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https://www.readbyqxmd.com/read/28529722/recent-advances-in-understanding-cushing-disease-resistance-to-glucocorticoid-negative-feedback-and-somatic-usp8-mutations
#1
REVIEW
Eleni Daniel, John Newell-Price
Cushing's disease is a rare disease with a characteristic phenotype due to significant hypercortisolism driven by over-secretion of adrenocorticotropic hormone and to high morbidity and mortality if untreated. It is caused by a corticotroph adenoma of the pituitary, but the exact mechanisms leading to tumorigenesis are not clear. Recent advances in molecular biology such as the discovery of somatic mutations of the ubiquitin-specific peptidase 8 ( USP8) gene allow new insights into the pathogenesis, which could be translated into exciting and much-needed therapeutic applications...
2017: F1000Research
https://www.readbyqxmd.com/read/28528397/perioperative-hypothalamic-pituitary-adrenal-function-in-patients-with-silent-corticotroph-adenomas
#2
Abdelle F Cheres, Nadine ElAsmar, Aman Rajpal, Warren R Selman, Baha M Arafah
BACKGROUND: Silent corticotroph adenomas (SCAs) are characterized by strong ACTH immunostaining without clinical manifestations of hypercortisolism. Patients with SCAs often present with mechanical symptoms related to tumor growth. This study investigates the hypothalamic pituitary adrenal axis (HPA) characteristics after adenomectomy in patients with SCAs. METHODS: Biochemical parameters of HPA function were monitored frequently after surgical resection of non-functioning macroadenomas...
May 20, 2017: Pituitary
https://www.readbyqxmd.com/read/28528327/three-quarters-adrenalectomy-for-infantile-onset-cushing-syndrome-due-to-bilateral-adrenal-hyperplasia-in-mccune-albright-syndrome
#3
Tomoyo Itonaga, Hironori Goto, Manabu Toujigamori, Yasuharu Ohno, Seigo Korematsu, Tatsuro Izumi, Satoshi Narumi, Tomonobu Hasegawa, Kenji Ihara
BACKGROUND: Bilateral adrenalectomy is performed in cases with infantile-onset Cushing syndrome due to bilateral adrenal hyperplasia in McCune-Albright syndrome (MAS) because severe Cushing syndrome with heart failure and liver dysfunction can have a lethal outcome. This procedure can completely ameliorate hypercortisolism, although lifetime steroid replacement therapy and steps to prevent adrenal crisis are necessary. Recently, the efficacy of unilateral adrenalectomy has been reported in adult cases of bilateral macronodular adrenal hyperplasia, but there is no consensus regarding the appropriate surgical treatment for bilateral adrenal hyperplasia in MAS...
May 19, 2017: Hormone Research in Pædiatrics
https://www.readbyqxmd.com/read/28515793/does-the-diurnal-cycle-of-cortisol-explain-the-relationship-between-physical-performance-and-cognitive-function-in-older-adults
#4
B Dijckmans, J Tortosa-Martínez, N Caus, G González-Caballero, B Martínez-Pelegrin, C Manchado-Lopez, J M Cortell-Tormo, I Chulvi-Medrano, A Clow
BACKGROUND: Regular physical activity is a promising strategy to treat and prevent cognitive decline. The mechanisms that mediate these benefits are not fully clear but physical activity is thought to attenuate the harmful effects of chronic psychological stress and hypercortisolism on cognition. However, the circadian pattern of cortisol secretion is complex and it is not known which aspects are most closely associated with increased cognitive function and better physical performance...
