Apraxia TX

AJSLP
Review Article
A Systematic Review of Treatment

Outcomes for Children With Childhood
Apraxia of Speech
Elizabeth Murray,a Patricia McCabe,a and Kirrie J. Ballarda
Purpose: To present a systematic review of single-case

experimental treatment studies for childhood apraxia of
speech (CAS).
Method: A search of 9 databases was used to find
peer-reviewed treatment articles from 1970 to 2012 of all
levels of evidence with published communication outcomes
for children with CAS. Improvement rate differences (IRDs)
were calculated for articles with replicated (n > 1), statistically
compared treatment and generalization evidence.
Results: Forty-two articles representing Phase I and II
single-case experimental designs (SCEDs; n = 23) or case
series or description studies (n = 19) were analyzed.
Six articles showed high CAS diagnosis confidence. Of the
13 approaches within the 23 SCED articles, treatments were
primarily for speech motor skills (n = 6), linguistic skills (n = 5),
or augmentative and alternative communication (n = 2). Most

participants responded positively to treatment, but only 7 of
13 approaches in SCED studies reported maintenance and/or
generalization of treatment effects. Three approaches had
preponderant evidence (Smith, 1981). IRD effect sizes were
calculated for Integral Stimulation/Dynamic Temporal and
Tactile Cueing, Rapid Syllable Transition Treatment, and
Integrated Phonological Awareness Intervention.
Conclusions: At least 3 treatments have sufficient evidence
for Phase III trials and interim clinical practice. In the future,
efficacy needs to be established via maintenance and
generalization measures.
especially in the realization of lexical or phrasal stress (ASHA,

2007, p. 4).
Although impaired movement planning and programming are considered to underlie CAS, there are also reports of
disrupted development of speech perception, language, and
phonology (including phonological awareness) in children
with CAS (Groenen, Maassen, Crul, & Thoonen, 1996; Lewis
et al., 2004; Maassen, Groenen, & Crul, 2003). It is unclear
whether these are primary deficits or flow-on effects from
CAS, comorbid impairments, or perhaps compensatory behaviors, as children with CAS develop their linguistic, phonological, and motor skills concurrently (Alcock, Passingham,
Watkins, & Vargha-Khadem, 2000; Marion, Sussman, &
Marquardt, 1993; Ozanne, 2005). Children with CAS can
therefore present with a range of difficulties requiring therapy
from speech-language pathologists (SLPs; Royal College of
Speech and Language Therapists, 2011).
The long-term functioning of people with CAS is
largely unreported. The available longitudinal studies suggest
that CAS is a persistent disorder that requires therapy (Hall,
Jordan, & Robin, 1993; Jacks, Marquardt, & Davis, 2006;
Stackhouse & Snowling, 1992). Children with CAS, like others
with persistent speech sound disorder, are also at risk for
hildhood apraxia of speech (CAS) is a developmental disorder of speech motor planning and/or programming (American Speech-Language-Hearing
Association [ASHA], 2007). It is also known as developmental verbal dyspraxia in the United Kingdom (excluding
being the result of any known neurological disorder) and
has previously been called developmental apraxia of speech
and dyspraxia. CAS causes reduced speech intelligibility
because of a hypothesized impairment in the transformation
of an abstract phonological code into motor speech commands (Terband, Maassen, Guenther, & Brumberg, 2009,
p. 1598). Such impairment leads to the current consensusbased core CAS features of (a) inconsistent errors on consonants and vowels in repeated productions of syllables or
words, (b) lengthened and disrupted coarticulatory transitions
between sounds and syllables, and (c) inappropriate prosody,
University of Sydney, New South Wales, Australia
Correspondence to Elizabeth Murray:

Elizabeth.murray@sydney.edu.au
Editor: Carol Scheffner Hammer
Associate Editor: Ken Bleile
Received March 27, 2013
Revision received July 7, 2013
Accepted December 1, 2013
DOI: 10.1044/2014_AJSLP-13-0035
486
Key Words: dyspraxia, intervention, efficacy,

methodological rigor, generalization
Disclosure: The authors have declared that no competing interests existed at the
time of publication.
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literacy, academic, social, and vocational difficulties (e.g.,

Lewis et al., 2004; Moriarty & Gillon, 2006).
A Cochrane systematic review, a subsequent journal
article, and a treatment review have reported no published
randomized controlled trials (RCTs) or nonrandomized
controlled trials (NRCTs) for any intervention for CAS
(Morgan & Vogel, 2008, 2009; Watts, 2009). Despite this
lack of high-level evidence, many published articles on the
treatment of CAS could facilitate practice and could help
identify potential lines of further research. Narrative reviews
have identified a range of treatment methods for children
reported to have CAS, likely reflecting the diversity of symptoms seen in these children and potentially the research and
clinical interests of the authors (ASHA, 2007; Strand &
Skinder, 1999). They encompass motor treatments (including
electropalatography), linguistic approaches, augmentative
and alternative communication (AAC), or some combination thereof (ASHA, 2007; Gillon & Moriarty, 2007; Hall,
2000; Morgan & Vogel, 2008). Few of these lower level
treatment studies have been examined rigorously, as they
were excluded on the basis of quality in previous systematic
reviews (Morgan & Vogel, 2008; Watts, 2009). Provision
of recommendations regarding which treatments have supportive evidence has therefore not been possible (ASHA,
2007; Morgan & Vogel, 2008; Pannbacker, 1988). This article
presents a systematic review of all levels of evidence that
may be critical to inform clinical practice until high-level
evidence becomes available.
Two primary challenges face a systematic review of
intervention for CAS. The first challenge is in identifying the
rigor of each study in terms of research phase (Robey, 2004),
the research design, the level of evidence generated (ASHA,
2004; Perdices et al., 2006), as well as the level of certainty that
the effects reported for a given treatment approach are real
(Smith, 1981). Treatment research often follows a developmental pathway that is associated with increasing research
rigor and different research questions. Robey (2004) defined
five phases in a research program. Phase I and Phase II
studies represent pilot or feasibility studies seeking to determine whether effects justify more rigorous study. These can
generate Level III or Level IIb evidence (ASHA, 2004;
Perdices et al., 2006). Level III evidence constitutes quasiexperimental group (case series) or single case reports with
pre- to posttreatment measurement and no within-subject
comparison or control conditions. Level IIb evidence comes
from more rigorous single-case experimental designs (SCEDs)
that systematically apply and withdraw treatment and establish control using a stable baseline phase or limited change
in control conditions (Byiers, Reichle, & Symons, 2012;
Olswang & Bain, 1994; Perdices & Tate, 2009).
The second challenge is that there is not yet a validated
assessment tool for diagnosing CAS. Thus, before evaluation
of the treatment in any intervention study, the descriptions
of participants must be scrutinized to determine the level of
confidence in the authors diagnosis. This involves determining to what extent the participants are described as meeting
the three consensus-based core features of CAS listed earlier
(ASHA, 2007).
Smiths (1981) level of certainty hierarchy considers the

research design and the possible effects of the intervention
to provide an overall judgment on how likely the results are to
be true. For the CAS literature, this can also be extended
to include confidence in diagnosis. SCEDs can show early
evidence for an intervention, with results from such studies
being classed as suggestive or possibly true. However,
statistically compared outcomes from SCEDs with confident
CAS diagnoses, replication (n > 1), and evidence of both
treatment effects and generalization of treatment effects could
be considered preponderant evidence or probably true.
SCED designs are usually designated as Phase II and Level IIb
evidence (but see Hegde, 2007; Kearns & de Riesthal, 2013).
Currently, Phase III studies (Robey, 2004) are typically RCTs
and NRCTs. These generate Level IIa evidence using groups
of participants to reduce bias and to eliminate individual
variance as a factor in treatment success. It is only through
meta-analyses and systematic reviews of several Phase III
studies of a given treatment approach, coupled with Phase IV
effectiveness studies in real-world clinical situations, that
results can be defined as conclusive or undoubtedly true
(Smith, 1981). As all studies of CAS treatments to date are
classified as Phase I and Phase II, the goal of this review is
to identify treatment approaches with suggestive or preponderant evidence.
Central to this review is treatment efficacy. Efficacy
considers clinical cause-and-effect relationships between
the provision of intervention and change in participant behavior (e.g., McReynolds & Kearns, 1983; Olswang & Bain,
2013). Demonstration of efficacy extends beyond treatment
effects, requiring assessment of maintenance and generalization of treatment effects that signify instrumental change.
Response generalization evaluates a childs performance on
untrained items that are somehow related to trained items,
to determine whether more widespread change is occurring
(Olswang & Bain, 1994). Stimulus generalization assesses
performance on untrained materials, people, or settings/
environments (Olswang & Bain, 1994). Such change is necessary to meet the overall goals of an intervention.
Aims
This systematic review evaluated studies of intervention, published between 1970 and October 2012, that state
an intention to treat children with CAS. The aims fall into
four broad areas:
1.
Study quality: to describe for each identified study the

research phase, the level of evidence, and the level of
confidence in CAS diagnosis;
2.
Treatment procedures: to define the behavioral goals

and structure of treatment (e.g., intensity and dosage
according to Warren, Fey, & Yoder, 2007) for each
SCED study (at/above Level IIb evidence) and to
group similar treatment types to facilitate treatment
outcome analysis;
3.
Treatment outcomes: to examine reported treatment,

maintenance, and generalization outcomes; and
Murray et al.: Systematic Review of Treatment Outcomes for CAS

