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Adrenal Mass

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141. A novel germline ARMC5 mutation in a patient with bilateral macronodular adrenal hyperplasia: a case report. Full Text available with Trip Pro

. The objective of this study was to report a case in which the patient presented with BMAH induced by a novel heterozygous germline ARMC5 mutation (c. 517C > T, p. Arg173*) alone rather than a two-hit mutation.A 51-year-old woman was identified with masses in the bilateral adrenals. Serum cortisol levels were increased significantly both in the morning (08:00 AM) and late at night (24:00 AM), while plasma adrenocorticotropic hormone was normal. The patient underwent a left adrenalectomy and histopathology (...) A novel germline ARMC5 mutation in a patient with bilateral macronodular adrenal hyperplasia: a case report. Bilateral macronodular adrenal hyperplasia (BMAH) is a rare cause of Cushing's syndrome (CS). BMAH is predominantly believed to be caused by two mutations, a germline and somatic one, respectively, as described in the two-hit hypothesis. In many familial cases of BMAH, mutations in armadillo repeat containing 5 (ARMC5), a putative tumor suppressor gene, are thought to induce the disorder

2018 BMC Medical Genetics

142. First Case of Bacteremia Due to Kytococcus schroeteri in a Child with Congenital Adrenal Hyperplasia. (Abstract)

First Case of Bacteremia Due to Kytococcus schroeteri in a Child with Congenital Adrenal Hyperplasia. Kytococcus schroeteri is a commensal organism of the human skin, which may cause serious infections in immunocompromised patients. We report the first case of K. schroeteri bacteremia in a child with congenital adrenal hyperplasia, identified by matrix-assisted laser desorption ionization-time of flight mass spectrometry (Bruker Daltonics, Germany). İdentification was confirmed by 16S rRNA gene

2018 Pediatric Infectious Dsease Journal

143. Bilateral Adrenalectomy in Congenital Adrenal Hyperplasia: A Systematic Review and Meta-Analysis. Full Text available with Trip Pro

of adrenalectomy, diagnosis, molecular abnormality, pre- and post-operative biochemistry, pre- and post-operative medications, pre- and post-operative body mass index (BMI), indication for adrenalectomy, surgical technique, gross and microscopic adrenal characteristics, follow-up duration, and short-term as well as long-term post-operative outcomes.Forty-eight cases of bilateral adrenalectomy in CAH were identified, with patients aged from four months to 56 years at time of surgery. The most common indication (...) Bilateral Adrenalectomy in Congenital Adrenal Hyperplasia: A Systematic Review and Meta-Analysis. Management of congenital adrenal hyperplasia (CAH) involves suppression of the hypothalamic-pituitary-adrenal (HPA) axis using supraphysiological doses of exogenous glucocorticoids. This can pose a challenge, with Cushing's syndrome being a frequent complication of adequate suppression. Bilateral adrenalectomy, with subsequent replacement of glucocorticoids and mineralocorticoids at physiological

2018 Journal of Clinical Endocrinology and Metabolism

144. Birth size, body composition, and adrenal androgens as determinants of bone mineral density in mid-childhood. Full Text available with Trip Pro

Birth size, body composition, and adrenal androgens as determinants of bone mineral density in mid-childhood. BackgroundBirth weight has an impact on adult bone mass. Higher birth weight is associated with greater bone mineral content (BMC) and children born small for gestational age (SGA) are at an increased risk for impaired accrual of bone mass. Our aim was to study whether the impact of birth size or early childhood growth on bone mass is visible already in mid-childhood.MethodsWe studied (...) 49 children born large for gestational age (LGA), 56 children born appropriate for gestational age (AGA), and 23 children born SGA at 5.0-8.7 years of age. Body composition was assessed by whole-body dual-energy X-ray absorptiometry. Fasting blood samples and anthropometric data were collected.ResultsThe children born SGA had lower bone mineral density (BMD) Z-score (P<0.001) and age- and sex-adjusted BMD (P<0.005) than the LGA and AGA children. Adjusted BMC, muscle mass, and body fat percentage

2018 Pediatric Research

145. Mice overexpressing chromogranin A display hypergranulogenic adrenal glands with attenuated ATP levels contributing to the hypertensive phenotype. Full Text available with Trip Pro

