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Self-Reported Communication Attitudes of Children with Childhood Apraxia of Speech

Sydney Keller

Department of Communication Sciences and Disorders, Temple University

February 22, 2022

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ABSTRACT

Much of the research literature on childhood apraxia of speech (CAS) has focused on

understanding, diagnosing, and treating the impairment, rather than examining its broader

impact. The present study focuses on the Personal Factors component of the World Health

Organization model. Two validated communication attitude questionnaires were administered to

12 children with CAS enrolled in an intensive speech-focused intervention. Children’s scores

were compared to the questionnaires’ typically developing norms. Relationships to CAS

severity, caregiver perceptions of communicative participation, frustration ratings during

therapy, and change over a brief period were also investigated. Preliminary findings indicate that

older but not younger children with CAS are more likely to have greater negative self-

perceptions about their speech. No significant correlation was found between caregivers’

perceptions of communicative participation in various contexts and communication attitudes,

highlighting the need to include more child self-report measures in research. Further implications

for CAS assessment and intervention are discussed.

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INTRODUCTION

Capturing child perspectives can provide researchers and clinicians with a unique

understanding of the contextual factors that influence how children experience the world. This is

particularly important for children with speech sound disorders who have distinct communicative

challenges compared to their typically developing peers. Speech sound disorders (SSD) are a

group of disorders that cause difficulty with motor production, perception, or phonological

representations of speech sounds or syllables (American Speech-Language-Hearing Association,

[ASHA] 2007). Examples of SSDs include articulation disorder, phonological disorder, and

childhood apraxia of speech. Articulation disorder impacts speech production at the sound level,

while phonological disorder impairs production at the syllable level (Dodd, 2014). These

disruptions cause predictable errors like sound substitutions and consonant cluster reductions.

Childhood apraxia of speech (CAS), a motor-based speech sound disorder, results from

inaccurate motor planning and programming of the speech musculature (ASHA, 2007). Though

estimates of prevalence are ongoing, approximately 1-2 children per 1,000 in the United States

are thought to be diagnosed with CAS (Shriberg et al., 2019). Children with CAS have difficulty

timing and orienting their speech musculature that cannot be accounted for by physical or

cognitive disabilities (Murray & Iuzzini-Seigel, 2017). The inability to accurately plan

movements causes several impairments including prosodic and stress errors, inconsistency over

repeated trials, and consonant and vowel distortions (Lewis et al., 2004). The unpredictability of

impairments poses a distinct challenge for treatment and distinguishes a CAS diagnosis from

other speech sound disorders. Lewis and colleagues (2004) found that school-age children with

CAS have more frequent and severe errors compared to their peers with other SSDs. A CAS

diagnosis also puts children at an increased risk of other expressive and receptive difficulties
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including reading and writing (Lewis et al., 2004). These distinguishing factors may mean that

CAS’s impact on children’s daily lives and personal attitudes differ from the experience of

children with other SSDs.

The World Health Organization’s International Classification of Functioning, Disability,

and Health (ICF) provides a framework for better understanding the complex array of factors that

impact individuals with various disorders (see Figure 1, Threats, 2006). This framework

recognizes the interconnections between the three levels and encourages clinicians to approach a

client’s care holistically. Level (1) is the identification of the disorder or disease. Level (2)

consists of body functions/structures, activity, and participation. CAS research thus far has

almost exclusively addressed Level (2) of the ICF, with many studies aimed at finding effective

treatments. Though no treatment has conclusive evidence of effectiveness, some show promising

outcomes. Current evidence suggests that the best CAS treatments target motor learning

principles to improve overall accuracy across syllables and words (Murray & Iuzzini-Seigel,

2017). Effective CAS treatments should also be deliberate in both how children practice during

therapy and what targets are chosen (Maas et al., 2014). One such treatment is Dynamic

Temporal and Tactile Cueing (DTTC), which relies on Integral Stimulation to target the planning

and programming of multiple speech sounds. A key feature of DTTC is its use of individualized

meaningful target words (Strand, 2020). In contrast, Rapid Syllable Transition treatment (ReST)

consists of varying the stress of nonsense words and probing for real words (McCabe et al.,

2017). The rationale for ReST is that nonsense targets can lead to more accurate planning across

multiple generalizable words. Outcomes measures for both DTTC and ReST are target accuracy.

In addition to target accuracy, other factors such as intelligibility, activity and participation, and
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changes in personal attitudes towards communication should also be considered to determine

degree of treatment success.

Figure 1: The World Health Organization’s International Classification of Functioning,

Disability, and Health

Neglected from current research on CAS is Level (3) of the ICF: environmental and

personal factors. Environmental factors are described as those influences that play a significant

role in the individual’s life beyond personal control. For instance, external factors such as public

policy or parental support can impact an individual’s functioning. Personal factors are those

more unique to the person’s life such as race, ethnicity, personality, and motivations (Threats,

2006). They can be divided into unchangeable factors (e.g., race and age) and factors that have

the possibility of changing such as lifestyle habits and coping styles (Howe, 2008). Personal

factors can help researchers and clinicians understand how different individuals are impacted by

the same diagnosis (Threats, 2006). Understanding the perspectives of children with CAS may

prove useful for informing treatment approaches and for developing functionally relevant goals
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(Murray & Iuzzini-Seigel, 2017). Given that personal factors were found to influence treatment

outcomes for children with phonological disorder (Baker & McLeod, 2011a, 2011b), it is critical

that researchers better understand how this translates for children with CAS.

