Catherine Adams, Elaine Lockton, Jenny Freed, Jacqueline Gaile, Gillian Earl, Kirsty McBean, Marysia Nash, Jonathan Green, Andy Vail and James Law

Transcription

1 INT J LANG COMMUN DISORD, MAY JUNE 2012, VOL. 47, NO. 3, Research Report The Social Communication Intervention Project: a randomized controlled trial of the effectiveness of speech and language therapy for school-age children who have pragmatic and social communication problems with or without autism spectrum disorder Catherine Adams, Elaine Lockton, Jenny Freed, Jacqueline Gaile, Gillian Earl, Kirsty McBean, Marysia Nash, Jonathan Green, Andy Vail and James Law Human Communication and Deafness Division, University of Manchester, Manchester, UK NHS Lothian, Edinburgh, UK Speech and Language Therapy Department, Royal Hospital for Sick Children, Edinburgh, UK Child and Adolescent Psychiatry, University of Manchester, Manchester, UK School of Education, Communication and Language Sciences, University of Newcastle, Newcastle, UK (Received August 2011; accepted December 2011) Abstract Background: Children who show disproportionate difficulty with the pragmatic as compared with the structural aspects of language are described as having pragmatic language impairment (PLI) or social communication disorder (SCD). Some children who have PLI also show mild social impairments associated with high-functioning autism or autism spectrum disorder (ASD). There is little robust evidence of effectiveness of speech language interventions which target the language, pragmatic or social communication needs of these children. Aims: To evaluate the effectiveness of an intensive manualized social communication intervention (SCIP) for children who have PLI with or without features of ASD. Methods & Procedures: In a single-blind RCT design, 88 children with pragmatic and social communication needs aged 5;11 10;8, recruited from UK speech and language therapy services, were randomly assigned in a 2:1 ratio to SCIP or to treatment-as-usual. Children in the SCIP condition received up to 20 sessions of direct intervention from a specialist research speech and language therapist working with supervised assistants. All therapy content and methodology was derived from an intervention manual. A primary outcome measure of structural language and secondary outcome measures of narrative, parent-reported pragmatic functioning and social communication, blind-rated perceptions of conversational competence and teacher-reported ratings of classroom learning skills were taken pre-intervention, immediately post-intervention and at 6-month follow-up. Analysis was by intention to treat. Outcomes & Results: No significant treatment effect was found for the primary outcome measure of structural language ability or for a measure of narrative ability. Significant treatment effects were found for blind-rated perceptions of conversational competence, for parent-reported measures of pragmatic functioning and social communication, and for teacher-reported ratings of classroom learning skills. Conclusions & Implications: There is some evidence of an intervention effect on blind and parent/teacher-reported communication outcomes, but not standardized language assessment outcomes, for 6 11-year-old children who have pragmatic and social communication needs. These findings are discussed in the context of the increasingly central role of service user outcomes in providing evidence for an intervention. The substantial overlap between the presence of PLI and ASD (75%) across the whole cohort suggests that the intervention may also be applicable to some verbally able children with ASD who have pragmatic communication needs. Keywords: language impairment, pragmatics, social communication disorder, treatment, randomized controlled trial Address correspondence to: Catherine Adams, Human Communication and Deafness Group, Ellen Wilkinson Building, University of Manchester, Oxford Road, Manchester M13 9PL, UK; catherine.adams@manchester.ac.uk International Journal of Language & Communication Disorders ISSN print/issn online c 2012 Royal College of Speech and Language Therapists DOI: /j x

