The Relationship among Oral Motor, Fine Motor, Simple, and Complex Speech Skills in Childhood of Speech

A Thesis Submitted to the Department of Communication Sciences and Disorders College of Allied Health Sciences

in partial fulfillment of the requirements for the degree of

Master of Arts

Allison R. Flynn, B.S. May 2011

Nancy Creaghead, Ph.D. Committee Chair

Sandra Grether, Ph.D. Committee Member

Erin Redle, Ph.D. Committee Member

Sandra Combs, Ph.D. Committee Member

Abstract

Childhood apraxia of speech (CAS) is a controversial and greatly debated diagnosis in the area of pediatric speech sound disorders. Currently there is a lack of understanding of the etiology of the disorder and disagreement over the core deficits. Deficits in oral motor and fine motor praxis are commonly reported in the literature in association with childhood apraxia of speech (ASHA, 2007; Dewey, 1995; Maassen, 2002; Newmeyer et al., 2007); however, they are not validated in the research. While there are reported relationships between oral motor, fine motor, and speech praxis, the role of these relationships is yet to be determined. This study aims to provide evidence regarding the relationships between the oral-motor, fine-motor and speech skills of children with CAS.

Subjects in this study were seen in the interdisciplinary apraxia clinic at Cincinnati

Children‟s Hospital Medical Center. To be included in the study the subjects had to be 2 to 5 years of age at the time of evaluation, receive a clinical diagnosis of childhood apraxia of speech

(CAS), have a standard receptive score greater than or equal to 85 to rule out receptive language deficits, and have received the Kaufman Speech Praxis Test. Subjects were excluded if they had a known neurological or or a known hearing loss.

Pearson correlations were run, using IBM SPSS© Statistics 19 software, to determine the relationships among the standard scores of the oral movement, simple phonemic and syllabic level, and complex phonemic and syllabic level parts of the KSPT and the fine-motor quotient of the PDMS-2. A statistically significant moderate correlation (r=.523; p=.001) was found between the KSPT oral-motor standard scores and the PDMS-2 fine-motor quotients. There were no other statistically significant correlations within the data.

ii The lack of a significant relationship between speech and non-speech motor control further supports current research that they are unique and independent of one another (Green,

Moore, & Reilly, 2002; Moore & Ruark, 1996; Ruark & Moore, 1997; Steeve, Moore, Green,

Reilly, & Ruark McMurtrey, 2008; Wilson, et al., 2008). Although oral-motor and fine-motor skills are commonly cited as diagnostic indicators for CAS, these data indicate there is no significant relationship between these skills and speech abilities. Furthermore, the results indicate that a child may have good oral and fine motor skills and still have significant deficits in their speech praxis at both the simple and complex levels. Clinically this indicates that when oral and fine motor deficits are involved with CAS it may be indicative of a global dyspraxia.

The results also indicate that some level of speech is required to evaluate and confirm a diagnosis of CAS. These findings further support evidence against the use of non-speech oral motor exercises to improve speech skills (Bunton, 2008; Lof & Watson, 2008; Ruscello, 2008).

iii

© 2011

Allison R. Flynn

ALL RIGHTS RESERVED

iv Acknowledgements

First of all I would like to thank my committee for their help and guidance. I‟ve gained a lot of insight into the research process and the need for quality research within the field of speech-language pathology. I would like to give special thanks to Dr. Grether and Dr. Redle. I am especially grateful for the hours they gave in meeting, developing, and editing this thesis with me. I would not have been able to accomplish this daunting task and still have a drive to do more if it were not for their constant support. Lastly, I must thank my family for their unending support and love during the stressful moments that came with the challenge of formulating, researching, writing, editing, and formatting my thesis.

v Table of Contents

Abstract …………………………………………………………………. ii Tables …………………………………………………………………. vii

Chapter 1 Introduction ……………………………………………………………. 1

Chapter 2 Literature Review……………………………………………………..... 4 Overview of Terminology……………………………………… 4 Overview of Etiology…………………………………………... 6 Diagnosis of CAS…………………………………………….… 8 Speech vs. Non-Speech Motor Controls……………………...… 10

Chapter 3 Methods…………………………………………………………………. 12 Subjects……………………………………………………….… 12 Procedure…………………………………………………….…. 13 Statistical Analysis……………………………………………… 15

Chapter 4 Results…………………………………………………………………… 16

Chapter 5 Discussion…………………………………………………………….….. 19 Limitations……………………………………………………..… 20 Future Research………………………………………………….. 21 Clinical Implications……………………………………...……… 21

Conclusion ……………………………………………………………. 23

References………………………………………………………………….. 24

vi List of Tables

Table 1 Diagnostic criteria cited within the CAS literature used in this study 9

Table 2 Mean scores and standard deviations of 49 children diagnosed with CAS on the

Kaufman Speech Praxis Test Parts 1, 2, and 3 and the Peabody Developmental Motor Scales-

2nd Ed. Comprised Fine-motor Quotient 18

Table 3 Pearson correlation values for correlations between oral motor and fine motor, oral motor and simple speech, oral motor and complex speech, fine motor and simple speech, fine motor and complex speech, and simple speech and complex speech 18

vii CHAPTER 1

Introduction

Childhood apraxia of speech (CAS) is a controversial and greatly debated diagnosis in the area of pediatric speech sound disorders. Currently there is a lack of understanding of the etiology of the disorder and disagreement over the core deficits. This leads to challenges in producing quality research on the prevalence, diagnosis, and treatment of the disorder (Morgan

& Vogel, 2009). However, there are a number of points of agreement within the field of speech language pathology regarding CAS. There is a universal understanding that CAS is a neurological deficit in children (ASHA, 2007). Childhood apraxia of speech has an early onset as well as a long-course to obtain normal speech even with intervention (Shriberg, Aram, &

Kwiatkowski, 1997a, 1997b). There is also agreement that the phonological errors associated with CAS are typically deviant rather than delayed (Maassen, 2002; Shriberg, et al., 1997b).

