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Research Article

Feedback Frequency in Treatment for


Childhood Apraxia of Speech
Edwin Maas,a Christine E. Butalla,a,b and Kimberly A. Farinellaa,c

Purpose: To examine the role of feedback frequency in Results: Findings were mixed, with 2 children showing an
treatment for childhood apraxia of speech (CAS). Reducing advantage for low-frequency feedback, 1 child showing a
the frequency of feedback enhances motor learning, and small advantage for high-frequency feedback, and 1 child
recently, such feedback frequency reductions have been showing no clear improvement in either condition.
recommended for the treatment of CAS. However, no published Conclusion: These findings suggest that reducing the
studies have explicitly compared different feedback frequencies frequency of feedback may be beneficial for some children
in this population. with CAS, although this may vary with the child’s age or
Method: Using an alternating treatments single-subject severity of apraxia. Caution is warranted in extrapolating from
design with multiple baselines across behaviors, retention and the nonspeech motor learning literature to speech treatment
transfer of learning were compared following high-frequency for CAS. Finally, this study contributes another replication
feedback and low-frequency feedback in 4 children with CAS. to the literature on the efficacy of integral stimulation treatment
Feedback frequency was manipulated in the context of an for children with CAS.
integral stimulation treatment. Changes in perceptual accuracy
were quantified with effect sizes and were compared across Key Words: childhood apraxia of speech, treatment,
conditions. motor learning, feedback, speech disorders

L
earning a new motor skill or improving an existing we manipulated the amount of feedback provided during
motor skill typically requires feedback regarding the treatment and examined the effects on retention and transfer
accuracy and performance of the movement during of learning.
practice. Such feedback enables the learner to determine
which aspects of the movement were successful, which were
Speech Motor Learning in CAS
not, and how the motor plan can be modified for success on
a future attempt. For children with childhood apraxia of The current consensus in the literature is that CAS re-
speech (CAS), learning the movements of speech is thought flects an impairment of speech motor programming and
to be particularly difficult (e.g., American Speech-Language- planning, which is evidenced by difficulties moving from
Hearing Association [ASHA], 2007). As such, feedback one speech sound to the next, inconsistent errors on vowels
may be especially critical for children with CAS, and a close and consonants, and prosodic problems (e.g., ASHA, 2007;
examination of different aspects of feedback implemented Grigos & Kolenda, 2010; Nijland et al., 2002; Skinder,
during treatment may contribute to improving speech motor Strand, & Mignerey, 1999; Terband, Maassen, van Lieshout,
learning in this population. The present study was part of & Nijland, 2011). In addition, it has been suggested that
a larger research program that systematically investigated children with CAS also have difficulties with speech motor
the effects of various conditions of practice in treatment for learning, based on their often noted slow, limited prog-
CAS (e.g., Maas & Farinella, 2012). In the current study, ress in treatment (e.g., Ballard, Robin, McCabe, & McDonald,
2010; Campbell, 1999; Shriberg, Aram, & Kwiatkowski,
1997; Strand, Stoeckel, & Baas, 2006) and on recent com-
a putational modeling simulations (e.g., Terband & Maassen,
University of Arizona, Tucson
b 2010).
Panacea Therapeutics, Tucson, AZ
c Although there are few published controlled treatment
Northern Arizona University, Flagstaff
efficacy studies that focus on CAS, the clinical observations
Correspondence to Edwin Maas: emaas@arizona.edu of slow and limited progress in treatment have motivated a
Editor: Carol Scheffner Hammer search for ways to optimize speech motor learning in chil-
Associate Editor: Ken Bleile dren with CAS. The literature on motor skill learning pro-
Received August 31, 2011 vides some potentially relevant ideas in this regard, in that
Revision received January 9, 2012 some conditions of practice and feedback have been found
Accepted March 9, 2012 optimal in promoting motor learning (sometimes referred to
DOI: 10.1044/1058-0360(2012/11-0119) as “principles of motor learning”; see Maas et al., 2008, for

American Journal of Speech-Language Pathology • Vol. 21 • 239–257 • August 2012 • A American Speech-Language-Hearing Association 239
review). Various authors have advocated the use of these items was negligible in all cases. These findings suggest that
conditions of practice and feedback in CAS treatment (e.g., although practice factors derived from the motor learning
Hall, 2000; Iuzzini & Forrest, 2010; McCauley & Strand, literature may influence speech motor learning in children
1999; Robin, Maas, Sandberg, & Schmidt, 2007), and sev- with CAS, the direction of those influences may be different.
eral studies have incorporated these conditions into CAS In other words, caution is warranted in extrapolating practice
treatment (e.g., Ballard et al., 2010; Edeal & Gildersleeve- principles from the motor learning literature to CAS treatment.
Neumann, 2011; Iuzzini & Forrest, 2010; Maas & Farinella, Thus, although the motor learning literature can provide
2012; Strand & Debertine, 2000; Strand, Stoeckel, & Baas, guidance for efforts to improve speech motor learning
2006). However, only two studies to date have explicitly outcomes in children with CAS, the few direct empirical
compared the effects of different practice conditions in CAS investigations of various practice factors in this population
treatment to determine whether the same advantages apply have yielded somewhat mixed results. As such, the con-
to this population (Edeal & Gildersleeve-Neumann, 2011; tinued direct study of various practice factors in treatment
Maas & Farinella, 2012). for CAS is necessary to determine optimal practice con-
Edeal and Gildersleeve-Neumann (2011) examined the ditions for children with CAS. The present study represents
role of practice amount, arguably the most powerful factor in a continuation along these lines by examining the role of
learning (Schmidt & Lee, 2005), in an integral stimulation feedback frequency in CAS treatment.
treatment for two children with CAS. Integral stimulation
treatment was originally developed for adults with apraxia of
speech (Rosenbek, Lemme, Ahern, Harris, & Wertz, 1973) Feedback Frequency Effects in Motor Learning
and involves auditory and visual models provided by the Models of motor control and learning assume that pro-
clinician (“listen to me, watch me, and say what I say”) and duction of a skilled movement involves exploiting knowl-
principles of motor learning (see Edeal & Gildersleeve- edge of the relations between various sources of information
Neumann, 2011, and Strand & Skinder, 1999, for excellent about a movement, such as motor commands, sensory con-
reviews of the integral stimulation treatment literature for sequences of the movement, and movement outcome (e.g.,
CAS). Edeal and Gildersleeve-Neumann defined practice Bullock, Grossberg, & Guenther, 1993; Schmidt, 1975;
amount in terms of the number of practice trials and used an Willingham, 1998; Wolpert, Ghahramani, & Flanagan,
alternating treatments single-subject design in which differ- 2001). Feedback is considered critical to allow the learner
ent sound targets were assigned to different practice con- to determine these relations and figure out which combina-
ditions (100–150 vs. 30–40 trials per session). They observed tions of motor commands produce the desired outcome in
improvements in speech sound accuracy for both practice a given situation. Intrinsic feedback refers to information
conditions, but retention and transfer were greater for speech about a movement that is inherent in performance of the
sounds that were practiced 100–150 times per session than motor act. For example, in throwing a dart, the visual system
for speech sounds that were practiced 30–40 times per ses- provides information about where the dart landed, and the
sion. These findings are consistent with the motor learning somatosensory system provides information about the grip
literature (e.g., Shea, Kohl, & Indermill, 1990; see Maas force and arm excursion used. In many situations, learners
et al., 2008, for review). can use intrinsic feedback to improve their performance, but
In an integral stimulation treatment with four children there are situations in which intrinsic feedback is not suf-
with CAS, Maas and Farinella (2012) investigated another ficient or optimal. For example, a learner may not have
promising variable, namely, practice schedule. Practice access to all relevant intrinsic information (e.g., in learning a
schedule refers to the order in which different targets are novel dance routine, the learner may not have access to, or be
practiced, and a random practice schedule has been found to able to focus on, information about arm movements when
enhance motor learning compared to a blocked practice focusing on foot movements), or a learner may not have a
schedule (in which different targets are practiced in separate frame of reference that is calibrated to some standard of
blocks). Maas and Farinella compared random and blocked correctness (e.g., the learner may not know how to interpret
practice in an alternating treatments design with multiple the scores on a dart board). In such situations, externally
baselines across behaviors. Treatment was provided in two provided (augmented) feedback from an instructor may be
phases, each lasting 4 weeks (È12 hr of treatment per phase) necessary to improve the skill (Wulf & Shea, 2004).
and separated by a 2-week maintenance phase following Considerable research effort has been devoted to finding
each treatment period (same design as present study). Indi- ways to optimize augmented feedback in the learning of
vidually tailored treatment targets and probe items were motor skills (see Maas et al., 2008; Wulf & Shea, 2004,
selected for each child depending on his or her particular for reviews). One feedback variable known to affect motor
difficulties, and treatment involved the dynamic temporal learning is feedback frequency, which refers to how often
and tactile cuing (DTTC) approach (Strand, Stoeckel, & learners receive augmented feedback on their motor perfor-
Baas, 2006). The findings were mixed: Three of the four mance during practice. A typical finding is that high-frequency
children showed a clear response to treatment (in both feedback (e.g., feedback after every practice trial) enhances
conditions) and retention following treatment in terms of practice performance but degrades motor learning, as com-
overall utterance accuracy, and two of those demonstrated pared to reduced frequency feedback (e.g., feedback only
a slight net advantage for blocked over random practice, after half of all practice trials) (e.g., Bruechert, Lai, & Shea,
whereas the third child showed a clear and consistent advan- 2003; Nicholson & Schmidt, 1991; Winstein & Schmidt,
tage for random over blocked practice. Transfer to untreated 1990).