2 234 Catherine Adams et al. What is already known? Children who have pragmatic and social communication needs are at risk of long term behavioural and social disadvantage. These children often have additional autism spectrum conditions and/or high-level language impairments. There are no randomized controlled trials of speech-language intervention for these children. What this paper adds: This paper reports evidence of an effect of intensive, specialist supervised speech and language therapy in improving overall conversational competence and functional social communication skills for school-age children who have persistent pragmatic and social communication needs. There was no effect of treatment on structural language skills. The majority of children with pragmatic difficulties also had a history of autism spectrum disorder or pervasive developmental disorder. Parent and teacher outcomes were important measures in the context of the implementation of a complex intervention where measurement of pragmatic and social communication skills remain challenging. Background Pragmatic language impairment (PLI) is present when children have disproportionate difficulty with the pragmatic domain of language in relation to relative strength in grammar and phonology (Bishop 2000). Pragmatic features of PLI include verbosity, excessive topic switching, a tendency to dominate verbal interactions, poor adjustment to listeners prior knowledge and limited application of inference in naturalistic interaction (Bishop and Adams 1989, Adams 2001). Symptoms characteristic of specific language impairment (SLI) are also present in PLI with some children presenting semantic errors, word-finding difficulties and persistent difficulty with receptive language. High-level language deficits such as poor comprehension of non-literal language and narratives and stories are also evident (Botting and Adams 2005). As a group, children who have PLI (CwPLI) present considerable heterogeneity in social communication and language skills, in both pattern and severity. The full profile of PLI emerges in the early school years as the disparity between structural and social language functioning becomes clear. The pragmatic characteristics of CwPLI are similar to those reported in high-functioning children who have autism spectrum disorder (HF-ASD) or pervasive developmental disorder (PDD 1 ) (Landa 2000). PLI and HF-ASD may share other abnormalities of communication such as use of stereotyped phrases, intonation and non-verbal interaction. As with CwPLI, a significant proportion of children who have HF-ASD also have a profile of language impairment similar to SLI (Kjelgaard and Tager-Flusberg 2001). There is consequently controversy surrounding the use of PLI as a diagnosis distinct from HF-ASD, since the two groups may not be distinguishable in terms of social communication and social interaction deficits. It has been suggested that CwPLI are typically differentiated from CwHF- ASD by the absence of repetitive/restricted behaviours (Bishop 1998, Leyfer et al. 2008). In addition, since the pattern of communication deficits seen in PLI clearly extends beyond the pragmatics domain, there is increasing consensus that the term social communication disorder (SCD) may be a more appropriate descriptor. In proposals for the fifth edition of the Diagnostic and Statistical Manual of the American Psychiatric Association (n.d.), SCD is categorized as a subtype of Language Impairment with features that closely match the description of PLI provided by Bishop and colleagues in earlier work. The diagnostic overlaps remain unresolved. In the current study, the term children who have PLI (CwPLI) will be used to describe children with a pattern of pragmatic and social communication needs similar to those described by Bishop (2000) and the SCD profile in DSM-5. The number of children who have significant needs in pragmatics and social communication is rising in line with increased identification of broader ASD conditions (Baird et al. 2006). Botting and Conti-Ramsden (1999) identified 22% of children attending UK specialist language units as having PLI. Emerging evidence suggests that pragmatic difficulties in the primary school years may be related to emotional and behaviour difficulties, high referral rates to educational psychology services (Mackie and Law 2010), longer-than-usual time spent in special provision (Botting et al. 1998), and specific difficulty in forming adult relationships in later life (Whitehouse et al. 2009). The need for adequate communication interventions as preventative measures is therefore considerable. Single case studies of CwPLI (Adams 2001) and SCD (Timler et al. 2005, Brinton et al. 2005) have shownpositiveeffectsofinterventiononspecificaspects of communication such as conversational skills; a case study series (Adams et al. 2006) reported a signal of change as a result of intensive speech language therapy for six CwPLI. There have been no trials of speech language interventions for CwPLI (Law et al. 2003). The aims of the present study were to examine the effectiveness of an intensive manualized social communication speech and language intervention in improving (1) language skills and (2) observed functional pragmatic ability and broader social communication of CwPLI within a small-scale randomized controlled trial.