Furthermore, neurological differences often exist prior to the awareness of the disorder, classifying it as a developmental rather than acquired disorder (Maassen, 2002). The etiology of

CAS is unknown; however, researchers are actively working toward identifying the neurological differences associated with a CAS diagnosis. Discrepancies in the diagnostic criteria are a significant challenge for developing a precise classification of CAS.

On the other hand, there are varied opinions regarding CAS within the field of speech language pathology. This lack of congruence was cause for a position statement and technical report from the American Speech-Language-Hearing Association (ASHA) in 2007. It was

ASHA‟s recommendation that the classification term for this specific pediatric speech sound disorder be CAS. A formal definition of childhood apraxia of speech was also formed based upon the literature (ASHA, 2007):

1 “Childhood apraxia of speech is a neurological childhood (pediatric) speech sound

disorder in which the precision and consistency of movements underlying speech are

impaired in the absence of neuromuscular deficits (e.g. abnormal reflexes, abnormal

tone). CAS may occur as a result of known neurological impairment in association with

complex neurobehavioral disorders of known or unknown origin, or as an idiopathic

neurogenic speech sound disorder. The core impairment in planning and/or programming

spatio/temporal parameters of movement sequences results in errors in speech sound

productions and prosody (p. 3-4).”

Despite this definition of CAS, there is still no validated list of diagnostic features to differentially diagnose CAS. Many speech language pathologists have different diagnostic criteria and have difficulty distinguishing CAS from a general speech sound delay, a phonological disorder, another severe speech sound disorder, or a (ASHA, 2007). A survey conducted at a workshop asked seventy-five speech language pathologists to list up to three diagnostic criteria they used to differentially diagnose CAS (Forrest, 2003). Fifty different criteria were identified, including general oral-motor difficulties (Forrest, 2003). However, general oral-motor difficulties, and many other diagnostic criteria listed, have yet to be validated as differential diagnostic criteria for CAS.

Deficits in oral-motor and fine motor praxis are reported in the literature in association with childhood apraxia of speech (ASHA, 2007; Dewey, 1995; Maassen, 2002; Newmeyer et al.,

2007); however, they are not validated in the research. While there are reported relationships between oral-motor, fine motor, and speech praxis, the role of these relationships is yet to be determined. This study aims to provide evidence regarding the relationships between the oral- motor, fine-motor and speech skills of children with CAS. The specific research questions are:

2 1. Is there a relationship between oral-motor skills and speech skills in children with CAS?

2. Is there a relationship between fine motor skills and speech skills in children with CAS?

3. Is there a relationship between oral-motor skills and fine-motor skills in children with

CAS?

3 CHAPTER 2

Literature Review

Overview of Terminology

Prior to ASHA‟s (2007) recommendation of childhood apraxia of speech (CAS) as the classification term for this population, there were a number of previous terms cited for CAS in the literature, including: developmental verbal dyspraxia, developmental apraxia of speech, developmental dyspraxia, developmental coordination disorder, and severe speech sound disorder. Childhood apraxia of speech is often considered synonymous with developmental verbal dyspraxia (DVD) and developmental apraxia of speech (DAS). The terminology of dyspraxia is more commonly cited in international literature (ASHA, 2007). Synonymous with developmental dyspraxia is developmental coordination disorder (Zwicker, Missiuna, & Boyd,

2009). With the confusion regarding a specific classification term and defined diagnostic criteria for this population, some professionals prefer to continue to refer to the diagnosis of a severe speech sound disorder, or will insert the term „suspected‟ in front of the diagnosis of CAS

(Newmeyer, et al., 2007).

The term, childhood apraxia of speech, stemmed from the similar diagnosis in the adult population, acquired apraxia of speech (AOS). Much of the current research for CAS is modeled after the understanding of acquired apraxia of speech. Acquired apraxia of speech can be defined as a “disturbed ability to produce purposeful, learned movements despite intact mobility, secondary to brain damage (Knollman-Porter, 2008, p. 484).” Individuals with acquired apraxia of speech do not demonstrate significant weakness, slowness, or difficulty with reflexive and automatic acts (Knollman-Porter, 2008; Maassen, 2002). Similarly, children with CAS exhibit

4 motor differences, but do not demonstrate weakness or a decrease in the range of motion of perioral structures (McCauley, Strand, Lof, Schooling, & Frymark, 2009). According to the

ASHA technical report, Childhood apraxia of speech is described as a neurological impairment affecting the precision and coordination of volitional speech motor movements. Childhood apraxia of speech is associated with deficits in timing, programming, and sensorimotor coordination. The literature also commonly associates CAS with fine and oral motor deficits

(ASHA, 2007). The classification of CAS includes idiopathic CAS, CAS as part of a larger , and a genetic form of CAS. Developmental verbal dyspraxia (DVD) and developmental apraxia of speech (DAS) specifically refer to the idiopathic type of CAS, indicating that speech praxis deficits exist without other associated motor praxis difficulties.

Meanwhile developmental dyspraxia and developmental coordination disorder are associated with global praxis deficits (Zwicker, Missiuna, & Boyd, 2009). Developmental dyspraxia is considered a disorder of gesture and is used to classify children with limb, orofacial, and verbal praxis deficits (Dewey, 1995).

The lack of a classification term for this population, in addition to undefined diagnostic standards, prior to the ASHA statement impacted research and has led to confusion among professionals as to who specifically is included within this population. With the variance in terms and what they encompass, an area of confusion is whether the population solely includes those with speech praxis deficits or those with more generalized motor praxis deficits. The

ASHA (2007) position on this issue should help to clarify much of the confusion; however, there still is no validated list of diagnostic features to clearly define the population included within this diagnostic classification.