487
literacy, academic, social, and vocational difficulties (e.g.,

Lewis et al., 2004; Moriarty & Gillon, 2006).
A Cochrane systematic review, a subsequent journal
article, and a treatment review have reported no published
randomized controlled trials (RCTs) or nonrandomized
controlled trials (NRCTs) for any intervention for CAS
(Morgan & Vogel, 2008, 2009; Watts, 2009). Despite this
lack of high-level evidence, many published articles on the
treatment of CAS could facilitate practice and could help
identify potential lines of further research. Narrative reviews
have identified a range of treatment methods for children
reported to have CAS, likely reflecting the diversity of symptoms seen in these children and potentially the research and
clinical interests of the authors (ASHA, 2007; Strand &
Skinder, 1999). They encompass motor treatments (including
electropalatography), linguistic approaches, augmentative
and alternative communication (AAC), or some combination thereof (ASHA, 2007; Gillon & Moriarty, 2007; Hall,
2000; Morgan & Vogel, 2008). Few of these lower level
treatment studies have been examined rigorously, as they
were excluded on the basis of quality in previous systematic
reviews (Morgan & Vogel, 2008; Watts, 2009). Provision
of recommendations regarding which treatments have supportive evidence has therefore not been possible (ASHA,
2007; Morgan & Vogel, 2008; Pannbacker, 1988). This article
presents a systematic review of all levels of evidence that
may be critical to inform clinical practice until high-level
evidence becomes available.
Two primary challenges face a systematic review of
intervention for CAS. The first challenge is in identifying the
rigor of each study in terms of research phase (Robey, 2004),
the research design, the level of evidence generated (ASHA,
2004; Perdices et al., 2006), as well as the level of certainty that
the effects reported for a given treatment approach are real
(Smith, 1981). Treatment research often follows a developmental pathway that is associated with increasing research
rigor and different research questions. Robey (2004) defined
five phases in a research program. Phase I and Phase II
studies represent pilot or feasibility studies seeking to determine whether effects justify more rigorous study. These can
generate Level III or Level IIb evidence (ASHA, 2004;
Perdices et al., 2006). Level III evidence constitutes quasiexperimental group (case series) or single case reports with
pre- to posttreatment measurement and no within-subject
comparison or control conditions. Level IIb evidence comes
from more rigorous single-case experimental designs (SCEDs)
that systematically apply and withdraw treatment and establish control using a stable baseline phase or limited change
in control conditions (Byiers, Reichle, & Symons, 2012;
Olswang & Bain, 1994; Perdices & Tate, 2009).
The second challenge is that there is not yet a validated
assessment tool for diagnosing CAS. Thus, before evaluation
of the treatment in any intervention study, the descriptions
of participants must be scrutinized to determine the level of
confidence in the authors diagnosis. This involves determining to what extent the participants are described as meeting
the three consensus-based core features of CAS listed earlier
(ASHA, 2007).
Smiths (1981) level of certainty hierarchy considers the

research design and the possible effects of the intervention
to provide an overall judgment on how likely the results are to
be true. For the CAS literature, this can also be extended
to include confidence in diagnosis. SCEDs can show early
evidence for an intervention, with results from such studies
being classed as suggestive or possibly true. However,
statistically compared outcomes from SCEDs with confident
CAS diagnoses, replication (n > 1), and evidence of both
treatment effects and generalization of treatment effects could
be considered preponderant evidence or probably true.
SCED designs are usually designated as Phase II and Level IIb
evidence (but see Hegde, 2007; Kearns & de Riesthal, 2013).
Currently, Phase III studies (Robey, 2004) are typically RCTs
and NRCTs. These generate Level IIa evidence using groups
of participants to reduce bias and to eliminate individual
variance as a factor in treatment success. It is only through
meta-analyses and systematic reviews of several Phase III
studies of a given treatment approach, coupled with Phase IV
effectiveness studies in real-world clinical situations, that
results can be defined as conclusive or undoubtedly true
(Smith, 1981). As all studies of CAS treatments to date are
classified as Phase I and Phase II, the goal of this review is
to identify treatment approaches with suggestive or preponderant evidence.
Central to this review is treatment efficacy. Efficacy
considers clinical cause-and-effect relationships between
the provision of intervention and change in participant behavior (e.g., McReynolds & Kearns, 1983; Olswang & Bain,
2013). Demonstration of efficacy extends beyond treatment
effects, requiring assessment of maintenance and generalization of treatment effects that signify instrumental change.
Response generalization evaluates a childs performance on
untrained items that are somehow related to trained items,
to determine whether more widespread change is occurring
(Olswang & Bain, 1994). Stimulus generalization assesses
performance on untrained materials, people, or settings/
environments (Olswang & Bain, 1994). Such change is necessary to meet the overall goals of an intervention.
Aims
This systematic review evaluated studies of intervention, published between 1970 and October 2012, that state
an intention to treat children with CAS. The aims fall into
four broad areas:
1.
Study quality: to describe for each identified study the

research phase, the level of evidence, and the level of
confidence in CAS diagnosis;
2.
Treatment procedures: to define the behavioral goals

and structure of treatment (e.g., intensity and dosage
according to Warren, Fey, & Yoder, 2007) for each
SCED study (at/above Level IIb evidence) and to
group similar treatment types to facilitate treatment
outcome analysis;
3.
Treatment outcomes: to examine reported treatment,

maintenance, and generalization outcomes; and

487
4.
Certainty of evidence: to determine the level of certainty for each treatment approach (Smith, 1981) and
to determine the effect size for any intervention approach classed as having preponderant evidence.
Method
Systematic Search Strategy
The search strategy used follows Preferred Reporting
Items for Systematic Reviews and Meta-Analyses (PRISMA)
search guidelines (Moher, Liberati, Tetzlaff, & Altman,
2009). The flow diagram of study selection is presented in
Figure 1.
Identification
Nine databases related to speech-language pathology
were comprehensively searched for peer-reviewed journal
articles. These were Allied and Complementary Medicine,
Cumulative Index to Nursing and Allied Health Literature,
Evidence-Based Medicine ReviewsCochrane Database
of Systematic Reviews, Education Resources Information
Center, Linguistic Language Behavior Abstracts, Medline,
PsycINFO, Scopus, and speechBITE. Specific search terms
varied on the basis of each database catalogue of terms in its
search directories. Key words used were as follows: apraxia
or dyspraxia or childhood apraxia of speech and
child* or develop* and motor speech therapy or
interven* or treat* or speech therapy/pathology or
efficacy or evaluation or effect and speech or communication or language or articulation impairments
or speech impairments or speech disorders or speech
intelligibility or prosody. A total of 1,301 studies were
identified from database searches.
Screening
All references were exported to EndNote X5 (Thomson
Reuters, 2011), where duplicates were removed. References
were also screened to ensure that authors stated an intention to
treat children with CAS (using synonyms; e.g., developmental
verbal dyspraxia). Thus, references were searched by title,
abstract, and key words in EndNote X5 and were excluded
if treatment articles involved other diagnoses without reference to CAS: cerebral palsy, dysarthria, cleft palate,
swallowing, ataxia, cochlear implants, deaf,
stutter, fluency, acquired, Down syndrome, autism,
phonolog*, and gait apraxia. All references that related
to assessment, diagnosis, or description/exploration of symptoms were excluded. Of the intervention articles that remained,
the intervention names and authors were searched again in
all the above databases as well as Google Scholar to ensure
that all relevant articles were found. The reference lists of
all review articles obtained were also searched to find any
additional articles. Screening removed 913 articles, leaving
119 to be assessed for eligibility.
Eligibility
Copies of articles were obtained and assessed against
the final inclusion criteria before being reviewed. These
criteria were as follows: (a) peer-reviewed articles published

