Differential Diagnosis of Children With Suspected Childhood Apraxia of Speech

JSLHR
Research Article
Differential Diagnosis of Children with

Suspected Childhood Apraxia of Speech
Elizabeth Murray,a Patricia McCabe,a Robert Heard,a and Kirrie J. Ballarda
Purpose: The gold standard for diagnosing childhood

apraxia of speech (CAS) is expert judgment of perceptual
features. The aim of this study was to identify a set of
objective measures that differentiate CAS from other speech
disorders.
Method: Seventy-two children (412 years of age)
diagnosed with suspected CAS by community speechlanguage pathologists were screened. Forty-seven
participants underwent diagnostic assessment including
presence or absence of perceptual CAS features. Twentyeight children met two sets of diagnostic criteria for CAS
(American Speech-Language-Hearing Association, 2007b;
Shriberg, Potter, & Strand, 2009); another 4 met the CAS
criteria with comorbidity. Fifteen were categorized as
non-CAS with phonological impairment, submucous cleft,
or dysarthria. Following this, 24 different measures from

the diagnostic assessment were rated by blinded raters.
Multivariate discriminant function analysis was used to
identify the combination of measures that best predicted
expert diagnoses.
Results: The discriminant function analysis model, including
syllable segregation, lexical stress matches, percentage
phonemes correct from a polysyllabic picture-naming task,
and articulatory accuracy on repetition of /ptk/, reached
91% diagnostic accuracy against expert diagnosis.
Conclusions: Polysyllabic production accuracy and an oral
motor examination that includes diadochokinesis may be
sufficient to reliably identify CAS and rule out structural
abnormality or dysarthria. Testing with a larger unselected
sample is required.
it remains difficult to differentially diagnose CAS from

other disorders. In the absence of a clinically available validated assessment procedure, the current gold standard for
diagnosis is expert opinion (Maas, Butalla, & Farinella,
2012). The purpose of this study was to determine, after expert diagnosis, if a quantitative measure or set of measures
differentiated CAS from non-CAS in a sample of children
referred from the community with suspected CAS.
hildhood apraxia of speech (CAS) is considered an

impairment of speech motor control or praxis. Most
researchers agree that the core deficit for children
with CAS is a reduced or degraded ability to convert abstract
phonological codes to motor speech commands, referred to
as motor planning and/or programming (American SpeechLanguage-Hearing Association [ASHA], 2007b; Nijland,
Maassen, & Van der Meulen, 2003; Shriberg, Lohmeier,
Strand, & Jakielski, 2012). This consensus has been supported
by behavioral studies (see ASHA, 2007b, and McCauley,
Tambyraja, & Daher-Twersky, 2012, for reviews), classification paradigms (Shriberg et al., 2010), and computational
modeling studies (Terband & Maassen, 2010; Terband,
Maassen, Guenther, & Brumberg, 2009). The impairment
then manifests itself as disordered articulation, difficulty
sequencing sounds and syllables, inconsistent production of
repeated sounds and syllables, and disruption at the suprasegmental level (i.e., dysprosody; ASHA, 2007b). Despite
the recent advances in our theoretical understanding of CAS,
The University of Sydney, Australia

Correspondence to Elizabeth Murray: Elizabeth.murray@sydney.edu.au
Editor: Jody Kreiman
Associate Editor: Ben A. M. Maassen
Received November 9, 2012
Revision received May 10, 2013
Accepted October 13, 2014
DOI: 10.1044/2014_JSLHR-S-12-0358
Diagnosis of Childhood Apraxia of Speech

CAS may occur as a result of known neurodevelopmental disorders (Kummer, Lee, Stutz, Maroney, & Brandt,
2007; Shriberg, Potter, & Strand, 2011; Spinelli, Rocha,
Giacheti, & Richieri-Costa, 1995) or deleterious genetic
mutation (e.g., Brunetti-Pierri et al., 2011; Carr et al., 2010;
Palka et al., 2012). The majority of reported cases, however,
are idiopathic.
Despite much research, there is currently no single
neurological or behavioral diagnostic marker for all cases
of CAS (ASHA, 2007b). Behavioral measures that have
been considered have included inconsistent speech features (e.g., Iuzzini, 2012), coarticulation and timing errors
(e.g., Sussman, Marquardt, & Doyle, 2000), prosody (e.g.,
Munson, Bjorum, & Windsor, 2003; Thoonen, Maassen,
Wit, Gabrels, & Schreuder, 1996), speech production
Disclosure: The authors have declared that no competing interests existed at the time
of publication.
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43
(e.g., Thoonen, Maassen, Gabrels, & Schreuder, 1999),

speech perception (e.g., Nijland, 2009), linguistic skills (e.g.,
Lewis, Freebairn, Hansen, Iyengar, & Taylor, 2004), and
nonspeech oral motor skills (e.g., Murdoch, Attard, Ozanne,
& Stokes, 1995). Diagnostic marker research has included
many contributions by Shriberg and colleagues utilizing their
Madison Speech Assessment Protocol and Speech Disorders
Classification System (SDCS) to attempt to differentiate
CAS and acquired apraxia of speech from normal development and other communication disorders (e.g., Shriberg,
Aram, & Kwiatkowski, 1997; Shriberg et al., 2010;
Shriberg, Green, Campbell, McSweeny, & Scheer, 2003;
Shriberg, Lohmeier, et al., 2009).
In the absence of a single pathognomonic feature, a
core set of validated differential diagnostic markers for CAS
is needed (ASHA, 2007b; Davis, Jakielski, & Marquardt,
1998). In 2007, ASHA released a position statement and
technical report on CAS (ASHA, 2007a, 2007b). The technical report provided a consensus position on a small set
of perceptual speech features associated with the primary
planning and programming deficits in CAS. Following a
thorough literature review of group-comparison diagnostic
studies and community consultation, three features with
some consensus were identified across investigators in apraxia:
(a) inconsistent errors on consonants and vowels in repeated productions of syllables or words, (b) lengthened and
disrupted coarticulatory transitions between sounds and
syllables, and (c) inappropriate prosody, especially in the
realization of lexical or phrasal stress (ASHA, 2007b,
pp. 4, 54, and 59).1 These features have been used for diagnosis in recent CAS treatment studies in which participants
had to demonstrate all three features to warrant diagnosis
(e.g., Ballard, Robin, McCabe, & McDonald, 2010; Maas
et al., 2012). Another recent checklist that is used for CAS
diagnosis in research and clinical settings is Strands 10-point
checklist (Shriberg et al., 2012; Shriberg, Potter, & Strand,
2009). This checklist provides 10 segmental and suprasegmental features that can be present in CAS, although
any combination of at least four out of the 10 features across
three tasks warrants the diagnosis of CAS.
As Strand and colleagues remarked, feature lists do not
directly lead to an assessment procedure (Strand, McCauley,
Weigand, Stoeckel, & Baas, 2013). Commercial normreferenced assessment tools offer clear assessment tasks
but often lack essential psychometric properties (McCauley &
Strand, 2008) and may also rely on feature lists or scales for
diagnosis of CAS (Blakeley, 2001; Hickman, 1997; Kaufman,
1995). The recently published Dynamic Evaluation of Motor
Skills is reliable but risks failing to identify some persons
with CAS (Strand et al., 2013). To date, there is no accepted, operationally defined, diagnostic testing protocol or
clinically available and validated set of behavioral features,
1
The Royal College of Speech and Language Therapists also released a

Policy Statement with a wider list of consensus features for CAS (Royal
College of Speech and Language Therapists, 2011). This statement was
published after data collection for the current study and was therefore
not considered.
44
with >90% sensitivity (identifying true cases of CAS) and