Parent perspectives are often used as a proxy for understanding the feelings and thoughts

of children with communication disorders. This is usually due to the perceived and inherent

difficulties that accompany collecting child perspectives. These difficulties include questioning

the reliability and validity of a child’s response and accounting for extra ethical considerations

when working with children (Harcourt, 2011). In many cases, parents provide accurate and

detailed accounts for their child. For example, Rusiewicz et al. (2018) used parent measures to

evaluate the daily impact of a CAS diagnosis, with parents being particularly concerned about

their child’s overall intelligibility and ability to navigate social situations. More generally,

McCormack et al. (2010) asked parents and speech-language pathologists to rate how they felt

about their children’s speech impairment using an ICF-CY based questionnaire. They found that

both populations expressed the greatest concern regarding interpersonal relationships and verbal

communication skills. These studies demonstrate the ability of parent perspectives to collect

more detailed information than would usually be possible from a child.

Solely collecting parent perspectives, however, relies on the assumption that they are

always accurate. Investigations into correlations between parent and child perspectives for

children with phonological disorder (McCormack et al., 2019) and children who stutter

(Vanryckeghem, 1995) have shown that parents do not always understand their child’s

communication attitudes. Disregarding child perspectives also fails to directly include children in

the conversation. According to Article 12 of the United Nations Conventions on the Rights of the

Child (1989) and the follow-up General Comment No. 7 (2005), children must be allowed to
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express their views and be included in decisions that directly impact them (Lundy et al., 2011).

These declarations underscore and reinforce the need to incorporate more child perspectives into

research and clinical practice in communication disorders. When collected correctly, children

can provide unique, distinct insights into their life experiences (Harcourt, 2011). It is worthwhile

to acknowledge that children’s perceptions may change frequently and reflect growing

development, but this does not imply that their responses at the given assessment are any less

relevant or valuable to examine (Harcourt & Einarsdóttir, 2011). Directly asking children with

speech and language disorders how they feel is also an effective way of understanding if children

are receiving adequate support (Roulstone & McLeod, 2011).

Relatively few self-report measures exist for understanding the self-perceptions of

children with communication disorders. Each have distinct strengths and weaknesses. The lack

of available measures is a contributing factor as to why there are few studies in this realm. For

the current study, relevant measures were examined for validity and feasibility.

The Speech Participation and Activity Assessment of Children

The Speech Participation and Activity Assessment of Children (SPAA-C) provides open

ended and Likert scale questions to uncover how speech difficulties influence children’s lives

(McLeod, 2004). It considers how other members of the child’s life (i.e., parents, friends,

siblings, parents, teachers, others) perceive the child with the speech difficulty. Questions range

from “Who do you like to play with?” to “Do you think your talking is different from other

children’s?” to gain a larger picture of the child’s perceptions. It is designed to be adapted for the

particular needs of the researcher and abilities and age of the child. McLeod, Daniel, and Barr

(2013) used the SPAA-C with school-aged children with speech sound disorders and emphasized

that children would be better supported if their perspectives were more often incorporated in
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assessment and intervention. Other applications of the SPAA-C have revealed the frustrations of

the child with an SSD in being misunderstood and communicative partners lack of listening

skills (McCormack, McLeod, Harrison et al., 2010) and incapacity to provide adequate support

in various settings (Daniel & McLeod, 2017). Though valuable, application of the SPAA-C can

be burdensome and time-consuming for participants and is less optimal for quantitative analysis.

The Pictorial Scale of Perceived Competence and Social Acceptance for Young Children

The Pictorial Scale of Perceived Competence and Social Acceptance for Young Children

(PSPCSA) asks children to compare pictures, a useful tool when examining child perspectives

(Elden, 2013), and decide which picture is more representative of themselves (Harter & Pike,

1984). A score of 1 to 4 is given for each image depending on the extent the child indicates that

picture applies to them. The PSPSCA has been given to children with specific language

impairment (SLI), with findings suggesting that older children with SLI struggle more with

scholastic competence, social acceptance, and behavioral conduct than their typically developing

peers (Jerome et al., 2002). However, question wording on the PSPSCA is outdated (i.e.,

“maternal acceptance” rather than “parental acceptance”), and there are doubts regarding its

application to diverse populations (Fantuzzo, 1996).

The Communication Attitude Test for School-age Children who Stutter

The Communication Attitude Test for School-age children who stutter (CAT), a

component of the Behavior Assessment Battery for Stuttering, is designed to assess the speech

attitudes of children ages 6;0 and older (Brutten & Dunham, 1989; Brutten & Vanryckeghem,

2007). It consists of 33 true/false statements such as “I like to talk” and “My parents like how I

talk.” Jones and colleagues (2021) found the CAT to possess among the highest measurement

properties compared to other measures of the psychological impacts of stuttering.

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The Communication Attitude Test for Preschool Age Children who Stutter

The Communication Attitude Test for Preschool Age children who stutter (KiddyCAT)

was developed as an extension of the CAT to assess how preschool children feel about their

speech (Vanryckeghem & Brutten, 2007). It asks children (ages 3;0-5;11) 12 yes/no questions

such as “Do you like how you talk?” and “Do words sometimes get stuck in your mouth?”

Both the CAT and KiddyCAT have been administered in several countries including

Australia (McCormack, McLeod, & Crowe, 2019), Italy (Bernadini et al., 2009), Belgium

(Vanryckeghem & Brutten, 1992; Vanryckeghem, De Niels et al., 2015), Japan (Kawai, 2012),

and Sweden (Johannisson, 2009), implicating its validity across cultures. Though developed for

stuttering, these assessments have been successfully administered to many clinical populations

including to children with phonological disorder (McCormack, McLeod, & Crowe, 2019)

articulation disorder (Luc & Brutten, 1990) and cleft palates (Havstam et al., 2011). These

applications are positive indications that the CAT and KiddyCAT can be administered to

populations other than to children who stutter.

