545685 research-article2014

AUT0010.1177/1362361314545685AutismHarrop

Review Article

Evidence-based, parent-mediated interventions for young children with autism spectrum disorder: The case of restricted and repetitive behaviors

Autism 1­–11 © The Author(s) 2014 Reprints and permissions: sagepub.co.uk/journalsPermissions.nav DOI: 10.1177/1362361314545685 aut.sagepub.com

Clare Harrop

Abstract Restricted and repetitive behaviors represent a core symptom of autism spectrum disorders. While there has been an increase in research into this domain in recent years, compared to social-communication impairments experienced by children with autism spectrum disorders, much less is known about their development, etiology, and management. Parent-mediated interventions have become increasingly popular in the field, with a surge of studies reporting significant findings in social communication and cognitive development in early childhood. Restricted and repetitive behaviors are often not specifically targeted or measured as an outcome within these interventions. This article reviews how 29 parent-mediated interventions approached the management, treatment, and measurement of restricted and repetitive behaviors. Recommendations for research and practice are presented. Keywords autism spectrum disorders, intervention, restricted and repetitive behaviors

Introduction Restricted and repetitive behaviors (RRBs) are a core symptom of autism spectrum disorders (ASD) (10th revision of the International Statistical Classification of Diseases and Related Health Problems (ICD-10); World Health Organization (WHO), 2009; Diagnostic and Statistical Manual of Mental Disorders, 5th ed. (DSM-5); American Psychiatric Association, 2013). Although research into this area has increased in recent years, compared to social-communication impairments, much less is known about the development and etiology of RRBs. While the field of social-communication intervention has progressed significantly (Kasari and Patterson, 2012; Oono et al., 2013), our understanding on how to treat RRBs within intervention is still poorly understood. Research has primarily focused on pharmacological treatments (Carrasco et al., 2012), comprehensive treatment models (CTMs) that are infrequently published in peerreviewed journals, or behavioral interventions that are predominantly single-case designs (SCDs) (Boyd et al., 2012; Patterson et al., 2010). RRBs in ASD have been categorized into two distinct (yet not mutually exclusive) categories through factor

analytic studies (Honey et al., 2012; Szatmari et al., 2006). In their earliest form, RRBs commonly manifest as lower order behaviors. These are characterized as repetitive motor actions and movements, and physical and/or sensory manipulation of objects. Lower order behaviors are more common in younger children who are developmentally delayed and have language impairments and/or intellectual disabilities (Barrett et al., 2004; Harrop et al., 2014; Lam et al., 2008). Higher order behaviors involve more advanced cognitive functions and are characterized by the presence of routines, an insistence on sameness, and circumscribed interests (Szatmari et al., 2006; Turner, 1999). Unlike social-communication impairments, it is important to acknowledge that not all RRBs are perceived as negative or debilitating for an individual with ASD; RRBs University of California, Los Angeles, USA Corresponding author: Clare Harrop, Center for Autism Research and Treatment, University of California, Los Angeles, 760 Westwood Plaza, Los Angeles, CA 90024, USA. Email: [email protected]

