Adipsic diabetes insipidus (ADI) is a rare and challenging complication secondary to several neurosurgical procedures. It can lead to severe hyperosmolarity and is associated with high rates of morbidity and mortality. Considering the variable time of thirst sensation recovery, long-term treatment is accompanied with the risk of extreme extracellular osmolality fluctuations. Herein, we report 3 cases of this rare presentation of hypothalamic injury. Â© 2017, Nephro-Urology Monthly

Akhtar, M.

Hashemi, S.M.R.

Mollahoseini, R.

Sabzghabaei, F.

eprints Iran University of Medical Sciences

Nephro-Urol Mon. 2018 January; 10(1):e14264.Published online 2017 December 16.doi: 10.5812/numonthly.14264.Case ReportAdipsic Diabetes Insipidus: A Single-Center Case SeriesForoogh Sabzghabaei,1,* Mahsa Akhtar,2 Seyed Mahmoud Ramak Hashemi,3 and Reza Mollahoseini41Assistant Professor of Nephrology, Department of Medicine, Firoozgar Hospital, Tehran, IR Iran2General Physician, Department of Medicine, Firoozgar Hospital, Tehran, IR Iran3Assistant Professor, Department of Neurosurgery, Firoozgar Hospital, Iran University of Medical Sciences, Tehran, IR Iran4Associate Professor, Department of Neurosurgery, Firoozgar Hospital, Iran University of Medical Sciences, Tehran, IR Iran*Corresponding author: Foroogh Sabzghabaei, Assistant Professor of Nephrology, Department of Medicine, Firoozgar Hospital, postal code: 1593748711, Tehran, IR Iran. E-mail:fresh1383@gmail.comReceived 2017 May 29; Revised 2017 August 25; Accepted 2017 November 05.AbstractAdipsic diabetes insipidus (ADI) is a rare and challenging complication secondary to several neurosurgical procedures. It can leadto severe hyperosmolarity and is associated with high rates of morbidity and mortality. Considering the variable time of thirstsensation recovery, long-term treatment is accompanied with the risk of extreme extracellular osmolality fluctuations. Herein, wereport 3 cases of this rare presentation of hypothalamic injury.Keywords: Adipsia, Diabetes Insipidus1. IntroductionThe occurrence of diabetes insipidus (DI) followingneurosurgical procedures for brain tumors may be tran-sient or permanent (1). Adipsia rarely accompanies DI(ADI) due to osmoreceptor destruction. These patientsbecome easily dehydrated and are at a high risk of fluidand electrolyte disorders (2). ADI is often associated withsignificant hypothalamic dysfunction and complications,such as obesity, sleep apnea, thermoregulatory disorders,seizure, venous thromboembolism, and muscle dysfunc-tion (3-5).Herein, we present our experiences in the manage-ment of 3 rare cases of ADI following neurosurgical proce-dures.2. Case Presentation2.1. Case 1A 68-year-old female patient underwent totaltranssphenoidal resection of pituitary adenoma. Onthe first postoperative day, the urine output was about 9L with an osmolality equal to 175 mosmol/kg. After 2 days,the sodium (Na) level increased to 160 - 175 mEq/L (Table 1),while blood pressure remained normal; the patient alsoshowed mild tachycardia. Other serum electrolytes, suchas potassium, calcium, magnesium, and blood sugar, werein the normal range.Despite high serum osmolality and full consciousness,the patient showed no desire to drink water. Desmopressinand fixed water intake were administered with the diag-nosis of ADI. After 3 days, the urine output and Na levelwere found to be in the normal range. In the regular 1-yearfollow-up, no recovery in thirst sensation was observed,whereas the patient showed a relatively stable serum Naand urine output with desmopressin and fixed water in-take.2.2. Case 2A 30-year-old female with headache, blurred vision,and diagnosis of suprasellar craniopharyngioma under-went surgery. On the first postoperative day, she was con-scious and hemodynamically stable with a urine outputof 11 L, Na level of 170 mEq/L (preoperative Na level, 144mEq/L), potassium level of 3.8 mEq/L, and calcium levelof 8.7 mg/dL. She showed no desire to drink water, andtreatment was