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Original Article
Hypothalamus and Pituitary Gland
Big Data Articles (National Health Insurance Service Database)
Descriptive Epidemiology and Survival Analysis of Prolactinomas and Cushing’s Disease in Korea
Jin Sun Park, Soo Jin Yun, Jung Kuk Lee, So Young Park, Sang Ouk Chin
Endocrinol Metab. 2021;36(3):688-696.   Published online June 28, 2021
DOI: https://doi.org/10.3803/EnM.2021.1000
  • 5,813 View
  • 153 Download
  • 4 Web of Science
  • 5 Crossref
AbstractAbstract PDFPubReader   ePub   
Background
Only a few studies have established the epidemiology of prolactinoma and Cushing’s disease in Korea. Furthermore, the incidence of these disease are increasing than before associated with the development of technologies. This study was designed to evaluate the epidemiology of prolactinoma and Cushing’s disease and their survival analysis according to treatment.
Methods
The nationwide, population-based study evaluated incidence and prevalence of prolactinoma and Cushing’s disease using de-identified claims data in The Korean Health Insurance Review and Assessment Service database between 2013 and 2017. The survival analysis investigated regarding treatment over a period of 6 years. A log-rank test and Cox proportional hazard regression analysis were used.
Results
The 6,056 patients with newly diagnosed prolactinoma and 584 patients with Cushing’s disease were recorded between 2013 and 2017. The annual incidence of prolactinoma was 23.5 cases per million, and its prevalence was 82.5 cases per million, and 2.3 cases per million/year and 9.8 cases per million for Cushing’s disease. The survival benefit was insignificant in prolactinoma according to treatment, but treatment of Cushing’s disease ameliorated the survival rate significantly.
Conclusion
Overall, the incidence of prolactinoma and Cushing’s disease was similar with those found previously, but the prevalence of two diseases were inconsistent when compared with the early studies. The present study also proposed necessity of treatment in Cushing’s disease for improving the survival rate.

