Research Article

Folia Phoniatr Logop Received: July 13, 2018

Accepted: April 18, 2019
DOI: 10.1159/000500554 Published online: July 22, 2019

Looking to the Future: Speech, Language, and

Academic Outcomes in an Adolescent with
Childhood Apraxia of Speech
Samantha J. Turner a, b Adam P. Vogel c, d Bronwyn Parry-Fielder e Rhonda Campbell f
       

Ingrid E. Scheffer a, g, h Angela T. Morgan a, b

   

a Department
of Paediatrics, The University of Melbourne, The Royal Children’s Hospital, Parkville, VIC, Australia;
b
Speech and Language Group, Clinical Sciences Theme, Murdoch Children’s Research Institute, Parkville, VIC,
 

Australia; c Centre for Neuroscience of Speech, The University of Melbourne, Melbourne, VIC, Australia; d Redenlab,
   

Melbourne, VIC, Australia; e Department of Speech Pathology, The Royal Children’s Hospital, Parkville, VIC, Australia;
 

f Bellfield Speech Pathology, Melbourne, VIC, Australia; g Epilepsy Research Centre, Department of Medicine, The
   

University of Melbourne, Austin Health, Melbourne, VIC, Australia; h Florey Institute of Neuroscience and Mental
 

Health, Melbourne, VIC, Australia

Keywords and writing at 14 years. Nonword reading, reading compre-

Childhood apraxia of speech · Longitudinal · Adolescence ·
Motor speech · Speech intelligibility · Speech prosody ·
Literacy · Language · Academic outcomes
Abstract
Objective: The clinical course of childhood apraxia of speech
(CAS) is poorly understood. Of the few longitudinal studies
in the field, only one has examined adolescent outcomes in
speech, language, and literacy. This study is the first to report
long-term speech, language, and academic outcomes in an
adolescent, Liam, with CAS. Methods: Speech, language, lit-
eracy, and academic outcome data were collected, including
3 research-based assessments. Overall, data were available
at 17 time points from 3; 10 to 15 years. Results: Liam had
moderate-to-severe expressive language impairment and
poor reading, writing, and spelling up to 10 years. His nu-
meracy was at or above the national average from 8 to 14
years. He made gains in preadolescence, with average ex-
pressive language at 11 years and above average reading
hension, and spelling remained areas of weakness. Recep-
tive language impairment was evident at 13 years, which
was an unexpected finding. Conclusion: Findings from sin-
gle cases can be hypothesis generating but require verifica-
tion in larger cohorts. This case shows that at least some chil-
dren with CAS may gain ground in adolescence, relative to
same age peers, in expressive language and academic areas
such as reading and writing. © 2019 S. Karger AG, Basel
Introduction
Childhood apraxia of speech (CAS) is a pediatric mo-
tor speech disorder, in which the “precision and consis-
tency of movements underlying speech are impaired” due
to deficits in speech motor planning and programming
[1]. The clinical course of the disorder is still poorly un-
derstood, with only a handful of studies examining long-
term outcomes [2–9]. Most studies have understandably
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© 2019 S. Karger AG, Basel Dr. Samantha Turner

Postdoctoral Research Fellow, Speech and Language Group, Clinical Sciences Theme
University of South Florida

Murdoch Children’s Research Institute, Flemington Road

E-Mail karger@karger.com
Parkville, VIC 3052 (USA)
www.karger.com/fpl
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E-Mail samantha.turner @ mcri.edu.au

