A Systematic Review of Treatment Outcome

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 or augmentative and alternative communication (n = 2). Most
experimental treatment studies for childhood apraxia of participants responded positively to treatment, but only 7 of
speech (CAS). 13 approaches in SCED studies reported maintenance and/or
Method: A search of 9 databases was used to find generalization of treatment effects. Three approaches had
peer-reviewed treatment articles from 1970 to 2012 of all preponderant evidence (Smith, 1981). IRD effect sizes were
levels of evidence with published communication outcomes calculated for Integral Stimulation/Dynamic Temporal and
for children with CAS. Improvement rate differences (IRDs) Tactile Cueing, Rapid Syllable Transition Treatment, and
were calculated for articles with replicated (n > 1), statistically Integrated Phonological Awareness Intervention.
compared treatment and generalization evidence. Conclusions: At least 3 treatments have sufficient evidence
Results: Forty-two articles representing Phase I and II for Phase III trials and interim clinical practice. In the future,
single-case experimental designs (SCEDs; n = 23) or case efficacy needs to be established via maintenance and
series or description studies (n = 19) were analyzed. generalization measures.
Six articles showed high CAS diagnosis confidence. Of the
13 approaches within the 23 SCED articles, treatments were Key Words: dyspraxia, intervention, efficacy,
primarily for speech motor skills (n = 6), linguistic skills (n = 5), methodological rigor, generalization
C
hildhood apraxia of speech (CAS) is a developmen- especially in the realization of lexical or phrasal stress” (ASHA,
tal disorder of speech motor planning and/or pro- 2007, p. 4).
gramming (American Speech-Language-Hearing Although impaired movement planning and program-
Association [ASHA], 2007). It is also known as develop- ming are considered to underlie CAS, there are also reports of
mental verbal dyspraxia in the United Kingdom (excluding disrupted development of speech perception, language, and
being the result of any known neurological disorder) and phonology (including phonological awareness) in children
has previously been called developmental apraxia of speech with CAS (Groenen, Maassen, Crul, & Thoonen, 1996; Lewis
and dyspraxia. CAS causes reduced speech intelligibility et al., 2004; Maassen, Groenen, & Crul, 2003). It is unclear
because of a hypothesized impairment in the “transformation whether these are primary deficits or flow-on effects from
of an abstract phonological code into motor speech com- CAS, comorbid impairments, or perhaps compensatory be-
mands” (Terband, Maassen, Guenther, & Brumberg, 2009, haviors, as children with CAS develop their linguistic, pho-
p. 1598). Such impairment leads to the current consensus- nological, and motor skills concurrently (Alcock, Passingham,
based core CAS features of “(a) inconsistent errors on con- Watkins, & Vargha-Khadem, 2000; Marion, Sussman, &
sonants and vowels in repeated productions of syllables or Marquardt, 1993; Ozanne, 2005). Children with CAS can
words, (b) lengthened and disrupted coarticulatory transitions therefore present with a range of difficulties requiring therapy
between sounds and syllables, and (c) inappropriate prosody, from speech-language pathologists (SLPs; Royal College of
Speech and Language Therapists, 2011).
a
The long-term functioning of people with CAS is
University of Sydney, New South Wales, Australia largely unreported. The available longitudinal studies suggest
Correspondence to Elizabeth Murray: that CAS is a persistent disorder that requires therapy (Hall,
[email protected] Jordan, & Robin, 1993; Jacks, Marquardt, & Davis, 2006;
Editor: Carol Scheffner Hammer Stackhouse & Snowling, 1992). Children with CAS, like others
Associate Editor: Ken Bleile with persistent speech sound disorder, are also at risk for
Received March 27, 2013
Revision received July 7, 2013
Accepted December 1, 2013 Disclosure: The authors have declared that no competing interests existed at the
DOI: 10.1044/2014_AJSLP-13-0035 time of publication.
486 American Journal of Speech-Language Pathology • Vol. 23 • 486–504 • August 2014 • A American Speech-Language-Hearing Association
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literacy, academic, social, and vocational difficulties (e.g., Smith’s (1981) level of certainty hierarchy considers the
Lewis et al., 2004; Moriarty & Gillon, 2006). research design and the possible effects of the intervention
A Cochrane systematic review, a subsequent journal to provide an overall judgment on how likely the results are to
article, and a treatment review have reported no published be true. For the CAS literature, this can also be extended
randomized controlled trials (RCTs) or nonrandomized to include confidence in diagnosis. SCEDs can show early
controlled trials (NRCTs) for any intervention for CAS evidence for an intervention, with results from such studies
(Morgan & Vogel, 2008, 2009; Watts, 2009). Despite this being classed as suggestive or “possibly true.” However,
lack of high-level evidence, many published articles on the statistically compared outcomes from SCEDs with confident
treatment of CAS could facilitate practice and could help CAS diagnoses, replication (n > 1), and evidence of both
identify potential lines of further research. Narrative reviews treatment effects and generalization of treatment effects could
have identified a range of treatment methods for children be considered preponderant evidence or “probably true.”
reported to have CAS, likely reflecting the diversity of symp- SCED designs are usually designated as Phase II and Level IIb
toms seen in these children and potentially the research and evidence (but see Hegde, 2007; Kearns & de Riesthal, 2013).
clinical interests of the authors (ASHA, 2007; Strand & Currently, Phase III studies (Robey, 2004) are typically RCTs
Skinder, 1999). They encompass motor treatments (including and NRCTs. These generate Level IIa evidence using groups
electropalatography), linguistic approaches, augmentative of participants to reduce bias and to eliminate individual
and alternative communication (AAC), or some combina- variance as a factor in treatment success. It is only through
tion thereof (ASHA, 2007; Gillon & Moriarty, 2007; Hall, meta-analyses and systematic reviews of several Phase III
2000; Morgan & Vogel, 2008). Few of these lower level studies of a given treatment approach, coupled with Phase IV
treatment studies have been examined rigorously, as they effectiveness studies in real-world clinical situations, that
were excluded on the basis of quality in previous systematic results can be defined as conclusive or “undoubtedly true”
reviews (Morgan & Vogel, 2008; Watts, 2009). Provision (Smith, 1981). As all studies of CAS treatments to date are
of recommendations regarding which treatments have sup- classified as Phase I and Phase II, the goal of this review is
portive evidence has therefore not been possible (ASHA, to identify treatment approaches with suggestive or prepon-
2007; Morgan & Vogel, 2008; Pannbacker, 1988). This article derant evidence.
presents a systematic review of all levels of evidence that Central to this review is treatment efficacy. Efficacy
may be critical to inform clinical practice until high-level considers clinical cause-and-effect relationships between
evidence becomes available. the provision of intervention and change in participant be-
Two primary challenges face a systematic review of havior (e.g., McReynolds & Kearns, 1983; Olswang & Bain,
intervention for CAS. The first challenge is in identifying the 2013). Demonstration of efficacy extends beyond treatment
rigor of each study in terms of research phase (Robey, 2004), effects, requiring assessment of maintenance and generaliza-
the research design, the level of evidence generated (ASHA, tion of treatment effects that signify instrumental change.
2004; Perdices et al., 2006), as well as the level of certainty that Response generalization evaluates a child’s performance on
the effects reported for a given treatment approach are real untrained items that are somehow related to trained items,
(Smith, 1981). Treatment research often follows a develop- to determine whether more widespread change is occurring
mental pathway that is associated with increasing research (Olswang & Bain, 1994). Stimulus generalization assesses
rigor and different research questions. Robey (2004) defined performance on untrained materials, people, or settings/
five phases in a research program. Phase I and Phase II environments (Olswang & Bain, 1994). Such change is neces-
studies represent pilot or feasibility studies seeking to deter- sary to meet the overall goals of an intervention.
mine 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 quasi-
Aims
experimental group (case series) or single case reports with This systematic review evaluated studies of interven-
pre- to posttreatment measurement and no within-subject tion, published between 1970 and October 2012, that state
comparison or control conditions. Level IIb evidence comes an intention to treat children with CAS. The aims fall into
from more rigorous single-case experimental designs (SCEDs) four broad areas:
that systematically apply and withdraw treatment and es- 1. Study quality: to describe for each identified study the
tablish control using a stable baseline phase or limited change research phase, the level of evidence, and the level of
in control conditions (Byiers, Reichle, & Symons, 2012; confidence in CAS diagnosis;
Olswang & Bain, 1994; Perdices & Tate, 2009).
The second challenge is that there is not yet a validated 2. Treatment procedures: to define the behavioral goals
assessment tool for diagnosing CAS. Thus, before evaluation and structure of treatment (e.g., intensity and dosage
of the treatment in any intervention study, the descriptions according to Warren, Fey, & Yoder, 2007) for each
of participants must be scrutinized to determine the level of SCED study (at/above Level IIb evidence) and to
confidence in the authors’ diagnosis. This involves determin- group similar treatment types to facilitate treatment
ing to what extent the participants are described as meeting outcome analysis;
the three consensus-based core features of CAS listed earlier 3. Treatment outcomes: to examine reported treatment,
(ASHA, 2007). maintenance, and generalization outcomes; and
Murray et al.: Systematic Review of Treatment Outcomes for CAS 487