Administration of the CAT and KiddyCAT to children who do and do not stutter have

revealed that children who stutter are more likely to have negative speech perceptions and that

younger children tend to feel more positively than older children about their speech (Brutten &

Vanryckeghem, 2007). Influence of age differences beyond stuttering was highlighted by

McCormack, McLeod, and Crowe (2019), who found that preschool age children with

phonological disorder did not have more negative perceptions of their speech compared to the

established norms.

Those administering both assessments are provided specific directions, which minimizes

issues with test-retest reliability and distortion of results. The CAT and KiddyCAT were chosen
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for the present study because they are standardized, encompass the age range (4;0-10;0), are easy

to track changes over time, and have been found to be reliable and valid across a variety of

languages, cultures, and clinical populations. Despite its widespread use, neither the CAT nor

KiddyCAT have been administered to a group of children with CAS. Given that CAS differs in

consistency of impairment and response to treatment compared to other SSDs, it is clearly

necessary to further understand these children’s communication attitudes.

The Present Study

The present study was designed to fill this critical gap in the literature by administering

the CAT and KiddyCAT to children with CAS. This study took place in the context of a

randomized controlled trial of (impairment-focused) speech therapy for CAS, which was

conducted as an intensive summer camp (see Methods section for further description of the

broader study context). This setting afforded the opportunity to address the following questions:

1. Do communication attitudes of children with CAS differ from those of typically

developing children, based on the tests’ normative values?

2. Do communication attitudes of children with CAS relate to impairment severity,

as operationalized by DEMSS score?

3. Do communication attitudes of children with CAS relate to caregivers’

perceptions of their child’s communicative participation, as operationalized by

FOCUS-34 score?

4. Do communication attitudes of children with CAS relate to average frustration

level during therapy?

5. Do communication attitudes of children with CAS change over the course of a

brief intervention period?

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METHODS

Study Context

This study took place in the context of a larger (“parent”) study involving a randomized

controlled trial to test initial efficacy of ASSIST (Apraxia of Speech Systematic Integral

Stimulation Treatment; Maas et al., 2019) for children with CAS. ASSIST is an impairment-

focused speech treatment approach based on integral stimulation (“watch me, listen to me, say

what I say”) and principles of motor learning (Maas et al., 2008). Speech targets are words and

phrases selected based on personal functional relevance and contain only sounds in the child’s

phonetic inventory. The treatment involves drill-based repetition of targets, with feedback and

cues provided by the treating clinician to help the child achieve movement patterns for accurate

speech.

In this parent study, children participated in an intensive 4-week virtual summer camp

program, during which they received 16 hours of individual ASSIST in 32 sessions of 30

minutes each. Children were randomly assigned to receive all their individual treatment in the

first two weeks (immediate massed), the second two weeks (delayed massed), or divided over all

four weeks (distributed) (see Figure 1 for an overview of the design of the parent study). Group

activities and individual ASSIST were delivered via videoconference. Before, at midpoint, and

after the 4-week period, children attended individual data collection sessions in person in the

Speech, Language, and Brain Lab at Temple University. Details and results from the parent

clinical trial are not further reported here, except insofar as relevant to the present study on child-

reported outcome measures (CAT and KiddyCAT), which were administered at T2 and T3. All

study procedures were approved by the Temple University IRB (protocol #25807) and informed

consent was obtained from at least one parent and all children provided assent.
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Participants

Twelve children ages 4-10 years old with a primary diagnosis of CAS participated in this

study. Three expert speech-language pathologists (SLPs) provided independent ratings of

presence of CAS, using a 3-point scale where 0 = no CAS, 1 = possible CAS, and 2 = CAS.

These ratings were based on the presence of current consensus features of CAS (ASHA, 2007),

including inconsistent errors on consonants and vowels, difficulties achieving or transitioning

into articulatory configurations, and abnormal prosody such as lexical stress errors. At least one

SLP made this judgment on the basis of the live assessment sessions, whereas the remaining

SLPs made their judgments from video-recordings of the assessment sessions. To be included in

the study, children were required to receive an average rating of >1 across the three expert SLPs

(M=1.69) and an Apraxia score of 1 or 2 on a Maximum Performance protocol (Thoonen et al.,

1996, 1999), the only prospectively validated protocol to date with acceptable sensitivity and

specificity.

Inclusion criteria were (a) English as primary home language per parent report, (b)

normal hearing as determined by parent report and/or pure tone audiometric screening, (c)

nonverbal cognition in the typical range based on the Reynolds Intellectual Assessment Scales

(Reynolds & Kamphaus, 2003), and (d) verbal output of 50+ words and communicative intent,

per SLP and parent report. Exclusionary criteria were (a) co-occurring neurobehavioral diagnosis

(e.g., autism) per parent report, (b) significant visual impairment per parent report, (c) primary
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speech diagnosis of dysarthria based on SLP judgment, (d) structural-anatomical abnormalities

of the oral structures (e.g., cleft palate) based on parent report and SLP judgment from an oral

mechanism evaluation, and (e) inability to operate or attend to virtual sessions, per parent and

SLP judgment.

