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PubMed2004

[Familial hypomagnesemia with hypercalciuria and nephrocalcinosis. Association with ocular abnormalities].

BACKGROUND: Familial hypomagnesemia with hypercalciuria and nephrocalcinosis is an unusual disease that usually leads to end-stage renal failure. There is no specific treatment and, to a variable degree, patients with this disease present ocular abnormalities. The illness is due to a defect in the reabsorption of magnesium and calcium at the thick ascending limb of Henle because of a mutation of the PCLN-1 gene, which encodes a protein, paracellin-1, which intervenes in the reabsorption of both cations. OBJECTIVE: To review outcome and the incidence of ocular abnormalities in our patients and in cases described in Spain and to compare the incidence found with that in groups from other countries. METHOD: Retrospective study of a group of patients with familial hypomagnesemia with hypercalciuria and nephrocalcinosis diagnosed at a hospital. RESULTS: There were six girls and three boys with clinical symptoms of polyuria, polydipsia, and less frequently, urinary tract infections and lithiasis. All had hypomagnesemia, hypercalciuria and nephrocalcinosis. Five of the patients had renal failure at diagnosis and four underwent transplantation without recurrence. Eight patients had diverse ocular abnormalities. Eighty-one percent of Spanish patients had ocular abnormalities compared with 24 % of those from other countries. There was no evidence of successful medical treatment. CONCLUSIONS: Almost half of the patients presented chronic renal failure at diagnosis and most of the patients reached end-stage renal failure in the second or third decade of life. Normal glomerular filtration rate was found only in patients diagnosed at an early age. The most frequent extra-renal association in Spanish patients (81 %) corresponded to ocular abnormalities. Effective treatment consists of kidney transplantation that completely corrects the tubular disorder.

Adolescent↗

PubMed2002

Hypercalcemia in Langerhans' cell granulomatosis with elevated 1,25 dihydroxyvitamin D (calcitriol) level.

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.

Antineoplastic Agents, Phytogenic↗

PubMed2001

Hemorheology and renal function during cardiopulmonary bypass in infants.

BACKGROUND: Acute renal failure is an occasional complication after cardiopulmonary bypass in infants. Whereas it is well known that postoperative hemodynamics inflict acute renal failure, the influence of extracorporeal circulation on the kidney is less clear. Moreover, changes in blood viscosity occur during and after surgery, which may influence renal dysfunction. For this reason, we investigated the impact of blood viscosity on renal function during cardiopulmonary bypass. METHODS: In 34 patients weighting less than 10 kg, we performed repeated analysis of urine, blood, and plasma viscosity. RESULTS: Polyuria and proteinuria that appeared during cardiopulmonary bypass indicated an elevated transglomerular filtration gradient, which recovered within 24 hours. The appearance of N-acetyl-beta-D-glucosaminidase in the urine, and elevated excretion of sodium, were additionally indicative of mild tubular damage. Elevation of blood viscosity during hypothermic perfusion showed a statistical correlation with proteinuria and N-acetyl-beta-D-glucosaminidaseuria. With hypothermia, the relation of blood viscosity to plasma viscosity became stronger, while the relation to the hematocrit decreased compared to normothermia. CONCLUSIONS: During cardiopulmonary bypass perfusion, the kidney can be stressed by proteinuria and mild tubular damage. Our data provide evidence that the kidneys can be protected by improved blood viscosity during cardioplegia, but this needs confirmation in a prospective interventional study.

Acetylglucosaminidase↗

PubMed2000

Cytogenetic profile of primary pituitary germinoma.

We present a case of a germinoma in the sellar region of a 10-year-old female patient who presented with a history of polydipsia, polyuria and visual disturbances. The tumor was resected and histologically analyzed. Interphase cytogenetics was performed using chromosome specific (peri)-centromeric DNA probes for all the somatic and X chromosomes on fresh frozen tissues. Fluorescent in situ cell hybridization demonstrated accumulated cytogenetic abnormalities involving significant alterations of chromosome 1, 4, 5/19 and 15. The child was treated postoperatively by radiation and now appears well with only minor neurological deficits. At 3-year follow-up no recurrent tumor mass could be demonstrated.

