 4 declined to participate 128 families randomized 64 Allocated to Intervention 61 Rec d PLAY + Community Services 2 Selected ABA 1 Caregiver went to jail 64 Allocated to Control 63 Rec d Community Services 1 Did not receive CS 1 Chose not to continue after allocation 57 Completed Study 4 Discontinued, 1 did ABA, 2 Dropped out, 1 had custody but other parent took child 55 Completed study 8 discontinued (1 rec d intensive intervention, 2 did PLAY, 2 moved, 3 lost

Methods II: Demographics MSU: IRB issues addressed and consent obtained Age avg. 50 months (32 to 71) About 20% children of color in CS, 30% in PLAY Mother primary care give in 90% Married about 90% (both groups) high propor#on Bachelor Degree 45 /53% Au#sm/ASD about 70/30% (i.e. more severe group) Income less than 60K: 54/56% All values non- significant i.e. randomiza#on successful. The groups were equivalent (apples to apples)

Community Services Control group received the usual community services Special educa#on preschool 12 hours per week of educa#onal services Supplement by SLP and OT Private OT Private SLP Not allowed to provide intensive interven#on greater than 10 hours per week. Allowed of course to drop out at any #me.

PLAY PROJECT INTERVENTION PROGRAM

Key Elements of PLAY Parent- mediated Home based 3 hour monthly home visits Adult learning: coaching, modeling, video and wriren feedback Intensive (parents expected to PLAY 2 hours/day) Developmental and rela#onship- based Based on the DIR (Greenspan and Wieder) theore#cal framework

Key Elements of PLAY Much less costly than professionally delivered models Trains master level child development professionals Complementary to exis#ng community services Used as a primary interven#on in Ohio Birth to Three program Cost $3500-4500/year/child Well established clinical program.

Well Established Clinical Program Over 100 licensed agencies in nearly 30 states and 9 countries A variety of seings: Strong Easter Seals Affilia#on Rehab Centers and private prac#ce Educa#on: Birth- 3, Special Ed Pre- Schools, Schools Hospitals and Health Centers Community Mental Health Centers 500 Professionals trained over the last 5 years 1000s of children per year provided PLAY Project Home Consultant Services State- wide EI training in Ohio

U.S. PLAY Project Consulta#on Programs

What We Do PLAY Project Organiza#on PLAY Project Early Interven#on Program Program Development PLAY Project Consulta#on Community Involvement Catalog of Training Opportuni#es (Workshops, webinars, etc.) Research

Vision/Mission Our Vision All parents will be supported in developing a joyous rela#onship with their children with au#sm spectrum disorders in a way that will help each child reach their full poten#al. Our Mission To train a global network of pediatric professionals to deliver an evidence- based, low- cost, intensive developmental interven#on to families of young children with au#sm spectrum disorders.

Profile of a PLAY Project Consultant Masters Degree or equivalent Experience working in child development Occupa#onal Therapists Speech/Language Pathologists Social Workers Early Interven#on Specialists Teachers & Special Educators Psychologists Community mental health professionals

Cer#fica#on Process: Prepara#on Cer#fied PLAY Project Consultant Curriculum: Func#onal Developmental Levels Consulta#on process 7 Circles of The PLAY Project Taking videos, analyzing videos Preparing reports for families Administra#ve marers

Cer#fica#on Process: Readiness Aler the 4- Day Cer#fica#on Training: Consultants are ready to begin providing early interven#on program Recommend caseload: 5-25 young children (up to age 6) with ASD

Cer#fica#on: Comple#on Supervision, Cer#fica#on, & Licensing: Complete 20 Supervision Tasks: 12-18 months Video and wriren reports ü submired online to PLAY supervisors ü Use HIPAA- compliant Sharefile ü Supervisors provide support and feedback Online Knowledge Exams CerCficaCon upon comple#on of supervision tasks Organiza#on/Agency licensed annually

PLAY Project: Skill Sequence 1. List Principles & Strategies Based on Comfort Zone (CZ) Sensory Profile (SP) and Functional Developmental Level (FDL) 2.Assess Child s unique: CZ Activities, SP & FDL 3. Define Daily & Weekly Curriculum/Activities 4. Follow Child s Cues, Lead & Intent to Increase Circles 5. Create Menu Of Specific Techniques 6. Video Tape/Critically Review Interactions and Progress 7. Refine Curriculum, Methods & Techniques

