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Hormone Research in Paediatrics May 2024Stimulated copeptin may provide an alternative to water deprivation testing (WDT) in the evaluation of polyuria-polydipsia syndrome (PPS). Though best studied, arginine...
INTRODUCTION
Stimulated copeptin may provide an alternative to water deprivation testing (WDT) in the evaluation of polyuria-polydipsia syndrome (PPS). Though best studied, arginine stimulation alone produces a modest copeptin response in children. We investigated the effectiveness of the arginine + LevoDopa/Carbidopa stimulation test (ALD-ST) for copeptin.
METHODS
47 healthy short children (controls), 10 children with primary polydipsia, and 10 children with AVP deficiency received arginine hydrochloride (500 mg/kg intravenously over 30 min) and Levodopa/carbidopa (10:1 ratio; 175 mg of
l -Dopa/m2 BSA) orally. Serum copeptin was measured at 0, 60, 90, and 120 min.RESULTS
In controls, ALD-ST increased copeptin from a median of 7.0 pmol/L (IQR 5.0-10.0) to a peak of 44.0 pmol/L (IQR 21.4-181.0) between 60 and 120 min (p < 0.001). Copeptin peak was higher in subjects who experienced nausea or vomiting (57%) than in those who did not (131.0 pmol/L [IQR 42.5-193.8] vs. 22.7 pmol/L [IQR 16.0-33.7], p < 0.001). While subjects with primary polydipsia had similar baseline (8.5 pmol/L [IQR 8.0-11.0]) and stimulated (125.2 pmol/L [IQR 87.6-174.0]) copeptin levels as controls, subjects with AVP deficiency had lower baseline (2.5 pmol/L [IQR 2.0-3.1]) and peak levels (4.6 pmol/L [IQR 2.4-6.0]). A peak copeptin of ≥9.3 pmol/L best predicted absence of complete or partial AVP deficiency with a sensitivity of 100% and specificity of 80%.
CONCLUSIONS
ALD-ST induced a robust peak copeptin in healthy short children and children with primary polydipsia. Nausea/vomiting, a side effect of ALD-ST, amplified the copeptin response. The ALD-ST may be a suitable initial screening test in children with PPS.
PubMed: 38705144
DOI: 10.1159/000539208 -
Annals of Medicine and Surgery (2012) May 2024Teucrium polium is one of the aromatic plants that grows in the Mediterranean region, and had been used as an herbal treatment for diabetes due to its hypoglycaemia...
INTRODUCTION AND IMPORTANCE
Teucrium polium is one of the aromatic plants that grows in the Mediterranean region, and had been used as an herbal treatment for diabetes due to its hypoglycaemia effect. Although this plant is being studied now for its therapeutic role, its side effects are not taken enough into consideration, so this unique case can shed the light on serious toxic effects of this plant.
CASE PRESENTATION
A 68-year-old woman presented to the hospital with generalized fatigue, malaise, nausea, vomiting, abdominal pain, polydipsia, polyuria, breathlessness, and no defecation for 2 days after drinking big amounts of teucrium polium. The diagnosis was diabetic ketoacidosis (DKA), complete heart block, acute liver and kidney damage, and urinary tract infection (UTI). The patient was admitted to the ICU and treated for the DKA with an insulin pump, an antibiotic treatment for UTI, in addition to a dopamine pump and atropine, and then a temporary pacemaker was placed. The patient's DKA, liver and kidney damage were improved on day 9, heart rate returned normal and she was discharged to insert a permanent pacemaker. However, the patient passed away at the end.
CLINICAL DISCUSSION
Most studies made on this plant focused on the hypoglycaemia effect, with no attention to its toxic effects, so only few studies showed that teucrium polium can cause hepatic, renal toxicity and hyperglycaemia and most of them were studied in animals. While cardiac toxicity has never been noticed before.
CONCLUSION
For this reason, herbal treatment should be used with caution to avoid catastrophic side effects.
PubMed: 38694375
DOI: 10.1097/MS9.0000000000001760 -
Nature Reviews. Endocrinology May 2024Polyuria-polydipsia syndrome can be caused by central diabetes insipidus, nephrogenic diabetes insipidus or primary polydipsia. To avoid confusion with diabetes... (Review)
Review
Polyuria-polydipsia syndrome can be caused by central diabetes insipidus, nephrogenic diabetes insipidus or primary polydipsia. To avoid confusion with diabetes mellitus, the name 'central diabetes insipidus' was changed in 2022 to arginine vasopressin (AVP) deficiency and 'nephrogenic diabetes insipidus' was renamed as AVP resistance. To differentiate the three entities, various osmotic and non-osmotic copeptin-based stimulation tests have been introduced in the past decade. The hypertonic saline test plus plasma copeptin measurement emerged as the test with highest diagnostic accuracy, replacing the water deprivation test as the gold standard in differential diagnosis of the polyuria-polydipsia syndrome. The mainstay of treatment for AVP deficiency is AVP replacement with desmopressin, a synthetic analogue of AVP specific for AVP receptor 2 (AVPR2), which usually leads to rapid improvements in polyuria and polydipsia. The main adverse effect of desmopressin is dilutional hyponatraemia, which can be reduced by regularly performing the so-called desmopressin escape method. Evidence from the past few years suggests an additional oxytocin deficiency in patients with AVP deficiency. This potential deficiency should be further evaluated in future studies, including feasible provocation tests for clinical practice and interventional trials with oxytocin substitution.
