Double trouble: two cases of dual adrenal pathologies in one adrenal mass

Context: Adrenal incidentalomas (AI) represent an increasingly common problem in modern endocrine practice. The diagnostic approach to AIs can be challenging and occasionally reveals surprising features. Here we describe two rare cases of complex adrenal lesions consisting of phaeochromocytomas wi...

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Main Authors: Vasileios Chortis, Christine J H May, Kassiani Skordilis, John Ayuk, Wiebke Arlt, Rachel K Crowley
Format: Article
Language:English
Published: Bioscientifica 2019-03-01
Series:Endocrinology, Diabetes & Metabolism Case Reports
Online Access:https://edm.bioscientifica.com/view/journals/edm/2019/1/EDM18-0151.xml
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author Vasileios Chortis
Christine J H May
Kassiani Skordilis
John Ayuk
Wiebke Arlt
Rachel K Crowley
author_facet Vasileios Chortis
Christine J H May
Kassiani Skordilis
John Ayuk
Wiebke Arlt
Rachel K Crowley
author_sort Vasileios Chortis
collection DOAJ
description Context: Adrenal incidentalomas (AI) represent an increasingly common problem in modern endocrine practice. The diagnostic approach to AIs can be challenging and occasionally reveals surprising features. Here we describe two rare cases of complex adrenal lesions consisting of phaeochromocytomas with synchronous metastases from extra-adrenal primaries. Case descriptions: Patient 1 – a 65-year-old gentleman with a newly diagnosed malignant melanoma was found to harbour an adrenal lesion with suspicious radiographic characteristics. Percutaneous adrenal biopsy was consistent with adrenocortical adenoma. After excision of the skin melanoma and regional lymphatic metastases, he was followed up without imaging. Three years later, he presented with abdominal discomfort and enlargement of his adrenal lesion, associated with high plasma metanephrines. Adrenalectomy revealed a mixed tumour consisting of a large phaeochromocytoma with an embedded melanoma metastasis in its core. Patient 2 – a 63-year-old lady with a history of NF-1-related phaeochromocytoma 20 years ago and previous breast cancer presented with a new adrenal lesion on the contralateral side. Plasma normetanephrine was markedly elevated. Elective adrenalectomy revealed an adrenal tumour consisting of chromaffin cells intermixed with breast carcinoma cells. Conclusions: Adrenal incidentalomas require careful evaluation to exclude metastatic disease, especially in the context of a history of previous malignancy. Adrenal biopsy provides limited and potentially misleading information. Phaeochromocytomas are highly vascularised tumours that may function as a sieve, extracting and retaining irregularly shaped cancer cells, thereby yielding adrenal masses with intriguing dual pathology.
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spelling doaj.art-32fde7914e694c6c9ea9bdbff99a7bd62022-12-22T02:18:40ZengBioscientificaEndocrinology, Diabetes & Metabolism Case Reports2052-05732052-05732019-03-01111510.1530/EDM-18-0151Double trouble: two cases of dual adrenal pathologies in one adrenal massVasileios Chortis0Christine J H May1Kassiani Skordilis2John Ayuk3Wiebke Arlt4Rachel K Crowley5Institute of Metabolism and Systems Research, University of Birmingham, Birmingham, UK; Centre for Endocrinology, Diabetes and Metabolism, Birmingham Health Partners, Birmingham, UK; Department of Endocrinology, University Hospital Birmingham NHS Foundation Trust, Birmingham, UKDepartment of Endocrinology, University Hospital Birmingham NHS Foundation Trust, Birmingham, UKCentre for Endocrinology, Diabetes and Metabolism, Birmingham Health Partners, Birmingham, UK; Department of Cellular Pathology, University Hospital Birmingham NHS Foundation Trust, Birmingham, UKCentre for Endocrinology, Diabetes and Metabolism, Birmingham Health Partners, Birmingham, UK; Department of Endocrinology, University Hospital Birmingham NHS Foundation Trust, Birmingham, UKInstitute of Metabolism and Systems Research, University of Birmingham, Birmingham, UK; Centre for Endocrinology, Diabetes and Metabolism, Birmingham Health Partners, Birmingham, UK; Department of Endocrinology, University Hospital Birmingham NHS Foundation Trust, Birmingham, UKSt. Vincent’s University Hospital and University College Dublin, Dublin, IrelandContext: Adrenal incidentalomas (AI) represent an increasingly common problem in modern endocrine practice. The diagnostic approach to AIs can be challenging and occasionally reveals surprising features. Here we describe two rare cases of complex adrenal lesions consisting of phaeochromocytomas with synchronous metastases from extra-adrenal primaries. Case descriptions: Patient 1 – a 65-year-old gentleman with a newly diagnosed malignant melanoma was found to harbour an adrenal lesion with suspicious radiographic characteristics. Percutaneous adrenal biopsy was consistent with adrenocortical adenoma. After excision of the skin melanoma and regional lymphatic metastases, he was followed up without imaging. Three years later, he presented with abdominal discomfort and enlargement of his adrenal lesion, associated with high plasma metanephrines. Adrenalectomy revealed a mixed tumour consisting of a large phaeochromocytoma with an embedded melanoma metastasis in its core. Patient 2 – a 63-year-old lady with a history of NF-1-related phaeochromocytoma 20 years ago and previous breast cancer presented with a new adrenal lesion on the contralateral side. Plasma normetanephrine was markedly elevated. Elective adrenalectomy revealed an adrenal tumour consisting of chromaffin cells intermixed with breast carcinoma cells. Conclusions: Adrenal incidentalomas require careful evaluation to exclude metastatic disease, especially in the context of a history of previous malignancy. Adrenal biopsy provides limited and potentially misleading information. Phaeochromocytomas are highly vascularised tumours that may function as a sieve, extracting and retaining irregularly shaped cancer cells, thereby yielding adrenal masses with intriguing dual pathology.https://edm.bioscientifica.com/view/journals/edm/2019/1/EDM18-0151.xml
spellingShingle Vasileios Chortis
Christine J H May
Kassiani Skordilis
John Ayuk
Wiebke Arlt
Rachel K Crowley
Double trouble: two cases of dual adrenal pathologies in one adrenal mass
Endocrinology, Diabetes & Metabolism Case Reports
title Double trouble: two cases of dual adrenal pathologies in one adrenal mass
title_full Double trouble: two cases of dual adrenal pathologies in one adrenal mass
title_fullStr Double trouble: two cases of dual adrenal pathologies in one adrenal mass
title_full_unstemmed Double trouble: two cases of dual adrenal pathologies in one adrenal mass
title_short Double trouble: two cases of dual adrenal pathologies in one adrenal mass
title_sort double trouble two cases of dual adrenal pathologies in one adrenal mass
url https://edm.bioscientifica.com/view/journals/edm/2019/1/EDM18-0151.xml
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AT johnayuk doubletroubletwocasesofdualadrenalpathologiesinoneadrenalmass
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