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do not occur pervasively in all children with ASD and may vary in the context in which they are expressed. When observed, RRBs may not interfere with the child’s ability to function socially. However, RRBs have been reported by parents as the most difficult symptom domain to manage (Bishop et al., 2007; Lecavalier et al., 2006; South et al., 2005), while exerting a significant impact on family stress levels (Gabriels et al., 2005; Lecavalier et al., 2006; Lounds et al., 2007). RRBs are known to interfere with child functioning (Gabriels et al., 2005; Nadig et al., 2010) and have been shown to lead to developmental consequences through missed learning opportunities (Pierce and Courchesne, 2001). These behaviors are often stigmatizing for children and families (Lam et al., 2008; South et al., 2005) and have direct consequences on the ability to engage socially (Loftin et al., 2008). The relationship between RRBs and the socialcommunication impairments is poorly understood; while some research supports the notion that RRBs are phenotypically distinct from social-communication impairments in ASD (Happé et al., 2006; Hus et al., 2007; Mandy and Skuse, 2008; Mundy et al., 1994; Ronald et al., 2005, 2006), other studies have found associations between these domains, although these are often weak and not clear cut (Lam et al., 2008; Szatmari et al., 2006; Watt et al., 2008). Added to this, interactions with chronological age and IQ (Gabriels et al., 2005; Harrop et al., 2014; Kim and Lord, 2010; Lam et al., 2008; Militerni et al., 2002; Mooney et al., 2009; Szatmari et al., 2006) and vast heterogeneity (Harrop et al., 2014; Walker et al., 2004) further complicate the ability to make recommendations for intervention. As a result, social-communication impairments and cognitive development are potentially viewed as more concrete intervention targets. Early intervention (EI) is now recognized as key for developmental success and outcome in ASD. Typically, EI focuses on social-communication impairments seen in ASD. This is hypothesized to be due to the perceived primacy of social and communication impairments relative to RRBs (Boyd et al., 2012; Lord et al., 2000). No standardized recommendations exist for the treatment of RRBs despite their status as a core deficit in ASD and obligatory presence for a DSM-5 diagnosis. Aforementioned not all RRBs are detrimental to development or perceived as negative. For example, intense interests may not infer with functioning and actually serve to help a child engage socially through shared interests. RRBs may also serve to reduce anxiety and stress and thus allow the child to participate in social interactions. On the other hand, complex motor behaviors—such as flapping—have been found to disrupt play behaviors and social-communication opportunities (Boyd et al., 2007); thus, the heterogeneity and purpose of these behaviors make recommendations for intervention difficult. Parent-mediated treatment models are common within ASD intervention (Oono et al., 2013). The inclusion of

parents within treatment is promoted (National Research Council, 2001; WHO, 2013) and stems from the theoretical view that enhancing the primary early social relationship will promote naturalistic opportunities for generalization across contexts. While the use of the caregiver within treatment contexts is now popular, the nature, dosage, and design of this involvement vary considerably across studies. While RRBs have been directly targeted as a primary outcome within pharmacological trials (Carrasco et al., 2012; Soorya et al., 2008), the majority of behaviorally based interventions specifically targeting RRBs utilize single-subject designs which are inherently difficult to generalize from (Patterson et al., 2010). In addition, measures specifically focused on quantifying potential change in RRBs are rarely included as outcome measures within studies, possibly attributable to the lack of well-validated standardized measures (Scahill et al., 2013). This is especially true when the main focus of intervention is social communication.

Aims In the last decade, there has been a surge in the reporting of parent-mediated interventions with young children with ASD. Parents are viewed as central to the treatment of the disorder, acting as advocates for their children and often co-therapists in early childhood. Based on the popularity and potential efficacy of parent-mediated interventions, this review explores two main research themes. 1. How are RRBs approached and managed within parent-mediated interventions for young children with ASD? 2. How do parent-mediated interventions measure RRB severity/change?

Method Inclusion criteria Inclusion criteria were the following. 1. All children aged between 12 and 72 months at the start of intervention. 2. All children had a diagnosis of ASD (accurate based on criteria at the time of study). 3. Intervention strategies involved the active training or teaching of parents (in all or a proportion of the participants). 4. The study reported original data (i.e. had not been reported elsewhere previously or a reanalysis of an existing intervention). 5. One of the primary outcomes focused on a child outcome (rather than parent outcomes such as acceptability).