240 American Journal of Speech-Language Pathology • Vol. 21 • 239–257 • August 2012


Probably the most influential explanation for the learning Second, feedback frequency effects may differ for dif-
benefit of reduced frequency feedback has been the guidance ferent movement aspects (e.g., Lai & Shea, 1998; Wulf,
hypothesis (Salmoni, Schmidt, & Walter, 1984; cf. Wulf & Schmidt, & Deubel, 1993). For example, Wulf et al. (1993)
Shea, 2004, for a more recent review). According to the reported that reduced frequency feedback enhanced learning
guidance hypothesis, learners come to rely too much on the of underlying movement patterns (i.e., relative timing and
guiding role of feedback when it is present during practice amplitude), whereas high-frequency feedback enhanced
(perhaps even integrating feedback with the task; Schmidt learning of scaling parameters (i.e., absolute timing and
& Bjork, 1992), thereby impeding the development of in- amplitude). Perceptual judgments of speech accuracy do not
ternal response evaluation and error correction mechanisms separate these aspects (Maas et al., 2008), thus potentially
based on intrinsic feedback (cf. Bruechert et al., 2003). masking learning effects (although Austermann Hula et al.,
Reducing the frequency of feedback would encourage the 2008, did report feedback frequency effects based on per-
learner to use intrinsic information to evaluate a response in ceptual judgments).
no-feedback trials and would allow the learner to calibrate Third, and perhaps most relevant to the present study,
this internal judgment of correctness to the external standard there are indications that feedback frequency has different
provided by the instructor. It is this ability to use intrinsic effects in children than in adults (e.g., Chiviacowsky, Wulf,
feedback to evaluate movement performance that presum- Laroque de Medeiros, Kaefer, & Wally, 2008; Sullivan,
ably facilitates ongoing learning after practice ends (analo- Kantak, & Burtner, 2008). For example, Sullivan et al.
gous to the well-known proverb, “give hungry people fish (2008) compared 100% feedback and 62% feedback for an
and they will be fed for a day; teach them how to fish arm movement task and found that children (average age
and they will be fed for a lifetime”). Bruechert et al. (2003) 10 years) in the 100% feedback group performed better on a
showed that participants were better at estimating their errors retention task than children in the 62% feedback group.
after practice with 50% feedback than after practice with
100% feedback. An alternative explanation for the learn-
ing benefit of reduced frequency feedback is the stability The Present Study
hypothesis (e.g., Lai & Shea, 1998; Schmidt & Bjork, 1992), Feedback on accuracy is a critical component of virtually
which states that frequent feedback may result in attempts all behavioral treatment programs in speech-language pa-
at correction of small, relatively insignificant errors, creat- thology, including treatments for CAS. Decisions have to be
ing response instability that prevents the formation of made, either explicitly or implicitly, regarding the frequency
a clear, stable movement pattern. These explanations are of clinician-provided feedback. Currently, there are no
not mutually exclusive, and in fact, the importance of published studies that have systematically studied feedback
response stability was already noted by Salmoni et al. frequency effects in children with CAS. Thus, the main
(1984). research question of the present study was: Does feedback
Investigation of feedback frequency in speech motor frequency affect the retention and transfer of speech motor
learning has been limited (Maas et al., 2008). One study learning in children with CAS?
compared 100% feedback and 60% feedback in treatment for The preceding review of the literature indicates that
four adults with apraxia of speech (Austermann Hula, Robin, reduced frequency feedback appears to enhance learning of
Maas, Ballard, & Schmidt, 2008). Findings suggested that speech and nonspeech motor tasks for adults. However,
reduced frequency feedback produced greater learning recent research suggests that high-frequency feedback may
(evident on transfer and /or retention tests) in two of the be better for learning of nonspeech motor tasks in children.
four participants; data for the other two participants were As such, the expectations regarding feedback frequency
confounded with target-complexity effects and could not be effects in treatment for children with CAS are unclear. If
interpreted relative to feedback frequency. Benefits for re- children with CAS pattern with adults (for speech and non-
ducing feedback frequency were also reported by Steinhauer speech motor learning), then we would expect greater
and Grayhack (2000) in a task requiring speakers to achieve learning with reduced frequency feedback. If, on the other
a particular nasalance target for sustained vowels. Thus, hand, children with CAS pattern with children in nonspeech
initial findings for speech motor learning are promising. motor learning, then we would expect greater learning with
Although reducing the frequency of augmented feedback high-frequency feedback.
appears to enhance motor learning, there are several reasons The main purpose of the present study was to test these
to be cautious in extending these findings to treatment for opposing predictions by comparing the effects of high- and
children with CAS. First, feedback frequency may interact low-frequency feedback on speech motor learning in four
with task complexity. For example, Wulf, Shea, and Matschiner children with CAS. We implemented the feedback frequency
(1998; Wulf & Shea, 2002) showed that learning of complex manipulation in the context of an integral stimulation
motor tasks appeared to benefit from more frequent (i.e. treatment modeled after the DTTC approach (Strand &
100%) feedback compared to 50% feedback. If speech is Debertine, 2000; Strand, Stoeckel, & Baas, 2006) for two
considered a complex motor task, then reducing feedback main reasons. First, DTTC has replicated evidence to support
frequency may be detrimental to learning. However, initial its efficacy, thus making this approach the most likely to
findings from speech motor learning (e.g., Austermann produce gains (minimizing potential floor effects and max-
Hula et al., 2008) argue against this idea because reduced imizing potential gain for participants). Second, this ap-
frequency feedback did appear to enhance learning, at least proach explicitly incorporates principles of motor learning,
for adults. and therefore allowed us to manipulate one factor (feedback

