Saara Bryk1, Anniina Färkkilä2, Ralf Bützow3, Arto Leminen4, Johanna Tapper4, Markku Heikinheimo5, Leila Unkila-Kallio4, Annika Riska4. 1. Obstetrics and Gynecology, University of Helsinki and Helsinki University Hospital, Finland; Children's Hospital, University of Helsinki and Helsinki University Hospital, Finland. Electronic address: saara.bryk@hus.fi. 2. Obstetrics and Gynecology, University of Helsinki and Helsinki University Hospital, Finland; Children's Hospital, University of Helsinki and Helsinki University Hospital, Finland. 3. Pathology and HUSLAB, University of Helsinki and Helsinki University Hospital, Finland. 4. Obstetrics and Gynecology, University of Helsinki and Helsinki University Hospital, Finland. 5. Children's Hospital, University of Helsinki and Helsinki University Hospital, Finland; Department of Pediatrics, Washington University School of Medicine, St Louis Children's Hospital, St Louis, MO, United States.
Abstract
OBJECTIVE: Adult-type ovarian granulosa cell tumors (AGCTs) have an unpredictable tendency to relapse. In a carefully validated patient cohort, we evaluated the prognostic factors related to AGCT recurrence. METHODS: We identified all patients diagnosed with AGCT during 1956-2014 in Helsinki University Hospital, with a minimum follow-up of one year (n=240). After a histological review supplemented with FOXL2 (402C-G) mutation status analysis, we analyzed the clinical data for association with relapse. RESULTS: The final cohort included 164 (68%) molecularly defined AGCTs (MD-AGCTs). The majority of the women were postmenopausal (63%), and 92% of tumors were stage I. The median follow-up time was 15.5years. Fifty-two (32%) patients developed tumor recurrence, of whom 55% had successive recurrences. Multiple-site recurrences were common, and nearly half of the recurrences were asymptomatic. The median time to the first relapse was 7.4years, and 75% of relapses occurred within ten years after primary diagnosis. The median disease-free survival was 11.3years. Premenopausal status at initial diagnosis, FIGO stage Ic versus Ia, and tumor rupture associated with relapse. However, tumor rupture was the only independent predictive factor. Of the relapsed patients, 48% died of AGCT in a median time of 15.3years. CONCLUSION: Tumor rupture is the strongest predictive factor for recurrence, and these patients might benefit from a more aggressive initial treatment approach. AGCT requires active follow up for 10 to 15years after primary diagnosis, since recurrences may develop late, asymptomatically and in multiple anatomical locations.
OBJECTIVE:Adult-type ovarian granulosa cell tumors (AGCTs) have an unpredictable tendency to relapse. In a carefully validated patient cohort, we evaluated the prognostic factors related to AGCT recurrence. METHODS: We identified all patients diagnosed with AGCT during 1956-2014 in Helsinki University Hospital, with a minimum follow-up of one year (n=240). After a histological review supplemented with FOXL2 (402C-G) mutation status analysis, we analyzed the clinical data for association with relapse. RESULTS: The final cohort included 164 (68%) molecularly defined AGCTs (MD-AGCTs). The majority of the women were postmenopausal (63%), and 92% of tumors were stage I. The median follow-up time was 15.5years. Fifty-two (32%) patients developed tumor recurrence, of whom 55% had successive recurrences. Multiple-site recurrences were common, and nearly half of the recurrences were asymptomatic. The median time to the first relapse was 7.4years, and 75% of relapses occurred within ten years after primary diagnosis. The median disease-free survival was 11.3years. Premenopausal status at initial diagnosis, FIGO stage Ic versus Ia, and tumor rupture associated with relapse. However, tumor rupture was the only independent predictive factor. Of the relapsed patients, 48% died of AGCT in a median time of 15.3years. CONCLUSION:Tumor rupture is the strongest predictive factor for recurrence, and these patients might benefit from a more aggressive initial treatment approach. AGCT requires active follow up for 10 to 15years after primary diagnosis, since recurrences may develop late, asymptomatically and in multiple anatomical locations.
Authors: R Tyler Hillman; Joseph Celestino; Christopher Terranova; Hannah C Beird; Curtis Gumbs; Latasha Little; Tri Nguyen; Rebecca Thornton; Samantha Tippen; Jianhua Zhang; Karen H Lu; David M Gershenson; Kunal Rai; Russell R Broaddus; P Andrew Futreal Journal: Nat Commun Date: 2018-06-27 Impact factor: 14.919
Authors: Ulla-Maija Haltia; Marjut Pihlajoki; Noora Andersson; Lotta Mäkinen; Johanna Tapper; Alejandra Cervera; Hugo M Horlings; Ursula Turpeinen; Mikko Anttonen; Ralf Bützow; Leila Unkila-Kallio; Olli Carpén; David B Wilson; Markku Heikinheimo; Anniina Färkkilä Journal: J Endocr Soc Date: 2020-03-16
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