A case of adult granulosa cell tumor of the ovary with long-term survival after multiple recurrences

Objective: To illustrate the clinical course of a rare case of recurrent adult granulosa cell tumor (AGCT) and discuss the features and management for recurrences. Case report: A 56-year-old female was first diagnosed with AGCT in 2008 and had uneventful, regular follow-ups until 2013. Recurrence wa...

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Main Authors: Angel Hsin-Yu Pai, Ren-Chin Wu, Feng-Yuan Liu, Chiao-Yun Lin, Yenpo Lin, Chyong-Huey Lai
Format: Article
Language:English
Published: Elsevier 2022-09-01
Series:Taiwanese Journal of Obstetrics & Gynecology
Subjects:
Online Access:http://www.sciencedirect.com/science/article/pii/S102845592200225X
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author Angel Hsin-Yu Pai
Ren-Chin Wu
Feng-Yuan Liu
Chiao-Yun Lin
Yenpo Lin
Chyong-Huey Lai
author_facet Angel Hsin-Yu Pai
Ren-Chin Wu
Feng-Yuan Liu
Chiao-Yun Lin
Yenpo Lin
Chyong-Huey Lai
author_sort Angel Hsin-Yu Pai
collection DOAJ
description Objective: To illustrate the clinical course of a rare case of recurrent adult granulosa cell tumor (AGCT) and discuss the features and management for recurrences. Case report: A 56-year-old female was first diagnosed with AGCT in 2008 and had uneventful, regular follow-ups until 2013. Recurrence was suspected and proven by computed tomography-guided biopsy. After undergoing complete cytoreductive surgery (CRS) followed by adjuvant megestrol acetate then leuprolide acetate, another recurrence sprouted at the presacral area in 2017. On both occasions, CRS with no visible residual tumor were attained. The patient has remained in complete remission to date with progestin therapy. Conclusion: There are currently no standardized tumor markers, imaging exams, or therapies for managing AGCT recurrences. Whole exome sequencing analysis of our patient suggested possible association with triosephosphate isomerase 1 mutation. Regular follow-ups with at least two types of imaging exams and indefinite hormone therapy are crucial for this patient's remission.
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spelling doaj.art-09de4ae4e8cd4163b0832641e7ea54df2022-12-22T03:12:15ZengElsevierTaiwanese Journal of Obstetrics & Gynecology1028-45592022-09-01615883888A case of adult granulosa cell tumor of the ovary with long-term survival after multiple recurrencesAngel Hsin-Yu Pai0Ren-Chin Wu1Feng-Yuan Liu2Chiao-Yun Lin3Yenpo Lin4Chyong-Huey Lai5Department of Obstetrics and Gynecology, Chang Gung Memorial Hospital, Linkou Branch, and Chang Gung University College of Medicine, Taoyuan, TaiwanDepartment of Pathology, Chang Gung Memorial Hospital, Linkou Branch, and Chang Gung University College of Medicine, Taoyuan, Taiwan; Gynecologic Cancer Research Center, Chang Gung Memorial Hospital, TaiwanDepartment of Nuclear Medicine, Chang Gung Memorial Hospital, Linkou Branch, and Chang Gung University College of Medicine, Taoyuan, Taiwan; Gynecologic Cancer Research Center, Chang Gung Memorial Hospital, TaiwanGynecologic Cancer Research Center, Chang Gung Memorial Hospital, TaiwanGynecologic Cancer Research Center, Chang Gung Memorial Hospital, Taiwan; Department of Medical Imaging and Intervention, Clinical Metabolomics Core Laboratory, Chang Gung Memorial Hospital, Linkou Branch, and Chang Gung University College of Medicine, Taoyuan, TaiwanDepartment of Obstetrics and Gynecology, Chang Gung Memorial Hospital, Linkou Branch, and Chang Gung University College of Medicine, Taoyuan, Taiwan; Gynecologic Cancer Research Center, Chang Gung Memorial Hospital, Taiwan; Corresponding author. Department of Obstetrics and Gynecology, Chang Gung Memorial Hospital, Linkou Branch, 5, Fu-Shin St, Kueishan, Tao-Yuan, 33305, Taiwan.Objective: To illustrate the clinical course of a rare case of recurrent adult granulosa cell tumor (AGCT) and discuss the features and management for recurrences. Case report: A 56-year-old female was first diagnosed with AGCT in 2008 and had uneventful, regular follow-ups until 2013. Recurrence was suspected and proven by computed tomography-guided biopsy. After undergoing complete cytoreductive surgery (CRS) followed by adjuvant megestrol acetate then leuprolide acetate, another recurrence sprouted at the presacral area in 2017. On both occasions, CRS with no visible residual tumor were attained. The patient has remained in complete remission to date with progestin therapy. Conclusion: There are currently no standardized tumor markers, imaging exams, or therapies for managing AGCT recurrences. Whole exome sequencing analysis of our patient suggested possible association with triosephosphate isomerase 1 mutation. Regular follow-ups with at least two types of imaging exams and indefinite hormone therapy are crucial for this patient's remission.http://www.sciencedirect.com/science/article/pii/S102845592200225XAdult granulosa cell tumorComplete cytoreductive surgeryMegestrol acetatePositron emission tomographyRecurrence
spellingShingle Angel Hsin-Yu Pai
Ren-Chin Wu
Feng-Yuan Liu
Chiao-Yun Lin
Yenpo Lin
Chyong-Huey Lai
A case of adult granulosa cell tumor of the ovary with long-term survival after multiple recurrences
Taiwanese Journal of Obstetrics & Gynecology
Adult granulosa cell tumor
Complete cytoreductive surgery
Megestrol acetate
Positron emission tomography
Recurrence
title A case of adult granulosa cell tumor of the ovary with long-term survival after multiple recurrences
title_full A case of adult granulosa cell tumor of the ovary with long-term survival after multiple recurrences
title_fullStr A case of adult granulosa cell tumor of the ovary with long-term survival after multiple recurrences
title_full_unstemmed A case of adult granulosa cell tumor of the ovary with long-term survival after multiple recurrences
title_short A case of adult granulosa cell tumor of the ovary with long-term survival after multiple recurrences
title_sort case of adult granulosa cell tumor of the ovary with long term survival after multiple recurrences
topic Adult granulosa cell tumor
Complete cytoreductive surgery
Megestrol acetate
Positron emission tomography
Recurrence
url http://www.sciencedirect.com/science/article/pii/S102845592200225X
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