Mice overexpressing chromogranin A display hypergranulogenic adrenal glands with attenuated ATP levels contributing to the hypertensive phenotype. Elevated circulating chromogranin A (CHGA) is observed in human hypertension. CHGA is critical for granulogenesis and exocytosis of catecholamine stores from secretory large dense core vesicles (LDCV). This study aims to understand the morphological, molecular and phenotypic changes because of excess CHGA and the mechanistic link eventuating in hyper (...) -adrenergic hypertension.Blood pressure and heart rate was monitored in mouse models expressing normal and elevated level of CHGA by telemetry. Catecholamine and oxidative stress radicals were measured. Adrenal ultrastructure, LDCV content and mitochondrial abundance were compared and respiration analyzed by Seahorse assay. Effect of CHGA dosage on adrenal ATP content, electron transport chain components and uncoupling protein 2 (UCP-2) were compared in vivo and in vitro.Mice with excess-CHGA displayed

2018 Journal of Hypertension

146. Adrenal relapse of primary central nervous system diffuse large B-cell lymphoma: A case report. Full Text available with Trip Pro

was diagnosed with endometrial cancer. Interestingly, a radiological study following the treatment of endometrial cancer demonstrated a right adrenal mass, which was suspicious for malignancy. Morphologic examination and immunohistochemistry studies confirmed the diagnosis of diffuse large B-cell lymphoma. A fluorescent in situ hybridization panel for lymphoma showed rearrangement of Immunoglobulin heavy chain (IGH) and B-cell lymphoma 6 (BCL6), respectively, suggesting fusion of BCL6/IGH. Immunoglobulin (...) Adrenal relapse of primary central nervous system diffuse large B-cell lymphoma: A case report. Primary central nervous system lymphoma (PCNSL) is a rare form of non-Hodgkin lymphoma with a dismal outcome. Most patients relapse in intracranial sites and <5% of patients relapse in extracranial sites. Here, we present the first case of PCNSL with an adrenal relapse.A 72-year-old woman, first presented 7 years ago with complaints of headache and dizziness.Enhanced magnetic resonance imaging

2018 Medicine

147. Non-α-fetoprotein-producing adrenal hepatoid adenocarcinoma: A case report and literature review. Full Text available with Trip Pro

Non-α-fetoprotein-producing adrenal hepatoid adenocarcinoma: A case report and literature review. Adrenal hepatoid adenocarcinoma typically secretes alpha-fetoprotein (AFP). Here, we report a case of non-AFP-producing adrenal hepatoid adenocarcinoma. Next-generation sequencing (NGS) was conducted to identify gene mutations.A 64-year-old man presented with mild back pain and unexplained weight loss for 3 months.Contrast-enhanced magnetic resonance imaging (MRI) showed a mass (9.9 × 9.7 × 9.1 mm (...) ) above the upper pole of the left kidney. The left renal artery and vein were compressed. The tumor was positive for CK8/18, CK19, CK7, hepatocyte marker (Hepatocyte), and Hep Par 1, but negative for AFP. Plasma AFP was 2.75 ng/mL (normal range: 0-7 ng/mL). NGS revealed mutations of the following genes: ATM, CDKN2A, EGFR, STK11, TP53, BIM, and MLH1. A diagnosis of adrenal hepatoid adenocarcinoma was established.The treatment included 4 cycles of the mFOLFOX6 regimen (oxaliplatin, leucovorin

2018 Medicine

148. MR-guided Gated Stereotactic Radiation Therapy Delivery for Lung, Adrenal, and Pancreatic Tumors: A Geometric Analysis. (Abstract)

, relative to PTV-center of mass (COM), were, respectively, -3.3 mm to 2.8 mm, -2.5 mm to 3.7 mm, and -4.4 mm to 2.9 mm. Corresponding distributions in the craniocaudal direction were -2.6 mm to 4.6 mm, -4.1 mm to 4.4 mm, and -4.4 mm to 4.5 mm, respectively. Mean GTV areas encompassed during beam-on for all fractions were 94.6%, 94.3%, and 95.3% for lung, adrenal, and pancreas tumors, respectively. Mean treatment duty cycle efficiency ranged from 67% to 87% for these tumors. Use of higher threshold-ROI (...) MR-guided Gated Stereotactic Radiation Therapy Delivery for Lung, Adrenal, and Pancreatic Tumors: A Geometric Analysis. We implemented magnetic resonance-guided breath-hold stereotactic body radiation therapy in combination with visual feedback using the MRIdian system. Both accuracy of gated delivery and reproducibility of tumor positions were studied.Tumor tracking is realized through repeated magnetic resonance imaging in a single sagittal plane at 4 frames per second with deformable image

