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Successful treatment of tubulointerstitial nephritis in immunoglobulin G4-related disease with rituximab: A case report

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World Journal of Clinical Cases 2019年第16期7卷 2309-2315页

作者： Eray Eroglu Murat Hayri Sipahioglu Soner Senel Sule Ketenci Ertas Seyma Savas figen ozturk Ismail Kocyigit Bulent Tokgoz Oktay Oymak Division of Nephrology Department of Internal MedicineErciyes University School of MedicineKayseri 38039Turkey Division of Rheumatology Department of Internal MedicineErciyes University School of MedicineKayseri 38039Turkey Department of Internal Medicine Erciyes University School of MedicineKayseri 38039Turkey Department of Pathology Erciyes University School of MedicineKayseri 38039Turkey

BACKGROUND Immunoglobulin G4-related disease (IgG4-RD) is an immune-mediated condition that consisted of disorders that share particular clinical,serologic and pathologic properties.The common presentation of disease includes tumor-like swelling of involved organs and the histopathological findings are a lymphoplasmacytic infiltrate enriched with IgG4-positive plasma cells,and a variable degree of fibrosis that has a characteristic "storiform" pattern in biopsy specimens of tumorlike masses.Major presentations of this disease,which often affects more than one organ,include autoimmune pancreatitis,salivary gland disease (sialadenitis),orbital disease and retroperitoneal fibrosis.The steroid treatment is essential for the treatment of the disease however,other immunosuppressive drugs including cyclophosphamide or rituximab could be an option in resistant cases.CASE SUMMARY Herein,we reported a 34-year-old woman whom previously had diagnosed with asthma,rheumatoid arthritis and Sj?gren’s syndrome (SS) referred our nephrology department due to acute kidney failure development at the last rheumatology visit.After kidney biopsy she has been diagnosed with IgG4-RD and tubuluointerstitial nephritis.She had been accepted resistant to steroid,mycophenolate mofetil,methotrexate and azathioprine therapies due to receiving in last two years.She refused to receive cyclophosphamide due to potential gonadotoxicity of the drug.Thus,rituximab therapy was considered.She received 1000 mg infusion,15 d apart and 6 mo later it has been administered same protocol.After one year from the last rituximab dose serum creatinine decreased from 4.4 mg/dL to 1.6 mg/dL,erythrocyte sedimentation rate decreased from 109 mm/h to 13 mm/h [reference range (RR) 0-20],and Creactive protein decreased from 55.6 mg/L to 5 mg/L (RR 0–6).All pathologic lymph nodes and masses were also disappeared.CONCLUSION Patients with IgG4-RD usually misdiagnosed with rheumatologic diseases including systemic lupus erythematous or SS and also they were screened for the

关键词： Immunoglobulin G4-related disease Tubulointerstitial nephritis Rituximab Case report

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