2017: European Review of Aging and Physical Activity
https://www.readbyqxmd.com/read/28458898/efficacy-of-pasireotide-in-controlling-severe-hypercortisolism-until-cardiac-transplantation
#5
Roberto Attanasio, Liana Cortesi, Daniela Gianola, Claudia Vettori, Fulvio Sileo, Roberto Trevisan
SUMMARY: Cushing's syndrome is associated with increased morbidity and mortality. Although surgery is the first-line treatment, drugs can still play a role as an ancillary treatment to be employed while waiting for surgery, after unsuccessful operation or in patients unsuitable for surgery. We were asked to evaluate a 32-year-old male waiting for cardiac transplantation. Idiopathic hypokinetic cardiomyopathy had been diagnosed since 6 years. He was on treatment with multiple drugs, had a pacemaker, an implantable cardioverter and an external device for the support of systolic function...
2017: Endocrinology, Diabetes & Metabolism Case Reports
https://www.readbyqxmd.com/read/28458897/armc5-mutation-in-a-portuguese-family-with-primary-bilateral-macronodular-adrenal-hyperplasia-pbmah
#6
Teresa Rego, Fernando Fonseca, Stéphanie Espiard, Karine Perlemoine, Jérôme Bertherat, Ana Agapito
SUMMARY: PBMAH is a rare etiology of Cushing syndrome (CS). Familial clustering suggested a genetic cause that was recently confirmed, after identification of inactivating germline mutations in armadillo repeat-containing 5 (ARMC5) gene. A 70-year-old female patient was admitted due to left femoral neck fracture in May 2014, in Orthopedics Department. During hospitalization, hypertension (HTA) and hypokalemia were diagnosed. She presented with clinical signs of hypercortisolism and was transferred to the Endocrinology ward for suspected CS...
2017: Endocrinology, Diabetes & Metabolism Case Reports
https://www.readbyqxmd.com/read/28444400/effects-of-insufficient-sleep-on-pituitary-adrenocortical-response-to-crh-stimulation-in-healthy-men
#7
Aurore Guyon, Lisa L Morselli, Marcella L Balbo, Esra Tasali, Rachel Leproult, Mireille L'Hermite-Baleriaux, Eve Van Cauter, Karine Spiegel
Study Objectives: Severe sleep restriction results in elevated evening cortisol levels. We examined whether this relative hypercortisolism is associated with alterations in the pituitary-adrenocortical response to evening corticotropin-releasing hormone (CRH) stimulation. Methods: Eleven subjects participated in 2 sessions (2 nights of 10h versus 4h in bed) in randomized order. Sleep was polygraphically recorded. After the second night of each session, blood was sampled at 20-min intervals from 09:00 to 24:00 for adrenocorticotropic hormone (ACTH) and cortisol measurements, and perceived stress was assessed hourly...
April 21, 2017: Sleep
https://www.readbyqxmd.com/read/28440767/zinc-alpha-2-glycoprotein-is-overproduced-in-cushing-s-syndrome
#8
Xavier Escoté, Gloria B Aranda, Mireia Mora, Gregori Casals, Joaquim Enseñat, Oscar Vidal, Yaiza Esteban, Irene Halperin, Felicia A Hanzu
INTRODUCTION: Cushing syndrome (CS), an endogenous hypercortisolemic condition with increased cardiometabolic morbidity, leads to development of abdominal obesity, insulin resistance, diabetes and proatherogenic dyslipidemia. Zinc alpha-2 glycoprotein (ZAG) is a recently characterized lipolytic adipokine implicated in regulation of adipose tissue metabolism and fat distribution. In vitro and animal studies suggest that glucocorticoids interact with ZAG secretion and action. To assess the relationship between ZAG and glucocorticoids in a human model of hypercortisolism, circulating ZAG levels were tested in patients with CS and its counterpart controls...
