Expressive language sampling and outcome measures for treatment trials in fragile X and down syndromes: composite scores and psychometric properties.
Journal
Scientific reports
ISSN: 2045-2322
Titre abrégé: Sci Rep
Pays: England
ID NLM: 101563288
Informations de publication
Date de publication:
07 06 2023
07 06 2023
Historique:
received:
14
08
2022
accepted:
29
05
2023
medline:
9
6
2023
pubmed:
8
6
2023
entrez:
7
6
2023
Statut:
epublish
Résumé
The lack of psychometrically sound outcome measures has been a barrier to evaluating the efficacy of treatments proposed for core symptoms of intellectual disability (ID). Research on Expressive Language Sampling (ELS) procedures suggest it is a promising approach to measuring treatment efficacy. ELS entails collecting samples of a participant's talk in interactions with an examiner that are naturalistic but sufficiently structured to ensure consistency and limit examiner effects on the language produced. In this study, we extended previous research on ELS by analyzing an existing dataset to determine whether psychometrically adequate composite scores reflecting multiple dimensions of language can be derived from ELS procedures administered to 6- to 23-year-olds with fragile X syndrome (n = 80) or Down syndrome (n = 78). Data came from ELS conversation and narration procedures administered twice in a 4-week test-retest interval. We found that several composites emerged from variables indexing syntax, vocabulary, planning processes, speech articulation, and talkativeness, although there were some differences in the composites for the two syndromes. Evidence of strong test-retest reliability and construct validity of two of three composites were obtained for each syndrome. Situations in which the composite scores would be useful in evaluating treatment efficacy are outlined.
Identifiants
pubmed: 37286643
doi: 10.1038/s41598-023-36087-3
pii: 10.1038/s41598-023-36087-3
pmc: PMC10247708
doi:
Types de publication
Journal Article
Research Support, N.I.H., Extramural
Langues
eng
Sous-ensembles de citation
IM
Pagination
9267Subventions
Organisme : NCATS NIH HHS
ID : UL1 TR001860
Pays : United States
Organisme : NICHD NIH HHS
ID : R01 HD074346
Pays : United States
Organisme : NICHD NIH HHS
ID : P50 HD103526
Pays : United States
Informations de copyright
© 2023. The Author(s).
Références
Cogn Neuropsychiatry. 1997 May 1;2(2):81-99
pubmed: 25420198
Genet Med. 2001 Sep-Oct;3(5):359-71
pubmed: 11545690
Am J Intellect Dev Disabil. 2019 May;124(3):220-233
pubmed: 31026204
J Autism Dev Disord. 2015 Mar;45(3):846-57
pubmed: 25234481
J Neurodev Disord. 2009 Mar;1(1):33-45
pubmed: 19865612
J Neurodev Disord. 2017 Jun 12;9:7
pubmed: 28616096
Sci Transl Med. 2012 Sep 19;4(152):152ra127
pubmed: 22993294
Am J Speech Lang Pathol. 2013 May;22(2):S285-97
pubmed: 23695905
J Speech Lang Hear Res. 2013 Feb;56(1):295-309
pubmed: 22744141
J Dev Behav Pediatr. 2022 Jan 1;43(1):1-8
pubmed: 34001744
Neurology. 2020 Mar 24;94(12):e1229-e1240
pubmed: 32094241
Mol Syndromol. 2021 Jul;12(4):202-218
pubmed: 34421499
J Neurodev Disord. 2014;6(1):16
pubmed: 26491488
Pediatrics. 2017 Jun;139(Suppl 3):S194-S206
pubmed: 28814540
Lang Speech Hear Serv Sch. 2022 Oct 6;53(4):1088-1100
pubmed: 35930679
Clin Linguist Phon. 2019;33(6):547-569
pubmed: 30696277
Brain Sci. 2020 Jan 26;10(2):
pubmed: 31991905
J Fluency Disord. 2008 Sep;33(3):220-40
pubmed: 18762063
J Neurodev Disord. 2020 Apr 22;12(1):12
pubmed: 32316911
Front Rehabil Sci. 2021 Nov;2:
pubmed: 35036992
Ment Retard. 1995 Oct;33(5):279-88
pubmed: 7476250
J Speech Lang Hear Res. 2002 Oct;45(5):902-15
pubmed: 12381048
Brain Sci. 2020 Jun 06;10(6):
pubmed: 32517224
J Autism Dev Disord. 2019 Mar;49(3):1131-1141
pubmed: 30430320
J Dev Behav Pediatr. 2013 Sep;34(7):508-22
pubmed: 24042082
J Neurodev Disord. 2016 Sep 06;8(1):35
pubmed: 27602170
Transl Sci Rare Dis. 2021;5(3-4):99-129
pubmed: 34268067
Brain Sci. 2021 Sep 08;11(9):
pubmed: 34573200
J Neurodev Disord. 2021 Apr 8;13(1):13
pubmed: 33827417
Psychol Rev. 1998 Jan;105(1):158-73
pubmed: 9450375
PLoS One. 2015 Apr 29;10(4):e0124120
pubmed: 25923140
Nat Rev Drug Discov. 2018 Apr;17(4):280-299
pubmed: 29217836
J Neurodev Disord. 2020 Mar 24;12(1):10
pubmed: 32204695
J Fluency Disord. 2003 Summer;28(2):115-41; quiz 141-2
pubmed: 12809748
Lang Speech Hear Serv Sch. 2010 Oct;41(4):393-404
pubmed: 20601531
Neurology. 2019 Apr 16;92(16):741-742
pubmed: 30918096
Ment Retard Dev Disabil Res Rev. 2007;13(3):262-71
pubmed: 17910079
Am J Speech Lang Pathol. 2018 Feb 6;27(1):123-135
pubmed: 29222570
J Multidiscip Healthc. 2016 May 04;9:211-7
pubmed: 27217764
J Autism Dev Disord. 2020 Jul;50(7):2287-2306
pubmed: 29873016
J Neurodev Disord. 2017 Jun 12;9:14
pubmed: 28616097
J Pediatr. 2013 Oct;163(4):1163-8
pubmed: 23885965
Am J Intellect Dev Disabil. 2017 May;122(3):247-281
pubmed: 28452584
J Dev Behav Pediatr. 2013 May;34(4):245-51
pubmed: 23669871
J Child Lang. 1990 Oct;17(3):651-75
pubmed: 2269702
J Speech Lang Hear Res. 2006 Aug;49(4):793-808
pubmed: 16908875
Int J Lang Commun Disord. 2006 Nov-Dec;41(6):675-93
pubmed: 17079222
Brain Commun. 2020;2(1):
pubmed: 32924010
Sci Transl Med. 2016 Jan 13;8(321):321ra5
pubmed: 26764156
Am J Hum Genet. 2009 Oct;85(4):503-14
pubmed: 19804849
J Speech Hear Res. 1981 Jun;24(2):154-61
pubmed: 7265928
J Speech Lang Hear Res. 2000 Apr;43(2):366-79
pubmed: 10757690
J Mol Diagn. 2009 Jul;11(4):324-9
pubmed: 19460941
J Intellect Disabil Res. 2022 Mar;66(3):265-281
pubmed: 34984734
J Speech Hear Res. 1982 Sep;25(3):421-7
pubmed: 7176616