Making a Writing Plan

REVIEW PAPER

Treatment of Echolalia in Individuals with Autism Spectrum Disorder: a Systematic Review

Leslie Neely1 & Stephanie Gerow2 & Mandy Rispoli3 & Russell Lang4 & Nathan Pullen4

Received: 15 October 2015 /Accepted: 25 November 2015 /Published online: 4 December 2015 # Springer Science+Business Media New York 2015

Abstract Echolalia can lead to communication breakdowns that increase the likelihood of social failure and stigmatization in children with autism spectrum disorder (ASD). In an effort to facilitate evidenced-based intervention and inform future research, this systematic review analyzes peer-reviewed stud- ies involving the treatment of echolalia in individuals with ASD. Using predetermined inclusion criteria, a total of 11 studies were identified, reviewed, and summarized in terms of the following: (a) participant characteristics (e.g., verbal and cognitive functioning), (b) type of echolalia (e.g., delayed or immediate), (c) intervention procedures, (d) intervention outcomes, (e) maintenance and generalization of outcomes, and (f) research design and other indicators of rigor (i.e., cer- tainty of evidence). Nine studies successfully reduced echola- lia in a total of 17 participants. However, only six of those nine studies met criteria to be classified as providing the highest level of certainty (i.e., conclusive). The findings of this review suggest that a number of treatment options can be considered promising practices for the treatment of echolalia in children with ASD. Although no single treatment package can be de- scribed as well-established evidence-based practice, all 11 studies involved behavior analytic intervention components, suggesting strong support for operant-based treatments. In particular, behavior analytic interventions demonstrating

conclusive levels of evidence included cues-pause-point, dif- ferential reinforcement of lower rates of behavior, script train- ing plus visual cues, and verbal modeling plus positive rein- forcement for appropriate responses. Implications for practi- tioners and directions for future research are offered.

Keywords Autism spectrum disorder . Echolalia .

Treatment . Systematic review

Autism spectrum disorder (ASD) is a developmental disorder characterized by deficits in social communication skills and excesses in repetitive and restrictive patterns of behaviors (Diagnostic and Statistical Manual of Mental Disorders [5th ed.; DSM-5]; American Psychiatric Association [APA] 2013). The combination of behavioral excesses and deficits can man- ifest as a repetitive, restricted pattern of vocal behavior called echolalia (Stribling et al. 2007). Echolalia is typically defined as the socially awkward or inappropriate verbatim repetition of part or all of a previously spoken utterance (Karmali et al. 2005; Stribling et al. 2007; Valentino et al. 2012). The initial utterance, that is then repeated, may come from another per- son in the environment or from a recording (television or audio source) and maybe immediate or delayed. Immediate echolalia occurs when the latency between initial utterance and repetition is within a few seconds, whereas delayed echolalia occurs when the time between the initial utterance and the repetition involves longer durations, inclusive of repetitions occurring days after the initial utterance being echoed (Foxx et al. 2004; Hetzroni and Tannous 2004). Another form of echolalia is palilalia. Palilalia involves the repeating of one’s own words in a quiet whispered voice immediately following the initial typical volume utterance (Karmali et al. 2005).

* Leslie Neely [email protected]

1 Department of Educational Psychology, The University of Texas at San Antonio, 501 W. Cesar E. Chavez Blvd., San Antonio, TX 78207-4415, USA

2 Texas A&M University, College Station, TX, USA 3 Purdue University, West Lafayette, IN, USA 4 Texas State University, San Marcos, TX, USA

Rev J Autism Dev Disord (2016) 3:82–91 DOI 10.1007/s40489-015-0067-4

Although language repetition is part of typical child development (Howlin 1982). some children with ASD en- gage in echolalia that persists past the early childhood developmental period (Barrera and Sulzer-Azaroff 1983; Neely 2014; Fay 1969). In addition, children with ASD often engage in echolalia that lacks social context Lovaas et al. 1973) and occurs at a higher rate than in typically developing children (Fay 1973).

Echolalia may (a) complicate educational programs de- signed to improve speech, (b) contribute to communication breakdowns, (c) increase the likelihood of social failure or stigmatization, and (d) increase the risk of challenging behav- ior (Light et al. 1998; Valentino et al. 2012). For example, Valentino et al. (2012) identified a 3-year-old male with ASD who repeated the instruction Bsay^ during echoic train- ing. The immediate echolalia was interfering with instruction and complicating the educational program aimed at teaching the child to tact. Previous research aimed at identifying the operant function of echolalia suggests that the complete range of functions found to maintain other behaviors (e.g., automatic reinforcement, socially mediated positive reinforcement, and socially mediated negative reinforcement) may also reinforce and maintain echolalia (Goren et al. 1977; Healy and Leader 2011).