January 2017: Endocrinol Diabetes Nutr
https://www.readbyqxmd.com/read/28438531/altered-gray-and-white-matter-microstructure-in-cushing-s-disease-a-diffusional-kurtosis-imaging-study
#9
Hong Jiang, Na-Ying He, Yu-Hao Sun, Fang-Fang Jian, Liu-Guan Bian, Jian-Kang Shen, Fu-Hua Yan, Si-Jian Pan, Qing-Fang Sun
Exposure to chronic hypercortisolism has multiple adverse effects on brain biology in humans. Cushing's disease (CD) represents a unique and natural human model for examining the effects of hypercortisolism on the brain. This cross-sectional study used Diffusional Kurtosis Imaging (DKI) to investigate the microstructure alterations in both white matter (WM) and gray matter (GM) of CD patients and to determine the relationship of these changes with clinical characteristics. DKI images were obtained from 15 active CD patients...
April 21, 2017: Brain Research
https://www.readbyqxmd.com/read/28435794/cushing-disease-in-a-patient-with-multiple-endocrine-neoplasia-type-2b
#10
Kannan Kasturi, Lucas Fernandes, Martha Quezado, Mary Eid, Leigh Marcus, Prashant Chittiboina, Mark Rappaport, Constantine A Stratakis, Brigitte Widemann, Maya Lodish
CONTEXT: Multiple endocrine neoplasia type 2B (MEN2B) is a rare autosomal-dominant cancer syndrome characterized in part by metastatic medullary thyroid cancer (MTC) and pheochromocytoma. Cushing disease is a rare cause of endogenous hypercortisolism in children. CASE DESCRIPTION: We describe a 21-year-old African-American male who was diagnosed at age 10 with an ACTH-secreting pituitary microadenoma. At age 16 he developed medullary thyroid cancer and was found to have multiple endocrine neoplasia type 2B with the characteristic M918T mutation of the RET proto-oncogene...
June 2017: J Clin Transl Endocrinol Case Rep
https://www.readbyqxmd.com/read/28431167/performance-of-18f-fdg-pet-ct-in-the-characterization-of-adrenal-masses-in-non-cancer-patients-a-prospective-study
#11
Carole Guerin, François Pattou, Laurent Brunaud, Jean-Christophe Lifante, Eric Mirallié, Magalie Haissaguerre, Damien Huglo, Pierre Olivier, Claire Houzard, Catherine Ansquer, Elif Hindié, Anderson Loundou, Cendrine Archange, Antoine Tabarin, Fréderic Sebag, Karine Baumstarck, David Taïeb
Context: Few prospective studies have evaluated the role of 18F-Fluorodeoxyglucose positron emission tomography/computed tomography (18F-FDG PET/CT) in the characterization of adrenal masses. Objective: To assess the performance of 18F-FDG PET/CT in the malignancy diagnosis of adrenal masses in non-cancer patients. Design: Prospective multicenter study. Material and methods: The study population consisted of 87 patients (87 adrenal masses) referred to endocrine surgeons: 56 with mass diameter ≥40 mm and 31 with a diameter <40 mm and of indeterminate nature based on unenhanced and washout CT attenuation densities...
April 20, 2017: Journal of Clinical Endocrinology and Metabolism
https://www.readbyqxmd.com/read/28407311/biological-variability-in-serum-cortisol-concentration-post-adrenocorticotropic-hormone-stimulation-in-healthy-dogs
#12
A Gal, K Weidgraaf, J P Bowden, N Lopez-Villalobos, N J Cave, J P Chambers, F Castillo-Alcala
BACKGROUND: The ACTH stimulation has low sensitivity for the diagnosis of hypercortisolism possibly as a result of biological and analytical variability. HYPOTHESIS/OBJECTIVES: To report the components of biological and analytical variability in serum cortisol concentration post-ACTH stimulation ([cortisol]) in healthy dogs. ANIMALS: Fourteen healthy harrier hound dogs. METHODS: The data were extracted from a separate, prospective, randomized, double-blinded, controlled discovery study in which dogs treated with vehicle control and 4 different doses of cortisone acetate (CA) for 7 days had an ACTH stimulation test performed to confirm the dose-dependent effect of CA...
