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National Guideline Alliance (UK). Cerebral palsy in under 25s: assessment and management. London: National Institute for Health and Care Excellence (NICE); 2017 Jan. (NICE Guideline, No. 62.)
Review question: In children and young people with cerebral palsy, what interventions are effective in improving speech intelligibility?
15.1. Introduction
Motor speech disorders are common in cerebral palsy, resulting in problems with speech intelligibility. There is an association between the overall severity of the movement difficulty and the level of intelligibility. Children and young people with little or no speech often have multiple challenges, including learning disability (intellectual disability), epilepsy, vision or hearing impairments, and feeding difficulties. Speech disorders are more prevalent in the dyskinetic forms of cerebral palsy, including children and young people with unilateral spastic cerebral palsy.
A lack of intelligibility can be a barrier to social engagement, education, employment and impact on self-esteem and quality of life for an individual. Even when intelligibility difficulties are present, speech is still often the quickest and most effective way for children and young people to communicate, particularly within families. Interventions aimed at improving speech intelligibility are therefore important to consider.
Speech production is a challenging area to study as its development is prolonged and varied in a child’s early years. Speech development is also influenced by other factors such as cognition, so a child’s potential for speech may be difficult to determine. Consequently, the same intervention in different children may have very different outcomes.
The Committee not only looked at areas such as speech production, intelligibility and expressive language but also quality of life, participation and self-confidence. These areas were identified based on existing guidelines, published reviews and personal experience. Those then felt to be most important were prioritised for detailed systematic review.
The objective of this review was to assess the clinical and cost effectiveness of interventions in improving speech intelligibility in children and young people with cerebral palsy.
Systematic reviews of randomised controlled trials or single experimental trials were considered the most appropriate study designs to answer the review question. Where possible, the Committee identified the most recent systematic review available and updated it with the latest studies that matched the criteria specified in the review.
15.2. Description of clinical evidence
One Cochrane review was identified on speech and language therapy in children and young people with cerebral palsy (Pennington 2004). The Cochrane review aimed to assess the effectiveness of speech and language therapy to improve the communication skills of children and young people with cerebral palsy including, but not limited to, speech intelligibility. Although the focus of this review was on speech, the results presented address wider communication skills. Both group and single-case experimental designs were included. Single-case experimental designs were included if communication behaviours were allocated to treatment or control and both behaviours were measured at baseline, intervention and follow-up phases in order to allow for causal inference. The review looked at any child or young person under 20 years of age with any communication disorder associated with cerebral palsy, including: dysarthria, dyspraxia and mixed syndromes; or their communication partners. Similarly, the review studied both interventions given directly to the child or young person with the aim of developing their communication skills, as well as those therapies given to familiar communication partners (families, teachers, teaching assistants, peers) that aim to change the communication partners’ conversation style and help the child or young person’s communication. The outcome measures considered were:
- measures of communication
- measures of family stress and coping
- children’s quality of life
- children’s participation
- satisfaction of patient and family with treatment
- non-compliance with treatment.
Of the 17 papers included by this review, the 9 papers focusing on therapies given directly to children (8 single-case studies and 1 interrupted time series study) have been considered in order to address the current review question.
Four additional studies have been found (Fox 2012, Miller 2013, Pennington 2013, Ward 2014) that looked at speech and language therapy interventions in children and young people with cerebral palsy. Miller and colleagues used a group design pre- versus post-intervention and looked at change in voice quality, whereas the paper by Ward used a single-subject A1BCA2 multiple baseline design to study effects on speech production accuracy. Fox 2012 conducted a study to examine the effect of intensive voice treatment (LSVT LOUD) in 5 children with spastic cerebral palsy, measuring results at baseline, post-treatment and 6-week follow-up. Finally, Pennington 2013 conducted a study in 15 children with dysarthria and cerebral palsy who received 3 sessions of therapy per week for 6 weeks. Results have been compared at baseline, and at 1, 6 and 12 weeks after therapy. For the detailed description of the included studies, see Table 63.
