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

作　　者：Eray Eroglu Murat Hayri Sipahioglu Soner Senel Sule Ketenci Ertas Seyma Savas Figen Ozturk Ismail Kocyigit Bulent Tokgoz Oktay Oymak 

机构地区：[1]Division of Nephrology,Department of Internal Medicine,Erciyes University School of Medicine,Kayseri 38039,Turkey [2]Division of Rheumatology,Department of Internal Medicine,Erciyes University School of Medicine,Kayseri 38039,Turkey [3]Department of Internal Medicine,Erciyes University School of Medicine,Kayseri 38039,Turkey [4]Department of Pathology,Erciyes University School of Medicine,Kayseri 38039,Turkey

出　　处：《World Journal of Clinical Cases》2019年第16期2309-2315,共7页世界临床病例杂志

摘　　要：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 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

关 键 词：IMMUNOGLOBULIN G4-related DISEASE Tubulointerstitial NEPHRITIS RITUXIMAB Case report 

分 类 号：R[医药卫生]