Adaptive functioning in children and adolescents with Trisomy X: An exploratory analysis.
Adaptation, Physiological
/ genetics
Adolescent
Attention Deficit Disorder with Hyperactivity
/ genetics
Autism Spectrum Disorder
/ genetics
Child
Chromosomes, Human, X
/ genetics
Cognition
/ physiology
Executive Function
/ physiology
Female
Humans
Intelligence
/ genetics
Sex Chromosome Aberrations
Sex Chromosome Disorders of Sex Development
/ genetics
Trisomy
/ genetics
Triple X syndrome
Trisomy X
adaptive skills
inattention
prenatal
Journal
American journal of medical genetics. Part C, Seminars in medical genetics
ISSN: 1552-4876
Titre abrégé: Am J Med Genet C Semin Med Genet
Pays: United States
ID NLM: 101235745
Informations de publication
Date de publication:
06 2020
06 2020
Historique:
received:
08
02
2020
revised:
29
04
2020
accepted:
11
05
2020
pubmed:
18
6
2020
medline:
6
5
2021
entrez:
18
6
2020
Statut:
ppublish
Résumé
Identifying the factors related to adaptive functioning will improve the information available to families and providers of females with Trisomy X. Cognitive and behavioral features were assessed in 50 females ages 12.2 ± 3.6 years using the Behavior Assessment System for Children Second Edition (BASC-2) and Wechsler Scales of Intelligence. Executive functioning, social skills, and autistic traits were evaluated in a subset. Adaptive functioning was assessed using the BASC-2 adaptive skills composite score (ASC). Participants were classified as average adaptive skills (ASC T-score > 40) or deficits (ASC T-score < 40). Group comparisons were conducted. Multiple linear regression examined which factors contributed to ASC score. Twenty-eight females (55.6%) had adaptive skills deficits with functional communication being the most commonly affected adaptive domain. The group with ASC in the average range had higher verbal IQ (VIQ) and lower rates of numerous behavioral concerns. Internalizing behavior composite, DSM-IV inattentive symptoms score, and VIQ were significant predictors of ASC. Prenatally diagnosed females comprised over 70% of those with average adaptive skills. In this study, internalizing behaviors, inattentive ADHD symptoms, and VIQ were associated with poorer adaptive functioning. Early interventions targeting internalizing behaviors, attention/executive functioning, and communication skills may improve adaptive skills and deserve further study.
Identifiants
pubmed: 32548885
doi: 10.1002/ajmg.c.31803
pmc: PMC8363475
mid: NIHMS1721736
doi:
Types de publication
Journal Article
Research Support, N.I.H., Extramural
Langues
eng
Sous-ensembles de citation
IM
Pagination
456-468Subventions
Organisme : NICHD NIH HHS
ID : R01 HD091251
Pays : United States
Organisme : NICHD NIH HHS
ID : R01 HD042974
Pays : United States
Organisme : NCATS NIH HHS
ID : UL1 TR001082
Pays : United States
Organisme : NINDS NIH HHS
ID : K23NS070337
Pays : United States
Organisme : NINDS NIH HHS
ID : K23 NS070337
Pays : United States
Organisme : NICHD NIH HHS
ID : L40 HD051024
Pays : United States
Organisme : NCATS NIH HHS
ID : UL1 TR002535
Pays : United States
Organisme : NCATS NIH HHS
ID : UL1TR001082
Pays : United States
Informations de copyright
© 2020 Wiley Periodicals LLC.
