The research landscape of tuberous sclerosis complex–associated neuropsychiatric disorders (TAND)—a comprehensive scoping review

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dc.contributor.authorVanclooster, Stephanie
dc.contributor.authorBissell, Stacey
dc.contributor.authorvan Eeghen, Agnies M
dc.contributor.authorChambers, Nola
dc.contributor.authorDe Waele, Liesbeth
dc.contributor.authorByars, Anna W
dc.contributor.authorCapal, Jamie K
dc.contributor.authorCukier, Sebastián
dc.contributor.authorDavis, Peter
dc.contributor.authorFlinn, Jennifer
dc.contributor.authorGardner-Lubbe, Sugnet
dc.contributor.authorGipson, Tanjala
dc.contributor.authorHeunis, Tosca-Marie
dc.contributor.authorHook, Dena
dc.contributor.authorKingswood, J C
dc.contributor.authorKrueger, Darcy A
dc.contributor.authorKumm, Aubrey J
dc.contributor.authorSahin, Mustafa
dc.contributor.authorSchoeters, Eva
dc.contributor.authorSmith, Catherine
dc.contributor.authorSrivastava, Shoba
dc.contributor.authorTakei, Megumi
dc.contributor.authorWaltereit, Robert
dc.contributor.authorJansen, Anna C
dc.contributor.authorde Vries, Petrus J
dc.date.accessioned2022-04-09T07:06:18Z
dc.date.available2022-04-09T07:06:18Z
dc.date.issued2022-02-13
dc.date.updated2022-02-14T07:35:11Z
dc.description.abstractBackground Tuberous sclerosis complex (TSC)–associated neuropsychiatric disorders (TAND) is an umbrella term for the behavioural, psychiatric, intellectual, academic, neuropsychological and psychosocial manifestations of TSC. Although TAND affects 90% of individuals with TSC during their lifetime, these manifestations are relatively under-assessed, under-treated and under-researched. We performed a comprehensive scoping review of all TAND research to date (a) to describe the existing TAND research landscape and (b) to identify knowledge gaps to guide future TAND research. Methods The study was conducted in accordance with stages outlined within the Arksey and O’Malley scoping review framework. Ten research questions relating to study characteristics, research design and research content of TAND levels and clusters were examined. Results Of the 2841 returned searches, 230 articles published between 1987 and 2020 were included (animal studies = 30, case studies = 47, cohort studies = 153), with more than half published since the term TAND was coined in 2012 (118/230; 51%). Cohort studies largely involved children and/or adolescents (63%) as opposed to older adults (16%). Studies were represented across 341 individual research sites from 45 countries, the majority from the USA (89/341; 26%) and the UK (50/341; 15%). Only 48 research sites (14%) were within low–middle income countries (LMICs). Animal studies and case studies were of relatively high/high quality, but cohort studies showed significant variability. Of the 153 cohort studies, only 16 (10%) included interventions. None of these were non-pharmacological, and only 13 employed remote methodologies (e.g. telephone interviews, online surveys). Of all TAND clusters, the autism spectrum disorder–like cluster was the most widely researched (138/230; 60%) and the scholastic cluster the least (53/200; 27%). Conclusions Despite the recent increase in TAND research, studies that represent participants across the lifespan, LMIC research sites and non-pharmacological interventions were identified as future priorities. The quality of cohort studies requires improvement, to which the use of standardised direct behavioural assessments may contribute. In human studies, the academic level in particular warrants further investigation. Remote technologies could help to address many of the TAND knowledge gaps identified.en_US
dc.identifier.apacitationVanclooster, S., Bissell, S., van Eeghen, A. M., Chambers, N., De Waele, L., Byars, A. W., ... de Vries, P. J. (2022). The research landscape of tuberous sclerosis complex–associated neuropsychiatric disorders (TAND)—a comprehensive scoping review. <i>Journal of Neurodevelopmental Disorders</i>, 14(1), 13. http://hdl.handle.net/11427/36308en_ZA
dc.identifier.chicagocitationVanclooster, Stephanie, Stacey Bissell, Agnies M van Eeghen, Nola Chambers, Liesbeth De Waele, Anna W Byars, Jamie K Capal, et al "The research landscape of tuberous sclerosis complex–associated neuropsychiatric disorders (TAND)—a comprehensive scoping review." <i>Journal of Neurodevelopmental Disorders</i> 14, 1. (2022): 13. http://hdl.handle.net/11427/36308en_ZA
dc.identifier.citationVanclooster, S., Bissell, S., van Eeghen, A.M., Chambers, N., De Waele, L., Byars, A.W., Capal, J.K. & Cukier, S. et al. 2022. The research landscape of tuberous sclerosis complex–associated neuropsychiatric disorders (TAND)—a comprehensive scoping review. <i>Journal of Neurodevelopmental Disorders.</i> 14(1):13. http://hdl.handle.net/11427/36308en_ZA
