Literature DB >> 28911001

Whole-exome analysis of a Li-Fraumeni family trio with a novel TP53 PRD mutation and anticipation profile.

Sara Franceschi1, Laura Spugnesi2, Paolo Aretini1, Francesca Lessi1, Rosa Scarpitta2, Alvaro Galli3, Caterina Congregati4, Maria Adelaide Caligo5, Chiara Maria Mazzanti1.   

Abstract

Li-Fraumeni syndrome is a clinically heterogeneous familial cancer predisposition syndrome with autosomal-dominant inheritance caused by heterozygous germline mutations in the TP53 gene. We here analyze the genetic background of a family with a 4-year-proband presented with a Li-Fraumeni tumor. The mother developed breast cancer at age 37 and the proband died at age 8. We performed Sanger sequencing and whole-exome sequencing on peripheral blood DNA from proband and relatives. Data analysis selected only high-quality score and depth reads, rare variants and protein impact involving missense, non-sense, frameshift and splice disrupt mutations. Disease implicated variants and predicted deleterious alterations were also chosen. TP53 genetic testing revealed a never reported TP53 deletion arose as de novo mutation in the mother and inherited by the proband. We then performed whole-exome analysis of the trio to uncover inherited variants from the father that potentially worsen the already altered genetic background in the proband. No pathogenic variants were inherited in autosomal recessive, de novo dominant or X-linked recessive manner. Comparing proband and father exome we detected 25 predicted deleterious variants including a nonsense mutation in ERCC3. Those inherited mutations are possible candidate modifiers linked to TP53, explaining the proband accelerated tumor onset compared to the mother and providing a possible explanation of the genetic anticipation event in this Li-Fraumeni family.
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Year:  2017        PMID: 28911001     DOI: 10.1093/carcin/bgx069

Source DB:  PubMed          Journal:  Carcinogenesis        ISSN: 0143-3334            Impact factor:   4.944


  3 in total

Review 1.  A Systematic Literature Review of Whole Exome and Genome Sequencing Population Studies of Genetic Susceptibility to Cancer.

Authors:  Alisa M Goldstein; Elizabeth M Gillanders; Melissa Rotunno; Rolando Barajas; Mindy Clyne; Elise Hoover; Naoko I Simonds; Tram Kim Lam; Leah E Mechanic
Journal:  Cancer Epidemiol Biomarkers Prev       Date:  2020-05-28       Impact factor: 4.254

Review 2.  Next Generation Sequencing and Bioinformatics Analysis of Family Genetic Inheritance.

Authors:  Aquillah M Kanzi; James Emmanuel San; Benjamin Chimukangara; Eduan Wilkinson; Maryam Fish; Veron Ramsuran; Tulio de Oliveira
Journal:  Front Genet       Date:  2020-10-23       Impact factor: 4.599

3.  Li-Fraumeni syndrome in Tunisian carriers with different and rare tumor phenotype: genotype-phenotype correlation.

Authors:  Hela Sassi; Rym Meddeb; Mohamed Aziz Cherif; Chiraz Nasr; Aouatef Riahi; Samia Hannachi; Neila Belguith; Ridha M'rad
Journal:  BMC Med Genomics       Date:  2022-03-04       Impact factor: 3.063

  3 in total

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