between 1970 and October 2012; (b) in English (to allow
analysis by monolingual English speaking authors); (c) treating at least one child/adolescent under 18 years of age with
CAS or suspected CAS; and (d) reporting quantitative participant data outcomes focused on speech (i.e., articulation,
phonology, prosody, intelligibility, rate), communication (i.e.,
pragmatics, social communication, AAC use), or language
(i.e., phonology, phonological awareness, grammar, morphology, receptive or expressive language, reading, spelling
or writing). There was no exclusion on the basis of the type
of therapy provided or who provided it. All named and
unnamed interventions were included, as were all levels of
evidence, except for systematic reviews, as they do not contain individual participant data (ASHA, 2004). This yielded
42 articles for review. Intrarater reliability (first author) for
inclusion of these studies was 96% (n = 1,032) with >2 months
between assessments. Interrater reliability with an independent rater was 91% (see Supplemental Appendix 1 in the
online supplemental materials for the list of excluded articles
and reason for exclusion). Raters were not blinded to article
title or authors, and raters did not review their own publications. Excluded articles were not further analyzed.
Finally, an additional search was undertaken for other
documents, such as published treatment manuals and theoretical or opinion articles on the individual treatment approaches, to confirm the type of behavior(s) targeted (e.g.,
speech motor or phonological), cues, and stimuli used. This
search used reference lists of already identified articles as well
as database and Google searches using intervention names
and key authors as search terms.
Data Analysis
Review of studies for Aims 1, 3, and 4 was based on
information provided in each article (e.g., Moseley, Herbert,
Maher, Sherrington, & Elkins, 2008). In one case, only group
data were published, and we contacted an author (Brigid
McNeill) for individual data (from McNeill, Gillon, & Dodd,
2009a). Addressing Aim 2 at times required reference to treatment manuals and other publications describing a treatments theoretical framework.
Aim 1: Study Quality
Each study was assigned to a phase of research (Robey,
2004), from Phase I to Phase V. The experimental design
and level of evidence were defined on the basis of published
guidelines (ASHA, 2004; Perdices et al., 2006). In addition, confidence in CAS diagnosis was assessed as detailed
below.
Confidence in CAS diagnosis. A 5-point rating scale
was used to rate confidence in CAS diagnosis (see Table 1;
Wambaugh, Duffy, McNeil, Robin, & Rogers, 2006). This was
based on description of primary versus nondiscriminative
features (McCabe, Rosenthal, & McLeod, 1998; see the
online supplemental materials, Supplemental Table 1). Primary features were the three consensus-based features listed
in ASHAs (2007) technical report, hypothesized to represent
488 American Journal of Speech-Language Pathology Vol. 23 486504 August 2014

Figure 1. Flow diagram of study selection (adapted from Preferred Reporting Items for Systematic Reviews and Meta-Analyses [PRISMA]; Moher
et al., 2009). CAS = childhood apraxia of speech; Ax = assessment; Dx = diagnosis; RCT = randomized controlled trial; NRCT = nonrandomized
controlled trial; SCED = single-case experimental design; AAC = augmentative and alternative communication.
impaired speech motor planning and/or programming. Nondiscriminative features were those shared with other disorders, such as poor intelligibility, slow progress, or delayed
language (ASHA, 2007; McCabe et al., 1998). Clear cases
of comorbid disorders were also noted, such as receptive
language impairment or dysarthria. On the basis of this
analysis, participants were classified either as CAS only or as
CAS+, in which CAS was the primary diagnosis, but other

disorders were present. A rating of 1 indicated high confidence in CAS diagnosis, and a rating of 5 indicated no
confidence. Intrarater reliability (first author) on four judgments (presence of each of the three primary features and
CAS diagnosis) for 83/83 children studied was 94%. Interrater reliability between the first and second authors for a

489
Table 1. Five-point rating scale of confidence in diagnosis of CAS (adapted from the Academy of Neurologic Communication Disorders and
Sciences guidelines; Wambaugh et al., 2006).
Level
Level 1
Level 2
Level 3
Level 4
Level 5
Primary characteristics
All the primary characteristics were
described as follows:
Inconsistency and
Lengthened and disrupted
coarticulatory transitions between
sounds and syllables and
Inappropriate prosody.
All the primary characteristics were
described as follows:
Inconsistency and
sounds and syllables and
Two of the three primary characteristics
were described:
Inconsistency and/or
sounds and syllables and/or
Only one of the three primary

characteristics was reported, or
incomplete/inadequate description of
the primary characteristics of CAS
was provided.
Diagnosis of CAS was reported or
implied, but no primary characteristics
were described.
Nondiscriminative characteristics
Comorbidity
CAS was the primary diagnosis

described. Any characteristics that
were attributable to other disorders
may have been described but
were not used to diagnose CAS.
CAS without another comorbid disorder

was reported (excluding expressive
language delay).
CAS was the primary diagnosis

described. Other characteristics
attributable to other disorders (e.g.,
dysarthria) were described and may
have been used to diagnose CAS.
This includes the following:

CAS without another comorbid
disorder (excluding expressive
language delay) or
Clear cases of comorbid CAS,
in which CAS was the primary
diagnosis.
This includes the following:
CAS without another comorbid
disorder or
Clear cases of comorbid CAS, in
which CAS was the primary
diagnosis or
Cases of CAS in which another
comorbid disorder had the same
severity (e.g., language delay,
dysarthria).
Unclear whether CAS was the primary
diagnosis.
CAS was described. Other

characteristics that were attributable
to other disorders (e.g., dysarthria)
were described and may have been
used to diagnose CAS.
Other characteristics that were

attributable to other disorders may
have been described, and it is unclear
whether these were used to diagnose
CAS.
Unclear whether CAS diagnosis was
likely and/or whether CAS was the
primary diagnosis.
random 33/83 children was 91%. Discrepancies were resolved

by consensus.
Exclusions. Articles that lacked experimental control
(Level III evidence; n = 19) were excluded, as they could
not be used to determine treatment outcomes. The remaining 23 Level IIb articles were analyzed to address Aims 2, 3,
and 4. No articles were excluded because of confidence in
CAS diagnosis; however, confidence in CAS diagnosis was
a factor in determining certainty of evidencesee Aim 4
below.
Aim 2: Treatment Procedures
Articles designed with adequate experimental control
(see Aim 1 above) were analyzed descriptively regarding the
nature of the treatment. Using the stated treatment goals,
selected stimuli, and specific cueing strategies reported, treatments were categorized as primarily (a) motor, (b) linguistic/
phonological (including literacy), or (c) AAC (ASHA,
2007; Gillon & Moriarty, 2007; Hall, 2000; Martikainen &
Korpilahti, 2011). The structure of treatment delivery was
also determined (Warren et al., 2007), including dose (trials
per session), dose frequency (number of times a dose is
provided over days or weeks), and total intervention time
(number of sessions). When reported, home practice and
service delivery model were documented.
Aim 3: Treatment Outcomes

Reported treatment, maintenance, and generalization
outcomes for each intervention were analyzed for (a) number
of participants with a treatment gain (change immediately
after treatment compared with baseline); (b) assessment
measures and statistics used in determining treatment effects;
(c) maintenance of treatment gains at least 2 weeks posttreatment, from report or by comparing treatment data with
performance in maintenance probes; (d) response generalization data, when statistical analysis was used; and (e) stimulus
generalization data.
Aim 4: Certainty of Evidence
Smiths (1981) three levels of certainty were applied, on
the basis of design (i.e., level of evidence, research design,
confidence in CAS diagnosis, and statistical comparison) and
possible effects of the intervention/outcomes. A treatment
approach was categorized as having preponderant evidence
when it showed Level IIb or better evidence (SCEDs), replicated cases, diagnostic confidence ratings of 13, statistically
significant treatment and generalization effects (or at least
moderate effect sizes), and clear maintenance of treatment
gains at least 2 weeks posttreatment. Any Level IIb or better
evidence that did not meet all the above criteria received the
lowest rating of suggestive evidence.

Other comorbid disorders may be

present.
For studies with preponderant evidence, effect sizes

were calculated using improvement rate difference (IRD
[also known as risk difference]; Parker, Vannest, & Brown,
2009), a valid and reliable tool for SCEDs used frequently in
medical research. This method utilizes visual analysis and has
reduced assumptions in comparison with other effect size
calculations allowing use over a range of SCED designs. It is
more discriminative to change than other nonoverlap techniques (e.g., percentage of nonoverlapping data) and is at
least moderately correlated with commonly used effect sizes
(Parker et al., 2009). An IRD is the difference between the
improvement rates of the treatment and baseline/withdrawal/
maintenance phases. The improvement rate was calculated
for each phase by determining the number of improved
points in each phase (those exceeding the data points of the
adjacent phase) divided by all the data points of the phase
(Parker et al., 2009). Thus, an IRD eliminates overlap across
phases in its determination of effect size.
IRDs were determined for treatment effects for each
participant and each behavior within an article. The IRDs
were then averaged across participants for the treated behavior(s) to determine an omnibus IRD for each article, as per
Parker et al. (2009), with 95% confidence intervals determined
using WinPEPI (Abramson, 2011). Effects for each article
were averaged again to determine an omnibus IRD for a given
treatment approach.
IRDs were also calculated for generalization effects
for each participant and each behavior/condition within an
article. No further omnibus IRDs were determined for generalization because of the various and often heterogeneous
measures used.
Results
Aim 1: Study Quality
The 42 studies that met the inclusion criteria were
classified as Phase I or Phase II. Of these, 23 represented
Level IIb evidence (SCEDs), and 19 represented Level III
evidence (one quasi-experimental case series and 18 case
reports or descriptions; see Figure 1). There was a shift toward higher quality single case studies over time, with six
Level IIb and 12 Level III articles prior to 2006, and with
17 Level IIb and five Level III articles from 2006 to 2012.
No RCT or NRCT designs have been published to date.
Overall, there were 83 participants across the 42 single
case studies. Within the 23 SCED articles, 32 participants
were reported to have CAS, and 19 participants were reported
to have comorbid CAS (CAS+). Of the 23 articles, four articles (17.4%) received a Level 1 rating for high confidence
in diagnosis, two articles (8.7%) received a Level 2 rating for
clear cases of CAS with comorbid disorders, seven articles
(30.4%) received a Level 3 rating, seven articles (30.4%) received a Level 4 rating, and three articles (13.0%) received a
Level 5 rating for no confidence (see Table 2).
Within the 19 case series and description articles,
25 participants were reported to have CAS, and eight participants were reported to have comorbid CAS (CAS+).
Of the 19 articles, three articles (15.8%) received a Level 1

rating for high confidence in diagnosis, zero articles (0.0%)
received a Level 2 rating, five articles (26.3%) received a
Level 3 rating, five articles (26.3%) received a Level 4 rating,
and six articles (31.5%) received a Level 5 rating for no confidence (see the online supplemental materials, Supplemental Table 2). The 19 Level III articles were not analyzed
further because of lack of experimental control.
Aim 2: Treatment Procedures

