A Rare Case of Cavernous Haemangioma of the Adrenal Gland: A Case Report and Review of Literature

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Abstract

Cavernous haemangiomas, also known as cavernoma or cavernous angiomas, are clusters of vasculature malformations arising from the endothelial layer of blood vessels. They are commonly found in the central nervous systems, skin, or liver. Rarely, they can also affect adrenal glands, a phenomenon with only 66 cases since the first case was reported in the literature in 1955 and 2018. Adrenal cavernous haemangiomas are typically non-functioning and found incidentally on radiological imaging.

Here, we present the case of a 79-year-old male who was referred by a district general hospital to our tertiary centre with an incidentaloma of the left adrenal gland which was first noted in 2014 measuring 6 cm. A repeat computed tomography in 2020 revealed the mass was 20.8 cm. In 2020, the growing mass was causing anaemia and abdominal discomfort due to displacement of the surrounding viscera.

The initial radiological impression performed in another hospital of the indeterminate mass was highly suspicious of primary adrenal malignancy. The decision was made to operate prior to biopsy as biopsy was deemed high risk for dissemination of primary malignancy of the adrenal.

The patient subsequently underwent a radical open left adrenalectomy in September 2020. The diagnosis of adrenal cavernous haemangioma was made on histopathological examination post-surgery. Published literature on this rare adrenal tumour between 2019 and 2021 is also reviewed in this paper.

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Most cited references 17

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Management of adrenal incidentalomas: European Society of Endocrinology Clinical Practice Guideline in collaboration with the European Network for the Study of Adrenal Tumors.

Martin Fassnacht, Wiebke Arlt, Irina Bancos … (2016)

: By definition, an adrenal incidentaloma is an asymptomatic adrenal mass detected on imaging not performed for suspected adrenal disease. In most cases, adrenal incidentalomas are nonfunctioning adrenocortical adenomas, but may also represent conditions requiring therapeutic intervention (e.g. adrenocortical carcinoma, pheochromocytoma, hormone-producing adenoma or metastasis). The purpose of this guideline is to provide clinicians with best possible evidence-based recommendations for clinical management of patients with adrenal incidentalomas based on the GRADE (Grading of Recommendations Assessment, Development and Evaluation) system. We predefined four main clinical questions crucial for the management of adrenal incidentaloma patients, addressing these four with systematic literature searches: (A) How to assess risk of malignancy?; (B) How to define and manage low-level autonomous cortisol secretion, formerly called 'subclinical' Cushing's syndrome?; (C) Who should have surgical treatment and how should it be performed?; (D) What follow-up is indicated if the adrenal incidentaloma is not surgically removed? SELECTED RECOMMENDATIONS: (i) At the time of initial detection of an adrenal mass establishing whether the mass is benign or malignant is an important aim to avoid cumbersome and expensive follow-up imaging in those with benign disease. (ii) To exclude cortisol excess, a 1mg overnight dexamethasone suppression test should be performed (applying a cut-off value of serum cortisol ≤50nmol/L (1.8µg/dL)). (iii) For patients without clinical signs of overt Cushing's syndrome but serum cortisol levels post 1mg dexamethasone >138nmol/L (>5µg/dL), we propose the term 'autonomous cortisol secretion'. (iv) All patients with '(possible) autonomous cortisol' secretion should be screened for hypertension and type 2 diabetes mellitus, to ensure these are appropriately treated. (v) Surgical treatment should be considered in an individualized approach in patients with 'autonomous cortisol secretion' who also have comorbidities that are potentially related to cortisol excess. (vi) In principle, the appropriateness of surgical intervention should be guided by the likelihood of malignancy, the presence and degree of hormone excess, age, general health and patient preference. (vii) Surgery is not usually indicated in patients with an asymptomatic, nonfunctioning unilateral adrenal mass and obvious benign features on imaging studies. We provide guidance on which surgical approach should be considered for adrenal masses with radiological findings suspicious of malignancy. Furthermore, we offer recommendations for the follow-up of patients with adrenal incidentaloma who do not undergo adrenal surgery, for those with bilateral incidentalomas, for patients with extra-adrenal malignancy and adrenal masses and for young and elderly patients with adrenal incidentalomas.

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CT and MRI of adrenal gland pathologies.

Fuqin Wang, Junwei Liu, Ruoxi Zhang … (2018)

Besides ultrasound and nuclear medicine techniques, computed tomography (CT) and magnetic resonance imaging (MRI) are commonly used to examine adrenal lesions in both symptomatic and asymptomatic patients. Some adrenal lesions have characteristic radiological features. If an adrenal nodule is discovered incidentally, determining whether the lesion is benign or malignant is of great importance. According to their biological behavior, lesions can be divided into benign (mainly: adenoma, hyperplasia, pheochromocytoma, cyst, hemorrhage, cystic lymphangioma, myelolipoma, hemangioma, ganglioneuroma, teratoma) and malignant (mainly: metastases, adrenal cortical carcinoma, neuroblastoma, lymphoma) conditions. In this paper, we review CT/MRI findings of common adrenal gland lesions.

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Hemangioma of the adrenal.

C C Johnson, F JEPPESEN (1955)

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Author and article information

Journal

Journal ID (nlm-ta): Cureus

Journal ID (iso-abbrev): Cureus

Journal ID (issn): 2168-8184

Title: Cureus

Publisher: Cureus (Palo Alto (CA) )

ISSN (Electronic): 2168-8184

Publication date (Electronic, pub): 4 October 2022

Publication date (Electronic, collection): October 2022

Volume: 14

Issue: 10

Electronic Location Identifier: e29917

Affiliations

[1 ] General Surgery, James Cook University Hospital, Middlesbrough, GBR

[2 ] Pathology, James Cook University Hospital, Middlesbrough, GBR

Author notes

Yunli Chua yunli.chua@ 123456gmail.com

Article

DOI: 10.7759/cureus.29917

PMC ID: 9633059

PubMed ID: 36348862

SO-VID: 58d21a6f-5d40-44b8-83cc-4ef8d5e77f34

License:

This is an open access article distributed under the terms of the Creative Commons Attribution License, which permits unrestricted use, distribution, and reproduction in any medium, provided the original author and source are credited.

History

Date accepted : 4 October 2022

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