Citations

Citations to this article as recorded by  
  • For the Forthcoming Winning Shot in the Battle against Cushing Disease
    Sang Ouk Chin
    Endocrinology and Metabolism.2024; 39(4): 573.     CrossRef
  • The 24-Hour Urinary Cortisol/Urinary Creatinine Ratio Helps Differentiate Cushing's Syndrome from Simple Obesity
    Meng Wang, Xueting Sun, Shiwei Li, Xin Li, Jingqiu Cui
    Endocrine Practice.2024;[Epub]     CrossRef
  • Big Data Research in the Field of Endocrine Diseases Using the Korean National Health Information Database
    Sun Wook Cho, Jung Hee Kim, Han Seok Choi, Hwa Young Ahn, Mee Kyoung Kim, Eun Jung Rhee
    Endocrinology and Metabolism.2023; 38(1): 10.     CrossRef
  • Cushing Syndrome
    Martin Reincke, Maria Fleseriu
    JAMA.2023; 330(2): 170.     CrossRef
  • Clinical Biology of the Pituitary Adenoma
    Shlomo Melmed, Ursula B Kaiser, M Beatriz Lopes, Jerome Bertherat, Luis V Syro, Gerald Raverot, Martin Reincke, Gudmundur Johannsson, Albert Beckers, Maria Fleseriu, Andrea Giustina, John A H Wass, Ken K Y Ho
    Endocrine Reviews.2022; 43(6): 1003.     CrossRef
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Case Reports
A Case with Isolated ACTH Deficiency.
Myoung Sik Kim, Byung Doo Lee, Sang Min Shin, Young Il Kim, Byung Oh Jeong, Hong Jib Choi, Phil Ho Kim, Kyung Soo Ko, Jae Hong Park
J Korean Endocr Soc. 1996;11(4):538-543.   Published online November 7, 2019
  • 1,342 View
  • 55 Download
AbstractAbstract PDF
Isolated ACTH deficiency is a rare cause of secondary adrenocortical insufficiency. Adrenal crisis in isolated ACTH deficiency is less common compared to primary adrenal insufficiency, but isolated ACTH deficiency is an important cause of hypoglycemia. Recently we experienced a 41-year-old man admitted because of mental confusion. On admission, plasma glucose and sodium concentration were 1.7, 132 mmol/L, respectively. Basal plasma ACTH and cortisol levels were low and other pituitary hormone showed normal response to combined pituitary stimulation test except growth hormone. Plasma ACTH concentration remained low even after intravenous injection of ovine corticotropin releasing factor. It suggest that the defect of ACTH secretion was apparently due to intrinsic pituitary rather than hypothalamic disease. The sellar CT showed the fossa to be filled by cerebrospinal fluid. After treatment with glucocorticoid, he had no further evidence of hypoglycemia and hyponatremia. In conclusion, we report a case of isolated ACTH deficiency with empty sella.
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A Case of Isolated ACTH Deficiency.
Sang Jin Kim, Kyung Mook Choi, Sei Hyun Baik, Dong Seop Choi
J Korean Endocr Soc. 1995;10(4):445-450.   Published online November 7, 2019
  • 1,224 View
  • 23 Download
AbstractAbstract PDF
solated ACTH deficiency is a very uncommon cause of hypoadrenocorticism, with less than 200 cases reported in the literatures. The clinical presentation can be similar to that of primary adrenal insufficiency, but there is a greater tendency for hypoglycemia and absence of hyperpigmentation. The diagnosis is established by demonstrating hypocortisolism with undetectable serum levels of ACTH, normal adrenal responsiveness to prolonged ACTH infusion, and an absent ACTH response to insulin-induced hypoglycemia. Other endocrine function is normal.We experienced a case of isolated ACTH deficiency in 34 years old female who was admitted due to fever and drowsy mentality.So we present this case with a review of literatures.
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Original Articles
Cushing's Disease Due to ACTH Producing Pituitary Carcinoma.
Young Kee Shong, Joong Yeol Park, Ghi Su Kim, Won Kyoung Cho, Jung Kyo Lee, Ghee Young Choe, Mun Ho Lee, Ki Up Lee
J Korean Endocr Soc. 1994;9(2):136-140.   Published online November 6, 2019
  • 1,191 View
  • 20 Download
AbstractAbstract PDF
Primary carcinoma of the pituitary is only rarely reported. About half of the reported cases are nonfunctional. It is generally agreed that the presence of distant metastasis is required to clearly establish the diagnosis of pituitary carcinoma. We have experienced a case of ACTH-producing pituitary carcinoma causing Cushing's syndrome which could be diagnosed by histologic features only without evidence of distant metastasis. A 35-year-old female with Cushingoid appearance was diagnosed as Cushing's disease after biochemical and neuroradiological evaluation. Near total removal of the pituitary tumor was performed. By histopathologic examination, the tumor revealed evidences of histological malignancy such as prominent nuclear pleomorphism, frequent mitoses and extensive tumor necrosis. Pseudosarcomatous components were also noted. By immunohistochemical studies, the tumor cells expressed ACTH. However, there was no evidence of distant metastasis at the initial operation. She was diagnosed as having ACTH producing pituitary carcinoma by histologic feature only. One month later, she complained progressive nausea and vomiting, and follow-up brain MRI revealed regrowing pituitary mass. She was reoperated and near total tumor bulk was removed again followed by radiotherapy with 5580 rads. Four month after the second operation, she developed generalized tonic clonic seizure. Brain CT showed multiple enhancing nodules on left temporal and frontal lobes, and around falx cerebri. She refused further treatment and was managed only with anticonvalsants. About month after discharge she died at home.
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Endocrine Research
Effects of Oxytocin on Cell Proliferation in a Corticotroph Adenoma Cell Line
Jung Soo Lim, Young Woo Eom, Eun Soo Lee, Hyeong Ju Kwon, Ja-Young Kwon, Junjeong Choi, Choon Hee Chung, Young Suk Jo, Eun Jig Lee
Endocrinol Metab. 2019;34(3):302-313.   Published online September 26, 2019
DOI: https://doi.org/10.3803/EnM.2019.34.3.302
  • 6,413 View
  • 80 Download
  • 3 Web of Science
  • 2 Crossref
AbstractAbstract PDFSupplementary MaterialPubReader   ePub   
Background

Oxytocin (OXT) has been reported to act as a growth regulator in various tumor cells. However, there is a paucity of data on the influence of OXT on cell proliferation of corticotroph adenomas. This study aimed to examine whether OXT affects cell growth in pituitary tumor cell lines (AtT20 and GH3 cells) with a focus on corticotroph adenoma cells.

