Efficacy of tolvaptan in an infant with syndrome of inappropriate antidiuretic hormone secretion associated with holoprosencephaly:A case report

BACKGROUND Holoprosencephaly(HPE)is a congenital malformation with various degrees of incomplete separation of the cerebral hemispheres due to differentiation disorders of the forebrain.Although HPE with diabetes insipidus due to associated pituitary dysfunction has been reported,HPE with the syndrome of inappropriate antidiuretic hormone secretion(SIADH)is very rare.Tolvaptan,a vasopressin V2 receptor antagonist,is effective in adults with SIADH.However,there is no report of its efficacy in infants with SIADH.The purpose of this report is to demonstrate that tolvaptan is effective for SIADH in infants and that administration of tolvaptan eliminates the need for restriction of water intake and sodium administration.CASE SUMMARY A 2414-g female infant was born at 38 wk by normal vaginal delivery.Facial anomalies and head magnetic resonance imaging indicated semilobar HPE.After birth,she had hyponatremia due to SIADH and was treated using water and sodium restriction.However,she developed an exaggerated response to the fluid restrictions,resulting in large fluctuations in serum sodium levels.Subsequent administration of tolvaptan improved the fluctuations in serum sodium levels without the need for adjustment of water or sodium administration.Serum sodium was maintained within the normal range after discontinuation of tolvaptan at 80 d of life.There were no side effects,such as hypernatremia or liver dysfunction,during the administration of tolvaptan.CONCLUSION This is the first report on the safety and efficacy of tolvaptan in an infant with SIADH associated with HPE.

抗利尿激素不适当分泌综合征(Treatment of the syndrome of inappropriate secretion of antidiuretic hormone)的治疗

抗利尿激素不适当分泌综合征(SIADH)是因多种病理因素引起的抗利尿激素(ADH)分泌异常增多,导致体内水潴留,持续性低钠血症、低渗透压血症、尿呈反常的高渗透压、尿钠排泄增多等现象。由于低钠血症和低血浆渗透压,其临床特征则以脑细胞肿胀(脑水肿)所致中枢神经系统的功能紊乱为主要表现。

Nedaplatin-induced syndrome of inappropriate secretion of antidiuretic hormone:A case report and review of the literature

BACKGROUND Syndrome of inappropriate secretion of antidiuretic hormone(SIADH)is relatively common in several cancers,such as small cell lung cancer.However,nedaplatin-induced SIADH is rare.We describe a case of SIADH mediated by nedaplatin.CASE SUMMARY A 54-year-old female with nasopharyngeal carcinoma was treated with nedaplatin and developed severe hyponatremia due to SIADH.The side effects were successfully treated by fluid restriction and sodium supplementation.CONCLUSION This case report highlights the importance of cautiously treating life-threatening hyponatremia in patients treated with nedaplatin.

Natural killer/T-cell lymphoma with concomitant syndrome of inappropriate antidiuretic hormone secretion: A case report and review of literature

We report a case of natural killer(NK)/T-cell lymphoma with concomitant syndrome of inappropriate antidiuretic hormone secretion(SIADH).The patient was a 64-yearold woman with a history of nasopharyngeal carcinoma of over 30 years.She was admitted with a chief complaint of intermittent fever for 2 mo.Palpation after admission indicated a swollen lymph node below the left jaw.Multiple imaging examinations on admission indicated multiple enlarged lymph nodes throughout the body.We performed a left submandibular lymph node biopsy,and the results revealed NK/T-cell lymphoma.A biochemical examination indicated Epstein-Barr virus positivity.At the same time,the patient developed hyponatremia.Based on her laboratory examination and clinical manifestation,decreased plasma osmolality,urine osmolality greater than plasma osmolality,lack of skin swelling,normal blood pressure,normal renal function,no adrenal function detected on serology,and no abnormalities in imaging examination of the adrenal glands,the likelihood of SIADH in the patient was high.After fluid restriction and administration of sodium chloride,the patient’s blood sodium level gradually increased.Subsequently,the immune function of the patient declined,there were severe symptoms of infection,and she died of respiratory failure.NK/T-cell lymphoma associated with SIADH has not,to our knowledge,been previously reported in PubMed.This case emphasizes the importance of monitoring serum ion levels,especially serum sodium,in patients with NK/T-cell lymphoma.

Pazopanib-induced posterior reversible encephalopathy syndrome with possible syndrome of inappropriate secretion of antidiuretic hormone: an incidental or pathophysiological association?