Maas et al.: Feedback Frequency in Treatment for CAS 241


frequency) while retaining the essential ingredients of the in segmental accuracy with reductions in speaking rate. Se-
overall approach as much as possible. verity judgments were based on the expert clinician’s
A secondary purpose of the study was to provide an- perceptual judgment of speech motor control difficulty and
other systematic replication of this integral stimulation treat- intelligibility using a 5-point verbal scale (i.e., mild, mild-
ment (cf. Maas & Farinella, 2012). As noted by Edeal and moderate, moderate, moderate-severe, severe). In addition,
Gildersleeve-Neumann (2011), integral stimulation is cur- the percentage of consonants correct (PCC) and percent-
rently the approach with the most evidence to support its use. age of vowels correct (PVC; Shriberg, Austin, Lewis,
However, there are very few peer-reviewed treatment studies McSweeny, & Wilson, 1997) based on narrow transcription
for CAS overall (ASHA, 2007), and even fewer studies of repetition of lists of words and phrases are provided as
that were primarily based on integral stimulation (Edeal & a more quantified metric of the severity of each child’s
Gildersleeve-Neumann, 2011; Maas & Farinella, 2012; speech impairment.
Strand & Debertine, 2000; Strand, Stoeckel, & Baas, 2006; Formal testing of receptive language was based on the
see Strand & Skinder, 1999, for review). Thus, additional Concepts & Following Directions subtest of the Clinical
replications across participants and research groups are nec- Evaluation of Language Fundamentals, Fourth Edition
essary to establish the generalizability of the efficacy of (CELF–4; Semel, Wiig, & Secord, 2003); the Auditory
integral stimulation treatment (cf. ASHA, 2007); the present Comprehension subtest of the Preschool Language Scale,
study adds to the evidence base in this regard by contributing Fourth Edition (PLS–4; Zimmerman, Steiner, & Pond,
another well-controlled single-subject design study involv- 2002); and /or the Peabody Picture Vocabulary Test, Fourth
ing four participants with CAS. Edition (PPVT–4; Dunn & Dunn, 2007). Expressive lan-
guage was assessed based on core language subtests
(Word Structure, Recalling Sentences, and Formulated Sen-
Method tences) of the CELF–4 (Semel et al., 2003) and language
sampling.
Participants Children ranged in age from 5;4 (years;months) to 8;4
This study included four monolingual English children at study onset, and all had normal hearing based on a pure-
with a diagnosis of CAS who were recruited from the tone hearing screening or parent report. All children demon-
University of Arizona’s Grunewald-Blitz Clinic for Com- strated speech sound production difficulties as reflected in
munication Disorders in Children and from local schools. standard scores on the GFTA–2 <85 (<12th percentile; see
Diagnosis of CAS was confirmed by a licensed and certified Table 1). None of the children had a neurological or medical
speech-language pathologist (SLP; third author) with ex- diagnosis at the time of the study. As is not uncommon in
tensive experience in differential diagnosis of childhood this population, the children had varying degrees of CAS
speech disorders. Diagnosis of CAS was based on the pres- severity and concomitant diagnoses; however, CAS was a
ence of all three characteristics proposed by the ASHA major diagnosis in all cases and formed the focus of treat-
technical report (ASHA, 2007); namely, inconsistent errors ment. Three of the four children (CAS001, CAS002, and
on vowels and consonants, difficulty transitioning between CAS005) had participated in a previous treatment study
speech sounds, and prosodic errors including incorrect (Maas & Farinella, 2012).1 Each child is described sepa-
or equal stress and segmented speech. In the absence of rately below; a summary is presented in Table 1. All study
validated standardized assessment instruments for diagnos- procedures were approved by the University of Arizona’s
ing and/or quantifying CAS (McCauley & Strand, 2008), the Institutional Review Board.
current gold standard is clinical expert opinion. For the CAS001. CAS001 was a non-Hispanic White girl, age
present study, CAS features and severity were determined 8;4, with moderate-severe CAS. Her speech exhibited incon-
perceptually by an experienced clinician based on connected sistent vowel and consonant substitutions and distortions,
speech samples (e.g., narrative samples) as well as dynamic segmentation of sounds and syllables, equal and incorrect
assessment procedures in structured, single-word or single- stress in multisyllabic words, and intermittent hypernasality,
phrase production tasks, including the Goldman-Fristoe Test all of which were consistent with a diagnosis of CAS. No
of Articulation 2 (GFTA–2; Goldman & Fristoe, 2000) and formal intelligibility assessment was undertaken, but clin-
the Dynamic Evaluation of Motor Speech Skill (DEMSS; ical judgment indicated severely reduced intelligibility.
Strand, McCauley, & Stoeckel, 2006). The DEMSS requires CAS001 demonstrated persistent use of age-inappropriate
the child to directly imitate stimulus items of increasing phonological error patterns, including consistent prevocalic
complexity, beginning with very simple consonant–vowel voicing, final consonant deletion, and fronting, as well as
(CV) and CVC words (e.g., up, day). Using a dynamic inconsistent deaffrication, velar assimilation (e.g., gog for
scoring procedure (scores depend on level of support needed), dog), and weak syllable deletion. Based on analysis of a
the DEMSS specifically measures vowel accuracy, move- word repetition list, CAS001’s PCC was 53% (particular
ment gesture accuracy (moving from one articulatory position problems with fricatives, affricates, and velar sounds), and
to another), consistency of sound production, and prosody.
Judgments of inconsistent errors were based on between 1
two and five repetitions of the same utterance and required For all three children, the time between studies was 1 week (5 weeks
between treatment sessions). To enable fair comparisons between condi-
at least one production that differed from the others. Tran- tions, entirely new speech targets, transfer items, and control items were
sitioning difficulties were judged based on the presence of selected and baselined for each child. The same participant numbers refer to
inter- and intrasyllable pauses and /or noted improvement the same individuals between studies.

242 American Journal of Speech-Language Pathology • Vol. 21 • 239–257 • August 2012


TABLE 1. Study participant information.

Age Childhood apraxia


ID Sex (years;months) of speech Dysarthria Language Speech

CAS001 Female 8;4 Moderate-severe None Moderate expressive delay; GFTA–2 SS: <40
low average receptive skills (<1st percentile)
PPVT–4 SS: 97 (42nd percentile) PCC: 53%
CELF–4 Core SS: 76 (5th percentile) PVC: 46%
Concepts & Following Directions = 9/10
Word Structure = 4/10
Recalling Sentences = 4/10
Formulated Sentences =7/10
CAS002 Male 5;4 Severe Very mild unilateral Expressive not testable; receptive GFTA–2 SS: <40
upper motor neuron language within normal limits (<1st percentile)
PPVT–4 SS: 92 (30th percentile) PCC: 42%
PLS–4 SS: 95 (37th percentile) PVC: 11%
CAS005 Female 7;3 Moderate-severe Moderate Severe expressive delay; GFTA–2 SS: 62
spastic-flaccid mild-moderate receptive delay (<1st percentile)
PPVT–4 SS: 79 (8th percentile) PCC: 55%
CELF–4 Core SS: 46 (< 0.1st percentile) PVC: 34%
Concepts & Following Directions = 4/10
Word Structure = 1/10
Recalling Sentences = 1/10
Formulated Sentences = 1/10
CAS012 Male 6;4 Moderate None Severe expressive delay; moderate GFTA–2 SS: 84
to severe receptive delay (12th percentile)
CELF–4 Core SS: 46 (<0.1st percentile) PCC: 59%
Concepts & Following Directions = 4/10 PVC: 62%
Word Structure = 1/10
Recalling Sentences = 1/10
Formulated Sentences = 1/10
Sentence Structure = 3/10

Note. SS = standard score; PCC = percentage of consonants correct; PVC = percentage of vowels correct; PPVT–4 = Peabody Picture
Vocabulary Test, Fourth Edition (Dunn & Dunn, 2007); CELF–4 = Clinical Evaluation of Language Fundamentals, Fourth Edition (Semel, Wiig, &
Secord, 2003); GFTA–2 = Goldman-Fristoe Test of Articulation 2 (Goldman & Fristoe, 2000); PLS–4 = Auditory Comprehension subtest of the
Preschool Language Scale, Fourth Edition (Zimmerman, Steiner, & Pond, 2002).

her PVC was 46% (particular difficulties with diphthongs PVC of 11% (difficulties with most vowels and diphthongs,
and rhotic vowels). A structural–functional oral mechanism except nonrhotic central vowels and /u/). A mild breathy/
exam (Duffy, 2005) revealed no neuromuscular deficits harsh voice quality was noted, and a structural–functional
suggestive of dysarthria. oral mechanism exam (Duffy, 2005) revealed very mild left
CAS001’s expressive language as assessed with the facial asymmetry, suggesting a possible mild unilateral upper
CELF–4 revealed a moderate delay. She demonstrated motor neuron dysarthria. Spontaneous utterances and ver-
difficulty with correct production of various morphological balizations were characterized by adequate respiratory func-
markers (e.g., possessive pronouns, regular and irregular past tion for purposes of speech breathing and by grossly normal
tense). Use of complex sentence structure was noted on loudness, pitch, and rate.
occasion (e.g., The girl who has a soccer ball was third). Her CAS002’s expressive language could not be formally
receptive language was evaluated using the CELF–4 and the assessed due to his markedly severe CAS, but language
PPVT–4 and was determined to be in the low average range. sample analysis revealed evidence of expressive language
CAS002. CAS002 was a non-Hispanic White boy, age delay. There was little evidence for correct use of age-
5;4, with severe CAS and normal receptive language. Ex- appropriate morphological markers (e.g., absence of third
pressive language was not formally tested due to his severely person singular, copula omission). When asked to describe
reduced intelligibility, but informal observation suggested and tell a story about a picture book, CAS002 verbalized
a severe expressive language delay. His CAS was charac- with high frequency and clearly was “telling” the story;
terized by inconsistent errors on vowels and consonants, however, the majority of his utterances were completely
difficulty achieving initial articulatory configurations, trial unintelligible. Occasionally, an intelligible word was noted
and error groping behaviors, inconsistent productions following (e.g., jumping), indicating that he likely had the concept of
repetition of stimulus items, intermittent hypernasality, and certain grammatical markers (i.e., verb + ing). His pragmatic
regular use of a stereotypical nonword (e.g., dunch). His language skills and play schemes were age appropriate.
speech was facilitated by verbal, visual, and tactile cues. Receptive language as assessed using the PPVT–4 and the
Performance on a word repetition list revealed a PCC of Auditory Comprehension subtest of the PLS–4 was deter-
42% (particular difficulty with fricatives and liquids) and a mined to be in the low average range. Difficulties were noted