3 RCT of speech/language therapy for children with pragmatic language impairment 235 Methods The two-arm parallel-group randomized controlled trial was carried out in accordance with the NHS Research Governance Framework for Health and Social Care (NHS 2005) and was approved by the Northern and Yorkshire NHS Research Ethics Committee (REC No. 07/MRE03/3). All parents gave written informed consent for their child s participation. Informed consent was also gained from the child (where able), their school, class teacher and learning support assistant (LSA) and the relevant local authorities. Participants The study was powered on the basis of a pilot study of the intervention (n = 6; Adams et al. 2006). This demonstrated a clinically significant standardized effect size for the Clinical Evaluation of Language Fundamentals (CELF-4; Semel et al. 2006) of 0.62 (difference in means of 3.1 with an SD of 5, based on the sum of two subtests only). Therefore, target recruitment for the current study was set at 99 participants, providing > 80% power to detect a standardized effect size of 0.6 in CELF-4 as the primary outcome. Speech and language therapists (SLTs) across the North West of England and South East Scotland referred to the trial any children on their caseloads aged 6 years to 10 years 11 months who met the following inclusion criteria: Pragmatic communication problems as observed by the child s SLT (including a minimum of two out of five pragmatic behaviours from a social communication behaviour checklist (SCBC, see appendix A). Attending mainstream primary education provision and identified as having Special Educational Needs (England) or Additional Support Needs (Scotland). English as the primary language of communication and learning. Agreement with the child s school to accommodate intervention and assessment visits. No current diagnosis of core autism. Currently receiving regular, on-going attention from SLT services and able to cooperate with direct intervention. No evidence of severe difficulties in emotional development, behaviour needs, unintelligibility or hearing. All referred children were assessed for eligibility and included in the study if they met the following screening criteria: A score in the communication impaired range ( 58) on the General Communication Composite (GCC) of the Children s Communication Checklist Second Edition (CCC-2; Bishop 2003), a measure functional communication including pragmatics completed by parents. A score 5th centile on Raven s Coloured Progressive Matrices (RCPM; Raven 1979), a test of non-verbal perceptual/analogical reasoning skills. Screening assessments were carried out by a researcher in the child s school with one or both parents present. The Social Interaction Deviance Composite (SIDC) of the CCC-2 and the Social Communication Questionnaire Lifetime version (SCQ) (Rutter et al. 2003), a parent questionnaire which asks about the history of diagnostic features of autism, were also completed to provide information on group characteristics but were not used as part of the inclusion criteria. An SCQ cutoff score of 15 is considered indicative of a history of PDD, and a cut-off score of 22 is considered indicative of a history of ASD (Berument et al. 1999). 2 Randomization, blinding and schedule of assessments Following screening and baseline (T1) assessments, a researcher at the University of Manchester, who was not independent of the study, randomly assigned each child to SCIP intervention or treatment-as-usual (TAU) in a 2:1 ratio, stratified by age group (6;00 8;11 or 9;00 10;11). A 2:1 ratio was used to improve power for later moderator and mediator exploratory analyses. Allocation used three possible permuted blocks of size three: 1: SCIP TAU SCIP 2: SCIP SCIP TAU 3: TAU SCIP SCIP A new block was selected after every three children in the same age band. All assessments (delivery, coding and scoring) were completed by a research assistant (RA) blind to treatment allocation; however, families, schools and those delivering intervention could not be blind to treatment allocation. At assessment points, children, parents and schools were reminded that the RA was blind to treatment allocation and that anything related to this should not be discussed with them. Questionnaires completed by parents or teachers (CCC-2, PRO and TRO see the outcome measures) were returned

4 236 Catherine Adams et al. by post to a researcher who was not involved with their child s intervention. Reassessments were carried out in the child s school within 2 weeks following the completion of intervention/tau (T2) and at 6 months following the completion of intervention/tau (T3). SCIP intervention began within 4 weeks of pre-intervention (T1) assessment. Interventions Participants in the SCIP group ceased their usual treatment as provided by their local SLT services for the experimental intervention period. The experimental treatment was an intensive manualized social communication intervention with content within a principled framework aimed at remediation of impairments in semantics and high-level language skills, pragmatic difficulties, and social interaction and social cue interpretation (Adams and Gaile 2012). The research intervention manual provided procedures for planning intervention and establishing appropriate goals as well as all intervention activities. For each child, between 