TY - JOUR
T1 - Antineutrophil cytoplasmic antibody- associated renal vasculitis treated with autologous mesenchymal stromal cells
T2 - Evaluation of the contribution of immune-mediated mechanisms
AU - Gregorini, Marilena
AU - Maccario, Rita
AU - Avanzini, Maria Antonietta
AU - Corradetti, Valeria
AU - Moretta, Antonia
AU - Libetta, Carmelo
AU - Esposito, Pasquale
AU - Bosio, Francesca
AU - Dal Canton, Antonio
AU - Rampino, Teresa
PY - 2013
Y1 - 2013
N2 - We report the first case of renal antineutrophil cytoplasmic antibody (ANCA)eassociated vasculitis treated with autologous mesenchymal stromal cells (MSCs). A 73-year-old man was admitted to the hospital for malaise, weight loss, and oliguria. His serum creatinine level was 2.7 mg/dL but it rapidly increased to 7.8 mg/dL; urinalysis showed proteinuria and hematuria, and the ANCA to myeloperoxidase with a perinuclear pattern (pANCA) titer was high (132 IU/mL). Renal biopsy showed necrotizing crescentic glomerulonephritis. Standard immunosuppressive therapy (cyclophosphamide and corticosteroids) was ineffective. Rituximab therapy was started, but it was discontinued after the third dose to minimize the risk of systemic spread of a severe oral Candida infection and to prevent superinfections that were facilitated by leukopenia. The patient received autologous MSCs, 1.5 × 106 cells/kg body weight, intravenously. After 7 days, his serum creatinine level decreased to 2.2 mg/dL, pANCA titer decreased to 75 IU/mL, and urinalysis findings normalized. Eight months later, he received a second MSC infusion because his serum creatinine level increased. In 1 week, his creatinine level decreased to 1.9 mg/dL and his pANCA titer decreased to 14 IU/mL. Immunosuppressive therapy was subsequently withdrawn. At the last follow-up visit, 12 months after the second MSC infusion, the patient remained in clinical remission without any therapy. Infusion of MSCs induced expansion of the T-lymphocyte subset expressing a regulatory T-cell phenotype (CD4+CD25+Foxp3+) and a notable reduction in interferon-g, interleukin 6, and tumor necrosis factor serum levels.
AB - We report the first case of renal antineutrophil cytoplasmic antibody (ANCA)eassociated vasculitis treated with autologous mesenchymal stromal cells (MSCs). A 73-year-old man was admitted to the hospital for malaise, weight loss, and oliguria. His serum creatinine level was 2.7 mg/dL but it rapidly increased to 7.8 mg/dL; urinalysis showed proteinuria and hematuria, and the ANCA to myeloperoxidase with a perinuclear pattern (pANCA) titer was high (132 IU/mL). Renal biopsy showed necrotizing crescentic glomerulonephritis. Standard immunosuppressive therapy (cyclophosphamide and corticosteroids) was ineffective. Rituximab therapy was started, but it was discontinued after the third dose to minimize the risk of systemic spread of a severe oral Candida infection and to prevent superinfections that were facilitated by leukopenia. The patient received autologous MSCs, 1.5 × 106 cells/kg body weight, intravenously. After 7 days, his serum creatinine level decreased to 2.2 mg/dL, pANCA titer decreased to 75 IU/mL, and urinalysis findings normalized. Eight months later, he received a second MSC infusion because his serum creatinine level increased. In 1 week, his creatinine level decreased to 1.9 mg/dL and his pANCA titer decreased to 14 IU/mL. Immunosuppressive therapy was subsequently withdrawn. At the last follow-up visit, 12 months after the second MSC infusion, the patient remained in clinical remission without any therapy. Infusion of MSCs induced expansion of the T-lymphocyte subset expressing a regulatory T-cell phenotype (CD4+CD25+Foxp3+) and a notable reduction in interferon-g, interleukin 6, and tumor necrosis factor serum levels.
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U2 - 10.1016/j.mayocp.2013.06.021
DO - 10.1016/j.mayocp.2013.06.021
M3 - Article
C2 - 24079687
AN - SCOPUS:84888631332
VL - 88
SP - 1174
EP - 1179
JO - Mayo Clinic Proceedings
JF - Mayo Clinic Proceedings
SN - 0025-6196
IS - 10
ER -