ArticleBrain pathology (Zurich, Switzerland)2017
Adipsic hypernatremia without hypothalamic lesions accompanied by autoantibodies to subfornical organ.
Article in Brain pathology (Zurich, Switzerland), 2017. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 20 papers.
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Who cites it
20 citing papers in PubMed, 37 citations in OpenAlex.
- Adipsic hypernatremia associated with hypothalamic dysfunction and sleep disorders.JCEM case reports · 2026Article
- 'Un-thirsty' hypernatremia.Endocrinology, diabetes & metabolism case reports · 2025Article
- Understanding of Thirst in Medical Science.Yonago acta medica · 2025Review
- Autoimmunity related to anti-NaEndocrine journal · 2024Review
- Article
- Brain sodium sensing for regulation of thirst, salt appetite, and blood pressure.Physiological reports · 2024Review
- High-Titer Anti-ZSCAN1 Antibodies in a Toddler Clinically Diagnosed with Apparent Rapid-Onset Obesity with Hypothalamic Dysfunction, Hypoventilation, and Autonomic Dysregulation Syndrome.International journal of molecular sciences · 2024Article
- Anti-ZSCAN1 Autoantibodies Are a Feasible Diagnostic Marker for ROHHAD Syndrome Not Associated with a Tumor.International journal of molecular sciences · 2024Article
- Adipsic hypernatremia with marked hyperprolactinemia and GH deficiency in a 9-year-old boy.Clinical pediatric endocrinology : case reports and clinical investigations : official journal of the Japanese Society for Pediatric Endocrinology · 2024Article
- Case report: Psychosis and catatonia in an adolescent patient with adipsic hypernatremia and autoantibodies against the subfornical organ.Frontiers in psychiatry · 2023Article
- Idiopathic partial central diabetes insipidus.Einstein (Sao Paulo, Brazil) · 2023Article
- A rare cause of hypernatremia in an adolescent girl: Answers.Pediatric nephrology (Berlin, Germany) · 2022Article
- Autoimmunity Related to Adipsic Hypernatremia and ROHHAD Syndrome.International journal of molecular sciences · 2022Review
- Central regulation of body fluid homeostasis.Proceedings of the Japan Academy. Series B, Physical and biological sciences · 2022Review
- Severe Hypernatremia in an Adolescent With Anorexia Nervosa.Clinical pediatrics · 2021Article
- Hyponatraemia and hypernatraemia: Disorders of Water Balance in Neurosurgery.Neurosurgical review · 2021Review
- Endocrinological Features of Hartsfield Syndrome in an Adult Patient With a Novel Mutation ofJournal of the Endocrine Society · 2020Article
- Adipsic Diabetes Insipidus-The Challenging Combination of Polyuria and Adipsia: A Case Report and Review of Literature.Frontiers in endocrinology · 2019Article
- Characteristic clinical features of adipsic hypernatremia patients with subfornical organ-targeting antibody.Clinical pediatric endocrinology : case reports and clinical investigations : official journal of the Japanese Society for Pediatric Endocrinology · 2017Article
- Successful treatment of hypodipsic/adipsic hypernatremia in a cat with lobar holoprosencephaly using oral desmopressin.JFMS open reportsArticle
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Authors and funding
19 authors at 5 institutions in 1 country.
Funding
No grant is acknowledged in the PubMed record.
Abstract
Adipsic (or essential) hypernatremia is a rare hypernatremia caused by a deficiency in thirst regulation and vasopressin release. In 2010, we reported a case in which autoantibodies targeting the sensory circumventricular organs (sCVOs) caused adipsic hypernatremia without hypothalamic structural lesions demonstrable by magnetic resonance imaging (MRI); sCVOs include the subfornical organ (SFO) and organum vasculosum of the lamina terminalis (OVLT), which are centers for the monitoring of body-fluid conditions and the control of water and salt intakes, and harbor neurons innervating hypothalamic nuclei for vasopressin release. We herein report three newly identified patients (3- to 8-year-old girls on the first visit) with similar symptoms. The common features of the patients were extensive hypernatremia without any sensation of thirst and defects in vasopressin response to serum hypertonicity. Despite these features, we could not detect any hypothalamic structural lesions by MRI. Immunohistochemical analyses using the sera of the three patients revealed that antibodies specifically reactive to the mouse SFO were present in the sera of all cases; in one case, the antibodies also reacted with the mouse OVLT. The immunoglobulin (Ig) fraction of serum obtained from one patient was intravenously injected into wild-type mice to determine whether the mice developed similar symptoms. Mice injected with a patient's Ig showed abnormalities in water/salt intake, vasopressin release, and diuresis, which resultantly developed hypernatremia. Prominent cell death and infiltration of reactive microglia was observed in the SFO of these mice. Thus, autoimmune destruction of the SFO may be the cause of the adipsic hypernatremia. This study provides a possible explanation for the pathogenesis of adipsic hypernatremia without demonstrable hypothalamus-pituitary lesions.
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