Intro to the PLAY Project 6 7 Change and Growth: Revising the Plan as Child Develops Visit Review: Video and Written Feedback 5 Engagement: PLAY time Between Parent and Child 1 Ready, Set PLAY! An Introduction to Principles and Methods 7 Circles of The PLAY Project 4 Family Guidance: Coaching, Modeling, and Feedback 2 Understanding Your Child: Creating a Unique Profile 3 The PLAY Plan: Individualized Techniques & Activities

7 Circles of the PLAY Project 6 7 Change and Growth: Revising the Plan as Child Develops Visit Review: Video and Written Feedback 5 Engagement: PLAY time Between Parent and Child 1 Ready, Set PLAY! An Introduction to Principles and Methods 7 Circles of The PLAY Project 4 Family Guidance: Coaching, Modeling, and Feedback 2 Understanding Your Child: Creating a Unique Profile 3 The PLAY Plan: Individualized Techniques & Activities

Circle 1: PLAY Project Methods Read the child's cues and intent Slow the pace of play, observing and wai#ng for the child s idea Follow the child's lead, responding to what the child wants Open and close circles of communica#on (back and forth interac#ons) Build on the child s interests

Julian get engaged (FDL 1-2) (Houston, we have contact!)

Circle 2: Crea#ng a Child s Unique Profile Functional Developmental Levels (FDLs) Sensory Motor Profile (SMP) Comfort Zone (CZ)

Circle 3: PLAY Plan Techniques The Purpose of the Techniques: Provide parents and professionals with ideas for engagement Increase alertness and awareness Improve ini#a#ve & flexibility Increase numbers & complexity of circles of communica#on Improve ability to solve problems

Sample Ac#vi#es by Func#onal Developmental Level Levels 1 & 2 Gently shaking arms or legs Gently squeezing arms, leg, head. Rolling child up in a rug Swinging in a blanket Levels 3 & 4 Chase: I m gonna get you Levels 5 & 6 Pretend: dress up, crashing cars, tea party, dolly sleeping, dinosaurs chasing a man, etc. Real hide and seek, not just peek a boo. Hide a doll and say Where is the dolly? Get the bubbles, balloon, etc. Duck, duck, goose Ball play (rolling it back and forth)

Julian Moves Up (FDL 3-4)

6 Func#onal Developmental Levels Self regula#on and shared aren#on (FDL 1) Engagement (FDL 2) Two- way Communica#on (FDL 3) Complex two- way Communica#on (FDL 4) Shared Meanings & Symbolic Play (FDL 5) Emo#onal Thinking (FDL 6)

Circle 4: Family Guidance Gold standard is home visi#ng Can be adapted to clinic seing PLAY Consultant Models PLAY Techniques Observes and Coaches PLAY Partners (Parents) Provides wriren Feedback on PLAY Session

Seven 7 Circles of of the the PLAY PLAY Project Project 6 7 Change and Growth: Revising the Plan as Child Develops Visit Review: Video and Written Feedback 5 Engagement: PLAY time Between Parent and Child 1 Ready, Set PLAY! An Introduction to Principles and Methods 7 Circles of The PLAY Project 4 Family Guidance: Coaching, Modeling, and Feedback 2 Understanding Your Child: Creating a Unique Profile 3 The PLAY Plan: Individualized Techniques & Activities

Circle 5: PLAY Time Engagement Between Parent and Child Playful & fun: When you do what the child loves, the child will love to be with you. 2 hours per day, broken up into 10-20 minute PLAY sessions Daily rou#nes such as meal #me, bath #me, and bed #me

Circle 6: Visit Review Video & Wriren Feedback Use 7 Circles of PLAY Project as guide Video shows caregivers and Consultant playing with child Parent/Video Report Form: Gives parents feedback about interac#on Gives parents feedback about child progress

Julian Becomes Symbolic (FDL 5!)

Circle 7: PLAY Clinical Goals Joyful rela#ng Simple and complex nonverbal gestures Long interac#ve sequences (e.g. 50+) Circles of spontaneous verbal communica#on Shared social aren#on (FDL 1-3) Symbolic language related to feelings (FDL 4-6) With us con#nuously, not fragmented/stuck in CZ. Socially func#onal & interested in others.

