Diabetes Insipidus, Neurogenic

尿崩症,神经性
  • 文章类型: Case Reports
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  • 文章类型: Case Reports
    背景:希恩综合征是一种罕见的疾病,其典型特征是产后休克或出血后垂体前叶功能减退。虽然尿崩症(DI)通常与希汉综合征无关,我们介绍了一例罕见的经产女性在严重产后出血后发展为快速发作的全垂体功能减退症和DI的病例。
    方法:一位以前健康的39岁女性,gravida5,para4,表现为阴道分娩后低血容量性休克,归因于严重的产后出血,需要紧急子宫切除术。尽管她的休克发作在住院期间得到了缓解,她出现了间歇性发热,后来被诊断为肾上腺功能不全。给予氢化可的松有效地解决了发烧。然而,她随后发展为尿崩症。通过功能性激素检查和MRI检查结果证实了Sheehan综合征合并中枢性尿崩症的诊断。治疗包括激素替代疗法,在10年的随访期内发现持续性全垂体功能减退。
    结论:希汉综合征是产后出血的罕见并发症。应该怀疑中枢尿崩症,虽然不常见,患者出现多尿和多饮。此外,应考虑长期激素替代疗法的潜在必要性.
    BACKGROUND: Sheehan\'s syndrome is a rare condition, which is classically characterized by anterior pituitary hypofunction following postpartum shock or hemorrhage. While diabetes insipidus (DI) is not commonly associated with Sheehan\'s syndrome, we present a rare case of a multiparous female developing rapid-onset panhypopituitarism and DI following severe postpartum hemorrhage.
    METHODS: A previously healthy 39-year-old woman, gravida 5, para 4, presented with hypovolemic shock after vaginal delivery, attributed to severe postpartum hemorrhage, necessitating emergent hysterectomy. Although her shock episodes resolved during hospitalization, she developed intermittent fever, later diagnosed as adrenal insufficiency. Administration of hydrocortisone effectively resolved the fever. However, she subsequently developed diabetes insipidus. Diagnosis of Sheehan\'s syndrome with central diabetes insipidus was confirmed through functional hormonal tests and MRI findings. Treatment consisted of hormone replacement therapy, with persistent panhypopituitarism noted during a ten-year follow-up period.
    CONCLUSIONS: Sheehan\'s syndrome is a rare complication of postpartum hemorrhage. Central diabetes insipidus should be suspected, although not commonly, while the patient presented polyuria and polydipsia. Besides, the potential necessity for long-term hormonal replacement therapy should be considered.
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  • 文章类型: Systematic Review
    目的:中心性尿崩症或加压素缺乏症(AVP-D)是经蝶窦手术(TSS)后最常见的水分平衡障碍,在研究中患病率不同。我们旨在确定使用TSS治疗的垂体肿瘤患者中新出现的短暂性或永久性AVP-D的发生率。
    方法:我们对Medline,Embase和CochraneLibrary于2000/01-2021/01/01/31之间进行,用于报告垂体腺瘤结局的研究,TSS术后颅咽管瘤和Rathke裂隙囊肿(RCC),并提供术后AVP-D的定义。我们使用Freeman-Tukey转化随机效应荟萃分析将结果合并为具有95%置信区间(CI)的比例。
    结果:来自11694项研究,包括51个。短暂性或永久性AVP-D的发生率为:全组的17%(95%CI,13-21)和3%(95%CI,2-5),16%(95%CI,12-21)和2%(95%CI,2-3)在垂体腺瘤,颅咽管瘤的31%(95%CI,24-39)和30%(95%CI,22-39),RCC中35%(95%CI,16-57)和14%(95%CI,6-23),分别。根据诊断标准,短暂性或永久性AVP-D的发生率为:低渗多尿,14%(95%CI,8-22)和3%(95%CI,1-4),低渗多尿和高钠血症,21%(95%CI,13-29)和5%(95%CI,2-11),去氨加压素给药,22%(95%CI,15-29)和9%(95%CI,0-30),分别。
    结论:在TSS之后,一小部分垂体腺瘤患者具有永久性AVP-D(2%),但是颅咽管瘤的患病率达到30%,RCC的患病率达到14%。术后AVP-D的诊断标准仍然可变,影响报告的这种情况的发生率。
    OBJECTIVE: Central diabetes insipidus or vasopressin deficiency (AVP-D) is the most frequent water balance disorder after transsphenoidal surgery (TSS) with variable prevalence amongst studies. We aimed to determine rates of newly developed transient or permanent AVP-D in patients with pituitary tumours treated with TSS.
