Case Report: Successful Treatment of Steroid-Refractory Immune Checkpoint Inhibitor-Related Pure Red Cell Aplasia With Cyclosporin
Anemia associated with Immune checkpoint inhibitor (ICI) is usually hemolytic and regenerative. Cases of non-regenerative pure red cell aplasia are rare, and typically improve upon drug discontinuation and after corticotherapy. We herein report a case of nivolumab-related erythroblastopenia refracto...
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| Format: | Article |
| Language: | English |
| Published: |
Frontiers Media S.A.
2020-08-01
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| Series: | Frontiers in Oncology |
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| Online Access: | https://www.frontiersin.org/article/10.3389/fonc.2020.01760/full |
| _version_ | 1829484451709583360 |
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| author | Alexandre Gérard Serena Romani Elise Van-Obberghen Audrey Fresse Marine Muzzone Nadège Parassol Annick Boscagli Fanny Rocher Delphine Borchiellini Milou-Daniel Drici |
| author_facet | Alexandre Gérard Serena Romani Elise Van-Obberghen Audrey Fresse Marine Muzzone Nadège Parassol Annick Boscagli Fanny Rocher Delphine Borchiellini Milou-Daniel Drici |
| author_sort | Alexandre Gérard |
| collection | DOAJ |
| description | Anemia associated with Immune checkpoint inhibitor (ICI) is usually hemolytic and regenerative. Cases of non-regenerative pure red cell aplasia are rare, and typically improve upon drug discontinuation and after corticotherapy. We herein report a case of nivolumab-related erythroblastopenia refractory to steroids in a melanoma patient that improved only after treatment with cyclosporin. Nivolumab had been well tolerated for 2 months after being introduced as an adjuvant treatment. Hemoglobin level then progressively decreased from 12.7 g/dl as baseline value to a nadir of 4.3 g/dL despite transfusion with a total of 29 packed red blood cells in 3 months. Extensive workup including repeated bone marrow examinations led to the diagnosis of pure red cell aplasia. Anemia persisted despite nivolumab discontinuation and over a month of corticotherapy, but improved dramatically 3 days after cyclosporin initiation and did not recur upon cyclosporin tapering. The patient remains cancer-free 9 months after nivolumab withdrawal. This case highlights the under-recognized risk of erythroblastopenia in patients treated with ICI and proves cyclosporin is a valid alternative for the treatment of steroid-refractory cases. |
| first_indexed | 2024-12-14T22:25:52Z |
| format | Article |
| id | doaj.art-1d0e62a7917347e6ac6cc8f071546d5f |
| institution | Directory Open Access Journal |
| issn | 2234-943X |
| language | English |
| last_indexed | 2024-12-14T22:25:52Z |
| publishDate | 2020-08-01 |
| publisher | Frontiers Media S.A. |
| record_format | Article |
| series | Frontiers in Oncology |
| spelling | doaj.art-1d0e62a7917347e6ac6cc8f071546d5f2022-12-21T22:45:21ZengFrontiers Media S.A.Frontiers in Oncology2234-943X2020-08-011010.3389/fonc.2020.01760579824Case Report: Successful Treatment of Steroid-Refractory Immune Checkpoint Inhibitor-Related Pure Red Cell Aplasia With CyclosporinAlexandre Gérard0Serena Romani1Elise Van-Obberghen2Audrey Fresse3Marine Muzzone4Nadège Parassol5Annick Boscagli6Fanny Rocher7Delphine Borchiellini8Milou-Daniel Drici9Pharmacovigilance, Department of Clinical Pharmacology, Pasteur Hospital, Centre Hospitalier Universitaire de Nice, Université Côte d’Azur, Nice, FrancePharmacovigilance, Department of Clinical Pharmacology, Pasteur Hospital, Centre Hospitalier Universitaire de Nice, Université Côte d’Azur, Nice, FrancePharmacovigilance, Department of Clinical Pharmacology, Pasteur Hospital, Centre Hospitalier Universitaire de Nice, Université Côte d’Azur, Nice, FrancePharmacovigilance, Department