Advances in the treatment of endocrine disorders.
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Adrenal lymphangiectatic cyst is a very rare pathological and clinical disease entity, and its clinical silence and lack of characteristic symptoms and signs makes it difficult to diagnose preoperatively. We experienced a case of adrenal lymphangiectatic cyst, accompanied by severe refractory hypertension, which was corrected by surgical removal of the cyst. We report it with a review of the literature.
INTRODUCTION: Adrenal masses discovered by imaging techniques for reasons unrelated to adrenal diseases are called adrenal incidentalomas (Al). The aim of this study was to find out the clinical outcome of 28 patients operated for incidentally discovered adrenal mass and to update the literature concerning this topic. PATIENTS AND METHODS: From September 1976 to December 1999 we operated on 28 patients for adrenal incidentaloma. Adrenal masses were unilateral in 25 cases and bilateral in 5. Average age was 57 years (range 10-73). Hormonal study was performed in all patients. All patients underwent adrenalectomy by the transabdominal subcostal approach. RESULTS: Histopathology assessed the adrenal masses as primary in 19 patients and secondary in 9. 24-hour urinary vanillylmandelic acid (VMA) excretion was elevated in 2 patients. Adrenal insufficiency was detected in 1 case. Average tumor diameter resulted 5.8 cm (range 2-17). Histopathologic features of primary adrenal masses included pheochromocytoma in 5 cases, cysts in 4, myelolipomas in 3. nodular hyperplasia in 2, tuberculous mass in 1, cortical adenoma in 1, extra-bone marrow hemopoiesis in 1, cortical carcinoma in 1 and neuroendocrine tumor of the adrenal medulla in 1. The 9 adrenal metastasis resulted by renal cell carcinoma in 7 patients, urothelial carcinoma of the upper urinary tract in I and primary renal lymphoma in 1. Average follow-up was 68 months (range 6-246). Patients alive were 18 (64%), deal 10 (36%). Of the 19 patients with primary adrenal tumors 16 (84%) were alive and disease free and 3 (16%) died (I for disease and 2 for reasons unrelated to the primary tumor). Of the 9 patients with adrenal metastasis 2 (22%) were alive (I disease free and I with progression of the disease) and 7 (78%) died for disease. Replacement therapy for adrenocortical hormones was given 5 patients. CONCLUSIONS: Management of Al need CT or MRI and hormonal investigation in order to detect malignancy and subclinical hypersecretory syndromes. Subclinical functional adrenal masses, single adrenal metastasis and primary nonhypersecretory adrenal tumors sized 4 cm are treated by surgery. A close morpho-functional follow-up is indicated for primary adrenal incidentalomas when nonhypersecretory and smaller than 4 cm.
A woman aged 62 developed a septic shock and pulmonary embolism after skin grafting for extensive burns. She was put on anticoagulants. A second shock led to renal insufficiency. Hypercalcaemia developed. A CT scan of the upper abdomen disclosed enlarged adrenal glands. An acute adrenal haemorrhage was suspected. The levels of cortisol were low in the plasma and urine and did not respond to ACTH stimulation. Cortisone replacement therapy improved the condition of the patient and normalized plasma calcium levels. The mechanisms of hypercalcaemia in acute adrenal insufficiency are discussed. Multiple factors have been proposed: haemoconcentration, an increased affinity of plasma proteins for calcium, an increase in the filtrable calcium complexes, and an enhanced calcium mobilization of skeletal origin.
Fourteen cases are reported of monolateral adrenal hemorrhage in newborns who were submitted to US exam at 1 week, 2 weeks, 3 weeks, and 3 months of age. The variability is emphasized of US and pathologic findings according to the patients' age. A concise terminology is suggested allowing the description of morphological and echo-structural patterns in adrenal hemorrhage. Being familiar with US spectrum of neonatal adrenal hemorrhage appearances can provide useful data in the cases with atypical clinical features. Moreover, the differential diagnosis becomes easier of neonatal adrenal hemorrhage and other diseases such as neonatal neuroblastoma, adrenal abscess, cystic neuroblastoma, cortical renal cyst, and obstructed upper cortical renal cyst, and obstructed upper excretory tract in duplicated kidney. The most effective criterion for US differential diagnosis is probably the chronological variability of US findings. Sonography is stressed as an useful and effective imaging modality in the diagnosis and follow-up of adrenal hemorrhage which helps avoid X-ray investigations and unnecessary laparotomies.
