Effect of tacrolimus in a patient with pure red-cell aplasia

Yoshida, Shigetoshi; Konishi, Tetsuro; Nishizawa, Toshihiro; Yoshida, Yataro

doi:10.1111/j.1365-2257.2004.00654.x

Cited by 13 publications

(6 citation statements)

References 13 publications

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“…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 .…”

Section: Discussionsupporting

confidence: 87%

Rational management approach to pure red cell aplasia

et al. 2017

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Pure red cell aplasia is an orphan disease, and as such lacks rationally established standard therapies. Most cases are idiopathic; a subset is antibody-mediated. There is overlap between idiopathic cases and those with T-cell large granular lymphocytic leukemia, hypogammaglobulinemia, and low-grade lymphomas. In each of the aforementioned, the pathogenetic mechanisms may involve autoreactive cytotoxic responses. We selected 62 uniformly diagnosed pure red cell aplasia patients and analyzed their pathophysiologic features and responsiveness to rationally applied first-line and salvage therapies in order to propose diagnostic and therapeutic algorithms that may be helpful in guiding the management of prospective patients, 52% of whom were idiopathic, while the others involved large granular lymphocytic leukemia, thymoma, and B-cell dyscrasia. T-cell-mediated responses ranged between a continuum from polyclonal to monoclonal (as seen in large granular lymphocytic leukemia). During a median observation period of 40 months, patients received a median of two different therapies to achieve remission. Frequently used therapy included calcineurin-inhibitors with a steroid taper yielding a first-line overall response rate of 76% (53/70). Oral cyclophosphamide showed activity, albeit lower than that produced by cyclosporine. Intravenous immunoglobulins were effective both in parvovirus patients and in hypogammaglobulinemia cases. In salvage settings, alemtuzumab is active, particularly in large granular lymphocytic leukemia-associated cases. Other potentially useful salvage options include rituximab, anti-thymocyte globulin and bortezomib. The workup of acquired pure red cell aplasia should include investigations of common pathological associations. Most effective therapies are directed against T-cell-mediated immunity, and therapeutic choices need to account for associated conditions that may help in choosing alternative salvage agents, such as intravenous immunoglobulin, alemtuzumab and bortezomib.

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Section: Discussionsupporting

confidence: 87%

Rational management approach to pure red cell aplasia

et al. 2017

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“…Several reports have indicated that in PRCA T lymphocytes play a key role in the production of autoantibodies directed to suppress erythropoiesis (1)(2)(3), and CsA has recently become accepted as the first-line drug for treatment of this disease in Japan (4,5). Tacrolimus can also be used for treatment of PRCA because of the mechanism similar to CsA with selective suppression of the activation of T lymphocytes by acting as a calcineurin inhibitor (6), but our patient was successfully treated with the latter but not with the former. Although the precise reason is unclear, a difference in the intracytoplasmic binding protein between CsA and tacrolimus may be related to these therapeutic outcomes (7).…”

Section: Pure Red Cell Aplasia (Prca) Is An Autoimmune Disorder Charamentioning

confidence: 99%

Pure Red Cell Aplasia Developing after Treatment of Pleural Recurrence of Thymoma, Successfully Treated with Cyclosporin A but not with Tacrolimus

et al. 2006

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“…Cyclosporine is reportedly effective in patients with PRCA secondary to thymoma, whereas tacrolimus is not [9]. Although tacrolimus was later reported effective in patients with thymoma-associated PRCA [12], the effectiveness of tacrolimus therapy in patients with PRCA remains unclear. There have been no reports of tacrolimus therapy for anti-EPO PRCA.…”

Section: Introductionmentioning

confidence: 98%

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

Hashimoto

Harada

Kamijo³

2016

Int J Hematol

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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.

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Effect of tacrolimus in a patient with pure red-cell aplasia

Cited by 13 publications

References 13 publications

Rational management approach to pure red cell aplasia

Rational management approach to pure red cell aplasia

Pure Red Cell Aplasia Developing after Treatment of Pleural Recurrence of Thymoma, Successfully Treated with Cyclosporin A but not with Tacrolimus

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

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