RESEARCH DESIGN

Research Timeline Pre- Post Study 2000-2005 Pub: Au6sm (2007) SBIR Phase 1 Feasibility 2005-6 SBIR Phase 2 RCT 2009-2013 Pub: JDBP Oct, 2014

8 Trained HCs At 5 ES sites Provide 12 HVs Over 12 months CS gets Pre-school Including SLP and OT /FEAS /CES-D Aim 1 Parent Interaction Aim 2 Child Outcomes Aim 3 Parent Implementation Aim 4 HC Fidelity

AIMS Aim 1: Improve Parent InteracCon with their child with ASD Sensi#ve, responsive, effec#ve Non- direc#ve Aim 2: Improve Child Development More con#ngent, reciprocal, social interac#ons Berer cogni#ve func#oning Improved recep#ve and expressive language Berer adap#ve behavior Reduce overall au#sm severity

AIMS Aim 3: PLAY is feasible for Parents to Implement Cogni#ve screen Stress Sa#sfac#on Implementa#on Aim 4: Home Consultants have fidelity to the model. Training model Fidelity Manual

MEASURES

MEASURES Pre and post measures in two 1 year cohorts. ADOS evaluators trained to research reliability All raters blinded to group and #me Measures de- iden#fied before being sent to MSU for analysis All measures pre- specified by grant aims according to original grant protocol.

MEASURES BY AIM Aim 1: Improve Parent InteracCon Maternal Behavioral Ra#ng Scale (Mahoney) Aim 2: Improve Child Development MacArthur CD Inventories Mullen Scales of Early Development Pivotal Behavior Ra#ng Scale (Mahoney) Fun#onal Emo#onal Ra#ng Scale (DiGangi, Greenspan) Vineland II (not filled out correctly and abandoned) Social Communica#on Ques#onnaire (SCQ) Au#sm Diagnos#c Observa#on Scale- G (Lord) Module 1 (little or not speech), Module 2 (some speech)

MEASURES BY AIM Aim 3: PLAY is feasible for parents to implement Peabody Picture Vocabulary Test- 4 (Adult IQ Screen) Paren#ng Stress Index CES- D (Depression) Parent Percep#on of Training and Interven#on (Sa#sfac#on) Aim 4: Home Consultants have fidelity to the model Home Consultant Ra#ng Scale (Fidelity)

RESULTS

MODEL EFFECTS Home Consultants Are True to the Model (AIM 4) Parent Effec#veness Improves (AIM 1) Parents Implement PLAY Project (AIM 3) Child Development Improves (AIM 2)

FIDELITY RESULTS A random sample of 20% of HC videos and write ups from each site was reviewed by supervisors Supervisors showed high inter- rater reliability in using the fidelity manual ra#ng system All 6 Home Consultants consistently completed all necessary elements and Scored 3 or above on quality of key components One HC scored an overall score of 2 on one write up Rated a 2 by two supervisors independently Conclusion: Home Consultants showed fidelity to the PLAY Project training model

AIMS Home Consultants Are True to the Model (AIM 4) Parent Effec#veness Improves (AIM 1) Parents Implement PLAY Project (AIM 3) Child Development Improves (AIM 2)

AIM 1: PARENT EFFECTIVENESS Dose: Parents par#cipated in 11 visits per year on average Reported using the PLAY interven#on for 621.9 hours over the year or 1.7 hours per day. Both groups reported about 100 hours per year in other therapies (SLP, OT, ABA, etc.) None more than 10 hours per week. Maternal Behavior Ra#ng Scale Results Sta#s#cal Analysis: MANCOVA (Mul#variate Analysis)

MBRS: Responsiveness Total Sensi#vity, Responsiveness, Effec#veness all sig. N = 112, p <.001, partial η 2 =.25

MBRS: Direc#ve Total Direc#veness sig., Pace not sig; both increase N = 112, p <.05, partial η 2 =.05

MODEL EFFECTS Home Consultants Are True to the Model (AIM 4) Parent Effec#veness Improves (AIM 1) Parents Implement PLAY Project (AIM 3) Child Development Improves (AIM 2)

η 2 =.04 AIM 3: RESULTS PARENT STRESS PSI Child Score 150 140 130 120 110 100 90 Control PLAY 80 Pre Post Time x group interac#on: p =.15 (ns), par#al η 2 =.043.

AIM 3: RESULTS 16 PARENT DEPRESSION CESD scores 14 12 10 8 6 4 2 Control PLAY 0 Pre Post Time x group interac#on: p =.24 (ns), par#al η 2 =.028.

