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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 or dysarthria. Following this, 24 different measures from
apraxia of speech (CAS) is expert judgment of perceptual the diagnostic assessment were rated by blinded raters.
features. The aim of this study was to identify a set of Multivariate discriminant function analysis was used to
objective measures that differentiate CAS from other speech identify the combination of measures that best predicted
disorders. expert diagnoses.
Method: Seventy-two children (4–12 years of age) Results: The discriminant function analysis model, including
diagnosed with suspected CAS by community speech- syllable segregation, lexical stress matches, percentage
language pathologists were screened. Forty-seven phonemes correct from a polysyllabic picture-naming task,
participants underwent diagnostic assessment including and articulatory accuracy on repetition of /pǝtǝkǝ/, reached
presence or absence of perceptual CAS features. Twenty- 91% diagnostic accuracy against expert diagnosis.
eight children met two sets of diagnostic criteria for CAS Conclusions: Polysyllabic production accuracy and an oral
(American Speech-Language-Hearing Association, 2007b; motor examination that includes diadochokinesis may be
Shriberg, Potter, & Strand, 2009); another 4 met the CAS sufficient to reliably identify CAS and rule out structural
criteria with comorbidity. Fifteen were categorized as abnormality or dysarthria. Testing with a larger unselected
non-CAS with phonological impairment, submucous cleft, sample is required.

C
hildhood apraxia of speech (CAS) is considered an it remains difficult to differentially diagnose CAS from
impairment of speech motor control or praxis. Most other disorders. In the absence of a clinically available vali-
researchers agree that the core deficit for children dated assessment procedure, the current gold standard for
with CAS is a reduced or degraded ability to convert abstract diagnosis is expert opinion (Maas, Butalla, & Farinella,
phonological codes to motor speech commands, referred to 2012). The purpose of this study was to determine, after ex-
as motor planning and/or programming (American Speech- pert diagnosis, if a quantitative measure or set of measures
Language-Hearing Association [ASHA], 2007b; Nijland, differentiated CAS from non-CAS in a sample of children
Maassen, & Van der Meulen, 2003; Shriberg, Lohmeier, referred from the community with suspected CAS.
Strand, & Jakielski, 2012). This consensus has been supported
by behavioral studies (see ASHA, 2007b, and McCauley, Diagnosis of Childhood Apraxia of Speech
Tambyraja, & Daher-Twersky, 2012, for reviews), classifi-
cation paradigms (Shriberg et al., 2010), and computational CAS may occur as a result of known neurodevelop-
modeling studies (Terband & Maassen, 2010; Terband, mental disorders (Kummer, Lee, Stutz, Maroney, & Brandt,
Maassen, Guenther, & Brumberg, 2009). The impairment 2007; Shriberg, Potter, & Strand, 2011; Spinelli, Rocha,
then manifests itself as disordered articulation, difficulty Giacheti, & Richieri-Costa, 1995) or deleterious genetic
sequencing sounds and syllables, inconsistent production of mutation (e.g., Brunetti-Pierri et al., 2011; Carr et al., 2010;
repeated sounds and syllables, and disruption at the supra- Palka et al., 2012). The majority of reported cases, however,
segmental level (i.e., dysprosody; ASHA, 2007b). Despite are idiopathic.
the recent advances in our theoretical understanding of CAS, Despite much research, there is currently no single
neurological or behavioral diagnostic marker for all cases
of CAS (ASHA, 2007b). Behavioral measures that have
a
The University of Sydney, Australia been considered have included inconsistent speech fea-
Correspondence to Elizabeth Murray: Elizabeth.murray@sydney.edu.au tures (e.g., Iuzzini, 2012), coarticulation and timing errors
Editor: Jody Kreiman (e.g., Sussman, Marquardt, & Doyle, 2000), prosody (e.g.,
Associate Editor: Ben A. M. Maassen Munson, Bjorum, & Windsor, 2003; Thoonen, Maassen,
Received November 9, 2012 Wit, Gabreëls, & Schreuder, 1996), speech production
Revision received May 10, 2013
Accepted October 13, 2014 Disclosure: The authors have declared that no competing interests existed at the time
DOI: 10.1044/2014_JSLHR-S-12-0358 of publication.

Journal of Speech, Language, and Hearing Research • Vol. 58 • 43–60 • February 2015 • Copyright © 2015 American Speech-Language-Hearing Association 43
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(e.g., Thoonen, Maassen, Gabreëls, & Schreuder, 1999), with >90% sensitivity (identifying true cases of CAS) and
speech perception (e.g., Nijland, 2009), linguistic skills (e.g., specificity (identifying true cases of non-CAS), discriminat-
Lewis, Freebairn, Hansen, Iyengar, & Taylor, 2004), and ing CAS from a range of other expressive communication
nonspeech oral motor skills (e.g., Murdoch, Attard, Ozanne, disorders (Shriberg et al., 2012). The closest we have is
& Stokes, 1995). Diagnostic marker research has included the contribution of 89% sensitivity and 100% specificity by
many contributions by Shriberg and colleagues utilizing their Thoonen and colleagues (1996, 1997, 1999) in discriminating
Madison Speech Assessment Protocol and Speech Disorders spastic dysarthria and CAS using maximum performance
Classification System (SDCS) to attempt to differentiate tasks. However, the procedure may not discriminate across
CAS and acquired apraxia of speech from normal develop- subtypes of dysarthria and other speech sound disorders (SSD)
ment and other communication disorders (e.g., Shriberg, because the validation sample used was small (n = 11).
Aram, & Kwiatkowski, 1997; Shriberg et al., 2010; Developing a reliable testing protocol that captures
Shriberg, Green, Campbell, McSweeny, & Scheer, 2003; the small set of agreed behavioral features of CAS is chal-
Shriberg, Lohmeier, et al., 2009). lenging when one considers a number of methodological
In the absence of a single pathognomonic feature, a constraints of previous research. These include lack of spec-
core set of validated differential diagnostic markers for CAS ificity of features within checklists used for diagnosis of
is needed (ASHA, 2007b; Davis, Jakielski, & Marquardt, CAS, various participant selection criteria, insufficient
1998). In 2007, ASHA released a position statement and operationalization of methods for replication, and different
technical report on CAS (ASHA, 2007a, 2007b). The tech- comparison groups. Checklists of features, such as Strand’s
nical report provided a consensus position on a small set 10-point checklist (Shriberg, Potter, et al., 2009) and others,
of perceptual speech features associated with the primary guide the clinician in making presence or absence decisions
planning and programming deficits in CAS. Following a for a range of speech, oromotor, and linguistic behaviors;
thorough literature review of group-comparison diagnostic however, for the most part, these checklists do not include
studies and community consultation, three features with explicit definitions of the features or methods for determining
some consensus were identified across investigators in apraxia: how much or how often any single behavior must be observed
“(a) inconsistent errors on consonants and vowels in re- (e.g., Crary, 1995; Davis & Velleman, 2000; cf. Thoonen
peated productions of syllables or words, (b) lengthened and et al., 1999; see Rvachew, Hodge, & Ohberg, 2005, for a tu-
disrupted coarticulatory transitions between sounds and torial), which affects application or replication of the results.
syllables, and (c) inappropriate prosody, especially in the The wide range of behaviors that have been considered for
realization of lexical or phrasal stress” (ASHA, 2007b, these checklists reflects (a) the belief that symptoms of CAS
pp. 4, 54, and 59).1 These features have been used for diag- change with age and/or maturation and (b) the different
nosis in recent CAS treatment studies in which participants theoretical perspectives as to whether CAS has core or co-
had to demonstrate all three features to warrant diagnosis occurring motor, oral motor, phonological or linguistic
(e.g., Ballard, Robin, McCabe, & McDonald, 2010; Maas memory, and literacy impairments (Alcock, Passingham,
et al., 2012). Another recent checklist that is used for CAS Watkins, & Vargha-Khadem, 2000; Marion, Sussman, &
diagnosis in research and clinical settings is Strand’s 10-point Marquardt, 1993; Shriberg et al., 2012). Furthermore, many
checklist (Shriberg et al., 2012; Shriberg, Potter, & Strand, surface behavioral features are shared by a range of commu-
2009). This checklist provides 10 segmental and supra- nication disorders, making discrimination of phonological-
segmental features that can be present in CAS, although linguistic and phonetic-motoric errors difficult (see Ballard,
any combination of at least four out of the 10 features across Granier, & Robin, 2000; McCabe, Rosenthal, & McLeod,
three tasks warrants the diagnosis of CAS. 1998; McNeil, Robin, & Schmidt, 2009).
As Strand and colleagues remarked, feature lists do not The methods of participant selection have also dif-
directly lead to an assessment procedure (Strand, McCauley, fered across studies, contributing to confusion. Some studies
Weigand, Stoeckel, & Baas, 2013). Commercial norm- have included participants with CAS and concomitant
referenced assessment tools offer clear assessment tasks hearing impairment, intellectual disability, and language
but often lack essential psychometric properties (McCauley & impairment (Shriberg et al., 2012), whereas others have
Strand, 2008) and may also rely on feature lists or scales for used participants with idiopathic CAS and no known
diagnosis of CAS (Blakeley, 2001; Hickman, 1997; Kaufman, comorbid disorders (e.g., Thoonen et al., 1997). When
1995). The recently published Dynamic Evaluation of Motor comparison groups have been used, specifically selected diag-
Skills is reliable but risks failing to identify some persons noses have been compared to CAS. Groups of participants
with CAS (Strand et al., 2013). To date, there is no ac- have included those with “speech delay” or “speech sound
cepted, operationally defined, diagnostic testing protocol or disorder” with or without language delay or impairment
clinically available and validated set of behavioral features, (e.g., Lewis et al., 2011; Shriberg, Green, et al., 2003; Shriberg
et al., 2012), dysarthria (e.g., Morley, Court, & Miller,
1
The Royal College of Speech and Language Therapists also released a
1954; Rosenbek & Wertz, 1972; Thoonen et al., 1999),
Policy Statement with a wider list of consensus features for CAS (Royal phonological impairment (e.g., Williams, Ingham, &
College of Speech and Language Therapists, 2011). This statement was Rosenthal, 1981; Yoss & Darley, 1974), specific language
published after data collection for the current study and was therefore impairment (e.g., Lewis et al., 2004), or stuttering (Byrd &
not considered. Cooper, 1989). Ultimately a set of quantifiable measures is