A descriptive review by Kavon and McLaughlin (1995) identified two interventions with preliminary support for the treatment of echolalia (i.e., cues-pause-point and more general verbal prompting interventions). Cues-pause-point is a behav- ioral intervention that has been evaluated for the treatment of immediate echolalia. Cues-pause-point was introduced by McMorrow and Foxx (1986) in their treatment of a 21-year- old male with ASD. The cues-pause-point intervention con- sists of a trainer providing a visual cue to the learner to remain silent (cue). The trainer then maintains the visual cue while providing instructions about the upcoming teaching session. The trainer poses a question and provides a short pause fol- lowing the question (pause). Finally, the trainer points to a card to prompt the learner to verbalize the answer to the ques- tion (point). For example, to teach the individual with ASD to respond appropriately to the question, BWhat is your name?,^ the trainer held up an index finger to cue silence (cue), stated, BI’m going to ask you some questions, do your best to answer them correctly^ (pause), asked the question, pointed to a card with the person’s name prompting the correct response (point), and then provided reinforcement contingent on the correct response. Following the initial study by McMorrow and Foxx, follow-up studies extended the procedure to indi- viduals with less developed language skills (McMorrow et al. 1987) and individuals with intellectual disabilities (Foxx, Faw, McMorrow, Kyle, & Bittle 1988) and then demonstrated that reductions of echolalia following cues-pause-point could be maintained up to 57 months following the cessation of the intervention (Foxx and Faw 1990).

Kavon and McLaughlin’s (1995) review categorized the remaining interventions as verbal prompting interventions. Studies in that category used a combination of reinforcement, prompting, and error correction to reduce echolalia but did not utilize the more specific sequence of cues-pause-point (e.g., Freeman et al. 1975; Lovaas 1977). For example, Freeman et al. (1975) used positive reinforcement for correct responding to questions and an error prevention procedure (consisting of interrupting the echolalia) to treat the echolalia of a 5-year-old male with autism. The intervention produced decreases in echolalia that maintained following the with- drawal of intervention. Although the review by Kavon and McLaughlin provides evidence in support of these interven- tions, additional studies have emerged over the last 20 years and an updated systematic review appears warranted.

Therefore, the purpose of this review is to update and ex- tend the previous review by Kavon and McLaughlin by (a) utilizing broader inclusion criteria not limited to behavioral (operant) approaches, (b) conducting a systematic review of the literature, (c) rating each included study’s certainty of ev- idence (quality of research design and controls) so results can be considered in light of each study’s methodological rigor, and (d) identifying advances in treatment that may have de- veloped since the previous review. A review of this nature is intended to offer directions for future research and to provide guidance to practitioners interested in the use of evidence- based treatments for echolalia in children with ASD.

Method

Search Procedures

Four electronic databases were searched to identify potential studies for this review: ERIC (EBSCO), Medline, Psychology and Behavioral Sciences Collection, and PsycINFO. There were no limitations on publication year, but results were lim- ited to English language, peer-reviewed research. Terms to describe individuals with an ASD were combined with terms to describe echolalia. The terms for individuals with an ASD included BAsperger,^ Bautis*,^ Bdevelopmental disab*,^ BASD,^ and BPDD-NOS.^ The terms searched to describe echolalia included Becholal*,^ Brepetitive speech,^ Brepetitive verbal*,^ Brepetitive talking,^ Brepetitive communication*,^ and Bpalilalia.^ Following the initial search, the last name of the first author of each included study was also entered into PsychINFO to identify any other potentially relevant studies that had been published by that author. Finally, the reference list of Kavon and McLaughlin (1995) was examined for ad- ditional studies meeting inclusion criteria.

These search procedures were conducted in May 2014, updated in April 2015, and yielded a total of 568 articles (534 from the original search and 34 from the updated search).

Rev J Autism Dev Disord (2016) 3:82–91 83

The title and abstracts of the 568 articles were screened using the predetermined inclusion criteria (see BInclusion Criteria^ section) to identify articles for potential inclusion in this re- view. Following this screening of title and abstracts, a total of 46 articles were identified for further review.

Inclusion Criteria

The 46 articles were then downloaded and evaluated based on the pre-set inclusion criteria. Studies were included if they (a) included a participant diagnosed with ASD or was described as an individual with Bautistic-like behaviors^ (included due to the age of the literature base), (b) implemented an interven- tion and reported outcomes for echolalia (inclusive of palilalia and defined as repetition of a previously spoken word or phrase) as a dependent variable, (c) employed an experimental design (inclusive of single-case and group experimental de- signs), and (d) echolalia outcomes for the individual with ASD could be disaggregated from participants without ASD and target behaviors other than echolalia. Studies which im- plemented interventions for individuals with ASD who uti- lized echolalic speech but did not present outcomes related to the echolalia were excluded (e.g., Barrera and Sulzer- Azaroff 1983; Charlop-Christy and Kelso 2003; Charlop 1983). Studies which evaluated echolalia under different con- ditions but did not implement an intervention to address echo- lalia were also excluded (e.g., Rydell and Mirenda 1994; Violette and Swisher 1992). In addition, studies which imple- mented interventions to treat other repetitive speech (i.e., noncontextual vocal stereotypy, such as a sound rather than a word or phrase) were excluded (e.g., Mancia et al. 2000; Ahearn et al. 2000; Taylor et al. 2005). Studies excluded because data on echolalia were not disaggregated from other outcomes in- volving other topographies of behavior were Arntzen et al. (2006) and Mancia et al. (2000). For example, Arntzen et al. (2006) taught a 44-year-old woman functional verbal responses and tracked subsequent decreases in aberrant verbal behavior. Although aberrant verbal behavior included repetitive echolalic responses, the aberrant verbal behavior also included Bpsychotic^ verbalizations and results for the two were collapsed into one dependent variable. Finally, Cohen (1981) was excluded because the figure referenced in the article was not included in the article and was not accessible to the authors after multiple attempts to locate the figure through university- based library services. Ultimately, a total of 11 studies met inclusion criteria and were included in this review.

