Cushing’s syndrome due to ectopic adrenocorticotropin secretion by a parotid carcinoma

Detalhes bibliográficos
Autor(a) principal: Antonacio,Fernanda F.
Data de Publicação: 2022
Outros Autores: Harada,Guilherme, Vilela,Rafael S., Freitas,Thais C., Lima Jr.,Jose V., Kowalski,Luiz Paulo, Almeida,Madson Q., Castro Junior,Gilberto de
Tipo de documento: Relatório
Idioma: eng
Título da fonte: Arquivos de Endocrinologia e Metabolismo (Online)
Texto Completo: http://old.scielo.br/scielo.php?script=sci_arttext&pid=S2359-39972022000100092
Resumo: SUMMARY We report a rare case of Cushing’s syndrome in a 37-year-old female who initially presented with localized acinic cell carcinoma of the parotid gland. In January 2014, she underwent a right parotidectomy with facial nerve preservation and adjuvant radiotherapy. In August 2018, she presented a histologically-proven local regional relapse. The patient was considered for salvage surgery with facial nerve sacrifice and remained with no evidence of disease. One year later the patient developed pulmonary dissemination and started to gain weight and developed facial plethora and acne on the face and upper trunk. In a physical examination, the patient presented moon face, buffalo hump, acne and stage 2 hypertension. Biochemical evaluation confirmed ACTH-dependent Cushing’s syndrome. IHC for ACTH in the lung biopsy revealed strong positive staining for ACTH confirming a diagnosis of ectopic ACTH secretion by a metastatic parotid acinic cell carcinoma. Ketoconazole (600 mg/d) was started to treat the CS. In addition, as chemotherapy was initiated to treat the metastatic disease. After the fifth cycle of chemotherapy, ketoconazole was suspended and the patient remained in remission of CS for four months, when CS recurred. A unique feature of this case is related to the clinical CS relapse associated with disease progression, which needed prompt treatment with ketoconazole, resulting in a significant improvement in the patient’s condition. Although rare, should be attentive for possible CS features in patients with high-grade salivary gland carcinomas, since the diagnosis of ectopic secretion of ACTH may significantly impact their management and outcomes.
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spelling Cushing’s syndrome due to ectopic adrenocorticotropin secretion by a parotid carcinomaSUMMARY We report a rare case of Cushing’s syndrome in a 37-year-old female who initially presented with localized acinic cell carcinoma of the parotid gland. In January 2014, she underwent a right parotidectomy with facial nerve preservation and adjuvant radiotherapy. In August 2018, she presented a histologically-proven local regional relapse. The patient was considered for salvage surgery with facial nerve sacrifice and remained with no evidence of disease. One year later the patient developed pulmonary dissemination and started to gain weight and developed facial plethora and acne on the face and upper trunk. In a physical examination, the patient presented moon face, buffalo hump, acne and stage 2 hypertension. Biochemical evaluation confirmed ACTH-dependent Cushing’s syndrome. IHC for ACTH in the lung biopsy revealed strong positive staining for ACTH confirming a diagnosis of ectopic ACTH secretion by a metastatic parotid acinic cell carcinoma. Ketoconazole (600 mg/d) was started to treat the CS. In addition, as chemotherapy was initiated to treat the metastatic disease. After the fifth cycle of chemotherapy, ketoconazole was suspended and the patient remained in remission of CS for four months, when CS recurred. A unique feature of this case is related to the clinical CS relapse associated with disease progression, which needed prompt treatment with ketoconazole, resulting in a significant improvement in the patient’s condition. Although rare, should be attentive for possible CS features in patients with high-grade salivary gland carcinomas, since the diagnosis of ectopic secretion of ACTH may significantly impact their management and outcomes.Sociedade Brasileira de Endocrinologia e Metabologia2022-02-01info:eu-repo/semantics/reportinfo:eu-repo/semantics/publishedVersiontext/htmlhttp://old.scielo.br/scielo.php?script=sci_arttext&pid=S2359-39972022000100092Archives of Endocrinology and Metabolism v.66 n.1 2022reponame:Arquivos de Endocrinologia e Metabolismo (Online)instname:Sociedade Brasileira de Endocrinologia e Metabologia (SBEM)instacron:SBEM10.20945/2359-3997000000426info:eu-repo/semantics/openAccessAntonacio,Fernanda F.Harada,GuilhermeVilela,Rafael S.Freitas,Thais C.Lima Jr.,Jose V.Kowalski,Luiz PauloAlmeida,Madson Q.Castro Junior,Gilberto deeng2022-03-15T00:00:00Zoai:scielo:S2359-39972022000100092Revistahttps://www.aem-sbem.com/https://old.scielo.br/oai/scielo-oai.php||aem.editorial.office@endocrino.org.br2359-42922359-3997opendoar:2022-03-15T00:00Arquivos de Endocrinologia e Metabolismo (Online) - Sociedade Brasileira de Endocrinologia e Metabologia (SBEM)false
dc.title.none.fl_str_mv Cushing’s syndrome due to ectopic adrenocorticotropin secretion by a parotid carcinoma
title Cushing’s syndrome due to ectopic adrenocorticotropin secretion by a parotid carcinoma
spellingShingle Cushing’s syndrome due to ectopic adrenocorticotropin secretion by a parotid carcinoma
Antonacio,Fernanda F.