4. Certainty of evidence: to determine the level of cer- criteria were as follows: (a) peer-reviewed articles published
tainty for each treatment approach (Smith, 1981) and between 1970 and October 2012; (b) in English (to allow
to determine the effect size for any intervention ap- analysis by monolingual English speaking authors); (c) treat-
proach classed as having preponderant evidence. ing at least one child/adolescent under 18 years of age with
CAS or suspected CAS; and (d) reporting quantitative par-
ticipant data outcomes focused on speech (i.e., articulation,
Method phonology, prosody, intelligibility, rate), communication (i.e.,
Systematic Search Strategy pragmatics, social communication, AAC use), or language
The search strategy used follows Preferred Reporting (i.e., phonology, phonological awareness, grammar, mor-
Items for Systematic Reviews and Meta-Analyses (PRISMA) phology, receptive or expressive language, reading, spelling
search guidelines (Moher, Liberati, Tetzlaff, & Altman, or writing). There was no exclusion on the basis of the type
2009). The flow diagram of study selection is presented in of therapy provided or who provided it. All named and
Figure 1. unnamed interventions were included, as were all levels of
evidence, except for systematic reviews, as they do not con-
Identification tain individual participant data (ASHA, 2004). This yielded
Nine databases related to speech-language pathology 42 articles for review. Intrarater reliability (first author) for
were comprehensively searched for peer-reviewed journal inclusion of these studies was 96% (n = 1,032) with >2 months
articles. These were Allied and Complementary Medicine, between assessments. Interrater reliability with an indepen-
Cumulative Index to Nursing and Allied Health Literature, dent rater was 91% (see Supplemental Appendix 1 in the
Evidence-Based Medicine Reviews–Cochrane Database online supplemental materials for the list of excluded articles
of Systematic Reviews, Education Resources Information and reason for exclusion). Raters were not blinded to article
Center, Linguistic Language Behavior Abstracts, Medline, title or authors, and raters did not review their own publi-
PsycINFO, Scopus, and speechBITE. Specific search terms cations. Excluded articles were not further analyzed.
varied on the basis of each database catalogue of terms in its Finally, an additional search was undertaken for other
search directories. Key words used were as follows: “apraxia” documents, such as published treatment manuals and the-
or “dyspraxia” or “childhood apraxia of speech” and oretical or opinion articles on the individual treatment ap-
“child*” or “develop*” and “motor speech therapy” or proaches, to confirm the type of behavior(s) targeted (e.g.,
“interven*” or “treat*” or “speech therapy/pathology” or speech motor or phonological), cues, and stimuli used. This
“efficacy” or “evaluation” or “effect” and “speech” or “com- search used reference lists of already identified articles as well
munication” or “language” or “articulation impairments” as database and Google searches using intervention names
or “speech impairments” or “speech disorders” or “speech and key authors as search terms.
intelligibility” or “prosody.” A total of 1,301 studies were
identified from database searches. Data Analysis
Review of studies for Aims 1, 3, and 4 was based on
Screening
information provided in each article (e.g., Moseley, Herbert,
All references were exported to EndNote X5 (Thomson
Maher, Sherrington, & Elkins, 2008). In one case, only group
Reuters, 2011), where duplicates were removed. References
data were published, and we contacted an author (Brigid
were also screened to ensure that authors stated an intention to
McNeill) for individual data (from McNeill, Gillon, & Dodd,
treat children with CAS (using synonyms; e.g., developmental
2009a). Addressing Aim 2 at times required reference to treat-
verbal dyspraxia). Thus, references were searched by title,
ment manuals and other publications describing a treat-
abstract, and key words in EndNote X5 and were excluded
ment’s theoretical framework.
if treatment articles involved other diagnoses without ref-
erence to CAS: “cerebral palsy,” “dysarthria,” “cleft palate,”
“swallowing,” “ataxia,” “cochlear implants,” “deaf,” Aim 1: Study Quality
“stutter,” “fluency,” “acquired,” “Down syndrome,” “autism,” Each study was assigned to a phase of research (Robey,
“phonolog*,” and “gait apraxia.” All references that related 2004), from Phase I to Phase V. The experimental design
to assessment, diagnosis, or description/exploration of symp- and level of evidence were defined on the basis of published
toms were excluded. Of the intervention articles that remained, guidelines (ASHA, 2004; Perdices et al., 2006). In addi-
the intervention names and authors were searched again in tion, confidence in CAS diagnosis was assessed as detailed
all the above databases as well as Google Scholar to ensure below.
that all relevant articles were found. The reference lists of Confidence in CAS diagnosis. A 5-point rating scale
all review articles obtained were also searched to find any was used to rate confidence in CAS diagnosis (see Table 1;
additional articles. Screening removed 913 articles, leaving Wambaugh, Duffy, McNeil, Robin, & Rogers, 2006). This was
119 to be assessed for eligibility. based on description of primary versus nondiscriminative
features (McCabe, Rosenthal, & McLeod, 1998; see the
Eligibility online supplemental materials, Supplemental Table 1). Pri-
Copies of articles were obtained and assessed against mary features were the three consensus-based features listed
the final inclusion criteria before being reviewed. These in ASHA’s (2007) technical report, hypothesized to represent
488 American Journal of Speech-Language Pathology • Vol. 23 • 486–504 • 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. Non- CAS+, in which CAS was the primary diagnosis, but other
discriminative features were those shared with other dis- disorders were present. A rating of 1 indicated high con-
orders, such as poor intelligibility, slow progress, or delayed fidence in CAS diagnosis, and a rating of 5 indicated no
language (ASHA, 2007; McCabe et al., 1998). Clear cases confidence. Intrarater reliability (first author) on four judg-
of comorbid disorders were also noted, such as receptive ments (presence of each of the three primary features and
language impairment or dysarthria. On the basis of this CAS diagnosis) for 83/83 children studied was 94%. Inter-
analysis, participants were classified either as CAS only or as rater reliability between the first and second authors for a

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 Primary characteristics Nondiscriminative characteristics Comorbidity
Level 1 All the primary characteristics were CAS was the primary diagnosis CAS without another comorbid disorder
described as follows: described. Any characteristics that was reported (excluding expressive
• Inconsistency and were attributable to other disorders language delay).
• Lengthened and disrupted may have been described but
coarticulatory transitions between were not used to diagnose CAS.
sounds and syllables and
• Inappropriate prosody.
Level 2 All the primary characteristics were CAS was the primary diagnosis This includes the following:
described as follows: described. Other characteristics • CAS without another comorbid
• Inconsistency and attributable to other disorders (e.g., disorder (excluding expressive
• Lengthened and disrupted dysarthria) were described and may language delay) or
coarticulatory transitions between have been used to diagnose CAS. • Clear cases of comorbid CAS,
sounds and syllables and in which CAS was the primary
• Inappropriate prosody. diagnosis.
Level 3 Two of the three primary characteristics CAS was described. Other This includes the following:
were described: characteristics that were attributable • CAS without another comorbid
• Inconsistency and/or to other disorders (e.g., dysarthria) disorder or
• Lengthened and disrupted were described and may have been • Clear cases of comorbid CAS, in
coarticulatory transitions between used to diagnose CAS. which CAS was the primary
sounds and syllables and/or diagnosis or
• Inappropriate prosody. • Cases of CAS in which another
comorbid disorder had the same
severity (e.g., language delay,
dysarthria).
Level 4 Only one of the three primary Other characteristics that were Unclear whether CAS was the primary
characteristics was reported, or attributable to other disorders may diagnosis.
incomplete/inadequate description of have been described, and it is unclear
the primary characteristics of CAS whether these were used to diagnose
was provided. CAS.
Level 5 Diagnosis of CAS was reported or Unclear whether CAS diagnosis was Other comorbid disorders may be
implied, but no primary characteristics likely and/or whether CAS was the present.
were described. primary diagnosis.
random 33/83 children was 91%. Discrepancies were resolved Aim 3: Treatment Outcomes
by consensus. Reported treatment, maintenance, and generalization
Exclusions. Articles that lacked experimental control outcomes for each intervention were analyzed for (a) number
(Level III evidence; n = 19) were excluded, as they could of participants with a treatment gain (change immediately
not be used to determine treatment outcomes. The remain- after treatment compared with baseline); (b) assessment
ing 23 Level IIb articles were analyzed to address Aims 2, 3, measures and statistics used in determining treatment effects;
and 4. No articles were excluded because of confidence in (c) maintenance of treatment gains at least 2 weeks post-
CAS diagnosis; however, confidence in CAS diagnosis was treatment, from report or by comparing treatment data with
a factor in determining certainty of evidence—see Aim 4 performance in maintenance probes; (d) response generaliza-
below. tion data, when statistical analysis was used; and (e) stimulus
generalization data.
Aim 2: Treatment Procedures
Articles designed with adequate experimental control Aim 4: Certainty of Evidence
(see Aim 1 above) were analyzed descriptively regarding the Smith’s (1981) three levels of certainty were applied, on
nature of the treatment. Using the stated treatment goals, the basis of design (i.e., level of evidence, research design,
selected stimuli, and specific cueing strategies reported, treat- confidence in CAS diagnosis, and statistical comparison) and
ments were categorized as primarily (a) motor, (b) linguistic/ possible effects of the intervention/outcomes. A treatment
phonological (including literacy), or (c) AAC (ASHA, approach was categorized as having preponderant evidence
2007; Gillon & Moriarty, 2007; Hall, 2000; Martikainen & when it showed Level IIb or better evidence (SCEDs), rep-
Korpilahti, 2011). The structure of treatment delivery was licated cases, diagnostic confidence ratings of 1–3, statistically
also determined (Warren et al., 2007), including dose (trials significant treatment and generalization effects (or at least
per session), dose frequency (number of times a dose is moderate effect sizes), and clear maintenance of treatment
provided over days or weeks), and total intervention time gains at least 2 weeks posttreatment. Any Level IIb or better
(number of sessions). When reported, home practice and evidence that did not meet all the above criteria received the
service delivery model were documented. lowest rating of suggestive evidence.

