Early Colorectal Cancers Provide New Evidence for a Lynch Syndrome-to-CMMRD Phenotypic Continuum.

CMMRD Lynch syndrome genetic modifiers phenotypic continuum whole-exome sequencing

Journal

Cancers
ISSN: 2072-6694
Titre abrégé: Cancers (Basel)
Pays: Switzerland
ID NLM: 101526829

Informations de publication

Date de publication:
30 Jul 2019
Historique:
received: 11 07 2019
revised: 18 07 2019
accepted: 25 07 2019
entrez: 2 8 2019
pubmed: 2 8 2019
medline: 2 8 2019
Statut: epublish

Résumé

Lynch syndrome (LS) is the most common hereditary colorectal cancer (CRC) syndrome, caused by heterozygous mutations in the mismatch repair (MMR) genes. Biallelic mutations in these genes lead however, to constitutive mismatch repair deficiency (CMMRD). In this study, we follow the diagnostic journey of a 12-year old patient with CRC, with a clinical phenotype overlapping CMMRD. We perform molecular and functional assays to discard a CMMRD diagnosis then identify by exome sequencing and validation in a cohort of 134 LS patients, a candidate variant in the

Identifiants

pubmed: 31366136
pii: cancers11081081
doi: 10.3390/cancers11081081
pmc: PMC6721314
pii:
doi:

Types de publication

Journal Article

Langues

eng

Subventions

Organisme : Spanish National Centre for Genomic Analysis
ID : 300-exomes to elucidate Rare-Diseases

Déclaration de conflit d'intérêts

The authors declare no conflict of interest.

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Auteurs

Ceres Fernandez-Rozadilla (C)

Fundación Pública Galega de Medicina Xenómica SERGAS, Grupo de Medicina Xenómica_USC, IDIS, 15706 Santiago de Compostela, Spain.

Miriam Alvarez-Barona (M)

Fundación Pública Galega de Medicina Xenómica SERGAS, Grupo de Medicina Xenómica_USC, IDIS, 15706 Santiago de Compostela, Spain.

Esther Schamschula (E)

Division of Human Genetics, Medical University Innsbruck, 6020 Innsbruck, Austria.
Department of Biosciences, Paris-Lodron University of Salzburg, 5020 Salzburg, Austria.

Sahra Bodo (S)

Sorbonne Université, Inserm, Centre de Recherche Saint-Antoine, CRSA, 75571 Paris, France.

Anael Lopez-Novo (A)

Fundación Pública Galega de Medicina Xenómica SERGAS, Grupo de Medicina Xenómica_USC, IDIS, 15706 Santiago de Compostela, Spain.

Andres Dacal (A)

Servicio de Gastroenterología, Hospital Universitario Lucus Augusti, IDIS, 27003 Lugo, Spain.

Consuelo Calviño-Costas (C)

Servicio de Pediatría, Hospital Universitario Lucus Augusti, 27003 Lugo, Spain.

Angel Lancho (A)

Servicio de Gastroenterología, Hospital Universitario Lucus Augusti, IDIS, 27003 Lugo, Spain.

Jorge Amigo (J)

Fundación Pública Galega de Medicina Xenómica SERGAS, Grupo de Medicina Xenómica_USC, IDIS, 15706 Santiago de Compostela, Spain.

Xabier Bello (X)

Fundación Pública Galega de Medicina Xenómica SERGAS, Grupo de Medicina Xenómica_USC, IDIS, 15706 Santiago de Compostela, Spain.

Jose Manuel Cameselle-Teijeiro (JM)

Servicio de Anatomía Patológica, Hospital Clínico Universitario, USC, 15706 Santiago de Compostela, Spain.

Angel Carracedo (A)

Fundación Pública Galega de Medicina Xenómica SERGAS, Grupo de Medicina Xenómica_USC, IDIS, 15706 Santiago de Compostela, Spain.
Grupo de Medicina Xenómica, Centro de Investigación Biomédica en Red de Enfermedades Raras (CIBERER), Universidade de Santiago de Compostela, 15782, Santiago de Compostela, Spain.

Chrystelle Colas (C)

Department of Genetics Institut Curie, Centre de Recherche Saint-Antoine, Sorbonne Université, 75571 Paris, France.

Martine Muleris (M)

Sorbonne Université, Inserm, Centre de Recherche Saint-Antoine, CRSA, 75571 Paris, France.

Katharina Wimmer (K)

Division of Human Genetics, Medical University Innsbruck, 6020 Innsbruck, Austria.

Clara Ruiz-Ponte (C)

Fundación Pública Galega de Medicina Xenómica SERGAS, Grupo de Medicina Xenómica_USC, IDIS, 15706 Santiago de Compostela, Spain. clara.ruiz.ponte@usc.es.
Grupo de Medicina Xenómica, Centro de Investigación Biomédica en Red de Enfermedades Raras (CIBERER), Universidade de Santiago de Compostela, 15782, Santiago de Compostela, Spain. clara.ruiz.ponte@usc.es.

Classifications MeSH