dc.identifier.ris TY - Journal Article AU - Vanclooster, Stephanie AU - Bissell, Stacey AU - van Eeghen, Agnies M AU - Chambers, Nola AU - De Waele, Liesbeth AU - Byars, Anna W AU - Capal, Jamie K AU - Cukier, Sebastián AU - Davis, Peter AU - Flinn, Jennifer AU - Gardner-Lubbe, Sugnet AU - Gipson, Tanjala AU - Heunis, Tosca-Marie AU - Hook, Dena AU - Kingswood, J C AU - Krueger, Darcy A AU - Kumm, Aubrey J AU - Sahin, Mustafa AU - Schoeters, Eva AU - Smith, Catherine AU - Srivastava, Shoba AU - Takei, Megumi AU - Waltereit, Robert AU - Jansen, Anna C AU - de Vries, Petrus J AB - Background Tuberous sclerosis complex (TSC)–associated neuropsychiatric disorders (TAND) is an umbrella term for the behavioural, psychiatric, intellectual, academic, neuropsychological and psychosocial manifestations of TSC. Although TAND affects 90% of individuals with TSC during their lifetime, these manifestations are relatively under-assessed, under-treated and under-researched. We performed a comprehensive scoping review of all TAND research to date (a) to describe the existing TAND research landscape and (b) to identify knowledge gaps to guide future TAND research. Methods The study was conducted in accordance with stages outlined within the Arksey and O’Malley scoping review framework. Ten research questions relating to study characteristics, research design and research content of TAND levels and clusters were examined. Results Of the 2841 returned searches, 230 articles published between 1987 and 2020 were included (animal studies = 30, case studies = 47, cohort studies = 153), with more than half published since the term TAND was coined in 2012 (118/230; 51%). Cohort studies largely involved children and/or adolescents (63%) as opposed to older adults (16%). Studies were represented across 341 individual research sites from 45 countries, the majority from the USA (89/341; 26%) and the UK (50/341; 15%). Only 48 research sites (14%) were within low–middle income countries (LMICs). Animal studies and case studies were of relatively high/high quality, but cohort studies showed significant variability. Of the 153 cohort studies, only 16 (10%) included interventions. None of these were non-pharmacological, and only 13 employed remote methodologies (e.g. telephone interviews, online surveys). Of all TAND clusters, the autism spectrum disorder–like cluster was the most widely researched (138/230; 60%) and the scholastic cluster the least (53/200; 27%). Conclusions Despite the recent increase in TAND research, studies that represent participants across the lifespan, LMIC research sites and non-pharmacological interventions were identified as future priorities. The quality of cohort studies requires improvement, to which the use of standardised direct behavioural assessments may contribute. In human studies, the academic level in particular warrants further investigation. Remote technologies could help to address many of the TAND knowledge gaps identified. DA - 2022-02-13 DB - OpenUCT DP - University of Cape Town IS - 1 J1 - Journal of Neurodevelopmental Disorders KW - Tuberous sclerosis complex KW - Scoping review KW - TSC-associated neuropsychiatric disorders KW - Autism KW - Behaviour KW - Psychiatric KW - Intellectual KW - Scholastic KW - Neuropsychological KW - Psychosocial LK - https://open.uct.ac.za PY - 2022 T1 - The research landscape of tuberous sclerosis complex–associated neuropsychiatric disorders (TAND)—a comprehensive scoping review TI - The research landscape of tuberous sclerosis complex–associated neuropsychiatric disorders (TAND)—a comprehensive scoping review UR - http://hdl.handle.net/11427/36308 ER - en_ZA
dc.identifier.urihttps://doi.org/10.1186/s11689-022-09423-3
dc.identifier.urihttp://hdl.handle.net/11427/36308
dc.identifier.vancouvercitationVanclooster S, Bissell S, van Eeghen AM, Chambers N, De Waele L, Byars AW, et al. The research landscape of tuberous sclerosis complex–associated neuropsychiatric disorders (TAND)—a comprehensive scoping review. Journal of Neurodevelopmental Disorders. 2022;14(1):13. http://hdl.handle.net/11427/36308.en_ZA
dc.language.rfc3066en
dc.publisher.departmentDivision of Child and Adolescent Psychiatryen_US
dc.publisher.facultyFaculty of Health Sciencesen_US
dc.rights.holderThe Author(s)
dc.rights.urihttp://creativecommons.org/licenses/by/4.0/en_US
dc.sourceJournal of Neurodevelopmental Disordersen_US
dc.source.journalissue1en_US
dc.source.journalvolume14en_US
dc.source.pagination13en_US
dc.source.urihttps://jneurodevdisorders.biomedcentral.com/
dc.subjectTuberous sclerosis complexen_US
dc.subjectScoping reviewen_US
dc.subjectTSC-associated neuropsychiatric disordersen_US
dc.subjectAutismen_US
dc.subjectBehaviouren_US
dc.subjectPsychiatricen_US
dc.subjectIntellectualen_US
dc.subjectScholasticen_US
dc.subjectNeuropsychologicalen_US
dc.subjectPsychosocialen_US
dc.titleThe research landscape of tuberous sclerosis complex–associated neuropsychiatric disorders (TAND)—a comprehensive scoping reviewen_US
dc.typeJournal Articleen_US
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