specificity (identifying true cases of non-CAS), discriminating CAS from a range of other expressive communication
disorders (Shriberg et al., 2012). The closest we have is
the contribution of 89% sensitivity and 100% specificity by
Thoonen and colleagues (1996, 1997, 1999) in discriminating
spastic dysarthria and CAS using maximum performance
tasks. However, the procedure may not discriminate across
subtypes of dysarthria and other speech sound disorders (SSD)
because the validation sample used was small (n = 11).
Developing a reliable testing protocol that captures
the small set of agreed behavioral features of CAS is challenging when one considers a number of methodological
constraints of previous research. These include lack of specificity of features within checklists used for diagnosis of
CAS, various participant selection criteria, insufficient
operationalization of methods for replication, and different
comparison groups. Checklists of features, such as Strands
10-point checklist (Shriberg, Potter, et al., 2009) and others,
guide the clinician in making presence or absence decisions
for a range of speech, oromotor, and linguistic behaviors;
however, for the most part, these checklists do not include
explicit definitions of the features or methods for determining
how much or how often any single behavior must be observed
(e.g., Crary, 1995; Davis & Velleman, 2000; cf. Thoonen
et al., 1999; see Rvachew, Hodge, & Ohberg, 2005, for a tutorial), which affects application or replication of the results.
The wide range of behaviors that have been considered for
these checklists reflects (a) the belief that symptoms of CAS
change with age and/or maturation and (b) the different
theoretical perspectives as to whether CAS has core or cooccurring motor, oral motor, phonological or linguistic
memory, and literacy impairments (Alcock, Passingham,
Watkins, & Vargha-Khadem, 2000; Marion, Sussman, &
Marquardt, 1993; Shriberg et al., 2012). Furthermore, many
surface behavioral features are shared by a range of communication disorders, making discrimination of phonologicallinguistic and phonetic-motoric errors difficult (see Ballard,
Granier, & Robin, 2000; McCabe, Rosenthal, & McLeod,
1998; McNeil, Robin, & Schmidt, 2009).
The methods of participant selection have also differed across studies, contributing to confusion. Some studies
have included participants with CAS and concomitant
hearing impairment, intellectual disability, and language
impairment (Shriberg et al., 2012), whereas others have
used participants with idiopathic CAS and no known
comorbid disorders (e.g., Thoonen et al., 1997). When
comparison groups have been used, specifically selected diagnoses have been compared to CAS. Groups of participants
have included those with speech delay or speech sound
disorder with or without language delay or impairment
(e.g., Lewis et al., 2011; Shriberg, Green, et al., 2003; Shriberg
et al., 2012), dysarthria (e.g., Morley, Court, & Miller,
1954; Rosenbek & Wertz, 1972; Thoonen et al., 1999),
phonological impairment (e.g., Williams, Ingham, &
Rosenthal, 1981; Yoss & Darley, 1974), specific language
impairment (e.g., Lewis et al., 2004), or stuttering (Byrd &
Cooper, 1989). Ultimately a set of quantifiable measures is
Journal of Speech, Language, and Hearing Research Vol. 58 4360 February 2015

needed that will reliably discriminate CAS from a wide

range of communication disorders that could present with
overlapping symptoms, whether within individuals or between groups of individuals.
The predominant approach in current treatment research studies of CAS is, first, to use a range of standardized assessments to rule out frank receptive and expressive
language impairment or orofacial structural abnormality
and, second, to use a checklist to note CAS-type behaviors
observed and so justify a diagnosis of CAS (e.g., Ballard
et al., 2010; Martikainen & Korpilahti, 2011). However, as
stated above, the feature lists have not been operationalized,
validated, or standardized, and no standard approach or
battery for assessment of these specific features has yet been
developed (ASHA, 2007b; Shriberg et al., 2012). As such,
it is likely that expert and novice clinicians would demonstrate low interrater reliability, as already shown in studies
of CAS (Shriberg, Campbell, et al., 2003) and dysarthria
(Zeplin & Kent, 1996; Zyski & Weisiger, 1987) using perceptual, criterion-based diagnostic methods. This uncertainty
results in clinicians in practice making diagnoses of suspected CAS, with risk of under- or overidentification
(Davis et al., 1998; Forrest, 2003; Shriberg, Campbell,
et al., 2003). The result can be selection of inappropriate
treatment approaches and the frequent complaint that children with CAS progress slowly in therapy (Forrest, 2003).
This study furthers the CAS diagnostic literature by attempting to determine if clinically available measures can discriminate CAS from other expressive communication disorders
in a sample of verbal children.
Aim
The primary aim of this study was to use objective
statistical methods to identify one or more quantitative
measures of speech that reproduce the expert classification
of children as having CAS or not. This approach has potential to provide a clinically feasible assessment protocol that
reliably identifies features of CAS and reduces reliance on
expert opinion.
Method
This study formed part of a larger clinical trial:
Unique Trial Number: U1111-1132-5952, Trial Registration
Number: ACTRN12612000744853. The research protocol
was approved by the Human Research Ethics Committee of
the University of Sydney (12924) and is published (Murray,
McCabe, & Ballard, 2012).
This diagnostic study was conducted in three phases
to address the research aim. First, children were recruited
and assessed according to the protocol below. Second, the
first two authors (E. M. and P. M.) judged presence or absence of CAS by listening to speech samples from the assessment protocol and completing two published checklists
of CAS characteristics. Third, the first author (E. M.) and
independent raters blinded to the diagnosis of CAS generated 24 quantitative measures from assessment data. These
measures were analyzed using bivariate and hierarchical

multivariate discriminant function analysis (DFA) to determine if any combination of the 24 measures reliably predicted the assigned CAS diagnosis.
Participants
Participants were recruited via a website advertisement
and flyers as well as e-mails and listserv posts to speechlanguage pathologists (SLPs), inviting them to volunteer for
a research treatment study. The treatment component of this
study is not discussed in this article. The inclusion criteria for
participants were (a) a clinical diagnosis of suspected CAS
by a community-based SLP, (b) age between 4 and 12 years,
(c) no previously identified language comprehension difficulty, (d) normal or adjusted-to-normal hearing and vision,
(e) native English speaker, and (f ) no other developmental
diagnoses not associated with CAS (e.g., intellectual disability,
autism, cerebral palsy). All participants had received speechlanguage pathology intervention prior to this study, and
all had normal hearing. Participants needed to attend the
on-campus clinic at the University of Sydney, Australia, to
participate. Parents or caregivers provided informed consent
on behalf of the children prior to participation.
Of the 72 participants who inquired regarding the
study, 47 passed the initial screening. All 47 were AustralianEnglish speaking, 33 (70%) were boys, 14 were girls (30%),
and the average age was 70.5 months (SD = 25.7 months).
No information on race, ethnicity, or socioeconomic status
was collected.
Clinical Assessment
The first author responded to all inquiries to participate in the study and followed a two-tiered assessment
protocol (see Figure 1). The first tier involved screening of
parents or caregivers and SLPs by phone and previous
speech pathology reports to determine the childs suitability
for entry into the study. The criteria were applied conservatively, in that children with another developmental disorder that could be associated with symptoms or consequences
of CAS (e.g., selective mutism) were included. For those
who passed the initial screening, the second tier involved administration of a 2-hour speech and language assessment
battery. Responses to these tests were used for all analyses
of CAS features and to determine the presence of comorbid
conditions such as dysarthria or language impairment. The
assessment battery was video- and audio-recorded using
a Sony IC Recorder ICD-UX71F and either an Echo Layla
24/96 multitrack recording system, Marantz PMD660 solidstate recorder, or Roland Quad-Capture UA-55 at the sampling rate 48000 Hz with 16-bit resolution with an AKG
C520 headset microphone at 5 cm mouth-to-microphone
distance.
Speech and Language Assessment Battery

First, a parent or caregiver case history questionnaire
and hearing screening was completed (ASHA, 1978). Next,
Murray et al.: Differential Diagnosis of CAS

45
Figure 1. Results of differential diagnostic process. Participants with childhood apraxia of speech (CAS) shown in white, comorbid CAS in yellow,
and non-CAS or excluded in blue.
No articulation or prosodic impairment noted by parents, no CAS suspected by SLP.
the diagnostic assessment utilized five published tests commonly available in clinical settings and culturally appropriate
for Australian children (see Table 1). The five tests were the
following:
1.
the Diagnostic Evaluation of Articulation and

Phonology (DEAP) Inconsistency subtest (Dodd, Hua,
Crosbie, Holm, & Ozanne, 2002), used to assess the
word-level token-to-token inconsistency in naming
25 pictured words over three test administrations, each
administration separated by other assessment tasks;
2.
the Single-Word Test of Polysyllables (Gozzard, Baker,

& McCabe, 2004, 2008), a 50-item picture-naming
task used to assess articulation, sound and syllable
sequencing, and lexical stress accuracy (i.e., prosody);
3.
a connected speech sample of at least 50 utterances

recorded over at least 10 minutes (McLeod, 1997), for
detection of perceptual features of CAS in connected
speech and calculation of articulation rate;
4.
the Oral and Speech Motor Control Protocol, a

published oral motor assessment (OMA) including
diadochokinesis (DDK; Robbins & Klee, 1987), used
to rule out any structural or functional abnormalities
in the oral mechanism; and
5.
46
the Clinical Evaluation of Language Fundamentals

(CELF; 4th edition or Preschool2nd edition,
Australian versions), used to assess receptive and
expressive language skills (Semel, Wiig, & Secord,

2006; Wiig, Secord, & Semel, 2006).
All tests were administered and scored by the first
author according to the relevant manual. For the DEAP
Inconsistency subtest, broad transcription was used and distortions and developmental errors were scored as correct,
according to Dodd et al. (2002, p. 25).
Initially, responses to the assessments were used for
expert qualitative judgments of presence or absence of CAS
and, following this, for generating 24 quantitative measures
for statistical analyses in order to address the study aim.
There was overlap in the speech samples used to make the
expert judgments and to generate the quantitative measures;
however, expert judgments were made prior to extracting the
wider range of measures from the assessment, and measure
calculations did not influence the initial classifications made.
In addition, three raters scored 100% of the data: the first
author and two independent raters blinded to expert diagnosis.
A detailed description of reliability calculations is provided
in the Reliability section.
Expert Diagnosis of CAS

