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Benign hormone-secreting adenoma within a larger adrenocortical mass showing intensely increased activity on (18)F-FDG PET/CT.

Fri, 07/14/2017 - 00:19
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Benign hormone-secreting adenoma within a larger adrenocortical mass showing intensely increased activity on (18)F-FDG PET/CT.

Endocrine. 2016 Oct;54(1):269-270

Authors: Papadakis GZ, Millo C, Stratakis CA

Abstract
Adrenal adenomas usually show (18)F-FDG activity less than that of the liver parenchyma. However, lipid-poor and hormone-secreting adenomas have been reported to show mild (18)F-FDG avidity. We report on a 51-year-old female with clinical symptoms of hypercortisolemia and a large right adrenal mass detected on CT. Post-contrast CT images showed an enhancing focus in the lower pole of the mass, with corresponding markedly increased activity on (18)F-FDG PET/CT. Right adrenalectomy was performed and histology revealed a benign adenoma, indicating that functioning benign adenomas can show intensely increased metabolic activity on (18)F-FDG mimicking malignancy.

PMID: 27154873 [PubMed - indexed for MEDLINE]

Visual Vignette.

Fri, 07/14/2017 - 00:19
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Visual Vignette.

Endocr Pract. 2017 Apr 02;23(4):510

Authors: Paschou SA, Tzioras K, Nika C, Papatheodorou A, Samaras V, Vryonidou A

PMID: 27631846 [PubMed - indexed for MEDLINE]

Melatonin regulates catecholamine biosynthesis by modulating bone morphogenetic protein and glucocorticoid actions.

Fri, 07/14/2017 - 00:19
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Melatonin regulates catecholamine biosynthesis by modulating bone morphogenetic protein and glucocorticoid actions.

J Steroid Biochem Mol Biol. 2017 Jan;165(Pt B):182-189

Authors: Komatsubara M, Hara T, Hosoya T, Toma K, Tsukamoto-Yamauchi N, Iwata N, Inagaki K, Wada J, Otsuka F

Abstract
Melatonin is functionally involved in the control of circadian rhythm and hormonal secretion. In the present study, we investigated the roles of melatonin in the interaction of catecholamine synthesis with adrenocortical steroids by focusing on bone morphogenetic protein (BMP)-4 expressed in the adrenal medulla using rat pheochromocytoma PC12 cells. Melatonin treatment significantly reduced the mRNA expression of catecholamine synthases, including the rate-limiting enzyme tyrosine hydroxylase (Th), 3,4-dihydroxyphenylalanine decarboxylase and dopamine-β-hydroxylase expressed in PC12 cells. In accordance with changes in the expression levels of enzymes, dopamine production and cAMP synthesis determined in the culture medium and cell lysate were also suppressed by melatonin. The MT1 receptor, but not the MT2 receptor, was expressed in PC12 cells, and luzindole treatment reversed the inhibitory effect of melatonin on Th expression, suggesting that MT1 is a functional receptor for the control of catecholamine synthesis. Interestingly, melatonin enhanced the inhibitory effect of BMP-4 on Th mRNA expression in PC12 cells. Melatonin treatment accelerated BMP-4-induced phosphorylation of SMAD1/5/8 and transcription of the BMP target gene Id1. Of note, melatonin significantly upregulated Alk2 and Bmpr2 mRNA levels but suppressed inhibitory Smad6/7 expression, leading to the enhancement of SMAD1/5/8 signaling in PC12 cells, while BMP-4 did not affect Mt1 expression. Regarding the interaction with adrenocortical steroids, melatonin preferentially enhanced glucocorticoid-induced Th mRNA through upregulation of the glucocorticoid receptor and downregulation of Bmp4 expression, whereas melatonin repressed Th mRNA expression induced by aldosterone or androgen without affecting expression levels of the receptors for mineralocorticoid and androgen. Collectively, the results indicate that melatonin plays a modulatory role in catecholamine synthesis by cooperating with BMP-4 and glucocorticoid in the adrenal medulla.

PMID: 27267863 [PubMed - indexed for MEDLINE]

Group for Research and Assessment of Psoriasis and Psoriatic Arthritis 2015 Treatment Recommendations for Psoriatic Arthritis.

Fri, 07/14/2017 - 00:19
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Group for Research and Assessment of Psoriasis and Psoriatic Arthritis 2015 Treatment Recommendations for Psoriatic Arthritis.