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needed that will reliably discriminate CAS from a wide measures were analyzed using bivariate and hierarchical
range of communication disorders that could present with multivariate discriminant function analysis (DFA) to deter-
overlapping symptoms, whether within individuals or be- mine if any combination of the 24 measures reliably pre-
tween groups of individuals. dicted the assigned CAS diagnosis.
The predominant approach in current treatment re-
search studies of CAS is, first, to use a range of standard- Participants
ized assessments to rule out frank receptive and expressive
language impairment or orofacial structural abnormality Participants were recruited via a website advertisement
and, second, to use a checklist to note CAS-type behaviors and flyers as well as e-mails and listserv posts to speech-
observed and so justify a diagnosis of CAS (e.g., Ballard language pathologists (SLPs), inviting them to volunteer for
et al., 2010; Martikainen & Korpilahti, 2011). However, as a research treatment study. The treatment component of this
stated above, the feature lists have not been operationalized, study is not discussed in this article. The inclusion criteria for
validated, or standardized, and no standard approach or participants were (a) a clinical diagnosis of suspected CAS
battery for assessment of these specific features has yet been by a community-based SLP, (b) age between 4 and 12 years,
developed (ASHA, 2007b; Shriberg et al., 2012). As such, (c) no previously identified language comprehension diffi-
it is likely that expert and novice clinicians would demon- culty, (d) normal or adjusted-to-normal hearing and vision,
strate low interrater reliability, as already shown in studies (e) native English speaker, and (f ) no other developmental
of CAS (Shriberg, Campbell, et al., 2003) and dysarthria diagnoses not associated with CAS (e.g., intellectual disability,
(Zeplin & Kent, 1996; Zyski & Weisiger, 1987) using per- autism, cerebral palsy). All participants had received speech-
ceptual, criterion-based diagnostic methods. This uncertainty language pathology intervention prior to this study, and
results in clinicians in practice making diagnoses of “sus- all had normal hearing. Participants needed to attend the
pected CAS,” with risk of under- or overidentification on-campus clinic at the University of Sydney, Australia, to
(Davis et al., 1998; Forrest, 2003; Shriberg, Campbell, participate. Parents or caregivers provided informed consent
et al., 2003). The result can be selection of inappropriate on behalf of the children prior to participation.
treatment approaches and the frequent complaint that chil- Of the 72 participants who inquired regarding the
dren with CAS progress slowly in therapy (Forrest, 2003). study, 47 passed the initial screening. All 47 were Australian-
This study furthers the CAS diagnostic literature by attempt- English speaking, 33 (70%) were boys, 14 were girls (30%),
ing to determine if clinically available measures can discrimi- and the average age was 70.5 months (SD = 25.7 months).
nate CAS from other expressive communication disorders No information on race, ethnicity, or socioeconomic status
in a sample of verbal children. was collected.

Aim Clinical Assessment


The first author responded to all inquiries to partic-
The primary aim of this study was to use objective
ipate in the study and followed a two-tiered assessment
statistical methods to identify one or more quantitative
protocol (see Figure 1). The first tier involved screening of
measures of speech that reproduce the expert classification
parents or caregivers and SLPs by phone and previous
of children as having CAS or not. This approach has poten-
speech pathology reports to determine the child’s suitability
tial to provide a clinically feasible assessment protocol that
for entry into the study. The criteria were applied conser-
reliably identifies features of CAS and reduces reliance on
vatively, in that children with another developmental disor-
expert opinion.
der that could be associated with symptoms or consequences
of CAS (e.g., selective mutism) were included. For those
Method who passed the initial screening, the second tier involved ad-
ministration of a 2-hour speech and language assessment
This study formed part of a larger clinical trial: battery. Responses to these tests were used for all analyses
Unique Trial Number: U1111-1132-5952, Trial Registration of CAS features and to determine the presence of comorbid
Number: ACTRN12612000744853. The research protocol conditions such as dysarthria or language impairment. The
was approved by the Human Research Ethics Committee of assessment battery was video- and audio-recorded using
the University of Sydney (12924) and is published (Murray, a Sony IC Recorder ICD-UX71F and either an Echo Layla
McCabe, & Ballard, 2012). 24/96 multitrack recording system, Marantz PMD660 solid-
This diagnostic study was conducted in three phases state recorder, or Roland Quad-Capture UA-55 at the sam-
to address the research aim. First, children were recruited pling rate 48000 Hz with 16-bit resolution with an AKG
and assessed according to the protocol below. Second, the C520 headset microphone at 5 cm mouth-to-microphone
first two authors (E. M. and P. M.) judged presence or ab- distance.
sence of CAS by listening to speech samples from the as-
sessment protocol and completing two published checklists
of CAS characteristics. Third, the first author (E. M.) and Speech and Language Assessment Battery
independent raters blinded to the diagnosis of CAS gener- First, a parent or caregiver case history questionnaire
ated 24 quantitative measures from assessment data. These and hearing screening was completed (ASHA, 1978). Next,

Murray et al.: Differential Diagnosis of CAS 45


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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.

