Natural clusters of tuberous sclerosis complex (TSC)-associated neuropsychiatric disorders (TAND) : New findings from the TOSCA TAND research project
| Title: | Natural clusters of tuberous sclerosis complex (TSC)-associated neuropsychiatric disorders (TAND) : New findings from the TOSCA TAND research project |
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| Authors: | De Vries P. J.; Belousova E.; Benedik M. P.; Carter T.; Cottin V.; Curatolo P.; D'Amato L.; Beure D'Augeres G.; Ferreira J. C.; Feucht M.; Fladrowski C.; Hertzberg C.; Jozwiak S.; Lawson J. A.; Macaya A.; Marques R.; Nabbout R.; O'Callaghan F.; Qin J.; Sander V.; Sauter M.; Shah S.; Takahashi Y.; Touraine R.; Youroukos S.; Zonnenberg B.; Kingswood J. C.; Jansen A. C.; Shinohara N.; Horie S.; Kubota M.; Tohyama J.; Imai K.; Kaneda M.; Kaneko H.; Uchida Y.; Kirino T.; Endo S.; Inoue Y.; Uruno K.; Serdaroglu A.; Yapici Z.; Anlar B.; Altunbasak S.; Lvova O.; Belyaev O. V.; Agranovich O.; Levitina E. V.; Maksimova Y. V.; Karas A.; Jiang Y.; Zou L.; Xu K.; Zhang Y.; Luan G.; Wang Y.; Jin M.; Ye D.; Liao W.; Zhou L.; Liu J.; Liao J.; Yan B.; Deng Y.; Jiang L.; Liu Z.; Huang S.; Li H.; Kim K.; Chen P. -L.; Lee H. -F.; Tsai J. -D.; Chi C. -S.; Huang C. -C.; Riney K.; Yates D.; Kwan P.; Likasitwattanakul S.; Nabangchang C.; Chomtho L. T. K.; Katanyuwong K.; Sriudomkajorn S.; Wilmshurst J.; Segel R.; Gilboa T.; Tzadok M.; Fattal-Valevski A.; Papathanasopoulos P.; Papavasiliou A. S.; Giannakodimos S.; Gatzonis S.; Pavlou E.; Tzoufi M.; Vergeer A. M. H.; Dhooghe M.; Verhelst H.; Roelens F.; Nassogne M. C.; Defresne P.; De Waele L.; Leroy P.; Demonceau N.; Legros B.; Van Bogaert P.; Ceulemans B.; Dom L.; Castelnau P.; Martin A. D. S.; Riquet A.; Milh M.; Cances C.; Pedespan J. -M.; Ville D.; Roubertie A.; Auvin S.; Berquin P.; Richelme C.; Allaire C.; Gueden S.; Tich S. N. T.; Godet B.; Rojas M. L. R. F.; Planas J. C.; Bermejo A. M.; Dura P. S.; Aparicio S. R.; Gonzalez M. J. M.; Pison J. L.; Barca M. O. B.; Laso E. L.; Luengo O. A.; Rodriguez F. J. A.; Dieguez I. M.; Salas A. C.; Carrera I. M.; Salcedo E. M.; Petri M. E. Y.; Candela R. C.; Da Conceicao Carrilho I.; Vieira J. P.; Da Silva Oliveira Monteiro J. P.; De Oliveira Ferreira Leao M. J. S.; Luis C. S. M. R.; Mendonca C. P.; Endziniene M.; Strautmanis J.; Talvik I.; Canevini M. P.; Gambardella A.; Pruna D.; Buono S.; Fontana E.; Bernardina B. D.; Burloiu C.; Cosma I. S. B.; Vintan M. A.; Popescu L.; Zitterbart K.; Payerova J.; Bratsky L.; Zilinska Z.; Gruber-Sedlmayr U.; Baumann M.; Haberlandt E.; Rostasy K.; Pataraia E.; Elmslie F.; Johnston C. A.; Crawford P.; Uldall P.; Dahlin M.; Uvebrant P.; Rask O.; Bjoernvold M.; Brodtkorb E.; Sloerdahl A.; Solhoff R.; Jaatun M. S. G.; Mandera M.; Radzikowska E. J.; Wysocki M.; Fischereder M.; Kurlemann G.; Wilken B.; Wiemer-Kruel A.; Budde K.; Marquard K.; Knuf M.; Hahn A.; Hartmann H.; Merkenschlager A.; Trollmann R. |
| Contributors: | P.J. De Vrie; E. Belousova; M.P. Benedik; T. Carter; V. Cottin; P. Curatolo; L. D'Amato; G. Beure D'Augere; J.C. Ferreira; M. Feucht; C. Fladrowski; C. Hertzberg; S. Jozwiak; J.A. Lawson; A. Macaya; R. Marque; R. Nabbout; F. O'Callaghan; J. Qin; V. Sander; M. Sauter; S. Shah; Y. Takahashi; R. Touraine; S. Yourouko; B. Zonnenberg; J.C. Kingswood; A.C. Jansen; N. Shinohara; S. Horie; M. Kubota; J. Tohyama; K. Imai; M. Kaneda; H. Kaneko; Y. Uchida; T. Kirino; S. Endo; Y. Inoue; K. Uruno; A. Serdaroglu; Z. Yapici; B. Anlar; S. Altunbasak; O. Lvova; O.V. Belyaev; O. Agranovich; E.V. Levitina; Y.V. Maksimova; A. Kara; Y. Jiang; L. Zou; K. Xu; Y. Zhang; G. Luan; Y. Wang; M. Jin; D. Ye; W. Liao; L. Zhou; J. Liu; J. Liao; B. Yan; Y. Deng; L. Jiang; Z. Liu; S. Huang; H. Li; K. Kim; P.-. Chen; H.-. Lee; J.-. Tsai; C.-. Chi; C.-. Huang; K. Riney; D. Yate; P. Kwan; S. Likasitwattanakul; C. Nabangchang; L.T.K. Chomtho; K. Katanyuwong; S. Sriudomkajorn; J. Wilmshurst; R. Segel; T. Gilboa; M. Tzadok; A. Fattal-Valevski; P. Papathanasopoulo; A.S. Papavasiliou; S. Giannakodimo; S. Gatzoni; E. Pavlou; M. Tzoufi; A.M.H. Vergeer; M. Dhooghe; H. Verhelst; F. Roelen; M.C. Nassogne; P. Defresne |
| Publisher Information: | BioMed Central |
| Publication Year: | 2020 |
| Collection: | The University of Milan: Archivio Istituzionale della Ricerca (AIR) |
| Subject Terms: | ASD; Cluster analysi; Factor analysi; Natural TAND cluster; Neuropsychiatric; Registry; TAND; TOSCA; Tuberous sclerosis complex; Settore MED/39 - Neuropsichiatria Infantile |
| Description: | 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 ... |
| Document Type: | article in journal/newspaper |
| Language: | English |
| Relation: | info:eu-repo/semantics/altIdentifier/pmid/32873244; info:eu-repo/semantics/altIdentifier/wos/WOS:000566155000001; volume:12; issue:1; firstpage:1; lastpage:13; numberofpages:13; journal:JOURNAL OF NEURODEVELOPMENTAL DISORDERS; https://hdl.handle.net/2434/784997 |
| DOI: | 10.1186/s11689-020-09327-0 |
| Availability: | https://hdl.handle.net/2434/784997; https://doi.org/10.1186/s11689-020-09327-0 |
| Rights: | info:eu-repo/semantics/openAccess |
| Accession Number: | edsbas.F7D25C37 |
| Database: | BASE |