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Nonparathyroid Hypercalcemia.

Wed, 01/16/2019 - 11:16
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Nonparathyroid Hypercalcemia.

Front Horm Res. 2019;51:77-90

Authors: Goltzman D

Abstract
Primary hyperparathyroidism is among the most common causes of hypercalcemia. However, ingestion of medication, including hydrochlorathiazide, lithium, and foscarnet, excessive vitamin A ingestion, endocrinopathies such as hyperthyroidism, adrenal insufficiency, and acromegaly, abnormal nutrient intake such as parenteral nutrition in preterm infants and milk-alkali syndrome, and prolonged immobilization have all been associated with hypercalcemia. The most common cause of nonparathyroid hypercalcemia is neoplasia. Hypercalcemia is generally due to the secretion of parathyroid hormone (PTH)-related peptide (PTHrP) by a wide variety of nonmetastatic solid tumors, including squamous cell tumors but also hematologic tumors. PTHrP, although encoded by a distinct gene, shares amino acid sequence homology with PTH in the amino-terminal domain, which allows it to cross-react at a common G protein receptor, the type 1 PTH/PTHrP receptor (PTHR1), resulting in similar skeletal effects and effects on calcium and phosphorus metabolism. Increased PTHrP action with hypercalcemia may be seen in the benign disease Jansen's metaphyseal chondrodysplasia due to a gain-of-function mutation in PTHR1. Another humoral factor, 1,25-dihyroxyvitamin D [1,25(OH)2D] may be produced by lymphomas, but also by benign granulomatous disorders and may also cause hypercalcemia when its metabolism is genetically impaired. Vitamin D intoxication may cause hypercalcemia due to overproduction of the metabolite, 25 hydroxyvitamin D, apparently in the absence of conversion to 1,25(OH)2D. Malignancies metastatic to bone or arising in bone (such as multiple myeloma) may produce a variety of growth factors and cytokines, in addition to PTHrP, which can contribute to tumor growth as well as osteolysis and hypercalcemia.

PMID: 30641526 [PubMed - in process]

Familial and Hereditary Forms of Primary Hyperparathyroidism.

Wed, 01/16/2019 - 11:16
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Familial and Hereditary Forms of Primary Hyperparathyroidism.

Front Horm Res. 2019;51:40-51

Authors: Cetani F, Saponaro F, Borsari S, Marcocci C

Abstract
Individuals with a familial predisposition to the development of parathyroid tumors constitute a small minority of all patients with primary hyperparathyroidism (PHPT). These familial syndromes exhibit Mendelian inheritance patterns and the main causative genes in most families have been identified. They include multiple endocrine neoplasia (MEN; types 1, 2A, and 4), hyperparathyroidism-jaw tumor (HPT-JT) syndrome, familial isolated hyperparathyroidism, familial hypocalciuric hypercalcemia (FHH), and neonatal severe PHPT. Each MEN type is associated with the various combinations of specific tumors. MEN1 is characterized by the occurrence of parathyroid, enteropancreatic, and pituitary tumors; MEN2A is characterized by medullary thyroid carcinoma and pheochromocytoma, and MEN4 is characterized by a pathological spectrum similar to that of MEN1 in association with tumors of the adrenal, kidney, and reproductive organs. HPT-JT is characterized by PHPT, ossifying fibromas of maxillary bones, kidney disease, and uterine neoplasias. The prompt diagnosis of these diseases is of great importance for planning appropriate surveillance of the mutant carriers and correct surgical management. The search for mutation is also useful for the identification of the family members who do not carry the mutation and can avoid unnecessary biochemical and instrumental evaluations. Surgery remains the treatment of choice in all familial forms except FHH.

PMID: 30641519 [PubMed - in process]

Pheochromocytoma-related cardiomyopathy presenting as broken heart syndrome: Case report and literature review.

Wed, 01/16/2019 - 11:16
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Pheochromocytoma-related cardiomyopathy presenting as broken heart syndrome: Case report and literature review.