5 Overview of Etiology

Currently the etiology of CAS is unknown and widely debated among professionals.

There is general agreement that childhood apraxia of speech is neurological in nature. There is also evidence that CAS may have an underlying genetic component; however, how these genes present in children with CAS is variable. A genetic marker was first identified in the KE family in 1998, which has been further researched since (Fisher, Lai, & Monaco, 2003; Fisher, Vargha-

Khadem, Watkins, Monaco, & Pembrey, 1998; Lai et al., 2000; Lai, Fisher, Hurst, Vargha-

Khadem, & Monaco, 2001). However, the findings have not been found to generalize to the overall population with CAS, and the phenotypes within the population were variable (ASHA,

2007).

While it appears that overall CAS has a neurological basis, CAS is often described as either idiopathic or part of a more global neurological disorder (ASHA, 2007). CAS in its idiopathic form affects speech praxis specifically without other deficits in motor praxis (ASHA,

2007). The motor speech theory suggests that CAS is linked to deficits in planning and programming the motor movements for speech. This theory associates with the theory of CAS as idiopathic, affecting speech praxis alone (Ackermann & Riecker, 2004; Maassen, 2002;

Nijland, Maassen, & van der Meulen, 2003; Riecker et al., 2000; Riecker, Brendel, Ziegler, Erb,

& Ackermann, 2008; Riecker et al., 2005). A computational neural model of CAS, the DIVA model, has been used to test the hypothesis regarding the neurological differences and sensorimotor deficits in CAS. This testing found that a possible neurological deficit in CAS is imprecise feed-forward commands, reducing somatosensory information and increasing neural noise. This testing specifically looked at speech motor controls and was based upon current behavioral data and hypotheses of the underlying deficits (Terband & Maassen, 2010; Terband,

6 Maassen, Guenther, & Brumberg, 2009). A contradictory view is that CAS is a secondary characteristic of a larger and more global neurologic deficit affecting all areas of motor praxis, which often co-occur with other known neurological disorders (ASHA, 2007). Global deficits are commonly described by professionals in the literature in association with speech praxis deficits. Other deficits within a global dyspraxia include: non-speech oral-motor, fine motor, and gross motor (Dewey, 1995; Forrest, 2003; Maassen, 2002; Morgan & Vogel, 2009; Newmeyer, et al., 2007; Zwicker, et al., 2009). Some researchers have gone on to look more closely at the underlying processes that may lead to a global praxis deficit. Currently there is research to support that CAS may be a central timing disorder affecting accuracy across modalities (Peter &

Stoel-Gammon, 2005, 2008). Theory of CAS is often derived from research in AOS. A study of individuals with AOS found that CAS may be related to difficulties with organizing the internal structure of units and noted that this reflects an underlying motor programming problem which affects both speech and non-speech tasks (Maas, Robin, Wright, & Ballard, 2008). Although there is cited research regarding the neurological basis of both forms of CAS, the findings are limited secondary to the insufficient diagnostic criteria.

Regardless of whether childhood apraxia of speech is idiopathic or part of a larger neurological disorder, how the underlying neurological deficits affect speech motor control is not fully understood. There are current theories to support both sides of CAS; however, the studies are limited by an undefined population. There is great variance in the presentation of childhood apraxia and how the motor controls are affected. Some researchers have found that the speech motor controls are affected by deficits in somatosensory processes (Terband & Maassen, 2010;

Terband, et al., 2009). Meanwhile, other researchers have found that general motor control is affected by a central timing deficit (Peter & Stoel-Gammon, 2005, 2008). Although there are

7 theories regarding the etiology of the motor deficits associated with CAS, the effects of these deficits within the overall population remains unknown.

Diagnosis of CAS

Currently there is inconsistency in the accurate and reliable diagnosis of childhood apraxia of speech across professionals and researchers. The challenge lies in the lack of specific diagnostic criteria for CAS (ASHA, 2007). This is evident by the survey conducted by Forrest, indicating fifty different criteria listed by seventy five speech language pathologists used to identify children with CAS. The most commonly cited included: inconsistent productions, groping/effortful productions, general oral-motor difficulties, inability to imitate sounds, increasing difficulty with sound production as the utterance length increases, and poor sequencing of sounds (Forrest, 2003). Many items on the list were contradictory indicating confusion among clinicians regarding the criteria for diagnosis of CAS. Many clinicians struggle with the ability to differentially diagnose CAS from a severe speech sound disorder, phonological disorder, general , or dysarthria (ASHA, 2007).

The lack of validated diagnostic criteria poses a challenge in identifying an appropriate population within the research (Shriberg, et al., 1997a). Table 1 shows the diagnostic criteria cited to define the population within the literature used within the current study. While there are many overlaps within the literature there are also multiple outlier characteristics. The diagnostic criteria listed in table 1 were often cited as frequently reported characteristics rather than diagnostic markers. Without true diagnostic markers the progress toward a „gold standard‟ of testing and good methodology for treatment is impeded (McCauley, et al., 2009; McCauley &

Strand, 2008; Morgan & Vogel, 2009).