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

the diagnostic assessment utilized five published tests com- expressive language skills (Semel, Wiig, & Secord,
monly available in clinical settings and culturally appropriate 2006; Wiig, Secord, & Semel, 2006).
for Australian children (see Table 1). The five tests were the All tests were administered and scored by the first
following: author according to the relevant manual. For the DEAP
Inconsistency subtest, broad transcription was used and dis-
1. the Diagnostic Evaluation of Articulation and
tortions and developmental errors were scored as correct,
Phonology (DEAP) Inconsistency subtest (Dodd, Hua,
according to Dodd et al. (2002, p. 25).
Crosbie, Holm, & Ozanne, 2002), used to assess the
Initially, responses to the assessments were used for
word-level token-to-token inconsistency in naming
expert qualitative judgments of presence or absence of CAS
25 pictured words over three test administrations, each
and, following this, for generating 24 quantitative measures
administration separated by other assessment tasks;
for statistical analyses in order to address the study aim.
2. the Single-Word Test of Polysyllables (Gozzard, Baker, There was overlap in the speech samples used to make the
& McCabe, 2004, 2008), a 50-item picture-naming expert judgments and to generate the quantitative measures;
task used to assess articulation, sound and syllable however, expert judgments were made prior to extracting the
sequencing, and lexical stress accuracy (i.e., prosody); wider range of measures from the assessment, and measure
3. a connected speech sample of at least 50 utterances calculations did not influence the initial classifications made.
recorded over at least 10 minutes (McLeod, 1997), for In addition, three raters scored 100% of the data: the first
detection of perceptual features of CAS in connected author and two independent raters blinded to expert diagnosis.
speech and calculation of articulation rate; A detailed description of reliability calculations is provided
in the Reliability section.
4. the Oral and Speech Motor Control Protocol, a
published oral motor assessment (OMA) including
diadochokinesis (DDK; Robbins & Klee, 1987), used Expert Diagnosis of CAS
to rule out any structural or functional abnormalities The first and second authors, each with more than
in the oral mechanism; and 10 years’ experience in differential diagnosis of pediatric
5. the Clinical Evaluation of Language Fundamentals speech sound disorders, independently diagnosed the pres-
(CELF; 4th edition or Preschool–2nd edition, ence and severity of CAS for all children based on their per-
Australian versions), used to assess receptive and ceptual ratings of each child’s speech samples, using the

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Table 1. Assessment tasks and diagnostic features used for assigning an expert diagnosis of childhood apraxia of speech.

ASHA consensus-based
feature list a Strand’s 10-point checklistb Assessment tasks considered in diagnosisc

1. Inconsistent errors on Not in list DEAP Inconsistency subtest (inconsistency defined


consonants and vowels in as a score ≥40%),d (Polysyllable Test,e connected
repeated productions of speech samplef )
syllables or words
2. Lengthened and disrupted 1. Difficulty achieving initial articulatory Polysyllable Test,e connected speech samplef
coarticulatory transitions configurations and transitions into vowels (DEAP Inconsistency subtestd)
between sounds and syllables 2. Syllable segregation
3. Inappropriate prosody, 3. Lexical stress errors or equal stress Polysyllable Teste (DEAP Inconsistency subtest,d
especially in the realization of Connected speech samplef )
lexical or phrasal stress 4. Vowel or consonant distortions including Polysyllable Teste (DEAP Inconsistency subtest,d
distorted substitutions Connected speech samplef )
5. Groping (nonspeech) Oral and Speech Motor Control Protocol (any three
nonspeech tasks)g
6. Intrusive schwa Polysyllable Teste (DEAP Inconsistency subtest,d
Connected speech samplec)
7. Voicing errors Polysyllable Teste (DEAP Inconsistency subtest,d
Connected speech samplef )
8. Slow rate Connected speech samplef Polysyllable Teste
(DEAP Inconsistency subtestd)
9. Slow DDK rate Polysyllable Teste (DEAP Inconsistency subtest,d
Connected speech samplef )
10. Increased difficulty with longer or more Polysyllable Teste (DEAP Inconsistency subtest,d
phonetically complex words Connected speech samplef )

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


a
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, Appendix A shows the CAS features judged to be
participants had to demonstrate (a) the three consensus-based present and the expert decision on presence or absence of
features listed in the ASHA Technical Report (2007b) and CAS for each of the 47 participants. Of these 47, 28 (60%)
(b) any four of the 10 features in Strand’s 10-point checklist were classified as having CAS (ASHA, 2007b; Shriberg,
(Shriberg, Potter, et al., 2009) over at least three assessment Potter, et al., 2009). Another four children (8%) met the
tasks as shown in Table 1. For the 10-point checklist, a verbal criteria for CAS but presented with comorbid dysarthria and
behavior was marked as present when perceptually detected receptive and/or expressive language disorder (CAS+),
in speech samples from three tasks: the DEAP Inconsistency similar to the Shriberg et al. (2010) classification of motor
assessment, the Single-Word Test of Polysyllables, and the speech disorder–apraxia of speech plus (MSD-AOS+).
connected speech sample. Neither published list provides Children diagnosed with CAS or CAS+ showed a median
operational definitions or cutoffs for the listed features. of 6 out of 10 features on the Strand 10-point checklist
The first and second authors listened to the speech (Shriberg, Potter, et al., 2009). Fifteen (32%) of the 47 chil-
samples and independently made a perceptual judgment as dren did not demonstrate all three features in the ASHA
to whether each feature was present or absent, except in the feature list and were classified as non-CAS in this study.
case of inconsistency of speech errors. For this one feature Non-CAS diagnoses included ataxic dysarthria (n = 1), flaccid
only, decisions were based on the score from the DEAP dysarthria (n = 1), submucous cleft (n = 3), or primarily
Inconsistency subtest, the only published test that defines phonological disorder (n = 10; see Figure 1). Phonological
inconsistency in the same way as the ASHA Technical impairment was identified in those who had clear phono-
Report (ASHA, 2007b). Syllable segregation was defined logical processes (Rvachew & Brosseau-Lapré, 2012) and
by the authors as “noticeable gaps between syllables.” Two an absence of oromotor or speech-motor deficits (e.g., DDK
criteria showed potential overlap, and, because no previous deficits) excluding them from motor speech diagnosis.
definitions were available, the authors defined difficulty
achieving initial articulatory configurations and transitions
into vowels to include within-speech groping, false starts, Quantitative Measures
restarts, and hesitations and defined groping as nonspeech Quantitative measures were extracted from the five
oral groping. Nonspeech groping was determined over tests administered in the clinical assessment. Table 2 lists
three oral motor tasks within the Robbins and Klee protocol the 24 measures generated and the associated assessment
(1987). For expert perceptual diagnosis of CAS, interrater task and CAS checklist feature. For measures that were
agreement between the first two authors was 100%. not standard scores from norm-referenced tools, nine raters

Murray et al.: Differential Diagnosis of CAS 47


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48

Table 2. Extracted measures from assessment tasks completed with means and standard deviations for the CAS, CAS+, and non-CAS groups.
Journal of Speech, Language, and Hearing Research • Vol. 58 • 43–60 • February 2015

CAS (n = 28) CAS+ (n = 4) Non-CAS (n = 15)


