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International Journal of Speech-Language Pathology Publication details, including instructions for authors and subscription information: http://www.tandfonline.com/loi/iasl20
Motor speech impairment, activity, and participation in children with cerebral palsy ab
Cristina Mei , Sheena Reilly
abc
abc
, Dinah Reddihough
, Fiona Mensah
abc
& Angela
abc
Morgan a
Murdoch Childrens Research Institute, Melbourne, Australia
b
Department of Paediatrics, University of Melbourne, Melbourne, Australia
c
Royal Children’s Hospital, Melbourne, Australia Published online: 09 Jun 2014.
Click for updates To cite this article: Cristina Mei, Sheena Reilly, Dinah Reddihough, Fiona Mensah & Angela Morgan (2014) Motor speech impairment, activity, and participation in children with cerebral palsy, International Journal of Speech-Language Pathology, 16:4, 427-435 To link to this article: http://dx.doi.org/10.3109/17549507.2014.917439
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International Journal of Speech-Language Pathology, 2014; 16(4): 427–435
INVITED ARTICLE
Motor speech impairment, activity, and participation in children with cerebral palsy
CRISTINA MEI1,2, SHEENA REILLY1,2,3, DINAH REDDIHOUGH1,2,3, FIONA MENSAH1,2,3 & ANGELA MORGAN1,2,3
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1Murdoch
Childrens Research Institute, Melbourne, Australia, 2Department of Paediatrics, University of Melbourne, Melbourne, Australia, and 3Royal Children’s Hospital, Melbourne, Australia
Abstract The present study used a population-based sample of children with cerebral palsy (CP) to estimate the prevalence of motor speech impairment and its association with activity and participation. A sample of 79 Victorian children aged 4 years 11 months to 6 years 5 months was recruited through the Victorian CP Register. The presence of motor speech impairment was recorded using the Viking Speech Scale (VSS). Activity and participation outcomes included speech intelligibility (the National Technical Institute for the Deaf rating scale, NTID), the Functional Communication Classification System (FCCS) and Communication Function Classification System (CFCS). A parent completed rating scale was used to examine the association between motor speech impairment and participation. Ninety per cent (71/79) of children demonstrated a motor speech impairment. Strong associations were found between the VSS and NTID ( .001), CFCS ( .001), and FCCS levels ( .001). VSS levels III–IV were significantly associated with restrictions in home, school, and communitybased participation as perceived by parents. Although some diversity in activity and participation outcomes was observed within specific VSS levels, the results of this study suggested that children with mild motor speech impairments are more likely to demonstrate superior activity and participation outcomes compared to children with moderate or severe deficits.
Keywords: Cerebral palsy, motor speech disorders, activity and participation.
Introduction The motor limitations associated with cerebral palsy (CP) pose many challenges during development, including the production of clear and intelligible speech. Recent population-based data suggest that 33–63% of children with CP experience motor speech impairment (Andersen, Mjoen, & Vik, 2010; Cockerill, Elbourne, Allen, Scrutton, Will, McNee, et al., 2014; Nordberg, Miniscalco, Lohmander, & Himmelmann, 2013; Parkes, Hill, Platt, & Donnelly, 2010). At present, there is no consensus on how communication impairment (affecting speech or language) should be defined in children with CP (Hidecker, Paneth, Rosenbaum, Kent, Lillie, Eulenberg, et al., 2011). Consequently, studies have used various definitions to examine the occurrence of motor speech impairment, including level of speech intelligibility (Cockerill et al., 2014) and whether children’s speech is indistinct (Andersen et al., 2010). In some instances, a clear definition is not reported.
Several classification systems have been developed recently that provide a common means to measure motor speech impairment and communication functioning at an activity and participation level. These include the Viking Speech Scale (VSS; Pennington, Virella, Mjoen, da Graça Andrada, Murray, Colver, et al., 2013b), the Functional Communication Classification System (FCCS; Barty & Caynes, 2009), and the Communication Function Classification System (CFCS; Hidecker et al., 2011). These scales are specifically designed for children with CP and correspond to various aspects of the International Classification of Functioning, Disability, and Health model (ICF; World Health Organization, 2001). For instance, the VSS specifically focuses at the impairment and activity levels of speech, while the FCCS and CFCS measure communication at an activity and participation level to determine the functional or daily impact of impairments. The CFCS classifies communication according to how effectively an individual sends and receives messages with familiar and unfamiliar communication partners. In comparison,
Correspondence: Ms Cristina Mei, BSpPath (Hons), Murdoch Children’s Research Institute, Flemington Road, Parkville, Melbourne, 3052, Australia. Email: [email protected] ISSN 1754-9507 print/ISSN 1754-9515 online © 2014 The Speech Pathology Association of Australia Limited Published by Informa UK, Ltd. DOI: 10.3109/17549507.2014.917439
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communication is classified by the FCCS according to a child’s ability to communicate messages/topics and needs/wants and takes into consideration both the familiarity of the communication partner and setting. The activity and participation restrictions specifically associated with motor speech impairment in children with CP have not been determined. Within this population, children’s abilities have largely been characterized at an impairment level; however, recent studies have incorporated activity and participation outcomes. Using the social function domain of the Paediatric Evaluation of Disability Inventory (PEDI), McFadd and Hustad (2013) reported that isolated motor speech impairment (in the absence of language impairment) did not result in significantly reduced social outcomes compared to children with CP with no speech motor involvement. Social restrictions were only evident when language abilities were also affected (McFadd & Hustad, 2013). This led the authors to conclude that the PEDI is sensitive to language impairment but not to reductions in speech intelligibility. However, the authors did not investigate the association between severity of motor speech impairment and social function. A growing body of evidence has highlighted the activity and participation restrictions associated with anarthria or complex communication needs (Clarke, Newton, Griffiths, Price, Lysley, & Petrides, 2011; Clarke, Newton, Petrides, Griffiths, Lysley, & Price, 2012; McFadd & Hustad, 2013; Raghavendra, Virgo, Olsson, Connell, & Lane, 2011; Thirumanickam, Raghavendra, & Olsson, 2011). Whether these limitations are also associated with mild or less severe forms of motor speech impairment is unknown. Children’s participation within various environments (e.g., home, school, and community) is influenced by multiple variables (Colver, Thyen, Arnaud, Beckung, Fauconnier, Marcelli, et al., 2012; Forsyth, Colver, Alvanides, Woolley, & Lowe, 2007; Hammal, Jarvis, & Colver, 2004; Lawlor, Mihaylov, Welsh, Jarvis, & Colver, 2006). One factor that is likely to play an important role in shaping the participation of children with motor speech impairment is their level of intelligibility. Speech intelligibility is influenced by the familiarity of the listener (Flipsen, 1995). Therefore, an assumption could be made that the participation of children with reduced intelligibility is affected more during activities outside of the home or with unfamiliar people than in the home or with familiar people. However, research suggests that even social outcomes within the home are likely to be reduced due to poor speech intelligibility (Pennington & McConachie, 2001). The impact of a child’s level of speech intelligibility on participation in various contexts (home, school, and community) has not been widely investigated. Investigation of participation across settings has focused predominately on children with severe communication difficulties where participation is lower in frequency
and restricted to certain settings (Clarke et al., 2012; Raghavendra et al., 2011; Thirumanickam et al., 2011). Until recently, a core challenge in investigating the broader effects of motor speech impairment in children with CP has been the lack of measures specific to this population addressing activity and participation, or, more specifically, communicative participation.1 The CFCS and FCCS are activity and participation measures that are designed for children with CP. As these scales were developed recently, limited research is available regarding their use, and little is known about the association between motor speech impairment and CFCS and FCCS levels (Virella, Pennington, da Graça Andrada, Greitane, Prasauskiene, Rackauskaite, et al., 2012). Identifying these associations is important for establishing the communication profiles of children with CP and understanding the functional therapeutic needs associated with varying degrees of motor speech involvement. To this end, the present study used a population-based sample of children with CP to (1) estimate the frequency of motor speech impairment using the VSS; (2) examine the association between VSS, speech intelligibility, CFCS, and FCCS ratings; and (3) examine the association between VSS ratings and parent ratings of children’s participation within the home, school, and community.
Methods Participants Participants were recruited through the Victorian Cerebral Palsy Register (VCPR). The VCPR contains information about all individuals with CP living or born in the state of Victoria since 1970. The VCPR identifies cases of CP by reviewing the medical records of children attending two tertiary paediatric hospitals in Victoria and through referrals from clinicians and families (Reid, Carlin, & Reddihough, 2011). The birth range that was of interest to this study was 25 August 2005 to 24 August 2007 (the age range of 5–6 years was chosen as by these ages children typically demonstrate a substantial degree of functional speech and language skills). Children with CP due to post-neonatal injury (occurring up to 2 years of age) were included. Based on these criteria, 232 eligible children were identified. Of these, 176 (76%) were contacted (six children had died and 50 were unable to be contacted). Consent to participate in the study was obtained for 84 children, representing 48% of the contacted families and 37% of the known living population of this age group on the VCPR. The presence/absence of motor speech impairment could not be confirmed for five children due to respiratory infections (n 4) or the inability to obtain a speech sample (n 1). This resulted in a sample size of 79 children. Ethics
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Motor speech impairment associated with CP approval for the study was obtained from Human Research Ethics Committees at The Royal Children’s Hospital (#30048) and Southern Health (#11380), Melbourne. Participants were aged between 4 years 11 months and 6 years 5 months (mean 5.4, SD .5) at the time of their assessment. Key characteristics of the 79 participants are presented in Table I. All motor types, Gross Motor Function Classification System (GMFCS; Palisano, Rosenbaum, Walter, Russell, Wood, & Galuppi, 1997) levels, and Manual Ability Classification System (MACS; Eliasson, KrumlindeSundholm, Rosblad, Beckung, Arner, Ohrvall, et al., 2006) levels were represented. Thirteen participants (15%) were classified as having mixed CP, which included spastic-dystonia (n 9), spastic-athetoid (n 1), spastic-hypotonia (n 1), ataxic-hypotonia (n 1), and ataxic-spastic (n 1). Language impairment was identified in 65% (51/79) of children. To determine the representativeness of the recruited sample, characteristics of the participants were compared against those of the non-participants. Non-participants were children who were not contacted by the VCPR, declined study participation, were lost to follow-up, or for whom the presence/ absence of motor speech impairment could not be determined. It excluded the six children that had died. Participants and non-participants were compared across key variables that included CP motor type and distribution, GMFCS level, and associated impairments. The chi-square test was used to determine statistically significant differences between participants and non-participants, as indicated by a p-value .05. As seen in Table I, participants included in the study did not differ significantly to non-participants (n 147) in terms of CP type and distribution, GMFCS level and the presence of epilepsy, hearing, vision, and cognitive impairment (Table I).
