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See detailEctopic Hormones-Secreting Pheochromocytoma: A Francophone Observational Study
KIRKBY-BOTT, James; BRUNAUD, Laurent; MATHONET, Muriel et al

in World Journal of Surgery (2012), 36

Background Ectopic hormone-secreting pheochromocytomas <br />are rare; only case reports exist in the literature. This <br />condition has been linked with increased malignancy, <br />familial syndromes ... [more ▼]

Background Ectopic hormone-secreting pheochromocytomas <br />are rare; only case reports exist in the literature. This <br />condition has been linked with increased malignancy, <br />familial syndromes, and ACTH secretion. We wanted to <br />test these hypotheses and shed light on the nature of ectopic <br />hormone-secreting pheochromocytomas. <br />Methods This is a multicenter (francophone) observational <br />study. Inclusion was based upon abnormal preoperative <br />hormone tests in patients with pheochromocytoma <br />that normalized after removal of the tumor. Where <br />possible, immunohistochemistry was performed to confirm <br />that ectopic secretion came from the tumor. <br />Results Sixteen cases were found: nine female and seven <br />male patients. Median age was 50.5 (range 31–89) years. <br />Most presented with hypertension, diabetes, or cushingoid <br />features. Ten patients had specific symptoms from the <br />ectopic hormone secretion. Two had a familial syndrome. <br />Of eight patients with excess cortisol secretion, three died <br />as a result of the tumor resection: two had pheochromocytomas[ <br />15 cm and their associated cortisol hypersecretion <br />complicated their postoperative course. The other died <br />from a torn subhepatic vein. The 13 survivors did not <br />develop any evidence of malignancy during follow-up <br />(median 50 months). Symptoms from the ectopic secretion <br />resolved after removal of the tumor. Immunohistochemistry <br />was performed and was positive in eight tumors: five <br />ACTH, three calcitonins, and one VIP. <br />Conclusions Most pheochromocytomas with ectopic <br />secretion are neither malignant nor familial. Most ectopic <br />hormone-secreting pheochromocytoma cause hypercortisolemia. <br />Patients with a pheochromocytoma should be <br />worked up for ectopic hormones, because removal of the <br />pheochromocytoma resolves those symptoms. Associated <br />cortisol secretion needs careful attention. [less ▲]

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See detailRisk factors and causes of death in MEN1 disease. A GTE (Groupe d'Etude des Tumeurs Endocrines) cohort study among 758 patients.
Goudet, P.; Murat, A.; Binquet, C. et al

in World Journal of Surgery (2010), 34(2), 249-255

Background - The natural history of multiple endocrine neoplasia type 1 (MEN1) is known through single-institution or single-family studies. We aimed to analyze the risk factors and causes of death in a ... [more ▼]

Background - The natural history of multiple endocrine neoplasia type 1 (MEN1) is known through single-institution or single-family studies. We aimed to analyze the risk factors and causes of death in a large cohort of MEN1 patients. Methods - Overall, 758 symptomatic MEN1 patients were identified through the GTE network (Groupe d’étude des Tumeurs Endocrines), which involves French and Belgian genetics laboratories responsible for MEN1 diagnosis and 80 clinical reference centers. The causes of death were analyzed. A frailty model, including time-dependent variables, was used to assess the impact of each clinical lesion, except for hyperparathyroidism, on survival. Results - The median follow-up was 6.3 years. Female gender, family history of MEN1, and recent diagnosis were associated with a lower risk of death. Compared with nonaffected patients, those with thymic tumors (hazard ratio [HR] = 4.64, 95% CI = 1.73-12.41), glucagonomas–vipomas–somatostatinomas (HR = 4.29, 95% CI = 1.54-11.93), nonfunctioning pancreatic tumors (HR = 3.43, 95% CI = 1.71-6.88), and gastrinoma (HR = 1.89, 95% CI = 1.09-3.25) had a higher risk of death after adjustment for age, gender, and diagnosis period. The increased risk of death among patients with adrenal tumors was not significant, but three patients died from aggressive adrenal tumors. Pituitary tumors, insulinomas, and bronchial tumors did not increase the risk of death. The proportion of MEN1-related deaths decreased from 76.8 to 71.4% after 1990. Conclusions - The prognosis of MEN1 disease has improved since 1980. Thymic tumors and duodenopancreatic tumors, including nonsecreting pancreatic tumors, increased the risk of death. Rare but aggressive adrenal tumors may also cause death. Most deaths were related to MEN1. New recommendations on abdominal and thoracic imaging are required. [less ▲]

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See detailMulticenter Study of 19 Aortopulmonary Window parathyroid Tumors : The callenge of Embryologic origin
Arnault, Vincent; Beaulieu, Anthony; Lifante, Jean-Christophe et al

in World Journal of Surgery (2010), 34

Background Ectopic abnormal parathyroid glands are relatively common in the superior mediastinum but are rarely situated in the aortopulmonary window (APW). The embryological origin of these abnormal ... [more ▼]