16 and 20 individual face-to-face one hour sessions of intervention (up to three sessions per week) were delivered in school over the course of one school term. Parent/teacher input was solicited throughout the setting of goals and intervention period. Each child therefore received an individualized intervention derived from the manual, but within a specified framework, as outlined above, so as to ensure that intervention choices were consistent. Two specialist research speech and language therapists (RSLT) (one at each of the two locations) and five specially trained therapy assistants (ThAs) (England n = 4, Scotland n = 1) delivered the experimental treatment. Treatment fidelity, measured by audit of planned intervention sessions versus received sessions and adherence to written activity procedure as stated in the manual (both across 10% of sample) was at 80% for both measures. A detailed and precise account of the experimental SCIP intervention, including rationale, the process of manualization, implementation, intervention components, individualization procedures, required level of practitioner expertise and treatment fidelity measures, is provided in Adams et al. (2012). Children allocated to TAU continued with the treatment being provided by their local SLT services. This provision was documented through telephone liaison with these services (data obtained for 27/29 children). Thirteen of these children (48%) received Learning Support Assistant (LSA) support provided by the Education Authority plus SLT contact or contact from another communication professional. Ten of these children received direct individual or group-based intervention. Seventeen SLTs reported general training for LSAs; three had provided training to LSAs aimed at the management of an individual child. Children in both arms continued to receive support from their LSAs (and/or classroom support for learning) throughout the study. Post-intervention, children in the SCIP intervention arm were referred back to their local SLT services for on-going care. Provision post-intervention to 6-month follow-up was documented through telephone liaison with local SLT services obtained for 21/29 children in the TAU arm and for 47/59 children in the SCIP arm. Of these children 17 (81%) in the TAU arm, and 30 (63%) in the SCIP arm, received SLT contact. Of these children, 12 (57% of TAU; 26% of SCIP) children from each arm were reported to have received direct individual or group specialist intervention. After 6-month follow-up (T3) measures, all SLTs of children allocated to TAU received a manual intervention plan based on what the child would have received had they been in the SCIP intervention arm. Outcome measures The primary outcome measure was predefined as the CELF-4 (Semel et al. 2006) Core Language Standard Score (CLSS) as a recognized, reliable and standardized measure of general language ability, which can be administered blind to treatment allocation. The relevant subtests were completed with each participant (taking into account age at that time point) to allow the calculation of composite core language scores (see appendix A). Secondary outcome measures were: Targeted Observation of Pragmatics in Children s Conversation (TOPICC; Adams et al. 2011): a semi-structured task that allows for the rating of overall quality of interaction in conversation (the same task was used in Bishop and Adams 1989). Each participant was video recorded discussing a standard series of three photographs of events with an RA who used a series of question prompts. The aim of the task was to obtain a sample of natural conversation with the child. Three sets of photographs were used in rotation for each participant at T1, T2 and T3. Following completion of all outcome measurement, the video recorded conversations were rated by a trained, independent assessor, blind to treatment allocation, at the University of Manchester. The assessor was asked to make a judgement of overall conversational skills of the child, rating them as improved, stayed the same or got worse from T1 to T3. A proportion of T1 and T3 videos (37% of total available; n = 29, TAU n = 10, SCIP n = 19) were

5 RCT of speech/language therapy for children with pragmatic language impairment 237 rated independently by a second assessor also blind to group status. Raters were asked to consider the overall impact of any pragmatic behaviours on the overall quality of interaction. The interrater reliability for the raters was Cohen s κ = 0.68, indicating a substantial inter-rater agreement for overall impression of conversation change. A pragmatics rating scale (CCC-PRAG), derived, a priori, from the CCC-2. Two highly specialist RSLTs provided face validity for this scale by identifying a list of 18 items which in their view was closely related to the content of SCIP intervention. An autism-type communication problems scale (CCC-AUT), consisting of 18 items not targeted by SCIP intervention directly, was also identified as a control for predictable bias by parents in the experimental intervention arm. Parents completing the CCC-2 would be unaware of separate PRAG/AUT lists embedded within it. CCC-PRAG and AUT lists are shown in appendix A. Scores are sums of rated items, converted from CCC-2 scoring to a single polarity, with higher scores indicating greater impairment. Split-half reliability (internal