Maas et al.: Feedback Frequency in Treatment for CAS 243


with understanding qualitative concepts (i.e., longest, pointed, delay. His referral indicated a history of ear infections and
thin), quantity concepts (e.g., half, whole, each), passive difficulties with sequential tasks (e.g., auditory and working
voice, rhyming sounds, and adding and subtracting numbers memory), which may have contributed to the severity of his
to five. He did not seem to have difficulty understanding speech and language difficulties. He had no medical or
contextually based questions and comments; however, he neurological diagnoses at the time of the study, but he did
was often unable to respond using intelligible words, often display some features consistent with attention deficit/
relying on the stereotypical nonword dunch. hyperactivity disorder based on parent and teacher rating
CAS005. CAS005 was a non-Hispanic White girl, age scales and educational psychological testing that had been
7;3, with moderate-severe CAS, moderate-severe dysarthria completed previously.
(mixed spastic–flaccid), a mild-moderate receptive language CAS012’s CAS was characterized by frequent frank
delay, and a severe expressive language delay. Diagnosis vowel errors, inconsistent errors, prosodic errors (e.g., in-
of CAS was based primarily on the presence of inconsistent correct lexical stress), difficulty achieving initial articulatory
errors on consonants and vowels (especially diphthongs). configurations, greater difficulty with longer utterances, and
Her PCC was 55% (particular difficulties with fricatives, intermittent hypernasality. Of note, his productions im-
affricates, and liquids), and her PVC was 34% (particular proved with slow, simultaneous clinician models. CAS012’s
difficulties with diphthongs and rhotic vowels). Other speech PCC based on a word/phrase repetition list was 59% (par-
features consistent with a CAS diagnosis were also noted ticular difficulties with fricatives, affricates, and liquids), and
(including speech sound distortions, sound and syllable his PVC was 62% (particular difficulties with diphthongs
segmentation, intermittent hypernasality, prosodic abnor- and rhotic vowels). He demonstrated idiosyncratic errors
malities, and reduced speech rate), although these features (e.g., distortion of initial /w/) and several phonological
may also be attributable to her dysarthria. Patterned speech patterned errors, including gliding and consonant cluster
errors included deaffrication, labialization, stopping, gliding, reduction. His spontaneous utterances were characterized
and consonant cluster reduction (especially /l/ and /a/ by mildly abnormal pitch variability, high pitch, inconsis-
clusters). Deletion of final /z/ and final /v/ was also noted. tent hypernasality, and normal loudness and voice quality.
CAS005 also exhibited a markedly severe frontal lisp and Direct observation of his oral articulators revealed grossly
consistently distorted /s/ and /z/ due to marked tongue normal range of motion, strength, and speed of the jaw,
protrusion on these sounds. lips, and tongue. Based on a structural–functional oral
CAS005’s referral stated a history of hypotonia and exam (Duffy, 2005), there was no evidence of nonverbal
developmental delay but no specific medical–neurological oral apraxia and no evidence of frank weakness, nor was
diagnosis. Her voice quality was intermittently hoarse and there evidence of dysarthria. Speech alternating motion
breathy, and difficulties with other fine motor tasks (e.g., rates ( papapa, tatata, kakaka) were adequate; sequential
writing) were noted. Spontaneous utterances were charac- motion rates ( pataka) were sequenced and produced
terized by mildly reduced respiratory drive for purposes incorrectly.
of speech production, slow speech rate, hypernasality, CAS012’s expressive language was assessed using the
monoloudness, and monopitch. A structural–functional oral CELF–4, which revealed a markedly severe delay. During
exam (Duffy, 2005) revealed evidence of moderate tongue contextual interactions, CAS012 tended to respond using
weakness in lateral tongue movements and tongue protrusion short, simple, unelaborated utterances (e.g., The potato good;
and moderately restricted range of motion on tongue pro- Me have lots of toys). The only grammatical markers noted
trusion. No tongue fasciculations or atrophy were observed; included verb + ing, plurals, articles, and irregular third
however, fasciculations of the chin were noted on the left person (e.g., does). He consistently substituted me for I and
side. Movement of the soft palate was symmetrical but re- did not use any complex sentence structure.
duced during production of /a/. Speech alternating motion CAS012’s receptive language was assessed using the
rates ( papapa, tatata, kakaka) were slow, and sequencing Concepts & Following Directions subtest and the Sentence
difficulties were noted during speech sequential motion Structure subtest of the CELF–4, which revealed a severe
rates ( pataka). delay. He had difficulty understanding directions containing
CAS005’s expressive language was evaluated using several linguistic concepts (e.g., farthest, before/after, be-
the CELF–4 expressive core language subtests. Many age- tween, all but one, separated by, neither). In addition, he
appropriate morphological markers were absent (e.g., regular struggled with longer, multipart directions, and sometimes
plural, third person singular) or were used inconsistently also with understanding contextually based questions and
(e.g., irregular plural, auxiliary + ing). CAS005 also con- comments during communicative interactions. Based on
sistently used me for I. These difficulties were also noted in educational psychological testing completed previously,
her spontaneous speech. his auditory processing/memory impairments possibly con-
CAS005’s receptive language was assessed using the tributed to the severity of his language and speech deficits,
CELF–4 and the PPVT–4. She had difficulty following particularly his receptive language.
directions containing many different linguistic concepts
(e.g., before/after, closest to), and the PPVT–4 revealed a
moderate to severe delay in her receptive vocabulary. Design
CAS012. CAS012 was a Hispanic left-handed boy, age We used an alternating treatments design with multiple
6;4, with moderate CAS, a markedly severe expressive baselines across behaviors over two phases (Kearns, 1986).
language delay, and a moderate to severe receptive language The independent variable was the amount of feedback, with

