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Bao-Ping Wang, Lei-Lei Yang, Hao Wang, Qing He, Zhong-Shu Ma, Yi Lin, Chang-Xin Jiang, Hao-Ran Sun, Ming Liu
BACKGROUND: Pheochromocytoma, especially for noncatecholamine-secreting pheochromocytoma, is an extremely rare cause of ectopic corticotrophin-releasing hormone (CRH) syndrome. CASE PRESENTATION: A 27-year-old Chinese woman was administered dexamethasone for a skin allergy, but her general condition rapidly deteriorated over a month. She was subsequently hospitalized for typical clinical features of Cushing's syndrome. Endocrinological investigation confirmed severe hypercortisolism along with elevated plasma adrenocorticotropin hormone (ACTH)...
June 19, 2018: BMC Endocrine Disorders
Maria Francesca Cassarino, Alberto G Ambrogio, Andrea Cassarino, Maria Rosa Terreni, Davide Gentilini, Antonella Sesta, Francesco Cavagnini, Marco Losa, Francesca Pecori Giraldi
ACTH-secreting pituitary adenomas give rise to a severe endocrinological disorder, i.e., Cushing's disease, with multifaceted clinical presentation and treatment outcomes. Experimental studies suggested that disease variability is inherent to the pituitary tumor, thus pointing to the need for further studies into tumor biology. Aim of the present study was to evaluate transcriptome expression pattern in a large series of ACTH-secreting pituitary adenoma specimens, in order to identify molecular signatures of these tumors...
June 19, 2018: Journal of Neuroendocrinology
Mattia Barbot, Filippo Ceccato, Carla Scaroni
Associated with important comorbidities that significantly reduce patients' overall wellbeing and life expectancy, Cushing's disease (CD) is the most common cause of endogenous hypercortisolism. Glucocorticoid excess can lead to diabetes, and although its prevalence is probably underestimated, up to 50% of patients with CD have varying degrees of altered glucose metabolism. Fasting glycemia may nevertheless be normal in some patients in whom glucocorticoid excess leads primarily to higher postprandial glucose levels...
2018: Frontiers in Endocrinology
Constanza Navarro Moreno, Amaury Delestienne, Etienne Marbaix, Selda Aydin, Konstanze Hörtnagel, Sarah Lechner, Yves Sznajer, Véronique Beauloye, Dominique Maiter, Philippe A Lysy
Cushing syndrome (CS) is a rare disease in children, frequently associated with subtle or periodic symptoms that may delay its diagnosis. Weight gain and growth failure, the hallmarks of hypercortisolism in pediatrics, may be inconsistent, especially in ACTH-independent forms of CS. Primary pigmented nodular adrenocortical disease (PPNAD) is the rarest form of ACTH-independent CS, and can be associated with endocrine and nonendocrine tumors, forming the Carney complex (CNC). Recently, phenotype/genotype correlations have been described with particular forms of CNC where PPNAD is isolated or associated only with skin lesions...
June 15, 2018: Hormone Research in Pædiatrics
Rowan S Hardy, Hong Zhou, Markus J Seibel, Mark S Cooper
Osteoporosis associated with long-term glucocorticoid therapy remains a common and serious bone disease. In addition, in recent years it has become clear that more subtle states of endogenous glucocorticoid excess may have a major impact on bone health. Adverse effects can be seen with mild systemic glucocorticoid excess but there is also evidence of tissue-specific regulation of glucocorticoid action within bone as a mechanism of disease. This review article will examine a) the role of endogenous glucocorticoids in normal bone physiology, b) the skeletal effects of endogenous glucocorticoid excess in the context of endocrine conditions such as Cushing's disease/syndrome and autonomous cortisol secretion (subclinical Cushing's syndrome), and c) the actions of therapeutic (exogenous) glucocorticoids on bone...
June 13, 2018: Endocrine Reviews
Justine C R Shotton, William S M Justice, Francisco J Salguero, Alan Stevens, Barbara Bacci
  Pituitary pars intermedia dysfunction (PPID), also known as equine Cushing's disease, is widely reported in middle-aged to older domestic equids but to date reported in only one nondomestic equid, the onager ( Equus hemionus onager). This case series reports clinical, hematological, and pathological findings consistent with PPID in two further equid species: one Chapman's zebra ( Equus quagga chapmani) and five Przewalski's horses ( Equus ferus przewalskii). The case series reports basal adrenocorticotropic hormone (ACTH) testing as a method to diagnose and monitor PPID in zoological equids and the use of pergolide mesylate to reduce basal ACTH concentration and reduce clinical signs associated with PPID...
