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Zona Batacchi, Nicole K Andeen, Subbulaxmi Trikudanathan
A 62-year-old woman presented with an 11-month history of worsening nasal symptoms of rhinorrhoea, anosmia, nasal congestion and intermittent epistaxis. MRI revealed a large mass in the upper nasal vault. Biopsy of the mass revealed an olfactory neuroblastoma. While waiting resection, she acutely developed severe proximal muscle weakness, lethargy and lower extremity oedema. Blood glucose was elevated, and hypokalaemic metabolic alkalosis was noted. Elevated serum cortisol level of 95.7 µg/dL (8.7-22.4 µg/dL) and markedly elevated 24-hour urinary cortisol level of 6962...
March 13, 2018: BMJ Case Reports
Marloes Langelaan, Jérôme Kisters, Mirjam Oosterwerff-Suiker, Arjen-Kars Boer
Saliva as a diagnostic tool is patient friendly and offers analytical advantages. Hormonal analysis of saliva is not influenced by changes in concentrations of binding globulins as the free concentration of the hormones is measured. Analysis of salivary cortisol is common practice in the diagnostic work-up of hypercortisolism. We investigated the potential role of measuring salivary cortisol when adrenal insufficiency (AI) is suspected, to reduce the numbers of ACTH stimulation tests. Over a period of 6 years, patients undergoing an ACTH stimulation test (tetracosactide, 250 μg) in our hospital were included...
March 12, 2018: Endocrine Connections
Thierry Brue, Vincent Amodru, Frédéric Castinetti
With fewer than 200 reported cases, Cushing's syndrome (CS) in pregnancy remains a diagnostic and therapeutic challenge. In normal pregnancies, misleading signs may be observed such as striae or hypokalemia, while plasma cortisol and urinary free cortisol may rise up to 2-3-fold. While the dexamethasone suppression test is difficult to use, reference values for salivary cortisol appear valid. The predominant cause is adrenal adenoma (sometimes without decreased ACTH), rather than Cushing's disease. There are considerable imaging pitfalls in Cushing's disease...
March 9, 2018: European Journal of Endocrinology
Femke M van Haalen, Elon H C van Dijk, Olaf M Dekkers, Maurice B Bizino, Greet Dijkman, Nienke R Biermasz, Camiel J F Boon, Alberto M Pereira
Objective: Central serous chorioretinopathy (CSC), a specific form of macular degeneration, has been reported as presenting manifestation of Cushing's syndrome. Furthermore, CSC has been associated with both exogenous hypercortisolism and endogenous Cushing's syndrome. It is important to know whether CSC patients should be screened for Cushing's syndrome. Although hypothalamic-pituitary-adrenal (HPA) axis hyperactivity in CSC has been suggested, no detailed evaluation of the HPA axis has been performed in a large cohort of CSC patients...
2018: Frontiers in Endocrinology
Emilia Sbardella, Marianna Minnetti, Denise D'Aluisio, Laura Rizza, Maria Rosaria Di Giorgio, Fabio Vinci, Riccardo Pofi, Elisa Giannetta, Mary Anna Venneri, Annarita Vestri, Sergio Morelli, Andrea Lenzi, Andrea M Isidori
BACKGROUND: Low-grade incomplete post-dexamethasone cortisol suppression in patients with adrenal incidentalomas -recently defined as possible autonomous cortisol secretion (pACS)- has been associated with increased cardiovascular events and mortality. However, prospective studies documenting cardiac abnormalities in these patients are lacking. SUBJECTS AND METHODS: Between July 2016 and September 2017, 71 consecutive patients with adrenal lesions were prospectively screened for hypercortisolism by dexamethasone suppression test (NCT02611258)...
March 6, 2018: European Journal of Endocrinology
Ahsan Zil-E-Ali, Omer Hanif Janjua, Aiza Latif, Muhammad Aadil
Cushing's syndrome is a collection of signs and symptoms due to hypercortisolism. Prolong use of topical steroid may cause this syndrome and suppression of hypothalamic and pituitary function, however such events are more common with oral and parenteral route. There are very few cases of Cushing's syndrome with a topical application amongst which triamcinolone is the rarest drug. We report a case of 11-year-old boy is presented who developed Cushing's disease by topical application. The child had body rashes for which the caregiver consulted a local quack, a topical cream of triamcinolone was prescribed...
January 2018: Journal of Ayub Medical College, Abbottabad: JAMC
Gautam U Mehta, Dale Ding, Amitabh Gupta, Hideyuki Kano, Nathaniel D Sisterson, Nuria Martinez-Moreno, Michal Kršek, Huai-Che Yang, Cheng-Chia Lee, Roman Liščák, Roberto Martinez-Alvarez, L Dade Lunsford, Mary Lee Vance, Jason P Sheehan
Stereotactic radiosurgery (SRS) is frequently used for Cushing's disease (CD) after failed pituitary surgery. Management of patients with persistent CD after failed SRS is complex, as the alternative therapeutic options harbor significant risks. The outcomes of repeat pituitary radiosurgery, however, have not been described. We sought to determine the outcomes of repeat SRS in patients with CD. We pooled data from five institutions participating in the International Gamma Knife Research Foundation for patients with recurrent or persistent CD ≥ 12 months after initial SRS...
