Are Oral and Verbal Motor Functions Related to Manual and Overall Motor Performance in Children with Developmental Apraxia of Speech?

Transkript

1 Institutionen för klinisk vetenskap, intervention och teknik Logopedprogrammet, kurs 27 Huvudämne Logopedi Examensarbete D-nivå, 30 poäng Vårterminen 2009 Are Oral and Verbal Motor Functions Related to Manual and Overall Motor Performance in Children with Developmental Apraxia of Speech? Författare: Helena Björelius Hort Handledare: Docent Gunilla Henningsson, Leg. Logoped Enheten för logopedi och foniatri, Karolinska Institutet Docent Anita McAllister, Leg. Logoped Enheten för Logopedi, Linköpings Universitet Professor Ann-Christin Eliasson, Leg. Arbetsterapeut Institutionen för kvinnors och barns Hälsa, Karolinska Institutet

2 ABSTRACT The aim of this study was to determine whether or not oral and verbal motor functions are related to manual and overall motor performances in children with developmental apraxia of speech (DAS). Background: DAS is a speech disorder characterized by deficits in speech motor planning for sequenced speech output in the absence of overt neuromuscular impairment. Due to the unclear criteria for diagnosing DAS, data on occurrence differ and researchers have not yet been able to establish the underlying cause of DAS. The motor deficit theory proposes a joint underlying neurophysical system, responsible for motor planning of both speech and other motor functions. Method: 16 children (aged years) with a clear-cut diagnosis of DAS, chosen from a convenient sample, were assessed with a test battery that included tests for oral and verbal function (VMPAC), manual and overall motor performance (BOT-2), oral stereognosis (STORM) and manual stereognosis (SCSIT). The results from each child with DAS were compared with VMPAC standardized means from typically developed children. The raw scores from each subtest of VMPAC and BOT-2 were correlated in all possible combinations. Results: Of the 16 children with DAS, 75% achieved below average of typically developed children in manual and/or overall motor performance areas. The oral and verbal motor function area that the children with DAS found most difficult was sequencing maintenance control. Partial correlation with age as a controlling factor showed tendencies towards significance between fine manual control and sequencing maintenance control (p= 0.053). These results were also confirmed by stepwise regression analyses, showing that fine manual control was the most powerful predictor variable for sequencing maintenance control (p= 0.000) and for connected speech and language control (p= 0.000). These results must be interpreted with caution due to the significant age factor. Conclusion: Children with DAS seem to have deficits in other motor performance areas. Regarding speech motor function they have greatest difficulties with speech sequences. The findings in this study might be seen as a support for the theory of a joint underlying neurophysiological system, responsible for motor planning of both verbal and motor functions. SAMMANFATTNING Syftet med denna studie var att utröna om det fanns samband mellan orala och verbala funktioner och fin- och grovmotorik hos barn med diagnosen dyspraxi. Bakgrund: Dyspraxi är en talstörning karaktäriserad av nedsatt motorisk planering av tal och talsekvenser utan neuromuskulär nedsättning. Forskare har ännu inte funnit någon bakomliggande orsak till dyspraxi och det finns inga klara riktlinjer för diagnostisering vilket ger varierade uppgift om förekomst. Motor-deficit teorin har föreslagit en underliggande neurofysiologisk komponent som är ansvarig för motorplanering av både tal och övriga motoriska funktioner. Metod: 16 barn med diagnosen dyspraxi valdes enligt ett bekvämlighetsurval och blev testade med ett testbatteri som innefattade; oral och verbal function (VMPAC); fin och grovmotorik (BOT-2); oral steregnosi (STORM) och hand stereognosi (SCSIT). Resultaten för barnen med dyspraxi jämfördes med VMPAC s standardiserade medelvärden från typiskt utvecklade barn. Råpoängen från deltesten av VMPAC och BOT-2 korrelerades i samtliga möjliga kombinationer. Resultat: 75% av barnen med dyspraxi presterade under normalfördelningen i finmotorik och/eller grovmotorik. Det orala och verbala motorfunktionsområdet som innebar störst svårigheter var sequencing maintenance control. Genom partiell korrelation hittades ett visst samband mellan deltestet fine manual control och deltesten sequencing maintenance control (p= 0,053). Dessa resultat bekräftades även genom en stegvis regressions analys. Fine manual control var den variabel som hade störst inflytande på sequencing maintenance control (p= 0,000) och connected speech and language control (p= 0,000). Resultaten av dessa fynd bör tolkas med försiktighet beroende på signifikant inverkan av ålders faktorn. Slutsats: Barn med dyspraxi förefaller ha nedsättningar även i andra motoriska färdigheter. Vid tal har de störst svårighet med talrörelser som innebär sekvensering. Fynden i denna studie stödjer eventuellt teorin att det finns en underliggande neurofysiologisk komponent ansvarig för motorplanering av både verbala och motoriska funktioner.

