Complex Genetic Architecture in <scp>RASopathies</scp> : Constitutional <scp> <i>PTPN11</i> </scp> and Mosaic <scp> <i>RIT1</i> </scp> Pathogenic Variants Underlying Severe Noonan Syndrome With Adult‐Onset Acute Myeloid Leukemia
Noonan syndrome (NS) is a genetically heterogeneous disorder characterized by a broad spectrum of clinical features resulting from dysregulation of the RAS/MAPK pathway. Although complex genotypes are increasingly recognized in NS, cases harboring two distinct pathogenic variants in different NS genes remain extremely rare. We describe the case of a 53-year-old female presenting a severe NS phenotype-including short stature, facial dysmorphism, congenital heart defect, and developmental delay-and concurrent acute myeloid leukemia (AML). Targeted NGS analysis of a RASopathy-specific gene panel identified a constitutional heterozygous PTPN11 variant (c.1472C>T, p.(Pro491Leu)) and a mosaic RIT1 variant (c.229G>C, p.(Ala77Pro), 16.45% VAF), both meeting criteria for pathogenicity. The RIT1 variant was validated via PCR-RFLP across multiple tissues, excluding leukemia-driven clonal expansion, and further quantified by high-depth amplicon-based sequencing. To our knowledge, this case represents a unique example of NS associated with pathogenic variants in two distinct RASopathy genes. Our findings underscore that comprehensive molecular characterization and multi-tissue validation are essential for accurate diagnosis, genetic counseling, and personalized management, while also revealing that combinatorial RAS/MAPK alterations may influence disease severity and clinical outcomes.
Authors
- Dario Seif Ali
- Davide Calosci (ORCID: https://orcid.org/0009-0005-9106-9816)
- Chiara Rigon (ORCID: https://orcid.org/0000-0001-7820-2161)
- Monica Forzan (ORCID: https://orcid.org/0000-0003-2653-7300)
- C. Piccolo (ORCID: https://orcid.org/0000-0002-2719-0014)
- Carmela Gurrieri (ORCID: https://orcid.org/0000-0001-8233-6007)
- Leonardo Salviati (ORCID: https://orcid.org/0000-0001-5642-9963)
- Francesco Prevedello (ORCID: https://orcid.org/0009-0004-8950-8595)
- Elena Tacchetto
- Roberta Palmitessa
- Eva Trevisson
Institutions
- University of Verona (IT)
- Marche Polytechnic University (IT)
- University of Padua (IT)
- Città della Speranza Foundation (IT)
Publication Details
- Journal
- American Journal of Medical Genetics Part A
- Published
- 2026-03-22
- DOI
- https://doi.org/10.1002/ajmg.a.70135
- Primary Topic
- Protein Tyrosine Phosphatases
- Type
- article
- Field-Weighted Citation Impact
- 0.00