March 3, 2018: Journal of Neuro-oncology
Serkan Yener, Mustafa Secil, Omer Demir, Basak Ozgen, Kutsal Yorukoglu
CONTEXT: To investigate whether any association between chemical shift magnetic resonance (MRI) findings, cortisol secretion and pathological findings exist that could predict subclinical hypercortisolism (SCH) in patients with adrenal incidentalomas (AI). DESIGN: Retrospective, cross sectional study in a tertiary centre. PATIENTS: Sixty-eight subjects with AIs and 13 patients with Cushing's syndrome (CS). Patients with AIs were categorized according to cortisol levels post 1mg dexamethasone (post-DST)...
March 1, 2018: Clinical Endocrinology
Geraldine Vitellius, Séverine Trabado, Christine Hoeffel-Fornes, Jerome Bouligand, Antoine Bennet, Frédéric Castinetti, Bénédicte Decoudier, Anne Guiochon-Mantel, Marc Lombes, Brigitte Delemer
Recently discovered mutations of NR3C1 gene, encoding for the Glucocorticoid Receptor (GR), in patients with glucocorticoid resistance and bilateral adrenal incidentalomas prompted us to investigate whether GR mutations might be associated with adrenal hyperplasia. OBJECTIVE: The multicenter French Clinical Research Program (Muta-GR) was set up to determine the prevalence of GR mutations and polymorphisms in patients harboring bilateral adrenal incidentalomas associated with hypertension and/or biological hypercortisolism without clinical Cushing's signs...
February 14, 2018: European Journal of Endocrinology
Liping Yu, Junqing Zhang, Xiaohui Guo, Xiaoyu Chen, Zhisong He, Qun He
To investigate Armadillo repeat-containing 5 (ARMC5) mutations in Chinese patients with familial and sporadic primary bilateral macronodular adrenal hyperplasia (PBMAH), we performed clinical data collection and ARMC5 sequencing for three PBMAH families and 23 sporadic PBMAH patients. ARMC5 pathogenic germline mutations were identified in all 3 PBMAH families. Secondary ARMC5 somatic mutations were found in two adrenal nodules from two PBMAH family members with ARMC5 germline mutations. PBMAH family members with ARMC5 pathogenic germline mutations displayed various clinical manifestations...
2018: PloS One
Prapai Dejkhamron, Karn Wejaphikul, Tuanjit Mahatumarat, Suchaya Silvilairat, Pimlak Charoenkwan, Suwit Saekho, Kevalee Unachak
BACKGROUND: Vitamin D deficiency is common in patients with thalassemia. Vitamin D deficiency could be related to cardiac dysfunction. Increased parathyroid hormone (PTH) is also known to be associated with heart failure. OBJECTIVES: To determine the prevalence of Vitamin D deficiency and to explore the impact of Vitamin D deficiency on cardiac iron and function in patients with transfusion-dependent thalassemia. METHOD: A cross-sectional study in patients with Transfusion-dependent thalassemia was conducted...
January 23, 2018: Pediatric Hematology and Oncology
Bram Peeters, Lies Langouche, Greet Van den Berghe
Critically ill patients have elevated plasma cortisol concentrations, in proportion to illness severity. This was traditionally attributed exclusively to a central activation of the hypothalamus-pituitary axis. However, low rather than high plasma ACTH concentrations have been reported in critically ill patients, with loss of diurnal ACTH and cortisol rhythm. Low ACTH together with high cortisol is referred to as "ACTH-cortisol dissociation." Although cortisol production is somewhat increased with inflammation, a reduced cortisol breakdown explains to a larger extent the hypercortisolism during critical illness...
December 12, 2017: Comprehensive Physiology
Justin M Moore, Elisa Sala, Alvaro Amorin, Hector Martinez, Aprotim C Bhowmik, Steven D Chang, Scott G Soltys, Griffith R Harsh, Laurence Katznelson
BACKGROUND: Surgery is the primary treatment for Cushing disease. When surgery is unsuccessful in normalizing hypercortisolism, adjuvant radiation, such as stereotactic radiosurgery, may be useful to improve biochemical control. METHODS: This retrospective study included a cohort of consecutive patients treated with CyberKnife (CK) radiosurgery for active Cushing disease at Stanford Hospital and Clinics. RESULTS: As first-line treatment, all patients underwent transsphenoidal surgery with histologic demonstration of an adrenocorticotropic hormone-producing pituitary adenoma...
January 31, 2018: World Neurosurgery
Anat Ben-Shlomo, Odelia Cooper
PURPOSE: Silent corticotroph adenomas (SCAs) present clinically as non-functioning adenomas (NFAs) but are immunopositive for adrenocorticotrophic hormone (ACTH) without biochemical and clinical manifestation of hypercortisolism. Pathologic examination of resected NFAs that demonstrate positive ACTH and/or TPIT expression confirms its corticotroph lineage. SCAs comprise up to 20% of NFAs and exhibit a higher rate of recurrence. Studies of molecular mechanisms have generated multiple hypotheses on SCA tumorigenesis, pathophysiology, and growth that as yet remain to be proven...