For studies with preponderant evidence, effect sizes Of the 19 articles, three articles (15.8%) received a Level 1
were calculated using improvement rate difference (IRD rating for high confidence in diagnosis, zero articles (0.0%)
[also known as risk difference]; Parker, Vannest, & Brown, received a Level 2 rating, five articles (26.3%) received a
2009), a valid and reliable tool for SCEDs used frequently in Level 3 rating, five articles (26.3%) received a Level 4 rating,
medical research. This method utilizes visual analysis and has and six articles (31.5%) received a Level 5 rating for no con-
reduced assumptions in comparison with other effect size fidence (see the online supplemental materials, Supplemen-
calculations allowing use over a range of SCED designs. It is tal Table 2). The 19 Level III articles were not analyzed
more discriminative to change than other nonoverlap tech- further because of lack of experimental control.
niques (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 Aim 2: Treatment Procedures
improvement rates of the treatment and baseline/withdrawal/ The first section details the classification of treatments
maintenance phases. The improvement rate was calculated on the basis of their approach. The second section presents
for each phase by determining the number of improved results on service delivery models used.
points in each phase (those exceeding the data points of the
adjacent phase) divided by all the data points of the phase Classification of Treatment Goals and Approaches
(Parker et al., 2009). Thus, an IRD eliminates overlap across Eleven of 23 SCED studies were classified as primarily
phases in its determination of effect size. motor approaches (see Table 3). They included primary
IRDs were determined for treatment effects for each measures for accuracy of articulation and/or prosody. Two
participant and each behavior within an article. The IRDs studies included secondary measures of speech intelligibility
were then averaged across participants for the treated be- or comprehensibility (Strand & Debertine, 2000; Strand,
havior(s) to determine an omnibus IRD for each article, as per Stoeckel, & Baas, 2006). All utilized articulatory placement
Parker et al. (2009), with 95% confidence intervals determined and imitation cues as well as multimodal cues (e.g., kinesthetic/
using WinPEPI (Abramson, 2011). Effects for each article touch cues, manipulating speech rate and timing, picture
were averaged again to determine an omnibus IRD for a given or orthographic stimuli). The majority (90%) made explicit
treatment approach. reference to incorporating principles of motor learning (PML;
IRDs were also calculated for generalization effects see Maas et al., 2008, for a review).
for each participant and each behavior/condition within an Ten studies were classified as primarily linguistic ap-
article. No further omnibus IRDs were determined for gen- proaches. Of these, six studies reported primary measures of
eralization because of the various and often heterogeneous speech sound production (phonological processes or stimul-
measures used. ability), phonological awareness accuracy, and spoken lan-
guage utterance length, which also used some motor cueing
(Iuzzini & Forrest, 2010; Krauss & Galloway, 1982; McNeill,
Results Gillon, & Dodd, 2009a, 2009b, 2010; Moriarty & Gillon, 2006).
The other four studies targeted expressive language skills
Aim 1: Study Quality (e.g., multisymbol messages or elaborated phrase structures)
The 42 studies that met the inclusion criteria were using AAC systems in children with previously established
classified as Phase I or Phase II. Of these, 23 represented AAC use (Binger, Kent-Walsh, Berens, Del Campo, & Rivera,
Level IIb evidence (SCEDs), and 19 represented Level III 2008; Binger & Light, 2007; Binger, Maguire-Marshall, &
evidence (one quasi-experimental case series and 18 case Kent-Walsh, 2011; Harris, Doyle, & Haaf, 1996).
reports or descriptions; see Figure 1). There was a shift to- Two studies were classified as AAC, measuring com-
ward higher quality single case studies over time, with six municative effectiveness in children with severe comorbid
Level IIb and 12 Level III articles prior to 2006, and with CAS (CAS+), by introducing AAC systems (Bornman, Alant,
17 Level IIb and five Level III articles from 2006 to 2012. & Meiring, 2001; Culp, 1989). These children had reportedly
No RCT or NRCT designs have been published to date. shown slow or minimal progress in speech production at-
Overall, there were 83 participants across the 42 single tempts and/or used AAC to alleviate frustration and behav-
case studies. Within the 23 SCED articles, 32 participants ioral problems due to communication failure.
were reported to have CAS, and 19 participants were reported A total of 13 treatment approaches were identified
to have comorbid CAS (CAS+). Of the 23 articles, four ar- across the 23 studies; six were solely or primarily motor,
ticles (17.4%) received a Level 1 rating for high confidence five were linguistic, and two were AAC (see Table 3). Seven
in diagnosis, two articles (8.7%) received a Level 2 rating for of these 13 approaches represented combined approaches.
clear cases of CAS with comorbid disorders, seven articles Of these, two combined two motor treatments (Lundeborg
(30.4%) received a Level 3 rating, seven articles (30.4%) re- & McAllister, 2007; Rosenthal, 1994), and another two
ceived a Level 4 rating, and three articles (13.0%) received a combined linguistic treatments (Iuzzini & Forrest, 2010;
Level 5 rating for no confidence (see Table 2). McNeill et al., 2009a), not affecting their classification.
Within the 19 case series and description articles, Another two combined a linguistic treatment (melodic
25 participants were reported to have CAS, and eight par- intonation therapy [MIT]) with a motor treatment, either
ticipants were reported to have comorbid CAS (CAS+). touch cue method (TCM; Martikainen & Korpilahti, 2011)

Table 2. Research design, level of evidence, participant description, and diagnosis analysis of the 23 Level IIb (SCED) articles.
Intervention name/ Level of Participant Diagnostic

approach Published articles Research design evidence description Diagnosis confidence rating
Aided AAC modeling Binger and Light (2007) Multiple baseline across IIb 2/5 (4;2 [years;months] Severe CAS+ (GDD) 4 (dysprosody and
3 participants SCED and 4;4, male, had sequencing NR)
previous SLP)
Binger, Kent-Walsh, Multiple baseline IIb 1/3 (3;4, female, previous Severe CAS+ 4 (dysprosody and
Berens, Del Campo, across probes SCED SLP NR) (suspected VCFS inconsistency NR)
and Rivera (2008) with profound VPI)

Binger, Maguire- Multiple baseline IIb 1/3 (6 years, female, Severe CAS+ (receptive 5
Marshall, and across 3 participants previous SLP NR) and expressive LD)
Kent-Walsh (2011) SCED
Articulation with Stokes and Griffiths (2010) ABA single case IIb 1 (7 years, male, Mild SSD (Hx of CAS) 4 (dysprosody and
facilitative vowel design 1 year previous SLP) nconsistency NR)
contexts
Combined intraoral Lundeborg and ABABABABA single IIb 1 (5;1, female, 1.50 years Severe CAS 3 (dysprosody NR)
stimulation, McAllister (2007) case design previous SLP)
Electropalatography
(EPG) with NDP
Combined melodic Martikainen and Multiple baseline IIb 1 (4;7, female, Severe CAS 1
intonation therapy Korpilahti (2011) across participants 1 year previous SLP)
(MIT) and touch cue SCED
method (TCM)
Combined stimulability Iuzzini and Forrest (2010) Multiple baseline IIb 4 (3;7–6;10, 2 males, Severe CAS 4 (dysprosody and

(STP) and modified SCED 2 females, previous sequencing NR)
core vocabulary (mCVT) SLP for 3/4)
Computer-based Harris, Doyle, Multiple baseline IIb 1 (5 years, male, Severe CAS+ 3 (dysprosody NR)
and Haaf (1996) across discourse approximately 3 years (Hx OME, receptive
contexts SCED previous SLP) and expressive LD)
(table continues)
Table 2 (Continued).

Integral Stimulation/ Strand and Debertine Multiple baseline SCED IIb 1 (5 years, female, Severe CAS (Hx of VPI) 1
Dynamic Temporal (2000) 4 years previous SLP)
and Tactile Cueing Strand, Stoeckel, Multiple baseline SCED IIb 4 (5;5–6;1, all male, Severe CAS+ 4 (for all cases;
(DTTC) and Baas (2006) 2–4 years (2 with mild spastic dysprosody
previous SLP) and/or ataxic and inconsistency
dysarthria, 1 with mild NR, clearly comorbid)
intellectual disability,
and 1 with OME)
Baas, Strand, Elmer, Multiple baseline SCED IIb 1 (12;8, male, 10 years Severe CAS+ (CHARGE 5
and Barbaresi (2008) previous SLP) syndrome intellectual
disability)
Edeal and Gildersleeve- AB—alternating IIb 2 (6;2 and 3;4, male, 6;2—severe CAS+ 1

Neumann (2011) treatments single 1–4 years (repaired CLP, severe
design (with three previous SLP) receptive LD),
stable baselines) 3;4—severe CAS
Maas and Farinella Multiple baseline SCED IIb 4 (5;0–7;9, 2 females, CAS001—moderate– 3 (by consensus);
(2012) 2 males, previous severe CAS; CAS001 = 1, CAS002 = 3
SLP NR) CAS002—severe (dysprosody NR, clearly
CAS+ (dysarthria); comorbid), CAS005 = 2
CAS005—moderate– (clearly comorbid),
severe CAS+ CAS010 = 4 (inconsistency
(dysarthria and coarticulation NR,
and receptive LD); clearly comorbid)
CAS010—mild-
moderate CAS+
(sensory processing,
fine and gross

motor skill delay,
hypotonia,
moderate–severe
receptive LD)
Maas, Butalla, and Multiple baseline SCED IIb 4 (3 in common with CAS001; 2 (by consensus); as
Farinella (2012) Maas & Farinella, above and CAS012 = 2
2012) (clearly comorbid)
(5;4–8;4, 2 females, CAS002;
2 males, previous CAS005—see above;
SLP NR) CAS012—CAS+
(moderate–severe
receptive LD,
Hx OME)
(table continues)
Murray et al.: Systematic Review of Treatment Outcomes for CAS

