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. 2023 Oct:147:101-123.
doi: 10.1016/j.pediatrneurol.2023.07.001. Epub 2023 Jul 7.

Development and Feasibility of the Self-Report Quantified Tuberous Sclerosis Complex-Associated Neuropsychiatric Disorders Checklist (TAND-SQ)

Tosca-Marie Heunis  1 , Nola Chambers  2 , Stephanie Vanclooster  1 , Stacey Bissell  3 , Anna W Byars  4 , Jamie K Capal  5 , Sebastián Cukier  6 , Peter E Davis  7 , Magdalena C de Vries  2 , Liesbeth De Waele  8 , Jennifer Flinn  9 , Sugnet Gardner-Lubbe  10 , Tanjala Gipson  11 , J Christopher Kingswood  12 , Darcy A Krueger  13 , Aubrey J Kumm  2 , Mustafa Sahin  14 , Eva Schoeters  15 , Catherine Smith  16 , Shoba Srivastava  17 , Megumi Takei  18 , Agnies M van Eeghen  19 , Robert Waltereit  20 , Anna C Jansen  21 , Petrus J de Vries  22
Affiliations

Development and Feasibility of the Self-Report Quantified Tuberous Sclerosis Complex-Associated Neuropsychiatric Disorders Checklist (TAND-SQ)

Tosca-Marie Heunis et al. Pediatr Neurol. 2023 Oct.

Abstract

Background: Tuberous sclerosis complex-associated neuropsychiatric disorders (TAND) are often present but underidentified and undertreated in individuals with tuberous sclerosis complex (TSC). The clinician-completed TAND-Lifetime Checklist (TAND-L) was developed to address this identification and treatment gap. Stakeholder engagement identified the need for a TAND Checklist that can (1) be completed by caregivers or individuals with TSC and (2) quantify TAND difficulties. The aim of this study was to develop a self-report quantified TAND Checklist (TAND-SQ) and conduct feasibility and acceptability testing.

Methods: This aim was addressed in three phases: (1) development of the TAND-SQ Checklist, (2) feasibility and acceptability testing of the "near-final" TAND-SQ Checklist, and (3) preparation of the final TAND-SQ Checklist. Participants included 23 technical experts from the TAND consortium in all phases and 58 lived experts (caregivers and individuals with TSC) in phase 2. All participants completed a TAND-SQ Checklist and a checklist feedback form.

Results: Phase 1 additions to the TAND-SQ, when compared with the TAND-L, included four new items and a quantification rating. Phase 2 showed high ratings for the "near-final" TAND-SQ Checklist on comprehensiveness, clarity, ease of use, and overall acceptability. In phase 3, questions on strengths, strategies, and a TAND Cluster Profile were added.

Conclusion: The TAND-SQ Checklist is presented here for use by individuals with TSC and their caregivers. The next steps as part of the TANDem project include internal and external validation of the checklist and linking of TAND Cluster Profiles generated from the checklist to evidence-informed consensus recommendations within a smartphone application.

Keywords: Acceptability; Feasibility; Self-report; Stakeholder engagement; TAND; TAND Checklist; TSC-associated neuropsychiatric disorders; Tuberous sclerosis complex.

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Figures

FIGURE 1.
FIGURE 1.
Development of the TAND-SQ Checklist.
FIGURE 2.
FIGURE 2.
Quantitative responses on the TAND-SQ Checklist feasibility and acceptability testing. (A-G) The percentage respondents per group for each item of the checklist feedback form.
FIGURE 3.
FIGURE 3.
The TAND-SQ Checklist.
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FIGURE 3.
The TAND-SQ Checklist.
FIGURE 3.
FIGURE 3.
The TAND-SQ Checklist.
FIGURE 3.
FIGURE 3.
The TAND-SQ Checklist.
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FIGURE 3.
The TAND-SQ Checklist.
FIGURE 3.
FIGURE 3.
The TAND-SQ Checklist.
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FIGURE 3.
The TAND-SQ Checklist.
FIGURE 3.
FIGURE 3.
The TAND-SQ Checklist.
FIGURE 3.
FIGURE 3.
The TAND-SQ Checklist.
FIGURE 3.
FIGURE 3.
The TAND-SQ Checklist.
FIGURE 3.
FIGURE 3.
The TAND-SQ Checklist.
FIGURE 3.
FIGURE 3.
The TAND-SQ Checklist.
FIGURE 3.
FIGURE 3.
The TAND-SQ Checklist.
FIGURE 3.
FIGURE 3.
The TAND-SQ Checklist.
See this image and copyright information in PMC

References

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