Methods

Reverse transcription polymerase chain reaction and enzyme-linked immunosorbent assay were conducted with AtT20 cells to confirm the effects of OXT on hormonal activity; flow cytometry was used to assess changes in the cell cycle after OXT treatment. Moreover, the impact of OXT on proliferating cell nuclear antigen (PCNA), nuclear factor κB, and mitogen-activated protein kinase signaling pathway was analyzed by Western blot.

Results

OXT treatment of 50 nM changed the gene expression of OXT receptor and pro-opiomelanocortin within a short time. In addition, OXT significantly reduced adrenocorticotropic hormone secretion within 1 hour. S and G2/M populations of AtT20 cells treated with OXT for 24 hours were significantly decreased compared to the control. Furthermore, OXT treatment decreased the protein levels of PCNA and phosphorylated extracellular-signal-regulated kinase (P-ERK) in AtT20 cells.

Conclusion

Although the cytotoxic effect of OXT in AtT20 cells was not definite, OXT may blunt cell proliferation of corticotroph adenomas by altering the cell cycle or reducing PCNA and P-ERK levels. Further research is required to investigate the role of OXT as a potential therapeutic target in corticotroph adenomas.

Citations

Citations to this article as recorded by  
  • Increased proliferation and neuronal fate in prairie vole brain progenitor cells cultured in vitro: effects by social exposure and sexual dimorphism
    Daniela Ávila-González, Italo Romero-Morales, Lizette Caro, Alejandro Martínez-Juárez, Larry J. Young, Francisco Camacho-Barrios, Omar Martínez-Alarcón, Analía E. Castro, Raúl G. Paredes, Néstor F. Díaz, Wendy Portillo
    Biology of Sex Differences.2023;[Epub]     CrossRef
  • Anterior pituitary gland synthesises dopamine from l‐3,4‐dihydroxyphenylalanine (l‐dopa)
    Santiago Jordi Orrillo, Nataly de Dios, Antonela Sofía Asad, Fernanda De Fino, Mercedes Imsen, Ana Clara Romero, Sandra Zárate, Jimena Ferraris, Daniel Pisera
    Journal of Neuroendocrinology.2020;[Epub]     CrossRef
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Review Articles
Recent Progress in the Medical Therapy of Pituitary Tumors
Fabienne Langlois, Shirley McCartney, Maria Fleseriu
Endocrinol Metab. 2017;32(2):162-170.   Published online May 19, 2017
DOI: https://doi.org/10.3803/EnM.2017.32.2.162
  • 6,390 View
  • 69 Download
  • 27 Web of Science
  • 27 Crossref
AbstractAbstract PDFPubReader   ePub   

Management of pituitary tumors is multidisciplinary, with medical therapy playing an increasingly important role. With the exception of prolactin-secreting tumors, surgery is still considered the first-line treatment for the majority of pituitary adenomas. However, medical/pharmacological therapy plays an important role in controlling hormone-producing pituitary adenomas, especially for patients with acromegaly and Cushing disease (CD). In the case of non-functioning pituitary adenomas (NFAs), pharmacological therapy plays a minor role, the main objective of which is to reduce tumor growth, but this role requires further studies. For pituitary carcinomas and atypical adenomas, medical therapy, including chemotherapy, acts as an adjuvant to surgery and radiation therapy, which is often required to control these aggressive tumors. In the last decade, knowledge about the pathophysiological mechanisms of various pituitary adenomas has increased, thus novel medical therapies that target specific pathways implicated in tumor synthesis and hormonal over secretion are now available. Advancement in patient selection and determination of prognostic factors has also helped to individualize therapy for patients with pituitary tumors. Improvements in biochemical and “tumor mass” disease control can positively affect patient quality of life, comorbidities and overall survival. In this review, the medical armamentarium for treating CD, acromegaly, prolactinomas, NFA, and carcinomas/aggressive atypical adenomas will be presented. Pharmacological therapies, including doses, mode of administration, efficacy, adverse effects, and use in special circumstances are provided. Medical therapies currently under clinical investigation are also briefly discussed.

