Regional Differences in the Frequency of BRCA1 and BRCA2 Variants in Northeastern Japan: A Cohort Study

ABSTRACT Background Germline mutations in BRCA1/2 are known to cause hereditary tumors in the breast, ovary, and other organs. With the widespread adoption of comprehensive diagnostics, including comprehensive genomic profiling (CGP) tests for solid tumors, many patients with BRCA1/2 variants have b...

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Main Authors: Hidekazu Shirota, Akimitsu Miyake, Maako Kawamura, Shuhei Suzuki, Kensuke Saito, Jun Yasuda, Hiroyuki Shibata, Motonobu Saito, Takeshi Iwaya, Hiroshi Tada, Muneaki Shimada, Naoki Kawamorita, Masayuki Kanamori, Eisaku Miyauchi, Hidetaka Niizuma, Tomoyuki Iwasaki, Yuki Kasahara, Hiroo Imai, Ken Saijo, Keigo Komine, Masanobu Takahashi, Tetsuya Niihori, Yoko Aoki, Toru Furukawa, Gen Tamiya, Chikashi Ishioka
Format: Article
Language:English
Published: Wiley 2025-04-01
Series:Cancer Medicine
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Online Access:https://doi.org/10.1002/cam4.70443
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author Hidekazu Shirota
Akimitsu Miyake
Maako Kawamura
Shuhei Suzuki
Kensuke Saito
Jun Yasuda
Hiroyuki Shibata
Motonobu Saito
Takeshi Iwaya
Hiroshi Tada
Muneaki Shimada
Naoki Kawamorita
Masayuki Kanamori
Eisaku Miyauchi
Hidetaka Niizuma
Tomoyuki Iwasaki
Yuki Kasahara
Hiroo Imai
Ken Saijo
Keigo Komine
Masanobu Takahashi
Tetsuya Niihori
Yoko Aoki
Toru Furukawa
Gen Tamiya
Chikashi Ishioka
author_facet Hidekazu Shirota
Akimitsu Miyake
Maako Kawamura
Shuhei Suzuki
Kensuke Saito
Jun Yasuda
Hiroyuki Shibata
Motonobu Saito
Takeshi Iwaya
Hiroshi Tada
Muneaki Shimada
Naoki Kawamorita
Masayuki Kanamori
Eisaku Miyauchi
Hidetaka Niizuma
Tomoyuki Iwasaki
Yuki Kasahara
Hiroo Imai
Ken Saijo
Keigo Komine
Masanobu Takahashi
Tetsuya Niihori
Yoko Aoki
Toru Furukawa
Gen Tamiya
Chikashi Ishioka
author_sort Hidekazu Shirota
collection DOAJ
description ABSTRACT Background Germline mutations in BRCA1/2 are known to cause hereditary tumors in the breast, ovary, and other organs. With the widespread adoption of comprehensive diagnostics, including comprehensive genomic profiling (CGP) tests for solid tumors, many patients with BRCA1/2 variants have been identified. Methods In this study, we extracted and analyzed cases of BRCA1/2 variants that were presumed to be germline, which were repeatedly detected using the CGP test for solid tumors in northeastern Japan. The frequencies of BRCA1/2 variants in regional areas were compared with those of healthy individuals or nationwide cancer cohorts to investigate regional distribution. Results Our findings revealed regional disparities in BRCA1/2 pathogenic germline variants, while variants of unknown significance (VUS) showed no such differences. The regional distribution of BRCA1 and BRCA2 variants showed distinct patterns: pathogenic variants of BRCA1 exhibited regional differences and were less prevalent compared to VUS, whereas BRCA2 variants, including both pathogenic variants and VUS, did not exhibit such clear regional localization. This discrepancy in regional distribution between BRCA1 and BRCA2 variants could be attributed to factors such as the diversity of the genome, gender differences, and cancer types. Conclusions These results highlight the importance of considering regional differences in comparative cohort studies, particularly in assessing the differential extension of mutations in pathogenic changes and VUS. Moreover, a presumption of pathogenicity variants would need to be discussed at the regional level.
