A Cortisol-Secreting Adrenal Adenoma Combined With a Micro-Pheochromocytoma: Case Report and Literature Review

Cushing’s syndrome and pheochromocytomas (PCCs) are associated with endocrine hypertension. Cortisol-producing adrenal adenomas are a major cause of Cushing’s syndrome. Simultaneous occurrence of cortisol-producing adrenal adenomas and PCCs is rare. Additionally, a PCC generally produces catecholami...

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Main Authors: Kimiko Sakai, Mai Asano, Masahide Hamaguchi, Hidefumi Taniguchi, Osamu Ukimura, Michiaki Fukui
Format: Article
Language:English
Published: SAGE Publishing 2023-01-01
Series:Clinical Medicine Insights: Endocrinology and Diabetes
Online Access:https://doi.org/10.1177/11795514221148556
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author Kimiko Sakai
Mai Asano
Masahide Hamaguchi
Hidefumi Taniguchi
Osamu Ukimura
Michiaki Fukui
author_facet Kimiko Sakai
Mai Asano
Masahide Hamaguchi
Hidefumi Taniguchi
Osamu Ukimura
Michiaki Fukui
author_sort Kimiko Sakai
collection DOAJ
description Cushing’s syndrome and pheochromocytomas (PCCs) are associated with endocrine hypertension. Cortisol-producing adrenal adenomas are a major cause of Cushing’s syndrome. Simultaneous occurrence of cortisol-producing adrenal adenomas and PCCs is rare. Additionally, a PCC generally produces catecholamines in proportion to its size; therefore, micro-PCCs are rarely found in clinical practice. It is unknown whether micro-PCCs produce excess catecholamines during the pre-biochemical phase. Herein, we report the case of a 53-year-old woman who was referred to our hospital for further evaluation of left adrenal incidentaloma. She had been suffering from hypertension for 7 years. Endocrine tests indicated autonomous cortisol secretion, and she was diagnosed with cortisol-producing adrenal adenoma. A laparoscopic left adrenalectomy was performed. The final pathological examination revealed an adrenocortical adenoma measuring 26 × 24 mm. In addition, a micro-PCC measuring 3 × 2 mm was incidentally found near the cortisol-secreting adrenal adenoma in the ipsilateral adrenal gland. All catecholamine biosynthetic enzymes, tyrosine hydroxylase, aromatic l -amino acid decarboxylase, dopamine β-hydroxylase, and phenyl ethanolamine N-methyltransferase, were detected in this micro-PCC by immunohistochemical analyses. Although catecholamine levels were not biochemically elevated, the PCC expressed catecholamine biosynthetic enzymes. This is the first immunohistochemical report to show that a micro-PCC produces excess catecholamines in the pre-biochemical phase.
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spelling doaj.art-8de874fbe4c4475caada9aac137b2a252023-01-16T14:03:19ZengSAGE PublishingClinical Medicine Insights: Endocrinology and Diabetes1179-55142023-01-011610.1177/11795514221148556A Cortisol-Secreting Adrenal Adenoma Combined With a Micro-Pheochromocytoma: Case Report and Literature ReviewKimiko Sakai0Mai Asano1Masahide Hamaguchi2Hidefumi Taniguchi3Osamu Ukimura4Michiaki Fukui5Department of Endocrinology and Metabolism, Graduate School of Medical Science, Kyoto Prefectural University of Medicine, Kyoto, JapanDepartment of Endocrinology and Metabolism, Graduate School of Medical Science, Kyoto Prefectural University of Medicine, Kyoto, JapanDepartment of Endocrinology and Metabolism, Graduate School of Medical Science, Kyoto Prefectural University of Medicine, Kyoto, JapanDepartment of Urology, Graduate School of Medical Science, Kyoto Prefectural University of Medicine, Kyoto, JapanDepartment of Urology, Graduate School of Medical Science, Kyoto Prefectural University of Medicine, Kyoto, JapanDepartment of Endocrinology and Metabolism, Graduate School of Medical Science, Kyoto Prefectural University of Medicine, Kyoto, JapanCushing’s syndrome and pheochromocytomas (PCCs) are associated with endocrine hypertension. Cortisol-producing adrenal adenomas are a major cause of Cushing’s syndrome. Simultaneous occurrence of cortisol-producing adrenal adenomas and PCCs is rare. Additionally, a PCC generally produces catecholamines in proportion to its size; therefore, micro-PCCs are rarely found in clinical practice. It is unknown whether micro-PCCs produce excess catecholamines during the pre-biochemical phase. Herein, we report the case of a 53-year-old woman who was referred to our hospital for further evaluation of left adrenal incidentaloma. She had been suffering from hypertension for 7 years. Endocrine tests indicated autonomous cortisol secretion, and she was diagnosed with cortisol-producing adrenal adenoma. A laparoscopic left adrenalectomy was performed. The final pathological examination revealed an adrenocortical adenoma measuring 26 × 24 mm. In addition, a micro-PCC measuring 3 × 2 mm was incidentally found near the cortisol-secreting adrenal adenoma in the ipsilateral adrenal gland. All catecholamine biosynthetic enzymes, tyrosine hydroxylase, aromatic l -amino acid decarboxylase, dopamine β-hydroxylase, and phenyl ethanolamine N-methyltransferase, were detected in this micro-PCC by immunohistochemical analyses. Although catecholamine levels were not biochemically elevated, the PCC expressed catecholamine biosynthetic enzymes. This is the first immunohistochemical report to show that a micro-PCC produces excess catecholamines in the pre-biochemical phase.https://doi.org/10.1177/11795514221148556
spellingShingle Kimiko Sakai
Mai Asano
Masahide Hamaguchi
Hidefumi Taniguchi
Osamu Ukimura
Michiaki Fukui
A Cortisol-Secreting Adrenal Adenoma Combined With a Micro-Pheochromocytoma: Case Report and Literature Review
Clinical Medicine Insights: Endocrinology and Diabetes
title A Cortisol-Secreting Adrenal Adenoma Combined With a Micro-Pheochromocytoma: Case Report and Literature Review
title_full A Cortisol-Secreting Adrenal Adenoma Combined With a Micro-Pheochromocytoma: Case Report and Literature Review
title_fullStr A Cortisol-Secreting Adrenal Adenoma Combined With a Micro-Pheochromocytoma: Case Report and Literature Review
title_full_unstemmed A Cortisol-Secreting Adrenal Adenoma Combined With a Micro-Pheochromocytoma: Case Report and Literature Review
title_short A Cortisol-Secreting Adrenal Adenoma Combined With a Micro-Pheochromocytoma: Case Report and Literature Review
title_sort cortisol secreting adrenal adenoma combined with a micro pheochromocytoma case report and literature review
url https://doi.org/10.1177/11795514221148556
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