Adipsic Hypernatremia after Clipping of a Ruptured Aneurysm in the Anterior Communicating Artery: A Case Report

Kim, Won Ki; Lee, Tae‐Ho; Kim, Ae Jin; Ro, Han; Chang, Jae Hyun; Lee, Hyun Hee; Chung, Wookyung; Jung, Ji Yong

doi:10.5049/ebp.2021.19.2.56

Cited by 2 publications

(1 citation statement)

References 11 publications

Supporting

Mentioning

Contrasting

Order By: Relevance

“…We assume that chemotherapy improved the mechanical compression or infiltration of the hypothalamus related to OTT, leading to the recovery of the sense of thirst. Previous studies have shown that i) adipsia in a patient with neurosarcoidosis resolved following anti-tumor necrosis factor-α therapy ( 10 ) and ii) postoperative impairment of OTT resolved over time in 10 patients with craniopharyngioma or anterior communicating artery aneurysm ( 11 , 12 , 13 , 14 , 15 ). The resolution of impaired OTT in these cases suggests the plasticity of the OTT, thereby supporting our speculation.…”

Section: Discussionmentioning

confidence: 99%

Potential indication of chemotherapy for hypodipsia and arginine vasopressin deficiency secondary to hypothalamic-pituitary Langerhans cell histiocytosis: a case report and literature review

Ota,

Sato,

Nakano

et al. 2024

Clin Pediatr Endocrinol

View full text Add to dashboard Cite

Hypothalamic-pituitary Langerhans cell histiocytosis (HP-LCH) is often associated with arginine vasopressin deficiency (AVD). Patients with AVD caused by HP-LCH rarely develop an impaired osmotic threshold for thirst (OTT). Improvement in OTT among such patients has not been reported in the literature. To our knowledge, here we report the first case of AVD due to HP-LCH in which hypodipsia resolved during chemotherapy. A nine-year-old Japanese girl presented with polydipsia, polyuria, anorexia, and hypernatremia (149.8 mEq/L) and was diagnosed with AVD secondary to HP-LCH. Visual analog scale examination showed a reduced OTT following the water deprivation test. During chemotherapy for Langerhans cell histiocytosis (LCH), serum sodium concentrations became stable between 138.9 and 142.9 mEq/L under the replacement of desmopressin. Repeated visual analog scale examinations showed that she experienced a sense of thirst at a serum sodium concentration of 142.3-144.6 mEq/L, at which she did not experience any thirst prior to the initiation of chemotherapy. These data suggest that chemotherapy directly improved the OTT in our patient. Improved mechanical compression or infiltration of the hypothalamus related to OTT may lead to the recovery of the sense of thirst. This report highlights the potential role of chemotherapy for solitary HP-LCH in patients with hypodipsia and AVD.

show abstract