The first and second authors, each with more than
10 years experience in differential diagnosis of pediatric
speech sound disorders, independently diagnosed the presence and severity of CAS for all children based on their perceptual ratings of each childs speech samples, using the

Table 1. Assessment tasks and diagnostic features used for assigning an expert diagnosis of childhood apraxia of speech.
ASHA consensus-based
feature list a
1. Inconsistent errors on
consonants and vowels in
repeated productions of
syllables or words
2. Lengthened and disrupted
coarticulatory transitions
between sounds and syllables
3. Inappropriate prosody,
especially in the realization of
lexical or phrasal stress
Strands 10-point checklistb
Assessment tasks considered in diagnosisc
Not in list
DEAP Inconsistency subtest (inconsistency defined

as a score 40%),d (Polysyllable Test,e connected
speech samplef )
1. Difficulty achieving initial articulatory

configurations and transitions into vowels
2. Syllable segregation
3. Lexical stress errors or equal stress
Polysyllable Test,e connected speech samplef

(DEAP Inconsistency subtestd)
4. Vowel or consonant distortions including

distorted substitutions
5. Groping (nonspeech)
6. Intrusive schwa
7. Voicing errors
8. Slow rate
9. Slow DDK rate
10. Increased difficulty with longer or more
phonetically complex words
Polysyllable Teste (DEAP Inconsistency subtest,d

Connected speech samplef )
Oral and Speech Motor Control Protocol (any three
nonspeech tasks)g
Connected speech samplec)
Connected speech samplef Polysyllable Teste
(DEAP Inconsistency subtestd)
Note. DEAP = Diagnostic Evaluation of Articulation and Phonology; DDK = diadochokinesis.

ASHA, 2007b, p. 4. ASHA criteria = 3/3 needed for CAS diagnosis. bShriberg, Potter, et al., 2009, p. 8. Strand criteria = 4/10 needed for CAS
diagnosis over three tasks. c The primary task or tasks used for detection of each feature are written first, with the secondary tasks needed to determine
features over three tasks shown in brackets. dDodd et al. (2002). eGozzard et al. (2004, 2008). fMcLeod (1997). gRobbins and Klee (1987).
following procedure. To be diagnosed with CAS in this study,

participants had to demonstrate (a) the three consensus-based
features listed in the ASHA Technical Report (2007b) and
(b) any four of the 10 features in Strands 10-point checklist
(Shriberg, Potter, et al., 2009) over at least three assessment
tasks as shown in Table 1. For the 10-point checklist, a verbal
behavior was marked as present when perceptually detected
in speech samples from three tasks: the DEAP Inconsistency
assessment, the Single-Word Test of Polysyllables, and the
connected speech sample. Neither published list provides
operational definitions or cutoffs for the listed features.
The first and second authors listened to the speech
samples and independently made a perceptual judgment as
to whether each feature was present or absent, except in the
case of inconsistency of speech errors. For this one feature
only, decisions were based on the score from the DEAP
Inconsistency subtest, the only published test that defines
inconsistency in the same way as the ASHA Technical
Report (ASHA, 2007b). Syllable segregation was defined
by the authors as noticeable gaps between syllables. Two
criteria showed potential overlap, and, because no previous
definitions were available, the authors defined difficulty
achieving initial articulatory configurations and transitions
into vowels to include within-speech groping, false starts,
restarts, and hesitations and defined groping as nonspeech
oral groping. Nonspeech groping was determined over
three oral motor tasks within the Robbins and Klee protocol
(1987). For expert perceptual diagnosis of CAS, interrater
agreement between the first two authors was 100%.
Appendix A shows the CAS features judged to be

present and the expert decision on presence or absence of
CAS for each of the 47 participants. Of these 47, 28 (60%)
were classified as having CAS (ASHA, 2007b; Shriberg,
Potter, et al., 2009). Another four children (8%) met the
criteria for CAS but presented with comorbid dysarthria and
receptive and/or expressive language disorder (CAS+),
similar to the Shriberg et al. (2010) classification of motor
speech disorderapraxia of speech plus (MSD-AOS+).
Children diagnosed with CAS or CAS+ showed a median
of 6 out of 10 features on the Strand 10-point checklist
(Shriberg, Potter, et al., 2009). Fifteen (32%) of the 47 children did not demonstrate all three features in the ASHA
feature list and were classified as non-CAS in this study.
Non-CAS diagnoses included ataxic dysarthria (n = 1), flaccid
dysarthria (n = 1), submucous cleft (n = 3), or primarily
phonological disorder (n = 10; see Figure 1). Phonological
impairment was identified in those who had clear phonological processes (Rvachew & Brosseau-Lapr, 2012) and
an absence of oromotor or speech-motor deficits (e.g., DDK
deficits) excluding them from motor speech diagnosis.
Quantitative Measures
Quantitative measures were extracted from the five
tests administered in the clinical assessment. Table 2 lists
the 24 measures generated and the associated assessment
task and CAS checklist feature. For measures that were
not standard scores from norm-referenced tools, nine raters

47
48
Table 2. Extracted measures from assessment tasks completed with means and standard deviations for the CAS, CAS+, and non-CAS groups.
CAS (n = 28)
Assessment task
Case history
DEAP Inconsistency
subtesta
Single-Word Test of
Polysyllablesb
Relation to diagnostic criteria

None
Inconsistent errors on consonants and
vowels in repeated productions of
syllables or wordsf
Inappropriate prosody, especially in the
realization of lexical or phrasal stressf
and equal or lexical stress errorsg
Vowel or consonant distortions including
distorted substitutionsg
Syllable segregationg
Intrusive schwag
g
Voicing errors
DEAP Inconsistencya vs.

Polysyllablesb
Connected speech
samplec
Oral and Speech Motor

Control Protocold
None
None
None
Increased difficulty with longer or more
phonetically complex wordsg
Slow rateg
Lengthened and disrupted coarticulatory
transitions between sounds and
syllablesf and difficulty achieving initial
articulatory configurations and
transitions into vowelsg
Groping (nonspeech)g
Slow DDK rateg
Slow DDK rateg
Slow DDK rateg
Slow DDK rateg
Slow DDK rateg
None
CELFe
None
None
None
None
None
Measure
Age in months
Percentage inconsistency (across three
repetitions of 25 words)a
Percentage of lexical stress matches
(relative to gloss)h
Distortion occurrences (out of a possible
328 phones)h
Syllable segregation occurrences (out of a
possible 114 opportunities)i
Intrusive schwa occurrences (out of a
possible 15 clusters)i
Voicing error occurrences (out of a possible
90 opportunities)h
PPCh
PCC-Rh,j
PVC on polysyllable testh
Magnitude of change score: NPC on 12
monosyllables/NPC on 12 polysyllables
(>1 = difficulty with polysyllables)
Articulation rate (syllables per minute)k
Presence of false articulatory starts and
restarts and/or inaudible within-speech
groping and/or audible within-speech
groping and/or hesitations (min. = 0,
max. = 1)l,m
Presence of nonspeech groping in lip and
tongue oral function tasks (min. = 0,
max. = 1)
/p/ rate over 3 s on two trialsd
/t/ rate over 3 s on two trialsd
/k/ rate over 3 s on two trialsd
/ptk/ rate over 3 s on two trialsd
/ptikek/ rate over 3 s on two trialsd
Accuracy on /ptk/ DDK task on
two trialsd
Oral structure scored
Oral function scored
Maximum Phonation Timed
Receptive Language Scoree
Expressive Language Scoree
CAS+ (n = 4)
Non-CAS (n = 15)
SD
SD
SD
66.5
63.8
24.8
12.8
72.5
75.0
26.0
15.1
77.3
39.9
27.6
24.5
9.8
9.1
16.3
17.5
67.3
22.4
39.4
5.8
45.2
8.8
46.24
22.9
30.2
8.8
8.5
8.4
1.2
2.1
1.1
1.3
0.5
1.0
0.0
0.0
3.4
4.0
6.0
5.0
1.7
1.5
52.2
54.0
50.5
1.23
15.0
20.0
11.4
0.2
24.3
22.8
24.8
2.52
20.6
23.8
19.0
1.9
73.1
70.9
75.1
2.07
26.3
26.4
27.3
2.91
1.7
0.9
1.0
0.4
0.9
1.0
0.6
0.0
2.6
0.1
1.0
0.3
0.5
0.5
0.5
0.6
0.0
0.0
3.6
3.4
2.9
0.9
1.0
48.5
1.0
1.0
1.2
0.6
0.4
22.6
3.0
2.3
1.8
0.5
0.7
23.8
1.7
1.3
1.0
0.1
0.4
9.0
4.5
4.4
4.3
1.4
1.3
75.9
2.0
1.7
1.9
0.8
0.7
23.2
23.4
76.6
9.6
100.3
85.5
.91
13.8
5.5
12.2
20.1
21.0
60.0
6.8
65.5
55.5
2.6
12.4
4.1
18.4
7.3
21.8
88.5
12.9
91.4
79.7
2.4
18.7
6.7
20.5
19.2
Note. CAS = childhood apraxia of speech; M = mean; SD = standard deviation; PPC = percentage phonemes correct; PCC-R = percentage consonants correctrevised; PVC = percentage
vowels correct; NPC = number of phonemes correct; CELF = Clinical Evaluation of Language Fundamentals (Semel, Wiig, & Secord, 2006; Wiig, Secord, & Semel, 2006).
a
Dodd et al. (2002). bGozzard et al. (2004, 2008). cMcLeod (1997). dRobbins and Klee (1987) includes some norms. eSemel et al. (2006) with norms. fASHA (2007b). gShriberg, Potter, et al.
(2009). hComputerized Profiling (Long et al., 2006). iCounted by hand from transcription. jShriberg, Austin, Lewis, McSweeny, and Wilson (1997). kLogan et al. (2011) includes norms.
l
McNeil et al. (2009). mDuffy (2012).

