Characterization of VHL promoter variants in patients suspected of Von Hippel-Lindau disease.


Journal

European journal of medical genetics
ISSN: 1878-0849
Titre abrégé: Eur J Med Genet
Pays: Netherlands
ID NLM: 101247089

Informations de publication

Date de publication:
Mar 2019
Historique:
received: 29 12 2017
revised: 10 06 2018
accepted: 09 07 2018
pubmed: 15 7 2018
medline: 9 3 2019
entrez: 15 7 2018
Statut: ppublish

Résumé

Von Hippel-Lindau (VHL) disease is a hereditary tumor syndrome in which carriers are at an increased risk of developing a variety of tumors in multiple organ systems. A clinical diagnosis of VHL is determined by the presence of specific clinical manifestations while a molecular genetic diagnosis results from a pathogenic variant in the VHL gene. The majority of mutations occur in VHL coding exons and DNA analysis of these regions has a reported sensitivity of nearly 100%. However, rare variants in the VHL gene promoter may be detected in some cases of suspected VHL disease. We report two cases where VHL promoter variants were detected and describe the role of multi-step mRNA and protein analysis in the diagnostic evaluation of these cases.

Identifiants

pubmed: 30006056
pii: S1769-7212(17)30880-7
doi: 10.1016/j.ejmg.2018.07.006
pii:
doi:

Substances chimiques

RNA, Messenger 0
Von Hippel-Lindau Tumor Suppressor Protein EC 2.3.2.27
VHL protein, human EC 6.3.2.-

Types de publication

Case Reports Journal Article

Langues

eng

Sous-ensembles de citation

IM

Pagination

177-181

Informations de copyright

Copyright © 2018 Elsevier Masson SAS. All rights reserved.

Auteurs

Saleh Albanyan (S)

Division of Clinical and Metabolic Genetics, Department of Pediatrics, University of Toronto, The Hospital for Sick Children, Toronto, Canada.

Rachel H Giles (RH)

Department of Nephrology and Hypertension, Regenerative Medicine Center Utrecht, University Medical Center Utrecht, The Netherlands.

Enric Mocholi Gimeno (EM)

Department of Cell Biology, Regenerative Medicine Center Utrecht, University Medical Center Utrecht, The Netherlands.

Josh Silver (J)

Fred A Litwin Family Centre in Genetic Medicine, University Health Network, Canada; Department of Molecular Genetics, University of Toronto, Toronto, Canada.

Jillian Murphy (J)

Fred A Litwin Family Centre in Genetic Medicine, University Health Network, Canada; Department of Molecular Genetics, University of Toronto, Toronto, Canada.

Hanna Faghfoury (H)

Fred A Litwin Family Centre in Genetic Medicine, University Health Network, Canada; Division of Medical Oncology and Hematology, Department of Medicine, University of Toronto, Toronto, Canada.

Chantal F Morel (CF)

Fred A Litwin Family Centre in Genetic Medicine, University Health Network, Canada; Division of Medical Oncology and Hematology, Department of Medicine, University of Toronto, Toronto, Canada.

Jerry Machado (J)

Prevention Genetics, Marshfield, WI, USA.

Raymond H Kim (RH)

Fred A Litwin Family Centre in Genetic Medicine, University Health Network, Canada; Division of Medical Oncology and Hematology, Department of Medicine, University of Toronto, Toronto, Canada. Electronic address: raymond.kim@utoronto.ca.

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Classifications MeSH