493

Integrated Phonological Moriarty and Gillon Multiple baseline SCED IIb 3 (6;3–6;10, 2 males, Male 1—Severe CAS+ 3 (Male 1 = 4 inconsistency
Awareness Intervention (2006) 1 female, up to (receptive and and dysprosody NR;
(N = 5) 2 years previous expressive LD); Male 2 and Female = 3)
SLP) Male 2—Severe
CAS+ (receptive
and expressive LD);
Female—mild–
moderate CAS
McNeill, Gillon, and Multiple baseline SCED IIb 12 (4;2–7;6, 3 females, Mild–moderate to 3 (dysprosody NR)
Dodd (2009a) 9 males, previous severe CAS
SLP NR) (no other diagnoses
reported)
McNeill, Gillon, and Multiple baseline SCED IIb 2 (identical twins also CAS (small interstitial 3 (dysprosody NR)

Dodd (2009b) in McNeill et al., deletion on
2009a) chromosome
(4;5, male, moderate– 10 (deletion at
severe) 10q21.2–22.1)
McNeill, Gillon, and Quasi-experimental group IIb Same participants as Mild–moderate 3 (dysprosody NR)
Dodd (2010) (following SCED) McNeill et al. (2009a) to severe CAS
(no other diagnoses
reported)
Rate control therapy Rosenthal (1994) ABAB single case design IIb 4 (10–14 years, 3 males, CAS (severity not 4 (dysprosody and
(with alternating 1 female, all had stated) inconsistency NR)
treatments) previous SLP)
MIT Krauss and Galloway ABAA single case design IIb 2 (6 and 5 years, male, CAS (severity not 4 (dysprosody and
(1982) had previous SLP) stated) inconsistency NR)
Partners in augmentative Culp (1989) ABA single case design IIb 1 (8 years, female, Severe CAS+ 2 (comorbid CAS)
communication 5 years previous SLP) (intellectual disability,

training (PACT) Hx tube insertion,
congenital
heart defect)
Rapid Syllable Transition Ballard, Robin, McCabe, Multiple baseline IIb 3 (7;8–10;10, 2 males, Mild or mild– 1
Treatment (ReST) and McDonald (2010) across behaviors and 1 female, 1–5 years moderate CAS
participants design previous SLP)
Voice output devices Bornman, Alant, and ABA single case design IIb 1 (6;6, male, 2.50 years CAS+ (anoxia causing 5
Meiring (2001) previous SLP) slight left hemiplegia,
grand mal fits)
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.
Table 3. Treatment outcomes for the 23 SCED articles.
Treatment across all participants

Response Stimulus
Cases with Maintenance generalization generalization
Therapy Therapy reported (Significant in no. (Significant in no. Judgment
type approach Rx effect Measures Statistics used? Attained? Time of participants) of participants) of certainty
R
Motor Articulation with 1/1 Accuracy (/ /) Yes—significant Yes 2 weeks 1/1 NR Suggestive
with facilitative vowel effects post
cueing contextsa
Combined intraoral 1/1 (1) PCC, Yes—significant NR NR NR NR Suggestive
stimulation and (2) PPC, effects for all
EPG (with NDP)b (3) PWC, measures
(4) intelligibility,
(5) assessment of
visual deviancy
Combined MIT and 1/1 (1) PVC, Yes†—1/5 post-MIT Varied—PVC 12 weeks NR NR Suggestive
TCMc (PVC; however, maintained. post
(2) PCC, PCC declined)— PCC and PMLU

(3) PMLU, 3/5 significant only significant
(4) PWP, post-TCM after MIT
(5) PWC withdrawn.
Greater changes
after withdrawal.
Integral Stimulation/ 11/13 Rx accuracy Yes—9/13 Yes for 5/7 (6 NR) 2–4 weeks 6/7 (6 NR) NR Preponderant
DTTCd moderate–large post
effect sizes
ReSTe 3/3 Perceptual stress Yes—significant Yes for 2/3 4 weeks 3/3 NR Preponderant
matches effects post
Rate Control 4/4 Rx accuracy No NR NR NR 0/4 to discourse Suggestive
Therapyf
Linguistic Combined STP 4/4 (only 3/4 (1) PCC, No NR NR NR NR Suggestive
with and mCVTg for CSIP)
some

motor
aspects
(2) phones added to
inventory, (3) ↓
inconsistency
(CSIP),
(4) ↓ inconsistency
(ISP)
Integrated 11/15 (1) % suppression of Yes—significant Yes—as group 6 months 11/15 NR Preponderant
Phonological process usage, effects of 12 (3 NR) post
Awareness (PA) (2) PA accuracy
Interventionh
MIT with traditional 2/2 (1) Porch Index of Yes—significant NR NR NR NR Suggestive
therapyi Communicative verbal naming and
Ability in imitation
Children, (2) MLU
Murray et al.: Systematic Review of Treatment Outcomes for CAS

(table continues)
495
Treatment across all participants

Response Stimulus
Cases with Maintenance generalization generalization
Therapy Therapy reported (Significant in no. (Significant in no. Judgment
type approach Rx effect Measures Statistics used? Attained? Time of participants) of participants) of certainty
Linguistic Aided AAC 4/4 Rx frequency Yes—moderate– Yes for all 2, 4, and NR (1) 3/3, Suggestive
with modeling (with large effect sizes 8 weeks
some communication post
AAC board or voice
output devices)j (1) multisymbol (2) NR

messages
(2) morpheme ↑ speech and
accuracy participation,
↓ frustration
Computer-based 1/1 Rx accuracy No NR NR NR NR Suggestive
AACk (1) book reading
(2) discourse
AAC Voice output 1/1 No. of appropriate No Yes 4 weeks NR Spoke intelligibly Suggestive
devices— responses post after 1 year
Macawl
PACTm 1/1 Communicative No NR NR NR ↑ participation Suggestive
effectiveness
(frequency
of turns)

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.
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).
†
MIT completed in the first block, and TCM completed in the second block.
or traditional articulation therapy (Krauss & Galloway, 1982). and Farinella (2012) and Maas and Farinella (2012) for three
The first was classified as a motor approach because of the of four participants following Dynamic Temporal and Tactile
goals and PML incorporated; the second was classified as a Cueing (DTTC; in any PML condition). The combined
linguistic approach, as MIT was the primary experimental MIT/TCM treatment showed significantly improved percentage
approach, and linguistic outcomes were primarily sought and of vowels correct; however, it significantly reduced percent-
reported. Finally, for one motor treatment, the participant age of consonants correct for the participant immediately
continued regular AAC therapy during the research (Edeal post the first block of MIT. The greatest gains were noted
& Gildersleeve-Neumann, 2011). after withdrawal of treatment. Despite the authors suggesting
this to be due to the treatments given, this equally may be due
Structure of Treatment Delivery to maturation or improvement after withdrawal of treatment
All 23 treatments were delivered individually, with providing unclear evidence to the effect of these treatments.
22 delivered in a clinic and one delivered at the participant’s Six studies of linguistic-based treatment reported a
home (Lundeborg & McAllister, 2007). Caregiver and child treatment effect for speech measures for 17 of 21 participants,
training sessions were utilized in the two AAC studies within a with 16 of 21 participants supported by statistical compar-
consultative-collaboration service delivery model (Bornman ison (Iuzzini & Forrest, 2010; Krauss & Galloway, 1982;
et al., 2001; Culp, 1989). Inclusion of parent training and McNeill et al., 2009a, 2009b, 2010; Moriarty & Gillon, 2006).
home practice protocols or activities was more prevalent in With Integrated Phonological Awareness Intervention, 11 of
AAC-based treatments and was used in six of 23 articles. 15 participants were reported to reduce phonological pro-
For motor treatments reporting dose frequency, the cesses and to improve phonological awareness skills. Another
median was three times a week, with a maximum of once a four articles reported that five of five participants increased
day and minimum of twice a week. Sessions were between use of multisymbol messages (phrases or morphemes) with
20 and 60 min long. Most linguistic and AAC approaches linguistic-based treatment utilizing AAC (Binger et al., 2008;
provided treatment two to three times a week for between Binger & Light, 2007; Binger et al., 2011; Harris et al., 1996).
15- and 60-min sessions. A small number gave intensive daily, Finally, AAC treatment studies focusing on communi-
short-term training. Dose in terms of treatment trials com- cative effectiveness reported treatment effects for two of two
pleted within sessions was adequately described in five of participants, with no statistical analyses conducted (Bornman
23 articles or three of 13 approaches and ranged from 60 to et al., 2001; Culp, 1989). The children reportedly increased
120 trials for motor approaches and from 10 to 30 trials appropriate responses and frequency of turns in conversation.
for linguistic and AAC approaches (Ballard, Robin, McCabe,
& McDonald, 2010; Binger et al., 2008; Binger & Light, Generalization
2007; Binger et al., 2011; Stokes & Griffiths, 2010). Further Seven articles considered response generalization, and
details of the treatment procedure analyses for each study another five considered stimulus generalization. No article
and approach are provided in the online supplemental mate- measured both response and stimulus generalization.
rials, Supplemental Table 3. Response generalization. All treatments measuring
generalization used statistical analysis. For motor-based
treatments, significant improvement in articulation accuracy
Aim 3: Treatment Outcomes for untrained responses was noted for one participant after
Analyses of treatment, maintenance, and generalization facilitative vowel treatment (Stokes & Griffiths, 2010) and for
outcomes for the 23 SCED articles are reported in Table 3. four of seven participants across any behavior/condition in
All studies used baseline phases, and 91% incorporated un- three studies applying Integral Stimulation/DTTC (Edeal
trained control items intended to demonstrate some exper- & Gildersleeve-Neumann, 2011; Maas et al., 2012; Maas
imental control. & Farinella, 2012). Significant generalization was reported
for three of three participants for lexical stress accuracy
Treatment and Maintenance Data in untrained three syllable pseudowords and for one of
All articles reported treatment effects for the majority three participants in untrained real word production for ReST
of the participants, despite a range of goals and measures treatment (Ballard et al., 2010).
being used. Statistical comparison of at least one key outcome Only one linguistic-based approach, the Integrated
was provided for 16 of 23 studies. Phonological Awareness Intervention, reported response
Of the 23 participants given motor-based treatment, 21 generalization. The same 11 of 15 children who demonstrated
were reported to demonstrate positive treatment effects, and a treatment gains also showed significant improvement in
statistical analysis of effects was reported for 17 (see Table 3). speech intelligibility, mean length of utterance, and phono-
Not all participants showed significant changes in all mea- logical awareness skills (phoneme segregation, manipulation,
sures assessed. The majority of measures consisted of percent- nonword reading, reading accuracy, and letter–sound cor-
age of accuracy on treated items or percentage of consonant, respondences; McNeill et al., 2009a, 2009b, 2010; Moriarty
vowel, phonemes, or words correct. Three studies demon- & Gillon, 2006).
strated improvement for treated prosodic accuracy: Ballard Stimulus generalization. Only five articles (22%) re-
et al. (2010) for three of three participants using the Rapid ported stimulus generalization, with four of these utiliz-
Syllable Transition Treatment (ReST) and Maas, Butalla, ing AAC treatments. Three participants, who increased

