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JCEM Case Reports Jan 2024Insulin edema is a poorly understood complication of insulin therapy. It has been reported in patients with both type 1 and 2 diabetes mellitus and typically occurs in...
Insulin edema is a poorly understood complication of insulin therapy. It has been reported in patients with both type 1 and 2 diabetes mellitus and typically occurs in patients with newly diagnosed or poorly controlled diabetes mellitus either after initiation or intensification of insulin therapy. A 20-year-old man presented with anorexia, polydipsia, and weight loss. Serum glucose on admission was 824 mg/dL (45.8 mmol/L) and hemoglobin A1c was >14.0. Additional workup was notable for positive anti-IA2 antibodies and low C-peptide of 0.5 ng/mL (1.1-4.4 ng/mL). He was diagnosed with type 1 diabetes mellitus and was started on insulin therapy with glargine and lispro. Within 4 days after insulin initiation, he developed bilateral leg swelling and reported a 25-pound (11.3-kg) weight gain over the next 10 days. After excluding other systemic causes of edema such as heart failure, renal failure, and liver failure, a diagnosis of insulin edema was made. Insulin glargine was switched to insulin degludec. Complete resolution of edema occurred within 3 days of switching the insulins. Insulin edema is a diagnosis of exclusion. Insulin's role in renal sodium handling, vasodilation, and increased vascular permeability have been postulated as possible mechanisms. Clinicians should be aware of this rare complication.
PubMed: 38116161
DOI: 10.1210/jcemcr/luad158 -
International Journal of Surgery Case... Jan 2024Tuberculosis of the central nervous system is unusual and accounts for 1 % of all cases of tuberculosis in the world. The pituitary location is even scarcer.
INTRODUCTION AND IMPORTANCE
Tuberculosis of the central nervous system is unusual and accounts for 1 % of all cases of tuberculosis in the world. The pituitary location is even scarcer.
CASE PRESENTATION
A 14-year-old girl presented with polyuria-polydipsia syndrome and menstrual irregularity. MRI showed an intrasellar lesion of the pituitary gland. She underwent transsphenoidal surgery for histopathological diagnosis and removal of the lesion. Histological findings were consistent with a tuberculoma. She was put on anti-tuberculosis drugs and is being followed up.
CLINICAL DISCUSSION
In endemic areas, pituitary tuberculosis should be considered in the differential diagnosis of pituitary tumors. The histological examination will guide the diagnosis. Sometimes, other complementary examinations such as the tuberculin skin test can be of great help when the histology is not conclusive. Medical treatment can be curative, however, surgery can be necessary for decompression.
CONCLUSION
In addition to being the first case of histologically proven primary pituitary tuberculosis in a child reported in Morocco, the present case is unique in the way that the extensive radiological examinations did not reveal any evidence of other systemic or pulmonary tuberculosis.
PubMed: 38113568
DOI: 10.1016/j.ijscr.2023.109144 -
Zhejiang Da Xue Xue Bao. Yi Xue Ban =... Dec 2023A 2-year-old boy was admitted to Shanghai Children's Medical Center, Shanghai Jiao Tong University School of Medicine in Nov 30, 2018, due to polydipsia, polyphagia,...
A 2-year-old boy was admitted to Shanghai Children's Medical Center, Shanghai Jiao Tong University School of Medicine in Nov 30, 2018, due to polydipsia, polyphagia, polyuria accompanied with increased glucose levels for more than 2 weeks. He presented with symmetrical short stature [height 81 cm (-2.2 SD), weight 9.8 kg (-2.1 SD), body mass index 14.94 kg/m (-)], and with no special facial or physical features. Laboratory results showed that the glycated hemoglobin A1c was 14%, the fasting C-peptide was 0.3 ng/mL, and the islet autoantibodies were all negative. Oral glucose tolerance test showed significant increases in both fasting and postprandial glucose, but partial islet functions remained (post-load C-peptide increased 1.43 times compared to baseline). A heterozygous variant c.1366C>T (p.R456C) was detected in gene, thereby the boy was diagnosed with a specific type of diabetes mellitus. The boy had congenital heart disease and suffered from transient hyperosmolar hyperglycemia after a patent ductus arteriosus surgery at 11 months of age. Insulin replacement therapy was prescribed, but without regular follow-up thereafter. The latest follow-up was about 3.5 years after the diagnosis of diabetes when the child was 5 years and 11 months old, with the fasting blood glucose of 6.0-10.0 mmol/L, and the 2 h postprandial glucose of 17.0-20.0 mmol/L.