Int J Surg Case Rep. 2019 Jan 09;55:7-10

Authors: Diaz B, Elkbuli A, Ehrhardt JD, McKenney M, Boneva D, Hai S

Abstract
INTRODUCTION: Pheochromocytoma are neuroendocrine tumors that arise from sympathetic chromaffin cells within the adrenal medulla. They principally secrete catecholamines, potentially causing life-threatening cardiovascular complications. A myriad of symptomatology and clinical findings are associated with pheochromocytoma, including a catecholamine-induced dilated cardiomyopathy.
PRESENTATION OF CASE: A 50-year-old woman presented with retrosternal chest pain and underwent diagnostic evaluation for acute coronary syndrome. Cardiac catheterization demonstrated patent coronary arteries and a pattern of ventricular hypokinesis consistent with takotsubo cardiomyopathy, also known as broken heart syndrome. Further imaging with abdominal CT revealed an adrenal mass. Laboratory markers supported the clinical picture of pheochromocytoma. Right adrenalectomy was performed and our patient was symptom-free at discharge on post-operative day three.
DISCUSSION: Alpha and beta adrenergic blockade are used in a critical care setting to prevent perioperative hemodynamic instability as well as catecholamine-induced heart failure in the setting of pheochromocytoma. Patients commonly require vasopressors in the postoperative period due to the rapid reduction in circulating catecholamines following resection. Discharge planning should include recommendations for genetic counseling to screen for syndromic causes of pheochromocytoma that increase the risk for other neoplasms.
CONCLUSION: We present a case report of a rare adrenal tumor in a middle-aged woman that manifested as acute coronary syndrome. A presumptive diagnosis of takotsubo cardiomyopathy on cardiac catheterization led to further investigation. Abdominal imaging located an adrenal mass that correlated with laboratory studies positive for high levels of catecholamines and their metabolites. The tumor was excised and the patient recovered without complications.

PMID: 30641325 [PubMed - as supplied by publisher]

A Case of Stage IV Chromophobe Renal Cell Carcinoma Treated with the Oncolytic ECHO-7 Virus, Rigvir®.

Wed, 01/16/2019 - 11:16
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A Case of Stage IV Chromophobe Renal Cell Carcinoma Treated with the Oncolytic ECHO-7 Virus, Rigvir®.

Am J Case Rep. 2019 Jan 12;20:48-52

Authors: Ismailov Z, Rasa A, Bandere K, Brokāne L, Tilgase A, Olmane E, Nazarovs J, Alberts P

Abstract
BACKGROUND Renal cell carcinoma is the most commonly diagnosed primary malignant tumor of the kidney in adults, and includes the variant of chromophobe renal cell carcinoma. Despite new targeted therapies that improve progression-free survival (PFS) and overall survival (OS) for early-stage renal cell carcinoma, the 5-year survival for patients with stage IV renal cell carcinoma remains below 10%, and the 50% OS is less than one year. Metastatic renal cell carcinoma can be resistant to cytotoxic chemotherapy. This report is of a case of stage IV chromophobe renal cell carcinoma that responded well to treatment with the oncolytic ECHO-7 virus, Rigvir®. CASE REPORT In December 2015, a 59-year-old man presented with a right-sided chromophobe renal cell carcinoma stage IV (pT₁N₀M₁) with adrenal gland metastasis. He underwent right nephro-adrenalectomy followed by treatments with Rigvir® (≥10⁶ TCID₅₀/ml) by intramuscular (i.m.) injection on three consecutive days. Treatment with Rigvir® continued once per week for three months, and from March 2016, once per month, with continued treatment until computed tomography (CT) scans confirmed that the tumor metastases had stabilized. CONCLUSIONS This case report has demonstrated that the oncolytic ECHO-7 virus, Rigvir® should be evaluated further as a potential treatment for advanced renal carcinoma.

PMID: 30635548 [PubMed - in process]

Suprasellar chordoid glioma: a report of two cases.

Wed, 01/16/2019 - 11:16
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Suprasellar chordoid glioma: a report of two cases.