grammatical constituents in treatment, also generalized use of subject experimental designs with Level IIb evidence to identify
symbol combinations across different scenarios or discourse promising treatment approaches for further study and for
types (Binger et al., 2008; Binger & Light, 2007). Anecdotal cautious application in clinical settings. Twenty-three studies
reports, predominately from caregivers, suggested improved qualified for in-depth review, and three treatment approaches,
expressive language skills or communication across settings, tested in seven studies, reached the level of preponderant
reduced frustration due to AAC device use (Binger & Light, evidence with promising evidence of efficacy across several
2007; Culp, 1989), and transition to reliance on speech participants diagnosed with CAS (confidence rating of 1–3).
(Bornman et al., 2001). There was one reported instance of
poor stimulus generalization; rate control therapy showed Study Quality
minimal generalization from the treated reading task to un-
treated conversation (Rosenthal, 1994). 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
Aim 4: Certainty of Evidence may be a consequence of critical narrative reviews in the late
The level of certainty that the effects of each treatment 1980s and 1990s (e.g., Hall et al., 1993; McCabe et al., 1998;
were true (Smith, 1981) is reported in Table 3. No treat- Pannbacker, 1988) and greater awareness of research design
ment approaches met the criteria for conclusive evidence. and evidence-based practice (ASHA, 2004). The body of
Three treatment approaches, two motor (Integral Stimulation/ research reflects Phase I and Phase II studies (Robey, 2004)
DTTC, ReST) and one linguistic (Integrated Phonological designed to test the feasibility and early efficacy of treatments.
Awareness Intervention), met the criteria for preponderant At this stage, no Phase III RCT or NRCT reports are avail-
evidence (replicated evidence across participants with prom- able to contribute to conclusive evidence (Smith, 1981).
ising treatment, maintenance, and generalization data). The Therefore, no conclusions as to which treatments are more
remaining approaches qualified as suggestive evidence. These efficacious than others for CAS are currently possible (ASHA,
included studies with questionable effects (e.g., combined 2007; Morgan & Vogel, 2009; Pannbacker, 1988). This review
MIT/TCM treatment), as there was not a specific category for identified preponderant evidence and well-designed, quasi-
these within Smith’s (1981) framework. Two studies with experimental studies that can guide clinical decisions and that
suggestive evidence approached preponderant evidence: Aided are suitable to pursue in more substantive comparative ef-
AAC modeling met all the criteria except for confidence in ficacy studies.
CAS diagnosis, and the facilitative vowel contexts treatment
reported only one case with low confidence in CAS diagnosis. Certainty of Evidence
The overall treatment effect size for each of the three
approaches with preponderant evidence was determined Two motor treatments (Integral Stimulation /DTTC
using IRD (Parker et al., 2009; see Table 4). Integral Stimulation/ and ReST) and one linguistic treatment (Integrated Phono-
DTTC demonstrated a moderate effect size for articulation logical Awareness Intervention) demonstrated preponderant
and/or prosodic accuracy (IRD = 0.60) for seven participants evidence (Smith, 1981) with positive treatment and generali-
ranging in age from 3;4 (years;months) to 8;4 with mild– zation effects across several children. Only Integral Stimulation/
moderate to severe CAS or CAS+. Integrated Phonological DTTC is supported currently by studies across independent
Awareness Intervention also demonstrated moderate effect research groups (Baas, Strand, Elmer, & Barbaresi, 2008; Edeal
sizes for percentage of phonemes correct (IRD = 0.51) for & Gildersleeve-Neumann, 2011; Maas et al., 2012; Maas &
15 participants ranging in age from 4;2 to 7;6 with mild– Farinella, 2012; Strand & Debertine, 2000; Strand et al., 2006),
moderate to severe CAS. Finally, ReST demonstrated a large although such replication was not directly analyzed here.
effect size for prosodic accuracy (IRD = 0.78) for three par- SCED ratings in the future could be elaborated beyond rep-
ticipants ranging in age from 7;8 to 10;10 with mild to mild– lication for external validity and application of the same
moderate CAS. protocol in direct replication studies (Tate et al., 2013) to also
Generalization effects varied according to the treat- include independence of research groups. Currently, no direct
ment and measures used, and thus a separate effect size was replication studies exist for CAS treatment.
calculated per measure/condition (see Table 5). The motor The remaining articles were classified as suggestive
treatments of Integral Stimulation/DTTC and ReST showed evidence. With future well-controlled investigation, some of
predominantly small effect sizes, with some moderate-to-large these treatments, as well as others excluded from this review,
effects (ranging from IRD = –0.20 to IRD = 0.84). The Inte- will likely emerge as promising options for CAS. Addition-
grated Phonological Awareness Intervention showed a large ally, an extra level of certainty should be considered for future
effect size for percentage of phonemes correct (IRD = 0.80). 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 A critical component of any treatment study is a clear
systematic review of treatment efficacy studies for children definition of the study participants to convince readers that
with CAS. Unlike previous reviews, we considered within- the appropriate population was targeted. When the 2007

Table 4. Omnibus improvement rate differences (IRDs) for preponderant evidence—treatment effects.
Treatment No. of Confidence Omnibus Interpretation

type Treatment approach Article cases in CAS Dxa IRD 95% CI of effect
Motor Integral Stimulation/ Strand and Debertine (2000) 1 1 1.00 [0.97, 1.00] Very large
DTTCb Edeal and Gildersleeve- 2 1 0.98 [0.88, 1.00] Very large
Neumann (2011)
Maas and Farinella (2012) 4c 3 0.18 [0.03, 0.33] Small or questionable
Maas et al. (2012) 4c 2 0.22 [0.08, 0.36] Small or questionable
Overall 0.60 [0.53, 0.67] Moderate
ReST Ballard et al. (2010) 3 1 0.78 [0.54, 1.00] Large (prosody—
PVI duration)
Linguistic Integrated Phonological Moriarty and Gillon (2006) 3 3 1.00 [0.89, 1.00] Very large
Awareness McNeill et al. (2009a) 12 3 0.10 [–0.06, 0.24] Small or questionable
Intervention Overall 0.51 [0.39, 0.58] Moderate
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.
consensus-based diagnostic features (ASHA, 2007) were ap- primarily conveyed on vowels, we counted mention of vowel
plied, only 16.6% achieved a rating of high confidence. How- errors in participants as possible evidence of dysprosody to
ever, most of the studies reviewed were published prior to accommodate older articles. Despite the chance that dys-
2007 and were using common descriptors for their time, now prosody would be overestimated because of this decision (as
considered by many to be nondiscriminative (e.g., ASHA, only a subset of vowel errors or distortions would be indi-
2007; McCabe et al., 1998). The most commonly overlooked cative of stress errors), only 50% of the articles (21 of 42)
CAS characteristic across studies was dysprosody, which reported vowel errors. Furthermore, the high rate of comor-
was not considered a core feature of CAS in many checklists bidity in CAS (41% in this sample) complicates diagnosis and
prior to 2007. As prosody (e.g., lexical or phrasal stress) is could partially account for lower confidence ratings.
Table 5. Omnibus IRDs for preponderant evidence—generalization.
Confidence
Therapy Therapy No. of in CAS Dx Generalization to Interpretation
type approach Article cases (1 = highest) untrained itemsa IRD 95% CI of effect
Motor Integral Stimulation/ Strand and 1 1 NR NR NA NA

DTTCb Debertine
(2000)
Edeal and 2 1 MFF sounds 0.05 [10.47, 0.57] Small or
Gildersleeve- questionable
Neumann (2011) HFF sounds 0.60 [0.18, 1.00] Moderate
Maas and Farinella 4c 3 Blocked practice items 0.04 [–0.16, 0.24] Small or
(2012) questionable
Random practice items 0.20 [0.01, 0.39] Small or
questionable
Maas et al. (2012) 4c 2 100% feedback items –0.15 [–0.13, 0.43] Small or
questionable
60% feedback items 0.03 [–0.14, 0.20] Small or
questionable
ReST Ballard et al. (2010) 3 1 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
Linguistic Integrated Moriarty and Gillon 3 3 NR NR NA NA
Phonological (2006)
Awareness McNeill et al. 12 3 Untrained PPC 0.80 [0.64, 0.88] Large
Intervention (2009a)
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.

