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Published in: BMC Nephrology 1/2018

Open Access 01-12-2018 | Case report

TINU-associated Fanconi syndrome: a case report and review of literature

Authors: Bernard Vô, Jean Cyr Yombi, Selda Aydin, Nathalie Demoulin, Halil Yildiz

Published in: BMC Nephrology | Issue 1/2018

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Abstract

Background

Tubulo-interstitial Nephritis and Uveitis (TINU) syndrome is a rare oculo-renal inflammatory disease. Renal tubular defects are usually found, but full proximal tubular abnormalities have rarely been described.

Case presentation

We report the case of a 55-year old woman, native from Morocco, presenting with bilateral, non-granulomatous, anterior uveitis, mild renal insufficiency, leucocyturia and glycosuria. Further work-up showed hypophosphatemia and hyperphosphaturia, hypouricemia and hyperuricosuria, and hyper aminoaciduria, consistent with Fanconi syndrome. A kidney biopsy was obtained and showed diffuse interstitial infiltrates with tubular necrosis. The patient improved after the initiation of a corticosteroid therapy, with tapering dose.

Conclusions

We reviewed the literature and found nine similar cases. This association mostly occurs in adult woman, without current evidence for an ethnic predilection, unlike previously reported. The renal prognosis seems favorable after corticosteroid therapy, even in case of severe renal injury. Nonetheless mild tubular defects may persist after treatment or spontaneous remission.
Literature
1.
go back to reference Mandeville JT, Levinson RD, Holland GN. The tubulointerstitial nephritis and uveitis syndrome. Surv Ophthalmol. 2001;46(3):195–208.CrossRef Mandeville JT, Levinson RD, Holland GN. The tubulointerstitial nephritis and uveitis syndrome. Surv Ophthalmol. 2001;46(3):195–208.CrossRef
4.
go back to reference Koike K, Lida S, Usui M, Matsumoto Y, Fukami K, Ueda S, Tamaki K, Kato S, Okuda S. Adult-onset acute tubulointerstitial nephritis and uveitis with Fanconi syndrome. Case report and review of the literature. Clin Nephrol. 2007;67(4):255–9.CrossRef Koike K, Lida S, Usui M, Matsumoto Y, Fukami K, Ueda S, Tamaki K, Kato S, Okuda S. Adult-onset acute tubulointerstitial nephritis and uveitis with Fanconi syndrome. Case report and review of the literature. Clin Nephrol. 2007;67(4):255–9.CrossRef
6.
go back to reference Legendre M, Devilliers H, Perard L, Groh M, Nefti H, Dussol B, Trad S, Toure F, Abad S, Boffa JJ, et al. Clinicopathologic characteristics, treatment, and outcomes of tubulointerstitial nephritis and uveitis syndrome in adults: a national retrospective strobe-compliant study. Medicine (Baltimore). 2016;95(26):e3964. https://doi.org/10.1097/MD.0000000000003964.CrossRef Legendre M, Devilliers H, Perard L, Groh M, Nefti H, Dussol B, Trad S, Toure F, Abad S, Boffa JJ, et al. Clinicopathologic characteristics, treatment, and outcomes of tubulointerstitial nephritis and uveitis syndrome in adults: a national retrospective strobe-compliant study. Medicine (Baltimore). 2016;95(26):e3964. https://​doi.​org/​10.​1097/​MD.​0000000000003964​.CrossRef
7.
go back to reference Tanaka H, Waga S, Nakahata T, Suzuki K, Ito T, Onodera N, Iwami S, Monma N, Ito E: Tubulointerstitial nephritis and uveitis syndrome in two siblings. Tohoku J Exp Med 194(1):71–74.CrossRef Tanaka H, Waga S, Nakahata T, Suzuki K, Ito T, Onodera N, Iwami S, Monma N, Ito E: Tubulointerstitial nephritis and uveitis syndrome in two siblings. Tohoku J Exp Med 194(1):71–74.CrossRef
9.
go back to reference Wakaki H, Sakamoto H, Awazu M. Tubulointerstitial nephritis and uveitis syndrome with autoantibody directed to renal tubular cells. Pediatrics. 2001;107(6):1443–6.CrossRef Wakaki H, Sakamoto H, Awazu M. Tubulointerstitial nephritis and uveitis syndrome with autoantibody directed to renal tubular cells. Pediatrics. 2001;107(6):1443–6.CrossRef
12.
go back to reference Takeda M, Hirata M, Koni I, Miyazaki R, Tohuku Y, Takeda R. A case of renal-ocular syndrome with Fanconi syndrome. Nihon Jinzo Gakkai shi. 1988;30(1):103–9.PubMed Takeda M, Hirata M, Koni I, Miyazaki R, Tohuku Y, Takeda R. A case of renal-ocular syndrome with Fanconi syndrome. Nihon Jinzo Gakkai shi. 1988;30(1):103–9.PubMed
13.
go back to reference Lessard M, Smith JD. Fanconi syndrome with uveitis in an adult woman. Am J Kidney Dis. 1989;13(2):158–9.CrossRef Lessard M, Smith JD. Fanconi syndrome with uveitis in an adult woman. Am J Kidney Dis. 1989;13(2):158–9.CrossRef
14.
go back to reference Igarashi T, Kawato H, Kamoshita S, Nosaka K, Seiya K, Hayakawa H. Acute tubulointerstitial nephritis with uveitis syndrome presenting as multiple tubular dysfunction including Fanconi's syndrome. Pediatr Nephrol. 1992;6(6):547–9.CrossRef Igarashi T, Kawato H, Kamoshita S, Nosaka K, Seiya K, Hayakawa H. Acute tubulointerstitial nephritis with uveitis syndrome presenting as multiple tubular dysfunction including Fanconi's syndrome. Pediatr Nephrol. 1992;6(6):547–9.CrossRef
15.
go back to reference Yao YH, Lin CC, Chung YM, Yang AH, Li SY, Lin CC, Lin YP, Yang WC, Yang CY. Tubulointerstitial nephritis and uveitis syndrome (TINU) with Fanconi's syndrome. Clin Nephrol. 2011;75(Suppl 1):75–8.PubMed Yao YH, Lin CC, Chung YM, Yang AH, Li SY, Lin CC, Lin YP, Yang WC, Yang CY. Tubulointerstitial nephritis and uveitis syndrome (TINU) with Fanconi's syndrome. Clin Nephrol. 2011;75(Suppl 1):75–8.PubMed
Metadata
Title
TINU-associated Fanconi syndrome: a case report and review of literature
Authors
Bernard Vô
Jean Cyr Yombi
Selda Aydin
Nathalie Demoulin
Halil Yildiz
Publication date
01-12-2018
Publisher
BioMed Central
Published in
BMC Nephrology / Issue 1/2018
Electronic ISSN: 1471-2369
DOI
https://doi.org/10.1186/s12882-018-1077-0

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