were employed to make measurements blinded to the childrens expert diagnosis of CAS presence or absence for reliability purposes. The raters were qualified SLPs with an
average of 3 years of clinical experience. The first author
trained each rater on the methods of measurement until a
minimum of 85% interrater reliability was achieved on a
training sample not included in the study. Raters were randomly assigned samples from 12 to 13 children (M = 12.70,
SD = 4.83). Each participants results were rated by at least
two raters. In the event of any discrepancies, a third independent rater measured the sample(s), and the first authors
scores were retained based on above 85% agreement for all
measures (see the Reliability section).
The DEAP Inconsistency subtest was transcribed
using broad transcription following the manual (Dodd et al.,
2002). Responses to the Single-Word Test of Polysyllables
were transcribed from an audio recording into the PROPH
module in Computerized Profiling (Long, Fey, & Channell,
2006). Transcription included broad transcription supplemented with International Phonetic Alphabet diacritics to
mark distortions where clearly present. Measures extracted
from PROPH were percentage consonants correctrevised
(PCC-R; Shriberg, Austin, Lewis, McSweeny, & Wilson,
1997), percentage vowels correct (PVC), percentage phonemes
correct (PPC), occurrences of intrusive schwa, distorted
substitutions and voicing errors, and percentage of syllable
stress matches compared to the gloss for each word. Syllable
segregation (transcribed as a plus sign + between syllables) and intrusive schwas between cluster elements were
recorded by simple counts of occurrence.
PCC-R is a revised metric of PCC in which typical and
atypical distortions are removed from the index (Shriberg,
Austin, et al., 1997). PCC-R was used as a general index of
speech sound disorder severity, regardless of specific diagnosis, for all three groups. This metric was used in this study
because it was more sensitive for the participant sample with
a diverse range of age and speech status (Shriberg, Austin,
et al., 1997). Distortions were counted as their own measure
because it may facilitate differential diagnosis of speech
disorders. Thus, using PCC-R allowed us to identify any
potential effects related to general severity rather than a specific diagnosis of CAS. Accordingly, distortions were not
part of the PCC-R measure, yet were measured separately
under the Occurrence of Consonant and Vowel Distortions
Including Distorted Substitutions measurement according
to Shriberg, Potter, and Strand (2009).
The Oral and Motor Speech Protocol and connected
speech sample were transcribed or scored from a video recording. Articulation rate was calculated by counting syllables per second produced in 1 min of monologue connected
speech. This was undertaken using Adobe Audition software
(Version 1.5), in which the sample was isolated and vowels
identified and counted to determine syllable rate following
Logan, Byrd, Mazzocchi, and Gillam (2011).
Strands criterion of increased difficulty with polysyllabic words (Shriberg, Potter, et al., 2009) was determined
using a magnitude-of-change score. This score was generated for each participant, comparing the mean number of
phonemes correct from the 12 monosyllable items in the

DEAP Inconsistency subtest to the first 12 items from the
Single-Word Test of Polysyllables. A magnitude of change
score greater than 1 indicated that production of polysyllabic
words was more difficult than monosyllabic words. The only
binary measures used were for presence of (a) false articulatory starts, restarts, or within-speech groping and (b) nonspeech groping movements.
Reliability
Intrarater and interrater transcription reliability was
calculated on a point-to-point basis for all unstandardized
dependent measures for each participant. Intrarater reliability was calculated on 20% of each measure for every participants data with each rater calculating the measure once
and then a second time at least 2 weeks after the initial calculation. Intrarater reliability for the first author was 94.6%
(SD = 3.6, range = 8898) and across all nine independent
raters was 95.2% (SD = 3.1, range = 88.399.1). Interrater
reliability was calculated first between the first author
and the nine raters and then between the nine independent
raters. Mean interrater reliability with the first author across
all measures was 94.2% (SD = 1.7, range = 91.396.2) and
between the nine raters was 93.3% (SD = 2.9, range = 86.4
98.1). For a breakdown of each raters and measures reliability, see online Supplemental Appendices 1 and 2.
Statistical Analyses
Simple bivariate followed by hierarchical multivariate, discriminant function analysis (DFA; McKean &
Hettmansperger, 1976) was used to determine whether one
or more of the 24 quantitative measures could reliably predict
the expert assignment of children to CAS or non-CAS
groups. Therefore, the results from the expert judgment of
CAS presence or absence and the quantitative measures were
compared in this phase to address the primary research aim.
The sample size was sufficient to minimize Type I
and Type II errors (Serlin & Harwell, 2004). Before completion of the analysis, data were screened for normality, linearity, and homoscedasticity. No violations of assumptions
were noted. Mahalanobis Distance was calculated to check
for outliers (Allen & Bennett, 2008) and did not exceed the
critical c2 of 26.13 (df = 8; a = .001) for any cases. Finally,
screening for multicollinearity above a conservative 0.80
(cf. 0.90; Allen & Bennett, 2008; Poulsen & French, 2004;
Tabachnick & Fidell, 2007) identified the following highly
correlated measures: PVC, PCC-R, and PPC; PVC and percentage stress matches; PPC and oral function score; PCC-R
and oral function score, percentage stress matches and
presence of articulatory false starts, restarts and/or withinspeech groping; and finally, /p/, /t/, and /k/ rates. When
two measures showed multicollinearity, only one measure
at a time was entered into the DFA as a predictor. There is
no consensus on the minimum ratio of predictor variables
to cases in DFA. Given this, we followed guidelines by
Poulsen and French (2004) and Tabachnick and Fidell

49
(2007) and used up to eight predictors at a time. Bivariate

DFA (for each measure predicting the given diagnosis) was
initially completed to determine the top eight theoretical
measures in discriminating CAS in this sample, taking into
account multicollinearity, for entry into the multivariate
DFA. Results for all 24 measures are presented in Table 3
for all participants and Table 4 when CAS+ and submucous clefts were excluded. When a potential predictor
correlated highly with another potential predictor, the DFA
was run twice, each time with only one of the highly correlated predictors. For example, the measures that were initially entered as predictors into the first multivariate DFA
were (a) presence of articulatory false starts, restarts, and/or
within-speech groping; (b) syllable segregation; (c) DEAP
(Inconsistency subtest); (d) PPC; (e) DDK accuracy; (f) nonspeech groping; (g) intrusive schwa; and (h) /k/ rate. The
predictor percentage lexical stress matches correlated highly
(i.e., was multicollinear with) the variable presence of articulatory false starts. Similarly, PVC was multicollinear with
the variable PPC and /ptk/ rate was multicollinear with
the variable /k/ rate. Analyses were rerun, substituting percentage stress matches, PVC or PCC-R, and /ptk/ for
multicollinear variables. As measures were identified as not
making a significant contribution to the hierarchical DFA,
they were replaced by other measures of potential clinical
importance (r < .5) to see whether they increased the predictive accuracy of the model. For ease of reading, DFA analyses are presented in the more familiar layout of multiple
regression, rather than as traditional discriminant functions.
a relatively straightforward manner with visual inspection

of the palate before differential diagnosis of CAS. The unstandardized (B) and standardized (b) regression coefficients
are reported for the two hierarchical DFAs in Table 5. The
standardized b coefficient indicates the contribution of each
measure in predicting the initial diagnosis. A high score on
a positive coefficient and a low score on a negative coefficient predict CAS.
For Model 1 (full data set), the hierarchical DFA
showed that together the percentage of stress matches and
occurrence of syllable segregation, both from the SingleWord Test of Polysyllables, accounted for a significant 82%
of the variance in CAS diagnosis in this sample, R2 = .82,
adjusted R2 = .81, F(2, 45) = 96.26, p .001. Additional
measures increased the overall predictive accuracy by only
1%2%, at most.
For Model 2 (limited data set), results indicated that
four measures in conjunction accounted for 91% of the CAS
diagnosis, R2 = .91, adjusted R2 = .90, F(4, 38) = 87.45,
p .001. These measures were syllable segregation, percentage of stress matches, PPC on the Single-Word Test of
Polysyllables, and accuracy on DDK tasks (/ptk/) from
the Oral and Speech Motor Control Protocol. A formula
can be derived from Model 2 for predicting an individuals
group membership. This involves multiplying each individuals raw scores on the four measures in the DFA with their
standardized b coefficient (i.e., a weighting), then summing
the products and the model constant, and rounding the
resulting single value to zero decimal places. A result of 1
indicates likely CAS, a score of 0 indicates likely non-CAS:
Results
Diagnosis Constant % lexical stress matches its weight
Table 4 reports the descriptive statistics for each of