Child↗

PubMed1999

A case of lymphocytic infundibuloneurohypophysitis: histophathological studies.

A 57-year-old woman presented with 2-year history of polyuria and polydipsia. Hormonal studies revealed almost normal anterior pituitary function and central diabetes insipidus. Magnetic resonance imaging showed thickening of the pituitary stalk and enlargement of the neurohypophysis without high intensity of the posterior lobe on T1-weighted images, which were compatible with lymphocytic infundibuloneurohypophysitis. Transsphenoidal biopsy was done and histological examination disclosed moderate fibrosis and lymphocytic infiltration not only in the posterior pituitary, but also in the adjacent anterior pituitary part of the gland. The lymphocytes both in the anterior and posterior pituitary were mainly T cells that were positive for UCHL 1, CD 3, and CD 8. Immunofluorescence of frozen tissue detected immunecomplex deposition in small vessels and the interstitium. These findings suggested that allergic reactions may play an important role in the pathogenesis of lymphocytic infundibuloneurohypophysitis.

CD3 Complex↗

PubMed2002

Toxic effect of single treatment with bromantane on neurological status of experimental animals.

Neurotoxicological profile of actoprotector bromantane was studied on rats using S. Irwin's protocol of multi-test observation. The drug in doses of 30-300 mg/kg stimulated and in doses of 600-9,600 mg/kg suppressed behavioral activity. Spontaneous motor activity increased after single treatment with bromantane in doses of 30-300 mg/kg, did not change after treatment in doses of 600 mg/kg, and was inhibited after treatment in doses above 600 mg/kg. In doses of 300-600 mg/kg the drug reduced pain sensitivity threshold and in doses above 600 mg/kg elevated the pain threshold and tactile sensitivity and reaction to knock. Bromantane induced mydriasis in all studied doses; in doses above 10 g/kg the preparation induced blepharoptosis. In doses above 5 g/kg bromantane slightly increased respiration rate and depth (Kussmaul-like respiration). In some animals bromantane in high doses induced regurgitation, diarrhea, and polyuria. Rectal temperature decreased by 0.5-1 degrees C after virtually all doses. Behavioral effects of bromantane in doses of 30 and 600 mg/kg were associated with stimulation of the central dopamine and suppression of muscarinic and nicotinic cholinergic structures, n-cholinolytic effects of bromantane was more pronounced at a dose of 30 mg/kg than at a dose of 600 mg/kg.

Amantadine↗

PubMed2004

Diabetes insipidus: historical aspects.

The contributions to our present knowledge and understanding of diabetes insipidus are briefly surveyed. Though a disease presenting with polyuria and thirst had been recognized since Antiquity, it was not until the 17. Century the distinction was made between diabetes insipidus and diabetes mellitus. At the beginning of the 20. Century almost nothing was known about the function of the pituitary. It was generally believed that diabetes insipidus was a renal disease. Two clinical observations in 1912 suggested an association between the hypophysis and diabetes insipidus. This view was supported by the recognition in 1913 that extract of the posterior lobe of the pituitary was effective in diabetes insipidus. Despite much evidence to the contrary, it was assumed that the antidiuretic hormone was produced in the intermediate lobe of the pituitary. Around 1950 it was finally established that 'the posterior lobe hormones' are in fact secreted in the hypothalamus. At the same time the antidiuretic hormone was isolated and synthesized. More recently, progress within genetics has made it possible to characterize in details other rare types of diabetes insipidus.

Diabetes Insipidus↗

PubMed2004

Clinical and pathological aspects of experimental oleander (Nerium oleander) toxicosis in sheep.

Dried Nerium oleander leaves at single lethal dose of 110 mg/kg body weight were administered orally to six native male sheep. Clinical signs of toxicosis in sheep began to appear about 30 min after receiving the oleander and included decrease of the heart rate followed by cardiac pauses and tachyarrhythmias; ruminal atony, mild to moderate tympany, abdominal pain, polyuria and polakiuria. Electrocardiography revealed bradycardia, atrio-ventricular blocks, depression of S-T segments, ventricular premature beats and tachycardia, and ventricular fibrillation. Five sheep died within 4-12 h and one survived. At necropsy there were varying degrees of haemorrhages in different organs and gastroenteritis. Histopathological examination of tissue sections revealed myocardial degeneration and necrosis, degeneration and focal necrosis of hepatocytes, necrosis of tubular epithelium in kidneys, oedema in the lungs, and ischemic changes in the cerebrum.

