Literature DB >> 10586173

Patterns of cognitive functioning in school-aged children with Noonan syndrome associated with variability in phenotypic expression.

I van der Burgt1, G Thoonen, N Roosenboom, C Assman-Hulsmans, F Gabreels, B Otten, H G Brunner.   

Abstract

OBJECTIVE: To evaluate the cognitive profiles of children with Noonan syndrome (NS) and to relate these profiles to measures of overall clinical severity. STUDY
DESIGN: Thirty-five children with NS between the ages of 7 and 18 years were tested on their intellectual, psychosocial, and academic functioning. The diagnosis of NS was established on the presence of a typical face, the characteristic heart defect, thorax deformity, short stature, affected first-degree relative(s), and cryptorchidism in male subjects.
RESULTS: The total group of children with NS (n = 35) achieved significantly lower mean full-scale IQ, verbal IQ (VIQ), and performance IQ (PIQ) scores (between 85.9 and 89.3) than expected based on normative data. The individual full-scale IQ scores varied between 48 and 130. Because of this wide range of individual scores, the mean group values are not extremely informative. The mean full-scale IQ for the group with moderate NS (n = 19) is 90.8; for the children with severe NS (n = 16) the mean full-scale IQ is 80.6. The patterns of discrepancies between VIQ and PIQ are: (1) an extreme discrepancy between VIQ and PIQ is most likely to emerge in children with severe NS with (low) average intellectual abilities; (2) children with moderate NS are more likely to attain similarities in VIQ and PIQ scores; and (3) children with moderate NS demonstrate a particular pattern of discrepancy between VIQ and PIQ (ie, VIQ > PIQ).
CONCLUSION: For children with NS, the findings on physical examination are indicative of the pattern of cognitive abilities. NS is not associated with substantial deficits in the level of intellectual functioning or with a single/unitary cognitive pattern. Severe NS expression, however, predicts in part a specific pattern of deficits and capacities in cognitive functioning.

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Year:  1999        PMID: 10586173     DOI: 10.1016/s0022-3476(99)70089-2

Source DB:  PubMed          Journal:  J Pediatr        ISSN: 0022-3476            Impact factor:   4.406


  17 in total

1.  The language phenotype of children and adolescents with Noonan syndrome.

Authors:  Elizabeth I Pierpont; Susan Ellis Weismer; Amy E Roberts; Erica Tworog-Dube; Mary Ella Pierpont; Nancy J Mendelsohn; Mark S Seidenberg
Journal:  J Speech Lang Hear Res       Date:  2010-06-11       Impact factor: 2.297

2.  Effects of germline mutations in the Ras/MAPK signaling pathway on adaptive behavior: cardiofaciocutaneous syndrome and Noonan syndrome.

Authors:  Elizabeth I Pierpont; Mary Ella Pierpont; Nancy J Mendelsohn; Amy E Roberts; Erica Tworog-Dube; Katherine A Rauen; Mark S Seidenberg
Journal:  Am J Med Genet A       Date:  2010-03       Impact factor: 2.802

3.  Noonan syndrome in diverse populations.

Authors:  Paul Kruszka; Antonio R Porras; Yonit A Addissie; Angélica Moresco; Sofia Medrano; Gary T K Mok; Gordon K C Leung; Cedrik Tekendo-Ngongang; Annette Uwineza; Meow-Keong Thong; Premala Muthukumarasamy; Engela Honey; Ekanem N Ekure; Ogochukwu J Sokunbi; Nnenna Kalu; Kelly L Jones; Julie D Kaplan; Omar A Abdul-Rahman; Lisa M Vincent; Amber Love; Khadija Belhassan; Karim Ouldim; Ihssane El Bouchikhi; Anju Shukla; Katta M Girisha; Siddaramappa J Patil; Nirmala D Sirisena; Vajira H W Dissanayake; C Sampath Paththinige; Rupesh Mishra; Eva Klein-Zighelboim; Bertha E Gallardo Jugo; Miguel Chávez Pastor; Hugo H Abarca-Barriga; Steven A Skinner; Eloise J Prijoles; Eben Badoe; Ashleigh D Gill; Vorasuk Shotelersuk; Patroula Smpokou; Monisha S Kisling; Carlos R Ferreira; Leon Mutesa; Andre Megarbane; Antonie D Kline; Amy Kimball; Emmy Okello; Peter Lwabi; Twalib Aliku; Emmanuel Tenywa; Nonglak Boonchooduang; Pranoot Tanpaiboon; Antonio Richieri-Costa; Ambroise Wonkam; Brian H Y Chung; Roger E Stevenson; Marshall Summar; Kausik Mandal; Shubha R Phadke; María G Obregon; Marius G Linguraru; Maximilian Muenke
Journal:  Am J Med Genet A       Date:  2017-07-27       Impact factor: 2.802

4.  Genotype differences in cognitive functioning in Noonan syndrome.

Authors:  E I Pierpont; M E Pierpont; N J Mendelsohn; A E Roberts; E Tworog-Dube; M S Seidenberg
Journal:  Genes Brain Behav       Date:  2008-12-11       Impact factor: 3.449

5.  Hodgkin's lymphoma in a patient with Noonan syndrome with germ-line PTPN11 mutations.

Authors:  Fu-Sung Lo; Tseng-Tong Kuo; Chao-Jan Wang; Min-Tzu Kuo; Ming-Chung Kuo
Journal:  Int J Hematol       Date:  2008-08-30       Impact factor: 2.490

6.  A QTL on chromosome 3q23 influences processing speed in humans.

Authors:  Emma E M Knowles; Samuel R Mathias; Josephine Mollon; Amanda Rodrigue; Marinka M G Koenis; Thomas D Dyer; Harald H H Goring; Joanne E Curran; Rene L Olvera; Ravi Duggirala; Laura Almasy; John Blangero; David C Glahn
Journal:  Genes Brain Behav       Date:  2018-11-16       Impact factor: 3.449

7.  Objective differential diagnosis of Noonan and Williams-Beuren syndromes in diverse populations using quantitative facial phenotyping.

Authors:  Antonio R Porras; Marshal Summar; Marius George Linguraru
Journal:  Mol Genet Genomic Med       Date:  2021-03-27       Impact factor: 2.183

8.  Clinical manifestations of Noonan syndrome.

Authors:  M Digilio; B Marino
Journal:  Images Paediatr Cardiol       Date:  2001-04

Review 9.  Noonan syndrome.

Authors:  Ineke van der Burgt
Journal:  Orphanet J Rare Dis       Date:  2007-01-14       Impact factor: 4.123

10.  Top-down or bottom-up: Contrasting perspectives on psychiatric diagnoses.

Authors:  Willem Ma Verhoeven; Siegfried Tuinier; Ineke van der Burgt
Journal:  Biologics       Date:  2008-09
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