Experimental Control due to the hypothesized links between speech motor and
linguistic difficulties in children with CAS (Souza, Payão, &
Experimental control is essential to ensure that treat-
Costa, 2009). However, the studies using combined approaches
ment effects are attributable to the intervention approach
had participants without comorbid CAS and often combined
provided and are an essential element to calculating effect
approaches within the same therapy type (e.g., two motor
sizes for single-subject designs (Olswang & Bain, 1994). Only
treatments). The theoretical motivation for combining such
experimental designs with some experimental control were
treatments is unclear, and no comparisons of single versus
included in the treatment outcome analysis. Experimental
combined treatments for children with clear diagnoses of
control was best demonstrated when SCEDs reported change
CAS are yet available.
in at least one condition beyond baseline levels after with-
drawal of treatment and when control data were used to
estimate improvement due to maturation (Byiers et al., 2012; Treatment Outcomes
Olswang & Bain, 1994; Perdices & Tate, 2009). Not all of
It is well accepted that the strongest evidence for a
the studies analyzed here demonstrated clear experimental
treatment efficacy is demonstration of skill maintenance
control and treatment effects on the basis of these conditions.
beyond the treatment period and generalization of treatment
In future research, CAS SCEDs would ideally use more
effects to related behaviors and/or communication contexts
than two phases (i.e., beyond just baseline and treatment but
(e.g., McReynolds & Kearns, 1983; Schmidt & Lee, 2011).
also withdrawal and other treatment phases), including at
Such effects constitute instrumental clinical change (Olswang
least three data points per phase (including baselines) and
& Bain, 1994, 2013). Few of the 23 studies evaluated reported
replication across cases (Tate et al., 2013).
maintenance of treatment or generalization effects. As
Within this review, 22% (five of 23) of the articles at
such, the robustness of most reported treatment effects over
Level IIb of evidence reported some changes in untreated
time is not known.
speech behaviors, which were hypothesized to be unrelated to
The six approaches that demonstrated maintenance
the treated behaviors. This phenomenon reflects a loss of
and response or stimulus generalization were the three ap-
experimental control and may undermine the claim of posi-
proaches with preponderant evidence (Integral Stimulation/
tive treatment effects. However, it may also reflect the
DTTC, ReST, and Integrated Phonological Awareness
underspecification of theories of speech motor control, in that
Intervention) and the aided AAC modeling and facilitative
there is limited evidence to guide how and why speech
vowel context interventions. Degree of generalization varied,
behaviors are related motorically (Folkins & Bleile, 1990).
seemingly influenced by specific stimuli chosen, incorpora-
This issue has been more fully discussed in studies of treat-
tion of PML, dosage, frequency and intensity of sessions,
ment of acquired apraxia of speech (Ballard, 2001; Ballard,
potential critical thresholds for skill mastery, and participant
Maas, & Robin, 2007). These unexpected generalization
characteristics. Notably, only those participants demon-
outcomes may provide interesting directions for exploring
strating strong gains for treated behaviors tended to maintain
relationships between different speech skills and for guiding
or generalize skills (e.g., Maas & Farinella, 2012; McNeill
the selection of generalization and control stimuli in future
et al., 2009a). The exception to this was the phonological
treatment trials.
treatment within the Integrated Phonological Awareness
Intervention, which showed large generalization effects despite
demonstrating moderate treatment effects. This is an expected
Treatment Approaches outcome in interventions that focus on learning linguistic
The reviewed treatments for CAS can be categorized as rules that can be rapidly generalized across a range of contexts
targeting motor or linguistic skills or using AAC to provide (Gierut, 1998; Gierut & Hulse, 2010) versus learning new
a primary means of communication. The approaches used motor skills. There are at least two possible explanations for
can also be viewed in light of the International Classification why children with probable CAS, a motor speech disorder,
of Functioning, Disability, and Health (ICF; World Health responded to a phonological treatment. First, phonological
Organization, 2002). Motor and linguistic approaches were therapy involves production of speech targets and so pro-
primarily directed at the impairment (body functions/structure) vides practice in planning, programming, and executing the
level of the ICF (McLeod & McCormack, 2007), commonly movements for these targets. Second, the participants had a
addressing articulation, prosody, phonological awareness, or number of phonological processes, and perhaps their con-
expressive language skills. AAC approaches were instead di- comitant phonological disorders were a primary concern for
rected at the activity/participation levels of the ICF, facilitating treatment at this time. These findings warrant thoughtful
communicative effectiveness with a greater emphasis on con- comparison of specific participant characteristics, each inter-
sultation, training, and home practice. The literature suggests vention’s theoretical framework, the stimuli and activities used
that the primary concern in CAS is developing intelligible during intervention, and stimuli used to assess generalization
speech, either through addressing articulatory and prosodic of skill to determine whether there is in fact any benefit in one
accuracy or through improving phonology, although concen- approach over the other.
tration on AAC and expressive language may be required. Likewise, stimulus generalization provides important
The majority of SCEDs intentionally combined treat- information about how skills treated in therapy generalize to
ments, a trend that appears to be increasing. This may be other contexts or situations for improvement in everyday

communication. Only rate control therapy (Rosenthal, 1994) Implications
and AAC approaches considered such generalization. Such
Despite a continued need for well-designed RCTs,
measures should be employed as we move toward Phase III
NRCTs, and additional SCED studies, existing evidence is
and Phase IV studies to demonstrate real change in com-
available to guide clinical practice in the treatment of CAS.
munication skills in children with CAS.
Comparing treatments is a future priority, considering the
It is important to note that, despite most children in the
range of treatments that are available. The treatments best
studies showing positive effects in treatment, all children
suited to inclusion in an RCT or NRCT are those that have
required additional therapy to work on other communica-
demonstrated maintenance and generalization effects. Fur-
tion goals after treatment blocks spanning between nine
ther research is needed for valid and reliable differential
and 195 sessions. This demonstrates that children with CAS
diagnosis of CAS and for understanding which client groups
often require ongoing therapy, spanning many goals and
would benefit from which type of treatment.
needs.
In terms of clinical practice, currently two motor
Further investigation is warranted to explore inter-
treatments (Integral Stimulation/DTTC and ReST) and one
actions between participant variables and the degree of
linguistic treatment (Integrated Phonological Awareness
treatment outcome to help tailor treatments, particularly for
Intervention) are best suited to interim clinical use, with ses-
those who did not demonstrate treatment effects. For exam-
sions at least twice a week and dose above 60 trials per session.
ple, Integral Simulation/DTTC was initially designed for
DTTC appears to work better for clients with more severe
children with severe CAS using functional core vocabulary
CAS, Integrated Phonological Awareness Intervention ap-
stimuli (Strand & Skinder, 1999). Whereas studies addressing
pears to work better for children 4–7 years of age with mild to
severe CAS have reported positive treatment effects across
severe CAS, and ReST appears to work better for children
multiple participants, those addressing mild-to-moderate
7–10 years of age with mild-to-moderate CAS.
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 Limitations
approaches can promote speech gains over and above speech- Because of the developing nature of this literature, this
based interventions (as described in Bornman et al., 2001). systematic review utilized some narrative review methodol-
The current literature suggests that when a child experienced ogy to ensure greater coverage of SCED designs that differed
frustration over low intelligibility or comprehensibility, considerably (Collins & Fauser, 2004). Potential bias was
AAC approaches may increase communicative success as reduced through the use of systematic review principles.
well as stimulate development of language skills that cannot We considered only peer-reviewed published reports
be practiced through speaking. 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 presenta-
Structure of Treatment Delivery tions or dissertations was excluded, thus excluding other
Intensive treatment delivery in impairment-based approaches with potential suggestive evidence, such as Prompts
intervention appears crucial for obtaining positive treatment for Restructuring Oral Muscular Phonetic Targets (Dale &
outcomes. These treatments provided therapy at least 2–3 times Hayden, 2011; now see Dale & Hayden, 2013) or the Nuffield
a week, with sessions of up to 60 min. The dose of treatment, Dyspraxia Programme (Belton, 2006). As in all reviews, there
defined as the “number of properly administrated teaching is a risk that studies with negative treatment outcomes are
episodes during a single intervention session” (Warren et al., underrepresented because of the difficulty publishing such
2007, p. 71), should probably also be high (Edeal & Gildersleeve- studies, potentially risking overestimation of treatment effects.
Neumann, 2011). This review suggests that at least 60 trials Our IRD analysis examined preliminary effect sizes
per session represents a “high” dose. Williams (2012) sug- but did not use moderator variables because of the small
gested that, with phonological therapy for speech sound number of articles (n = 7) and participants (n = 26). Here, we
disorder, ≥50 trials per session over ≥30 sessions is effec- provide some preliminary observations about which treat-
tive, although dose and intensity need to increase as impairments appear useful for different age and severity levels. As
ment severity increases. Further research is indicated to additional studies are published, it is recommended that such
allow reliable estimates of the overall amount of therapy moderator variables be evaluated more fully.
needed, dose, and intensity for CAS, considering age and It is important to note the IRD is calculated and inter-
severity. preted differently to other effect sizes. For example, the recent
SCED studies in this review all delivered treatment DTTC studies (Maas et al., 2012; Maas & Farinella, 2012)
in individual sessions, and 40% utilized home practice to used Beeson and Robey’s (2006) effect size method and found
increase dose and intensity. The high intensity that is indi- large effect sizes for some participants and conditions and
cated would clearly require significant resources. Thus, de- found no effects for others. As the majority of articles did
termining effective treatments with engaging home practice not report means and standard deviations, such effect size
activities should maximize maintenance and generalization calculations were not possible across all preponderant arti-
of skills. cles. The IRDs reported here differ from those reported

