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SAT367 A Rare Case of Ovarian Cellular Fibroma Producing Testosterone

Disclosure: H.K. Rai: None. P. Dandona: None. Background: Ovarian fibromas are hormonally inactive, benign sex cord-stromal tumors. However, the occurrence of ovarian fibromas in a woman presenting with post-menopausal bleeding, hirsutism, and elevated testosterone levels raises suspicion of such tu...

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Autores principales: Rai, Harpreet Kaur, Dandona, Paresh
Formato: Online Artículo Texto
Lenguaje:English
Publicado: Oxford University Press 2023
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC10554879/
http://dx.doi.org/10.1210/jendso/bvad114.1672
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author Rai, Harpreet Kaur
Dandona, Paresh
author_facet Rai, Harpreet Kaur
Dandona, Paresh
author_sort Rai, Harpreet Kaur
collection PubMed
description Disclosure: H.K. Rai: None. P. Dandona: None. Background: Ovarian fibromas are hormonally inactive, benign sex cord-stromal tumors. However, the occurrence of ovarian fibromas in a woman presenting with post-menopausal bleeding, hirsutism, and elevated testosterone levels raises suspicion of such tumors being functionally active.Case: 62-year-old menopausal woman presented for evaluation of worsening facial hair for the past year. She reported vaginal spotting for the past two months and was following up with a gynecologist. She denied acne, male pattern baldness, or any other virilizing features. She was also recently diagnosed with polycythemia; hematological workup was remarkable for elevated testosterone. Her blood work was significant for elevated testosterone levels and hemoglobin, results as follows: total testosterone: 160ng/dl (2-45 ng/dL), free testosterone: 25.4pg/ml (0.1-6.4 pg/ml), sex hormone binding globulin (SHBG): 26 nmol/L (14-73 nmol/L), estradiol: <15 pg/ml (post-menopausal range: </=31 pg/ml), FSH: 17.3mIU/ml (23.0-116.3mIU/ml), LH: 19.1mIU/L (10.0-54.7 mIU/mL), prolactin: 5.7 ng/mL (2.0-20.0 ng/mL), DHEAS: 78 mcg/dL (12-133 mcg/dL), 8 AM cortisol: 17 mcg/dL (10-20 mcg/dL), ACTH: 11pg/ml (6-50pg/ml), Hb: 16.6g/dl (11.7-15.5 g/dL) and hematocrit: 49.8% (35.0-45.0%). Ultrasound pelvis showed heterogeneous thickened endometrium, small left-sided intramural leiomyoma of the uterus and ovaries were not well identified. MRI pelvis with and without contrast showed a 2.4 cm left uterine fibroid. A simple cyst was noted in the right ovary and two para-ovarian cysts were noted on the left ovary. The overall appearance of both ovaries was similar to a CT scan done a year earlier. She underwent a total hysterectomy with bilateral salpingo-oophorectomy. Surgical pathology was remarkable for cellular fibroma of both ovaries. After surgery, the patient's facial hair significantly improved, and she was noted to have normal testosterone levels (total testosterone: 23, free testosterone: 2.6, SHBG: 50). Her hemoglobin normalized as well, reaching a value of 13 g/dL. Conclusion: Our patient presented with post-menopausal bleeding, hirsutism, polycythemia, and increased testosterone levels. After she underwent total hysterectomy with bilateral salpingo-oopherectomy, her symptoms resolved. Testosterone and hemoglobulin levels returned to normal. Surprisingly, ovarian cellular fibroma on both sides was identified by surgical pathology. Ovarian cellular fibromas are rarely linked to endocrine activity, but with this case, we want to emphasize that its hormonal activity needs further exploration in the future. Presentation Date: Saturday, June 17, 2023