Introduction:Pazopanib is an oral protein kinase inhibitor(PKI)that targets vascular endothelial growth factor(VEGF)receptors,fibroblastic growth factor receptors,platelet-derived growth factor receptors,and stem cell factor that inhibits VEGF-induced cellular proliferation.Pazopanib is approved for use in advanced renal cell carcinoma and subtypes of advanced soft-tissue sarcoma(Deguchi et al.,2018).Major adverse drug reactions of pazopanib include hypertension,high-grade hyponatremia and posterior reversible encephalopathy syndrome(PRES)(Berardi et al.,2016;Deguchi et al.,2018).In clinical trials,few investigations have been conducted to determine the aetiology of PKI-associated hyponatremia,the mechanism remains therefore unknown.Only rare cases of PKI-induced syndrome of inappropriate secretion of antidiuretic hormone(SIADH)(Largeau et al.,2019),and none with pazopanib,have been reported.PRES is a clinical and radiological entity where a bilateral white matter oedema,occurring predominantly in the posterior occipital and parietal lobes,is associated with several neurologic symptoms.Interestingly,a recent review suggests that SIADH could be a symptom of PRES(Largeau et al.,2019).To our knowledge,this is the first case published where pazopanib-induced PRES occurs contemporaneously with possible SIADH.

Inappropriate secretion of antidiuretic hormone:a rare complication after common bile duct exploration

BACKGROUND: The syndrome of inappropriate secretion of antidiuretic hormone (SIADH) as a dilutional hyponatremia is due to a pathological increase of antidiuretic hormone (ADH). It is characterized by hyponatremia and decreased serum osmolarity as well as an increase in urinary osmolarity. The most common etiological factors of this syndrome include diseases or trauma of the central nervous system and malignant tumor or inflammation of the lung. SIADH following abdominal surgery is rare. METHODS: We report the case of a 68-year-old woman who developed, 24 hours after common bile duct exploration and stone removal, continuous hyponatremia for 20 days and clinical manifestations of nausea, vomiting, and lethargy without focal neurological signs. RESULT: Laboratory examinations supported the diagnosis of SIADH. After therapy with fluid restriction, the patient recovered. CONCLUSION: There are diverse causes for SIADH. It is important to have kept this clinical possibility in mind in the differential diagnosis of refractory hyponatremia under any circumstances.

Changes of atrial natriuretic peptide and antidiuretic hormone in children with postural tachycardia syndrome and orthostatic hypertension： a case control study

Background The abnormal blood volume regulation is one of the most important pathogenesis in postural tachycardia syndrome in children.This study was designed to investigate the plasma atrial natriuretic peptide and antidiuretic hormone levels in postural tachycardia syndrome children,and their associations with the changes in heart rate and blood pressure in head-up test.Methods Twenty-one postural tachycardia syndrome patients （（12±2） years） and 26 healthy children （（12±1） years） were included.According to blood pressure changes in head-up test,the postural tachycardia syndrome patients were divided into two subgroups：postural tachycardia syndrome with orthostatic hypertension and postural tachycardia syndrome without orthostatic hypertension.The plasma atrial natriuretic peptide and antidiuretic hormone levels were measured using enzyme-linked immunosorbent assay.Results The plasma atrial natriuretic peptide level in postural tachycardia syndrome patients was higher than the control （P=0.004）,whereas the difference in plasma antidiuretic hormone level between postural tachycardia syndrome and controls was not significant （P=0.222）.The plasma antidiuretic hormone level of patients suffering from postural tachycardia syndrome with orthostatic hypertension was much higher than that of children having postural tachycardia syndrome without orthostatic hypertension （P ＜0.05）.In postural tachycardia syndrome patients,the updght max heart rate was positively correlated with the plasma atrial natriuretic peptide level （r=0.490,P＜0.05） and the upright systolic blood pressure was positively correlated with the plasma antidiuretic hormone levels （r=0.472,P ＜0.05）.Conclusions There was a disturbance of plasma atrial natriuretic peptide and antidiuretic hormone in postural tachycardia syndrome children.

Inappropriate antidiuretic hormone secretion in a patient with pulmonary lymphoepithelioma like carcinoma

Antidiuretic hormone （ADH） is produced by an area of he brain called the hypothalamus. The hormone is stored in and released by the pituitary gland. When ADH （also called vasopressin） is produced somewhere other than the hypothalamus, the condition is called syndrome of inappropriate antidiuretic hormone （SIADH）. A variety of conditions can trigger abnormal ADH production, but the main cause is cancer. It is frequently one of the first signs of lung cancer, especially small cell carcinoma, which produces ADH ectopically.l

去氨加压素片服用方法影响尿崩症治疗效果的病例分析

中枢性尿崩症（diabetes insipidus,DI）是由抗利尿激素（antidiuretic hormone,ADH）缺乏所致的肾脏浓缩功能障碍,可出现多尿、夜尿增多和烦渴等症状。去氨加压素是一种ADH类似物,几乎是所有中枢性尿崩症患者的首选药物[1]。去氨加压素片为成功应用于临床的肽类激素口服剂型治疗药物[2],临床效果明显,本文报道1例由于个体差异,进食导致药物敏感度降低的患者。