focused on speech outcomes and largely in primary
school age children. Understanding how the broader
phenotype associated with CAS may evolve into adoles-
cence is crucial to differential diagnosis of older individu-
als and will provide important prognostic information to
families.
Speech Changes over Time in CAS
Several studies demonstrate that speech improves with
age in CAS. Taking these findings together, 10 preschool-
ers with CAS (7 male, 3 female) who were unintelligible
at 4–6 years, had improved articulation of single words,
and were mostly intelligible in conversational speech by
school age (8–10 years) [4, 6]. This trend was also noted
in older children with CAS. Two school age children (Mi-
chael, 10;7 years; Caroline, 11;9 years) became more intel-
ligible and had better performance on a single word ar-
ticulation test (Edinburgh Articulation Test) at 14;6 and
15; 8 years, respectively [4, 6]. Improvements in speech
intelligibility were attributed to the development of word
specific articulatory programs in these older children.
While consonant and vowel accuracy generally improved
in 3 children with CAS (4;6, 5;6, and 5;10 years) over a
3-year period, their production of later developing sounds
(Late 8 – /ʃ, ʒ, l, r, s, z, θ, ð/) was poor up to school age
(6;5, 7;5, and 7;7 years) [5]. This was in the context of on-
going therapy from an early age focused on phoneme ac-
curacy and other speech production targets. Over time,
they demonstrated higher word token accuracy in con-
versational speech and decreased total word token vari-
ability (ratio of number of different variant productions
to total number of tokens produced), yet marked session-
to-session variability remained a core feature [3, 5].
Children with CAS appear to have persistent deficits
in syllable construction. Consonant omissions have been
frequently reported in preschool, and sound and syllable
deletions increasingly apparent in school age and adoles-
cence [4–7, 10]. Syllable sequencing deficits also persisted
in school age children with CAS [4]. Delayed and atypical
errors most commonly heard in older children included
liquid simplification, vowel and voicing errors, sound in-
trusion, and metathesis [2, 4–7]. Little improvement was
seen in nonword repetition, with impaired performance
into adolescence [4, 6].
Language and Literacy Changes over Time in CAS
Persistent expressive language impairment has been
reported to occur up to school age in children with CAS
[4, 7, 8]. One child (MV) showed little improvement in
expressive language over a 2 years period (5;6–7;6 years)
2 Folia Phoniatr Logop
DOI: 10.1159/000500554
with lexical diversity and expressive grammar significant-
ly below age expectations [8]. Another child (LH) had
persistent and severe expressive language impairment
from 6 to 11;4 years [7]. Deficits in expressive syntax and
receptive morphology were noted, with severe short-term
memory impairment impacting on sentence and number
repetition tasks [7]. In a larger cohort of 10 pre-schoolers
with CAS, all children tested had expressive language im-
pairment, with only 2 scoring in the average range at
school age (8–10 years) [4]. Of those with average recep-
tive language skills in preschool, 3 subsequently demon-
strated impaired receptive language on the school age as-
sessment. These impairments were seen despite improve-
ments in speech-sound production, associated with poor
nonverbal problem-solving skills and speed of informa-
tion processing. This downward trend was also noted
in LH, who had severely impaired receptive language at
11;4 years, having previously been in the average range
[7]. MV had average receptive language up to 7 years [8].
One study has examined literacy skills over time in
CAS [4, 7, 11]. Two children showed little improvement
in reading and spelling despite 4 years of intensive pho-
nics therapy, with severely impaired skills in adolescence
(Michael, 14;5 years; Caroline, 15 years). These children
had difficulty discriminating between words and non-
words, had poor letter-sound correspondence, and were
unable to use phonological reading and spelling strate-
gies. The majority of their spelling errors appeared to be
related to their residual speech errors, including sound
and syllable deletions and insertions. In contrast, LH
made progress in literacy after 5.5 years of weekly therapy
[7]. Her reading skills were within the average range at 11;
4 years, with reading fluency, accuracy, rate and compre-
hension standard scores falling within one SD of the
mean. She was able to spell sight words better than regu-
lar words and sentences (97 vs. 40% correct). Her spelling
errors also mirrored her speech production deficits, in-
cluding epenthesis, substitutions, and sequencing errors.
Deficits in reading comprehension and decoding of real
and nonsense words were evident in other school aged
children with CAS [4, 11].
Taken together, these studies demonstrate that chil-
dren with CAS can become more intelligible as they ma-
ture, yet have persistent sound and syllable-level errors
and poor repetition of nonwords. Expressive language
impairment may be evident up to school age and associ-
ated with deficits in reading and spelling. How language
and literacy evolve in adolescence, and how children with
CAS perform in other academic areas such as numeracy
and writing require further examination. This is critical
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information to guide speech pathology input as children
with CAS progress through high school, helping to max-
imize their performance in the final school years.
Here we address these gaps in the literature and de-
scribe long-term speech, language and academic out-
comes in an adolescent boy with CAS. “Liam” was chosen
for investigation as he had relatively pure CAS without
comorbid dysarthria, and no other cognitive or neuro-
logical comorbidities. Rich phenotypic data were avail-
able on Liam from over a decade, including the results of
research-based and clinical speech pathology assess-
ments, national academic assessment, and speech pathol-
ogy intervention. This was a rare opportunity to examine
longitudinal change in one child with CAS from pre-
school to adolescence.
Case History
Liam was induced at 41 weeks of gestation after an un-
eventful pregnancy. He weighed 4,175 g at birth and was
well in the neonatal period. There was no history of feed-
ing difficulties, and he breastfed until 12 months of age.
He met appropriate motor milestones, although he was
described by his mother as “clumsy” with some motor
tasks (writing, riding a bike). His fine and gross motor
skills have not been formally assessed. Liam’s speech de-
velopment was delayed. He did not babble as an infant,
said his first words around 3 years of age and put 2 words
together at 3;9 years. There was no history of regression.
His hearing was normal at 3;6 years. Motor speech plan-
ning difficulties were first noted at 3;10 years, including
groping during sound production and inconsistent use of
phonemes in words. At this age, the assessing speech pa-
thologist reported that Liam had “difficulty getting his
lips, tongue, and teeth in the right position at the right
time.” Frequent omission errors were also noted. Expres-
sive language impairment was first diagnosed at 3; 10
years, with his score in the 3rd percentile on the Clinical
Evaluation of Language Fundamentals (CELF) – Pre-
school. He was diagnosed with CAS at 4;4 years by the
third author (B.P.-F.); a speech pathologist with over 20
years of experience in CAS at the time of diagnosis. At that
time, Liam’s spontaneous speech was frequently unintel-
ligible, he had a reduced consonant inventory (no frica-
tives apart from glottal fricative /h/; no affricates), reduced
syllable shapes, sound substitutions and omissions (17%
consonants correct), distorted vowels, atypical phonolog-
ical patterns (e.g., initial consonant deletion), and incon-
sistent word attempts. He had increased errors in multi-
syllabic words and phrases. He was able to produce iso-
lated vowels and some consonants on the sequencing
Adolescent Outcomes in CAS
subtest of the Verbal Motor Production Assessment for
Children, however, had difficulty sequencing 2 or more
sounds. Groping was evident in nonspeech oral motor
tasks. Cognitive assessment at 5 years (Wechsler Pre-
school and Primary Scale of Intelligence, 3rd Edition) re-
vealed borderline verbal intelligence (IQ 74), with average
nonverbal intelligence (IQ 100) and processing speed (IQ
86). Liam was severely delayed in the Communication do-
main on the Vineland Adaptive Behaviour Scales (stan-
dard score 46). He had average verbal (IQ 94) and nonver-
bal (IQ 84) intelligence on subsequent testing at 7;1 years
(Kaufman Brief Intelligence Test, 2nd Edition). Difficul-
ties in basic attention and working memory were evident
at this time (Wechsler Intelligence Scale for Children, 4th
Edition Digit Span Forward score 6; Backward score 5).
Oral motor assessment at 7;5 years revealed mandibu-
lar retrognathia, reduced tongue strength against mild re-
sistance, and involuntary tongue movement at rest.
Groping was evident in nonspeech tasks. His scores on
3 subtests of the Verbal Motor Production Assessment
for Children were in the severe range (Global Oromotor
Control, 80%; Focal Oromotor Control, 67%; Sequenc-
ing, 52%).
Liam has had regular speech pathology intervention
from 3 years of age. He had fortnightly therapy in a com-
munity health setting for around 1 year and attended a
speech and language program in Prep. He commenced
private speech therapy at 6;7 years – details regarding the
frequency of session and goals of therapy are included in
Table 1. He received 4 years of language disorder funding
from Prep to Grade 3 through State Support Services,
which funded 1: 1 speech therapy sessions. He also had
regular integration aide support in the classroom.
Methods
Liam was initially referred to a research study of the genetics of
speech disorders, as he had CAS and a history of speech and lan-
guage disorder in his extended family. The study had approval
from The Royal Children’s Hospital Human Research Ethics
Committee (27053), and written informed consent was obtained
from Liam’s parents. He was assessed on 3 separate occasions
(7;4, 9;4, and 10;5 years) by the first author (S.J.T.) as part of his
participation in the research study. A battery of standardized age-
appropriate assessments was used to phenotype Liam’s speech,
language, phonological processing, and literacy skills as described
below. Audiovisual recordings of the assessments were made using
a Marantz PMD671 digital recorder, Countryman Isomax headset
microphone, and a Sony DCR-SR85 digital camera. Two speech
pathologists (S.J.T., A.T.M.) independently rated research-based
speech and oral motor assessments, then reached consensus on
discrepant ratings.
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Table 1. Speech therapy targets and frequency of intervention from 6 to 15 years