A battery of tests were administered prior to allocation to groups to determine eligibility

and to characterize participant profiles. These are reported in Table 1, but most of these will not

be discussed in detail here, except for the Dynamic Evaluation of Motor Speech Skill (DEMSS;

Strand & McCauley, 2019), as the DEMSS score represents an index of CAS severity that will

be used in the subsequent analyses. The DEMSS is a criterion-referenced tool designed to assess

children, ages 3 and older, with moderate to severe speech impairment, prosodic or vowel errors,

poor speech intelligibility, or who have minimal or no verbal communication skills (Strand &

McCauley, 2019). The assessment can confirm or rule out a CAS diagnosis, be an estimate of

severity and prognosis, and/or demonstrate the effectiveness of various cues. Administration

generally takes less than 30 minutes. The test requires the child to imitate an SLP across 60

utterances. The child is given additional support and cues if the initial attempt is incorrect.

Possible scores range from 0 to 426, where lower scores indicate greater severity. Scores

between 0 and 323 indicate significant evidence for CAS, scores from 324 to 373 indicate some

evidence of at least mild CAS, and scores from 374 to 426 reflect little to no evidence for CAS

(Strand & McCauley, 2019).

Participants were recruited through local community referrals, existing databases of

individuals who expressed prior interest in research opportunities, community outreach and

education events, and advertisements in newsletters and websites. Of the 16 children assessed for
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eligibility, 12 children met the selection criteria, including 9 boys and 3 girls (see Figure 1 and

Table 1).
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Table 1. Participant information.

Chil Ag Se Cond CAS DEM GFT GFT DEAPf DEAP RIA RIAS EVT PPV CELF
d ea x .b ratin SS Ae Ae SS Phon. f S NV i SS Tj SS k CLS

gc scored raw SS Incons NIX Memor

. g yh
301 6;0 M D 1.00 383 43 63 60 32% 50 50 82 96 75
306 4;2 M D 2.00 215 107 49 65 80% 83 63 117 93 NAl
309 7;1 M D 1.33 298 73 40 55 68% 42 64 81 85 66
0
310 4;1 M D 2.00 DNCm 131 40 CNCn 72% 66 75 83 71 NAl
314 10; M D 2.00 401 22 59 NAl 40% 40 38 80 95 72
0
322 8;3 F D 1.00 395 10 70 105 36% 42 61 87 86 93

304 5;5 M M1 2.00 295 40 72 75 44% 78 56 98 90 93

312 5;9 F M1 1.67 143 109 40 55 64% 47 81 81 76 79
321 6;4 M M1 2.00 262 83 40 55 56% 43 49 89 94 93

305 7;0 F M2 2.00 382 70 40 55 44% 46 49 86 78 75

m
313 4;1 M M2 2.00 DNC 78 49 65 84% 67 59 96 86 NAl
1
324 5;2 M M2 1.33 357 76 47 65 52% 53 66 98 118 100
a
Age at T2 in years; months
b
Condition: D = Distributed; M1 = Massed Immediate (phase 1); M2 = Massed Delayed (phase 2)
c
Mean of 3 expert speech-language pathologists’ independent ratings regarding presence of CAS (0 = no CAS; 1 = possible CAS; 2 =
CAS)
d
Score on the Dynamic Evaluation of Motor Speech Skill (Strand & McCauley, 2019)
e
Raw score and Standard Score (SS) on the Goldman-Fristoe Test of Articulation, 3rd Edition (Goldman & Fristoe, 2015)
f
Diagnostic Evaluation of Articulation and Phonology (Dodd et al., 2006) phonology standard score and inconsistency percentage.
g
Nonverbal Intelligence Quotient (NIX) T-score based on the Reynolds Intellectual Assessment Scales (Reynolds & Kamphaus,
2003)
h
Nonverbal Memory T-score based on the Reynolds Intellectual Assessment Scales (Reynolds & Kamphaus, 2003)
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i
Standard score (SS) on the Expressive Vocabulary Test, 3rd Edition (Williams, 2019)
j
Standard score (SS) on the Peabody Picture Vocabulary Test, 5th Edition (Dunn, 2019)
k
Core Language Score (CLS) Standard Score on the Clinical Evaluation of Language Fundamentals, 5th Edition (Wiig et al., 2013)
l
NA = not administered due to age outside normative range
m
DNC = did not complete (no total score available)
n
CNC = could not compute (score outside of normative range)
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Instruments

CAT and KiddyCAT

As described in the Introduction, the CAT and KiddyCAT have been used to provide

insight into how children with a range of speech disorders feel about their speech. The CAT is

designed for children 6 and older and consists of 33 true/false statements to be answered by the

child. The KiddyCAT is designed for children between 3 and 6 years old and consists of 12

yes/no questions to be answered by the child. Both the CAT and KiddyCAT include

statements/questions where an approximately even number of true and false (or yes and no)

responses indicate negative self-perceptions. Each answer that indicates a negative self-

perception is awarded one point. Thus, higher scores on both assessments indicate more negative

speech perceptions. The average KiddyCAT score for children who do not stutter is 1.79

(SD=1.78) and the average CAT score for children who do not stutter is 6.38 (SD=5.21). Per

each manual, a score of 5 or higher on the KiddyCAT or a score of 17 or higher on the CAT is

indicative of a negative speech perception. However, clinicians and researchers are also advised

to consider and investigate slightly lower scores on both assessments during analyses.

Focus on Outcomes of Communication Under Six (FOCUS)-34

The Focus on Outcomes of Communication Under Six (FOCUS-34; Oddson et al., 2019;

Washington et al., 2012) is a parent-rated 34-question assessment of child participation across

various communicative contexts. It consists of two parts: Part 1 asks about how well a statement

applies to the child, and Part 2 asks about the amount of help a child needs to accomplish

communicative tasks. Example items from Part 1 include “My child is confident communicating

with adults who do not know my child well” and “My child uses words to ask for things”. Each
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item is scored from 1 (not at all like my child) to 7 (exactly like my child). Example items from

Part 2 include “My child is included in games by other children” and “My child joins in

conversations with other children”. Items are rated from 1 (cannot do at all) to 7 (can always do

without help). The FOCUS-34 takes approximately ten minutes to complete.

