Pure red cell aplasia induced by anti-erythropoietin antibodies, well-controlled with tacrolimus

Abstract

Anti-erythropoietin (anti-EPO) antibody-related pure red cell aplasia (PRCA) is a rare but serious complication that can occur during the administration of erythropoiesis-stimulating agents. Treatment with the calcineurin inhibitor cyclosporine has shown benefits in patients with anti-EPO PRCA. The efficacy of tacrolimus, another calcineurin inhibitor, in patients with anti-EPO PRCA has not been determined. The present report is the first our knowledge to describe the successful treatment of a patient with anti-EPO PRCA using tacrolimus. A 73-year-old man was markedly anemic 8 months after starting epoetin beta treatment. He was diagnosed with anti-EPO PRCA. Cyclosporine was started, but he experienced side effects. He was switched from cyclosporine to tacrolimus. No side effects were observed, and his anti-EPO PRCA was improved 6 months later, despite the persistence of anti-EPO antibodies. Tacrolimus was continued until the disappearance of the antibodies. Following the cessation of tacrolimus, PRCA did not relapse. Antibody remained detectable at the time of clinical remission, indicating that immunosuppressive therapy should be continued while monitoring the antibody titer. When the antibody titer decreases to the negative range, cessation of the immunosuppressive therapy does not result in disease relapse.

Rational management approach to pure red cell aplasia

“…T-LGL-associated PRCA. Tacrolimus showed activity as a salvage option in a few reports, 37 , 38 and we were able to substantiate those findings in our study. While no head-to-head comparisons were conducted, previous reports suggested lower response rates to CTX vs .…”

Case report: Dynamic antibody monitoring in a case of anti-recombinant human erythropoietin-mediated pure red cell aplasia with prolonged course after kidney transplantation

“…Recently, studies had recommended initial treatment, including cyclosporine or cyclophosphamide combined with prednisone in PRCA ( 9 , 11 , 20 ). Tacrolimus may be considered as a substitute for cyclosporine ( 21 ). Chen et al.…”

“…Recently, studies had recommended initial treatment, including cyclosporine or cyclophosphamide combined with prednisone in PRCA (9,11,20). Tacrolimus may be considered as a substitute for cyclosporine (21). Chen et al also reported the therapeutic effect of sirolimus in PRCA patients with a complete response of 58.3% and a median time of 4 (1-7) months to achieve the optimal effect (22).…”

Antibody-mediated pure red cell aplasia related with epoetin-beta pegol (C.E.R.A.) as an erythropoietic agent: case report of a dialysis patient

“…Especially, the peak incidence of PRCA related to ESA therapy occurred during 2002–2003 [ 1 ]. The majority of those cases were caused by a preparation of epoetin-alfa (Eprex/Erypo) [ 5 , 6 ], though a small case series caused by epoetin-beta was also reported [ 7 – 10 ]. This transient increase of PRCA related to Eprex during 2002–2003 was associated with the use of the polysorbate-80(PS-80) formulation in prefilled syringes with uncoated rubber stoppers [ 6 , 11 ].…”