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      A Case of Tubulointerstitial Nephritis and Uveitis (TINU) Syndrome with High ASLO Titer

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          Abstract

          Tubulointerstitial nephritis and uveitis (TINU) syndrome is a rare autoimmune disease and the pathogenesis is still unknown. We report a case of TINU syndrome with high ASLO titer.

          Uveitis improved and urine β 2-MG normalized with low dose systemic predonisolone and cyclosporin A. The high ASLO titer in early phase suggested that streptococcal infection might have triggered TINU syndrome. Lymphocyte phenotypes normalized after treatment with low dose systemic predonisolone and cyclosporin A.

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          The tubulointerstitial nephritis and uveitis syndrome.

          The world's medical literature on tubulointerstitial nephritis and uveitis (TINU) syndrome was reviewed, and data on 133 patients with TINU syndrome were identified. The median age of onset was 15 years (range 9-74 years) with a 3:1 female-to-male predominance. Common laboratory abnormalities included elevated Westergren erythrocyte sedimentation rates and elevated urinary beta-2-microglobulin levels. Ocular symptoms preceded systemic symptoms in 21% of cases, and followed systemic symptoms by up to 14 months in 65% of cases. Uveitis involved only the anterior segment in 80% of cases. Uveitis was bilateral at presentation in 77% of cases. Patients were treated with systemic corticosteroids in 80% of cases and with immunosuppressive drugs in 9% of cases. Uveitis recurred or followed a chronic course in 56% of patients and persisted for several years in some cases. Ocular complications (including posterior synechiae, cataracts, and elevated intraocular pressure) were reported in 21% of cases. The visual prognosis appears to be good. Persistent renal dysfunction was reported in 11% of cases, including five patients who required renal dialysis. TINU syndrome is a distinct clinical entity that may be under-recognized and may account for some cases of unexplained chronic or recurrent uveitis. It is important for ophthalmologists, nephrologists, and primary care providers to be familiar with this disorder to ensure early diagnosis and appropriate treatment.
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            Acute eosinophilic interstitial nephritis and renal failure with bone marrow-lymph node granulomas and anterior uveitis. A new syndrome.

            We describe two patients with a unique granulomatous syndrome who presented with renal failure secondary to diffuse eosinophilic interstitial nephritis. Both had bilateral anterior uveitis, bone marrow granulomas, hypergammaglobulinemia and an increased sedimentation rate. One patient had lymph node granulomas and an immunoglobulin G (IgG) rheumatoid factor. An extensive investigation for an etiologic agent was unrewarding, and neither patient could be placed into any existing diagnostic category. Over a period of 2 years both patients have experienced improved renal function and dissolution of their bone marrow granulomas.
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              Course and outcome of tubulointerstitial nephritis and uveitis syndrome.

              We report here the clinical features and outcomes of 10 patients, aged 11 to 21 years (median, 13.0), with idiopathic tubulointerstitial nephritis (TIN) and uveitis syndrome (TINU syndrome). The initial symptoms were visual impairment in 7 patients and prolonged fever, anemia, or asthenia in 4 patients. An increase in urinary beta(2)-microglobulin was noticed at the initial checkup in all patients, including 2 patients who showed the normal ranges of 24-hour protein excretion. Creatinine clearance was decreased in 8 patients. TIN was found simultaneously with ocular symptoms in 7 patients and preceded these symptoms in the remaining 3 patients. Percutaneous renal biopsy indicated tubulointerstitial lesions in varying degrees. The histological grade of TIN was correlated with urinary beta(2)-microglobulin levels. Systemic steroid therapy was performed in 7 patients because of the progression of uveitis. The 10 patients were followed-up for 16 to 94 months (median, 31.0 months). In all patients, creatinine clearance recovered to the normal ranges (>/=70 mL/min/1.73 m(2)) mostly within 1 year. Urinary beta(2)-microglobulin excretion gradually declined but was slightly elevated in 4 patients at the latest checkup. Uveitis recurred in all 10 patients, which did not affect the renal status. Our findings indicate that early referral of patients from ophthalmologists and determination of beta(2)-microglobulin in the urine is helpful for the early discovery of TINU syndrome. In children and adolescents with this syndrome, TIN spontaneously resolves and its long-term prognosis is good, but uveitis often relapses. Systemic steroids may be required for uveitis, but not for TIN.
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                Author and article information

                Journal
                Clin Med Case Rep
                Clin Med Case Rep
                Clinical Medicine. Case Reports
                Libertas Academica
                1178-6450
                2009
                09 April 2009
                : 2
                : 27-30
                Affiliations
                Department of Pediatrics, Tokyo Medical University, Tokyo, Japan.
                Author notes
                Correspondence: Yasuyo Kashiwagi, MD, Department of Pediatrics, Tokyo Medical University, 6-7-1 Nishishinjuku, Shinjuku-ku, Tokyo, Japan. Tel: +81-3-3342-6111; Fax: +81-3-3344-0643; Email: hoyohoyo@ 123456tokyo-med.ac.jp
                Article
                ccrep-2-2009-027
                3785316
                24179369
                6ba351cf-0e7d-4467-8873-76dc0e5d51c0
                © 2009 by the authors

                This article is an open-access article distributed under the terms and conditions of the Creative Commons Attribution license ( http://creativecommons.org/licenses/by/3.0/).

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                Categories
                Case Report

                tinu syndrome,aslo titer,lymphocyte phenotype
                tinu syndrome, aslo titer, lymphocyte phenotype

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