maas-2024-treatment-for-childhood-apraxia-of-speech-past-present-and-future

You might also like

Download as pdf or txt
Download as pdf or txt
You are on page 1of 26

Review Article

Treatment for Childhood Apraxia of Speech:


Past, Present, and Future
Edwin Maasa
a
Department of Communication Sciences and Disorders, Temple University, Philadelphia, PA

ARTICLE INFO ABSTRACT


Article History: Purpose: The purposes of this review article were to provide an introduction to
Received April 7, 2023 and “bird’s-eye” overview of the current evidence base for treatment of child-
Revision received September 15, 2023 hood apraxia of speech (CAS), identify some gaps and trends in this rapidly
Accepted March 21, 2024 growing literature, and formulate some future research directions, in order to
advance the evidence base and clinical practice for children with CAS.
Editor-in-Chief: Cara E. Stepp Method: Following a brief introduction outlining important concepts, a narrative
Editor: Maria Grigos review of the CAS treatment literature is provided, and trends and future direc-
tions are identified based on this review. The review is organized around four
https://doi.org/10.1044/2024_JSLHR-23-00233 fundamental treatment research questions: (a) “Does Treatment X work?”, (b)
“Does Treatment X work better than Treatment Y?”, (c) “For whom does Treat-
ment X work?”, and (d) “What does ‘work’ mean, anyway?”
Results: A wide range of CAS treatments with varying degrees of evidence for
efficacy exists. Research is beginning to emerge that compares different treat-
ments and seeks to determine optimal treatment parameters. Few studies to
date have explored child-level predictors of treatment response, and the evi-
dence base currently is limited in scope with respect to populations and out-
comes studied.
Conclusions: A growing evidence base supports the efficacy of a number of
treatments for CAS. However, many important gaps in the literature were identi-
fied that warrant redoubled and sustained research attention. Research is
beginning to emerge that addresses treatment optimization, comparison, candi-
dacy, and outcomes. Suggestions for future research are offered, and the con-
cept of a hypothesized pathway was applied to CAS to illustrate how compo-
nents of an intervention can effect change in a clinical goal and can help guide
development and refinement of treatments for children with CAS.

At the time of the first Apraxia Kids Research Sym- research agenda. Since then, and since the initiation of
posium (AKRS) in 2002, no cohesive research community Apraxia Kids’ annual Research Grant competition in 2007,
existed dedicated to the study of childhood apraxia of the CAS treatment literature has grown rapidly, as illus-
speech (CAS). That first research symposium, and Apraxia trated by a simple PubMed search with the search term
Kids (then known as the Childhood Apraxia of Speech “childhood apraxia of speech treatment” (see Figure 1).
Association of North America) more generally, was instru-
mental in bringing together scientists with research interests
in or related to CAS and identifying priorities for a Purpose

This review article, based in part on an invited presen-


Correspondence to Edwin Maas: emaas@temple.edu. Publisher Note: tation at AKRS 2022, overviews the current state of CAS
This article is part of the Special Issue: Selected Papers From the treatment research evidence. While significant advances
2022 Apraxia Kids Research Symposium. Disclosure: The author have been made in other areas as well, including diagnostics
serves on the Professional Advisory Council of Apraxia Kids and has
received grant funding from Apraxia Kids in the past. The author has
(e.g., Murray et al., 2021) and genetics (e.g., Kaspi et al.,
declared that no other competing financial or nonfinancial interests 2023), the focus of this review article is specifically on CAS
existed at the time of publication. treatment research. The overall goal is to encourage further

Journal of Speech, Language, and Hearing Research  1–26  Copyright © 2024 The Author 1
This work is licensed under a Creative Commons Attribution-NonCommercial 4.0 International License.
Downloaded from: https://pubs.asha.org 5.210.215.88 on 06/08/2024, Terms of Use: https://pubs.asha.org/pubs/rights_and_permissions
Figure 1. Number of peer-reviewed articles published on the topic of childhood apraxia of speech (CAS) treatment since 1990 (source:
PubMed; search term “childhood apraxia of speech treatment”). Key events that stimulated CAS treatment research are indicated on this
timeline. AKRS = Apraxia Kids Research Symposium; ASHA = American Speech-Language-Hearing Association.

treatment research and thus optimize clinical practice and big-picture, “bird’s-eye level” perspective of the current
outcomes for children with CAS. As a record of AKRS evidence base and trends. In the spirit of the AKRS, occa-
2022, this review article serves as a mile marker and sign- sional references to presentations at the 2022 symposium
post along the road of CAS treatment research and has the and other recent conferences are included to highlight
following specific purposes: emerging developments and help readers anticipate publi-
cations in the near future by identifying research groups
1. To introduce basic concepts of clinical trials for
engaged in cutting-edge CAS treatment research.
those in the CAS community who are less familiar
with such research. This introduction will help The literature review is structured along four main
appreciate the overview to follow. treatment research questions, each involving different
research designs and approaches, and each related to an
2. To review and synthesize the CAS treatment litera-
element of the PICO1 question format: (a) “Does Treat-
ture and identify gaps and trends.
ment X work?”, (b) “Does Treatment X work better than
3. To formulate some possible future directions for the Treatment Y?”, (c) “For whom does Treatment X
CAS treatment research agenda. work?”, (d) “What does ‘work’ mean, anyway?” This
organization is for expository purposes and does not
Scope and Organization imply independence. Clearly, the question of whether a
treatment works is intertwined with the question of for
An important caveat is in order at the outset: An whom it works, and the question of what “work” means
overview and synthesis of the by-now sizable and complex is integral to all others. Moreover, undoubtedly, readers
CAS treatment research literature required some selection will identify additional gaps and future directions. Never-
and structure. Therefore, although an attempt was made theless, the hope is that this review and structure
to offer broad coverage, this is a narrative rather than a
systematic or scoping review (see Murray et al., 2014, for 1
PICO: P = Population; I = Intervention; C = Comparison (no or
a systematic review). As such, the review is not exhaustive other intervention); O = Outcome (e.g., For children with CAS [P],
and does not discuss studies in detail but rather provides a does Treatment X [I] or Treatment Y [C] lead to better outcomes [O]?)

2 Journal of Speech, Language, and Hearing Research  1–26

Downloaded from: https://pubs.asha.org 5.210.215.88 on 06/08/2024, Terms of Use: https://pubs.asha.org/pubs/rights_and_permissions


provides a useful foundation for the next decade of CAS 2 can support claims of treatment effect and efficacy (i.e.,
treatment research. claims of causality) because study designs at these levels
involve systematic manipulation of conditions, often includ-
The remainder of the review article is organized as
ing a control condition. In contrast, Level 3 designs can only
follows. First, fundamental concepts related to clinical tri-
document change (positive or negative) from before to after
als research are introduced to help the reader interpret the
treatment, but not treatment effects (change attributable to
evidence base. Next, the CAS treatment research literature
treatment). Thus, despite a focus on individual participants
is surveyed, in subsections by the four treatment research
and the word “case” in the name, there is an important dis-
questions. Each subsection is divided into a review of past
tinction between case studies (uncontrolled) and SCEDs
and present literature as well as directions for future
(controlled) in terms of claims that can be supported.
research. Finally, the review article concludes with a brief
summary and conclusions. Evidence hierarchies are not without criticism (e.g.,
J. C. Rosenbek, 2016; Roulstone, 2011), and it is impor-
tant to note the qualifier “well-designed.” For example,
Clinical Trials Research: evidence from a well-designed SCED may be stronger
Important Concepts than evidence from a poorly designed RCT, and not all
RCTs (or SCEDs) are equally rigorous methodologically.
Study Designs and Levels of Evidence In concurrence with J. C. Rosenbek (2016), absence of
RCTs does not constitute absence of evidence, and even
Not all evidence is created equal: Some study expert opinion (lowest level of evidence) can be an impor-
designs can provide stronger evidence than others. tant source of evidence. Nevertheless, all else being equal
Although different evidence hierarchies exist, varying in (e.g., well-controlled design), Level 1 evidence is stronger
details, there is a general agreement about the ranking of than evidence from Level 2, Level 2 evidence is stronger
main study designs (e.g., American Speech-Language- than evidence from Level 3, and so forth, and controlled
Hearing Association [ASHA], 2004; Murad et al., 2016; evidence should be given greater weight than expert opin-
OCEBM Levels of Evidence Working Group, 2011). This ion (Dollaghan, 2004).
review article follows the evidence hierarchy used by
ASHA (see Table 1), in which evidence from well-
Phases of Clinical Trials
controlled meta-analyses of multiple randomized con-
trolled trials (RCTs) represents the highest level of evi-
Developing effective treatments takes time, money,
dence (Level 1a), followed by evidence from a single well-
and other resources that are often limited. To make judi-
controlled RCT (Level 1b). Level 2 includes well-designed
cious use of limited resources, development and testing of
quasi-experimental designs, such as group designs with
a treatment typically proceeds in phases with different
nonrandom group assignment and single-case experimen-
intermediate goals, increasing resource demands, and
tal designs (SCEDs). Next are nonexperimental designs
greater methodological rigor (e.g., stronger designs, larger
such as case studies and correlational studies (e.g., one-
sample sizes). As with evidence hierarchies, there are
group pre–post group designs), followed by other uncon-
slightly different conceptualizations of these phases across
trolled and/or unsystematic data (e.g., expert opinion).
and within health domains (e.g., Czajkowski et al., 2015;
Such hierarchies are important to keep in mind Fey & Finestack, 2009; Robey, 2004). Although not
when evaluating the evidence base and level of support for intended to be rigidly prescriptive, and although some
or against a given treatment. Evidence from Levels 1 and studies may address goals of multiple phases, a general

Table 1. Levels of evidence for treatment research.

General description Level Study design categories


True experimental designs 1a Well-designed meta-analysis of > 1 RCT
1b Well-designed RCT
Quasi-experimental designs 2a Well-designed controlled study without randomization
2b Well-designed SCED
Nonexperimental designs 3 Well-designed correlational and case studies
No controlled, systematic data 4 Expert committee report, consensus conference,
clinical experience of respected authorities
Note. © American Speech-Language-Hearing Association (2004), used and adapted with permission (original adaptation from the Scottish
Intercollegiate Guideline Network; https://www.sign.ac.uk/). RCT = randomized controlled trial; SCED = single-case experimental design.

Maas: CAS Treatment Review 3


Downloaded from: https://pubs.asha.org 5.210.215.88 on 06/08/2024, Terms of Use: https://pubs.asha.org/pubs/rights_and_permissions
implication is that conducting a later-phase study is con- an intervention’s definitive efficacy under optimal circum-
tingent on achieving the goals in the preceding phase (e.g., stances, applied to the population defined in Phase II, and
Bergþórsdóttir & Ingham, 2017). Furthermore, consider- using the protocol and measures developed in Phase II.
ation of clinical trial phases places the evidence base in a Study designs typically include quasi- or true experimental
larger, longer-term context and clarifies goals for the group designs with relatively large samples, and statistical
future. This section largely follows Robey’s (2004) concep- testing is stringent to minimize Type I error. Comparison
tualization of phases. conditions typically involve an alternative treatment, often
“standard care” (treatment received in regular clinical
In Phase I, the main goal is to establish the potential
practice) or “standard of care” (the current gold standard
benefit of the intervention and detect a treatment effect
treatment, if there is one).
(referred to here as “initial efficacy”). Study designs may
include case studies, SCEDs, and small2 group designs, Once definitive efficacy has been demonstrated,
where the comparison condition may be absence of treat- determining the effectiveness of the intervention is the
ment. Outcomes measurement may be limited to immedi- main goal in Phase IV. Effectiveness refers to the likeli-
ate posttreatment time points with no longer-term follow- hood of a benefit under routine (i.e., less-controlled) clini-
up, and statistical testing may be limited or relatively cal circumstances. This may involve expanding the popu-
lenient (i.e., greater tolerance for Type II error) in order lation criteria, service delivery methods, dosages, and so
to avoid prematurely abandoning the treatment. Phase I forth. Whereas the main concern in Phases I–III is inter-
studies also provide estimates of the likely effect size, nal validity, the main concern in Phase IV is external
which inform design and sample size for subsequent stud- validity. Phase IV studies may involve pre–post group
ies. An additional goal in Phase I is to develop a hypothe- designs with or without comparison groups, and statistical
sized pathway that specifies the mechanism of action by testing is stringent.
which the treatment is thought to exert its effect on the
outcome of interest (Czajkowski et al., 2015; Fridriksson Finally, Phase V involves determining the efficiency
et al., 2022; see example in Figure 2). An explicitly formu- of the intervention, for example, by considering the bene-
lated pathway can help to refine and improve the treatment fits relative to the costs associated with the treatment
by identifying factors that facilitate (“active ingredients”), (cost–benefit analysis). Phase V research typically involves
hinder (“counteractive ingredients”), or do not affect other disciplines such as health economics and is impor-
(“inactive ingredients”) improvement. A final important tant for informing and directing public policy, insurance
Phase I goal is to establish the safety of the intervention. coverage, and so forth.
Although not typically considered in our field, there may Although the phase of a study is not always clear
be potential negative side effects of speech therapy (see also (some studies address goals from multiple phases), a
the What Does “Work” Mean, Anyway? section below). framework that systematically outlines phases and their
Assuming a positive or promising effect, research goals can help make optimal use of limited resources. For
would progress to Phase II, where the main goal is to instance, without evidence of a possible benefit (Phase I)
determine the intervention’s “preliminary efficacy” and and clear effect size estimates (Phase II), it makes little
prepare for a Phase III study of “definitive efficacy.” sense to invest the considerable resources involved in con-
Phase II studies seek to operationalize outcome measures, ducting a large-scale Phase III RCT of definitive efficacy.
define the target population and optimal dosage, and Similarly, studying effectiveness of a treatment without
finalize a standardized treatment protocol. Phase II studies established efficacy makes little sense: If a treatment does
also provide data to establish likely effect sizes to calculate not work under optimal conditions, it is unlikely to work in
sample sizes for Phase III studies. In Phase II, longer-term a routine clinical environment with more variability (in
maintenance of effects may also be assessed. Study designs fidelity of delivery, client characteristics, etc.). Thus,
may involve SCEDs, small pre–post group designs, and although the phase of a study is not always clear, this
small quasi- or true experimental group designs, and sta- framework provides a “roadmap” to systematically develop,
tistical testing becomes more rigorous (i.e., less tolerance refine, and test interventions with progressively greater rigor
for Type II error). Comparison conditions may involve a and impact.
sham treatment or an alternative treatment.
If findings in Phase II are positive, a Phase III study
is warranted. The goal of Phase III studies is to determine
CAS Treatment Research
This section provides an overview and synthesis of
2
For the purpose of this article, “small” group studies are operation- the CAS treatment literature in order to situate our
ally defined as those with N < 15. knowledge in the present and chart directions for the

4 Journal of Speech, Language, and Hearing Research  1–26

Downloaded from: https://pubs.asha.org 5.210.215.88 on 06/08/2024, Terms of Use: https://pubs.asha.org/pubs/rights_and_permissions


Figure 2. Example of a hypothesized pathway of how a treatment effects change in a clinical goal (after Czajkowski et al., 2015). In this
example, the goal is speech accuracy as measured on an overall rating scale (e.g., Strand et al., 2006). Barriers are factors that, when lim-
ited, affect the clinical goal. In this example, barriers include prosodic, consonant, and vowel accuracy. These factors are, in turn, affected
or supported by underlying knowledge and skills (e.g., speech motor planning skills, phoneme representations) that may be targeted in treat-
ment. The treatment focuses on underlying skills thought to be the primary source of the barrier for the clinical goal. In the example, integral
stimulation treatment elements are designed to target speech motor planning and sensory feedback processing (the presumed impairment
in childhood apraxia of speech), as indicated by black arrows. The gray text and arrows indicate additional underlying skills and knowledge
that may affect the barriers but that are not directly targeted by the treatment. This example could be expanded with further details about
which elements of the treatment target which specific focus (e.g., speech motor planning, sensory feedback processing) to make explicit the
mechanisms of action and identify specific active ingredients.

