Symptom rates and profile clustering in tuberous sclerosis complex-associated neuropsychiatric disorders (TAND) / Samuel ALPERIN in Journal of Neurodevelopmental Disorders, 13 (2021)  

Public ISBD [article]  inJournal of Neurodevelopmental Disorders > 13 (2021) Titre : Symptom rates and profile clustering in tuberous sclerosis complex-associated neuropsychiatric disorders (TAND) Type de document : texte imprimé Auteurs : Samuel ALPERIN, Auteur ; Darcy A. KRUEGER, Auteur ; David N. FRANZ, Auteur ; Karen D. AGRICOLA, Auteur ; Gabrielle STIRES, Auteur ; Paul S. HORN, Auteur ; Jamie K. CAPAL, Auteur Langues : Anglais (eng) Mots-clés : Adult  Checklist  Child  Cluster Analysis  Cohort Studies  Humans  Mental Disorders/complications/epidemiology  Tuberous Sclerosis/complications/psychology  Intellectual disability  TSC-associated neuropsychiatric disorders (TAND)  Tuberous sclerosis complex  disclosures. Darcy Krueger reports consulting fees and/or research support from  Novartis Pharmaceuticals, TS Alliance, Druggability Technologies, Greenwich  Biosciences, and Marinus Pharmaceuticals. Dr. Krueger also has received speaking  fees from Novartis Pharmaceuticals and Greenwich Biosciences. David N. Franz  reports travel support and honoraria from Novartis (more than 2 years ago). His  employer has also received support for clinical trials and consulting work from  Novartis (more than 2 years ago). Jamie K. Capal receives research support from  Roche. Index. décimale : PER Périodiques Résumé : BACKGROUND: Tuberous Sclerosis Complex (TSC) is associated with a range of neuropsychiatric difficulties, appropriately termed TSC-Associated Neuropsychiatric Disorders (TAND). The objectives of the study were to analyze the rates of TAND symptoms in a cohort of patients seen at the TSC Center of Excellence at Cincinnati Children's Hospital and to identify clinically meaningful profiles based on TAND symptoms. METHODS: Data from the TAND Checklist was obtained from participants seen at the TSC Center of Excellence at Cincinnati Children's Hospital Medical Center from June 2015 to August 2018. Cluster and factor analyses for each TAND symptom were performed. Factor scores were then calculated for participants, and a K-means cluster analysis of these scores was used to empirically identify distinct overall TAND symptom profiles occurring in TSC. RESULTS: A total of 1545 checklists was completed for 668 participants (37% adults and 63% children). Approximately 90% of participants reported at least one TAND symptom with an average of 12 symptoms (out of 29). Symptom rates ranged between 5 and 60%. The most common symptoms were neuropsychologic symptoms. A seven-cluster and seven-factor solution were found to be optimal. K-means cluster analysis resulted in a seven-profile solution, ranging from low to high symptom burden. CONCLUSION: This study is the first to identify natural phenotypic profiles of TAND symptoms. Study of specific TAND subpopulations with shared profiles may facilitate better understanding of the underlying biology of TAND and better assessment of more targeted treatments. En ligne : https://dx.doi.org/10.1186/s11689-021-09408-8 Permalink : https://www.cra-rhone-alpes.org/cid/opac_css/index.php?lvl=notice_display&id=574 [article] Symptom rates and profile clustering in tuberous sclerosis complex-associated neuropsychiatric disorders (TAND) [texte imprimé] / Samuel ALPERIN, Auteur ; Darcy A. KRUEGER, Auteur ; David N. FRANZ, Auteur ; Karen D. AGRICOLA, Auteur ; Gabrielle STIRES, Auteur ; Paul S. HORN, Auteur ; Jamie K. CAPAL, Auteur. Langues : Anglais (eng) in Journal of Neurodevelopmental Disorders > 13 (2021) Mots-clés : Adult  Checklist  Child  Cluster Analysis  Cohort Studies  Humans  Mental Disorders/complications/epidemiology  Tuberous Sclerosis/complications/psychology  Intellectual disability  TSC-associated neuropsychiatric disorders (TAND)  Tuberous sclerosis complex  disclosures. Darcy Krueger reports consulting fees and/or research support from  Novartis Pharmaceuticals, TS Alliance, Druggability Technologies, Greenwich  Biosciences, and Marinus Pharmaceuticals. Dr. Krueger also has received speaking  fees from Novartis Pharmaceuticals and Greenwich Biosciences. David N. Franz  reports travel support and honoraria from Novartis (more than 2 years ago). His  employer has also received support for clinical trials and consulting work from  Novartis (more than 2 years ago). Jamie K. Capal receives research support from  Roche. Index. décimale : PER Périodiques Résumé : BACKGROUND: Tuberous Sclerosis Complex (TSC) is associated with a range of neuropsychiatric difficulties, appropriately termed TSC-Associated Neuropsychiatric Disorders (TAND). The objectives of the study were to analyze the rates of TAND symptoms in a cohort of patients seen at the TSC Center of Excellence at Cincinnati Children's Hospital and to identify clinically meaningful profiles based on TAND symptoms. METHODS: Data from the TAND Checklist was obtained from participants seen at the TSC Center of Excellence at Cincinnati Children's Hospital Medical Center from June 2015 to August 2018. Cluster and factor analyses for each TAND symptom were performed. Factor scores were then calculated for participants, and a K-means cluster analysis of these scores was used to empirically identify distinct overall TAND symptom profiles occurring in TSC. RESULTS: A total of 1545 checklists was completed for 668 participants (37% adults and 63% children). Approximately 90% of participants reported at least one TAND symptom with an average of 12 symptoms (out of 29). Symptom rates ranged between 5 and 60%. The most common symptoms were neuropsychologic symptoms. A seven-cluster and seven-factor solution were found to be optimal. K-means cluster analysis resulted in a seven-profile solution, ranging from low to high symptom burden. CONCLUSION: This study is the first to identify natural phenotypic profiles of TAND symptoms. Study of specific TAND subpopulations with shared profiles may facilitate better understanding of the underlying biology of TAND and better assessment of more targeted treatments. En ligne : https://dx.doi.org/10.1186/s11689-021-09408-8 Permalink : https://www.cra-rhone-alpes.org/cid/opac_css/index.php?lvl=notice_display&id=574

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