in Maas et al. (2012) and Maas and Farinella (2012) in two Alcock, K. J., Passingham, R. E., Watkins, K. E., & Vargha-
ways. First, in our calculation of IRD, only nonoverlapping Khadem, F. (2000). Oral dyspraxia in inherited speech and
data between baseline/withdrawal and treatment phases language impairment and acquired dysphasia. Brain and
Language, 75, 17–33.
were considered indicative of improvement. Second, the
American Speech-Language-Hearing Association. (2004). Evidence-
IRDs here were averaged across participants within studies; based practice in communication disorders: An introduction
thus, greater variability across participants conservatively [Technical report]. Available from www.asha.org/policy
reduced the magnitude of the omnibus IRD. American Speech-Language-Hearing Association. (2007). Childhood
Other aspects that could be investigated were beyond the apraxia of speech [Technical report]. Available from www.asha.
scope of the current study. This includes SCED methodolog- org/policy
ical quality as per reported guidelines (e.g., Tate et al., 2013; Baas, B. S., Strand, E. A., Elmer, L. M., & Barbaresi, W. J. (2008).
see Wendt & Miller, 2012, for a review) and analysis of motor Treatment of severe childhood apraxia of speech in a 12-year-old
learning principles utilized in each treatment (e.g., Bislick, male with CHARGE association. Journal of Medical Speech-
Language Pathology, 16, 181–190.
Weir, Spencer, Kendall, & Yorkston, 2012). We did not eval-
*Ballard, K. J. (2001). Response generalization in apraxia of speech
uate the theoretical stance of each treatment approach, which treatments: Taking another look. Journal of Communication
might have revealed further information as to why some Disorders, 34, 3–20.
treatments generated stronger effects than others. Ballard, K. J., Maas, E., & Robin, D. A. (2007). Treating control of
voicing in apraxia of speech with variable practice. Aphasiology,
21, 1195–1217.
Conclusion Ballard, K. J., Robin, D. A., McCabe, P., & McDonald, J. (2010).
This review identified 23 SCED articles reporting 13 A treatment for dysprosody in childhood apraxia of speech.
treatment approaches classified as primarily motor, linguistic, Journal of Speech, Language, and Hearing Research, 53,
or AAC. Three treatments were judged to have preponderant 1227–1245.
evidence to support their efficacy, indicating that the re- Beeson, P. M., & Robey, R. R. (2006). Evaluating single-subject
treatment research: Lessons learned from the aphasia literature.
ported effects were “probably true”: Integral Stimulation/
Neuropsychological Review, 16, 161–169.
DTTC, ReST, and Integrated Phonological Awareness Belton, E. (2006). Evaluation of the effectiveness of the Nuffield Dys-
Intervention. These treatments had moderate-to-large treat- praxia Programme as a treatment approach for children with severe
ment effects and small-to-large generalization effects, making speech disorders. London, England: University College London.
them strong candidates for Phase III research, comparison Binger, C., Kent-Walsh, J., Berens, J., Del Campo, S., & Rivera, D.
in RCTs, and interim clinical use. Larger scale rigorous RCTs (2008). Teaching Latino parents to support the multi-symbol
and NRCTs are critically needed to compare treatments with message productions of their children who require AAC.
larger sample sizes to potentially inform a greater number Augmentative & Alternative Communication, 24, 323–338.
of clients with CAS (Morgan & Vogel, 2009). Such efforts will Binger, C., & Light, J. (2007). The effect of aided AAC modeling on
the expression of multi-symbol messages by preschoolers who
serve to generate conclusive evidence through well-controlled
use AAC. AAC: Augmentative and Alternative Communication,
experimental designs, including measures of maintenance 23, 30–43. doi:10.1080/07434610600807470
and generalization of treatment effects and manipulation of Binger, C., Maguire-Marshall, M., & Kent-Walsh, J. (2011). Using
treatment intensity and other motor learning principles. This aided AAC models, recasts, and contrastive targets to teach
work will be facilitated by parallel work toward a reliable grammatical morphemes to children who use AAC. Journal
diagnostic system and exploration of participant variables of Speech, Language, and Hearing Research, 54, 160–176.
that influence treatment response. doi:10.1044/1092-4388(2010/09-0163)
Bislick, L. P., Weir, P. C., Spencer, K., Kendall, D., & Yorkston,
K. M. (2012). Do principles of motor learning enhance retention
Acknowledgments and transfer of speech skills? A systematic review. Aphasiology,
This research was supported by the Douglas and Lola Douglas 26, 709–728. doi:10.1080/02687038.2012.676888
Scholarship on Child and Adolescent Health, the Speech Pathology Bornman, J., Alant, E., & Meiring, E. (2001). The use of a digital
Australia Nadia Verrall Memorial Research Grant and Postgraduate voice output device to facilitate language development in a child
Research Award, and the University of Sydney James Kentley with developmental apraxia of speech: A case study. Disability
Memorial Scholarship and Postgraduate Research Support Scheme and Rehabilitation, 23, 623–634.
awarded to the first author. This research was also supported by an Byiers, B. J., Reichle, J., & Symons, F. J. (2012). Single-subject
Australian Research Council Future Fellowship awarded to the experimental design for evidence-based practice. American
third author. We thank Catherine Mason and Donna Thomas for Journal of Speech-Language Pathology, 21, 397–414. doi:10.1044/
assistance coding the data and completing reliability. 1058-0360(2012/11-0036)
Collins, J. A., & Fauser, B. C. J. M. (2004). Balancing the strengths
of systematic and narrative reviews. Human Reproduction
References Update, 11, 103–104.
References marked with an asterisk indicate studies included in the Culp, D. M. (1989). Developmental apraxia and augmentative or
improvement rate difference analysis. alternative communication: A case example. AAC: Augmenta-
tive and Alternative Communication, 5, 27–34. doi:10.1080/
Abramson, J. H. (2011). WINPEPI updated: Computer programs 07434618912331274936
for epidemiologists, and their teaching potential. Epidemiologic Dale, P. S., & Hayden, D. A. (2011, June). The efficacy of PROMPT
Perspectives & Innovations, 8, 1–9. for treating childhood apraxia of speech. Paper presented at

the 6th International Conference on Speech Motor Control, *Maas, E., & Farinella, K. A. (2012). Random versus blocked
Nijmegen-Groningen, the Netherlands. practice in treatment for childhood apraxia of speech. Journal
Dale, P. S., & Hayden, D. A. (2013). Treating speech subsystems in of Speech, Language, and Hearing Research, 55, 561–578.
childhood apraxia of speech with tactual input: The PROMPT doi:10.1044/1092-4388(2011/11-0120)
approach. American Journal of Speech-Language Pathology, Maas, E., Robin, D. A., Austermann Hula, S. N., Freedman, S. E.,
22, 644–661. doi:10.1044/1058-0360(2013/12-0055) Wulf, G., Ballard, K. J., & Schmidt, R. A. (2008). Principles of
*Edeal, D. M., & Gildersleeve-Neumann, C. E. (2011). The impor- motor learning in treatment of motor speech disorders. American
tance of production frequency in therapy for childhood apraxia Journal of Speech-Language Pathology, 17, 277–298. doi:10.1044/
of speech. American Journal of Speech-Language Pathology, 1058-0360(2008/025)
20, 95–110. Maassen, B., Groenen, P., & Crul, T. (2003). Auditory and phonetic
Folkins, J. W., & Bleile, K. M. (1990). Taxonomies in biology, perception of vowels in children with apraxic speech disorders.
phonetics, phonology, and speech motor control. Journal of Clinical Linguistics & Phonetics, 17, 447–467.
Speech and Hearing Disorders, 55, 596–611. Marion, M. J., Sussman, H. M., & Marquardt, T. P. (1993). The
Gierut, J. A. (1998). Treatment efficacy: Functional phonological perception and production of rhyme in normal and develop-
disorders in children. Journal of Speech, Language, and Hearing mentally apraxic children. Journal of Communication Disorders,
Research, 41, S85–S100. 26, 129–160.
Gierut, J. A., & Hulse, L. E. (2010). Evidence-based practice: Martikainen, A.-L., & Korpilahti, P. (2011). Intervention for child-
A matrix for predicting phonological generalization. Clinical hood apraxia of speech: A single-case study. Child Language
Linguistics & Phonetics, 24, 323–334. doi:10.3109/ Teaching and Therapy, 27, 9–20.
02699200903532490 McCabe, P., Rosenthal, J. B., & McLeod, S. (1998). Features of
Gillon, G. T., & Moriarty, B. C. (2007). Childhood apraxia of developmental dyspraxia in the general speech-impaired popu-
speech: Children at risk for persistent reading and spelling lation? Clinical Linguistics & Phonetics, 12, 105–126.
disorder. Seminars in Speech and Language, 28, 48–57. McLeod, S., & McCormack, J. (2007). Application of the ICF and
Groenen, P., Maassen, B., Crul, T., & Thoonen, G. (1996). The ICF-Children and Youth in children with speech impairment.
specific relation between perception and production errors Seminars in Speech & Language, 28, 254–264.
for place of articulation in developmental apraxia of speech. *McNeill, B. C., Gillon, G. T., & Dodd, B. (2009a). Effectiveness of
Journal of Speech and Hearing Research, 39, 468–482. an integrated phonological awareness approach for children with
Hall, P. K. (2000). A letter to the parent(s) of a child with devel- childhood apraxia of speech (CAS). Child Language Teaching
opmental apraxia of speech: Part IV. Treatment of DAS. and Therapy, 25, 341–366.
Language, Speech, and Hearing Services in Schools, 31, 179–181. McNeill, B. C., Gillon, G. T., & Dodd, B. (2009b). A longitudinal
Hall, P. K., Jordan, L. S., & Robin, D. A. (1993). Developmental case study of the effects of an integrated phonological aware-
apraxia of speech: Theory and clinical practice. Austin, TX: Pro-Ed. ness program for identical twin boys with childhood apraxia
Harris, L., Doyle, E. S., & Haaf, R. (1996). Language treatment of speech (CAS). International Journal of Speech-Language
approach for users of AAC: Experimental single-subject inves- Pathology, 11, 482–495.
tigation. Augmentative and Alternative Communication, 12, McNeill, B. C., Gillon, G. T., & Dodd, B. (2010). The longer term
230–243. doi:10.1080/07434619612331277698 effects of an integrated phonological awareness intervention
Hegde, M. N. (2007). A methodological review of randomized for children with childhood apraxia of speech. Asia Pacific
clinical trials. Communicative Disorders Review, 1, 15–36. Journal of Speech, Language & Hearing, 13, 145–161.
Iuzzini, J., & Forrest, K. (2010). Evaluation of a combined treatment McReynolds, L. V., & Kearns, K. P. (1983). Single-subject experi-
approach for childhood apraxia of speech. Clinical Linguistics & mental designs in communicative disorders. Baltimore, MD:
Phonetics, 24, 335–345. University Park Press.
Jacks, A., Marquardt, T. P., & Davis, B. L. (2006). Consonant Moher, D., Liberati, A., Tetzlaff, J., & Altman, D. G. (2009).
and syllable structure patterns in childhood apraxia of speech: Preferred reporting items for systematic reviews and meta-
Developmental change in three children. Journal of Communi- analyses: The PRISMA statement. PLoS Med, 6(7), e1000097.
cation Disorders, 39, 424–441. doi:10.1371/journal.pmed.1000097
Kearns, K. P., & de Riesthal, M. (2013). Applying single-subject Morgan, A. T., & Vogel, A. P. (2008). Intervention for childhood
experimental research to inform clinical practice. In L. A. C. apraxia of speech. Cochrane Database of Systematic Reviews, 16,
Golper & C. Frattali (Eds.), Outcomes in speech-language 1–19. doi:10.1002/14651858.CD006278.pub2
pathology (2nd ed., pp. 279–297). New York, NY: Thieme. Morgan, A. T., & Vogel, A. P. (2009). A Cochrane review of treat-
Krauss, T., & Galloway, H. (1982). Melodic intonation therapy with ment for childhood apraxia of speech. European Journal of
language delayed apraxic children. Journal of Music Therapy, Physical & Rehabilitation Medicine, 45, 103–110.
19, 102–113. *Moriarty, B., & Gillon, G. (2006). Phonological awareness interven-
Lewis, B. A., Freebairn, L. A., Hansen, A., Taylor, H., Iyengar, S., tion for children with childhood apraxia of speech. International
& Shriberg, L. D. (2004). Family pedigrees of children with Journal of Language & Communication Disorders, 41, 713–734.
suspected childhood apraxia of speech. Journal of Communica- Moseley, A. M., Herbert, R. D., Maher, C. G., Sherrington, C., &
tion Disorders, 37, 157–175. doi:10.1016/j.jcomdis.2003.08.003 Elkins, M. R. (2008). PEDro scale can only rate what papers
Lundeborg, I., & McAllister, A. (2007). Treatment with a combi- report. Australian Journal of Physiotherapy, 54(4), 288.
nation of intraoral sensory stimulation and electropalatography doi:10.1016/S0004-9514(08)70017-5
in a child with severe developmental dyspraxia. Logopedics Olswang, L. B., & Bain, B. A. (1994). Data collection: Monitoring
Phoniatrics Vocology, 32, 71–79. children’s treatment progress. American Journal of Speech-
*Maas, E., Butalla, C. E., & Farinella, K. A. (2012). Feedback fre- Language Pathology, 3, 55–66.
quency in treatment for childhood apraxia of speech. American Olswang, L. B., & Bain, B. A. (2013). Treatment research. In L. A.
Journal of Speech-Language Pathology, 21, 239–257. doi:10.1044/ C. Golper & C. Frattali (Eds.), Outcomes in speech-language
1058-0360(2012/11-0119) pathology (2nd ed., pp. 245–264). New York, NY: Thieme.