    METHODS: We performed systematic review of Medline, Embase, and Cochrane Library between January 1, 2000 and January 31, 2021 for studies reporting on outcomes for pituitary adenoma, craniopharyngioma, and Rathke\'s cleft cyst (RCC) after TSS and providing definition of post-operative AVP-D. We pooled the results as proportions with 95% confidence intervals (CIs) using Freeman-Tukey transformation random effects meta-analysis.
    RESULTS: From 11 694 studies, 51 were included. Rates of transient or permanent AVP-D were: 17% (95% CI, 13-21) and 3% (95% CI, 2-5) in total group, 16% (95% CI, 12-21) and 2% (95% CI, 2-3) in pituitary adenomas, 31% (95% CI, 24-39) and 30% (95% CI, 22-39) in craniopharyngiomas, and 35% (95% CI, 16-57) and 14% (95% CI, 6-23) in RCCs, respectively. Based on diagnostic criteria, rates of transient or permanent AVP-D were: For hypotonic polyuria, 14% (95% CI, 8-22) and 3% (95% CI, 1-4), for hypotonic polyuria and hypernatraemia, 21% (95% CI, 13-29) and 5% (95% CI, 2-11), and for desmopressin administration, 22% (95% CI, 15-29) and 9% (95% CI, 0-30), respectively.
    CONCLUSIONS: Following TSS, a small proportion of patients with pituitary adenoma have permanent AVP-D (2%), but prevalence reaches 30% in ones with craniopharyngioma and 14% in those with RCC. Diagnostic criteria for post-operative AVP-D remain variable affecting reported rates of this condition.
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  • 文章类型: Case Reports
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  • 文章类型: Case Reports
    继发性自发性气胸发生在已知潜在肺部疾病的患者中。肺气肿患者,bullae,肺部的囊性病变有发生气胸的高风险。像朗格汉斯细胞组织细胞增生症(LCH)这样的囊性肺病可能会出现气胸等并发症。其他常见的表现特征包括斑丘疹和骨病变。它也可能与内分泌疾病有关,最常见的中枢尿崩症(CDI)。我们在这里介绍一个22岁的男性,他出现了气胸,多尿,和多饮。他在经支气管肺活检时被诊断为LCH,与CDI相关,并采用胸腔镜引导自体血补片治疗持续性漏气和皮下阿糖胞苷。
    Secondary spontaneous pneumothoraces occur in patients with known underlying lung disease. Patients with emphysema, bullae, and cystic lesions in the lungs are at high risk of developing pneumothorax. Cystic lung diseases like Langerhans cell histiocytosis (LCH) can present with complications like pneumothorax. Other common presenting features include maculopapular rashes and bone lesions. It can also be associated with endocrinopathies, most commonly central diabetes insipidus (CDI). We here present a case of a 22-year-old male who presented with pneumothorax, polyuria, and polydipsia. He was diagnosed with LCH on transbronchial lung biopsy, associated with CDI, and was treated with thoracoscopy-guided autologous blood patch for persistent air leak and subcutaneous cytarabine.
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  • 文章类型: Case Reports
    一名78岁的男子抱怨亚急性全身疲劳和厌食症,复视和步态障碍。他表现出广泛的步态和小步步态,客观上没有异常的眼球运动。脑MRI显示垂体柄和腺体增大,对比度均匀增强。PET-CT显示垂体吸收FDG,纵隔淋巴结,和左肺门淋巴结。血液检查显示垂体功能减退症和高血清IgG4水平,最高可达265µmg/dl。组织病理学检查显示活检纵隔淋巴结中无IgG4阳性细胞浸润。然而,我们根据临床症状和MRI表现怀疑IgG4相关的垂体炎,用类固醇显着解决。出现了中央掩盖尿崩症,但口服去氨加压素改善。我们应该密切关注IgG4相关的垂体炎可能会出现各种症状,这些症状被认为是与衰老或潜在疾病有关的不确定的主诉,尤其是老年多发病患者。
    A 78-year-old man complained of subacute general fatigue and anorexia, following diplopia and gait disturbance. He demonstrated wide-based and small-stepped gait without objectively abnormal ocular movements. Brain ‍MRI showed enlargement of the pituitary stalk and gland with uniform contrast enhancement. PET-CT showed FDG ‍uptake in the pituitary gland, mediastinal lymph nodes, and left hilar lymph nodes. Blood investigations revealed panhypopituitarism and high serum IgG4 levels up to 265 ‍mg/dl. Histopathological examination revealed no IgG4-positive cell infiltration in the biopsied mediastinal lymph nodes. However, we suspected IgG4-associated hypophysitis based on the clinical symptoms and MRI findings, which were markedly resolved with steroid. Central masked diabetes insipidus was manifested, but was improved with oral desmopressin. We should pay close attention to the fact that IgG4-related hypophysitis may present with various symptoms regarded as indefinite complaints related to aging or underlying diseases, especially in elderly patients with multimorbidity.