8 Table 1. Diagnostic criteria cited within the CAS literature used in this study.

Diagnostic Criteria for CAS ASHA (2007)  Groping  Differences in performance of automatic and volitional activities with volitional activities more affected  Inconsistency of errors  Increased number of errors with longer or more complex word and syllable shapes  Slow speech development  Vowel errors  Reduced vowel inventory  Reduced phonemic/phonetic repertoire  Multiple speech sound errors  Errors in the ordering of sound, syllables, morphemes, or words  Reduced percentage of correct consonants  Unintelligibility  Unusual errors that „defy process analysis‟  Persistent or frequent regressions Shriberg, L., Aram, D.,  Groping & Kwiatkowski, J.  Failure to achieve on command isolated and sequenced oral-motor movements  Inability to volitionally produce isolated phonemes or sequences of phonemes that (1997) have been previously produced correctly  Inconsistent pattern of articulation errors  Increased articulation errors with increased utterance length  Speech development shows a deviant pattern  Unable to perform diadochokinetic tasks  Struggle  Trial and error behavior when producing phonemes Morgan, A. & Vogel, A.  Inconsistent errors patterns on consonants and vowels in repeated words or (2009) syllables  Inappropriate prosody  Lengthened and impaired coarticulatory transitions McCauley, R. & Strand,  Inconsistent errors with repeated productions E. (2008)  Presence of vowel errors  Abnormal prosody  Difficulty with smooth transitions between articulatory configurations Nijland, L., Maassen, B.,  Inconsistent error patterns van der Meulen, S.  Difficulty with complex phonemic sequences  Difficulty with sequencing (2003)  Complete phonemic repertoire with multiple phonemic errors  High frequency of substitutions and consonant cluster omissions Terband, H., Maassen,  Searching articulation B., Guenther, F.,  Increased variability  Deviant coarticulation Brumberg, J. (2009)  Distortion of speech sounds Peter, B. & Stoel-  Impaired volitional oral movements Gammon, C. (2005)  Inconsistent articulation errors  Increased errors with longer units of speech output  Vowel errors  Limited phonemic inventory  Reduced diadochokinetic rates  Disordered prosody, voice quality and fluency  Omission errors  Difficulty imitating speech  Predominant use of simple syllable shapes  Expressive language skills lower than receptive

9 Speech vs. Non-Speech Motor Controls

In childhood apraxia of speech many researchers and clinicians identify non-speech oral motor difficulties as diagnostic markers. One view among professionals is that the motor controls of speech and non-speech actions are related and interdependent as they share many of the same active muscles (Wilson, Green, Yunusova, & Moore, 2008). A similar view is that speech oral-motor controls emerge from earlier non-speech oral-motor controls. This perspective is associated with the connection of speech motor controls with a central pattern generator that influences the organization of rhythmic and complex behaviors such as mastication, respiration, phonation, swallowing and sucking (Moore & Ruark, 1996). On the contrary, within the community there is the view that speech and non-speech oral motor controls are distinct from one another (Green, Moore, & Reilly, 2002; Moore & Ruark, 1996; Ruark &

Moore, 1997; Steeve, et al., 2008; Wilson, et al., 2008).

There is evidence to support the idea that speech and non-speech oral motor controls are unique and independent (Green, Moore, & Reilly, 2002; Moore & Ruark, 1996; Ruark & Moore,

1997; Steeve, Moore, Green, Reilly, & Ruark McMurtrey, 2008; Wilson, et al., 2008). A strong indicator for task specific development of oral motor control is the involvement of sensory information. Sensory feedback is critical in the differential development of motor control for speech vs. non-speech behaviors with distinct differences in sensory information involved

(Wilson, et al., 2008). When infants and young children are developing control for motor speech actions both visual and auditory feedbacks play a role. There are indications that infants as young as 4.5 months of age are learning visual cues for speech (Wilson, et al., 2008).

Computational modeling using the Directions into Velocities of Articulators (DIVA) model also supports the essential role of auditory feedback for the development of speech specific oral-

10 motor controls (Terband, et al., 2009). Additionally, it has been found that a link between auditory perception and activation of speech motor areas of the brain develops between 6 and 12 months of age (Imada, et al., 2006). In contrast there is no evidence to support the integration of auditory or visual feedback in the development of non-speech oral motor controls (Wilson, et al.,

2008).

Furthermore, the coordination of the lip muscles and mandible have been studied in children ages 9 months to 2 years, comparing the coordination in speech vs. non-speech tasks.

Overall it was found that by 12 months of age, oral motor coordination of both the lip and mandible was markedly distinct for speech versus non-speech behaviors (Moore & Ruark, 1996;

Ruark & Moore, 1997). At 9 months of age task specific oral motor coordination and organization were found to be poorly specified though they shared similar characteristics with the task-specific patterns seen in the older infants and adults (Steeve, et al., 2008).

While there is strong evidence to support the unique oral motor controls for speech vs. non-speech tasks, research and clinical findings suggest that deficits in oral-motor control manifest in both speech and non-speech tasks. The purpose of this study is to provide evidence regarding the relationships among the oral-motor, fine-motor and speech skills of children with

CAS. This research is guided by the following questions:

1. Is there a relationship between oral-motor skills and speech skills in children with CAS?

2. Is there a relationship between fine motor skills and speech skills in children with CAS?

3. Is there a relationship between oral-motor skills and fine-motor skills in children with

CAS?

11 CHAPTER 3

Methods

This study is part of a larger retrospective study using data collected from the interdisciplinary apraxia clinic at Cincinnati Children‟s Hospital Medical Center (CCHMC) through the Division of Developmental and Behavioral Pediatrics. Approval was obtained from the Institutional Review Board of the University of Cincinnati, as well as the Institutional

Review Board of Cincinnati Children‟s Hospital Medical Center.

Subjects

Patients seen in the interdisciplinary apraxia clinic at CCHMC between July 2003 and

October 2007 were included in this study. Subjects participated in a standardized clinical evaluation process. The process included an evaluation with a developmental pediatrician, a speech language pathologist, and an occupational therapist. The number of examiners included in this study was limited to one developmental pediatrician, two speech language pathologists, and three occupational therapists. The subjects completed a battery of standardized tests; each of the selected tests had available published normative values.