Measures The following measures were completed during a comprehensive face-to-face assessment of communication that included a range of further speech and language measures not reported on here. Ratings were completed as per the user guidelines for each scale. Motor speech impairment, speech intelligibility, and functional communication ratings were completed by the examiner (CM) and the child’s parent. Motor speech impairment. Motor speech impairment was recorded using the Viking Speech Scale (VSS), a newly developed speech classification system specifically designed for children with CP over 4 years of age (Pennington et al., 2013b). The VSS consists of the following four levels: (I) speech is not affected; (II) speech is imprecise but usually under-
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Table I. Characteristics of participants (n 79) and nonparticipants (n 147). Participants, Non-participants, n (%) n (%) p-value Gender Female Male CP motor type Spastic Dyskinesia Hypotonia Ataxic Mixed Unknown CP distribution Monoplegia Hemiplegia Diplegia Triplegia Quadriplegia Unknown GMFCS level I II III IV V Unknown MACS levela I II III IV V Epilepsy No Yes Resolved Unknown Cognitive impairmentb No Yes Unknown Hearing impairment No Yes Unknown Vision impairment No Yes Unknown Language impairmentc No Yes Unknown
34 (43) 45 (57)
62 (42) 85 (58)
.90
61 1 3 1 13 0
(77) (1) (4) (1) (16) (0)
123 1 4 2 15 2
(84) (1) (3) (1) (10) (1)
.69
1 29 23 1 25 0
(1) (37) (29) (1) (32) (0)
2 58 44 3 38 2
(1) (39) (30) (2) (26) (1)
.93
31 14 12 15 7 0
(39) (18) (15) (19) (9) (0)
54 34 12 22 14 11
(37) (23) (8) (15) (10) (7)
.50
28 27 11 7 6
(35) (34) (14) (9) (8)
61 17 1 0
(77) (22) (1) (0)
— — — — — 104 36 1 6
(71) (24) (1) (4)
.74
49 (62) 22 (28) 8 (10)
59 (40) 40 (27) 48 (33)
.21
73 (92) 6 (8) 0 (0)
105 (71) 18 (12) 24 (16)
.13
52 (66) 27 (34) 0 (0)
67 (46) 47 (32) 33 (22)
.32
27 (34) 51 (65) 1 (1)
— — —
p-value calculated using the chi-square test. GMFCS, Gross Motor Function Classification System level (Palisano et al., 1997); MACS, Manual Ability Classification System (Eliasson et al., 2006). aMACS levels for non-participants are not reported due to the high percentage of missing data on the Victorian Cerebral Palsy Register. bCognitive status (recorded as “no impairment”, “probably no impairment”, “probably some impairment”, “unknown”) was based on data obtained by the Victorian Cerebral Palsy Register. cLanguage data not available for non-participants. Language was measured using the Preschool Language Scale, 4th edition (Zimmerman, Steiner, & Pond, 2002). Language impairment was defined as a standard score greater than one standard deviation below the mean on the Auditory Comprehension and/or Expressive Communication sub-scales.
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standable to unfamiliar listeners (e.g., imprecise articulation, breathy or harsh voice); (III) speech is unclear and not usually understandable to unfamiliar listeners out of context (e.g., difficulties controlling breathing for speech, small range of consonants produced); and (IV) no understandable speech. The identification of motor speech impairment was based on a rating of II–IV. Speech intelligibility. The National Technical Institute for the Deaf (NTID) rating scale (Johnson, 1975) was used to measure children’s connected speech intelligibility (samples were obtained during a parent–child free-play interaction). The scale’s use can extend beyond its original population of interest (i.e., hearing impaired children) (Kent, Miolo, & Bloedel, 1994) and has previously been used with children with motor speech disorders (Liégeois, Morgan, Cross, Stewart, Vogel, & Vargha-Khadem, 2010). The five levels of the NTID rating scale are summarized as follows: (1) speech is unintelligible; (2) only isolated words or phrases are intelligible; (3) half of the message is understood; (4) speech is intelligible with some exceptions; and (5) speech is completely intelligible. The NTID scale is similar to the VSS in that they both classify speech intelligibility; however, they differ in that the VSS also takes into consideration the familiarity of the communication partner, the need for contextual cues, and the child’s impairment within each of the motor speech sub-systems. Functional communication. Children’s communicative abilities at an activity and participation level were measured using the CFCS (Hidecker et al., 2011) and the FCCS (Barty & Caynes, 2009). Each scale has five-levels and is designed specifically for children with CP. The CFCS consists of the following levels: (I) effective sender and receiver; (II) effective but slower paced sender and/or receiver; (III) effective sender and receiver with familiar partners only; (IV) inconsistent sender and/or receiver with familiar partners; and (V) seldom effective sender and receiver, even with familiar partners. The levels of the FCCS include: (I) an effective communicator in most situations; (II) an effective communicator in most situations with help; (III) an effective communicator in some situations; (IV) assistance required in most situations, particularly with unfamiliar people and environments; and (V) communicates unintentionally using movement and behaviour. Inter-rater agreement (weighted kappa) between examiner and parent ratings for the above scales was moderate-to-high (VSS .72, 95% confidence interval [CI] .68–.74; NTID .82, 95% CI .73–.90; CFCS .61, 95% CI .54–.67; FCCS .74, 95% CI .65–.84). Participation within the home, school, and community. The ease with which children were able to partici-
pate because of their ability to communicate was rated by parents. This measure aimed to address children’s overall participation, whereas the previously described measures focused on communicative activity and participation. Participation within the home, school, and community were each rated using a 10-point scale (see Appendix). For each of the scales parents were asked to rate “how easy it is for your child to participate because of their ability to communicate”. A score of 1 corresponded to “extremely easy” and a score of 10 represented “extremely difficult”. The three scales formed part of an activity and participation questionnaire completed by the child’s parent during the face-to-face assessment. This questionnaire was developed by the research team and was based on individual interviews conducted with 13 parents of children with CP aged 4–9 years (Mei, Morgan, Reilly, Reddihough, Mensah, Green, et al., 2012). Participation was described to parents as their child’s involvement in life situations as per the ICF. Examples of participation provided to parents included interacting or playing with others and being socially involved in activities or events. Analysis Associations between children’s ratings for motor speech impairment (VSS) with speech intelligibility (NTID) and functional communication (CFCS and FCCS) were examined using contingency tables and the Chi-squared test. A one-way analysis of variance (ANOVA) was used to compare children’s participation (as measured by the 10-point scales) according to VSS and NTID levels. Mean scores on the 10-point scales were compared across each level, with VSS level I and NTID level 5 (children without impairment) as the reference groups. Statistically significant associations were indicated by a p-value .05.
Results Frequency of motor speech impairment Motor speech impairment was identified in 90% (71/79) of children based on a VSS rating of II–IV. Fifty-four per cent (43/79) of children demonstrated mild deficits (level II). The proportion of children classified at the remaining levels was 10% (8/79, level I), 11% (9/79, level III), and 24% (19/79, level IV). Functional communication and speech intelligibility Ratings from the CFCS, FCCS, and the NTID speech intelligibility scale are presented in Table II. Just over half of the sample (54%, 43/79) demonstrated highly intelligible speech, corresponding to levels 4 and 5 of the NTID scale. Level 1 (completely unintelligible) was comprised of children unable to produce meaningful speech (23%, 18/79).
Motor speech impairment associated with CP Results from the FCCS indicated that 62% (49/79) of children were effective communicators in the majority of situations (levels I–II). In regard to the CFCS, 38% (30/79) of children were effective sender and receivers with familiar and unfamiliar people (levels I–II). A small number of children demonstrated severe activity and participation limitations, corresponding to level V of the CFCS (5%, 4/79) and FCCS (4%, 3/79).
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Association between motor speech impairment, speech intelligibility, and functional communication Strong associations were found between VSS and NTID (p .001), CFCS (p .001), and FCCS ratings (p .001) (Table III). Children at VSS level II typically demonstrated minor reductions in speech intelligibility ratings and demonstrated more favourable activity and participation outcomes. However, as evidenced in Table III, there were some exceptions as a small number of participants within VSS level II demonstrated greater functional restrictions (CFCS and FCCS level IV). VSS levels III–IV were associated with greater reductions in speech intelligibility and were always associated with reduced functional communication outcomes (Table III). Similarly, strong associations between NTID speech intelligibility ratings with CFCS (p .001) and FCCS levels (p .001) were found (Table IV). Children within levels 1–3 of the NTID scale always exhibited functional communication deficits (Table IV). Participation within the home, school, and community Data demonstrating the association between VSS and NTID levels and parents’ ratings of their child’s Table II. National Technical Institute for the Deaf (NTID) speech intelligibility, Functional Communication Classification System (FCCS), and Communication Function Classification System (CFCS) ratings. Measure
n (%)
NTID speech intelligibility scale 5 4 3 2 1
9 34 11 7 18
(11) (43) (14) (9) (23)
CFCS I II III IV V
17 13 35 10 4
(22) (16) (44) (13) (5)
FCCS I II III IV V
10 39 9 18 3
(13) (49) (11) (23) (4)
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Table III. Association between the Viking Speech Scale (VSS), National Technical Institute for the Deaf (NTID) speech intelligibility, Functional Communication Classification System (FCCS), and Communication Function Classification System (CFCS). VSS level, n (%) I (n 8) II (n 43) III (n 9) IV (n 19) p-value NTID speech intelligibility scale 5 7 (88) 2 (5) 4 1 (13) 33 (77) 3 0 (0) 8 (19) 2 0 (0) 0 (0) 1 0 (0) 0 (0)
0 0 3 6 0
(0) (0) (33) (67) (0)
0 0 0 1 18
(0) (0) (0) (5) (95)
.001a
CFCS I II III IV V
7 1 0 0 0
(88) (13) (0) (0) (0)
10 12 19 2 0
(23) (28) (44) (5) (0)
0 0 8 1 0
(0) (0) (89) (11) (0)
0 0 8 7 4
(0) (0) (42) (37) (21)
.001b
FCCS I II III IV V
3 5 0 0 0
(38) (63) (0) (0) (0)
7 34 1 1 0
(16) (79) (2) (2) (0)
0 0 7 2 0
(0) (0) (78) (22) (0)
0 0 1 15 3
(0) (0) (5) (79) (16)
.001c
p-value calculated using the chi-square test. aχ2 173.17, df 12. bχ2 62.60, df 12. cχ2 114.63, df 12.