Case of inappropriate ADH syndrome:Hyponatremia due to polyethylene glycol bowel preparation

Colonoscopic screening has been reported to reduce deaths from colorectal cancer.Adequate bowel preparation is essential for this and safety is an important issue in choosing the methods.Polyethylene glycol(PEG)is regarded as a safe method for cleansing,especially compared with oral sodium phosphate.Here,we present a case of hyponatremia caused by the syndrome of inappropriate antidiuretic hormone(ADH)syndrome after PEG precolonoscopic cleansing resulting in generalized tonic-clonic seizures.A 62-year-old women had ingested PEG for precolonoscopic bowel cleansing.While waiting for the colonoscopy,she developed a stuporous mentality and generalized tonic-clonic seizures,which did not correlate with brain magnetic resonance imaging.Her serum sodium level was 113 mEq per liter and laboratory analyses were consistent with inappropriate ADH syndrome.Her thyroid and adrenal functions were normal.There were no malignancies,infections,respiratory disorders or central nervous disorders and she had no history of taking either diuretics or other medications,which might have caused inappropriate ADH syndrome.She was treated with 3%hypertonic saline and showed a complete neurological recovery as her sodium levels recovered.Follow-up visits showed the patient to have a normal sodium level without neurologic deficits.This case shows that inappropriate ADH syndrome can be caused by PEG preparation,which implies that physicians have to be aware of the possible side effects of this colonic cleansing approach and mindful of the possible ensuing symptoms.

Desmopression is an effective adjunct treatment for reversing excessive hyponatremia overcorrection

We report a case of a 50-year-old malnourished African American male with hiccups, nausea and vomiting who was brought to the Emergency Department after repeated seizures at home. Laboratory evaluations revealed sodium(Na+) 107 mmol/L, unmeasurably low potassium, chloride < 60 mmol/L, bicarbonate of 38 mmol/L and serum osmolality 217 m Osm/kg. Seizures were controlled with 3% saline Ⅳ. Once nausea was controlled with iv antiemetics, he developed large volume free water diuresis with 6 L of dilute urine in 8 h(urine osmolality 40-60 m Osm/kg) and serum sodium rapidly rose to 126 mmol/L in 12 h. Both intravenous desmopressin and 5% dextrose in water was given to achieve a concentrated urine and to temporarily reverse theacute rise of sodium, respectively. Serum Na+ was gradually re-corrected in 2-3 mmol/L daily increments from 118 mmol/L until 130 mmol/L. Hypokalemia was slowly corrected with resultant auto-correction of metabolic alkalosis. The patient discharged home with no neurologic sequaele on the 11 th hospital day. In euvolemic hyponatremic patient, controlling nausea may contribute to unpredictable free water diuresis. The addition of an antidiuretic hormone analog, such as desmopressin can limit urine output and prevent an unpredictable rise of the serum sodium.

Respiratory tract tumor

930125 Dynamic study on plasma antidiureticChina Medical Abstracts(Internal Medicine) hormone before and after resection of lung can-cers.YANG Jun(杨俊),et al.Nanfang Hosp,Guangzhou.Chin J Oncol 1992;14(5):360-361.Plasma antidiuretic hormone(ADH)was as-sayed before and after resection of lung cancers.The results showed that the plasma ADH in thecontrol group was 11.6±4.8Pg/ml in contrastto higher levels in the lung cancer patients.TheADH level was the highest in patients

The management of hyponatremia in cancer patients:a practical view in Spain

Hyponatremia is a common disorder among cancer patients and is associated with a poor prognosis in several malignancies.It is classified by volemic status into hypovolemic,euvolemic,and hypervolemic hyponatremia.Clinical history,physical examination,and blood and urine tests are important for a correct classification and diagnosis of hyponatremia,to assure correct management.Treatment of hyponatremia in cancer patients depends on the etiology of hyponatremia,as well as on the chosen therapy for the tumor.Supportive care is also a factor to be taken into account.

Acute Intermittent Porphyria： A Diagnostic Challenge for Endocrinologist

INTRODUCTION Acute intermittent porphyria （AIP） is an autosomal dominant inborn error of metabolism caused by deficiency of porphobilinogen （PBG） deaminase, also known as hydroxymethylbilane synthase （HMBS）, the third enzyme in the heine biosynthetic pathway.

DOES ADRENAL GLAND PARTICIPATE IN PRESSOR RESPONSE OF VAGUS POSTPITUITARY REFLEX?

H. C. Chang et al. observed the release of acetycholine (ACH) in the central nervous system during stimulating the central end of the vagus nerve on the isolated head preparation of the dog. It is this ACH that releases the vasopressin, oxytocin and antidiuretic hormone in neurohypophysis. The pathway of this reflex may