May 2017: Journal of Veterinary Internal Medicine
https://www.readbyqxmd.com/read/28405879/histopathological-and-genetic-characterization-of-aldosterone-producing-adenomas-with-concurrent-subclinical-cortisol-hypersecretion-a-case-series
#13
Francesco Fallo, Isabella Castellano, Celso E Gomez-Sanchez, Yara Rhayem, Catia Pilon, Valentina Vicennati, Donatella Santini, Valeria Maffeis, Ambrogio Fassina, Paolo Mulatero, Felix Beuschlein, Martin Reincke
PURPOSE: Aldosterone-producing adenomas with concurrent subclinical cortisol hypersecretion are reported in an increasing number of patients. Five aldosterone-producing adenomas from patients with primary aldosteronism and subclinical hypercortisolism were examined. THE AIMS OF OUR STUDY WERE: (1) to analyze pathological features and immunohistochemical expression of CYP11B1 (11β-hydroxylase) and CYP11B2 (aldosterone synthase) in these tumors; (2) to investigate somatic mutations involved in adrenal steroid hypersecretion and/or tumor growth...
April 12, 2017: Endocrine
https://www.readbyqxmd.com/read/28404594/health-related-quality-of-life-of-patients-with-hypothalamic-pituitary-adrenal-axis-dysregulations-a-cohort-study
#14
Charlotte De Bucy, Laurence Guignat, Tanya Niati, Jerome Bertherat, Joel Coste
OBJECTIVE: Health-related quality of life (HrQoL) is increasingly considered to be an important outcome of care for hypothalamic-pituitary-adrenal (HPA) axis dysregulation. The objective of this study was to assess the influence of type of HPA axis dysregulation and cortisol status on HrQOL and its evolution with time and treatment. DESIGN: Prospective cohort study. METHODS: Between September 2007 and April 2014, HrQoL questionnaires were administered during routine management to all patients with HPA axis dysregulation hospitalized in a single department and this was repeated after 6- 12-, 24- and 36-month during standard follow-up...
April 12, 2017: European Journal of Endocrinology
https://www.readbyqxmd.com/read/28393326/psychiatric-symptoms-in-patients-with-cushing-s-syndrome-prevalence-diagnosis-and-management
#15
Alicia Santos, Eugenia Resmini, Juan Carlos Pascual, Iris Crespo, Susan M Webb
Cushing's syndrome (CS) results from chronic exposure to cortisol excess, produced by the adrenal cortex. Hypercortisolism predisposes to psychiatric and neurocognitive disorders, mainly to depression and anxiety disorders. Screening tools to identify psychiatric symptoms are available for clinicians in their daily practice, although a specific diagnosis should be performed by specialists. Even if psychiatric symptoms improve after remission of hypercortisolism, complete recovery may not be achieved. Given the burden of these symptoms, psychiatric or psychological monitoring and treatment should be offered through all phases of CS, with a multidisciplinary approach...
May 2017: Drugs
https://www.readbyqxmd.com/read/28382019/transcriptome-analysis-showed-a-differential-signature-between-invasive-and-non-invasive-corticotrophinomas
#16
Leonardo Jose Tadeu de Araújo, Antonio Marcondes Lerario, Margaret de Castro, Clarissa Silva Martins, Marcello Delano Bronstein, Marcio Carlos Machado, Ericka Barbosa Trarbach, Maria Candida Barisson Villares Fragoso
ACTH-dependent hypercortisolism caused by a pituitary adenoma [Cushing's disease (CD)] is the most common cause of endogenous Cushing's syndrome. CD is often associated with several morbidities, including hypertension, diabetes, osteoporosis/bone fractures, secondary infections, and increased cardiovascular mortality. While the majority (≈80%) of the corticotrophinomas visible on pituitary magnetic resonance imaging are microadenomas (MICs, <10 mm of diameter), some tumors are macroadenomas (MACs, ≥10 mm) with increased growth potential and invasiveness, exceptionally exhibiting malignant demeanor...