Animals↗

PubMed2002

[Pseudohypoadrenocorticism in two Siberian huskies with gastrointestinal parasitoses].

Two Siberian Huskies were presented because of anorexia, chronic diarrhoea, polydipsia and polyuria. The most significant clinical finding was a severe dehydration. Laboratory results showed severe electrolyte disturbances with sodium: potassium ratios of 13.2 and 15.4, respectively. Hypoadrenocorticism could be excluded in both dogs with an ACTH stimulation test. The plasma aldosterone concentrations before and after stimulation were very high, which might be due to a stimulation by the electrolyte disturbances. The final diagnosis was an infection with the whipworm Trichuris vulpis and secondary pseudohypoadrenocorticism. A life-threatening dehydration syndrome with hyponatremia, hyperkalemia and metabolic acidosis due to intestinal parasitism has been reported in earlier studies. Up till now sodium: potassium ratios less than 14, however, have only been seen in animals with Morbus Addison. The severe hyperkalemia in these dogs might be due to a particular sensitivity in this breed.

Adrenal Insufficiency↗

PubMed2006

[Leiomyoma of a kidney in a dog: a rare diagnosis].

A five-year-old, neutered male, Bernese Mountain Dog was referred because of a two-week history of hematuria and a one-week history of polydipsia and polyuria. Palpation of the dorsomedial mesogastrium revealed a mass on the left side, which was seen via radiography and ultrasonography to be attached to the left kidney. The left kidney and the attached mass were excised surgically, and the dog had an uneventful recovery. Histological examination of the mass revealed a dense proliferation of non-polymorphic spindle cells, which stained positive for smooth muscle actin via immunohistochemistry. Leiomyoma of the kidney was diagnosed. Postoperatively, there was mild azotaemia, which did not affect the general condition of the dog and was still present 15 months after surgery. Leiomyoma of the kidney is a benign tumor, and hematuria may be the only clinical sign.

Animals↗

PubMed2001

Varying clinical presentations at onset of type 1 diabetes mellitus in children--epidemiological evidence for different subtypes of the disease?

OBJECTIVE: On the basis of 2121 case observations between 1987 and 1997, we describe the clinical and laboratory characteristics of diabetes mellitus type 1 at its onset. Our objective is to analyze whether clinical presentation follows a uniform pattern or whether there is evidence for different subtypes. RESEARCH DESIGN AND METHODS: Thirty-one pediatric hospitals and one diabetes center in Baden-Wuerttemberg (BW), Germany, participated in this study. The hospital records of 2121 children below 15 yr of age were examined retrospectively. Statistical analysis was done after logarithmic transformation into a normal distribution. RESULTS: The average duration of symptoms was found to be 15.2 d (95% CI (Confidence Intervals) = 14.3-16.1) ranging between 2.0 and 180 d (95% central range). The most frequent symptoms were polyuria and polydipsia; 7.2% presented with altered level of consciousness. The mean blood glucose value was 407.9 mg/dL (95% CI = 400.0-416.0), corresponding to 23.3 mmol/L (95% CI = 22.8-23.8). The median pH value was 7.35 (95% CI = 7.34-7.36), and the median base excess was -5 mmol/L (95% CI =-5 to -4). The younger patients had a shorter duration of symptoms and suffered most frequently from ketoacidosis. CONCLUSIONS: Although the symptoms of diabetes at its onset follow a uniform pattern, the clinical presentation and duration of symptoms indicate that there may be various forms of type 1 diabetes.

Journal Article↗

PubMed2002

Secretion of natriuretic peptides caused by an epileptic attack.

