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Whole-exome analysis of a Li-Fraumeni family trio with a novel TP53 PRD mutation and anticipation profile.
Franceschi, Sara; Spugnesi, Laura; Aretini, Paolo; Lessi, Francesca; Scarpitta, Rosa; Galli, Alvaro; Congregati, Caterina; Caligo, Maria Adelaide; Mazzanti, Chiara Maria.
Afiliação
  • Franceschi S; FPS - Fondazione Pisana per la Scienza, Pisa 56121, Italy.
  • Spugnesi L; Section of Genetic Oncology, Department of Laboratory Medicine, University Hospital of Pisa, Pisa 56100, Italy.
  • Aretini P; FPS - Fondazione Pisana per la Scienza, Pisa 56121, Italy.
  • Lessi F; FPS - Fondazione Pisana per la Scienza, Pisa 56121, Italy.
  • Scarpitta R; Section of Genetic Oncology, Department of Laboratory Medicine, University Hospital of Pisa, Pisa 56100, Italy.
  • Galli A; Yeast Genetics and Genomics Group, Laboratory of Functional Genetics and Genomics, Institute of Clinical Physiology CNR, Pisa 56124, Italy.
  • Congregati C; Cytogenetics and Molecular Genetic Unit, University Hospital of Pisa, Pisa 56100, Italy.
  • Caligo MA; UO Medical Genetics, Department of Laboratory Medicine, University Hospital of Pisa, Pisa 56100, Italy.
  • Mazzanti CM; FPS - Fondazione Pisana per la Scienza, Pisa 56121, Italy.
Carcinogenesis ; 38(9): 938-943, 2017 09 01.
Article em En | MEDLINE | ID: mdl-28911001
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.
Assuntos

Texto completo: 1 Base de dados: MEDLINE Assunto principal: Neoplasias da Mama / Proteína Supressora de Tumor p53 / Síndrome de Li-Fraumeni / Antecipação Genética Tipo de estudo: Prognostic_studies Limite: Child, preschool / Female / Humans / Male Idioma: En Ano de publicação: 2017 Tipo de documento: Article

Texto completo: 1 Base de dados: MEDLINE Assunto principal: Neoplasias da Mama / Proteína Supressora de Tumor p53 / Síndrome de Li-Fraumeni / Antecipação Genética Tipo de estudo: Prognostic_studies Limite: Child, preschool / Female / Humans / Male Idioma: En Ano de publicação: 2017 Tipo de documento: Article