Clinical profile and etiologies of children with central diabetes insipidus: a single-center experience from Turkey

Çatlı, Gönül; Abacı, Ayhan; Demir, Korcan; Ulusoy, Emel; Altıncık, Ayça; Büyükgebiz, Atilla; Böber, Ece

doi:10.1515/jpem-2012-0006

Cited by 15 publications

(9 citation statements)

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“…Because of its rarity, the numbers of CDI patients in previous studies were less than 100 [4][5][6], and the investigation of CDI prognosis has been difficult. In the present study, using databases of 3 hospital electronic medical recording systems, we evaluated 149 patients with CDI for the prognosis.…”

Section: Discussionmentioning

confidence: 99%

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Adipsia increases risk of death in patients with central diabetes insipidus

et al. 2014

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Arginine vAsopressin (Avp) is an antidiuretic hormone which is synthesized in the supraoptic and paraventricular nuclei in the hypothalamus, and released into the systemic circulation from the posterior pituitary [1,2]. Secretion of AVP is precisely controlled by plasma osmolality so that even as little as 1% increases in plasma osmolality or serum sodium levels significantly increase plasma AVP levels [1,2]. Increases in plasma osmolality are sensed via the osmoreceptors in the hypothalamus, and induce not Abstract. Central diabetes insipidus (CDI) is caused by deficiency of arginine vasopressin, an antidiuretic hormone. Patients with CDI manifest polyuria which is usually compensated for by increases in water intake. However, some patients are not able to sense thirst due to the destruction of osmoreceptors in the hypothalamus. These adipsic CDI patients are easily dehydrated and the consequent dehydration could be life-threatening. The objective of this study was to investigate the prognosis of adipsic CDI patients. We have reviewed 149 patients with CDI in three hospitals using databases of the electronic medical recording systems, and examined whether adipsia could affect the morbidity and mortality in CDI patients with multivariable analyses. Twenty-three patients with CDI were adipsic while the remaining 126 patients were non-adipsic. The multivariate analyses showed that the incidence of serious infections which required hospitalization was significantly higher in the adipsic CDI patients compared to that in non-adipsic CDI patients (p <0.001). A total of 6 patients with CDI died during the follow-up (median duration; 60 months, range 1 to 132 months). Four of them were adipsic, three of whom died of infection. The statistical analyses revealed that the risk of death in adipsic CDI patients was significantly higher than in non-adipsic patients (p =0.007). It is thus suggested that adipsic CDI patients were susceptible to serious infections which could be the causes of death.

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Section: Discussionmentioning

confidence: 99%

“…Central diabetes insipidus (CDI) is caused by deficiency of AVP, and the etiologies include several diseases in the hypothalamus such as germ cell tumor of the central nervous system, craniopharyngioma and inflammatory diseases [4][5][6]. Patients with CDI manifest polyuria, which is compensated for by increases in water intake if thirst sensation is intact.…”

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confidence: 99%

Adipsia increases risk of death in patients with central diabetes insipidus

et al. 2014

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“…Previous studies about pediatric CDI mainly focused on etiology and common features, while our study mainly focused on CDI caused by SSR lesions, and summarized the detailed clinical manifestations, tumor markers, radiology characteristics and histopathology about each etiology, trying to offer evidence for diagnosis and differential diagnosis. Acquired infiltrative disease or tumor including CP, GCT, LCH, pituitary adenoma, lymphoma, astrocytoma, and cavernous hemangioma were all the etiology of CDI reported before (9,(11)(12)(13)(14). In our cohort, the causes of SSR lesions only included GCT, LCH and CP, whose proportions were 74.5, 18.2, and 7.3%, respectively.…”

Section: Discussionmentioning

confidence: 69%

“…Liu et al (14) also found that GCT was the most common cause (52.6%) among acquired infiltrative disease or tumor in Taiwanese children with CDI, which was consistent with our results. This might indicate that GCT would be the most likely to invade the hypothalamic- (13), the proportion of GCT among acquire infiltrative disease or tumor was much lower (21.3, 21.1, and 26.7%, respectively). Considering that PUMCH is both a general hospital and the center for managing rare and complicated disorders, patients with endocrine problems more commonly visit this hospital, so the higher prevalence of GCT and less variety of the etiology in our study might be explained by potential selection bias.…”

Section: Discussionmentioning

confidence: 99%

Clinical Characteristics of Pediatric Patients With Sellar and Suprasellar Lesions Who Initially Present With Central Diabetes Insipidus: A Retrospective Study of 55 Cases From a Large Pituitary Center in China

Wang

et al. 2020

Front. Endocrinol.