OBJECTIVE: To describe clinical features of a patient with secretions of atrial natriuretic peptide (ANP) and brain natriuretic peptide (BNP) during an epileptic attack. PATIENT: A 65-year-old woman experienced frequent bouts of polyuria, pyrexia and general fatigue after several years of a cerebral contusion involving the left fronto-temporal lobe caused by a traffic accident. Her urine output and urinary sodium excretion increased, and plasma ANP and BNP concentrations were markedly high during each attack. Electroencephalography (EEG) showed serial seizure discharge in the left anterior temporal region during the attacks, indicative of epileptic focus. CONCLUSION: ANP and BNP secretions probably were triggered by epileptic stimulation on the diencephalon beyond the focus.

Aged↗

PubMed2002

Myelodysplastic syndrome in transformation to acute myeloid leukemia presenting with diabetes insipidus: due to pituitary infiltration association with abnormalities of chromosomes 3 and 7.

A 31-yr-old woman with myelodysplastic syndrome (MDS) in transformation to acute myeloid leukemia (AML) presented with initial symptoms of polyuria and polydipsia. Cytogenetics revealed monosomy 7 and translocation (3;3)(q21;q26). The initial symptoms, in conjunction with a low serum level of anti-diuretic hormone (ADH) and magnetic resonance imaging (MRI) findings demonstrating loss of the "bright spot" of the neurohypophysis, indicated diabetes insipidus (DI), e.g. caused by leukemic infiltration of the neurohypophysis. After induction chemotherapy the patient's bone marrow revealed blast persistence, and following a second course of chemotherapy and normalisation of MRI, an allogeneic peripheral blood stem cell transplantation (PBSCT) from the patient's HLA-identical brother was performed, resulting in ongoing complete remission. Recently, Lavabre-Bertrand et al. reported an association of AML with DI, elevated platelet counts, and monosomy 7 and chromosome 3 abnormalities in three patients (Eur. J. Haematol. 2001: 66: 66-69). Our report of an MDS with trilineage dysplasia and these karyotypic changes associated with DI indicates that this new entity may also include preleukemic cases.

Acute Disease↗

PubMed2003

Protective effects of glycyrrhizin on gentamicin-induced acute renal failure in rats.

The aim of this study was to investigate the effects of glycyrrhizin (200 mg/kg/day) on renal function in association with the regulation of aquaporin 2 water channel in rats with gentamicin (100 mg/kg/day)-induced acute renal failure. Polyuria in rats with gentamicin-induced acute renal failure was associated with down-regulation of renal aquaporin 2 in the inner and outer renal medulla, and cortex. Glycyrrhizin administration restored the expression of aquaporin 2 with paralleled changes in urine output. Changes in renal functional parameters, such as creatinine clearance, urinary osmolality, and solute-free reabsorption, accompanying acute renal failure were also partially restored after administration of glycyrrhizin. Histological changes in rats with gentamicin-induced acute renal failure were also abrogated by glycyrrhizin treatment. The above results suggest that glycyrrhizin treatment could ameliorate renal defects in rats with acute renal failure induced by gentamicin.

Acute Kidney Injury↗

PubMed1979

Captopril (SQ 14225) depresses drinking and aldosterone in rats lacking vasopressin.

Angiotensin II is dipsogenic, and vasopressin (ADH) regulates renal water excretion. Together, these hormones govern overall mammalian water balance. The Brattleboro rat with inherited diabetes insipidus (DI) lacks ADH and is therefore a convenient model with which to elucidate mechanisms regulating water metabolism. In the present studies, angiotensin II has also been removed from DI rats by the administration of an inhibitor (captopril, SQ 14225; D-2-methyl-3-mercaptopropanoyl-L-proline) of the enzyme which converts angiotensin I, the relatively inert component of the renin-angiotensin system, to angiotensin II, the biologically active substance. SQ 14225 reduced the drinking rates, and after 6 days lowered peripheral plasma aldosterone concentrations were associated with hyperkalaemia. We conclude that the polydipsia of diabetes insipidus partly results from elevated plasma renin activities and angiotensin II concentrations seen in this syndrome. Further, the apparent hypoaldosteronism of DI Brattleboro rats reflects differences in both tissue usage of the steroid and adrenocortical sensitivities associated with polyuria, hyperosmolarity and possibly potassium wasting.

