Expressive language sampling and outcome measures for treatment trials in fragile X and down syndromes: composite scores and psychometric properties.

Leonard Abbeduto, Laura Del Hoyo Soriano, Elizabeth Berry-Kravis, Audra Sterling, Jamie O Edgin, Nadia Abdelnur, Andrea Drayton, Anne Hoffmann, Debra Hamilton, Danielle J Harvey, Angela John Thurman
Author Information
  1. Leonard Abbeduto: MIND Institute and Department of Psychiatry and Behavioral Sciences, University of California Davis Health, 2828 50Th St., Sacramento, CA, 95817, USA. LJabbeduto@ucdavis.edu.
  2. Laura Del Hoyo Soriano: MIND Institute and Department of Psychiatry and Behavioral Sciences, University of California Davis Health, 2828 50Th St., Sacramento, CA, 95817, USA.
  3. Elizabeth Berry-Kravis: Department of Neurology, Rush University Medical Center, Chicago, IL, USA.
  4. Audra Sterling: Waisman Center and Department of Communication Sciences and Disorders, University of Wisconsin-Madison, Madison, WI, USA.
  5. Jamie O Edgin: Department of Psychology, Sonoran UCEDD, UA Family and Community Medicine, University of Arizona, Phoenix, AZ, USA.
  6. Nadia Abdelnur: MIND Institute and Department of Psychiatry and Behavioral Sciences, University of California Davis Health, 2828 50Th St., Sacramento, CA, 95817, USA.
  7. Andrea Drayton: MIND Institute and Department of Psychiatry and Behavioral Sciences, University of California Davis Health, 2828 50Th St., Sacramento, CA, 95817, USA.
  8. Anne Hoffmann: Department of Communication Disorders and Sciences, Rush University, Chicago, IL, USA.
  9. Debra Hamilton: Department of Human Genetics, Emory University School of Medicine, Atlanta, GA, USA.
  10. Danielle J Harvey: Department of Public Health Sciences, University of California, Davis, USA.
  11. Angela John Thurman: MIND Institute and Department of Psychiatry and Behavioral Sciences, University of California Davis Health, 2828 50Th St., Sacramento, CA, 95817, USA.

Abstract

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.

