Cases reported "Adrenal Gland Neoplasms"

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1/15. Modern imaging methods and preoperative management of pheochromocytoma: review of the literature and case report.

    OBJECTIVES: To discuss the modern imaging techniques and preoperative management of pheochromocytoma and to report on one additional case. methods: A 66-year-old male with an incidentally discovered left adrenal mass is described. The adrenal medulla strongly accumulate 131 I-metaiodobenzylguanidine (MIBG). The patient underwent left adrenalectomy after preoperative therapy with alpha and beta-blockers. The recent literature on pheochromocytoma modern imaging techniques and preoperative management is reviewed. RESULTS: MIBG scintigraphy diagnosed a benign functioning adrenal pheochromocytoma, allowing preoperative medical management. Postoperative workup was unremarkable. diagnosis of pheochromocytoma was confirmed by immunohistopathology. At 18 months follow-up, the patient is alive and disease-free. CONCLUSIONS: Incidentally discovered adrenal masses have to be investigated to detect malignancy and subtle hormonal overproduction. MIBG scintigraphy has a high specificity (100%) in detecting pheochromocytoma, metastasis, surgical residual tumor, local relapse and other adrenal crest tumors. Positive results of octreotide scintigraphy in detecting malignant pheochromocytoma have been reported. Currently, pheochromocytoma removal is a safe operation with mortality rates ranging from 0 to less than 3%. Preoperative alpha adrenergic blockage with phenoxybenzamine or prazosin is important in decreasing the operative risk. Beta-blockers may be necessary for cardiac arrhythmia. Intraoperative invasive monitoring of hemodynamic variables may be both diagnostic and therapeutic of inadequate preoperative management. Lifelong follow-up for patients with pheochromocytoma is important.
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2/15. Molecular and biochemical screening for the diagnosis and management of medullary thyroid carcinoma in multiple endocrine neoplasia type 2a.

    patients with Multiple Endocrine Neoplasia (MEN) type 2A are at risk for early medullary thyroid carcinoma (MTC). We performed different screening tests for MTC--a recently reported biochemical screening test using omeprazole-induced calcitonin (CT) stimulation and dna analysis--in fifteen members of two non-consanguineous Brazilian families with MEN 2A. RET proto-oncogene analysis was carried out by direct dna sequencing of PCR-amplified products for exons 10 and 11. family 1 showed a germline mutation (C634Y) in three individuals; a sister and a brother with symptomatic MTC; the former also presented with pheochromocytoma and hyperparathyroidism, and her son was a nine-year-old boy of previously unknown status. family 2 showed the C634R mutation only in the index case, who presented with cutaneous lichen amyloidosis in addition to MTC, pheochromocytoma and hyperparathyroidism. Neither her parents nor her four brothers showed this genetic abnormality, suggesting a de novo RET proto-oncogene mutation in this patient. The controls and patients presented normal basal gastrin levels and a significant increase after omeprazole. Basal CT levels were elevated in patients with MTC and undetectable in control and asymptomatic family members. No subject showed any increase in CT levels after omeprazole treatment. In conclusion, the two most frequent RET proto-oncogene mutations in MEN 2A are present in Brazilian families. In addition, the specificity of basal and omeprazole-stimulated calcitonin is rather limited, and the efficacy of the omeprazole test still needs to be systematically examined. Therefore, RET proto-oncogene analysis must be the first choice for a screening procedure to identify gene carriers in MEN 2A family members and to permit early prophylactic treatment of MTC.
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3/15. Diagnostic accuracy of radionuclide imaging using 131I nor-cholesterol or meta-iodobenzylguanidine in patients with hypersecreting or non-hypersecreting adrenal tumours.

