主页 文献库文献详情
PMID: 42261585 已发表 · ppublish 英语

Clinical and Molecular Portraits of Pediatric RASopathies: A Study of 118 Genotype-Confirmed Cases.

Clinical genetics ·第 110 卷 ·第 3 期 ·2026-09-00

Genç A, Sarıkaya E, Ceylan AC, Çavdarlı B, Çetin İİ, Erdoğan İ, Kılıç E

摘要

RASopathies are clinically and genetically heterogeneous disorders resulting from dysregulation of the RAS/MAPK pathway. We present a single-center retrospective cohort of 118 genotype-confirmed pediatric patients diagnosed with RASopathy. Noonan syndrome was the most common clinical diagnosis, followed by NF-Noonan syndrome, Legius syndrome, LEOPARD syndrome, Costello syndrome, and cardiofaciocutaneous syndrome. The most frequent clinical features included short stature, pulmonary stenosis, pectus deformities, and neurocognitive delay. Through molecular analysis, we identified distinct variants across major RAS/MAPK pathway genes, with a considerable proportion of patients carrying rare or less frequently reported variants, including those in LZTR1, RIT1, RAF1, HRAS, BRAF, RASA2, KRAS, CBL, SHOC2, and MAP2K2. In addition to well-established genotype-phenotype correlations, we report several uncommon or previously unrecognized features: microcephaly in multiple PTPN11-positive patients and renal agenesis in a child with a novel HRAS variant diagnosed with Costello syndrome. Furthermore, we identified three patients harboring RASA2 variants, providing additional support for its emerging role in Noonan syndrome. This study expands the clinical and molecular spectrum of pediatric RASopathies. It emphasizes the need for comprehensive genetic evaluation and regular follow-up in the context of overlapping clinical features, supporting accurate diagnosis and identifying candidates for emerging targeted therapies.

关键词
Noonan syndrome RASopathy genotype–phenotype correlations rare variants
文献信息
期刊
Clinical genetics
期刊简称
Clin Genet
ISSN
1399-0004
发表日期
2026-09-00
语言
英语
国家/地区
Denmark
NLM ID
0253664
分析服务
分析服务

联系地址

山东省济南市章丘区文博路2号

齐鲁师范学院 genelibs生信实验室

山东省济南市高新区舜华路750号

大学科技园北区F座4单元2楼

电话: 0531-88819269

微信公众号

关注微信订阅号,实时查看信息,关注医学生物学动态。


商务邮箱

E-mail: product@genelibs.com