Literature DB >> 35531113

Ectopic adrenocorticotrophic hormone syndrome initially presenting as abnormal mental behavior caused by thymic carcinoid: a case report.

Xu Han1, Qihe Liu1, Yan Yang1, Xixian Ke1, Cheng Chen1.   

Abstract

Background: Thymic carcinoid is a rare highly differentiated neuroendocrine neoplasm, which can manifest as endocrine disorders caused by ectopic adrenocorticotrophic hormone (ACTH) syndrome. Although clinical manifestations such as hypertension and hypokalemia are common manifestations in patients with ectopic ACTH syndrome, clinicians should also be aware of the mental and behavioral abnormalities that may initially appear in patients. It is extremely rare for patients with ectopic ACTH syndrome caused by thymic carcinoid to concurrently exhibit abnormal mental behavior, especially as the initial clinical manifestation of the tumor. Studies have suggested that abnormal mental behavior may be related to elevated blood cortisol levels. Case Description: A patient was admitted to hospital due to abnormal mental behavior, manifesting as hyperphasia involving gibberish and illogical language, trance, and a state of suspicion. The patient had experienced persecutory delusion. Auxiliary examination revealed elevated cortisol and ACTH. Chest computed tomography (CT) showed right anterior mediastinal tumor. After discussion, the multi-disciplinary team (MDT) concluded that ectopic ACTH syndrome derived from the thymus should be considered. After excluding surgical contraindications, a thymic tumor was resected, and the postoperative pathology confirmed that it was thymic carcinoid. At 6 postoperative months, the results were as follows: cortisol at 8:00 am 196.50 nmol/L; and ACTH at 8:00 am 28.63 pg/mL. The patient's mental behavior had returned to normal, and normal communication was possible. The postoperative symptoms and signs of the patient were improved, which reiterated the presence ectopic ACTH syndrome caused by thymic carcinoid. Conclusions: Thymic carcinoid with ectopic ACTH syndrome is very rare in clinical practice, and it is easily missed and misdiagnosed. Although clinical manifestations such as hypertension and hypokalemia are common manifestations in patients with Cushing's syndrome, clinicians should be aware that patients with Cushing's syndrome may initially exhibit abnormal mental behavior. Clinically, if the patient exhibits abnormal mental behavior accompanied by symptoms such as hypokalemia, hypertension, and diabetes, blood cortisol and ACTH hormone levels should be screened without delay. If the levels are found to be significantly increased, ACTH syndrome should be highly suspected. 2022 Gland Surgery. All rights reserved.

Entities:  

Keywords:  Thymic carcinoid; case report; ectopic adrenocorticotrophic hormone syndrome (ectopic ACTH syndrome); multi-disciplinary team (MDT)

Year:  2022        PMID: 35531113      PMCID: PMC9068539          DOI: 10.21037/gs-22-173

Source DB:  PubMed          Journal:  Gland Surg        ISSN: 2227-684X


  15 in total

1.  Carcinoid tumors of the thymus and Cushing's syndrome: Clinicopathologic features and current best evidence regarding the cell of origin of these unusual neoplasms.

Authors:  Ann E Walts; Joseph Frye; David M Engman; Alberto M Marchevsky
Journal:  Ann Diagn Pathol       Date:  2018-11-22       Impact factor: 2.090

2.  Prognostic factors in ectopic Cushing's syndrome due to neuroendocrine tumors: a multicenter study.

Authors:  Maria Vittoria Davi'; Elisa Cosaro; Serena Piacentini; Giuseppe Reimondo; Nora Albiger; Giorgio Arnaldi; Antongiulio Faggiano; Giovanna Mantovani; Nicola Fazio; Alessandro Piovesan; Emanuela Arvat; Franco Grimaldi; Letizia Canu; Massimo Mannelli; Alberto Giacinto Ambrogio; Francesca Pecori Giraldi; Chiara Martini; Andrea Lania; Manuela Albertelli; Diego Ferone; Maria Chiara Zatelli; Davide Campana; Annamaria Colao; Carla Scaroni; Massimo Terzolo; Laura De Marinis; Sara Cingarlini; Rocco Micciolo; Giuseppe Francia
Journal:  Eur J Endocrinol       Date:  2017-04       Impact factor: 6.664

3.  Thymic neuroendocrine tumors: a SEER database analysis of 160 patients.

Authors:  Puja Gaur; Colleen Leary; James C Yao
Journal:  Ann Surg       Date:  2010-06       Impact factor: 12.969

4.  Mediastinal endocrine neoplasm, of probable thymic origin, related to carcinoid tumor. Clinicopathologic study of 8 cases.

Authors:  J Rosai; E Higa
Journal:  Cancer       Date:  1972-04       Impact factor: 6.860

Review 5.  MANAGEMENT OF ENDOCRINE DISEASE: Cushing's syndrome due to ectopic ACTH secretion: an expert operational opinion.

Authors:  Jacques Young; Magalie Haissaguerre; Oceana Viera-Pinto; Olivier Chabre; Eric Baudin; Antoine Tabarin
Journal:  Eur J Endocrinol       Date:  2020-04       Impact factor: 6.664

Review 6.  Ectopic Cushing's syndrome due to thymic neuroendocrine tumours: a systematic review.

Authors:  Fernando Guerrero-Pérez; Inmaculada Peiró; Agustina Pia Marengo; Alex Teulé; José Carlos Ruffinelli; Roger Llatjos; Teresa Serrano; Ivan Macia; Nuria Vilarrasa; Pedro Iglesias; Carles Villabona
Journal:  Rev Endocr Metab Disord       Date:  2021-05-07       Impact factor: 6.514

Review 7.  Severe psychosis due to Cushing's syndrome in a patient with a carcinoid tumour in the lung: a case report and review of the current management.

Authors:  Mohamad Baba; Debamalya Ray
Journal:  World J Surg Oncol       Date:  2015-04-30       Impact factor: 2.754

8.  An unusual case of ectopic corticotrophin-releasing hormone syndrome caused by an adrenal noncatecholamine-secreting pheochromocytoma: a case report.

Authors:  Bao-Ping Wang; Lei-Lei Yang; Hao Wang; Qing He; Zhong-Shu Ma; Yi Lin; Chang-Xin Jiang; Hao-Ran Sun; Ming Liu
Journal:  BMC Endocr Disord       Date:  2018-06-19       Impact factor: 2.763

9.  A unique case of ectopic Cushing's syndrome from a thymic neuroendocrine carcinoma.

Authors:  Lima Lawrence; Peng Zhang; Humberto Choi; Usman Ahmad; Valeria Arrossi; Andrei Purysko; Vinni Makin
Journal:  Endocrinol Diabetes Metab Case Rep       Date:  2019-02-22

10.  Primary thymic atypical carcinoid with rare multiple bone metastasis: A case report and literature review.

Authors:  Xinrui Rao; Weibing Chen; Ji Li; Gang Peng; Gang Wu; Rui Zhou; Qian Ding
Journal:  Mol Clin Oncol       Date:  2021-02-25
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