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SAT-260 Resolution of a Cystic Macroprolactinoma with Dopamine Agonist Therapy

Background. Prolactinomas with cystic regions occupying ≥ 50% of tumor volume are classified as cystic prolactinomas. They appear to arise from tumor necrosis or hemorrhage. Though Pituitary Society guidelines recommend surgery as first-line treatment for cystic prolactinomas, there is evidence that...

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Autores principales: Ansari, Mohammad Jamal Uddin, Abdelkarim, Jumana, Baral, Shailesh, Parveen, Sanober, Mariam, Murtaza Ali, Jabri, Hadoun, Rehman, Anis, Jakoby, Michael G
Formato: Online Artículo Texto
Lenguaje:English
Publicado: Oxford University Press 2020
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC7209175/
http://dx.doi.org/10.1210/jendso/bvaa046.327
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author Ansari, Mohammad Jamal Uddin
Abdelkarim, Jumana
Baral, Shailesh
Parveen, Sanober
Mariam, Murtaza Ali
Jabri, Hadoun
Rehman, Anis
Jakoby, Michael G
author_facet Ansari, Mohammad Jamal Uddin
Abdelkarim, Jumana
Baral, Shailesh
Parveen, Sanober
Mariam, Murtaza Ali
Jabri, Hadoun
Rehman, Anis
Jakoby, Michael G
author_sort Ansari, Mohammad Jamal Uddin
collection PubMed
description Background. Prolactinomas with cystic regions occupying ≥ 50% of tumor volume are classified as cystic prolactinomas. They appear to arise from tumor necrosis or hemorrhage. Though Pituitary Society guidelines recommend surgery as first-line treatment for cystic prolactinomas, there is evidence that cystic prolactinomas are hormonally and anatomically responsive to treatment with dopamine agonists (DA). We present a case of a cystic macroprolactinoma fully responding to treatment with cabergoline. Case. A 31 year old male was referred for evaluation of gynecomastia, diminished libido, and erectile dysfunction that occurred over the course of eight months. Bilateral glandular breast tissue with a slight right asymmetry was present. Testes were 15–20 mL, soft, and without masses. Phallus and pubic hair were unremarkable. Morning (8 AM) testosterone, estradiol, and LH levels were 92 ng/dL (348–1197), 13.4 pg/mL (7.6–42.6), and 2.0 mIU/mL (1.7–8.6), respectively. Prolactin level was 331 ng/mL (4–15), and MRI with pituitary protocol revealed a 1.4 x 1.0 x 1.5 cm cystic mass with peripheral rim enhancement and extension into the suprasellar cistern. No mass effect on the optic chiasm was observed. Somatotroph, thyrotroph, and corticotroph axes were unaffected. Cystic macroprolactinoma causing hyponadotropic hypogonadism complicated by gynecomastia was diagnosed. The patient chose initial management with cabergoline 0.25 mg twice weekly. Three months after starting treatment, libido and erectile function had recovered to baseline, 8 AM testosterone was 607 ng/dL, and hyperprolactinemia was well controlled (4.2 ng/mL). At six months, anatomic resolution of the cystic macroprolactinoma was demonstrated on repeat pituitary MRI. Plastic surgery was required for management of gynecomastia. Conclusions. Surgery is recommended for management of cystic prolactinomas due to concern that a lack of dopamine receptors in the cystic components of tumors will make DA ineffective reducing tumor size. However, this case joins a growing body of evidence that DA can treat hyperprolactinemia and induce regression of cystic macroprolactinomas. For example, in a retrospective case review of patients with cystic macroprolactinomas at Massachusetts General Hospital, persistent cyst reduction occurred in 20 of 22 patients treated initially with DA, and median reduction in cyst volume exceeded 80 percent. Median time to documented cyst reduction was approximately six months, and there was no difference in degree of cyst reduction for patients treated with bromocriptine or cabergoline. The response of cystic prolactinomas to DA is similar to solid prolactinomas, and DA are reasonable initial treatment for cystic macroprolactinomas without clear indications for surgery.