PubMed: 38693275
DOI: 10.1038/s41574-024-00985-x -
Revista Medica de Chile May 2023Langerhans-cells Histiocytosis (LCH) is a rare disease characterized by the clonal proliferation of immature dendritic cells that locally or diffusely infiltrate...
Langerhans-cells Histiocytosis (LCH) is a rare disease characterized by the clonal proliferation of immature dendritic cells that locally or diffusely infiltrate different systems, mainly affecting children. We present the case of a 38-year-old man with a long-standing history of multiple tooth loss without association with trauma, smoking, or poor dental hygiene. Skull computed tomography (CT) showed multiple lytic jaw lesions. Jaw biopsy and immunohistochemical results were compatible with the finding of LCH. The patient evolved with polyuria, polydipsia, and nocturia, confirming the presence of diabetes insipidus and panhypopituitarism through hormonal studies. Magnetic resonance imaging (MRI) of the brain shows a hypothalamic neoplasm with infundibular involvement and an image suggestive of a pituitary granuloma. The treatment consisted of maxillobuccofacial defocusing, hormonal supplementation, chemotherapy, and radiotherapy with favorable evolution.
Topics: Humans; Male; Hypopituitarism; Adult; Histiocytosis, Langerhans-Cell; Magnetic Resonance Imaging; Tomography, X-Ray Computed
PubMed: 38687548
DOI: 10.4067/s0034-98872023000500659 -
Journal of Avian Medicine and Surgery Apr 2024Diabetes mellitus (DM) is an uncommon, poorly documented metabolic disorder of birds. Extrapolating knowledge from DM in mammals is challenging because of marked... (Review)
Review
Diabetes mellitus (DM) is an uncommon, poorly documented metabolic disorder of birds. Extrapolating knowledge from DM in mammals is challenging because of marked differences in avian physiology and metabolism. A literature review from December 1991 to January 2022 identified 14 publications covering 16 diabetic birds, 63% (10/16) of which belonged to the order Psittaciformes with as the predominant genus. No sex predilection was noted, but males generally presented at a younger age. Commonly reported clinical signs included polyuria 94% (15/16), polydipsia 88% (14/16), weight loss 75% (12/16), lethargy 63% (10/16), and polyphagia 38% (6/16). Diagnosis of DM was based on the presence of clinical signs and persistent hyperglycemia 100% (16/16), often with glucosuria 93% (13/14), response to insulin therapy 80% (8/10), and pancreatic pathology 90% (9/10). Specific treatment for DM was initiated in 14 patients, but blood glucose regulation for 6 months or longer was only achieved in 6 birds. Five of the regulated birds were managed with injectable long-acting insulin and 1 with oral glipizide combined with dietary modifications. However, glipizide yielded poor results in other cases, likely attributable to a lack of functional beta cells. Three diabetic birds progressed to remission. Treatment proved unsuccessful for 7 patients with a mean survival time of 36 days from diagnosis. One patient was lost to follow-up, and 2 were euthanized immediately following diagnosis. Histological examination of the pancreas frequently (90%, 9/10) revealed abnormalities including atrophy, fibrosis, and vacuolization of the endocrine islets with or without lymphoplasmacytic pancreatitis. Comorbidities, including hemosiderosis and infection, were common. This review suggests that birds diagnosed with DM are primarily affected by a type I diabetes as observed in dogs and humans. In contrast to mammalian species, avian DM is often associated with underlying disease and a complete clinical workup is essential to diagnose and address secondary disease conditions prior to initiating long-term insulin therapy.
Topics: Animals; Bird Diseases; Birds; Diabetes Mellitus
PubMed: 38686885
DOI: 10.1029/AVIANMS-D-22-00057 -
Journal of the American Veterinary... Apr 2024To describe a novel presentation of paraneoplastic hypercalcemia caused by a canine salivary carcinoma.
OBJECTIVE
To describe a novel presentation of paraneoplastic hypercalcemia caused by a canine salivary carcinoma.
ANIMAL
A 6-year-old intact male Husky with hypercalcemia and a spontaneous salivary carcinoma, stage III.