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Harrop 6. Study was published in a peer-reviewed journal— no gray literature, dissertations, or book chapters were included. 7. Study was not a case study, follow-up of an existing intervention, or retrospective study. 8. Due to translation restrictions, only articles in English were included. A range of methodologies were reviewed. All group designs that met the inclusion criteria were included. This encompassed within-subject group designs, quasiexperimental studies, and randomized controlled trials (RCTs). Given the breadth of the SCD literature, difficulty in generalization, and methodological concerns such as the lack of blind assessment, criteria developed by Reichow et al. (2008) for evaluating the quality of SCDs were implemented. This scale has been used previously in reviews of ASD interventions (Kasari and Lawton, 2010; Kasari and Patterson, 2012; Siegel and Beaulieu, 2012) and for inclusion within meta-analysis studies (Makrygianni and Reed, 2010). Reichow et al. (2008) rate indices of quality, separated by primary and secondary indicators. For SCDs, the primary criteria are set at clearly defined participant characteristics, independent and dependent variables. Baselines have to be defined with replicable precision, encompass three measurement points, and appear stable (with no trend in the data). Data have to be visually plotted for each participant, appear stable with minimal overlap between conditions, and show a large shift or trend coincided with the implementation (or removal) or the independent variable (in this case, treatment). The final primary criteria rate indicators of experimental control with at least three visible demonstrations of an experimental effect, over different data points with changes in participant outcomes co-varied with the manipulation of the independent variable (i.e. treatment). Additionally, six secondary indices of quality rate the implementation of inter-observer agreement, reporting of kappa and fidelity, the use of blind raters, the social validity of the study, and attempts to measure generalization and/or maintenance. A rating of strong was set for the inclusion of SCDs within this review. This entailed the study receiving highquality ratings for all primary quality indicators and evidence of three or more secondary quality indicators.

Literature search Articles were located via a systematic and thorough review of the literature. Databases (PubMed, ERIC, MEDLINE, PsycINFO and CENTRAL) were searched using the keywords autism, autism spectrum disorder, pervasive developmental disorder, intervention, treatment, parent, and caregiver on a regular basis from December 2013 to April 2014. Google Scholar was also searched for articles that were not yet indexed. Reference lists of review articles relevant to this review (e.g. Boyd et al., 2012; Oono et al., 2013; Patterson et al., 2012) were also searched.

Two evaluators (the author (CH) and a graduate research assistant) reviewed the results generated from the search. Both have extensive training and experience working with young children with ASD. The author holds a PhD and has published in the field of RRBs in ASD. All citations generated by the search were reviewed firsthand by the author (CH) through their title and abstract to identify those that should be retrieved for further review. The second evaluator reviewed 20% of the abstracts generated to ensure consensus on further inclusion.

Search results A total of 148 articles were identified for further review; 29 studies were included in the review. An overview of these studies is included in Table 1; 119 articles were excluded from further review (see Figure 1); 112 studies did not meet one or more of the established inclusion criteria. The reasons for exclusion are outlined in Figure 1. A total of 27 group design studies were included following initial review; 9 SCDs met all primary inclusion criteria and were all reviewed based on the quality criteria established by Reichow et al. (2008). Anderson et al. (1987) was included in the quality review due to the authors reporting the implementation of a within-subject group design yet reporting multiple-baseline methodology to establish an experimental effect. Thus, it was unclear whether this study was a true group design or a multiple-baseline design. Two SCDs were rated as high using the Reichow et al. (2008) criteria by both evaluators. These were included within the review. This is in line with ratings of quality in previous reviews with most receiving a rating of adequate or poor using this criteria (e.g. Kasari and Lawton, 2010; Patterson et al., 2012). A list of excluded studies can be obtained from the author (CH).

Article review The focus of this review was to explore two areas: first how the management of RRBs was addressed within parent-mediated interventions and second how potential RRB change was measured. To answer the first question, all 29 articles and associated manuals (if available) were reviewed for descriptions of techniques and strategies aimed at reducing/managing RRBs within intervention sessions or through parent training. The evaluators searched articles for terms such as “repetitive behavior,” “self-stimulatory,” and “behavioral management” as well as referring to all available manuals. To answer the question of measurement, primary and secondary outcome measures were reviewed. When the article did not specify primary and secondary outcomes, all reported variables and measures were reviewed. The author (CH) reviewed all eligible articles; 20% were selected at random and reviewed by the second evaluator. These were discussed to ensure consensus on the identification of variables of interest.