Descriptive Synthesis

Included studies were reviewed and summarized based on the following categories: (a) participant characteristics (e.g., ver- bal and cognitive functioning), (b) type of echolalia (e.g., de- layed or immediate), (c) intervention procedures, (d)

intervention outcomes, (e) maintenance and generalization of outcomes, and (f) research design and other indicators of rigor (i.e., certainty of evidence). Participant description in- cluded the number of participants with ASD, their ages, and gender. Participant verbal and cognitive functioning was cod- ed using reported standardized assessments or was gleaned from detailed descriptions of participant functioning. Echolalia was coded as either immediate, delayed, or palilalia, and when noted in the reviewed study, the operant function of echolalia was noted. Various procedural aspects were coded to identify intervention protocols or components (e.g., cues- pause-point protocol, script training, or reinforcement procedures)

Intervention outcomes were summarized and coded as nega- tive, mixed, or positive. As all 11 studies employed single-case research designs, study outcomes were determined based on vi- sual analysis criteria for single-case research outlined by Kennedy (2005). A study was rated as having negative results if there was no reduction observed in echolalia as indicated by a flat or increasing trend in the intervention phase as compared to the baseline phase. Studies were coded as having mixed results if some, but not all, of the participants demonstrated a reduction in echolalia during the intervention phase relative to the baseline phase. Positive results indicated that echolalia decreased in all participants during intervention phase as relative baseline.

The study’s capacity to provide a certainty of evidence was rated as suggestive, preponderant, or conclusive, with conclu- sive being the highest rating (Schlosser 2009; Simeonsson and Bailey 1991; Smith 1981). Studies rated as conclusive had the following: (a) an experimental design capable of establishing experimental control (e.g., ABAB, multiple-baseline design, alternating treatments design), (b) sufficient interobserver agreement (IOA) collected on the observed educator behav- iors (i.e., agreement coefficients above 80 % and IOA collect- ed for a minimum of 20 % of the sessions), (c) intervention procedures detailed enough to promote replication of the pro- cedures, (d) operationalized descriptions of the dependent var- iable, and (e) demonstrated convincing effects of the interven- tion for every participant (i.e., received a rating of positive results). A study rated as preponderant met most of the criteria for a Bconclusive^ study, but results may have demonstrated Bmixed^ effects of the intervention for some or all of the participants with ASD. Any study that (a) lacked an experi- mental design capable of establishing experimental control, (b) did not meet the minimum IOA criterion, (c) did not operationally define the intervention procedures, (d) or did not operationally define the dependent variable were auto- matically rated as offering suggestive evidence.

Interrater Reliability

Inclusion Criteria To ensure accurate application of the in- clusion criteria, two raters reviewed each of the 46 articles,

84 Rev J Autism Dev Disord (2016) 3:82–91

resulting from the systematic search and initial title/abstract review, for potential inclusion. Agreement was reached on whether to include or exclude a study on 100 % of the articles.

Descriptive Synthesis To establish interrater reliability (IRR) for the data summaries, two independent raters coded five of the 11 included articles (46 %). A third rater reviewed the independent data summaries and made a decision as to wheth- er the summaries agreed. IRR was calculated based on wheth- er the two raters agreed on the extracted data. There were a total of 30 items in which there could be agreement or dis- agreement (i.e., five studies with six data categories each). IRR was calculated using percent agreement by dividing the total number of agreements by the sum of the agreements and disagreements and multiplying by 100 % to convert to a per- centage. Initial agreement for the coding of studies was 90 %. In instances of disagreement, the raters discussed until 100 % agreement was reached.

Results

Table 1 created from the coded study summarizes and displays each study according to the following: (a) participant charac- teristics (e.g., verbal and cognitive functioning), (b) type of echolalia (e.g., delayed or immediate), (c) intervention proce- dures, (d) intervention outcomes, (e) maintenance and gener- alization of outcomes, and (f) research design and other indi- cators of rigor (i.e., certainty of evidence).

Participant Characteristics

The 11 studies included a total of 25 participants with ASD. Ten of the 11 studies reported the gender of their participants with 17 male and 5 female participants. One study did not report participants’ gender (Laski et al. 1988). All of the stud- ies reported the participants’ ages, with a mean reported age of 8 years (range 3–21 years) across studies.

