Kazmierski D, Palomba M L, Barsigian C
Geisinger Commonwealth School of Medicine, 521 Pine Street, Scranton, PA 18510, USA.
Division of Hematology and Oncology, Memorial Sloan-Kettering Cancer Center, New York, NY, USA.
Case Rep Hematol. 2017;2017:5183646. doi: 10.1155/2017/5183646. Epub 2017 Feb 12.
The MYD88 L265P somatic variant (MYD88) has a high prevalence in Waldenstrom's Macroglobulinemia (WM), a form of lymphoplasmacytic lymphoma (LPL) associated with monoclonal IgM. Although the role of MYD88 in WM was initially reported in 2012, it was not until 2016 that MYD88 testing was included in the National Cancer Care Network (NCCN) Guidelines. We present a case illustrating the utility of MYD88 status in distinguishing atypical forms of WM from marginal zone lymphoma (MZL) and in selecting second-line therapy with ibrutinib. In 2012, a 64-year-old male presented with dyspnea on exertion, a hemoglobin of 5.6 g/dL, a platelet count of 86,000, and monoclonal IgM kappa on serum immunofixation but no detectable M-spike. Bone marrow biopsy revealed 95% monoclonal B-lymphocytes with lymphoplasmacytic differentiation favoring a diagnosis of LPL/WM over MZL, with a favorable response to chemotherapy. This diagnosis was called into question 3 years later following relapse, and MZL was favored based on the lack of MYD88 mutation. One year later, however, repeat bone marrow biopsy detected the MYD88 mutation and therapy with ibrutinib yielded a favorable response. The distinction between certain lymphomas can be problematic and in this case MYD88 was helpful in clarifying a diagnosis of atypical LPL/WM from MZL and in selecting effective second-line therapy.
MYD88 L265P体细胞变异(MYD88)在华氏巨球蛋白血症(WM)中具有较高的发生率,WM是一种与单克隆IgM相关的淋巴浆细胞淋巴瘤(LPL)形式。尽管MYD88在WM中的作用最初于2012年被报道,但直到2016年MYD88检测才被纳入美国国立综合癌症网络(NCCN)指南。我们报告一例病例,说明MYD88状态在鉴别非典型WM与边缘区淋巴瘤(MZL)以及选择依鲁替尼二线治疗中的作用。2012年,一名64岁男性出现劳力性呼吸困难,血红蛋白为5.6 g/dL,血小板计数为86,000,血清免疫固定电泳显示单克隆IgM κ,但未检测到M蛋白峰。骨髓活检显示95%为具有淋巴浆细胞分化的单克隆B淋巴细胞,支持LPL/WM而非MZL的诊断,对化疗反应良好。3年后复发,该诊断受到质疑,基于未检测到MYD88突变更倾向于MZL诊断。然而,1年后,重复骨髓活检检测到MYD88突变,依鲁替尼治疗产生了良好反应。某些淋巴瘤之间的鉴别可能存在问题,在本病例中,MYD88有助于明确非典型LPL/WM与MZL的诊断,并有助于选择有效的二线治疗。