January 17, 2018: Pituitary
Harumi Sawada, Akihiro Mori, Peter Lee, Shiho Sugihara, Hitomi Oda, Toshinori Sako
The purpose of this study was to determine the therapeutic and/or adverse effects of radiation therapy (RT) against pituitary tumors in dogs with pituitary-dependent hypercortisolism, as monitored by frequent post-RT detailed MRI examinations, clinical signs, and changes in hormone concentrations. Nine dogs with an adrenocorticotropic hormone (ACTH)-secreting pituitary mass diagnosed by magnetic resonance imaging (MRI) underwent RT for 4weeks (total of 48Gy in 4-Gy fractions). Pituitary height/brain area (P/B) value, clinical signs, basal plasma ACTH concentrations, serum cortisol concentrations (pre- and post-ACTH stimulation test) and adverse effects of RT were evaluated before and post-RT...
January 10, 2018: Research in Veterinary Science
Franziska Ritschel, Sabine Clas, Daniel Geisler, Verena Haas, Maria Seidel, Julius Steding, Veit Roessner, Clemens Kirschbaum, Stefan Ehrlich
Anorexia nervosa (AN) is a severe mental disorder accompanied by extensive metabolic and endocrine abnormalities. It has been associated with hypercortisolism using short-term measurement methods such as 24 h-urine, saliva, and blood. The aim of this study was to examine whether the proposed hypercortisolism in acutely underweight AN (acAN) is also reflected in a long-term measure: hair cortisol (HCC). To gain further insight, we compared hair cortisol to a well-established classical cortisol measure (24 h-urine; UCC) longitudinally in acAN...
December 21, 2017: Journal of Psychiatric Research
K Sanders, J A Mol, A Slob, H S Kooistra, S Galac
Hypercortisolism is one of the most commonly diagnosed endocrinopathies in dogs, and new targeted medical treatment options are desirable. Steroidogenic factor-1 (SF-1), an orphan nuclear hormone receptor, is a key regulator of adrenal steroidogenesis, development, and growth. In pituitary-dependent hypercortisolism (PDH), high plasma ACTH concentrations increase the transcriptional activity of SF-1. In adrenal-dependent hypercortisolism, SF-1 expression is significantly greater in dogs with recurrence after adrenalectomy than in those without recurrence...
April 2018: Domestic Animal Endocrinology
Steven B Carr, Bette K Kleinschmidt-DeMasters, Janice M Kerr, Katja Kiseljak-Vassiliades, Margaret E Wierman, Kevin O Lillehei
OBJECTIVE The authors report their single-institution experience with the pathological findings, rates of remission, and complications in patients with presumed Cushing's disease (CD) who underwent a two-thirds pituitary gland resection when no adenoma was identified at the time of transsphenoidal surgery (TSS). The authors also review the literature on patients with CD, negative surgical exploration, and histological findings. METHODS This study is a retrospective analysis of cases found in neurosurgery and pathology department databases between 1989 and 2011...
December 8, 2017: Journal of Neurosurgery
Gloria Aranda, Cristina Lopez, Rebeca Fernandez-Ruiz, Yaiza Esteban, Guillermo Garcia-Eguren, Mireia Mora, Irene Halperin, Gregori Casals, Joaquim Enseñat, Felicia A Hanzu
Glucocorticoids (GC) induce cardiometabolic risk while atherosclerosis is a chronic inflammation involving immunity. GC are immune suppressors, and the adrenocorticotrophic hormone (ACTH) has immune modulator activities. Both may act in atherothrombotic inflammation involving immune cells (IMNC). Aim . To investigate adhesion and activation surface cell markers (CDs) of peripheral IMNC in endogenous Cushing syndrome (CS) and the immune modulator role of ACTH. Material and Methods . 16 ACTH-dependent CS (ACTH-D), 10 ACTH-independent (ACTH-ID) CS, and 16 healthy controls (C) were included...
2017: International Journal of Endocrinology
Graeme Eisenhofer, Jimmy Masjkur, Mirko Peitzsch, Guido Di Dalmazi, Martin Bidlingmaier, Matthias Grüber, Julia Fazel, Andrea Osswald, Felix Beuschlein, Martin Reincke
BACKGROUND: Diagnosis of Cushing syndrome requires a multistep process that includes verification of hypercortisolism followed by identification of the cause of adrenocortical hyperfunction. This study assessed whether pituitary, ectopic, and adrenal subtypes of Cushing syndrome were characterized by distinct plasma steroid profiles that might assist diagnosis. METHODS: In this retrospective cross-sectional study, mass spectrometric measurements of a panel of 15 plasma steroids were applied to 222 patient samples tested for Cushing syndrome...
March 2018: Clinical Chemistry
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