Ten studies (90 %) reported information regarding partici- pants’ verbal or cognitive functioning. Four studies (36 %) reported results from standardized cognitive assessments (i.e., Stanford-Binet, Merrill-Palmer, and Peabody Picture Vocabulary Test (PPVT)) with three studies including four participants with extremely low cognitive functioning (16 %; Handen et al. 1984; McMorrow and Foxx 1986; Nientimp and Cole 1992) and one study including one participant with below-average to average cognitive functioning (4 %; Freeman et al. 1975). Three studies (27 %) reported results from standardized language assessments (i.e., Alpern-Boll and PPVT-III) indicating extremely low verbal abilities and below-average cognitive functioning for eight of the 25 par- ticipants (32 %; Foxx et al. 2004; Karmali et al. 2005; Palyo et al. 1979). Five of the studies reported descriptive

information regarding the language functioning of partici- pants, all of which suggested below-average verbal abilities for 16 of the 25 participants (64 %; Ganz et al. 2008; Hetzroni and Tannous 2004; Karmali et al. 2005; Laski et al. 1988; Valentino et al. 2012).

Type of Echolalia

Across the 11 studies, five targeted immediate echolalia only (45 %; Foxx et al. 2004; McMorrow and Foxx 1986; Nientimp and Cole 1992; Palyo et al. 1979; Valentino et al. 2012), two targeted delayed echolalia only (18 %; Ganz et al. 2008; Handen et al. 1984), and three studies (27 %) targeted both immediate and delayed echolalia (Freeman et al. 1975; Hetzroni and Tannous 2004; Laski et al. 1988). Finally, one study targeted palilalia (Karmali et al. 2005). No study report- ed operant functions of target behaviors.

Intervention Procedures

All of the 11 studies employed an intervention with behavioral analytic components (e.g., differential reinforcement, model- ing, prompting). Five of the 11 studies (45 %) evaluated the effects of specific treatment package on echolalia (i.e., cues- pause-point; Natural Language Paradigm, and computer- based intervention). Cues-pause-point was the most frequent treatment package evaluated (n=3; 27 %; Foxx et al. 2004; McMorrow and Foxx 1986; Valentino et al. 2012). For exam- ple, Valentino and colleagues (2012) evaluated the use of cues-pause-point to treat a 3-year-old male child who echoed the instruction say during echoic training. Decreased echolalia and increase appropriate responding (e.g., repeating the target word without echoing say) were noted.

Laski et al. (1988) trained parents to implement the Natural Language Paradigm within a clinical setting and then assessed the effects of the parent implemented program on child com- munication outcomes. Parents were taught to use direct rein- forcement of verbal attempts, to promote turn-taking with play items, to vary stimuli and exemplars, and to utilize shared control (i.e., rotating between child-led and parent-led activi- ties). Although appropriate vocalizations were the primary dependent variable, child engagement in echolalia was mea- sured as an ancillary variable with mixed results noted for the effects of the Natural Language Paradigm on echolalia.

The final treatment package was a computer program enti- tled BI Can Word It Too^ that was available in both Arabic and Hebrew (Hetzroni and Tannous 2004). The program presented participants with a simulated situation in which a parent asked the participant a question. The participant would then choose the appropriate sentence or question option, and an animation of their choice would be played. For example, if the question was Bwhat would you like to play with^ and the participant selected the option BI want to play ball with you,^ an

Rev J Autism Dev Disord (2016) 3:82–91 85

T ab

le 1

In te rv en ti on s st ud ie s to

d ec re as e ec h o la li a fo r in d iv id u al s w it h A S D

A rt ic le

P ar ti ci p an t ch ar ac te ri st ic s

T y p e o f ec h o la li a

In te rv en ti o n p ro ce d u re s

In te rv en ti o n

o u tc o m es

M ai n te n an ce

an d g en er al iz at io n

o f o u tc o m es

C er ta in ty

o f ev id en ce

F o x x et al . (2 0 0 4 )

n = 2 ; m al es ; 5 an d 6 y ea rs

P 1 : P P V T -I II 4 0 (s ta n d ar d sc or e) ;

E O W V T 5 5 (s ta n d ar d sc o re );

C A R S 3 7 ; G A R S A u ti sm

Q u ot ie n t 9 7

P 2 : P P V T -I II 4 0 (s ta n d ar d sc or e) ;

E O W V T 5 8 (s ta n d ar d sc o re );

C A R S 4 0 ; G A R S A u ti sm

Q u ot ie n t 8 3

R ep ea te d w o rd s co n ta in ed

in an

as k ed

q u es ti o n ;

im m ed ia te ec h o la li a

C ue s- p au se -p o in t

P os it iv e

P o si ti v e u si n g a g ra d u al

in te rv en ti o n fa d in g

p ro ce d ur e) / p o si ti v e ac ro ss

n o v el se tt in g , an d n o ve l

tr ai ne r

C o n cl u si v e

F re em

an et al .

(1 9 75 )

n = 1 ; m al e; 5 y ea rs

N o st an d ar d iz ed

la n g u ag e

as se ss m en t; IQ

as se ss m en ts

ra n g ed

fr o m 8 1 (S ta nf o rd -B in et )

to 1 2 0 (M

er ri ll -P al m er ).