All participants’ parents in the current study, regardless of age, were given this form at

each time point. While this assessment was designed and normed for children under age six,

there is no significant reason to question its validity for older children, and there is precedent for

use of the FOCUS-34 with children older than six (e.g., Rusiewicz et al., 2018). The same parent

completed the form at each time point.

Frustration Ratings

As part of the parent study’s safety protocol, potential negative side effects of treatment

were monitored, with a particular emphasis on child frustration. The treating clinician provided a

numerical score reflecting the child’s frustration level, based on their judgment informed by their

knowledge of the child and the child’s behavior in the session. Because frustration may be

expressed differently by different children (e.g., some children may cry, others may refuse to

continue, others may display avoidance behaviors), judgment by the treating clinician was

deemed to be an adequate measure to capture these potentially different expressions. Possible

scores were 0 (no frustration, full compliance with procedures throughout the session), 1 (some

frustration, occasional noncompliance during session), 2 (significant frustration, frequent

noncompliance during session), and 3 (marked frustration, noncompliance during entire session).
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Procedures

At the end of T2 and T3 data collection sessions, trained research assistants, blinded to

treatment status, administered the CAT (for children 6;0 and older; n=6) or KiddyCAT (for

children 4;0-5;11; n=6). Per the CAT and KiddyCAT manual, children were instructed that there

were no right or wrong answers because the questions were about what they thought about their

own speech. Before administration, two practice questions were given to establish adequate

understanding of the task. Prior to T2 administration, any child given the KiddyCAT and deemed

to have a questionable understanding of the concepts ‘easy’ and ‘difficult’ was provided two jars

and asked to distinguish which was easy to open and which was hard to open (n=1). The child

had no trouble understanding this concept.

Each question was read aloud by research assistants for both the CAT and KiddyCAT.

Participants were required to respond true/false for the CAT and yes/no for the KiddyCAT. For

the CAT, right/wrong and yes/no were also deemed acceptable responses in place of true/false.

Administration took approximately 5-15 minutes for the KiddyCAT and 15-30 minutes for the

CAT. All sessions were audio- and video-recorded for subsequent analysis and reliability check.

Reliability

CAT and KiddyCAT questionnaires were scored independently by a second scorer (who

was not blinded to condition) from session recordings. Each answer was screened for adequate

understanding. A few specific questions were excluded due to missing items; for this reason, all

scores were converted to percentages for analysis. 1 The scores of the second scorer were

1
At T2, two CAT questions were excluded for Participant 309, one CAT question was excluded for Participant 322,
and one KiddyCAT question was excluded for Participant 310. At T3, one CAT question was excluded for
Participant 309 and Participant 314. All analyses were conducted both on raw scores and percentaged scores; the
pattern of results did not differ in any analysis.
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compared with the original forms scored by the examiner who administered the questionnaires 2

to evaluate inter-rater reliability. Inter-rater reliability of total scores was assessed via intraclass

correlation coefficients (ICC) with 95% confidence intervals, calculated with the irr package

(Gamer et al., 2012) in R software (R Studio version 1.2.5042), based on a single-measures,

absolute agreement, two-way mixed effects model (McGraw & Wong, 1996a, 1996b). Resulting

ICCs were in the good to excellent range (Koo & Li, 2016) for all four comparisons (CAT T2,

CAT T3, KiddyCAT T2, KiddyCAT T3; see Table 2 below).

Discrepancies between scorers were reviewed and resolved by a third independent scorer

and were attributed to human error by the administrator. For all subsequent analyses, scores from

the second scorer were used because this scorer had opportunity to re-watch the video to increase

confidence in the child’s response.

Table 2. Interrater reliability across tests and timepoints based on total score converted to
percentage. ICC = intraclass correlation coefficient; CI = confidence interval [lower bound,
upper bound]. Interpretation of ICC values based on Koo and Li (2016).

Test T2 T3

ICC 95% CI Interpretation ICC 95% CI Interpretation

KiddyCAT 0.836 [0.304, 0.975] good 0.886 [0.339, 0.987] good

CAT 0.896 [0.397, 0.985] good 0.995 [0.962, 0.999] excellent

Analyses

The research questions were addressed as follows. For all inferential statistical analyses,

an alpha level of 0.05 was adopted to determine statistical significance.

2
During T2, the 35-question form of the CAT was administered to 3 participants. The updated 33- question version
was administered to all participants at T3. The additional two questions from the 35-question form were not
considered in analyses. The KiddyCAT form was consistent across both timepoints.
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Research Question 1 (comparison with normative values) was addressed descriptively.

Specifically, individual CAT and KiddyCAT total scores (percentages) at each timepoint as well

as their means across the two timepoints were compared to their respective normative values

(also converted to percentages for this comparison), and children whose scores were more than 1

standard deviation from the normative mean were identified.

For Research Question 2 (relation with CAS severity), CAT and KiddyCAT scores at

each timepoint and their means across timepoints were correlated with the DEMSS total score

using non-parametric Spearman correlations, given the small sample sizes. Note that more severe

CAS is reflected by lower DEMSS scores. Thus, if more severe CAS is associated with more

negative self-perceptions, we expect a negative correlation, because more negative self-

perceptions are reflected by higher CAT/KiddyCAT scores.

For Research Question 3 (relation with parent perception of communicative

participation), CAT and KiddyCAT scores at each timepoint were correlated with the FOCUS-34

total scores from those same timepoints using non-parametric Spearman correlations. If children

with more negative self-perceptions are less likely to participate in communicative activities (as

judged by their parent), then we expect a negative correlation, because higher FOCUS-34 scores

indicate greater communicative participation.