initiated with desmopressin and regularwater intake, regardless of adipsia. One week later, theurine output was 2400 ml/day, and Na level decreased to122 mEq/L. Subsequently, she started developing a sense ofthirst. Treatment with desmopressin was ceased, and Nalevel recovered to normal (Table 1). After 3 years, she pre-sented with permanent DI, requiring desmopressin to thesame extent.2.3. Case 3A 57-year-old man presenting with severe headachewas admitted with a diagnosis of intracranial hemorrhage.Upon admission, urine osmolality was 140 mosmol/kg,Copyright © 2017, Nephro-Urology Monthly. This is an open-access article distributed under the terms of the Creative Commons Attribution-NonCommercial 4.0International License (http://creativecommons.org/licenses/by-nc/4.0/) which permits copy and redistribute the material just in noncommercial usages, provided theoriginal work is properly cited.Sabzghabaei F et al.Table 1. The Patients’ Characteristics and Laboratory InformationPatient Pre Operation Second Post-Operation Day At DischargeGCS SerumSodium,Meq/LUrineOsmolalitymosm,ol/kgSerumCreatinine,mg/dLGCS SerumSodium,Meq/LUrineOsmolality,Mosmol/kgSerumCreatinine,mg/dLGCS SerumSodium,Meq/LUrineOsmolality,Mosmol/kgSerumCreatinine,mg/dLPatient 1Female 68years15 143 980 1.1 15 177 280 0.9 15 144 490 0.9Patient 2Female 30years15 145 875 0.7 15 159 240 0.8 15 141 385 0.7Patient 3Male 57 years15 130 140 1.2 15 167 235 1.3 15 140 525 1.2urine volume was 7300 mL/24 h, and serum Na level was130 mEq/L. On the following day, he underwent surgery dueto the aneurysm of anterior communicating artery. On thefirst postoperative day, Na level was 144 mEq/L and urinevolume was normal. On the second day, polyuria appearedand Na level started increasing. On the third day, bloodpressure dropped to 90 mmHg, without any evidence ofbleeding or fluid loss. The urine volume decreased, andserum Na level increased to 167 mEq/L.The patient was conscious, but had no thirst sensation.Accordingly, diagnosis of adipsia was confirmed. Treat-ment with intravenous hypoosmolar fluids and pure wa-ter was initiated. After 24 hours, blood pressure was 130/80mmHg, and polyuria with diluted urine appeared (urineosmolality, 240 mosmol/kg). Therapy with desmopressinwas started and after 2 days, thirst sensation recovered andDI disappeared.3. DiscussionADI is a rare disorder, which is generally secondary tohypothalamic lesions (6). It is attributed to the loss ofthirst reflex due to surgical trauma to lamina terminalis,where the osmoreceptor neurons are located (7-9). Limitedstudies have reported a significantly higher risk of morbid-ity and mortality in ADI patients (2, 9, 10). Therefore, earlydiagnosis of this disorder in patients with neurosurgicalinterventions near the hypophysis or hypothalamus is crit-ical.In almost all cases, including our patients, diagnosis isbased on the evaluation of thirst sensation while the pa-tient is awake and hypernatremic. In these cases, water de-privation test is not required for diagnosis. Increased mor-bidity may be due to treatable hypothalamic abnormali-ties, such as obesity, sleep apnea, seizure, and thermoreg-ulatory disorders, which should be examined in patients.One of the limitations of the present study was that we didnot evaluate the patients for these disorders.An interesting point about our patients was the recov-ery time of thirst sensation. In comparison with case No.1, who was diagnosed with permanent pituitary adenomaand adipsia, recovery was faster in the other 2 patients.This finding is in contrast to our speculations and previ-ous reports, which suggest that damage to anterior andposterior hypophysis in craniopharyngioma managementis more severe than in other surgical interventions caus-ing ADI (1). Management of ADI is generally challenging, asthe time of adipsia recovery is variable, and central DI per-sists despite adipsia recovery. Detection of improvementand treatment cessation are important factors since fatalNa