participation within the home, school, and community are provided in Table V. Children at VSS level II demonstrated mean scores that were not statistically different to children with no motor speech involvement (level I). VSS levels III and IV, representing moderate and severe profiles of motor speech impairment, respectively, were associated with significant difficulties in participation within
Table IV. Association between ratings on the National Technical Institute for the Deaf (NTID) speech intelligibility, Functional Communication Classification System (FCCS), and Communication Function Classification System (CFCS). Speech intelligibility (NTID rating), n (%) 5 (n 9) 4 (n 34) 3 (n 11) 2 (n 7) 1 (n 18) p-value CFCS I II III IV V
7 2 0 0 0
(78) (22) (0) (0) (0)
10 11 13 0 0
(29) (33) (38) (0) (0)
0 0 9 2 0
(0) (0) (82) (18) (0)
0 0 6 1 0
(0) (0) (86) (14) (0)
0 0 7 7 4
(0) (0) (39) (39) (22)
.001a
FCCS I II III IV V
4 5 0 0 0
(44) (56) (0) (0) (0)
6 28 0 0 0
(18) (82) (0) (0) (0)
0 6 4 1 0
(0) (55) (36) (9) (0)
0 0 4 3 0
(0) (0) (57) (43) (0)
0 0 1 14 3
(0) (0) (6) (78) (17)
.001b
p-value calculated using the chi-square test. a χ2 72.95, df 16. b χ2 102.69, df 16.
on a scale of 1–10 (1 extremely easy, 10 extremely difficult), Mean difference difference in mean scores on the 10-point scale compared to the reference (Ref) group representing children without impairment. home: F 11.00, df 3; school F 6.96, df 3; community: F 18.20, df 3. cData missing for two participants. dANOVA: home: F 8.82, df 4; school F 6.67, df 4; community: F 17.29, df 4. *Significant at p .05. bANOVA:
aBased
NTID speech intelligibility ratingd (1) speech is unintelligible; (2) only isolated words or phrases are intelligible; (3) half of the message is understood; (4) speech is intelligible with some exceptions; and (5) speech is completely intelligible. 1–5 1.7 (1.3) Ref Ref 1–7 2.4 (2.0) Ref Ref 1–8 3.0 (2.5) Ref Ref 5 (n 9) 1–6 2.1 (1.3) .30 1–9 3.0 (2.0) .70 1–8 3.5 (2.1) 4 (n 34) 1.1 (3.0, .8) .4 (2.4, 1.6) 2.1 (4.1, .0) .05* 1–7 3.5 (2.2) 3–10 4.9 (2.1) .30 1–10 5.4 (2.3) 3 (n 11) 2.0 (3.7, .4) .9 (2.6, .8) 3.3 (5.0, 1.5) .02* .001* 2–10 4.4 (2.7) 3–10 5.4 (2.5) 5–10 6.6 (1.8) 2 (n 7) 3.4 (4.7, 2.1) 2.8 (4.2, 1.5) 5.2 (6.5, 3.8) .001* .001* .001* 1–10 5.5 (3.2) 1–10 5.8 (2.8) 1–10 8.6 (2.3) 1 (n 16)c 3.8 (5.6, 2.1) 3.4 (5.2, 1.5) 5.6 (7.5, 3.8) .001* .001* .001*
(I) speech is not affected; (II) speech is imprecise but usually understandable to unfamiliar listeners; (III) speech is unclear and not usually understandable to unfamiliar listeners out of context; and (IV) no understandable speech. VSS 1–2 1.3 (.5) Ref Ref 1–4 2.0 (1.1) Ref Ref 1–8 2.8 (2.4) Ref Ref I (n 8) 1–7 2.4 (1.7) .15 1–10 3.5 (2.3) .10 1–10 3.9 (2.4) .20 II (n 43) 1.2 (.4, 2.8) 1.5 (.3, 3.2) 1.2 (.6, 3.0) 2–10 4.0 (2.5) 2.8 (.7, 4.8) 3–10 5.1 (2.3) 3.1 (1.0, 5.3) 4–10 6.1 (1.8) 3.4 (1.1, 5.6) III (n 9) .009* .006* .004* 1–10 5.5 (3.1) 4.2 (2.4, 6.0) 1–10 5.8 (2.7) 3.8 (1.8, 5.7) 1–10 8.4 (2.4) 5.7 (3.7, 7.7) IV (n 17)c .001* .001* .001*
Mean difference (95% CI) Range p Mean difference (95% CI) Range
Meana (SD)
Mean difference (95% CI)
p
Range
Mean (SD)
School Home
Level of difficulty for the child participating because of their communication, in the:
Table V. Impact of motor speech impairment, speech intelligibility, and parent perception of communication on participation within the home, school, and community.
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Mean (SD)
Community
p
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levelb
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the home, school, and community. Similar findings were identified in regard to speech intelligibility (i.e., children with poorer speech intelligibility typically showed greater difficulties in participation), although some evidence was found to suggest that slight reductions in speech intelligibility (NTID level 4) influences children’s participation within the community (mean difference 2.1, 95% CI 4.1 to .0, p .05; Table V).
Discussion In this study, 90% of children with CP were found to have some degree of motor speech impairment at 5–6 years of age. This figure is higher than recent population-based studies suggest (33–63%; Andersen et al., 2010; Cockerill et al., 2014; Nordberg et al., 2013; Parkes et al., 2010), which may be due to the different aspects of speech measured. In our study, the VSS was used to measure speech, which takes into consideration all motor speech sub-systems. Various definitions of motor speech impairment have been used previously, making it difficult to compare findings across studies. In order to understand the precise frequency of motor speech impairment, it is essential that studies use consistent definitions. Wider use of the VSS may potentially address this current limitation as it has been identified as a suitable tool for surveillance purposes (Pennington et al., 2013b; Virella et al., 2012). In a study based on VSS ratings for 155 children with CP (completed by parents, speech pathologists, and other health professionals), it was found that the scale was easier to apply and had higher levels of inter-rater reliability than the CFCS and FCCS (Virella et al., 2012). However, the developers of the VSS acknowledge that the CFCS or FCCS may compliment the scale given its specific focus on speech (Pennington et al., 2013b; Virella et al., 2012). Given the similarities between the VSS and NTID scales, it is not unexpected that motor speech impairment was associated with reduced speech intelligibility, with higher VSS levels corresponding to greater reductions in intelligibility (i.e., lower levels on the NTID scale). The same pattern was evident in regard to children’s functional communication (CFCS and FCCS) outcomes in that VSS level II and NTID level 4 were associated with more favourable CFCS and FCCS ratings. However, it is important to consider that these general trends may not apply to the entire population. Within the present sample, a small number of children with mild deficits (VSS level II) presented with considerable functional communication limitations (CFCS and FCCS level IV). This suggests that some caution is needed in presuming that all children within VSS level II will demonstrate more positive activity and participation outcomes, and that these areas should be measured on an
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Motor speech impairment associated with CP individual basis. It is as yet unknown whether the impact of mild speech deficits changes over time. Based on the adult literature concerning populations other than CP (e.g., traumatic brain injury, multiple sclerosis, and Parkinson’s disease), speech intelligibility and severity of dysarthria do not necessarily predict difficulty in communicative participation (Hartelius, Elmberg, Holm, Lovberg, & Nikolaidis, 2008; McAuliffe, Carpenter, & Moran, 2010; Yorkston, Klasner, & Swanson, 2001). With regard to children’s participation within the home, school, and community, participation became increasingly more difficult in each environment as VSS ratings increased and NTID speech intelligibility ratings decreased. This is not surprising and is consistent with previous research reporting a link between severe communication deficits and reduced social participation (Clarke et al., 2012; McFadd & Hustad, 2013). The general pattern of results indicated that participation was easiest in familiar environments (i.e., at home), compared to school and community settings. However, for children within VSS levels III–IV and NTID levels 3–1, home-based participation was significantly more difficult compared to children with no motor speech or intelligibility issues.These restrictions may potentially be related to parents’ interaction with children (Pennington & McConachie, 2001) and in turn can detrimentally impact on children’s quality-of-life (Dickinson, Parkinson, Ravens-Sieberer, Schirripa, Thyen, Arnaud, et al., 2007). It is likely, however, that a range of parent, child, and environmental factors influence children’s participation within the home. An encouraging finding is that parents rated the ease in which children at VSS level II were able to participate within the home, school, and community similarly to children with no motor speech impairment. This suggests that mild speech deficits do not substantially affect children’s ability to participate in a range of environments. Although some exceptions may exist, these findings imply that intervention focused on enhancing participation is warranted predominately for children with moderate or severe motor speech impairments (VSS levels III–IV). Further exploration is needed to determine the ways to achieve this as few studies have investigated the effects of speech therapy on the participation of children with CP. One study to do so found that, whilst some children with moderate-to-severe dysarthria demonstrated improvements in communicative participation following speech therapy, increases in speech intelligibility were not associated with changes in participation (Pennington, Roelant, Thompson, Robson, Steen, & Miller, 2013a). Thus, intervention at an impairment level (i.e., targeting breath support, phonation, and speech rate) resulting in improved intelligibility does not necessarily translate into improved participation. This suggests that intervention strategies specifically targeting participation are needed to improve outcomes in this area.