Arch Endocrinol Metab. 2018;62(6):648-654

Authors: Danilowicz K, Abbati SG, Sosa S, Witis FL, Sevlever G

Abstract
Chordoid glioma (CG) is considered a slow growing glial neoplasm. We report two new cases with endocrinological presentation, management and outcome. Case reports: 1) An 18 year-old female patient was admitted due to headaches, nausea and vomiting and visual abnormalities. She was in amenorrhea. A brain magnetic resonance imaging (MRI) demonstrated a 35 mm-diameter sellar and suprasellar mass. An emergency ventricular peritoneal valve was placed due to obstructive hydrocephalus. Transcraneal surgery was performed. The patient developed central hypothyroidism, adrenal insufficiency and transient diabetes insipidus; she never recovered spontaneous menstrual cycles. Histopathologic study showed cells in cords, inside a mucinous stroma, positive for glial fibrillary acidic protein (GFAP). Due to residual tumor gamma knife radiosurgery was performed. Three years after surgery, the patient is lucid, with hypopituitarism under replacement. 2) A 46 year-old woman complained about a three year-history of amenorrhea, galactorrhea and headache. An MRI showed a solid-cystic sellar mass 40 mm-diameter that extended to the suprasellar cistern. She had hypogonatropic hypogonadism and mild hyperprolactinemia. The tumor mass was removed via nasal endoscopic approach. Histopathological study reported cellular proliferation of glial lineage positive for GFAP. The patient evolved with central hypothyroidism and diabetes insipidus. She was re-operated for fistula and again under the diagnosis of extradural abscess. She evolved with cardiorespiratory descompensation and death, suspected to be due to a thromboembolism. In conclusion, the first case confirms that best treatment for CG is surgery considering radiotherapy as an adjuvant therapy. The other case, on the contrary, illustrates the potentially fatal evolution due to surgical complications.

PMID: 30624507 [PubMed - in process]

Postmenopausal androgen-secreting ovarian tumors: challenging differential diagnosis in two cases.

Wed, 01/16/2019 - 11:16
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Postmenopausal androgen-secreting ovarian tumors: challenging differential diagnosis in two cases.

Climacteric. 2019 Jan 09;:1-5

Authors: Arteaga E, Martinez A, Jaramilo J, Villaseca P, Cuello M, Valenzuela P, Gejman R, Blumel JE

Abstract
Postmenopausal hyperandrogenism constitutes a very rare condition of tumoral or non-tumoral origin primarily residing either in the ovary or in the adrenal glands. We present herein two cases with this condition; one with abnormal postmenopausal genital bleeding and mild increase in facial hair, and the second with slow-developing hirsutism and virilization. Both cases shared a notorious increase in libido. The laboratory tests showed high levels of testosterone (>100 ng/ml). A normal value of dehydroepiandrosterone sulfate and a normal cortisol level at 9 am after 1 mg of dexamethasone administered at midnight (Nugent test) made an adrenal etiology very unlikely. On the other hand, a high level of inhibine B oriented to an ovarian source. Transvaginal sonography failed to demonstrate an ovarian tumor, but an abdominal and pelvic computed tomography scan or magnetic resonance imaging detected an ovarian tumor and normal adrenal glands. A laparoscopic oophorectomy was performed, and the histological study demonstrated a steroidal cell tumor in the first case and a Leydig cell tumor in the second.

PMID: 30623686 [PubMed - as supplied by publisher]

MicroRNA351 targeting TRAF6 alleviates dexamethasone-induced myotube atrophy.

Wed, 01/16/2019 - 11:16
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MicroRNA351 targeting TRAF6 alleviates dexamethasone-induced myotube atrophy.