The first section details the classification of treatments
on the basis of their approach. The second section presents
results on service delivery models used.
Classification of Treatment Goals and Approaches
Eleven of 23 SCED studies were classified as primarily
motor approaches (see Table 3). They included primary
measures for accuracy of articulation and/or prosody. Two
studies included secondary measures of speech intelligibility
or comprehensibility (Strand & Debertine, 2000; Strand,
Stoeckel, & Baas, 2006). All utilized articulatory placement
and imitation cues as well as multimodal cues (e.g., kinesthetic/
touch cues, manipulating speech rate and timing, picture
or orthographic stimuli). The majority (90%) made explicit
reference to incorporating principles of motor learning (PML;
see Maas et al., 2008, for a review).
Ten studies were classified as primarily linguistic approaches. Of these, six studies reported primary measures of
speech sound production (phonological processes or stimulability), phonological awareness accuracy, and spoken language utterance length, which also used some motor cueing
(Iuzzini & Forrest, 2010; Krauss & Galloway, 1982; McNeill,
Gillon, & Dodd, 2009a, 2009b, 2010; Moriarty & Gillon, 2006).
The other four studies targeted expressive language skills
(e.g., multisymbol messages or elaborated phrase structures)
using AAC systems in children with previously established
AAC use (Binger, Kent-Walsh, Berens, Del Campo, & Rivera,
2008; Binger & Light, 2007; Binger, Maguire-Marshall, &
Kent-Walsh, 2011; Harris, Doyle, & Haaf, 1996).
Two studies were classified as AAC, measuring communicative effectiveness in children with severe comorbid
CAS (CAS+), by introducing AAC systems (Bornman, Alant,
& Meiring, 2001; Culp, 1989). These children had reportedly
shown slow or minimal progress in speech production attempts and/or used AAC to alleviate frustration and behavioral problems due to communication failure.
A total of 13 treatment approaches were identified
across the 23 studies; six were solely or primarily motor,
five were linguistic, and two were AAC (see Table 3). Seven
of these 13 approaches represented combined approaches.
Of these, two combined two motor treatments (Lundeborg
& McAllister, 2007; Rosenthal, 1994), and another two
combined linguistic treatments (Iuzzini & Forrest, 2010;
McNeill et al., 2009a), not affecting their classification.
Another two combined a linguistic treatment (melodic
intonation therapy [MIT]) with a motor treatment, either
touch cue method (TCM; Martikainen & Korpilahti, 2011)

491

Articulation with
facilitative vowel
contexts
Combined intraoral
stimulation,
Electropalatography
(EPG) with NDP
Combined melodic
intonation therapy
(MIT) and touch cue
method (TCM)
Combined stimulability
(STP) and modified
core vocabulary (mCVT)
Computer-based
ABABABABA single
case design
Multiple baseline
across participants
SCED
Multiple baseline
SCED
Multiple baseline
across discourse
contexts SCED
Lundeborg and
McAllister (2007)
Martikainen and
Korpilahti (2011)
Iuzzini and Forrest (2010)
Harris, Doyle,
and Haaf (1996)
IIb
IIb
IIb
IIb
4 (3;76;10, 2 males,
2 females, previous
SLP for 3/4)
1 (5 years, male,
approximately 3 years
previous SLP)
1 (4;7, female,
1 year previous SLP)
1 (5;1, female, 1.50 years

previous SLP)
1 (7 years, male,
1 year previous SLP)
IIb
IIb
1/3 (6 years, female,

previous SLP NR)
IIb
Binger, Kent-Walsh,
Multiple baseline
Berens, Del Campo,
across probes SCED
and Rivera (2008)
Binger, MaguireMultiple baseline
Marshall, and
across 3 participants
Kent-Walsh (2011)
SCED
Stokes and Griffiths (2010) ABA single case
design
Participant
description
2/5 (4;2 [years;months]
and 4;4, male, had
previous SLP)
1/3 (3;4, female, previous
SLP NR)
Level of
evidence
IIb
Binger and Light (2007)
Aided AAC modeling
Research design
Multiple baseline across
3 participants SCED
Published articles
Intervention name/
approach
Table 2. Research design, level of evidence, participant description, and diagnosis analysis of the 23 Level IIb (SCED) articles.
Severe CAS+
(Hx OME, receptive
and expressive LD)
Severe CAS
Severe CAS
Severe CAS
Mild SSD (Hx of CAS)
Severe CAS+
(suspected VCFS
with profound VPI)
Severe CAS+ (receptive
and expressive LD)
Severe CAS+ (GDD)
Diagnosis
(table continues)
3 (dysprosody NR)
4 (dysprosody and
sequencing NR)
3 (dysprosody NR)
4 (dysprosody and
nconsistency NR)
4 (dysprosody and
inconsistency NR)
4 (dysprosody and
sequencing NR)
Diagnostic
confidence rating

493
Integral Stimulation/
Dynamic Temporal
and Tactile Cueing
(DTTC)
Intervention name/
approach
Table 2 (Continued).
Maas, Butalla, and

Farinella (2012)
Maas and Farinella

(2012)
Multiple baseline SCED
ABalternating
treatments single
design (with three
stable baselines)
Edeal and GildersleeveNeumann (2011)
IIb
IIb
IIb
IIb
IIb
IIb
Level of
evidence
Research design
Baas, Strand, Elmer,

and Barbaresi (2008)
Strand and Debertine

(2000)
Strand, Stoeckel,
and Baas (2006)
Published articles
4 (3 in common with
Maas & Farinella,
2012)
(5;48;4, 2 females,
2 males, previous
SLP NR)
4 (5;07;9, 2 females,
2 males, previous
SLP NR)
2 (6;2 and 3;4, male,

14 years
previous SLP)
1 (12;8, male, 10 years

previous SLP)
1 (5 years, female,
4 years previous SLP)
4 (5;56;1, all male,
24 years
previous SLP)
Participant
description
CAS002;
CAS005see above;
CAS012CAS+
(moderatesevere
receptive LD,
Hx OME)
Severe CAS+
(2 with mild spastic
and/or ataxic
dysarthria, 1 with mild
intellectual disability,
and 1 with OME)
Severe CAS+ (CHARGE
syndrome intellectual
disability)
6;2severe CAS+
(repaired CLP, severe
receptive LD),
3;4severe CAS
CAS001moderate
severe CAS;
CAS002severe
CAS+ (dysarthria);
CAS005moderate
severe CAS+
(dysarthria
and receptive LD);
CAS010mildmoderate CAS+
(sensory processing,
fine and gross
motor skill delay,
hypotonia,
moderatesevere
receptive LD)
CAS001;
Severe CAS (Hx of VPI)
Diagnosis
(table continues)
2 (by consensus); as
above and CAS012 = 2
(clearly comorbid)
3 (by consensus);
CAS001 = 1, CAS002 = 3
(dysprosody NR, clearly
comorbid), CAS005 = 2
(clearly comorbid),
CAS010 = 4 (inconsistency
and coarticulation NR,
clearly comorbid)
4 (for all cases;

dysprosody
and inconsistency
NR, clearly comorbid)
Diagnostic
confidence rating

Bornman, Alant, and

Meiring (2001)
Voice output devices
Multiple baseline
across behaviors and
participants design
ABA single case design
ABA single case design
IIb
IIb
IIb
IIb
IIb
IIb
IIb
IIb
IIb
Level of
evidence
3 (7;810;10, 2 males,
1 female, 15 years
previous SLP)
1 (6;6, male, 2.50 years
previous SLP)
4 (1014 years, 3 males,

1 female, all had
previous SLP)
2 (6 and 5 years, male,
had previous SLP)
1 (8 years, female,
5 years previous SLP)
2 (identical twins also

in McNeill et al.,
2009a)
(4;5, male, moderate
severe)
Same participants as
McNeill et al. (2009a)
12 (4;27;6, 3 females,
9 males, previous
SLP NR)
3 (6;36;10, 2 males,
1 female, up to
2 years previous
SLP)
Participant
description
CAS+ (anoxia causing

slight left hemiplegia,
grand mal fits)
CAS (severity not

stated)
Severe CAS+
(intellectual disability,
Hx tube insertion,
congenital
heart defect)
Mild or mild
moderate CAS
Male 1Severe CAS+