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An important strength of this study is that we recruited a community cohort of children with CP drawn from a representative sample. Further, participants were similar to non-participants across key variables (e.g., CP motor type and distribution, and the presence of epilepsy, cognitive, hearing, and vision impairment). This provides some confidence that findings reflect the general population of children with CP aged 5 and 6 years. However, as the current study forms part of a broader project investigating speech and language in children with CP, it is possible that parents who were concerned about their child’s development in these areas were more likely to provide consent, potentially resulting in an over-representation of motor speech impairment being reported here. There are important factors that were not addressed here that warrant consideration in future research. For instance, the impact of co-existing communication impairments (e.g., language; Liégeois, Mahony, Connelly, Pigdon, Tournier, & Morgan, 2013) and environmental factors were not addressed. Potentially associated factors such as children’s cognition, socioeconomic status, and non-English speaking background may also be considered in future research. These additional factors may explain why some children within VSS level I demonstrated reduced outcomes on the CFCS and FCCS. Based on the work by McFadd and Hustad (2013), which showed that restrictions in social participation were only evident when language was affected, it is possible that, within the current sample, co-morbid language impairment influenced children’s activity and participation ratings. Although not the focus of this paper, this is evidently likely, given that 93% (26/28) of children within VSS levels III–IV demonstrated language impairment. As the measures used in this study are classification tools, detailed descriptions of children’s motor speech impairment and activity and participation were not possible. Further research is needed to examine the features of motor speech impairment in children with CP and the specific restrictions in activity and participation they may experience. A limitation of the present study is the use of an equal appearing interval scale as a measure of speech intelligibility. It is acknowledged that measures such as Percent Phonemes Correct or those that rely on word identification provide a more objective estimation of children’s intelligibility. An objective standardized measure of speech intelligibility was not included in the current study in order to reduce the burden of the assessment length on participants. The NTID scale provided a means to estimate intelligibility without any inconvenience to children and families. Given the absence of an objective measure of intelligibility in this study, further research is needed to examine the association between standardized intelligibility scores with ratings from the VSS, CFCS, and FCCS. Additionally, future research may evaluate the association between the CFCS and FCCS scales. Taken from our data, participants assigned to level II of the
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FCCS were more likely to be rated at level III of the CFCS, which may imply an association between these levels. As the scales included in this study were all completed by the same rater, it is possible that ratings on one scale may have influenced ratings on another. A final factor to consider when interpreting the present findings is that the perception of communication on the ease with which children participate within the home, school, and community was measured using parent report. Although parents know their child best, it is possible that they may have under- or over-estimated the difficulty children experience within each setting, particularly within the school setting which they may not regularly attend. The use of self-report to measure the participation of children with disabilities has been advocated (Michelsen, 2013). Future research investigating the activity and participation of children with motor speech impairment should consider the opinions of children if at all possible. This would provide critical insight into children’s level of enjoyment during activities, which was not captured by the current study. Research suggests that children with severe communication difficulties demonstrate higher levels of enjoyment during activities compared to matched peers (Thirumanickam et al., 2011). Therefore, although our findings highlight the negative impact of motor speech impairment on children’s participation, children may have still demonstrated high levels of enjoyment during activities.
Conclusions Motor speech impairment is commonly experienced by children with CP. Findings indicate that an estimated 90% of children with CP aged 5–6 years demonstrate motor speech deficits based on the VSS. The general pattern of results indicated that activity and participation limitations increased with greater reductions in motor speech ability and speech intelligibility. The finding that children with mild motor speech impairment (VSS level II) had comparable participation to children with no motor speech impairment, suggests a relatively positive functional outcome for this study’s age group compared to those children at VSS levels III–IV.
Note 1.
Defined by Eadie, Yorkston, Klasner, Dudgeon, Deitz, Baylor, et al. (2006) as “taking part in life situations where knowledge, information, ideas, or feelings are exchanged” (p. 309).
Acknowledgements We are grateful to all the families who participated in this study. We kindly thank Sue Reid (Manager of
the VCPR) and the following departments at the Royal Children’s Hospital, Melbourne, for their generous assistance in recruiting families: Developmental Medicine, Orthopaedics, and the Victorian Paediatric Rehabilitation Service. This project was funded by a National Health and Medical Research Council (NHMRC) Postgraduate Scholarship (#607448) and a Speech Pathology Australia Nadia Verrall Research Grant. The following authors were funded by NHMRC: AM (Career Development Award #607315); SR (Practitioner Fellowship #1041892); and FM (Population Health Capacity Building Grant #436914 and Early Career Fellowship #1037449). Support was also received from the Victorian Government’s Operational Infrastructure Support Program. Declaration of interest: The authors report no conflicts of interest. The authors alone are responsible for the content and writing of the paper. References Andersen, G., Mjoen, T. R., & Vik, T. (2010). Prevalence of speech problems and the use of augmentative and alternative communication in children with cerebral palsy: A registry-based study in Norway. Perspectives on Augmentative and Alternative Communication, 19, 12–20. Barty, E., & Caynes, K. (2009). Development of the Functional Communication Classification Scale. Paper presented at the 3rd International Cerebral Palsy Conference, Sydney, Australia. Clarke, M., Newton, C., Petrides, K., Griffiths, T., Lysley, A., & Price, K. (2012). An examination of relations between participation, communication and age in children with complex communication needs. Augmentative and Alternative Communication, 28, 44–51. Clarke, M. T., Newton, C., Griffiths, T., Price, K., Lysley, A., & Petrides, K. V. (2011). Factors associated with the participation of children with complex communication needs. Research in Developmental Disabilities, 32, 774–780. Cockerill, H., Elbourne, D., Allen, E., Scrutton, D., Will, E., McNee, A., et al. (2014). Speech, communication and use of augmentative communication in young people with cerebral palsy: The SH & PE population study. Child: Care, Health and Development, 40, 149–157. Colver, A., Thyen, U., Arnaud, C., Beckung, E., Fauconnier, J., Marcelli, M., et al. (2012). Association between participation in life situations of children with cerebral palsy and their physical, social and attitudinal environment: A cross-sectional multi-centre European study. Archives of Physical Medicine and Rehabilitation, 93, 2154–2164. Dickinson, H. O., Parkinson, K. N., Ravens-Sieberer, U., Schirripa, G., Thyen, U., Arnaud, C., et al. (2007). Self-reported quality of life of 8–12-year-old children with cerebral palsy: A crosssectional European study. Lancet, 369, 2171–2178. Eadie, T. L., Yorkston, K. M., Klasner, E. R., Dudgeon, B. J., Deitz, J. C., Baylor, C. R., et al. (2006). Measuring communicative participation: A review of self-report instruments in speech-language pathology. American Journal of SpeechLanguage Pathology, 15, 307–320. Eliasson, A. C., Krumlinde-Sundholm, L., Rosblad, B., Beckung, E., Arner, M., Ohrvall, A. M., et al. (2006). The Manual Ability Classification System (MACS) for children with cerebral palsy: Scale development and evidence of validity and reliability. Developmental Medicine and Child Neurology, 48, 549–554.
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Motor speech impairment associated with CP Flipsen, P. (1995). Speaker-listener familiarity: Parents as judges of delayed speech intelligibility. Journal of Communication Disorders, 28, 3–19. Forsyth, R., Colver, A., Alvanides, S., Woolley, M., & Lowe, M. (2007). Participation of young severely disabled children is influenced by their intrinsic impairments and environment. Developmental Medicine and Child Neurology, 49, 345–349. Hammal, D., Jarvis, S. N., & Colver, A. F. (2004). Participation of children with cerebral palsy is influenced by where they live. Developmental Medicine and Child Neurology, 46, 292–298. Hartelius, L., Elmberg, M., Holm, R., Lovberg, A. S., & Nikolaidis, S. (2008). Living with dysarthria: Evaluation of a self-report questionnaire. Folia Phoniatrica et Logopaedica, 60, 11–19. Hidecker, M. J. C., Paneth, N., Rosenbaum, P. L., Kent, R. D., Lillie, J., Eulenberg, J. B., et al. (2011). Developing and validating the Communication Function Classification System for individuals with cerebral palsy. Developmental Medicine and Child Neurology, 53, 704–710. Johnson, D. D. (1975). Communication characteristics of NTID students. Journal of the Academy of Rehabilitative Audiology, 8, 17–32. Kent, R. D., Miolo, G., & Bloedel, S. (1994). The intelligibility of children’s speech: A review of evaluation procedures. American Journal of Speech-Language Pathology, 3, 81–95. Lawlor, K., Mihaylov, S., Welsh, B., Jarvis, S., & Colver, A. (2006). A qualitative study of the physical, social and attitudinal environments influencing the participation of children with cerebral palsy in northeast England. Developmental Neurorehabilitation, 9, 219–228. Liégeois, F., Morgan, A. T., Cross, J. H., Stewart, L. H., Vogel, A. P., & Vargha-Khadem, F. (2010). Speech and oral motor profile after childhood hemispherectomy. Brain and Language, 114, 126–134. Liégeois, F. J., Mahony, K., Connelly, A., Pigdon, L.,Tournier, J. D., & Morgan, A. T. (2013). Pediatric traumatic brain injury: Language outcomes and their relationship to the arcuate fasciculus. Brain and Language, 127, 388–398. McAuliffe, M. J., Carpenter, S., & Moran, C. (2010). Speech intelligibility and perceptions of communication effectiveness by speakers with dysarthria following traumatic brain injury and their communication partners. Brain Injury, 24, 1408–1415. McFadd, E., & Hustad, K. C. (2013). Assessment of social function in four-year-old children with cerebral palsy. Developmental Neurorehabilitation, 16, 102–112. Mei, C., Morgan, A., Reilly, S., Reddihough, R., Mensah, F., Green, J., et al. (2012). Barriers and facilitators affecting the activities and participation of children with cerebral palsy: Parent perspectives. Developmental Medicine and Child Neurology, 54 (Suppl 5), 84. Michelsen, S. (2013). Participation in children with disabilities – a public health perspective. Developmental Medicine and Child Neurology, 55, 684–685.