School Frequency Goals

grade
level speech language literacy

Prep Unknown Articulation Receptive and expressive language development

Phonology

Grade 1 31 × 30 min sessions /ʃ/, /tʃ/, /dʒ/ (traditional Subject-verb-object, complement, adverb (colourful
articulation therapy) semantics)
Fronting and cluster reduction Following instructions of increasing length
(minimal pairs therapy)

Grade 2 29 × 30 min sessions /θ/, /r/, /æ/, /ʌ/ Comparatives/superlatives Thrass

Past tense
Passive sentences
Following instructions of increasing length
Sentence recall
Absurdities

Grade 3 16 × 30 min sessions /r/, /l/ clusters Inferencing skills

Semantic relationships
Conjunctions
Auditory processing

Grade 4 9 × 30 min sessions /θ/ Accuracy of word retrieval

Increasing speech volume Word decoding
Spelling

Grade 5 19 × 30 min sessions /t/, /d/, /n/, /θ/, /l/, /r/ Sentence structure Word decoding
Moving across syllables Word classes Spelling
WH and conditional questions Rapid naming
Absurdities
True/false statements
Comparatives/superlatives
Semantics – odd one out

Grade 6 14 × 30 min sessions Moving across syllables Semantic relationships Word decoding
Noun, verb, prepositional and adjective phrases (CLIP) Reading comprehension
Conjunctions Rapid naming

Year 7 16 × 30 min sessions Conjunctions Reading comprehension

Complex sentences Spelling
Spoken and written grammar
Figurative language

Year 8 16 × 30 min sessions /d/, /z/, /dʒ/, /l/ clusters Copula – is/are Reading fluency
vowels /æ/, /i/, /ʌ/, /ɛ/ Has/have Reading comprehension
Intonation Pronouns/possessives
Sentence-level syntax (Rainbow sentences)

Year 9 4 × 30 min sessions Intonation and articulatory Pronouns/possessives Reading fluency

+ intensive group accuracy in sentences Present tense
therapy Rainbow sentences

Intensive therapy group to strengthen the development of oral

language for writing

Research Assessment Battery
Speech production in single words was examined using the
Goldman-Fristoe Test of Articulation, Second Edition (GFTA-2)
[12]. The GFTA-2 uses a series of colored pictures of objects and
actions to elicit all of the sounds of English.
Connected speech tasks included reading a short passage (The
Rainbow Passage), a picture description task (The Cookie Theft),
and a 10-minute conversation with the researcher. These tasks
were regarded as more indicative of Liam’s functional communi-
cation [13, 14].
Single word and connected speech tasks were analyzed for ar-
ticulation and phonological speech error patterns and features of
CAS. Phonological errors were classified with reference to the nor-
mative study by Dodd et al. [15, 16].
Consistency of speech production was examined using the in-
consistency assessment of the Diagnostic Evaluation of Articulation
and Phonology (DEAP) [16]. Liam was asked to name 25 pictures 3
times in one session, with other tasks administered in between.
Challenging speech tasks included repetition of multisyllabic real
words and nonwords. Australian recordings of the stimuli were pre-
sented, and Liam was instructed to repeat exactly what he had heard.
The Nonword Memory Test [17] consists of 28 nonwords. The total
number of words correctly repeated was compared to mean scores
(± SD) for the normative sample. The Multisyllabic Word Repetition
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Table 2. CAS speech features (categorised under the 3 ASHA consensus diagnostic criteria) present in Liam

Criteria Speech features associated with each criteria Feature present?

Inconsistent errors Same word/syllable different on repetitions

Lengthened and
disrupted coarticulatory
transitions between
sounds and/or syllables
Inappropriate prosody
Same C/V different across different words
Any one of these
Speech motor behaviours, including groping
Slowed DDK rates/disrupted DDK sequence
Difficulty sequencing phonemes and syllables
Difficulty achieving initial articulatory configurations/transitory movement gestures
Any 2 or more of these
Syllable segregation
Intrusive schwa
Epenthesis
Frequent omission errors
Addition errors
Prolongation errors
Repetitions of sounds and syllables
Voicing errors
Non-phonemic productions or distorted substitutions
Nasality and/or nasal emissions
Errors increase with word length and phonological complexity
Metathesis
Difficulty maintaining syllable integrity
Equal stress or lexical stress errors
Prolongation errors
Vowel errors
Vowel distortion
Altered suprasegmental characteristics

Shaded box indicates that the speech feature is present. CAS, childhood apraxia of speech; DDK, diadochokinetic.

