title: Heterogenous patterns of recovery of thirst in adult patients with adipsic diabetes insipidus.
abstract: BACKGROUND
  The natural history of adipsic diabetes insipidus (ADI) is not well described, and reports of recovery of thirst are rare.
  
  
  METHODS
  Case histories presentation. ADI was identified by demonstrating absent thirst and arginine vasopressin (AVP) responses to hypertonic saline infusion.
  
  
  RESULTS
  Twelve patients with ADI were identified (craniopharyngioma 5, anterior communicating artery aneurysm (ACOM) repair 4, congenital 1, neurosarcoidosis 1, prolactinoma 1). Three patients died. Six patients had permanent ADI. Three patients had recovery of thirst, with a heterogenous pattern of recovery. In the first case, ADI had developed after clipping of an ACOM aneurysm. Ten years after surgery; he sensed the return of thirst; repeated hypertonic saline infusion showed recovery of thirst and AVP secretion. In the second case, a 41-year-old female with an intrasellar craniopharyngioma developed post-operative ADI with persistent hypernatremia. Two years post-operatively, she complained of thirst, and hypertonic saline infusion showed normalization of thirst but absent AVP responses, confirming recovery of thirst, but with persistent diabetes insipidus (DI). In the third case, a 29-year-old Caucasian had craniotomy and radiotherapy for craniopharyngioma and developed ADI post-operatively. Eight years post-op, she presented with thirst, seizures and pNa of 112 mmol/l. Hypertonic saline infusion showed persistent DI but thirst responses typical of compulsive water drinking; she has had recurrent hyponatraemia since then.
  
  
  CONCLUSIONS
  We report that 3/12 patients with ADI recovered thirst after longstanding adipsia with heterogenous pattern of recovery. Both the mortality of 25% and the recovery rate of 25% should be considered when planning long-term surveillance.
authors.affiliation: From the Academic Department of Endocrinology, Beaumont Hospital/RCSI Medical School, Dublin, Ireland and.
authors.forename: M
authors.initials: M
authors.lastname: Cuesta
authors.affiliation: From the Academic Department of Endocrinology, Beaumont Hospital/RCSI Medical School, Dublin, Ireland and.
authors.forename: S
authors.initials: S
authors.lastname: Gupta
authors.affiliation: From the Academic Department of Endocrinology, Beaumont Hospital/RCSI Medical School, Dublin, Ireland and.
authors.forename: R
authors.initials: R
authors.lastname: Salehmohamed
authors.affiliation: From the Academic Department of Endocrinology, Beaumont Hospital/RCSI Medical School, Dublin, Ireland and.
authors.forename: R
authors.initials: R
authors.lastname: Dineen
authors.affiliation: From the Academic Department of Endocrinology, Beaumont Hospital/RCSI Medical School, Dublin, Ireland and.
authors.forename: M J
authors.initials: MJ
authors.lastname: Hannon
authors.affiliation: Academic Department of Chemical Pathology, Beaumont Hospital/RCSI Medical School, Dublin, Ireland.
authors.forename: W
authors.initials: W
authors.lastname: Tormey
authors.affiliation: From the Academic Department of Endocrinology, Beaumont Hospital/RCSI Medical School, Dublin, Ireland and christhompson@beaumont.ie.
authors.forename: C J
authors.initials: CJ
authors.lastname: Thompson
mesh_terms: D000328:Adult; D001127:Arginine Vasopressin / Q000378:metabolism*; D003681:Dehydration / Q000209:etiology / Q000503:physiopathology* / Q000628:therapy; D020790:Diabetes Insipidus, Neurogenic / Q000150:complications / Q000188:drug therapy / Q000503:physiopathology*; D005260:Female; D006801:Humans; D007010:Hyponatremia / Q000209:etiology / Q000503:physiopathology* / Q000628:therapy; D007262:Infusions, Intravenous; D008297:Male; D008875:Middle Aged; D020127:Recovery of Function / Q000502:physiology*; D012462:Saline Solution, Hypertonic / Q000627:therapeutic use*; D013894:Thirst / Q000502:physiology*; D016896:Treatment Outcome; D014883:Water-Electrolyte Imbalance
chemical_list: D012462:Saline Solution, Hypertonic; D001127:Arginine Vasopressin