A silent corticotroph adenoma: making the case for a pars intermedia origin. Illustrative case

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Abstract

BACKGROUND

Silent corticotroph adenomas (SCAs) are the only pituitary adenomas thought to originate from the pars intermedia. This case report presents the rare finding of a multimicrocystic corticotroph macroadenoma displacing the anterior and posterior lobes of the pituitary gland on magnetic resonance imaging (MRI). This finding supports the hypothesis that silent corticotroph adenomas may originate from the pars intermedia and should be considered in the differential for tumors arising from this location.

OBSERVATIONS

A 55-year-old man presented with an episode of confusion and blurred vision. MRI demonstrated separation of the anterior and posterior glands by a solid-cystic lesion located within the pars intermedia that superiorly displaced the optic chiasm. Endocrinologic evaluation was unremarkable. The differential diagnosis included pituitary adenoma, Rathke cleft cyst, and craniopharyngioma. The tumor was confirmed to be an SCA on pathology and was completely removed through the endoscopic endonasal transsphenoidal approach.

LESSONS

The case highlights the importance of preoperative screening for subclinical hypercortisolism for tumors arising from this location. Knowledge of a patient’s preoperative functional status is critical and dictates their postoperative biochemical assessment to determine remission. The case also illustrates surgical strategies for resecting pars intermedia lesions without injuring the gland.

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Most cited references 21

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Overview of the 2017 WHO Classification of Pituitary Tumors.

Ozgur Mete, M Lopes (2017)

This review focuses on discussing the main changes on the upcoming fourth edition of the WHO Classification of Tumors of the Pituitary Gland emphasizing histopathological and molecular genetics aspects of pituitary neuroendocrine (i.e., pituitary adenomas) and some of the non-neuroendocrine tumors involving the pituitary gland. Instead of a formal review, we introduced the highlights of the new WHO classification by answering select questions relevant to practising pathologists. The revised classification of pituitary adenomas, in addition to hormone immunohistochemistry, recognizes the role of other immunohistochemical markers including but not limited to pituitary transcription factors. Recognizing this novel approach, the fourth edition of the WHO classification has abandoned the concept of "a hormone-producing pituitary adenoma" and adopted a pituitary adenohypophyseal cell lineage designation of the adenomas with subsequent categorization of histological variants according to hormone content and specific histological and immunohistochemical features. This new classification does not require a routine ultrastructural examination of these tumors. The new definition of the Null cell adenoma requires the demonstration of immunonegativity for pituitary transcription factors and adenohypophyseal hormones Moreover, the term of atypical pituitary adenoma is no longer recommended. In addition to the accurate tumor subtyping, assessment of the tumor proliferative potential by mitotic count and Ki-67 index, and other clinical parameters such as tumor invasion, is strongly recommended in individual cases for consideration of clinically aggressive adenomas. This classification also recognizes some subtypes of pituitary neuroendocrine tumors as "high-risk pituitary adenomas" due to the clinical aggressive behavior; these include the sparsely granulated somatotroph adenoma, the lactotroph adenoma in men, the Crooke's cell adenoma, the silent corticotroph adenoma, and the newly introduced plurihormonal Pit-1-positive adenoma (previously known as silent subtype III pituitary adenoma). An additional novel aspect of the new WHO classification was also the definition of the spectrum of thyroid transcription factor-1 expressing pituitary tumors of the posterior lobe as representing a morphological spectrum of a single nosological entity. These tumors include the pituicytoma, the spindle cell oncocytoma, the granular cell tumor of the neurohypophysis, and the sellar ependymoma.

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Pathohistological classification of pituitary tumors: 10 years of experience with the German Pituitary Tumor Registry.

Stephan Petersenn, Dieter Lüdecke, Michael Buchfelder … (2007)

In 1996, the German Registry of Pituitary Tumors was founded by the Pituitary Section of the German Society of Endocrinology as a reference center for collection and consultant pathohistological studies of pituitary tumors. The experiences of the first 10 years of this registry based on 4122 cases will herein be reported. The data supplement former collections of the years 1970-1995 with 3480 surgically removed tumors or lesions of the pituitary region. The cases were studied using histology, immunostainings and in some cases also molecular pathology or electron microscopy. The adenomas were classified according to the current World Health Organization classification in the version of 2004. From 1996 on 3489 adenomas (84.6%), 5 pituitary carcinomas (0.12%), 133 craniopharyngiomas (3.2%), 39 meningiomas (0.94%), 25 metastases (0.6%), 22 chordomas (0.5%), 115 cystic non-neoplastic lesions (2.8%), and 46 inflammatory lesions (1.1%, 248 other lesions or normal tissue (6.0%)) were collected by us. The adenomas (100%) were classified into densely granulated GH cell adenomas (9.2%), sparsely granulated GH cell adenomas (6.3%), sparsely granulated prolactin (PRL) cell adenomas (8.9%), densely granulated PRL cell adenomas (0.3%), mixed GH/PRL cell adenomas (5.2%), mammosomatotropic adenomas (1.1%), acidophilic stem cell adenomas (0.2%), densely granulated ACTH cell adenomas (7.2%), sparsely granulated ACTH cell adenomas (7.9%), Crooke cell adenomas (0.03%), TSH cell adenomas (1.5%), FSH/LH cell adenomas (24.8%), null cell adenomas (19.3%), null cell adenoma, oncocytic variant (5.8%), and plurihormonal adenomas (1.3%). Following the WHO classification of 2004, the new entity 'atypical adenoma' was found in 12 cases in 2005. Various prognostic parameters and clinical implications are discussed.