Table 63
Summary of included studies (Cochrane review).
As GRADE was not done for this question; the quality of the evidence was reported by study based on the study design and risk of bias. Included studies have all been assigned a very low-quality evidence status, as their study design does not allow for generalisation of the results (RCT would have been the most appropriate design for this intervention review). For more details, please see section 15.5.4 on the quality of the evidence.
Given the very wide range of communication aspects targeted, interventions used in the review, and the methodology employed by the Cochrane review, a narrative summary of the evidence has been used in this evidence review.
For full details, see protocol in Appendix E. See also the study selection flow chart in Appendix F, study evidence tables in Appendix J and the exclusion list in Appendix K.
15.3. Economic evidence
No economic evaluations of interventions relevant improving speech intelligibility were identified in the literature search conducted for this guideline and this review question was not prioritised for additional economic analysis. Full details of the search and economic article selection flow chart can be found in Appendix E and Appendix F, respectively.
15.4. Evidence statements
15.4.1. Speech production and Intelligibility
There is very low-quality evidence from 1 pre-post intervention study with 16 participants with cerebral palsy (Miller 2013) that observed differences pre- versus post-intervention in speech intelligibility. The study focused on examining the correlation between voice quality (grade, roughness, breathiness, asthenia and strain) and speech intelligibility, but mean intelligibility scores for each participant were also reported. Intelligibility scores were based on the mean percentage intelligibility score per participant from the single word and connected speech (cartoon strip) results obtained from the multiple unfamiliar listeners. An overall improvement in speech intelligibility was observed, as the mean score increased from 29.70 pre-intervention to 45.70 post-intervention.
There is very low-quality evidence from 1 study with interrupted time series design (Pennington 2010) to suggest an overall improvement in speech production in 15 children aged 12 to 18 years (mean=14, SD=2) and able to comprehend simple instructions, who received intervention focusing on respiratory and phonatory control, and control of speech rate and phrase length. No changes in speech production that were understandable to familiar and unfamiliar adults were observed at baseline (6 weeks and 1 week prior to treatment). Following treatment the estimated increase in intelligibility to familiar listeners was 14.7% (95% CI 9.8–19.5) for single words and 12.1% (95% CI 4.3–20.0) for connected speech. For unfamiliar listeners the immediate post-intervention estimated increase was 15.0% (95% CI 11.73–18.17) for single words and 15.9% (95% CI 11.8–20.0) for words in connected speech. No differences were observed between post-intervention scores and follow-up scores taken at 1 and 6 weeks after intervention completion for either single words or connected speech when heard by either a familiar or unfamiliar listener.
There is low-quality evidence from another single-case multiple baseline study (Ward 2014) with 6 children with cerebral palsy who received speech and language therapy using a tactile-kinethetic therapy programme (PROMPT) to suggest a significant change in performance level post-intervention. Weekly speech probes containing trained and untrained words were administered individually to each participant. The speech probes were used to analyse motor-speech movement pattern (MSMP) and perceptual accuracy (PA). Data on MSMP showed that between phases A1-B and B-C 6 out of 6 and 4 out of 6 children, respectively, recorded a significant increase; 5 participants achieved a significant increase at phase A2 (12 weeks follow-up) as compared to phase A1. Data on PA showed that between phases A1-B and B-C, 4 out of 6 and 1 out of 6 children, respectively, recorded a significant change in performance; all participants achieved a significant increase at phase A2 as compared to phase A1.
There is very low-quality evidence from 1 multiple baseline single-subject design study (Fox 2012) with 5 children with spastic cerebral palsy and dysarthria that showed that changes in acoustic measures of vocal functioning after Lee Silverman Voice Treatment (LSVT LOUD) were not consistent across participants. Although an improved perception of vocal loudness immediately after treatment was reported by parents, maintenance of such changes at 6-week follow-up varied across participants.