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spelling pubmed-105548792023-10-06 SAT367 A Rare Case of Ovarian Cellular Fibroma Producing Testosterone Rai, Harpreet Kaur Dandona, Paresh J Endocr Soc Reproductive Endocrinology Disclosure: H.K. Rai: None. P. Dandona: None. Background: Ovarian fibromas are hormonally inactive, benign sex cord-stromal tumors. However, the occurrence of ovarian fibromas in a woman presenting with post-menopausal bleeding, hirsutism, and elevated testosterone levels raises suspicion of such tumors being functionally active.Case: 62-year-old menopausal woman presented for evaluation of worsening facial hair for the past year. She reported vaginal spotting for the past two months and was following up with a gynecologist. She denied acne, male pattern baldness, or any other virilizing features. She was also recently diagnosed with polycythemia; hematological workup was remarkable for elevated testosterone. Her blood work was significant for elevated testosterone levels and hemoglobin, results as follows: total testosterone: 160ng/dl (2-45 ng/dL), free testosterone: 25.4pg/ml (0.1-6.4 pg/ml), sex hormone binding globulin (SHBG): 26 nmol/L (14-73 nmol/L), estradiol: <15 pg/ml (post-menopausal range: </=31 pg/ml), FSH: 17.3mIU/ml (23.0-116.3mIU/ml), LH: 19.1mIU/L (10.0-54.7 mIU/mL), prolactin: 5.7 ng/mL (2.0-20.0 ng/mL), DHEAS: 78 mcg/dL (12-133 mcg/dL), 8 AM cortisol: 17 mcg/dL (10-20 mcg/dL), ACTH: 11pg/ml (6-50pg/ml), Hb: 16.6g/dl (11.7-15.5 g/dL) and hematocrit: 49.8% (35.0-45.0%). Ultrasound pelvis showed heterogeneous thickened endometrium, small left-sided intramural leiomyoma of the uterus and ovaries were not well identified. MRI pelvis with and without contrast showed a 2.4 cm left uterine fibroid. A simple cyst was noted in the right ovary and two para-ovarian cysts were noted on the left ovary. The overall appearance of both ovaries was similar to a CT scan done a year earlier. She underwent a total hysterectomy with bilateral salpingo-oophorectomy. Surgical pathology was remarkable for cellular fibroma of both ovaries. After surgery, the patient's facial hair significantly improved, and she was noted to have normal testosterone levels (total testosterone: 23, free testosterone: 2.6, SHBG: 50). Her hemoglobin normalized as well, reaching a value of 13 g/dL. Conclusion: Our patient presented with post-menopausal bleeding, hirsutism, polycythemia, and increased testosterone levels. After she underwent total hysterectomy with bilateral salpingo-oopherectomy, her symptoms resolved. Testosterone and hemoglobulin levels returned to normal. Surprisingly, ovarian cellular fibroma on both sides was identified by surgical pathology. Ovarian cellular fibromas are rarely linked to endocrine activity, but with this case, we want to emphasize that its hormonal activity needs further exploration in the future. Presentation Date: Saturday, June 17, 2023 Oxford University Press 2023-10-05 /pmc/articles/PMC10554879/ http://dx.doi.org/10.1210/jendso/bvad114.1672 Text en © The Author(s) 2023. Published by Oxford University Press on behalf of the Endocrine Society. https://creativecommons.org/licenses/by-nc-nd/4.0/This is an Open Access article distributed under the terms of the Creative Commons Attribution-NonCommercial-NoDerivs licence (https://creativecommons.org/licenses/by-nc-nd/4.0/), which permits non-commercial reproduction and distribution of the work, in any medium, provided the original work is not altered or transformed in any way, and that the work is properly cited. For commercial re-use, please contact journals.permissions@oup.com
spellingShingle Reproductive Endocrinology
Rai, Harpreet Kaur
Dandona, Paresh
SAT367 A Rare Case of Ovarian Cellular Fibroma Producing Testosterone
title SAT367 A Rare Case of Ovarian Cellular Fibroma Producing Testosterone
title_full SAT367 A Rare Case of Ovarian Cellular Fibroma Producing Testosterone
title_fullStr SAT367 A Rare Case of Ovarian Cellular Fibroma Producing Testosterone
title_full_unstemmed SAT367 A Rare Case of Ovarian Cellular Fibroma Producing Testosterone
title_short SAT367 A Rare Case of Ovarian Cellular Fibroma Producing Testosterone
title_sort sat367 a rare case of ovarian cellular fibroma producing testosterone
topic Reproductive Endocrinology
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC10554879/
http://dx.doi.org/10.1210/jendso/bvad114.1672
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