task [18] consists of 52 words that are generally familiar to school age
children. Raw scores were calculated as the number of items cor-
rectly repeated, and compared to the mean scores (± SD) of indi-
viduals with a history of moderate-severe speech disorder and aged-
matched individuals with typical speech [19].
Receptive and expressive language was examined using the
CELF-4 [20]. The Receptive Language Index score measures per-
formance on tasks assessing comprehension of grammatical rules,
relationships between words, and the ability to follow oral com-
mands containing functional language. The Expressive Language
index score measures performance on tasks evaluating the ability
to recall and reproduce sentences of varying length and complexity,
to formulate complex sentences and to complete sentences using
grammatical rules. A standard score of 80 or below on the CELF-4
Receptive or Expressive Language Index indicated impairment, as
per recently proposed criteria, that is, language ability > 1.25 SD
below the population mean on standardized language tests [21].
Phonological processing skills including phonological aware-
ness (the ability to attend to, identify and manipulate sounds in
spoken words), phonological memory (use of the speech sound
system to store information in short-term memory), and rapid
naming (retrieval of sound-based information from long-term
memory) were examined using the Comprehensive Test of Pho-
nological Processing (CTOPP) [22]. Composite standard scores
were calculated with reference to normative data.
Literacy was assessed using the Wide Range Achievement Test,
4th Edition (WRAT-4) [23]. The Word Reading subtest examined
letter identification and recognition of single words, while the
Spelling subtest examined the ability to encode sounds into written
form through a dictated spelling format. Standard scores for each
subtest were calculated with reference to normative data.
Liam’s CAS diagnosis was confirmed by 2 speech pathologists
(S.J.T., A.T.M.). Diagnostic criteria were adapted from Murray et al.
[24] based on the 3 consensus features of CAS proposed by the Amer-
ican Speech-Language-Hearing Association; (1) inconsistent errors;
(2) lengthened and disrupted coarticulatory transitions between
sounds/syllables; and (3) inappropriate prosody [1] (Table 2).
At this time, all available clinical speech, language, and literacy
assessments were obtained from Liam’s past and current treating
speech pathologists. Original test forms from clinical assessments
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 Color version available online
10;9 years^
Literacy
Schonell

3;10 years^ 6;7 years^ 7;10 years^ 13;4 years^

Speech and language Speech Speech 10;5 years Language
Articulation Survey Quick screener Articulation survey Speech CELF-4
CELF-P DEAP/NWR/MSW

7;4 years 8;11 years^ 11;10 years^ 15;11 years^

5;3 years^
Speech and language Language Language and Language
Speech and language
GFTA CELF-4 literacy CELF-4
Quick screener
CELF-4 CELF-4
CELF-4
CTOPP Schonell
9;4 years 12;10 years
Literacy Literacy
WRAT-4 WRMT-III

Fig. 1. Timeline of research and clinical speech pathology assessments from age 3–15 years. NAPLAN testing was
conducted in Year 3 (8 years), Year 5 (10 years), Year 7 (12 years) and Year 9 (14 years) (indicated by stars).
DEAP, Diagnostic Evaluation of Articulation and Phonology Inconsistency Assessment; NWR, Non-word Mem-
ory Test; GFTA, Goldman-Fristoe Test of Articulation; CTOPP, Comprehensive Test of Phonological Processing;
WRAT, Wide Range Achievement Test; WRMT, Woodcock Reading Mastery Test; MSW, Multisyllabic Word
Repetition task; Quick Screener, A Quick Test of Articulation and Phonology; Schonell, Schonell Graded Word
Reading and Spelling test. ^ External clinical assessment.

Table 3. Liam’s speech features from 3;10 to 10;5 years

Age, Consonant inventory Syllable Phonological processes Other

years, shapes
months FCD voicing stopping WSD fronting CR deaffrication gliding atypical

3;10 Early-8 CV ** ** ** ICD Consonants correct

/m, n, j, b, w, d, p, h/ VC [k] for /g/ [b] for /z/ MCD 17%
Middle-8 CVC [b] for /p/ Nasals for stops Vowel errors
/t, ŋ, k, g, f/ VCV Favourite sound /j/ Assimilation
Late-8 CVCV Metathesis
/l, z/ CVCVC
VCVC
VCVCVC

5;3 ** ** ** ** ** [w] for /r, l/ ICD Vowel distortions

[d] for /t/ [t], [d] for [t] for /k/ Frequent omissions
/s, z, ʃ, θ/ [d] for /g/
[b] for /v/

6;7 Early-8 as above + ** ** ** ** ** ** [f] for /ts/ Consonants correct

/m, n, j, b, w, d, p, h/ CVCC [b] for /p/ [d], [t] for /s, z/ [t], [d] for /k, g/ [d] for /dʒ/ [w] for /r/ /s/ for /θ/ 61%
Middle-8 CCVC [s] for /ʃ/ [t] for /tʃ/ [h] for /j/ Vowels 94%
/t, ŋ, k, g, f, v, dʒ/ [ts] for /tʃ/
Late-8 Age appropriate
/ʒ, l, s, z/ [f] for /θ/

7;5 Early-8 as above + ** ** ** ** [s] for /f/ Consonants correct

/m, n, j, b, w, d, p, h/ CCVCC [s] for /z/ [b] for /v/ [s] for [f] [w] for /r, l/ [ʃ] for /s/ 80%
Middle-8 CVCVC Age appropriate Insert schwa in Vowels 88%
/t, ŋ, k, g, f, v, tʃ, dʒ/ CVCVCC [f] for /θ/ clusters
Late-8 CVCCVC [v] for /ð/
/ʃ, ʒ, l, r, s, z, θ, ð/ CVCCVCC
CVCVCVC

7;10 ** ** ** [n] for /m/ Consonants correct

[s] for /ʃ/ [d] for /dʒ/ [w] for /r/ Insert schwa in 95%
[z] for /ʒ/ [t] for /tʃ/ clusters Vowels 94%
[d] for /g/
Age appropriate
[f] for /θ/
[v] for /ð/