Im m ed ia te an d d el ay ed

ec h o la li a

P o si ti v e re in fo rc em

en t

fo r co rr ec t an sw

er s

an d er ro r co rr ec ti o n

p ro ce d u re

to b lo ck

ec h o la li a

P os it iv e

N o t re p o rt ed /n o t re p o rt ed

S u g ge st iv e; A B A

d es ig n ; IO

A w as

n o t as se ss ed

G an z et al .

(2 0 08 )

n = 2 m al es ; 7 an d 1 2 y ea rs b

N o st an d ar d la n g u ag e as se ss m en ts .

N o co g n it iv e as se ss m en ts .

P 1 : d if fi cu lt y w it h W h - q u es ti o n s;

ag e- ap p ro p ri at e p h o n o lo g ic al

an d se m an ti c sp ee ch

P 2 : A g e- ap p ro p ri at e p ho n o lo g ic

an d se m an ti c sk il ls ; ra re ly

in it ia te d co n v er sa ti o n .

P 1 : R ep ea te d p h ra se s fr o m

te le v is io n an d v id eo

g am

es ; d el ay ed

ec h ol al ia

P 2 : R ep ea te d p h ra se s fr o m

so n g s an d b o o k s; de la y ed

ec h o la li a

S cr ip t tr ai n in g an d

v is u al cu es

P os it iv e

N o t re p o rt ed /n o t re p o rt ed

C o n cl u si v e

H an d en

et al .

(1 9 84 )

n = 1 ; m al e; 1 6 y ea rs

N o st an d ar d iz ed

la n g u ag e

as se ss m en t; m en ta l ag e o f

5 y ea rs 11

m o n th s

(S ta n fo rd -B in et ).

R ep ea te d st at em

en ts an d /o r

as k in g sa m e q u es ti o n

m u lt ip le ti m es

a d ay ;

d el ay ed

ec h o la li a

D if fe re n ti al re in fo rc em

en t

o f lo w er

ra te s

P os it iv e

P o si ti v e at 9 an d 1 4 m o n th s

fo ll o w -u p /n o t re po rt ed

C o n cl u si v e

H et zr o n i an d

T an n o u s (2 0 0 4 )

n = 5 ; 3 m al es

an d 2 fe m al es ;

7 .8 , 8 , 8 .5 , 11 .5 , an d 1 2 .5

y ea rs

N o st an d ar d iz ed

la n g u ag e

as se ss m en ts . N o co g n it iv e

as se ss m en ts .

Im m ed ia te an d d el ay ed

ec h o la li a

S o ft w ar e p ro g ra m

(I C an

W o rd

it T o o )

M ix ed

N o t re p o rt ed /r es u lt s

g en er al iz ed

to au th en ti c se tt in g s

fo r so m e p ar ti ci p an ts

S u g ge st iv e; o n e le g

o f ev er y p ar ti ci p an t’ s

M B D d id

n o t

d em

o n st ra te ef fe ct s;

ad d it io n al in fo rm

at io n

n ec es sa ry

fo r re p li ca ti o n

of in te rv en ti on

K ar m al i et al .( 2 0 0 5 )

N = 5 ; 4 m al e an d 1 fe m al e;

3 to

4 y ea rs

N o rm

-r ef er en ce d as se ss m en ts

in d ic at ed

sp ee ch

d el ay s fo r al l

p ar ti ci p an ts ; n o co gn it iv e

as se ss m en ts re p o rt ed

P al il al ia w as

al l re la te d to

ch il d re n ’s m o v ie s o r

so n g s; d el ay ed

p al il al ia

T ac t m od el in g p lu s

p o si ti ve

re in fo rc em

en t

o f ap p ro p ri at e re sp o ns es

P os it iv e

N o t re p o rt ed /p o si ti v e

ac ro ss

se tt in g s

C o n cl u si v e

L as k i et al . (1 9 8 8 )

n = 3 ; g en de r n o t sp ec if ie d ;

5 .8 , 6 .2 , an d 8 .1 1 y ea rs a

Im m ed ia te an d d el ay ed

ec h o la li a

N at u ra l L an g u ag e

P ar ad ig m

M ix ed

N o t re p o rt ed /n o t re p o rt ed

S u g ge st iv e (e ch o la li a w as

an an ci ll ar y d ep en d en t

v ar ia b le )

86 Rev J Autism Dev Disord (2016) 3:82–91

T ab

le 1

(c o n ti n u ed )

A rt ic le

P ar ti ci p an t ch ar ac te ri st ic s

T y p e o f ec h o la li a

In te rv en ti o n p ro ce d u re s

In te rv en ti o n

o u tc o m es

M ai n te n an ce

an d g en er al iz at io n

o f o ut co m es

C er ta in ty

o f ev id en ce

N o st an d ar d iz ed

la n g u ag e

as se ss m en ts ; P 1:

co u ld

im it at e

so un d s an d a fe w w o rd s o n

re q u es t, ra re ly

in it ia te d , an d

re ce p ti v e v o ca b u la ry

le ss

th an

1 5 w o rd s. P 6 an d P 8 : Bl ar g er

v o ca b u la ri es ^ an d us ed

sh o rt

p h ra se s; ra re ly

sp o k e

sp on ta n eo u sl y.

