Natural clusters of tuberous sclerosis complex (TSC)-associated neuropsychiatric disorders (TAND): new findings from the TOSCA TAND research project

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dc.contributor.authorde Vries, Petrus J
dc.contributor.authorBelousova, Elena
dc.contributor.authorBenedik, Mirjana P
dc.contributor.authorCarter, Tom
dc.contributor.authorCottin, Vincent
dc.contributor.authorCuratolo, Paolo
dc.contributor.authorD’Amato, Lisa
dc.contributor.authorBeure d’Augères, Guillaume
dc.contributor.authorFerreira, José C
dc.contributor.authorFeucht, Martha
dc.contributor.authorFladrowski, Carla
dc.contributor.authorHertzberg, Christoph
dc.contributor.authorJozwiak, Sergiusz
dc.contributor.authorLawson, John A
dc.contributor.authorMacaya, Alfons
dc.contributor.authorMarques, Ruben
dc.contributor.authorNabbout, Rima
dc.contributor.authorO’Callaghan, Finbar
dc.contributor.authorQin, Jiong
dc.contributor.authorSander, Valentin
dc.contributor.authorSauter, Matthias
dc.contributor.authorShah, Seema
dc.contributor.authorTakahashi, Yukitoshi
dc.contributor.authorTouraine, Renaud
dc.contributor.authorYouroukos, Sotiris
dc.contributor.authorZonnenberg, Bernard
dc.contributor.authorKingswood, J. Chris
dc.contributor.authorJansen, Anna C
dc.date.accessioned2021-04-01T06:54:19Z
dc.date.available2021-04-01T06:54:19Z
dc.date.issued2020-09-01
dc.date.updated2020-09-06T04:04:32Z
dc.description.abstractAbstract Background Tuberous sclerosis complex (TSC)-associated neuropsychiatric disorders (TAND) have unique, individual patterns that pose significant challenges for diagnosis, psycho-education, and intervention planning. A recent study suggested that it may be feasible to use TAND Checklist data and data-driven methods to generate natural TAND clusters. However, the study had a small sample size and data from only two countries. Here, we investigated the replicability of identifying natural TAND clusters from a larger and more diverse sample from the TOSCA study. Methods As part of the TOSCA international TSC registry study, this embedded research project collected TAND Checklist data from individuals with TSC. Correlation coefficients were calculated for TAND variables to generate a correlation matrix. Hierarchical cluster and factor analysis methods were used for data reduction and identification of natural TAND clusters. Results A total of 85 individuals with TSC (female:male, 40:45) from 7 countries were enrolled. Cluster analysis grouped the TAND variables into 6 clusters: a scholastic cluster (reading, writing, spelling, mathematics, visuo-spatial difficulties, disorientation), a hyperactive/impulsive cluster (hyperactivity, impulsivity, self-injurious behavior), a mood/anxiety cluster (anxiety, depressed mood, sleep difficulties, shyness), a neuropsychological cluster (attention/concentration difficulties, memory, attention, dual/multi-tasking, executive skills deficits), a dysregulated behavior cluster (mood swings, aggressive outbursts, temper tantrums), and an autism spectrum disorder (ASD)-like cluster (delayed language, poor eye contact, repetitive behaviors, unusual use of language, inflexibility, difficulties associated with eating). The natural clusters mapped reasonably well onto the six-factor solution generated. Comparison between cluster and factor solutions from this study and the earlier feasibility study showed significant similarity, particularly in cluster solutions. Conclusions Results from this TOSCA research project in an independent international data set showed that the combination of cluster analysis and factor analysis may be able to identify clinically meaningful natural TAND clusters. Findings were remarkably similar to those identified in the earlier feasibility study, supporting the potential robustness of these natural TAND clusters. Further steps should include examination of larger samples, investigation of internal consistency, and evaluation of the robustness of the proposed natural clusters.
dc.identifier.urihttps://doi.org/10.1186/s11689-020-09327-0
dc.identifier.urihttp://hdl.handle.net/11427/33209
dc.language.rfc3066en
dc.rights.holderThe Author(s)
dc.sourceJournal of Neurodevelopmental Disorders
dc.source.journalissue1
dc.source.journalvolume12
dc.source.pagination24
dc.source.urihttps://jneurodevdisorders.biomedcentral.com/
dc.subjectASD
dc.subjectCluster analysis
dc.subjectFactor analysis
dc.subjectNatural TAND clusters
dc.subjectTAND
dc.subjectTuberous sclerosis complex
dc.subjectTOSCA
dc.subjectRegistry
dc.subjectNeuropsychiatric
dc.titleNatural clusters of tuberous sclerosis complex (TSC)-associated neuropsychiatric disorders (TAND): new findings from the TOSCA TAND research project
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