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Hanen’s “More Than Words” (MTW) Social communication

Carter et al. (2011)

Preschool Autism Communication Trial (PACT) Parenting training focused on social communication: developmental and behavioral strategies Autism Preschool Program (APP)

Green et al. (2010)

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Self-Directed Learning of Pivotal Response Treatment (PRT): Behavioral Joint attention and language

Nefdt et al. (2010)

Pajareya and Nopmaneejumruslers (2011) Rickards et al. (2007)

Ozonoff and Cathcart (1998)

Oosterling et al. (2010)

Comprehensive treatment model: home-based TEACCH Developmental, IndividualDifference, RelationshipBased (DIR)/Floortime™ Home-based program

Responsive Parent Training

Mahoney and Perales (2003)

Lovaas (1987)

Joint Attention and Play Intervention Lovaas Method: Applied Behavioral Analysis (ABA)

Kasari et al. (2010)

Jocelyn et al. (1998)

Ingersoll and Wainer (2013)

Joint attention routines

Drew et al. (2002)

Early Start Denver Model (ESDM)

Social communication

Aldred et al. (2004)

Casenhiser et al. (2013) Dawson et al. (2010)

Parent-mediated approach

Study

RCT: Home Based + Center Based versus Center Based

Quasi-Experimental: Homebased TEACCH versus Control RCT: DIR/Floortime™ versus TAU

Cluster RCT: Focused Parent Training versus TAU

59

32

22

75

27

20

38

38

25

RCT: Day care + APP versus Day care RCT: Joint Attention Intervention versus Waitlist Quasi-Experimental: Intensive ABA with Treatment Team (including parent) versus Less Intensive One-to-One Control Quasi-experimental: Responsive Training (no controls) RCT: PRT versus Waitlist

27

152

24

48

51

62

28

N

Quasi-Experimental: Project ImPACT (no control)

RCT: Parent Training versus TAU RCT: TAU + PACT versus TAU

RCT: ESDM versus TAU

RCT: MEHRIT versus TAU

RCT: TAU + SocialCommunication Intervention versus TAU RCT: MTW versus TAU

Design

Table 1.  Characteristics of parent-mediated interventions included within review.

Child Functional Emotional Development Cognitive Functioning

Developmental Abilities

Child Language

Child Language

Child Social–Emotional Functioning

Child Engagement; Joint Attention Cognitive Functioning

IQ

Child Language: SocialCommunication Skills; Social Responsiveness

Autism Symptomatology

Child Language

Parent–Child Interaction Cognitive Functioning; Adaptive Functioning

Child Communication

Autism Symptomatology

Primary child outcomes

Adaptive Behavior; Child Behavior

Autism Symptomatology; Cognitive Functioning

Autism Characteristics

Autism Symptomatology; Global Clinical Improvement

Free play

Autism Symptomatology

Autism Symptomatology; Repetitive Behaviors; Cognitive and Adaptive Functioning Sub-Scales Non-Verbal IQ (NVIQ); Autism Symptomatology Parent–Child Interaction; Child Language; Adaptive Functioning

Cognitive Functioning; Adaptive Functioning; Autism Symptomatology Child Language

Child Language; Communicative Initiation; Parent–Child Interaction

Secondary child outcomes

(Continued)

ADOS and ADI-R explored as mediating factors but not RRB subdomain CARS scores explored as potential mediator but not by symptom domain CARS scores reported pre and post but not by symptom domain Not reported

Measure of temperament and atypical behavior (not ASD or RRBs specific) Not reported

Free play scored for selfstimulatory behaviors. Descriptively report change but no follow-up data reported

CARS scores reported pre and post but not by symptom domain Not reported

Items on SRS address RRBs but not reported by symptom domain

Repetitive and stereotyped behavior domain on ADI-R Stereotyped and restricted behavior domain of ADOS

RBS

Not reported

Not reported

Stereotyped and restricted behavior domain of ADOS

Measurement of RRB at exit

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Denver Model (DM)/ Prompts for Restoring Speech (PROMPT)

Parent-Mediated Early Start Denver Model (P-ESDM)

Focused Playtime Intervention (FPI)