For Research Question 4 (relation with frustration during treatment), CAT and

KiddyCAT scores at each timepoint as well as their means across timepoints were correlated

with the mean frustration rating across all treatment sessions and with the percentage of sessions

during which children received a frustration rating of 3 (marked frustration). If children with

more negative self-perceptions are more prone to frustration, we expect a positive correlation.
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For Research Question 5 (change following brief intervention period), descriptive

analysis was used given the small and unequal sample sizes in the different treatment conditions.

The number of children whose scores increased, remained the same, and decreased numerically

were identified.

RESULTS

Results are described below by research question. Within each research question, CAT

and KiddyCAT results are described separately. For the KiddyCAT, one participant (P306) did

not return for T3 testing; thus, analyses involving T3 are based on data from the remaining five

children in this age range.

Research Question 1: Comparison to Normative Values

Figure 2: KiddyCAT Scores

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Figure 3: CAT Scores

Among the younger children (KiddyCAT), one child fell above the normative range at

T2, but none did at T3. One child (P324) fell below the normative range at both timepoints. 3

Among the older children (CAT), 3 out of 6 children had higher (i.e., more negative) scores

compared to the normative sample at T2, and 5 out of 6 children at T3; one child showed a lower

(i.e., more positive) score at T3 compared to the normative sample.

Per the CAT and KiddyCAT manual, participants’ scores on these assessments were also

compared to +/- 2 standard deviations from the normative range. For the KiddyCAT, no child

fell outside of 2 standard deviations of the mean (a score of 5 or above) at either timepoint. For

the CAT, one participant at T2 (P301) and one participant at T3 (P305) fell above 2 standard

deviations of the normative range (a score of 17 or above).

3
P324 scored 0 at both timepoints, whereas P306, who did not return for testing, does not have a score at T3.
 24

Research Question 2: Relationship with CAS Severity

Figure 4: Comparison to DEMSS Scores

KiddyCAT% x DEMSS: T2 & T3

For the CAT, correlations with baseline DEMSS scores were negative but none were

significant (T2: rho = -0.486, p = 0.329; T3: rho = -0.261, p = 0.618; mean: rho = -0.486, p =

0.329). For the KiddyCAT, correlations with baseline DEMSS scores were negative and also

significant (rho = -1.000, p < 0.0001, for T2, T3, and mean). It should be noted that these

correlations are based on small sample sizes (n = 4 for T2, n = 3 for T3 and mean) due to missing

T3 data for Child 306 and incomplete DEMSS scores for two other children.

Research Question 3: Relationship with Parent-Rated Communicative Participation

For the CAT, a moderate negative correlation was observed at T2, but this correlation

was not significant (rho = -0.657, p = 0.156). At T3, a small and nonsignificant negative

correlation was observed (rho = -0.232, p = 0.658).

 25

For the KiddyCAT, analysis revealed a small but nonsignificant positive correlation at T2

(rho = 0.176, p = 0.738) and a small nonsignificant negative correlation at T3 (rho = -0.103, p =

0.870).

Research Question 4: Relationship with Frustration Level during Treatment

For the CAT, small to moderate positive correlations were obtained with mean frustration

scores; however, none of these correlations were significant (T2: rho = 0.143, p = 0.787; T3:

0.406, p = 0.425; mean: 0.543, p = 0.266). However, when considering the proportion of

treatment sessions with a frustration level of 3, there was a strong and significant positive

correlation with CAT scores at T3 (rho = 0.857, p = 0.029). Correlations with T2 and average

scores were not significant (T2: rho = 0.068, p = 0.899; mean: rho = 0.541, p = 0.269).

For the KiddyCAT, small to moderate positive correlations were observed with mean

frustration ratings, but none of these were significant (T2: rho = 0.319, p = 0.538; T3: rho =

0.359, p = 0.553; mean: 0.400, p = 0.505). Similar to the CAT analyses, when considering the

proportion of sessions with a frustration level rating of 3, a strong positive correlation was

obtained at T3, although this failed to reach significance (rho = 0.860, p = 0.061). Correlations

with KiddyCAT scores at T2 and the mean across timepoints were moderate to strong but were

also nonsignificant (T2: rho = 0.678, p = 0.139; mean: rho = 0.783, p = 0.118).
 26

Research Question 5: Change following a Brief Intervention Period

Figure 5: KiddyCAT Scores across timepoints

Figure 6: CAT Scores across timepoints

For younger children (KiddyCAT), the majority of scores remain the same across

timepoints. One score in the massed-immediate group decreased from T2 to T3 (P312). One

child who completed the KiddyCAT was assigned to the distributed group; that participant’s

score slightly decreased between timepoints (P310).

For the older children (CAT), all scores varied from T2 to T3. In the distributed group,

scores of three participants decreased (P301, P309, P314) and one participant’s score increased
 27

(P322). Only one child who completed the CAT was assigned to the massed-immediate group;

that participant’s score increased slightly from T2 to T3 (P321). One child who completed the

CAT was assigned to the massed-delayed group; that participant’s score showed the greatest

increase from T2 to T3 (P305, 27% to 52%).

DISCUSSION

This study was the first to examine self-reported attitudes from children with CAS about

their speech, by administering the CAT and KiddyCAT to a group of children with CAS. By

relating scores on these assessments to severity of diagnoses, parents’ perceptions of

participation across contexts, and average frustration ratings during therapy, a more

comprehensive understanding of how-to best support children with CAS is gained. Because of

the novelty of this research, scores that fell outside one standard deviation of the normative range

were identified as possible negative attitudes.