Arthritis Rheumatol. 2016 May;68(5):1060-71

Authors: Coates LC, Kavanaugh A, Mease PJ, Soriano ER, Laura Acosta-Felquer M, Armstrong AW, Bautista-Molano W, Boehncke WH, Campbell W, Cauli A, Espinoza LR, FitzGerald O, Gladman DD, Gottlieb A, Helliwell PS, Husni ME, Love TJ, Lubrano E, McHugh N, Nash P, Ogdie A, Orbai AM, Parkinson A, O'Sullivan D, Rosen CF, Schwartzman S, Siegel EL, Toloza S, Tuong W, Ritchlin CT

Abstract
OBJECTIVE: To update the 2009 Group for Research and Assessment of Psoriasis and Psoriatic Arthritis (GRAPPA) treatment recommendations for the spectrum of manifestations affecting patients with psoriatic arthritis (PsA).
METHODS: GRAPPA rheumatologists, dermatologists, and PsA patients drafted overarching principles for the management of PsA, based on consensus achieved at face-to-face meetings and via online surveys. We conducted literature reviews regarding treatment for the key domains of PsA (arthritis, spondylitis, enthesitis, dactylitis, skin disease, and nail disease) and convened a new group to identify pertinent comorbidities and their effect on treatment. Finally, we drafted treatment recommendations for each of the clinical manifestations and assessed the level of agreement for the overarching principles and treatment recommendations among GRAPPA members, using an online questionnaire.
RESULTS: Six overarching principles had ≥80% agreement among both health care professionals (n = 135) and patient research partners (n = 10). We developed treatment recommendations and a schema incorporating these principles for arthritis, spondylitis, enthesitis, dactylitis, skin disease, nail disease, and comorbidities in the setting of PsA, using the Grading of Recommendations, Assessment, Development and Evaluation process. Agreement of >80% was reached for approval of the individual recommendations and the overall schema.
CONCLUSION: We present overarching principles and updated treatment recommendations for the key manifestations of PsA, including related comorbidities, based on a literature review and consensus of GRAPPA members (rheumatologists, dermatologists, other health care providers, and patient research partners). Further updates are anticipated as the therapeutic landscape in PsA evolves.

PMID: 26749174 [PubMed - indexed for MEDLINE]

adrenal tumor; +22 new citations

Thu, 07/06/2017 - 21:56

22 new pubmed citations were retrieved for your search. Click on the search hyperlink below to display the complete search results:

adrenal tumor

These pubmed results were generated on 2017/07/06

PubMed comprises more than millions of citations for biomedical literature from MEDLINE, life science journals, and online books. Citations may include links to full-text content from PubMed Central and publisher web sites.

adrenal tumor; +22 new citations

Thu, 06/29/2017 - 19:24

22 new pubmed citations were retrieved for your search. Click on the search hyperlink below to display the complete search results:

adrenal tumor

These pubmed results were generated on 2017/06/29

PubMed comprises more than millions of citations for biomedical literature from MEDLINE, life science journals, and online books. Citations may include links to full-text content from PubMed Central and publisher web sites.

Epigenetics of pheochromocytoma and paraganglioma.

Thu, 06/22/2017 - 16:59
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Epigenetics of pheochromocytoma and paraganglioma.

Mol Cell Endocrinol. 2017 Jun 16;:

Authors: Björklund P, Backman S

Abstract
Pheochromocytomas and paragangliomas (PPGLs) are neuroendocrine tumors arising in the medullae of the adrenal glands or in paraganglia. The knowledge of the tumor biology of these lesions has increased dramatically during the past two decades and more than a dozen recurrently mutated genes have been identified. Different clusters have been described that share epigenetic signatures. Mutations in the succinate dehydrogenase complex subunit genes play a pivotal role in reprogramming the epigenetic state of these tumors by inhibiting epigenetic regulators such as TET enzymes and histone demethylases. Another subgroup of tumors carries hypomethylated genomes, and overexpression of several micro-RNAs has been described. While much remains to be investigated regarding the epigenetics of PPGLs, it is clear that it plays an important role in PPGL biology.

PMID: 28630023 [PubMed - as supplied by publisher]

A Case of Retroperitoneal Paraganglioma.

Thu, 06/22/2017 - 16:59
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A Case of Retroperitoneal Paraganglioma.