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Nordberg, A., Miniscalco, C., Lohmander, A., & Himmelmann, K. (2013). Speech problems affect more than one in two children with cerebral palsy: Swedish population-based study. Acta Paediatrica, 102, 161–166. Palisano, R., Rosenbaum, P., Walter, S., Russell, D., Wood, E., & Galuppi, B. (1997). Development and reliability of a system to classify gross motor function in children with cerebral palsy. Developmental Medicine and Child Neurology, 39, 214–223. Parkes, J., Hill, N., Platt, M. J., & Donnelly, C. (2010). Oromotor dysfunction and communication impairments in children with cerebral palsy: A register study. Developmental Medicine and Child Neurology, 52, 1113–1119. Pennington, L., & McConachie, H. (2001). Predicting patterns of interaction between children with cerebral palsy and their mothers. Developmental Medicine and Child Neurology, 43, 83–90. Pennington, L., Roelant, E., Thompson, V., Robson, S., Steen, N., & Miller, N. (2013a). Intensive dysarthria therapy for younger children with cerebral palsy. Developmental Medicine and Child Neurology, 55, 464–471. Pennington, L., Virella, D., Mjoen, T., da Graça Andrada, M., Murray, J., Colver, A., et al. (2013b). Development of The Viking Speech Scale to classify the speech of children with cerebral palsy. Research in Developmental Disabilities, 34, 3202–3210. Raghavendra, P., Virgo, R., Olsson, C., Connell, T., & Lane, A. E. (2011). Activity participation of children with complex communication needs, physical disabilities and typicallydeveloping peers. Developmental Neurorehabilitation, 14, 145–155. Reid, S. M., Carlin, J. B., & Reddihough, D. S. (2011). Classification of topographical pattern of spasticity in cerebral palsy: A registry perspective. Research in Developmental Disabilities, 32, 2909–2915. Thirumanickam, A., Raghavendra, P., & Olsson, C. (2011). Participation and social networks of school-age children with complex communication needs: A descriptive study. Augmentative and Alternative Communication, 27, 195–204. Virella, D., Pennington, L., da Graça Andrada, M., Greitane, A., Prasauskiene, A., Rackauskaite, G., et al. (2012). Scales for assessment of communication ability in the surveillance of children with cerebral palsy. Paper presented at the 4th International Cerebral Palsy Conference, Pisa, Italy. World Health Organization (WHO). (2001). International classification of functioning, disability and health. Geneva: WHO. Yorkston, K. M., Klasner, E. R., & Swanson, K. M. (2001). Communication in context: A qualitative study of the experiences of individuals with multiple sclerosis. American Journal of Speech-Language Pathology, 10, 126–137. Zimmerman, I. L., Steiner, V. G., & Pond, R. E. (2002). Preschool language scale, fourth edition. San Antonio, TX: Harcourt Assessment, Inc.
Appendix: Scale for parent ratings of their child’s participation within the home, school, and community Please rate how easy it is for your child to participate because of their ability to communicate. (a) In the home 1 Extremely easy
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(b) At school/kindergarten 1 2 Extremely easy
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International Journal of Speech-Language Pathology Publication details, including instructions for authors and subscription information: http://www.tandfonline.com/loi/iasl20
Motor speech impairment, activity, and participation in children with cerebral palsy ab
Cristina Mei , Sheena Reilly
abc
abc
, Dinah Reddihough
, Fiona Mensah
abc
& Angela
abc
Morgan a
Murdoch Childrens Research Institute, Melbourne, Australia
b
Department of Paediatrics, University of Melbourne, Melbourne, Australia
c
Royal Children’s Hospital, Melbourne, Australia Published online: 09 Jun 2014.
Click for updates To cite this article: Cristina Mei, Sheena Reilly, Dinah Reddihough, Fiona Mensah & Angela Morgan (2014) Motor speech impairment, activity, and participation in children with cerebral palsy, International Journal of Speech-Language Pathology, 16:4, 427-435 To link to this article: http://dx.doi.org/10.3109/17549507.2014.917439
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International Journal of Speech-Language Pathology, 2014; 16(4): 427–435
INVITED ARTICLE
Motor speech impairment, activity, and participation in children with cerebral palsy
CRISTINA MEI1,2, SHEENA REILLY1,2,3, DINAH REDDIHOUGH1,2,3, FIONA MENSAH1,2,3 & ANGELA MORGAN1,2,3
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1Murdoch
Childrens Research Institute, Melbourne, Australia, 2Department of Paediatrics, University of Melbourne, Melbourne, Australia, and 3Royal Children’s Hospital, Melbourne, Australia
Abstract The present study used a population-based sample of children with cerebral palsy (CP) to estimate the prevalence of motor speech impairment and its association with activity and participation. A sample of 79 Victorian children aged 4 years 11 months to 6 years 5 months was recruited through the Victorian CP Register. The presence of motor speech impairment was recorded using the Viking Speech Scale (VSS). Activity and participation outcomes included speech intelligibility (the National Technical Institute for the Deaf rating scale, NTID), the Functional Communication Classification System (FCCS) and Communication Function Classification System (CFCS). A parent completed rating scale was used to examine the association between motor speech impairment and participation. Ninety per cent (71/79) of children demonstrated a motor speech impairment. Strong associations were found between the VSS and NTID ( .001), CFCS ( .001), and FCCS levels ( .001). VSS levels III–IV were significantly associated with restrictions in home, school, and communitybased participation as perceived by parents. Although some diversity in activity and participation outcomes was observed within specific VSS levels, the results of this study suggested that children with mild motor speech impairments are more likely to demonstrate superior activity and participation outcomes compared to children with moderate or severe deficits.
Keywords: Cerebral palsy, motor speech disorders, activity and participation.
Introduction The motor limitations associated with cerebral palsy (CP) pose many challenges during development, including the production of clear and intelligible speech. Recent population-based data suggest that 33–63% of children with CP experience motor speech impairment (Andersen, Mjoen, & Vik, 2010; Cockerill, Elbourne, Allen, Scrutton, Will, McNee, et al., 2014; Nordberg, Miniscalco, Lohmander, & Himmelmann, 2013; Parkes, Hill, Platt, & Donnelly, 2010). At present, there is no consensus on how communication impairment (affecting speech or language) should be defined in children with CP (Hidecker, Paneth, Rosenbaum, Kent, Lillie, Eulenberg, et al., 2011). Consequently, studies have used various definitions to examine the occurrence of motor speech impairment, including level of speech intelligibility (Cockerill et al., 2014) and whether children’s speech is indistinct (Andersen et al., 2010). In some instances, a clear definition is not reported.
Several classification systems have been developed recently that provide a common means to measure motor speech impairment and communication functioning at an activity and participation level. These include the Viking Speech Scale (VSS; Pennington, Virella, Mjoen, da Graça Andrada, Murray, Colver, et al., 2013b), the Functional Communication Classification System (FCCS; Barty & Caynes, 2009), and the Communication Function Classification System (CFCS; Hidecker et al., 2011). These scales are specifically designed for children with CP and correspond to various aspects of the International Classification of Functioning, Disability, and Health model (ICF; World Health Organization, 2001). For instance, the VSS specifically focuses at the impairment and activity levels of speech, while the FCCS and CFCS measure communication at an activity and participation level to determine the functional or daily impact of impairments. The CFCS classifies communication according to how effectively an individual sends and receives messages with familiar and unfamiliar communication partners. In comparison,
Correspondence: Ms Cristina Mei, BSpPath (Hons), Murdoch Children’s Research Institute, Flemington Road, Parkville, Melbourne, 3052, Australia. Email: [email protected] ISSN 1754-9507 print/ISSN 1754-9515 online © 2014 The Speech Pathology Association of Australia Limited Published by Informa UK, Ltd. DOI: 10.3109/17549507.2014.917439
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communication is classified by the FCCS according to a child’s ability to communicate messages/topics and needs/wants and takes into consideration both the familiarity of the communication partner and setting. The activity and participation restrictions specifically associated with motor speech impairment in children with CP have not been determined. Within this population, children’s abilities have largely been characterized at an impairment level; however, recent studies have incorporated activity and participation outcomes. Using the social function domain of the Paediatric Evaluation of Disability Inventory (PEDI), McFadd and Hustad (2013) reported that isolated motor speech impairment (in the absence of language impairment) did not result in significantly reduced social outcomes compared to children with CP with no speech motor involvement. Social restrictions were only evident when language abilities were also affected (McFadd & Hustad, 2013). This led the authors to conclude that the PEDI is sensitive to language impairment but not to reductions in speech intelligibility. However, the authors did not investigate the association between severity of motor speech impairment and social function. A growing body of evidence has highlighted the activity and participation restrictions associated with anarthria or complex communication needs (Clarke, Newton, Griffiths, Price, Lysley, & Petrides, 2011; Clarke, Newton, Petrides, Griffiths, Lysley, & Price, 2012; McFadd & Hustad, 2013; Raghavendra, Virgo, Olsson, Connell, & Lane, 2011; Thirumanickam, Raghavendra, & Olsson, 2011). Whether these limitations are also associated with mild or less severe forms of motor speech impairment is unknown. Children’s participation within various environments (e.g., home, school, and community) is influenced by multiple variables (Colver, Thyen, Arnaud, Beckung, Fauconnier, Marcelli, et al., 2012; Forsyth, Colver, Alvanides, Woolley, & Lowe, 2007; Hammal, Jarvis, & Colver, 2004; Lawlor, Mihaylov, Welsh, Jarvis, & Colver, 2006). One factor that is likely to play an important role in shaping the participation of children with motor speech impairment is their level of intelligibility. Speech intelligibility is influenced by the familiarity of the listener (Flipsen, 1995). Therefore, an assumption could be made that the participation of children with reduced intelligibility is affected more during activities outside of the home or with unfamiliar people than in the home or with familiar people. However, research suggests that even social outcomes within the home are likely to be reduced due to poor speech intelligibility (Pennington & McConachie, 2001). The impact of a child’s level of speech intelligibility on participation in various contexts (home, school, and community) has not been widely investigated. Investigation of participation across settings has focused predominately on children with severe communication difficulties where participation is lower in frequency
and restricted to certain settings (Clarke et al., 2012; Raghavendra et al., 2011; Thirumanickam et al., 2011). Until recently, a core challenge in investigating the broader effects of motor speech impairment in children with CP has been the lack of measures specific to this population addressing activity and participation, or, more specifically, communicative participation.1 The CFCS and FCCS are activity and participation measures that are designed for children with CP. As these scales were developed recently, limited research is available regarding their use, and little is known about the association between motor speech impairment and CFCS and FCCS levels (Virella, Pennington, da Graça Andrada, Greitane, Prasauskiene, Rackauskaite, et al., 2012). Identifying these associations is important for establishing the communication profiles of children with CP and understanding the functional therapeutic needs associated with varying degrees of motor speech involvement. To this end, the present study used a population-based sample of children with CP to (1) estimate the frequency of motor speech impairment using the VSS; (2) examine the association between VSS, speech intelligibility, CFCS, and FCCS ratings; and (3) examine the association between VSS ratings and parent ratings of children’s participation within the home, school, and community.