Assessment task Relation to diagnostic criteria Measure M SD M SD M SD

Case history None Age in months 66.5 24.8 72.5 26.0 77.3 27.6
DEAP Inconsistency Inconsistent errors on consonants and Percentage inconsistency (across three 63.8 12.8 75.0 15.1 39.9 24.5
subtesta vowels in repeated productions of repetitions of 25 words)a
syllables or wordsf
Single-Word Test of Inappropriate prosody, especially in the Percentage of lexical stress matches 9.8 9.1 16.3 17.5 67.3 22.4
Polysyllablesb realization of lexical or phrasal stressf (relative to gloss)h
and equal or lexical stress errorsg
Vowel or consonant distortions including Distortion occurrences (out of a possible 39.4 5.8 45.2 8.8 46.24 22.9
distorted substitutionsg 328 phones)h
Syllable segregationg Syllable segregation occurrences (out of a 30.2 8.8 8.5 8.4 1.2 2.1
possible 114 opportunities)i
Intrusive schwag Intrusive schwa occurrences (out of a 1.1 1.3 0.5 1.0 0.0 0.0
possible 15 clusters)i
g
Voicing errors Voicing error occurrences (out of a possible 3.4 4.0 6.0 5.0 1.7 1.5
90 opportunities)h
None PPCh 52.2 15.0 24.3 20.6 73.1 26.3
None PCC-Rh,j 54.0 20.0 22.8 23.8 70.9 26.4
None PVC on polysyllable testh 50.5 11.4 24.8 19.0 75.1 27.3
DEAP Inconsistencya vs. Increased difficulty with longer or more Magnitude of change score: NPC on 12 1.23 0.2 2.52 1.9 2.07 2.91
Polysyllablesb phonetically complex wordsg monosyllables/NPC on 12 polysyllables
(>1 = ↑ difficulty with polysyllables)
Connected speech Slow rateg Articulation rate (syllables per minute)k 1.7 1.0 0.9 0.6 2.6 1.0
samplec Lengthened and disrupted coarticulatory Presence of false articulatory starts and 0.9 0.4 1.0 0.0 0.1 0.3
transitions between sounds and restarts and/or inaudible within-speech
syllablesf and difficulty achieving initial groping and/or audible within-speech
articulatory configurations and groping and/or hesitations (min. = 0,
transitions into vowelsg max. = 1)l,m
Oral and Speech Motor Groping (nonspeech)g Presence of nonspeech groping in lip and 0.5 0.5 0.5 0.6 0.0 0.0
Control Protocold tongue oral function tasks (min. = 0,
max. = 1)
Slow DDK rateg /pǝ/ rate over 3 s on two trialsd 3.6 1.0 3.0 1.7 4.5 2.0
Slow DDK rateg /tǝ/ rate over 3 s on two trialsd 3.4 1.0 2.3 1.3 4.4 1.7
Slow DDK rateg /kǝ/ rate over 3 s on two trialsd 2.9 1.2 1.8 1.0 4.3 1.9
Slow DDK rateg /pǝtǝkǝ/ rate over 3 s on two trialsd 0.9 0.6 0.5 0.1 1.4 0.8
Slow DDK rateg /pætikeɪk/ rate over 3 s on two trialsd 1.0 0.4 0.7 0.4 1.3 0.7
None Accuracy on /pǝtǝkǝ/ DDK task on 48.5 22.6 23.8 9.0 75.9 23.2
two trialsd
None Oral structure scored 23.4 .91 21.0 2.6 21.8 2.4
None Oral function scored 76.6 13.8 60.0 12.4 88.5 18.7
None Maximum Phonation Timed 9.6 5.5 6.8 4.1 12.9 6.7
CELFe None Receptive Language Scoree 100.3 12.2 65.5 18.4 91.4 20.5
None Expressive Language Scoree 85.5 20.1 55.5 7.3 79.7 19.2

Note. CAS = childhood apraxia of speech; M = mean; SD = standard deviation; PPC = percentage phonemes correct; PCC-R = percentage consonants correct–revised; 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).

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were employed to make measurements blinded to the chil- phonemes correct from the 12 monosyllable items in the
dren’s expert diagnosis of CAS presence or absence for reli- DEAP Inconsistency subtest to the first 12 items from the
ability purposes. The raters were qualified SLPs with an Single-Word Test of Polysyllables. A magnitude of change
average of 3 years of clinical experience. The first author score greater than 1 indicated that production of polysyllabic
trained each rater on the methods of measurement until a words was more difficult than monosyllabic words. The only
minimum of 85% interrater reliability was achieved on a binary measures used were for presence of (a) false articula-
training sample not included in the study. Raters were ran- tory starts, restarts, or within-speech groping and (b) non-
domly assigned samples from 12 to 13 children (M = 12.70, speech groping movements.
SD = 4.83). Each participant’s results were rated by at least
two raters. In the event of any discrepancies, a third inde-
pendent rater measured the sample(s), and the first author’s Reliability
scores were retained based on above 85% agreement for all Intrarater and interrater transcription reliability was
measures (see the Reliability section). calculated on a point-to-point basis for all unstandardized
The DEAP Inconsistency subtest was transcribed dependent measures for each participant. Intrarater reliabil-
using broad transcription following the manual (Dodd et al., ity was calculated on 20% of each measure for every partici-
2002). Responses to the Single-Word Test of Polysyllables pant’s data with each rater calculating the measure once
were transcribed from an audio recording into the PROPH and then a second time at least 2 weeks after the initial cal-
module in Computerized Profiling (Long, Fey, & Channell, culation. Intrarater reliability for the first author was 94.6%
2006). Transcription included broad transcription supple- (SD = 3.6, range = 88–98) and across all nine independent
mented with International Phonetic Alphabet diacritics to raters was 95.2% (SD = 3.1, range = 88.3–99.1). Interrater
mark distortions where clearly present. Measures extracted reliability was calculated first between the first author
from PROPH were percentage consonants correct–revised and the nine raters and then between the nine independent
(PCC-R; Shriberg, Austin, Lewis, McSweeny, & Wilson, raters. Mean interrater reliability with the first author across
1997), percentage vowels correct (PVC), percentage phonemes all measures was 94.2% (SD = 1.7, range = 91.3–96.2) and
correct (PPC), occurrences of intrusive schwa, distorted between the nine raters was 93.3% (SD = 2.9, range = 86.4–
substitutions and voicing errors, and percentage of syllable 98.1). For a breakdown of each rater’s and measure’s reli-
stress matches compared to the gloss for each word. Syllable ability, see online Supplemental Appendices 1 and 2.
segregation (transcribed as a plus sign “+” between sylla-
bles) and intrusive schwas between cluster elements were
recorded by simple counts of occurrence. Statistical Analyses
PCC-R is a revised metric of PCC in which typical and Simple bivariate followed by hierarchical multi-
atypical distortions are removed from the index (Shriberg, variate, discriminant function analysis (DFA; McKean &
Austin, et al., 1997). PCC-R was used as a general index of Hettmansperger, 1976) was used to determine whether one
speech sound disorder severity, regardless of specific diag- or more of the 24 quantitative measures could reliably predict
nosis, for all three groups. This metric was used in this study the expert assignment of children to CAS or non-CAS
because it was more sensitive for the participant sample with groups. Therefore, the results from the expert judgment of
a diverse range of age and speech status (Shriberg, Austin, CAS presence or absence and the quantitative measures were
et al., 1997). Distortions were counted as their own measure compared in this phase to address the primary research aim.
because it may facilitate differential diagnosis of speech The sample size was sufficient to minimize Type I
disorders. Thus, using PCC-R allowed us to identify any and Type II errors (Serlin & Harwell, 2004). Before comple-
potential effects related to general severity rather than a spe- tion of the analysis, data were screened for normality, line-
cific diagnosis of CAS. Accordingly, distortions were not arity, and homoscedasticity. No violations of assumptions
part of the PCC-R measure, yet were measured separately were noted. Mahalanobis Distance was calculated to check
under the Occurrence of Consonant and Vowel Distortions for outliers (Allen & Bennett, 2008) and did not exceed the
Including Distorted Substitutions measurement according critical c2 of 26.13 (df = 8; a = .001) for any cases. Finally,
to Shriberg, Potter, and Strand (2009). screening for multicollinearity above a conservative 0.80
The Oral and Motor Speech Protocol and connected (cf. 0.90; Allen & Bennett, 2008; Poulsen & French, 2004;
speech sample were transcribed or scored from a video re- Tabachnick & Fidell, 2007) identified the following highly
cording. Articulation rate was calculated by counting sylla- correlated measures: PVC, PCC-R, and PPC; PVC and per-
bles per second produced in 1 min of monologue connected centage stress matches; PPC and oral function score; PCC-R
speech. This was undertaken using Adobe Audition software and oral function score, percentage stress matches and
(Version 1.5), in which the sample was isolated and vowels presence of articulatory false starts, restarts and/or within-
identified and counted to determine syllable rate following speech groping; and finally, /pə/, /tə/, and /kə/ rates. When
Logan, Byrd, Mazzocchi, and Gillam (2011). two measures showed multicollinearity, only one measure
Strand’s criterion of increased difficulty with polysyl- at a time was entered into the DFA as a predictor. There is
labic words (Shriberg, Potter, et al., 2009) was determined no consensus on the minimum ratio of predictor variables
using a magnitude-of-change score. This score was gen- to cases in DFA. Given this, we followed guidelines by
erated for each participant, comparing the mean number of Poulsen and French (2004) and Tabachnick and Fidell