10;5 ** ** ** Poor repetition of

[s] for /z/ [m] for /n, ŋ/ [w] for /l/ multisyllabic and
[f] for /θ/ [l] for /r/ non-words
Inconsistency 32%
(DEAP)

** Delayed phonological process.

FCD, final consonant deletion; WSD, weak syllable deletion; CR, cluster reduction; ICD, initial consonant deletion; MCD, medial consonant deletion; DEAP, Diagnostic Evaluation of Articulation and Phonol-
ogy. Shaded box indicates that the phonological process is present.
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were collected where possible. His performance results on the Na-
tional Assessment Program – Literacy and Numeracy (NAPLAN)
were obtained from his school. NAPLAN is an assessment of aca-
demic performance conducted annually for all Australian students
in years 3, 5, 7, and 9, across the domains of reading, writing, spell-
ing, grammar/punctuation, and numeracy [25].
Overall, speech pathology and NAPLAN data were available at
17 timepoints from 3;10 to 15 years (Fig. 1).
Lexical Stress
Liam’s lexical stress production in multisyllabic words was ex-
amined using an acoustic measure of stress contrastivity, namely,
the pairwise variability index (PVI) [26, 27]. PVI quantifies relative
stress across adjacent syllables within a word. It provides a separate
measure for each of the suprasegmentals that denote lexical
stress – vowel duration (ms), peak intensity of the vowel (dB), and
fundamental frequency. Typically developing children use these
suprasegmentals to contrast weak and strong syllables within words
at around 2–3 years of age [28]. Normalized PVI is derived by cal-
culating the difference in vowel duration (or vowel intensity) be-
tween adjacent vowels, divided by the average duration multiplied
by 100.
Liam’s single word productions on the GFTA-2 conducted at
7;4 years, and the DEAP Inconsistency Assessment and multisyl-
labic word repetition task conducted at 10;5 years were used for
lexical stress analysis. Three and 4 syllable real words were ana-
lyzed, with a trochaic (strong-weak; SW) or iambic (weak-strong;
WS) stress pattern across the first 2 syllables. A total of 47 word
tokens were included in the analysis (34 SW words; 13 WS words);
acoustic measures were derived using Praat [29] according to pre-
viously published methods. PVIs for vowel duration (PVI_dura-
tion) and peak vowel intensity (PVI_intensity) were calculated.
Results
Speech
Liam’s consonant inventory, syllable shapes, phono-
logical patterns, and consonant and vowel accuracy from
3;10 to 10;5 years are shown in Table 3.
He had acquired all consonant sounds by 7;5 years and
was able to produce a variety of syllable shapes. Consonant
accuracy measured in percent consonants correct (PCC)
improved with age (17% PCC at 3;10 years; 95% at 7;10
years). He had a number of delayed and atypical phono-
logical patterns in his speech, with voicing, fronting, and
gliding still evident at 10;5 years. Atypical patterns includ-
ed initial consonant deletion. He had impaired perfor-
mance of a number of speech tasks at 10;5 years, including
poor repetition of nonwords and multisyllabic words and
high token-to-token variability (DEAP inconsistency score
32%). The presence of inconsistent errors past 7 years of
age is regarded as atypical (B. Dodd, personal communica-
tion). Errors evident on word repetition tasks included
sound substitutions and omissions (e.g., /vʌlnrəbəl/ →
Adolescent Outcomes in CAS
Table 4. Mean normalised PVI for vowel duration and intensity at
2 different ages
Age, SW WS
years,
months PVI_duration PVI_intensity PVI_duration PVI_intensity
7;4 –5.74a 0.42 –23.6 –0.01
10;5 5.26 0.79 –7.86 –0.78
PVI_duration, normalised PVI for vowel duration (ms); PVI_intensity,
normalised PVI for peak vowel intensity (dB). SW, strong-weak; WS, weak-
strong; PVI, pairwise variability index
a Positive normalised PVI is expected for words with a target SW stress
pattern.
/vʌnəbl/), syllable deletions (e.g., /pærəlɛl/ → /pærɛl/),
metathesis (e.g., /ɛnəmi/ → /ɛməni/), and vowel errors,
with more errors noted as syllable length increased (non-
word repetition: 2 syllables 43% correct; 3 syllables 29%
correct; 4 syllables 14% correct; 5 syllables 0% correct).
Maximum repetition rate of monosyllables was slow (/ta/
4.3 syllables/s; /ka/ 4.0 syllables/s), and he was unable to
correctly repeat a trisyllabic sequence (pataka).
Lexical Stress Analysis (Table 4)
Words with a target SW stress pattern across the first 2
syllables should be produced with a positive normalized
PVI, while words with a target WS stress pattern should
be produced with a negative normalized PVI. At 7;4 years,
Liam produced SW words with positive mean PVI_inten-
sity but negative mean PVI_duration on average. The data
in Table 4 suggest that he had difficulty coordinating du-
ration and intensity parameters to produce the target pro-
sodic patterns across multisyllable words. Children typi-
cally achieve adult-like stress contrastivity in words with
a SW stress pattern by 3 years of age [28]. He had negative
mean normalized PVI_duration and PVI_intensity at 7;4
and 10;5 years, indicating appropriate placement of stress
on the second syllable in words with WS stress.
The value of normalized PVI may also give an indication
of lexical stress, with a larger value (either positive or nega-
tive) indicating a greater level of stress contrastivity. A PVI
approaching zero represents more equal stress. At 7;4 years,
the value of Liam’s PVI_duration means were small in mag-
nitude for both SW (–5.74) and WS (–23.6) words. As a
point of reference, PVI duration means between 22.4 (SD
25.8) and 49.6 (SD 26.8) for SW words, and –94.3 (SD 25.5)
and –116.23 (SD 17.9) for WS words were reported in typ-
ically developing children aged 3–7 years [28], although
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 Color version available online
110
Receptive
105
Expressive
100

95

90

85
CELF standard score

80

75

70

65

60

55

50

45
3;10 5;3 7;4 8;11 11;10 13;4 15;11
Years

Fig. 2. Liam’s Receptive Language Index (blue) and Expressive Language Index (orange) standard scores on the
Clinical Evaluation of Language Fundamentals from 3 to 15 years of age. Index scores are on a normalised stan-
dard score scale that has a mean of 100 and a standard deviation of 15. Error bars reflect confidence intervals at
95% level.