title_short Cushing’s syndrome due to ectopic adrenocorticotropin secretion by a parotid carcinoma
title_full Cushing’s syndrome due to ectopic adrenocorticotropin secretion by a parotid carcinoma
title_fullStr Cushing’s syndrome due to ectopic adrenocorticotropin secretion by a parotid carcinoma
title_full_unstemmed Cushing’s syndrome due to ectopic adrenocorticotropin secretion by a parotid carcinoma
title_sort Cushing’s syndrome due to ectopic adrenocorticotropin secretion by a parotid carcinoma
author Antonacio,Fernanda F.
author_facet Antonacio,Fernanda F.
Harada,Guilherme
Vilela,Rafael S.
Freitas,Thais C.
Lima Jr.,Jose V.
Kowalski,Luiz Paulo
Almeida,Madson Q.
Castro Junior,Gilberto de
author_role author
author2 Harada,Guilherme
Vilela,Rafael S.
Freitas,Thais C.
Lima Jr.,Jose V.
Kowalski,Luiz Paulo
Almeida,Madson Q.
Castro Junior,Gilberto de
author2_role author
author
author
author
author
author
author
dc.contributor.author.fl_str_mv Antonacio,Fernanda F.
Harada,Guilherme
Vilela,Rafael S.
Freitas,Thais C.
Lima Jr.,Jose V.
Kowalski,Luiz Paulo
Almeida,Madson Q.
Castro Junior,Gilberto de
description SUMMARY We report a rare case of Cushing’s syndrome in a 37-year-old female who initially presented with localized acinic cell carcinoma of the parotid gland. In January 2014, she underwent a right parotidectomy with facial nerve preservation and adjuvant radiotherapy. In August 2018, she presented a histologically-proven local regional relapse. The patient was considered for salvage surgery with facial nerve sacrifice and remained with no evidence of disease. One year later the patient developed pulmonary dissemination and started to gain weight and developed facial plethora and acne on the face and upper trunk. In a physical examination, the patient presented moon face, buffalo hump, acne and stage 2 hypertension. Biochemical evaluation confirmed ACTH-dependent Cushing’s syndrome. IHC for ACTH in the lung biopsy revealed strong positive staining for ACTH confirming a diagnosis of ectopic ACTH secretion by a metastatic parotid acinic cell carcinoma. Ketoconazole (600 mg/d) was started to treat the CS. In addition, as chemotherapy was initiated to treat the metastatic disease. After the fifth cycle of chemotherapy, ketoconazole was suspended and the patient remained in remission of CS for four months, when CS recurred. A unique feature of this case is related to the clinical CS relapse associated with disease progression, which needed prompt treatment with ketoconazole, resulting in a significant improvement in the patient’s condition. Although rare, should be attentive for possible CS features in patients with high-grade salivary gland carcinomas, since the diagnosis of ectopic secretion of ACTH may significantly impact their management and outcomes.
publishDate 2022
dc.date.none.fl_str_mv 2022-02-01
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dc.publisher.none.fl_str_mv Sociedade Brasileira de Endocrinologia e Metabologia
publisher.none.fl_str_mv Sociedade Brasileira de Endocrinologia e Metabologia
dc.source.none.fl_str_mv Archives of Endocrinology and Metabolism v.66 n.1 2022
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