3 INDEX 1 BACKGROUND Introduction Etiology The FOXP2 theory The motor deficit theory Motor performance relationships Phonology and DAS Oral and manual stereognosis Motor performance Tests for oral and verbal motor functions in children with DAS AIM AND QUESTIONS Aim Questions MATERIALS AND METHOD Ethics Participants Drop-outs from the subject group Exclusion criteria Inclusion criteria Materials Test of oral and verbal motor function Procedures for the translations of words and sentences Test for manual and overall motor performance Stereognosis for mouth Stereognosis for hand Participation procedures Procedures for the test group Procedures for the subjects Intra-rater reliability in assessing VMPAC Statistics Statistics for VMPAC Statistics for BOT Statistics for oral and manual stereognosis Statistics for VMPAC and BOT RESULTS Results of VMPAC Descriptive statistics of VMPAC Results for the children with DAS compared with standardization mean for the three subtests (VMPAC) Correlations for all subtests of VMPAC Results of BOT Descriptive statistics of BOT Frequencies and results of the three subtests from BOT Correlations between the three subtests of BOT Results of Oral and Manual Stereognosis Descriptive statistics of oral and manual stereognosis Results for oral and manual stereognosis Results of combined analyses of oral and verbal functions and motor performance Linear regression analyses Partial correlation and stepwise regression analyses DISCUSSION Discussion of method Selection of criteria Assessment of oral and verbal function (VMPAC)... 22

4 5.1.3 Assessment of manual and overall motor performance (BOT-2) Assessment of oral and manual stereognosis Assessment situation Statistics Discussion of results Oral and verbal motor functions in children with DAS Manual and overall motor performance in children with DAS? Relationships between oral and verbal motor functions and manual and overall motor performance in children with DAS Limitations Conclusions Future studies REFERENCES Appendix I (1) Appendix I (2) Appendix II Appendix III (1) Appendix III (2) Appendix IV Appendix V (1) Appendix V (2) Appendix VI Appendix VII Appendix VIII Appendix IX (1) Appendix IX (2)... 42

5 1 Background 1.1 Introduction Developmental apraxia of speech (DAS) is a speech disorder characterized by deficits in speech motor planning for sequenced speech output in the absence of overt neuromuscular impairment; the notion of soft neurological signs is often mentioned as well (Hall, Jordan & Robin, 1993; Davis, 2003). Various denotations (labels) are in use today to circumscribe the symptoms of developmental apraxia in children: developmental apraxia of speech (DAS), childhood apraxia of speech (CAS) and developmental verbal dyspraxia (DVD).The unclear criteria for diagnosing DAS lead to differences in data on occurrence. There is, however, agreement about the impairment being more prevalent among boys, 3 4:1 (Hall et al., 1993; Portwood, 1999). DAS requires a large amount of treatment, which often takes longer than other types of articulatory speech impairment. In order to achieve the desired results, the patient is usually prescribed exercises more or less daily for a number of years (Hall, et al., 1993). This places heavy demands on both the child and the parents. The diagnosis DAS is much disputed. The general ambiguity of the diagnostic criteria for DAS is evident from a report by Forrest (2003), who identified 50 different characteristics that were included in the diagnoses of DAS made by 75 speech-language pathologists. According to The Childhood Apraxia of Speech Association of North America (CASANA), with support from Forrest (2003), some of the key characteristics are limited repertoire of vowels, errors increase with the length or complexity of utterances, such as multi-syllabic or phonetically challenging words. Depending on the level of severity, the child may be able to produce the same target utterance accurately in one context but not in a different context. Additional features are impaired rate/accuracy on diadochokinetic tasks (alternating movement accuracy or maximum repetition rate of same sequences, such as /pa/, /pa/, /pa/, and multiple phoneme sequences, such as /pa/ /ta/ /ka/) and disturbances in prosody, including overall slow rate, timing deficits in duration of sounds and pauses between and within syllables, contributing to the perception of excess and/or equal stress, "choppy" and monotone speech. The search for an instrument with which clinicians can differentiate between children with DAS and children with other speech and language disorders is a very important matter that has been thoroughly addressed by Shriberg, Aram and Kwiatkowski (1997 a, b and c). They suggest that the only linguistic domain which distinguishes a majority of the children with suspected DAS from those with other forms of speech and language disorders is inappropriate stress. These findings, among others, can lead to more accurate diagnosing systems for children with speech disorders. The American Speech and Hearing Association (2007) recently reached consensus on three features that have diagnostic validity: (1) inconsistent error production on both consonants and vowels across repeated production of syllables and words, (2) lengthened and impaired co-articulation transitions between sounds and syllables, and (3) inappropriate prosody. One interesting finding by Skov (2008) is that difficulties and articulatory symptoms found in 12 Danish children with DAS tended to resemble those described in English, American and Swedish studies on DAS. This suggests that DAS is not language-specific. The controversy about diagnosing DAS makes the choice of intervention even more of a challenge for the Speech-Language Pathologist clinician. In a review in the Cochrane Collaboration, Morgan and Vogel (2008) found a critical lack of well-controlled treatment 1