M cM

o rr o w

an d F ox x (1 9 8 6 )

& F o x x an d F aw

(1 9 9 0 )

n = 1 ; m al e; 2 1 y ea rs

N o st an d ar d iz ed

la n g u ag e

as se ss m en t. N o v er b al

in it ia ti on s; d id

n o t re sp o n d to

q u es ti o n s; n ea rl y al l

v er b al iz at io n s w er e ec h o la li c.

IQ o f 4 0 u si n g th e P P V T .

R ep ea te d st at em

en ts ;

im m ed ia te ec h o la li a

E x p er im

en t 1 : cu es -p au se -

po in t an d th en

p au se

o n ly

E x p er im

en t2

an d 3 :m

o d el in g

P o si ti v e

P o si ti v e at 5 7 m o n th s fo ll o w -u p

(F o x x an d F aw

1 9 9 0 )/ re su lt s

d id

n o t g en er al iz e to

n ew

q u es ti o n (c u es -p au se -p o in t

n o t u se d in

g en er al iz at io n

p ro b es )

C o nc lu si v e

N ie n ti m p an d

C o le

1 9 9 2

n = 3 ; 2 m al es

an d 1 fe m al e; 1 2 ,

1 2 .8 , 1 3 .4

y ea rs

N o st an d ar d iz ed

la n g u ag e

as se ss m en t. A ll d es cr ib ed

as v er b al b u t p ro m p t d ep en d en t.

P 1 : IQ

3 8 an d P 2 : IQ

3 2

(S ta n fo rd -B in et ). N o IQ

o n

fe m al e p ar ti ci p an t

R ep ea te d al l o r p ar t o f a

g re et in g ; im

m ed ia te

ec h o la li a

C o n st an t ti m e d el ay

an d

co n ti n g en t v er b al pr ai se

P o si ti v e

P o si ti v e fo r tw o o f th e

p ar ti ci p an ts d u ri n g

im m ed ia te w it h d ra w al o f

in te rv en ti o n /m

ix ed

w it h

g en er al iz at io n to

n o v el p ee rs

fo r tw o o f th re e p ar ti ci p an ts

S u g g es ti v e; p re -

ex p er im

en ta l d es ig n

(A B d es ig n ; o n e

p ar ti ci p an t) an d A B A

d es ig n a (t w o p ar ti ci p an ts )

P al y o et al . (1 97 9 )

n = 1 ; fe m al e; 5 .7

y ea rs b

V in el an d S o ci al Q u o ti en t 63 ;

A lp er n -B o ll IQ

61 ; A lp er n -

B o ll co m m u n ic at io n ag e

eq u iv al en t 2 ye ar s

Im m ed ia te ec h o la li a

P u n is h m en t fo r ec h o la li a,

po si ti v e re in fo rc em

en t

fo r ap p ro p ri at e re sp o n se s,

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Rev J Autism Dev Disord (2016) 3:82–91 87

animation of a father and child playing ball would appear. The participant was then observed in their classroom, and data were collected on appropriate and inappropriate verbal behavior. Results were mixed with some participants demonstrating improvement in echolalia and some demon- strating no improvement.

The remaining six studies employed a variety of behavior analytic interventions to treat echolalia. Behavioral compo- nents included error correction and differential reinforcement (n=2; Freeman et al. 1975; Palyo et al. 1979). differential reinforcement of lower rates (n=1; Handen et al. 1984). modeling and positive reinforcement (n=2; Karmali et al. 2005; Palyo et al. 1979). modeling (n=1; McMorrow and Foxx 1986). time delay and differential reinforcement (n=1; Nientimp and Cole 1992). and visual cues with differential reinforcement (n=1; Ganz et al. 2008). For example, Handen and colleagues (1984) implemented differential rein- forcement of lower rates (DRL) of behavior to decrease the echolalia of a 16-year-old male with ASD. The intervention occurred over an 18-month time frame and involved pro- viding the participant with tokens for engaging in lower rates of echolalia than a predetermined criterion. When the participant engaged in echolalia below the target rate, he exchanged the tokens for a tangible item from his reinforcement menu. The intervention was effective in reducing the participant’s engagement in echolalia; however, after intervention was removed, the partici- pant’s echolalia returned to baseline levels.

In another study, Ganz et al. (2008) taught two children with ASD who engaged in echolalia to engage in reciprocal social-communicative responses (e.g., compliments, ques- tions, and statements corresponding to the current activity). Responses were taught by providing visual scripts of the target response and systematically fading scripts over three phases. To reduce echolalia, a visual cue was presented which sig- naled to the participant that they should cease talking (i.e., a 3″×3″ line drawing of a face with a finger in front of the mouth indicating Bquiet^). This visual cue was introduced only if the participant engaged in echolalia. Results indicated clear decreases in echolalia.