J Thorac Dis. 2018 Nov;10(11):6238-6246

Authors: Qiu J, Wang L, Wang Y, Zhang Q, Ma W, Fang Q, Sun H, Ding F

Abstract
Background: Glucocorticoids, including dexamethasone (Dex), are corticosteroids secreted by the adrenal gland, which are used as potent anti-inflammatory, anti-shock, and immunosuppressive agents. Dex is commonly used in patients with malignant tumors, such lung cancer. However, administration of high-dose Dex induces severe atrophy of the skeletal muscle, and the underlying mechanisms of this skeletal muscle atrophy remain unclear. Abundant miRNAs of skeletal muscle, such as miR-351, play an important role in the regulation of extenuating the process of muscle atrophy.
Methods: The mRNA and protein expression of TRAF6, MuRF1, MAFbx was determined by real-time PCR and western blot, while the expression of miR-351 was detected by real-time PCR. The myotubes were transfected with miR-351 mimic, negative control, or miR-351 inhibitor. The C2C12 myotubes diameter was measured.
Results: MicroRNA351 (miR-351) level was markedly reduced and the mRNA and protein levels of tumor necrosis factor (TNF) receptor-associated factor 6 (TRAF6) were increased in Dex-induced C2C12 myotube atrophy. miR-351 directly interacted with the 3'-untranslated region (3'UTR) of TRAF6. Interestingly, miR-351 administration notably inhibited the reduction of the C2C12 myotube diameter induced by Dex treatment and reduced the levels of TRAF6, muscle-RING-finger protein-1 (MuRF1), and muscle atrophy F-box (MAFbx).
Conclusions: miR-351 counteracts Dex-induced C2C12 myotube atrophy by repressing the TRAF6 expression as well as E3 ubiquitin ligase MuRF1 and MAFbx. miR-351 maybe a potential target for development of a new strategy for skeletal muscle atrophy.

PMID: 30622796 [PubMed]

Laparoscopic Excision of an Adrenal Ganglioneuroma Presented as an Incindentaloma of the Retro Peritoneum.

Wed, 01/16/2019 - 11:16
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Laparoscopic Excision of an Adrenal Ganglioneuroma Presented as an Incindentaloma of the Retro Peritoneum.

Curr Health Sci J. 2018 Jan-Mar;44(1):71-75

Authors: Sapalidis K, Mandalovas S, Kesisoglou I

Abstract
Adrenal ganglioneuroma (GN) is a benign tumor of retro-peritoneum that is presented as an incidentaloma. GNs most commonly have been diagnosed by imaging examinations notably by CT scan. Differential diagnosis from other malignant adrenal tumors is often difficult. The only examination that confirms diagnosis is only the histopathological one. Surgical excision of a GN has been proposed as the best solution for both diagnostic and therapeutic purposes. This case report is about a 68-year old male patient presenting with a non-typical abdominal pain. The imaging investigation showed an incidentaloma over 4cm at its large axis. The patient underwent laparoscopic excision of the tumor. We present the steps followed during the diagnosis the appropriate treatment and the follow up examinations as a result of this rare finding.

PMID: 30622759 [PubMed]

Efficient screening of patients with aldosterone-producing adenoma using the ACTH stimulation test.

Wed, 01/16/2019 - 11:16
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Efficient screening of patients with aldosterone-producing adenoma using the ACTH stimulation test.

Hypertens Res. 2019 Jan 08;:

Authors: Kita T, Furukoji E, Sakae T, Kitamura K

Abstract
Adrenal venous sampling (AVS) is the gold standard test for distinguishing between unilateral and bilateral primary aldosteronism (PA); however, AVS requires advanced and time consuming technique. The needs for AVS have been increasing due to the increased utilization of screening for PA. An efficient selection of unilateral PA, such as aldosterone-producing adenoma (APA), before AVS is useful to avoid undesirable AVS in bilateral PA, such as idiopathic hyperaldosteronism. In this study, 40 patients who received all three confirmatory tests, including the captopril challenge test, furosemide upright test and adrenocorticotropin (ACTH) stimulation test (AST), and who were diagnosed as having PA by AVS were recruited. Subjects were diagnosed as having unilateral aldosterone excess (n = 22) or bilateral aldosterone excess (n = 18) by AVS. All patients with unilateral PA underwent an operation and were finally diagnosed with APA. Major differences were detected in serum potassium level, basal plasma aldosterone concentration (PAC), presence of adrenal tumor, and AST results between the two groups. The PAC/cortisol ratio at 120 min in the AST showed the highest diagnostic capability for distinguishing the subtypes of PA according to a receiver operating characteristic (ROC) curve analysis (area under the ROC curve was 0.956). At a cutoff value of 1.20 for the PAC/cortisol ratio at 120 min on the AST, the sensitivity was 95.5%, and the specificity was 88.9%. This sufficiently high sensitivity suggests that the PAC/cortisol ratio at 120 min in the AST could be useful for the screening of patients with PA who are suitable for AVS.