6 studies addressing treatment efficacy for DAS, making it impossible to draw conclusions about which intervention is most effective for treating DAS in children or adolescents. 1.2 Etiology Researchers have not yet established the underlying cause of DAS (Davis, Jacks & Marquardt 2005). As with many other diagnoses, DAS probably includes an inheritable factor. Lewis, Freebairn, Hansen & Gerry-Taylor (2004) report that familial aggregation for speech and language disorders was demonstrated in 86% of the children with speech and language disorder; moreover, 13 out of 22 children had one affected parent. Mothers of the children with DAS demonstrated a higher affection rate than mothers of children with other speech disorders The FOXP2 theory One theory that is gaining ground stems from discoveries of mutations in the FOXP2 gene located in chromosome 7q31. A possible genetic disruption in the FOXP2 gene has been found in the three-generation KE family; all the members of this family who are affected by a pronounced verbal dyspraxia have a mutation of the FOXP2 gene (Vargha-Kadem, Watkins, Price & Ashburner, 1998; Vernes, Nicod, Elahi & Coventry, 2006; Lennon, Cooper, Peiffer & Gundersson, 2007). In a language experimental study, the authors found significant underactivation in Broca s area, among other cortical language-related regions, in the affected members of the KE family relative to members unaffected by the speech disorder. (Liegeois, Baldeweg, Connelly & Gadian, 2003) The motor deficit theory Researchers propose a motor programming deficit in the light of the inconsistency in repeated utterances and the slow speaking rate found in children with DAS (Maasen, Nijland & van der Meulen, 2001; Nijland, & Maasen, 2003). In a study comparing adults with apraxia of speech and typically speaking adults, Strand & McNiel (1996) found that apraxic speakers consistently produced longer vowel and between-word segment durations in sentence contexts than in word contexts, which the authors suggested might indicate a different mechanism for executing motor programs. Nijland & Maasen (2003) investigated articulatory compensation using bite-blocks in a comparison of adults and children with typical speech and children with DAS. The participants produced utterances in normal speaking conditions and in a bite-block condition where the mandible was kept in a fixed position. The results showed that the biteblock condition in typically-speaking children, as in adult women, did not affect the extent of anticipatory co-articulation. In the speech of children with DAS, the bite block had large effects both on co-articulatory patterns and on vowel quality, which, contrary to expectations, was improved. The results were interpreted as a clear demonstration of deficient motor programming in DAS. 1.3 Motor performance relationships There are few reports on children with DAS and possible relations with fine and gross motor impairments. It is noteworthy that quite a few recent studies have investigated possible comorbidity in speech and language disorders and fine and gross motor performance. In a study by Gaines & Missiuna (2006), 18 out of 40 children with speech and language disorders showed evidence of significant motor impairment; 12 of the 18 children were diagnosed with developmental coordination disorder (DCD). Similarly, Webster, Majnemer, Platt & Shevell (2005) found that 52% of children with speech and language disorder had motor impairment. Another study highlighted the possible risk of associated deficits in fine motor function among preschool-aged children with speech-sound disorders. Relationships were found 2

7 between the Oral Movement assessment subtest of KSPT (Kaufman Speech Praxis Test for Children, 1995), the visual motor integration subtest from PDMS-2 (Peabody Developmental Motor Scale, 2000) and the Objective Manipulation subtest (PMDS ), (Newmeyer, Grether, Grasha & White, 2007). Dewey (1993) found similar errors in the motor planning of verbal and motor skills in children identified as having a Developmental Motor Deficit (DMD). In a study of children with DAS, Bradford & Dodd (1996) looked for the possible cooccurrence of deficiencies in oral/verbal and fine motor tasks. They compared speechdisordered children with a typically developed control group on tasks assessing volitional and non-volitional oral movements, fine motor skills and speech-motor planning. Only children (n=5) with DAS had difficulties with fine motor tasks compared to other speech-impairments and to a control group of typically developed children. The authors suggested possible deficits at the levels of integrating sensory information into plan and action, and of coordinating speed and dexterity of intricate movements. Comparing adults with acquired apraxia and children with DAS, Poole, Gallagher, Janosky & Qualls (1997) found that the type of error patterns made in verbal and motor skills were similar in the two groups with different diagnoses. Recently, an interesting study on relationships was conducted in patients with autism spectrum disorders (Gernsbacher, Sauer, Geye, & Schweigert 2008). Regarding relationships between oral and manual functions among patients with autism spectrum disorders, subjects with impaired speech were found to have less capacity for communication with gestures. In a study with neuroimaging and repetitive transcranial magnetic stimulation (rtms), Gentilucci & Volta (2008) suggested that the system governing both speech and gesture is located in Broca s area. Their data support the hypothesis that the hand motor control system is involved in higher order cognition According to speech and language pathologists working at Karolinska University Hospital, Huddinge, clinicians have long been aware that many children with DAS also have difficulties with manual functions. These findings may support the hypothesis of a joint underlying neurophysical system that is responsible for motor planning of both verbal and motor functions (Newmeyer et al., 2007). 1.4 Phonology and DAS Phonological awareness and the auditory ability to discriminate language sounds are very important for grasping language and learning to read and write. Many children with speech impairment and hearing deficits have problems with auditive perception (Raitano, Pennington, Tunick & Boada, 2004; Nettelblad, Samuelsson, Sahlén & Ors, 2008). Lewis, Freebairn, Hansen & Iyengar (2004) found that children with DAS also exhibited co-morbid disorders of reading and spelling. Group comparisons revealed that the DAS group was similar to children with speech and language disorder during preschool years. By school age, however, the group with speech and language disorders displayed more positive changes in language skills than the DAS group. The authors suggested that language disorders persist in the latter children despite partial resolution of articulation problems. 1.5 Oral and manual stereognosis Oral stereognosis is the ability to identify and discriminate shapes intra-orally (Jacobs, Bou Serhal, & van Steenberghe 1998; Norström & Sjöberg, 2003). The term is mainly used when examining oral sensory functions in children and adults with oral motor problems. Calhoun, Gibson, Hartley & Minton (1992) describe oral stereognosis as the identification of a threedimensional object from just a very light touch, two-point discrimination and proprioception. Proprioceptors provide information about the relative positions and movements of the limbs. 3