Qigong Sensory Training (QST; massage intervention)

QST (massage intervention)

ABA

Nova Scotia (NS) EIBI (parenting training and oneto-one PRT) Parent Education and Behavior Management (PEBM)

Rogers et al. (2006)

Rogers et al. (2012)

Siller et al. (2013)

Silva et al. (2009)

Silva et al. (2011)

Smith et al. (2000)

Smith et al. (2010)

Quasi-experimental: Parent Responsiveness Training versus Waitlist Quasi-experimental: HTP versus Waitlist (single-subject design also reported) RCT: Social Communication versus TAU

SCD: ESDM

RCT: PEBM versus NonSpecific Parent Education versus TAU

Quasi-RCT: NS EIBI versus Waitlist

RCT: Parent Training versus Intensive Intervention (ABA)

Quasi-experimental: QST versus Waitlist

17

20

14

8

70

45

28

47

46

70

RCT: FPI versus Parent Education RCT: QST versus Waitlist

98

10

67

N

RCT: P-ESDM versus TAU

SCD: DM versus PROMPT

RCT: HB versus Center Based with Parent Training (vs Waitlist)

Design

Autism Symptomatology

Cognitive Functioning; Adaptive Functioning

Autism Symptomatology and Psychopathology; Adaptive Functioning Child Social Communication Child Language

Child Language

Child Language; Child Social Skills; Adaptive Functioning; Psychopathology Child Functional Speech; Social Communicative Functioning Autism Symptomatology; Cognitive Functional Parent–Child Interaction; Child Expressive Language Child Social, Language and Maladaptive Behaviors; Autism Symptomatology Autism Symptomatology: Child Language; Maladaptive Behaviors; Child Regulation Cognitive Functioning

Primary child outcomes

Child Communication and Social Behavior; Child Play

Child Engagement

Child Language; Adaptive Functioning; Social–Emotional Functioning; Academic Achievement; Parent Ratings of Change Behavioral Problems; Cognitive Functioning; Adaptive Functioning; Autism Symptomatology Cognitive Functioning; Child Language

Child Regulation

Child Receptive Language; NVIQ; Child Joint Attention

Child Language; Adaptive Functioning; Child Moderating Variables

Expressive and Receptive Language

Secondary child outcomes

Not reported

Not reported

Not reported

Not reported

Items on SRS address RRBs but not reported by symptom domain Not reported

Not reported

ABC completed pre and post but not separated by symptom domain

ABC completed pre and post but not separated by symptom domain

Not reported

ADOS conducted pre and post, but only socialcommunication scores reported Stereotyped and restricted behavior domain of ADOS

Not reported

Measurement of RRB at exit

ASD: autism spectrum disorders; ADOS: Autism Diagnostic Observation Schedule; RRB: restricted and repetitive behaviors; TEACCH: Treatment and Education of Autistic and Related Communication Handicapped Children; ADI-R: Autism Diagnostic Interview–Revised; RCT: randomized controlled trial; MEHRIT: Milton and Ethel Harris Research Initiative Treatment Program; SRS: Social Responsiveness Scale; CARS: Childhood Autism Rating Scale; EIBI: Early Intensive Behavioral Intervention; RBS: Repetitive Behavior Scale; ImPACT: Improving Parents as Communication Teachers; SCD: single-case designs; TAU: Treatment As Usual; ABC: Autism Behavior Checklist.

Wong and Kwan (2010)

Welterlin et al. (2012)

Venker et al. (2012)

Vismara et al. (2009)

ESDM: Brief 12 week, 1 h/ week Parent verbal responsiveness (adapted from MTW) Parent training: Home TEACCHing Program (HTP) Social communication

Home-based individualized program (HB)

Roberts et al. (2011)

Tonge et al. (2012)

Parent-mediated approach

Study

Table 1. (Continued)

Harrop 5

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Stage 1 Studies retrieved for further review N = 148

