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Investigating the Pathogenesis of MYH7 Mutation Gly823Glu in Familial Hypertrophic Cardiomyopathy using a Mouse Model
Published on: August 8, 2022
A Novel HRAS Mutation Independently Contributes to Left Ventricular Hypertrophy in a Family with a Known MYH7
Maria Elena Sana1,2, Lawrence A Quilliam3, Andrea Spitaleri4
1USSD Laboratorio di Genetica Medica, Azienda Socio Sanitaria Territoriale Papa Giovanni XXIII, Bergamo, Italy.
Insights
A novel HRAS gene mutation causes left ventricular hypertrophy (LVH) and RASopathy, mimicking inherited hypertrophic cardiomyopathy (HCM). This finding underscores the importance of comprehensive genetic testing for accurate diagnosis of cardiac conditions.
Area of Science:
- Genetics
- Cardiology
- Molecular Biology
Background:
- Left ventricular hypertrophy (LVH) can stem from genetic conditions, most commonly hypertrophic cardiomyopathy (HCM) due to sarcomere gene mutations.
- RASopathies, linked to RAS-MAPK pathway mutations, are rare disorders with potential cardiac manifestations like LVH.
Observation:
- A 62-year-old male presented with severe obstructive LVH but lacked typical HCM-associated sarcomere mutations found in relatives.
- Exome sequencing revealed a novel HRAS gene mutation (p.Arg68Trp) in the patient and his daughter, who exhibited mild LVH and intellectual disability.
Findings:
- The identified HRAS mutation leads to constitutive activation of the RAS-MAPK pathway, confirmed by in silico and in vitro studies.
- This activation drives cellular proliferation, contributing to LVH and demonstrating a significant role for RAS signaling in cardiac hypertrophy.
Implications:
- This study highlights the diagnostic challenges in differentiating RASopathies from HCM, especially when phenotypes overlap.
- Comprehensive genetic analysis is crucial for accurate diagnosis, revealing the complex genetic underpinnings of cardiac hypertrophy and RASopathies.
Abstract:
Several genetic conditions can lead to left ventricular hypertrophy (LVH). Among them, hypertrophic cardiomyopathy (HCM), caused by mutations in sarcomere genes, is the most common inherited cardiac disease. Instead, RASopathies, a rare class of disorders characterized by neuro-cardio-facial-cutaneous abnormalities and sometimes presenting with LVH, are caused by mutations in the RAS-MAPK pathway. We report on a 62-years-old male who presented isolated severe obstructive LVH but did not carry the sarcomere mutation previously identified in his affected relatives. By exome sequencing, we detected a novel mutation in HRAS gene (NM_005343.2:p.Arg68Trp), present also in the proband's daughter, who showed mild LVH and severe intellectual disability. The cardiac phenotype was indistinguishable between family members carrying either mutation. In silico studies suggested that the mutated HRAS protein is constitutionally activated. Consistently, functional characterization in vitro confirmed elevated HRAS-GTP accumulation and downstream RAS-MAPK pathway activation that are known to drive cell proliferation in LVH. Our study emphasizes the role of RAS signaling in cardiac hypertrophy and highlights the complexity in differential diagnosis of RASopathies. In fact, the mild features of RASopathy and the recurrence of sarcomeric HCM in this family delayed the correct diagnosis until comprehensive genetic testing was performed.
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