Literature DB >> 9332653

Discriminant analysis of the Ullrich-Turner syndrome neurocognitive profile.

J L Ross1, H Kushner, A R Zinn.   

Abstract

Ullrich-Turner syndrome (UTS), or monosomy X, is a genetic disorder characterized by short stature, gonadal dysgenesis, and a particular neurocognitive profile of normally developed language abilities (particularly verbal IQ) and impaired visual-spatial and/or visual-perceptual abilities. The most frequently described profile in UTS includes difficulty with tasks involving memory and attention, decreased arithmetic skills, and impaired visual spatial processing. We used discriminant function analysis (DFA) to distinguish between the neurocognitive profiles of girls with UTS vs. controls matched for age, height, IQ, and socioeconomic status. DFA is a statistical method for deriving a linear function that optimally weights parameters to permit sensitive and specific differentiation among groups. We developed a modified discriminant function, based on seven cognitive test scores, that successfully discriminated between the UTS and control subjects with a sensitivity of 0.45 and a specificity of 0.97. To validate its performance, we applied the discriminant function to a small group of 45,X UTS subjects (n = 13) and control female subjects (n = 25), ages 7-16 years, who were not part of the previous analyses. The discriminant function (DF) identified 54% of these 13 UTS subjects as having the "UTS neurocognitive profile" and 92% of the 25 control subjects as not having the profile. We also compared the DF scores of UTS girls with various mosaic karyotypes and found that the group with 46,XX mosaicism had significantly higher scores (i.e., closer to normal controls) than the other two mosaic groups (t = 2.86, P < 0.005). The results of this study should be useful for genetic counseling and planning educational programs for girls with UTS.

Entities:  

Mesh:

Year:  1997        PMID: 9332653     DOI: 10.1002/(sici)1096-8628(19971031)72:3<275::aid-ajmg4>3.0.co;2-q

Source DB:  PubMed          Journal:  Am J Med Genet        ISSN: 0148-7299


  12 in total

Review 1.  Behavioural phenotypes: what do they teach us?

Authors:  D H Skuse
Journal:  Arch Dis Child       Date:  2000-03       Impact factor: 3.791

2.  The Turner syndrome-associated neurocognitive phenotype maps to distal Xp.

Authors:  J L Ross; D Roeltgen; H Kushner; F Wei; A R Zinn
Journal:  Am J Hum Genet       Date:  2000-08-08       Impact factor: 11.025

3.  Impact of cognitive profile on social functioning in prepubescent females with Turner syndrome.

Authors:  Jean-François Lepage; Bria Dunkin; David S Hong; Allan L Reiss
Journal:  Child Neuropsychol       Date:  2012-02-28       Impact factor: 2.500

Review 4.  Turner syndrome.

Authors:  Shelli R Kesler
Journal:  Child Adolesc Psychiatr Clin N Am       Date:  2007-07

5.  Executive Functions in Children and Adolescents with Turner Syndrome: A Systematic Review and Meta-Analysis.

Authors:  Claire Mauger; Céline Lancelot; Arnaud Roy; Régis Coutant; Nicole Cantisano; Didier Le Gall
Journal:  Neuropsychol Rev       Date:  2018-04-27       Impact factor: 7.444

Review 6.  Clinical developmental, neuropsychological, and social-emotional features of Turner syndrome.

Authors:  Christa Hutaff-Lee; Elizabeth Bennett; Susan Howell; Nicole Tartaglia
Journal:  Am J Med Genet C Semin Med Genet       Date:  2019-02-14       Impact factor: 3.908

7.  Psychosocial functioning and social cognitive processing in girls with Turner syndrome.

Authors:  David S Hong; Bria Dunkin; Allan L Reiss
Journal:  J Dev Behav Pediatr       Date:  2011-09       Impact factor: 2.225

Review 8.  Cognitive profile of Turner syndrome.

Authors:  David Hong; Jamie Scaletta Kent; Shelli Kesler
Journal:  Dev Disabil Res Rev       Date:  2009

Review 9.  Sex differences in psychiatric disorders: what we can learn from sex chromosome aneuploidies.

Authors:  Tamar Green; Shira Flash; Allan L Reiss
Journal:  Neuropsychopharmacology       Date:  2018-07-16       Impact factor: 7.853

10.  Developmental malformation of the corpus callosum: a review of typical callosal development and examples of developmental disorders with callosal involvement.

Authors:  Lynn K Paul
Journal:  J Neurodev Disord       Date:  2010-09-23       Impact factor: 4.025

View more

北京卡尤迪生物科技股份有限公司 © 2022-2023.