• • • • • • • •

Stage 2 N = 36

• • • •

Excluded studies based on inclusion criteria N = 112 Age of children (N = 30) No diagnosis of ASD in some or all of children (N = 12) Retrospecve study (N= 6) Case study/descripve (N = 13) Reanalysis of previous intervenon (N = 2) No intervenon provided/unclear how intervenon provided (N = 7) Reported elsewhere previously (N = 4) Role of parent unclear/not parent mediated (N = 17) Follow up study (N = 1) Primary outcome not child focused (N = 11) Not available in English (N = 2) Unpublished/not peer reviewed (N = 7)

Excluded SCDs based on Reichow et al. (2008) Quality Rangs N=7 Included studies: 29 Group Design N = 27 SCD N = 2

Figure 1.  Included and excluded studies: reasons for exclusion. SCD: single-case design; ASD: autism spectrum disorders.

Results 1.  How are RRBs approached and managed within parent-mediated interventions for young children with ASD? Overall, little systematic and/or manualized recommendations within the parent-mediated literature were found. Of the studies reviewed, intervention approaches broadly fell into three categories: behavioral, social communication/ developmental (often with embedded behavioral strategies), and CTMs. A total of 13 studies reported specific strategies for managing RRBs. Behavioral interventions reported strategies based on Applied Behavioral Analysis (ABA) principles. Both Lovaas (1987) and Smith et al. (2000) targeted the reduction of unwanted behaviors (including RRBs) using the University of California, Los Angeles (UCLA) Lovaas method. While older studies (e.g. Lovaas, 1987) report the use of aversive consequences to selfstimulatory behaviors, more recent ABA-based approaches (Nefdt et al., 2010; Smith et al., 2000, 2010) report shaping and reinforcing more socially acceptable behaviors through Discrete Trail Training (DTT) or Pivotal Response Treatment (PRT). Four studies (Dawson et al., 2010; Rogers et al., 2006, 2012; Vismara et al.,

2009) report developmental behavioral strategies within the Early Start Denver Model (ESDM; termed Denver Model in Rogers et al., 2006), which integrates a relationship-focused developmental intervention with ABA principles. Two studies (Carter et al., 2011; Venker et al., 2012) implemented interventions based on Hanen’s More Than Words (MTW; Sussman, 1999). This approach favors building sensory behaviors into people games to engage the child within social routines. Pajareya and Nopmaneejumruslers (2011) employed a Developmental, Individual-Difference, Relationship-Based (DIR) approach (Floortime™; Greenspan and Wieder, 1997). Floortime discusses the uses of individualized strategies for dealing with perseverative behaviors while also joining with the child in these behaviors to make them more social. Two studies (Ozonoff and Cathcart, 1998; Welterlin et al., 2012) reported the findings of the implementation of Treatment and Education of Autistic and Related Communication Handicapped Children (TEACCH), involving parents within the therapy team or via a homebased approach. CTMs, such as TEACCH, target a range of developmental areas through multiple intervention components and specifically target structured teaching of appropriate skills. Two studies mentioned behavioral management strategies and tips given to parents to address similar problems, such as problematic or unwanted behaviors, but did not specifically mention RRBs (Ingersoll and Wainer, 2013; Jocelyn et al., 1998). Two studies implemented a Qigong Sensory Massage technique, which is hypothesized to reduce sensory stimulation and increase regulation (Silva et al., 2009, 2011). The remaining 12 studies made no specific reference to the management of RRBs. The majority of these studies focused on increasing engagement and social communication between the dyads. It is important to acknowledge that many of the studies implemented parent-training/mediation within free play sessions (e.g. Aldred et al., 2004; Drew et al., 2002; Kasari et al., 2010; Green et al., 2010; Venker et al., 2012). Higher frequencies of RRBs are known to associate negatively with advancing play skills (Azrin et al., 1973; Bruckner and Yoder, 2007; Harrop et al., 2014; Honey et al., 2007); therefore, interventions employing this form of approach may lead to a reduction of RRBs as a result of increased play skills and engagement. How do parent-mediated interventions measure 2.  RRB severity/change? No study included a measure solely focused on RRBs as a primary outcome. Parent-mediated approaches generally included measures of social communication, autism severity, child language, cognitive functioning, and adaptive behavior (see Table 1).

