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This thesis investigates social competence and social information processing in children with neurofibromatosis type 1 (NF1). The aims of this thesis were threefold: 1) to investigate day to day social competence in children with NF1 (Paper 1), 2) to better understand facial emotion recognition abilities in children with NF1 (Paper 2) and 3) to explore face perception and attention to faces in children with NF1 (Papers 2 and 3). In relation to the first aim, the results of Paper 1 indicated that, as a group, children with NF1 displayed significantly poorer day to day social competence than their typically developing peers. Children with NF1 who displayed high levels of autistic symptomatology and socially anxious behaviour were particularly at risk of social competence problems. Pertaining to the second aim, the findings of Paper 2 suggested that children with NF1 displayed poorer recognition of threatening emotions, but not non-threatening emotions, compared to typically developing children. Children with NF1 displayed particular deficits in identifying fear, but also had difficulty identifying anger. Facial emotion recognition problems were not significantly related to the manner in which children with NF1 viewed faces or their ability to perceive and discriminate between faces, although a non-significant trend was observed whereby poorer face perception skills were associated with reduced proficiency in identifying anger. In relation to the third aim, results suggested that children with NF1 spent less time attending to faces than typically developing children both when presented in isolation (Paper 2) and when presented in the context of a static social scene (Paper 3). As a group, children with NF1 also displayed significantly poorer face perception abilities than typically developing children (Papers 2 and 3). Taken together, these findings indicate that children with NF1 are at significant risk of day to day social competence problems. Moreover, findings indicate that impairments in perceiving, interpreting, and attending to information from faces may be important aspects of the social-cognitive phenotype of NF1.

Supplemental material, sj-DOCX-1-aut-10.1177_13623613211029116 for Adapting and validating the Autism Diagnostic Interview - Revised for use with deaf children and young people by Barry Wright, Helen Phillips, Victoria Allgar, Jennifer Sweetman, Rachel Hodkinson, Emily Hayward, Amelia Ralph-Lewis, Catarina Teige, Martin Bland and Ann Le Couteur in Autism

Purpose: We developed and evaluated in a randomized controlled trial a computerized speechreading training program to determine (a) whether it is possible to train speechreading in deaf children and (b) whether speechreading training results in improvements in phonological and reading skills. Previous studies indicate a relationship between speechreading and reading skill and further suggest this relationship may be mediated by improved phonological representations. This is important since many deaf children find learning to read to be very challenging.Method: Sixty-six deaf 5- to 7-year-olds were randomized into speechreading and maths training arms. Each training program was composed of a 10-min sessions a day, 4 days a week for 12 weeks. Children were assessed on a battery of language and literacy measures before training, immediately after training, and 3 months and 11 months after training.Results: We found no significant benefits for participants who completed the speechreading training, compared to those who completed the maths training, on the speechreading primary outcome measure. However, significantly greater gains were observed in the speechreading training group on one of the secondary measures of speechreading. There was also some evidence of beneficial effects of the speechreading training on phonological representations; however, these effects were weaker. No benefits were seen to word reading.Conclusions: Speechreading skill is trainable in deaf children. However, to support early reading, training may need to be longer or embedded in a broader literacy program. Nevertheless, a training tool that can improve speechreading is likely to be of great interest to professionals working with deaf children.Supplemental Material S1. CONSORT 2010 checklist of information to include when reporting a randomised trial.Supplemental Material S2. Further details regarding training arms and assessments.Pimperton, H., Kyle, F., Hulme, C., Harris, M., Beedie, I., Ralph-Lewis, A., ... MacSweeney, M. (2019). Computerized speechreading training for deaf children: A randomized controlled trial. Journal of Speech, Language, and Hearing Research, 62, 2882–2894. https://doi.org/10.1044/2019_JSLHR-H-19-0073