Citations

Citations to this article as recorded by  
  • Multiomics Approach to Acromegaly: Unveiling Translational Insights for Precision Medicine
    Kyungwon Kim, Cheol Ryong Ku, Eun Jig Lee
    Endocrinology and Metabolism.2023; 38(5): 463.     CrossRef
  • PDCD10 promotes the aggressive behaviors of pituitary adenomas by up-regulating CXCR2 and activating downstream AKT/ERK signaling
    Jingdian Liu, Junwen Wang, Weidong Tian, Yu Xu, Ran Li, Kai Zhao, Chao You, Yuan Zhu, Joerg Walter Bartsch, Hongquan Niu, Huaqiu Zhang, Kai Shu, Ting Lei
    Aging.2022; 14(15): 6066.     CrossRef
  • LncRNA MYMLR promotes pituitary adenoma development by upregulating carbonyl reductase 1 via sponging miR-197-3p
    Tuo Wang, Ping Mao, Yan Zhang, Bo Cui, Mao-De Wang, Ya Li, Ke Gao
    Anti-Cancer Drugs.2022; 33(10): 1058.     CrossRef
  • Comparative Proteomic Study Shows the Expression of Hint-1 in Pituitary Adenomas
    Carolina Carrillo-Najar, Daniel Rembao-Bojórquez, Martha L. Tena-Suck, Sergio Zavala-Vega, Noemí Gelista-Herrera, Miguel A. Ramos-Peek, Juan L. Gómez-Amador, Febe Cazares-Raga, Fidel de la Cruz Hernández-Hernández, Alma Ortiz-Plata
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  • Recent Understanding and Future Directions of Recurrent Corticotroph Tumors
    José Miguel Hinojosa-Amaya, César Ernesto Lam-Chung, Daniel Cuevas-Ramos
    Frontiers in Endocrinology.2021;[Epub]     CrossRef
  • What to Do with Incidentally Discovered Pituitary Abnormalities?
    Fabienne Langlois, Maria Fleseriu
    Medical Clinics of North America.2021; 105(6): 1081.     CrossRef
  • Aggressive prolactinoma (Review)
    Ana Valea, Florica Sandru, Aida Petca, Mihai Dumitrascu, Mara Carsote, Razvan-Cosmin Petca, Adina Ghemigian
    Experimental and Therapeutic Medicine.2021;[Epub]     CrossRef
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    O. A. Beylerli, Zhao Shiguang, I. F. Gareev, Chen Xin
    Creative surgery and oncology.2020; 9(4): 311.     CrossRef
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    Cristina Capatina, José Miguel Hinojosa-Amaya, Catalina Poiana, Maria Fleseriu
    Expert Review of Endocrinology & Metabolism.2020; 15(5): 321.     CrossRef
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    Ashraf Abdali, Ludmila Astafeva, Pavel Kalinin, Yuriy Trunin, I. V. Chernov, G. E. Сhmutin, Abdali Badshahzar, GulSharif Sherzad, Simfukwe Keith
    Vestnik nevrologii, psihiatrii i nejrohirurgii (Bulletin of Neurology, Psychiatry and Neurosurgery).2020; (10): 67.     CrossRef
  • Effects of Oxytocin on Cell Proliferation in a Corticotroph Adenoma Cell Line
    Jung Soo Lim, Young Woo Eom, Eun Soo Lee, Hyeong Ju Kwon, Ja-Young Kwon, Junjeong Choi, Choon Hee Chung, Young Suk Jo, Eun Jig Lee
    Endocrinology and Metabolism.2019; 34(3): 302.     CrossRef
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    Domenico Solari, Rosario Pivonello, Chiara Caggiano, Elia Guadagno, Carmela Chiaramonte, Giovanni Miccoli, Luigi M. Cavallo, Marialaura Del Basso De Caro, Annamaria Colao, Paolo Cappabianca
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    World Neurosurgery.2019; 121: e45.     CrossRef
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Close layer
Adrenal gland
Clinical Guidelines for the Diagnosis and Treatment of Cushing's Disease in Korea
Kyu Yeon Hur, Jung Hee Kim, Byung Joon Kim, Min-Seon Kim, Eun Jig Lee, Sung-Woon Kim
Endocrinol Metab. 2015;30(1):7-18.   Published online March 27, 2015
DOI: https://doi.org/10.3803/EnM.2015.30.1.7
  • 8,683 View
  • 203 Download
  • 11 Web of Science
  • 16 Crossref
AbstractAbstract PDFPubReader   

Cushing's disease (CD) is a rare disorder characterized by the overproduction of adrenocorticotropic hormone due to a pituitary adenoma that ultimately stimulates excessive cortisol secretion from the adrenal glands. Prior to the detection of pituitary adenomas, various clinical signs of CD such as central obesity, moon face, hirsutism, and facial plethora are usually already present. Uncontrolled hypercortisolism is associated with metabolic, cardiovascular, and psychological disorders that result in increased mortality. Hence, the early detection and treatment of CD are not only important but mandatory. Because its clinical manifestations vary from patient to patient and are common in other obesity-related conditions, the precise diagnosis of CD can be problematic. Thus, the present set of guidelines was compiled by Korean experts in this field to assist clinicians with the screening, diagnoses, and treatment of patients with CD using currently available tests and treatment modalities.