J UOEH. 2017;39(2):161-166

Authors: Amaike T, Tamura T, Koga A, Shibao K, Higure A, Hirata K

Abstract
We report a surgical case of retroperitoneal paraganglioma. A paraganglioma is a catecholamine-producing tumor originating in the chromaffin cells of the sympathetic ganglion. It is a kind of pheochromocytoma which occurs on the outside of the adrenal gland. The patient was a 72 year old male with a history of hypertension and a pacemaker implantation. A mass in the ventral side of the right iliopsoas muscle was detected during a routine contrasting computed tomography (CT) examination for checking his pacemaker. The mass was considered to be malignant, and a laparotomy and mass enucleation was performed. It was diagnosed as phaeochromocytoma, based on the pathology and immunestology of the excised specimen. The hypertension was cured soon after the surgery. Nine months after surgery, there is no evidence of any abnormality or recurrence. There is a previous report of a recurrence 25 years after surgery, so a careful follow-up of this patient will be necessary in the future.

PMID: 28626127 [PubMed - in process]

[A Case of Bilateral Adrenal Incidentaloma in which MTX-Related Lymphoproliferative Disease Could Be Diagnosed by Computed Tomography-Guided Biopsy and MTX Administration History].

Thu, 06/22/2017 - 16:59
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[A Case of Bilateral Adrenal Incidentaloma in which MTX-Related Lymphoproliferative Disease Could Be Diagnosed by Computed Tomography-Guided Biopsy and MTX Administration History].

Hinyokika Kiyo. 2017 May;63(5):201-206

Authors: Kitakaze H, Matsushita M, Okada K, Minato N, Mori N, Yoshioka T

Abstract
The patient was a 76-year-old man. Because bilateral adrenal tumor (right adrenal gland 7 cm, left adrenal gland 1.5 cm) was detected in by computed tomography (CT) in methotrexate (MTX) administration for articular rheumatism from 2011, he was referred to this hospital in February, 2016. An endocrine examination, and imaging study did not lead to a definitive diagnosis and CT-guided lower needle biopsy was performed. The pathological diagnosis was diffuse large B cell lymphoma. Also, in situ hybridization revealed EBER-positive and the diagnosis of MTX-related lymphoproliferative disease (MTXLPD) was made in conjunction with the medical history. After MTX cancellation, the tumor became markedly smaller. The annual incidence of this disorder in the RA patients during MTX internal use is reported as 0.06%. According to the site of origin, lymphatic extranodal disease accounts for approximately half of the cases, but this is the third case of primary adrenal origin reported.

PMID: 28625027 [PubMed - in process]

Collision and composite tumors; radiologic and pathologic correlation.

Thu, 06/22/2017 - 16:59
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Collision and composite tumors; radiologic and pathologic correlation.

Abdom Radiol (NY). 2017 Jun 16;:

Authors: Sung CT, Shetty A, Menias CO, Houshyar R, Chatterjee S, Lee TK, Tung P, Helmy M, Lall C

Abstract
The terms composite and collision tumors have been used interchangeably throughout radiological literature. Both composite and collision tumors involve two morphologically and immunohistochemically distinct neoplasms coexisting within a single organ. However, collision tumors lack the histological cellular intermingling seen in composite tumors. Composite tumors often arise from a common driver mutation that induces a divergent histology from a common neoplastic source while collision tumors may arise from coincidental neoplastic change. The purpose of this review is to provide an overview of abdominal composite and collision tumors by discussing hallmark radiographic and pathological presentations of rare hepatic, renal, and adrenal case studies. A better understanding of the presentation of each lesion is imperative for proper recognition, diagnosis, and management of these unique tumor presentations.

PMID: 28623377 [PubMed - as supplied by publisher]

Laparoscopic Partial Adrenalectomy.

Thu, 06/22/2017 - 16:59
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Laparoscopic Partial Adrenalectomy.

Chirurgia (Bucur). 2017 Jan-Feb;112(1):77-81

Authors: Miron A, Giulea C, Nădrăgea M, Enciu O, -

Abstract
Laparoscopic adrenalectomy became the gold standard for adrenal disease, from incidentaloma to cancer. Partial adrenalectomy is difficult to accept due to its technical difficulties as well as hemorrhagic risk and a consensus has not been reached. On the other hand, in selected cases of benign adrenal tumors, adrenalectomy may be futile, partial resections being perfectly justified and with lower hemorrhagic risks. For functioning tumors smaller than 3 cm with an anterior or lateral location, partial adrenalectomy may be indicated. The key points reside in adenoma identification, preservation of the remaining glandular parenchyma and its blood supply with dissection in the space between the adenoma and the normal parenchyma. Laparoscopic partial adrenalectomy is feasible and effective for the treatment of benign tumors. Although partial resections have clear-cut advantages over conventional adrenalectomy especially for bilateral tumors, it remains a difficult intervention.