future. As will become clear, the road has been long, and as the likely importance of practice in naturalistic set-
while there is much road yet ahead of us, it is important tings (M. E. Morley, 1969) and emphasizing movement
to appreciate how far we have already come. sequences and visual cues (J. Rosenbek et al., 1974; Yoss
& Darley, 1974). Of course, these authors recognized that
A Brief Historical Context: The Early Days their recommendations were necessarily tentative and
were offered “not so much as answers to the sometimes
In the early days of CAS treatment (arbitrarily cho- perplexing questions of apraxia therapeutics but as devel-
sen here as pre-1990), important initial guiding light in the opmental milestones so that we can look back one day
darkness involved case reports and clinical expert opinion and see how far we have come” (J. C. Rosenbek &
by such luminaries as Muriel Morley (M. Morley et al., Wertz, 1976, p. 197). Correspondingly, there was recogni-
1955; M. E. Morley, 1969), Jay Rosenbek (J. Rosenbek tion of the need for further research, as expressed clearly
et al., 1974; J. C. Rosenbek & Wertz, 1972), and Kathe by J. C. Rosenbek and Wertz (1972, p. 32): “Results of
Yoss (Yoss & Darley, 1974). These researchers made rec- experimental therapy with these children are badly needed.”
ommendations based on their own and others’ clinical And so the stage was set. Fast forward to the present day
experience, evidence from adults with apraxia of speech, for a look at how far we have come and what our next
and case reports (i.e., mostly Level 4 evidence) for a pop- milestones for CAS treatment development may be.
ulation that back then was even less well understood
than it is today. It is not surprising that some recommen- Does Treatment X Work?
dations and opinions may no longer be considered Approaches and Evidence
appropriate, such as suggestions to begin by targeting
“production of one phonetic sound in isolation” (M. E. This first question is perhaps the most basic and the
Morley, 1969, p. 161), the pessimistic notion that one most commonly addressed in treatment research: Does
“attempts to teach a child to speak will produce little Treatment X work? This section first provides a brief narra-
result” (M. Morley et al., 1955, p. 467), and recommen- tive review of CAS treatment approaches (“Treatments X”)
dations that “compensatory behaviors such as equal and the current evidence base, followed by discussion of
stress and the use of pauses to break up phrases and sen- some emerging trends and directions for future research.
tences may be appropriate” (J. C. Rosenbek & Wertz,
1972, p. 32) or to begin therapy “with mirror work and The Present
all kinds of tongue and lip movements” (Yoss & Darley,
1974, p. 30). Rather, it is perhaps surprising how pre- Today, there are quite a few specific intervention
scient these authors were in their recommendations, such approaches designed specifically for children with CAS

Maas: CAS Treatment Review 5


Downloaded from: https://pubs.asha.org 5.210.215.88 on 06/08/2024, Terms of Use: https://pubs.asha.org/pubs/rights_and_permissions
(see Maas et al., 2014; Murray et al., 2014, for reviews), stimulation treatment represents Phase I and II studies,
with varying degrees of evidence. Increased understanding mostly involving SCED studies (Level 2b). There are at
of speech production, development, and CAS in recent least 10 SCED studies (total N = 29 children with CAS),
years has fostered development of theoretically informed with replication across independent research groups
treatments. Most CAS interventions target aspects of (Baas et al., 2008; Edeal & Gildersleeve-Neumann, 2011;
speech motor planning, with others focusing on language Gildersleeve-Neumann & Goldstein, 2015; Leonhartsberger
aspects or, more broadly, on communication using aug- et al., 2022; Lim et al., 2019; Maas & Farinella, 2012;
mentative and alternative communication (AAC) systems Maas et al., 2012, 2019; Strand & Debertine, 2000; Strand
(Murray et al., 2014). Most studies represent Phase I or II et al., 2006). These studies support the preliminary efficacy
studies (Murray et al., 2014), but Phase III studies are of integral stimulation treatment in terms of speech accu-
beginning to appear as well (e.g., Murray et al., 2015). racy, with retention shown up to 2 months posttreatment
Study designs have become more rigorous, with more con- (Maas et al., 2019). No Phase III studies have yet been
trolled SCEDs and group designs. Most studies are Level published, but several RCTs are underway (Iuzzini-Seigel
2 (mostly SCEDs), but Level 1 studies (RCTs) are also et al., 2023; Maas, Caspari, Beiting, Gildersleeve-Neumann,
emerging (McCabe et al., 2023; Murray et al., 2015; Stoeckel, & Wu, 2022; McCabe et al., 2022). Integral stim-
Namasivayam et al., 2015). The overview below does not ulation approaches have mostly been studied in children
provide a detailed critical appraisal of the evidence but 4–12 years old, although recent work documented applica-
broadly characterizes the nature and extent of the evi- tion to children as young as 2;5 (years;months; Grigos
dence base. et al., 2023).
Approaches targeting syllable sequencing and pros-
Motor-Based Approaches ody in the context of multisyllabic nonwords have
Motor-based interventions represent the majority of emerged in recent years as promising alternatives with
approaches for CAS, and there are many different spe- a relatively strong and growing evidence base. As with
cific treatments, including integral stimulation-based integral stimulation, there are different named approaches,
approaches (e.g., Maas et al., 2012; Strand, 2020), syllable including Rapid Syllable Transition treatment (ReST;
sequencing approaches (e.g., Ballard et al., 2010; H. E. Murray et al., 2015) and Treatment for Establishing
Miller et al., 2021), Prompts for Restructuring Oral Muscu- Motor Program Organization (TEMPO; H. E. Miller
lar Phonetic Targets (PROMPT; e.g., Chumpelik, 1984; et al., 2021). ReST and TEMPO share many features,
Dale & Hayden, 2013), ultrasound biofeedback-based treat- including targeting of multisyllabic nonwords to empha-
ment (e.g., Preston et al., 2013, 2017), the Nuffield Dys- size sequencing, prosody, and fluency of speech move-
praxia Programme (McKechnie et al., 2020; Williams & ments; a large number of attempts per session; and incor-
Stephens, 2010), Melodic Intonation Therapy (Helfrich- poration of principles of motor learning. Evidence to date
Miller, 1994; LaGasse, 2012), the Kaufman Speech to Lan- comes from at least nine studies (total N = 56 children
guage Protocol (Gomez et al., 2018; Kaufman, 2013), the with CAS; Ballard et al., 2010; McCabe et al., 2014, 2023;
Speech Motor Learning program (van der Merwe & Steyn, H. E. Miller et al., 2021; Murray et al., 2015; Scarcella
2018), auditory–motor integration treatment (Rvachew & et al., 2021; D. C. Thomas et al., 2014, 2016, 2018). Stud-
Matthews, 2019), and Concurrent Treatment (Skelton & ies primarily involve SCED studies (Level 2b) representing
Hagopian, 2014). The most prominent approaches to date Phases I and II, although there are also several small-scale
are integral stimulation approaches, syllable sequencing– RCT design studies (Level 1b) representing Phase II or III
oriented approaches, ultrasound biofeedback approaches, (McCabe et al., 2023; H. E. Miller et al., 2021; Murray
and PROMPT (Maas et al., 2014). et al., 2015). Evidence from these studies supports the pre-
liminary efficacy of these approaches with respect to speech
Integral stimulation approaches are among the most
accuracy, with generalization to untrained multisyllabic real
replicated, although there are different versions, including
words and retention demonstrated up to 4 months post-
Dynamic Temporal and Tactile Cueing (DTTC; Strand,
treatment (e.g., Murray et al., 2015). Similar to integral
2020), Apraxia of Speech Systematic Integral Stimulation
stimulation, these syllable sequencing approaches have been
Treatment (ASSIST; Maas et al., 2019), and unnamed
studied primarily in children between 4 and 12 years old
approaches (Edeal & Gildersleeve-Neumann, 2011). Despite
(McCabe et al., 2020).
some differences, these approaches all involve integral stim-
ulation as a cueing or elicitation method (“Watch me, listen Ultrasound biofeedback treatment involves speech
to me, say what I say”; Milisen, 1954), focus on speech motor practice with visual feedback about tongue position
movement accuracy of words or phrases, elicit many pro- and movement (e.g., Preston et al., 2013) and thus mainly
duction attempts, and incorporate principles of motor focuses on articulatory placement rather than prosody or
learning (Maas et al., 2008). The evidence for integral sequencing. The rationale is that children with CAS may

6 Journal of Speech, Language, and Hearing Research  1–26

Downloaded from: https://pubs.asha.org 5.210.215.88 on 06/08/2024, Terms of Use: https://pubs.asha.org/pubs/rights_and_permissions


be less able to use auditory or somatosensory feedback to noted that CAS diagnostic criteria differ from those used in
establish or refine speech motor plans (e.g., Terband most other studies. Retention has been shown up to
et al., 2014). Feedback via the intact visual modality may 6 months in a follow-up study (McNeill et al., 2010) for
circumvent such difficulties to support speech motor learn- the children in McNeill et al.’s (2009b) study. IPA treat-
ing. Evidence comes from at least five studies (total N = ment has been studied primarily in children between 4 and
25 children with CAS), including a case series (Preston, 7 years of age (e.g., McNeill et al., 2009b).
Leece, & Maas, 2016), three SCED studies (Preston et al.,
Finally, AAC interventions for CAS focus on com-
2013, 2017; Preston, Maas, et al., 2016), and two RCTs
munication rather than speech specifically, to provide chil-
(McCabe et al., 2023; Preston et al., 2023), representing
dren with CAS a means to communicate as part of an
Phases I and II. Although not all children responded
approach that also includes speech therapy (Binger, 2007).
(Preston, Maas, et al., 2016), the weight of evidence sup-
Beyond expert opinion (Binger, 2007), the evidence base
ports the preliminary efficacy of biofeedback treatment
includes five SCED studies (total N = 8; Binger & Light,
for CAS, with retention shown up to 2 months posttreat-
2007; Binger et al., 2008, 2011, 2017; Kent-Walsh et al.,
ment (Preston et al., 2017). Ultrasound treatment for CAS
2015) representing Phase I. Results to date support the ini-
has been studied primarily with children ages 9–17 years
tial efficacy of AAC methods for CAS, although it should
(e.g., Preston et al., 2023).
be noted that the diagnosis of CAS was not always clear
PROMPT, an approach that focuses on speech (Murray et al., 2014) and outcome measures varied across
movement accuracy, was developed in the 1980s (Chum- studies (e.g., production of yes/no questions, different gram-
pelik, 1984). It involves speech practice and tactile cues, matical morphemes). AAC for CAS has been studied pri-
with a systematic progression from a focus on respiratory marily in 3- to 4-year-old children (e.g., Binger et al., 2017).
control to prosody. With respect to CAS, the evidence
base to date consists of three Level 3 studies, including Summary
two case studies (Chumpelik, 1984; Grigos et al., 2010) Taking stock of the current evidence base, it is
and a pre-experimental (one-group pre/post) design study encouraging to see the increase in both the quantity and
(Kadis et al., 2014), as well as a Level 2b SCED study quality of CAS treatment research. There are now many
(Dale & Hayden, 2013) and a Level 1b RCT study approaches (not all reviewed above), with varying but
(Namasivayam et al., 2015). Evidence from these two con- growing evidence from SCED studies and even some
trolled studies (total N = 41 children with CAS) supports RCTs, representing mostly Phase I and II stages of devel-
the preliminary efficacy of PROMPT for CAS, with reten- opment. Most studies focus on motor-based approaches,
tion demonstrated up to 3 months posttreatment (Dale & of which different types exist. Although different in
Hayden, 2013). PROMPT for CAS has been studied pri- details, these approaches share several key features such
marily in children under 5 years old (as young as 2;8; as a focus on speech movements and sequencing, incorpo-
Namasivayam et al., 2015). ration of principles of motor learning, use of multimodal
cueing and feedback, and grounding intervention within
Other Approaches theories of speech production and CAS. Despite these
Beyond motor-based treatments, there are interven- encouraging developments, much remains to be done.
tions that focus on other aspects of communication. One With this backdrop, the next section sketches some direc-
example is Integrated Phonological Awareness (IPA) treat- tions for future development and study of interventions
ment (Moriarty & Gillon, 2006), a linguistic approach for CAS.
with a combined focus on speech production accuracy and
phonological awareness, based on the observation that The Future
children with CAS are at an elevated risk for difficulties
with literacy and reading (Lewis et al., 2004; G. J. Miller One obvious direction for future research is to fur-
et al., 2019). IPA treatment focuses on consistent error ther refine existing approaches and study their effects in
patterns and combines phonological awareness activities more rigorous study designs with larger samples, to align
(e.g., separating, blending sounds) with production of with a move toward later clinical trial phases. Further
treatment words. The main focus in terms of speech pro- refinement of existing approaches includes identifying and
duction is on phonological structure (e.g., omission errors), enhancing active ingredients as well as identifying and
with no attention paid to distortions or errors of place or eliminating counteractive or inactive ingredients. This will
manner of articulation. Evidence comes from two SCED involve further Phase I and II studies to “tweak” the pro-
studies (total N = 15; McNeill et al., 2009b; Moriarty & tocol in preparation for Phase III tests of definitive effi-
Gillon, 2006) and supports initial efficacy, with improve- cacy of a fixed protocol. Phase III studies will likely
ment in both literacy and speech production. It should be require multisite RCT studies (Iuzzini-Seigel et al., 2023;

Maas: CAS Treatment Review 7


Downloaded from: https://pubs.asha.org 5.210.215.88 on 06/08/2024, Terms of Use: https://pubs.asha.org/pubs/rights_and_permissions
McCabe et al., 2022) to achieve sufficiently large sample 2010; n = 13 per group), a standard care intervention in
sizes, given the relatively low prevalence of CAS. Further Australia. Murray et al. reported greater improvement in
into the future, Phase IV studies of routine clinical prac- speech accuracy for NDP3 immediately posttreatment but
tice effectiveness are needed, which will require consider- greater improvement for ReST 4 months posttreatment.
ation of practice barriers and opportunities as well as These findings illustrate the importance of comparative
pragmatic clinical trial designs (cf. Ford & Norrie, 2016). effectiveness trials because longer-term and maintained
gains are more desirable. Without well-conducted and sys-
In addition to refining existing approaches, the field
tematic comparison studies on meaningful and long-term
is likely to witness development of novel approaches,
outcomes, speech-language pathologists (SLPs) and chil-
based on new insights from basic science; address practical
dren and their families lack the information necessary for
constraints; or take advantage of practical opportunities.
optimal decision making.
Examples, some of which already in development, include
combining behavioral intervention with neurostimulation More recently, McCabe et al. (2023) compared
techniques (e.g., transcranial direct current stimulation; ReST with ultrasound biofeedback treatment in a small
Mehta & Moorer, 2022; Nakamura-Palacios et al., 2024), pilot RCT (n = 7 per group). Findings showed compara-
preventative interventions in prelinguistic children at risk ble improvements, although the lack of a control group
for CAS (Peter et al., 2019, 2022), and interprofessional made it difficult to rule out maturation. In addition, the
intervention (e.g., speech and music therapy; van Tellingen randomization of a small sample resulted in some non-
et al., 2023). equivalence at baseline. Nevertheless, these findings are
promising in beginning to tackle the need for comparing
Does Treatment X Work Better Than different treatments.
Treatment Y? Treatment Optimization This relative dearth of comparative effectiveness tri-
als of treatment packages makes sense in light of the clini-
The second question is closely related to the first
cal trial phases, according to which an intervention is first
one: Does Treatment X work better than Treatment Y?
tested against a no-treatment condition or a sham treat-
Beyond comparing overall treatment packages with fixed
ment to determine the presence of an effect (Phases I and
protocols (e.g., DTTC vs. ReST) in Phase III studies, this
II). Once an intervention has been shown to produce a
question also encompasses comparisons between different
benefit compared to no treatment or sham treatment,
versions of a given treatment in order to optimize a treat-
more stringent tests and comparisons with other effica-
ment protocol (i.e., part of Phase I and II studies). This
cious interventions are warranted. As most treatments are
section first reviews efforts to compare (versions of) treat-
in Phase I and II development, few are ready for compar-
ments and then sketches some directions for future
ative effectiveness trials, which require larger sample sizes
research. For simplicity of exposition, this discussion is
due to the smaller effect sizes when compared to other
limited to comparisons between (versions of) treatments
active interventions (vs. when compared to no treatment).
for children with comparable profiles. It is taken as a
given that some treatments are likely better suited to some
children than others (e.g., ultrasound biofeedback may be Treatment Optimization
more appropriate for children with specific sounds in Given that most treatments are in Phase I and II
error); however, it is beyond the scope of this review arti- development, a number of studies have focused on factors
cle to discuss the many intersections of child characteris- that may enhance outcomes within the context of a given
tics and different treatments. Instead, the focus here is on treatment. Two main areas of focus are (a) principles of
comparing (versions of) treatments for children who are, motor learning and (b) service delivery models.
in principle, suitable candidates for multiple treatments Principles of motor learning. Principles of motor
(e.g., Murray et al., 2015; see the For Whom Does Treat- learning refer to relatively predictable learning benefits of
ment X Work? section for further discussion). some practice or feedback conditions compared to other
practice/feedback conditions (Maas et al., 2008). Several
The Present principles have been examined in CAS treatment to date,
mainly in SCED studies, including practice amount (more
Treatment Comparisons vs. less; Edeal & Gildersleeve-Neumann, 2011; Maas
To date, very few studies have directly compared et al., 2019; Namasivayam et al., 2015), variability (con-
overall treatment packages. Two exceptions are the studies stant vs. variable; Preston et al., 2017), distribution
by McCabe et al. (2023) and Murray et al. (2015). Murray (massed vs. distributed; Leonhartsberger et al., 2022;
et al. compared ReST with the Nuffield Dyspraxia Maas et al., 2019; Namasivayam et al., 2015; Preston
Programme–Third Edition (NDP3; Williams & Stephens, et al., 2023; D. C. Thomas et al., 2014), schedule (random