Intervention Outcomes

The data from nine of the studies indicated that the results were positive for all participants (Foxx et al. 2004; Freeman et al. 1975; Ganz et al. 2008; Handen et al. 1984; Karmali et al. 2005; McMorrow and Foxx 1986; Nientimp and Cole 1992; Palyo et al. 1979; Valentino et al. 2012). Data from two of the studies suggested mixed results with some participants dem- onstrating improved behavior and some demonstrating no im- provement (Hetzroni and Tannous 2004; Laski et al. 1988). The first study( Laski et al. 1988) measured echolalia as an ancillary dependent variable and provided pre- and post-

treatment means for echolalia, with no differences noted in one setting (i.e., the break room). The second study (Hetzroni and Tannous 2004) utilized a multiple baseline de- sign across settings to evaluate the effects of their technology- based intervention on participant echolalia. However, de- creases in echolalia were not demonstrated for all three set- tings for any of the participants undercutting the experimental control of the multiple baseline design. The two studies with mixed results represented 8 of the 25 participants.

Maintenance and Generalization

Five of studies assessed maintenance of behavior change (Foxx et al. 2004; Handen et al. 1984; Nientimp and Cole 1992; Palyo et al. 1979; Valentino et al. 2012). and one study was published as a long-term follow-up to the McMorrow and Foxx study (1986; Foxx and Faw 1990). The timing of the collection of maintenance data ranged from immediately fol- lowing the conclusion of the intervention (Foxx et al. 2004; McMorrow and Foxx 1986; Nientimp and Cole 1992) to 57 months after the intervention (Foxx and Faw 1990). All studies reported that echolalia levels at maintenance were be- low baseline levels. Seven studies assessed stimulus general- ization (Foxx et al. 2004; Hetzroni and Tannous 2004; Karmali et al. 2005; McMorrow and Foxx 1986; Nientimp and Cole 1992; Palyo et al. 1979; Valentino et al. 2012) in- cluding generalization across settings, people, materials, and different preceding utterances (questions). Four studies report- ed positive results for generalization (Foxx et al. 2004; Karmali et al. 2005; Palyo et al. 1979; Valentino et al. 2012). Two studies reported that generalization occurred for some participants but not for all (Hetzroni and Tannous 2004; Nientimp and Cole 1992). One study found that results did not generalize for the participants (McMorrow and Foxx 1986).

Certainty of Evidence

Six of the studies were categorized as offering a conclusive level of evidence with positive results, sufficient research de- sign and IOA data, and detailed procedural descriptions (Foxx et al. 2004; Ganz et al. 2008; Handen et al. 1984; Karmali et al. 2005; McMorrow and Foxx 1986; Valentino et al. 2012). Five studies were categorized as suggestive (Freeman et al. 1975; Hetzroni and Tannous 2004; Laski et al. 1988; Nientimp and Cole 1992; Palyo et al. 1979). Of the five studies, three did not demonstrate experimental control (Freeman et al. 1975; Nientimp and Cole 1992; Palyo et al. 1979). two studies had mixed results (Hetzroni and Tannous 2004; Laski et al. 1988). and one study did not assess IOA (Freeman et al. 1975). None of the studies was classified at the preponderant level of evidence.

88 Rev J Autism Dev Disord (2016) 3:82–91

Discussion

The purpose of this review was to identify promising practices for decreasing echolalia in individuals with ASD. This sys- tematic literature review synthesized 11 studies which employed a variety of behavioral interventions. Of the 11 studies, nine reported positive results for 17 participants, and two of the studies reported mixed results for two participants. When examining the quality of the literature base, six of the 11 studies were classified as providing conclusive evidence. Ultimately, the findings of this review indicate that the litera- ture base cannot conclusively support any one approach for the treatment of echolalia in individuals with ASD.

Implications for Research

The first purpose of this review was to update the previous review by Kavon and McLaughlin (1995) to identify effective interventions for the treatment of echolalia in individuals with ASD. Although no single intervention procedure or package met any of the commonly used criteria for classification as a well-established or evidence-based practice (e.g., Chambless and Holland 1998; Odom and Wolery 2003). a number of themes emerged. First, of the six studies classified as conclu- sive, all contained behavioral analytic intervention compo- nents (e.g., programmed reinforcement contingencies) sug- gesting support for operant-based treatments in the reduction of echolalia for individuals with ASD. When considering im- mediate echolalia, the cues-pause-point intervention was iden- tified as effective in three studies (Foxx et al. 2004; McMorrow and Foxx 1986; Valentino et al. 2012). This con- clusion supports the previous descriptive review identifying cues-pause-point as a potentially effective intervention for im- mediate echolalia. Of note, only two studies have evaluated the effects of cues-pause-point since the previous review (Kavon and McLaughlin 1995) highlighting the need for more research in this area.