consistency) of both CCC-PRAG and CCC-AUT scales were in the acceptable range (Cronbach s α for CCC-PRAG = 0.82; for CCC-AUT α = 0.80). Parents completed CCC-PRAG/AUT lists at home within 2 weeks following the completion of intervention (T2) and at 6 months following the completion of intervention (T3). Completed CCCs were returned by post to a researcher who was not involved in the child s intervention. The Expression, Reception and Recall of Narrative Instrument (ERRNI; Bishop 2004) was used as a measure of ability to interpret, remember and tell a pictured narrative. Standardized scores are given for initial telling of the story (ERRNI-I), story recall (ERRNI-R) and comprehension (ERRNI- C). The ERRNI story was alternated at each time point (therefore the same story was used at T1 and T3). Parent-reported outcome (PRO): parents were asked, via a questionnaire, to state their judgments about the current status of their child s Language Skills (PRO-LS), Social Communication (PRO- SC), Social Situations (PRO-SS) and Peer Relationships (PRO-PR) and to rate whether these have improved, stayed the same or got worse since pre-intervention (T1). Parents completed PRO questionnaires at home within 2 weeks following the completion of intervention (T2) and at 6 months following the completion of intervention (T3). Teacher-reported outcome (TRO): teachers were asked, via a questionnaire, to state their judgments about the current status of the child s classroom learning skills (TRO-CLS) and to rate whether these have improved, stayed the same or got worse since pre-intervention (T1). Teachers completed TRO questionnaires at school at 6 months following the completion of intervention (T3) only. Statistical analysis Analysis was by intention to treat 3 and was undertaken after completion of all T3 assessments. For the primary outcome, and secondary outcomes CCC-2 PRAG/AUT and ERRNI, analysis was by linear regression with adjustment for age and the corresponding T1 measure. Analysis for TOPICC, PRO and TRO was by logistic regression with similar adjustment for age and the corresponding T1 measure. Results Participants were recruited between April 2007 and April 2008 in England and between October 2007 and September 2008 in Scotland. All assessments occurred within schedule. Figure 1 shows the trial profile. The study underrecruited participants (final n = 88) compared with the target. Fifty-nine participants were randomly assigned to the SCIP group and 29 participants were randomly assigned to the TAU group. In the SCIP group 57 participants received intended treatment and were analysed for the primary outcome. In the TAU group 28 participants received the intended treatment and were analysed for the primary outcome. Attrition was therefore 3/88 (3%) for the primary endpoint. One participant who withdrew from the study during intervention also withdrew consent for their T1 data to be used. Table 1 shows baseline demographic and clinical characteristics for each group. The groups were well matched on age, demographics, CELF-4 CLSS and CCC-2 GCC. Mean Raven s Progressive Coloured Matrices mid-point percentile score were slightly higher in the SCIP than in the TAU group. Scores on the SCQ showed that 75% of participants scores (76% of SCIP and 73% of TAU) fell within the range indicative of the child having PDD or ASD features at some stage in development. Primary outcome The primary outcome measure was CELF-4 CLSS. Figure 2 shows the spread of CELF-4 CLSS scores at T1, T2 and T3 for SCIP and TAU groups.

6 238 Catherine Adams et al. Figure 1. Trial profile. SCIP, Social Communication Intervention Project. Figure 2. CELF-4 Core Language Standard Score at T1, T2 and T3 for TAU and SCIP intervention groups. The dotted line is at the CELF-4 CLSS population mean of 100; and the dashed line is at the cut-off between normal range ( 80) and language impaired (< 80).

7 RCT of speech/language therapy for children with pragmatic language impairment 239 Table 1. Baseline demographic and clinical characteristics of each group c Whole group SCIP TAU n = 87 n = 59 n = boys, 12 girls 52 boys, 7 girls 23 boys, 5 girls Mean SD Range Mean SD Range Mean SD Range Age (months) NRS ABC1 demographics b Middle to high area a 30 (34%) 18 (31%) 12 (43%) Middle to low area a 49 (56%) 34 (58%) 15 (54%) Mixed area a 8 (9%) 7 (12%) 1 (4%) SCBC (n = 80) CCC-2 GCC RCPM percentile e CELF-4 CLSS d Language impaired CELF CLSS d < 80 (n = 56) Not language impaired CELF-4 CLSS d 80 (n = 31) CCC-PRAG (n = 86) CCC-AUT ERRNI-I d ERRNI-R d ERRNI-C d SCQ (n = 84) Non-ASD SCQ 14 a 21 (25%) 14 (24%) 7 (27%) PDD-NOS SCQ a 23 (27%) 17 (29%) 6 (23%) ASD SCQ 22 a 40 (48%) 27 (47%) 13 (50%) Notes: a n (%). b Area based on postcode. Middle to high areas include C2 only; middle to low areas include C1 only; and mixed areas included a range from A/B to D/E. c n = 87 as one child withdrew without giving permission to use the data. d Standard scores have a population mean = 100. e RCPM percentile ranges transformed into percentile midpoints, e.g. 5th 10th percentile becomes 7.5th percentile SCQ scores 15 are considered indicative of PDD-NOS; and scores 22 are considered indicative of ASD. NRS, National Readership Survey (NRS Ltd); SCBC, Social Communication Behaviour Checklist; CCC-2 GCC, Children s Communication Checklist-2 General Communication Composite; RCPM, Ravens