the 24 variables measured across the three groups: CAS
( expressive language disorder), CAS+ (CAS with comorbid
dysarthria and language impairment), and non-CAS (submucous cleft, phonological disorder, and dysarthria). First,
the effects of severity and age were determined. In terms
of PCC-R, an index of general severity of speech sound disorder, the groups were significantly different, F(2, 45) =
7.865, p = .001. A Scheff post-hoc analysis adjusted for
multiple comparisons revealed that the CAS+ group had
significantly lower PCC-R scores than the CAS, p = .042,
and non-CAS, p = .02, groups, but importantly, the latter
two were not differentiated. This is consistent with the
CAS+ group having more complex speech disorders. There
was no effect of age between groups, F(2, 45) = 0.853,
p = .433, and age was not correlated above .5 with any of
the 24 measures.
Two models are provided that represent the set of
variables with highest predictive accuracy for (a) all participants with CAS (n = 32) against all non-CAS (n = 15) and
(b) participants with CAS (CAS+ cases removed, n = 28)
against non-CAS (cases with submucous cleft removed,
n = 12). Model 2 removed cases with CAS+ (where participants shared features across both groups and confounded
the analysis) and participants in the non-CAS group with
submucous cleft palates, which could be discriminated in
50
syllable segregation score its weight PPC

its weight=ptk=DDK accuracyits weight
Round0
All participants used to create the model were correctly
diagnosed using the formula (100% sensitivity, or true positive
rate, and 100% specificity, or true negative rate). The model
had high positive and zero negative likelihood ratios and a
diagnostic odds ratio of 1,425; see Appendix B). The strength
of Model 2 was further tested with data from the four CAS+
children and three children with submucous cleft who had
been excluded from the DFA. Sensitivity remained high
at 97%, with one CAS+ participant being misdiagnosed as
non-CAS; specificity remained at 100%, with all submucous
cleft participants accurately classified as non-CAS. Again,
the positive likelihood ratio was high (16), and there was a
zero negative ratio, with a diagnostic odds ratio of 651 (see
Appendix B). Table 6 demonstrates use of this formula for
four participants in this sample, two with CAS and two without CAS. The formula can be used in Microsoft Excel to
calculate automatically from evaluation test score input.
Discussion
The purpose of the current study was to determine
whether expert diagnosis of CAS in a sample of 47 children

Table 3. Model 1: Results from bivariate DFA for all measures in order from highest to lowest accuracy in discriminating CAS/CAS+ group from
the non-CAS group participants.
Measure
Syllable segregation
Percentage of stress matches (lexical stress) on polysyllable test
Presence of false articulatory starts, restarts, or within-speech groping
DEAP (Inconsistency subtest)
PVC on the polysyllable test
/ptk/ accuracy
Nonspeech groping movements
PPC on the polysyllable test
Oral structure score
Intrusive schwa
/k/ rate
Articulation rate (syllables per second) in connected speech
Oral function score
PCC-R on the polysyllable test
/ptk/ rate
/t/ rate
/ptikek / rate
Vowel or consonant distortions
Maximum phonation time /a / (MPT)
CELF receptive language score
CELF expressive language score
/p/ rate
Voicing errors
Increased difficulty with polysyllable words (change score)
Bivariate DFA
Accuracy in diagnosing
CAS /CAS+ vs. non-CAS
0.890
0.882
0.764
0.551
0.546
0.535
0.536
0.515
0.492
0.477
0.445
0.428
0.386
0.389
0.356
0.352
0.321
0.067
0.261
0.270
0.142
0.291
0.231
0.205
<.001
<.001
<.001
<.001
<.001
<.001
.002
<.001
.004
<.001
.002
.006
.007
.007
.014
.024
.028
.669
.091
.080
.365
.058
.136
.196
79%
77%
58%
30%
30%
29%
29%
27%
24%
23%
17%
17%
15%
15%
13%
12%
10%
Not significant
Not significant
Not significant
Not significant
Not significant
Not significant
Not significant
Note. DFA = discriminant function analysis; DEAP = Diagnostic Evaluation of Articulation and Phonology; PVC = percentage vowels correct;
PPC = percentage phonemes correct; PCC-R = percentage consonants correctrevised.
Table 4. Model 2: Results from bivariate DFA for all measures in order from highest to lowest accuracy in discriminating the CAS group
(excluding CAS+) from the non-CAS group (excluding submucous cleft) participants.
Measure
Percentage of stress matches (lexical stress) on polysyllable test
Presence of false articulatory starts, restarts, or within-speech groping
DEAP (Inconsistency subtest)
PVC on the polysyllable test
/ptk/ accuracy
Nonspeech groping movements
PPC on the polysyllable test
Oral function score
Vowel or consonant distortions
Intrusive schwa
PCC-R on the polysyllable test
Articulation rate (syllables per second) in connected speech
/k/ rate
CELF receptive language score
/t/ rate
Oral structure score
/ptk/ rate
/ptikek / rate
Maximum phonation time /a / (MPT)
CELF expressive language score
/p/ rate
Voicing errors
Increased difficulty with polysyllable words (change score)
Bivariate DFA
CAS vs. non-CAS
0.893
0.873
0.732
0.670
0.615
0.606
0.507
0.540
0.467
0.464
0.449
0.429
0.429
0.388
0.362
0.326
0.280
0.258
0.214
0.166
0.215
0.269
0.237
0.208
<.001
<.001
<.001
<.001
<.001
<.001
.002
<.001
.002
.003
.004
.007
.006
.013
.022
.040
.081
.109
.185
.306
.183
.093
.141
.199
80%
76%
54%
45%
38%
37%
26%
29%
22%
22%
20%
18%
18%
15%
13%
12%
Not significant
Not significant
Not significant
Not significant
Not significant
Not significant
Not significant
Not significant

51
Table 5. Unstandardized (B) and standardized () regression coefficients for each predictive measure in a regression model predicting CAS
diagnosis.
Variable/measure
(Constant)
Percentage of stress matches on
polysyllable test
(Constant)
Percentage of stress matches on
polysyllable test
PPC on polysyllable test
Accuracy on DDK task /ptk/
0.707
0.009
0.575
Participants included
Model 1
CAS and CAS+ group (n = 32) vs.
non-CAS (n = 15)
Model 2
CAS (n = 28) vs. non-CAS (n = 12)
(CAS+ and submucous cleft
omitted)
0.382
p
<.001
<.001
0.012
0.504
0.011
0.729
.001
<.001
<.001
0.012
0.007
0.003
0.383
0.300
0.145
<.001
.001
.039
CAS in this sample
82%
91%
a
The values indicate the contribution (weight) of each task in diagnosis of CAS. A high score on a positive coefficient predicts CAS is more
likely, whereas, on a negative coefficient, a low score predicts CAS is more likely. Taking all the measures and weightings together gives the
overall accuracy in diagnosing CAS in this sample.
could be predicted from a combination of quantitative measures often collected as part of standard clinical practice.
The current standard for diagnosing CAS utilizes experts
making judgments of presence or absence of a small set
of speech behaviors, with no operational definitions or standardized testing protocol for eliciting the behaviors. In
the current sample, 28 children were judged by this method
to have CAS ( expressive language disorder; ASHA,
2007b; Shriberg et al., 2012), four were judged to have CAS+
(CAS plus dysarthria and receptive and/or expressive language
impairment), and 15 were judged to have either submucous
cleft, phonological impairment, or dysarthria (non-CAS).

Following the diagnosis, 24 quantitative measures were extracted from clinically based assessments by raters blinded to
expert diagnosis. A series of multivariate DFAs revealed that
a combination of the two quantitative measures of percentage of stress matches and occurrence of syllable segregation
showed 82% diagnostic accuracy with expert diagnosis of
CAS and CAS+ versus non-CAS. However, the highest
accuracy (91%) was obtained when four CAS+ children and
three non-CAS children with structural impairment (i.e.,
submucous cleft) were excluded from analyses.
Table 6. Using the DFA Model 2 formula to diagnose CAS.

