From ACTH-Dependent to ACTH-Independent Cushing’s Syndrome from a Malignant Mixed Corticomedullary Adrenal Tumor: Potential Role of Embryonic Stem Cells

Ramírez‐Rentería, Claudia; Espinosa-de-los-Monteros, Ana Laura; Espinosa-Cárdenas, Etual; Marrero‐Rodríguez, Daniel; Castellanos, Guillermo; Arreola-Rosales, Rocío Lorena; Montoya-Martínez, Guillermo; Taniguchi‐Ponciano, Keiko; Briseño-Díaz, Paola; Padilla-Cristerna, Mayte Lizeth; Figueroa-Corona, M.D.P.; Mercado, Moisés

doi:10.1155/2020/4768281

Cited by 4 publications

(3 citation statements)

References 25 publications

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“…Another hypothesis is that these 2 tumors of different origins may have been derived from a single stem cell [ 8 ]. This hypothesis has been reported to clearly explain the simultaneous occurrence of tumors derived from different germ layers in other organs [ 19 ].…”

Section: Discussionmentioning

confidence: 99%

Mixed Corticomedullary Tumor Accompanied by Unilateral Aldosterone-Producing Adrenocortical Micronodules: A Case Report

Yoshida

Babaya

Ito

et al. 2021

Journal of the Endocrine Society

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Mixed corticomedullary tumors (MCMTs) are rare and comprise medullary and cortical cells in a single adrenal tumor. The mechanisms underlying their development have not been fully elucidated. Here, we report a case of MCMT in a 42-year-old woman. Based on the preoperative clinical findings, the patient was diagnosed as having a pheochromocytoma with subclinical Cushing syndrome. Postoperative pathological diagnosis revealed that the tumor demonstrated morphologically distinct medullary and cortical components, which produced catecholamines and cortisol, respectively. Hybrid tumor cells producing both catecholamines and cortisol were not detected. Adrenocorticotropin (ACTH)-positive tumor cells were identified to be present in the pheochromocytoma. This ectopic production of ACTH can contribute to an autonomous cortisol production in a paracrine manner. In addition, micronodules producing aldosterone were detected in the adrenal tissue adjacent to the tumor. The simultaneous development of these 2 lesions may not be correlated with each other; however, this case confirms the importance of a detailed histopathological examination of the adrenal lesions harboring complicated hormonal abnormalities by providing pivotal and indispensable information on their pathogenesis and the possible interaction of the hormones produced in the adrenal gland.

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Section: Discussionmentioning

confidence: 99%

Mixed Corticomedullary Tumor Accompanied by Unilateral Aldosterone-Producing Adrenocortical Micronodules: A Case Report

Yoshida

Babaya

Ito

et al. 2021

Journal of the Endocrine Society

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“…The etiology and mechanisms(pathophysiology) of these tumors is not well understood. Many proposed mechanisms have been cited such as “collision theory”, overproduction of adrenocorticotrophic hormone (ACTH) in a pheochromocytoma, pluripotent stem cells, or an underlying oncogene component [ 6 , 7 , 8 , 9 ]. Given the scarcity at which MCMT is seen, it is difficult to study these tumors in a full investigative fashion.…”

Section: Introductionmentioning

confidence: 99%

Current Understanding of “Mixed Corticomedullary Adrenal Tumor” and an Insight into Genomic Profiling

Ullah

Mohamed

Khan

et al. 2022

Clinics and Practice

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Background: Malignant mixed corticomedullary adrenal tumors (MCMTs) are extremely rare, with limited cases reported in the literature. The pathophysiology of malignant MCMTs is not well understood; the most prevailing theories are that it is a composite tumor of embryologically derived mesodermal (adrenal cortex) and neural crest (medulla) origin, perpetuating as two distinct cell lines forming a singular mass. Clinical features and laboratory diagnosis are associated with hypersecretions of the adrenal cortex and medulla. Surgical resection is curative in an isolated tumor. We reviewed and compared cases in the literature highlighting the pathogenesis and genetics of benign and malignant MCMT. Methods: Comprehensive literature analysis was conducted on PubMed and all the cases of mixed corticomedullary adrenal tumor published in English were included. Results: Most patients were female (73.1%) with a median age of 49 in women and 50 in men. Surgery was performed in all patients, and in four patients with malignant disease, chemotherapy was used as well. Clinically, most patients presented with hypertension (69%) followed by Cushing syndrome (42%) and diabetes (19%). Tumors often produced cortisol (74%), catecholamines (50%), and adrenocorticotrophic hormone (ACTH) (38%), with lower incidence of aldosterone- (7%) or dopamine (4%)-producing tumors. Immunohistochemical staining of 96% of cases showed Chromogranin-A (73%) and Synaptophysin (62%), followed by Inhibin-α (50%), Melan-A (31%), and S-100 (23%). Of the reported four cases with malignant disease, three showed a Ki-67 index of 40–50% with one showing less than 5%. Conclusion: Mixed corticomedullary adrenal tumors rarely present as a malignant disease requiring chemotherapy. Most MCMTs confer a good prognosis and respond well to surgical resection, though their pathogenesis is largely up to speculation because of limited data. Current theories regarding MCMT pathogenesis should be investigated further with genetic testing. Future research on MCMT may provide ways to guide physician diagnosis and subsequent treatment for refractory cases.

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“…In the article titled “From ACTH-Dependent to ACTH-Independent Cushing's Syndrome from a Malignant Mixed Corticomedullary Adrenal Tumor: Potential Role of Embryonic Stem Cells” [ 1 ], there was a spelling error in the author's name in the author list, where Mercado Moises should have read as Moises Mercado. The revised authors' list is shown above.…”

mentioning

confidence: 99%

Erratum to “From ACTH-Dependent to ACTH-Independent Cushing’s Syndrome from a Malignant Mixed Corticomedullary Adrenal Tumor: Potential Role of Embryonic Stem Cells”

From ACTH-Dependent to ACTH-Independent Cushing’s Syndrome from a Malignant Mixed Corticomedullary Adrenal Tumor: Potential Role of Embryonic Stem Cells

Cited by 4 publications

References 25 publications

Mixed Corticomedullary Tumor Accompanied by Unilateral Aldosterone-Producing Adrenocortical Micronodules: A Case Report

Mixed Corticomedullary Tumor Accompanied by Unilateral Aldosterone-Producing Adrenocortical Micronodules: A Case Report

Current Understanding of “Mixed Corticomedullary Adrenal Tumor” and an Insight into Genomic Profiling

Erratum to “From ACTH-Dependent to ACTH-Independent Cushing’s Syndrome from a Malignant Mixed Corticomedullary Adrenal Tumor: Potential Role of Embryonic Stem Cells”

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