Cargando…

Successful Pregnancies in an Acromegalic Woman After Non-Radical Pituitary Adenomectomy for Somatoprolactinoma

Background: Pregnancy is unusual in patients with acromegaly due to somatotropinomas or somatoprolactinomas. Fertility is impaired because of hormonal hypersecretion, pituitary damage by tumor compression or both. Managing somatoprolactinomas and fertility issues are often challenging. Clinical Case...

Descripción completa

Detalles Bibliográficos
Autores principales: Gatzk Arruda, Anna Catarina, Outuki, Giovana, Dias, Marcos Antonio, Carrilho, Alexandre Jose Faria, Mazzuco, Tania Longo
Formato: Online Artículo Texto
Lenguaje:English
Publicado: Oxford University Press 2021
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC8090536/
http://dx.doi.org/10.1210/jendso/bvab048.1248
_version_ 1783687307165958144
author Gatzk Arruda, Anna Catarina
Outuki, Giovana
Dias, Marcos Antonio
Carrilho, Alexandre Jose Faria
Mazzuco, Tania Longo
author_facet Gatzk Arruda, Anna Catarina
Outuki, Giovana
Dias, Marcos Antonio
Carrilho, Alexandre Jose Faria
Mazzuco, Tania Longo
author_sort Gatzk Arruda, Anna Catarina
collection PubMed
description Background: Pregnancy is unusual in patients with acromegaly due to somatotropinomas or somatoprolactinomas. Fertility is impaired because of hormonal hypersecretion, pituitary damage by tumor compression or both. Managing somatoprolactinomas and fertility issues are often challenging. Clinical Case: A 20-year woman with primary amenorrhea and headache was diagnosed with hypogonadotrophic hypogonadism secondary to hyperprolactinemia (2500 µg/L, n<23 µg/L). No other abnormalities were found on the pituitary function screening tests. MRI revealed an intra and suprasellar adenoma (2.5x1.8x1.8 cm) with optic chiasm compression. The onset of menses occurred after 11 months under dopaminergic treatment, and tumor size diminished (1.9x1.5x1.5 cm), bringing on optic chiasm decompression. She remained under dopamine agonist treatment for 6 years, when she realized extremities enlargement and height increase by 3 cm. Acromegaly was confirmed by blood levels of IGF-1 (3.37xULN), GH (8 µg/L, n<8 µg/L), and GH nadir (4.3 µg/L, n<1 µg/L) during OGTT. Then, octreotide LAR was added to cabergoline treatment while waiting for elective surgical treatment. She underwent to transsphenoidal endonasal neurosurgical microscopy approach guided by neuronavigation, with the removal of a large portion of tumor. However, it was not possible to extract the part of invasive adenoma close to right carotid artery due to the risk of vascular and intracavernous cranial nerves injury. Immunohistochemistry analysis of the adenoma was positive only for GH cells with low Ki67 index (<1%). Due to the poor biochemical control (unsuppressed post-OGTT GH, IGF-1 1.66xULN and PRL 301 µg/L) and the presence of a small stable tumor residue, treatment with cabergoline and somatostatin analogues was maintained (3-year octreotide LAR, transitioned to lanreotide in an attempt to achieve a better biochemical response). After 14 years of the initial diagnosis and 5 years post-surgery, the patient expressed the desire to get pregnant and all medications in use were suspended. In the following 3 years, she had two uneventful gestation without complications or worsen of acromegaly; she only breastfed for few months after her first pregnancy. The second one was a twin pregnancy. After one year, the MRI revealed no increase of tumor mass (1.0x0.3x1.0 cm), and PRL levels withing normal range, IGF-1 slightly elevated, but GH not suppressed by OGTT. Cabergoline was reintroduced and the biochemical control of acromegaly was achieved. Conclusion: We reported the very unusual spontaneous conception and normal course of pregnancies in a woman with acromegaly, who was submitted to successful transsphenoidal neurosurgical microscopy approach in which large part of the tumor was removed and the normal pituitary tissue was preserved, allowing fertility restoration.
format Online
Article
Text
id pubmed-8090536
institution National Center for Biotechnology Information
language English
publishDate 2021
publisher Oxford University Press
record_format MEDLINE/PubMed