8 Journal of Speech, Language, and Hearing Research  1–26

Downloaded from: https://pubs.asha.org 5.210.215.88 on 06/08/2024, Terms of Use: https://pubs.asha.org/pubs/rights_and_permissions


vs. blocked; Maas & Farinella, 2012), feedback type compatible with, and effective in, common clinical realities
(knowledge of results vs. knowledge of performance; (e.g., Iuzzini-Seigel et al., 2023; McCabe et al., 2022).
McKechnie et al., 2020), and frequency (100% vs. 60%;
Maas et al., 2012). These studies reveal that, despite some The Future
individual variability and lack of differences in some
cases, effects of most principles generally align with the Treatment Comparisons
motor learning literature. For example, more practice leads Once efficacy has been established, additional Phase
to greater learning than less practice (Edeal & Gildersleeve- III studies are warranted to compare relative efficacy of
Neumann, 2011; Maas et al., 2019; Namasivayam et al., different treatments. Because different treatments may tar-
2015), variable practice enhances learning compared to get different skills, there may be an optimal staging or
constant practice (Preston et al., 2017), and less feedback sequencing of interventions depending on a child’s age,
enhances learning compared to feedback on all trials severity, comorbidities, profile, and progress. Studying
(Maas et al., 2012). However, no clear differences were such adaptive intervention sequences will require more
observed for practice schedule (Maas & Farinella, 2012) complex designs, such as Sequential Multiple Assignment
and feedback type (McKechnie et al., 2020), although Randomized Trial designs (Almirall et al., 2014). Further-
some children in these studies did show benefits for one more, comparing treatments that target different skills will
condition or another. require careful consideration of primary outcome mea-
sures that are not biased toward one or the other. For
More critically, for practice distribution (“inten-
example, ReST primarily targets prosody and sequencing,
sity”), findings to date are largely contrary to expectations
whereas the IPA treatment primarily targets syllable struc-
from the motor learning literature, where distributed prac-
ture and consistent error processes. If these two treatments
tice has a robust advantage over massed practice (Maas
are compared directly, a primary outcome measure of pro-
et al., 2019). Instead, evidence from children with CAS
sodic accuracy would likely favor ReST, whereas a pri-
thus far indicates a relatively consistent benefit for massed
mary outcome measure of syllable structure accuracy
over distributed practice (Maas et al., 2019; Namasivayam
would favor the IPA treatment (see the What Does
et al., 2015; Preston et al., 2023; D. C. Thomas et al.,
“Work” Mean, Anyway? section for further discussion).
2014). Thus, while it is encouraging that most principles
of motor learning appear to apply to speech motor learn-
Treatment Optimization
ing in CAS, it cannot simply be assumed that the same
Principles of motor learning. Replication and exten-
effects will be observed. In the absence of evidence to the
sion of research on principles of motor learning is war-
contrary, we may rely on external evidence from the motor
ranted. Recently, there has been increased focus on treat-
learning literature, but it is important to continue to
ment intensity, reflected in the AKRS 2022 program (e.g.,
develop an evidence base specifically for children with CAS.
Maas, Caspari, Beiting, Gildersleeve-Neumann, Niculae-
Service delivery models. Most studies to date have Caxi, et al., 2022; Preston et al., 2023; D. Thomas et al.,
been conducted in university clinics and research labs, 2023). Thus far, most studies suggest that more intensive
involving individual face-to-face sessions in which an SLP (massed) practice is better than less intensive (distributed)
or a graduate student clinician delivers the treatment practice, but the optimal distribution remains unclear in
(Murray et al., 2014; but see Namasivayam et al., 2015, part because different studies compare different intensities
for an exception). This is understandable given the state and may define intensity differently (e.g., sessions per day
of development of most treatments, as the focus in early or week [Preston et al., 2023; D. Thomas et al., 2023] vs.
phases is on efficacy (likelihood of effect under optimal trials per session [Maas et al., 2019]). Preliminary data
circumstances) rather than effectiveness (likelihood of from a recent small-scale RCT further suggest that distrib-
effect under routine clinical circumstances). Optimal cir- uted practice may lead to greater improvement than
cumstances can be implemented more easily in a research massed practice (Maas, Caspari, Beiting, Gildersleeve-
lab or university clinic than in a busy routine clinical Neumann, Niculae-Caxi, et al., 2022; Maas et al., 2023),
environment. However, it is important to recognize that although the distributed practice condition in this study
this model of service delivery assumes availability of was relatively intensive (1 hr/day for 4 weeks; vs. massed
resources—including transportation means, time and cost, practice: 2 hr/day for 2 weeks). If replicated, such findings
and sufficient number of SLPs to provide treatment at an suggest that there may be a range of optimal intensity
optimal amount and intensity. Such resources are not with upper bounds (perhaps 2 hr/day is too intensive) and
available in all clinical settings or for all children, posing lower bounds (perhaps one session per week is insufficient;
significant barriers to implementing evidence-based inter- D. Thomas et al., 2023). Furthermore, a changing level of
vention protocols in clinical settings. Thus, there is a great intensity may be even more effective. Preston et al. (2023)
need to develop and study interventions that are more compared a fixed distributed schedule (two sessions per

Maas: CAS Treatment Review 9


Downloaded from: https://pubs.asha.org 5.210.215.88 on 06/08/2024, Terms of Use: https://pubs.asha.org/pubs/rights_and_permissions
week for 10 weeks) with a “tapered” intensity condition in color of a ball in a bowling task (Abdollahipour et al.,
which treatment was initially delivered at high intensity 2017; Lewthwaite et al., 2015; see Wulf & Lewthwaite,
(10 sessions in Week 1) and then tapered off to a more 2016, for a review). Although not yet examined in CAS,
distributed regimen (two sessions per week in Weeks 4 studies of some of these principles in the speech domain
and 5). Findings suggested that the “tapered” version led have begun to emerge and generally align with findings
to greater and longer-lasting gains than the fully distrib- from the motor learning literature (attentional focus:
uted version. Lisman & Sadagopan, 2013; McAllister Byun et al.,
2016; self-controlled feedback: Potkovac, 2020).
Similar replication and extension for other principles
is also justified. Here too, there is reason to compare One final point regarding the investigation of princi-
sequenced with fixed conditions. For example, the motor ples of motor learning is that different principles may
learning literature suggests that starting with constant interact with one another (Maas et al., 2008, 2012) or pro-
followed by variable practice enhances learning compared vide additive benefits. For example, Abdollahipour et al.
to either constant or variable alone or variable followed by (2017) showed that an external focus of attention and
constant practice (Lai et al., 2000); similar suggestions have autonomy of task-incidental choice (ball color) provided
been made for blocked versus random practice (Guadagnoli additive benefits for children learning a bowling task.
& Lee, 2004; Porter & Magill, 2010; Shea et al., 2001; cf. Studies of such interactive or additive effects will require
Jones & Croot, 2016; Maas & Farinella, 2012). factorial group designs.
In addition, several promising principles of motor Service delivery models. There are multiple reasons
learning have not yet been investigated in CAS. These to expand the investigation of service delivery models
include several principles proposed within the OPTIMAL3 beyond in-person intervention by an SLP in the lab or
theory of motor learning (Wulf & Lewthwaite, 2016), clinic. Most of these reasons relate to a limitation of
which emphasizes the roles of attention, enhanced expec- resources, either at a systems level (e.g., not enough SLPs
tancies, and autonomy. Although too numerous to pro- for the caseload or in a geographic area to provide suffi-
vide an exhaustive list, I note here several examples that ciently intensive treatment) or at the individual (family)
may be, or have been, shown to apply to children, in level (e.g., no insurance or money for private SLP ser-
some cases with even stronger effects (see Wulf & vices, no transportation, inability to take time off work to
Lewthwaite, 2016, for a review). With respect to attention, bring a child to the clinic). Additional reasons include
a robust finding in the motor learning literature is that an challenges with keeping children engaged and attentive
external focus (on the effect of the movement, e.g., golf (boredom or fatigue, especially for intensive treatments)
club) enhances performance and learning compared to an and health concerns (e.g., concern about exposure to
internal focus (on the moving body parts, e.g., arm; infectious diseases such as COVID-19, especially for chil-
Abdollahipour et al., 2017; Freedman et al., 2007; Shea & dren who may be at an elevated risk due to underlying
Wulf, 1999; Wulf et al., 1999, 2001; see Chua et al., 2021, conditions).
for a meta-analysis). With respect to enhanced expectan-
cies, increasing the learner’s expectations for successful In recent years, different service delivery models
performance enhances learning. This may be accom- have begun to be explored in order to mitigate some of
plished, for example, by providing positive feedback after the concerns with in-person treatment by an SLP in the
accurate attempts compared to negative feedback after clinic or lab. One model that has gained some attention
inaccurate attempts (Chiviacowsky & Wulf, 2007) or by is an intensive summer camp format, which enables
telling the learner that their performance is above average intensive treatment delivery (e.g., multiple sessions per
compared to others (Lewthwaite & Wulf, 2010). With day). Summer camps have become more common as a
respect to autonomy, greater learning is observed when clinical model (Preston, Leece, & Maas, 2016; see the
learners are given some autonomy over aspects of the list of summer camps at https://www.apraxia-kids.org/
learning situation. This may be accomplished, for exam- summer-speech-language-camps-for-apraxia/), and some
ple, by allowing the learner (rather than the instructor) to studies have implemented this model as well (Keller &
choose which attempts receive feedback (Chiviacowsky & Maas, 2023; cf. Levy et al., 2021, for a camp for children
Wulf, 2005; Chiviacowsky et al., 2008; Janelle et al., 1997; with dysarthria). However, no studies to date have directly
see Patall et al., 2008, for a meta-analysis) or by allowing compared this format with the more traditional format.
the learner to choose incidental task aspects such as the As such, it is unknown whether intensive summer camps
result in more positive outcomes than traditional formats,
3
OPTIMAL is an acronym for Optimizing Performance through
nor whether any differences are due to greater intensity,
Intrinsic Motivation and Attention for Learning (Wulf & Lewthwaite, the social element (e.g., interacting with other children
2016). with CAS in camp), or both.

10 Journal of Speech, Language, and Hearing Research  1–26

Downloaded from: https://pubs.asha.org 5.210.215.88 on 06/08/2024, Terms of Use: https://pubs.asha.org/pubs/rights_and_permissions


Even before, but especially since the COVID-19 pan- target words correctly. Although the game continues to be
demic, interest in remote (virtual) delivery of speech ther- refined and improved, initial results from small-scale Level
apy has increased (Bahar et al., 2022; Beiting & Nicolet, 3 studies (e.g., one-group pre–post designs) support the
2020), and several initial studies have examined the feasi- feasibility of implementation, accuracy of the automatic
bility and efficacy of virtual treatment for CAS (Maas, speech recognition system, and user engagement and satis-
Caspari, Beiting, Gildersleeve-Neumann, Niculae-Caxi, faction (Ahmed et al., 2018; Hair et al., 2021). Further-
et al., 2022; Parnandi et al., 2015; D. C. Thomas et al., more, clinically significant improvements have been dem-
2016). Although findings to date suggest that such a onstrated that were comparable to in-person gains
delivery model is feasible and may have positive effects, observed in previous studies (Hair et al., 2021). Future
only one study has directly compared in-person treatment work using more controlled designs is needed to determine
with mostly-virtual treatment (Parnandi et al., 2015). whether observed improvements represent treatment effects
Their results suggested greater improvement for children and to determine the efficacy of gamified speech therapy.
in the in-person condition than in the mostly-virtual con- Development of such approaches will likely include further
dition, although children improved in both conditions. optimization of automatic speech recognition and analysis
However, this was a small-scale study (N = 6) with sev- algorithms to provide reliable, accurate automatic feedback
eral important limitations, such as lack of blinded assess- (e.g., Quach et al., 2006; Valentine et al., 2023).
ment (Bahar et al., 2022). Thus, further and more rigor-
ous research is needed to compare in-person and virtual One additional service delivery model that may war-
treatment, including equivalence and/or noninferiority rant empirical study is group-based speech therapy. Due
trials (Christensen, 2007). to large caseloads, SLPs in schools frequently provide
group-based rather than individual (one-on-one) services
Other options to increase the amount and intensity
(ASHA, 2002). Data from the ASHA National Outcomes
of speech treatment and mitigate access limitations (e.g.,
Measurement System suggest that children receiving indi-
means and time to travel to the SLP) include providing
vidual speech therapy make greater progress on speech
services in or near the child’s home by an itinerant SLP,
goals than those who receive group-based speech therapy
or training caregivers to provide treatment, either instead
(ASHA, 2002). However, these data are not from con-
of or to supplement the SLP’s services. An ongoing multi-
trolled studies but based on outcomes in clinical practice.
site RCT of DTTC (McCabe et al., 2022) offers the
Two recent studies with children with speech sound disor-
option for in-home delivery by an itinerant SLP at some
ders (SSDs) demonstrated a positive effect of group-based
sites, and although not designed to compare in-home ver-
treatment (Skelton & Richard, 2016) and no differences in
sus in-clinic delivery models, this study will enable a post
gains between children receiving individual versus group
hoc examination of treatment effects following in-home
speech therapy (Byers et al., 2021), but it is not clear
delivery and, possibly, a comparison between these
whether or to what extent these findings apply to children
models. With respect to caregiver training, D. C. Thomas
with CAS specifically. It may, at first glance, seem obvi-
et al. (2018) showed that a combined (SLP + caregiver)
ous that individual speech therapy would be more effec-
delivery of ReST may be efficacious for some children but
tive because children have more practice opportunities (vs.
also reported that the effects were smaller compared to
sharing therapy time with other children), and this is con-
historical data from in-person ReST studies. Thus, study
sistent with the idea that more practice enhances learning
of these delivery models is in its infancy and will likely
(Edeal & Gildersleeve-Neumann, 2011; Maas et al., 2019).
expand in the next decade.
However, group treatment may induce greater distribution
Another option that aims to mitigate systemic and of practice (Skelton & Richard, 2016), which may benefit
individual resource limitations as well as reduced engage- learning. Observing other children with speech disorders
ment due to boredom or fatigue is the gamification of (or CAS specifically) also affords opportunities for obser-
speech therapy. By creating engaging video games that vational learning from attempts and strategies used by,
embed speech practice, especially older children with CAS and feedback provided to, other children (cf. Byers et al.,
may achieve higher amounts of practice than is possible 2021). This may include enhancing speech perception,
with in-person treatment. An interesting and promising which may predict treatment response (Beiting, 2022b;
example is Say Bananas!, an elaborate multilevel video Hitchcock et al., 2023). Furthermore, there may be social
game that integrates speech practice structured along prin- benefits from sharing the speech therapy experience with
ciples of motor learning and includes an automatic speech other children. Thus, given that group speech therapy is a
recognition system to provide feedback (Ahmed et al., common service delivery model and that there are reasons
2018; Hair et al., 2021). In the game, children control a why group speech therapy may offer benefits, controlled
monkey to get bananas and cross the finish line; children study and comparison of these service delivery models is
get more rewards and avoid time-outs if they say their well justified.

Maas: CAS Treatment Review 11


Downloaded from: https://pubs.asha.org 5.210.215.88 on 06/08/2024, Terms of Use: https://pubs.asha.org/pubs/rights_and_permissions
For Whom Does Treatment X Work? (Namasivayam et al., 2015), IPA treatment (McNeill
Candidacy, Prognosis, and Generalizability et al., 2009b), the Kaufman program (Gomez et al.,
2018), biofeedback treatment (Preston, Maas, et al., 2016),
This third fundamental treatment research question and others (Rvachew & Matthews, 2019).6
is essentially a question of external validity and is impor-
It is critical that we understand why some children
tant to determine whether a particular child is a good can-
respond well to a treatment while others do not. This is
didate for a given treatment and to establish a likely prog-
important to inform clinical decision making and select
nosis. If multiple treatment approaches are available, the
appropriate treatments for an individual child, which in
question may extend to determine which of the treatments
turn is important for making judicious use of limited
works better for which children. It is beyond the scope of
available resources within the health care system. How-
this review article to discuss the presumed optimal candi-
ever, at present, we know very little about the factors that
dacy characteristics for each of the specific approaches
predict treatment response. This state of affairs is in part
mentioned.4 To be sure, identifying such characteristics
related to the study designs reported in the literature to
for each approach is an important undertaking, but one
date, which primarily involve case studies or SCED stud-
more suitable for a focused systematic or scoping review,
ies. On the face of it, SCED studies are ideally suited to
replication studies with populations different from the
determine predictive factors because, contrary to group
originally proposed optimal candidates,5 or post hoc anal-
designs, they provide rich detail about background factors
ysis of multiple published studies of a specific approach
and treatment response of individual children and can rep-
(see Ng et al., 2022, for an example). Rather, the discus-
resent a broader range of participants (vs. requiring a rela-
sion here focuses on the broader issues applicable to all
tively homogeneous sample in group studies). This allows
treatments, but illustrated with a brief review of existing
clinicians to determine the degree to which their clients
studies about specific approaches. The subsections below
match those in the literature (J. C. Rosenbek, 2016). How-
are each divided into two different, albeit interrelated,
ever, the small sample sizes preclude systematic examina-
parts: (a) Predictors of Response to Treatment and (b)
tion of predictive factors because such factors often
Underrepresented Populations.
covary, making it difficult to determine which of these
factors (or combination thereof) is the most relevant one.
The Present
For example, in the study of Maas and Farinella (2012),
one of four children did not show improvement. This
Predictors of Response to Treatment
child was both the youngest and judged to have the most
It is a truism that children vary in their response to
severe CAS; he was also the only one with a possible mild
treatment, and an unfortunate but common observation is
dysarthria per clinical judgment. Which (combination) of
that not all children with CAS improve. Similarly, not all
these factors was responsible for his lack of progress?
children with CAS show the same response to different
Were there other factors? With such a small sample, it is
treatment conditions, and opposite response patterns have
impossible to know. Increasing sample size by pooling
been reported for practice schedule (Maas & Farinella,
across studies may be an option (Ng et al., 2022),
2012), feedback variables (Maas et al., 2012; McKechnie
although differences in methodology, treatment parame-
et al., 2020), and practice intensity (Maas et al., 2019).
ters, and settings introduce further variability that may
The observation that not all children respond similarly, or
prevent discovery of predictive factors.
at all, is not specific to any particular approach (nor to
CAS) but applies across treatments, including integral The advantage of group studies with large samples
stimulation (Maas et al., 2012; Strand et al., 2006), ReST is that different variables can be dissociated analytically
(Ng et al., 2022), the Motor Speech Treatment Protocol across children who all underwent the same protocol
under the same circumstances, for example, via regression
approaches to uncover predictive factors without introduc-
4
Readers are referred to the original sources for specific inclusion/ ing interstudy variability. A drawback is that such studies
exclusion criteria and rationale for treatment candidacy. However, require relatively homogeneous samples (J. C. Rosenbek,
the fact that an approach was originally designed for (or studied in) a
specific population does not mean that the treatment is not or less
2016) and thus are limited in the range of variables that
effective for children with other profiles. can be considered. As suggested by Rosenbek, there is a
5
Examples include the studies of Edeal and Gildersleeve-Neumann role for both SCED and group studies in identifying pre-
(2011) and Maas et al. (2012), who studied integral stimulation treat- dictive participant factors (i.e., a “both/and” rather than
ment for children with moderate rather than severe CAS (Strand,
2020); Korkalainen et al. (2023), who studied ReST in children with
6
dysarthria; and Grigos et al. (2023), who studied DTTC in children For group design studies, interindividual variation in treatment
with CAS younger than in the original studies (Strand et al., 2006; response can be inferred from inspection of means and standard devi-
Strand & Debertine, 2000). ations of change scores (e.g., Namasivayam et al., 2015).