Criteria for inclusion included an age of two to five years at the time of evaluation, a standard receptive language score greater than or equal to 85, and a final clinical diagnosis of childhood apraxia of speech (CAS). Subjects were excluded from the study if they had a known diagnosis of a neurological or developmental disorder, or had a known hearing loss. Subjects who were clinically diagnosed with CAS presented with the following characteristics indicated on the KSPT Diagnostic Rating Scale Continuum (Kaufman, 1995):

 Oral scanning or groping

12  Difficulty maintaining the same motor-speech patterns twice  Inability to imitate motor-speech patterns of increased length or complexity  Intact isolated consonant production, with a breakdown at the word level  Vowel distortions  A consonant repertoire limited to simple consonants /m p b t d n h/  Sound errors (distortions/weak targets)  Inconsistent attempts on words of increased length or complexity Procedure

Each client who underwent testing in the CCHMC Apraxia Clinic completed a battery of assessments. The clients completed a neurological examination that included a complete family and past medical history inventory, a standardized speech and language evaluation, and a fine motor evaluation. The demographic information, family history, past medical history, and results from the standardized evaluations were compiled into an electronic database.

The neurological examination and patient information was gathered by the developmental pediatrician with the parent and child present. One of two trained speech-language pathologist conducted the speech and language evaluations. The two speech-language pathologists completed reliability training on the first five patients, comparing results and discussing differences to determine consistent criteria for rating items that differed. The standardized and norm-referenced language assessment tool used was the Preschool Language Scale- 4th edition

(PLS-4). This assessment evaluates receptive and expressive language skills in children aged 2 weeks to 6 years 11 months. The PLS-4 provides a standard score (mean 100; SD 15), percentile rank, and age equivalent for both auditory comprehension and expressive communication, as well as a total language score (Zimmerman, Steiner, & Pond, 2002). The Kaufman Speech Praxis

Test for Children (KSPT) was completed to assess the subjects‟ speech skills. The KSPT is a norm-referenced standardized assessment tool used to clinically identify characteristics of

13 abnormal speech praxis in children ages two to six years. The KSPT has been norm-referenced for the normal speaking pediatric population as well as for the pediatric population with speech and language deficits. The KSPT parts yield a raw score, standard score (mean 100; SD15), percentile rank, and age equivalent for each of the four parts. The four parts of the KSPT include: oral movement, simple phonemic and syllabic level, complex phonemic and syllabic level, and spontaneous length and complexity (Kaufman, 1995). The parts are designed so that the degree of task difficulty increases with each part; therefore, based upon the severity of the , not all subjects completed all parts. According to the KSPT guidelines, Part 2

(simple phonemic and syllabic level) was presented regardless of performance on Part 1 (oral movement) (Kaufman, 1995). Part 3 was presented to each client if they were capable of completing the tasks. However, the parts may not have been completed and scored depending on the child‟s ability to imitate given tasks, or due to behavioral challenges as the tasks increased in difficulty. Subjects with a minimum of a score for part 1 of the KSPT were included in the current study.

Fine motor skills were evaluated by the current clinic occupational therapist. During the time frame that data were collected three occupational therapists participated in the clinic. As one transitioned the position to the next therapist, the two therapists scored the initial protocols together to insure consistency and reliability. The clients were evaluated using the Peabody

Developmental Motor Scales- 2nd edition (PDMS-2). The PDMS-2 evaluates motor development in children ages birth to five years (Folio & Fewell, 2000). Subjects were administered three of the six subtests from the PDMS-2 which most closely associate with fine motor sequencing and planning. The subtests administered were object-manipulation, grasping and visual-motor integration, providing standard scores (mean 10; SD 3). The standard scores

14 for the grasping and visual-motor integration subtests are included in the composition of the fine- motor quotient (mean 100; SD15).

Statistical Analysis

The data collected were entered into a Microsoft Access database. Selected data were then entered into IBM SPSS© Statistics 19 software to be analyzed. Pearson correlations were run to determine the relationships among the standard scores of the oral movement, simple phonemic and syllabic level, and complex phonemic and syllabic level parts of the KSPT and the fine-motor quotient of the PDMS-2. Pearson correlations were completed for each of the scores paired against one another. A regression analysis was not used as it was not appropriate to answer the study‟s research questions which examined the relationships between individual pairs of data: oral motor vs. fine motor, oral motor vs. simple speech, oral motor vs. complex speech, fine motor vs. simple speech, fine motor vs. complex speech, and simple speech vs. complex speech.

15 CHAPTER 4

Results

The results from 197 clients‟ initial evaluations were included within the Childhood

Apraxia of Speech database at CCHMC. Criteria for inclusion in this study included an age of two to five years at the time of evaluation, which excluded 36 subjects from this study. Another

23 subjects had not received a final clinical diagnosis of childhood apraxia of speech (CAS). A standard receptive language score greater than or equal to 85 was required to rule out any conflicting receptive language deficits, this removed an additional 69 subjects. Of the remaining

69 subjects, 11 had not received the Kaufman Speech Praxis Test eliminating them from the study. Subjects with a known diagnosis of a neurological, neuromuscular, or developmental disorder were also excluded. Therefore the 8 subjects identified with hypotonia and 1 with a seizure disorder were excluded. None of the remaining subjects had abnormal reflexes, a diagnosis of fragile X, cerebral palsy, spectrum disorder, or a known hearing loss.

The remaining 49 subject population was comprised of 8 females (16%) and 40 males

(81.6%). The average age of subjects was 42.7 months with a range of 24-70 months. There were 39 Caucasian, 4 Asian, 2 African American, and 4 other subjects. Past medical histories revealed that 13 subjects had received MRI studies and 7 had abnormal results, 10 subjects had a history of ear tubes. Prenatal and early infancy histories were significant for 7 subjects with a history of prematurity (1 pre-31 weeks, 6 pre-36 weeks), and 9 subjects with a history of a NICU stay. None of the subjects had a known history of prenatal alcohol or other drug exposure. In addition, 9 subjects were described to have a feeding problem and 5 were described to have a drooling problem in their past medical history.