8 They are activated by stimuli from inside the body, inform the central nervous system of external loading and serve in a variety of functional capacities, including sensation, composite sensory experiences, reflex initiation and modulation of patterned motor behaviour (Calhoun et al., 1992). In studies of oral stereognostic ability, the experimental design of the test is of primary importance as both the method used and the material applied may dramatically influence the results. The form, size and surface characteristics of the test piece, the order of presentation, subject-related factors and the scoring method all affect the results. An oral stereognostic ability test cannot be said to measure stereognostic ability in general (Jacobs et al., 1998). Manual stereognosis is the ability to identify and discriminate shapes manually. It has mainly being used in studies on cerebral palsy to detect sensory deficits (Cooper, Majnemer, Rosenblatt & Birnbaum 1995) or hand impairments and their relationship with manual ability (Arnould, Penta, & Thonnard 2007). According to Landt (1976), there is no clear relationship between manual and oral stereognosis. 1.6 Motor performance Motor skill development should be a key strategy in childhood interventions aiming to promote long-term physical activity (Barnett, van Beurden, Morgan, & Brooks 2009). A cross-sectional study has demonstrated that proficiency in motor skills favours participation in physical activity. According to Bruininx & Bruininx (2005), fine manual control involves motor performance such as control and coordination of the distal musculature of the hands and fingers. Manual coordination involves motor performance, such as control and coordination of the arms and hands, especially for object manipulation. Body coordination involves control and coordination of the large musculature used in maintaining posture, balance, strength and agility, which gives support in casual play, competitive sports, and other physical activities. 1.7 Tests for oral and verbal motor functions in children with DAS In Sweden, no standardized test is available for assessing presumed DAS. Only a few tests exist for measuring oral motor functions and articulation disorders. One of them, NOT-S (Bakke, Bergendal, McAllister & Sjögreen, 2007), is a screening instrument that can be used to identify orofacial dysfunction in individuals from 3 years of age and older. The screening process, which consists of a structured interview and a clinical examination, can be performed by dental and medical professionals. Another test is SVANTE (Svenskt Artikulations och Nasalitetstest) (Lohmander, Borell, Havstam Henningsson, Lundberg & Persson 2005), constructed to assess articulation, resonance, performance and deviances due to oral structural anomalies, with norms for 3, 5, 7 and 10 years of age. In addition there are some assessment protocols, such as STORM (Stockholms Oralmotoriska Bedömningsprotokoll) (Henningsson, McAllister, Hartstein, & Raud Westberg 2008) and ORIS (Holmberg & Bergström, 1996), which measure oral motor dysfunctions but have not yet been standardized. As none of the available Swedish tests meet the demands of this study in terms of appropriate assessment items and being standardized, efforts were made to find a suitable test. In a review, McCauley and Strand (2008) recently demonstrated that only a few tests are diagnostically dependable for motor speech disorders. Verbal motor production assessment for children (VMPAC) (Hayden & Square 1999) was the only test that met McCauley and Strand s operational definition on adequate norms. It also had test-retest and interexaminer reliability as well as content and construct validity. VMPAC is designed to aid in the systematic assessment of the neuromotor integrity of the motor speech system in children 4

9 aged 3 through 12 years who have speech production disorders. It is not a test of articulation or language but a step-by-step assessment of the speech production system at rest and when engaged in vegetative and volitional non-speech and speech tasks. Speech and language pathologists can use the VMPAC in determining whether or not a motor disruption such as dysarthria or DAS is a dimension of a child s speech production disorder (Hayden, 1994; Hayden & Square, 1999). Because one aim was to describe children with DAS in terms of oral and verbal motor functions and another was to look for relationships between those functions and other motor performance areas, it was important to find a test that could be supportive in fulfilling the need for the study. On the basis of the information acquired from McCauley and Strand s review (2008) and the article by Hayden (1994), it was decided to implement the VMPAC in this study. 2 Aim and questions 2.1 Aim The primary aim of the present study was to describe children with DAS (Developmental Apraxia of Speech) in terms of oral and verbal motor functions and manual and overall motor performance. The second aim was to investigate potential correlations between these motor areas. The third aim was to study and correlate sensory functions in terms of tests for oral and manual stereognosis. 2.2 Questions The following questions where formulated: Are there differences in oral and verbal motor functions between children with DAS and the standardized means for typically developed children constituted from the norms in the VMPAC manual? Are there relationships between oral and verbal motor functions in children with DAS? Are there relationships between manual and overall motor performance in children with DAS? Are there relationships between oral and verbal motor functions and manual and overall motor performance in children with DAS? Are there relationships between oral and manual stereognosis? 5