Research Question 1: Communication Attitudes of Preschool Children with CAS

As a group, young children with CAS do not appear to feel negatively about their speech.

This is on par with previous literature regarding the communication attitudes of young children

who stutter (Guttormsen et al., 2015) and who have phonological disorder (McCormack,

McLeod, & Crowe, 2019). Though preliminary, this finding is reassuring. Differences in type

and consistency of impairment were expected to translate to negative attitudes; yet 5 out of 6

participants at T2 and 5 out of 5 participants at T3 indicated feeling more positive than negative

about their communication. It is worth noting that one child’s score at T2 (P312) fell above one
 28

standard deviation of the normative range. At T3 the child’s score was within norms. Alone, the

T2 score is not indicative of a negative speech attitude but warrants further investigation.

There were several KiddyCAT questions where all answers indicated positive speech

attitudes. At T2 and T3, all participants answered yes to the question (Q2), “Do you think you

talk right?” It is possible that this relates to age; younger children may not fully understand their

diagnosis or recognize the reason for going to therapy. Regardless, the consistency of this answer

across timepoints is helpful for researchers, clinicians, and families to know. Participation in

intensive speech therapy did not seem to affect young children’s perception of the ‘rightness’ of

their speech.

Questions commonly answered at individual timepoints were also identified. At T2, all

children answered no to the question (Q4) “Do you think people need to help you talk?”, which

is a further indication that participation in speech therapy did not influence young children’s

concerns about their speech. At T3, all participants answered yes to Q3, “Do mom and dad like

how you talk?”, indicating that children were not equating parental concern regarding speech

(i.e., putting the child in speech therapy) to their parents disliking how they talk. Additionally, all

children answered yes to Q7, “Do you talk well with everybody?” This question relates to

children’s participation levels across contexts and indicates that young children may not feel

their diagnosis impacts their ability to communicate with different people. The final two

commonly answered questions both relate to if children think speaking is “difficult” (Q8) and

whether words are “hard” to say (Q11). Of the KiddyCAT questions, Q8 and Q11 are the most

demonstrative of awareness, or lack thereof, of speech difficulties. By the end of treatment all

younger children did not think “talking is difficult” or that “words are hard for them to say.”
 29

Communication Attitudes of School-Age Children with CAS

Scores of school-age children with CAS are suggestive of more negative communication

attitudes as compared to normative values and compared to the younger children. Half of the

children who completed the CAT scored more than one standard deviation above the normative

range at both timepoints. Two additional children fell above the normative range at T3. Prior

research on children with cleft palates (Havstam et al., 2011), children who have articulation

disorder (Luc & Brutten, 1990), and children who stutter (Vanryckeghem & Brutten, 2007) has

assumed that older children become more aware of speech differences and individual abilities

once they enter school, and therefore develop more negative speech attitudes. While this may be

the case for children with CAS, it is also worth considering that differences in impairment and

treatment may be influencing factors. CAS impairments are unpredictable, which can make

children more difficult to understand, interfere with normal conversations, and make the disorder

hard to treat.

There were several CAT questions where all answers indicated positive speech attitudes.

At both timepoints, all children responded false to Q1 “I don’t talk right,” and true to Q7 “I like

the way I talk.” At T2, all children answered false to Q27 “I am not a good talker” and Q28 “I

wish I could talk like other children.” These answers indicate that when asked directly, school-

age children with CAS feel like there is nothing wrong with how they speak. At T2, 5 of the 6

children answered true to Q9, “My parents like the way I talk” and at T3 all six children

responded true to that question. This parallels with younger children’s responses to the question,

“Do your parents like how you talk?” and is an assuring sign that both older and younger

children do not feel their parents think negatively of their speech because of their diagnosis.
 30

Q18: “Other kids wish they could talk like me,” was the only question in which all

question answered false, indicating a negative speech attitude. This question will be further

discussed in following sections regarding its applicability to overall attitudes.

Research Question 2: CAS Severity and Speech Attitudes

When CAT and KiddyCAT assessments were related to assessments of severity, findings

were mixed. No significant correlation was found between the speech attitudes for school-age

children with CAS and their DEMSS score. Increasing severity of diagnoses was expected to be

associated with negative speech attitudes. For the KiddyCAT, a significant negative correlation

was identified, meaning that preschool age children with CAS’s attitudes about their speech are

correlated with the severity of diagnoses. This finding, though preliminary, is critical. Clinicians

and families informed of a possible correlation between severity of CAS and negative speech

perceptions will be better able to provide necessary support for the child. For one child (P312),

this correlation helps contextualize a borderline KiddyCAT score. This participant scored just

above one standard deviation of the normative range at T2, and on the DEMSS, had significant

evidence of CAS (a score of 323 or less).

Research Question 3: Caregiver Perceptions of Participation and Communication Attitudes

Though designed to assess Level (2) of the ICF: life participation, the FOCUS-34 is often

used as a proxy for understanding the influence of communication disorders on Level (3) of the

ICF: personal factors. Identifying if this holds true for children with CAS was of great interest. In

this study, caregiver perceptions of participation across contexts were not significantly correlated

with children’s attitudes about their speech. Low statistical power due to a small sample size may
 31

have occluded the detection of a significant correlation between these assessments. If this pattern

remains true with a larger sample size, it will indicate that these assessments measure two

distinct constructs - meaning parents’ perceptions of participation across contexts are not

accurate indicators of how children with CAS feel about their speech. It is worth noting that a

study of young children with phonological disorder also found no significant correlation of

parental perception of participation across contexts and children’s communication attitudes

(McCormack, McLeod, & Crowe, 2019). In conjunction, both findings suggest caregiver ratings

of child participation are not adequate substitutes for information gained from self-report

assessments of communication attitudes.