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spelling pubmed-72091752020-05-13 SAT-260 Resolution of a Cystic Macroprolactinoma with Dopamine Agonist Therapy Ansari, Mohammad Jamal Uddin Abdelkarim, Jumana Baral, Shailesh Parveen, Sanober Mariam, Murtaza Ali Jabri, Hadoun Rehman, Anis Jakoby, Michael G J Endocr Soc Neuroendocrinology and Pituitary Background. Prolactinomas with cystic regions occupying ≥ 50% of tumor volume are classified as cystic prolactinomas. They appear to arise from tumor necrosis or hemorrhage. Though Pituitary Society guidelines recommend surgery as first-line treatment for cystic prolactinomas, there is evidence that cystic prolactinomas are hormonally and anatomically responsive to treatment with dopamine agonists (DA). We present a case of a cystic macroprolactinoma fully responding to treatment with cabergoline. Case. A 31 year old male was referred for evaluation of gynecomastia, diminished libido, and erectile dysfunction that occurred over the course of eight months. Bilateral glandular breast tissue with a slight right asymmetry was present. Testes were 15–20 mL, soft, and without masses. Phallus and pubic hair were unremarkable. Morning (8 AM) testosterone, estradiol, and LH levels were 92 ng/dL (348–1197), 13.4 pg/mL (7.6–42.6), and 2.0 mIU/mL (1.7–8.6), respectively. Prolactin level was 331 ng/mL (4–15), and MRI with pituitary protocol revealed a 1.4 x 1.0 x 1.5 cm cystic mass with peripheral rim enhancement and extension into the suprasellar cistern. No mass effect on the optic chiasm was observed. Somatotroph, thyrotroph, and corticotroph axes were unaffected. Cystic macroprolactinoma causing hyponadotropic hypogonadism complicated by gynecomastia was diagnosed. The patient chose initial management with cabergoline 0.25 mg twice weekly. Three months after starting treatment, libido and erectile function had recovered to baseline, 8 AM testosterone was 607 ng/dL, and hyperprolactinemia was well controlled (4.2 ng/mL). At six months, anatomic resolution of the cystic macroprolactinoma was demonstrated on repeat pituitary MRI. Plastic surgery was required for management of gynecomastia. Conclusions. Surgery is recommended for management of cystic prolactinomas due to concern that a lack of dopamine receptors in the cystic components of tumors will make DA ineffective reducing tumor size. However, this case joins a growing body of evidence that DA can treat hyperprolactinemia and induce regression of cystic macroprolactinomas. For example, in a retrospective case review of patients with cystic macroprolactinomas at Massachusetts General Hospital, persistent cyst reduction occurred in 20 of 22 patients treated initially with DA, and median reduction in cyst volume exceeded 80 percent. Median time to documented cyst reduction was approximately six months, and there was no difference in degree of cyst reduction for patients treated with bromocriptine or cabergoline. The response of cystic prolactinomas to DA is similar to solid prolactinomas, and DA are reasonable initial treatment for cystic macroprolactinomas without clear indications for surgery. Oxford University Press 2020-05-08 /pmc/articles/PMC7209175/ http://dx.doi.org/10.1210/jendso/bvaa046.327 Text en © Endocrine Society 2020. http://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 (http://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 Neuroendocrinology and Pituitary
Ansari, Mohammad Jamal Uddin
Abdelkarim, Jumana
Baral, Shailesh
Parveen, Sanober
Mariam, Murtaza Ali
Jabri, Hadoun
Rehman, Anis
Jakoby, Michael G
SAT-260 Resolution of a Cystic Macroprolactinoma with Dopamine Agonist Therapy
title SAT-260 Resolution of a Cystic Macroprolactinoma with Dopamine Agonist Therapy
title_full SAT-260 Resolution of a Cystic Macroprolactinoma with Dopamine Agonist Therapy
title_fullStr SAT-260 Resolution of a Cystic Macroprolactinoma with Dopamine Agonist Therapy
title_full_unstemmed SAT-260 Resolution of a Cystic Macroprolactinoma with Dopamine Agonist Therapy
title_short SAT-260 Resolution of a Cystic Macroprolactinoma with Dopamine Agonist Therapy
title_sort sat-260 resolution of a cystic macroprolactinoma with dopamine agonist therapy
topic Neuroendocrinology and Pituitary
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC7209175/
http://dx.doi.org/10.1210/jendso/bvaa046.327
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