Topics: Male; Child; Humans; Child, Preschool; Infant; Diabetes Mellitus, Type 2; Mutation, Missense; C-Peptide; China; Insulin; Glucose; Blood Glucose; GATA6 Transcription Factor
PubMed: 38105674
DOI: 10.3724/zdxbyxb-2023-0351 -
BMC Endocrine Disorders Dec 2023The nucleoside transport capabilities of the human equilibrative nucleoside transporter-3 (hENT3) are disrupted by mutations in SLC29A3 (10q22.2), which are genes for... (Review)
Review
BACKGROUND
The nucleoside transport capabilities of the human equilibrative nucleoside transporter-3 (hENT3) are disrupted by mutations in SLC29A3 (10q22.2), which are genes for the nucleoside transporter and are the cause of the unusual autosomal recessive disease known as H syndrome. As a result, histiocytic cells invade a number of organs.
CASE PRESENTATION
A 17-year-old Syrian male was admitted to the internal medicine department with a one-month history of polyuria, polydipsia, general weakness, and pallor. He had a history of progressive bilateral sensorineural hearing loss and failure to gain weight for three years. Physical examination revealed various abnormalities, including scrotal mass, small penis and testicles, absence of pubic and axillary hair, joint abnormalities, short stature, hallux valgus, fibrous protrusion near the navel, and hyperpigmented non-itchy painful skin plaques. Clinical signs along with laboratory test results confirmed hyperglycemia, primary hypogonadism, osteopenia, and growth hormone deficiency. After a review of the relevant medical literature, this patient's presentation of hyperglycemia with hypogonadism, hyperpigmentation, hallux valgus, hearing loss, hematological abnormalities, and short stature suggested the diagnosis of H syndrome. The patient received treatment with insulin and testosterone, leading to a significant improvement in his presenting symptoms.
CONCLUSIONS
H syndrome is a very rare condition, and the fact that the first case has only recently been reported in Syria serves to emphasize how rare it is. H Syndrome should be suspected if a patient has short stature with signs of hyperglycemia and other endocrine and cutaneous abnormalities. We are reporting this case to increase physicians' awareness of this exceedingly rare and unique syndrome.
Topics: Humans; Male; Adolescent; Syria; Hallux Valgus; Hyperpigmentation; Hearing Loss, Sensorineural; Dwarfism; Hypogonadism; Nucleoside Transport Proteins; Hyperglycemia; Growth Hormone
PubMed: 38093297
DOI: 10.1186/s12902-023-01525-w -
Reviews in Endocrine & Metabolic... Jun 2024Arginine vasopressin deficiency (AVP-D) is one of the main entities of the polyuria-polydipsia syndrome. Its correct diagnosis and differentiation from the other two... (Review)
Review
Arginine vasopressin deficiency (AVP-D) is one of the main entities of the polyuria-polydipsia syndrome. Its correct diagnosis and differentiation from the other two causes - AVP resistance and primary polydipsia - is crucial as this determines the further management of these patients.Over the last years, several new diagnostic tests using copeptin, the stable surrogate marker of AVP, have been introduced. Among them, hypertonic saline stimulated copeptin was confirmed to reliably and safely improve the diagnostic accuracy to diagnose AVP-D. Due to its simplicity, arginine stimulated copeptin was put forward as alternative test procedure. Glucagon-stimulated copeptin also showed promising results, while the oral growth hormone secretagogue Macimorelin failed to provide a sufficient stimulus. Interestingly, an approach using machine learning techniques also showed promising results concerning diagnostic accuracy.Once AVP-D is diagnosed, further workup is needed to evaluate its etiology. This will partly define the further treatment and management. In general, treatment of AVP-D focuses on desmopressin substitution, with oral formulations currently showing the best tolerance and safety profile. However, in addition to desmopressin substitution, recent data also showed that psychopathological factors play an important role in managing AVP-D patients.
Topics: Humans; Arginine Vasopressin; Polyuria; Polydipsia; Deamino Arginine Vasopressin; Glycopeptides
PubMed: 38087160
DOI: 10.1007/s11154-023-09862-w -
Peptides Jan 2024The orexin (hypocretin) neuropeptide system is an important regulator of ingestive behaviors, i.e., it promotes food and water intake. Here, we investigated the role of...