Murray et al.: Differential Diagnosis of CAS 49


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(2007) and used up to eight predictors at a time. Bivariate a relatively straightforward manner with visual inspection
DFA (for each measure predicting the given diagnosis) was of the palate before differential diagnosis of CAS. The un-
initially completed to determine the top eight theoretical standardized (B) and standardized (b) regression coefficients
measures in discriminating CAS in this sample, taking into are reported for the two hierarchical DFAs in Table 5. The
account multicollinearity, for entry into the multivariate standardized b coefficient indicates the contribution of each
DFA. Results for all 24 measures are presented in Table 3 measure in predicting the initial diagnosis. A high score on
for all participants and Table 4 when CAS+ and sub- a positive coefficient and a low score on a negative coeffi-
mucous clefts were excluded. When a potential predictor cient predict CAS.
correlated highly with another potential predictor, the DFA For Model 1 (full data set), the hierarchical DFA
was run twice, each time with only one of the highly corre- showed that together the percentage of stress matches and
lated predictors. For example, the measures that were ini- occurrence of syllable segregation, both from the Single-
tially entered as predictors into the first multivariate DFA Word Test of Polysyllables, accounted for a significant 82%
were (a) presence of articulatory false starts, restarts, and/or of the variance in CAS diagnosis in this sample, R2 = .82,
within-speech groping; (b) syllable segregation; (c) DEAP adjusted R2 = .81, F(2, 45) = 96.26, p ≤ .001. Additional
(Inconsistency subtest); (d) PPC; (e) DDK accuracy; (f) non- measures increased the overall predictive accuracy by only
speech groping; (g) intrusive schwa; and (h) /kə/ rate. The 1%–2%, at most.
predictor percentage lexical stress matches correlated highly For Model 2 (limited data set), results indicated that
(i.e., was multicollinear with) the variable presence of artic- four measures in conjunction accounted for 91% of the CAS
ulatory false starts. Similarly, PVC was multicollinear with diagnosis, R2 = .91, adjusted R2 = .90, F(4, 38) = 87.45,
the variable PPC and /pətəkə/ rate was multicollinear with p ≤ .001. These measures were syllable segregation, percent-
the variable /kə/ rate. Analyses were rerun, substituting per- age of stress matches, PPC on the Single-Word Test of
centage stress matches, PVC or PCC-R, and /pətəkə/ for Polysyllables, and accuracy on DDK tasks (/pətəkə/) from
multicollinear variables. As measures were identified as not the Oral and Speech Motor Control Protocol. A formula
making a significant contribution to the hierarchical DFA, can be derived from Model 2 for predicting an individual’s
they were replaced by other measures of potential clinical group membership. This involves multiplying each individ-
importance (r < .5) to see whether they increased the predic- ual’s raw scores on the four measures in the DFA with their
tive accuracy of the model. For ease of reading, DFA anal- standardized b coefficient (i.e., a weighting), then summing
yses are presented in the more familiar layout of multiple the products and the model constant, and rounding the
regression, rather than as traditional discriminant functions. resulting single value to zero decimal places. A result of 1
indicates likely CAS, a score of 0 indicates likely non-CAS:
Results Diagnosis ¼ ðConstant þ ð% lexical stress matches  its weightÞ
Table 4 reports the descriptive statistics for each of þ ðsyllable segregation score  its weightÞþ ðPPC
the 24 variables measured across the three groups: CAS  its weightÞþð=pətəkə=DDK accuracyits weightÞ
(± expressive language disorder), CAS+ (CAS with comorbid
dysarthria and language impairment), and non-CAS (sub- Roundð0ÞÞ
mucous cleft, phonological disorder, and dysarthria). First, All participants used to create the model were correctly
the effects of severity and age were determined. In terms diagnosed using the formula (100% sensitivity, or true positive
of PCC-R, an index of general severity of speech sound dis- rate, and 100% specificity, or true negative rate). The model
order, the groups were significantly different, F(2, 45) = had high positive and zero negative likelihood ratios and a
7.865, p = .001. A Scheffé post-hoc analysis adjusted for diagnostic odds ratio of 1,425; see Appendix B). The strength
multiple comparisons revealed that the CAS+ group had of Model 2 was further tested with data from the four CAS+
significantly lower PCC-R scores than the CAS, p = .042, children and three children with submucous cleft who had
and non-CAS, p = .02, groups, but importantly, the latter been excluded from the DFA. Sensitivity remained high
two were not differentiated. This is consistent with the at 97%, with one CAS+ participant being misdiagnosed as
CAS+ group having more complex speech disorders. There non-CAS; specificity remained at 100%, with all submucous
was no effect of age between groups, F(2, 45) = 0.853, cleft participants accurately classified as non-CAS. Again,
p = .433, and age was not correlated above .5 with any of the positive likelihood ratio was high (16), and there was a
the 24 measures. zero negative ratio, with a diagnostic odds ratio of 651 (see
Two models are provided that represent the set of Appendix B). Table 6 demonstrates use of this formula for
variables with highest predictive accuracy for (a) all partici- four participants in this sample, two with CAS and two with-
pants with CAS (n = 32) against all non-CAS (n = 15) and out CAS. The formula can be used in Microsoft Excel to
(b) participants with CAS (CAS+ cases removed, n = 28) calculate automatically from evaluation test score input.
against non-CAS (cases with submucous cleft removed,
n = 12). Model 2 removed cases with CAS+ (where partici-
pants shared features across both groups and confounded Discussion
the analysis) and participants in the non-CAS group with The purpose of the current study was to determine
submucous cleft palates, which could be discriminated in whether expert diagnosis of CAS in a sample of 47 children

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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.

Accuracy in diagnosing
Measure Bivariate DFA p CAS /CAS+ vs. non-CAS

Syllable segregation 0.890 <.001 79%


Percentage of stress matches (lexical stress) on polysyllable test −0.882 <.001 77%
Presence of false articulatory starts, restarts, or within-speech groping 0.764 <.001 58%
DEAP (Inconsistency subtest) 0.551 <.001 30%
PVC on the polysyllable test −0.546 <.001 30%
/pǝtǝkǝ/ accuracy −0.535 <.001 29%
Nonspeech groping movements 0.536 .002 29%
PPC on the polysyllable test −0.515 <.001 27%
Oral structure score 0.492 .004 24%
Intrusive schwa 0.477 <.001 23%
/kǝ/ rate −0.445 .002 17%
Articulation rate (syllables per second) in connected speech −0.428 .006 17%
Oral function score −0.386 .007 15%
PCC-R on the polysyllable test −0.389 .007 15%
/pǝtǝkǝ/ rate −0.356 .014 13%
/tǝ/ rate −0.352 .024 12%
/pætikeɪk / rate −0.321 .028 10%
Vowel or consonant distortions 0.067 .669 Not significant
Maximum phonation time /a / (MPT) −0.261 .091 Not significant
CELF receptive language score 0.270 .080 Not significant
CELF expressive language score 0.142 .365 Not significant
/pǝ/ rate −0.291 .058 Not significant
Voicing errors 0.231 .136 Not significant
Increased difficulty with polysyllable words (change score) 0.205 .196 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 correct–revised.

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.

Accuracy in diagnosing
Measure Bivariate DFA p CAS vs. non-CAS

Percentage of stress matches (lexical stress) on polysyllable test −0.893 <.001 80%
Syllable segregation 0.873 <.001 76%
Presence of false articulatory starts, restarts, or within-speech groping 0.732 <.001 54%
DEAP (Inconsistency subtest) 0.670 <.001 45%
PVC on the polysyllable test −0.615 <.001 38%
/pǝtǝkǝ/ accuracy −0.606 <.001 37%
Nonspeech groping movements 0.507 .002 26%
PPC on the polysyllable test −0.540 <.001 29%
Oral function score −0.467 .002 22%
Vowel or consonant distortions 0.464 .003 22%
Intrusive schwa 0.449 .004 20%
PCC-R on the polysyllable test −0.429 .007 18%
Articulation rate (syllables per second) in connected speech −0.429 .006 18%
/kǝ/ rate −0.388 .013 15%
CELF receptive language score 0.362 .022 13%
/tǝ/ rate −0.326 .040 12%
Oral structure score 0.280 .081 Not significant
/pǝtǝkǝ/ rate −0.258 .109 Not significant
/pætikeɪk / rate −0.214 .185 Not significant
Maximum phonation time /a / (MPT) −0.166 .306 Not significant
CELF expressive language score 0.215 .183 Not significant
/pǝ/ rate −0.269 .093 Not significant
Voicing errors 0.237 .141 Not significant
Increased difficulty with polysyllable words (change score) −0.208 .199 Not significant

Murray et al.: Differential Diagnosis of CAS 51


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Table 5. Unstandardized (B) and standardized (β) regression coefficients for each predictive measure in a regression model predicting CAS
diagnosis.