    The aim of this retrospective study was to evaluate the diagnostic accuracy of nor-cholesterol and meta-iodobenzylguanidine radionuclide imaging in two separate groups of patients with adrenal tumours to characterize lesions as adenoma or pheochromocytoma. We studied 75 patients (22 male and 53 female, mean age 47 /- 15 years) with hypersecreting (n = 32) or non-hypersecreting (n = 43) unilateral adrenal tumours detected by computerized tomography or magnetic resonance scans. 131I nor-cholesterol adrenal scintigraphy was performed in 41 patients. Meta-[131I]iodobenzylguanidine (131I-MIBG) imaging was acquired in the other 34 patients. pathology examinations (n = 58) or computerized tomography follow-up studies (n = 17) were obtained. Adrenal lesions were represented by 44 adenomas, four cysts, one myelolipoma, one pseudotumour, one ganglioneuroma, 16 pheochromocytomas, three carcinomas, four metastases and one sarcoma. Radionuclide studies were qualitatively evaluated and the corresponding results were classified as true positive, true negative, false positive and false negative. Diagnostic sensitivity, specificity and accuracy as well as positive and negative predictive values were calculated. The diagnostic values of nor-cholesterol scintigraphy in identifying adrenal adenomas were sensitivity 100%, specificity 71%, accuracy 95%, positive predictive value 94% and negative predictive value 100%; of note, two false positive cases were observed represented by a pheochromocytoma and a myelolipoma. The diagnostic values of MIBG scintigraphy in recognizing pheochromocytoma were sensitivity 100%, specificity 95%, accuracy 97%, positive predictive value 94% and negative predictive value 100%; only one false positive case occurred consisting of a carcinoma. It is concluded that, in the large majority of cases, adrenal scintigraphy using nor-cholesterol or MIBG is able to characterize specific lesions such as adenoma and pheochromocytoma, respectively. These findings show relevant clinical impact, particularly in patients with non-hypersecreting adrenal lasions. Radiotracer selection depends on clinical patient history and department availability; since benign adenomas are the most common cause of non-hypersecreting tumours, nor-cholesterol should be the first choice followed by MIBG if nor-cholesterol shows normal images. However, rare as well as unusual findings may be observed; nor-cholesterol uptake may occasionally be also found in non-adenoma tumours such as myelolipoma and pheochromocytoma. Similarly, MIBG accumulation may occur not only in lesions arising from medullary chromaffin tissue, but also rarely in cortical adrenal carcinoma.
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4/15. adrenal cortex, tumor, and peripheral production of deoxycorticosterone.

    A method is reported for the measurement of the urine excretion rates of tetrahydro-11-deoxycorticosterone (3 alpha,5 beta-THDOC), an important metabolite of 11-deoxycorticosterone (DOC). Quantification using gas chromatography/mass spectrometry (GC/MS) was achieved by comparing the ion fragment response for the molecular ion (m/z 507) of the analyte (as methyloxime trimethylsilyl ether derivative) to that of a fixed amount of an isomer of THDOC added to urine as internal standard. To improve the specificity of measuring THDOC in clinical samples, an additional Sephadex LH-20 chromatography step was introduced to separate 11-deoxycortisol and some progesterone metabolites. In the luteal phase of the menstrual cycle, THDOC excretion was higher than in the follicular phase; it was also higher than in women taking oral contraceptives. The correlation of THDOC with progesterone production, independent of a constant cortisol output, supports an ovarian or peripheral conversion of progesterone to DOC. The assay proved useful (1) in monitoring for the recurrence of a mineralocorticoid-secreting tumor and (2) when adrenal production of DOC was not fully suppressed in congenital adrenal hyperplasia due to 11 beta-hydroxylase deficiency. Under the latter circumstances, the renin-angiotensin system seemed to be an important regulator of DOC production.
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5/15. in vitro synthesis of steroids by a feminising adrenocortical carcinoma: effect of prolactin and other protein hormones.

    The study describes the effects of ACTH, prolactin and other protein hormones on the synthesis and secretion of steroid hormones by tissue from a feminising adrenocortical carcinoma removed from a post-menopausal female. Steroid production by the tissue was determined by high resolution-mass fragmentography and by radioimmunoassay. prolactin and ACTH stimulated the synthesis of estrogens by the tissue whereas GH, LH and ACTH were more effective than prolactin in stimulating androgen synthesis. The effect of protein hormones, other than ACTH, on adenylate cyclase activity of this tumour tissue indicated a lack of specificity of the membrane receptor sites.
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6/15. pheochromocytoma: diagnosis by scintigraphy using iodine 131 metaiodobenzylguanidine.