Citations

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    Aysun Seker, Dilek Gogas Yavuz
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    Jong Chul Won, Ki-Hyun Baek
    Endocrinology and Metabolism.2022; 37(6): 851.     CrossRef
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    L. I. Astafyeva, I. V. Chernov, I. V. Chekhonin, E. I. Shults, I. N. Pronin, P. L. Kalinin
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Case Report
Thyroid
Medullary Thyroid Carcinoma with Ectopic Adrenocorticotropic Hormone Syndrome
Hong Seok Choi, Min Joo Kim, Chae Ho Moon, Jong Ho Yoon, Ha Ra Ku, Geon Wook Kang, Im Il Na, Seung-Sook Lee, Byung-Chul Lee, Young Joo Park, Hong Il Kim, Yun Hyi Ku
Endocrinol Metab. 2014;29(1):96-100.   Published online March 14, 2014
DOI: https://doi.org/10.3803/EnM.2014.29.1.96
  • 4,528 View
  • 41 Download
  • 4 Web of Science
  • 5 Crossref
AbstractAbstract PDFPubReader   

Ectopic adrenocorticotropic hormone (ACTH) syndrome is caused most frequently by a bronchial carcinoid tumor or by small cell lung cancer. Medullary thyroid carcinoma (MTC) is a rare etiology of ectopic ACTH syndrome. We describe a case of Cushing syndrome due to ectopic ACTH production from MTC in a 48-year-old male. He was diagnosed with MTC 14 years ago and underwent total thyroidectomy, cervical lymph node dissection and a series of metastasectomies. MTC was confirmed by the pathological examination of the thyroid and metastatic mediastinal lymph node tissues. Two years after his last surgery, he developed Cushingoid features, such as moon face and central obesity, accompanied by uncontrolled hypertension and new-onset diabetes. The laboratory results were compatible with ectopic ACTH syndrome. A bilateral adrenalectomy improved the clinical and laboratory findings that were associated with Cushing syndrome. This is the first confirmed case of ectopic ACTH syndrome caused by MTC in Korea.

Citations

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  • Medullary thyroid carcinoma with ACHT-dependent Cushing's syndrome: Therapeutic possibilities
    Dušan Ilić, Sanja Ognjanović, Bojana Popović, Valentina Elezović-Kovačević, Milica Opalić-Palibrk, Lena Radić, Katarina Krstić, Đuro Macut
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    Won-Young Lee
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Close layer
Review Article
ACTH-Independent Macronodular Adrenal Hyperplasia.
Livia M Mermejo, Tania L Mazzuco, Solange Grunenwald, Maria Candida B V Fragoso, Isabelle Bourdeau, Andre Lacroix
Endocrinol Metab. 2011;26(1):1-11.   Published online March 1, 2011
DOI: https://doi.org/10.3803/EnM.2011.26.1.1
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AbstractAbstract PDF
ACTH-independent macronodular adrenal hyperplasia (AIMAH) is an uncommon cause of Cushing's syndrome (CS). The pathophysiology of this disorder is heterogeneous in its molecular origin and also in its clinical presentation. AIMAH can present mainly as an incidental radiological finding with sub-clinical CS or rarely with overt CS. In a few familial cases reported with AIMAH, specific aberrant G-protein coupled receptors were expressed in the adrenals of all affected members, but sporadic cases are more common. The aberrant adrenal function of G-protein coupled receptors can lead to cell proliferation and abnormal regulation of steroidogenesis. Unilateral or bilateral adrenalectomy has been the most frequently used treatment for this adrenal disorder; alternatively, the identification of aberrant receptors using in vivo protocol of investigation can offer specific pharmacological approach to control abnormal steroidogenesis and possibly prevent AIMAH progression.