PMID: 28266298 [PubMed - indexed for MEDLINE]

[Value of single-source dual-energy CT (ssDECT) in differentiating lipid-poor adrenal adenomas from metastatic lesions].

Thu, 06/22/2017 - 16:59
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[Value of single-source dual-energy CT (ssDECT) in differentiating lipid-poor adrenal adenomas from metastatic lesions].

Zhonghua Zhong Liu Za Zhi. 2016 Nov 23;38(11):826-832

Authors: Ju Y, Liu AL, Wang HQ, Liu YJ, Liu JH, Chen AL, Chen LH, Li Y, Han Z

Abstract
Objective: To evaluate the value of single-source dual-energy CT (ssDECT) in differentiating lipid-poor adenomas from metastases in adrenal glands. Methods: From August 2011 to Oct 2014, 63 patients with 73 adrenal nodules (53 metastases proven by 5-6 months follow-up, and 20 histopathologically proven adenomas, CT value > 10 HU) underwent ssDECT scanning. The CT values of conventional polychromatic CT and virtual monochromatic images (40-140 keV) , fat-water density images and effective atomic number (eff-Z) were reconstructed on an ADW4.5 workstation and ROC curves were then constructed to evaluate the effectiveness of each parameter. The slope of spectral curve was measured and divided into 3 types: increment curve (K>0.1), straight curve (-0.1≤K≤0.1) and decrement curve (K< -0.1) according to the slope (the value of K) of spectral curve, and the curve patterns in the two groups were compared statistically. Results: There was no statistical difference between the mean CT values of metastases (35.12±5.29)HU and lipid-poor adenomas (32.48±6.94)HU by conventional polychromatic CT (P>0.05). The range of single-energy CT values of metastases [from (53.00±15.12) HU to (33.38±5.67) HU] was significantly higher than that of lipid-poor adenomas [from (26.90±26.94) HU to (28.77±10.66) HU] at energy levels ranging from 40 to 80 keV (P<0.05). There was no significant difference between the single-energy CT value of metastases and lipid-poor adenomas at energy levels ranging from 90 to 140 keV (P>0.05). The median fat-water concentration of metastases was -164.61 μg/cm(3,) significantly lower than that of lipid-poor adenomas (114.32 μg/cm(3,)P<0.05). The eff-Z of metastases (7.76±0.15) was also significantly higher than that of lipid-poor adenomas (7.50±0.25, P<0.05). When the threshold of fat-water concentration was -143.89 μg/cm(3,) the sensitivity, specificity and accuracy rate of metastasis diagnosis was 70.0%, 66.0%, and 76.7%, respectively. When the threshold of eff-Z was 7.63, the sensitivity, specificity and accuracy rate of metastasis diagnosis was 83.0%, 65.0%, and 80.4%, respectively. The lower the energy, the higher the diagnostic accuracy at energy levels ranging from 40 to 80 keV, and that of 40 keV was the highest. The spectral curves of metastases included 2 (3.8%) ascending curves, 9 (17.0%) straight curves and 42 (79.2%) descending curves, while in the 20 lipid-poor adenomas, there were 9 (45.0%) ascending curves, 4 (20.0%) straight curves and 7 (35.0%) descending curves, showing significant differences between the two groups (P<0.05). Conclusions: Single-source dual-energy CT provides an effective multi-parameter approach for differentiating lipid-poor adrenal adenomas from metastases.

PMID: 27998440 [PubMed - indexed for MEDLINE]

Postoperative Complications after Ileocecal Resection in Crohn's Disease: A Prospective Study From the REMIND Group.

Thu, 06/22/2017 - 16:59
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Postoperative Complications after Ileocecal Resection in Crohn's Disease: A Prospective Study From the REMIND Group.

