Cargando…

Case Report: Progression of a Silent Corticotroph Tumor to an Aggressive Secreting Corticotroph Tumor, Treated by Temozolomide. Changes in the Clinic, the Pathology, and the β-Catenin and α-SMA Expression

Clinically silent corticotroph tumors are usually macroadenomas that comprise 20% of ACTH tumors. They frequently progress to aggressive tumors with high recurrence, invasiveness, and on rare occasions, they may become hormonally active causing Cushing’s disease. Trustable biomarkers that can predic...

Descripción completa

Detalles Bibliográficos
Autores principales: Demarchi, Gianina, Perrone, Sofía, Esper Romero, Gaela, De Bonis, Cristian, Casasco, Juan Pablo, Sevlever, Gustavo, Berner, Silvia Ines, Cristina, Carolina
Formato: Online Artículo Texto
Lenguaje:English
Publicado: Frontiers Media S.A. 2022
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC9344046/
https://www.ncbi.nlm.nih.gov/pubmed/35928898
http://dx.doi.org/10.3389/fendo.2022.870172
_version_ 1784761129278373888
author Demarchi, Gianina
Perrone, Sofía
Esper Romero, Gaela
De Bonis, Cristian
Casasco, Juan Pablo
Sevlever, Gustavo
Berner, Silvia Ines
Cristina, Carolina
author_facet Demarchi, Gianina
Perrone, Sofía
Esper Romero, Gaela
De Bonis, Cristian
Casasco, Juan Pablo
Sevlever, Gustavo
Berner, Silvia Ines
Cristina, Carolina
author_sort Demarchi, Gianina
collection PubMed
description Clinically silent corticotroph tumors are usually macroadenomas that comprise 20% of ACTH tumors. They frequently progress to aggressive tumors with high recurrence, invasiveness, and on rare occasions, they may become hormonally active causing Cushing’s disease. Trustable biomarkers that can predict their aggressive course, as well as their response to traditional or new therapies, are paramount. Aberrant β-Catenin expression and localization have been proposed as responsible for several malignancies including pituitary tumors. Nevertheless, the role of β-Catenin in the aggressive transformation of silent corticotropinomas and their response to Temozolomide salvage treatment have not been explored yet. In this work, we present a case of a silent corticotroph tumor that invaded cavernous sinus and compressed optic chiasm and, after a first total resection and tumor remission it recurred six years later as an aggressive ACTH-secreting tumor. This lesion grew with carotid compromise and caused Cushing’s signs. It required multiple medical treatments including Cabergoline, Ketoconazole, TMZ, and radiotherapy. Besides, other two surgeries were needed until it could be controlled. Interestingly, we found α-SMA vascular area reduction and differential β-Catenin cell localization in the more aggressive tumor stages characterized by high Ki-67 indexes and p53 expression. Our results may indicate a role of angiogenesis and β-Catenin trigged events in the pituitary tumor progression, which could in turn affect the response to TMZ and/or conventional treatments. These molecular findings in this unusual case could be useful for future management of aggressive pituitary tumors.
format Online
Article
Text
id pubmed-9344046
institution National Center for Biotechnology Information
language English
publishDate 2022
publisher Frontiers Media S.A.
record_format MEDLINE/PubMed
spelling pubmed-93440462022-08-03 Case Report: Progression of a Silent Corticotroph Tumor to an Aggressive Secreting Corticotroph Tumor, Treated by Temozolomide. Changes in the Clinic, the Pathology, and the β-Catenin and α-SMA Expression Demarchi, Gianina Perrone, Sofía Esper Romero, Gaela De Bonis, Cristian Casasco, Juan Pablo Sevlever, Gustavo Berner, Silvia Ines Cristina, Carolina Front Endocrinol (Lausanne) Endocrinology Clinically silent corticotroph tumors are usually macroadenomas that comprise 20% of ACTH tumors. They frequently progress to aggressive tumors with high recurrence, invasiveness, and on rare occasions, they may become hormonally active causing Cushing’s disease. Trustable biomarkers that can predict their aggressive course, as well as their response to traditional or new therapies, are paramount. Aberrant β-Catenin expression and localization have been proposed as responsible for several malignancies including pituitary tumors. Nevertheless, the role of β-Catenin in the aggressive transformation of silent corticotropinomas and their response to Temozolomide salvage treatment have not been explored yet. In this work, we present a case of a silent corticotroph tumor that invaded cavernous sinus and compressed optic chiasm and, after a first total resection and tumor remission it recurred six years later as an aggressive ACTH-secreting tumor. This lesion grew with carotid compromise and caused Cushing’s signs. It required multiple