Literature DB >> 30430033

Co-occurrence of Noonan and Cardiofaciocutaneous Syndrome Features in a Patient with KRAS Variant.

Fernando Rodríguez1, Carla Vallejos1, Víctor M Bolanos-Garcia2, Diana Ponce1, Nancy Unanue1, Francisco Garay3, Fernando Cassorla1, Mariana Aracena3,4.   

Abstract

We report the case of a 3-year-old girl, who is the third child of nonconsanguineous parents, with short stature, hypertrophic cardiomyopathy, and mild dysmorphic features; all suggestive of Noonan syndrome. In addition, the patient presents with feeding difficulties, deep palmar and plantar creases, sparse hair, and delayed psychomotor and language development, all characteristics frequently observed in cardiofaciocutaneous syndrome. Molecular analysis of the Ras/ MAPK pathway genes using high-resolution melting curve analysis and gene sequencing revealed a de novo KRAS amino acid substitution of leucine to tryptophan at codon 53 (p.L53W). This substitution was recently described in an Iranian patient with Noonan syndrome. The findings described in this report expand the phenotypic heterogeneity observed in RASopathy patients harboring a KRAS substitution, and advocate for the inclusion of genes with low mutational frequency in genetic screening protocols for Noonan syndrome and other RASopathies.

Entities:  

Keywords:  KRAS mutations ; Noonan syndrome; cardiofaciocutaneous syndrome

Year:  2018        PMID: 30430033      PMCID: PMC6234039          DOI: 10.1055/s-0038-1653977

Source DB:  PubMed          Journal:  J Pediatr Genet        ISSN: 2146-460X


  16 in total

1.  KRAS gene mutations in Noonan syndrome familial cases cluster in the vicinity of the switch II region of the G-domain: report of another family with metopic craniosynostosis.

Authors:  Amanda S Brasil; Alexsandra C Malaquias; Chong A Kim; José Eduardo Krieger; Alexander A L Jorge; Alexandre C Pereira; Débora R Bertola
Journal:  Am J Med Genet A       Date:  2012-04-09       Impact factor: 2.802

Review 2.  Noonan syndrome: clinical features, diagnosis, and management guidelines.

Authors:  Alicia A Romano; Judith E Allanson; Jovanna Dahlgren; Bruce D Gelb; Bryan Hall; Mary Ella Pierpont; Amy E Roberts; Wanda Robinson; Clifford M Takemoto; Jacqueline A Noonan
Journal:  Pediatrics       Date:  2010-09-27       Impact factor: 7.124

3.  Characterization of a novel KRAS mutation identified in Noonan syndrome.

Authors:  Md Abdur Razzaque; Yuta Komoike; Tsutomu Nishizawa; Kei Inai; Michiko Furutani; Toru Higashinakagawa; Rumiko Matsuoka
Journal:  Am J Med Genet A       Date:  2012-02-02       Impact factor: 2.802

4.  Expansion of the genotypic and phenotypic spectrum in patients with KRAS germline mutations.

Authors:  Martin Zenker; Katarina Lehmann; Anna Leana Schulz; Helmut Barth; Dagmar Hansmann; Rainer Koenig; Rudolf Korinthenberg; Martina Kreiss-Nachtsheim; Peter Meinecke; Susanne Morlot; Stefan Mundlos; Anne S Quante; Salmo Raskin; Dirk Schnabel; Lars-Erik Wehner; Christian P Kratz; Denise Horn; Kerstin Kutsche
Journal:  J Med Genet       Date:  2006-10-20       Impact factor: 6.318

Review 5.  The guanine nucleotide-binding switch in three dimensions.

Authors:  I R Vetter; A Wittinghofer
Journal:  Science       Date:  2001-11-09       Impact factor: 47.728

6.  Features and development of Coot.

Authors:  P Emsley; B Lohkamp; W G Scott; K Cowtan
Journal:  Acta Crystallogr D Biol Crystallogr       Date:  2010-03-24

Review 7.  Recent advances in RASopathies.

Authors:  Yoko Aoki; Tetsuya Niihori; Shin-ichi Inoue; Yoichi Matsubara
Journal:  J Hum Genet       Date:  2015-10-08       Impact factor: 3.172

8.  Germline missense mutations affecting KRAS Isoform B are associated with a severe Noonan syndrome phenotype.

Authors:  Claudio Carta; Francesca Pantaleoni; Gianfranco Bocchinfuso; Lorenzo Stella; Isabella Vasta; Anna Sarkozy; Cristina Digilio; Antonio Palleschi; Antonio Pizzuti; Paola Grammatico; Giuseppe Zampino; Bruno Dallapiccola; Bruce D Gelb; Marco Tartaglia
Journal:  Am J Hum Genet       Date:  2006-05-01       Impact factor: 11.025

9.  Noonan syndrome gain-of-function mutations in NRAS cause zebrafish gastrulation defects.

Authors:  Vincent Runtuwene; Mark van Eekelen; John Overvoorde; Holger Rehmann; Helger G Yntema; Willy M Nillesen; Arie van Haeringen; Ineke van der Burgt; Boudewijn Burgering; Jeroen den Hertog
Journal:  Dis Model Mech       Date:  2011-01-24       Impact factor: 5.758

10.  The Phyre2 web portal for protein modeling, prediction and analysis.

Authors:  Lawrence A Kelley; Stefans Mezulis; Christopher M Yates; Mark N Wass; Michael J E Sternberg
Journal:  Nat Protoc       Date:  2015-05-07       Impact factor: 13.491

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  1 in total

Review 1.  Cardiofaciocutaneous syndrome - a longitudinal study of a case over 33 years: case report and review of the literature.

Authors:  Maria Claudia Jurcă; Oana Alexandra Iuhas; Maria Puiu; Adela Chiriţă-Emandi; Nicoleta Ioana Andreescu; Codruţa Diana Petcheşi; Alexandru Daniel Jurcă; Ioan Magyar; Sânziana Iulia Jurcă; Kinga Kozma; Emilia Maria Severin; Marius Bembea
Journal:  Rom J Morphol Embryol       Date:  2021 Apr-Jun       Impact factor: 1.033

  1 in total

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