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Harrop Only one study included a separate measure focused specifically on RRBs as a secondary outcome. Dawson et al. (2010) used the Repetitive Behavior Scale (RBS; Bodfish et al., 1999) to characterize change following intervention. A total of 16 studies looked at severity of autism symptoms as a primary or secondary outcome; though only four reported RRBs as a separate symptom domain (Aldred et al., 2004; Drew et al., 2002; Green et al., 2010; Rogers et al., 2012) either through direct observation (Autism Diagnostic Observation Schedule (ADOS); Lord et al., 2002) or parent report (Autism Diagnostic Interview–Revised (ADI-R); Lord et al., 1994). Studies were often vague in their reporting of behavioral change; for example, Rickards et al. (2007) reported change in both researcher- and parent-reported behaviors; however, questionnaire measures were not ASD specific and did not report RRBs. Despite implementing very specific strategies to reduce RRBs, Lovaas (1987) descriptively reported change in RRBs at exit; however, no data or coding scheme were available.

Conclusion and recommendations No parent-mediated intervention included in this review focused on RRBs as a primary outcome, both in terms of an intervention target and outcome measurement. Nearly half (44%) of the studies reported specific strategies for managing RRBs, with most focusing on ABA principles. Over a third of studies (41%) made no reference to the management of RRBs with a primary focus on engagement and social communication. Overall, parent-mediated approaches were split in their reporting of RRB management strategies with behaviorally based approaches reporting specific strategies. It seems unlikely that the discussion of RRBs would not occur between parents and therapists; therefore, it is possible that more naturalistic parent-mediated approaches target RRBs in a less systematic and more individualized way within the context of the intervention targets. It should be noted that the interventions included in this review varied dramatically in their dosage of treatment, with some reporting 40 h a week (Lovaas, 1987), comprehensive treatment approaches (Ozonoff and Cathcart, 1998) and others relying on self-directed (Nefdt et al., 2010) or 1 h a week of direct parent teaching (Vismara et al., 2009). No study included a measure solely focused on RRBs as a primary outcome with outcomes focused on autism severity, social-communication outcomes, language, and cognitive functioning. One study included a measure focused solely on RRBs as a secondary outcome. Only four studies separated ASD symptomatology to include a separate analysis of RRBs at end point.

Current state of interventions for RRBs Much of what we know about the treatment of RRBs in young children with ASD comes from the SCD literature.

The majority of the evidence base for treating and managing these behaviors comes from studies applying ABA principles. Boyd et al. (2012) provide an excellent review of the current state of behavioral interventions; however, they conclude that currently no empirically established behavioral or pharmacological intervention exists for RRBs. Additionally, the majority of the studies included in their review do not include parents as mediators of treatment. The lack of standardized (and well-documented) approaches to treat RRBs is possibly due to the vast heterogeneity observed in these behaviors; at least two distinct subtypes of RRBs have been identified (e.g. Turner, 1999), and strategies vary by the type of RRB within each subtype. For lower order RRBs, suggested strategies include blocking, interrupting, and redirecting behaviors. Removal of positive consequences and reinforcement of alternative behaviors are also promoted, as well as antecedent-based strategies. However, these strategies often contrast with the theoretical underpinnings of more naturalistic socialcommunication interventions that promote following the child’s lead and promoting engagement in a shared activity. Interventions promoting social engagement in peer activities have shown promising collateral effects on the reduction of RRBs in some children (e.g. Lee et al., 2007), and this is an area that requires further research. For higher order RRBs (often not seen in very young children with ASD), methods include cognitive behavioral therapy, differential reinforcement, and consequence- or antecedentbased approaches.