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  • 文章类型: Letter
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  • 文章类型: Journal Article
    背景:术后中枢性尿崩症(CDI)通常见于颅咽管瘤(CP)患者,炎症反应在CPs中起重要作用。我们旨在评估术前外周炎症标志物及其组合对CPsCDI发生的预测价值。
    方法:回顾性分析208例接受手术治疗的CP患者的临床资料,包括术前外周炎症指标。术前外周血白细胞(WBC),中性粒细胞,淋巴细胞,单核细胞,血小板(PLT),中性粒细胞与淋巴细胞比率(NLR),派生NLR(dNLR),在总共208例CP患者以及不同年龄和手术方式的CP患者亚组中评估了单核细胞与淋巴细胞比率(MLR)和PLT与淋巴细胞比率(PLR).通过受试者操作员特征曲线分析评估其预测值。
    结果:术前外周白细胞,中性粒细胞,NLR,dNLR,MLR,与PLR呈正相关,淋巴细胞与CP患者术后CDI的发生呈负相关,特别是当白细胞≥6.66×109/L或淋巴细胞≤1.86×109/L时同时,多因素logistic回归分析显示,年龄>18岁的患者WBC>6.39×109/L,经颅入路患者WBC>6.88×109/L或淋巴细胞≤1.85×109/L与术后CDI发生率升高密切相关。此外,从受试者操作特征曲线分析获得的曲线下面积显示,炎症标志物的最佳预测因子是总CP患者的NLR,≤18岁年龄组和经蝶窦组的MLR,>18岁年龄组的NLR和经颅组的dNLR。值得注意的是,NLR+dNLR组合指数在所有组中显示出最有价值的预测因子。
    结论:术前外周炎症标志物,尤其是WBC,淋巴细胞和NLR+dNLR,是CP术后CDI的有希望的预测因子。
    BACKGROUND: Postoperative central diabetes insipidus (CDI) is commonly observed in craniopharyngioma (CP) patients, and the inflammatory response plays an important role in CPs. We aimed to evaluate the predictive value of preoperative peripheral inflammatory markers and their combinations regarding CDI occurrence in CPs.
    METHODS: The clinical data including preoperative peripheral inflammatory markers of 208 CP patients who underwent surgical treatment were retrospectively collected and analyzed. The preoperative peripheral white blood cells (WBC), neutrophils, lymphocytes, monocytes, platelet (PLT), neutrophil-to-lymphocyte ratio (NLR), derived-NLR (dNLR), monocyte-to-lymphocyte ratio (MLR) and PLT-to-lymphocyte ratio (PLR) were assessed in total 208 CP patients and different age and surgical approach CP patient subgroups. Their predictive values were evaluated by the receiver operator characteristic curve analysis.
    RESULTS: Preoperative peripheral WBC, neutrophils, NLR, dNLR, MLR, and PLR were positively correlated and lymphocyte was negatively associated with postoperative CDI occurrence in CP patients, especially when WBC ≥ 6.66 × 109/L or lymphocyte ≤ 1.86 × 109/L. Meanwhile, multiple logistic regression analysis showed that WBC > 6.39 × 109/L in the > 18 yrs age patients, WBC > 6.88 × 109/L or lymphocytes ≤ 1.85 × 109/L in the transcranial approach patients were closely associated with the elevated incidence of postoperative CDI. Furthermore, the area under the curve obtained from the receiver operator characteristic curve analysis showed that the best predictors of inflammatory markers were the NLR in total CP patients, the MLR in the ≤ 18 yrs age group and the transsphenoidal group, the NLR in the > 18 yrs age group and the dNLR in the transcranial group. Notably, the combination index NLR + dNLR demonstrated the most valuable predictor in all groups.
    CONCLUSIONS: Preoperative peripheral inflammatory markers, especially WBC, lymphocytes and NLR + dNLR, are promising predictors of postoperative CDI in CPs.