(receptive and
expressive LD);
Male 2Severe
CAS+ (receptive
and expressive LD);
Femalemild
moderate CAS
Mildmoderate to
severe CAS
(no other diagnoses
reported)
CAS (small interstitial
deletion on
chromosome
10 (deletion at
10q21.222.1)
Mildmoderate
to severe CAS
(no other diagnoses
reported)
CAS (severity not
stated)
Diagnosis
4 (dysprosody and
inconsistency NR)
2 (comorbid CAS)
4 (dysprosody and
inconsistency NR)
3 (dysprosody NR)
3 (dysprosody NR)
3 (dysprosody NR)
3 (Male 1 = 4 inconsistency
and dysprosody NR;
Male 2 and Female = 3)
Diagnostic
confidence rating
Note. Please see the online supplemental materials, Supplemental Table 3, for the 19 Level III articles. SLP = speech-language pathology; CAS+ = comorbid childhood apraxia of speech;
GDD = global developmental delay; NR = not reported; VCFS = velocardiofacial syndrome; VPI = velopharyngeal incompetence; LD = language delay/disorder; A = baseline/withdrawal
phase; B = treatment/intervention phase; SSD = speech sound disorder; Hx = history of; NDP = Nuffield Dyspraxia Programme; OME = otitis media with effusion/glue ear; CHARGE
syndrome = coloboma, heart disease, atresia of the choanae, retarded growth and mental development, genital anomalies, and ear malformations and hearing loss; CLP = cleft lip
and palate.
Ballard, Robin, McCabe,

and McDonald (2010)
Rapid Syllable Transition

Treatment (ReST)
Partners in augmentative
communication
training (PACT)
Krauss and Galloway

(1982)
Culp (1989)
Quasi-experimental group
(following SCED)
McNeill, Gillon, and

Dodd (2010)
MIT

Dodd (2009b)
ABAB single case design

(with alternating
treatments)
ABAA single case design

Dodd (2009a)
Rosenthal (1994)
Research design
Moriarty and Gillon

(2006)
Published articles
Rate control therapy
Integrated Phonological
Awareness Intervention
(N = 5)
Intervention name/
approach

495
Linguistic
with
some
motor
aspects
Articulation with
facilitative vowel
contextsa
Combined intraoral
stimulation and
EPG (with NDP)b
Motor
with
cueing
Integrated
Phonological
Awareness (PA)
Interventionh
MIT with traditional
therapyi
4/4
Rate Control
Therapyf
Combined STP
and mCVTg
2/2
11/15
No
Yes9/13
moderatelarge
effect sizes
Yessignificant
effects
No
Yes1/5 post-MIT
(PVC; however,
PCC declined)
3/5 significant
post-TCM
Yessignificant
effects for all
measures
Yessignificant
effects
Statistics used?
Attained?
Yesas group
of 12 (3 NR)
NR
NR
Yes for 2/3
Time
NR
NR
1/1
NR
6 months
post
NR
4 weeks
post
NR
NR
11/15
NR
NR
3/3
NR
NR
NR
0/4 to discourse
NR
NR
NR
NR
NR
Response
Stimulus
generalization
generalization
(Significant in no. (Significant in no.
of participants)
of participants)
24 weeks 6/7 (6 NR)

post
12 weeks
post
NR
2 weeks
post
Maintenance
VariedPVC
maintained.
PCC and PMLU
only significant
after MIT
withdrawn.
Greater changes
after withdrawal.
Yes for 5/7 (6 NR)
NR
Yes
(1) Porch Index of

Yessignificant
NR
Communicative
verbal naming and
Ability in
imitation
Children, (2) MLU
(2) phones added to

inventory, (3)
inconsistency
(CSIP),
(4) inconsistency
(ISP)
(1) % suppression of Yessignificant
process usage,
effects
(2) PA accuracy
4/4 (only 3/4 (1) PCC,

for CSIP)
Perceptual stress
matches
Rx accuracy
3/3
ReSTe
(2) PCC,
(3) PMLU,
(4) PWP,
(5) PWC
(1) PCC,
(2) PPC,
(3) PWC,
(4) intelligibility,
(5) assessment of
visual deviancy
(1) PVC,
R
Accuracy (/ /)
Rx accuracy
1/1
1/1
1/1
Measures
Integral Stimulation/ 11/13

DTTCd
Combined MIT and

TCMc
Therapy
approach
Therapy
type
Cases with
reported
Rx effect
Treatment across all participants
Table 3. Treatment outcomes for the 23 SCED articles.
(table continues)
Suggestive
Preponderant
Suggestive
Suggestive
Preponderant
Preponderant
Suggestive
Suggestive
Suggestive
Judgment
of certainty

No
No
No
Yesmoderate
large effect sizes
Statistics used?
NR
Yes
NR
Yes for all
Attained?
Time
NR
4 weeks
post
NR
NR
NR
NR
Suggestive
Suggestive
participation
Suggestive
Suggestive
Judgment
of certainty
Spoke intelligibly
after 1 year
speech and
participation,
frustration
NR
(2) NR
(1) 3/3,
Response
Stimulus
generalization
generalization
(Significant in no. (Significant in no.
of participants)
of participants)
2, 4, and
NR
8 weeks
post
Maintenance
MIT completed in the first block, and TCM completed in the second block.
a
Stokes and Griffiths (2010). bLundeborg and McAllister (2007). cMartikainen and Korpilahti (2011). dStrand and Debertine (2000), Strand et al. (2006), Baas et al. (2008), Edeal and
Gildersleeve-Neumann (2011), Maas and Farinella (2012), and Maas et al. (2012). eBallard et al. (2010). fRosenthal (1994). gIuzzini and Forrest (2010). hMoriarty and Gillon (2006) and McNeill
et al. (2009a, 2009b, 2010). iKrauss and Galloway (1982). jBinger and Light (2007) and Binger et al. (2008, 2011). kHarris et al. (1996). lBornman et al. (2001). mCulp (1989).
Note. Rx = medical prescription; PCC = percentage of consonants correct; PPC = percentage of phonemes correct; PWC = percentage of words correct; PVC = percentage of vowels
correct; PMLU = phonological mean length of utterance; PWP = proportion of whole-word proximity; CSIP = consonant substitute inconsistency percentage; ISP = inconsistency severity
percentage; MLU = mean length of utterance.
1/1
Communicative
effectiveness
(frequency
of turns)
Voice output
devices
Macawl
PACTm
1/1
AAC
Rx accuracy
(1) book reading
(2) discourse
No. of appropriate
responses
1/1
Computer-based
AACk
(1) multisymbol
messages
(2) morpheme
accuracy
Rx frequency
4/4
Aided AAC
modeling (with
communication
board or voice
output devices)j
Linguistic
with
some
AAC
Measures
Therapy
approach
Cases with
reported
Rx effect
Treatment across all participants
Therapy
type
or traditional articulation therapy (Krauss & Galloway, 1982).

The first was classified as a motor approach because of the
goals and PML incorporated; the second was classified as a
linguistic approach, as MIT was the primary experimental
approach, and linguistic outcomes were primarily sought and
reported. Finally, for one motor treatment, the participant
continued regular AAC therapy during the research (Edeal
& Gildersleeve-Neumann, 2011).
Structure of Treatment Delivery
All 23 treatments were delivered individually, with
22 delivered in a clinic and one delivered at the participants
home (Lundeborg & McAllister, 2007). Caregiver and child
training sessions were utilized in the two AAC studies within a
consultative-collaboration service delivery model (Bornman
et al., 2001; Culp, 1989). Inclusion of parent training and
home practice protocols or activities was more prevalent in
AAC-based treatments and was used in six of 23 articles.
For motor treatments reporting dose frequency, the
median was three times a week, with a maximum of once a
day and minimum of twice a week. Sessions were between
20 and 60 min long. Most linguistic and AAC approaches
provided treatment two to three times a week for between
15- and 60-min sessions. A small number gave intensive daily,
short-term training. Dose in terms of treatment trials completed within sessions was adequately described in five of
23 articles or three of 13 approaches and ranged from 60 to
120 trials for motor approaches and from 10 to 30 trials
for linguistic and AAC approaches (Ballard, Robin, McCabe,
& McDonald, 2010; Binger et al., 2008; Binger & Light,
2007; Binger et al., 2011; Stokes & Griffiths, 2010). Further
details of the treatment procedure analyses for each study
and approach are provided in the online supplemental materials, Supplemental Table 3.
Aim 3: Treatment Outcomes