2018 Biology and Physics

149. Transesophageal approach to lung, adrenal biopsy and fiducial placement using endoscopic ultrasonography (EUS): An interventional pulmonology experience. Initial experience of the UCSF-FRETOC (fresno tracheobronchial & oesophageal center) study group. (Abstract)

Transesophageal approach to lung, adrenal biopsy and fiducial placement using endoscopic ultrasonography (EUS): An interventional pulmonology experience. Initial experience of the UCSF-FRETOC (fresno tracheobronchial & oesophageal center) study group. Routine lung cancer surveillance has resulted in early detection of pulmonary nodules and masses. Combined endobronchial ultrasound (EBUS) and trans-esophageal endoscopic ultrasound (EUS) are approved methods for sampling lymph nodes or masses (...) . All patients underwent a post-procedure chest radiograph and were followed outpatient.20 of 39 patients underwent sampling of a suspicious lung mass. An adequate sample was obtained in 19 of 20 patients. In all 19 patients a definitive diagnosis was achieved (95%). In all 13 patients who underwent adrenal sampling, presence or absence of metastasis was conclusively established. 6 patients successfully underwent fiducial placement. In all 39 patients, no major procedure related complications were

2018 Respiratory medicine

150. Feminizing adrenocortical carcinoma - the source of estrogens production and the role of adrenal-gonadal de-differentiation. Full Text available with Trip Pro

manifestations, high levels of estradiol that were normalized after surgery, and histological Weiss score. Gonadal steroidogenic biomarkers including CYP19A1, HSD17B3, and LHCGR were markedly elevated in the feminizing ACC tissues. Adrenal vein sampling and liquid chromatography-tandem mass spectrometry suggested high CYP19A1 activity in the adrenal mass. β-catenin expression was also elevated. When treated with niclosamide and PNU-74654, the H295R cell line showed a decrease in β-catenin expression, cell (...) Feminizing adrenocortical carcinoma - the source of estrogens production and the role of adrenal-gonadal de-differentiation. Feminizing adrenocortical carcinoma (ACC) is rare. The source of estrogen production and the underlying mechanism remain unclear.In the current study, we investigated the source and the molecular mechanism of estrogen production in feminizing ACC.A total of 46 consecutive patients with a diagnosis of ACC were recruited in our center. We described the clinical

2018 Journal of Clinical Endocrinology and Metabolism

151. Unusual Techniques for Preserving Surgical and Oncologic Safety in Hepatectomy of Advanced Adrenal Malignancy with Vena Cava and Liver Invasion. (Abstract)

and repaired the IVC side wall using 4-0 Prolene. Case 2: A 54-year-old woman who complained of abdominal discomfort visited our hospital. Abdominal computed tomography (CT) scan revealed huge adrenal mass with liver and IVC invasion. Thrombosis inside the IVC extended to the right atrium. We decided to carry out veno-veno bypass during operation in collaboration with heart surgeon. After application of veno-veno bypass, the right atrium wall was opened and the tumor thrombus removed. We then carried out (...) Unusual Techniques for Preserving Surgical and Oncologic Safety in Hepatectomy of Advanced Adrenal Malignancy with Vena Cava and Liver Invasion. Status in terms of major vascular structure invasion is a crucial factor for successful major hepatic resection. In particular, surgery for advanced tumors with inferior vena cava (IVC) invasion is difficult and may even be dangerous for the patient, having high risk of massive bleeding and greater chance of embolic complications such as stroke, bowel

2018 Annals of Surgical Oncology

152. Elevated heat shock proteins in bipolar disorder patients with hypothalamic pituitary adrenal axis dysfunction. Full Text available with Trip Pro