Am J Gastroenterol. 2017 Feb;112(2):337-345

Authors: Fumery M, Seksik P, Auzolle C, Munoz-Bongrand N, Gornet JM, Boschetti G, Cotte E, Buisson A, Dubois A, Pariente B, Zerbib P, Chafai N, Stefanescu C, Panis Y, Marteau P, Pautrat K, Sabbagh C, Filippi J, Chevrier M, Houze P, Jouven X, Treton X, Allez M, REMIND study group investigators

Abstract
OBJECTIVES: We sought to determine the frequency of and risk factors for early (30-day) postoperative complications after ileocecal resection in a well-characterized, prospective cohort of Crohn's disease patients.
METHODS: The REMIND group performed a nationwide study in 9 French university medical centers. Clinical-, biological-, surgical-, and treatment-related data on the 3 months before surgery were collected prospectively. Patients operated on between 1 September 2010 and 30 August 2014 were included.
RESULTS: A total of 209 patients were included. The indication for ileocecal resection was stricturing disease in 109 (52%) cases, penetrating complications in 88 (42%), and medication-refractory inflammatory disease in 12 (6%). A two-stage procedure was performed in 33 (16%) patients. There were no postoperative deaths. Forty-three (21%) patients (23% of the patients with a one-stage procedure vs. 9% of those with a two-stage procedure, P=0.28) experienced a total of 54 early postoperative complications after a median time interval of 5 days (interquartile range, 4-12): intra-abdominal septic complications (n=38), extra-intestinal infections (n=10), and hemorrhage (n=6). Eighteen complications (33%) were severe (Dindo-Clavien III-IV). Reoperation was necessary in 14 (7%) patients, and secondary stomy was performed in 8 (4.5%). In a multivariate analysis, corticosteroid treatment in the 4 weeks before surgery was significantly associated with an elevated postoperative complication rate (odds ratio (95% confidence interval)=2.69 (1.15-6.29); P=0.022). Neither preoperative exposure to anti-tumor necrosis factor (TNF) agents (n=93, 44%) nor trough serum anti-TNF levels were significant risk factors for postoperative complications.
CONCLUSIONS: In this large, nationwide, prospective cohort, postoperative complications were observed after 21% of the ileocecal resections. Corticosteroid treatment in the 4 weeks before surgery was significantly associated with an elevated postoperative complication rate. In contrast, preoperative anti-TNF therapy (regardless of the serum level or the time interval between last administration and surgery) was not associated with an elevated risk of postoperative complications.

PMID: 27958285 [PubMed - indexed for MEDLINE]

A successfully treated case of cardiac arrest after Caesarean section complicated by pheochromocytoma crisis and amniotic fluid embolism.

Thu, 06/22/2017 - 16:59
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A successfully treated case of cardiac arrest after Caesarean section complicated by pheochromocytoma crisis and amniotic fluid embolism.

J Anesth. 2017 Feb;31(1):140-143

Authors: Mita K, Tsugita K, Yasuda Y, Matsuki Y, Obata Y, Matsuki Y, Kamisawa S, Shigemi K

Abstract
Both pheochromocytoma and amniotic fluid embolism (AFE) are important causes of maternal mortality. We present a case of a 29-year-old woman who developed cardiac arrest after Caesarean section, complicated by both pheochromocytoma crisis and AFE. After resuscitation, the patient developed multiple organ dysfunction, rhabdomyolysis and disseminated intravascular coagulation (DIC). After institution of multidisciplinary interventions (including the use of an intra-aortic balloon pump, extracorporeal membrane oxygenation, continuous hemodiafiltration, and neuroprotective therapeutic hypothermia) the patient made a full recovery without any apparent neurological deficit.

PMID: 27832332 [PubMed - indexed for MEDLINE]

Fourth-Generation Progestins Inhibit 3β-Hydroxysteroid Dehydrogenase Type 2 and Modulate the Biosynthesis of Endogenous Steroids.

Thu, 06/22/2017 - 16:59
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Fourth-Generation Progestins Inhibit 3β-Hydroxysteroid Dehydrogenase Type 2 and Modulate the Biosynthesis of Endogenous Steroids.