Analyses of treatment, maintenance, and generalization
outcomes for the 23 SCED articles are reported in Table 3.
All studies used baseline phases, and 91% incorporated untrained control items intended to demonstrate some experimental control.
Treatment and Maintenance Data
All articles reported treatment effects for the majority
of the participants, despite a range of goals and measures
being used. Statistical comparison of at least one key outcome
was provided for 16 of 23 studies.
Of the 23 participants given motor-based treatment, 21
were reported to demonstrate positive treatment effects, and a
statistical analysis of effects was reported for 17 (see Table 3).
Not all participants showed significant changes in all measures assessed. The majority of measures consisted of percentage of accuracy on treated items or percentage of consonant,
vowel, phonemes, or words correct. Three studies demonstrated improvement for treated prosodic accuracy: Ballard
et al. (2010) for three of three participants using the Rapid
Syllable Transition Treatment (ReST) and Maas, Butalla,
and Farinella (2012) and Maas and Farinella (2012) for three
of four participants following Dynamic Temporal and Tactile
Cueing (DTTC; in any PML condition). The combined
MIT/TCM treatment showed significantly improved percentage
of vowels correct; however, it significantly reduced percentage of consonants correct for the participant immediately
post the first block of MIT. The greatest gains were noted
after withdrawal of treatment. Despite the authors suggesting
this to be due to the treatments given, this equally may be due
to maturation or improvement after withdrawal of treatment
providing unclear evidence to the effect of these treatments.
Six studies of linguistic-based treatment reported a
treatment effect for speech measures for 17 of 21 participants,
with 16 of 21 participants supported by statistical comparison (Iuzzini & Forrest, 2010; Krauss & Galloway, 1982;
McNeill et al., 2009a, 2009b, 2010; Moriarty & Gillon, 2006).
With Integrated Phonological Awareness Intervention, 11 of
15 participants were reported to reduce phonological processes and to improve phonological awareness skills. Another
four articles reported that five of five participants increased
use of multisymbol messages (phrases or morphemes) with
linguistic-based treatment utilizing AAC (Binger et al., 2008;
Binger & Light, 2007; Binger et al., 2011; Harris et al., 1996).
Finally, AAC treatment studies focusing on communicative effectiveness reported treatment effects for two of two
participants, with no statistical analyses conducted (Bornman
et al., 2001; Culp, 1989). The children reportedly increased
appropriate responses and frequency of turns in conversation.
Generalization
Seven articles considered response generalization, and
another five considered stimulus generalization. No article
measured both response and stimulus generalization.
Response generalization. All treatments measuring
generalization used statistical analysis. For motor-based
treatments, significant improvement in articulation accuracy
for untrained responses was noted for one participant after
facilitative vowel treatment (Stokes & Griffiths, 2010) and for
four of seven participants across any behavior/condition in
three studies applying Integral Stimulation/DTTC (Edeal
& Gildersleeve-Neumann, 2011; Maas et al., 2012; Maas
& Farinella, 2012). Significant generalization was reported
for three of three participants for lexical stress accuracy
in untrained three syllable pseudowords and for one of
three participants in untrained real word production for ReST
treatment (Ballard et al., 2010).
Only one linguistic-based approach, the Integrated
Phonological Awareness Intervention, reported response
generalization. The same 11 of 15 children who demonstrated
treatment gains also showed significant improvement in
speech intelligibility, mean length of utterance, and phonological awareness skills (phoneme segregation, manipulation,
nonword reading, reading accuracy, and lettersound correspondences; McNeill et al., 2009a, 2009b, 2010; Moriarty
& Gillon, 2006).
Stimulus generalization. Only five articles (22%) reported stimulus generalization, with four of these utilizing AAC treatments. Three participants, who increased

497
grammatical constituents in treatment, also generalized use of

symbol combinations across different scenarios or discourse
types (Binger et al., 2008; Binger & Light, 2007). Anecdotal
reports, predominately from caregivers, suggested improved
expressive language skills or communication across settings,
reduced frustration due to AAC device use (Binger & Light,
2007; Culp, 1989), and transition to reliance on speech
(Bornman et al., 2001). There was one reported instance of
poor stimulus generalization; rate control therapy showed
minimal generalization from the treated reading task to untreated conversation (Rosenthal, 1994).
Aim 4: Certainty of Evidence

The level of certainty that the effects of each treatment
were true (Smith, 1981) is reported in Table 3. No treatment approaches met the criteria for conclusive evidence.
Three treatment approaches, two motor (Integral Stimulation/
DTTC, ReST) and one linguistic (Integrated Phonological
Awareness Intervention), met the criteria for preponderant
evidence (replicated evidence across participants with promising treatment, maintenance, and generalization data). The
remaining approaches qualified as suggestive evidence. These
included studies with questionable effects (e.g., combined
MIT/TCM treatment), as there was not a specific category for
these within Smiths (1981) framework. Two studies with
suggestive evidence approached preponderant evidence: Aided
AAC modeling met all the criteria except for confidence in
CAS diagnosis, and the facilitative vowel contexts treatment
reported only one case with low confidence in CAS diagnosis.
The overall treatment effect size for each of the three
approaches with preponderant evidence was determined
using IRD (Parker et al., 2009; see Table 4). Integral Stimulation/
DTTC demonstrated a moderate effect size for articulation
and/or prosodic accuracy (IRD = 0.60) for seven participants
ranging in age from 3;4 (years;months) to 8;4 with mild
moderate to severe CAS or CAS+. Integrated Phonological
Awareness Intervention also demonstrated moderate effect
sizes for percentage of phonemes correct (IRD = 0.51) for
15 participants ranging in age from 4;2 to 7;6 with mild
moderate to severe CAS. Finally, ReST demonstrated a large
effect size for prosodic accuracy (IRD = 0.78) for three participants ranging in age from 7;8 to 10;10 with mild to mild
moderate CAS.
Generalization effects varied according to the treatment and measures used, and thus a separate effect size was
calculated per measure/condition (see Table 5). The motor
treatments of Integral Stimulation/DTTC and ReST showed
predominantly small effect sizes, with some moderate-to-large
effects (ranging from IRD = 0.20 to IRD = 0.84). The Integrated Phonological Awareness Intervention showed a large
effect size for percentage of phonemes correct (IRD = 0.80).
subject experimental designs with Level IIb evidence to identify

promising treatment approaches for further study and for
cautious application in clinical settings. Twenty-three studies
qualified for in-depth review, and three treatment approaches,
tested in seven studies, reached the level of preponderant
evidence with promising evidence of efficacy across several
participants diagnosed with CAS (confidence rating of 13).
Study Quality
The vast majority of studies examining treatment for
CAS are single case studies, with an increasing trend toward
more rigorous experimental designs over time. This trend
may be a consequence of critical narrative reviews in the late
1980s and 1990s (e.g., Hall et al., 1993; McCabe et al., 1998;
Pannbacker, 1988) and greater awareness of research design
and evidence-based practice (ASHA, 2004). The body of
research reflects Phase I and Phase II studies (Robey, 2004)
designed to test the feasibility and early efficacy of treatments.
At this stage, no Phase III RCT or NRCT reports are available to contribute to conclusive evidence (Smith, 1981).
Therefore, no conclusions as to which treatments are more
efficacious than others for CAS are currently possible (ASHA,
2007; Morgan & Vogel, 2009; Pannbacker, 1988). This review
identified preponderant evidence and well-designed, quasiexperimental studies that can guide clinical decisions and that
are suitable to pursue in more substantive comparative efficacy studies.
Certainty of Evidence
Two motor treatments (Integral Stimulation /DTTC
and ReST) and one linguistic treatment (Integrated Phonological Awareness Intervention) demonstrated preponderant
evidence (Smith, 1981) with positive treatment and generalization effects across several children. Only Integral Stimulation/
DTTC is supported currently by studies across independent
research groups (Baas, Strand, Elmer, & Barbaresi, 2008; Edeal
& Gildersleeve-Neumann, 2011; Maas et al., 2012; Maas &
Farinella, 2012; Strand & Debertine, 2000; Strand et al., 2006),
although such replication was not directly analyzed here.
SCED ratings in the future could be elaborated beyond replication for external validity and application of the same
protocol in direct replication studies (Tate et al., 2013) to also
include independence of research groups. Currently, no direct
replication studies exist for CAS treatment.
The remaining articles were classified as suggestive
evidence. With future well-controlled investigation, some of
these treatments, as well as others excluded from this review,
will likely emerge as promising options for CAS. Additionally, an extra level of certainty should be considered for future
reviews flagging studies with questionable effects (i.e., those
that did not demonstrate clear treatment effects).
Discussion
Confidence in CAS Diagnosis
The aim of this study was to conduct an in-depth and

systematic review of treatment efficacy studies for children
with CAS. Unlike previous reviews, we considered within-
A critical component of any treatment study is a clear

definition of the study participants to convince readers that
the appropriate population was targeted. When the 2007

Table 4. Omnibus improvement rate differences (IRDs) for preponderant evidencetreatment effects.
Treatment
type
Motor
Linguistic
Treatment approach
Article
ReST
Strand and Debertine (2000)

Edeal and GildersleeveNeumann (2011)
Maas and Farinella (2012)
Maas et al. (2012)
Overall
Ballard et al. (2010)
Integrated Phonological
Awareness
Intervention
Moriarty and Gillon (2006)