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Labelled steroid precursors have been found to be valuable in scintigraphic imaging of localized adrenal disease or even in adrenal hyperplasia. Scanning procedures are non-invasive diagnostic methods and, in the case of the adrenals the images reflect anatomical as well as functional changes in the organs, constituting an advantage over angiographic methods. However, careful preparation of the patient, sophisticated technical prerequisites and a case-oriented programme of the procedure favouring either dexamethasone suppression, comparsion with phantom measurements adrenal profiles and/or multichannel analysis are necessary for the attainment of optimum results on scintigraphy.
Adrenal scintigraphy after i.v. injection of 131I-19-iodocholesterol has been performed in 4 patients with primary aldosteronism, 5 with Cushing's syndrome and 1 patient with phaeochromocytoma. In primary aldosteronism a unilaternal adrenocortical adenoma was demonstrated in 2 patients, while the method failed in 1 patient to visualize a tumour that was localized by measurements of aldosterone concentrations in the adrenal veins and by adrenal venography; in 1 patient none of the methods demonstrated a tumour. In Cushing's syndrome, adrenal scinitgraphy indicated bilateral adrenocortical hyperplasia in 1 patient and visualized the tumour in 2 patients with adrenocortical adenoma. In all patients with Cushing's syndrome due to unilateral adrenocortical tumour, the accumulation of radioactivity in the contralateral adrenal was suppressed. However, a delayed and slight accumulation of the isotope in the suppressed gland contralateral to an adrenocortical carcinoma was misinterpreted and led to exploration on the wrong side since the tumour did not concentrate radioactivity at all. The method failed in 1 patient to localize the adrenocortical tissue responsible for the relapse of Cushing's syndrome after bilateral adrenalectomy for hyperplasia. In the patient with phaeochromocytoma, no radioactivity was found on the tumour. It is conculded that adrenal scintigraphy is a safe and valuable method for localization of adrenal tumours and their differentiation from adrenocortical hyperplasia. Some diagnostic pitfalls do, however, exist, as demonstrated in this series of patients.
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Women with congenital adrenal hyperplasia due to 21-hydroxylase deficiency often have a polycystic ovary-like syndrome, consisting of hyperandrogynism, infertility, menstrual irregularities, and elevated LH levels. This is generally considered secondary to poor control of the congenital adrenal hyperplasia. However, our experience led us to suspect that ovarian hyperandrogenism occurs even when congenital adrenal hyperplasia is well controlled on glucocorticoid therapy. Therefore, we tested the hypothesis that congenital adrenal virilizing disorders result in ovarian hyperandrogenism. We studied eight women with congenital adrenal virilizing disorders, seven with well controlled classic 21-hydroxylase deficiency and one with congenital virilizing adrenal carcinoma removed at 1.7 yr of age. We also studied six women with late-onset 21-hydroxylase deficiency, without signs of congenital virilization. An ovarian source of androgens was assessed after suppressing adrenal function with dexamethasone and then testing pituitary-ovarian function by a GnRH agonist (nafarelin) test. Five women with congenital adrenal virilizing disorders (four with classic 21-hydroxylase deficiency and one with congenital virilizing adrenal carcinoma) and one women with late-onset 21-hydroxylase deficiency had ovarian hyperandrogenism as determined by subnormal suppression of free testosterone after dexamethasone and/or by increased 17-hydroxyprogesterone response to nafarelin while on dexamethasone. All women with congenital adrenal virilization and ovarian hyperandrogenism had elevated LH levels after dexamethasone or elevated early LH response to nafarelin, which suggests that LH excess is the cause of their ovarian hyperandrogenism. This was not the case for the late-onset 21-hydroxylase-deficient woman. Our data are compatible with the hypothesis that congenital adrenal virilization programs the hypothalamic-pituitary axis for hypersecretion of LH at puberty. This is postulated to frequently cause ovarian hyperandrogenism even when adrenal androgen excess is subsequently controlled by glucocorticoid therapy.
The article presents a brief essay dedicated to the development of endocrinology in our country since pre-war time. The article covers the most promising areas of endocrinology, from endemic goiter to the introduction of the newest technologies to endocrinology, and informs the readers about structural and organizational changes in the diabetological service, including the establishment of Institute of Diabetes and Institute of Pediatric Endocrinology, the first ones in Russia, the establishment of diabetological service, including State Registry of Diabetic Patients, a school for adult patients, etc. Another issue is the establishment of Growth Center and the introduction of genetic engineering growth hormone preparations into the practice of treatment of short children. Once again, the authors emphasize the issue of fighting endemic goiter. Many tasks of Russian endocrinology are now considered weighty matters of state.