Recommendations for intervention Interventions (including parent-mediated approaches) need to be more transparent in their reporting of approaches for targeting RRBs within practice, particularly those with a social-communication focus. Recent observational research demonstrates that young children with ASD demonstrate a wide range of RRBs even within a short observation timeframe (Barber et al., 2012; Harrop et al., 2014; Militerni et al., 2002; Watt et al., 2008), and these behaviors appear to be relatively stable in early childhood (Harrop et al., 2014; Richler et al., 2010). Many of the interventions reported used play sessions to implement parent-mediated strategies. Play complexity has been shown to associate with RRBs (Bruckner and Yoder, 2007; Harrop et al., 2014; Honey et al., 2007), and repetitive behaviors and routines are likely to impact engagement and advancing play skills (Boyd et al., 2007). Therefore, it is unlikely that some (if not the majority of) parents did not raise concerns or discuss the presence of RRBs either within the context of the intervention or in the home environment and beyond. A key recommendation for future research is the inclusion of manualized strategies and/or examples on how these behaviors were approached even if they were not the specific target of the intervention.

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Recommendations for research and measurement Given the popularity of parent-mediated interventions, further systematic research is required to explore if and how these approaches impact RRBs. This will allow us to further our understanding of how the different areas of ASD are conceptually related and how to approach these behaviors within intervention. Given the vast heterogeneity of RRBs in children with ASD, these behaviors may be viewed as more difficult to assess and change. The lack of a sensitive and standardized outcome measure is an issue within RRB intervention research (Scahill et al., 2013); while the ADOS is the gold standard for diagnosis and study inclusion within ASD, it may not be sensitive to change within a short intervention timeframe (e.g. Green et al., 2010). Additionally, many parent-mediated interventions report main outcome measures contingent with the mode of intervention and thus do not include a measure of RRBs. While RRBs are a core symptom of ASD, the expression of these behaviors may not be pervasive in all children or limited to specific contexts and activities. Therefore, observational coding of these behaviors may fail to capture these behaviors and change over time. A combination of examiner-rated, parent-reported, and observational measures would be beneficial to assess potential change in RRBs post-intervention (irrespective of delivery method and approach). Further research to disentangle how RRBs relate to other core impairments in ASD will also advance our understanding as to how to approach these behaviors within EI. Currently, the picture is mixed, with some research suggestive of a separation of RRBs and socialcommunication abilities (e.g. Hus et al., 2007; Ronald et al., 2005, 2006) and others finding associations between these domains (Lam et al., 2008; Watt et al., 2008).

Limitations The majority of studies included in this review were group designs. While the use of the criteria developed by Reichow et al. (2008) provided a systematic way to include only SCDs with a quality rating of high, only two SCDs were included within this review. This was expected based on previous reviews implementing these criteria (e.g. Kasari and Lawton, 2010). Group designs were automatically included in the review without a pre-defined quality benchmark. Thus, while the SCDs included were of the highest quality in terms of their methodology, the same assumption cannot be made for the group designs. The majority of SCDs did not meet high-quality criteria due to the instability of baseline conditions and visual data. However, SCDs provide a wealth of data on specific intervention strategies and are particularly informative for the development of treatment approaches (Patterson et al., 2012). The majority of SCDs provided clear information

regarding the participants and the intervention methodology. In terms of developing intervention practices for RRBs, both larger controlled and smaller SCD studies will be equally informative for moving the field forward.

Conclusion Parent-mediated interventions are currently favored within intervention research due to the increased likelihood of generalization outside the immediate treatment context. Despite promising findings on social-communication skills (Oono et al., 2013), RRBs remain the forgotten symptom. Few studies explicitly outlined techniques for the management of RRBs. Even fewer measured specific change in RRBs pre- and post-intervention. Only one study (Dawson et al., 2010) reported specific techniques to approach unwanted behaviors while also directly measuring change post-intervention through the RBS. Recommendations for research and practice include transparency in the reporting of advice given to parents and a greater focus on the measurement of RRBs pre- and post-intervention. Acknowledgements The author thanks Helen McConachie for her comments and discussion and Lilit Hovespyan for her assistance with literature searches and reviewing.

Funding This research received no specific grant from any funding agency in the public, commercial, or not-for-profit sectors.

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