spelling pubmed-80905362021-05-05 Successful Pregnancies in an Acromegalic Woman After Non-Radical Pituitary Adenomectomy for Somatoprolactinoma Gatzk Arruda, Anna Catarina Outuki, Giovana Dias, Marcos Antonio Carrilho, Alexandre Jose Faria Mazzuco, Tania Longo J Endocr Soc Neuroendocrinology and Pituitary Background: Pregnancy is unusual in patients with acromegaly due to somatotropinomas or somatoprolactinomas. Fertility is impaired because of hormonal hypersecretion, pituitary damage by tumor compression or both. Managing somatoprolactinomas and fertility issues are often challenging. Clinical Case: A 20-year woman with primary amenorrhea and headache was diagnosed with hypogonadotrophic hypogonadism secondary to hyperprolactinemia (2500 µg/L, n<23 µg/L). No other abnormalities were found on the pituitary function screening tests. MRI revealed an intra and suprasellar adenoma (2.5x1.8x1.8 cm) with optic chiasm compression. The onset of menses occurred after 11 months under dopaminergic treatment, and tumor size diminished (1.9x1.5x1.5 cm), bringing on optic chiasm decompression. She remained under dopamine agonist treatment for 6 years, when she realized extremities enlargement and height increase by 3 cm. Acromegaly was confirmed by blood levels of IGF-1 (3.37xULN), GH (8 µg/L, n<8 µg/L), and GH nadir (4.3 µg/L, n<1 µg/L) during OGTT. Then, octreotide LAR was added to cabergoline treatment while waiting for elective surgical treatment. She underwent to transsphenoidal endonasal neurosurgical microscopy approach guided by neuronavigation, with the removal of a large portion of tumor. However, it was not possible to extract the part of invasive adenoma close to right carotid artery due to the risk of vascular and intracavernous cranial nerves injury. Immunohistochemistry analysis of the adenoma was positive only for GH cells with low Ki67 index (<1%). Due to the poor biochemical control (unsuppressed post-OGTT GH, IGF-1 1.66xULN and PRL 301 µg/L) and the presence of a small stable tumor residue, treatment with cabergoline and somatostatin analogues was maintained (3-year octreotide LAR, transitioned to lanreotide in an attempt to achieve a better biochemical response). After 14 years of the initial diagnosis and 5 years post-surgery, the patient expressed the desire to get pregnant and all medications in use were suspended. In the following 3 years, she had two uneventful gestation without complications or worsen of acromegaly; she only breastfed for few months after her first pregnancy. The second one was a twin pregnancy. After one year, the MRI revealed no increase of tumor mass (1.0x0.3x1.0 cm), and PRL levels withing normal range, IGF-1 slightly elevated, but GH not suppressed by OGTT. Cabergoline was reintroduced and the biochemical control of acromegaly was achieved. Conclusion: We reported the very unusual spontaneous conception and normal course of pregnancies in a woman with acromegaly, who was submitted to successful transsphenoidal neurosurgical microscopy approach in which large part of the tumor was removed and the normal pituitary tissue was preserved, allowing fertility restoration. Oxford University Press 2021-05-03 /pmc/articles/PMC8090536/ http://dx.doi.org/10.1210/jendso/bvab048.1248 Text en © The Author(s) 2021. 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 (http://creativecommons.org/licenses/by-nc-nd/4.0/ (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 Neuroendocrinology and Pituitary
Gatzk Arruda, Anna Catarina
Outuki, Giovana
Dias, Marcos Antonio
Carrilho, Alexandre Jose Faria
Mazzuco, Tania Longo
Successful Pregnancies in an Acromegalic Woman After Non-Radical Pituitary Adenomectomy for Somatoprolactinoma
title Successful Pregnancies in an Acromegalic Woman After Non-Radical Pituitary Adenomectomy for Somatoprolactinoma
title_full Successful Pregnancies in an Acromegalic Woman After Non-Radical Pituitary Adenomectomy for Somatoprolactinoma
title_fullStr Successful Pregnancies in an Acromegalic Woman After Non-Radical Pituitary Adenomectomy for Somatoprolactinoma
title_full_unstemmed Successful Pregnancies in an Acromegalic Woman After Non-Radical Pituitary Adenomectomy for Somatoprolactinoma
title_short Successful Pregnancies in an Acromegalic Woman After Non-Radical Pituitary Adenomectomy for Somatoprolactinoma
title_sort successful pregnancies in an acromegalic woman after non-radical pituitary adenomectomy for somatoprolactinoma
topic Neuroendocrinology and Pituitary
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC8090536/
http://dx.doi.org/10.1210/jendso/bvab048.1248
work_keys_str_mv AT gatzkarrudaannacatarina successfulpregnanciesinanacromegalicwomanafternonradicalpituitaryadenomectomyforsomatoprolactinoma
AT outukigiovana successfulpregnanciesinanacromegalicwomanafternonradicalpituitaryadenomectomyforsomatoprolactinoma
AT diasmarcosantonio successfulpregnanciesinanacromegalicwomanafternonradicalpituitaryadenomectomyforsomatoprolactinoma
AT carrilhoalexandrejosefaria successfulpregnanciesinanacromegalicwomanafternonradicalpituitaryadenomectomyforsomatoprolactinoma
AT mazzucotanialongo successfulpregnanciesinanacromegalicwomanafternonradicalpituitaryadenomectomyforsomatoprolactinoma