12 Journal of Speech, Language, and Hearing Research  1–26

Downloaded from: https://pubs.asha.org 5.210.215.88 on 06/08/2024, Terms of Use: https://pubs.asha.org/pubs/rights_and_permissions


an “either/or” approach). SCEDs, by their nature, can sizes, but they are promising and point to the importance
examine a wider range of populations and therefore are of evaluating speech perception in children with CAS
ideal for generating hypotheses about predictive factors. In (Hitchcock et al., 2023) and possibly incorporating audi-
contrast, group designs, by virtue of larger samples and tory speech perception training elements in CAS treat-
consistent procedures for all participants, are better suited ments (e.g., see Rvachew & Matthews, 2019).
for testing hypotheses about predictive factors. For exam-
ple, a group study could disentangle the contributions of Underrepresented Populations
age and CAS severity (as in the Maas and Farinella exam- The previous section briefly reviewed potential pre-
ple above) or test the hypothesis that poor phonological dictive factors for treatment response based on children
processing skills limit response to treatment (a hypothesis who participated in treatment studies. However, these
generated from an SCED study; Preston, Maas, et al., study samples do not adequately represent the population
2016). Thus, SCED and group design studies complement of children with CAS at large. Group studies tend to
each other. involve relatively narrow selection criteria in order to
achieve homogeneity within the sample and group equiv-
At present, very few studies exist that have systema- alence at baseline. Although not necessary in SCED
tically examined potential predictive factors for response studies, these often also set narrow selection criteria,
to CAS treatment (Beiting, 2022b; Grigos et al., 2023; Ng especially if such studies include multiple children about
et al., 2022). Ng et al. (2022) examined several predictors whom some generalization about efficacy is to be made
of response to ReST treatment based on data from multi- (Dale & Hayden, 2013; Maas et al., 2019; Rvachew &
ple published and some (as-yet) unpublished studies, Matthews, 2019). While understandable and scientifically
involving 36 children. Regression analyses showed that justifiable for internal validity, these narrow criteria
several baseline variables predicted higher posttreatment mean that we have little to no evidence about treatment
performance, including higher expressive language scores, efficacy for many children with CAS. Among the limita-
higher consonant accuracy, lower vowel accuracy, greater tions of external validity in the CAS treatment literature
speech consistency, and higher baseline accuracy on tar- are those related to comorbidities, language diversity,
gets (cf. Murray et al., 2015). However, none of the exam- language backgrounds, and demographics.
ined variables predicted change scores.
Regarding comorbidities, co-occurring language dis-
Grigos et al. (2023) reported that, in a sample of orders are not typically considered exclusionary criteria,
seven young children with severe CAS who received presumably due to the frequent comorbidity of language
DTTC, degree of improvement was not associated with disorders and CAS (Iuzzini-Seigel et al., 2022). However,
nonverbal cognitive and receptive language skills, despite the vast majority of studies exclude children with dysar-
the sample representing a considerable range on these thria, cognitive impairments, neurobehavioral disorders
measures. Such findings suggest that low nonverbal cogni- such as autism or attention-deficit/hyperactivity disorder,
tion and receptive vocabulary are not contraindications visual or hearing impairments, and atypical oral structures
for successful application of DTTC in young children with (e.g., cleft palate). Exceptions exist, but those represent case
severe CAS. The only measure that appeared to predict studies or SCED studies with small sample sizes (N ≤ 3;
improvement was baseline accuracy, such that lower base- Baas et al., 2008; Beiting & Maas, 2021; Caspari et al.,
line accuracy was associated with greater improvement. 2008) and/or low confidence in CAS diagnosis (Beathard &
Krout, 2008; Binger & Light, 2007; Binger et al., 2008;
Beiting (2022b) studied possible predictors for chil-
Murray et al., 2014). Comorbidities represented in the CAS
dren enrolled in two small-scale RCT studies of ASSIST,
treatment literature to date include developmental delay or
involving a total of 27 unique children (Maas, Caspari,
intellectual disability (Baas et al., 2008; Beathard & Krout,
Beiting, Gildersleeve-Neumann, Niculae-Caxi, et al., 2022;
2008; Binger & Light, 2007; Caspari et al., 2008;
Maas, Caspari, Beiting, Gildersleeve-Neumann, Stoeckel,
Nakamura-Palacios et al., 2024), velopharyngeal insuffi-
& Wu, 2022). Predictor variables included age, baseline
ciency (Binger et al., 2008), dysarthria (Maas et al., 2019;
speech accuracy and speech inconsistency, and perceptual
Strand et al., 2006; D. C. Thomas et al., 2014), galacto-
ability based on an auditory discrimination task. Percep-
semia (Peter et al., 2019, 2022), and autism (Beiting &
tual ability was available only for children in the second
Maas, 2021).
study (n = 12). Neither age, baseline speech accuracy, nor
speech inconsistency significantly predicted change score. With respect to language diversity, the vast majority
However, perceptual ability strongly and significantly pre- of CAS treatment research has been conducted in English,
dicted improvement, such that children with greater per- in particular American or Australian English (but see
ceptual ability showed greater improvement. Needless to Namasivayam et al., 2015, and Rvachew & Matthews,
say, these findings require replication with larger sample 2019, for Canadian English; McNeill et al., 2009a, for

Maas: CAS Treatment Review 13


Downloaded from: https://pubs.asha.org 5.210.215.88 on 06/08/2024, Terms of Use: https://pubs.asha.org/pubs/rights_and_permissions
New Zealand English; van der Merwe & Steyn, 2018, for No systematic analysis is available of CAS treatment stud-
South African English). Other languages studied in CAS ies after 2012 (the last year covered in Murray et al.’s sys-
treatment include Afrikaans (Bornman et al., 2001), tematic review); thus, it is unclear whether reporting of
Dutch (van Tellingen et al., 2023), Finnish (Martikainen this information has increased. Some recent studies
& Korpilahti, 2011), German (Leonhartsberger et al., included at least some of this information (Gildersleeve-
2022), Italian (Scarcella et al., 2021), Brazilian Portu- Neumann & Goldstein, 2015; Maas et al., 2019; Peter
guese (Nakamura-Palacios et al., 2024), and Swedish et al., 2022; Preston et al., 2013, 2023), but many still do
(Lundeborg & McAllister, 2007). Thus, except for a few not, so it is unknown whether or to what extent sample
single studies on Finnish, Italian, and Portuguese, the demographics represent the population at large. It is not
CAS treatment literature is entirely based on languages unreasonable to suspect that children from non-White,
from the Germanic branch of the Indo-European language and/or Hispanic, and/or low-SES backgrounds are under-
family. This significantly constrains generalizability and represented in the current CAS treatment evidence base,
understanding of how treatments may be used or adapted for example, due to resource limitations restricting access
for other languages. to research opportunities or higher levels of mistrust of
researchers given a history of research-related injustices
With respect to language backgrounds, virtually all
and mistreatment (“the Tuskegee effect”; Green et al.,
CAS treatment studies to date have included only mono-
1997; Shavers et al., 2000). Given the well-known existence
lingual children (and mostly English). However, over half
of health care disparities, including in speech-language
of the world population speaks more than one language
pathology (Ellis, 2009; Morgan et al., 2016), correlations
(Grosjean, 2012), and over 21% of people older than
between race/ethnicity and SES (Caldas & Bankston, 1997),
5 years old in the United States speak a language other
and elevated risk of lower literacy and academic out-
than English at home (U.S. Census Bureau, 2022). There-
comes in CAS (Lewis et al., 2004; G. J. Miller et al.,
fore, the current CAS treatment literature woefully underre-
2019), children with CAS from these underrepresented
presents the population, and little to no controlled evidence
backgrounds may have an even higher risk. Thus, this
exists regarding efficacy or application of treatment for
lack of information poses an obstacle for determining
bilingual or multilingual children with CAS to support clini-
(lack of) generalizability of the evidence base and identi-
cal decision making (cf. Simon-Cereijido, 2018). One excep-
fying the unique needs, challenges, and opportunities for
tion is a study by Gildersleeve-Neumann and Goldstein
children from different backgrounds in terms of clinical
(2015), who provided integral stimulation treatment in both
service delivery (cf. Hammer, 2011).
Spanish and English (with emphasis on Spanish) for a bilin-
gual Spanish–English boy with CAS. Findings indicated
comparable gains in both languages, despite the greater The Future
emphasis on Spanish. The authors suggest that bilingual
treatment that emphasizes the dominant language may lead Predictors of Response to Treatment
to more widespread gains than targeting the nondominant Given that few clear predictors of treatment response
language, although this intriguing hypothesis needs further have been identified, mostly due to the limited number of
investigation in controlled studies. studies with sufficient sample sizes, the variability in treat-
ment response among children with CAS remains largely
Finally, with respect to demographic factors, most unexplained—for any treatment approach. Additional rep-
studies include children between 4 and 10 years old, with lications and group design studies will offer opportunities
few studies of younger children (e.g., see Grigos & to generate and test hypotheses about potential predic-
Kolenda, 2010, and Grigos et al., 2023, for exceptions) or tors (e.g., SES, treatment history). Future studies should,
older children and adolescents (e.g., see Preston et al., for example, assess, and evaluate the role of, speech per-
2013, 2017, 2023, for exceptions). While age and sex are ception abilities in children with CAS because speech
commonly reported, other factors such as race, ethnicity, perception was identified as a potentially strong predictor
and socioeconomic status (SES) are not. An informal (Beiting, 2022b).
analysis of 20 articles reviewed in Murray et al.’s (2014)
study reveals that sex was reported for 43 (of 52) children Furthermore, additional hypotheses about predictive
in these studies, of whom 35% were girls and 65% were factors may emerge from a more detailed theoretical
boys. However, race and ethnicity were reported in only understanding of speech motor planning impairments in
four studies (20%). Among the eight children with CAS in CAS. In recent years, awareness and appreciation have
these studies, one was Black, one was Asian, and one was grown for the potential value of identifying underlying
Native American (12.5% each), and five (62.5%) were processes that may be impaired in CAS and for the possi-
White. None of these children (0%) were identified as His- bility that different subtypes of CAS exist depending on
panic. SES was rarely reported (Binger & Light, 2007). which motor planning processes are affected (Littlejohn &

14 Journal of Speech, Language, and Hearing Research  1–26

Downloaded from: https://pubs.asha.org 5.210.215.88 on 06/08/2024, Terms of Use: https://pubs.asha.org/pubs/rights_and_permissions


Maas, 2023; Shriberg et al., 2012; Terband et al., 2019; cf. language are also sorely needed, given that the majority of
Mailend & Maas, 2021). Given the complex, multifaceted the world population and a sizable portion of the U.S.
nature of speech motor planning (Tourville & Guenther, population speak more than one language. The dearth of
2011), it is plausible that not all children with the studies on treatment for bilingual children with CAS hin-
“umbrella label” of CAS present with the same underlying ders SLPs’ ability to select the most appropriate interven-
impairment. Different underlying impairments may respond tion approach, as they must rely on the clinical expertise,
differently to the same treatment, and different treatments client values and preferences, and theoretical rationale pil-
may be more effective for different impairments. Further lars of evidence-based practice without the benefit of exter-
development of process-oriented approaches to under- nal evidence. Thus, until direct research becomes available,
standing and assessment of CAS may yield process- clinicians may need to resort to “off-label” use of treat-
specific measures that could explain heterogeneity in treat- ments found to be efficacious for monolingual children.
ment response and lead to development or adaptation of
Finally, it is unknown whether efficacious interven-
interventions to target specific impairments. A recent
tions generalize to younger children (under 4 years of age) or
study by Rvachew and Matthews (2019) illustrates this
children from underrepresented populations. Future studies
idea nicely: They identified children with CAS who pri-
should actively work to recruit and retain children from a
marily had motor planning impairments and children with
diverse population in terms of racial/ethnic and socioeco-
CAS who primarily had phonological planning impair-
nomic backgrounds and examine whether there are system-
ments. In a single-subject randomization design, children
atic differences in efficacy. If so, treatments must be adapted
received an auditory–motor integration (AMI), a phonolo-
or developed to optimize outcomes for these children. This
gical memory and planning (PMP), and a control version
call for action is consistent with increased priority of includ-
of an integral stimulation intervention. Results under-
ing underrepresented minorities in research in our field
scored the value of differentiating profiles: Children with
(cf. special funding opportunities at the National Institute
motor planning impairments responded best to the AMI
on Deafness and Other Communication Disorders, grants.
version, whereas children with phonological planning
nih.gov/grants/guide/notice-files/NOT-DC-21-003.html). As a
impairments responded best to the PMP version. These
bare minimum first step, studies should report demographic
interesting findings support further development of
information beyond age and sex, so that these variables can
methods and measures to identify underlying impairments
be documented and analyzed in future reviews. Future stud-
and/or CAS subtypes that can inform treatment candidacy
ies should also consider community-based participatory
and development of more targeted interventions.
designs (e.g., Horowitz et al., 2009).
Underrepresented Populations
With respect to underrepresented populations, much What Does “Work” Mean, Anyway?
work remains to be done in order to determine whether Outcome Measures
and to what extent the available evidence base can be gen-
eralized. In terms of comorbidities, additional studies are Up to this point, I have avoided this question, which
needed involving a broader spectrum of comorbidities. is the most fundamental one because it underlies the other
Although large-scale studies may be a longer-term goal, three treatment questions. The narrative review thus far
there is ample need for well-controlled SCED studies to has simply relied on the outcome measures reported in the
examine the efficacy of existing treatments and/or treat- literature. This section delves further into what it means
ments designed or adapted for children with various to say that a treatment works, or works better, for which
comorbidities (Beiting, 2022a; Beiting & Maas, 2021). children with CAS.
Such SCED studies can then help set the stage for larger
group design studies. The Present
There is also a great need to examine treatment effi-
The outcome measures used in CAS treatment stud-
cacy for children with CAS who speak other languages—
ies to date only capture a narrow, albeit important, view
and in particular, non-Germanic languages—as it cannot
of function, with very few exceptions. Specifically, virtu-
simply be assumed that intervention approaches apply in
ally all studies only report outcomes at the Body
the same way and have similar effects. There may be lan-
Structures/Functions (impairment) level of the World
guage or cultural differences that require modifications of
Health Organization’s (2007) International Classification
existing protocols (e.g., tone language vs. nontone lan-
of Functioning, Disability and Health (WHO ICF; see
guage; Wong et al., 2023).
Figure 3 for application to CAS). For example, Kearney
Research on efficacy or optimal intervention et al. (2015) reported that out of 66 studies for motor-
approaches for children who speak more than one based SSDs (not limited to CAS), all 66 studies reported