PLoS One. 2016;11(10):e0164170

Authors: Louw-du Toit R, Perkins MS, Snoep JL, Storbeck KH, Africander D

Abstract
Progestins used in contraception and hormone replacement therapy are synthetic compounds designed to mimic the actions of the natural hormone progesterone and are classed into four consecutive generations. The biological actions of progestins are primarily determined by their interactions with steroid receptors, and factors such as metabolism, pharmacokinetics, bioavailability and the regulation of endogenous steroid hormone biosynthesis are often overlooked. Although some studies have investigated the effects of select progestins on a few steroidogenic enzymes, studies comparing the effects of progestins from different generations are lacking. This study therefore explored the putative modulatory effects of progestins on de novo steroid synthesis in the adrenal by comparing the effects of select progestins from the respective generations, on endogenous steroid hormone production by the H295R human adrenocortical carcinoma cell line. Ultra-performance liquid chromatography/tandem mass spectrometry analysis showed that the fourth-generation progestins, nestorone (NES), nomegestrol acetate (NoMAC) and drospirenone (DRSP), unlike the progestins selected from the first three generations, modulate the biosynthesis of several endogenous steroids. Subsequent assays performed in COS-1 cells expressing human 3βHSD2, suggest that these progestins modulate the biosynthesis of steroid hormones by inhibiting the activity of 3βHSD2. The Ki values determined for the inhibition of human 3βHSD2 by NES (9.5 ± 0.96 nM), NoMAC (29 ± 7.1 nM) and DRSP (232 ± 38 nM) were within the reported concentration ranges for the contraceptive use of these progestins in vivo. Taken together, our results suggest that newer, fourth-generation progestins may exert both positive and negative physiological effects via the modulation of endogenous steroid hormone biosynthesis.

PMID: 27706226 [PubMed - indexed for MEDLINE]

Solitary adrenal metastasis from early-stage dedifferentiated endometrial carcinoma: CT findings and review of the literature.

Thu, 06/22/2017 - 16:59
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Solitary adrenal metastasis from early-stage dedifferentiated endometrial carcinoma: CT findings and review of the literature.

J Obstet Gynaecol. 2016 10;36(7):881-882

Authors: Mouka V, Tsili AC, Messinis T, Papoudou-Bai A, Kamina S, Argyropoulou MI

PMID: 27624653 [PubMed - indexed for MEDLINE]

Nonsurgical options to treat giant-cell tumors of the head and neck: A case report and brief review of the literature.

Thu, 06/22/2017 - 16:59
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Nonsurgical options to treat giant-cell tumors of the head and neck: A case report and brief review of the literature.

Ear Nose Throat J. 2016 Jul;95(7):E29-34

Authors: Rasband-Lindquist AN, Lindquist JD, Larsen CG, Thiessen A, Girod D

Abstract
Central giant-cell granulomas (CGCGs) are relatively uncommon. When they do occur, they typically arise in the mandible and maxilla. Some lesions are more destructive than others, and the destructive subtype has a tendency to recur. Unfortunately, there is no reproducible way to differentiate aggressive from nonaggressive subtypes. Treatment of CGCG has historically been based on surgical curettage or wide local excision. However, surgery has been associated with significant morbidity, disfigurement, and expense, as well as a high recurrence rate. Pharmacologic treatments-either as an alternative or an adjunct to surgery-have been shown to yield acceptable results. These agents include intralesional and/or systemic corticosteroids, bisphosphonates, calcitonin, and interferon alfa. These options are typically less expensive than surgery, and they are associated with few side effects, which makes them potentially more desirable. We report the case of a 36-year-old woman with a CGCG who was successfully treated with a combination of an intralesional steroid and an oral steroid over a period of 5 months. As evidenced by this case, medical management can be effective for tumor regression in treating CGCG of the head and neck, and it is ultimately associated with less morbidity and is less costly. To the best of our knowledge, no randomized controlled studies have been published on this topic. Such a study would be welcome, particularly considering the presence of both aggressive and nonaggressive subtypes of CGCG. We also briefly review the literature.

PMID: 27434482 [PubMed - indexed for MEDLINE]

Epidemiologic, Clinicopathologic, Diagnostic, and Management Challenges of Pityriasis Rubra Pilaris: A Case Series of 100 Patients.

Thu, 06/22/2017 - 16:59
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Epidemiologic, Clinicopathologic, Diagnostic, and Management Challenges of Pityriasis Rubra Pilaris: A Case Series of 100 Patients.