CLINICAL PRESENTATION, PROGRESSION, AND PROCEDURES
The dog presented with polyuria, polydipsia, and hypercalcemia. Physical examination revealed a 37 X 43-mm firm mass in the ventrolateral aspect of the right-hand side of the neck, caudal to the temporomandibular joint. Incisional biopsy was suspicious of metastatic carcinoma to the mandibular lymph node. A full-body CT scan found a large, heterogenous, contrast-enhancing mass on the right ventrolateral neck that appeared to be originating from either the mandibular lymph node or right mandibular salivary gland. Parathyroid hormone-related protein was considered within normal reference intervals, and both parathyroid glands appeared ultrasonographically normal.
TREATMENT AND OUTCOME
The patient was treated with a marginal surgical excision of the mass, without immediate complications. Histopathology confirmed the presence of a salivary carcinoma with narrow margins of excision and invasion of the mandibular lymph node. Twenty-four hours after surgery, ionized calcium returned to normal reference values and clinical signs completely resolved.
CLINICAL RELEVANCE
Hypercalcemia is an urgent pathology with important systemic implications requiring prompt diagnosis and intervention. In this case report, we identify the first salivary carcinoma associated with a paraneoplastic hypercalcemia, including this pathology as a new differential diagnosis. The hypercalcemia resolved with marginal surgical excision, but interestingly the parathyroid hormone-related protein was not overexpressed, meaning that this neoplasia could mediate hypercalcemia by another pathophysiological mechanism.
PubMed: 38663442
DOI: 10.2460/javma.24.01.0058 -
Journal of Cellular and Molecular... Apr 2024X-linked nephrogenic diabetes insipidus (X-NDI) is a rare congenital disease caused by inactivating mutations of the vasopressin type-2 receptor (AVPR2), characterized...
X-linked nephrogenic diabetes insipidus (X-NDI) is a rare congenital disease caused by inactivating mutations of the vasopressin type-2 receptor (AVPR2), characterized by impaired renal concentrating ability, dramatic polyuria, polydipsia and risk of dehydration. The disease, which still lacks a cure, could benefit from the pharmacologic stimulation of other GPCRs, activating the cAMP-intracellular pathway in the kidney cells expressing the AVPR2. On the basis of our previous studies, we here hypothesized that the β3-adrenergic receptor could be such an ideal candidate. We evaluated the effect of continuous 24 h stimulation of the β3-AR with the agonist BRL37344 and assessed the effects on urine output, urine osmolarity, water intake and the abundance and activation of the key renal water and electrolyte transporters, in the mouse model of X-NDI. Here we demonstrate that the β3-AR agonism exhibits a potent antidiuretic effect. The strong improvement in symptoms of X-NDI produced by a single i.p. injection of BRL37344 (1 mg/kg) was limited to 3 h but repeated administrations in the 24 h, mimicking the effect of a slow-release preparation, promoted a sustained antidiuretic effect, reducing the 24 h urine output by 27%, increasing urine osmolarity by 25% and reducing the water intake by 20%. At the molecular level, we show that BRL37344 acted by increasing the phosphorylation of NKCC2, NCC and AQP2 in the renal cell membrane, thereby increasing electrolytes and water reabsorption in the kidney tubule of X-NDI mice. Taken together, these data suggest that human β3-AR agonists might represent an effective possible treatment strategy for X-NDI.
Topics: Male; Animals; Mice, Inbred C57BL; Disease Models, Animal; Adrenergic beta-3 Receptor Agonists; Antidiuretic Agents; Kidney Concentrating Ability; Polydipsia
PubMed: 38652212
DOI: 10.1111/jcmm.18301 -
Medizinische Klinik, Intensivmedizin... Apr 2024Max Reger was an organist, university teacher and composer whose life, illnesses, death and dying are not or hardly known to many.
BACKGROUND
Max Reger was an organist, university teacher and composer whose life, illnesses, death and dying are not or hardly known to many.
OBJECTIVES
Which illnesses determined Reger's life and did his lifestyle and illnesses influence his compositional work? Could his early death have been avoided? From today's point of view, could modern intensive care medicine have helped him?
MATERIAL AND METHODS
A detailed analysis of Reger's diseases was performed using scientific databases (medline, pubmed). All published articles were evaluated and examined in detail.
RESULTS
Max Reger was born in Brand in 1873 and received early lessons in violin, piano and organ playing. From 1890 he studied at the conservatory in Sondershausen, later at the conservatory in Wiesbaden. In 1901 he moved to Munich, and in 1907 to Leipzig, where he became university director and professor at the conservatory. Four years later he took over the court chapel in Meiningen, but ended this activity again in 1914. A year later he moved to Jena and wrote his late works in the "Jenaish style". Reger suffered from many illnesses, especially bipolar disorder with manic and depressive phases. He had metabolic syndrome with arterial hypertension, was overweight and smoked incredibly heavily. Overeating ("binge eating" syndrome) and polydipsia were other prominent findings. Reger's life was characterized by alcohol abuse, often aggravated by professional and/or human crises. In 1916 Reger died suddenly and unexpectedly in Leipzig of cardiovascular failure.