Maas: CAS Treatment Review 15


Downloaded from: https://pubs.asha.org 5.210.215.88 on 06/08/2024, Terms of Use: https://pubs.asha.org/pubs/rights_and_permissions
Figure 3. The World Health Organization (WHO) International Clas- measures beyond the Body Structures/Functions level, per-
sification of Functioning, Disability and Health model (WHO, 2007)
as applied to childhood apraxia of speech (CAS). Adapted with haps on the (implicit or explicit) assumption that improve-
permission from World Health Organization (2007). ICF-CY: Inter- ments in speech accuracy will more or less automatically
national Classification of Functioning, Disability and Health: Chil- translate into improved intelligibility, which in turn would
dren and youth version. WHO is not responsible for the content or
accuracy of this adaptation. cascade into communicative participation, development of
peer relationships, and emotional well-being.
However, although this assumption is reasonable,
these assumed relationships have not (yet) been well estab-
lished empirically for children with CAS. For example,
only two studies have explicitly examined the relationship
between speech accuracy and single-word intelligibility
(Activity) in children with CAS (Chenausky et al., 2022;
Skoog & Maas, 2020). Both studies did suggest a positive
relationship between accuracy and intelligibility and noted
that consonant accuracy had a greater impact on intellig-
ibility than vowel accuracy. However, Skoog and Maas
only included three children, and Chenausky et al. used
SLP/graduate student listeners who were likely familiar
with the stimuli from a common articulation test, likely
inflating intelligibility. Neither study examined relation-
ships with connected speech intelligibility or communica-
impairment-level outcome measures, but only three reported
tive participation. Only one recent study examined associ-
measures of Activity or Participation, and none reported out-
ations between children’s self-reported communication
come measures related to Environmental or Personal Factors.
attitudes (Personal Factors) and caregiver-rated measures
Impairment is most commonly captured in measures of communicative participation (Participation; Keller &
of speech accuracy (e.g., whole-word accuracy ratings, Maas, 2023). The small sample prevented formal analysis,
percent phonemes correct) as judged by trained research but scatter plots showed no relationship, suggesting that
assistants or SLPs. This means that when we say that a communicative participation does not predict, nor is pre-
treatment “works,” we generally refer to effects on speech dicted by, children’s self-reported communication attitudes.
accuracy judged by trained listeners. However, speech
To date, only a handful of CAS treatment studies
accuracy may not be the most meaningful outcome for chil-
included outcome measures beyond the impairment level
dren and their families—the most important people
(Dale & Hayden, 2013; Maas, Caspari, Beiting, Gildersleeve-
involved. In fact, both anecdotal and systematic (Rusiewicz
Neumann, Niculae-Caxi, et al., 2022; Namasivayam et al.,
et al., 2018) reports suggest that caregivers are more con-
2015; Wang & Grigos, 2023). Dale and Hayden included
cerned about their child’s emotional well-being (e.g., confi-
measures of word and sentence intelligibility (Activity)
dence, frustration; Personal Factors) and their child’s ability
and peer socialization (Participation). The design (essen-
to be understood (e.g., intelligibility, comprehensibility;
tially an AB SCED) did not permit establishing causality
Activity) as well as form peer relationships and participate
for these measures, but all four children showed more
in activities (e.g., social, academic; Participation).
than 16.7% gain on word and sentence7 intelligibility after
Evidence-based practice (ASHA, 2004) requires con- treatment, and two showed improvement for peer sociali-
sideration of client values and preferences in setting mean- zation. Namasivayam et al. (2015) included measures of
ingful goals. Baylor and Darling-White (2020) have pro- intelligibility and participation. Neither the low- nor
posed that communicative participation represents the pri- high-intensity group showed improved intelligibility,
mary goal of planning and implementing an intervention although the high-intensity group showed improved artic-
and provide a conceptual framework and resources for ulation accuracy. However, the high-intensity group did
shared decision making in this regard. They note that show significant gains in communicative participation,
impairment severity is often not, or only weakly, corre- again underscoring that participation is not predictable
lated with measures of communicative participation, from intelligibility. Maas, Caspari, Beiting, Gildersleeve-
underscoring one of the key insights of the WHO ICF, Neumann, Niculae-Caxi, et al. (2022) included caregiver-
namely, that there are many interacting factors that deter- rated measures of comprehensibility and communicative
mine someone’s functioning, and functioning at one level
does not necessarily predict functioning at another level. 7
Only three children completed the sentence intelligibility test; all
Yet, CAS treatment studies rarely include outcome three showed significant improvement.

16 Journal of Speech, Language, and Hearing Research  1–26

Downloaded from: https://pubs.asha.org 5.210.215.88 on 06/08/2024, Terms of Use: https://pubs.asha.org/pubs/rights_and_permissions


participation in a study of intensity of integral stimula- because the treatments are thought to exert their effect
tion teletherapy. Although neither the massed (16 hr in most directly and clearly on those proximal measures and
2 weeks) nor distributed (16 hr in 4 weeks) group because reduced speech accuracy impacts intelligibility
improved on comprehensibility, the distributed group sig- and naturalness. However, as argued above, speech accu-
nificantly improved on communicative participation, racy may not be the most meaningful to the children and
with a significant advantage of distributed over massed their families, for whom they may be relevant only insofar
practice. Finally, Wang and Grigos (2023) examined as they form part of a causal chain, leading to clinically
intelligibility for five young children with severe CAS in significant improvements in intelligibility, communicative
a treatment study of DTTC and reported improved intel- participation, emotional well-being, and so forth. See Fig-
ligibility that was maintained 6 weeks posttreatment, ure 4 for an example of such a causal chain in the form of
based on unfamiliar listeners rather than SLPs to a hypothesized pathway.
enhance ecological validity. Although the design does not
allow interpretation of this improvement as a treatment The value of a hypothesized pathway is that it
effect, these findings are important in that they document makes explicit our thinking about these relationships and
gains in a measure that may be more relevant to client/ facilitates understanding of different outcomes and how to
family goals than accuracy and set the stage for further improve the clinical impact of a treatment if we collect
controlled studies. measures of different parts of the pathway. For example,
if a clinical trial shows improvement in the focus (speech
The Future accuracy), barrier (intelligibility), and clinical goal (partici-
pation), this would support the hypothesized relationship
Outcome Measures: Measuring What Matters between these constructs. If a clinical trial shows improve-
Given that most current treatments for CAS are ment in the clinical goal but not in measures of the focus
impairment focused (targeting speech motor planning or barrier, this would suggest success by an unknown path-
skills in some form or other), it is likely—and appropriate way and would require further thinking to understand
—that outcome measures in future treatment studies will how the change came about. If a clinical trial fails to
continue to include impairment-level measures such as show improvement in the clinical goal (a negative trial),
speech accuracy. Obtaining such measures is important but there is improvement in the focus or barrier, this

Figure 4. Hypothesized pathway of how a treatment effects change in a clinical goal at the WHO ICF Participation level (after Czajkowski
et al., 2015; see also Figure 2). Colors correspond to levels of the WHO ICF as in Figure 3 (red = Body Structure/Function, blue = Activity,
green = Participation, yellow = Environmental Factors, orange = Personal Factors). In this example, the (more distal) clinical goal is commu-
nicative participation (Baylor & Darling-White, 2020) measured on a caregiver-reported instrument (e.g., Thomas-Stonell et al., 2012). Barriers
include intelligibility and comprehensibility (Activity), willingness to take a risk and engage in a communicative situation (Personal Factors),
and listener effort and willingness and ability to accommodate the speaker (Environmental Factors). Note that speech accuracy (the clinical
goal in Figure 2) is now relegated to a more proximal focus of treatment among others that may require additional treatments or compo-
nents; integral stimulation is used here as one example of an impairment-focused intervention. Note further that different elements within a
domain (focus of treatment, barrier) may influence each other as well. To be clear, these examples are hypotheses that require further test-
ing, but this pathway illustrates how (elements of) treatments could be added to maximize the clinical goal of communicative participation.
AAC = augmentative and alternative communication; ICF = International Classification of Functioning, Disability and Health; WHO = World
Health Organization.

Maas: CAS Treatment Review 17


Downloaded from: https://pubs.asha.org 5.210.215.88 on 06/08/2024, Terms of Use: https://pubs.asha.org/pubs/rights_and_permissions
would suggest a failed hypothesis: The assumption that existing interventions and development of novel manage-
improvements in accuracy or intelligibility will lead to ment approaches designed to optimize impact on outcome
increased participation would not be supported, and addi- measures that matter most.
tional factors would need to be considered that may have
prevented change in the clinical goal. Alternatively, lack The “Dark Side”? Measuring Side
of change in accuracy or intelligibility in a negative trial Effects of Treatment
would constitute a failed trial: The hypothesized relation- Consideration of children’s functioning in the con-
ship cannot be determined because the expected change in text of the WHO ICF also extends to the contextual fac-
proximal functions did not occur. In that case, further tin- tors. In addition to advancing our understanding of rela-
kering is needed to ensure the treatment does impact accu- tionships among outcome measures at the impairment,
racy and/or intelligibility, so that the relationship between limitation, and restriction levels, future research should
them and the clinical goal can be tested. also examine relationships with measures of Personal and
Environmental Factors. To date, little treatment research
Thus, it is important that treatment research begin
has systematically investigated impact on, and of, broader
to include measures of constructs related to other levels of
personal factors (e.g., psychosocial well-being, confidence,
the WHO ICF, in order to demonstrate (not merely
anxiety) or the environment (e.g., family dynamics, care-
assume) whether, and how, improved speech accuracy
giver burden, health economics). These relationships are
translates into functionally meaningful gains. As a first
expected to be positive, such that treatments that improve
step, measures of these other levels may be included as
speech accuracy also lead to improvements in these other
secondary outcome measures, in order to document
domains. However, it is possible that these relationships
whether or to what extent these measures covary or
may in some cases be negative, such that a treatment that
change with treatment. Documenting changes on these
improves speech accuracy leads to negative effects in these
measures as secondary outcomes will generate estimates of
other domains.
likely effect sizes, which are necessary to determine
required sample sizes for studies in which these measures Contrary to surgical or pharmacological treatment,
serve as a primary outcome measure. As treatments prog- behavioral interventions such as speech therapy generally
ress toward Phase III studies, it can be argued that mea- have minimal to no risk of physical harm. Perhaps for this
sures such as intelligibility or communicative participation reason, CAS treatment studies to date (regardless of
are more appropriate, meaningful, and relevant primary phase) have not systematically examined possible negative
outcomes for which to determine definitive efficacy and side effects of treatment. Rather, the focus has been on
claim that a treatment “works” (Baylor & Darling-White, documenting and establishing positive effects, in particular
2020). It may be that different treatments achieve compa- on impairment-level measures. Although this focus is nec-
rable improvements on speech accuracy (albeit perhaps essary and understandable, broadening our view beyond
via different mechanisms) yet differ in their impact on Body Structures/Functions raises important questions
intelligibility or communicative participation (McCabe about potential negative effects at other WHO ICF levels,
et al., 2023). Of course, intelligibility and communicative including Personal Factors (e.g., potential negative emo-
participation are examples here and not proposed as end- tional effects). To be clear, there is no evidence that CAS
all-be-all measures. It is likely that the next decade will treatment has negative side effects or that impairment-
also see more qualitative research in CAS that centers on focused CAS treatment is harmful—far from it. Rather,
the lived experience of children with CAS and their families the lack of evidence either way is a major gap in the liter-
to inform development of meaningful outcome measures ature. It is important to know whether there are trade-
and optimal intervention methods to target them (e.g., offs, such that treatment procedures designed to improve
McCormack et al., 2019, 2022; Rusiewicz et al., 2018). speech accuracy may reduce rather than improve (for
example) a child’s confidence or well-being. Two exam-
To reiterate, I am in no way suggesting that we
ples, related to frustration and communication attitudes,
abandon impairment-level measures. The point here is
illustrate the point.
merely that future research should take the WHO ICF
seriously and begin to systematically and rigorously It is a common clinical observation that impairment-
address outcome measures beyond the impairment level focused speech therapy can lead to frustration in children
and consider the pathways and mechanisms of action that with CAS because most such approaches require children
lead to clinically significant changes in more distal clinical to repeatedly practice targets that are difficult for them to
goals. Beyond adding and studying such outcome mea- say. Despite SLPs’ high level of skill in behavioral man-
sures in studies with impairment-focused primary out- agement and emotional support, frustration is often
comes, consideration of children’s functioning in the con- impossible to avoid completely. Taking breaks may allevi-
text of the WHO ICF will also inform modification of ate frustration but also reduces the amount of speech

18 Journal of Speech, Language, and Hearing Research  1–26

Downloaded from: https://pubs.asha.org 5.210.215.88 on 06/08/2024, Terms of Use: https://pubs.asha.org/pubs/rights_and_permissions


practice. In recent work from our lab, we have begun to the course of the treatment study, one child reported more
document children’s frustration levels during treatment as negative communication attitudes after a 2-week bout of
a way to (a) operationally define a stopping rule (discon- intensive speech therapy, and another reported more posi-
tinue treatment if a child experiences sustained significant tive communication attitudes following a less intensive 2-
frustration), (b) understand individual variation in treat- week bout of speech therapy. Although these changes can-
ment response, and (c) enable selection of (versions of) not be attributed to the treatment given the small sample
treatments that strike an optimal balance between speech and design, the findings warrant further controlled study
accuracy and frustration (Cook, 2021). If two treatments to determine whether or how impairment-focused speech
show comparable gains on speech accuracy (or intelligibil- therapy may impact self-reported communication attitudes
ity, etc.), but one engenders more frustration, it would be and whether or how self-reported communication attitudes
appropriate to choose the other treatment. In our work, may impact the efficacy of treatment.
the clinician rates the child’s frustration level after each
In summary, CAS treatment research must continue
session on a 4-point scale from 0 (no frustration) to 3
to document and establish positive effects of treatment,
(marked frustration). Preliminary findings indicate low
including on outcome measures beyond the impairment
frustration levels overall (mean across 16 sessions < 0.5)
level, but should also begin to consider potential side
and significantly higher frustration for afternoon than for
effects and trade-offs between outcome domains. As in
morning sessions in an intensive format of four 30-min
drug development, a complete understanding of treatment
sessions per day (Cook, 2021). No relationship was found
effects (positive and negative) is needed for SLPs and care-
between frustration and speech accuracy gains. Clearly,
givers to make well-informed decisions about appropriate
frustration is a complex construct, and further research
treatment for their children. Existing treatments may need
with larger samples is needed to determine relationships
to be supplemented or adapted to achieve positive out-
between frustration levels and treatment parameters,
comes across domains. To reiterate, there is no evidence
speech accuracy, and other factors such as child tempera-
of negative effects of treatment for children with CAS at
ment, past experiences, and environmental support. These
this time, so this discussion should not be construed as an
preliminary observations suggest that such exploration is
argument to abandon, deny, or restrict (impairment-
warranted.
focused or other) treatments. Rather, absence of evidence
is not evidence of absence, and it will be important to
A second example relates to children’s self-reported
obtain evidence—one way or another—about potential
attitudes about their own speech and communication
negative effects as well as positive effects.
skills. Potential negative effects of SSD have been docu-
mented for children with other, non-CAS SSDs (e.g.,
McCormack et al., 2010, 2019, 2022; Owen et al., 2004).
Overall, children hold positive attitudes about their
Summary and Conclusion
speech, but some children may experience negative effects
This “bird’s-eye” view of the CAS treatment litera-
related to their speech (e.g., negative self-attitudes, embar-
ture surveyed and organized the evidence base, identified
rassment, frustration), which may be related to speech
emerging trends, and outlined research directions. This
severity (McCormack et al., 2010, 2019). For CAS specifi-
was not a systematic review but a narrative review orga-
cally, emerging research indicates that adolescents and
nized around four fundamental treatment research ques-
adults with a history of CAS self-report higher rates of
tions, as a guiding framework and foundation for future
social difficulties (Lewis et al., 2021) and anxiety (Cassar
research. This organization was for expository reasons
et al., 2023). A suggestion for more negative self-reported
and does not imply that these issues are separate. For
social–emotional well-being was also reported for older
example, questions of which treatment works better, or
children with CAS (Swartz, 2022). However, these studies
which outcome measures are most suitable, are inter-
were not conducted in the context of treatment and did
twined with child characteristics (e.g., age, severity,
not include children with CAS younger than 9 years old.
comorbidity). Nevertheless, the hope is that this review
A recent exploratory study of self-reported communication
and organization provide a useful starting point for future
attitudes among children with CAS (age range: 4–9 years)
CAS treatment research.
enrolled in an intensive integral stimulation treatment
showed that children under 6 years of age did not differ In terms of “Does Treatment X work?,” the evi-
from normative values of children without communication dence base is expanding in quantity, study quality, and
disorders, whereas children older than 6 years of age were range of treatment approaches. While most approaches
more likely to report negative communication attitudes continue to be motor based (targeting the speech motor
(Keller & Maas, 2023). Moreover, while most children’s planning impairment), other promising approaches are
communication attitudes did not appreciably change over beginning to emerge as well.