Ozanne, A. (2005). Childhood apraxia of speech. In B. Dodd (Ed.), Strand, E. A., & Skinder, A. (1999). Treatment of developmental
Differential diagnosis and treatment of children with speech apraxia of speech: Integral stimulation methods. In A. Caruso &
disorder (pp. 71–82). London, England: Whurr. E. Strand (Eds.), Clinical management of motor speech disorders
Pannbacker, M. (1988). Management strategies for developmental in children (pp. 109–148). New York, NY: Thieme.
apraxia of speech: A review of literature. Journal of Communi- Strand, E. A., Stoeckel, R., & Baas, B. S. (2006). Treatment of
cation Disorders, 21, 363–371. severe childhood apraxia of speech: A treatment efficacy study.
Parker, R., Vannest, K., & Brown, L. (2009). The improvement rate Journal of Medical Speech-Language Pathology, 14, 297–307.
difference for single-case research. Exceptional Children, 75, Tate, R. L., Perdices, M., Rosenkoetter, U., Wakim, D., Godbee, K.,
135–150. Togher, L., & McDonald, S. (2013). Revision of a method
Perdices, M., Schultz, R., Tate, R., McDonald, S., Togher, L., quality rating scale for single-case experimental designs and
Savage, S., . . . Smith, K. (2006). The evidence base of neuro- n-of-1 trials: The 15-item Risk of Bias in N-of-1 Trials (RoBiNT)
psychological rehabilitation in acquired brain impairment (ABI): Scale. Neuropsychological Rehabilitation, 23, 619–638.
How good is the research? Brain Impairment, 7, 119–132. doi:10.1080/09602011.2013.824383
Perdices, M., & Tate, R. L. (2009). Single-subject designs as a tool Terband, H., Maassen, B., Guenther, F. H., & Brumberg, J. (2009).
for evidence-based clinical practice: Are they unrecognised and Computational neural modeling of speech motor control in
undervalued? Neuropsychological Rehabilitation, 19, 904–927. childhood apraxia of speech (CAS). Journal of Speech, Language,
doi:10.1080/09602010903040691 and Hearing Research, 52, 1595–1609.
Robey, R. R. (2004). A five-phase model for clinical-outcome Thomson Reuters. (2011). EndNote X5 [Computer software]. New
research. Journal of Communication Disorders, 37, 401–411. York, NY: Author.
Rosenthal, J. B. (1994). Rate control therapy for developmental Wambaugh, J. L., Duffy, J. R., McNeil, M. R., Robin, D. A., &
apraxia of speech. Clinics in Communication Disorders, 4, Rogers, M. A. (2006). Treatment guidelines for acquired apraxia
190–200. of speech: A synthesis and evaluation of the evidence. Journal
Royal College of Speech and Language Therapists. (2011). RCSLT of Medical Speech-Language Pathology, 14, xv–xxxiii.
policy statement. Retrieved from www.ndp3.org/documents/ Warren, S. F., Fey, M. E., & Yoder, P. J. (2007). Differential
rcslt2011dvdPolicyStatement.pdf treatment intensity research: A missing link to creating optimally
Schmidt, R. A., & Lee, T. D. (2011). Motor control and learning: A effective communication interventions. Mental Retardation
behavioral emphasis (5th ed.). Champaign, IL: Human Kinetics. and Developmental Disabilities Research Reviews, 13, 70–77.
Smith, N. L. (1981). The certainty of judgments in health evalua- doi:10.1002/mrdd.20139
tions. Evaluation and Program Planning, 4, 273–278. Watts, C. R. (2009). Lack of randomized controlled trials prohibits
doi:10.1016/0149-7189(81)90028-8 analysis of effectiveness for the treatment of childhood apraxia
Souza, T. N., Payão, M. C., & Costa, R. C. (2009). Childhood of speech: A systematic review of quasi-experimental group
speech apraxia in focus: Theoretical perspectives and present designs and single-subject experimental designs is needed.
tendencies. Pró-fono: Revista de atualização científica, 21, Evidence-Based Communication Assessment and Intervention, 3,
76–80. 8–10. doi:10.1080/17489530902732924
Stackhouse, J., & Snowling, M. (1992). Developmental verbal Wendt, B., & Miller, B. (2012). Quality appraisal of single-subject
dyspraxia: II. A developmental perspective on two case studies. experimental designs: An overview and comparison of differ-
European Journal of Disorders of Communication, 27, 35–54. ent appraisal tools. Education and Treatment of Children, 35,
Stokes, S. F., & Griffiths, R. (2010). The use of facilitative vowel 235–268.
contexts in the treatment of post-alveolar fronting: A case study. Williams, A. L. (2012). Intensity in phonological intervention: Is there
International Journal of Language & Communication Disorders, a prescribed amount? International Journal of Speech-Language
45, 368–380. doi:10.3109/13682820903094737 Pathology, 14, 456–461. doi:10.3109/17549507.2012.68886
*Strand, E. A., & Debertine, P. (2000). The efficacy of integral World Health Organization. (2002). Towards a common language
stimulation intervention with developmental apraxia of speech. for functioning, disability and health: ICF. Geneva, Switzerland:
Journal of Medical Speech-Language Pathology, 8, 295–300. Author.


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A Systematic Review of Treatment

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Murray et al.: Systematic Review of Treatment Outcomes for CAS 487

488 American Journal of Speech-Language Pathology • Vol. 23 • 486–504 • August 2014

Murray et al.: Systematic Review of Treatment Outcomes for CAS 489

Level Primary characteristics Nondiscriminative characteristics Comorbidity

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Murray et al.: Systematic Review of Treatment Outcomes for CAS 491

Intervention name/ Level of Participant Diagnostic

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Intervention name/ Level of Participant Diagnostic

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

Intervention name/ Level of Participant Diagnostic

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Treatment across all participants

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

Treatment across all participants

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Discussion Confidence in CAS Diagnosis

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Treatment No. of Confidence Omnibus Interpretation

Note. CI = confidence interval; PVI = pairwise variability index (acoustic measure).

Table 5. Omnibus IRDs for preponderant evidence—generalization.

Motor Integral Stimulation/ Strand and 1 1 NR NR NA NA

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