June 2018: Journal of Zoo and Wildlife Medicine: Official Publication of the American Association of Zoo Veterinarians
Mengxue Yang, Biao Long, Jie Xu, Jie Yu, Xianwen Li, Fanhao Ye, Bo Yang, Yulan Liao, Sicheng Li, Ya Li, Xue Zhou
Carney complex (CNC) is a rare genetic disease. Here, we report a case of CNC and explore clinical manifestations and gene mutation studies of CNC. A male patient with CNC at the age of 16 yr was admitted to Affiliated Hospital of Zunyi Medical University in July, 2015. Although the patient had typical signs of Cushing's syndrome, he also presented with certain rare signs of Cushing's syndrome, such as "freckle-like" scattered spots of pigmentation on the face and around the lips. In addition, concomitant severe osteoporosis led to flattened vertebrae and the compression of corresponding levels of the spinal cord...
April 2018: Iranian Journal of Public Health
Ruchika Goel, Eshan U Patel, Melissa M Cushing, Steven M Frank, Paul M Ness, Clifford M Takemoto, Ljiljana V Vasovic, Sujit Sheth, Marianne E Nellis, Beth Shaz, Aaron A R Tobian
Importance: Increasing evidence supports the role of red blood cells (RBCs) in physiological hemostasis and pathologic thrombosis. Red blood cells are commonly transfused in the perioperative period; however, their association with postoperative thrombotic events remains unclear. Objective: To examine the association between perioperative RBC transfusions and postoperative venous thromboembolism (VTE) within 30 days of surgery. Design, Setting, and Participants: This analysis used prospectively collected registry data from the American College of Surgery National Surgical Quality Improvement Program (ACS-NSQIP) database, a validated registry of 525 teaching and nonteaching hospitals in North America...
June 13, 2018: JAMA Surgery
Francesco Ferraù, Márta Korbonits
Cushing's syndrome (CS), including visceral obesity, dyslipidemia, hypertension and diabetes among its many manifestations, is "a model" of metabolic syndrome. Glucocorticoid (GC) excess, through a combination of effects on liver, muscle, adipose tissue and pancreas, increases gluconeogenesis and impairs insulin sensitivity, leading to carbohydrate abnormalities. Dyslipidemia is a common finding in CS as a consequence of GC-related increased lipolysis, lipogenesis and adipogenesis. CS patients experience typical changes in body composition, with fat redistribution resulting in accumulation of visceral adipose tissue...
2018: Frontiers of Hormone Research
Wakako Fujiwara, Tomohiro Haruki, Yoshiteru Kidokoro, Takashi Ohno, Yohei Yurugi, Ken Miwa, Yuji Taniguchi, Hiroshige Nakamura
BACKGROUND: Ectopic ACTH-producing thymic carcinoid tumors are rare, but often behave aggressively with local invasion and distant metastasis. We herein report a case of ACTH-producing thymic typical carcinoid tumor with lymph node metastasis treated by surgery and postoperative radiation therapy. CASE PRESENTATION: A 61-year-old woman was admitted to be evaluated for hypoglycemia and hypokalemia. Laboratory data revealed elevation of serum cortisol and ACTH levels...
June 11, 2018: Surgical Case Reports
Lin Lu, Yu Yue Zhao, Hong Bo Yang, Xin Lun Tian, Zuo Jun Xu, Zhao Lin Lu
BACKGROUND: Patients with Cushing's disease (CD) with hypercortisolism have an increased risk of opportunistic infection. However, most CD patients exposed to infections are diagnostic latency, leading to a poor prognosis. METHODS: Six patients in our hospital and an additional six patients in the literature were included in this study. Clinical information of CD patients with pulmonary Cryptococcus neoformans are reviewed. RESULTS: The average baseline total cortisol and ACTH in serum at 8 am of all the patients was 44...
June 5, 2018: Journal of the Formosan Medical Association, Taiwan Yi Zhi
Mariana Fuertes, Julieta Tkatch, Josefina Rosmino, Leandro Nieto, Mirtha Adriana Guitelman, Eduardo Arzt
Cushing's disease (CD) is an endocrine disorder originated by a corticotroph tumor. It is linked with high mortality and morbidity due to chronic hypercortisolism. Treatment goals are to control cortisol excess and achieve long-term remission, therefore, reducing both complications and patient's mortality. First-line of treatment for CD is pituitary's surgery. However, 30% of patients who undergo surgery experience recurrence in long-term follow-up. Persistent or recurrent CD demands second-line treatments, such as pituitary radiotherapy, adrenal surgery, and/or pharmacological therapy...
2018: Frontiers in Endocrinology
Anna Babinska, Mariusz Kaszubowski, Krzysztof Sworczak
Due to the fact that overweight or obesity is accompanied by hormonally active adrenal tumors: Cushing Syndrome-(CS) and Subclinical Cushing Syndrome (SCS), it is of high interest the correlation between different adipokines and cytokines secreted by adipose tissue, with metabolic disorders and hormonal activity in this group. Even in non-functioning adrenal incidentalomas (NFAI) elevated risk for cardiovascular disease and metabolic syndrome was demonstrated. The aim of the study was to investigate plasma adiponectin, leptin, resistin, tumor necrosis factor α (TNFα), interleukin 6 (IL6) and monocyte chemoattractant protein 1 (MCP1) levels in patients with NFAIs and healthy subjects...