Research Question 4: Frustration During Therapy and Communication Attitudes

Higher average frustration ratings during therapy for both preschool and school-age

children with CAS did not correlate with negative speech perceptions. However, at T3, the

number of sessions in which school-age children were clearly frustrated and noncompliant (a

score of 3) did correlate with negative communication attitudes. Marked frustration during

sessions could have been due to several reasons (i.e., general unhappiness, tiredness, boredom,

stress, etc.). Higher levels of frustration during treatment could have exacerbated negative

attitudes about speech abilities. Due to the novelty and intensity of this treatment approach, this

is an important consideration. Given the small sample size and lack of findings at T2 or for

average scores, more research is needed before drawing further conclusions.

 32

Research Question 5: Change in Scores over a Brief Intervention Period

When younger children’s scores were compared across timepoints, no trends emerged.

KiddyCAT scores remained the same at T2 and T3 for the majority of participants. Similarly,

when treatment groups were compared, no trends emerged. Whether the child received treatment

in the first two weeks, last two weeks, or throughout all four weeks of the study did not impact

communication attitudes for younger children. This implies that the ASSIST protocol and camp

format did not negatively or positively change how young children viewed their speech.

For older children with CAS, there was more variation in scores across timepoints and

treatment groups. Scores of children in the massed-immediate and massed-delayed groups

increased from T2 to T3. In particular, the score of the singular child in the massed-delayed

group increased substantially between timepoints. This may mean that the intensive treatment

that this child received between timepoints heightened awareness of any communication

differences. Scores of those who received distributed treatment both increased and decreased,

signifying that intensity of treatment was not a good predictor of changes in communication

attitudes. Further research is needed before determining if this treatment protocol impacts how

children view their speech.

Limitations

As the first investigation into communication attitudes as self-reported by children with

CAS, there were a number of limitations to this study. First, this study had a small sample size;

the CAT and KiddyCAT were only administered to a total of 12 children with CAS. This

increased the risk of Type II errors due to low statistical power. Second, only children with CAS

were included in this study, meaning that scores were only compared to established norms and
 33

not current peers. Third, no communication attitudes assessments were administered at the first

data collection timepoint (T1); scores may have already been influenced by the first two weeks

of camp attendance. Finally, administrative errors required that some questions be excluded from

analyses, which is a deviation from typical use of these assessments.

Future directions

Future research should include a larger sample size of children with and without CAS.

Should these assessments be incorporated in future treatment studies for children with CAS, they

should be administered at all data collection timepoints. On future distributions of the CAT and

KiddyCAT, all questions should be included during analysis.

Wording on specific CAT questions should be reconsidered for future studies. During test

administration, participants had difficulty understanding the meaning of Q2: “I don’t mind

asking a teacher the question in class.” For young children, using ambiguous language like “I

don’t mind” is not ideal. For example, one participant answered false, I don’t mind, and another

participant responded true, I don’t mind, but upon additional cueing, both meant that they were

had no reservations about asking the teacher a question in class. Future revisions should consider

changing ambiguous language like “I don’t mind…” to wording more suitable for young

children like “I’m okay with….” The connotation remains the same, which means that scoring

does not change.

Question 18 on the CAT, “Other kids wish they could talk like me”, has questionable

translation to overall speech attitudes. In this study, all school-age children with CAS indicated

that they did not think that other children wanted to talk like they do. Thinking that others do not

want to talk like them, however, does not necessarily denote a negative speech attitude. If future
 34

research distributes the CAT to children with and without CAS, it would be of interest to

understand how typically developing children respond to this question and the underlying

reasons for those responses.

It is also of interest to understand why this test requires true/false responses. Though

school-age children can understand true and false statements, it would be clearer for the CAT to

adopt yes/no statements, like the KiddyCAT. In this study, children often responded with yes/no

or right/wrong despite being directed to respond true/false. This is a particularly important

consideration when discussing future administration of the CAT to larger samples of children

with CAS, as many of those children are at in increased risk for comorbid language disorders.

Future studies should also consider including a qualitative component. Several

participants volunteered comments regarding their speech perceptions during data collection that

provided context that quantitative assessments cannot provide. For instance, one participant

(P314) made various remarks regarding his speaking improvements since the beginning of camp

including “I’m working on [speaking] and getting better at it” and remarked that reading out loud

in class “was hard but now it’s easy.” Incorporating a qualitative component in future studies of

children with CAS will help provide reasons as to why children feel the way they do.

Given that several problems arose when the KiddyCAT and CAT were administered to

children with CAS, future research would benefit from the development of a new self-report

speech attitudes measure designed for children with SSDs. This measure should include

questions better geared towards SSDs and be adaptable for administration to children with

comorbid disorders.
 35

CONCLUSION

Because of this study, researchers and clinicians can begin to understand how children with

CAS feel about their speech. Understanding communication attitudes is critical - for many

children, living with CAS means more than disrupted intelligibility or poor sound accuracy.

Negative attitudes may impact interactions with peers, participation in school activities, or

progress in treatment. When given the opportunity, children with CAS can provide credible,

meaningful perspectives. In this study, scores of more than half of school-age children with CAS

were indicative of negative speech attitudes. Though preliminary, these findings have clinical

and familial implications. A speech-language pathologist who identifies a child with CAS to

have negative speech attitudes may more adeptly create relevant treatment goals. Families who

are aware of their child’s attitudes will be better able to provide psychological support. Including

communication attitude measures in future studies is a critical step towards understanding how to

best treat and support children with CAS.

 36

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