16 Table 2 shows the mean scores and standard deviations of 49 children whose data were included n the study. Pearson correlations were completed, using IBM SPSS© Statistics 19 software, to determine the relationships between each of the following measures paired with each other: the standard scores of the oral movement, simple phonemic and syllabic level, and complex phonemic and syllabic level parts of the KSPT and the fine-motor quotient of the

PDMS-2. Table 3 shows the Pearson correlation values.

1. Is there a relationship between oral-motor skills and speech skills in children with CAS?

The Pearson correlation values for oral motor vs. simple speech (r=.194, p=.243) and oral

motor vs. complex speech (r=.020, p=.945) were not found to be statistically significant.

2. Is there a relationship between fine motor skills and speech skills in children with CAS?

The Pearson correlation values for fine motor vs. simple speech (r=.317, p=.114) and fine

motor vs. complex speech (r=.328, p=.354) were not found to be statistically significant.

3. Is there a relationship between oral-motor skills and fine-motor skills in children with

CAS?

The analysis showed a statistically significant correlation (r=.523, p=.001) between the

KSPT oral-motor standard scores and the PDMS-2 fine-motor quotients. This was a

moderate correlation, with mean standard scores of 84.29 (SD 34.49) on the KSPT oral

movement part and 90.47 (SD 12.12) on the PDMS-2 tests of fine-motor.

17 Table 2. Mean scores and standard deviations of 49 children diagnosed with CAS on the

Kaufman Speech Praxis Test Parts 1, 2, and 3 and the Peabody Developmental Motor Scales-

2nd Ed. Comprised Fine-motor Quotient

Mean Scores and Standard Deviations PQuotient KSPOMSS KSPSPSS KSPCPSS Mean 90.47 84.29 50.59 55.40 SD 12.12 24.49 39.35 28.19 PQuotient: PDMS-2 fine-motor quotient; KSPSPSS: KSPT simple speech standard score; KSPOMSS: KSPT oral-motor standard score; KSPCPSS: KSPT complex speech standard score

Table 3. Pearson correlation values for correlations between oral motor and fine motor, oral motor and simple speech, oral motor and complex speech, fine motor and simple speech, fine motor and complex speech, and simple speech and complex speech

Pearson Correlation Values PQuotient KSPOMSS KSPSPSS KSPCPSS PQuotient Pearson Correlation 1.000 .523 .317 .328 Sig. (2-tailed) .001*** .114 .354 N 34 34 26 10 KSPOMSS Pearson Correlation .523 1.000 .194 .020 Sig. (2-tailed) .001*** .243 .945 N 34 48 38 14 KSPSPSS Pearson Correlation .317 .194 1.000 .422 Sig. (2-tailed) .114 .243 .117 N 26 38 39 15 KSPCPSS Pearson Correlation .328 .020 .422 1.000 Sig. (2-tailed) .354 .945 .117 N 10 14 15 15 PQuotient: PMDS-2 fine-motor quotient; KSPSPSS: KSPT simple speech standard score; KSPOMSS: KSPT oral-motor standard score; KSPCPSS: KSPT complex speech standard score ***Correlation significant at the 0.01 level (2-tailed)

18 CHAPTER 5

Discussion

Oral-motor and fine-motor skill deficits are commonly cited as diagnostic indicators for

CAS. Within the literature these deficits are often reported as observed skill deficits or frequently reported characteristics (ASHA, 2007; Dewey, 1995; Maassen, 2002; Newmeyer, et al., 2007). There is also theory to support the notion that oral-motor and speech movements are controlled by the same central pattern generator as they activate the same muscles (Moore &

Ruark, 1996; Wilson, et al., 2008). To date these skills have not been validated within the research literature as confirmed diagnostic criteria or to be correlated with speech skills. This retrospective study examined the relationship between these non-speech motor controls and the speech motor controls impaired in children diagnosed with CAS.

From the sample of 49 children with CAS ages 24-70 months, it was found that there is no significant relationship between non-speech motor skills and speech abilities. The results of the Pearson correlations indicate that there is a lack of significant relationships between speech motor control and non-speech motor control. This information reflects findings from previous research indicating that speech and non-speech motor controls are unique and independent of one another (Green, et al., 2002; Moore & Ruark, 1996; Ruark & Moore, 1997; Steeve, et al., 2008;

Terband, et al., 2009; Wilson, et al., 2008). Previous research supporting these findings have been based on the influence of auditory and visual feedback used for the development of speech, in contrast to the lack of feedback used when developing non-speech oral-motor skills (Wilson, et al., 2008). In addition, the findings that the development of speech skills is task specific and distinct in comparison to non-speech oral-motor skills support the results that there is no

19 significant relationship between non-speech and speech motor controls (Moore & Ruark, 1996;

Ruark & Moore, 1997).

This research targeted a population that would likely indicate an idiopathic type of CAS, in contrast to a more global dyspraxia, through its selection of exclusion criteria including: below average auditory comprehension scores, other known neurological, neuromuscular, and developmental disorders, and hearing loss. The findings of this research study indicate that while there is no relationship between non-speech and speech motor controls, there is a significant relationship (p=.001) between oral-motor and fine-motor skills. These results lead to the view that oral and fine motor tasks are part of a global generalized neurological motor control network, while speech develops from specific specialized neurological motor controls. From review of the scores, the mean standard scores indicate that the non-speech motor skills of this population fall within the low-average to average range. However, there is a disparity between motor skills and speech skills when reviewing the mean standard scores across speech specific task scores, which were significantly below average. These results indicate that in this idiopathic population, a child may have age appropriate oral and fine motor skills, while their speech praxis abilities at both the simple and complex levels can show significant deficits.

Limitations

In line with previous research in CAS, this study is limited by the lack of universally defined diagnostic criteria for CAS (McCauley, et al., 2009; McCauley & Strand, 2008; Morgan

& Vogel, 2009; Shriberg, et al., 1997a). Without universal guidelines for a diagnosis, this study‟s ability to differentiate and compare this population from previous and future research populations is limited.

