American journal of medical genetics. Part A · 2024
New Insights Into the Spectrum of RASopathies: Clinical and Genetic Data in a Cohort of 121 Spanish Patients.
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Barbero Ana Isabel Sánchez, Valenzuela Irene, Fernández-Alvarez Paula, Vazquez Élida, Cueto-Gonzalez Anna Maria, Lasa-Aranzasti Amaia, Trujillano Laura, Masotto Bárbara, Arumí Elena García, Tizzano Eduardo F
Аннотация
Noonan syndrome and related disorders are a group of well-known genetic conditions caused by dysregulation of the Ras/mitogen-activated protein kinase (RAS/MAPK) pathway. Because of the overlap of clinical and molecular features, they are now called RASopathies. In this study, we retrospectively analyzed the clinical data of 121 patients with a molecularly confirmed diagnosis of RASopathy, describing frequencies for clinical features in all organ systems as well as molecular data. The most common clinical diagnosis was Noonan Syndrome and the most frequently affected gene was PTPN11 followed by SOS1, RAF1, LZTR1, and RIT1. All patients had distinctive craniofacial features indicative of the RASopathy spectrum but we report some atypical features regarding craniofacial shape, such as craniosynostosis and microcephaly. We also describe uncommon clinical characteristics such as aortic dilation, multivalvular heart disease, abnormalities of the posterior fossa, and uterine congenital anomalies in female patients. Furthermore, the presence of multiple giant cell granulomas was observed specifically in patients with SOS1 variants. This comprehensive evaluation allows broadening the phenotypic spectrum of our population and their correlation with the genotype, which are essential to improve the recognition and the follow up of RASopathies as a multisystemic disease.
Предметные рубрики MeSH
Noonan SyndromeHumansRetrospective StudiesSpainMaleFemaleInfantChild, PreschoolAdultMiddle AgedAgedInfant, NewbornCraniofacial AbnormalitiesNeurodevelopmental DisordersCardiovascular AbnormalitiesCohort StudiesNeoplasmsHematologic DiseasesMAP Kinase Signaling SystemProtein Tyrosine Phosphatase, Non-Receptor Type 11MutationAbnormalities, Multiple