Maas: CAS Treatment Review 19


Downloaded from: https://pubs.asha.org 5.210.215.88 on 06/08/2024, Terms of Use: https://pubs.asha.org/pubs/rights_and_permissions
In terms of “Does Treatment X work better than that we are well on our way and indeed likely to see
Treatment Y?,” a growing number of studies examine fac- another drastic expansion in quantity, quality, and range
tors that may optimize treatment outcomes or alternative of CAS treatment research in years to come.
service delivery formats. Such developments are particu-
larly important in light of the many logistical and finan-
cial barriers that limit the ability to implement existing Data Availability Statement
treatments in clinical settings. Furthermore, as develop-
ment of new and existing treatments progresses, we are As this is a review article, there are no primary data.
likely to see an increasing number of comparative effec-
tiveness studies of established treatments.
Acknowledgments
With respect to “For whom does Treatment X
work?,” few studies exist at the moment that have syste- This work was supported by the National Institute on
matically investigated potential predictive factors for treat- Deafness and Other Communication Disorders (NIDCD;
ment response. Given the common observation that not R01 DC017768; Principal Investigator: Maas). The content
all children respond equally to a treatment, it is important is solely the responsibility of the author and does not neces-
to understand and identify factors that predict response in sarily represent the official views of NIDCD. This article is
order to make well-informed clinical decisions. Further- based on an invited plenary presentation at the 2022
more, the generalizability of findings is currently limited Apraxia Kids Research Symposium in Las Vegas, NV. The
by the relatively narrow study samples in terms of comor- author is grateful to Apraxia Kids and the symposium
bidities and demographic factors (including age). organizers for the opportunity to present and discuss a ver-
Finally, with respect to “What does ‘work’ mean, sion of this review article at the symposium.
anyway?,” a critical gap in the literature is that virtually
all CAS treatment studies have only measured outcomes References
at the Body Structures/Functions level of the WHO ICF,
such as speech accuracy. Relatively ignored, to date, are Abdollahipour, R., Nieto, M. P., Psotta, R., & Wulf, G. (2017).
outcome measures more clearly or closely aligned with the External focus of attention and autonomy support have addi-
concerns of the most important people involved (i.e., the tive benefits for motor performance in children. Psychology of
children themselves and their families), such as intelligibil- Sport and Exercise, 32, 17–24. https://doi.org/10.1016/j.
psychsport.2017.05.004
ity, participation, and psychosocial well-being. This is a Ahmed, B., Monroe, P., Hair, A., Tan, C. T., Gutierrez-Osuna,
critical area for rigorous research in the near future to R., & Ballard, K. J. (2018). Speech-driven mobile games for
ensure that our evidence base is appropriately responsive speech therapy: User experiences and feasibility. International
to and aligned with the concerns and goals of children Journal of Speech-Language Pathology, 20(6), 644–658.
with CAS and their families. https://doi.org/10.1080/17549507.2018.1513562
Almirall, D., Nahum-Shani, I., Sherwood, N. E., & Murphy, S. A.
Looking back at the literature through this narrative (2014). Introduction to SMART designs for the development
of adaptive interventions: With application to weight loss
review lens, it is clear that CAS treatment research has research. Translational Behavioral Medicine, 4(3), 260–274.
expanded drastically since the first AKRS in 2002, the ini- https://doi.org/10.1007/s13142-014-0265-0
tiation of Apraxia Kids’ Research Grant competition, and American Speech-Language-Hearing Association. (2002). A work-
the 2013 AKRS. Perhaps at the next AKRS, we will wit- load analysis approach for establishing speech-language caseload
ness another notable increase. The hope is that the present standards in the schools [Position statement]. https://www.asha.
org/policy/ps2002-00122/
review article will provide a useful foundation and frame- American Speech-Language-Hearing Association. (2004). Evidence-
work to guide future CAS treatment research, spark or based practice in communication disorders: An introduction
renew enthusiasm in both seasoned and more junior [Technical report]. https://www.asha.org/policy/tr2004-00001/
researchers to continue to advance the CAS treatment evi- Baas, B. S., Strand, E. A., Elmer, L. M., & Barbaresi, W. J.
dence base, and, ultimately, improve outcomes for chil- (2008). Treatment of severe childhood apraxia of speech in a
12-year-old male with CHARGE association. Journal of Med-
dren with CAS and their families. In the spirit of J. C. ical Speech-Language Pathology, 16, 181–191.
Rosenbek and Wertz (1976), the suggested directions for Bahar, N., Namasivayam, A. K., & van Lieshout, P. (2022). Tele-
future research by no means represent an exhaustive list. health intervention and childhood apraxia of speech: A scop-
Rather, these suggestions are offered as potential goals, ing review. Speech, Language and Hearing, 25(4), 450–462.
“so that we can look back one day and see how far we https://doi.org/10.1080/2050571X.2021.1947649
Ballard, K. J., Robin, D. A., McCabe, P., & McDonald, J.
have come” (p. 197). We have come a long way, but there (2010). A treatment for dysprosody in childhood apraxia of
are many roads yet ahead of us. The cutting-edge research speech. Journal of Speech, Language, and Hearing Research,
presented at the 2022 AKRS is encouraging and suggests 53(5), 1227–1245. https://doi.org/10.1044/1092-4388(2010/09-0130)

20 Journal of Speech, Language, and Hearing Research  1–26

Downloaded from: https://pubs.asha.org 5.210.215.88 on 06/08/2024, Terms of Use: https://pubs.asha.org/pubs/rights_and_permissions


Baylor, C., & Darling-White, M. (2020). Achieving participation- intervention intensity and service delivery models with school-
focused intervention through shared decision making: Pro- age children with speech sound disorders in a school setting.
posal of an age- and disorder-generic framework. American Language, Speech, and Hearing Services in Schools, 52(2),
Journal of Speech-Language Pathology, 29(3), 1335–1360. 529–541. https://doi.org/10.1044/2020_LSHSS-20-00057
https://doi.org/10.1044/2020_AJSLP-19-00043 Caldas, S. J., & Bankston, C. (1997). Effect of school population
Beathard, B., & Krout, R. E. (2008). A music therapy clinical socioeconomic status on individual academic achievement.
case study of a girl with childhood apraxia of speech: Finding The Journal of Educational Research, 90(5), 269–277. https://
Lily’s voice. The Arts in Psychotherapy, 35(2), 107–116. doi.org/10.1080/00220671.1997.10544583
https://doi.org/10.1016/j.aip.2008.01.004 Caspari, S. S., Strand, E. A., Kotagal, S., & Bergqvist, C. (2008).
Beiting, M. (2022a). Diagnosis and treatment of childhood Obstructive sleep apnea, seizures, and childhood apraxia of
apraxia of speech among children with autism: Narrative speech. Pediatric Neurology, 38(6), 422–425. https://doi.org/10.
review and clinical recommendations. Language, Speech, and 1016/j.pediatrneurol.2008.03.002
Hearing Services in Schools, 53(4), 947–968. http://doi.org/10. Cassar, C., McCabe, P., & Cumming, S. (2023). “I still have
34944/dspace/7963 issues with pronunciation of words”: A mixed methods inves-
Beiting, M. (2022b). The impact of individual-level factors on tigation of the psychosocial and speech effects of childhood
progress in speech therapy for children with childhood apraxia apraxia of speech in adults. International Journal of Speech-
of speech (CAS) [Doctoral dissertation, Temple University]. Language Pathology, 25(2), 193–205. https://doi.org/10.1080/
TUScholarShare. https://scholarshare.temple.edu 17549507.2021.2018496
Beiting, M., & Maas, E. (2021). Autism-Centered Therapy for Chenausky, K. V., Gagné, D., Stipancic, K. L., Shield, A., &
Childhood Apraxia of Speech (ACT4CAS): A single-case Green, J. R. (2022). The relationship between single-word
experimental design study. American Journal of Speech- speech severity and intelligibility in childhood apraxia of
Language Pathology, 30(3S), 1525–1541. https://doi.org/10. speech. Journal of Speech, Language, and Hearing Research,
1044/2020_AJSLP-20-00131 65(3), 843–857. https://doi.org/10.1044/2021_JSLHR-21-00213
Beiting, M., & Nicolet, G. (2020). Screenless teletherapy and Chiviacowsky, S., & Wulf, G. (2005). Self-controlled feedback is
silent telesupervision: Leveraging technology for innovative effective if it is based on the learner’s performance. Research
service delivery and clinician training in speech-language Quarterly for Exercise and Sport, 76(1), 42–48. https://doi.org/
pathology during the COVID-19 era. CommonHealth, 1(3), 10.1080/02701367.2005.10599260
106–120. https://doi.org/10.15367/ch.v1i3.413 Chiviacowsky, S., & Wulf, G. (2007). Feedback after good trials
Bergþórsdóttir, Í. Ö., & Ingham, R. J. (2017). Putting the cart enhances learning. Research Quarterly for Exercise and Sport,
before the horse: A cost effectiveness analysis of treatments 78(2), 40–47. https://doi.org/10.1080/02701367.2007.10599402
for stuttering in young children requires evidence that the Chiviacowsky, S., Wulf, G., de Medeiros, F. L., Kaefer, A., &
treatments analyzed were effective. Journal of Communica- Tani, G. (2008). Learning benefits of self-controlled knowl-
tion Disorders, 65, 65–67. https://doi.org/10.1016/j.jcomdis. edge of results in 10-year-old children. Research Quarterly for
2016.04.006 Exercise and Sport, 79(3), 405–410. https://doi.org/10.1080/
Binger, C. (2007). Aided AAC intervention for children with sus- 02701367.2008.10599505
pected childhood apraxia of speech. Perspectives on Augmen- Christensen, E. (2007). Methodology of superiority vs. equiva-
tative and Alternative Communication, 16(1), 10–12. https:// lence trials and non-inferiority trials. Journal of Hepatology,
doi.org/10.1044/aac16.1.10 46(5), 947–954. https://doi.org/10.1016/j.jhep.2007.02.015
Binger, C., Kent-Walsh, J., Berens, J., Del Campo, S., & Rivera, Chua, L. K., Jimenez-Diaz, J., Lewthwaite, R., Kim, T., & Wulf,
D. (2008). Teaching Latino parents to support the multi- G. (2021). Superiority of external attentional focus for motor
symbol message productions of their children who require performance and learning: Systematic reviews and meta-analy-
AAC. Augmentative and Alternative Communication, 24(4), ses. Psychological Bulletin, 147(6), 618–645. https://doi.org/10.
323–338. https://doi.org/10.1080/07434610802130978 1037/bul0000335
Binger, C., Kent-Walsh, J., King, M., & Mansfield, L. (2017). Chumpelik, D. (1984). The PROMPT system of therapy: Theoret-
Early sentence productions of 3- and 4-year-old children who ical framework and applications for developmental apraxia of
use augmentative and alternative communication. Journal of speech. Seminars in Speech and Language, 5(2), 139–156.
Speech, Language, and Hearing Research, 60(7), 1930–1945. https://doi.org/10.1055/s-0028-1085172
https://doi.org/10.1044/2017_JSLHR-L-15-0408 Cook, S. J. (2021). The role of frustration in intensive treatment
Binger, C., & Light, J. (2007). The effect of aided AAC modeling of childhood apraxia of speech [Master’s thesis, Temple Uni-
on the expression of multi-symbol messages by preschoolers versity]. TUScholarShare. https://scholarshare.temple.edu
who use AAC. Augmentative and Alternative Communication, Czajkowski, S. M., Powell, L. H., Adler, N., Naar-King, S.,
23(1), 30–43. https://doi.org/10.1080/07434610600807470 Reynolds, K. D., Hunter, C. M., Laraia, B., Olster, D. H.,
Binger, C., Maguire-Marshall, M., & Kent-Walsh, J. (2011). Perna, F. M., Peterson, J. C., Epel, E., Boyington, J. E., &
Using aided AAC models, recasts, and contrastive targets to Charlson, M. E. (2015). From ideas to efficacy: The ORBIT
teach grammatical morphemes to children who use AAC. model for developing behavioral treatments for chronic dis-
Journal of Speech, Language, and Hearing Research, 54(1), eases. Health Psychology, 34(10), 971–982. https://doi.org/10.
160–176. https://doi.org/10.1044/1092-4388(2010/09-0163) 1037/hea0000161
Bornman, E., Alant, E., & Meiring, E. (2001). The use of a digital Dale, P. S., & Hayden, D. A. (2013). Treating speech subsys-
voice output device to facilitate language development in a tems in childhood apraxia of speech with tactual input:
child with developmental apraxia of speech: A case study. The PROMPT approach. American Journal of Speech-
Disability and Rehabilitation, 23(14), 623–634. https://doi.org/ Language Pathology, 22(4), 644–661. https://doi.org/10.
10.1080/09638280110036517 1044/1058-0360(2013/12-0055)
Byers, B. A., Bellon-Harn, M. L., Allen, M., Saar, K. W., Dollaghan, C. A. (2004). Evidence-based practice in communica-
Manchaiah, V., & Rodrigo, H. (2021). A comparison of tion disorders: What do we know, and when do we know it?

Maas: CAS Treatment Review 21


Downloaded from: https://pubs.asha.org 5.210.215.88 on 06/08/2024, Terms of Use: https://pubs.asha.org/pubs/rights_and_permissions
Journal of Communication Disorders, 37(5), 391–400. https:// conditions in motor learning. Journal of Motor Behavior,
doi.org/10.1016/j.jcomdis.2004.04.002 36(2), 212–224. https://doi.org/10.3200/JMBR.36.2.212-224
Edeal, D. M., & Gildersleeve-Neumann, C. E. (2011). The impor- Hair, A., Ballard, K. J., Markoulli, C., Monroe, P., McKechnie,
tance of production frequency in therapy for childhood apraxia J., Ahmed, B., & Gutierrez-Osuna, R. (2021). A longitudinal
of speech. American Journal of Speech-Language Pathology, evaluation of tablet-based child speech therapy with Apraxia
20(2), 95–110. https://doi.org/10.1044/1058-0360(2011/09-0005) World. ACM Transactions on Accessible Computing (TAC-
Ellis, C. (2009). Does race/ethnicity really matter in adult neuro- CESS), 14(1), 1–26. https://doi.org/10.1145/3433607
genics? American Journal of Speech-Language Pathology, 18(4), Hammer, C. S. (2011). Broadening our knowledge about diverse
310–314. https://doi.org/10.1044/1058-0360(2009/08-0039) populations. American Journal of Speech-Language Pathology,
Fey, M. E., & Finestack, L. H. (2009). Research and develop- 20(2), 71–72. https://doi.org/10.1044/1058-0360(2011/ed-02)
ment in children’s language intervention: A 5-phase model. In Helfrich-Miller, K. R. (1994). A clinical perspective: Melodic
R. G. Schwartz (Ed.), Handbook of child language disorders. intonation therapy for developmental apraxia. Clinics in Com-
The Psychology Press. munication Disorders, 4(3), 175–182.
Ford, I., & Norrie, J. (2016). Pragmatic trials. The New England Hitchcock, E. R., Swartz, M. T., & Cabbage, K. L. (2023). Pre-
Journal of Medicine, 375(5), 454–463. https://doi.org/10.1056/ liminary speech perception performance profiles of school-age
NEJMra1510059 children with childhood apraxia of speech, speech sound dis-
Freedman, S. E., Maas, E., Caligiuri, M. P., Wulf, G., & Robin, order, and typical development. Journal of Speech, Language,
D. A. (2007). Internal versus external: Oral-motor perfor- and Hearing Research. Advance online publication. https://
mance as a function of attentional focus. Journal of Speech, doi.org/10.1044/2023_JSLHR-22-00634
Language, and Hearing Research, 50(1), 131–136. https://doi. Horowitz, C. R., Robinson, M., & Seifer, S. (2009). Community-
org/10.1044/1092-4388(2007/011) based participatory research from the margin to the main-
Fridriksson, J., Basilakos, A., Boyle, M., Cherney, L. R., DeDe, stream: Are researchers prepared? Circulation, 119(19), 2633–
G., Gordon, J. K., Harnish, S. M., Hoover, E. L., Hula, 2642. https://doi.org/10.1161/CIRCULATIONAHA.107.729863
W. D., Pompon, R. H., Johnson, L. P., Kiran, S., Murray, Iuzzini-Seigel, J., Case, J., Grigos, M. I., Velleman, S. L.,
L. L., Rose, M. L., Obermeyer, J., Salis, C., Walker, G. M., Thomas, D., & Murray, E. (2023). Dose frequency random-
& Martin, N. (2022). Demystifying the complexity of aphasia ized controlled trial for dynamic temporal and tactile cueing
treatment: Application of the Rehabilitation Treatment Specifi- (DTTC) treatment for childhood apraxia of speech: Protocol
cation Systemx. Archives of Physical Medicine and Rehabilita- paper. BMC Pediatrics, 23(1), Article 263. https://doi.org/10.
tion, 103(3), 574–580. https://doi.org/10.1016/j.apmr.2021.08.025 1186/s12887-023-04066-2
Gildersleeve-Neumann, C., & Goldstein, B. A. (2015). Cross-lin- Iuzzini-Seigel, J., Delaney, A. L., & Kent, R. D. (2022). Retro-
guistic generalization in the treatment of two sequential spective case–control study of communication and motor abil-
Spanish–English bilingual children with speech sound disor- ities in 143 children with suspected childhood apraxia of
ders. International Journal of Speech-Language Pathology, speech: Effect of concomitant diagnosis. Perspectives of the
17(1), 26–40. https://doi.org/10.3109/17549507.2014.898093 ASHA Special Interest Groups, 7(1), 45–55. https://doi.org/10.
Gomez, M., McCabe, P., Jakielski, K., & Purcell, A. (2018). 1044/2021_PERSP-20-00283
Treating childhood apraxia of speech with the Kaufman Janelle, C. M., Barba, D. A., Frehlich, S. G., Tennant, L. K., &
Speech to Language Protocol: A Phase I pilot study. Lan- Cauraugh, J. H. (1997). Maximizing performance feedback
guage, Speech, and Hearing Services in Schools, 49(3), 524– effectiveness through videotape replay and a self-controlled
536. https://doi.org/10.1044/2018_LSHSS-17-0100 learning environment. Research Quarterly for Exercise and Sport,
Green, B. L., Maisiak, R., Wang, M. Q., Britt, M. F., & Ebeling, 68(4), 269–279. https://doi.org/10.1080/02701367.1997.10608008
N. (1997). Participation in health education, health promotion, Jones, K., & Croot, K. (2016). The effect of blocked, random and
and health research by African Americans: Effects of the Tus- mixed practice schedules on speech motor learning of tongue
kegee Syphilis Experiment. Journal of Health Education, 28(4), twisters in unimpaired speakers. Motor Control, 20(4), 350–
196–201. https://doi.org/10.1080/10556699.1997.10603270 379. https://doi.org/10.1123/mc.2013-0102
Grigos, M. I., Case, J., Lu, Y., & Lyu, Z. (2023). Dynamic tem- Kadis, D. S., Goshulak, D., Namasivayam, A., Pukonen, M.,
poral and tactile cueing: Quantifying speech motor changes Kroll, R., De Nil, L. F., Pang, E. W., & Lerch, J. P. (2014).
and individual factors that contribute to treatment gains in Cortical thickness in children receiving intensive therapy for
childhood apraxia of speech. Journal of Speech, Language, idiopathic apraxia of speech. Brain Topography, 27(2), 240–
and Hearing Research. Advance online publication. https:// 247. https://doi.org/10.1007/s10548-013-0308-8
doi.org/10.1044/2023_JSLHR-22-00658 Kaspi, A., Hildebrand, M. S., Jackson, V. E., Braden, R., van
Grigos, M. I., Hayden, D., & Eigen, J. (2010). Perceptual and Reyk, O., Howell, T., Debono, S., Lauretta, M., Morison, L.,
articulatory changes in speech production following PROMPT Coleman, M. J., Webster, R., Coman, D., Goel, H., Wallis,
treatment. Journal of Medical Speech-Language Pathology, M., Dabscheck, G., Downie, L., Baker, E. K., Parry-Fielder,
18(4), 46–53. B., Ballard, K., . . . Morgan, A. T. (2023). Genetic aetiologies
Grigos, M. I., & Kolenda, N. (2010). The relationship between for childhood speech disorder: Novel pathways co-expressed
articulatory control and improved phonemic accuracy in during brain development. Molecular Psychiatry, 28(4), 1647–
childhood apraxia of speech: A longitudinal case study. Clini- 1663. https://doi.org/10.1038/s41380-022-01764-8
cal Linguistics & Phonetics, 24(1), 17–40. https://doi.org/10. Kaufman, N. (2013). Kaufman Speech to Language Protocol treat-
3109/02699200903329793 ment kits 1 & 2 [Manual]. Northern Speech Services.
Grosjean, F. (2012). Bilingualism: A short introduction. In F. Kearney, E., Granata, F., Yunusova, Y., van Lieshout, P.,
Grosjean & P. Li (Eds.), The psycholinguistics of bilingualism Hayden, D., & Namasivayam, A. (2015). Outcome measures
(pp. 5–25). Wiley. in developmental speech sound disorders with a motor basis.
Guadagnoli, M. A., & Lee, T. D. (2004). Challenge point: A Current Developmental Disorders Reports, 2(3), 253–272.
framework for conceptualizing the effects of various practice https://doi.org/10.1007/s40474-015-0058-2