Table 5. CELF subtest scaled scores

Age, Receptive subtests Expressive subtests

years, LC BC SS CFD WC-R USP FL RS WS FS WC-E
months

3;10 6 5 9 5 3 3
5;3 6 7 8 1 5 4
7;4 7 8 10 6 1 5 8
8;11 10 9 4 9 6 3 8
11;10 11 8 6 8 7 7
13;4 6 2 9 6 5
15;11 9 8 8 6 7

Subtest scores are on a normalised score scale that has a mean of 10 and a SD of 3. Scaled scores >1 SD below
the mean are highlighted in bold, indicating performance below the average range.
LC, Linguistic Concepts; BC, Basic Concepts; SS, Sentence Structure; CFD, Concepts and Following Direc-
tions; WC-R, Word Classes, Receptive; USP, Understanding Spoken Paragraphs; RS, Recalling Sentences; FL,
Formulating Labels; WS, Word Structure; FS, Formulated Sentences; WC-E, Word Classes, Expressive; CELF,
Clinical Evaluation of Language Fundamentals.
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Band 8
Band 7
Band 6
Band 5
Band 4
Band 3
Band 2
Band 1
Reading Writing Spelling
Fig. 3. Liam’s NAPLAN scores (denoted by x) in Grades 3, 5, 7, and 9 compared to the middle 60% of students
in Australia in reading, writing, spelling, grammar/punctuation, and numeracy.
these values are not directly comparable to Liam’s means as
different tokens were used. Liam’s PVI intensity means
were close to zero (SW 0.42; WS –0.01) indicating more
equal stress. Similar means were evident at 10;5 years. Tak-
en together, these findings suggest that Liam was producing
speech with reduced stress contrastivity between adjacent
syllables and that this persisted up to 10 years of age.
Language (Fig. 2; Table 5)
Liam had moderate-severe expressive language im-
pairment from 3 to 8 years of age, with scores at or below
the 5th percentile on the CELF. A shift was noted at 11
years, with average expressive language (standard score
85 at 11 years; 80 at 13 years; 83 at 15 years). This was in
contrast to average receptive language skills (standard
scores 80 or above) from 3 to 15 years. Of note, his recep-
tive language scores fell significantly at 13 years with per-
formance in the 4th percentile.
Analysis of his performance on the different receptive
and expressive language subtests revealed impairments
across the domains of syntax, semantics, and morphology.
His ability to apply word structure rules (Word Structure)
and form semantically and grammatically correct spoken
sentences (Formulated Sentences) were areas of relative
weakness. Repetition of spoken sentences improved from
8;11 years (Recalling Sentences). He was able to interpret
spoken sentences and directions of increasing length and
complexity (Concepts and Following Directions; Sentence
Structure), yet had difficulty listening to spoken para-
graphs and understanding and answering questions about
the text.
Adolescent Outcomes in CAS
Color version available online
Grammar and punctuation Numeracy
Academic Outcomes
NAPLAN results are reported on a 10-band scale span-
ning Grade 3 to Year 9, comparing an individual student’s
performance to grade-level peers across Australia. Liam’s
NAPLAN scores across all domains at age 8 years (Grade
3), 10 years (Grade 5), 12 years (Year 7), and 14 years
(Year 9) relative to Australian students nationwide are
shown in Figure 3.
Liam’s reading, writing, spelling, and grammar/punc-
tuation were below the national average in Grades 3 and 5.
Reading and writing were particularly poor, with his per-
formance at the minimum Australian standard for reading
in Grade 3 (band 2) and for writing in Grade 5 (band 4).
His phonological awareness skills were in the average range
at 7;5 years (CTOPP Phonological Awareness standard
score 97; Phonological Memory 70). A literacy assessment
at 9;4 years revealed average reading and spelling (WRAT-
4 Word Reading Standard score 89, Sentence Comprehen-
sion 92, Spelling 96). This disparity with the NAPLAN re-
sults likely reflects differences in test administration
(WRAT spelling to dictation vs. NAPLAN error correction
and proofreading) with more words spelt correctly when
dictated [30]. An independent clinical evaluation of literacy
skills in Years 5 and 6 revealed reading and spelling skills
were 1–2 years below age expectations (Schonell Word
Reading age equivalent 9 years and Spelling 8;9 years in
Year 5/10;9 years and Word Reading age equivalent 9.11
years and Spelling 10.2 years in Year 6/11;10 years).
Improvements in all areas were evident in secondary
school, and by his final year of NAPLAN assessment
(Year 9), Liam was performing above the national average
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in reading, writing, and grammar/punctuation. He had
average word reading (Word Identification standard
score 87), reading fluency (Oral Reading fluency score
91), and listening comprehension (Listening Compre-
hension score 84) on an independent clinical evaluation
of reading at 12; 10 years (Woodcock Reading Mastery
Test, 3rd Edition; WRMT-III). Yet difficulties with non-
sense word reading (Word Attack standard score 77) and
reading comprehension (Word comprehension standard
score 78; Passage comprehension 61) were evident. His
spelling scores on the NAPLAN continued to fall below
the national average up to 14 years of age. His numeracy
skills were strong relative to language-based domains,
with scores at or above the national average in both pri-
mary and secondary school (8–14 years).
Discussion
Our study is the first to report long-term speech, lan-
guage, and academic outcomes in CAS from preschool to
adolescence (3–15 years). Previously studies report 3 or 4
years follow-up, predominately from preschool (4–6 years)
to school age (6–11 years). Liam made significant improve-
ment over time, and at 15 years of age had intelligible
speech, average receptive and expressive language skills,
and scored above the national average in reading, writing,
grammar/punctuation, and mathematics. Deficits in read-
ing comprehension, decoding of nonsense words, and
spelling persisted into adolescence. Liam had speech pa-
thology intervention for a number of years, including sev-
eral bursts of intensive therapy, thus it is important that
these longitudinal changes are considered in light of this.
Speech Outcomes
Liam’s speech development mirrored other reported
cases of CAS [3–7]. Over time, his speech became more in-
telligible and his consonant accuracy improved, with 95%
PCC by 7 years. He had delayed and atypical phonological