Percentage
of lexical
stress
Syllable
/ptk/
matches segregation PPC accuracy
Participant characteristic
Participant No. 2216: CAS,

4 years, female, severe
articulation severity
Participant No. 1217: non-CAS
(ataxic dysarthria), 4 years,
female, severe articulation
severity
Participant No. 2219: CAS,
12 years, male, mild
articulation severity
Participant No. 1311: non-CAS
(residual phonological
disorder), 12 years, male,
mild articulation severity
0.011
6
0.012
45
0.007
46
0.003
77
Raw score
33
20
25
66
Raw score
26
19
79
72
Raw score
88
97
100
Weighting ( values)
Raw score
Formula as entered
into Excel
Resulta
= round(0.504 + (6 0.011)
+ (45 0.012) + (46 0.007)
+ (77 0.003), 0)
= round(0.504 + (33 0.011)
+ (2 0.012) + (25 0.007)
+ (66 0.003), 0)
= round(0.504 + (26 0.011)

+ (19 0.012) + (79 0.007)
+ (72 0.003), 0)
= round(0.504 + (88 0.011)
+ (0 0.012) + (97 0.007)
+ (100 0.003), 0)
Note. Note the importance of the whole pattern of responses, rather than considering cutoffs variable by variable. Participants No. 2216 and
no. 2219, both diagnosed with CAS, had better scores for PPC on the polysyllable test than Participant No. 1311, diagnosed without CAS.
Classification of Participants No. 2216 and No. 2219 as CAS, using the formula, occurs because their substantially poorer scores on percentage
of stress matches on polysyllable test, syllable segregation, and accuracy on DDK task tip them over the cutoff for CAS classification.
Participant No. 1311s better scores on these three measures and his mildly compromised PPC on the polysyllable test all indicate he does not
fit the pattern of CAS in this sample.
a
1 = likely CAS; 0 = likely non-CAS.
52

The procedures used here for expert judgment on

presence of CAS resulted in 32% of this sample being diagnosed as non-CAS, from a group of children referred with
suspected CAS according to Australian community-based
SLPs. This is consistent with American studies (Davis et al.,
1998; Forrest, 2003; Shriberg et al., 2011) in which children
with CAS seemed to be over- rather than underdiagnosed.
However, we did not determine if CAS was underdiagnosed
in a general SSD population here. This finding is not surprising, given the lack of validated and/or published assessment batteries currently available for this population (ASHA,
2007b).
Of concern, there was a subset of children who presented with frank structural and neurological deficits that
were not identified prior to the community clinician assigning the diagnosis of suspected CAS. An OMA is a practical,
inexpensive screen to check for any overt structural deficits
or functional impairments related to muscle strength and
tone. If such deficits are found, referral to a neurologist or
head and neck team should follow. This may seem obvious,
yet there were participants as old as 12 years in this sample
whose structural and/or dysarthric symptoms had not been
previously detected. This is relevant for those with a CAS
diagnosis as well because such diagnoses can co-occur. Furthermore, only 52% (22 out of 42) of the studies reviewed
by McCauley et al. (2012) included OMAs. OMAs should
be part of standard pediatric practice, reflected in policy
guidelines and clinical pathways. In this sample, those with
dysarthria exhibited low oral structure scores on our oral
structure and function test (Robbins & Klee, 1987) due to
lack of tongue symmetry, the presence of tongue fasciculations, and tongue atrophy. In the submucous cleft cases in
the non-CAS group, all three had low oral structure scores
due to poor palatal juncture and one had a bifid uvula.
Given that the primary aim of this study was to identify a set of measures for identifying children with CAS in
a group with no other known diagnoses (e.g., hearing impairment, structural abnormality, or other diagnosed developmental diagnoses), the final analysis excluded those
with comorbid CAS and submucous clefts. This exclusion
allowed us to determine how performance on a specific set
of measures, taken from existing clinical tests, interacted
with each other to differentiate children with CAS from
children with other speech sound disorders. This differentiation is likely to be the difficulty SLPs face in identifying
true cases of CAS in order to provide effective management. Importantly, age and severity of speech sound disorder (PCC-R) of these 40 children did not explain the
variance in the CAS and non-CAS groups. Discriminant
function analysis revealed that four measures in combination had 91% predictive accuracy: (a) syllable segregation,
(b) percentage of lexical stress matches, (c) PPC, and (d) accuracy on repetition of /ptk/ in the DDK task. The
four measures are considered together in a hierarchical format to make a diagnosis; individual cutoff scores for each
measure, although clinically appealing, do not capture
the relationships between the variables and therefore the
diagnosis. These four measures were derived from just
two assessment tasks: a polysyllabic word picture-naming

task (Gozzard et al., 2004) and an oral mechanism examination (Robbins & Klee, 1987), taking less than 30 min to
complete.
Two of the four informative measuressyllable segregation and percentage of lexical stress matchesmeasure
prosodic features now considered core symptoms of CAS.
These perceptual features are strongly related to acoustic
measures identified as potential markers for CAS, including
the lexical stress ratio (Shriberg, Campbell, et al., 2003), the
coefficient of variation ratio (Shriberg, Green, et al., 2003),
and the pairwise variability index (Ballard et al., 2010). Our
results support dysprosody as a potential core feature of
CAS in children with some ability to produce polysyllable
combinations. Prosody was a discriminant measure in both
DFAs regardless of whether participants with neurological,
linguistic, or structural impairments were included.
The Strand 10-point checklist (Shriberg, Potter, et al.,
2009) includes both syllable segregation and lexical
stress errors as features. PPC was not used to measure any
criterion from this feature list. Instead, six individual segmental error features were noted (out of the 10 features; see
Table 1), but they were not individually discriminative. A
more global measure such as PPC could have teased apart
differences between groups, particularly because the nonCAS group was diverse. Accuracy on the /ptk/ DDK
task was not used to measure any criterion from this checklist. However, Thoonen et al. (1999) adds support for this
finding. They found trisyllabic repetition of /pataka/ was
discriminative for CAS against controls. However, they
found it did not discriminate CAS from dysarthria, which
was achieved using fricative maximum phonation. This
measure was not calculated here but could be added to an
OMA as an assessment measure in future studies. Importantly, Strands checklist (Shriberg, Potter et al., 2009) does
not require children to exhibit these features to be diagnosed with CAS: Children need to show evidence of any
four of the 10 features on the list, with no differential
weighting of any feature. To our knowledge, the sensitivity
and specificity of Strands checklist has not been tested, and
it may not be sufficiently specific, with potential risk of diagnosing negative cases with CAS. The present results show
that two participants with clear dysarthria and one with a
submucous cleft were diagnosed with CAS using the Strand
checklist.
The inclusion of lexical stress errors in the ASHA list
of strong discriminative behaviors of CAS was supported
by our analyses, but the other two features (inconsistency
and lengthened and disrupted coarticulatory transitions)
were not supported. The second feature, lengthened and
disrupted coarticulatory transitions between sounds and
syllables, was measured as a binary score if false starts, restarts, hesitations, or within-speech groping were demonstrated by a participant in connected speech and on the
polysyllable test. In the bivariate DFA, this measure had
54% accuracy in diagnosis when used independently but did
not add any additional information to the Model 2 (limited
data set) analysis. This diagnostic information may have

53
been captured in the DFA by syllable segregation, which

is associated with disrupted coarticulatory transitions as
well as dysprosody. Additionally, the motorically complex
DDK task of repetitions of /ptk/ also examined disrupted
transitions and within-speech groping as part of its score.
Descriptions of such coarticulation, sequencing, and groping errors are common in CAS (e.g., ASHA, 2007b; Grigos
& Kolenda, 2010; Thoonen et al., 1996, 1999). These results
suggest that some behaviors on perceptually based feature
checklists are not independent.
The third ASHA criterion, inconsistent errors on
consonants and vowels in repeated productions of words
and syllables (ASHA, 2007b, p. 4), was most notably absent from the models of discriminative measures. Forrest
(2003) found that inconsistency was the feature most used
by clinicians to identify CAS. The present study measured
inconsistency with the DEAP Inconsistency subtest, as it
most closely matched the definition of inconsistency used
in the ASHA Technical Report (2007b)that is, token-totoken inconsistency across separate productions of the same
words. This has also been referred to as target stability
(Marquardt, Jacks, & Davis, 2004). When used as a sole
predictor in a bivariate DFA, inconsistency discriminated
the groups with 30% accuracy and did not contribute significantly to predicting diagnosis in the multivariate analyses.
Other available measures that capture a different type of
inconsistency might yield a different result. In Model 2
(limited data set), inconsistency may have been captured by
the repeated productions of the /ptk/ DDK task, which
would be more similar to token accuracy whereby greater
accuracy demonstrates less inconsistency (Marquardt et al.,
2004). Alternatively, the Inconsistency Severity Percentage
(Iuzzini & Forrest, 2010), for example, is a measure of phonemic and phonetic inconsistency, similar to total token
variability (Marquardt et al., 2004), from multiple elicitations of all consonants in English across varied syllable
or word contexts. Iuzzini (2012) reported that the Inconsistency Severity Percentage differentiated CAS from phonological disorder better than the DEAP Inconsistency measure.
Further investigation of the construct of inconsistency and
factors influencing its nature and degree would help to determine the most informative measure and whether that
might have greater predictive accuracy when combined
with measures of other features associated with CAS. Given
the debate over inconsistency in the acquired AOS literature (see Staiger, Finger-Berg, Aichert, & Ziegler, 2012, for
a review) and the absence of this feature in Strands 10-point
checklist, further research is required on this feature, one
that is strongly represented in clinicians impressions of both
CAS and AOS.
Nonspeech groping (i.e., groping during nonspeech
tasks) was another noteworthy measure. Such nonspeech
oromotor tasks have been used in CAS checklists over time,
presumably as a feature of CAS (e.g., Ozanne, 2005; Royal
College of Speech and Language Therapists, 2011), and nonspeech groping was the second most prevalent feature in
diagnosis in a survey of clinicians (Forrest, 2003). Despite
this, the current study results suggest that nonspeech groping
54
had low diagnostic accuracy independently and did not add