10 3 Materials and Method The study will be incorporated in a larger investigation in cooperation with occupational and physio-therapist Sermin Tükel (ST) from the Department of Women and Child Health, Karolinska University Hospital Solna. All the research data in this cooperation were collected during the present study. 3.1 Ethics An ethical application was approved by EPN (Etiska Prövnings Nämnden) in Stockholm (Protocol 2009/4:2). The parents of the subject and test group received explicit information about the purpose and implementation of the study and how collected data would be stored. Only age, gender and group data will be published. 3.2 Participants The test battery was applied to a pilot group of three children, aged 4, 8 and 10 years, respectively, before testing the subjects. The subjects consisted of a group of 18 children 4 to 10 years of age, five girls and 11 boys, with diagnosed developmental apraxia of speech (R.48.2) who has been treated by the same speech and language pathologist at Karolinska University Hospital, Stockholm, during (Table 1). A group of 11 children (test group), between 4:3 and 6:5 years of age, four girls and seven boys, who according to their parents had a typical development, had not been treated for speech and language delay or impairment and had Swedish as their first language, participated as a group for testing the translated word and sentence task in VMPAC (Fig. 2) Drop-outs from the subject group Of the 18 subjects, one failed the auditory discrimination test and another decided not to participate, so 16 subjects finally participated. 6

11 Table 1. Age, treatment period and number of treatment occasions for the sixteen children with DAS participating in the study. Completed = treatment completed. Age (years:months) Treatment period (years:months) Number of treatment occasions 04:05 0: :10 1: :00 3: :00 2: :07 2: :11 2: :02 2: :01 2: :01 1: :06 2:6 Completed 35 07:10 4: :02 4: :10 3:11 Completed 28 08:10 3:11 Completed 30 10:01 4: : Exclusion criteria To keep the study group as uniform as possible, we excluded children with any supplementary diagnosis, such as marked language impairment, neurological disease, diagnosed or suspected intellectual disability, structural anomalies orally or manually, and phonological disorders according to the assessment of auditory perception for children (Bedömning av språk. B.A.S, Frylmark, 2002), (Fig. 1) Inclusion criteria To be included in this study, all subjects had to have Swedish as their first language and also had to pass an Auditory Discrimination test (B.A.S, Bedömning av språk, Frylmark, OrdAF 2002) assessed by their SLP at the Speech and Language Department, Karolinska University Hospital. The test includes ten pairs of words that the listener has to distinguish auditorily, for example Sporta Spotta, Mor Mår. The test has norms for children aged 5:5 6 years with a maximum score of 10 (Andersson & Schück 1999). A minimum score of 7/10 was set for subjects under the age of 5:0 years to be included (Fig. 1). Auditory Discrimination Test Results :05 04:10 05:00 05:00 05:07 05:11 06:02 07:01 07:01 07:06 07:10 08:02 08:10 08:10 10:01 10:07 Figure 1. Results of the 10-item auditory discrimination test for the sixteen children with DAS. Each item gives one score point. Subjects presented by age (years:months) and score. 7

12 3.3 Materials Test of oral and verbal motor function The Verbal Motor Production Assessment for Children, VMPAC (Hayden & Square 1999), was used to assess non-speech and speech oromotor control. The speech tasks consist of linguistically meaningful and non-meaningful vocal production. The VMPAC uses a form that lists all the items for each subtest. It provides information about how to conduct the assessment and what criteria each item needs. The scores for the child are entered directly on the form. The VMPAC contains five subtests, organised into three main areas and two supplementary areas. All subtests have standardized norms, in percent with the standard deviation, for 3, 4, 5, 6 and 7-12 years of age. The main areas are: 1: Global Motor Control. 2: Focal Oromotor Control 3: Sequencing Maintenance Control The supplementary areas are: 4: Connected Speech and Language Control 5: Speech Characteristics The subtests that were implemented in this study were: 2: Focal Oromotor Control 3: Sequencing Maintenance Control 4: Connected Speech and Language Control The two subtests that were excluded are used primarily for the differential diagnosis of dysarthria and DAS, which was not an aim of this study. Another reason for excluding them was the low age of some of the participants and their capacity to endure the time and burden of tasks for the whole study. 2: Focal Oromotor Control area consists of 46 items and assesses the child s volitional oromotor control for three individual subsystems: mandibular, labial-facial, and lingual control, both in isolation and in combination with each other. The items comprise non-speech oromotor movements (17 items) and speech oromotor movements (29 items). Examples of Non-speech Oromotor Movement are assessed by the clinician asking the subject to show me how you kiss (vertical plane/labial facial protrusion), show me how you bite (horizontal plane/jaw), and show me how you smile (vertical plane/labial facial retraction). Examples of Speech Oromotor Movements (single Oromotor-Phoneme) are movements when the child produces one oromotor posture at a time in response to requests to say [a] or say [k]. Double and Triple Oromotor-Phoneme Movements: the child produces two or three oromotor postures in succession in response to requests to say [u-i] or say [pa-ta-ka]. In Oromotor Production of Word Sequences and Sentences, the clinician observes the precision of motor control in meaningful words as well as in a sequence and in simple sentences when the child produces three- and four-word sequences and five-word sentences (see ) (App. VIII). Production of the words and sentences is supported by stimulus cards. 8