PMID: 30622316 [PubMed - as supplied by publisher]

A case report of late local relapse of adrenocortical carcinoma 18 years after adrenalectomy.

Wed, 01/16/2019 - 11:16
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A case report of late local relapse of adrenocortical carcinoma 18 years after adrenalectomy.

Exp Oncol. 2018 Oct;40(3):251-253

Authors: Tkachenko R, Golovko A, Kuryk O

Abstract
Adrenocortical cancer is an extremely rare tumor presenting with extensive locoregional spread at the time of diagnosis. Due to the diagnostic difficulties preoperatively and a lack of effective treatment options, patients have poor prognosis. Patients succumb to metastases within a couple of months. Only 20 cases have been so far reported in the literature with a medium disease-free survival up to 2 years. We present a case of a locoregional recurrence of adrenocortical cancer 18 years after left adrenalectomy.

PMID: 30285002 [PubMed - indexed for MEDLINE]

Biological diagnosis of pheochromocytoma: is there an interest in urinary metanephrines measurement over 3 consecutive days?

Wed, 01/16/2019 - 11:16
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Biological diagnosis of pheochromocytoma: is there an interest in urinary metanephrines measurement over 3 consecutive days?

Ann Biol Clin (Paris). 2018 Oct 01;76(5):537-544

Authors: Bettacchioli É, Leven C, Thuillier P, Plée-Gautier E, Lapègue M, Carré JL, Padelli M

Abstract
Pheochromocytoma and paraganglioma are neuroendocrine tumors characterized by a catecholamine production potential. The biochemical diagnosis for this type of tumor is carried out through the metanephrine titration on 24-hours urines. Some authors have suggested that the sensitivity of the test could be improved by sampling and analyzing urines 3 days in a row (cycle) versus a unique measurement but this method has never been fully evaluated. The goal of this study was to establish a comparison of diagnosis performances between urinary metanephrines measurement for 3 consecutive days, and metanephrines measurement on a unique 24-hour sample. Patients of Brest Regional University Hospital whose 3-consecutive day 24-hour urine samples had been analyzed from January 2011 to May 2017 were included in this study. The primary endpoint was the comparison of diagnostic performances of urinary metanephrine titration over a single day versus 3 consecutive days. Eighty-two patients for a total of 103 cycles among which 7 revealed a pheochromocytoma were analyzed. ROC curve analysis shows that the metanephrine cycle titration method is more efficient than the metanephrine single titration method (metanephrine: AUC=0.881 against 0.826 respectively; normetanephrine: AUC=0.946 against 0.901 respectively). Urinary titration over 3 days allows the diagnosis of 100% (7/7) of pheochromocytomas against 85.7% (6/7) for the the single urinary titration. In conclusion, metanephrine titration over 3 consecutive days of 24-hours urine samples shows a better sensitivity and better diagnosis performances for detecting pheochromocytoma and paraganglioma than the single titration method.

PMID: 30174318 [PubMed - indexed for MEDLINE]

Primary fibroblast co-culture stimulates growth and metabolism in Sdhb-impaired mouse pheochromocytoma MTT cells.

Wed, 01/16/2019 - 11:16
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Primary fibroblast co-culture stimulates growth and metabolism in Sdhb-impaired mouse pheochromocytoma MTT cells.

Cell Tissue Res. 2018 Dec;374(3):473-485

Authors: Richter S, D'Antongiovanni V, Martinelli S, Bechmann N, Riverso M, Poitz DM, Pacak K, Eisenhofer G, Mannelli M, Rapizzi E