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  • 文章类型: Journal Article
    多尿多饮综合征可由中枢性尿崩症引起,肾性尿崩症或原发性多饮。为了避免与糖尿病混淆,在2022年将“中心性尿崩症”的名称更改为精氨酸加压素(AVP)缺乏症,并将“肾源性尿崩症”更名为AVP抵抗。为了区分这三个实体,在过去的十年中,已经引入了各种基于和肽素的渗透和非渗透刺激测试。高渗盐水测试加血浆和肽素测量成为诊断准确性最高的测试,取代缺水试验作为多尿多饮综合征鉴别诊断的金标准。治疗AVP缺乏的主要方法是用去氨加压素替代AVP,一种对AVP受体2(AVPR2)具有特异性的AVP合成类似物,这通常会导致多尿和多饮的快速改善。去氨加压素的主要不良反应是稀释性低钠血症,可以通过定期执行所谓的去氨加压素逃逸方法来减少。过去几年的证据表明,AVP缺乏症患者存在催产素缺乏症。这种潜在的缺陷应该在未来的研究中进一步评估。包括临床实践的可行激发试验和催产素替代的介入试验。
    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.
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  • 文章类型: Journal Article
    大麻和合成大麻素的消费正在全球范围内增加。大麻含有许多植物大麻素,作用于全身表达的G蛋白偶联大麻素受体1型(CB1R)和2型(CB2R),包括肾脏.基本上每个器官,包括肾脏,产生内源性大麻素(EC),这些受体的内源性配体。大麻素急剧增加啮齿动物和人类的尿量,因此可能影响全身水和电解质稳态。由于肾脏集合管(CD)调节全身水分,酸/碱,和电解质平衡通过主要电池(PC)和嵌入电池(IC)的特定功能,我们检查了CB1R在小鼠CD中的细胞特异性免疫定位。针对CB1R的C端或N端一致标记的皮质和髓样CD中AQP2(-)细胞的抗体,因此,大概是IC。鉴于ICs在尿酸中的作用已经确立,我们在7d的小鼠和用大麻素受体激动剂治疗的非酸负荷小鼠中使用了清除方法,WIN55,212-2(WIN),或车辆控制。虽然WIN对尿酸没有影响,与对照组相比,这些经WIN治疗的小鼠在PC中的根尖+根尖下AQP2表达较少,并出现与大量稀释尿液排泄相关的急性尿崩症(DI).当WIN+1-去氨基-8-d-精氨酸-加压素(去氨加压素,DDAVP)共同给药,与中枢而非肾性DI一致。尽管IC表示CB1R,CB1R在这种细胞类型中的生理作用尚待确定.
    Cannabis and synthetic cannabinoid consumption are increasing worldwide. Cannabis contains numerous phytocannabinoids that act on the G protein-coupled cannabinoid receptor type 1 (CB1R) and cannabinoid receptor type 2 expressed throughout the body, including the kidney. Essentially every organ, including the kidney, produces endocannabinoids, which are endogenous ligands to these receptors. Cannabinoids acutely increase urine output in rodents and humans, thus potentially influencing total body water and electrolyte homeostasis. As the kidney collecting duct (CD) regulates total body water, acid/base, and electrolyte balance through specific functions of principal cells (PCs) and intercalated cells (ICs), we examined the cell-specific immunolocalization of CB1R in the mouse CD. Antibodies against either the C-terminus or N-terminus of CB1R consistently labeled aquaporin 2 (AQP2)-negative cells in the cortical and medullary CD and thus presumably ICs. Given the well-established role of ICs in urinary acidification, we used a clearance approach in mice that were acid loaded with 280 mM NH4Cl for 7 days and nonacid-loaded mice treated with the cannabinoid receptor agonist WIN55,212-2 (WIN) or a vehicle control. Although WIN had no effect on urinary acidification, these WIN-treated mice had less apical + subapical AQP2 expression in PCs compared with controls and developed acute diabetes insipidus associated with the excretion of large volumes of dilute urine. Mice maximally concentrated their urine when WIN and 1-desamino-8-d-arginine vasopressin [desmopressin (DDAVP)] were coadministered, consistent with central rather than nephrogenic diabetes insipidus. Although ICs express CB1R, the physiological role of CB1R in this cell type remains to be determined.NEW & NOTEWORTHY The CB1R agonist WIN55,212-2 induces central diabetes insipidus in mice. This research integrates existing knowledge regarding the diuretic effects of cannabinoids and the influence of CB1R on vasopressin secretion while adding new mechanistic insights about total body water homeostasis. Our findings provide a deeper understanding about the potential clinical impact of cannabinoids on human physiology and may help identify targets for novel therapeutics to treat water and electrolyte disorders such as hyponatremia and volume overload.
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