Accuracy in diagnosing
Participants included Variable/measure B βa p CAS in this sample

Model 1 (Constant) 0.707 <.001 82%


CAS and CAS+ group (n = 32) vs. Percentage of stress matches on –0.009 –0.575 <.001
non-CAS (n = 15) polysyllable test
Syllable segregation 0.012 0.382 .001
Model 2 (Constant) 0.504 <.001 91%
CAS (n = 28) vs. non-CAS (n = 12) Percentage of stress matches on –0.011 –0.729 <.001
(CAS+ and submucous cleft polysyllable test
omitted) Syllable segregation 0.012 0.383 <.001
PPC on polysyllable test 0.007 0.300 .001
Accuracy on DDK task /pǝtǝkǝ/ –0.003 –0.145 .039
a
The β 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 mea- cleft, phonological impairment, or dysarthria (non-CAS).
sures often collected as part of standard clinical practice. Following the diagnosis, 24 quantitative measures were ex-
The current standard for diagnosing CAS utilizes experts tracted from clinically based assessments by raters blinded to
making judgments of presence or absence of a small set expert diagnosis. A series of multivariate DFAs revealed that
of speech behaviors, with no operational definitions or stan- a combination of the two quantitative measures of percent-
dardized testing protocol for eliciting the behaviors. In age of stress matches and occurrence of syllable segregation
the current sample, 28 children were judged by this method showed 82% diagnostic accuracy with expert diagnosis of
to have CAS (± expressive language disorder; ASHA, CAS and CAS+ versus non-CAS. However, the highest
2007b; Shriberg et al., 2012), four were judged to have CAS+ accuracy (91%) was obtained when four CAS+ children and
(CAS plus dysarthria and receptive and/or expressive language three non-CAS children with structural impairment (i.e.,
impairment), and 15 were judged to have either submucous submucous cleft) were excluded from analyses.

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

Percentage
of lexical
stress Syllable /pǝtǝkǝ/ Formula as entered
Participant characteristic matches segregation PPC accuracy into Excel Resulta

Weighting (β values) −0.011 0.012 0.007 −0.003


Participant No. 2216: CAS, Raw score 6 45 46 77 = round(0.504 + (6 × −0.011) 1
4 years, female, severe + (45 × 0.012) + (46 × 0.007)
articulation severity + (77 × −0.003), 0)
Participant No. 1217: non-CAS Raw score 33 20 25 66 = round(0.504 + (33 × −0.011) 0
(ataxic dysarthria), 4 years, + (2 × 0.012) + (25 × 0.007)
female, severe articulation + (66 × −0.003), 0)
severity
Participant No. 2219: CAS, Raw score 26 19 79 72 = round(0.504 + (26 × −0.011) 1
12 years, male, mild + (19 × 0.012) + (79 × 0.007)
articulation severity + (72 × −0.003), 0)
Participant No. 1311: non-CAS Raw score 88 0 97 100 = round(0.504 + (88 × −0.011) 0
(residual phonological + (0 × 0.012) + (97 × 0.007)
disorder), 12 years, male, + (100 × −0.003), 0)
mild articulation severity

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. 1311’s 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.

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The procedures used here for expert judgment on two assessment tasks: a polysyllabic word picture-naming
presence of CAS resulted in 32% of this sample being diag- task (Gozzard et al., 2004) and an oral mechanism exam-
nosed as non-CAS, from a group of children referred with ination (Robbins & Klee, 1987), taking less than 30 min to
“suspected CAS” according to Australian community-based complete.
SLPs. This is consistent with American studies (Davis et al., Two of the four informative measures—syllable seg-
1998; Forrest, 2003; Shriberg et al., 2011) in which children regation and percentage of lexical stress matches—measure
with CAS seemed to be over- rather than underdiagnosed. prosodic features now considered core symptoms of CAS.
However, we did not determine if CAS was underdiagnosed These perceptual features are strongly related to acoustic
in a general SSD population here. This finding is not sur- measures identified as potential markers for CAS, including
prising, given the lack of validated and/or published assess- the lexical stress ratio (Shriberg, Campbell, et al., 2003), the
ment batteries currently available for this population (ASHA, coefficient of variation ratio (Shriberg, Green, et al., 2003),
2007b). and the pairwise variability index (Ballard et al., 2010). Our
Of concern, there was a subset of children who pre- results support dysprosody as a potential core feature of
sented with frank structural and neurological deficits that CAS in children with some ability to produce polysyllable
were not identified prior to the community clinician assign- combinations. Prosody was a discriminant measure in both
ing the diagnosis of suspected CAS. An OMA is a practical, DFAs regardless of whether participants with neurological,
inexpensive screen to check for any overt structural deficits linguistic, or structural impairments were included.
or functional impairments related to muscle strength and The Strand 10-point checklist (Shriberg, Potter, et al.,
tone. If such deficits are found, referral to a neurologist or 2009) includes both “syllable segregation” and “lexical
head and neck team should follow. This may seem obvious, stress errors” as features. PPC was not used to measure any
yet there were participants as old as 12 years in this sample criterion from this feature list. Instead, six individual seg-
whose structural and/or dysarthric symptoms had not been mental error features were noted (out of the 10 features; see
previously detected. This is relevant for those with a CAS Table 1), but they were not individually discriminative. A
diagnosis as well because such diagnoses can co-occur. Fur- more global measure such as PPC could have teased apart
thermore, only 52% (22 out of 42) of the studies reviewed differences between groups, particularly because the non-
by McCauley et al. (2012) included OMAs. OMAs should CAS group was diverse. Accuracy on the /pətəkə/ DDK
be part of standard pediatric practice, reflected in policy task was not used to measure any criterion from this check-
guidelines and clinical pathways. In this sample, those with list. However, Thoonen et al. (1999) adds support for this
dysarthria exhibited low oral structure scores on our oral finding. They found trisyllabic repetition of /pataka/ was
structure and function test (Robbins & Klee, 1987) due to discriminative for CAS against controls. However, they
lack of tongue symmetry, the presence of tongue fascicula- found it did not discriminate CAS from dysarthria, which
tions, and tongue atrophy. In the submucous cleft cases in was achieved using fricative maximum phonation. This
the non-CAS group, all three had low oral structure scores measure was not calculated here but could be added to an
due to poor palatal juncture and one had a bifid uvula. OMA as an assessment measure in future studies. Impor-
Given that the primary aim of this study was to iden- tantly, Strand’s checklist (Shriberg, Potter et al., 2009) does
tify a set of measures for identifying children with CAS in not require children to exhibit these features to be diag-
a group with no other known diagnoses (e.g., hearing im- nosed with CAS: Children need to show evidence of any
pairment, structural abnormality, or other diagnosed de- four of the 10 features on the list, with no differential
velopmental diagnoses), the final analysis excluded those weighting of any feature. To our knowledge, the sensitivity
with comorbid CAS and submucous clefts. This exclusion and specificity of Strand’s checklist has not been tested, and
allowed us to determine how performance on a specific set it may not be sufficiently specific, with potential risk of di-
of measures, taken from existing clinical tests, interacted agnosing negative cases with CAS. The present results show
with each other to differentiate children with CAS from that two participants with clear dysarthria and one with a
children with other speech sound disorders. This differentia- submucous cleft were diagnosed with CAS using the Strand
tion is likely to be the difficulty SLPs face in identifying checklist.
true cases of CAS in order to provide effective manage- The inclusion of lexical stress errors in the ASHA list
ment. Importantly, age and severity of speech sound dis- of strong discriminative behaviors of CAS was supported
order (PCC-R) of these 40 children did not explain the by our analyses, but the other two features (inconsistency
variance in the CAS and non-CAS groups. Discriminant and lengthened and disrupted coarticulatory transitions)
function analysis revealed that four measures in combina- were not supported. The second feature, “lengthened and
tion had 91% predictive accuracy: (a) syllable segregation, disrupted coarticulatory transitions between sounds and
(b) percentage of lexical stress matches, (c) PPC, and (d) ac- syllables,” was measured as a binary score if false starts, re-
curacy on repetition of /pətəkə/ in the DDK task. The starts, hesitations, or within-speech groping were demon-
four measures are considered together in a hierarchical for- strated by a participant in connected speech and on the
mat to make a diagnosis; individual cutoff scores for each polysyllable test. In the bivariate DFA, this measure had
measure, although clinically appealing, do not capture 54% accuracy in diagnosis when used independently but did
the relationships between the variables and therefore the not add any additional information to the Model 2 (limited
diagnosis. These four measures were derived from just data set) analysis. This diagnostic information may have