error patterns, including fronting, gliding, and voicing,
which are commonly reported in school age children and
adolescents with CAS [4, 7, 10, 31]. He also had persistent
syllable structure deficits identified in other CAS cases,
with frequent omissions (initial, medial, and final conso-
nant deletion) evident up to 5;3 years and reduction of ini-
tial and final consonant clusters up to 6;7 years. Sound and
syllable omissions were still apparent at 10 years in his re-
sponses to word and nonword repetition tasks, along with
metathesis and vowel errors. Several studies report these
types of errors (sound and syllable omissions, sound rever-
10 Folia Phoniatr Logop
DOI: 10.1159/000500554
sal and insertions, vowel errors) in the speech of older in-
dividuals with CAS [6, 31, 32]. These error types are diag-
nostic of CAS and may serve as a useful sign of the disorder
in adolescents and adults whose conversational speech er-
rors have largely resolved. This observation is supported by
a recent study which found adults with a probable history
of CAS had significantly more sequencing errors (e.g., as-
similations, migration, metathesis, omissions, and inser-
tions) than controls [33]. Prosodic errors also persisted,
with reduced stress contrastivity evident at 10 years. This
was not unexpected given that more than half of children
with suspected CAS have excessive, equal, or misplaced
stress, and these errors may be apparent as late as 14 years
of age [34, 35]. Liam performed poorly on multisyllabic
word and nonword repetition tasks, which is typically re-
ported in CAS and a core deficit in cases with CAS and
FOXP2 mutations [36, 37]. Deficits in nonword repetition
and sequencing are postulated to be part of a sequential
processing deficit underlying CAS [33].
Language Outcomes
Liam’s receptive and expressive language skills fol-
lowed distinct trajectories over a 10-year period, with a
shift noted around preadolescence (11–13 years). His ex-
pressive language improved, with standard scores pro-
gressing from below the 5th percentile at the start of pri-
mary school into the average range by his final year. His
expressive language scores remained stable into adoles-
cence. Other longitudinal studies suggest that the major-
ity of children with CAS show little improvement at school
age [4, 7, 8]. In a longitudinal cohort of 10 children with
CAS, only one made gains in expressive language from 5
to 7 years, moving from the 5th to the 42nd percentile [4].
A second child had average expressive language at 9 years
(standard score 86). These studies do not report language
outcomes past 11 years of age, so it may be that changes in
adolescence would have been seen given longer follow-up.
In contrast to expressive language, Liam’s receptive
language skills declined sharply in adolescence, with
scores below the 5th percentile at 13 years. Receptive lan-
guage had previously been in the average range through-
out preschool and primary school. He had a 6-month
break in therapy at this time. A very low score on the Un-
derstanding Spoken Paragraphs subtest primarily drove
Liam’s poor receptive language score at 13 years. Different
modes of assessment vary in the cognitive load they place
on the test-taker’s working memory, with attentional re-
source capacity/allocation and processing speed crucial to
the understanding of spoken narrative [38]. Liam evi-
denced working memory impairments at 7 years that may
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have degraded his performance on the subtest. The low
Understanding Spoken Paragraphs score may also be due
to measurement error. The reliability of this subtest is not
high (test–retest stability coefficient 0.76; internal consis-
tency coefficient 0.70). There is also a degree of error re-
flected in the CELF scores, with Liam’s true receptive lan-
guage score falling within a range from 65 and 83 (95%
CI). Of note, Liam performed poorly on other receptive
language tasks around 12–13 years (WRMT-III Passage
Comprehension standard score 61, Word comprehension
78 at 12;10 years; CELF-4 Word Classes-Receptive 6 at 13;
4 years) and deterioration in receptive language was evi-
dent in younger cohorts with CAS [4, 7]. Liam’s receptive
language skills returned to the average range at 15 years.
Academic Outcomes
Liam’s poor NAPLAN performance from Grade 3 to
Year 7 is likely a direct result of his speech and language
impairment. Significant differences in NAPLAN out-
comes were found for children in the Longitudinal Study
of Australian Children who had speech and language
problems, compared with typically developing peers [39].
Despite improvements in their NAPLAN performance
over time, they had consistently lower scores across all
tests (reading, writing, spelling, grammar, and numeracy)
up to Year 7 and did not “close the gap.” Around preado-
lescence, Liam’s expressive language skills improved and
by his final year of NAPLAN assessment (Year 9), he was
performing above the national average in reading, writing,
and grammar/punctuation. His spelling scores continued
to fall below the national average up to 14 years of age.
Liam’s numeracy skills were strong relative to lan-
guage-based domains, with scores at or above the nation-
al average in both primary and secondary school (8–14
years). Numeracy in CAS has received little attention to
date. Around one-third of parents report their child with
CAS has difficulties with mathematics [40]. One individ-
ual with persistent speech impairment in a large CAS kin-
dred (PM family) scored 1.8 SD below the mean on the
Numerical Operations subtest of the Wechsler Individu-
al Achievement Test [41]. Ten other affected family mem-
bers had standard scores of 80 or above. Of note, 6 PM
family members with persistent speech deficits (aged
9–55 years) had specific learning disorder with impair-
ments in reading and written expression.
There are limited data available on how language and
literacy abilities evolve in CAS past primary school age. A
handful of multiplex family studies have examined lan-
guage skills in adults with CAS or suspected CAS in child-
hood. In the KE family with a FOXP2 mutation, affected
Adolescent Outcomes in CAS
family members (aged 9–75 years, mean 25.3 years) had
comparable performance to adults with aphasia on tests of
receptive grammar, nonword repetition, object naming,
production of inflectional and derivational morphology,
