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Germline variant in REXO2 is a novel candidate gene in familial pheochromocytoma

Pheochromocytoma (PCC) is a rare, mostly benign tumour of the adrenal medulla. Hereditary PCC accounts for ~35% of cases and has been associated with germline mutations in several cancer susceptibility genes (e.g., KIF1B, SDHB, VHL, SDHD, RET). We performed whole-exome sequencing in a family with fo...

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Autores principales: Laitman, Yael, Tzur, Shay, Attai, Ruben, Tirosh, Amit, Friedman, Eitan
Formato: Online Artículo Texto
Lenguaje:English
Publicado: Cambridge University Press 2020
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC7214532/
https://www.ncbi.nlm.nih.gov/pubmed/32354376
http://dx.doi.org/10.1017/S0016672320000038
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author Laitman, Yael
Tzur, Shay
Attai, Ruben
Tirosh, Amit
Friedman, Eitan
author_facet Laitman, Yael
Tzur, Shay
Attai, Ruben
Tirosh, Amit
Friedman, Eitan
author_sort Laitman, Yael
collection PubMed
description Pheochromocytoma (PCC) is a rare, mostly benign tumour of the adrenal medulla. Hereditary PCC accounts for ~35% of cases and has been associated with germline mutations in several cancer susceptibility genes (e.g., KIF1B, SDHB, VHL, SDHD, RET). We performed whole-exome sequencing in a family with four PCC-affected patients in two consecutive generations and identified a potential novel candidate pathogenic variant in the REXO2 gene that affects splicing (c.531-1G>T (NM 015523.3)), which co-segregated with the phenotype in the family. REXO2 encodes for RNA exonuclease 2 protein and localizes to 11q23, a chromosomal region displaying allelic imbalance in PCC. REXO2 protein has been associated with DNA repair, replication and recombination processes and thus its inactivation may contribute to tumorigenesis. While the study suggests that this novel REXO2 gene variant underlies PCC in this family, additional functional studies are required in order to establish the putative role of the REXO2 gene in PCC predisposition.
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spelling pubmed-72145322020-05-18 Germline variant in REXO2 is a novel candidate gene in familial pheochromocytoma Laitman, Yael Tzur, Shay Attai, Ruben Tirosh, Amit Friedman, Eitan Genet Res (Camb) Research Paper Pheochromocytoma (PCC) is a rare, mostly benign tumour of the adrenal medulla. Hereditary PCC accounts for ~35% of cases and has been associated with germline mutations in several cancer susceptibility genes (e.g., KIF1B, SDHB, VHL, SDHD, RET). We performed whole-exome sequencing in a family with four PCC-affected patients in two consecutive generations and identified a potential novel candidate pathogenic variant in the REXO2 gene that affects splicing (c.531-1G>T (NM 015523.3)), which co-segregated with the phenotype in the family. REXO2 encodes for RNA exonuclease 2 protein and localizes to 11q23, a chromosomal region displaying allelic imbalance in PCC. REXO2 protein has been associated with DNA repair, replication and recombination processes and thus its inactivation may contribute to tumorigenesis. While the study suggests that this novel REXO2 gene variant underlies PCC in this family, additional functional studies are required in order to establish the putative role of the REXO2 gene in PCC predisposition. Cambridge University Press 2020-05-01 /pmc/articles/PMC7214532/ /pubmed/32354376 http://dx.doi.org/10.1017/S0016672320000038 Text en © The Author(s) 2020 http://creativecommons.org/licenses/by/4.0/ http://creativecommons.org/licenses/by/4.0/This is an Open Access article, distributed under the terms of the Creative Commons Attribution licence (http://creativecommons.org/licenses/by/4.0/), which permits unrestricted re-use, distribution, and reproduction in any medium, provided the original work is properly cited.
spellingShingle Research Paper
Laitman, Yael
Tzur, Shay
Attai, Ruben
Tirosh, Amit
Friedman, Eitan
Germline variant in REXO2 is a novel candidate gene in familial pheochromocytoma
title Germline variant in REXO2 is a novel candidate gene in familial pheochromocytoma
title_full Germline variant in REXO2 is a novel candidate gene in familial pheochromocytoma
title_fullStr Germline variant in REXO2 is a novel candidate gene in familial pheochromocytoma
title_full_unstemmed Germline variant in REXO2 is a novel candidate gene in familial pheochromocytoma
title_short Germline variant in REXO2 is a novel candidate gene in familial pheochromocytoma
title_sort germline variant in rexo2 is a novel candidate gene in familial pheochromocytoma
topic Research Paper
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC7214532/
https://www.ncbi.nlm.nih.gov/pubmed/32354376
http://dx.doi.org/10.1017/S0016672320000038
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