to the diagnostic models. The current data support the findings of Strand and McCauley (2008), as well as studies of
acquired AOS and oral apraxia (see Ballard et al., 2000, for
a review), in that nonspeech features are indicative of oral
apraxia rather than CAS. Therefore nonspeech difficulties
are best considered as concomitant rather than core features
of CAS (ASHA, 2007b; Strand et al., 2013).
The findings from this study indicate that four measures, in combination, diagnosed CAS with high accuracy
against the current gold standard of expert opinion. The
final model (Model 2) did not include children with CAS+
or with the frank oral structural impairment of submucous
cleft, and data from these seven children were used to further test the robustness of the model (see Appendix B). This
shows that although Model 2 was not being developed with
those participants with CAS+dysarthria, it can be used to
determine presence of CAS contributions to their speech
impairment. Only one participant with CAS+ (Participant
No. 1328) was misdiagnosed by the models formula as
non-CAS. This diagnosis was most likely due to his comorbid flaccid dysarthria and a lack of syllable segregation,
considering he was unable to produce any polysyllabic words.
Three important points are demonstrated here. First, replication of this study is required with an unselected sample
that includes more children with dysarthria. Second, an OMA
would be required before using these criteria in the future
to determine frank dysarthria and/or submucous clefts in
those who may also have CAS. Third, children with minimal verbal output may not be diagnosed accurately using the
measures described here because they are based on attempts
at polysyllabic words. In future, syllable segregation could
also be examined in utterances between words rather than
just within polysyllable words using, for example, Shribergs
inappropriate pauses measure (Shriberg, 2013).
Data from four participants are included as case studies in Table 6 to illustrate the use of the criteria and further
explain the weightings and use of the DFA results. The
first two cases are 4-year-old girls, each with severe articulation difficulties, one of whom was diagnosed with CAS
and the other with ataxic dysarthria by the first and second
authors. These cases illustrate the importance all four criteria and weightings of the measures: Each girl had poor
lexical stress matches and a high occurrence of syllable segregation, both of which are features of CAS and ataxic dysarthria (ASHA, 2007b; Duffy, 2012). However, although
the child with CAS had poor (46%) accuracy for the PPC
measure, this child was relatively better than the participant
with ataxic dysarthria (with imprecise articulation), who
had 25% accuracy. This may have helped discriminate these
cases, because the child with ataxic dysarthria exhibited
many more distortions and articulation errors, despite the
first two measures showing a similar pattern. The second
pair of cases illustrates the contrast between two 12-year-old
boys with mild articulation difficulties, one with CAS and
the other with residual phonological impairment. The boy
with CAS had fewer lexical stress matches (26%, compared
to 88% for the child with phonological impairment), more

instances of syllable segregation, and poorer /ptk/ accuracy than the boy with phonological impairment. These
three features are hypothesized to reflect an underlying impairment of motor planning and programming or praxis
(e.g., ASHA, 2007b; Shriberg, Green, et al., 2003; Thoonen
et al., 1999). The criteria provide confidence in discriminating those with some underlying praxis deficits, demonstrating CAS is at least part of the childs presentation.
Therefore, the combination of the four discriminant measures
and their weightings appear crucial in separating verbal
participants with different types of expressive communication disorder and wide ranges of age and severity.
It is important to consider that the assessment procedures used here assessed children at a single time point.
Any developmental disorder can vary in its surface presentation across time. Regular review is therefore indicated to
ensure early detection of other clinical behaviors that might
emerge with therapy and maturation (e.g., literacy concerns)
and ultimately to ensure that management strategies match
presenting and prognostic features (Stackhouse & Snowling,
1992; Zaretsky, Velleman, & Curro, 2010).
Our findings advance the field in this area by identifying two tests that appear central to differentiating CAS
from other disorders, at least in this sample: namely, a complete OMA including a DDK task and a sufficiently large
sample of polysyllabic single-word production (e.g., Gozzard
et al., 2004). Both the real-word polysyllabic test and the
nonword /ptk/ DDK task from the OMA were motorically challenging and appear to successfully elicit behaviors
that reflected the underlying motor planning and programming deficits in CAS at both the segmental and suprasegmental level (Shriberg, Lohmeier, et al., 2009; Thoonen
et al., 1996, 1999). These two tests alone may be sufficient
for reliable diagnosis of CAS in verbal children. However,
SLPs using these tasks still need to consider normal acquisition and development in terms of both segmental and prosody
accuracy (Ballard, Djaja, Arciuli, James, & van Doorn,
2012; James, 2006; Shriberg, Lohmeier, et al., 2009).
Limitations
The major limitation of this study is that it utilized
a selected clinical sample of children with suspected CAS
and selection criteria designed to find idiopathic CAS
rather than comorbid CAS. This approach is not uncommon (e.g., McCauley et al., 2012; Rosenbek & Wertz, 1972;
Yoss & Darley, 1974) and was warranted in exploring and
identifying quantitative measures for further investigation.
As in any multivariate DFA, the results are heavily reliant
on the characteristics of the sample used and the measures
selected for analysis, and there are inherent risks that the
findings might not generalize to a similar but larger sample
or to a different group of children (e.g., children with comorbid intellectual disability and receptive language impairment). For example, the formula used PPC to separate
CAS from ataxic dysarthria. However, there is no reason
that a child with CAS may not have severe articulation
errors. Also, the participant sample here all demonstrated
prosodic errors being features on both the ASHA consensusbased (2007b) and Strand 10-point feature lists (Shriberg,
Potter, & Strand, 2009). However, other samples that demonstrate dysprosody may only account for a subset of children with CAS (e.g., Shriberg, Aram, & Kwiatkowski, 1997)
or younger children may demonstrate other errors of greater
prevalence, such as inconsistency (Iuzzini & Forrest, 2010).
The Model 2 DFA formula based on the limited data set is
reported here to facilitate replication, but caution must be
used in applying it to clinical populations or cases that differ
from the studied sample.
In this study, we relied on perceptual measures of
speech behaviors. This was a deliberate decision considering
that perceptual measures are most commonly used in clinical practice (Duffy, 2012) and have been shown to correlate
highly with acoustic measures of lexical stress (e.g., pairwise variability indices; Ballard et al., 2010). However, the
relatively subjective nature of these measures could result
in discrepancies and therefore errors in the formulas result,
particularly in untrained SLPs. In the current study, training of raters was required to maintain interrater reliability
above 85%. To assist replication in future studies, efforts to
make the measures more objective would be warranted by
providing defined features, measures, and scoring criteria in
a manual or by finding more objective kinematic or acoustic measures. There are promising acoustic measures (e.g.,
Ballard et al., 2012; McKechnie et al., 2008; Shriberg, Green,
et al., 2003) and potential for automation (Hosom, Shriberg,
& Green, 2004; Rosen et al., 2010). However, manual
acoustic measures can be time-intensive and also require a period of training. It may be some time before such acoustic
measures are available in a format that allows rapid automated analysis in clinical settings.
Studies aiming to identify quantitative measures that
differentiate disorders suffer an inherent circularity (ASHA,
2007b; Guyette & Diedrich, 1981). However, here the aim
was not to validate the original checklists (ASHA, 2007b;
Shriberg et al., 2009) used for initial diagnosis. Instead, the
aim was to test the assessment samples to determine whether
a more replicable and efficient method (or set of measures
in combination) could reproduce the initial expert diagnosis
with high accuracy. Our findings suggest that a set of just
four measures strongly predicts presence of CAS in verbal
children. This is a more parsimonious solution than a
checklist of multiple features, of which only a subset is required for diagnosis (e.g., Hickman, 1997; Kaufman, 1995;
Shriberg, Potter, & Strand, 2009).
Further Directions
This preliminary study used a sample of children ages
412 years with idiopathic and comorbid CAS and compared them to others who had been suspected to have CAS
but were instead diagnosed with phonological or language
impairment, submucous cleft, or dysarthria. Further research is necessary to test the robustness of the sets of criteria and the resultant formula identified when diagnosing
an unselected community sample of children with suspected