References

  1. Cogn Neuropsychiatry. 1997 May 1;2(2):81-99 [PMID: 25420198]
  2. Genet Med. 2001 Sep-Oct;3(5):359-71 [PMID: 11545690]
  3. Am J Intellect Dev Disabil. 2019 May;124(3):220-233 [PMID: 31026204]
  4. J Autism Dev Disord. 2015 Mar;45(3):846-57 [PMID: 25234481]
  5. J Neurodev Disord. 2009 Mar;1(1):33-45 [PMID: 19865612]
  6. J Neurodev Disord. 2017 Jun 12;9:7 [PMID: 28616096]
  7. Sci Transl Med. 2012 Sep 19;4(152):152ra127 [PMID: 22993294]
  8. Am J Speech Lang Pathol. 2013 May;22(2):S285-97 [PMID: 23695905]
  9. J Speech Lang Hear Res. 2013 Feb;56(1):295-309 [PMID: 22744141]
  10. J Dev Behav Pediatr. 2022 Jan 1;43(1):1-8 [PMID: 34001744]
  11. Neurology. 2020 Mar 24;94(12):e1229-e1240 [PMID: 32094241]
  12. Mol Syndromol. 2021 Jul;12(4):202-218 [PMID: 34421499]
  13. J Neurodev Disord. 2014;6(1):16 [PMID: 26491488]
  14. Pediatrics. 2017 Jun;139(Suppl 3):S194-S206 [PMID: 28814540]
  15. Lang Speech Hear Serv Sch. 2022 Oct 6;53(4):1088-1100 [PMID: 35930679]
  16. Clin Linguist Phon. 2019;33(6):547-569 [PMID: 30696277]
  17. Brain Sci. 2020 Jan 26;10(2): [PMID: 31991905]
  18. J Fluency Disord. 2008 Sep;33(3):220-40 [PMID: 18762063]
  19. J Neurodev Disord. 2020 Apr 22;12(1):12 [PMID: 32316911]
  20. Front Rehabil Sci. 2021 Nov;2: [PMID: 35036992]
  21. Ment Retard. 1995 Oct;33(5):279-88 [PMID: 7476250]
  22. J Speech Lang Hear Res. 2002 Oct;45(5):902-15 [PMID: 12381048]
  23. Brain Sci. 2020 Jun 06;10(6): [PMID: 32517224]
  24. J Autism Dev Disord. 2019 Mar;49(3):1131-1141 [PMID: 30430320]
  25. J Dev Behav Pediatr. 2013 Sep;34(7):508-22 [PMID: 24042082]
  26. J Neurodev Disord. 2016 Sep 06;8(1):35 [PMID: 27602170]
  27. Transl Sci Rare Dis. 2021;5(3-4):99-129 [PMID: 34268067]
  28. Brain Sci. 2021 Sep 08;11(9): [PMID: 34573200]
  29. J Neurodev Disord. 2021 Apr 8;13(1):13 [PMID: 33827417]
  30. Psychol Rev. 1998 Jan;105(1):158-73 [PMID: 9450375]
  31. PLoS One. 2015 Apr 29;10(4):e0124120 [PMID: 25923140]
  32. Nat Rev Drug Discov. 2018 Apr;17(4):280-299 [PMID: 29217836]
  33. J Neurodev Disord. 2020 Mar 24;12(1):10 [PMID: 32204695]
  34. J Fluency Disord. 2003 Summer;28(2):115-41; quiz 141-2 [PMID: 12809748]
  35. Lang Speech Hear Serv Sch. 2010 Oct;41(4):393-404 [PMID: 20601531]
  36. Neurology. 2019 Apr 16;92(16):741-742 [PMID: 30918096]
  37. Ment Retard Dev Disabil Res Rev. 2007;13(3):262-71 [PMID: 17910079]
  38. Am J Speech Lang Pathol. 2018 Feb 6;27(1):123-135 [PMID: 29222570]
  39. J Multidiscip Healthc. 2016 May 04;9:211-7 [PMID: 27217764]
  40. J Autism Dev Disord. 2020 Jul;50(7):2287-2306 [PMID: 29873016]
  41. J Neurodev Disord. 2017 Jun 12;9:14 [PMID: 28616097]
  42. J Pediatr. 2013 Oct;163(4):1163-8 [PMID: 23885965]
  43. Am J Intellect Dev Disabil. 2017 May;122(3):247-281 [PMID: 28452584]
  44. J Dev Behav Pediatr. 2013 May;34(4):245-51 [PMID: 23669871]
  45. J Child Lang. 1990 Oct;17(3):651-75 [PMID: 2269702]
  46. J Speech Lang Hear Res. 2006 Aug;49(4):793-808 [PMID: 16908875]
  47. Int J Lang Commun Disord. 2006 Nov-Dec;41(6):675-93 [PMID: 17079222]
  48. Brain Commun. 2020;2(1): [PMID: 32924010]
  49. Sci Transl Med. 2016 Jan 13;8(321):321ra5 [PMID: 26764156]
  50. Am J Hum Genet. 2009 Oct;85(4):503-14 [PMID: 19804849]
  51. J Speech Hear Res. 1981 Jun;24(2):154-61 [PMID: 7265928]
  52. J Speech Lang Hear Res. 2000 Apr;43(2):366-79 [PMID: 10757690]
  53. J Mol Diagn. 2009 Jul;11(4):324-9 [PMID: 19460941]
  54. J Intellect Disabil Res. 2022 Mar;66(3):265-281 [PMID: 34984734]
  55. J Speech Hear Res. 1982 Sep;25(3):421-7 [PMID: 7176616]

Grants

  1. UL1 TR001860/NCATS NIH HHS
  2. R01 HD074346/NICHD NIH HHS
  3. P50 HD103526/NICHD NIH HHS

MeSH Term

Humans
Psychometrics
Reproducibility of Results
Language
Vocabulary
Outcome Assessment, Health Care

Word Cloud

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