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.

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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
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article

Complex Genetic Architecture in RASopathies : Constitutional PTPN11 and Mosaic RIT1 Pathogenic Variants Underlying Severe Noonan Syndrome With Adult‐Onset Acute Myeloid Leukemia

Dario Seif Ali, Davide Calosci, Chiara Rigon, Monica Forzan et al.
American Journal of Medical Genetics Part A
Protein Tyrosine Phosphatases
article

Complex Genetic Architecture in RASopathies : Constitutional PTPN11 and Mosaic RIT1 Pathogenic Variants Underlying Severe Noonan Syndrome With Adult‐Onset Acute Myeloid Leukemia

Dario Seif Ali, Davide Calosci, Chiara Rigon, Monica Forzan, C. Piccolo, Carmela Gurrieri, Leonardo Salviati, Francesco Prevedello, Elena Tacchetto, Roberta Palmitessa, Eva Trevisson
article en

Abstract

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.

American Journal of Medical Genetics Part A
University of Verona (IT), Marche Polytechnic University (IT), University of Padua (IT), Città della Speranza Foundation (IT)
Gender equality
Openalex Percentile: Top 11%
Protein Tyrosine Phosphatases
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