There is very low-quality evidence from 1 study (Pennington 2013) with 15 children with cerebral palsy and dysarthria that showed that children’s mean speech intelligibility (both single words and connected speech) improved post-treatment when rated by both familiar listeners and unfamiliar listeners.
15.4.2. Pre-intentional, non-verbal communication
There is very low-quality evidence from 1 single-case study (Richman & Kozlowski 1977) to suggest an increase in pre-intentional communication in a 9-year-old child with severe cognitive impairment who received intervention aimed to increase her amount of eye contact, time she kept her head in upright position, and her imitative vocalisations. Wide variation was observed at baseline in each of the 3 behaviours. Increases were observed during the intervention phase. Behaviours reduced during the reversal phase, and increased again once the treatment was recommenced. Increased scores were also observed at 12-month follow-up.
15.4.3. Expressive language
There is very low-quality evidence from 1 single-case study (Campbell & Stremel-Campbell 1982) to suggest an improvement in expressive language in 1 child aged 10 years with CP and moderate language delay who received intervention aimed to teach the use of ‘is/are’ in 3 linguistic structures: ‘wh’ questions (what, why, who, where, etc.), ‘yes/no’ reversal questions and statements). Training criterion was established at 80% correct in each of 2 consecutive 5-session blocks for’ is/are’ use in each of the syntactic structures. At baseline measurement, ‘is/are’ were produced correctly in 0 to 10% of ‘wh’ questions, 0 to 10% of yes/no reversal questions and 0 to 35% of statements. During intervention, the percentage of correct productions rose steeply for all 3 targeted structures. However, the number of training sessions required to reach criterion performance on ‘is/are’ use for a given syntactic structure varied: the child required 70 sessions to reach criterion on ‘wh’ questions and 45 session on the ‘yes/no’ reversal questions. Because the participant transferred to a different school system prior to the completion of training, is/are use in statements was not trained. Level showed considerable variations during the maintenance phase. Generalisation to use in spontaneous speech showed increases from baseline for ‘yes/no’ questions, but much lower levels than observed with intervention.
15.4.4. Expressive communication (augmentative and alternative communication)
There is very low-quality evidence from 5 single-case studies that focused on the production of nonverbal messages, teaching children and young people to use individual communication functions.
One study (Hunt 1986) observed one 7-year-old child with severe intellectual impairment and multiple disabilities who communicated via vocalisation, 1 gesture, 2 manual signs, and by touching the listener. The child was taught to request 4 objects or events by eye pointing to line drawings symbolising the object or action. Baseline measurements of interactions were stable, showing infrequent use of any of the requests. The first request showed a steady increase and reached criterion (3 successive correctly produced requests) in 16 sessions; the second request was produced without direct teaching; the third request also increased steadily during the intervention phase and reached criterion in 13 sessions; the final request also generalised without direct teaching.
One study (Pinder & Olswang 1995) observed 4 children and young people with cerebral palsy who were taught to request either an object or ‘more’ by looking at the adult and the object, the untaught request acted as a control. Baseline measurements were stable for 3 of the children with requests made to less than 20% of probes. For all children, increases in the production of both taught and untaught requests were observed during intervention across both treatment and generalisation situations. Levels of requests were maintained for 4 weeks after therapy had been withdrawn.
One study (Sigafoos & Couzens 1995) observed a 6-year-old child with severe cerebral palsy of unspecified type, who had moderate cognitive impairment and required assistance for all activities of daily living, and who was taught to use 3 requests for objects by using micro-teaching strategies. Baseline percentages of correct production of the 3 requests (not separated) ranged from 0% to 35%. For the first request, correct production increased from 35% to 60% with verbal prompting and to 80% to 100% when expectant delay was used and verbal prompts were faded. Although requests increased from the first to the second phase of intervention, they showed a downward trend in the latter part of the second phase. The other target requests (tested after intervention for the first one) were correct for 65% and 30% of 17 trials.
