Inhibin A and inhibin B producing ovarian fibrothecoma revealed by suppression of follicle stimulating hormone (FSH) in a post-menopausal woman: report of the first case.

In this report, we describe the first case ever reported in the literature, of an inhibin-A (INHA) and inhibin-B (INHB) producing fibrothecoma. A post-menopausal woman was referred to our unit because of follicle stimulating hormone (FSH) level below the reference interval for postmenopausal women. By contrast luteinizing hormone, hCG, and estradiol levels were within normal range. This discrepancy suggested the secretion of FSH inhibitory factors. INHB and INHA levels were markedly elevated for age, 475 pg/mL and 100 pg/mL, respectively. Ultrasonography and MRI showed a pelvic mass of indeterminate nature. Abnormal inhibin secretion is generally observed in granulosa cell tumors. In this case this etiology was unlikely because of low estradiol and AMH levels. Surgical exploration revealed a 10 cm mass of the left ovary proven histologically to be an ovarian fibrothecoma (OFT). After tumor removal, INHB and INHA levels decreased rapidly. Only three cases of OFT with an important secretion of INHB have been reported to date. INHA secretion has never been associated with OFT. There is a need to develop coupled hormone and imaging strategies to diagnose the source of INH secretion in case of FSH/LH discrepancy.

Time to revise classification of phyllodes tumors of breast? Results of a French multicentric study.

OBJECTIVE: To assess prognostic factors of recurrence of phyllodes tumors (PT) of the breast. METHODS: We performed a retrospective, multicentric cohort study, including all patients who underwent breast surgery for grade 1 (benign), 2 (borderline) or 3 (malignant) PT between 2000 and 2016 in five tertiary University hospitals, diagnosed according to World Health Organisation classification. RESULTS: 230 patients were included: 144 (63%), 60 (26%) and 26 (11%) with grade 1, 2 and 3 PT, respectively. Recurrence occurred in 10 (7%), 7 (12%) and 5 (19%) patients with grade 1, 2 and 3 PT, respectively. In univariate analysis, moderate to severe nuclear stromal pleomorphism (HR 8.00 [95% CI: 1.65-38.73], p < 0.009) was correlated with recurrence in all groups including grade 1 (HR 14.3 [95% CI: 1.29-160], p = 0.031). In multivariate analysis, surgical margin >5 mm, (HR 0.20 [95% CI: 0.06-0.63], p = 0.013) were significantly correlated with less recurrence in all PT grades. For grade 1 PT, there was also significantly less recurrence with surgical margin >5 mm, (HR 0.09 [95% CI: 0.01-0.85], p = 0.047) in multivariate analysis. CONCLUSION: The surgical margin should be at least 5 mm whatever the grade of PT. Moderate to severe nuclear stromal pleomorphism identified a subgroup of grade 1 PT with a higher rate of recurrence. This suggests that the WHO classification could be revised with the introduction of nuclear stromal pleomorphism to tailor PT management.