Literature DB >> 16303835

The ectopic adrenocorticotropin syndrome: clinical features, diagnosis, management, and long-term follow-up.

Andrea M Isidori1, Gregory A Kaltsas, Carlotta Pozza, Vanni Frajese, John Newell-Price, Rodney H Reznek, Paul J Jenkins, John P Monson, Ashley B Grossman, G Michael Besser.   

Abstract

CONTEXT: There are few large series of patients with ectopic, nonpituitary, corticotropin (ACTH) secretion (EAS).
OBJECTIVE: The objective of this study was to analyze the clinical, biochemical, and radiological features, management, and treatment outcome of patients with EAS.
DESIGN: This was a retrospective case-record study.
SETTING: The setting for this study was a tertiary referral hospital center. PATIENTS: Forty patients with EAS were studied. MAIN OUTCOME MEASURES: Clinical, biochemical, and radiological features and response to therapy and survival were measured.
RESULTS: The median follow-up was 5 yr (range, 2-30 yr). None of the dynamic tests achieved 100% accuracy, but bilateral inferior petrosal sinus sampling showed an absent central gradient in all but one case (one of 12). Imaging correctly identified the lesion at first investigation in 65% of cases. Bronchial carcinoid tumors were the most common cause of EAS (n = 12; 30%), followed by other neuroendocrine tumors (n = 13, 32.5%). In 12.5% of patients, the source of EAS was never found. Octreotide scintigraphy and whole-body venous sampling were of limited value. Surgical attempt at curative resection was successful in 83% (10 of 12) of patients with bronchial carcinoid tumors; others responded generally well to adrenolytic therapy or bilateral adrenalectomy. Tumor histology and the presence of distant metastases were the main predictors of overall survival (P < 0.05).
CONCLUSIONS: A variety of tests and imaging studies are necessary for the correct diagnosis of the EAS, but even then, up to 20% of cases present a covert or occult EAS syndrome. These cases require a prolonged follow-up, review, and repetition of diagnostic tests and scans.

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Year:  2005        PMID: 16303835     DOI: 10.1210/jc.2005-1542

Source DB:  PubMed          Journal:  J Clin Endocrinol Metab        ISSN: 0021-972X            Impact factor:   5.958


  123 in total

1.  Tumor Lysis Syndrome in an Unusual Metastatic Pancreatic Neuroendocrine Tumor with Ectopic ACTH Secretion.

Authors:  Michael Shusterman; Noah A Bloomgarden; Susan T Sotardi; Aditi Shastri
Journal:  J Gastrointest Cancer       Date:  2016-12

Review 2.  Diagnostic utility of Gallium-68-somatostatin receptor PET/CT in ectopic ACTH-secreting tumors: a systematic literature review and single-center clinical experience.

Authors:  Elena Varlamov; José Miguel Hinojosa-Amaya; Madeleine Stack; Maria Fleseriu
Journal:  Pituitary       Date:  2019-10       Impact factor: 4.107

3.  A case of severe occult ectopic adrenocorticotropin syndrome treated with retrograde venous adrenal ablation using ethanol.

Authors:  A Gatti; M G Baroni; S Romeo; E Mandosi; M Fallarino; M Bezzi; L Paciucci; S Filetti; S Morano
Journal:  J Endocrinol Invest       Date:  2008-12       Impact factor: 4.256

Review 4.  Molecular basis of pharmacological therapy in Cushing's disease.

Authors:  Diego Ferone; Claudia Pivonello; Giovanni Vitale; Maria Chiara Zatelli; Annamaria Colao; Rosario Pivonello
Journal:  Endocrine       Date:  2013-11-23       Impact factor: 3.633

5.  Ectopic Cushing's syndrome due to retroperitoneal ACTH-producing paragangliomas.

Authors:  Fan Chen; Xiangyu Wang; Yang Wang; Hui Meng; Xinguo Hou; Yaofeng Zhu; Wei Gao; Xuewen Jiang; Shouzhen Chen; Zhaocun Zhang; Zhichuan Zou; Tianyi He; Yue Yang; Kejia Zhu; Yong Wang; Yaxiao Liu; Jianfeng Cui; Benkang Shi; Gang Yin
Journal:  Can Urol Assoc J       Date:  2016-09-13       Impact factor: 1.862

Review 6.  Cushing's syndrome: from physiological principles to diagnosis and clinical care.

Authors:  Hershel Raff; Ty Carroll
Journal:  J Physiol       Date:  2015-01-05       Impact factor: 5.182

7.  [Unexplained weight gain in a 41-year-old woman].

Authors:  I A Harsch; E G Hahn
Journal:  Internist (Berl)       Date:  2010-03       Impact factor: 0.743

8.  Utility of various functional and anatomic imaging modalities for detection of ectopic adrenocorticotropin-secreting tumors.

Authors:  Marina S Zemskova; Bhaskar Gundabolu; Ninet Sinaii; Clara C Chen; Jorge A Carrasquillo; Millie Whatley; Iffat Chowdhury; Ahmed M Gharib; Lynnette K Nieman
Journal:  J Clin Endocrinol Metab       Date:  2010-01-20       Impact factor: 5.958

9.  Cushing's syndrome due to ectopic adrenocorticotropic hormone production secondary to hepatic carcinoid: diagnosis, treatment, and improved quality of life.

Authors:  Susan G Coe; Winston W Tan; Thomas P Fox
Journal:  J Gen Intern Med       Date:  2008-04-02       Impact factor: 5.128

10.  Esthesioneuroblastoma (Olfactory Neuroblastoma) with Ectopic ACTH Syndrome: a multidisciplinary case presentation from the Joan Karnell cancer center of Pennsylvania Hospital.

Authors:  David M Mintzer; Sarah Zheng; Michiko Nagamine; Jason Newman; Maria Benito
Journal:  Oncologist       Date:  2010-01-06
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