One study (Davis 1998) observed an individualaged 15 years who communicated using vocalisation, gesture and word phrases via alternative and augmentative communication (AAC), who was taught to produce conversational responses to statements made by 3 communication partners. The participant communicated by yes/no responses only but had access to a voice output communication device with pre-stored phrases and spelling for novel words. At baseline, responses to statements were rare, being produced following 0% to 20% of statements by each of the 3 partners in communication (means=1.8%; 2.5%; 4.0%). During the intervention phase the percentage of responses immediately increased, following an average of 41.7% and 52% of statements by the first 2 partners. However, considerable variation was observed in the frequency of responses during intervention, ranging from 0% to 60% and from 20% to 80% with each partner. As intervention with the third partner was not carried out because of the child’s family moving away from the area where the research was conducted, it remained at baseline level and used as control.
Finally, 1 study (Hurlburt 1982) observed 3 children with severe cerebral palsy and cognitive impairments who were trained to use Blissymbols and iconic symbols to name objects. The proportions of Blissymbols and iconic symbols used to label taught and untaught items was calculated before and throughout training. Results showed that the children required approximately 4 times as many trials to acquire Blissymbols as iconic pictures. All children also produced iconic symbols more frequently than Blissymbols in maintenance and generalisation probes, and named more untaught objects using iconic symbols than Blissymbols. Finally, participants almost always showed more iconic symbols responses than Blissymbol responses in daily spontaneous usage.
15.4.5. Receptive vocabulary
There is very low-quality evidence from 1 single-case study (Dada 2009) to suggest an improvement in the identification of graphic symbols in 3 children, aged from 8 to 12 years of age, with cerebral palsy, who had fewer than 15 spoken words. During baseline, 2 children selected 2 out of the 24 items named. During the intervention, the percentage of correct identification rose steeply for all target items. During follow-up, children continued to select items from the first 2 sets of vocabulary items. However, follow-up was not long enough to show retention of the third set of taught words.
15.4.6. Quality of life
No evidence was retrieved for this outcome.
15.4.7. Self-confidence
There is very low-quality evidence from 1 study (Pennington 2013) with 15 children with cerebral palsy and dysarthria that showed that children’s communicative participation in interactions at home and school improved post-treatment, when measured by parents and teachers.
15.4.8. Family stress and coping
No evidence was retrieved for this outcome.
15.4.9. Satisfaction of patient and family with treatment
No evidence was retrieved for this outcome.
15.5. Evidence to recommendations
15.5.1. Relative value placed on the outcomes considered
The aim of this evidence review was to assess the clinical and cost effectiveness of interventions in improving speech intelligibility in children and young people with cerebral palsy. The Committee agreed that participation and speech intelligibility were to be the critical outcomes. In addition, quality of life, self-confidence, family stress and coping, and satisfaction of patient and family with the treatment were considered to be important outcomes.
15.5.2. Consideration of clinical benefits and harms
The Committee were aware that, despite the fact that a wide range of different interventions were available for children and young people with speech difficulties, good-quality evidence was very limited. In addition, evidence was not retrieved for the following outcomes listed in the review protocol: quality of life, self-confidence, family stress and coping, and satisfaction with the treatment.
This review focused specifically on interventions to improve speech intelligibility. Other studies, targeting a broader range of communication skills, were included, as intelligible speech may not be a realistic expectation for children and young people with severe functional disability, particularly if accompanied by cognitive impairments. The available evidence suggested there may be benefit from interventions addressing aspects of communication development, such as language understanding, and expressive communication in its widest sense (including functions such as making requests and participating in conversations) using a range of non-verbal methods: eye pointing, gesture, turn-taking, and the use of augmentative and alternative communication. Interventions may also target the skills of the people around the child or young person with cerebral palsy to create an environment that supports communication.