McNeill et al. (2009a)
Overall
Integral Stimulation/
DTTCb
No. of
cases
Confidence
in CAS Dxa
Omnibus
IRD
95% CI
1
2
1
1
1.00
0.98
[0.97, 1.00]
[0.88, 1.00]
Very large
Very large
4c
4c
3
2
0.18
0.22
0.60
0.78
[0.03, 0.33]
[0.08, 0.36]
[0.53, 0.67]
[0.54, 1.00]
3
12
3
3
1.00
0.10
0.51
[0.89, 1.00]
[0.06, 0.24]
[0.39, 0.58]
Small or questionable
Moderate
Large (prosody
PVI duration)
Very large
Moderate
Interpretation
of effect
Note. CI = confidence interval; PVI = pairwise variability index (acoustic measure).

a
1 = highest. bBaas et al. (2008) and Strand et al. (2006) were excluded from IRD because of diagnostic confidence ratings of 4 and 5.
c
Three participants in common across two studies.
primarily conveyed on vowels, we counted mention of vowel

errors in participants as possible evidence of dysprosody to
accommodate older articles. Despite the chance that dysprosody would be overestimated because of this decision (as
only a subset of vowel errors or distortions would be indicative of stress errors), only 50% of the articles (21 of 42)
reported vowel errors. Furthermore, the high rate of comorbidity in CAS (41% in this sample) complicates diagnosis and
could partially account for lower confidence ratings.
consensus-based diagnostic features (ASHA, 2007) were applied, only 16.6% achieved a rating of high confidence. However, most of the studies reviewed were published prior to
2007 and were using common descriptors for their time, now
considered by many to be nondiscriminative (e.g., ASHA,
2007; McCabe et al., 1998). The most commonly overlooked
CAS characteristic across studies was dysprosody, which
was not considered a core feature of CAS in many checklists
prior to 2007. As prosody (e.g., lexical or phrasal stress) is
Table 5. Omnibus IRDs for preponderant evidencegeneralization.
Therapy
type
Therapy
approach
Article
Generalization to
untrained itemsa
IRD
NR
95% CI
NA
Interpretation
of effect
NR
MFF sounds
4c
Maas et al. (2012)
4c
ReST
Ballard et al. (2010)
Linguistic Integrated
Phonological
Awareness
Intervention
Moriarty and Gillon

(2006)
McNeill et al.
(2009a)
0.05 [10.47, 0.57] Small or

questionable
HFF sounds
0.60 [0.18, 1.00] Moderate
Blocked practice items
0.04 [0.16, 0.24] Small or
questionable
Random practice items 0.20 [0.01, 0.39] Small or
questionable
100% feedback items 0.15 [0.13, 0.43] Small or
questionable
60% feedback items
0.03 [0.14, 0.20] Small or
questionable
Less complex
0.84 [0.62, 1.00] Large
pseudowords
(PVI duration)
Real words
0.13 [0.21, 0.47] Small or
(PVI duration)
questionable
NR
NR NA
NA
12
Untrained PPC
Motor
Integral Stimulation/ Strand and

Debertine
DTTCb
(2000)
Edeal and
GildersleeveNeumann (2011)
Maas and Farinella
(2012)
Confidence
No. of in CAS Dx
cases (1 = highest)
0.80 [0.64, 0.88]
NA
Large
Note. NA = not applicable; MFF = moderate frequency feedback; HFF = high frequency feedback.
a
Generalization items were individualized for each participant. bBaas et al. (2008) and Strand et al. (2006) were excluded from IRD because of
diagnostic confidence ratings of 4 and 5. cThree participants in common across two studies.

499
Experimental Control
Experimental control is essential to ensure that treatment effects are attributable to the intervention approach
provided and are an essential element to calculating effect
sizes for single-subject designs (Olswang & Bain, 1994). Only
experimental designs with some experimental control were
included in the treatment outcome analysis. Experimental
control was best demonstrated when SCEDs reported change
in at least one condition beyond baseline levels after withdrawal of treatment and when control data were used to
estimate improvement due to maturation (Byiers et al., 2012;
Olswang & Bain, 1994; Perdices & Tate, 2009). Not all of
the studies analyzed here demonstrated clear experimental
control and treatment effects on the basis of these conditions.
In future research, CAS SCEDs would ideally use more
than two phases (i.e., beyond just baseline and treatment but
also withdrawal and other treatment phases), including at
least three data points per phase (including baselines) and
replication across cases (Tate et al., 2013).
Within this review, 22% (five of 23) of the articles at
Level IIb of evidence reported some changes in untreated
speech behaviors, which were hypothesized to be unrelated to
the treated behaviors. This phenomenon reflects a loss of
experimental control and may undermine the claim of positive treatment effects. However, it may also reflect the
underspecification of theories of speech motor control, in that
there is limited evidence to guide how and why speech
behaviors are related motorically (Folkins & Bleile, 1990).
This issue has been more fully discussed in studies of treatment of acquired apraxia of speech (Ballard, 2001; Ballard,
Maas, & Robin, 2007). These unexpected generalization
outcomes may provide interesting directions for exploring
relationships between different speech skills and for guiding
the selection of generalization and control stimuli in future
treatment trials.
Treatment Approaches
The reviewed treatments for CAS can be categorized as
targeting motor or linguistic skills or using AAC to provide
a primary means of communication. The approaches used
can also be viewed in light of the International Classification
of Functioning, Disability, and Health (ICF; World Health
Organization, 2002). Motor and linguistic approaches were
primarily directed at the impairment (body functions/structure)
level of the ICF (McLeod & McCormack, 2007), commonly
addressing articulation, prosody, phonological awareness, or
expressive language skills. AAC approaches were instead directed at the activity/participation levels of the ICF, facilitating
communicative effectiveness with a greater emphasis on consultation, training, and home practice. The literature suggests
that the primary concern in CAS is developing intelligible
speech, either through addressing articulatory and prosodic
accuracy or through improving phonology, although concentration on AAC and expressive language may be required.
The majority of SCEDs intentionally combined treatments, a trend that appears to be increasing. This may be
due to the hypothesized links between speech motor and

linguistic difficulties in children with CAS (Souza, Payo, &
Costa, 2009). However, the studies using combined approaches
had participants without comorbid CAS and often combined
approaches within the same therapy type (e.g., two motor
treatments). The theoretical motivation for combining such
treatments is unclear, and no comparisons of single versus
combined treatments for children with clear diagnoses of
CAS are yet available.
Treatment Outcomes
It is well accepted that the strongest evidence for a
treatment efficacy is demonstration of skill maintenance
beyond the treatment period and generalization of treatment
effects to related behaviors and/or communication contexts
(e.g., McReynolds & Kearns, 1983; Schmidt & Lee, 2011).
Such effects constitute instrumental clinical change (Olswang
& Bain, 1994, 2013). Few of the 23 studies evaluated reported
maintenance of treatment or generalization effects. As
such, the robustness of most reported treatment effects over
time is not known.
The six approaches that demonstrated maintenance
and response or stimulus generalization were the three approaches with preponderant evidence (Integral Stimulation/
DTTC, ReST, and Integrated Phonological Awareness
Intervention) and the aided AAC modeling and facilitative
vowel context interventions. Degree of generalization varied,
seemingly influenced by specific stimuli chosen, incorporation of PML, dosage, frequency and intensity of sessions,
potential critical thresholds for skill mastery, and participant
characteristics. Notably, only those participants demonstrating strong gains for treated behaviors tended to maintain
or generalize skills (e.g., Maas & Farinella, 2012; McNeill
et al., 2009a). The exception to this was the phonological
treatment within the Integrated Phonological Awareness
Intervention, which showed large generalization effects despite
demonstrating moderate treatment effects. This is an expected
outcome in interventions that focus on learning linguistic
rules that can be rapidly generalized across a range of contexts
(Gierut, 1998; Gierut & Hulse, 2010) versus learning new
motor skills. There are at least two possible explanations for
why children with probable CAS, a motor speech disorder,
responded to a phonological treatment. First, phonological
therapy involves production of speech targets and so provides practice in planning, programming, and executing the
movements for these targets. Second, the participants had a
number of phonological processes, and perhaps their concomitant phonological disorders were a primary concern for
treatment at this time. These findings warrant thoughtful
comparison of specific participant characteristics, each interventions theoretical framework, the stimuli and activities used
during intervention, and stimuli used to assess generalization
of skill to determine whether there is in fact any benefit in one
approach over the other.
Likewise, stimulus generalization provides important
information about how skills treated in therapy generalize to
other contexts or situations for improvement in everyday

communication. Only rate control therapy (Rosenthal, 1994)

and AAC approaches considered such generalization. Such
measures should be employed as we move toward Phase III
and Phase IV studies to demonstrate real change in communication skills in children with CAS.
It is important to note that, despite most children in the
studies showing positive effects in treatment, all children
required additional therapy to work on other communication goals after treatment blocks spanning between nine
and 195 sessions. This demonstrates that children with CAS
often require ongoing therapy, spanning many goals and
needs.
Further investigation is warranted to explore interactions between participant variables and the degree of
treatment outcome to help tailor treatments, particularly for
those who did not demonstrate treatment effects. For example, Integral Simulation/DTTC was initially designed for
children with severe CAS using functional core vocabulary
stimuli (Strand & Skinder, 1999). Whereas studies addressing
severe CAS have reported positive treatment effects across
multiple participants, those addressing mild-to-moderate
CAS and specific sound, prosody, and/or word structure
goals have had small omnibus effect sizes (Maas et al., 2012;
Maas & Farinella, 2012).
Similarly, it is yet to be determined whether AAC
approaches can promote speech gains over and above speechbased interventions (as described in Bornman et al., 2001).
The current literature suggests that when a child experienced
frustration over low intelligibility or comprehensibility,
AAC approaches may increase communicative success as
well as stimulate development of language skills that cannot
be practiced through speaking.
Structure of Treatment Delivery

