Natural clusters of tuberous sclerosis complex (TSC)-associated neuropsychiatric disorders (TAND) : new findings from the TOSCA TAND research project
(2020) In Journal of Neurodevelopmental Disorders 12(1). p.24-24- Abstract
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... (More)
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.
(Less)
- author
- contributor
- Rask, Olof LU
- author collaboration
- organization
- publishing date
- 2020-09-01
- type
- Contribution to journal
- publication status
- published
- subject
- in
- Journal of Neurodevelopmental Disorders
- volume
- 12
- issue
- 1
- pages
- 24 - 24
- publisher
- BioMed Central (BMC)
- external identifiers
-
- pmid:32873244
- scopus:85090181194
- ISSN
- 1866-1955
- DOI
- 10.1186/s11689-020-09327-0
- language
- English
- LU publication?
- yes
- id
- 77ca53de-8765-4c38-a2d5-30a2e03fcdb8
- date added to LUP
- 2020-12-25 17:10:57
- date last changed
- 2024-05-17 00:07:38
@article{77ca53de-8765-4c38-a2d5-30a2e03fcdb8,
abstract = {{<p>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.</p><p>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.</p><p>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.</p><p>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.</p>}},
author = {{de Vries, Petrus J and Belousova, Elena and Benedik, Mirjana P and Carter, Tom and Cottin, Vincent and Curatolo, Paolo and D'Amato, Lisa and Beure d'Augères, Guillaume and Ferreira, José C and Feucht, Martha and Fladrowski, Carla and Hertzberg, Christoph and Jozwiak, Sergiusz and Lawson, John A and Macaya, Alfons and Marques, Ruben and Nabbout, Rima and O'Callaghan, Finbar and Qin, Jiong and Sander, Valentin and Sauter, Matthias and Shah, Seema and Takahashi, Yukitoshi and Touraine, Renaud and Youroukos, Sotiris and Zonnenberg, Bernard and Kingswood, J Chris and Jansen, Anna C}},
issn = {{1866-1955}},
language = {{eng}},
month = {{09}},
number = {{1}},
pages = {{24--24}},
publisher = {{BioMed Central (BMC)}},
series = {{Journal of Neurodevelopmental Disorders}},
title = {{Natural clusters of tuberous sclerosis complex (TSC)-associated neuropsychiatric disorders (TAND) : new findings from the TOSCA TAND research project}},
url = {{http://dx.doi.org/10.1186/s11689-020-09327-0}},
doi = {{10.1186/s11689-020-09327-0}},
volume = {{12}},
year = {{2020}},
}