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spelling doaj-art-30b130dee5d24e50adb920de018adf502025-08-20T03:15:05ZengWileyCancer Medicine2045-76342025-04-01148n/an/a10.1002/cam4.70443Regional Differences in the Frequency of BRCA1 and BRCA2 Variants in Northeastern Japan: A Cohort StudyHidekazu Shirota0Akimitsu Miyake1Maako Kawamura2Shuhei Suzuki3Kensuke Saito4Jun Yasuda5Hiroyuki Shibata6Motonobu Saito7Takeshi Iwaya8Hiroshi Tada9Muneaki Shimada10Naoki Kawamorita11Masayuki Kanamori12Eisaku Miyauchi13Hidetaka Niizuma14Tomoyuki Iwasaki15Yuki Kasahara16Hiroo Imai17Ken Saijo18Keigo Komine19Masanobu Takahashi20Tetsuya Niihori21Yoko Aoki22Toru Furukawa23Gen Tamiya24Chikashi Ishioka25Department of Medical Oncology Tohoku University Hospital Sendai JapanTohoku Medical Megabank Organization Tohoku University Sendai JapanPersonalized Medicine Center Tohoku University Hospital Sendai JapanDepartment of Clinical Oncology Yamagata University Faculty of Medicine Yamagata JapanDepartment of Medical Oncology Hirosaki University Graduate School of Medicine Hirosaki JapanDivision of Molecular Cellular Oncology Miyagi Cancer Center Research Institute Natori JapanDepartment of Clinical Oncology, Graduate School of Medicine Akita University Akita JapanDepartment of Gastrointestinal Tract Surgery Fukushima Medical University School of Medicine Fukushima JapanDepartment of Clinical Oncology Iwate Medical University School of Medicine Morioka JapanDepartment of Breast and Endocrine Surgical Oncology Tohoku University Graduate School of Medicine Sendai JapanDepartment of Obstetrics and Gynecology Tohoku University School of Medicine Sendai JapanDepartment of Urology Tohoku University School of Medicine Sendai JapanDepartment of Neurosurgery Tohoku University School of Medicine Sendai JapanDepartment of Respiratory Medicine Tohoku University Graduate School of Medicine Sendai JapanDepartment of Pediatrics Tohoku University School of Medicine Sendai JapanDepartment of Medical Oncology Tohoku University Hospital Sendai JapanDepartment of Medical Oncology Tohoku University Hospital Sendai JapanDepartment of Medical Oncology Tohoku University Hospital Sendai JapanDepartment of Medical Oncology Tohoku University Hospital Sendai JapanDepartment of Medical Oncology Tohoku University Hospital Sendai JapanDepartment of Medical Oncology Tohoku University Hospital Sendai JapanDepartment of Medical Genetics Tohoku University Graduate School of Medicine Sendai JapanDepartment of Medical Genetics Tohoku University Graduate School of Medicine Sendai JapanDepartment of Investigative Pathology Tohoku University Graduate School of Medicine Sendai JapanTohoku Medical Megabank Organization Tohoku University Sendai JapanDepartment of Medical Oncology Tohoku University Hospital Sendai JapanABSTRACT Background Germline mutations in BRCA1/2 are known to cause hereditary tumors in the breast, ovary, and other organs. With the widespread adoption of comprehensive diagnostics, including comprehensive genomic profiling (CGP) tests for solid tumors, many patients with BRCA1/2 variants have been identified. Methods In this study, we extracted and analyzed cases of BRCA1/2 variants that were presumed to be germline, which were repeatedly detected using the CGP test for solid tumors in northeastern Japan. The frequencies of BRCA1/2 variants in regional areas were compared with those of healthy individuals or nationwide cancer cohorts to investigate regional distribution. Results Our findings revealed regional disparities in BRCA1/2 pathogenic germline variants, while variants of unknown significance (VUS) showed no such differences. The regional distribution of BRCA1 and BRCA2 variants showed distinct patterns: pathogenic variants of BRCA1 exhibited regional differences and were less prevalent compared to VUS, whereas BRCA2 variants, including both pathogenic variants and VUS, did not exhibit such clear regional localization. This discrepancy in regional distribution between BRCA1 and BRCA2 variants could be attributed to factors such as the diversity of the genome, gender differences, and cancer types. Conclusions These results highlight the importance of considering regional differences in comparative cohort studies, particularly in assessing the differential extension of mutations in pathogenic changes and VUS. Moreover, a presumption of pathogenicity variants would need to be discussed at the regional level.https://doi.org/10.1002/cam4.70443BRCA1BRCA2CGP testgeographical distributionsolid tumor
spellingShingle Hidekazu Shirota
Akimitsu Miyake
Maako Kawamura
Shuhei Suzuki
Kensuke Saito
Jun Yasuda
Hiroyuki Shibata
Motonobu Saito
Takeshi Iwaya
Hiroshi Tada
Muneaki Shimada
Naoki Kawamorita
Masayuki Kanamori
Eisaku Miyauchi
Hidetaka Niizuma
Tomoyuki Iwasaki
Yuki Kasahara
Hiroo Imai
Ken Saijo
Keigo Komine
Masanobu Takahashi
Tetsuya Niihori
Yoko Aoki
Toru Furukawa
Gen Tamiya
Chikashi Ishioka
Regional Differences in the Frequency of BRCA1 and BRCA2 Variants in Northeastern Japan: A Cohort Study
Cancer Medicine
BRCA1
BRCA2
CGP test
geographical distribution
solid tumor
title Regional Differences in the Frequency of BRCA1 and BRCA2 Variants in Northeastern Japan: A Cohort Study
title_full Regional Differences in the Frequency of BRCA1 and BRCA2 Variants in Northeastern Japan: A Cohort Study
title_fullStr Regional Differences in the Frequency of BRCA1 and BRCA2 Variants in Northeastern Japan: A Cohort Study
title_full_unstemmed Regional Differences in the Frequency of BRCA1 and BRCA2 Variants in Northeastern Japan: A Cohort Study
title_short Regional Differences in the Frequency of BRCA1 and BRCA2 Variants in Northeastern Japan: A Cohort Study
title_sort regional differences in the frequency of brca1 and brca2 variants in northeastern japan a cohort study
topic BRCA1
BRCA2
CGP test
geographical distribution
solid tumor
url https://doi.org/10.1002/cam4.70443
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