Methods Participants Participants were recruited through the Victorian Cerebral Palsy Register (VCPR). The VCPR contains information about all individuals with CP living or born in the state of Victoria since 1970. The VCPR identifies cases of CP by reviewing the medical records of children attending two tertiary paediatric hospitals in Victoria and through referrals from clinicians and families (Reid, Carlin, & Reddihough, 2011). The birth range that was of interest to this study was 25 August 2005 to 24 August 2007 (the age range of 5–6 years was chosen as by these ages children typically demonstrate a substantial degree of functional speech and language skills). Children with CP due to post-neonatal injury (occurring up to 2 years of age) were included. Based on these criteria, 232 eligible children were identified. Of these, 176 (76%) were contacted (six children had died and 50 were unable to be contacted). Consent to participate in the study was obtained for 84 children, representing 48% of the contacted families and 37% of the known living population of this age group on the VCPR. The presence/absence of motor speech impairment could not be confirmed for five children due to respiratory infections (n 4) or the inability to obtain a speech sample (n 1). This resulted in a sample size of 79 children. Ethics
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Motor speech impairment associated with CP approval for the study was obtained from Human Research Ethics Committees at The Royal Children’s Hospital (#30048) and Southern Health (#11380), Melbourne. Participants were aged between 4 years 11 months and 6 years 5 months (mean 5.4, SD .5) at the time of their assessment. Key characteristics of the 79 participants are presented in Table I. All motor types, Gross Motor Function Classification System (GMFCS; Palisano, Rosenbaum, Walter, Russell, Wood, & Galuppi, 1997) levels, and Manual Ability Classification System (MACS; Eliasson, KrumlindeSundholm, Rosblad, Beckung, Arner, Ohrvall, et al., 2006) levels were represented. Thirteen participants (15%) were classified as having mixed CP, which included spastic-dystonia (n 9), spastic-athetoid (n 1), spastic-hypotonia (n 1), ataxic-hypotonia (n 1), and ataxic-spastic (n 1). Language impairment was identified in 65% (51/79) of children. To determine the representativeness of the recruited sample, characteristics of the participants were compared against those of the non-participants. Non-participants were children who were not contacted by the VCPR, declined study participation, were lost to follow-up, or for whom the presence/ absence of motor speech impairment could not be determined. It excluded the six children that had died. Participants and non-participants were compared across key variables that included CP motor type and distribution, GMFCS level, and associated impairments. The chi-square test was used to determine statistically significant differences between participants and non-participants, as indicated by a p-value .05. As seen in Table I, participants included in the study did not differ significantly to non-participants (n 147) in terms of CP type and distribution, GMFCS level and the presence of epilepsy, hearing, vision, and cognitive impairment (Table I).
Measures The following measures were completed during a comprehensive face-to-face assessment of communication that included a range of further speech and language measures not reported on here. Ratings were completed as per the user guidelines for each scale. Motor speech impairment, speech intelligibility, and functional communication ratings were completed by the examiner (CM) and the child’s parent. Motor speech impairment. Motor speech impairment was recorded using the Viking Speech Scale (VSS), a newly developed speech classification system specifically designed for children with CP over 4 years of age (Pennington et al., 2013b). The VSS consists of the following four levels: (I) speech is not affected; (II) speech is imprecise but usually under-
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Table I. Characteristics of participants (n 79) and nonparticipants (n 147). Participants, Non-participants, n (%) n (%) p-value Gender Female Male CP motor type Spastic Dyskinesia Hypotonia Ataxic Mixed Unknown CP distribution Monoplegia Hemiplegia Diplegia Triplegia Quadriplegia Unknown GMFCS level I II III IV V Unknown MACS levela I II III IV V Epilepsy No Yes Resolved Unknown Cognitive impairmentb No Yes Unknown Hearing impairment No Yes Unknown Vision impairment No Yes Unknown Language impairmentc No Yes Unknown
34 (43) 45 (57)
62 (42) 85 (58)
.90
61 1 3 1 13 0
(77) (1) (4) (1) (16) (0)
123 1 4 2 15 2
(84) (1) (3) (1) (10) (1)
.69
1 29 23 1 25 0
(1) (37) (29) (1) (32) (0)
2 58 44 3 38 2
(1) (39) (30) (2) (26) (1)
.93
31 14 12 15 7 0
(39) (18) (15) (19) (9) (0)
54 34 12 22 14 11
(37) (23) (8) (15) (10) (7)
.50
28 27 11 7 6
(35) (34) (14) (9) (8)
61 17 1 0
(77) (22) (1) (0)
— — — — — 104 36 1 6
(71) (24) (1) (4)
.74
49 (62) 22 (28) 8 (10)
59 (40) 40 (27) 48 (33)
.21
73 (92) 6 (8) 0 (0)
105 (71) 18 (12) 24 (16)
.13
52 (66) 27 (34) 0 (0)
67 (46) 47 (32) 33 (22)
.32
27 (34) 51 (65) 1 (1)
— — —
p-value calculated using the chi-square test. GMFCS, Gross Motor Function Classification System level (Palisano et al., 1997); MACS, Manual Ability Classification System (Eliasson et al., 2006). aMACS levels for non-participants are not reported due to the high percentage of missing data on the Victorian Cerebral Palsy Register. bCognitive status (recorded as “no impairment”, “probably no impairment”, “probably some impairment”, “unknown”) was based on data obtained by the Victorian Cerebral Palsy Register. cLanguage data not available for non-participants. Language was measured using the Preschool Language Scale, 4th edition (Zimmerman, Steiner, & Pond, 2002). Language impairment was defined as a standard score greater than one standard deviation below the mean on the Auditory Comprehension and/or Expressive Communication sub-scales.
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standable to unfamiliar listeners (e.g., imprecise articulation, breathy or harsh voice); (III) speech is unclear and not usually understandable to unfamiliar listeners out of context (e.g., difficulties controlling breathing for speech, small range of consonants produced); and (IV) no understandable speech. The identification of motor speech impairment was based on a rating of II–IV. Speech intelligibility. The National Technical Institute for the Deaf (NTID) rating scale (Johnson, 1975) was used to measure children’s connected speech intelligibility (samples were obtained during a parent–child free-play interaction). The scale’s use can extend beyond its original population of interest (i.e., hearing impaired children) (Kent, Miolo, & Bloedel, 1994) and has previously been used with children with motor speech disorders (Liégeois, Morgan, Cross, Stewart, Vogel, & Vargha-Khadem, 2010). The five levels of the NTID rating scale are summarized as follows: (1) speech is unintelligible; (2) only isolated words or phrases are intelligible; (3) half of the message is understood; (4) speech is intelligible with some exceptions; and (5) speech is completely intelligible. The NTID scale is similar to the VSS in that they both classify speech intelligibility; however, they differ in that the VSS also takes into consideration the familiarity of the communication partner, the need for contextual cues, and the child’s impairment within each of the motor speech sub-systems. Functional communication. Children’s communicative abilities at an activity and participation level were measured using the CFCS (Hidecker et al., 2011) and the FCCS (Barty & Caynes, 2009). Each scale has five-levels and is designed specifically for children with CP. The CFCS consists of the following levels: (I) effective sender and receiver; (II) effective but slower paced sender and/or receiver; (III) effective sender and receiver with familiar partners only; (IV) inconsistent sender and/or receiver with familiar partners; and (V) seldom effective sender and receiver, even with familiar partners. The levels of the FCCS include: (I) an effective communicator in most situations; (II) an effective communicator in most situations with help; (III) an effective communicator in some situations; (IV) assistance required in most situations, particularly with unfamiliar people and environments; and (V) communicates unintentionally using movement and behaviour. Inter-rater agreement (weighted kappa) between examiner and parent ratings for the above scales was moderate-to-high (VSS .72, 95% confidence interval [CI] .68–.74; NTID .82, 95% CI .73–.90; CFCS .61, 95% CI .54–.67; FCCS .74, 95% CI .65–.84). Participation within the home, school, and community. The ease with which children were able to partici-
pate because of their ability to communicate was rated by parents. This measure aimed to address children’s overall participation, whereas the previously described measures focused on communicative activity and participation. Participation within the home, school, and community were each rated using a 10-point scale (see Appendix). For each of the scales parents were asked to rate “how easy it is for your child to participate because of their ability to communicate”. A score of 1 corresponded to “extremely easy” and a score of 10 represented “extremely difficult”. The three scales formed part of an activity and participation questionnaire completed by the child’s parent during the face-to-face assessment. This questionnaire was developed by the research team and was based on individual interviews conducted with 13 parents of children with CP aged 4–9 years (Mei, Morgan, Reilly, Reddihough, Mensah, Green, et al., 2012). Participation was described to parents as their child’s involvement in life situations as per the ICF. Examples of participation provided to parents included interacting or playing with others and being socially involved in activities or events. Analysis Associations between children’s ratings for motor speech impairment (VSS) with speech intelligibility (NTID) and functional communication (CFCS and FCCS) were examined using contingency tables and the Chi-squared test. A one-way analysis of variance (ANOVA) was used to compare children’s participation (as measured by the 10-point scales) according to VSS and NTID levels. Mean scores on the 10-point scales were compared across each level, with VSS level I and NTID level 5 (children without impairment) as the reference groups. Statistically significant associations were indicated by a p-value .05.