There is wide variation in practice in the UK concerning interventions to improve speech intelligibility. Non-speech oral motor exercises, oral motor therapies and speech articulation therapies targeting specific speech sounds are in use. No evidence was identified to support the use of such interventions in cerebral palsy. Some low-quality evidence was found to suggest that therapy focusing on teaching children to produce slower, louder speech may be associated with increased speech intelligibility, voice quality and clarity. There was also low-quality evidence to suggest tactile-kinesthetic therapy may benefit some children. Based on this evidence, and their clinical experience, the Committee was confident in making a recommendation in this area. Other recommendations focused more broadly on the risk of speech, language and communication difficulties in children and young people with cerebral palsy, and the possible need for augmentative and alternative communication.
The Committee agreed that regular assessments should be carried out in children and young people with cerebral palsy in order to identify concerns regarding speech, language and communication skills. The Committee also agreed that these assessments should be carried out by a multidisciplinary team, including a speech and language therapist. This decision was derived by consensus based on the Committee’s clinical experience.
15.5.3. Consideration of economic benefits and harms
The Committee were unable to recommend any specific intervention because the plan would be individualised to the patient, which may involve several interventions. Despite this, if there were concerns about speech, language and communication, including speech intelligibility, the Committee agreed that all children and young people with cerebral palsy should be referred to a speech and language therapist. According to NHS Reference Costs 2015, the cost per consultant-led attendance with a speech and language therapist is £101 (WF01B, Non-Admitted Face to Face Attendance, First Attendance, Service Code 652).
15.5.4. Quality of evidence
One systematic review of 8 single-case studies and 1 interrupted time series study was included in this evidence review. In addition, 3 pre- versus post-intervention group studies and 1 study using single-subject multiple baseline design were retrieved following publication of the included systematic review. The quality of the included studies was very low. Main reasons of bias were that the study design used did not allow for generalisation of the results (randomised controlled trials [RCTs] would have been the most appropriate design for this intervention review) and many of the included studies reported low reliability scores between unblinded raters. In addition, there was a very wide range of communication aspects targeted and interventions used in the review.
15.5.5. Other considerations
In reviewing the evidence it was noted that interventions were often tailored to the communication skills profile of an individual child or small group of children and/or young people. Communication skills and needs are influenced by a complex interaction of variables, including type of cerebral palsy, severity of functional disability, level of cognition, the skills of conversational partners and the opportunities for communication in different environments. The role of skilled assessment, as a precursor to choosing appropriate interventions, was recognised by the Committee in the development of the recommendations.
The recommendations related to this evidence review were based on the evidence and the Committee’s clinical experience.
15.5.6. Key conclusions
Although the Committee recognised that there was no good-quality evidence to support speech and language therapy interventions for children with cerebral palsy, it believed that, on an individual basis, certain approaches might be effective for a particular child. For that reason it recommended a specialist assessment where there were concerns and access to interventions, particularly in the areas of speech intelligibility, augmentative and alternative communication, and training for families, carers and professionals in strategies to support communication. It noted that no evidence had been identified to suggest there were likely harmful effects associated with speech and language therapies for these children and young people.
15.6. Recommendations
- 58.
Regularly assess children and young people with cerebral palsy during routine reviews to identify concerns about speech, language and communication, including speech intelligibility.
- 59.
Refer children and young people with cerebral palsy for specialist assessment if there are concerns about speech, language and communication, including speech intelligibility.
- 60.
Specialist assessment of the communication skills, including speech intelligibility, of children and young people with cerebral palsy should be conducted by a multidisciplinary team that includes a speech and language therapist.
- 61.
Recognise the importance of early intervention to improve the communication skills of children and young people with cerebral palsy.
- 62.
Offer interventions to improve speech intelligibility, for example targeting posture, breath control, voice production and rate of speech, to children and young people with cerebral palsy:
- who have a motor speech disorder and some intelligible speech and
- for whom speech is the primary means of communication and
- who can engage with the intervention.
15.7. Research recommendations
None identified for this topic.
- Improving speech, language and communication: speech intelligibility - Cerebral ...Improving speech, language and communication: speech intelligibility - Cerebral palsy in under 25s: assessment and management
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