Literature DB >> 31173466

Severe Noonan syndrome phenotype associated with a germline Q71R MRAS variant: a recurrent substitution in RAS homologs in various cancers.

Hisato Suzuki1, Toshiki Takenouchi2, Tomoko Uehara1, Satoshi Takasago3, Satoshi Ihara4, Hiroshi Yoshihashi5, Kenjiro Kosaki1.   

Abstract

Activation of the RAS pathway through either the activation of genes that accelerate the pathway or the suppression of genes that inhibit the pathway leads to a group of disorders collectively referred to as RASopathies. The key molecules of the RAS pathway are KRAS, HRAS, and NRAS. Mutations in these three RAS homolog genes have been shown to be associated with RASopathies. Recently, two patients with a Noonan syndrome phenotype were shown to carry mutations in the yet another RASopathy gene, MRAS (muscle RAS oncogene homolog). Here, we report a patient with a severe Noonan syndrome phenotype associated with a germline Q71R MRAS variant, which represents a recurrent substitution in RAS homologs in various cancers. The patient's dysmorphic features included relative macrocephaly, a down-slanted palpebral fissure, hypertelorism, a depressed nasal bridge, and low-set ears with thick lobes; these facial features are strongly associated with RASopathy. We confirmed that the MRAS gene represents a causative gene for RASopathy.
© 2019 Wiley Periodicals, Inc.

Entities:  

Keywords:  Costello syndrome; MRAS; Noonan syndrome; RAS; RASopathy

Mesh:

Substances:

Year:  2019        PMID: 31173466     DOI: 10.1002/ajmg.a.61261

Source DB:  PubMed          Journal:  Am J Med Genet A        ISSN: 1552-4825            Impact factor:   2.802


  5 in total

1.  Structure of the SHOC2-MRAS-PP1C complex provides insights into RAF activation and Noonan syndrome.

Authors:  Daniel A Bonsor; Patrick Alexander; Kelly Snead; Nicole Hartig; Matthew Drew; Simon Messing; Lorenzo I Finci; Dwight V Nissley; Frank McCormick; Dominic Esposito; Pablo Rodriguez-Viciana; Andrew G Stephen; Dhirendra K Simanshu
Journal:  Nat Struct Mol Biol       Date:  2022-09-29       Impact factor: 18.361

2.  Structure of the MRAS-SHOC2-PP1C phosphatase complex.

Authors:  Zachary J Hauseman; Michelle Fodor; Anxhela Dhembi; Jessica Viscomi; David Egli; Melusine Bleu; Stephanie Katz; Eunyoung Park; Dong Man Jang; Kathryn A Porter; Fabian Meili; Hongqiu Guo; Grainne Kerr; Sandra Mollé; Camilo Velez-Vega; Kim S Beyer; Giorgio G Galli; Saveur-Michel Maira; Travis Stams; Kirk Clark; Michael J Eck; Luca Tordella; Claudio R Thoma; Daniel A King
Journal:  Nature       Date:  2022-07-13       Impact factor: 69.504

3.  Six years' accomplishment of the Initiative on Rare and Undiagnosed Diseases: nationwide project in Japan to discover causes, mechanisms, and cures.

Authors:  Yuji Takahashi; Hidetoshi Date; Hideki Oi; Takeya Adachi; Noriaki Imanishi; En Kimura; Hotake Takizawa; Shinji Kosugi; Naomichi Matsumoto; Kenjiro Kosaki; Yoichi Matsubara; Hidehiro Mizusawa
Journal:  J Hum Genet       Date:  2022-03-23       Impact factor: 3.755

4.  Integrated in silico MS-based phosphoproteomics and network enrichment analysis of RASopathy proteins.

Authors:  Javier-Fernando Montero-Bullón; Óscar González-Velasco; María Isidoro-García; Jesus Lacal
Journal:  Orphanet J Rare Dis       Date:  2021-07-06       Impact factor: 4.123

Review 5.  The Role of R-Ras Proteins in Normal and Pathologic Migration and Morphologic Change.

Authors:  Shannon M Weber; Steven L Carroll
Journal:  Am J Pathol       Date:  2021-06-07       Impact factor: 5.770

  5 in total

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