JAMA Dermatol. 2016 Jun 01;152(6):670-5

Authors: Ross NA, Chung HJ, Li Q, Andrews JP, Keller MS, Uitto J

Abstract
IMPORTANCE: Pityriasis rubra pilaris (PRP) is a rare papulosquamous disorder with limited epidemiologic and clinicopathologic data. Little information is available on long-term outcomes, comorbidities, and treatment efficacy.
OBJECTIVE: To evaluate objective and subjective disease experience metrics from the perspectives of patients and clinicians.
DESIGN, SETTING, AND PARTICIPANTS: One hundred patients with a putative diagnosis of PRP and who elected to participate completed a comprehensive survey, followed by acquisition of their medical records, including histopathology slides and reports. The data were analyzed separately from the health care clinician and the patient perspectives. Two academic dermatologists examined clinical notes, pathology reports, and photographs, confirming diagnoses via predetermined criteria. Patients were categorized into 4 levels of diagnostic certainty to allow stratification of the findings for subgroup analysis. Patients with a diagnosis of PRP were solicited through patient support organization websites.
MAIN OUTCOMES AND MEASURES: Clinical outcomes, unexpected association of comorbidities, and efficacy (or lack of it) of various treatment modalities.
RESULTS: Among the 100 patients, 50 were diagnosed as having classic, unquestionable PRP. The patients were a median of 61 years old (range, 5-87 years), and 46% were female. Fifty were categorized as level 1 diagnostic certainty, 15 as level 2, 30 as level 3, and 5 as level 4. Of the level 1 patients, 13 (26%) were correctly diagnosed at initial presentation; diagnosis was delayed, on average, by 29 months (range, 0.25-288 months; median, 2 months); and 27 (54%) having undergone 2 or more biopsies. At enrollment, PRP symptoms had persisted in 36 patients (72%) for an average of 58 months (range, 1-300 months; median, 30 months). Thirty-one patients (62%) had comorbidities, including hypothyroidism (20%). Nearly all patients (98%) received some form of therapy. Patients cited topical emollients, corticosteroids, and salicylic acid along with oral retinoids, methotrexate, and tumor necrosis factor inhibitors as most helpful.
CONCLUSIONS AND RELEVANCE: Pityriasis rubra pilaris remains a challenging diagnosis without established and specific treatment. Our data highlight new potential avenues for research with therapeutic perspective.

PMID: 26963004 [PubMed - indexed for MEDLINE]

Adrenal and thyroid metachronous metastases from renal cell carcinoma.

Thu, 06/22/2017 - 16:59
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Adrenal and thyroid metachronous metastases from renal cell carcinoma.

Endocrine. 2016 Jul;53(1):335-6

Authors: Taguchi T, Karashima T, Terada Y

PMID: 26611247 [PubMed - indexed for MEDLINE]

AN INDIVIDUALIZED APPROACH TO THE EVALUATION OF CUSHING SYNDROME.

Thu, 06/15/2017 - 14:02

AN INDIVIDUALIZED APPROACH TO THE EVALUATION OF CUSHING SYNDROME.

Endocr Pract. 2017 Jun;23(6):726-737

Authors: Sharma ST, AACE Adrenal Scientific Committee

Abstract
Cushing syndrome (CS) is caused by chronic exposure to excess glucocorticoids. Early recognition and treatment of hypercortisolemia can lead to decreased morbidity and mortality. The diagnosis of CS and thereafter, establishing the cause can often be difficult, especially in patients with mild and cyclic hypercortisolism. Surgical excision of the cause of excess glucocorticoids is the optimal treatment for CS. Medical therapy (steroidogenesis inhibitors, medications that decrease adrenocorticotropic hormone [ACTH] levels or glucocorticoid antagonists) and pituitary radiotherapy may be needed as adjunctive treatment modalities in patients with residual, recurrent or metastatic disease, in preparation for surgery, or when surgery is contraindicated. A multidisciplinary team approach, individualized treatment plan and long-term follow-up are important for optimal management of hypercortisolemia and the comorbidities associated with CS.
ABBREVIATIONS: ACTH = adrenocorticotropic hormone; BIPSS = bilateral inferior petrosal sinus sampling; CBG = corticosteroid-binding globulin; CD = Cushing disease; CRH = corticotropin-releasing hormone; CS = Cushing syndrome; Dex = dexamethasone; DST = dexamethasone suppression test; EAS = ectopic ACTH syndrome; FDA = U.S. Food & Drug Administration; HDDST = high-dose DST; IPS/P = inferior petrosal sinus to peripheral; MRI = magnetic resonance imaging; NET = neuroendocrine tumor; PET = positron emission tomography; UFC = urinary free cortisol.

PMID: 28614036 [PubMed - in process]

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