55
expressive communication disorders. Other measures could

also be assessed for their predictive accuracyfor example,
the Inconsistency Severity Percentage inconsistency measure (Iuzzini & Forrest, 2010).
Further research is also warranted for children with
suspected CAS and limited verbal output. The majority of
participants in this study used some polysyllabic words.
Use of dynamic assessment procedures is likely to be a better interim option for diagnosing children with only monosyllabic productions (e.g., observing the childs responses
to cues such as reducing speech rate and providing tactile
cues; see Strand et al., 2013). Moreover, efforts should be
directed toward finding and testing more objective, possibly
acoustic or kinematic measures of behaviors that correlate
highly with our perception. Likewise, structural and functional neuroimaging studies might shed light on the nature
of CAS and its response to different types of intervention.
Conclusions
This study commenced with expert diagnosis of children with suspected CAS and then extracted quantitative
measures that were analyzed to determine whether any combination predicted the expert diagnosis. CAS and non-CAS
in verbal 4- to 12-year-olds in this sample could be discriminated with 91% accuracy based on four measures, following completion of a thorough OMA including accuracy on
/ptk/ (Robbins & Klee, 1987) and a 50-word sample
of polysyllable words (Gozzard et al., 2004). The results met
Shribergs criteria of >90% sensitivity and specificity, but
within a selected sample. A formula based on the results of
DFA is provided to assist in application and replication.
Further research is required with a larger unselected sample to ensure that the four measures and the resultant formula can differentiate CAS in a wider population from
typical development and other speech, neurological, and
linguistic disorders. Investigation of kinematic, acoustic,
and other assessment tools for CAS features would also
be beneficial.
Acknowledgments
This research was supported by the Douglas and Lola Douglas
Scholarship on Child and Adolescent Health, the Nadia Verrall
Memorial Research Grant (2010), a Postgraduate Research Award
(2011) from Speech Pathology Australia, and the James Kentley
Memorial Scholarship and Postgraduate Research Support Schemes
to Elizabeth Murray; a University of Sydney International Program Development Fund Grant to Patricia McCabe and Kirrie
Ballard; and an Australian Research Council Future Fellowship
(FT120100255) to Kirrie Ballard. Parts of this article were presented
at the Motor Speech Conference in Santa Rosa, CA, 2012, and the
Speech Pathology Australia Conference in Hobart, Tasmania, 2012.
We thank research assistants Morin Beausoleil, Virginia Caravez,
Claire Formby, Jennifer Fortin Zornow, Sally Hanna, Claire Layfield,
Aimee-Kate Parkes, Gemma Patterson, Alyssa Piper, and Caitlin
Winkelman. Thank you also to Samantha Warhurst, Kate Anderson,
and Claire Layfield for editorial suggestions.
56
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Appendix A
Diagnostic results for all participants using the ASHA criteria (2007b) and the Strand 10-point checklist (Shriberg, Potter, &
Strand, 2009).
Participant no.
ASHA
criteria meta
Strand
criteria metb
1217
1314
2/3
2/3
5/10
6/10
0236
1313
0235
0/3
1/3
0/3
1/10
1/10
0/10
0333
0/3
3/10
0/3
0/3
0/3
0/3
0/3
1/3
1/3
1/3
0/3
0/15
1/10
2/10
2/10
1/10
2/10
6/10
2/10
2/10
1/10
3/15
3/3
7/10
2105
1214
1328
3/3
3/3
3/3
8/10
9/10
8/10
No. of CAS+ participants

that met criteria
CAS
1103
1101
1102
1104
2106
2107
2108
1210
1211
2212
1213
1215
2216
2217
2218
2219
2320
1321
2322
2323
1324
1325
2326
1329
1330
0331
1316
2109
No. of CAS participants
that met criteria
3/3
8/10
3/3
3/3
3/3
3/3
3/3
3/3
3/3
3/3
3/3
3/3
3/3
3/3
3/3
3/3
3/3
3/3
3/3
3/3
3/3
3/3
3/3
3/3
3/3
3/3
3/3
3/3
3/3
3/3
28/28
8/10
7/10
8/10
6/10
9/10
7/10
8/10
8/10
9/10
8/10
7/10
6/10
7/10
7/10
7/10
5/10
7/10
5/10
8/10
5/10
5/10
7/10
6/10
6/10
7/10
7/10
5/10
6/10
28/28
Group
Non-CAS
0334
0237
1311
2315
1312
0138
0139
1310
0335
No. of non-CAS participants
that met criteria
CAS+
0332
Diagnosis
Ataxic dysarthria (+ receptive & expressive language disorder)
Flaccid dysarthria (CNXII) (+ receptive & expressive language
disorder)
Phonological disorder (+ expressive language disorder)
Phonological disorder
Phonological disorder (+ receptive & expressive language disorder);
hoarse voice
Phonological disorder (+ receptive & expressive language disorder);
suspected global developmental delay
Phonological disorder (+ receptive & expressive language disorder)
Phonological disorder (+ receptive & expressive language disorder)
Comorbid phonological disorder and stuttering
Submucous cleft palate (+ expressive language disorder)
Submucous cleft palate
Submucous cleft palate
Phonological disorder (+ expressive language disorder)
CAS and ataxic dysarthria (+ receptive & expressive language
disorder)
CAS and flaccid dysarthria (CNXII) (+ expressive language disorder)
CAS and flaccid dysarthria (CNXII) (+ expressive language disorder)
CAS, flaccid dysarthria (CNXII) (+ receptive & expressive language
disorder)
CAS
CAS
CAS
CAS
CAS
CAS
CAS
CAS
CAS
CAS
CAS
CAS
CAS
CAS
CAS
CAS
CAS
CAS
CAS
CAS
CAS
CAS
CAS
CAS
CAS
CAS
CAS
CAS
(+ expressive language disorder)



ASHA criteria = 3/3 needed for CAS diagnosis. bStrand criteria = 4/10 needed for CAS diagnosis over three tasks.

59
Appendix B
Sensitivity and specificity based on the Model 2 DFA.
Model 2 participants
Predicted diagnosis (from DFA)
Expert diagnosis
CAS (n = 28) (excluding CAS+)
True positive = 100% (n = 28)
False negative = 0% (n = 0)
Non-CAS (n = 12) (excluding submucous clefts)

False positive = 0% (n = 0)
True negative = 100% (n = 12)
Note. Positive likelihood ratio = infinity; negative likelihood ratio = 0.
All participants
Predicted diagnosis (from DFA)
Expert diagnosis
CAS (n = 32) (including CAS+)
True positive = 97% (n = 31)
False negative = 3% (n = 1)
Non-CAS (n = 15) (including submucous clefts)

False positive = 0% (n = 0)
True negative = 100% (n = 15)
Note. Positive likelihood ratio = 16; negative likelihood ratio = 0.
60

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JSLHR

Differential Diagnosis of Children with

Purpose: The gold standard for diagnosing childhood

or dysarthria. Following this, 24 different measures from

it remains difficult to differentially diagnose CAS from

hildhood apraxia of speech (CAS) is considered an

The University of Sydney, Australia

Diagnosis of Childhood Apraxia of Speech

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(e.g., Thoonen, Maassen, Gabrels, & Schreuder, 1999),

The Royal College of Speech and Language Therapists also released a

with >90% sensitivity (identifying true cases of CAS) and

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needed that will reliably discriminate CAS from a wide

measures were analyzed using bivariate and hierarchical

Speech and Language Assessment Battery

Murray et al.: Differential Diagnosis of CAS

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No articulation or prosodic impairment noted by parents, no CAS suspected by SLP.

the Diagnostic Evaluation of Articulation and

the Single-Word Test of Polysyllables (Gozzard, Baker,

a connected speech sample of at least 50 utterances

the Oral and Speech Motor Control Protocol, a

the Clinical Evaluation of Language Fundamentals

expressive language skills (Semel, Wiig, & Secord,

Expert Diagnosis of CAS

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Strands 10-point checklistb

Assessment tasks considered in diagnosisc

DEAP Inconsistency subtest (inconsistency defined

1. Difficulty achieving initial articulatory

Polysyllable Test,e connected speech samplef

4. Vowel or consonant distortions including

Polysyllable Teste (DEAP Inconsistency subtest,d

Note. DEAP = Diagnostic Evaluation of Articulation and Phonology; DDK = diadochokinesis.

following procedure. To be diagnosed with CAS in this study,

Appendix A shows the CAS features judged to be

Murray et al.: Differential Diagnosis of CAS

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Relation to diagnostic criteria

DEAP Inconsistencya vs.

Oral and Speech Motor

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phonemes correct from the 12 monosyllable items in the

Murray et al.: Differential Diagnosis of CAS

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(2007) and used up to eight predictors at a time. Bivariate

a relatively straightforward manner with visual inspection

Diagnosis Constant % lexical stress matches its weight

Table 4 reports the descriptive statistics for each of

syllable segregation score its weight PPC

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Murray et al.: Differential Diagnosis of CAS

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cleft, phonological impairment, or dysarthria (non-CAS).

Table 6. Using the DFA Model 2 formula to diagnose CAS.

Participant No. 2216: CAS,

= round(0.504 + (26 0.011)

1 = likely CAS; 0 = likely non-CAS.

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The procedures used here for expert judgment on

two assessment tasks: a polysyllabic word picture-naming

Murray et al.: Differential Diagnosis of CAS

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