MODEL EFFECTS Home Consultants Are True to the Model (AIM 4) Parent Effec#veness Improves (AIM 1) Parents Implement PLAY Project (AIM 3) Child Development Improves (AIM 2)

CBRS: Aren#on Total Aren#on to Ac#vity (sig.), Persistence (not sig., same parern), Involvement (sig.), Compliance (sig.) N = 128, p <.001, partial η 2 =.10

CBRS: Ini#a#on Total Ac#vity, Adult, Affect all sig. N = 128, p <.001, partial η 2 =.10

Developmental Measures FEAS Total Score (PLAY vs CS): Func#onal development showed significant improvement (p =.05) SCQ: Parent report of ASD sx. Both groups showed significant improvement. No differences between PLAY and CS. MacArthur Child Development Inventories. Both groups showed significant improvement. No differences between PLAY and CS. Mullen: 20% of children could not get a score. No differences between PLAY and CS.

Among all children, regardless of ini#al diagnosis, the treatment group was significantly more likely to show improvement compared to the control group (N=128, p = 0.001, Odds Ra#o: 2.39)

ADOS 33% of CS children and 54% of PLAY improved by one category For more severely involved (Dx: Au#sm on ADOS) 29% of CS Children and 55% of PLAY children changed a category For higher func#oning children 41% in CS and 53% in PLAY changed a category Odds ra#on of 2.39 indicates that PLAY Children more than twice as likely to change a category

Julian Develops Rich Pretend Play

DISCUSSION

Summary This RCT evaluated the PLAY Project Home Consulta#on (PLAY) program: parent- mediated, intensive, developmental, rela#onship based approach for young children ages 2.5 to 6 years old with ASD. Aler 12 months of interven#on the following aims using a rigorous Intent to Treat (ITT analysis) were demonstrated:

Summary 1. Consultants show fidelity to the model 2. PLAY parents interact with more skill Improved ability to sensi#vely respond and effec#vely engage their child. 3. PLAY parent do NOT suffer more stress and significantly reduced depression 4. PLAY children improve in their development: Improved interac#on with increased aren#on and ini#a#on Improved func#onal development Less au#sm symptomatology with PLAY children more than twice as likely to improve in their diagnos#c category.

ADOS Results First study using a parent mediated model to show improvement in au#sm symptomatology. These results must be interpreted with cau#on. Interpreted clinically 41% in the CS group and 53% in PLAY who were more mildly affected (ASD) improved enough to be categorized as non- ASD. 16% of CS and 31% of PLAY moved from more severe to mild (Au#sm to ASD) Not consistent with clinical experience.

ADOS Results ADOS is only one component of a dx eval...should never be used in isola#on... ADOS- G has been revised to improve dx accuracy. Categorical cut points might have led raters to interpret the test items such that slight changes in score cuased categories to be crossed leading to the incongruous results.

Depression Results This is one of the first rigorous, long term studies to show a reduc#on in depression following intensive interven#on. Up to 40% of mothers with a child with ASD report depression. PLAY offers hope, child becomes more responsive, parents mo#vated to put in the #me?

Parent Satisfaction Parent 1: "This intervention helped our child reach potentials beyond our expectations and we are grateful for the opportunity to be a part of this project." Parent 2: "We are so grateful to have had this opportunity. It has been so rewarding to watch our child's growth with a few critical changes to how we interact with him."

Study Limita#ons Language as measured by the MCDI and DQ as measure by the Mullen Scales did not differ by group. Most of the children in the study were lower func#oning. Many could not complete the Mullen. Future studies may need to enroll children by cogni#ve and/or language levels to find differences.

Study Limita#ons Interven#on group received similar community services making it difficult to assess PLAY as an isolated interven#on. Combining parent- mediated interven#on with other locally available services however may improve the comprehensiveness of services. PLAY added only 1 home visit and 1 mid- month feedback component to CS which makes it easy to implement in most community seings

Study Limita#ons Finally, in terms of study limita#ons parents were more likely to be married, berer educated, and of somewhat higher SES than average. May limit the generaliza#on of findings. On the other hand this was a real world test of the model in 5 diverse sites in the U.S.

Research: Implica#ons PLAY uses an efficient training model with high fidelity. Parents consistently and effec#vely applied the program. PLAY offers a low cost op#on for parents and society Thus, the PLAY Project Home Consulta#on model can be broadly and quickly disseminated To serve a growing, unmet na#onal need.

The PLAY Project www.playproject.org