June 5, 2018: Endocrine Journal
Andrea Osswald, Timo Deutschbein, Christina Maria Berr, Eva Plomer, Anne Mickisch, Katrin Ritzel, Jochen Schopohl, Felix Beuschlein, Martin Fassnacht, Stefanie Hahner, Martin Reincke
OBJECTIVE Aim of our study was to analyze long-term outcome of patients with the ectopic Cushing's syndrome (ECS) compared to patients with Cushing's disease (CD) regarding cardiovascular, metabolic, musculoskeletal and psychiatric co-morbidities. DESIGN Cross-sectional study in patients with ECS and CD in two German academic tertiary-care centers. METHODS Standardized clinical follow-up examination was performed including health-related quality of life (QoL) in 21 ECS patients in long-term remission (≥ 18 months since successful surgery)...
June 6, 2018: European Journal of Endocrinology
Dariush Jahandideh, Brooke Swearingen, Lisa B Nachtigall, Anne Klibanski, Beverly M K Biller, Nicholas A Tritos
OBJECTIVE: To characterize a cohort of patients with cyclic Cushing's disease (CD) in comparison with non-cyclic CD using late night salivary cortisol (LNSC) and examine the diagnostic sensitivity of LNSC in comparison with that of 24 hour urine free cortisol (UFC) in this population. DESIGN: Retrospective study of patients with CD seen in our institution between 2008-2017 PATIENTS: 205 patients, including 17 (8%) with cyclic CD (based on a minimum of 3 peaks and 2 troughs in cortisol levels)...
June 6, 2018: Clinical Endocrinology
Yu Zhao, Patrick R Cushing, David C Smithson, Maria Pellegrini, Alexandre A Pletnev, Sahar Al-Ayyoubi, Andrew V Grassetti, Scott A Gerber, R Kiplin Guy, Dean R Madden
No abstract text is available yet for this article.
June 5, 2018: Bioscience Reports
Lucas Stetzik, Denis Ganshevsky, Michelle N Lende, Laura E Roache, Sergei Musatov, Bruce S Cushing
This study tested the hypothesis that site-specific estrogen receptor alpha (ERα) expression is a critical factor in the expression of male prosocial behavior and aggression. Previous studies have shown that in the socially monogamous prairie vole (Microtus ochrogaster) low levels of ERα expression, in the medial amygdala (MeA), play an essential role in the expression of high levels of male prosocial behavior and that increasing ERα expression reduced male prosocial behavior. We used an shRNA adeno-associated viral vector to knock down/inhibit ERα in the MeA of the polygynous male meadow vole (M...
May 30, 2018: Behavioural Brain Research
Kara Leigh Krajewski, Roman Rotermund, Jörg Flitsch
PURPOSE: Pituitary adenomas are rare in the pediatric population. We present a recent cohort of children operated transsphenoidally on pituitary adenoma from a single center. METHODS: Inclusion criteria were age < 21 years and histopathological diagnosis of adenoma after transsphenoidal surgery. The electronic file was used for prospective and retrospective data collection on symptoms, pituitary function before/after surgical intervention, and surgical complications...
May 30, 2018: Child's Nervous System: ChNS: Official Journal of the International Society for Pediatric Neurosurgery
Rehana Parvin, Erika Noro, Akiko Saito-Hakoda, Hiroki Shimada, Susumu Suzuki, Kyoko Shimizu, Hiroyuki Miyachi, Atsushi Yokoyama, Akira Sugawara
Although therapeutic effects of the peroxisome proliferator-activated receptor gamma (PPAR- γ ) agonists rosiglitazone and pioglitazone against Cushing's disease have been reported, their effects are still controversial and inconsistent. We therefore examined the effects of a novel PPAR- γ agonist, MEKT1, on Pomc expression/ACTH secretion using murine corticotroph-derived AtT20 cells and compared its effects with those of rosiglitazone and pioglitazone. AtT20 cells were treated with either 1 nM~10  μ M MEKT1, rosiglitazone, or pioglitazone for 24 hours...
2018: PPAR Research
Zahraa Abdul Sater, Smita Jha, Raven McGlotten, Iris Hartley, Mustapha El Lakis, Katherine Araque Triana, Lynnette K Nieman
Context: Patients taking exogenous glucocorticoids are at risk of gastrointestinal (GI) complications including peptic ulcer disease with perforation, and gastric bleeding. However, little is known about the GI co-morbidity in patients with endogenous hypercortisolemia. Case descriptions: We describe six patients with endogenous Cushing's Syndrome (CS) who developed sudden perforation of colonic diverticula requiring urgent exploratory laparotomy. Most of these patients shared the following features of Cushing's syndrome: significant skin thinning, severe hypercortisolemia (24-hour urine free cortisol ≥10X the upper limit of normal), ectopic secretion of ACTH, and severe hypokalemia...
May 28, 2018: Journal of Clinical Endocrinology and Metabolism
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