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Natural clusters of...
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de Vries, Petrus JUniversity of Cape Town
(författare)
Natural clusters of tuberous sclerosis complex (TSC)-associated neuropsychiatric disorders (TAND) : new findings from the TOSCA TAND research project
- Artikel/kapitelEngelska2020
Förlag, utgivningsår, omfång ...
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2020-09-01
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Springer Science and Business Media LLC,2020
Nummerbeteckningar
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LIBRIS-ID:oai:lup.lub.lu.se:77ca53de-8765-4c38-a2d5-30a2e03fcdb8
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https://lup.lub.lu.se/record/77ca53de-8765-4c38-a2d5-30a2e03fcdb8URI
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https://doi.org/10.1186/s11689-020-09327-0DOI
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Språk:engelska
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Sammanfattning på:engelska
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Ämneskategori:art swepub-publicationtype
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Ämneskategori:ref swepub-contenttype
Anmärkningar
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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.
Ämnesord och genrebeteckningar
Biuppslag (personer, institutioner, konferenser, titlar ...)
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Belousova, ElenaPirogov Russian National Research Medical University (RNRMU)
(författare)
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Benedik, Mirjana PUniversity Medical Centre Ljubljana
(författare)
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Carter, TomTSA Tuberous Sclerosis Association, UK
(författare)
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Cottin, VincentLaboratoire Lyon République
(författare)
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Curatolo, PaoloTor Vergata University Hospital
(författare)
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D'Amato, LisaNovartis Farma S.p.A
(författare)
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Beure d'Augères, GuillaumeAssociation Sclérose Tubéreuse de Bourneville
(författare)
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Ferreira, José CCentro Hospitalar de Lisboa Ocidental (CHLO)
(författare)
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Feucht, MarthaUniversitatsklinik fur Kinder-und Jugendheilkunde, Vienna
(författare)
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Fladrowski, CarlaAssociazione Sclerosi Tuberosa ONLUS
(författare)
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Hertzberg, ChristophVivantes Hospital Neukölln
(författare)
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Jozwiak, SergiuszMedical University of Warsaw
(författare)
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Lawson, John ASydney Children's Hospital
(författare)
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Macaya, AlfonsVall d'Hebron University Hospital
(författare)
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Marques, RubenNovartis Farma S.p.A
(författare)
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Nabbout, RimaParis Descartes University
(författare)
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O'Callaghan, FinbarUniversity College London Hospital
(författare)
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Qin, JiongPeking University People's Hospital
(författare)
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Sander, ValentinTallinn Children's Hospital
(författare)
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Sauter, MatthiasKlinikum Kempten-Oberallgäu
(författare)
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Shah, SeemaUniversity of Hyderabad
(författare)
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Takahashi, YukitoshiNational Epilepsy Center, Shizuoka Institute of Epilepsy and Neurological Disorders
(författare)
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Touraine, RenaudFox Chase Cancer Center
(författare)
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Youroukos, SotirisAghia Sophia Children's Hospital
(författare)
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Zonnenberg, BernardHanyang University Medical Center
(författare)
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Kingswood, J ChrisUniversity of London
(författare)
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Jansen, Anna CUniversitair Ziekenhuis Brussel
(författare)
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Rask, OlofLund University,Lunds universitet,Barn- och ungdomspsykiatri,Sektion IV,Institutionen för kliniska vetenskaper, Lund,Medicinska fakulteten,Child and Adolescent Psychiatry,Section IV,Department of Clinical Sciences, Lund,Faculty of Medicine(Swepub:lu)pedi-ora
(creator_code:cre_t)
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University of Cape TownPirogov Russian National Research Medical University (RNRMU)
(creator_code:org_t)
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TOSCA Consortium and TOSCA Investigators
Sammanhörande titlar
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Ingår i:Journal of Neurodevelopmental Disorders: Springer Science and Business Media LLC12:1, s. 24-241866-19551866-1947
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de Vries, Petrus ...
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Belousova, Elena
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Benedik, Mirjana ...
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Carter, Tom
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Cottin, Vincent
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Curatolo, Paolo
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visa fler...
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D'Amato, Lisa
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Beure d'Augères, ...
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Ferreira, José C
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Feucht, Martha
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Fladrowski, Carl ...
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Hertzberg, Chris ...
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Jozwiak, Sergius ...
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Lawson, John A
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Macaya, Alfons
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Marques, Ruben
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Nabbout, Rima
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O'Callaghan, Fin ...
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Qin, Jiong
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Sander, Valentin
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Sauter, Matthias
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Shah, Seema
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Takahashi, Yukit ...
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Touraine, Renaud
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Youroukos, Sotir ...
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Zonnenberg, Bern ...
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Kingswood, J Chr ...
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Jansen, Anna C
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Rask, Olof
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