22 Journal of Speech, Language, and Hearing Research  1–26

Downloaded from: https://pubs.asha.org 5.210.215.88 on 06/08/2024, Terms of Use: https://pubs.asha.org/pubs/rights_and_permissions


Keller, S., & Maas, E. (2023). Self-reported communication atti- Lundeborg, I., & McAllister, A. (2007). Treatment with a combi-
tudes of children with childhood apraxia of speech: An nation of intra-oral sensory stimulation and electropalatogra-
exploratory study. American Journal of Speech-Language phy in a child with severe developmental dyspraxia. Logope-
Pathology, 32(4S), 1806–1824. https://doi.org/10.1044/2022_ dics, Phoniatrics, Vocology, 32(2), 71–79. https://doi.org/10.
AJSLP-22-00163 1080/14015430600852035
Kent-Walsh, J., Binger, C., & Buchanan, C. (2015). Teaching chil- Maas, E., Butalla, C. E., & Farinella, K. A. (2012). Feedback fre-
dren who use augmentative and alternative communication to quency in treatment for childhood apraxia of speech. Ameri-
ask inverted yes/no questions using aided modeling. American can Journal of Speech-Language Pathology, 21(3), 239–257.
Journal of Speech-Language Pathology, 24(2), 222–236. https://doi.org/10.1044/1058-0360(2012/11-0119)
https://doi.org/10.1044/2015_AJSLP-14-0066 Maas, E., Caspari, S., Beiting, M., Gildersleeve-Neumann, C.,
Korkalainen, J., McCabe, P., Smidt, A., & Morgan, C. (2023). Niculae-Caxi, T., Stoeckel, R., & Wu, J. (2022, July). Virtual
The effectiveness of Rapid Syllable Transition treatment in ASSIST speech therapy for childhood apraxia of speech
improving communication in children with cerebral palsy: A (CAS): Effects of intensity on caregiver-rated functional out-
randomized controlled trial. Developmental Neurorehabilitation, comes [Paper presentation]. Childhood Apraxia of Speech
26(5), 309–319. https://doi.org/10.1080/17518423.2023.2218485 Research Symposium, Las Vegas, NV, United States.
LaGasse, B. (2012). Evaluation of melodic intonation therapy for Maas, E., Caspari, S., Beiting, M., Gildersleeve-Neumann, C.,
developmental apraxia of speech. Music Therapy Perspectives, Niculae-Caxi, T., Stoeckel, R., & Wu, J. (2023, November
30(1), 49–55. https://doi.org/10.1093/mtp/30.1.49 15–18). Effects of treatment intensity in virtual speech therapy
Lai, Q., Shea, C. H., Wulf, G., & Wright, D. L. (2000). Optimiz- for childhood apraxia of speech (CAS) [Paper presentation].
ing generalized motor program and parameter learning. American Speech-Language-Hearing Association Convention,
Research Quarterly for Exercise and Sport, 71(1), 10–24. Boston, MA, United States.
https://doi.org/10.1080/02701367.2000.10608876 Maas, E., Caspari, S., Beiting, M., Gildersleeve-Neumann, C. E.,
Leonhartsberger, S., Huber, E., Brandstötter, G., Stoeckel, R., Stoeckel, R., & Wu, J. (2022, November 17–19). Initial treat-
Baas, B., Weber, C., & Holzinger, D. (2022). Efficacy of treat- ment efficacy of ASSIST for childhood apraxia of speech: A
ment intensity in German-speaking children with childhood randomized controlled trial [Paper presentation]. American Speech-
apraxia of speech. Child Language Teaching and Therapy, Language-Hearing Association Convention, New Orleans, LA,
38(1), 43–58. https://doi.org/10.1177/02656590211035156 United States.
Levy, E. S., Chang, Y. M., Hwang, K., & McAuliffe, M. J. Maas, E., & Farinella, K. A. (2012). Random versus blocked
(2021). Perceptual and acoustic effects of dual-focus speech practice in treatment for childhood apraxia of speech. Journal
treatment in children with dysarthria. Journal of Speech, Lan- of Speech, Language, and Hearing Research, 55(2), 561–578.
guage, and Hearing Research, 64(6S), 2301–2316. https://doi. https://doi.org/10.1044/1092-4388(2011/11-0120)
org/10.1044/2020_JSLHR-20-00301 Maas, E., Gildersleeve-Neumann, C., Jakielski, K., Kovacs, N.,
Lewis, B. A., Benchek, P., Tag, J., Miller, G., Freebairn, L., Stoeckel, R., Vradelis, H., & Welsh, M. (2019). Bang for your
Taylor, H. G., Iyengar, S. K., & Stein, C. M. (2021). Psycho- buck: A single-case experimental design study of practice
social comorbidities in adolescents with histories of childhood amount and distribution in treatment for childhood apraxia of
apraxia of speech. American Journal of Speech-Language speech. Journal of Speech, Language, and Hearing Research,
Pathology, 30(6), 2572–2588. https://doi.org/10.1044/2021_ 62(9), 3160–3182. https://doi.org/10.1044/2019_JSLHR-S-18-0212
AJSLP-21-00035 Maas, E., Gildersleeve-Neumann, C. E., Jakielski, K. J., &
Lewis, B. A., Freebairn, L. A., Hansen, A. J., Iyengar, S. K., & Stoeckel, R. (2014). Motor-based intervention protocols in
Taylor, H. G. (2004). School-age follow-up of children with treatment of childhood apraxia of speech (CAS). Current
childhood apraxia of speech. Language, Speech, and Hearing Developmental Disorders Reports, 1(3), 197–206. https://doi.
Services in Schools, 35(2), 122–140. https://doi.org/10.1044/ org/10.1007/s40474-014-0016-4
0161-1461(2004/014) Maas, E., Robin, D. A., Austermann Hula, S. N., Freedman,
Lewthwaite, R., Chiviacowsky, S., Drews, R., & Wulf, G. (2015). S. E., Wulf, G., Ballard, K. J., & Schmidt, R. A. (2008). Prin-
Choose to move: The motivational impact of autonomy sup- ciples of motor learning in treatment of motor speech disor-
port on motor learning. Psychonomic Bulletin & Review, ders. American Journal of Speech-Language Pathology, 17(3),
22(5), 1383–1388. https://doi.org/10.3758/s13423-015-0814-7 277–298. https://doi.org/10.1044/1058-0360(2008/025)
Lewthwaite, R., & Wulf, G. (2010). Social-comparative feedback Mailend, M.-L., & Maas, E. (2021). To lump or to split? Possible
affects motor skill learning. Quarterly Journal of Experi- subtypes of apraxia of speech. Aphasiology, 35(4), 592–613.
mental Psychology, 63(4), 738–749. https://doi.org/10.1080/ https://doi.org/10.1080/02687038.2020.1836319
17470210903111839 Martikainen, A. L., & Korpilahti, P. (2011). Intervention for
Lim, J., McCabe, P., & Purcell, A. (2019). ‘Another tool in my childhood apraxia of speech: A single-case study. Child Lan-
toolbox’: Training school teaching assistants to use dynamic guage Teaching and Therapy, 27(1), 9–20. https://doi.org/10.
temporal and tactile cueing with children with childhood 1177/0265659010369985
apraxia of speech. Child Language Teaching and Therapy, McAllister Byun, T., Swartz, M. T., Halpin, P. F., Szeredi, D., &
35(3), 241–256. https://doi.org/10.1177/0265659019874858 Maas, E. (2016). Direction of attentional focus in biofeedback
Lisman, A. L., & Sadagopan, N. (2013). Focus of attention and treatment for /r/ misarticulation. International Journal of Lan-
speech motor performance. Journal of Communication Disorders, guage & Communication Disorders, 51(4), 384–401. https://doi.
46(3), 281–293. https://doi.org/10.1016/j.jcomdis.2013.02.002 org/10.1111/1460-6984.12215
Littlejohn, M., & Maas, E. (2023). How to cut the pie is no piece McCabe, P., Macdonald-D’Silva, A. G., van Rees, L. J., Ballard,
of cake: Toward a process-oriented approach to assessment K. J., & Arciuli, J. (2014). Orthographically sensitive treatment
and diagnosis of speech sound disorders. International Journal for dysprosody in children with childhood apraxia of speech
of Language & Communication Disorders. Advance online using ReST intervention. Developmental Neurorehabilitation,
publication. https://doi.org/10.1111/1460-6984.12934 17(2), 137–145. https://doi.org/10.3109/17518423.2014.906002

Maas: CAS Treatment Review 23


Downloaded from: https://pubs.asha.org 5.210.215.88 on 06/08/2024, Terms of Use: https://pubs.asha.org/pubs/rights_and_permissions
McCabe, P., Preston, J., Maas, E., Purcell, A., Thomas, D., Journal of Speech-Language Pathology, 28(4), 1432–1447.
Murray, E., Gomez, M., Heard, R., & Cronin, P. (2022, Feb- https://doi.org/10.1044/2019_AJSLP-18-0132
ruary 16–20). Protocol for a two-arm cross-over randomized Miller, H. E., Ballard, K. J., Campbell, J., Smith, M., Plante,
control trial examining dynamic temporal and tactile cueing A. S., Aytur, S. A., & Robin, D. A. (2021). Improvements in
(DTTC) and usual care in childhood apraxia of speech treat- speech of children with apraxia: The efficacy of Treatment for
ment [Poster presentation]. Conference on Motor Speech, Establishing Motor Program Organization (TEMPOSM).
Charleston, SC, United States. Developmental Neurorehabilitation, 24(7), 494–509. https://doi.
McCabe, P., Preston, J. L., Evans, P., & Heard, R. (2023). A org/10.1080/17518423.2021.1916113
pilot randomized control trial of motor-based treatments for Morgan, P. L., Hammer, C. S., Farkas, G., Hillemeier, M. M.,
childhood apraxia of speech: Rapid Syllable Transition treat- Maczuga, S., Cook, M., & Morano, S. (2016). Who receives
ment and ultrasound biofeedback. American Journal of speech/language services by 5 years of age in the United
Speech-Language Pathology, 32(2), 629–644. https://doi.org/ States? American Journal of Speech-Language Pathology,
10.1044/2022_AJSLP-22-00144 25(2), 183–199. https://doi.org/10.1044/2015_AJSLP-14-0201
McCabe, P., Thomas, D. C., & Murray, E. (2020). Rapid Sylla- Moriarty, B. C., & Gillon, G. T. (2006). Phonological awareness
ble Transition treatment—A treatment for childhood apraxia intervention for children with childhood apraxia of speech.
of speech and other pediatric motor speech disorders. Perspec- International Journal of Language & Communication Disorders,
tives of the ASHA Special Interest Groups, 5(4), 821–830. 41(6), 713–734. https://doi.org/10.1080/13682820600623960
https://doi.org/10.1044/2020_PERSP-19-00165 Morley, M., Court, D., Miller, H., & Garside, R. F. (1955).
McCormack, J., McLeod, S., & Crowe, K. (2019). What do chil- Delayed speech and developmental aphasia. British Medical
dren with speech sound disorders think about their talking? Journal, 2(4937), 463–467. https://doi.org/10.1136/bmj.2.4937.463
Seminars in Speech and Language, 40(2), 94–104. https://doi. Morley, M. E. (1969). Disorders of articulation: Theory and ther-
org/10.1055/s-0039-1677760 apy. British Journal of Disorders of Communication, 4(2), 151–
McCormack, J., McLeod, S., Harrison, L. J., & Holliday, E. L. 165. https://doi.org/10.3109/13682826909011484
(2022). Drawing talking: Listening to children with speech Murad, M. H., Asi, N., Alsawas, M., & Alahdab, F. (2016). New
sound disorders. Language, Speech, and Hearing Services in evidence pyramid. Evidence-Based Medicine, 21(4), 125–127.
Schools, 53(3), 713–731. https://doi.org/10.1044/2021_LSHSS- https://doi.org/10.1136/ebmed-2016-110401
21-00140 Murray, E., Iuzzini-Seigel, J., Maas, E., Terband, H., & Ballard,
McCormack, J., McLeod, S., McAllister, L., & Harrison, L. J. K. J. (2021). Differential diagnosis of childhood apraxia of
(2010). My speech problem, your listening problem, and my speech compared to other speech sound disorders: A system-
frustration: The experience of living with childhood speech atic review. American Journal of Speech-Language Pathology,
impairment. Language, Speech, and Hearing Services in 30(1), 279–300. https://doi.org/10.1044/2020_AJSLP-20-00063
Schools, 41(4), 379–392. https://doi.org/10.1044/0161-1461(2009/ Murray, E., McCabe, P., & Ballard, K. J. (2014). A systematic
08-0129) review of treatment outcomes for children with childhood
McKechnie, J., Ahmed, B., Gutierrez-Osuna, R., Murray, E., apraxia of speech. American Journal of Speech-Language Pathol-
McCabe, P., & Ballard, K. J. (2020). The influence of type ogy, 23(3), 486–504. https://doi.org/10.1044/2014_AJSLP-13-0035
of feedback during tablet-based delivery of intensive treat- Murray, E., McCabe, P., & Ballard, K. J. (2015). A randomized
ment for childhood apraxia of speech. Journal of Communi- controlled trial for children with childhood apraxia of speech
cation Disorders, 87, Article 106026. https://doi.org/10.1016/ comparing Rapid Syllable Transition treatment and the Nuf-
j.jcomdis.2020.106026 field Dyspraxia Programme–Third Edition. Journal of Speech,
McNeill, B. C., Gillon, G. T., & Dodd, B. (2009a). A longitudinal Language, and Hearing Research, 58(3), 669–686. https://doi.
case study of the effects of an integrated phonological awareness org/10.1044/2015_JSLHR-S-13-0179
program for identical twin boys with childhood apraxia of Nakamura-Palacios, E. M., Falçoni Júnior, A. T., Tanese, G. L.,
speech (CAS). International Journal of Speech-Language Pathol- Vogeley, A. C. E., & Namasivayam, A. K. (2024). Enhancing
ogy, 11(6), 482–495. https://doi.org/10.3109/17549500902842583 speech rehabilitation in a young adult with Trisomy 21: Integrat-
McNeill, B. C., Gillon, G. T., & Dodd, B. (2009b). Effectiveness ing transcranial direct current stimulation (tDCS) with Rapid
of an integrated phonological awareness approach for children Syllable Transition training for apraxia of speech. Brain Sci-
with childhood apraxia of speech (CAS). Child Language ences, 14(1), Article 58. https://doi.org/10.3390/brainsci14010058
Teaching and Therapy, 25(3), 341–366. https://doi.org/10.1177/ Namasivayam, A. K., Pukonen, M., Goshulak, D., Hard, J.,
0265659009339823 Rudzicz, F., Rietveld, T., Maassen, B., Kroll, R., & van
McNeill, B. C., Gillon, G. T., & Dodd, B. (2010). The longer Lieshout, P. (2015). Treatment intensity and childhood
term effects of an integrated phonological awareness interven- apraxia of speech. International Journal of Language & Com-
tion for children with childhood apraxia of speech. Asia munication Disorders, 50(4), 529–546. https://doi.org/10.1111/
Pacific Journal of Speech, Language, and Hearing, 13(3), 145– 1460-6984.12154
161. https://doi.org/10.1179/136132810805335074 Ng, W. L., McCabe, P., Heard, R., Park, V., Murray, E., &
Mehta, J., & Moorer, L. (2022, July 7–9). Effectiveness of trans- Thomas, D. (2022). Predicting treatment outcomes in Rapid
cranial direct current stimulation (tDCS) in childhood apraxia Syllable Transition treatment: An individual participant data
of speech (CAS) [Paper presentation]. 2022 Apraxia Kids meta-analysis. Journal of Speech, Language, and Hearing
Research Symposium, Las Vegas, NV, United States. Research, 65(5), 1784–1799. https://doi.org/10.1044/2022_
Milisen, R. (1954). A rationale for articulation disorders. Journal of JSLHR-21-00617
Speech and Hearing Disorders, 4(Monograph Supplement), 6–17. OCEBM Levels of Evidence Working Group. (2011). The Oxford
Miller, G. J., Lewis, B., Benchek, P., Freebairn, L., Tag, J., 2011 levels of evidence. Oxford Centre for Evidence-Based
Budge, K., Iyengar, S. K., Voss-Hoynes, H., Taylor, H. G., & Medicine. http://www.cebm.net/index.aspx?o=5653
Stein, C. (2019). Reading outcomes for individuals with histo- Owen, R., Hayett, L., & Roulstone, S. (2004). Children’s views of
ries of suspected childhood apraxia of speech. American speech and language therapy in school: Consulting children with