The orexin (hypocretin) neuropeptide system is an important regulator of ingestive behaviors, i.e., it promotes food and water intake. Here, we investigated the role of orexin in drinking induced by the potent dipsogen angiotensin II (ANG II). Specifically, male and female orexin-deficient mice received intracerebroventricular (ICV) injections of ANG II, followed by measuring their water intake within 15 min. We found that lower doses of ANG II (100 ng) significantly stimulated drinking in males but not in females, indicating a general sex-dependent effect that was not affected by orexin deficiency. However, higher doses of ANG II (500 ng) were sufficient to induce drinking in female wild-type mice, while female orexin-deficient mice still did not respond to the dipsogenic properties of ANG II. In conclusion, these results suggest sex-dependent effects in ANG II-induced drinking and further support the sexual dimorphism of orexin system functions.
Topics: Mice; Animals; Male; Female; Orexins; Angiotensin II; Drinking; Neuropeptides; Feeding Behavior; Injections, Intraventricular
PubMed: 38043589
DOI: 10.1016/j.peptides.2023.171127 -
Cureus Oct 2023We present a case of a 10-month-old male infant who initially presented with polyuria, polydipsia, drowsiness, and fever. Neuroimaging using non-contrast computed...
We present a case of a 10-month-old male infant who initially presented with polyuria, polydipsia, drowsiness, and fever. Neuroimaging using non-contrast computed tomography (NCCT) demonstrated obstructive hydrocephalus associated with a suprasellar mass, for which emergency neurosurgical intervention was performed with right parietal medium pressure ventriculoperitoneal (MPVP) shunting. For fever, no cause was found with sterile cerebrospinal fluid (CSF) analysis, and empirical antibiotics were administered. The patient exhibited polyuria with hypernatremia and was diagnosed with arginine vasopressin (AVP) deficiency, further complicated by visual impairment due to left optic atrophy. Hormonal workup revealed secondary hypothyroidism and hypocortisolism. Imaging by contrast-enhanced magnetic resonance imaging (CEMR) revealed a lobulated solid-cystic suprasellar mass with flow void, suggestive of adamantinomatous craniopharyngioma initially. However, despite multiple neurosurgical interventions, the patient's condition deteriorated with recurrent fever and seizures, leading to a revision of ventriculoperitoneal shunts. Repeat CEMR showed an increase in the size of the lesion with spinal leptomeningeal metastasis, suggesting a different pathology. Transventricular biopsy confirmed an atypical teratoid and rhabdoid tumor (AT/RT), World Health Organization Classification of Tumors of the Central Nervous System (CNS WHO) grade 4, characterized by diffuse growth pattern, moderate nuclear pleomorphism, clear cytoplasm, and prominent nucleoli. Immunohistochemistry revealed positive vimentin staining and loss of integrase interactor 1 (INI1) expression, consistent with AT/RT. The patient's parents were counseled on the need for multimodal management, including surgery and chemotherapy. However, due to socioeconomic constraints and a guarded prognosis, they chose to leave against medical advice. This case illustrates the diagnostic challenges in distinguishing AT/RT from other suprasellar masses and emphasizes the importance of a multidisciplinary approach in managing complex pediatric cases.
PubMed: 38022150
DOI: 10.7759/cureus.47632 -
Cureus Oct 2023Psychogenic polydipsia occurs during water or fluid intoxication and can lead to electrolyte disturbances, such as hyponatremia. Hyponatremia can give rise to signs and...
Psychogenic polydipsia occurs during water or fluid intoxication and can lead to electrolyte disturbances, such as hyponatremia. Hyponatremia can give rise to signs and symptoms, including lethargy, psychosis, seizures, or death. Psychogenic, or primary polydipsia, can be compared to other medical conditions that cause excessive thirst. This case report will focus on the symptoms, disease, and treatment involved in the care and hospitalization of a 30-year-old male patient who reported ingesting up to 40 liters of water a day for the last three years. This patient with psychogenic polydipsia, chronic schizophrenia, and active psychosis was diagnosed with metabolic encephalopathy secondary to severe hyponatremia (day one sodium level: 108 mEq/L). The management goal was to stabilize electrolytes and increase sodium levels without causing osmotic demyelination syndrome. During subsequent hospitalization, the psychiatry team worked towards the normalization of sodium levels and managed behavioral patterns contributing to water consumption. The patient achieved a normal sodium level on day 21 of inpatient psychiatric treatment with the following medication regimen: acetazolamide, candesartan, olanzapine, sodium chloride, and trazodone.