Adrenal Cortex Hormones↗

PubMed1998

Diuretic response to cyclophosphamide in rats bearing a matrix metalloproteinase-9-producing tumour.

When cyclophosphamide (CY) (100-120 mg kg(-1)) was administered intravenously (i.v.) to normal F-344 rats, oliguria occurred over the 5-day observation period. Conversely, in rats bearing matrix metalloproteinase-9 (MMP-9) producing 13762NF mammary adenocarcinoma (MTLn3 clone), polyuria occurred chiefly during the first 24 h after CY treatment. In parallel with urine volume, a decrease in the urinary excretion of N-acetyl-beta-D-glucosaminidase (NAG) was observed during the first 5 days after CY treatment in normal rats, but it increased in MTLn3-bearing rats. No elevation in blood urea nitrogen (BUN) or serum creatinine (Cr) values was observed for either group. Both urine volume and urinary excretion of NAG after CY treatment were lower in rats bearing the MTC clone (lower production of MMP-9) than for those bearing the MTLn3 clone. In the case of treatment with cisplatin (CDDP, 4-6 mg kg(-1)), urine volume, urinary NAG excretion and BUN and serum Cr values all increased in normal rats and were all found to be higher in MTLn3-bearing rats than in normal rats. The diuretic response to these drugs in tumour-bearing (TB) rats may be associated with MMP-9 produced by the tumour cells. This report suggests that the nephrotoxicity due to anti-cancer drugs may change when the drugs are used for the treatment of patients bearing a MMP-9-producing tumour.

Adenocarcinoma↗

PubMed1978

Nephron function of the transplanted rat kidney.

Tubular function in the early phase (one to three hours) after transplantation of rat kidneys was analyzed with respect to glomerular filtration, vascular and tubular pressures, and excretory variables. Kidneys exposed to a short period of cold ischemia (two hours) functioned almost normally, except for a polyuria. After 12 and 16 hr of cold ischemia, nephron heterogeneity appeared with 1) "normal" tubules, 2) dilated tubules, and 3) collapsed tubules. In the "normal" tubules, the pressure was increased to 20 mm Hg, and the filtration was reduced in proportion to the mean net driving force. The dilated tubules had no filtration due to a more or less complete tubular obstruction, probably located in the thin loop of Henle and in the collecting ducts. The collapsed tubules had no filtration due to glomerular ischemia, which in turn might be the consequence of afferent arteriolar constriction. The total GFR was greatly reduced since only the "normal" tubules contributed to the total filtration. Concentrating ability and potassium secretion were also impaired. We interpreted this impairment as being due to medullary dysfunction, which would explain the isosthenuria and the impaired potassium transport.

Animals↗

PubMed1986

Acute renal failure in hemorrhagic hypotension: cellular energetics and renal function.

In male Wistar rats, renal adenosine triphosphate (ATP), inorganic phosphate (Pi) and intracellular pH were measured by 31phosphorus nuclear magnetic resonance (31P NMR) and correlated with renal function before, during, and for one hour after a period of 30 to 40 minutes hemorrhagic hypotension. In animals which suffered no change in these metabolites during hypotension, retransfusion immediately restored normal renal function. When metabolite changes were observed during hypotension, they occurred suddenly with severe ATP depletion, Pi accumulation, and intracellular acidosis occurring almost concurrently. Metabolic changes of this magnitude were always associated with renal dysfunction in the post-hypotensive period, which occurred even when the period of biochemical change was only 10 to 15 minutes. The abnormalities in post-hypotensive renal function resemble the pattern of change seen in human acute tubular necrosis (ATN): depressed glomerular filtration rate (GFR), urine output varying from polyuria to oliguria, decreased urine to plasma inulin ratio, increased urinary sodium concentration, increased fractional excretion of sodium, and increased fractional excretion of potassium. It is postulated that changes in renal cellular energy status during hemorrhagic hypotension distinguish pre-renal failure from early or incipient ATN.

Acute Kidney Injury↗