244 American Journal of Speech-Language Pathology • Vol. 21 • 239–257 • August 2012


two conditions: high-frequency feedback (HFF; feedback on possibility of transfer between item sets. The first two
all trials) and low-frequency feedback (LFF; feedback on requirements were met by selecting target sets for each
È60% of trials; see Treatment Procedures for more detail). child that were equally difficult as determined based on a
Both conditions were administered every treatment session, pretreatment word repetition task. This word repetition task
with the order of conditions counterbalanced across sessions. was similar to the baseline and weekly probes but included
This design feature eliminated order/sequence effects a wider range of words and phrases expected to reveal
without relying on between-participant counterbalancing. difficulties, as well as items expected to be within a given
An initial baseline period with a minimum of three probe child’s capacity. As an additional control for possible con-
sessions over 2 weeks was implemented before any treat- founds due to the difficulty or learnability of targets, the
ment. To assess retention, a 2-week maintenance period design included a multiple baseline replication in a second
followed each treatment phase. A final probe was adminis- treatment phase in which target-condition pairings were
tered 1 month after the last treatment session. Thus, the total reversed where possible.
study duration from baseline to follow-up was 16 weeks (i.e., The third requirement was addressed primarily on theo-
2 weeks baseline, 4 weeks treatment, 2 weeks maintenance, retical grounds. In particular, we chose different target sets
4 weeks treatment, 2 weeks maintenance, follow-up after involving fundamentally different speech planning opera-
another 2 weeks). tions (e.g., sequencing consonants vs. sequencing syllables;
The main reason for implementing two phases of cf. Bohland & Guenther, 2006, for empirical support for
treatment was to control for unanticipated differences in this distinction) or different relative timing (stress) patterns.
difficulty or learnability between the target sets in Phase 1. The motor learning literature suggests that different relative
Given that each condition was paired with a different set of timing patterns are governed by different underlying motor
targets, any differential learning might be attributed to the programs (e.g., Schmidt, 1975; Shea & Wulf, 2005) and
targets rather than the feedback frequency condition. Im- are thus considered independent. Empirical support for the
plementing a second phase with a new set of targets in each notion of independence comes from motor learning studies
condition enabled us to provide a within-participant repli- in which limited or no transfer was observed between move-
cation of any effects (i.e., the multiple-baseline-across- ments with different relative timing patterns (e.g., Sakai,
behaviors component of the design). Although the use of Kitaguchi, & Hikosaka, 2003; Wilde & Shea, 2006). Given
multiple phases necessarily introduces the possibility of that stress patterns are defined in part by their relative timing
order and sequence effects, we attempted to minimize this structure, we conjectured that different stress patterns
possibility by reversing the pairing between condition and would be governed by different underlying motor programs
general target type in Phase 2 (e.g., clusters or adjacent stress (Maas et al., 2008; cf. also Skinder, Connaghan, Strand, &
in LFF in Phase 1, then clusters or adjacent stress in HFF in Betz, 2000) and therefore would not show cross-transfer.
Phase 2) and demonstrating stable baselines before Phase 2 Although efforts were made to include targets with per-
treatment. sonal functional relevance, the aforementioned target se-
Weekly probes were administered throughout the study to lection requirements prevented us from using personal
track retention and transfer of learning. The study included functional relevance as the primary selection criterion. In
both a performance-based criterion (80% correct on targets addition to treated targets for each condition in each phase
on two consecutive probes) and a time-based treatment (i.e., four treatment sets), we also included untreated item
termination criterion (4 weeks per phase). Because none of sets with similar properties to examine transfer, and un-
the children reached the performance-based criterion, treat- related untreated items to establish experimental control. The
ment phases lasted 4 weeks for all children,2 constituting item sets for each child are briefly described in the follow-
È10 hr of treatment per phase for each child. There were ing paragraphs; an overview of the item sets is provided
three weekly 50-min treatment sessions during treatment in Table 2.
phases for all children except CAS012, whose schedule CAS001. Given the prosodic difficulties noted in her
involved two weekly 1-hr sessions due to scheduling speech, targets for CAS001 included phrases with different
constraints. stress patterns. Phase 1 targets for CAS001 consisted of
five 3-syllable phrases per condition, with a strong–weak–
Treatment Target Selection strong (SwS) stress pattern (e.g., time to go) for the HFF
condition and a wSS stress pattern (e.g., before school)
Comparing conditions or treatments in a single-subject for the LFF condition. Phase 2 targets consisted of five
design context requires careful selection of different item 4-syllable phrases per condition, with a wSSw stress pattern
sets. Important requirements for treatment target sets include (e.g., begin reading) in the HFF condition and an SwwS
(a) low baseline performance, to justify treatment and enable stress pattern (e.g., tell me a joke) in the LFF condition. Five
detection of treatment effects; (b) equal difficulty level untreated phrases with the same target stress pattern were
before treatment, to enable a fair comparison between also generated for each of these four sets, and 48 untreated,
conditions; and (c) independent targets, to minimize the different control items were included as well. Finally, the
12 treated targets from the previous treatment study (Maas
2 & Farinella, 2012) were also included (but will not be
Note that due to technical difficulties, some probe sessions during treatment
were not recorded for CAS002 and CAS012. Thus, Figures 2 and 4 display reported here), for a total list of 100 items.
fewer probe points during treatment periods, even though four probes CAS002. Phase 1 targets for CAS002 included three
were administered in both phases as planned. 2-syllable words (e.g., circle) in the HFF condition and three

Maas et al.: Feedback Frequency in Treatment for CAS 245


TABLE 2. Target items for all study participants.

ID Condition Phase 1 targets Phase 2 targets

CAS001 HFF SwS phrases in the chair; make it quick; eat a cake; wSSw phrases without asking; the cute monkey; begin
time to go;comb your hair reading; I play soccer; escape summer
LFF wSS phrases before school; a big dog; avoid heat; SwwS phrases tell me a joke; give it away; finish the
his new bike; the small cup work; under my coat; never be late
Control SSw phrases (n = 5); wSwS phrases (n = 5); 5-syllable phrases (n = 15); 4–5-syllable words (n = 10); 1-syllable words (n = 13)
CAS002 HFF 2-syllable words tiger, balloon, circle Initial clusters broom, smoke, flame
LFF Final clusters mask, point, barn 3-syllable words bicycle, tomato, radio
Control 4-syllable words; fricative CVC words; affricate CVC words; plosive CVC words; liquid CVC words; sCC clusters (n = 10 all)
CAS005 HFF wSS phrases I don’t know; before school; a big dog; SwwS phrases give it away; never be late; thanks for
his new bike; my pink shirt your help; after the storm; tell me a joke
LFF SwS phrases this is fun; time to go; not today; wSSw phrases a hot summer; the cute monkey; without
brush your teeth; take a break asking; before Friday; begin reading
Control SSw phrases (n = 5); wSwS phrases (n = 5); 5-syllable phrases (n = 15); 4–5-syllable words (n = 10); 1-syllable words (n = 13)
CAS012 HFF Sww words bicycle, pyramid, astronaut wSwS words America, photographer, emergency
LFF wSw words flamingo, volcano, coyote SwSw words Arizona, caterpillar, helicopter
Control multisyllabic words (n = 21); multisyllabic phrases (n = 28); initial clusters (CCVC) (n = 10)

Note. HFF = high-frequency feedback, LFF = low-frequency feedback, S = strong, w = weak, C = consonant, V = vowel.

words with final consonant clusters (e.g., point) in the LFF (e.g., Arizona). In addition to three transfer items per set,
condition. Phase 2 targets consisted of three words with we included 59 untreated control items, for a total list of
initial clusters (e.g., broom) in the HFF condition and three 3- 83 items.
syllable words (e.g., bicycle) in the LFF condition. For each
of these four treatment sets, seven untreated words
with similar structures were generated as transfer sets. Procedure
With 60 unrelated control words, the total list consisted Treatment and probes were administered by a trained grad-
of 100 words. uate student clinician under direct supervision of the third
CAS005. Given her difficulties with multisyllabic utter- author, who is a certified and licensed SLP with extensive
ances, targets for CAS005 included phrases with different experience in the treatment of speech disorders in children.
stress patterns. Phase 1 focused on 3-syllable phrases. The Each child was treated by the same clinician throughout
five targets in the HFF condition had a wSS stress pattern the study.
(e.g., my pink shirt), and the five targets in the LFF condition Probe procedures. The data for this study were based on
had an SwS stress pattern (e.g., take a break). In Phase 2, productions on a probe task. The probe task was a repetition
treatment focused on 4-syllable phrases: five targets with task in which the clinician produced each item at a normal
an SwwS stress pattern in the HFF condition (e.g., never rate without any other cues. No feedback was provided
be late) and five targets with a wSSw stress pattern in the during the probe task. Treatment targets, transfer items, and
LFF condition (e.g., a hot summer). Five similar, untreated control items for each child were combined into a single list
phrases were selected for each treatment set (transfer), and were presented in one of four random orders. Probes
and 48 different untreated items served as controls. The were administered three to four times before any treatment
12 treated items from the previous treatment study (Maas started (baseline) and then once per week thereafter. Probes
& Farinella, 2012) were also included (but will not be were always administered at the beginning of a session
reported here), for a list of 100 items. and were audio- and video-recorded for subsequent data
CAS012. Given his noted difficulties with prosody, analysis and reliability scoring.
targets for CAS012 included multisyllabic words. Phase 1 Treatment procedures. We elected to study the effects of
targeted 3-syllable words. The HFF condition targets con- feedback frequency in the context of the DTTC approach
sisted of three words with an Sww stress pattern (e.g., (Strand, Stoeckel, & Baas, 2006) because this treatment
pyramid ), and the LFF condition targets consisted of three has replicated efficacy support (Maas & Farinella, 2012;
words with a wSw stress pattern (e.g., coyote). In Phase 2, Strand & Debertine, 2000; Strand, Stoeckel, & Baas, 2006)
six 4-syllable words were selected as targets: three HFF con- and because it incorporates principles of motor learning,
dition targets with a wSwS stress pattern (e.g., America) allowing us to systematically vary one of these while
and three LFF condition targets with an SwSw stress pattern keeping the treatment otherwise the same. Furthermore, by