13 Focal Oromotor Control scores the child s ability in motor control, with a maximum score of 2 points and a minimum of 0 in three possible modalities, depending on the modality in which the child is responding: (a) Auditory, (b) Visual and (c) Tactile (Table 2). The modality in which the child responds serves as a guideline to the clinician in choosing the most appropriate form of intervention. In this study it was decided to use the three modalities in non-speech tasks but only the auditory modalities for the speech tasks. If a subject made an articulatory error in the production of a vowel and consonant, she/he was scored minus 1 point when the test manual maximum was 2 points. Items (consistency score and motor control score) are scored as accurate (1 point) or inaccurate (0 points). 3: Sequencing Maintenance Control area consists of 23 items, constructed to assess the child s ability to produce non-speech and speech movements in the correct sequential order (Sequence Maintenance). The items comprise both non-speech and speech movements. They have combinations of Double and Triple Oromotor-Phoneme Movement sequences in Word Sequences and Sentences. The tasks are the same as in the Focal Oromotor Control area and the clinician examines the child to determine whether or not the correct order or sequence of movements is produced. Sequencing Maintenance Control scores the child s ability to produce non-speech and speech movements in the correct sequential order, with maximum 2 points and minimum 0 points (Table 2). 4: Connected Speech and Language Control area assesses the child s motor control as it varies with the complexity of language formulation. The area contains 5 items. The child formulates a story that corresponds to four sequenced pictures. The precision and coordination of jaw, lip and tongue movements and their interaction, such as co-articulation, are closely observed during the task. Connected Speech and Language Control in spontaneous speech scores the child s abilities in Motor Control, with maximum 7 points and minimum 0 points, and the presence or absence of correct spoken language (Syntax), with maximum 6 points and minimum 0 points. Table 2. Examples of scoring in the assessment of a subject using the VMPAC. A score of 0 indicates either no response or severe articulatory errors; 1 point indicates less severe articulatory errors; 2 points indicate no articulatory errors. Item: Part 1 Part 2 Sequence Motor Control Score Maintenance Score Auditory Visual Tactile 33. Show me how to bite and blow a. 0 b. 2 c. 2 d Part 1. Say /u-i-a/ a. 2 b. 2 c Part 2. Say /u-i-a, u-i-a, u-i-a, u-i-a/ d. 1 Sum: 2 Sum: 4 Sum: 4 MC Score sum: 10 SM Score sum: 3 The score sums for each subtest are transformed into percentage scores in the VMPAC form. The percentage for each subtest is plotted in the form on a graph that is appropriate for the 9

14 child s age and shows a curve of the child s abilities. From this curve the clinician can discern the subtest(s) where the child has problems and from that information draw conclusions about a possible diagnosis Procedures for the translations of words and sentences The words and sentences that are used in the VMPAC were translated into Swedish by a committee formed for this purpose, comprising the author, a speech and language pathologist and the supervisors Gunilla Henningsson and Anita McAllister. The translation of words and sentences was done with the aim to find articulatory variables that matched the oral and verbal motor control movements of the original items as closely as possible. Another aim was to match the semantic and non-semantic contexts of the words and sentences, which in most cases proved to be difficult and in some cases impossible. Effort towards solving the problem was made by keeping all the articulatory variables but making allowances for the variables to change position between the items (App. VIII). The VMPAC stimuli cards for assessing word and sentence tasks were not compatible with the Swedish translations. New stimuli cards were therefore created by the author to suit the Swedish version of words and sentences (App. IX). The translations were sampled on a test group of children age 4:3 to 6:5 years (see 3.4.1). Word sequence: Item 61. Look here is a pea with some tea holding a key. Say, pea, tea, key. Titta, Nanna får be om te som Dadda vill ge. Säg, be, te, ge. Sentence: Item 65. Look, Dad sat on a mat. Say, dad sat on a mat. Titta, Dadda satt på en matta. Säg, Dadda satt på en matta Test for manual and overall motor performance Fine motor skills were assessed with the Bruininx & Oseretsky Test of Motor Profiency, Second Edition (2006) (BOT-2). This test was developed to determine motor ability in individuals between 4 and 21 years of age. It has 8 subtests, normative interpretations of each subtest and composite scores. Clinical validity and reliability have been shown (Bruininx & Bruininx, 2005). Fine manual control, manual coordination motor composites and the short form of BOT-2 were used in this study to determine children s strengths and weaknesses as regards fine motor skills and overall motor performance. All subtests and the short form of BOT-2 have standard scores and percentiles dependent on male and female norms. A standard score of signifies average motor performance, a score of average motor performance (-1 SD), and a score of 30 or less well below average motor performance (-2 SD) according to BOT-2 norms. 1. Fine Manual Control Motor Composite consists of two subtests: Fine Motor Precision and Fine Motor Integration. 10

