Biomedical subjects
Publications and source records attributed to G Somers.
We describe a patient with Whipple's disease without apparent intestinal involvement at initial presentation. Electron microscopy demonstrated the typical bacilli in PAS-negative lymph node and muscle biopsy specimens.
A 37-year-old woman with a multiple endocrine neoplasia type 2a underwent bilateral adrenalectomy and total thyroidectomy with parathyroidectomy in 1977. The left-sided pheochromocytoma was considered to be malignant. Seven yr after the initial operation, an enhanced urinary excretion-ratio of N-methyladrenaline/adrenalin suggested the possible presence of either persistent or metastatic pheochromocytoma Iodine-123-labeled metaiodobenzylguanidine scintigraphy (I-123 MIBG) showed a hot spot in the right lung. However, repeated chest-X-rays and computed tomographic scanning of the thorax remained negative. Anatomical localization of the lesion was finally obtained with magnetic resonance imaging. The surgical removal of the small pheochromocytoma was guided by the gamma-ray emission of I-123 MIBG administered iv before the operation. This case report illustrates the usefulness of new analytical methods and of new imaging techniques in the diagnosis and management of metastatic pheochromocytoma.
This paper presents the findings on two patients with a final diagnosis of dermatomyositis in whom 111In-antimyosin imaging revealed diffuse muscle necrosis of the limbs. The potential application of this scintigraphic procedure for diagnosis and follow-up of patients with dermatomyositis-polymyositis is addressed.
Peripheral levels of basal and tolbutamide-induced somatostatin have been measured in patients with diabetes or impaired glucose tolerance (IGT) and compared with those in normal individuals. Basal somatostatin was significantly higher in patients with Type 1 diabetes than in age-matched control subjects. This increase was most pronounced at diagnosis, and appeared to be related to metabolic control in insulin-treated patients. No increase was noted in patients with Type 2 diabetes or with IGT. Intravenous bolus injection of tolbutamide enhanced peripheral somatostatin levels in healthy volunteers in a biphasic manner. Patients with IGT also exhibited a biphasic response but the amplitude of the first phase was higher. No secretory response was detected in 27/29 Type 1 diabetic patients at diagnosis; a somatostatin response to tolbutamide became detectable again in Type 1 patients with normalization of their basal somatostatin levels but was then paradoxically related to poor blood glucose control. In Type 2 diabetes, basal somatostatin levels were similar to age-matched control subjects, but decreased upon intravenous tolbutamide administration.
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The usefulness of radio-metaiodobenzylguanidine (MIBG), a specific radiopharmaceutical agent for scintigraphic imaging and treatment of phaeochromocytoma and neuroblastoma, has been extended to the location of carcinoid tumors. Scintigraphic evaluation with I-123 MIBG in a patient with a histologically proven endocrine tumor (apudoma) of unknown origin with liver and bone metastases is reported. Elevated plasma hormone levels of gastrin, pancreatic polypeptide, and serotonin were found. Tumoral content of these hormones was immunocytochemically confirmed on liver biopsy. I-123 MIBG uptake could be seen in those areas of the liver with deficient lesions in the Tc-99m colloid image with a maximal uptake in a large mass at the level of the left liver lobe. No abnormal uptake could be observed at any other level, which was in contrast with autopsy findings of generalized metastatic disease.
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A case of Sweet's syndrome treated with potassium iodide is hereby described. The patient responded well a few days after the initiation of therapy, but the evolution was complicated with a severe clinical deterioration two weeks later. Systemic vasculitis was diagnosed on the basis of significant impairment of renal function, involvement of the eyes (papillitis) and the skin biopsy which showed a leukocytoclastic vasculitis. This systemic vasculitis was attributed to the potassium iodide therapy.
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A patient is described in whom the diagnosis of pheochromocytoma was suspected on clinical grounds. He had normal urinary and plasma norepinephrine levels, with normal suppression of plasma norepinephrine by clonidine. However, plasma and urinary epinephrine levels were above normal, and plasma epinephrine was not suppressed but increased by clonidine. Selective venous sampling showed marked unilateral adrenal epinephrine excess. A large epinephrine secreting pheochromocytoma was surgically removed. This case suggests that, in pheochromocytomas releasing predominantly epinephrine, normal norepinephrine release from axon terminals of sympathetic postganglionic neurons is maintained and is suppressed by clonidine administration. It further reaffirms the value of plasma epinephrine estimations in the diagnosis of pheochromocytoma and indicates that normal suppression of plasma norepinephrine by clonidine does not preclude the presence of a predominantly epinephrine secreting tumor.
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A case of progressive diaphyseal dysplasia (Engelmann's disease) is described in a 23-year old woman. The disease is characterized by limb pain, muscular wasting, a waddling gait and easy fatiguability. It is diagnosed radiologically by cortical thickening, a narrowed medullary cavity, and increased diameter of the diaphyses of the long bones. The extension of the bone lesions was evaluated by scintigraphy using 99mTc methylene diphosphonate (MDP) and 99mTc colloids. Corticosteroid treatment resulted in clinical improvement and reduction of the diaphyseal uptake of MDP and colloids.
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A patient presented with the clinical and laboratory features of the ectopic ACTH syndrome. No ACTH-producing tumor was found, and bilateral adrenalectomy was performed to correct the hypercortisolism. Six months later, the removal of a pseudotumor containing only fat and inflammatory tissue resulted in normalization of both basal plasma ACTH levels and the ACTH feedback response to cortisol infusion. It is suggested that the leukocytes in the inflammatory tissue were the source of the ectopic ACTH production.
A case of MEN type IIa is described showing among other symptoms, three attacks of transient neurological symptoms. These were attributed to a localized, transient cerebral ischemia. The latter probably developed as a consequence of the concomitant occurrence of different phenomena: the existence of hypercalcemia and on the other hand the secretion of vasoactive substances by one of the three tumours forming the MEN type IIa syndrome. The pheochromocytoma played an essential role because the symptoms did not recur after the bilateral adrenalectomy.
Five cases of somatostatinoma are reported, four being primarily located in the pancreas and one in the duodenum. The diagnosis was based upon the histological and immunochemical characteristics of tumoral and metastatic tissue. A marked clinical heterogeneity was noted: one patient presented with gallstones, steatorrhea, and diabetes, two patients suffered from severe hypoglycemic attacks, and two cases were admitted for obstructive jaundice. This varying symptomatology was related to differences in the circulating levels of biologically active somatostatin and to a variable cellular composition of the tumor. In all cases, a basal and/or tolbutamide-induced hypersomatostatinemia was measured. It is concluded that the clinical and hormonal features of the earlier defined somatostatinoma syndrome are no requisite for the diagnosis of somatostatinoma; the analysis of plasma somatostatin immunoreactivity might lead to a higher detection rate of this endocrine tumor.