Background Ectopic abnormal parathyroid glands are relatively common in the superior mediastinum but are rarely situated in the aortopulmonary window (APW). The embryological origin of these abnormal parathyroid glands is controversial. The purpose of this investigation was to investigate the embryological origin and the surgical management of abnormal parathyroid glands situated in the APW. Methods The databases of patients operated on for primary, secondary, and tertiary hyperparathyroidism at eight European medical centers with a special interest in endocrine surgery were reviewed to identify those with APW adenomas. Demographic features, localization procedures, and perioperative and pathology findings were documented. The embryological origin was determined based on the number and position of identified parathyroid glands. Results Nineteen (0.24%) APW parathyroid tumors were identified in 7,869 patients who underwent an operation for hyperparathyroidism (HPT) and 181 patients (2.3%) with mediastinal abnormal parathyroid glands. Ten patients had primary, eight had secondary, and one had tertiary HPT. Sixteen patients had undergone previous unsuccessful cervical exploration. In three patients, an APW adenoma was suspected by preoperative localization studies and was cured at the initial operation. Sixteen patients had persistent HPTof whom 15 were reoperated, resulting in 6 failures. Evaluation of 17 patients who had bilateral neck exploration allowed us to determine the most probable origin of the APW parathyroid tumors: 12 were supernumerary, 4 appeared to originate from a superior, and 1 from an inferior gland. Conclusions Abnormal parathyroid glands situated in the APW are rare and usually identified after an unsuccessful cervical exploration. Preoperative imaging of the mediastinum and neck are essential. The origin of these ectopically situated tumors is probably, as suggested by our data, from a supernumerary fifth parathyroid gland or from abnormal migration of a superior parathyroid gland during the embryologic development. [less ▲]

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See detailHyperparathyroidism in multiple endocrine neoplasia type 1 : Surgical trends and results : a 256-patient series from the genem study group
Goudet, P.; Cougard, P.; Verges, B. et al

in World Journal of Surgery (2001), 25(7), 886-890

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See detailSurgical management of amiodarone-associated thyrotoxicosis: too risky or too effective?
Hamoir, Etienne ULg; Meurisse, Michel ULg; Defechereux, Thierry ULg et al

in World Journal of Surgery (1998), 22(6), 537-42542-3

Amiodarone-associated thyrotoxicosis, often clinically mild and resolutive after amiodarone discontinuation or under medical therapy, is sometimes drug unresponsive and not uncommonly follows a dramatic ... [more ▼]

Amiodarone-associated thyrotoxicosis, often clinically mild and resolutive after amiodarone discontinuation or under medical therapy, is sometimes drug unresponsive and not uncommonly follows a dramatic, even fatal course. Therefore, we considered a surgical solution in 15 severely amiodarone-associated thyrotoxic patients. Twelve men and three women (mean age 68 years, range 50-84 years) underwent radical thyroidectomy for clinical and biologically proved amiodarone-associated thyrotoxicosis. In six surgery was the first-line therapeutic option. In the other nine thyroidectomy seemed unavoidable considering the unresponsiveness to medical therapy and rapid deterioration of the patients' clinical condition, with life-threatening cardiac failure in three. In every patient surgery was conducted without immediate or delayed complications. Total thyroidectomy proved uniformly, definitively, and rapidly effective in controlling thyrotoxicosis in all patients, with a spectacular reversal of cardiac failure in the three most critical cases. Surgery was beneficial to our 15 patients and undoubtedly life-saving in the three most worrying cases. These results suggest that thyroidectomy should be more liberally regarded as an interesting alternative to conventional, but unpredictably effective, medical therapies. [less ▲]

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See detailAmiodarone-induced thyrotoxicosis: is there a place for surgery?
Meurisse, Michel ULg; Hamoir, Etienne ULg; D'Silva, Milbhor et al

in World Journal of Surgery (1993), 17(5), 622-6627

Amiodarone-induced hyperthyroidism has on most instances been reported as mild, and thyroid functions return to normal after discontinuation of the drug. Nevertheless, life-threatening amiodarone-induced ... [more ▼]

Amiodarone-induced hyperthyroidism has on most instances been reported as mild, and thyroid functions return to normal after discontinuation of the drug. Nevertheless, life-threatening amiodarone-induced thyrotoxicosis has also been described. Conventional treatments such as antithyroid drugs (thionamide) and corticosteroids are essentially ineffective or fail to alter the dramatic course of the thyroid crisis. This limited effectiveness of medical therapy, particularly in patients with previously neglected or unknown thyroid disease, prompted us to intervene surgically. We report a series of nine patients who underwent total or near-total thyroidectomy as a first-line therapy for five of them. Surgery resulted in rapid resolution of thyrotoxicosis with an uneventful postoperative course. This approach has the advantage of immediate effectivity, low risk of relapse, and appears to be the only treatment that permits continued therapy with amiodarone when the drug appears needed to control a life-threatening arrhythmia. [less ▲]

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