15 (a) Fine Motor Precision (e.g., cutting out a circle, connecting dots) has 7 items, the object of each item being to draw, cut or fold within a specified boundary: Performance is evaluated on how well the examinee remains within the boundary. (b) Fine Motor Integration (e.g., copying a star, copying a square) has 8 items, each of which requires the examinee to reproduce drawings of various geometric shapes that range in complexity from a single circle to overlapping pencils. It has a multi-faceted scoring approach in which characteristics of each geometric shape are scored separately. 2. Manual Coordination Motor Composite consists of Manual Dexterity and Upper-limb Coordination subtests. (a) Manual Dexterity (e.g., transferring pennies, sorting cards, stringing blocks) has 5 items and uses goal-directed activities that involve reaching, grasping and bimanual coordination with small objects. The emphasis is on accuracy but the items are timed and the examinee is told to perform the task as quickly as possible. (b) Upper-Limb Coordination (e.g., dropping and catching a ball, throwing a ball at a target) has 7 items and consists of activities designed to measure visual tracking with coordinated arm and hand movements. 3. Short Form is used for screening overall motor performance. It consists of 14 items that represent all the 8 subtests (Fine Motor Precision, Fine Motor Integration, Manual Dexterity, Upper-limb Coordination, Bilateral Coordination, Balance, Running Speed and Agility, Strength) Stereognosis for mouth The oral sensory function was assessed in terms of a stereognostic examination as part of STORM (Stockholms oralmotoriska bedömningsprotokoll). (Henningsson et al., 2008). With vision occluded, the subject is required to identify a flat metal (cobolt chrome) shape in the anterior part of the mouth and then point to its match among pictured shapes. Four shapes (star, triangle, circle and half circle in two sizes, = eight items) are separately presented to the subjects in a random order. The scores are correct (1 point) and incorrect responses (0 points). The oral stereognosis assessment has normative values for ages 4:0 5:5 years (Norström & Sjöberg, 2003) Stereognosis for hand The Manual Form Perception Test (MFPT) was used to assess stereognosis for hand. With vision occluded, the subject feels a plastic shape in one hand and points with the other hand to its matched form among a group of pictured shapes. Twelve shapes are separately presented to the subject in a given order according to test prescriptions. The scores are correct (1 point) and incorrect (0 points) (Ayres, Southern California Sensory Integration Tests: Manual. Los Angeles: Western Psychological Services). 11

16 3.4 Participation procedures Procedures for the test group A letter was sent to the heads of some day-care centers in Stockholm for permission to make announcements to the parents about the study and invite their children to participate as part of a test group (App. I). When the head of a day care centre granted permission (App. II) information about the study was posted on the day care centres notice board. The parents were then sent an information letter, including a form for informed consent (App. III & IV). The author (HBH) phoned the parents to give them an opportunity to have all their questions answered. If the parents agreed to participate with their child in the test group, they signed the consent form and returned it. The participants in the test group were tested individually at their day-care centre with items 61 66, Oromotor Production in Words and Sentences, of the VMPAC, translated into Swedish (App. VIII; IX). The testing took approximately 10 minutes/child and was audio recorded with iaudio U3 (Cowon). After listening to the audio-recorded material, the results were processed and listed into Excel. The results of the test group, measured at group level in percent, were the same or higher than the standardized means from the VMPAC manual (Fig. 2). The translated words and sentences were therefore implemented in the VMPAC assessment. The test group (n=11) 100% 90% 80% 70% 60% 50% 40% 30% 20% 10% Items WSeq MC sc Items WSeq SM sc Items 65 & 66 Sen MC sc Items 65 & 66 Sen SM sc Figure 2. Results in percent for items 61 66, translated words and sentences, assessed in 11 typically developed children in comparison with standardized means from VMPAC. W Seq MC sc (Word Sequence Motor Control score) 90% and 93%; W Seq SM sc (Word Sequence Maintenance score) 99 percent and 81 %; Sen MC sc (Sentence Motor Control Score) 100 percent and 93 % and Sen SM sc ( Sentence Sequence Maintenance Score) 100% and 81%.. 12

17 3.4.2 Procedures for the subjects All information and assessment procedures of the study and tests of the controls were handled by the author. Occupational and physio-therapist (ST) performed the video recording and the interpretation of results for BOT 2 and manual stereognosis. First, a letter was sent to the parents of the subjects with information about the study and asking whether they would be interested in letting their child participate (App. V). Another letter with information about the study was included, addressed to subjects from 7 years of age (App. VI). Second, the parents were contacted by phone and given an opportunity to put questions directly to the investigator/author. Third, when the parents had agreed to let their child participate, a letter was sent to them with a form for informed consent that both parents signed and returned (App VII). The parents of the subjects were also asked whether or not they could accept the use of the video recording of their child for educational purposes for speech and language students. If the parents did not want the video recording to be used for educational purposes, the video will be erased when the study has been completed. The subjects came to the Speech and Language Department at Karolinska University Hospital, Huddinge, to undergo the tests. Four subjects who did not have convenient transport facilities were tested in their homes. The subject was seated on one side of a table and a camera was positioned at an appropriate angle on the opposite side. The examiner (HBH) was seated beside the table to assess the child. The assessments took approximately 1 hour/child. The assessment was video recorded (Digital video, Canon, F5100) by ST so as to be able to certify the test results. There was a break for refreshments midway through the test period. The order of the assessments varied, depending on the subject s individual needs, mainly the capacity to concentrate. After watching the video recordings, the results from the VMPAC were processed by HBH and the results from BOT-2 were processed by ST after the test session. The results were listed into Excel files. 3.5 Intra-rater reliability in assessing VMPAC The intra-rater reliability was measured by the use of correlations. Test data scores from the VMPAC s three areas focal oromotor control; sequencing maintenance control and connected speech and language control were reanalyzed from video recordings of two subjects and tested for correlation using Spearman s rho. A significant correlation was found (p= 0.001). Table 3 Results for intra-rater reliability of the first and second analyses from VMPAC of subjects 1 and 11 in percent. Subject Age Analysis Focal Oromotor Control Sequencing Maintenance Control Connected Speech & Language Control S01 04:10 1 st 79% 59% 78% 2 nd 82% 54% 78% S11 05:11 1 st 86% 57% 80% 2 nd 83% 57% 78% 13