24 Journal of Speech, Language, and Hearing Research  1–26

Downloaded from: https://pubs.asha.org 5.210.215.88 on 06/08/2024, Terms of Use: https://pubs.asha.org/pubs/rights_and_permissions


communication difficulties. Child Language Teaching and Ther- Robey, R. R. (2004). A five-phase model for clinical-outcome
apy, 20(1), 55–73. https://doi.org/10.1191/0265659004ct263oa research. Journal of Communication Disorders, 37(5), 401–411.
Parnandi, A., Karappa, V., Lan, T., Shahin, M., McKechnie, J., https://doi.org/10.1016/j.jcomdis.2004.04.003
Ballard, K., Ahmed, B., & Gutierrez-Osuna, R. (2015). Devel- Rosenbek, J., Hansen, R., Baughman, C. H., & Lemme, M.
opment of a remote therapy tool for childhood apraxia of (1974). Treatment of developmental apraxia of speech: A case
speech. ACM Transactions on Accessible Computing (TAC- study. Language, Speech, and Hearing Services in Schools,
CESS), 7(3), 1–23. https://doi.org/10.1145/2776895 5(1), 13–22. https://doi.org/10.1044/0161-1461.0501.13
Patall, E. A., Cooper, H., & Robinson, J. C. (2008). The effects Rosenbek, J. C. (2016). Tyranny of the randomised clinical trial.
of choice on intrinsic motivation and related outcomes: A International Journal of Speech-Language Pathology, 18(3),
meta-analysis of research findings. Psychological Bulletin, 241–249. https://doi.org/10.3109/17549507.2015.1126644
134(2), 270–300. https://doi.org/10.1037/0033-2909.134.2.270 Rosenbek, J. C., & Wertz, R. T. (1972). A review of fifty cases of
Peter, B., Davis, J., Finestack, L., Stoel-Gammon, C., VanDam, developmental apraxia of speech. Language, Speech, and
M., Bruce, L., Kim, Y., Eng, L., Cotter, S., Landis, E., Hearing Services in Schools, 3(1), 23–33. https://doi.org/10.
Beames, S., Scherer, N., Knerr, I., Williams, D., Schrock, C., 1044/0161-1461.0301.23
& Potter, N. (2022). Translating principles of precision medi- Rosenbek, J. C., & Wertz, R. T. (1976). Treatment of apraxia of
cine into speech-language pathology: Clinical trial of a proac- speech in adults. In R. H. Brookshire (Ed.), Clinical aphasiol-
tive speech and language intervention for infants with classic ogy: Conference proceedings (pp. 191–198). BRK Publishers.
galactosemia. Human Genetics and Genomics Advances, 3(3), Roulstone, S. (2011). Evidence, expertise, and patient preference
Article 100119. https://doi.org/10.1016/j.xhgg.2022.100119 in speech-language pathology. International Journal of Speech-
Peter, B., Potter, N., Davis, J., Donenfeld-Peled, I., Finestack, L., Language Pathology, 13(1), 43–48. https://doi.org/10.3109/
Stoel-Gammon, C., Lien, K., Bruce, L., Vose, C., Eng, L., 17549507.2010.491130
Yokoyama, H., Olds, D., & VanDam, M. (2019). Toward a Rusiewicz, H. L., Maize, K., & Ptakowski, T. (2018). Parental
paradigm shift from deficit-based to proactive speech and lan- experiences and perceptions related to childhood apraxia of
guage treatment: Randomized pilot trial of the Babble Boot speech: Focus on functional implications. International Jour-
Camp in infants with classic galactosemia. F1000Research, 8, nal of Speech-Language Pathology, 20(5), 569–580. https://doi.
Article 271. https://doi.org/10.12688/f1000research.18062.2 org/10.1080/17549507.2017.1359333
Porter, J. M., & Magill, R. A. (2010). Systematically increasing Rvachew, S., & Matthews, T. (2019). An N-of-1 randomized con-
contextual interference is beneficial for learning sport skills. trolled trial of interventions for children with inconsistent
Journal of Sports Sciences, 28(12), 1277–1285. https://doi.org/ speech sound errors. Journal of Speech, Language, and Hear-
10.1080/02640414.2010.502946 ing Research, 62(9), 3183–3203. https://doi.org/10.1044/2019_
Potkovac, G. (2020). Feedback control in treatment for apraxia of JSLHR-S-18-0288
speech [Master’s thesis, Temple University]. TUScholarShare. Scarcella, I., Michelazzo, L., & McCabe, P. (2021). A pilot single-
https://scholarshare.temple.edu/ case experimental design study of Rapid Syllable Transition
Preston, J. L., Brick, N., & Landi, N. (2013). Ultrasound bio- treatment for Italian children with childhood apraxia of speech.
feedback treatment for persisting childhood apraxia of speech. American Journal of Speech-Language Pathology, 30(3S), 1496–
American Journal of Speech-Language Pathology, 22(4), 627– 1510. https://doi.org/10.1044/2021_AJSLP-20-00133
643. https://doi.org/10.1044/1058-0360(2013/12-0139) Shavers, V. L., Lynch, C. F., & Burmeister, L. F. (2000). Knowl-
Preston, J. L., Caballero, N. F., Leece, M. C., Wang, D., Herbst, edge of the Tuskegee study and its impact on the willingness
B. M., & Benway, N. R. (2023). A randomized controlled to participate in medical research studies. Journal of the
trial of treatment distribution and biofeedback effects on National Medical Association, 92(12), 563–572.
speech production in school-age children with apraxia of Shea, C. H., Lai, Q., Wright, D. L., Immink, M., & Black, C.
speech. Journal of Speech, Language, and Hearing Research. (2001). Consistent and variable practice conditions: Effects on
Advance online publication. https://doi.org/10.1044/2023_ relative and absolute timing. Journal of Motor Behavior,
JSLHR-22-00622 33(2), 139–152. https://doi.org/10.1080/00222890109603146
Preston, J. L., Leece, M. C., & Maas, E. (2016). Intensive treat- Shea, C. H., & Wulf, G. (1999). Enhancing motor learning
ment with ultrasound visual feedback for speech sound errors through external-focus instructions and feedback. Human
in childhood apraxia. Frontiers in Human Neuroscience, 10, Movement Science, 18(4), 553–571. https://doi.org/10.1016/
Article 440. https://doi.org/10.3389/fnhum.2016.00440 S0167-9457(99)00031-7
Preston, J. L., Leece, M. C., McNamara, K., & Maas, E. (2017). Shriberg, L. D., Lohmeier, H. L., Strand, E. A., & Jakielski,
Variable practice to enhance speech learning in ultrasound K. J. (2012). Encoding, memory, and transcoding deficits in
biofeedback treatment for childhood apraxia of speech: A sin- childhood apraxia of speech. Clinical Linguistics & Phonetics,
gle case experimental study. American Journal of Speech- 26(5), 445–482. https://doi.org/10.3109/02699206.2012.655841
Language Pathology, 26(3), 840–852. https://doi.org/10.1044/ Simon-Cereijido, G. (2018). Bilingualism, a human right in times
2017_AJSLP-16-0155 of anxiety: Lessons from California. International Journal of
Preston, J. L., Maas, E., Whittle, J., Leece, M. C, & McCabe, P. Speech-Language Pathology, 20(1), 157–160. https://doi.org/
(2016). Limited acquisition and generalisation of rhotics with 10.1080/17549507.2018.1392610
ultrasound visual feedback in childhood apraxia. Clinical Lin- Skelton, S. L., & Hagopian, A. L. (2014). Using randomized vari-
guistics & Phonetics, 30(3–5), 363–381. https://doi.org/10.3109/ able practice in the treatment of childhood apraxia of speech.
02699206.2015.1052563 American Journal of Speech-Language Pathology, 23(4), 599–
Quach, W., Ball, L. J., Rupp, D., & Beukelman, D. R. (2006). A 611. https://doi.org/10.1044/2014_AJSLP-12-0169
computer software prototype for monitoring speech motor Skelton, S. L., & Richard, J. T. (2016). Application of a motor
learning performance in (childhood) apraxia of speech (techni- learning treatment for speech sound disorders in small groups.
cal report). Journal of Medical Speech-Language Pathology, Perceptual and Motor Skills, 122(3), 840–854. https://doi.org/
14(4), 285–290. 10.1177/0031512516647693

Maas: CAS Treatment Review 25


Downloaded from: https://pubs.asha.org 5.210.215.88 on 06/08/2024, Terms of Use: https://pubs.asha.org/pubs/rights_and_permissions
Skoog, K., & Maas, E. (2020). Predicting intelligibility: An Tourville, J. A., & Guenther, F. H. (2011). The DIVA model: A
investigation of speech sound accuracy in childhood apraxia neural theory of speech acquisition and production. Language
of speech. CommonHealth, 1(2), 44–56. https://doi.org/10.15367/ and Cognitive Processes, 26(7), 952–981. https://doi.org/10.
ch.v1i2.397 1080/01690960903498424
Strand, E. A. (2020). Dynamic temporal and tactile cueing: A U.S. Census Bureau. (2022). Language Spoken at Home. American
treatment strategy for childhood apraxia of speech. American Community Survey, ACS 1-Year Estimates Subject Tables,
Journal of Speech-Language Pathology, 29(1), 30–48. https:// Table S1601. https://data.census.gov/table/ACSST1Y2022.S1601?q=
doi.org/10.1044/2019_AJSLP-19-0005 language
Strand, E. A., & Debertine, P. (2000). The efficacy of integral Valentine, H., MacAuslan, J., Grigos, M., & Speights, M. (2023).
stimulation intervention with developmental apraxia of My vowels matter: Formant automation tools for diverse
speech. Journal of Medical Speech-Language Pathology, 8, child speech. Proceedings of Interspeech, 674–675.
295–300. van der Merwe, A., & Steyn, M. (2018). Model-driven treatment
Strand, E. A., Stoeckel, R., & Baas, B. (2006). Treatment of of childhood apraxia of speech: Positive effects of the speech
severe childhood apraxia of speech: A treatment efficacy study. motor learning approach. American Journal of Speech-
Journal of Medical Speech-Language Pathology, 14, 297–307. Language Pathology, 27(1), 37–51. https://doi.org/10.1044/
Swartz, M. T. (2022). Exploring vowel space metrics and quality 2017_AJSLP-15-0193
of life measures in adolescents with typical speech, residual van Tellingen, M., Hurkmans, J., Terband, H., van de Zande,
speech sound disorder, and childhood apraxia of speech [Doctoral A. M., Maassen, B., & Jonkers, R. (2023). Speech and music
dissertation, Montclair State University]. Theses, Dissertations therapy in the treatment of childhood apraxia of speech: An
and Culminating Projects, Article 1068. https://digitalcommons. introduction and a case study. Journal of Speech, Language,
montclair.edu/etd/1068 and Hearing Research. Advance online publication. https://
Terband, H., Maassen, B., Guenther, F. H., & Brumberg, J. doi.org/10.1044/2023_JSLHR-22-00619
(2014). Auditory–motor interactions in pediatric motor speech Wang, E. W., & Grigos, M. I. (2023). Naive listener ratings of
disorders: Neurocomputational modeling of disordered devel- speech intelligibility over the course of motor-based interven-
opment. Journal of Communication Disorders, 47, 17–33. tion in children with childhood apraxia of speech. Journal of
https://doi.org/10.1016/j.jcomdis.2014.01.001 Speech, Language, and Hearing Research. Advance online pub-
Terband, H., Maassen, B., & Maas, E. (2019). A psycholinguistic lication. https://doi.org/10.1044/2023_JSLHR-22-00656
framework for diagnosis and treatment planning of develop- Williams, P., & Stephens, H. (2010). The Nuffield Centre Dys-
mental speech disorders. Folia Phoniatrica et Logopaedica, praxia Programme. In A. L. Williams, S. McLeod, & R. J.
71(5–6), 216–227. https://doi.org/10.1159/000499426 McCauley (Eds.), Interventions for speech sound disorders in
Thomas, D., Murray, E., Williamson, E., & McCabe, P. (2023). children (pp. 159–177). Brookes.
Weekly treatment for childhood apraxia of speech with Rapid Wong, E. C. H., Wong, M. N., Velleman, S. L., Tong, M. C. F.,
Syllable Transition treatment: A single-case experimental & Lee, K. Y. S. (2023). Lexical tone perception and production
design study. Journal of Speech, Language, and Hearing in Cantonese-speaking children with childhood apraxia of
Research. Advance online publication. https://doi.org/10.1044/ speech: A pilot study. Clinical Linguistics & Phonetics, 37(4–6),
2023_JSLHR-22-00665 316–329. https://doi.org/10.1080/02699206.2022.2074310
Thomas, D. C., McCabe, P., & Ballard, K. J. (2014). Rapid Syllable World Health Organization. (2007). ICF-CY: International Classi-
Transitions (ReST) treatment for childhood apraxia of speech: fication of Functioning, Disability and Health: Children and
The effect of lower dose-frequency. Journal of Communication Dis- youth version. https://iris.who.int/handle/10665/43737
orders, 51, 29–42. https://doi.org/10.1016/j.jcomdis.2014.06.004 Wulf, G., Lauterbach, B., & Toole, T. (1999). The learning
Thomas, D. C., McCabe, P., & Ballard, K. J. (2018). Combined advantages of an external focus of attention in golf. Research
clinician–parent delivery of Rapid Syllable Transition (ReST) Quarterly for Exercise and Sport, 70(2), 120–126. https://doi.
treatment for childhood apraxia of speech. International Jour- org/10.1080/02701367.1999.10608029
nal of Speech-Language Pathology, 20(7), 683–698. https://doi. Wulf, G., & Lewthwaite, R. (2016). Optimizing performance
org/10.1080/17549507.2017.1316423 through intrinsic motivation and attention for learning: The OPTI-
Thomas, D. C., McCabe, P., Ballard, K. J., & Lincoln, M. MAL theory of motor learning. Psychonomic Bulletin & Review,
(2016). Telehealth delivery of Rapid Syllable Transitions 23(5), 1382–1414. https://doi.org/10.3758/s13423-015-0999-9
(ReST) treatment for childhood apraxia of speech. Interna- Wulf, G., McNevin, N., & Shea, C. H. (2001). The automaticity
tional Journal of Language & Communication Disorders, 51(6), of complex motor skill learning as a function of attentional
654–671. https://doi.org/10.1111/1460-6984.12238 focus. The Quarterly Journal of Experimental Psychology,
Thomas-Stonell, N., Robertson, B., Walker, J., Oddson, B., 54(4), 1143–1154. https://doi.org/10.1080/713756012
Washington, K., & Rosenbaum, P. (2012). FOCUS©: Focus on Yoss, K. A., & Darley, F. L. (1974). Therapy in developmental
the Outcomes of Communication Under Six. Holland Bloorview apraxia of speech. Language, Speech, and Hearing Services in
Kids Rehabilitation Hospital. Schools, 5(1), 23–31. https://doi.org/10.1044/0161-1461.0501.23

26 Journal of Speech, Language, and Hearing Research  1–26

Downloaded from: https://pubs.asha.org 5.210.215.88 on 06/08/2024, Terms of Use: https://pubs.asha.org/pubs/rights_and_permissions

You might also like