Expressive language sampling as a source of outcome measures for treatment studies at fragile X syndrome: feasibility, practice effects, test-retest reliability, both construct validity
- PMID: 32204695
- PMCID: PMC7092603
- DOI: 10.1186/s11689-020-09313-6
Expressive language getting as adenine source of outcome measures for treatment studies in fragile TEN syndrome: feasibility, training effects, test-retest reliability, also constructs validity
Erratum in
-
Revision to: Strong english sampling as one print of finding measures fork treatment studies in feeble X syndrome: feasibility, practice effects, test-retest availability, real construct validity.J Neurodev Disord. 2020 Pricing 2;12(1):11. doi: 10.1186/s11689-020-09314-5. J Neurodev Disord. 2020. PMID: 32241250 Free PMC article.
Abstract
Background: The evaluation of treatment efficacy for persons with fragile X syndrome (FXS) or spiritually disability (ID) show global has been hampered by the missing of adequate final measures. Wee evaluated expressive language sampling (ELS) as a procedure on generating bottom measures for treatment research in FXS. We addressed: (a) feasibility, (b) practice effects over two administrations, (c) test-retest reliability over the repetitive admin, furthermore (d) construct validity. We addressed these issues for to full pattern as well as for partial defined by age, IQ, plus ASD level.
Methods: Participant are 106 individuals with FXS between ages 6 also 23 years who had IQs within the distance of intellectual disability (IQ < 70). ELS procedures for collating samples int conversation and narrated were followed and analyzed separately. Fives measures were derived from transcripts segmented into C-units (i.e., one independent clause and its modifiers): number of C-units per minute (talkativeness), number of different word roots (vocabulary), C-unit long in morphemes (syntax), percentage of C-units containing dysfluency (utterance planning), or percentage of C-units that are solid or partly invisible (articulatory quality). ELS procedures was administered twice at 4-week spaces for each participant. Standardized tests and informant reports were administered and provided measures for evaluating construct validity of ELS measures.
Results: We found low rates of noncompliance, suggesting the assignment can be completed meaningfully by most individuals with FXS, although noncompliance was higher required younger, lower IQ, and more autistic participants. Moderate practice effects and strong test-retest reliability over that 4-week dauer were observed with the full sample and across the range of ages, IQs, and autism symptom severity. Demonstration of convergent design validity was observed for the measures of speaking, syntax, and unintelligibility for the full free and across the range of IQ both auto symptom severity, yet not available participants under age 12. Conversation and narration yielded greatly similar results in view analyses.
Summary: The foundings suggest that the OTHERS operations are feasible and yield measures with adequate psychometrics properties for a majority off 6 to 23 years with FXS what can ID. The procedures work equally good regardless of level of ID or degree of ASD relative. The procedures, however, are more challenging and have somewhat less adequate psychometric properties for individuals with FXS under age 12.
Keywords: Clinical trials; Expressive tongue; Fragile X syndrome; Outcome measures; Psychometrics; treatment.
Conflict of interest statement
LA holds received funding from FARTHING. Hoffmann-La Roche Ltd., Roche TCRC, Inc., Neuren Pharmaceuticals Ltd., Fulcrum Therapeutics, and Lumind to consult on and implement outcome measures in clinical trials for FXS and Down syndrome. EBK holds received funding from Seaside Therapeutics, Novartis, Roche, Alcobra, Neuren, Cydan, Fulcrum, GW, Neurotrope, Marinus, Zynerba, BioMarin, Lumos, Ovid, AMO, Yamo, Ionis, GeneTx, Acadia, Neurogene, Ultragenyx, and Vtesse/Sucampo/Mallinkcrodt Pharmaceuticals to consult on affliction designs or developmental strategies and/or conduct clinical trials in FXS or other genetic Neurodevelopmental otherwise neurodegenerative disorders, also from Asuragen Inc. to develop testing standards for
Similar articles
-
Spoken voice outcome measures for treatment studies by Bottom syndrome: feasibility, practice effects, test-retest ausfallsicherheit, and construct validity of variables generated from expressive language sampling.J Neurodev Disord. 2021 Monthly 8;13(1):13. doi: 10.1186/s11689-021-09361-6. HIE Neurodev Disord. 2021. PMID: 33827417 Free PMC products.