20 In addition, background information did not include previous speech interventions and progress of speech intelligibility with intervention. This information would further support the diagnoses of CAS if limited progress was noted, as this is a frequently reported characteristic of

CAS and is often used to differentially diagnosis CAS from a severe phonological disorder

(ASHA, 2007).

Future Research

Further research in this area is needed to confirm the relationships noted in this study as well as to determine any role of the relationships within the diagnosis and treatment of CAS. For this, a larger sample size would be necessary. In addition, a greater span of ages, including an older population of children with CAS, would give a broader view of the population.

Clinical Implications

While these results are not indicative of oral-motor or fine-motor involvement with CAS in a population without other conflicting disorders, when these deficits do occur they may indicate a more global praxis deficit. Developmental dyspraxia, or developmental coordination disorder, is a global disorder of praxis involving limb, orofacial, and verbal skills (Dewey,

1995). Fine-motor or oral-motor deficits identified within an evaluation for CAS would suggest a referral for further evaluation of fine motor skills to rule-out a global praxis deficit, such as developmental coordination disorder or developmental dyspraxia (Ho & Wilmut, 2010;

Newmeyer, et al., 2007)

As noted by the results in this idiopathic population, oral-motor deficits are not indicative of a diagnosis of CAS. However, currently oral-motor deficits are commonly used as a diagnostic indicator for CAS (ASHA, 2007; Dewey, 1995; Forrest, 2003; Maassen, 2002).

When taking into consideration the findings of this study, it is important to note the need for a

21 speech sample and analysis to give a diagnosis of CAS. The data from this study would not support the diagnosis of CAS for a non-verbal child with oral-motor deficits. Evaluations for

CAS require more than an oral-motor examination involving imitation, as children‟s oral movement abilities are not always a reflection of their speech abilities. Therefore, it is critical that clinicians evaluate speech praxis prior to confirming a diagnosis of CAS.

Non-speech oral-motor exercises (NSOMEs) are currently used to attempt to improve articulation and speech skills by some speech-language pathologists. Sixty percent of these speech-language pathologists believe that speech skills emerge from early non-speech oral motor skills (Lof & Watson, 2008). This is in disagreement with current research indicating that speech and non-speech motor skills develop independently (Green, et al., 2002; Moore & Ruark,

1996; Ruark & Moore, 1997; Steeve, et al., 2008; Terband, et al., 2009; Wilson, et al., 2008).

This study supports that speech and non-speech praxis skills are independent from one another, as the results indicate that they do not have a significant relationship based on scores on the

Kaufman Speech Praxis Test and the Peabody Developmental Motor Scales- 2nd edition.

Furthermore, current motor theory indicates task-specific motor control, which is supported by the lack of a relationship between non-speech and speech skills in the idiopathic CAS population reviewed in this sample. Childhood apraxia of speech is reported to be the second most frequent population to be treated with the use of non-speech oral motor exercises (Lof & Watson, 2008); yet the population sample of this study indicates that non-speech skills are typically average in children with CAS. Current research does not support the use of non-speech oral motor exercises in the treatment of speech sound disorders as there is a lack of empirical evidence to support the notion that targeting non-speech oral-motor skills improves speech (Bunton, 2008;

Lof & Watson, 2008; Ruscello, 2008).

22 Conclusion

There is further research to be conducted to r examine the existence of non-speech motor praxis deficits in association with CAS. The population for this study was selected for idiopathic

CAS by excluding children with other diagnoses impacting motor praxis and speech skills. This study found no relationship between non-speech and speech skills in children with idiopathic

CAS, further supporting the argument for independent motor controls for non-speech and speech skills. However, there is a moderate correlation (r=.523) between oral-motor and fine-motor skills, with mean scores in the average range. This may indicate that oral and fine motor skills are correlated as general fine-motor praxis while speech is a specialized motor task.

Furthermore, these children had average non-speech motor skills, while their speech skills were significantly disordered.

23 References

Ackermann, H., & Riecker, A. (2004). The contribution of the insula to motor aspects of speech

production: a review and a hypothesis. Brain Lang, 89(2), 320-328.

American Speech-Language-Hearing Association. (2007). Childhood Apraxia of Speech

[Technical Report]. Available from www.asha.org/policy.

Bunton, K. (2008). Speech versus nonspeech: different tasks, different neural organization.

Semin Speech Lang, 29(4), 267-275.

Dewey, D. (1995). What is developmental dyspraxia? Brain Cogn, 29(3), 254-274.

Fisher, S. E., Lai, C. S., & Monaco, A. P. (2003). Deciphering the genetic basis of speech and

language disorders. Annu Rev Neurosci, 26, 57-80.

Fisher, S. E., Vargha-Khadem, F., Watkins, K. E., Monaco, A. P., & Pembrey, M. E. (1998).

Localisation of a gene implicated in a severe speech and . Nat Genet,

18(2), 168-170.

Folio, M. R, & Fewell, R. (2000). Peabody Developmental Motor Scale. 2nd ed. Austin, Tex:

Pro-Ed.

Forrest, K. (2003). Diagnostic criteria of developmental apraxia of speech used by clinical

speech-language pathologists. Am J Speech Lang Pathol, 12(3), 376-380.

Green, J. R., Moore, C. A., & Reilly, K. J. (2002). The sequential development of jaw and lip

control for speech. J Speech Lang Hear Res, 45(1), 66-79.

Ho, A. K., & Wilmut, K. (2010). Speech and oro-motor function in children with developmental

coordination disorder: a pilot study. Hum Mov Sci, 29(4), 605-614.

24 Imada, T., Zhang, Y., Cheour, M., Taulu, S., Ahonen, & A., Kuhl, P. K. (2006). Infant speech

perception activates Broca's area: a developmental magnetoencephalography study.