246 American Journal of Speech-Language Pathology • Vol. 21 • 239–257 • August 2012


selecting DTTC as the treatment, this study would contribute (2012), we operationally defined an effect to be present
yet another replication of this approach. when ES >1 (i.e., when magnitude of change exceeded the
Briefly, DTTC involves integral stimulation (“watch me, standard deviation). Mean level differences in percentages
listen carefully, and say what I say”) combined with inten- (gain scores; i.e. the numerator in the ES formula) are also
sive drill practice on a small set of vocabulary items. Slow provided.
speech and tactile gestures are used as needed to highlight
the movement patterns, and the amount of time between Reliability and Fidelity
the clinician’s model and the child’s attempt is gradually
increased to encourage greater reliance on independent re- The proportion of randomly selected double-scored ses-
trieval and planning of speech motor gestures. The number sions ranged from 18% for CAS001 to 61% for CAS005
of practice trials per session was maximized by using quick (CAS002: 27%; CAS012: 29%). As in previous studies
reinforcers (e.g., stickers). Targets were practiced in ran- using this scoring system (e.g., Maas & Farinella, 2012;
dom order throughout the study, in both feedback conditions. Strand et al., 2006), interrater reliability was acceptable
Children received È2 hr of treatment per week, divided over to high (CAS001: 86%; CAS002: 94%; CAS005: 88%;
two or three sessions. CAS012: 71%).
The two conditions being compared were HFF and LFF. An independent analyst examined several treatment as-
Feedback was defined as explicit verbal information pro- pects for fidelity (Kaderavek & Justice, 2010) based on
vided by the clinician about the accuracy (knowledge of viewing a randomly selected subset of video-recorded ses-
results; e.g., “that was almost right!”) and movement ges- sions. Fidelity aspects scored for each condition included
tures (knowledge of performance; e.g., “you didn’t round number of trials, amount of time, number of whole-word
your lips enough”). For each treatment target, 10 index cards production attempts per trial, feedback delay, and feedback
were created, some of which were marked to indicate feed- frequency. Proportion of trials adhering to protocol was also
back trials to the clinician. In the HFF condition, all cards assessed; deviations from protocol included failure to pro-
were marked; in the 60% condition, only six of the 10 cards vide feedback (HFF condition), inconsistent presence of
per target were marked. Before each session, the cards in feedback within a trial (e.g., feedback on first attempt but
each of the two piles were shuffled to randomize trial order. not on last attempt in a trial), and feedback delays <1 s or
Breaks were provided at the condition switch and as needed. >5 s. An overview of these fidelity measures is provided
All sessions were audio- and video-recorded for subse- in Appendix C. As can be seen, treatment fidelity was
quent analysis and fidelity evaluation. The treatment pro- not perfect. By and large, however, conditions were well
tocol is provided in Appendix A. matched.

Data Analysis Results


The dependent measure was percentage accuracy on Data for each child are plotted in Figures 1–4 and sum-
the probe lists, based on a 3-way perceptual scoring system marized in Table 3. In all figures, square markers (red and
that included both segmental and suprasegmental aspects of pink lines) represent the HFF condition, round markers (dark
entire target words or phrases, rather than only segmental and light blue lines) represent the LFF condition, solid lines
accuracy (cf. Maas & Farinella, 2012; Strand & Debertine, with filled markers denote treated items, and broken lines
2000; Strand, Stoeckel, & Baas, 2006). In this system, a with open markers denote untreated transfer items. Treatment
score of 2 denotes a correct production, a score of 1 denotes periods are demarcated by vertical lines (see also footnote 2).
a close approximation, and a score of 0 denotes an incorrect
production. These scores were operationally defined (see
Appendix B). Scores were converted to a percentage based CAS001
on the total possible points for each set. Each child’s data As Figure 1 shows, both conditions were at a stable 0%
were analyzed by one primary analyst who was unaware of correct during baseline. After onset of treatment, improve-
the treated targets, and sessions were analyzed in random ment was evident for the treated items in both conditions,
order to prevent a confound of increasing familiarity with a but more so for the LFF condition. After treatment, both
child’s speech. Reliability was assessed by a second blinded conditions showed performance above baseline levels, with
analyst for a randomly selected number of sessions (see higher accuracy for the LFF condition. The effect size mea-
Reliability and Fidelity section later). sures corroborate this visual analysis, indicating ES = 1.52
Data were plotted for visual analysis and were quantified (18% gain) for the HFF condition and ES = 2.51 (35% gain)
by calculating effect sizes (ES) using Beeson and Robey’s for the LFF condition. Neither condition showed transfer
(2006) modification of Busk and Serlin’s (1992) d metric: (HFF: ES = 0.15, 1% gain; LFF: ES = 0.37, 4% gain), nor did
ES = (mean score post treatment – mean score pre treatment)/ the control items show improvement (combined weighted
pooled standard deviation.3 Following Maas and Farinella ES = 0.72, 7% gain; 3-syllable SSw phrases only: ES = 1.35;
17% gain).
3 In Phase 2, CAS001 demonstrated a response to treat-
We chose to use the pooled standard deviation as the denominator rather
than the pretreatment standard deviation because the latter option would
ment in both conditions, following a stable baseline. The
result in many instances of incalculable effect sizes due to zero variance in performance on probes during the treatment phase appeared
baseline (cf. Beeson & Robey, 2006; Maas & Farinella, 2012). equivalent between conditions; performance on probes after

Maas et al.: Feedback Frequency in Treatment for CAS 247


FIGURE 1. CAS001 data for Phase 1 (top), Phase 2 (middle), and control items (bottom).

Note. S = strong (stressed), w = weak (unstressed), FB = feedback; Tx = treated items. Treatment periods are delineated with
vertical lines.

treatment showed greater accuracy for the LFF condition on untreated items. Inspection of the bottom graph in Figure 1
again. Effect size could not be calculated for the HFF con- reveals that this effect was driven primarily by improvements
dition due to zero variance in both pre- and posttreatment on 3-syllable phrases with SSw stress patterns (ES = 5.93,
data; for the LFF condition, effect size calculation showed 22% gain) and, to a lesser degree, by the 4- and 5-syllable
ES = 2.28. The mean level difference was larger for the LFF phrases (ES = 1.93, 3% gain).
condition (24% gain) than for the HFF condition (10% gain). When combining data across both phases, the LFF con-
Transfer was negligible for the LFF condition (ES = 0.99, dition shows an effect size of ES = 2.40 (30% average gain);
7% gain) and small for the HFF condition (ES = 1.18, 8% no combined HFF condition effect size could be com-
gain). The combined weighted effect size for control items in puted (14% average gain). No transfer is evident for either
Phase 2 indicated ES = 1.69 (5% gain), suggesting improvement condition, and control items show ES = 1.20 (6% gain).

248 American Journal of Speech-Language Pathology • Vol. 21 • 239–257 • August 2012


FIGURE 2. CAS002 data for Phase 1 (top), Phase 2 (middle), and control items (bottom).

Thus, the LFF condition consistently leads to greater changes (HFF: ES = 0.88, 4% gain; LFF: ES = 0.38, 1% gain), nor
on treated items than the HFF condition, and the effect sizes did control items improve (combined weighted ES = 0.70,
are larger than for the control items. 4% gain; liquids only ES = 1.64; 6% gain).
In Phase 2, a similar pattern emerged: a stable baseline,
followed by an increase in accuracy toward the end of the
CAS002 phase, with a larger increase for the HFF condition and a
For CAS002, a stable baseline was established before decrease in accuracy after treatment ended. Effect size cal-
Phase 1 treatment. As can be seen in Figure 2, an increase culations indicated treatment effects in both conditions, with
in accuracy for treated items emerged toward the end of the a relative advantage for HFF (ES = 1.91, 17% gain) com-
phase, with a larger increase for HFF items than for LFF pared to LFF (ES = 1.11, 6% gain). No transfer was evi-
items. However, these gains were not retained after cessation dent in either condition (0% change); control items also
of treatment, as evinced by the effect sizes (HFF: ES = 0.77, remained unchanged (combined weighted ES = 0.37, 0%
6% gain; LFF: ES = 0.58, 4% gain). No transfer was evident gain; liquids only ES = 1.48; 8% gain).

Maas et al.: Feedback Frequency in Treatment for CAS 249


FIGURE 3. CAS005 data for Phase 1 (top), Phase 2 (middle), and control items (bottom).

Taken together, CAS002 showed a small treatment effect items, except for HFF items in Phase 2 (ES = –0.51,
for HFF items (ES = 1.32, 12% gain) but not for LFF items –2% gain; 0% gain for other treated items in both con-
(ES = 0.83, 5% gain), with no transfer (ES not computable ditions and both phases). The increase in performance on
for both conditions; HFF 2% gain; LFF 1% gain) and no LFF transfer items following Phase 2 treatment (ES = 1.08)
change in control items (ES = 0.53, 2% gain; liquids only was minimal (5% gain). Some improvement was noted
ES = 1.56, 7% gain). for untreated control items in Phase 1 (ES = 1.75, 6% gain);
this effect was driven entirely by improvement on sim-
pler one-syllable words (ES = 2.09, 22% gain). In Phase 2,
CAS005 there was no change in combined control items (ES = 0.96,
CAS005 demonstrated a stable baseline for both con- 5% gain), although 3-syllable SSw phrases did improve
ditions in both phases, with no apparent change throughout (ES = 2.15, 11% gain) following treatment on 4-syllable
the study. Effect sizes could not be computed for treated phrases.