Coloured Progressive Matrices; CELF-4 CLSS, Clinical Evaluation of Language Fundamentals Core Language Standard Score; CCC-PRAG, Pragmatics rating scale; CCC-AUT, Autism-Communication Rating Scale; ERRNI-I/R/C, Expression, Reception and Recall of Narrative Instrument Initial Telling/Story Recall/Comprehension Standard Score; SCQ, Social Communication Questionnaire. Using linear regression, comparison was made between the TAU and SCIP groups on the CELF-4 CLSS at T2 and T3 with adjustment for age and the CELF-4 CLSS at T1. No significant treatment effect was found at T2 (p = 0.78) or T3 (p = 0.87). The estimated group difference at T2 was 0.5 (95% CI = 3.1 to 4.1) and at T3 was 0.3 (95% CI = 3.7to4.4). Given that a large proportion (36%) of participants were found to have CELF-4 CLSS well within the normal range (CELF-4 CLSS > 80) at baseline, a further exploratory comparison was made for the subgroup of children who scored in the low ability/language impaired range (CELF-4 CLSS < 80) at baseline. Conclusions were similar: the estimated group differences (95% CI) at T2 and T3 were 3.9 ( 1.3 to 8.9) and 1.4 ( 4.8 to 7.6) respectively. Secondary outcomes Table 2 shows the results of secondary outcomes at T2. No significant treatment effects were found for CCC- PRAG/AUT or ERRNI at T2. However, parent ratings (PRO) showed significant differences by treatment group in favour of SCIP at T2. Table 3 shows the results of secondary outcomes at T3. TOPICC, CCC-PRAG, PRO-SC and PRO- SS, and TRO-CLS showed significant differences by treatment group in favour of SCIP at T3. However, no significant treatment effects were found for CCC- AUT, ERRNI, PRO-LS or PRO-PR at T3. Note that for PRO-PR numbers in the control group were too small for analysis by logistic regression. Discussion This is the first randomized controlled trial investigating the effectiveness of intervention for children who have PLI. The first aim was to establish the effects of the SCIP intervention compared with TAU on standardized language assessment. The primary outcome, a standardized measure of overall language performance (CELF-4 CLSS), did

8 240 Catherine Adams et al. Table 2. Secondary outcomes at Time 2 SCIP TAU Effect a n Mean (SD) n Mean (SD) Mean difference (95% CI) p CCC-PRAG (10.2) (11.4) 0.7 ( 3.3 to 4.6) 0.74 CCC-AUT (10.8) (8.9) 1.6 ( 2.1 to 5.3) 0.39 ERRNI-I (17.8) (17.3) 0.31 ( 6.9 to 7.5) 0.93 ERRNI-R (20.2) (21.2) 3.7 ( 4.7 to 12) 0.38 ERRNI-C (13.9) (12.9) 2.4 ( 3.3 to 8.1) 0.41 n (%) improved n (%) improved Odds ratio (95% CI) PRO-LS (76) 19 4 (21.1) 11.9 ( ) < PRO-SC (72.9) 20 7 (35) 5 ( ) PRO-SS (57.4) 21 3 (14.3) 8.7 ( ) PRO-PR b (48.8) 18 1 (5.6) Notes: a Mean difference by linear regression or odds ratio by logistic regression. b Numbers improved in the control group too small to compute odds ratio. p < Abbreviations are as given in table 1, except for PRO-LS/SC/SS/PR, Parent Reported Outcome Language Skills/Social Communication/Behaviour in Social Situations/Peer Relationships. not show a significant intervention effect for SCIP compared with TAU, nor did a secondary standardized measure of narrative ability (ERRNI). The identification of a single, predefined standardized outcome measure, which was sensitive enough to capture change in pragmatic impairment, although highly desirable, was not easily achievable for this complex group. Over one-third of participants recruited were found to be performing well within the normal range on CELF-4 CLSS (and ERRNI) at pre-intervention assessment. A further exploratory comparison was therefore made for the subgroup of children who scored in the low ability/language impaired range (CELF-4 CLSS < 80) at baseline. This comparison showed a trend in favour of intervention, but had insufficient power to detect plausible differences between groups. Inspection of the standard score increments and confidence intervals on CELF-4 CLSS also indicate that very large changes in raw scores (larger than those considered clinically significant) would be required to show shift in standard scores. Despite CELF-4 CLSS being a recognized, reliable and objective test of general language function, some CwPLI in this study had functional difficulties with language processing that were evident to teachers and parents but which were not detected by standardized language tests. Further, some CwPLI functioned in the normal range across all language tasks and did not present concern to teachers or parents, except in the pragmatics and social communication domain. Further characterization of the PLI population may therefore be Table 3. Secondary outcomes at Time 3 SCIP TAU Effect a n Mean (SD) n Mean (SD) Mean difference (95% CI) p CCC-PRAG (12.9) (9.0) 5.5 ( ) CCC-AUT (11.4) (9.6) 0.13 ( 4.8 to 5.1) 0.96 ERRNI-I (14.8) (13.8) 3.3 ( 2.5 to 9.1) 0.27 ERRNI-R (20.9) (23.2) 0.58 ( 8.7 to 9.9) 0.90 ERRNI-C (14.5) (15.5) 1.4 ( 4.7 to 7.5) 0.64 n (%) improved n (%) improved Odds ratio (95% CI) TOPICC (43.1) 27 5 (18.5) 0.3 ( ) 0.04 PRO-LS (68.4) 16 7 (43.8) 2.9 ( ) 0.09 PRO-SC (73.7) 16 4 (25.0) 8.0 ( ) PRO-SS (63.2) 15 3 (20.0) 7.4 ( ) PRO-PR (63.2) 14 7 (50.0) 1.6 ( ) 0.46 TRO-CLS (75.0) 18 8 (44.4) 3.7 ( ) 0.03 Notes: a Mean difference by linear regression or odds ratio by logistic regression. p < 0.05; p < Abbreviations are as given in tables 1 and 2, except TRO-CLS, Teacher Reported Outcome Classroom Learning Skills.