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The Complementary Role of Transcription Factors in the Accurate Diagnosis of Clinically Nonfunctioning Pituitary Adenomas.

Noriaki Fukuhara, Sylvia L Asa, Kyohei Hayashi … (2015)

Clinically nonfunctioning pituitary adenomas (NFAs) may be hormonally inactive tumors of differentiated cells, mainly not only gonadotroph adenomas (GAs) but also silent corticotroph adenomas (SCAs) and other differentiated silent adenomas. Recently, the use of transcription factors has been recommended to confirm cytodiffererentiation of these neoplasms. Our objective was to assess the clinical significance of the new classification system using transcription factors. Five hundred sixteen consecutive NFAs were studied retrospectively. They were initially classified based on hormone immunohistochemistry as follows: 119 hormone-negative adenomas (23.1 %), 300 GAs (58.1 %), 51 SCAs (9.9 %), and 46 other silent adenomas. The 119 hormone-negative adenomas were further evaluated for expression of transcription factors including steroidogenic factor-1 (SF-1), estrogen receptor-α (ERα), pituitary-specific transcription factor 1 (Pit-1), and t-box transcription factor (Tpit). One hundred thirteen of 119 (95 %) hormone-negative adenomas showed mutually exclusive lineage-specific differentiation as gonadotrophs (SF-1 positive), corticotrophs (Tpit positive), or somatotrophs/mammosomatotrophs/lactotrophs/thyrotrophs (Pit-1 positive) in 79 cases (66.4 %), 32 cases (26.9 %), and 2 cases, respectively. The 32 ACTH-negative and Tpit-positive adenomas had higher pro-opiomelanocortin mRNA expression levels compared with GAs (P = 0.0001) on quantitative real-time PCR. They showed a female preponderance (P < 0.0001) and were more frequently giant adenomas (P = 0.0028) associated with marked cavernous sinus invasion (P < 0.0001) compared with GAs. These clinical features were identical to those of the 51 ACTH-positive SCAs. Our results justify the complementary role of transcription factors in the precise classification of NFAs that can more accurately characterize biological behavior. Our data suggest that more than one quarter of hormone-negative adenomas are SCAs that share distinct clinicopathological features with ACTH-expressing SCAs.

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Author and article information

Journal

Journal ID (nlm-ta): J Neurosurg Case Lessons

Journal ID (iso-abbrev): J Neurosurg Case Lessons

Journal ID (publisher-id): J Neurosurg Case Lessons

Title: Journal of Neurosurgery: Case Lessons

Publisher: American Association of Neurological Surgeons

ISSN (Electronic): 2694-1902

Publication date Collection: 15 May 2023

Publication date (Electronic): 15 May 2023

Volume: 5

Issue: 20

Electronic Location Identifier: CASE2350

Affiliations

Departments of [1 ]Otolaryngology–Head and Neck Surgery,

[2 ]Neurological Surgery, and

[6 ]Endocrinology, Diabetes, and Metabolism, Wexner Medical Center at The Ohio State University, Columbus, Ohio

[3 ]The James Cancer Hospital and Solove Research Institute, The Ohio State University, Columbus, Ohio

[4 ]Department of Otolaryngology, Faculty of Medicine, Mansoura University, Mansoura, Egypt; and

[5 ]Department of Neurosurgery, Hospital Universitario Torrecardenas, Almería, Spain

Author notes

Correspondence Daniel M. Prevedello: The Ohio State University, Columbus, OH. prev02@ 123456osumc.edu .

INCLUDE WHEN CITING Published May 15, 2023; DOI: 10.3171/CASE2350.

Disclosures The authors report no conflict of interest concerning the materials or methods used in this study or the findings specified in this paper.

Article

Publisher ID: CASE2350

DOI: 10.3171/CASE2350

PMC ID: 10550526

PubMed ID: 37212418

SO-VID: be350c30-d006-4b08-88c2-a083e8ea9b5f

License:

CC BY-NC-ND 4.0 ( http://creativecommons.org/licenses/by-nc-nd/4.0/)

History

Date received : 21 February 2023

Date accepted : 8 March 2023

Page count

Figures: 2, Tables: 0, References: 21, Pages: 4

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