Results Frequency of motor speech impairment Motor speech impairment was identified in 90% (71/79) of children based on a VSS rating of II–IV. Fifty-four per cent (43/79) of children demonstrated mild deficits (level II). The proportion of children classified at the remaining levels was 10% (8/79, level I), 11% (9/79, level III), and 24% (19/79, level IV). Functional communication and speech intelligibility Ratings from the CFCS, FCCS, and the NTID speech intelligibility scale are presented in Table II. Just over half of the sample (54%, 43/79) demonstrated highly intelligible speech, corresponding to levels 4 and 5 of the NTID scale. Level 1 (completely unintelligible) was comprised of children unable to produce meaningful speech (23%, 18/79).
Motor speech impairment associated with CP Results from the FCCS indicated that 62% (49/79) of children were effective communicators in the majority of situations (levels I–II). In regard to the CFCS, 38% (30/79) of children were effective sender and receivers with familiar and unfamiliar people (levels I–II). A small number of children demonstrated severe activity and participation limitations, corresponding to level V of the CFCS (5%, 4/79) and FCCS (4%, 3/79).
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Association between motor speech impairment, speech intelligibility, and functional communication Strong associations were found between VSS and NTID (p .001), CFCS (p .001), and FCCS ratings (p .001) (Table III). Children at VSS level II typically demonstrated minor reductions in speech intelligibility ratings and demonstrated more favourable activity and participation outcomes. However, as evidenced in Table III, there were some exceptions as a small number of participants within VSS level II demonstrated greater functional restrictions (CFCS and FCCS level IV). VSS levels III–IV were associated with greater reductions in speech intelligibility and were always associated with reduced functional communication outcomes (Table III). Similarly, strong associations between NTID speech intelligibility ratings with CFCS (p .001) and FCCS levels (p .001) were found (Table IV). Children within levels 1–3 of the NTID scale always exhibited functional communication deficits (Table IV). Participation within the home, school, and community Data demonstrating the association between VSS and NTID levels and parents’ ratings of their child’s Table II. National Technical Institute for the Deaf (NTID) speech intelligibility, Functional Communication Classification System (FCCS), and Communication Function Classification System (CFCS) ratings. Measure
n (%)
NTID speech intelligibility scale 5 4 3 2 1
9 34 11 7 18
(11) (43) (14) (9) (23)
CFCS I II III IV V
17 13 35 10 4
(22) (16) (44) (13) (5)
FCCS I II III IV V
10 39 9 18 3
(13) (49) (11) (23) (4)
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Table III. Association between the Viking Speech Scale (VSS), National Technical Institute for the Deaf (NTID) speech intelligibility, Functional Communication Classification System (FCCS), and Communication Function Classification System (CFCS). VSS level, n (%) I (n 8) II (n 43) III (n 9) IV (n 19) p-value NTID speech intelligibility scale 5 7 (88) 2 (5) 4 1 (13) 33 (77) 3 0 (0) 8 (19) 2 0 (0) 0 (0) 1 0 (0) 0 (0)
0 0 3 6 0
(0) (0) (33) (67) (0)
0 0 0 1 18
(0) (0) (0) (5) (95)
.001a
CFCS I II III IV V
7 1 0 0 0
(88) (13) (0) (0) (0)
10 12 19 2 0
(23) (28) (44) (5) (0)
0 0 8 1 0
(0) (0) (89) (11) (0)
0 0 8 7 4
(0) (0) (42) (37) (21)
.001b
FCCS I II III IV V
3 5 0 0 0
(38) (63) (0) (0) (0)
7 34 1 1 0
(16) (79) (2) (2) (0)
0 0 7 2 0
(0) (0) (78) (22) (0)
0 0 1 15 3
(0) (0) (5) (79) (16)
.001c
p-value calculated using the chi-square test. aχ2 173.17, df 12. bχ2 62.60, df 12. cχ2 114.63, df 12.
participation within the home, school, and community are provided in Table V. Children at VSS level II demonstrated mean scores that were not statistically different to children with no motor speech involvement (level I). VSS levels III and IV, representing moderate and severe profiles of motor speech impairment, respectively, were associated with significant difficulties in participation within
Table IV. Association between ratings on the National Technical Institute for the Deaf (NTID) speech intelligibility, Functional Communication Classification System (FCCS), and Communication Function Classification System (CFCS). Speech intelligibility (NTID rating), n (%) 5 (n 9) 4 (n 34) 3 (n 11) 2 (n 7) 1 (n 18) p-value CFCS I II III IV V
7 2 0 0 0
(78) (22) (0) (0) (0)
10 11 13 0 0
(29) (33) (38) (0) (0)
0 0 9 2 0
(0) (0) (82) (18) (0)
0 0 6 1 0
(0) (0) (86) (14) (0)
0 0 7 7 4
(0) (0) (39) (39) (22)
.001a
FCCS I II III IV V
4 5 0 0 0
(44) (56) (0) (0) (0)
6 28 0 0 0
(18) (82) (0) (0) (0)
0 6 4 1 0
(0) (55) (36) (9) (0)
0 0 4 3 0
(0) (0) (57) (43) (0)
0 0 1 14 3
(0) (0) (6) (78) (17)
.001b
p-value calculated using the chi-square test. a χ2 72.95, df 16. b χ2 102.69, df 16.
on a scale of 1–10 (1 extremely easy, 10 extremely difficult), Mean difference difference in mean scores on the 10-point scale compared to the reference (Ref) group representing children without impairment. home: F 11.00, df 3; school F 6.96, df 3; community: F 18.20, df 3. cData missing for two participants. dANOVA: home: F 8.82, df 4; school F 6.67, df 4; community: F 17.29, df 4. *Significant at p .05. bANOVA:
aBased
NTID speech intelligibility ratingd (1) speech is unintelligible; (2) only isolated words or phrases are intelligible; (3) half of the message is understood; (4) speech is intelligible with some exceptions; and (5) speech is completely intelligible. 1–5 1.7 (1.3) Ref Ref 1–7 2.4 (2.0) Ref Ref 1–8 3.0 (2.5) Ref Ref 5 (n 9) 1–6 2.1 (1.3) .30 1–9 3.0 (2.0) .70 1–8 3.5 (2.1) 4 (n 34) 1.1 (3.0, .8) .4 (2.4, 1.6) 2.1 (4.1, .0) .05* 1–7 3.5 (2.2) 3–10 4.9 (2.1) .30 1–10 5.4 (2.3) 3 (n 11) 2.0 (3.7, .4) .9 (2.6, .8) 3.3 (5.0, 1.5) .02* .001* 2–10 4.4 (2.7) 3–10 5.4 (2.5) 5–10 6.6 (1.8) 2 (n 7) 3.4 (4.7, 2.1) 2.8 (4.2, 1.5) 5.2 (6.5, 3.8) .001* .001* .001* 1–10 5.5 (3.2) 1–10 5.8 (2.8) 1–10 8.6 (2.3) 1 (n 16)c 3.8 (5.6, 2.1) 3.4 (5.2, 1.5) 5.6 (7.5, 3.8) .001* .001* .001*
(I) speech is not affected; (II) speech is imprecise but usually understandable to unfamiliar listeners; (III) speech is unclear and not usually understandable to unfamiliar listeners out of context; and (IV) no understandable speech. VSS 1–2 1.3 (.5) Ref Ref 1–4 2.0 (1.1) Ref Ref 1–8 2.8 (2.4) Ref Ref I (n 8) 1–7 2.4 (1.7) .15 1–10 3.5 (2.3) .10 1–10 3.9 (2.4) .20 II (n 43) 1.2 (.4, 2.8) 1.5 (.3, 3.2) 1.2 (.6, 3.0) 2–10 4.0 (2.5) 2.8 (.7, 4.8) 3–10 5.1 (2.3) 3.1 (1.0, 5.3) 4–10 6.1 (1.8) 3.4 (1.1, 5.6) III (n 9) .009* .006* .004* 1–10 5.5 (3.1) 4.2 (2.4, 6.0) 1–10 5.8 (2.7) 3.8 (1.8, 5.7) 1–10 8.4 (2.4) 5.7 (3.7, 7.7) IV (n 17)c .001* .001* .001*
Mean difference (95% CI) Range p Mean difference (95% CI) Range
Meana (SD)
Mean difference (95% CI)
p
Range
Mean (SD)
School Home
Level of difficulty for the child participating because of their communication, in the:
Table V. Impact of motor speech impairment, speech intelligibility, and parent perception of communication on participation within the home, school, and community.
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Mean (SD)
Community
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the home, school, and community. Similar findings were identified in regard to speech intelligibility (i.e., children with poorer speech intelligibility typically showed greater difficulties in participation), although some evidence was found to suggest that slight reductions in speech intelligibility (NTID level 4) influences children’s participation within the community (mean difference 2.1, 95% CI 4.1 to .0, p .05; Table V).