    We summarize our experience using iodine 131 metaiodobenzylguanidine (131I MIBG) imaging in 24 patients with suspected pheochromocytoma and compare 131I MIBG imaging with other imaging modalities such as computerized tomography and magnetic resonance imaging. Our results confirm the high sensitivity and excellent specificity reported from other centers experienced in 131I MIBG imaging. This radiopharmaceutical permits noninvasive, safe, accurate detection of pheochromocytomas of all types. iodine 131 MIBG scintigraphy is especially efficacious in the detection of extra-adrenal, recurrent, and metastatic tumors. We review precautions in the screening of patients for 131I MIBG examination, imaging techniques, and drugs that interfere with tumor uptake of 131I MIBG. Though 131I MIBG imaging has some limitations, it deserves strong consideration whenever the diagnosis of pheochromocytoma is suspected.
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7/15. Multiple endocrine neoplasia type IIB identified by magnetic resonance imaging.

    Ectopic, epinephrine-secreting pheochromocytomas are rare and difficult to diagnose. In our case, MRI was able to show the lesion, which could not be clearly identified by CT. The histologic specificity provided by T2-weighted images confirmed the biochemical diagnosis of pheochromocytoma. MRI should be considered as an alternative to CT in the diagnosis of familial pheochromocytomas.
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8/15. Carney's triad: guidelines for management.

    Carney's triad--gastric leiomyosarcoma, pulmonary chondroma, and extra-adrenal paraganglioma--is a syndrome that occurs primarily in young women. To date, 28 patients with at least two of these individually unusual or rare neoplasms have been described. This updated case report of one of the originally described patients with Carney's triad highlights several clinically important features of this unusual syndrome: (1) the multicentricity of both the paragangliomas and the epithelioid leiomyosarcomas, (2) the often indolent progression of metastatic leiomyosarcoma, (3) the potential for late recurrences, and (4) the importance of distinguishing intra-adrenal from periadrenal catecholamine-producing tumors (paragangliomas). Localization of paragangliomas is facilitated by two relatively new techniques--131I-metaiodobenzylguanidine scanning (a scintigraphic technique with high specificity for catecholamine-producing tumors) and two-dimensional echocardiography (which can noninvasively localize and demonstrate the anatomic relationships of aorticopulmonary paragangliomas). In patients with this syndrome, new or recurrent tumors frequently manifest after unusually long asymptomatic intervals. We outline an approach for continued follow-up of patients with one or more of the three neoplasms that constitute the syndrome. Rigorous long-term screening of these patients should not only lead to early recognition and resection of recurrent or new tumors but also enhance our understanding of this intriguing syndrome.
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9/15. Use of 131I-MIBG scintigraphy in the evaluation of suspected pheochromocytoma.

    Studies at the University of michigan have shown that 131I-metaiodobenzylguanidine (131I-MIBG) is an effective agent for the diagnosis and localization of pheochromocytomas and paragangliomas. We conducted a study that confirmed and expanded that finding. From January 1983 to March 1984, 48 patients at our institution had 51 131I-MIBG scans during the workup of suspected sporadic or metastatic pheochromocytoma. Scintigrams were obtained after 500 microCI of 131I-MIBG had been administered intravenously. The final diagnosis (true-positive, false-negative, or false-positive result) was made at operation and pathologic examination. A true-negative diagnosis was confirmed by normal plasma and fractionated urinary levels of catecholamines and metabolites and, in most patients, computed tomography (CT). There were 20 true-positive studies (6 pheochromocytomas, 4 paragangliomas, and 10 metastatic or recurrent pheochromocytomas) and 24 true-negative studies. One patient with a suspected recurrent paraganglioma near the bladder had a false-positive 131I-MIBG scan (and also a false-positive (CT). Among six patients with false-negative scintigrams (three pheochromocytomas, one paraganglioma, and two metastatic lesions), one also had a false-negative CT. The overall sensitivity of 131I-MIBG scanning was 77%, specificity was 96%, and accuracy was 86%. This test is fairly sensitive in the workup of patients with known or suspected recurrent or metastatic pheochromocytoma. It may also be helpful in the evaluation of suspected sporadic pheochromocytoma when CT findings are normal.
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10/15. Cold agglutinin autoimmune hemolytic anemia in nonhematologic malignancies.

    Four patients with nonhematologic malignancies had the simultaneous finding of hemolytic anemia due to high-titer cold agglutinins. In each patient, the cold agglutinin had "anti-I" specificity and was of the IgM kappa immunoglobulin class. Although patients with hematologic malignancies not uncommonly have cold agglutinins, the association between these antibodies and nonhematologic malignancies is unusual.
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