Abstract
Pheochromocytomas and paragangliomas (PGLs) due to mutations of succinate dehydrogenase (SDH) B, a subunit of the SDH complex with a role in the Krebs cycle and the respiratory chain, tend to be larger at diagnosis and more prone to metastatic disease than other tumors. This presentation contrasts with the behavior of some cell line models of SDHB impairment, which show reduced growth compared to wild type. We hypothesize that reduced growth of SDHB-impaired monolayer culture models might reflect lack of support from sources within the tumor microenvironment. The present study therefore investigates how the microenvironment, modeled here by fibroblast co-culture, modulates cell metabolism, growth and invasion in an Sdhb-impaired mouse pheochromocytoma cell line. We employed two different constructs of short hairpin RNA to knockdown Sdhb and compared growth in a monolayer with and without fibroblast co-culture. Sdhb-silenced cells showed functional impairment of SDH with elevated succinate to fumarate ratio and decreased oxidative capacity. Cell growth was delayed with an increase in doubling time of 2 h or 20 h. Clonogenic cell survival and viability, on the other hand, were either unchanged or increased compared to control. In standard monolayer culture, no differences in pro-metastatic features were present. Co-culture with primary mouse fibroblast reversed the difference of proliferation between control and Sdhb knockdown but was unable to significantly influence invasiveness under these culture conditions. Metabolic studies identified that lactate secreted by fibroblasts was taken up preferentially by Sdhb-silenced cells. In summary, the present study identified a potential role for the tumor microenvironment in influencing phenotypic features of SDHB-mutated PGLs, providing a basis for the use of therapies targeted towards the tumor microenvironment.

PMID: 30159755 [PubMed - indexed for MEDLINE]

18F-FDOPA PET/CT Uptake Parameters Correlate with Catecholamine Secretion in Human Pheochromocytomas.

Wed, 01/16/2019 - 11:16
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18F-FDOPA PET/CT Uptake Parameters Correlate with Catecholamine Secretion in Human Pheochromocytomas.

Neuroendocrinology. 2018;107(3):228-236

Authors: Moog S, Houy S, Chevalier E, Ory S, Weryha G, Rame M, Klein M, Brunaud L, Gasman S, Cuny T

Abstract
BACKGROUND: 18F-FDOPA positron emission tomography/computed tomography (PET/CT) is a sensitive nuclear imaging technology for the diagnosis of pheochromocytomas (PHEO). However, its utility in determining predictive factors for the secretion of catecholamines remains poorly studied.
METHODS: Thirty-nine histologically confirmed PHEO were included in this retrospective single-center study. Patients underwent 18F-FDOPA PET/CT before surgery, with an evaluation of several uptake parameters (standardized uptake values [SUVmax and SUVmean] and the metabolic burden [MB] calculated as follows: MB = SUVmean × tumor volume) and measurement of plasma and/or urinary metanephrine (MN), normetanephrine (NM), and chromogranin A. Thirty-five patients were screened for germline mutations in the RET, SDHx, and VHL genes. Once resected, primary cultures of 5 PHEO were used for real-time measurement of catecholamine release by carbon fiber amperometry.
RESULTS: The MB of the PHEO positively correlated with 24-h urinary excretion of NM (r = 0.64, p < 0.0001), MN (r = 0.49, p = 0.002), combined MN and NM (r = 0.75, p < 0.0001), and eventually plasma free levels of NM (r = 0.55, p = 0.006). In the mutated patients (3 SDHD, 2 SDHB, 3 NF1, 1 VHL, and 3 RET), a similar correlation was observed between MB and 24-h urinary combined MN and NM (r = 0.86, p = 0.0012). For the first time, we demonstrate a positive correlation between the PHEO-to-liver SUVmax ratio and the mean number of secretory granule fusion events of the corresponding PHEO cells revealed by amperometric spikes (p = 0.01).
CONCLUSION: While the 18F-FDOPA PET/CT MB of PHEO strongly correlates with the concentration of MN, amperometric recordings suggest that 18F-FDOPA uptake could be enhanced by overactivity of catecholamine exocytosis.

PMID: 29949805 [PubMed - indexed for MEDLINE]

Occurrence of Endocrine and Thyroid Cancers Among Alaska Native People, 1969-2013.

Wed, 01/16/2019 - 11:16
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Occurrence of Endocrine and Thyroid Cancers Among Alaska Native People, 1969-2013.