18 3.6 Statistics All research data were fed into Excel files and transferred to SPSS (Statistical Package for the Social Sciences) (17.0) for further analyses. In most of the analyses, non-parametric tests were chosen because of the small sample size and the large age range, between 4:5 to 10: Statistics for VMPAC Descriptive statistics was used to show median, minimum and maximum and the Wilcoxon signed-ranks test was used to analyse possible differences between group results of focal oromotor control, sequencing maintenance control and connected speech and language control in data percentage scores (Fig. 3). The Wilcoxon signed-ranks test was used to analyse possible differences between group results for focal oromotor control, sequencing maintenance control and connected speech and language control and standardizes mean of typically developed children (VMPAC) from each subtest in data percentage scores (Figs. 4, 5 and 6). Spearman s rho correlation coefficient was used to analyse possible relationships between focal motor control, sequencing maintenance control and connected speech and language control in data percentage scores (Table 4) Statistics for BOT-2 Descriptive statistics was used to show median, minimum and maximum (Fig. 7). Spearman s rho correlation coefficient was used to analyse possible relationships between fine manual control; manual coordination and overall motor performance in standard scores (Table 6) Statistics for oral and manual stereognosis The Wilcoxon signed-ranks test was used to analyse possible differences between oral and manual stereognosis in data percentage scores (Fig. 9). Linear regression was used to analyse possible relationships between oral and manual stereognosis in data percentage scores (Fig. 10) Statistics for VMPAC and BOT-2 Linear regression was used to analyse possible relationships between group results for focal oromotor control, sequencing maintenance control, connected speech and language control, fine manual control, manual coordination and overall motor performance in raw scores (Table 7). Partial correlation with age as the control variable was used to analyse possible relationships between group results for focal oromotor control, sequencing maintenance control, connected speech and language control, fine manual control, manual coordination and overall motor performance in raw scores (Table 8). Stepwise regression analyses were carried out to confirm the results of the partial regression analyses (Table 9). 14

19 4 Results 4.1 Results of VMPAC Descriptive statistics of VMPAC Figure 3. Oral and verbal functions for the sixteen children with DAS in focal oromotor control, sequencing maintenance control and connected speech and language control (median, minimum, maximum, 25% and 75% in percent). Figure 3 shows the results for the sixteen children with DAS in the three subtests from VMPAC, converted into percentage scores. The minimum and maximum values for focal oromotor control were 72 and 95, respectively (median= 88); for sequencing maintenance control they were 46 and 93 (median= 68.5); and for connected speech and language control they were 71 and 100 (median 90). The Wilcoxon signed-ranks test shows that the group of children with DAS were more impaired in sequencing maintenance control than in either focal oromotor control, (Z = 3.414, p = 0.001) or connected speech and language control (Z = - 3,519, p = 0.000) (Fig. 3) Results for the children with DAS compared with standardization mean for the three subtests (VMPAC) To be able to compare the results of the three subtests for the children with DAS (n=16) with typically developed children, the standardized mean from VMPAC (n=1040) is also presented in Figures 4, 5 and 6. Both the standardized mean from VMPAC and the children with DAS showed an age-related increase in performance. 100% 80% 60% 40% 20% 0% 04:05 04:10 Age Subtest 2. Focal Oromotor Control 05:00 05:00 05:07 05:11 06:02 07:01 07:01 07:06 07:10 08:02 08:10 08:10 10:01 10:07 Subject's performance Standardization mean Figure 4. Comparison of focal oromotor control between age-related standardized mean values for 1040typically developing children and the sixteen children with DAS in percent related to age; arrows indicate completed treatment. 15

20 For focal oromotor control, the results for the children with DAS ranged from 20 % below to much the same as for typically developed children (see Figure 4). The two children aged 10 had the same result and the children aged 7:6; 8:10 and 8:10 who completed treatment had the same or almost the same result as the standardized means. However, at group level, the Wilcoxon signed-ranks test showed that this difference was statistically significant (Z = 3.235, P = 0.001). Subtest 3. Sequencing Maintenance Control 100% 80% 60% 40% Subject's performance Standardization mean 20% 0% 04:05 04:10 05:00 05:00 05:07 05:11 06:02 07:01 07:01 07:06 07:10 08:02 08:10 08:10 10:01 10:07 Age Figure 5. Comparison of sequencing maintenance control between the age-related standardized mean values for 1040 typically developing children and the sixteen children with DAS in percent related to age; arrows indicate completed treatment. For sequencing maintenance control, the results for the children with DAS ranged from 35% below to much the same as for typically developing children (see Figure 5). Two of the children aged 8:10 who had completed treatment had a slightly lower or the same result, while for the child aged 7:6 who had completed treatment the result for sequencing maintenance control was still 15 % below the standardized mean. At group level, the Wilcoxon signedranks test showed that this difference was statistically significant (Z = 3.414, P = 0.001). 100% 80% 60% 40% 20% 0% Age 04:05 04:10 05:00 Subtest 4. Connected Speech & Language Control 05:00 05:07 05:11 06:02 07:01 07:01 07:06 07:10 08:02 08:10 08:10 10:01 10:07 Subject's performance Standardization mean Figure 6. Comparison of connected speech and language control between the age-related standardized mean values for 1040 typically developing children and the sixteen children with DAS in percent related to age; arrows indicate completed treatment. For connected speech and language control, the results for the children with DAS ranged from 20 % below to the same as for typically developing children (see Figure 6). The performance of the three children aged 7:6; 8:10 and 8:10 who had completed treatment matched that of 16