. The study also included 94 healthy controls. Among the BD patients, 33 had abnormal hypothalamic-pituitary-adrenal (HPA) axis activity. Blood samples were obtained from the patients and controls. The chemiluminescence method, mass spectrometry, and flow cytometry were used for analysis.The BD patients compared with the controls had a significantly lower level of CPN10 and significantly higher levels of CPN60 and CPN70. The BD patients with abnormal HPA axis activity had a significantly lower level (...) Elevated heat shock proteins in bipolar disorder patients with hypothalamic pituitary adrenal axis dysfunction. Heat shock proteins (HSP) might be useful as biomarkers for bipolar disorder (BD) which would be clinically valuable since no reliable biomarker for BD has so far been identified. The purpose of this study was to assess the heat shock proteins CPN10, CPN60, and CPN70 as potential biomarkers of BD.The study included 100 BD patients recruited from a hospital during 2012 and 2013

2018 Medicine

153. Role of daily plan adaptation in MR-guided stereotactic ablative radiotherapy for adrenal metastases. (Abstract)

Role of daily plan adaptation in MR-guided stereotactic ablative radiotherapy for adrenal metastases. To study interfractional organ changes during magnetic resonance (MR)-guided stereotactic ablative radiation therapy for adrenal metastases and to evaluate the dosimetric advantages of online plan adaptation.Seventeen patients underwent a total of 84 fractions of video-assisted, respiration-gated, MR-guided adaptive radiation therapy to deliver either 50 Gy (5 fractions), 60 Gy (8 fractions (...) changes within a 3 cm distance from the PTV surface, center of mass displacements, and the Dice similarity coefficient. Plan quality evaluation was based on target coverage (GTV and PTV) and high dose sparing of all OARs (V36Gy, V33Gy, and V25Gy).Substantial center of mass displacements were observed for stomach, bowel, and duodenum, 17, 27 and 36 mm, respectively. Maximum volume changes for the stomach, bowel, and duodenum within 3 cm of PTV were 23.8, 20.5, and 20.9 cm3, respectively. Dice

2018 Biology and Physics

154. An unusual case of ectopic corticotrophin-releasing hormone syndrome caused by an adrenal noncatecholamine-secreting pheochromocytoma: a case report. Full Text available with Trip Pro

clinical features of Cushing's syndrome. Endocrinological investigation confirmed severe hypercortisolism along with elevated plasma adrenocorticotropin hormone (ACTH). However, magnetic resonance imaging (MRI) revealed no pituitary adenoma. Abdominal contrast-enhanced computed tomography (CT) revealed a 6.5 cm heterogeneous right adrenal mass with mildly contrast enhancement. The tumor was found during a routine physical check-up at a local hospital 16 months ago; however, the patient did not have any (...) An unusual case of ectopic corticotrophin-releasing hormone syndrome caused by an adrenal noncatecholamine-secreting pheochromocytoma: a case report. Pheochromocytoma, especially for noncatecholamine-secreting pheochromocytoma, is an extremely rare cause of ectopic corticotrophin-releasing hormone (CRH) syndrome.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

2018 BMC Endocrine Disorders

155. The human fetal adrenal produces cortisol but no detectable aldosterone throughout the second trimester. Full Text available with Trip Pro

analysis of adrenal steroid levels and steroidogenic enzyme expression in normal second trimester human fetuses.This was an observational study of steroids, messenger RNA transcripts and proteins in adrenals from up to 109 second trimester fetuses (11 weeks to 21 weeks) at the Universities of Aberdeen and Glasgow. The study design was balanced to show effects of maternal smoking.Concentrations of 19 intra-adrenal steroids were quantified using liquid chromatography and mass spectrometry. Pregnenolone (...) The human fetal adrenal produces cortisol but no detectable aldosterone throughout the second trimester. Human fetal adrenal glands are highly active and, with the placenta, regulate circulating progesterone, estrogen and corticosteroids in the fetus. At birth the adrenals are essential for neonate salt retention through secretion of aldosterone, while adequate glucocorticoids are required to prevent adrenal insufficiency. The objective of this study was to carry out the first comprehensive

2018 BMC Medicine

156. Adrenal myelolipoma with hyperandrogenemia and schizophrenia Full Text available with Trip Pro

Adrenal myelolipoma with hyperandrogenemia and schizophrenia Adrenal myelolipoma with hyperandrogenemia is extremely rare. We report a case of a 26-year-old Chinese female with schizophrenia, who presented with a hormonally active tumor causing hyperandrogenemia. The mass was found by computerized tomography when she had her gynecologic examination for secondary amenorrhea, and it was confirmed to be an adrenal myelolipoma after a histopathological study. She was referred for a left adrenal (...) laparoscopic excision, and the size of adrenal myelolipoma was found to be more than 10 cm. We report this case because large adrenal myelolipomas with hyperandrogenemia and schizophrenia are rare, and adrenal myelolipoma associated with hyperandrogenemia might be determined by the enzymes involved in the production of hormones.