medical treatments including Cabergoline, Ketoconazole, TMZ, and radiotherapy. Besides, other two surgeries were needed until it could be controlled. Interestingly, we found α-SMA vascular area reduction and differential β-Catenin cell localization in the more aggressive tumor stages characterized by high Ki-67 indexes and p53 expression. Our results may indicate a role of angiogenesis and β-Catenin trigged events in the pituitary tumor progression, which could in turn affect the response to TMZ and/or conventional treatments. These molecular findings in this unusual case could be useful for future management of aggressive pituitary tumors. Frontiers Media S.A. 2022-07-19 /pmc/articles/PMC9344046/ /pubmed/35928898 http://dx.doi.org/10.3389/fendo.2022.870172 Text en Copyright © 2022 Demarchi, Perrone, Esper Romero, De Bonis, Casasco, Sevlever, Berner and Cristina https://creativecommons.org/licenses/by/4.0/This is an open-access article distributed under the terms of the Creative Commons Attribution License (CC BY). The use, distribution or reproduction in other forums is permitted, provided the original author(s) and the copyright owner(s) are credited and that the original publication in this journal is cited, in accordance with accepted academic practice. No use, distribution or reproduction is permitted which does not comply with these terms.
spellingShingle Endocrinology
Demarchi, Gianina
Perrone, Sofía
Esper Romero, Gaela
De Bonis, Cristian
Casasco, Juan Pablo
Sevlever, Gustavo
Berner, Silvia Ines
Cristina, Carolina
Case Report: Progression of a Silent Corticotroph Tumor to an Aggressive Secreting Corticotroph Tumor, Treated by Temozolomide. Changes in the Clinic, the Pathology, and the β-Catenin and α-SMA Expression
title Case Report: Progression of a Silent Corticotroph Tumor to an Aggressive Secreting Corticotroph Tumor, Treated by Temozolomide. Changes in the Clinic, the Pathology, and the β-Catenin and α-SMA Expression
title_full Case Report: Progression of a Silent Corticotroph Tumor to an Aggressive Secreting Corticotroph Tumor, Treated by Temozolomide. Changes in the Clinic, the Pathology, and the β-Catenin and α-SMA Expression
title_fullStr Case Report: Progression of a Silent Corticotroph Tumor to an Aggressive Secreting Corticotroph Tumor, Treated by Temozolomide. Changes in the Clinic, the Pathology, and the β-Catenin and α-SMA Expression
title_full_unstemmed Case Report: Progression of a Silent Corticotroph Tumor to an Aggressive Secreting Corticotroph Tumor, Treated by Temozolomide. Changes in the Clinic, the Pathology, and the β-Catenin and α-SMA Expression
title_short Case Report: Progression of a Silent Corticotroph Tumor to an Aggressive Secreting Corticotroph Tumor, Treated by Temozolomide. Changes in the Clinic, the Pathology, and the β-Catenin and α-SMA Expression
title_sort case report: progression of a silent corticotroph tumor to an aggressive secreting corticotroph tumor, treated by temozolomide. changes in the clinic, the pathology, and the β-catenin and α-sma expression
topic Endocrinology
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC9344046/
https://www.ncbi.nlm.nih.gov/pubmed/35928898
http://dx.doi.org/10.3389/fendo.2022.870172
work_keys_str_mv AT demarchigianina casereportprogressionofasilentcorticotrophtumortoanaggressivesecretingcorticotrophtumortreatedbytemozolomidechangesintheclinicthepathologyandthebcateninandasmaexpression
AT perronesofia casereportprogressionofasilentcorticotrophtumortoanaggressivesecretingcorticotrophtumortreatedbytemozolomidechangesintheclinicthepathologyandthebcateninandasmaexpression
AT esperromerogaela casereportprogressionofasilentcorticotrophtumortoanaggressivesecretingcorticotrophtumortreatedbytemozolomidechangesintheclinicthepathologyandthebcateninandasmaexpression
AT deboniscristian casereportprogressionofasilentcorticotrophtumortoanaggressivesecretingcorticotrophtumortreatedbytemozolomidechangesintheclinicthepathologyandthebcateninandasmaexpression
AT casascojuanpablo casereportprogressionofasilentcorticotrophtumortoanaggressivesecretingcorticotrophtumortreatedbytemozolomidechangesintheclinicthepathologyandthebcateninandasmaexpression
AT sevlevergustavo casereportprogressionofasilentcorticotrophtumortoanaggressivesecretingcorticotrophtumortreatedbytemozolomidechangesintheclinicthepathologyandthebcateninandasmaexpression
AT bernersilviaines casereportprogressionofasilentcorticotrophtumortoanaggressivesecretingcorticotrophtumortreatedbytemozolomidechangesintheclinicthepathologyandthebcateninandasmaexpression
AT cristinacarolina casereportprogressionofasilentcorticotrophtumortoanaggressivesecretingcorticotrophtumortreatedbytemozolomidechangesintheclinicthepathologyandthebcateninandasmaexpression