Citations

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  • Cushing Syndrome Caused by ACTH-independent Macronodular Adrenal Hyperplasia
    Yeon-Kyung Choi, In-Ryang Hwang, Jung-Bum Seo, Ji Yun Jeong, Jung-Guk Kim
    The Korean Journal of Medicine.2017; 92(2): 190.     CrossRef
  • Primary bilateral adrenal nodular disease with Cushing's syndrome: varying aetiology
    Kush Dev Singh Jarial, Rama Walia, Uma Nahar, Anil Bhansali
    BMJ Case Reports.2017; : bcr-2017-220154.     CrossRef
  • Multiple aberrant hormone receptors in Cushing's syndrome
    Nada El Ghorayeb, Isabelle Bourdeau, André Lacroix
    European Journal of Endocrinology.2015; 173(4): M45.     CrossRef
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Case Reports
Ectopic ACTH Syndrome with Bilateral Pheochromocytoma in Multiple Endocrine Neoplasia Type 2A.
Ji Mi Moon, Yoon Jung Kim, Young Jin Seo, Hye Yoon Choi, Joo Hyong Kim, Ju Ri Park, Yun Jeong Lee, Hee Young Kim, Sin Gon Kim, Dong Seop Choi
J Korean Endocr Soc. 2009;24(4):265-271.   Published online December 1, 2009
DOI: https://doi.org/10.3803/jkes.2009.24.4.265
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AbstractAbstract PDF
Multiple endocrine neoplasia type 2A (MEN 2A) is an autosomal dominant syndrome characterized by the presence of medullary thyroid carcinoma, pheochromocytoma, and hyperparathyroidism. MEN 2A arises due to a germline missense mutation of the RET proto-oncogene. Specific RET mutation analysis has revolutionized the diagnosis and therapy of this disorder, and early thyroidectomy may have lowered the morbidity and mortality associated with these diseases. Ectopic adrenocorticotropic hormone (ACTH) syndrome is characterized by hypercortisolism due to the hypersecretion of ACTH outside of the pituitary gland; the most common causes are malignancies, but rarely adrenal pheochromocytoma may be the cause.

Citations

Citations to this article as recorded by  
  • Pheochromocytoma With High Adrenocorticotropic Hormone Production Capacity Without Pigmentation and Cushingoid Symptoms: A Case Report With a Literature Review
    Gen Mizutani, Masashi Isshiki, Eisuke Shimizu, Daigo Saito, Akira Shimada
    Cureus.2024;[Epub]     CrossRef
  • Ectopic ACTH- and/or CRH-Producing Pheochromocytomas
    Patrick F Elliott, Thomas Berhane, Oskar Ragnarsson, Henrik Falhammar
    The Journal of Clinical Endocrinology & Metabolism.2021; 106(2): 598.     CrossRef
  • Severe Cushing Syndrome Due to an ACTH-Producing Pheochromocytoma: A Case Presentation and Review of the Literature
    Jenan N Gabi, Maali M Milhem, Yara E Tovar, Emhemmid S Karem, Alaa Y Gabi, Rodhan A Khthir
    Journal of the Endocrine Society.2018; 2(7): 621.     CrossRef
  • Adrenocorticotropic hormone (ACTH)-producing pheochromocytoma presented as Cushing syndrome and complicated by invasive aspergillosis
    Jae Ho Cho, Da Eun Jeong, Jae Young Lee, Jong Geol Jang, Jun Sung Moon, Mi Jin Kim, Ji Sung Yoon, Kyu Chang Won, Hyoung Woo Lee
    Yeungnam University Journal of Medicine.2015; 32(2): 132.     CrossRef
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A Case of Pneumocystis Carinii Pneumonia and Cutaneous Nocardiosis Associated with Ectopic ACTH Syndrome.