Purpose: We developed and evaluated in a randomized controlled trial a computerized speechreading training program to determine (a) whether it is possible to train speechreading in deaf children and (b) whether speechreading training results in improvements in phonological and reading skills. Previous studies indicate a relationship between speechreading and reading skill and further suggest this relationship may be mediated by improved phonological representations. This is important since many deaf children find learning to read to be very challenging.Method: Sixty-six deaf 5- to 7-year-olds were randomized into speechreading and maths training arms. Each training program was composed of a 10-min sessions a day, 4 days a week for 12 weeks. Children were assessed on a battery of language and literacy measures before training, immediately after training, and 3 months and 11 months after training.Results: We found no significant benefits for participants who completed the speechreading training, compared to those who completed the maths training, on the speechreading primary outcome measure. However, significantly greater gains were observed in the speechreading training group on one of the secondary measures of speechreading. There was also some evidence of beneficial effects of the speechreading training on phonological representations; however, these effects were weaker. No benefits were seen to word reading.Conclusions: Speechreading skill is trainable in deaf children. However, to support early reading, training may need to be longer or embedded in a broader literacy program. Nevertheless, a training tool that can improve speechreading is likely to be of great interest to professionals working with deaf children.Supplemental Material S1. CONSORT 2010 checklist of information to include when reporting a randomised trial.Supplemental Material S2. Further details regarding training arms and assessments.Pimperton, H., Kyle, F., Hulme, C., Harris, M., Beedie, I., Ralph-Lewis, A., ... MacSweeney, M. (2019). Computerized speechreading training for deaf children: A randomized controlled trial. Journal of Speech, Language, and Hearing Research, 62, 2882–2894. https://doi.org/10.1044/2019_JSLHR-H-19-0073

Purpose: We developed and evaluated in a randomized controlled trial a computerized speechreading training program to determine (a) whether it is possible to train speechreading in deaf children and (b) whether speechreading training results in improvements in phonological and reading skills. Previous studies indicate a relationship between speechreading and reading skill and further suggest this relationship may be mediated by improved phonological representations. This is important since many deaf children find learning to read to be very challenging.Method: Sixty-six deaf 5- to 7-year-olds were randomized into speechreading and maths training arms. Each training program was composed of a 10-min sessions a day, 4 days a week for 12 weeks. Children were assessed on a battery of language and literacy measures before training, immediately after training, and 3 months and 11 months after training.Results: We found no significant benefits for participants who completed the speechreading training, compared to those who completed the maths training, on the speechreading primary outcome measure. However, significantly greater gains were observed in the speechreading training group on one of the secondary measures of speechreading. There was also some evidence of beneficial effects of the speechreading training on phonological representations; however, these effects were weaker. No benefits were seen to word reading.Conclusions: Speechreading skill is trainable in deaf children. However, to support early reading, training may need to be longer or embedded in a broader literacy program. Nevertheless, a training tool that can improve speechreading is likely to be of great interest to professionals working with deaf children.Supplemental Material S1. CONSORT 2010 checklist of information to include when reporting a randomised trial.Supplemental Material S2. Further details regarding training arms and assessments.Pimperton, H., Kyle, F., Hulme, C., Harris, M., Beedie, I., Ralph-Lewis, A., ... MacSweeney, M. (2019). Computerized speechreading training for deaf children: A randomized controlled trial. Journal of Speech, Language, and Hearing Research, 62, 2882-2894. https://doi.org/10.1044/2019_JSLHR-H-19-0073

Purpose: We developed and evaluated in a randomized controlled trial a computerized speechreading training program to determine (a) whether it is possible to train speechreading in deaf children and (b) whether speechreading training results in improvements in phonological and reading skills. Previous studies indicate a relationship between speechreading and reading skill and further suggest this relationship may be mediated by improved phonological representations. This is important since many deaf children find learning to read to be very challenging.Method: Sixty-six deaf 5- to 7-year-olds were randomized into speechreading and maths training arms. Each training program was composed of a 10-min sessions a day, 4 days a week for 12 weeks. Children were assessed on a battery of language and literacy measures before training, immediately after training, and 3 months and 11 months after training.Results: We found no significant benefits for participants who completed the speechreading training, compared to those who completed the maths training, on the speechreading primary outcome measure. However, significantly greater gains were observed in the speechreading training group on one of the secondary measures of speechreading. There was also some evidence of beneficial effects of the speechreading training on phonological representations; however, these effects were weaker. No benefits were seen to word reading.Conclusions: Speechreading skill is trainable in deaf children. However, to support early reading, training may need to be longer or embedded in a broader literacy program. Nevertheless, a training tool that can improve speechreading is likely to be of great interest to professionals working with deaf children.Supplemental Material S1. CONSORT 2010 checklist of information to include when reporting a randomised trial.Supplemental Material S2. Further details regarding training arms and assessments.Pimperton, H., Kyle, F., Hulme, C., Harris, M., Beedie, I., Ralph-Lewis, A., ... MacSweeney, M. (2019). Computerized speechreading training for deaf children: A randomized controlled trial. Journal of Speech, Language, and Hearing Research. Advance online publication. https://doi.org/10.1044/2019_JSLHR-H-19-0073