Thyroid. 2018 04;28(4):481-487

Authors: Nash SH, Lanier AP, Southworth MB

Abstract
BACKGROUND: Nationwide, the incidence of thyroid cancer is lower among American Indian/Alaska Native (AI/AN) people than among U.S. whites (USW). However, little is known about the incidence of thyroid or other endocrine cancers specifically among Alaska Native (AN) people.
METHODS: Data were examined from the National Cancer Institute's Surveillance, Epidemiology, and End Results Alaska Native Tumor Registry on endocrine cancers diagnosed among AN people from 1969-2013, with a specific focus on thyroid cancers. Frequencies of endocrine cancers by site and also of thyroid cancers by histology, size, and stage at diagnosis were evaluated. Distributions were compared to USW (Surveillance, Epidemiology, and End Results 9 Registries) using the chi-square test. Five-year average annual age-adjusted incidence rates of thyroid cancers were calculated, stratified by histology, age, and five-year period of diagnosis, and compared to those observed among USW. Five-year cause-specific survival was evaluated using cause of death data from the National Death Index Plus from the National Center for Health Statistics.
RESULTS: During the 45-year period (1969-2013), 224 endocrine cancers were diagnosed among AN people, of which 210 (94%) were thyroid cancers. Compared to USW, AN people were diagnosed at a slightly younger age, had a higher proportion of thyroid cancers diagnosed with a size of 20-40 mm, and a larger proportion of patients with regional metastases. More than 85% of AN thyroid cancers were of papillary histology. The incidence of thyroid cancers was similar between AN people and USW, and appeared to increase among AN people over the period of surveillance. Finally, five-year cause-specific survival rate was 100% for papillary carcinoma patients and 86.3% [confidence interval 54.7-96.5] for follicular thyroid cancer patients.
CONCLUSIONS: This study is the first report of endocrine cancers and the first detailed examination of thyroid cancer among AN people. The incidence of thyroid cancer was similar among AN people and USW. However, compared to USW, AN people appear to be at risk for diagnosis at a younger age, larger size, and higher stage. Further research is needed to explore the causes of these differences.

PMID: 29439613 [PubMed - indexed for MEDLINE]

Anesthesia Case of the Month.

Wed, 01/16/2019 - 11:16
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Anesthesia Case of the Month.

J Am Vet Med Assoc. 2018 Feb 01;252(3):286-288

Authors: Katzman SA, Perez-Noguez M, Pypendop BH, Alex CE, Affolter VK

PMID: 29346057 [PubMed - indexed for MEDLINE]

Clinical effects of prophylactic use of phentolamine in patients undergoing pheochromocytoma surgery.

Wed, 01/16/2019 - 11:16
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Clinical effects of prophylactic use of phentolamine in patients undergoing pheochromocytoma surgery.

J Clin Anesth. 2018 02;44:119

Authors: Yu M, Han C, Zhou Q, Liu C, Ding Z

PMID: 29195099 [PubMed - indexed for MEDLINE]

The role of ARMC5 in human cell cultures from nodules of primary macronodular adrenocortical hyperplasia (PMAH).

Wed, 01/16/2019 - 11:16
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The role of ARMC5 in human cell cultures from nodules of primary macronodular adrenocortical hyperplasia (PMAH).

Mol Cell Endocrinol. 2018 01 15;460:36-46

Authors: Cavalcante IP, Nishi M, Zerbini MCN, Almeida MQ, Brondani VB, Botelho MLAA, Tanno FY, Srougi V, Chambo JL, Mendonca BB, Bertherat J, Lotfi CFP, Fragoso MCBV

Abstract
The participation of aberrant receptors and intra-adrenal ACTH in hyperplastic tissue are considered mechanisms that regulate hypercortisolism in PMAH. Additionally, germline ARMC5 mutations have been described as the most frequent genetic abnormality found in patients diagnosed with PMAH. Previous functional studies analyzed ARMC5 role using H295R cells. Therefore, we investigated the role of ARMC5 in cell cultures obtained from PMAH nodules containing steroidogenic cells, aberrant receptors and intra-adrenal ACTH. ARMC5 silencing in non-mutated PMAH cell cultures decreased steroidogenesis-related genes and increased CCNE1 mRNA expression and proliferative capacity without affecting cell viability. Additionally, ARMC5 overexpression induced cell death in PMAH mutated cell cultures, thereby decreasing cell viability. We confirmed the role of ARMC5 as an important pro-apoptotic protein involved in PMAH-related steroidogenesis. We also report for the first time the involvement of ARMC5 in controlling proliferation and regulating cell cycle in PMAH cell cultures; these effects need to be explored further.

