Literature DB >> 26607044

New Mutations Associated with Rasopathies in a Central European Population and Genotype-Phenotype Correlations.

M Čizmárová1, K Hlinková1, S Bertok2, P Kotnik2, H C Duba3, R Bertalan4, K Poločková5, Ľ Košťálová1, Z Pribilincová1, A Hlavatá1, L Kovács1, D Ilenčíková1,3.   

Abstract

We performed the genetic analysis of Rasopathy syndromes in patients from Central European by direct sequencing followed by next generation sequencing of genes associated with Rasopathies. All 51 patients harboured the typical features of Rasopathy syndromes. Thirty-five mutations were identified in the examined patients (22 in PTPN11, two in SOS1, one in RIT1, one in SHOC2, two in HRAS, three in BRAF, two in MAP2K1 and two in the NF1 gene). Two of them (p.Gly392Glu in the BRAF gene and p.Gln164Lys in the MAP2K1 gene) were novel with a potentially pathogenic effect on the structure of these proteins. Statistically significant differences in the presence of pulmonary stenosis (63.64% vs. 23.81%, P = 0.013897) and cryptorchidism (76.47% vs. 30%, P = 0.040224) were identified as the result of comparison of the prevalence of phenotypic features in patients with the phenotype of Noonan syndrome and mutation in the PTPN11 gene, with the prevalence of the same features in patients without PTPN11 mutation. Cryptorchidism as a statistically significant feature in our patients with PTPN11 mutation was not reported as significant in other European countries (Germany, Italy and Greece). The majority of mutations were clustered in exons 3 (45.45%), 8 (22.73%), and 13 (22.73%) of the PTPN11 gene.
© 2015 John Wiley & Sons Ltd/University College London.

Entities:  

Keywords:  BRAF; MAP2K1; PTPN11; Rasopathies; cardio-facio-cutaneous syndrome; central Europe; noonan syndrome

Mesh:

Substances:

Year:  2015        PMID: 26607044     DOI: 10.1111/ahg.12140

Source DB:  PubMed          Journal:  Ann Hum Genet        ISSN: 0003-4800            Impact factor:   1.670


  10 in total

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2.  Initial analysis of peripheral lymphocytic extracellular signal related kinase activation in autism.

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Review 3.  Evolving health care through personal genomics.

Authors:  Heidi L Rehm
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4.  The molecular functions of RIT1 and its contribution to human disease.

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5.  Integrating Functional Analysis in the Next-Generation Sequencing Diagnostic Pipeline of RASopathies.

Authors:  Gordon K C Leung; H M Luk; Vincent H M Tang; W W Gao; Christopher C Y Mak; Mullin H C Yu; W L Wong; Yoyo W Y Chu; W L Yang; Wilfred H S Wong; Alvin C H Ma; Anskar Y H Leung; D Y Jin; Kelvin Y K Chan; Judith Allanson; Ivan F M Lo; Brian H Y Chung
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7.  The spectrum of genetic variants and phenotypic features of Southeast Asian patients with Noonan syndrome.

Authors:  Ai-Ling Koh; Ee-Shien Tan; Maggie S Brett; Angeline H M Lai; Saumya Shekhar Jamuar; Ivy Ng; Ene-Choo Tan
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8.  Molecular and clinical studies in 107 Noonan syndrome affected individuals with PTPN11 mutations.

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9.  Intrinsically active MEK variants are differentially regulated by proteinases and phosphatases.

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  10 in total

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