disorders may occur in this period.In summary, delayed diagnosis of ADI, given the rarenature of this disease, leads to severe complications of thehyperosmolar state (11). Routine adipsia evaluation, com-bined with water deprivation and vasopressin tests (if nec-essary), is critical for the diagnosis and treatment of ADI.The fluid intake in patients diagnosed with ADI should besupervised daily, based on the constant volume of oral flu-ids, daily measurement of fluid balance, body weight, andsodium level (12), especially in patients who are physicallyunable to care for themselves.3.1. ConclusionsADI is a rare and challenging condition to manage.Therefore, careful monitoring and a high index of suspi-cion are required for its detection. The variable time of re-covery in adipsia and DI is the most important issue, espe-cially in the chronic management of patients.References1. Gonzalez Briceno L, Grill J, Bourdeaut F, Doz F, Beltrand J, BenabbadI, et al. Water and electrolyte disorders at long-term post-treatmentfollow-up in paediatric patients with suprasellar tumours include un-expected persistent cerebral salt-wasting syndrome. Horm Res Paedi-atr. 2014;82(6):364–71. doi: 10.1159/000368401. [PubMed: 25377653].2. Arima H, Wakabayashi T, Nagatani T, Fujii M, Hirakawa A, Murase T,et al. Adipsia increases risk of death in patients with central diabetesinsipidus. Endocr J. 2014;61(2):143–8. [PubMed: 24212879].3. Sabzghabaei F, Rastegar A. Adipsic hypernatremic myopathy. Iran JKidney Dis. 2015;9(3):256–8. [PubMed: 25957431].2 Nephro-Urol Mon. 2018; 10(1):e14264.Sabzghabaei F et al.4. Smith D, McKenna K, Moore K, Tormey W, Finucane J, Phillips J, et al.Baroregulation of vasopressin release in adipsic diabetes insipidus.J Clin Endocrinol Metab. 2002;87(10):4564–8. doi: 10.1210/jc.2002-020090.5. Miljic D, Miljic P, Doknic M, Pekic S, Stojanovic M, Petakov M, etal. Adipsic diabetes insipidus and venous thromboembolism (VTE):recommendations for addressing its hypercoagulability. Hormones(Athens). 2014;13(3):420–3. doi: 10.14310/horm.2002.1496. [PubMed:25079469].6. Verdin E, Smitz S, Thibaut A, Born J, Legros JJ, Luyckx A. Adipsic hy-pernatremia in a patient with pseudotumor cerebri and the primaryempty sella syndrome. J Endocrinol Invest. 1985;8(4):369–72. [PubMed:4067208].7. Macias Batista A, Martinez Martin FJ, de Pablos Velasco PL. [Diabetes in-sipidus and adipsic hypernatremia in a patient with a craniopharyn-gioma]. AnMed Interna. 1999;16(2):87–8. [PubMed: 10193001].8. Colleran K, Sanchez Goettler L, Sloan A. Hypothalamic obesity com-plicated by adipsic central diabetes insipidus following surgical re-section of a craniopharyngioma. J Clin Hypertens. 2009;11(10):608–10.doi: 10.1111/j.1751-7176.2009.00057.x.9. Raghunathan V, Dhaliwal MS, Gupta A, Jevalikar G. From cerebral saltwasting to diabetes insipidus with adipsia, case report of a child withcraniopharyngioma. J Pediatr Endocrinol Metab. 2015;28(3):323–6. doi:10.1515/jpem-2014-0224.10. Zantut-Wittmann DE, Garmes HM, Panzan AD, Lima Mde O, BaptistaMT. Severe rhabdomyolysis due to adipsic hypernatremia after cran-iopharyngioma surgery.ArqBrasEndocrinolMetabol. 2007;51(7):1175–9.[PubMed: 18157396].11. Hu MM, Liu M, Liu W. [Diagnosis and treatment of adipsic diabetesinsipidus accompanied with intracranial calcification]. Zhongguo YiXue Ke Xue Yuan Xue Bao. 2013;35(2):161–5. doi: 10.3881/j.issn.1000-503X.2013.02.007. [PubMed: 23643004].12. Janus DM, Wojcik M, Zygmunt-Gorska A, Wyrobek L, Urbanik A,Starzyk JB. Adipsic diabetes insipidus in pediatric patients. Indian JPediatr. 2014;81(12):1307–14. doi: 10.1007/s12098-014-1421-8. [PubMed:24788913].Nephro-Urol Mon. 2018; 10(1):e14264. 3

Adipsic diabetes insipidus: A single-center case series

http://eprints.iums.ac.ir/6980/1/Adipsic%20diabetes%20insipidus%20A%20single-center%20case%20series.pdf

Adipsic diabetes insipidus: A single-center case series

Abstract

Similar works

Full text

Available Versions

eprints Iran University of Medical Sciences