Ojeda-Uribe Mario, Rimelen Valérie, Marzullo Cathérine
Department of Hematology and Cellular Therapy Unit, Groupe Hospitalier Region Mulhouse-Sud-Alsace (GHRMSA), Mulhouse 68070, France.
Laboratory of Molecular Biology CHRU Strasbourg, Strasbourg 67200, France.
J Blood Med. 2020 Feb 13;11:67-72. doi: 10.2147/JBM.S233059. eCollection 2020.
Acquired von Willebrand syndrome (AVWS) in the setting of Waldenström macroglobulinemia (WM) is a challenging condition. No real standard of care is recommended for these patients, although the therapeutic strategy should include a rapid approach to the emergency bleeding events and to the underlying malignant lymphoid disorder. We report here our experience treating three elderly patients with these concomitant hematologic entities. The use of a bortezomib-based chemotherapy regimen showed a good profile of tolerance and efficacy even in a long-term follow-up period. These patients were treated for several years before switching their therapy to idelalisib, a targeted oral therapy that inhibits phosphatidylinositol 3-kinase isoform-delta (PI3KD), which is part of the signaling pathway downstream B-cell receptor. This approach was well tolerated and efficacious, although some adverse effects were observed, particularly at hepatic levels, but were all reversible. The same profile of tolerance/efficacy was observed in one very old patient who received idelalisib as a first-line therapy. We think that bortezomib-based therapy could be considered in refractory patients with AVWS associated with WM.
在华氏巨球蛋白血症(WM)背景下的获得性血管性血友病综合征(AVWS)是一种具有挑战性的病症。对于这些患者,尽管治疗策略应包括快速处理紧急出血事件和潜在的恶性淋巴样疾病,但目前尚无真正推荐的标准治疗方案。我们在此报告治疗三名患有这些并发血液学疾病的老年患者的经验。使用基于硼替佐米的化疗方案即使在长期随访期间也显示出良好的耐受性和疗效。这些患者在将治疗方案转换为idelalisib(一种靶向口服疗法,可抑制磷脂酰肌醇3激酶异构体δ(PI3KD),其是B细胞受体下游信号通路的一部分)之前已经接受了数年治疗。这种方法耐受性良好且有效,尽管观察到一些不良反应,特别是在肝脏水平,但都是可逆的。在一名接受idelalisib作为一线治疗的高龄患者中也观察到了相同的耐受性/疗效情况。我们认为,对于与WM相关的难治性AVWS患者,可以考虑基于硼替佐米的治疗。