2018 Cancer management and research

157. Surgical Management of a Giant Adrenal Pseudocyst: A Case Report and Review of the Literature in the Last Decade Full Text available with Trip Pro

material. The size of a mass was 11. 7 × 9.3 × 6.6 cm in diameter close to the pancreas, but the origin was from the left adrenal gland. The mass was excised with surgical laparotomy. Giant adrenal pseudocysts are rare entities. Final diagnosis usually confirmed with the pathology examination. Management of such adrenal lesions depends on the unique characteristics, the surgeon's experience, and local resources. (...) Surgical Management of a Giant Adrenal Pseudocyst: A Case Report and Review of the Literature in the Last Decade Adrenal pseudocysts are rare entities and occurred in the 5th and the 6th decades of life. They are discovered accidentally, while appearing with nonspecific clinical and imaging findings. We report a case of a 28-year-old woman presented in our Emergency Department complaining about upper abdomen pain. Computed tomography revealed a hypodense cystic lesion containing hyperdense

2018 Case reports in surgery

158. An Intrarenal Adrenocortical Carcinoma Arising in an Adrenal Rest Full Text available with Trip Pro

An Intrarenal Adrenocortical Carcinoma Arising in an Adrenal Rest We describe a case of a 61-year-old Korean man who was diagnosed with renal cell carcinoma that was discovered on abdominopelvic computed tomography obtained after the patient complained of back pain. A radical nephrectomy was performed, and the surgical specimen showed a relatively well-circumscribed and yellowish lobulated hard mass. Microscopically, the tumor showed sheets and nests of hypercellular pleomorphic cells (...) with thick fibrous septation, frequent mitoses, and areas of adrenal cortical-like tissue. Immunohistochemical staining revealed that the tumor cells were positive for inhibin-α, vimentin, synaptophysin, and melan A. It also revealed that the tumor cells were negative for pan-cytokeratin, epithelial membrane antigen, paired box 8, α-methylacyl-coenzyme A racemase, CD10, cytokeratin 7, carbonic anhydrase 9, c-Kit, renal cell carcinoma, transcription factor E3, human melanoma black 45, desmin, smooth

2018 Journal of Pathology and Translational Medicine

159. Giant nonfunctioning adrenal tumors: two case reports and review of the literature Full Text available with Trip Pro

Giant nonfunctioning adrenal tumors: two case reports and review of the literature There are an estimated 1-2 cases per million per year of adrenocortical carcinoma in the USA. It represents a rare and aggressive malignancy; it is the second most aggressive endocrine malignant disease after anaplastic thyroid carcinoma. Non-secretory adrenal masses are diagnosed late due to a mass effect or metastatic disease or found incidentally (adrenal incidentalomas).The first case report describes a 39 (...) and demands high clinical suspicion. The work-up for adrenal masses must include determination of whether the mass is functioning or nonfunctioning and whether it is benign or malignant.

2018 Journal of medical case reports

160. Assessment of Subclinical Cardiac Alterations and Atrial Electromechanical Delay by Tissue Doppler Echocardiography in Patients with Nonfunctioning Adrenal Incidentaloma Full Text available with Trip Pro

Assessment of Subclinical Cardiac Alterations and Atrial Electromechanical Delay by Tissue Doppler Echocardiography in Patients with Nonfunctioning Adrenal Incidentaloma Majority of the incidentally discovered adrenal masses, called adrenal incidentaloma (AI), are nonfunctioning adrenal adenomas. The appropriate management of AI is still a matter debate, so it is necessary to investigate their associated morbidity. However, data regarding morphological and functional cardiac alterations (...) echocardiography. Data were analyzed with Statistical Package for the Social Sciences (SPSS, Chicago, IL, United States) statistics, version 17.0 for Windows. P < 0.05 was considered statistically significant.Left ventricular (LV) mass index and LV myocardial performance index were significantly increased in AI group. Among atrial conduction times, both intra- and interatrial electromechanical delays were significantly prolonged in patients with nonfunctioning AI. Other laboratory and echocardiographic

2018 Arquivos brasileiros de cardiologia

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