250 American Journal of Speech-Language Pathology • Vol. 21 • 239–257 • August 2012


FIGURE 4. CAS012 data for Phase 1 (top), Phase 2 (middle), and control items (bottom).

Combined across phases, there was no improvement or (ES = 1.04, 24% gain) but not for the LFF condition (ES =
transfer in either condition, and a slight improvement on 0.83, 20% gain). There was no transfer for either condition
control items (ES = 1.36, 6% gain). (HFF: ES = 0.21, 4% gain; LFF: ES = 0.28, 7% gain), and
no change for control items (ES = 0.38, 5% gain).
The baseline for Phase 2 targets was stable, and there was
CAS012 a relatively small response to treatment in the HFF condi-
For CAS012, the Phase 1 baseline was more variable than tion that was not maintained after treatment (ES not com-
for the other participants, but was relatively low and with- putable, 0% gain). For the LFF condition, there was no
out an improving trend. Following initiation of treatment, an change during treatment, but performance following treat-
increase in accuracy on treated items was evident for both ment was consistently above baseline levels, which was
conditions, with a more sustained gain for HFF. Effect sizes corroborated by the effect size calculation (ES = 4.42, 22%
indicated a small treatment effect for the HFF condition gain). Regarding transfer, a small improvement was noted

Maas et al.: Feedback Frequency in Treatment for CAS 251


TABLE 3. Effect sizes per condition, phase, and child. condition for treated items in both phases. These effects
exceeded changes in the control items overall, indicating that
Child Condition Phase 1 Phase 2 Combined gains were likely attributable to treatment. However, some
change was noted in some of the control items (primarily
CAS001 HFF Treated 1.52 — —
Transfer 0.15 1.18 0.67 3-syllable SSw phrases in Phase 2). Although this suggests a
LFF Treated 2.51 2.28 2.40 loss of experimental control, an alternative interpretation is
Transfer 0.37 0.99 0.68 that these control items inadvertently showed transfer from
Control 0.72 1.69 1.20 the 4-syllable phrases treatment. This interpretation is sup-
CAS002 HFF Treated 0.77 1.91 1.32 ported by the fact that other control items remained un-
Transfer 0.88 — — changed and by the fact that the improvement was coincident
LFF Treated 0.58 1.11 0.83 with the onset of Phase 2 treatment. Thus, the results from
Transfer 0.38 — —
Control 0.70 0.37 0.53 CAS001 are consistent with the motor learning literature
on adults and with findings from research with adults with
CAS005 HFF Treated — –0.51 —
Transfer 0.51 — — apraxia of speech (Austermann Hula et al., 2008).
LFF Treated — — — CAS012 showed feedback frequency effects that were
Transfer 0.66 1.08 0.87 inconsistent across phases, with a small effect for HFF only
Control 1.75 0.96 1.36 in Phase 1 and a large effect for LFF only in Phase 2. This
CAS012 HFF Treated 1.04 — — pattern of findings suggests an interaction between feedback
Transfer 0.21 1.11 0.64 frequency and target difficulty, such that LFF was more
LFF Treated 0.83 4.42 2.55 effective than HFF, and words starting with a stressed syl-
Transfer 0.28 0.12 0.20
Control 0.43 0.67 0.55 lable (Sww in Phase 1 and SwSw in Phase 2) were easier
than words starting with an unstressed syllable (wSw in
Note. Effect size = (mean post) – (mean pre) / SD(pre+post). — = Phase 1 and wSwS in Phase 2). Thus, when the “easy”
ES could not be computed due to zero variance. targets were combined with the less effective HFF condition
(Phase 1), they counteracted the potential disadvantage of
HFF to produce a slight advantage over the LFF condition.
for the HFF transfer items (ES = 1.11, 6% gain) but not In Phase 2, when the “easy” targets were paired with the
for the LFF transfer items (ES = 0.12, 1% gain). Control optimal LFF condition, there was a larger difference be-
items did not show improvement as a group (ES = 0.90, 3% tween conditions, now in favor of LFF (a “double-whammy”
gain), although multisyllabic words did show some improve- effect). Overall, combined across both phases, there was a
ment (ES = 1.02, 6% gain). clear treatment effect in the LFF condition only, and this
Combined across both phases, CAS012 showed a treat- effect exceeded changes in the control items, suggesting that
ment effect for the LFF condition (ES = 2.55, 21% gain), the effects were due to treatment. Thus, the data from CAS012
whereas no effect size could be computed for the HFF con- also indicate an advantage for LFF over HFF.
dition (12% gain). There was no transfer or change in con- In contrast, data from CAS002 indicated a slight advan-
trol items (HFF transfer: 5% gain; LFF transfer: 4% gain; tage for HFF overall in terms of the retention of treated
control items: 4% gain). targets. Treatment effects in Phase 2 exceeded changes in
combined control items, supporting the idea that improve-
ments were due to treatment. This notion is further strength-
Discussion ened by the decrease in performance following treatment
The primary purpose of the present study was to compare (i.e., the maintenance period turned out to act as a withdrawal
the relative effects of different feedback frequencies (high design feature). Inspection of Figure 2 strongly suggests that
vs. low) in treatment for CAS with respect to retention and treatment was terminated too early (based on our a priori
transfer of learning. A secondary purpose was to replicate termination criterion) because notable increases in accuracy
a version of the DTTC treatment approach (e.g., Strand, began to appear on the last probe session during each treat-
Stoeckel, & Baas, 2006). Each of these issues will be dis- ment phase. Given the severity of his CAS and the notion
cussed in turn. that CAS often requires prolonged treatment (e.g., Ballard
et al., 2010; Shriberg, Aram, & Kwiatkowski, 1997), ex-
tending our relatively short treatment phases likely would
Feedback Frequency in CAS Treatment have resulted in greater, more sustained gains, especially
Summarizing the feedback frequency effects combined in the HFF condition, although this must remain speculative
across treatment phases, it is clear that findings were mixed. at this time. Nevertheless, it is worth noting that at least
Limiting our discussion to only the three children who showed minimal gains were seen in this child, contrary to a previous
improvement on treated items in any condition or phase (i.e., study in which he showed no gains (Maas & Farinella, 2012).
excluding CAS005), it appears that two children (CAS001 One possible explanation is that the previous study used
and CAS012) demonstrated an advantage for LFF compared LFF; the present findings suggest that CAS002 might have
to HFF, whereas the other child (CAS002) showed the shown gains if HFF had been used in the previous study.
reverse. The use of two separate treatment phases raises the pos-
For CAS001, both conditions resulted in a positive re- sibility that the Phase 2 effects were influenced by Phase 1
sponse to treatment, with a consistent advantage for the LFF treatment carryover. Given that we attempted to counterbalance

