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Social cognitive training for adults with Noonan syndrome: a feasibility study

PURPOSE: Noonan syndrome (NS) is a genetic disorder that is associated with social cognitive problems. While treatment aimed at the improvement of social cognition is available for other neuropsychiatric disorders, no such interventions yet exist for NS patients. In this study, the development of th...

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Autores principales: Roelofs, Renée L, Wingbermühle, Ellen, Kessels, Roy P C, Egger, Jos I M
Formato: Online Artículo Texto
Lenguaje:English
Publicado: Dove Medical Press 2019
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC6396661/
https://www.ncbi.nlm.nih.gov/pubmed/30880986
http://dx.doi.org/10.2147/NDT.S179527
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author Roelofs, Renée L
Wingbermühle, Ellen
Kessels, Roy P C
Egger, Jos I M
author_facet Roelofs, Renée L
Wingbermühle, Ellen
Kessels, Roy P C
Egger, Jos I M
author_sort Roelofs, Renée L
collection PubMed
description PURPOSE: Noonan syndrome (NS) is a genetic disorder that is associated with social cognitive problems. While treatment aimed at the improvement of social cognition is available for other neuropsychiatric disorders, no such interventions yet exist for NS patients. In this study, the development of the first social cognitive training for NS patients is described and its applicability and feasibility evaluated. METHODS: Eleven adult patients with NS participated in this controlled proof-of-principle study. Six patients were included in the treatment group and five in the control group. Neuropsychological testing was performed in both groups at baseline and posttreatment. Social cognition was a primary outcome measure and nonsocial cognition and psychopathology secondary outcome measures. Differences between pre- and posttest were investigated with Wilcoxon signed-rank tests, and a process evaluation was performed to aid interpretation of the results. RESULTS: Both groups were comparable with regard to age, estimated intelligence, and baseline performance. Although no significant differences were found between pre- and posttest scores on primary and secondary outcome measures in either group, a medium–large effect size was found on emotion recognition in the treatment group. Also, the process evaluation demonstrated the feasibility of the training. CONCLUSION: This first social cognitive training for adult patients with NS has proven to be feasible for this population and showed some encouraging results regarding emotion recognition, although the training protocol could be optimized. Further investigation is required using a randomized controlled design in a larger sample, in order to substantiate the overall effectiveness of the training.
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spelling pubmed-63966612019-03-15 Social cognitive training for adults with Noonan syndrome: a feasibility study Roelofs, Renée L Wingbermühle, Ellen Kessels, Roy P C Egger, Jos I M Neuropsychiatr Dis Treat Original Research PURPOSE: Noonan syndrome (NS) is a genetic disorder that is associated with social cognitive problems. While treatment aimed at the improvement of social cognition is available for other neuropsychiatric disorders, no such interventions yet exist for NS patients. In this study, the development of the first social cognitive training for NS patients is described and its applicability and feasibility evaluated. METHODS: Eleven adult patients with NS participated in this controlled proof-of-principle study. Six patients were included in the treatment group and five in the control group. Neuropsychological testing was performed in both groups at baseline and posttreatment. Social cognition was a primary outcome measure and nonsocial cognition and psychopathology secondary outcome measures. Differences between pre- and posttest were investigated with Wilcoxon signed-rank tests, and a process evaluation was performed to aid interpretation of the results. RESULTS: Both groups were comparable with regard to age, estimated intelligence, and baseline performance. Although no significant differences were found between pre- and posttest scores on primary and secondary outcome measures in either group, a medium–large effect size was found on emotion recognition in the treatment group. Also, the process evaluation demonstrated the feasibility of the training. CONCLUSION: This first social cognitive training for adult patients with NS has proven to be feasible for this population and showed some encouraging results regarding emotion recognition, although the training protocol could be optimized. Further investigation is required using a randomized controlled design in a larger sample, in order to substantiate the overall effectiveness of the training. Dove Medical Press 2019-02-26 /pmc/articles/PMC6396661/ /pubmed/30880986 http://dx.doi.org/10.2147/NDT.S179527 Text en © 2019 Roelofs et al. This work is published and licensed by Dove Medical Press Limited The full terms of this license are available at https://www.dovepress.com/terms.php and incorporate the Creative Commons Attribution – Non Commercial (unported, v3.0) License (http://creativecommons.org/licenses/by-nc/3.0/). By accessing the work you hereby accept the Terms. Non-commercial uses of the work are permitted without any further permission from Dove Medical Press Limited, provided the work is properly attributed.
spellingShingle Original Research
Roelofs, Renée L
Wingbermühle, Ellen
Kessels, Roy P C
Egger, Jos I M
Social cognitive training for adults with Noonan syndrome: a feasibility study
title Social cognitive training for adults with Noonan syndrome: a feasibility study
title_full Social cognitive training for adults with Noonan syndrome: a feasibility study
title_fullStr Social cognitive training for adults with Noonan syndrome: a feasibility study
title_full_unstemmed Social cognitive training for adults with Noonan syndrome: a feasibility study
title_short Social cognitive training for adults with Noonan syndrome: a feasibility study
title_sort social cognitive training for adults with noonan syndrome: a feasibility study
topic Original Research
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC6396661/
https://www.ncbi.nlm.nih.gov/pubmed/30880986
http://dx.doi.org/10.2147/NDT.S179527
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