9 RCT of speech/language therapy for children with pragmatic language impairment 241 required to understand the true nature and occurrence of underlying language impairments as well as development of sensitive, valid and objective measures of high level language outcomes. The content of CELF-4 CLSS was also ultimately relatively far removed from the intervention goals in SCIP which emerged for individuals over time. These tended to be at the levels of activity and participation, reflecting the priorities of parents and teachers whose input was solicited throughout the setting of goals and intervention period. The second aim was to explore any notable effects of the SCIP intervention compared with TAU, on observed functional pragmatic ability and broader social communication. Significant intervention effects were found for SCIP intervention compared with TAU for overall conversational quality (TOPICC) between baseline and 6- month follow-up. This finding is important given that it was achieved using blind rater perceptions. It is carefully noted, however, that approximately half of the intervention group were rated as not improved on this measure, indicating that only some CwPLI are likely to be able to adapt conversational style with a short period of intervention. It would be important to identify, in further research, variables associated with potential for change in order to adopt differential management strategies. TOPICC ratings were based on blind perceptions of change between baseline and 6- month follow-up suggesting the intervention effect is maintained well beyond the end of intensive therapy for these children who did show change. Significant intervention effects were also found for SCIP intervention compared with TAU for non-blind parent-reported social communication, social behaviour and language skills immediately following intervention. These effects were maintained at 6-month follow-up for social communication and behaviour in social situations. Significant intervention effects were also found for nonblind teacher ratings of classroom learning skills at 6- month follow-up (measured at T3 only). Non-blind parent-reported pragmatic functioning (CCC-PRAG) showed a significant intervention effect compared with TAU at 6-month follow-up, but not immediately following intervention. It might therefore be that skills learned in an intensive period of therapy take time to be consolidated in broader contexts, which would contribute to a possible sleeper effect where the effects of treatment may be undetectable on immediate post-test but become apparent on later testing. CCC- PRAG was composed of a diverse list of pragmatic skills, not all of which could have been addressed in any single individual s intervention. It was not unexpected therefore that this finding just achieved significance. It is important to note that between-condition effects on non-blind parent and teacher-reported measures were subject to bias and therefore need to be interpreted with great caution. In this study we aimed to mitigate bias by extracting two sets of items, CCC-PRAG (behaviours targeted in the intervention) and CCC-AUT (not targeted in the intervention) from CCC-2 assessments. The hypothesis that intervention which targeted pragmatics should show an effect on CCC-PRAG only was confirmed. Parent report was able to differentiate effects for these two aspects of communication. Differential effects across categories were also demonstrated within Parent Reported Outcomes, where findings were not universally in favour of intervention across individual items. Although careful attention was given to the method of reporting to attempt to control halo effects, it is possible that parents in the intervention group were biased to report positive changes in aspects of intervention which they perceived as central to therapy, but, in practice, intervention contained multiple integrated components and these were not explicitly labelled by therapists. Further, exploration of outcome measures indicates that there is convergence in the types of functional communication changes observed by parents across measures (CCC- PRAG/PRO) and good agreement between parent CCC raters and blind TOPICC raters (Adams et al. 2011). Delivering a complex individualized speech language intervention to a relatively heterogeneous population, in the context of all the variables of reallife school environments was exceptionally challenging and there are several potential limitations and possibilities for bias which have to be controlled. The TAU group received less direct specialist intervention than participants receiving SCIP (Adams et al. 2012) making it difficult to attribute intervention effects to the specific content of SCIP rather than increased specialist attention. However, the TAU group continued to receive SLT support via LSAs in a training/consultancy model which is widely used in UK mainstream SLT services. Thus, this was a fair comparison of the experimental treatment to the care that would have been received were the trial not to have taken place and therefore has ecological validity. In this study, the developer of the SCIP intervention and the TOPICC outcome measure is also the evaluator of the intervention in this trial. As such, a potential for systematic bias related to conflict of interest may exist (Petrosino and Soydan 2005). However, the trial was rigorous with respect to blinding and pre-definition of outcome measures. These findings highlight important implications for future studies of interventions for complex developmental constructs such as PLI and SCD. This complexity means that it is unlikely that there will be uniform