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been captured in the DFA by syllable segregation, which had low diagnostic accuracy independently and did not add
is associated with disrupted coarticulatory transitions as to the diagnostic models. The current data support the find-
well as dysprosody. Additionally, the motorically complex ings of Strand and McCauley (2008), as well as studies of
DDK task of repetitions of /pətəkə/ also examined disrupted acquired AOS and oral apraxia (see Ballard et al., 2000, for
transitions and within-speech groping as part of its score. a review), in that nonspeech features are indicative of oral
Descriptions of such coarticulation, sequencing, and grop- apraxia rather than CAS. Therefore nonspeech difficulties
ing errors are common in CAS (e.g., ASHA, 2007b; Grigos are best considered as concomitant rather than core features
& Kolenda, 2010; Thoonen et al., 1996, 1999). These results of CAS (ASHA, 2007b; Strand et al., 2013).
suggest that some behaviors on perceptually based feature The findings from this study indicate that four mea-
checklists are not independent. sures, in combination, diagnosed CAS with high accuracy
The third ASHA criterion, “inconsistent errors on against the current gold standard of expert opinion. The
consonants and vowels in repeated productions of words final model (Model 2) did not include children with CAS+
and syllables” (ASHA, 2007b, p. 4), was most notably ab- or with the frank oral structural impairment of submucous
sent from the models of discriminative measures. Forrest cleft, and data from these seven children were used to fur-
(2003) found that inconsistency was the feature most used ther test the robustness of the model (see Appendix B). This
by clinicians to identify CAS. The present study measured shows that although Model 2 was not being developed with
inconsistency with the DEAP Inconsistency subtest, as it those participants with CAS+dysarthria, it can be used to
most closely matched the definition of inconsistency used determine presence of CAS contributions to their speech
in the ASHA Technical Report (2007b)—that is, token-to- impairment. Only one participant with CAS+ (Participant
token inconsistency across separate productions of the same No. 1328) was misdiagnosed by the model’s formula as
words. This has also been referred to as target stability non-CAS. This diagnosis was most likely due to his comor-
(Marquardt, Jacks, & Davis, 2004). When used as a sole bid flaccid dysarthria and a lack of syllable segregation,
predictor in a bivariate DFA, inconsistency discriminated considering he was unable to produce any polysyllabic words.
the groups with 30% accuracy and did not contribute signif- Three important points are demonstrated here. First, repli-
icantly to predicting diagnosis in the multivariate analyses. cation of this study is required with an unselected sample
Other available measures that capture a different type of that includes more children with dysarthria. Second, an OMA
inconsistency might yield a different result. In Model 2 would be required before using these criteria in the future
(limited data set), inconsistency may have been captured by to determine frank dysarthria and/or submucous clefts in
the repeated productions of the /pətəkə/ DDK task, which those who may also have CAS. Third, children with mini-
would be more similar to token accuracy whereby greater mal verbal output may not be diagnosed accurately using the
accuracy demonstrates less inconsistency (Marquardt et al., measures described here because they are based on attempts
2004). Alternatively, the Inconsistency Severity Percentage at polysyllabic words. In future, syllable segregation could
(Iuzzini & Forrest, 2010), for example, is a measure of pho- also be examined in utterances between words rather than
nemic and phonetic inconsistency, similar to total token just within polysyllable words using, for example, Shriberg’s
variability (Marquardt et al., 2004), from multiple elicita- inappropriate pauses measure (Shriberg, 2013).
tions of all consonants in English across varied syllable Data from four participants are included as case stud-
or word contexts. Iuzzini (2012) reported that the Inconsis- ies in Table 6 to illustrate the use of the criteria and further
tency Severity Percentage differentiated CAS from phono- explain the weightings and use of the DFA results. The
logical disorder better than the DEAP Inconsistency measure. first two cases are 4-year-old girls, each with severe artic-
Further investigation of the construct of inconsistency and ulation difficulties, one of whom was diagnosed with CAS
factors influencing its nature and degree would help to de- and the other with ataxic dysarthria by the first and second
termine the most informative measure and whether that authors. These cases illustrate the importance all four cri-
might have greater predictive accuracy when combined teria and weightings of the measures: Each girl had poor
with measures of other features associated with CAS. Given lexical stress matches and a high occurrence of syllable seg-
the debate over inconsistency in the acquired AOS litera- regation, both of which are features of CAS and ataxic dys-
ture (see Staiger, Finger-Berg, Aichert, & Ziegler, 2012, for arthria (ASHA, 2007b; Duffy, 2012). However, although
a review) and the absence of this feature in Strand’s 10-point the child with CAS had poor (46%) accuracy for the PPC
checklist, further research is required on this feature, one measure, this child was relatively better than the participant
that is strongly represented in clinicians’ impressions of both with ataxic dysarthria (with imprecise articulation), who
CAS and AOS. had 25% accuracy. This may have helped discriminate these
Nonspeech groping (i.e., groping during nonspeech cases, because the child with ataxic dysarthria exhibited
tasks) was another noteworthy measure. Such nonspeech many more distortions and articulation errors, despite the
oromotor tasks have been used in CAS checklists over time, first two measures showing a similar pattern. The second
presumably as a feature of CAS (e.g., Ozanne, 2005; Royal pair of cases illustrates the contrast between two 12-year-old
College of Speech and Language Therapists, 2011), and non- boys with mild articulation difficulties, one with CAS and
speech groping was the second most prevalent feature in the other with residual phonological impairment. The boy
diagnosis in a survey of clinicians (Forrest, 2003). Despite with CAS had fewer lexical stress matches (26%, compared
this, the current study results suggest that nonspeech groping to 88% for the child with phonological impairment), more

