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      Congenital Nephrogenic Diabetes Insipidus Presented With Bilateral Hydronephrosis and Urinary Infection : A Case Report

      research-article
      Author(s): , MD, , MD, , MD
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      Publication date (Electronic):
      Journal: Medicine
      Publisher: Wolters Kluwer Health

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          Abstract

          Nephrogenic diabetes insipidus (NDI) is a condition resulting from the kidney's impaired response to circulating antidiuretic hormone (ADH), leading to polydipsia and polyuria. Urinary tract dilatation caused by NDI is a rare situation. Here, we report a case of congenital NDI presented with bilateral hydronephrosis.

          A 15-year-old boy complaining a history of intermittent fever was admitted to Peking Union Medical College Hospital. He voided 10 to 15 L of urine daily. Radiographic examination revealed severe dilatation of bilateral renal pelvis, ureter, and bladder. Urinalysis shows hyposthenuria.

          He was diagnosed NDI since born. Transient insertion of a urethral catheter helped to relieve fever. Medical therapy of hydrochlorothiazide and amiloride was prescribed and effective.

          Dilatation of urinary tract caused by diabetes insipidus is rare, but may be present in severe condition. Therefore, it is crucial for clinicians to perform early treatment to avoid impairment of renal function.

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          Congenital Nephrogenic Diabetes Insipidus Presented with Bilateral Hydronephrosis: Genetic Analysis of V2R Gene Mutations

          Tae-Hyun Yoo, Dong-Ryeol Ryu, Young Song … (2006)
          Most cases of hydronephrosis are caused by urinary tract obstruction. However, excessive polyuric syndrome rarely gives rise to non-obstructive hydronephrosis, megaureter, and a distended bladder. The authors report here on two cases of congenital nephrogenic diabetes insipidus (NDI) with severe bilateral hydronephrosis and megaureter. It is Interesting that the patients were symptomless except for their polyuria, and they both presented with bilateral hydronephrosis. Fluid deprivation testing revealed the presence of AVP resistant NDI. Gene analysis for these patients showed the AVP receptor 2 (V2R) missense mutations (Q225X and S126F), which have previously been reported on in other studies. We made the diagnosis of NDI by using a physiologic test, and we confirmed it by mutation analysis of the V2R gene.
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            Nephrogenic diabetes insipidus with dilatation of bilateral renal pelvis, ureter and bladder.

            Shan Chen, Liang Cai, Zhao Chen … (2008)
            Nephrogenic diabetes insipidus (NDI) with dilatation of upper urinary tract and bladder is rarely reported. This study evaluated the urological manifestations and treatment results of this rare condition. According to a fluid deprivation test, seven men and two women were diagnosed as NDI, and underwent ultrasonography, magnetic resonance urography, cystography, and urodynamic and laboratory examination. A micturition diary and symptoms were recorded before and during treatment. All patients were given hydrochlorothiazide and followed up with ultrasonography, the postvoiding residual volume (PVR), urinalysis and kidney function tests. All patients presented with polydipsia and polyuria with severe dilatation of upper urinary tract and bladder. Urodynamics showed that the mean values of the bladder capacity, bladder pressure at the end of filling, maximum flow rate (Q(max)), detrusor pressure and PVR were 1056.3 ml, 40.5 cmH(2)O, 30.8 ml/s, 51.3 cmH(2)O, 436.3 ml, respectively. The total urine volume in all patients decreased by more than 50% after administration of hydrochlorothiazide, which improved the symptoms and hydronephrosis in seven cases. The symptoms of other two cases were relieved after treatment with an electric transurethral incision of the bladder neck and a cystostomy. NDI should be considered in patients with dilatation of the urinary tract and polyuria. Higher bladder pressure at the end of filling may contribute to the dilatation of urinary tract. Normal detrusor contractility with large PVR is a unique manifestation of this condition. Controlling the urine volume and reducing the PVR are of key importance in the treatment of this condition.
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              Nonobstructive urinary tract dilatation in children with diabetes insipidus

              Daniel Colliver, Rowland Storey, Hannah Dickens … (2012)
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                Author and article information

                Journal
                Journal ID (nlm-ta): Medicine (Baltimore)
                Journal ID (iso-abbrev): Medicine (Baltimore)
                Journal ID (publisher-id): MEDI
                Title: Medicine
                Publisher: Wolters Kluwer Health
                ISSN (Print): 0025-7974
                ISSN (Electronic): 1536-5964
                Publication date Collection: May 2016
                Publication date (Electronic): 03 June 2016
                Volume: 95
                Issue: 22
                Electronic Location Identifier: e3464
                Affiliations
                From the Department of Urology (KZ), the First Affiliated Hospital of Wenzhou Medical University, Wenzhou Medical University; and Department of Urology (KZ, YX, HL), Peking Union Medical College Hospital, Chinese Academy of Medical Science & Peking Union Medical College, P.R. China.
                Author notes
                Correspondence: Hanzhong Li, Department of Urology, Peking Union Medical College Hospital, 1 Shuai Fu Yuan, Wangfujing, Dongcheng District, Beijing, 100730, P.R. China (e-mail: lihanzhong_pumc@ 123456163.com) .
                Article
                Accession ID: 03464
                DOI: 10.1097/MD.0000000000003464
                PMC ID: 4900698
                PubMed ID: 27258490
                SO-VID: 72bedfa1-357f-4f4d-95d0-a3875c596a01
                Copyright © Copyright © 2016 Wolters Kluwer Health, Inc. All rights reserved.
                License:

                This is an open access article distributed under the Creative Commons Attribution License 4.0, which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited. http://creativecommons.org/licenses/by/4.0

                History
                Date received : 15 December 2015
                Date revision received : 26 March 2016
                Date accepted : 31 March 2016
                Categories
                Subject: 7300
                Subject: Research Article
                Subject: Clinical Case Report
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                OPEN-ACCESS TRUE

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