10 242 Catherine Adams et al. outcome effects across participants due to differential profiles of impairment and rates of development (Koenig et al. 2009). Obtaining a balance in outcome measures between sensitivity to change in performance increments (where potential measureable outcomes are many) and meaningful functional communication change is a real challenge in intervention trials such as these. The TOPICC method of using blind rater perception of overall conversational quality holds promise as a way of capturing change across a complex population presenting with wide ranging impairments that impact on the quality of conversation in different ways. However, there is a need to develop measures which are specific and sensitive to the perceived needs of the children from the service users perspective too. Therefore, parent or teacher-reported outcomes may ultimately be a more powerful primary outcome measure in a study such as this. Conclusions and clinical implications The overall conclusions are that it is likely that the intervention provided in SCIP is effective at improving overall conversational quality (but not structural language skills) in 6 11 year olds who have significant pragmatic and social communication needs compared with TAU. SCIP is perceived by parents and teachers as effective at improving some functional pragmatic and social communication skills at home, and classroom learning skills, for these children. The implications are that, with carefully targeted specialist intervention, there is potential for some change in some school-aged children who have persistent pragmatic and social communication needs, even with a brief period of speech and language therapy. The amount of therapy offered was constrained by the experimental model; in reality, some children may require longer periods of intervention to consolidate gains made in the intensive period of therapy. Changes in individual children varied widely and a further stage of enquiry would be to investigate factors which contribute to this finding. These findings may provide some support for the conclusions of a recently reported trial of intervention for children with SLI (McCartney et al. 2011), which concluded that the effects of a specialist intervention can be lost whilst attempting to embed it within a consultancy training model. In the present study s TAU condition, children received less direct intervention but continued to receive SLT support from LSAs via a consultancy model. Many children had received this support for a long period of time and continued to do so. In the SCIP condition, direct therapy was intensively provided using a model of specialist SLT provision supported by specially trained assistants (with additional indirect support). In future work it will be important to include estimates of cost-effectiveness and the resources required to support alternative models of delivery. It would be essential to look at the balance between a more intensive, mixed direct/indirect, specialist-led provision which has the potential to produce outcomes desired by service users and the more indirect SLT models which are generally provided over longer periods and whose potential for achieving desired outcomes is unknown. Although resources prevented the use of gold standard autism diagnostic procedures such as the Autism Diagnostic Observation Schedule (ADOS; Lord et al. 2000), there is an indication that our PLI group showed a substantial overlap with children with HF- ASD/PDD and that SCIP may therefore also be a relevant intervention in addressing social communication skills in this group. We emphasize though that SCIP intervention is aimed at optimizing social communication, it does not aim to cure communication impairments or change autistic symptomatology. The findings are suggestive of changes in children s communication skills which are perceived as meaningful to those living and working with the children daily. Such indicators are now seen as central to the aspiration of delivering services which are designed around the family (Department for Children, Schools and Families (DCSF) 2008). Acknowledgements This study was funded by the Nuffield Foundation (Grant Reference: EDU/32953) and sponsored by the University of Manchester. The authors gratefully thank the participating children, parents, schools, and referring speech and language therapy services in the NorthWestofEnglandandintheSouthEastScotlandarea.They acknowledge the cooperation of the Centre for Integrated Healthcare Research, Queen Margaret University, Edinburgh; and the support and guidance from their study advisory committee (Sue Roulstone, Bonnie Brinton, Martin Fujiki and Geoff Lindsay). Declaration of interest: The authors report no conflicts of interest. The authors alone are responsible for the content and writing of the paper. The intervention described in this paper is currently in preparation for publication in book form by two of the authors (Adams and Gaile). Notes 1. The term PDD is likely to be subsumed under ASD in DSM-5; therefore, it will not be used from this point. Note that it does appear in the SCQ assessment used in this study. The proportion of children with historical ASD and PDD on this assessment will be interpreted as all falling into a broader ASD definition (see the Results). 2. SCQ Lifetime scores are based on parent report of autism features which have occurred in the child s lifetime; it is probable that the behavioural and social profile of the child in the current study will have changed to some degree over time, so no definitive diagnosis of current autism functioning can be derived. 3. Outcome data for all randomized participants were included in analysis where available.

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