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instances of syllable segregation, and poorer /pətəkə/ accu- prosodic errors being features on both the ASHA consensus-
racy than the boy with phonological impairment. These based (2007b) and Strand 10-point feature lists (Shriberg,
three features are hypothesized to reflect an underlying im- Potter, & Strand, 2009). However, other samples that dem-
pairment of motor planning and programming or praxis onstrate dysprosody may only account for a subset of chil-
(e.g., ASHA, 2007b; Shriberg, Green, et al., 2003; Thoonen dren with CAS (e.g., Shriberg, Aram, & Kwiatkowski, 1997)
et al., 1999). The criteria provide confidence in discrimi- or younger children may demonstrate other errors of greater
nating those with some underlying praxis deficits, demon- prevalence, such as inconsistency (Iuzzini & Forrest, 2010).
strating CAS is at least part of the child’s presentation. The Model 2 DFA formula based on the limited data set is
Therefore, the combination of the four discriminant measures reported here to facilitate replication, but caution must be
and their weightings appear crucial in separating verbal used in applying it to clinical populations or cases that differ
participants with different types of expressive communica- from the studied sample.
tion disorder and wide ranges of age and severity. In this study, we relied on perceptual measures of
It is important to consider that the assessment proce- speech behaviors. This was a deliberate decision considering
dures used here assessed children at a single time point. that perceptual measures are most commonly used in clini-
Any developmental disorder can vary in its surface presen- cal practice (Duffy, 2012) and have been shown to correlate
tation across time. Regular review is therefore indicated to highly with acoustic measures of lexical stress (e.g., pair-
ensure early detection of other clinical behaviors that might wise variability indices; Ballard et al., 2010). However, the
emerge with therapy and maturation (e.g., literacy concerns) relatively subjective nature of these measures could result
and ultimately to ensure that management strategies match in discrepancies and therefore errors in the formula’s result,
presenting and prognostic features (Stackhouse & Snowling, particularly in untrained SLPs. In the current study, train-
1992; Zaretsky, Velleman, & Curro, 2010). ing of raters was required to maintain interrater reliability
Our findings advance the field in this area by identify- above 85%. To assist replication in future studies, efforts to
ing two tests that appear central to differentiating CAS make the measures more objective would be warranted by
from other disorders, at least in this sample: namely, a com- providing defined features, measures, and scoring criteria in
plete OMA including a DDK task and a sufficiently large a manual or by finding more objective kinematic or acous-
sample of polysyllabic single-word production (e.g., Gozzard tic measures. There are promising acoustic measures (e.g.,
et al., 2004). Both the real-word polysyllabic test and the Ballard et al., 2012; McKechnie et al., 2008; Shriberg, Green,
nonword /pətəkə/ DDK task from the OMA were motori- et al., 2003) and potential for automation (Hosom, Shriberg,
cally challenging and appear to successfully elicit behaviors & Green, 2004; Rosen et al., 2010). However, manual
that reflected the underlying motor planning and program- acoustic measures can be time-intensive and also require a pe-
ming deficits in CAS at both the segmental and supra- riod of training. It may be some time before such acoustic
segmental level (Shriberg, Lohmeier, et al., 2009; Thoonen measures are available in a format that allows rapid auto-
et al., 1996, 1999). These two tests alone may be sufficient mated analysis in clinical settings.
for reliable diagnosis of CAS in verbal children. However, Studies aiming to identify quantitative measures that
SLPs using these tasks still need to consider normal acquisi- differentiate disorders suffer an inherent circularity (ASHA,
tion and development in terms of both segmental and prosody 2007b; Guyette & Diedrich, 1981). However, here the aim
accuracy (Ballard, Djaja, Arciuli, James, & van Doorn, was not to validate the original checklists (ASHA, 2007b;
2012; James, 2006; Shriberg, Lohmeier, et al., 2009). 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
Limitations in combination) could reproduce the initial expert diagnosis
The major limitation of this study is that it utilized with high accuracy. Our findings suggest that a set of just
a selected clinical sample of children with suspected CAS four measures strongly predicts presence of CAS in verbal
and selection criteria designed to find idiopathic CAS children. This is a more parsimonious solution than a
rather than comorbid CAS. This approach is not uncom- checklist of multiple features, of which only a subset is re-
mon (e.g., McCauley et al., 2012; Rosenbek & Wertz, 1972; quired for diagnosis (e.g., Hickman, 1997; Kaufman, 1995;
Yoss & Darley, 1974) and was warranted in exploring and Shriberg, Potter, & Strand, 2009).
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 Further Directions
selected for analysis, and there are inherent risks that the This preliminary study used a sample of children ages
findings might not generalize to a similar but larger sample 4–12 years with idiopathic and comorbid CAS and com-
or to a different group of children (e.g., children with co- pared them to others who had been suspected to have CAS
morbid intellectual disability and receptive language im- but were instead diagnosed with phonological or language
pairment). For example, the formula used PPC to separate impairment, submucous cleft, or dysarthria. Further re-
CAS from ataxic dysarthria. However, there is no reason search is necessary to test the robustness of the sets of cri-
that a child with CAS may not have severe articulation teria and the resultant formula identified when diagnosing
errors. Also, the participant sample here all demonstrated an unselected community sample of children with suspected

Murray et al.: Differential Diagnosis of CAS 55


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Ballard; and an Australian Research Council Future Fellowship Diagnostic evaluation of articulation and phonology (DEAP).
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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).

ASHA Strand
Group Participant no. criteria meta criteria metb Diagnosis

Non-CAS 1217 2/3 5/10 Ataxic dysarthria (+ receptive & expressive language disorder)
1314 2/3 6/10 Flaccid dysarthria (CNXII) (+ receptive & expressive language
disorder)
0236 0/3 1/10 Phonological disorder (+ expressive language disorder)
1313 1/3 1/10 Phonological disorder
0235 0/3 0/10 Phonological disorder (+ receptive & expressive language disorder);
hoarse voice
0333 0/3 3/10 Phonological disorder (+ receptive & expressive language disorder);
suspected global developmental delay
0334 0/3 1/10 Phonological disorder (+ receptive & expressive language disorder)
0237 0/3 2/10 Phonological disorder (+ receptive & expressive language disorder)
1311 0/3 2/10 Phonological disorder
2315 0/3 1/10 Phonological disorder
1312 0/3 2/10 Comorbid phonological disorder and stuttering
0138 1/3 6/10 Submucous cleft palate (+ expressive language disorder)
0139 1/3 2/10 Submucous cleft palate
1310 1/3 2/10 Submucous cleft palate
0335 0/3 1/10 Phonological disorder (+ expressive language disorder)
No. of non-CAS participants 0/15 3/15
that met criteria
CAS+ 0332 3/3 7/10 CAS and ataxic dysarthria (+ receptive & expressive language
disorder)
2105 3/3 8/10 CAS and flaccid dysarthria (CNXII) (+ expressive language disorder)
1214 3/3 9/10 CAS and flaccid dysarthria (CNXII) (+ expressive language disorder)
1328 3/3 8/10 CAS, flaccid dysarthria (CNXII) (+ receptive & expressive language
disorder)
No. of CAS+ participants 3/3 8/10
that met criteria
CAS 1103 3/3 8/10 CAS (+ expressive language disorder)
1101 3/3 7/10 CAS
1102 3/3 8/10 CAS (+ expressive language disorder)
1104 3/3 6/10 CAS
2106 3/3 9/10 CAS (+ expressive language disorder)
2107 3/3 7/10 CAS (+ expressive language disorder)
2108 3/3 8/10 CAS (+ expressive language disorder)
1210 3/3 8/10 CAS
1211 3/3 9/10 CAS (+ expressive language disorder)
2212 3/3 8/10 CAS (+ expressive language disorder)
1213 3/3 7/10 CAS (+ expressive language disorder)
1215 3/3 6/10 CAS
2216 3/3 7/10 CAS
2217 3/3 7/10 CAS
2218 3/3 7/10 CAS
2219 3/3 5/10 CAS
2320 3/3 7/10 CAS (+ expressive language disorder)
1321 3/3 5/10 CAS
2322 3/3 8/10 CAS (+ expressive language disorder)
2323 3/3 5/10 CAS
1324 3/3 5/10 CAS
1325 3/3 7/10 CAS (+ expressive language disorder)
2326 3/3 6/10 CAS
1329 3/3 6/10 CAS
1330 3/3 7/10 CAS
0331 3/3 7/10 CAS (+ expressive language disorder)
1316 3/3 5/10 CAS (+ expressive language disorder)
2109 3/3 6/10 CAS
No. of CAS participants 28/28 28/28
that met criteria
a
ASHA criteria = 3/3 needed for CAS diagnosis. bStrand criteria = 4/10 needed for CAS diagnosis over three tasks.

Murray et al.: Differential Diagnosis of CAS 59


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Appendix B
Sensitivity and specificity based on the Model 2 DFA.

Model 2 participants Expert diagnosis

CAS (n = 28) (excluding CAS+) Non-CAS (n = 12) (excluding submucous clefts)


Predicted diagnosis (from DFA) True positive = 100% (n = 28) False positive = 0% (n = 0)
False negative = 0% (n = 0) True negative = 100% (n = 12)

Note. Positive likelihood ratio = infinity; negative likelihood ratio = 0.

All participants Expert diagnosis

CAS (n = 32) (including CAS+) Non-CAS (n = 15) (including submucous clefts)


Predicted diagnosis (from DFA) True positive = 97% (n = 31) False positive = 0% (n = 0)
False negative = 3% (n = 1) True negative = 100% (n = 15)

Note. Positive likelihood ratio = 16; negative likelihood ratio = 0.

60 Journal of Speech, Language, and Hearing Research • Vol. 58 • 43–60 • February 2015

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