PMID: 28676429 [PubMed - indexed for MEDLINE]

[Catecholamine Cardiomyopathy Presenting Inverted-takotsubo Pattern Asynergy].

Wed, 01/16/2019 - 11:16
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[Catecholamine Cardiomyopathy Presenting Inverted-takotsubo Pattern Asynergy].

Masui. 2017 Apr;66(4):401-404

Authors: Okuno T, Hino M, Kiyama R, Shindo K

Abstract
Pheochromocytoma is complicated with catecholamine cardiomyopathy, and preoperative care becomes considerably more difficult We report anesthetic management for an 48-year-old man brought to our hospital by ambulance and immediately hospitalized due to pheochromocytoma crisis complicated with catechol- amine cardiomyopathy presenting inverted-takotsubo pattern asynergy. Before surgery, α and β blocking drugs were used to control hypertension and tachycardia, and continuous hemodiafiltration was initiated and the patient was intubated for severe pulmonary edema. Seven days later, he underwent laparoscopic adrenalectomy. Total intravenous anesthesia was achieved with propofol, remifentanil, fentanyl, and rocuronium. During the operation, continuous infusion of landiolol, phentolamine, and nitroglycerin, and addi- tional bolus injections of landiolol were given to control hypertension. After severing the right adrenal vein, noradrenaline (0.15 μg · kg⁻¹ · min⁻¹), and dopamine (4μg · kg⁻¹ · min⁻¹) were started and the patient was placed in the intensive care unit Inverted-takotsubo pattern asynergy is not very common, and treatment consists of supportive care, as in the usual takotsubo. Preoperative CHDF (continuous hemodiafiltration) may be useful when it is difficult to control hypertension or tachycardia with medications.

PMID: 30382641 [PubMed - indexed for MEDLINE]

adrenal tumor; +21 new citations

Wed, 01/09/2019 - 10:46

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Pheochromocytoma in Dogs Undergoing Adrenalectomy.

Wed, 01/02/2019 - 10:09
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Pheochromocytoma in Dogs Undergoing Adrenalectomy.

Vet Pathol. 2018 Dec 30;:300985818819174

Authors: Zini E, Nolli S, Ferri F, Massari F, Gerardi G, Nicoli S, Romanelli G, Montinaro V, Trez D, Cavicchioli L, Ferro S

Abstract
Pheochromocytoma is frequent in dogs and carries a guarded prognosis. Current histological criteria may not predict malignant behavior in dogs, similar to humans. In humans, characterization of tumors has been refined using the pheochromocytoma of the adrenal gland scaled score (PASS) and by immunohistochemistry. The study aim was to investigate PASS and immunohistochemical markers used in humans in 24 dogs with pheochromocytoma that underwent adrenalectomy. Dogs with pheochromocytomas were reviewed and tumors collected. Histological sections were evaluated to apply the PASS and were single-labeled for chromogranin A, Ki-67, COX-2, p53, BCL-2, c-erbB-2, vascular endothelial growth factor, and S100. Survival, age, and vascular and capsular invasion were compared for PASS and immunohistochemical markers; results of PASS were also compared for each marker. Associations between markers were tested. PASS and immunohistochemical markers did not differ for survival, age, and vascular and capsular invasion. Tumors showing BCL-2 expression in >50% cells had lower PASS than those with lower expression (PASS: 7 ± 2 vs 9 ± 2; P = .011). Tumors positive for S100 had higher PASS than those that were negative (PASS: 10 ± 2 vs 7 ± 2; P = .001). Results of the different markers were not associated. In conclusion, in the context of canine pheochromocytoma, PASS and the selected immunohistochemical markers are not associated with survival, age, or vascular or capsular invasion. The higher PASS in S100-positive tumors may indicate that pheochromocytomas developing morphologic changes acquire S100 expression. The significance of lower PASS in tumors with elevated BCL-2 expression is uncertain. Overall, the use of PASS and the present immunohistochemical markers may not be useful in dogs with pheochromocytoma.

PMID: 30595108 [PubMed - as supplied by publisher]

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