Hee Sun Jung, Seung Hwan Lee, Hee Yeon Kim, Eun Ho Chu, Kang Woo Lee, Mi Ja Kang, Ji Hyun Kim, Hyuk Sang Kwon, Kun Ho Yoon, Bong Yun Cha, Ho Young Son
J Korean Endocr Soc. 2008;23(1):44-50.   Published online February 1, 2008
DOI: https://doi.org/10.3803/jkes.2008.23.1.44
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AbstractAbstract PDF
Cushing's syndrome is characterized by hypercortisolism and impairment of cell-mediated immune function, increasing the risk of opportunistic infection, as occurs in other immunocompromised groups such as organ transplant recipients, patients with lymphoreticular malignancy, or acquired immunodeficiency syndrome. We report a case of a 68-year-old woman who was affected with pneumocystis carinii pneumonia (PCP) 8 years ago and cutaneous nocardiosis this year. The patient was diagnosed with ectopic ACTH syndrome 8 years ago, but no ectopic ACTH secreting focus was found in radiologic imaging studies. She recovered from PCP and cutaneous nocardiosis after antibiotic (trimethoprim-sulfamethoxazole) administration. After the infection resolved, she underwent laparoscopic bilateral adrenalectomy and was not longer hypercortisolemic.
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A Case of Ectopic ACTH Syndrome Associated with Small Cell Lung Cancer Presented with Hypokalemia.
Hong Jun Yang, Hea Jung Sung, Ji Eun Kim, Hyo Jin Lee, Jin Min Park, Chan Kwon Park, Eun Suk Roh, Jae Hyung Cho, Seung Hyun Ko, Ki Ho Song, Yu Bai Ahn
J Korean Endocr Soc. 2007;22(5):359-364.   Published online October 1, 2007
DOI: https://doi.org/10.3803/jkes.2007.22.5.359
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AbstractAbstract PDF
We report a case of a 73-year-old female patient who was diagnosed with ectopic ACTH syndrome caused by small cell lung cancer. We initially presumed that the patient was in a state of mineralocorticoid excess, because she had hypertension and hypokalemic alkalosis. This was however excluded because her plasma renin activity was not suppressed and her plasma aldosterone/plasma renin activity ratio was below 25. Moreover, her 24 hour urine free cortisol level was elevated and her serum cortisol levels after a low dose dexamethasone suppression test, were not suppressed. Furthermore, her basal plasma ACTH and serum cortisol levels increased and her serum cortisol level after a high dose dexamethasone suppression test was not suppressed. We performed studies to identify the source of ectopic ACTH syndrome and found a 3 cm-sized mass in the patient's right lower lobe of her lung, which was eventually diagnosed as small cell lung cancer following a bronchoscopic biopsy. In conclusion, Cushing's syndrome, and in particular ectopic ACTH syndrome, must be considered in the differential diagnosis of mineralocorticoid-induced hypertension. The excessive cortisol saturates the 11beta-hydroxysteroid dehydrogenase type 2 (11beta-HSD2) activity, which in turn, inactivates the conversion of cortisol to cortisone in the renal tubules. Moreover, excessive cortisol causes binding to the mineralocorticoid receptors, causing mineralocorticoid hypertension, characterized by severe hypercortisolism.