regular and irregular past tense production, non-word
reading and spelling [42]. In the PM family, family mem-
bers with persistent speech disorder had impaired expres-
sive language (CELF-4 Expressive Language Index 40–70),
while receptive language skills ranged from severely im-
paired to average (CELF-4 Receptive Language Index 63–
92) [41]. Interestingly, family members with resolved
speech sound disorder (13–51 years) had average receptive
and expressive language skills. Liam’s reading and spelling
outcomes appeared better than those of 2 adolescents with
CAS aged 14;5 and 15 years, who had severely impaired
skills despite years of phonics therapy [11]. Their spelling
errors (deletions, intrusive sounds, perseverations) were re-
portedly analogous to their speech errors. At 15 years, Liam
evidenced sound omissions, substitutions, and additions
when reading aloud (e.g., soloist read as “solist” and satirical
as “sartiriatical”) and spelling (e.g., permanent spelt “perna-
ment” and enthusiastic spelt “infusiatic”). Deficits in non-
word reading and spelling have also been reported in adult
family members from other CAS kindreds [31, 32].
Taken together with previous longitudinal studies, our
findings suggest that while diverse speech symptomatol-
ogy may be seen in individuals with CAS, they may share
a developmental pattern of speech improvement with
age. As CAS is a symptom complex with a range of cogni-
tive and other motor sequencing comorbidities [43, 44],
the long-term trajectory for the broader CAS phenotype
is more variable. This is likely due to distinct deficits un-
derlying CAS [43]. For example, our case AN with CAS
and a FOXP2 mutation had more severely impaired
speech and language than Liam at a similar age (39% PCC
at 8 years; receptive language 0.1 percentile, expressive
language <0.1 percentile) [37]. We have established that
both individuals have distinct underlying etiologies and
associated neural phenotypes [45, 46]. Variants in a num-
ber of different genes have been identified in CAS, includ-
ing FOXP2, GRIN2A, KANSL1, and BCL11A as well as
16p11.2 deletions and other copy number variations [37,
47–50]. This suggests multiple different subtypes of CAS
with distinct speech and language trajectories.
Limitations of the Study and Future Directions
This is a single descriptive case study and as such had
a number of limitations. Liam’s literacy skills were evalu-
ated in research and clinical settings using different as-
sessment tools, with variable findings. He performed in
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the average range on the WRAT-4 at 9;4 years; yet, his
reading and spelling skills were 1–2 years below age ex-
pectations in years 5 and 6 on the Schonell Graded Word
Reading and Spelling Test. The different psychometric
properties of the tests likely account for these discrepant
results. Scores on the Schonell are expressed as reading
ages, based on a sample of British children from one loca-
tion in the late 1960s/early 1970s. Conversely, the WRAT-
4 reports standard scores and percentile ranks, based on
a national sample of over 3,000 individuals. The limita-
tions of age equivalent scores vs standard scores are well
documented [51]. At this time, Liam scored below the
national average on NAPLAN reading and spelling. This
may due to the different mode of assessment used as high-
lighted previously. Assessment at 12; 10 years on the
WRMT III revealed difficulties with reading comprehen-
sion and reading nonsense words. Future studies examin-
ing reading and spelling using the same assessment tool
will further clarify the evolution of literacy skills in CAS.
While language was examined using the same assess-
ment tool from 3 to 15 years, the CELF provides only lim-
ited information regarding language skills. We have not
explored Liam’s pragmatic or higher-level language skills,
nor considered the impact of his metalinguistic skills on
his language and literacy development. Future longitudi-
nal studies investigating these aspects would provide a
more comprehensive view of language in CAS over time.
The research speech tasks were independently rated by
2 experienced speech pathologists and consensus reached
on discrepant ratings; however, inter-rater agreement
was not determined.
We have detailed Liam’s speech therapy from 3 years of
age, including frequency and targets of therapy, yet have not
examined the correlation between therapy and the evolu-
tion of Liam’s speech, language, and literacy skills over time.
While the magnitude of Liam’s PVI means suggested
deficits in stress contrastivity, his PVI data could not be di-
rectly compared to a sample of typically developing chil-
dren aged 3–7 years [28] and 8–11 years [52], as different
word tokens were used. Change in stress contrastivity with
age in CAS would be an interesting area of future study.
Further long-term studies in a larger subset of indi-
viduals are needed to confirm the trends seen here. Yet,
the results presented here a positive prognosis for speech,
receptive and expressive language and academic areas
such as writing and mathematics for a subset of individu-
als with CAS.
Acknowledgments
The authors thank Liam and his family for taking part in the
study.
Disclosure Statement
A.P.V. serves as chief science officer of Redenlab who provide
assistance with acoustic analysis. The other authors report no dis-
closures.
Funding Sources
S.J.T. supported by National Health and Medical Research
Council (NHMRC) Postgraduate Scholarship (101777), Austra-
lian National University Gowrie Scholarship, and Speech Pathol-
ogy Australia Nadia Verrall Memorial Research Grant. A.P.V. sup-
ported by NHMRC Fellowship (1135683). A.T.M. supported by
NHMRC Practitioner Fellowship (1105008). I.E.S. supported by
NHMRC Program Grant (628952) and Practitioner Fellowship
(1006110). Project also supported by NHMRC Centre for Research
Excellence Grant (1116976), NHMRC Project Grant (1127144),
and Australian Research Council Discovery Project (DP120100285)
to A.T.M. and I.E.S.
Author Contributions
S.J.T. designed the study and wrote the manuscript. A.P.V. per-
formed acoustic analysis. S.J.T and R.C. performed research and
clinical assessments. S.J.T., B.P.-F., and A.T.M. performed pheno-
typic analysis. A.T.M., and I.E.S. supervised the study.

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