2017: Frontiers in Endocrinology
https://www.readbyqxmd.com/read/28377460/diagnostic-tests-for-cushing-s-syndrome-differ-from-published-guidelines-data-from-ercusyn
#17
MULTICENTER STUDY
Elena Valassi, Holger Franz, Thierry Brue, Richard A Feelders, Romana Netea-Maier, Stylianos Tsagarakis, Susan M Webb, Maria Yaneva, Martin Reincke, Michael Droste, Irina Komerdus, Dominique Maiter, Darko Kastelan, Philippe Chanson, Marija Pfeifer, Christian J Strasburger, Miklós Tóth, Olivier Chabre, Antoine Tabarin, Michal Krsek, Carmen Fajardo, Marek Bolanowski, Alicia Santos, John A H Wass, Peter J Trainer
OBJECTIVE: To evaluate which tests are performed to diagnose hypercortisolism in patients included in the European Registry on Cushing's syndrome (ERCUSYN), and to examine if their use differs from the current guidelines. PATIENTS AND METHODS: We analyzed data on the diagnostic tests performed in 1341 patients with Cushing's syndrome (CS) who have been entered into the ERCUSYN database between January 1, 2000 and January 31, 2016 from 57 centers in 26 European countries...
May 2017: European Journal of Endocrinology
https://www.readbyqxmd.com/read/28368467/glucose-metabolism-abnormalities-in-cushing-s-syndrome-from-molecular-basis-to-clinical-management
#18
Carla Scaroni, Marialuisa Zilio, Michelangelo Foti, Marco Boscaro
An impaired glucose metabolism, which often leads to the onset of diabetes mellitus (DM), is a common complication of chronic exposure to exogenous and endogenous glucocorticoid (GC) excess, and plays an important part in contributing to morbidity and mortality in patients with Cushing's syndrome (CS).This article reviews the pathogenesis, epidemiology, diagnosis and management of changes in glucose metabolism associated with hypercortisolism, addressing both the pathophysiological aspects and the clinical and therapeutic implications...
March 20, 2017: Endocrine Reviews
https://www.readbyqxmd.com/read/28364356/diabetes-in-cushing-disease
#19
REVIEW
G Mazziotti, A M Formenti, S Frara, F Maffezzoni, M Doga, A Giustina
PURPOSE OF REVIEW: This review focuses on the pathophysiological and clinical aspects of diabetes mellitus occurring in patients with Cushing disease (CD). RECENT FINDINGS: Insulin resistance and impairment in insulin secretion are both involved in the pathogenesis of glucocorticoid-induced diabetes. Correction of glucocorticoid excess does not always resolve abnormalities of glucose homeostasis, and correction of hyperglycaemia is specifically required. In fact, insulin resistance may persist even after correction of glucocorticoid excess and diabetes needs to be treated for long term...
May 2017: Current Diabetes Reports
https://www.readbyqxmd.com/read/28325824/persistence-of-myopathy-in-cushing-s-syndrome-evaluation-of-the-german-cushing-s-registry
#20
Christina Maria Berr, Mareike R Stieg, Timo Deutschbein, Marcus Quinkler, Ralf Schmidmaier, Andrea Osswald, Nicole Reisch, Katrin Ritzel, Christina Dimopoulou, Julia Fazel, Stefanie Hahner, Gunter K Stalla, Felix Beuschlein, Martin Reincke
BACKGROUND: Cushing's syndrome (CS) is characterized by an excessive secretion of glucocorticoids that results in a characteristic clinical phenotype. One feature of clinical hypercortisolism is breakdown of protein metabolism translating into clinical consequences including glucocorticoid induced myopathy. While surgery is effective in control of cortisol excess, the effect of biochemical remission on muscular function is yet unclear. METHODS: In a cross-sectional study we analyzed 47 patients with CS during the florid phase (ActiveCS)...
March 21, 2017: European Journal of Endocrinology
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