A case report of tubulointerstitial nephritis and uveitis syndrome in children with an unfavorable outcome
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Tubulointerstitial nephritis and uveitis (TINU) syndrome is defined as the occurrence of tubulointerstitial nephritis and uveitis in the absence of other systemic diseases. Three pediatric cases have been reported in the Republic of Korea, and we now report a fourth case. A 15-year-old girl presented to the ophthalmology department with a 1-week history of bilateral ocular discomfort that worsened on the day of presentation with redness and pain in both eyes. She was diagnosed with bilateral uveitis, and her baseline examination revealed moderate renal dysfunction and mild proteinuria. A renal biopsy was performed and confirmed the diagnosis of TINU syndrome. She was started on steroid eye drops and a 12-week course of oral steroids at a dose of 40 mg/m2/day, which completely resolved the proteinuria and mild renal function to an estimated glomerular filtration rate of 60 mL/min/1.73 m2. However, the uveitis did not improve, and despite the addition of oral methotrexate as a second-line treatment, the uveitis remains unresponsive to treatment over 21 months. Further evaluation and treatment are ongoing, and active therapeutic intervention is suggested even at a pediatric age, considering the lack of improvement in renal function and uveitis to date.
Abstract
Tubulointerstitial nephritis and uveitis (TINU) syndrome is defined as the occurrence of tubulointerstitial nephritis and uveitis in the absence of other systemic diseases. Three pediatric cases have been reported in the Republic of Korea, and we now report a fourth case. A 15-year-old girl presented to the ophthalmology department with a 1-week history of bilateral ocular discomfort that worsened on the day of presentation with redness and pain in both eyes. She was diagnosed with bilateral uveitis, and her baseline examination revealed moderate renal dysfunction and mild proteinuria. A renal biopsy was performed and confirmed the diagnosis of TINU syndrome. She was started on steroid eye drops and a 12-week course of oral steroids at a dose of 40 mg/m2/day, which completely resolved the proteinuria and mild renal function to an estimated glomerular filtration rate of 60 mL/min/1.73 m2. However, the uveitis did not improve, and despite the addition of oral methotrexate as a second-line treatment, the uveitis remains unresponsive to treatment over 21 months. Further evaluation and treatment are ongoing, and active therapeutic intervention is suggested even at a pediatric age, considering the lack of improvement in renal function and uveitis to date.
참고문헌 (13)
- Dobrin RS, "Acute eosinophilic interstitial nephritis and renal failure with bone marrow-lymph node granulomas and anterior uveitis : a new syndrome", Am J Med, 59, pp.325~333 (1975)
- Okafor LO, "Tubulointerstitial nephritis and uveitis (TINU) syndrome: a systematic review of its epidemiology, demographics and risk factors", Orphanet J Rare Dis, 12, pp.128~ (2017)
- Mandeville JT, "The tubulointerstitial nephritis and uveitis syndrome", Surv Ophthalmol, 46, pp.195~208 (2001)
- Regusci A, "Tubulointerstitial nephritis and uveitis syndrome : a systematic review", Nephrol Dial Transplant, 37, pp.876~886 (2022)
- Clive DM, "The syndrome of tubulointerstitial nephritis with uveitis(TINU)", Am J Kidney Dis, 72, pp.118~128 (2018)
- Mortajil F, "Acute tubulointerstitial nephritis and anterior uveitis(TINU syndrome) : a report of two cases", Saudi J Kidney Dis Transpl, 17, pp.386~389 (2006)
- Li C, "Tubulointerstitial nephritis with uveitis in Chinese adults", Clin J Am Soc Nephrol, 9, pp.21~28 (2014)
- Mackensen F, "Enhanced recognition, treatment, and prognosis of tubulointerstitial nephritis and uveitis syndrome", Ophthalmology, 114, pp.995~999 (2007)
- Hahn HW, "A case of tubulointerstitial nephritis and uveitis(TINU)", J Korean Soc Pediatr Nephrol, 4, pp.166~169 (2000)
- Ji Eun Kim, "Successful treatment of tubulointerstitial nephritis and uveitis with steroid and azathioprine in a 12-year- old boy", Clinical and Experimental Pediatrics, 59, pp.99~102 (2016), 대한소아청소년과학회
- Lee JW, "A case of tubulointerstitial nephritis and uveitis syndrome with severe immunologic dysregulation", Pediatr Nephrol, 20, pp.1805~1808 (2005)
- Saarela V, "Tubulointerstitial nephritis and uveitis syndrome in children : a prospective multicenter study", Ophthalmology, 120, pp.1476~1481 (2013)
- Rytkonen S, "Long-term outcome of biopsy-proven idiopathic tubulointersitial nephritis with or without uveitis in children-a nationwide follow-up study", Pediatr Nephrol, 36, pp.3663~3671 (2021)