Lobbes Hervé
Service de médecine interne, Hôpital Estaing, Centre Hospitalier Universitaire de Clermont-Ferrand, Clermont-Ferrand, France; Université Clermont Auvergne, CHU Clermont-Ferrand, CNRS, Institut Pascal, 63000 Clermont-Ferrand, France.
Rev Med Interne. 2023 Jan;44(1):19-26. doi: 10.1016/j.revmed.2022.10.385. Epub 2022 Nov 3.
Pure red cell aplasia (PRCA) is a rare anemia characterised by profound reticulocytopenia caused by a marked reduction in bone marrow erythroblasts, without abnormalities in other blood lineages. Blackfan-Diamond anemia is an inherited ribosomopathy responsible for a hereditary form of PRCA. Acquired PRCA are separated in primary and secondary forms, including Parvovirus B19 infection, thymoma, lymphoproliferative disorders, autoimmune diseases (lupus) and drug-induced PRCA. The pathophysiology of PRCA is not fully understood and involves both humoral and T lymphocyte autoreactive cells. In Parvovirus B19-related PRCA, treatment is based on polyvalent immunoglobulins. Thymectomy for thymoma is mandatory but results in prolonged remission in a limited number of cases. The therapeutic strategy is based on expert opinion: corticosteroids in monotherapy provide few sustained responses. The choice of an additional immunosuppressant drug is guided by the presence of an underlying disease. In most cases, cyclosporine A is the first choice providing the best response rate but requires a concentration monitoring (150 to 250 ng/mL). The second choice is cyclophosphamide in large granular lymphocyte leukaemia. Sirolimus (mTOR inhibitor) seems to be a promising option especially in refractory cases. Transfusion independence is the main objective. If the patient receives numerous red blood cell transfusions (> 20 packs), iron overload assessment is crucial to initiate an iron chelation. A retrospective and prospective national cohort (EPIC-F) has been set up and is now available to include each case of PRCA to improve the knowledge of this disease and to optimize the therapeutic strategy.
纯红细胞再生障碍性贫血(PRCA)是一种罕见的贫血症,其特征是骨髓成红细胞显著减少导致网织红细胞严重减少,而其他血细胞谱系无异常。黑-戴二氏贫血是一种遗传性核糖体病,是PRCA的一种遗传形式。获得性PRCA分为原发性和继发性,包括细小病毒B19感染、胸腺瘤、淋巴增殖性疾病、自身免疫性疾病(狼疮)和药物性PRCA。PRCA的病理生理学尚未完全了解,涉及体液和T淋巴细胞自身反应性细胞。在与细小病毒B19相关的PRCA中,治疗基于多价免疫球蛋白。胸腺瘤患者必须进行胸腺切除术,但仅有少数病例能获得长期缓解。治疗策略基于专家意见:单一使用皮质类固醇很少能产生持续反应。是否选择额外的免疫抑制药物取决于潜在疾病。在大多数情况下,环孢素A是首选药物,缓解率最佳,但需要进行血药浓度监测(150至250 ng/mL)。第二种选择是在大颗粒淋巴细胞白血病中使用环磷酰胺。西罗莫司(一种mTOR抑制剂)似乎是一个有前景的选择,尤其是在难治性病例中。实现不依赖输血是主要目标。如果患者接受大量红细胞输血(>20单位),评估铁过载情况对于启动铁螯合治疗至关重要。现已建立了一项回顾性和前瞻性全国队列研究(EPIC-F),可纳入每一例PRCA病例,以增进对该疾病的了解并优化治疗策略。