DISCUSSION
Reger was an outstanding personality who left behind an extensive oeuvre. Among the highlights of Max Reger's oeuvre are his chorale fantasies such as on "Ein' feste Burg ist unser Gott" (op. 27) or also the "Fantasia and Fugue on B A C H" (op. 46), but other compositions such as the Mozart Variations (op. 132) and the Clarinet Quintet (op. 146) are also world-famous. His lifestyle certainly favored coronary heart disease, the consequences of which caused Reger's sudden, unexpected and much too early death. Today's modern intensive care medicine could probably have prolonged his life.
PubMed: 38652144
DOI: 10.1007/s00063-024-01144-w -
Case Reports in Endocrinology 2024Gestational diabetes insipidus (DI) is a very rare complication of pregnancy. We present a case of gestational DI combining two different types of DI. . A 39-year-old...
BACKGROUND
Gestational diabetes insipidus (DI) is a very rare complication of pregnancy. We present a case of gestational DI combining two different types of DI. . A 39-year-old pregnant woman suddenly presented with thirst, polydipsia, and polyuria after 31 gestation weeks (GWs). Based on laboratory findings of hypotonic urine (78 mOsm/kgHO) with higher plasma osmolality (298 mOsm/kgHO) and higher serum sodium levels (149 mEq/L), gestational DI was suspected, and the clinical course was monitored without therapy until the results of a measurement of plasma arginine vasopressin (AVP) levels were available. However, she subsequently developed acute prerenal failure and underwent an emergency cesarean section at 34 GWs. Her resected placenta weighed 920 g, nearly twice the normal weight. Immediately following delivery, intranasal 1-desamino-8-D-arginine vasopressin was administered, and her symptoms promptly disappeared. Afterward, her predelivery plasma AVP level was found to have been inappropriately low (0.7 pg/mL) given her serum sodium level. The patient's serum vasopressinase level just before delivery was 2,855 ng/mL, more than 1,000 times the upper limit of the normal range, suggesting excess vasopressinase-induced DI. The presence of anti-rabphilin-3A antibodies in the patient's blood, a hypertonic saline infusion test result, and loss of the high-intensity signal of the posterior pituitary on fat-suppressed T1-weighted magnetic resonance images without thickening of the stalk and enlargement of the neurohypophysis suggested concurrent central DI-like lymphocytic infundibulo-neurohypophysitis (LINH).
CONCLUSION
In addition to the degradation of AVP by excess placental vasopressinase due to the enlarged placenta, an insufficient compensatory increase in AVP secretion from the posterior pituitary gland due to LINH-like pathogenesis might have led to DI symptoms.
PubMed: 38646198
DOI: 10.1155/2024/8687054 -
Archives de Pediatrie : Organe Officiel... May 2024Adrenal insufficiency (AI) is one of the most life-threatening disorders resulting from adrenal cortex dysfunction. Symptoms and signs of AI are often nonspecific, and...
Adrenal insufficiency (AI) is one of the most life-threatening disorders resulting from adrenal cortex dysfunction. Symptoms and signs of AI are often nonspecific, and the diagnosis can be missed and lead to the development of AI with severe hypotension and hypovolemic shock. We report the case of a 13-year-old child admitted for cardiac arrest following severe hypovolemic shock. The patient initially presented with isolated mild abdominal pain and vomiting together with unexplained hyponatremia. He was discharged after an initial short hospitalization with rehydration but with persistent hyponatremia. After discharge, he had persistent refractory vomiting, finally leading to severe dehydration and extreme asthenia. He was admitted to pediatric intensive care after prolonged hypovolemic cardiac arrest with severe anoxic encephalopathy leading to brain death. After re-interviewing, the child's parents reported that he had experienced polydipsia, a pronounced taste for salt with excessive consumption of pickles lasting for months, and a darkened skin since their last vacation 6 months earlier. A diagnosis of autoimmune Addison's disease was made. Primary AI is a rare life-threatening disease that can lead to hypovolemic shock. The clinical symptoms and laboratory findings are nonspecific, and the diagnosis should be suspected in the presence of unexplained collapse, hypotension, vomiting, or diarrhea, especially in the case of hyponatremia.
Topics: Humans; Adolescent; Male; Addison Disease; Shock; Hyponatremia; Adrenal Insufficiency; Heart Arrest
PubMed: 38644058
DOI: 10.1016/j.arcped.2024.02.005