Discussion In this study, 90% of children with CP were found to have some degree of motor speech impairment at 5–6 years of age. This figure is higher than recent population-based studies suggest (33–63%; Andersen et al., 2010; Cockerill et al., 2014; Nordberg et al., 2013; Parkes et al., 2010), which may be due to the different aspects of speech measured. In our study, the VSS was used to measure speech, which takes into consideration all motor speech sub-systems. Various definitions of motor speech impairment have been used previously, making it difficult to compare findings across studies. In order to understand the precise frequency of motor speech impairment, it is essential that studies use consistent definitions. Wider use of the VSS may potentially address this current limitation as it has been identified as a suitable tool for surveillance purposes (Pennington et al., 2013b; Virella et al., 2012). In a study based on VSS ratings for 155 children with CP (completed by parents, speech pathologists, and other health professionals), it was found that the scale was easier to apply and had higher levels of inter-rater reliability than the CFCS and FCCS (Virella et al., 2012). However, the developers of the VSS acknowledge that the CFCS or FCCS may compliment the scale given its specific focus on speech (Pennington et al., 2013b; Virella et al., 2012). Given the similarities between the VSS and NTID scales, it is not unexpected that motor speech impairment was associated with reduced speech intelligibility, with higher VSS levels corresponding to greater reductions in intelligibility (i.e., lower levels on the NTID scale). The same pattern was evident in regard to children’s functional communication (CFCS and FCCS) outcomes in that VSS level II and NTID level 4 were associated with more favourable CFCS and FCCS ratings. However, it is important to consider that these general trends may not apply to the entire population. Within the present sample, a small number of children with mild deficits (VSS level II) presented with considerable functional communication limitations (CFCS and FCCS level IV). This suggests that some caution is needed in presuming that all children within VSS level II will demonstrate more positive activity and participation outcomes, and that these areas should be measured on an
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Motor speech impairment associated with CP individual basis. It is as yet unknown whether the impact of mild speech deficits changes over time. Based on the adult literature concerning populations other than CP (e.g., traumatic brain injury, multiple sclerosis, and Parkinson’s disease), speech intelligibility and severity of dysarthria do not necessarily predict difficulty in communicative participation (Hartelius, Elmberg, Holm, Lovberg, & Nikolaidis, 2008; McAuliffe, Carpenter, & Moran, 2010; Yorkston, Klasner, & Swanson, 2001). With regard to children’s participation within the home, school, and community, participation became increasingly more difficult in each environment as VSS ratings increased and NTID speech intelligibility ratings decreased. This is not surprising and is consistent with previous research reporting a link between severe communication deficits and reduced social participation (Clarke et al., 2012; McFadd & Hustad, 2013). The general pattern of results indicated that participation was easiest in familiar environments (i.e., at home), compared to school and community settings. However, for children within VSS levels III–IV and NTID levels 3–1, home-based participation was significantly more difficult compared to children with no motor speech or intelligibility issues.These restrictions may potentially be related to parents’ interaction with children (Pennington & McConachie, 2001) and in turn can detrimentally impact on children’s quality-of-life (Dickinson, Parkinson, Ravens-Sieberer, Schirripa, Thyen, Arnaud, et al., 2007). It is likely, however, that a range of parent, child, and environmental factors influence children’s participation within the home. An encouraging finding is that parents rated the ease in which children at VSS level II were able to participate within the home, school, and community similarly to children with no motor speech impairment. This suggests that mild speech deficits do not substantially affect children’s ability to participate in a range of environments. Although some exceptions may exist, these findings imply that intervention focused on enhancing participation is warranted predominately for children with moderate or severe motor speech impairments (VSS levels III–IV). Further exploration is needed to determine the ways to achieve this as few studies have investigated the effects of speech therapy on the participation of children with CP. One study to do so found that, whilst some children with moderate-to-severe dysarthria demonstrated improvements in communicative participation following speech therapy, increases in speech intelligibility were not associated with changes in participation (Pennington, Roelant, Thompson, Robson, Steen, & Miller, 2013a). Thus, intervention at an impairment level (i.e., targeting breath support, phonation, and speech rate) resulting in improved intelligibility does not necessarily translate into improved participation. This suggests that intervention strategies specifically targeting participation are needed to improve outcomes in this area.
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An important strength of this study is that we recruited a community cohort of children with CP drawn from a representative sample. Further, participants were similar to non-participants across key variables (e.g., CP motor type and distribution, and the presence of epilepsy, cognitive, hearing, and vision impairment). This provides some confidence that findings reflect the general population of children with CP aged 5 and 6 years. However, as the current study forms part of a broader project investigating speech and language in children with CP, it is possible that parents who were concerned about their child’s development in these areas were more likely to provide consent, potentially resulting in an over-representation of motor speech impairment being reported here. There are important factors that were not addressed here that warrant consideration in future research. For instance, the impact of co-existing communication impairments (e.g., language; Liégeois, Mahony, Connelly, Pigdon, Tournier, & Morgan, 2013) and environmental factors were not addressed. Potentially associated factors such as children’s cognition, socioeconomic status, and non-English speaking background may also be considered in future research. These additional factors may explain why some children within VSS level I demonstrated reduced outcomes on the CFCS and FCCS. Based on the work by McFadd and Hustad (2013), which showed that restrictions in social participation were only evident when language was affected, it is possible that, within the current sample, co-morbid language impairment influenced children’s activity and participation ratings. Although not the focus of this paper, this is evidently likely, given that 93% (26/28) of children within VSS levels III–IV demonstrated language impairment. As the measures used in this study are classification tools, detailed descriptions of children’s motor speech impairment and activity and participation were not possible. Further research is needed to examine the features of motor speech impairment in children with CP and the specific restrictions in activity and participation they may experience. A limitation of the present study is the use of an equal appearing interval scale as a measure of speech intelligibility. It is acknowledged that measures such as Percent Phonemes Correct or those that rely on word identification provide a more objective estimation of children’s intelligibility. An objective standardized measure of speech intelligibility was not included in the current study in order to reduce the burden of the assessment length on participants. The NTID scale provided a means to estimate intelligibility without any inconvenience to children and families. Given the absence of an objective measure of intelligibility in this study, further research is needed to examine the association between standardized intelligibility scores with ratings from the VSS, CFCS, and FCCS. Additionally, future research may evaluate the association between the CFCS and FCCS scales. Taken from our data, participants assigned to level II of the
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FCCS were more likely to be rated at level III of the CFCS, which may imply an association between these levels. As the scales included in this study were all completed by the same rater, it is possible that ratings on one scale may have influenced ratings on another. A final factor to consider when interpreting the present findings is that the perception of communication on the ease with which children participate within the home, school, and community was measured using parent report. Although parents know their child best, it is possible that they may have under- or over-estimated the difficulty children experience within each setting, particularly within the school setting which they may not regularly attend. The use of self-report to measure the participation of children with disabilities has been advocated (Michelsen, 2013). Future research investigating the activity and participation of children with motor speech impairment should consider the opinions of children if at all possible. This would provide critical insight into children’s level of enjoyment during activities, which was not captured by the current study. Research suggests that children with severe communication difficulties demonstrate higher levels of enjoyment during activities compared to matched peers (Thirumanickam et al., 2011). Therefore, although our findings highlight the negative impact of motor speech impairment on children’s participation, children may have still demonstrated high levels of enjoyment during activities.
Conclusions Motor speech impairment is commonly experienced by children with CP. Findings indicate that an estimated 90% of children with CP aged 5–6 years demonstrate motor speech deficits based on the VSS. The general pattern of results indicated that activity and participation limitations increased with greater reductions in motor speech ability and speech intelligibility. The finding that children with mild motor speech impairment (VSS level II) had comparable participation to children with no motor speech impairment, suggests a relatively positive functional outcome for this study’s age group compared to those children at VSS levels III–IV.
Note 1.
Defined by Eadie, Yorkston, Klasner, Dudgeon, Deitz, Baylor, et al. (2006) as “taking part in life situations where knowledge, information, ideas, or feelings are exchanged” (p. 309).
Acknowledgements We are grateful to all the families who participated in this study. We kindly thank Sue Reid (Manager of
the VCPR) and the following departments at the Royal Children’s Hospital, Melbourne, for their generous assistance in recruiting families: Developmental Medicine, Orthopaedics, and the Victorian Paediatric Rehabilitation Service. This project was funded by a National Health and Medical Research Council (NHMRC) Postgraduate Scholarship (#607448) and a Speech Pathology Australia Nadia Verrall Research Grant. The following authors were funded by NHMRC: AM (Career Development Award #607315); SR (Practitioner Fellowship #1041892); and FM (Population Health Capacity Building Grant #436914 and Early Career Fellowship #1037449). Support was also received from the Victorian Government’s Operational Infrastructure Support Program. Declaration of interest: The authors report no conflicts of interest. The authors alone are responsible for the content and writing of the paper. References Andersen, G., Mjoen, T. R., & Vik, T. (2010). Prevalence of speech problems and the use of augmentative and alternative communication in children with cerebral palsy: A registry-based study in Norway. Perspectives on Augmentative and Alternative Communication, 19, 12–20. Barty, E., & Caynes, K. (2009). Development of the Functional Communication Classification Scale. Paper presented at the 3rd International Cerebral Palsy Conference, Sydney, Australia. Clarke, M., Newton, C., Petrides, K., Griffiths, T., Lysley, A., & Price, K. (2012). An examination of relations between participation, communication and age in children with complex communication needs. Augmentative and Alternative Communication, 28, 44–51. Clarke, M. T., Newton, C., Griffiths, T., Price, K., Lysley, A., & Petrides, K. V. (2011). Factors associated with the participation of children with complex communication needs. Research in Developmental Disabilities, 32, 774–780. Cockerill, H., Elbourne, D., Allen, E., Scrutton, D., Will, E., McNee, A., et al. (2014). Speech, communication and use of augmentative communication in young people with cerebral palsy: The SH & PE population study. Child: Care, Health and Development, 40, 149–157. Colver, A., Thyen, U., Arnaud, C., Beckung, E., Fauconnier, J., Marcelli, M., et al. (2012). Association between participation in life situations of children with cerebral palsy and their physical, social and attitudinal environment: A cross-sectional multi-centre European study. Archives of Physical Medicine and Rehabilitation, 93, 2154–2164. Dickinson, H. O., Parkinson, K. N., Ravens-Sieberer, U., Schirripa, G., Thyen, U., Arnaud, C., et al. (2007). Self-reported quality of life of 8–12-year-old children with cerebral palsy: A crosssectional European study. Lancet, 369, 2171–2178. Eadie, T. L., Yorkston, K. M., Klasner, E. R., Dudgeon, B. J., Deitz, J. C., Baylor, C. R., et al. (2006). Measuring communicative participation: A review of self-report instruments in speech-language pathology. American Journal of SpeechLanguage Pathology, 15, 307–320. Eliasson, A. C., Krumlinde-Sundholm, L., Rosblad, B., Beckung, E., Arner, M., Ohrvall, A. M., et al. (2006). The Manual Ability Classification System (MACS) for children with cerebral palsy: Scale development and evidence of validity and reliability. Developmental Medicine and Child Neurology, 48, 549–554.
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Appendix: Scale for parent ratings of their child’s participation within the home, school, and community Please rate how easy it is for your child to participate because of their ability to communicate. (a) In the home 1 Extremely easy
2
3
4
5
6
7
8
9
10 Extremely difficult
(b) At school/kindergarten 1 2 Extremely easy
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10 Extremely difficult
(c) In the community 1 2 Extremely easy
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10 Extremely difficult