252 American Journal of Speech-Language Pathology • Vol. 21 • 239–257 • August 2012


target types across phases, such carryover effects would re- but not all children showed improvements (e.g., Maas &
sult in a reduction of condition differences when combining Farinella, 2012; Strand, Stoeckel, & Baas, 2006). Thus, this
both phases. The fact that we still observed condition dif- study adds to the evidence base by providing support for
ferences suggests that such order effects were minimal, if a modified version of the DTTC approach. The magnitude
present at all. In addition, the multiple baseline aspect of the of treatment effects was generally comparable to the treat-
design allowed us to demonstrate that Phase 2 targets did ment effects obtained in a previous study using this modified
not improve during Phase 1 treatment, corroborating the DTTC approach (Maas & Farinella, 2012), but smaller than in
notion that any potential carryover effects were negligible. the original DTTC studies (Strand & Debertine, 2000; Strand,
Although treatment fidelity was not perfect, the condi- Stoeckel, & Baas, 2006).
tions were generally well matched in this regard. Further- As discussed in Maas and Farinella (2012), the differ-
more, to the extent that differences were noted, they tended ence in magnitude of effects between the original and this
to favor the condition that resulted in the smallest improve- modified DTTC treatment may be related to one or more
ments (e.g., for CAS001, HFF had longer average duration factors, including treatment distribution or intensity (ten
than LFF, yet the LFF condition showed the greatest change). 30-min sessions per week for 6 weeks in Strand, Stoeckel, &
Thus, it is unlikely that fidelity discrepancies can account Baas, 2006; three 1-hr sessions per week for 4 weeks per
for the pattern of findings. phase in the present study), the nature of treatment targets
Taken together, two children benefited more from LFF, (relatively simple, functionally relevant items in Strand &
whereas the third benefited more from HFF. Based on this Debertine, 2000; relatively complex items that were not
small sample size, we can only speculate as to why these selected strictly based on personal functional relevance in
different patterns emerged. At least two possibilities present the present study), and the probe measurement procedures
themselves; namely, age (or age-related factors) and CAS (multiple opportunities per target in Strand, Stoeckel, &
severity. The two children who showed an LFF advantage Baas, 2006, and probes administered at the end of treatment
were older than the other child, and feedback frequency effects sessions in Strand & Debertine, 2000; one opportunity per
may shift with age (cf. Sullivan et al., 2008). Although Sullivan target, obtained at the beginning of a treatment session in
et al. (2008) studied older children (mean age = 10 years) in a the present study).
different motor task (arm movement task), it is possible that It is important to note that three of the four children in this
such age-related feedback frequency reversal effects occur study also participated in a previous treatment study (Maas
earlier for speech. Perhaps a certain minimum level of error & Farinella, 2012). Although we selected new targets
awareness—and a reference of correctness—is required to take (neither treated nor included on the probe lists in the previous
advantage of LFF, and perhaps our older children met this study) and demonstrated stable, low baseline performance
minimum level. Anecdotally, both children who showed an for these items, these children were already familiar with
advantage for LFF were aware of their errors, as evidenced by the general treatment procedures, which may have enhanced
attempts at self-correction. CAS002, who benefited more from the gains seen in the present study. However, this appears
HFF, initiated very few attempts at self-correction. unlikely for two main reasons. First, the treatment effects
Alternatively, CAS severity may play a role as well, with were comparable in magnitude to those observed in the prior
more severe CAS requiring practice conditions that are con- study (if anything, smaller than in the previous study). Sec-
sidered suboptimal in the motor learning literature. Consis- ond, one of these children (CAS005) showed considerable
tent with this idea is the observation in the previous study treatment effects in the previous study but showed no effects
(Maas & Farinella, 2012) that to the extent that CAS002 in the current study. In the case of CAS005, her previous
showed gains at all, these gains appeared to be in blocked participation may have reduced her motivation; anecdotal
rather than random practice. Again, the severity of his CAS observations suggested greater noncompliance during treat-
may have limited CAS002’s a priori reference of correct- ment compared to the other children, as well as compared
ness or his ability to detect and correct errors. HFF may have to herself in the prior study. This may explain her lack of
facilitated establishing such a reference of correctness or error improvement in the present study. Thus, although participa-
detection ability. Although we did not formally test speech tion in the previous study may have influenced the present
perception abilities, some research suggests that speech per- findings, such influences do not necessarily enhance treat-
ception may also be impaired in some children with CAS ment effects. Future studies are needed to replicate this treat-
(e.g., Groenen, Maassen, Crul, & Thoonen, 1996; Maassen, ment with additional children (who may or may not have
Groenen, & Crul, 2003; Nijland, 2009). Such deficits could received prior treatment).
conceivably interfere with the ability to detect errors and with With respect to transfer, the general conclusion is that
the ability to develop a reference of correctness. Future transfer was quite limited for all of the children studied. This
treatment studies might benefit from including formal, de- may be due to the nature of the relation between treated
tailed measures of speech perception and error detection and untreated targets. We defined our target sets in terms of
abilities. word/phrase length and stress pattern for three out of four
children, and it is possible that this type of similarity was not
sufficient to allow transfer. Perhaps similarity in terms of
Efficacy of Integral Stimulation Treatment for CAS place or manner of articulation classes is a more appropriate
Regarding our secondary purpose—to provide a replica- way to assess transfer; however, our materials were not de-
tion of an integral stimulation-based treatment for CAS—our signed or controlled in this way. We did note changes in
findings were consistent with previous reports in that most some control items for some children that seemed to coincide

Maas et al.: Feedback Frequency in Treatment for CAS 253


with onset of treatment on similar but more complex tar- (Pittsburgh, PA, 2010), and the Arizona Speech-Language-Hearing
gets. For example, untreated words with singleton liquids Association Convention (Phoenix, AZ, 2011); portions of these
showed some improvement for CAS002 during Phase 2 data were also part of Christine Butalla’s master’s thesis at the
treatment, in which the HFF condition targeted word-initial University of Arizona.
clusters with liquids. For CAS001, untreated 3-syllable SSw
phrases improved during Phase 2, which targeted 4-syllable
phrases. Nevertheless, limited transfer is not uncommon in
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Appendix A
Treatment protocol

Clinician: “Watch me, listen carefully, and repeat after me,” and models target
Child responds
Clinician waits 2–3 seconds
If child’s response is correct:
: FB trial: Provide FB – Knowledge of Results (KR) and Knowledge of Performance (KP)
: No FB trial: Present next target
If child’s response is incorrect:
: FB trial:
: Provide FB (KR and KP)
: Slow, simultaneous production (up to 2 times); tactile cues as needed
: Fade simultaneous cue (mouth the words)
: Immediate repetition
: Wait 2–3 seconds and provide FB (KR and KP)
: No FB trial:
: “Let’s do it slowly together”
: Slow simultaneous production (up to 2 times); tactile cues as needed
: Fade simultaneous cue (mouth the words)
: Immediate repetition
: Wait 2–3 seconds, then “Now let’s do another one”

As child improves, increase delay between clinician model and child’s response (i.e., delayed repetition instead of
immediate repetition) and eventually elicit targets without clinician model.

Note. FB = feedback.

256 American Journal of Speech-Language Pathology • Vol. 21 • 239–257 • August 2012


Appendix B
Scoring guidelines

Score = 0 Example Score = 1 Examples

More than one sound in error kæt Y gæd


(distorted, substituted, tbÃtɚflaI Y tbætɚfaI
omitted, added)
Omission of sound/syllable kæt Y kæ
tbÃtɚflaI Y tbÃflaI
tbÃtɚflaI Y tbÃtɚfaI
Addition of sound/syllable kæt Y kætəl Excessive plosive release and /or kæt Y kæth
tbÃtɚflaI Y tbÃtɚflaIp aspiration (unless also included
in the clinician’s model)
Vowel “substitution” (sounds kæt Y k I t Vowel distortion: kæt Y kæt æ/ɛ?
like a good exemplar tbÃtɚflaI Y tbætɚflaI (sounds like a bad exemplar of the
of a different vowel) target vowel, or between target and
other vowel)
Also includes excessive lengthening. tbÃtɚflaI Y tbÃtɚflaI Ã /I?
No other errors: kæt Y kæ:t
Rest of word should be entirely correct.
Gross consonant error kæt Y kæf, bæt Minor consonant errors: kæt Y gæt, kæp
(more than one tbÃtɚflaI Y tbÃzɚflaI No more than one feature off target tbÃtɚflaI Y tdÃtɚflaI, tmÃtɚflaI
feature off target) (may result in perceived substitution) tbÃtɚflaI Y tbÃtɚf:laI
for one consonant.
Also includes excessive lengthening.
No other errors: Rest of word should
be correct.
Sound error plus prosodic tbÃtɚflaI Y bætɚtflaI Minor prosodic error: tbÃtɚflaI Y bÃtɚtflaI
error (e.g., syllable Incorrect stress pattern or syllable tbÃtɚflaI Y tbÃ(.)tɚ(.)flaI
segregation, incorrect segregation (only for multisyllabic items).
stress pattern) No other errors: Rest of word should
be correct.
Any other errors that do Unintelligible
not qualify for a 1 Two minor prosodic errors

Appendix C
Treatment fidelity measures

Child Sessions checked Condition Duration (min.) # Trials # Attempts per trial FB delay (sec.) %FB % Adherence

CAS001 3/24 HFF 26 16 4.2 2.4 83% 76%


LFF 18 15 4.5 2.4 48% 77%
CAS002 3/24 HFF 26 19 7.8 2.1 81% 68%
LFF 28 21 6.8 2.0 45% 66%
CAS005 3/24 HFF 17 11 6.2 2.5 91% 64%
LFF 28 14 6.2 2.4 66% 83%
CAS012 3/12 HFF 22 14 4.5 2.4 91% 86%
LFF 19 16 4.6 2.6 63% 92%

Note. HFF= high-frequency feedback, LFF = low-frequency feedback.

Maas et al.: Feedback Frequency in Treatment for CAS 257


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