Citations

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  • Emergencia hipertensiva como debut de síndrome de Cushing paraneoplásico
    E. Rubio González, M. de Valdenebro Recio, M.I. Galán Fernández
    Hipertensión y Riesgo Vascular.2024; 41(2): 135.     CrossRef
  • Management of small cell lung cancer complicated with paraneoplastic Cushing’s syndrome: a systematic literature review
    Yanlong Li, Caiyu Li, Xiangjun Qi, Ling Yu, Lizhu Lin
    Frontiers in Endocrinology.2023;[Epub]     CrossRef
  • Ectopic Cushing Syndrome in Adenocarcinoma of the Lung: Case Report and Literature Review
    Rana Al-Zakhari, Safa Aljammali, Basma Ataallah, Svetoslav Bardarov, Philip Otterbeck
    Cureus.2021;[Epub]     CrossRef
  • A Case of Ectopic Adrenocorticotropic Hormone Syndrome in Small Cell Lung Cancer
    Chaiho Jeong, Jinhee Lee, Seongyul Ryu, Hwa Young Lee, Ah Young Shin, Ju Sang Kim, Joong Hyun Ahn, Hye Seon Kang
    Tuberculosis and Respiratory Diseases.2015; 78(4): 436.     CrossRef
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A Case of Silent Corticotroph-cell Adenoma with Elevated Serum ACTH.
Jeong Geun Moon, So Young Park, Byoung Chul Cho, Jung Min Lee, Si Hoon Lee, Yoo Mee Kim, Yu Mie Rhee, Bong Soo Cha, Hyun Chul Lee, Sung Kil Lim
J Korean Endocr Soc. 2004;19(5):535-541.   Published online October 1, 2004
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AbstractAbstract PDF
A 48 year-old man was referred to our Department with a headache, and also presented with an elevated serum ACTH level, but without an elevated serum cortisol. Although there was no clinical evidence of Cushing's syndrome, a brain CT and MRI showed a 4x4.5 cm sized pituitary mass, which was successfully removed by a transsphenoidal approach (TSA). A histopathological examination revealed the mass to have an ACTH positive reaction. Therefore, through hormonal and pathological evaluation, a silent corticotroph-cell adenoma (SCCA), with an elevated serum ACTH level, was diagnosed. Although reports on SCCA have been recently increased, this case is reported because these kinds of tumor are still rare, and those SCCA with an elevated serum ACTH even more so
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Original Article
Diagnostic Value of 1microgram Rapid ACTH Stimulation Test According to the Timing of Sampling of Serum Cortisol in Patients with Suspected Central Adrenal Insufficiency.
Sang Wan Kim, Young Min Cho, Do Joon Park, Chan Soo Shin, Kyung Soo Park, Seong Yeon Kim, Bo Youn Cho, Hong Kyu Lee
J Korean Endocr Soc. 2004;19(1):33-41.   Published online February 1, 2004
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AbstractAbstract PDF
BACKGROUND
Traditional testing of the hypothalamic-pituitary-adrenal axis function has essentially relied upon the insulin tolerance test or the metyrapone test(both tests are not only uncomfortable, but are also dangerous). The standard ACTH stimulation test uses an extremely supra-physiologic amount(250f microgram) of ACTH to evaluate a physiological response, which may result in a false normal response. The 1microgram rapid ACTH stimulation test is more physiological and more sensitive than the standard test, but there exist much controversy about when the serum cortisol should be measured or what the most appropriate cut-off point is for normality or whether the 1microgram ACTH commercial solution is needed. The aims of this study were to investigate 1) whether 1microgram of ACTH is an appropriate amount to stimulate the adrenal gland of patients that have suspected central adrenal insufficiency compared with insulin tolerance test(ITT) and 2) the diagnostic value of the 1microgram rapid ACTH stimulation test according to timing of sampling of serum cortisol. METHODS: In order to evaluate the dose-response relationship between ACTH and cortisol, we performed the ITT in 77 patients with suspected central adrenal insufficiency with serial measurements of serum cortisol and plasma ACTH. We drew the blood samples in 10 min intervals between 10 and 60 min after the administration of 1microgram ACTH in 39 patients with central adrenal insufficiency and in 38 pituitary control patients with pituitary. ITT was used to confirm the diseases for the patients of central adrenal insufficiency, but for pituitary control patients, the ITT indicated normality in the patients. Also, all subjects underwent the 250microgram rapid ACTH stimulation test, and we compared the diagnostic value of the 1microgram ACTH stimulation test with the 250microgram test. RESULTS: 1) The plasma ACTH level after the 1microgram ACTH stimulation test, even if it was be assumed as approximately 300pg/mL, was expected to be sufficient enough to stimulate the adrenal cortex normally(serum cortisol levels >18microgram/dL) compared to the plasma ACTH level in the ITT. 2) The sensitivity and specificity of the 1microgram rapid ACTH stimulation test was highest with 92.3% and 84.2%, respectively, when serum cortisol levels were measured at 20, 30, and 40 min after the ACTH injection. The 1microgram rapid ACTH stimulation test was more sensitive than the 250microgram ACTH test(sensitivity: 92.3%, specificity: 71.8%). CONCLUSION: The 1microgram rapid ACTH stimulation test was more sensitive test in patients with suspected central adrenal insufficiency, and blood samples for cortisol levels should be drawn at 20, 30, and 40 min after ACTH administration.
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Case Report
A Case of AVP Dependent Bilateral Macronodular Adrenal Hyperplasia.
Hyun Jin Kim, Se Hwa Kim, Yu Mie Rhee, Sung Eun Kim, Chul Woo Ahn, Bong Soo Cha, Kyung Rae Kim, Hyun Chul Lee, Kap Bum Huh, Sung Kil Lim
J Korean Endocr Soc. 2002;17(4):603-609.   Published online August 1, 2002
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AbstractAbstract PDF
Adrenocorticotropin (ACTH) independent bilateral macronodular adrenal hyperplasia (AIMAH) is a rare form of Cushing's syndrome, in which unique endocrinological, clinical and histopathological features have been described. In AIMAH, cortisol secretion is autonomous and independent of ACTH, thus plasma ACTH levels are persistently suppressed. Various etiological mechanisms have been proposed to explain the development of AIMAH, the development of aberrant adrenal sensitivity to gastric inhibitory polypeptide (GIP), vasopressin, beta-adrenergic receptor agonists or the presence of circulating adrenal stimulating immunoglobulins have been suggested. We report on a 46-year-old female who had Cushing's syndrome, due to AIMAH, with a positive response to vasopressin.
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