Conclusive studies evaluating interventions for delayed echolalia also utilized behavioral analytic components. Three interventions, DRL of behavior (Handen et al. 1984). script training plus visual cues (Ganz et al. 2008). and tact modeling plus positive reinforcement for appropriate responses (Karmali et al. 2005). were all identified as effective for de- layed echolalia. Of particular interest is the study by Ganz et al. (2008) which utilized visual cues to signal to the partic- ipant that they should cease talking. In addition, visual scripts were provided to prompt the target response. Although not inclusive of all the elements of cues-pause-point, Ganz et al. did implement a cue to remain silent and a point to prompt the correct answer. In addition, the DRL of behavior intervention by Handen et al. (1984) utilized differential reinforcement which was a contingency in effect in the cues-pause-point interventions. Overall, the research combined provides

preliminary support for operant-based behavioral interven- tions in general and cues-pause-point in particular.

As the interventions evaluated in these studies align with the operant conditioning paradigm, it is alarming that none of the studies assessed the operant function of echolalia. In other forms of challenging behavior (e.g., aggression, property de- struction, and self-injury), identifying the function of the be- havior via a functional analysis procedure (Iwata et al. 1982/1994; Lydon et al. 2012) is associated with better re- sponse to treatment (e.g., Didden et al. 2006). Of particular concern is that echolalia may serve various social as well as non-social communicative functions for individuals with ASD (Goren et al. 1977; Healy and Leader 2011). If echolalia is reduced during treatment but a functionally equivalent re- placement behavior is not taught, this could limit the mainte- nance and generalization of the behavioral change (Schreibman and Carr 1978). In addition, as there is diver- gence within the field regarding whether echolalia is nonfunc- tional (e.g., Lovaas, Schreibman, and Koegel 1974). a neces- sary part of developing functional communication (e.g., Roberts 2014). or serves a social function (e.g., Prizant and Duncan 1981). identifying the function of echolalia would help guide future research and practice.

One potential strength of this literature base is the assess- ment of maintenance of behavioral change following cessa- tion of the intervention phase. Of the six articles that assessed the maintenance of behavioral change, all reported positive results indicating levels of echolalia maintained below base- line levels during follow-up sessions. However, although sev- en of the studies did evaluate the generalization of the results across stimuli, people, and settings, results were mixed with only four reporting successful generalization. Future re- searchers might consider evaluating interventions to promote sustained behavioral change (e.g., fading, multiple exemplar training; Valentino et al. 2012).

Implications for Practice

A second purpose of this review was to offer guidance to practitioners interested in the use of evidence-based treatments for individuals with ASD. Given the limited number of con- clusive studies, recommendations as to an evidence-based in- tervention for decreasing echolalia cannot be drawn. However, the results of this review did suggest that cues- pause-point, which was investigated by three different studies with positive results and conclusive levels of evidence, is po- tentially effective for immediate echolalia (Foxx et al. 2004; McMorrow and Foxx 1986; Valentino et al. 2012). Although these results support the conclusions from previous research that cues-pause-point is potentially effective (Kavon and McLaughlin 1995). practitioners should use caution when implementing this intervention due to the limited number of studies. In particular, practitioners should closely monitor the

Rev J Autism Dev Disord (2016) 3:82–91 89

intervention effects through ongoing progress monitoring and rely on objective data to evaluate the effectiveness of the intervention.

Limitations of this Review

There are a couple of limitations of this review to consider. First, the definition of echolalia used by the authors was intended to distinguish echolalia from vocal stereotypy. However, it was difficult to identify a definition of echolalia that was accepted throughout the literature base. In addition, as none of the studies reported the function of the target echo- lalia behavior, it is uncertain whether echolalia was isolated from other forms of vocal stereotypy. A second limitation is the age of the literature base. Of the 11 studies reviewed, six of the studies were published over 20 years ago. As research quality indicators have evolved dramatically in the past 20 years, the age of this literature base may have been a factor in the conclusiveness of the evidence. In addition, since the review by Kavon and McLaughlin (1995). only five additional studies have been published on this topic. Therefore, there is a need to update and expand this literature base to promote the use of evidence-based practices in the treatment of echolalia for individuals with ASD. A third limitation is the procedures used to code intervention outcomes as applied to this literature base. Study results were rated as Bpositive,^ mixed, or Bnegative^ with mixed indicating that some but not all partic- ipants demonstrated improvements in behavior. Five of the 11 included studies contained only one subject with ASD, there- fore restricting the rating of the outcomes to either negative or positive. Although restricted codes could have negatively im- pacted intervention outcome ratings, all of the studies with one subject received positive ratings. However, the limited num- ber of subject limits the external validity of the conclusions. A fourth limitation is the absence of large-scale randomized con- trol trials in the resulting literature base. Although this might be a reflection of the exceptionality of the population, the exclusive use of single-subject designs limits the external va- lidity of this literature base. A fifth limitation of this review is the focus on individuals with ASD. Future researchers might consider expanding to include other developmental disabil- ities to ensure a more comprehensive review. Such an analysis might allow for distinctions relevant to the presentation and/or treatment of echolalia in children with ASD relative to indi- viduals with other forms of disability.

Compliance with Ethical Standards

Funding The authors report no funding for this manuscript.

Conflict of Interest The authors report no conflicts of interest.

Ethical Approval This article does not contain any studies with human participants performed by any of the authors.

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