Références
Birth Defects Orig Artic Ser. 1979;15(1):3-14
pubmed: 444646
Wellcome Open Res. 2019 Feb 15;4:32
pubmed: 31231689
Am J Med Genet A. 2016 Nov;170(11):2870-2881
pubmed: 27644018
J Neurodev Disord. 2015;7(1):30
pubmed: 26379794
Obstet Gynecol. 2016 May;127(5):e123-e137
pubmed: 26938574
Pediatr Endocrinol Rev. 2010 Dec;8 Suppl 1:151-9
pubmed: 21217607
Fertil Steril. 2003 Oct;80(4):1052-4
pubmed: 14556833
Pediatrics. 2014 Sep;134(3):e903-18
pubmed: 25157020
Pediatr Neurol. 2006 Jul;35(1):69-74
pubmed: 16814091
Fetal Diagn Ther. 2018;44(2):85-90
pubmed: 28873375
Sci Rep. 2019 May 23;9(1):7767
pubmed: 31123326
Orphanet J Rare Dis. 2010 May 11;5:8
pubmed: 20459843
Dev Med Child Neurol. 2010 Feb;52(2):119-29
pubmed: 20059514
J Child Psychol Psychiatry. 2012 Oct;53(10):1072-81
pubmed: 22827287
Am J Med Genet A. 2010 Feb;152A(2):367-72
pubmed: 20101696
J Dev Behav Pediatr. 2012 Nov-Dec;33(9):713-20
pubmed: 23117596
Birth Defects Orig Artic Ser. 1990;26(4):1-304
pubmed: 1982520
Eur J Med Genet. 2013 Jun;56(6):286-91
pubmed: 23542668
Pediatrics. 1988 Oct;82(4):619-30
pubmed: 2459656
J Am Acad Child Adolesc Psychiatry. 2001 Feb;40(2):168-79
pubmed: 11211365
J Am Acad Child Adolesc Psychiatry. 1987 Jul;26(4):499-503
pubmed: 3654500
Am J Med Genet A. 2018 Nov;176(11):2284-2291
pubmed: 30070765
Genes Brain Behav. 2014 Jun;13(5):459-67
pubmed: 24655419
Child Neuropsychol. 2018 Feb;24(2):277-286
pubmed: 27892807
Birth Defects Orig Artic Ser. 1990;26(4):209-23
pubmed: 2090319
Arch Dis Child. 2011 Oct;96(10):954-9
pubmed: 20656736
Clin Neuropsychol. 2013;27(3):386-95
pubmed: 23336208
Birth Defects Orig Artic Ser. 1982;18(4):1-5
pubmed: 7159715
Birth Defects Orig Artic Ser. 1986;22(3):1-328
pubmed: 3814774
Birth Defects Orig Artic Ser. 1979;15(1):203-41
pubmed: 444642
Eur J Hum Genet. 2017 Aug;25(8):930-934
pubmed: 28612834
Pediatrics. 1995 Aug;96(2 Pt 1):302-8
pubmed: 7630689
Autism Res. 2018 Feb;11(2):284-295
pubmed: 28960841
Child Neuropsychol. 2011;17(1):51-66
pubmed: 21218296
Orphanet J Rare Dis. 2019 Jan 14;14(1):16
pubmed: 30642344
J Clin Neurophysiol. 2004 Jul-Aug;21(4):249-53
pubmed: 15509914
J Dev Behav Pediatr. 2012 May;33(4):309-18
pubmed: 22333574
J Child Psychol Psychiatry. 2014 Apr;55(4):374-83
pubmed: 24256459
Am J Med Genet C Semin Med Genet. 2013 Feb 15;163C(1):27-34
pubmed: 23359595
J Autism Dev Disord. 2011 Aug;41(8):1007-18
pubmed: 21042872
Am J Med Genet. 2002 Jun 1;110(1):11-8
pubmed: 12116265
Eur J Hum Genet. 2010 Mar;18(3):265-71
pubmed: 19568271
Psychol Assess. 2001 Dec;13(4):566-71
pubmed: 11793899
Pediatrics. 1983 Feb;71(2):262-7
pubmed: 6823432
Am J Obstet Gynecol. 2017 Dec;217(6):691.e1-691.e6
pubmed: 29032050
Genes Brain Behav. 2015 Feb;14(2):200-8
pubmed: 25684214
Wellcome Open Res. 2018 Nov 8;3:143
pubmed: 30815537