Intensive treatment delivery in impairment-based
intervention appears crucial for obtaining positive treatment
outcomes. These treatments provided therapy at least 23 times
a week, with sessions of up to 60 min. The dose of treatment,
defined as the number of properly administrated teaching
episodes during a single intervention session (Warren et al.,
2007, p. 71), should probably also be high (Edeal & GildersleeveNeumann, 2011). This review suggests that at least 60 trials
per session represents a high dose. Williams (2012) suggested that, with phonological therapy for speech sound
disorder, 50 trials per session over 30 sessions is effective, although dose and intensity need to increase as impairment severity increases. Further research is indicated to
allow reliable estimates of the overall amount of therapy
needed, dose, and intensity for CAS, considering age and
severity.
SCED studies in this review all delivered treatment
in individual sessions, and 40% utilized home practice to
increase dose and intensity. The high intensity that is indicated would clearly require significant resources. Thus, determining effective treatments with engaging home practice
activities should maximize maintenance and generalization
of skills.
Implications
Despite a continued need for well-designed RCTs,
NRCTs, and additional SCED studies, existing evidence is
available to guide clinical practice in the treatment of CAS.
Comparing treatments is a future priority, considering the
range of treatments that are available. The treatments best
suited to inclusion in an RCT or NRCT are those that have
demonstrated maintenance and generalization effects. Further research is needed for valid and reliable differential
diagnosis of CAS and for understanding which client groups
would benefit from which type of treatment.
In terms of clinical practice, currently two motor
treatments (Integral Stimulation/DTTC and ReST) and one
linguistic treatment (Integrated Phonological Awareness
Intervention) are best suited to interim clinical use, with sessions at least twice a week and dose above 60 trials per session.
DTTC appears to work better for clients with more severe
CAS, Integrated Phonological Awareness Intervention appears to work better for children 47 years of age with mild to
severe CAS, and ReST appears to work better for children
710 years of age with mild-to-moderate CAS.
Limitations
Because of the developing nature of this literature, this
systematic review utilized some narrative review methodology to ensure greater coverage of SCED designs that differed
considerably (Collins & Fauser, 2004). Potential bias was
reduced through the use of systematic review principles.
We considered only peer-reviewed published reports
in English. We did not control for publication bias, as we
excluded evidence that had not undergone journal-level peer
review. This means that evidence from conference presentations or dissertations was excluded, thus excluding other
approaches with potential suggestive evidence, such as Prompts
for Restructuring Oral Muscular Phonetic Targets (Dale &
Hayden, 2011; now see Dale & Hayden, 2013) or the Nuffield
Dyspraxia Programme (Belton, 2006). As in all reviews, there
is a risk that studies with negative treatment outcomes are
underrepresented because of the difficulty publishing such
studies, potentially risking overestimation of treatment effects.
Our IRD analysis examined preliminary effect sizes
but did not use moderator variables because of the small
number of articles (n = 7) and participants (n = 26). Here, we
provide some preliminary observations about which treatments appear useful for different age and severity levels. As
additional studies are published, it is recommended that such
moderator variables be evaluated more fully.
It is important to note the IRD is calculated and interpreted differently to other effect sizes. For example, the recent
DTTC studies (Maas et al., 2012; Maas & Farinella, 2012)
used Beeson and Robeys (2006) effect size method and found
large effect sizes for some participants and conditions and
found no effects for others. As the majority of articles did
not report means and standard deviations, such effect size
calculations were not possible across all preponderant articles. The IRDs reported here differ from those reported

501
in Maas et al. (2012) and Maas and Farinella (2012) in two

ways. First, in our calculation of IRD, only nonoverlapping
data between baseline/withdrawal and treatment phases
were considered indicative of improvement. Second, the
IRDs here were averaged across participants within studies;
thus, greater variability across participants conservatively
reduced the magnitude of the omnibus IRD.
Other aspects that could be investigated were beyond the
scope of the current study. This includes SCED methodological quality as per reported guidelines (e.g., Tate et al., 2013;
see Wendt & Miller, 2012, for a review) and analysis of motor
learning principles utilized in each treatment (e.g., Bislick,
Weir, Spencer, Kendall, & Yorkston, 2012). We did not evaluate the theoretical stance of each treatment approach, which
might have revealed further information as to why some
treatments generated stronger effects than others.
Conclusion
This review identified 23 SCED articles reporting 13
treatment approaches classified as primarily motor, linguistic,
or AAC. Three treatments were judged to have preponderant
evidence to support their efficacy, indicating that the reported effects were probably true: Integral Stimulation/
DTTC, ReST, and Integrated Phonological Awareness
Intervention. These treatments had moderate-to-large treatment effects and small-to-large generalization effects, making
them strong candidates for Phase III research, comparison
in RCTs, and interim clinical use. Larger scale rigorous RCTs
and NRCTs are critically needed to compare treatments with
larger sample sizes to potentially inform a greater number
of clients with CAS (Morgan & Vogel, 2009). Such efforts will
serve to generate conclusive evidence through well-controlled
experimental designs, including measures of maintenance
and generalization of treatment effects and manipulation of
treatment intensity and other motor learning principles. This
work will be facilitated by parallel work toward a reliable
diagnostic system and exploration of participant variables
that influence treatment response.
Acknowledgments
This research was supported by the Douglas and Lola Douglas
Scholarship on Child and Adolescent Health, the Speech Pathology
Australia Nadia Verrall Memorial Research Grant and Postgraduate
Research Award, and the University of Sydney James Kentley
Memorial Scholarship and Postgraduate Research Support Scheme
awarded to the first author. This research was also supported by an
Australian Research Council Future Fellowship awarded to the
third author. We thank Catherine Mason and Donna Thomas for
assistance coding the data and completing reliability.
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AJSLP

A Systematic Review of Treatment

Purpose: To present a systematic review of single-case

or augmentative and alternative communication (n = 2). Most

especially in the realization of lexical or phrasal stress (ASHA,

University of Sydney, New South Wales, Australia

Correspondence to Elizabeth Murray:

Key Words: dyspraxia, intervention, efficacy,

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literacy, academic, social, and vocational difficulties (e.g.,

Smiths (1981) level of certainty hierarchy considers the

Study quality: to describe for each identified study the

Treatment procedures: to define the behavioral goals

Treatment outcomes: to examine reported treatment,

Murray et al.: Systematic Review of Treatment Outcomes for CAS

Downloaded From: http://ajslp.pubs.asha.org/ by a Proquest User on 08/12/2014

literacy, academic, social, and vocational difficulties (e.g.,

Smiths (1981) level of certainty hierarchy considers the

Study quality: to describe for each identified study the

Treatment procedures: to define the behavioral goals

Treatment outcomes: to examine reported treatment,

Murray et al.: Systematic Review of Treatment Outcomes for CAS

Downloaded From: http://ajslp.pubs.asha.org/ by a Proquest User on 08/12/2014

criteria were as follows: (a) peer-reviewed articles published

488 American Journal of Speech-Language Pathology Vol. 23 486504 August 2014

CAS+, in which CAS was the primary diagnosis, but other

Murray et al.: Systematic Review of Treatment Outcomes for CAS

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Only one of the three primary

CAS was the primary diagnosis

CAS without another comorbid disorder

CAS was the primary diagnosis

This includes the following:

CAS was described. Other

Other characteristics that were

random 33/83 children was 91%. Discrepancies were resolved

Aim 3: Treatment Outcomes

490 American Journal of Speech-Language Pathology Vol. 23 486504 August 2014

Other comorbid disorders may be

For studies with preponderant evidence, effect sizes

Of the 19 articles, three articles (15.8%) received a Level 1

Aim 2: Treatment Procedures

Murray et al.: Systematic Review of Treatment Outcomes for CAS

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492 American Journal of Speech-Language Pathology Vol. 23 486504 August 2014

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Iuzzini and Forrest (2010)

1 (5;1, female, 1.50 years

1/3 (6 years, female,

Binger and Light (2007)

Aided AAC modeling

Mild SSD (Hx of CAS)

Severe CAS+ (GDD)

Murray et al.: Systematic Review of Treatment Outcomes for CAS

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Maas, Butalla, and

Maas and Farinella

Multiple baseline SCED

Edeal and GildersleeveNeumann (2011)

Multiple baseline SCED

Multiple baseline SCED

Multiple baseline SCED

Baas, Strand, Elmer,

Strand and Debertine