of Clinical Pharmacology, Pasteur Hospital, Centre Hospitalier Universitaire de Nice, Université Côte d’Azur, Nice, FrancePharmacovigilance, Department of Clinical Pharmacology, Pasteur Hospital, Centre Hospitalier Universitaire de Nice, Université Côte d’Azur, Nice, FrancePharmacovigilance, Department of Clinical Pharmacology, Pasteur Hospital, Centre Hospitalier Universitaire de Nice, Université Côte d’Azur, Nice, FranceDepartment of Medical Oncology, Centre Antoine Lacassagne, Université Côte d’Azur, Nice, FrancePharmacovigilance, Department of Clinical Pharmacology, Pasteur Hospital, Centre Hospitalier Universitaire de Nice, Université Côte d’Azur, Nice, FranceDepartment of Medical Oncology, Centre Antoine Lacassagne, Université Côte d’Azur, Nice, FrancePharmacovigilance, Department of Clinical Pharmacology, Pasteur Hospital, Centre Hospitalier Universitaire de Nice, Université Côte d’Azur, Nice, FranceAnemia associated with Immune checkpoint inhibitor (ICI) is usually hemolytic and regenerative. Cases of non-regenerative pure red cell aplasia are rare, and typically improve upon drug discontinuation and after corticotherapy. We herein report a case of nivolumab-related erythroblastopenia refractory to steroids in a melanoma patient that improved only after treatment with cyclosporin. Nivolumab had been well tolerated for 2 months after being introduced as an adjuvant treatment. Hemoglobin level then progressively decreased from 12.7 g/dl as baseline value to a nadir of 4.3 g/dL despite transfusion with a total of 29 packed red blood cells in 3 months. Extensive workup including repeated bone marrow examinations led to the diagnosis of pure red cell aplasia. Anemia persisted despite nivolumab discontinuation and over a month of corticotherapy, but improved dramatically 3 days after cyclosporin initiation and did not recur upon cyclosporin tapering. The patient remains cancer-free 9 months after nivolumab withdrawal. This case highlights the under-recognized risk of erythroblastopenia in patients treated with ICI and proves cyclosporin is a valid alternative for the treatment of steroid-refractory cases.https://www.frontiersin.org/article/10.3389/fonc.2020.01760/fullimmune checkpoint inhibitornivolumabanemiapure red cell aplasiasteroid-refractorycyclosporin |
| spellingShingle | Alexandre Gérard Serena Romani Elise Van-Obberghen Audrey Fresse Marine Muzzone Nadège Parassol Annick Boscagli Fanny Rocher Delphine Borchiellini Milou-Daniel Drici Case Report: Successful Treatment of Steroid-Refractory Immune Checkpoint Inhibitor-Related Pure Red Cell Aplasia With Cyclosporin Frontiers in Oncology immune checkpoint inhibitor nivolumab anemia pure red cell aplasia steroid-refractory cyclosporin |
| title | Case Report: Successful Treatment of Steroid-Refractory Immune Checkpoint Inhibitor-Related Pure Red Cell Aplasia With Cyclosporin |
| title_full | Case Report: Successful Treatment of Steroid-Refractory Immune Checkpoint Inhibitor-Related Pure Red Cell Aplasia With Cyclosporin |
| title_fullStr | Case Report: Successful Treatment of Steroid-Refractory Immune Checkpoint Inhibitor-Related Pure Red Cell Aplasia With Cyclosporin |
| title_full_unstemmed | Case Report: Successful Treatment of Steroid-Refractory Immune Checkpoint Inhibitor-Related Pure Red Cell Aplasia With Cyclosporin |
| title_short | Case Report: Successful Treatment of Steroid-Refractory Immune Checkpoint Inhibitor-Related Pure Red Cell Aplasia With Cyclosporin |
| title_sort | case report successful treatment of steroid refractory immune checkpoint inhibitor related pure red cell aplasia with cyclosporin |
| topic | immune checkpoint inhibitor nivolumab anemia pure red cell aplasia steroid-refractory cyclosporin |
| url | https://www.frontiersin.org/article/10.3389/fonc.2020.01760/full |
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