PubMed: 38021912
DOI: 10.7759/cureus.47719 -
Medicina (Kaunas, Lithuania) Oct 2023There is growing interest in alternative therapies for type 2 diabetes mellitus (T2DM) because some patients refuse to receive conventional therapies. In East Asia,...
There is growing interest in alternative therapies for type 2 diabetes mellitus (T2DM) because some patients refuse to receive conventional therapies. In East Asia, herbal medicines are often used to treat T2DM, and modified Gangsimtang (mGST) is prescribed to treat a condition called wasting thirst (), which resembles T2DM. This study reported the treatment of hyperglycemia using herbal medicines without oral hypoglycemic agents or insulin therapy. : A 36-year-old man with obesity was diagnosed with T2DM four years prior to hospitalization and experienced blood glucose level reduction from 22.2-27.8 mmol/L (400-500 mg/dL) to 5.6-11.1 mmol/L (100-200 mg/dL) by using herbal medicines. He visited D Korean Medicine Hospital with chronic polydipsia and general weakness as chief complaints. He was diagnosed with T2DM on the basis of a hemoglobin A1c level of 11.7% and 2 h postprandial blood glucose level of >25.0 mmol/L (450 mg/dL). Moreover, he was diagnosed with a "dual deficiency of qi and yin" () because of ordinary symptoms (). During his 30-day inpatient treatment, the patient received mGST 120 mL thrice daily; as a result, his postprandial blood glucose level decreased from 25.3 mmol/L (455 mg/dL) to 8.6 mmol/L (154 mg/dL), polydipsia decreased (visual analog scale score decreased from six to one), and triglyceride levels decreased from 11.7 mmol/L (1031 mg/dL) to 2.0 mmol/L (174 mg/dL). Plasma glucose levels remained stable for 6 months after the treatment, and no adverse events were observed over 200 days. We administered an herbal decoction to decrease plasma glucose levels without using oral hypoglycemic agents or insulin. : Herbal decoctions such as mGST can reduce hyperglycemia in patients with T2DM who refuse conventional therapy.
Topics: Male; Humans; Adult; Hypoglycemic Agents; Diabetes Mellitus, Type 2; Blood Glucose; Insulin; Hyperglycemia; Polydipsia; Plant Extracts
PubMed: 38003968
DOI: 10.3390/medicina59111919 -
Journal of Pediatric Endocrinology &... Jan 2024Although AVP and its surrogate, copeptin, are mainly regulated by osmotic and volume stimuli, their secretion is also elicited by stress and growth hormone (GH)...
OBJECTIVES
Although AVP and its surrogate, copeptin, are mainly regulated by osmotic and volume stimuli, their secretion is also elicited by stress and growth hormone (GH) stimulating agents. The aim of this report is to describe unusual patterns of copeptin response in a subset of children undergoing GH stimulation tests (GH-ST).
METHODS
We conducted a secondary analysis of a cohort of 93 healthy short children with no polydipsia, polyuria or fluid/electrolyte abnormalities, undergoing GH-ST with intravenous arginine, insulin, oral clonidine, or L-Dopa/carbidopa in various combinations. Serum copeptin concentrations were measured 1-3 min after phlebotomy (0 min) and at 60, 90, 120 min during GH-ST.
RESULTS
In 85 subjects (normal response group, NRG) serum copeptin concentrations increased from a 0 min median of 9 pmol/L (IQR 6, 11.5) (all values ≤21) to a median peak between 60 and 120 min of 22 (IQR15, 38) pmol/L, which varied depending on the stimulating agent. Conversely, in the eight outliers, copeptin concentrations decreased gradually from a median of 154 (IQR 61, 439) pmol/L (all ≥40 pmol/L) to values as low as 14 % of the basal value, by 120 min. Test-associated anxiety was described in 17 subjects in the NRG (20 %) and five of the outliers (63 %).
CONCLUSIONS
A distinctive pattern of very elevated serum copeptin concentrations occurred in 9 % of children undergoing GH-ST, similar to reports in previous pediatric studies. Etiology may include pain or stress of phlebotomy. This phenomenon should be recognized for proper interpretation of copeptin values in children.
Topics: Humans; Child; Phlebotomy; Glycopeptides; Intercellular Signaling Peptides and Proteins; Polyuria
PubMed: 37991411
DOI: 10.1515/jpem-2023-0390