deamino-arginine-vasopressin and Hypercalcemia

Topics: Adolescent; Biopsy; Deamino Arginine Vasopressin; Diabetes Insipidus; Histiocytosis, Langerhans-Cell; Humans; Hypercalcemia; Hypothalamic Diseases; Magnetic Resonance Imaging; Muscle Weakness; Testosterone

A 48-year-old woman presented with a history of premature menopause, polyuria, polydipsia, fever, and diffuse bony tenderness. Her evaluation revealed central diabetes insipidus, hypothalamic amenorrhea, an elevated free calcium on multiple occasions with an elevated 1,25 dihydroxyvitamin D level, and osteoporosis by densitometry. Skeletal series revealed multiple lytic lesions involving the long bones. The diagnosis of Langerhans' cell granulomatosis was made. She was treated with hormone replacement therapy, radiotherapy, and vinblastine, with a dramatic improvement in her pain and a near normalization of her free calcium. Whereas hypercalcemia has been described in several granulomatous disorders and is secondary to unregulated extrarenal production of 1,25 dihydroxyvitamin D, it is, however, extremely rare in Langerhans' cell granulomatosis. This is the first case report of Langerhans' cell granulomatosis with hypercalcemia and documented elevated increased 1,25 dihydroxyvitamin D level that responded to the treatment of her primary disease.

Topics: Antineoplastic Agents, Phytogenic; Calcitriol; Calcium; Deamino Arginine Vasopressin; Diabetes Insipidus; Estrogen Replacement Therapy; Female; Histiocytosis, Langerhans-Cell; Humans; Hypercalcemia; Middle Aged; Osteoporosis; Vinblastine

We present here a case of prominent hypercalcemia accompanied by hypothalamic tumor and Graves' disease. A 24-year-old man with hypothalamic tumor showed hypopituitarism, central diabetes inspidus (DI) and hyperthyroidism. Nausea, loss of thirst and appetite, and general fatigue were found with the unveiling of hypercalcemia and hypernatremia. Parathyroid hormone (PTH) and 1alpha-dihydroxyvitamin D levels were suppressed with a normal range of PTH-related protein values. One-desamino-(8-D-arginine)-vasopressin (DDAVP) and half-saline administration normalized hypernatremia, while hypercalcemia was still sustained. Administration of cortisone acetate and thiamazole reduced the elevated serum Ca level. In the present case, concurrent hyperthyroidism was assumed to accelerate skeletal mobilization of calcium into the circulation. Hypocortisolism and central DI was also considered to contribute, to some extent, to the hypercalcemia through renal handling of Ca.

Topics: Adult; Antithyroid Agents; Calcitriol; Calcium; Cortisone; Craniotomy; Deamino Arginine Vasopressin; Diabetes Insipidus; Drug Therapy, Combination; Germinoma; Graves Disease; Humans; Hypercalcemia; Hypernatremia; Hyperthyroidism; Hypopituitarism; Hypothalamic Neoplasms; Magnetic Resonance Imaging; Male; Methimazole; Parathyroid Hormone; Parathyroid Hormone-Related Protein; Peptide Fragments; Proteins; Renal Agents; Sodium; Teratoma

Vasopressin function and thirst were studied in fourteen hypercalcaemic patients (ten hyperparathyroid and four disseminated malignant disease). Ten patients had decreased renal concentrating ability which reversed within a few days in the majority of patients whose hypercalcaemia was corrected by parathyroidectomy. Although eight patients complained of thirst, none showed a lowered threshold of thirst appreciation during hypertonic saline infusion. Osmoregulation of vasopressin secretion was not reduced in any patient, but the hyperparathyroid group had an exaggerated vasopressin response to osmotic stimulation. We conclude that a partial, reversible nephrogenic diabetes insipidus occurs in at least 70% of hypercalcaemic patients irrespective of cause, which accounts for the polyuria induced by hypercalcaemia.

Topics: Adult; Aged; Deamino Arginine Vasopressin; Female; Humans; Hypercalcemia; Hyperparathyroidism; Kidney; Kidney Concentrating Ability; Male; Middle Aged; Osmolar Concentration; Parathyroid Glands; Thirst; Vasopressins