NeuroReport 17(10), 957-962.

Knollman-Porter, K. (2008). Acquired apraxia of speech: a review. Top Rehabil, 15(5),

484-493.

Kaufman, N. (1995). Kaufman Speech Praxis Test for Children. Detroit, Mich: Wayne State

University Press.

Lai, C. S., Fisher, S. E., Hurst, J. A., Levy, E. R., Hodgson, S., Fox, M., et al. (2000). The

SPCH1 region on human 7q31: genomic characterization of the critical interval and

localization of translocations associated with speech and language disorder. Am J Hum

Genet, 67(2), 357-368.

Lai, C. S., Fisher, S. E., Hurst, J. A., Vargha-Khadem, F., & Monaco, A. P. (2001). A forkhead-

domain gene is mutated in a severe speech and language disorder. Nature, 413(6855),

519-523.

Lof, G. L., & Watson, M. M. (2008). A nationwide survey of nonspeech oral motor exercise use:

implications for evidence-based practice. Lang Speech Hear Serv Sch, 39(3), 392-407.

Maas, E., Robin, D. A., Wright, D. L., & Ballard, K. J. (2008). Motor programming in apraxia of

speech. Brain Lang, 106(2), 107-118.

Maassen, B. (2002). Issues contrasting adult acquired versus developmental apraxia of speech.

Semin Speech Lang, 23(4), 257-266.

McCauley, R. J., Strand, E., Lof, G. L., Schooling, T., & Frymark, T. (2009). Evidence-based

systematic review: effects of nonspeech oral motor exercises on speech. Am J Speech

Lang Pathol, 18(4), 343-360.

25 McCauley, R. J., & Strand, E. A. (2008). A review of standardized tests of nonverbal oral and

speech motor performance in children. Am J Speech Lang Pathol, 17(1), 81-91.

Moore, C. A., & Ruark, J. L. (1996). Does speech emerge from earlier appearing oral motor

behaviors? J Speech Hear Res, 39(5), 1034-1047.

Morgan, A. T., & Vogel, A. P. (2009). A Cochrane review of treatment for childhood apraxia of

speech. Eur J Phys Rehabil Med, 45(1), 103-110.

Newmeyer, A. J., Grether, S., Grasha, C., White, J., Akers, R., Aylward, C., et al. (2007). Fine

motor function and oral-motor imitation skills in preschool-age children with speech-

sound disorders. Clin Pediatr (Phila), 46(7), 604-611.

Nijland, L., Maassen, B., & van der Meulen, S. (2003). Evidence of motor programming deficits

in children diagnosed with DAS. J Speech Lang Hear Res, 46(2), 437-450.

Peter, B., & Stoel-Gammon, C. (2005). Timing errors in two children with suspected childhood

apraxia of speech (sCAS) during speech and music-related tasks. Clin Linguist Phon,

19(2), 67-87.

Peter, B., & Stoel-Gammon, C. (2008). Central timing deficits in subtypes of primary speech

disorders. Clin Linguist Phon, 22(3), 171-198.

Riecker, A., Ackermann, H., Wildgruber, D., Meyer, J., Dogil, G., Haider, H., et al. (2000).

Articulatory/phonetic sequencing at the level of the anterior perisylvian cortex: a

functional magnetic resonance imaging (fMRI) study. Brain Lang, 75(2), 259-276.

Riecker, A., Brendel, B., Ziegler, W., Erb, M., & Ackermann, H. (2008). The influence of

syllable onset complexity and syllable frequency on speech motor control. Brain Lang,

107(2), 102-113.

26 Riecker, A., Mathiak, K., Wildgruber, D., Erb, M., Hertrich, I., Grodd, W., et al. (2005). fMRI

reveals two distinct cerebral networks subserving speech motor control. ,

64(4), 700-706.

Ruark, J. L., & Moore, C. A. (1997). Coordination of lip muscle activity by 2-year-old children

during speech and nonspeech tasks. J Speech Lang Hear Res, 40(6), 1373-1385.

Ruscello, D. M. (2008). Nonspeech oral motor treatment issues related to children with

developmental speech sound disorders. Lang Speech Hear Serv Sch, 39(3), 380-391.

Shriberg, L. D., Aram, D. M., & Kwiatkowski, J. (1997a). Developmental apraxia of speech: I.

Descriptive and theoretical perspectives. J Speech Lang Hear Res, 40(2), 273-285.

Shriberg, L. D., Aram, D. M., & Kwiatkowski, J. (1997b). Developmental apraxia of speech: II.

Toward a diagnostic marker. J Speech Lang Hear Res, 40(2), 286-312.

Steeve, R. W., Moore, C. A., Green, J. R., Reilly, K. J., & Ruark McMurtrey, J. (2008).

Babbling, chewing, and sucking: oromandibular coordination at 9 months. J Speech Lang

Hear Res, 51(6), 1390-1404.

Terband, H., & Maassen, B. (2010). Speech motor development in childhood apraxia of speech:

generating testable hypotheses by neurocomputational modeling. Folia Phoniatr Logop,

62(3), 134-142.

Terband, H., Maassen, B., Guenther, F. H., & Brumberg, J. (2009). Computational neural

modeling of speech motor control in childhood apraxia of speech (CAS). J Speech Lang

Hear Res, 52(6), 1595-1609.

Wilson, E. M., Green, J. R., Yunusova, Y., & Moore, C. A. (2008). Task specificity in early oral

motor development. Semin Speech Lang, 29(4), 257-266.

27 Zimmerman, I. L., Steiner, V. G., & Pond, R. E. (2002). Preschool Language Scale. 4th ed.

Orlando, Fla: Harcourt.

Zwicker, J. G., Missiuna, C., & Boyd, L. A. (2009). Neural correlates of developmental

coordination disorder: a review of hypotheses. J Child Neurol, 24(10), 1273-1281.

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