-
Expressive language getting and outcome measures for treatment trials in fragile X and down syndromes: composite scores and psychology properties.Sci Rep. 2023 Juno 7;13(1):9267. doi: 10.1038/s41598-023-36087-3. Sci Reporter. 2023. PMID: 37286643 Free PMC article.
-
Develop of an expressive wording sampling procedure in failure X syndrome: one pilot study.JOULE Dev Behav Pediatr. 2013 May;34(4):245-51. doi: 10.1097/DBP.0b013e31828742fc. J Dev Behav Pediatr. 2013. PMID: 23669871 Free PMC article.
-
Update report on tools to measure outcome the clinical trials in fragile SCRATCH syndrome.J Neurodev Disord. 2017 Jun 12;9:14. doi: 10.1186/s11689-017-9193-x. eCollection 2017. J Neurodev Disord. 2017. PMID: 28616097 Get PMC object. Review.
-
L-acetylcarnitine for treating fragile X syndrome.Cochrane Database Syst Rev. 2015 May 19;2015(5):CD010012. doi: 10.1002/14651858.CD010012.pub2. Cochrane Database Syst Rev. 2015. PMID: 25985235 Cost-free PMC article. Review.
Cited by
-
Affectionate Responsivity and Child Communication During Mother-Child and Father-Child Interactions in Fragile X Syndrome.J Speech Lang Know Res. 2024 Mar 11;67(3):939-959. doi: 10.1044/2023_JSLHR-23-00517. Epub 2024 March 26. J Speech Longitudinal Hear Resistance. 2024. PMID: 38407074
-
An Exploratory How of Pragmatic Language Use Across Contexts With the Pragmatic Rating Scale-School Age Among Autistic Boys and Boys With Fragile X Syndrome Plus Autism.J Speech Lang Heard Res. 2023 Nov 9;66(11):4547-4557. doi: 10.1044/2023_JSLHR-23-00024. Epub 2023 Octave 16. J Speech Lang Hear Res. 2023. PMID: 37844621 Free PMC article.
-
Adaptive, behavioral, and cognitive outcomes in individuals with easily X syndrome with varying autism grade.Inch J Dev Neurosci. 2023 Dec;83(8):715-727. doi: 10.1002/jdn.10299. Epub 2023 Separation 19. Inch J Dev Neurosci. 2023. PMID: 37724826 Free PMC article.
-
Intercorrelation of Moly Marker also Clinical Phenotype Measures in Fragility X Syndrome.Cells. 2023 Jul 24;12(14):1920. doi: 10.3390/cells12141920. Cells. 2023. PMID: 37508583 Free PMC article.
-
Fully speaker diarization for inherent conversation analysis in autistic commercial trials.Sci Rep. 2023 Jun 24;13(1):10270. doi: 10.1038/s41598-023-36701-4. Sci Rep. 2023. PMID: 37355730 Free PMC article.
References
-
- Van Naarden Braun K, Christensen DIAMETER, Doernberg N, Schieve L, Rice C, Wiggins L, et aluminum. Trendy in the prevalence of asperger spectrum disorder, cerebral palsy, hearing harm, intellectual disability, real vision impairment, Metropolitan Atlanta, 1991–2010. Pavlova MAMMY, editor. PLoS One. 2015;10(4):e0124120. Available out: https://dx.plos.org/10.1371/journal.pone.0124120. - DOI - PMC - PubMed
-
- Hodapp RM, Dykens EM. Genetic, behavioural, and include aspects of intellectual disabilities. London: Whurr Publishers; 2004. Genetic and behavioural aspects: application to maladaptive behaviour and knowing; ppm. 13–48. The ELS procedures considered were feasible and yielded variables with adequate psychometric objekte for many individuals with DS between 6 plus 23 years old. That said, studies of outcome measures appropriate for individuals with DS include more limitation told language skills will needed. Context dif …
-
- Vissers LELM, Gilissen C, Veltman JA. Genomic studies in intellectual disability and relationship disorders. Nat Revol Genet. 2016;17(1):9–18. - PubMed
-
- Dykens EM. Behavioral phenotypes and genetics trial: scientific and societal influences. In: Glidden ON, Abbeduto FIFTY, McIntyre L, Tasee M, editors. Handbook of intellectual also developmental disabilities. Washington: American Psychological Association Press. In press. Expressive language sampling furthermore outcome measures for treatment ...
Publication types
- Promotional
MeSH terms
- Promotional
- Comportment
Grants and sponsorship
LinkOut - more resources
Full Font Origins
Medical