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Purpose: To increase knowledge for the early differential diagnosis and accurate therapeutic strategies for pediatric patients with sellar or suprasellar region (SSR) lesions who initially present with central diabetes insipidus (CDI).Methods: This is a retrospective review of 55 pediatric patients (≤14 years old) with identified lesions in the SSR who initially presented with CDI at a large pituitary center between 2012 and 2018. The following data were summarized: demographic, clinical, endocrine, and neuroimaging data, intraoperative findings, histopathological findings, treatments, and prognosis.Results: In our group, the etiologies of the SSR lesions included germ cell tumors (GCTs, 74.5%), Langerhans cell histiocytosis (LCH, 18.2%), and craniopharyngioma (CP, 7.3%). Almost all patients (50/55, 90.9%) showed anterior pituitary dysfunction [multiple axes dysfunction (38), and isolated axis dysfunction (14)], while the GH/IGF-I axis was the most affected. Most GCT patients presented with various clinical manifestations besides CDI and had elevated β-HCG, whereas LCH and CP patients mostly presented few non-specific symptoms besides CDI and most had normal level tumor markers. Sellar MRI demonstrated that posterior pituitary bright spot disappearance occurred in all patients, and pituitary stalk thickening was observed in 96.7% of patients. Treatment varied due to the different etiologies of the SSR lesions. After follow-up for 35.4 ± 20.2 months, the proportions of patients who needed AVP (arginine vasopressin) for GCT, LCH, and CP were 86.5, 100, and 75%, respectively, and the proportions of patients who needed HRT were 89.2, 50, and 75%, respectively. Ji et al. Pediatric SSR Lesions With CDIConclusion: For pediatric SSR lesions that first manifest as CDI, we should comprehensively consider clinical characteristics and imaging features to aid in their early differential diagnosis. Tumor markers and surgical histopathology are also great complements for the differential diagnosis. Additionally, various treatment strategies should be adopted according to different causes to improve the child's prognosis and quality of life.

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“…1 In a series of 34 children with CDI, it was reported that four patients (7%) had LHH in etiology. 5 Central diabetes insipidus is the distinctive finding of HPA infiltration in cases with LCH. It occurs in 25% of all patients with LCH or up to 50% of patients with multisystem disease.…”

Section: Discussionmentioning

confidence: 99%

Langerhans Cell Histiocytosis with Isolated Central Diabetes Insipidus, Low Grade Fever and Sellar Erosion

Okur¹,

Arı²,

Çeti̇nkaya³

et al. 2021

Trends in Pediatrics

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Langerhans cell histiocytosis (LCH) is a rare disease of the monocyte-macrophage system. Although it is known that bone involvement is seen very frequently in cases with LCH, our case is the first case with a lytic-destructive lesion in the bone structure forming sella turcica. A 4-year-old, 5-month-old male patient who applied to our outpatient clinic was diagnosed with Langerhans cell histiocytosis in further examination after the diagnosis of central diabetes insipidus (CDI) was made. On cranial magnetic resonance imaging (MRI), widespread lytic-destructive bone lesions were observed in the bone structure forming the sella (sphenoid bone), sellar destruction not previously described in the literature. Sellar erosion has not been reported before in cases diagnosed with LCH in the literature. The presence of low-grade fever in a patient presenting with isolated CDI is a warning sign for the diagnosis of LCH.

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Clinical profile and etiologies of children with central diabetes insipidus: a single-center experience from Turkey

Cited by 15 publications

References 0 publications

Adipsia increases risk of death in patients with central diabetes insipidus

Adipsia increases risk of death in patients with central diabetes insipidus

Clinical Characteristics of Pediatric Patients With Sellar and Suprasellar Lesions Who Initially Present With Central Diabetes Insipidus: A Retrospective Study of 55 Cases From a Large Pituitary Center in China

Langerhans Cell Histiocytosis with Isolated Central Diabetes Insipidus, Low Grade Fever and Sellar Erosion

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