It is demonstrated that oncogenesis in LFS can be driven by partial impairment of functional p53, rather than dominant-negative or gain-of-function mutations alone.
Abstract
Li-Fraumeni Syndrome (LFS) is a hereditary cancer predisposition syndrome caused by germline mutations in the
TP53
tumour suppressor gene, which encodes the multifunctional transcription factor p53. p53 is the most commonly mutated protein in human cancer, with the majority occurring within the DNA-binding domain, often disrupting transcriptional activity and resulting in a loss-of-function. Here, we characterise the novel p53
N263Tfs*7
truncated mutant, identified as a germline mutation from a patient with LFS who developed breast cancer. Functional assays revealed a partial loss-of-function across key cellular processes, including proliferation, cell death, cell motility, and transcriptional transactivation. This mutant lacked a dominant-negative effect, distinguishing it from common DNA-binding domain missense mutations. Our findings demonstrate that oncogenesis in LFS can be driven by partial impairment of functional p53, rather than dominant-negative or gain-of-function mutations alone. This underscores the clinical significance of recognising subtle
TP53
variants for the refined molecular classification and clinical prediction of cancer risk in
TP53
mutation carriers.
The role of the PI3K/AKT pathway in the PIK3CA-related overgrowth is clarified, with one pediatric patient carrying a novel mosaic de novo in-frame deletion mutation in the PIK3CA gene identified.
Jing-Heng Wu, Shan-Lin Chen, Yun-Hao Xue et al.· Yi chuan = Hereditas· 0 citations
Functional evidence is provided to fine-tune the classification of these SAMD9L variants and significantly advance the understanding of the molecular mechanisms by which SAMD9L variants drive inherited myeloid neoplasms.
E. Vuelta, A. Liquori, M. Morín et al.· British Journal of Haematolo...· 0 citations
No major functional difference was detected between PAX8-WT and L264P under the tested conditions; however, these findings do not establish functional equivalence or exclude smaller, tissue-specific, or developmental-stage-specific effects.
Lin He, Liang-Zhe Li, Yu-Xiao Li et al.· Genes· 0 citations
ASXL3 patient truncations in neurodevelopmental condition Bainbridge-Ropers syndrome are shown to mediates gain-of-function (GOF) by escaping nonsense-mediated decay and Cullin 4-dependent degradation, resulting in aberrant protein accumulation, widespread transcriptional dysregulation, and altered chromatin accessibil...
Y. Nakamura, T. Nguyen, N. Mor et al.· medRxiv· 0 citations
It is shown that cancer-associated DICER1 mutations shift microRNA processing and reactivate HERVH endogenous retroviruses and cause upregulation of transposable elements, including the human endogenous retrovirus H through miRNA-independent mechanisms.
K. Gordon, N. Bellora, Jeroen Witteveldt et al.· Nature Communications· 0 citations
Complementary strategies, including covalent stabilization and targeted degradation, further extend this framework, establishing mutation-encoded structural vulnerabilities as actionable therapeutic opportunities, in which oncogenic missense mutations can generate druggable neo-pockets.
Lin-Wan Song, Hua Wang· Medical Review· 0 citations
We use cookies to run the site and, with your consent, for analytics and to show ads.
See our Cookie Policy.