Epstein-Barr virus–positive, primary cutaneous marginal zone lymphoma, with transformation: Case report and review of the literature

IF 4.6 Q2 MATERIALS SCIENCE, BIOMATERIALS
Lori Soma, Liliana Crisan, Jack Reid, Winston Lee, Joo Song, Michelle Afkhami, Geoffrey Shouse, Fei Fei, Olga Danilova, Raju Pillai, Jasmin Zain, Christiane Querfeld
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引用次数: 0

Abstract

Epstein-Barr Virus (EBV) positive primary cutaneous marginal zone lymphoma (PCMZL) is uncommon and subsequent transformation is rare. Methods: We report a patient with EBV positive PCMZL with subsequent transformation to plasmablastic lymphoma and review the literature for transformed PCMZL to assess clinical and pathologic characteristics. In the case we describe, the patient presented with multifocal PCMZL, developed large B cell transformation with plasmacytic differentiation, followed by plasmablastic transformation (PBL), and ultimately died of disease progression despite multiple lines of therapy. Past history was significant for psoriatic arthritis (multiple prior lines of immunomodulatory therapy). The lymphomas and non-involved bone marrow share the same somatic DNMT3A and TET2 mutations, suggesting clonal relatedness and an association with clonal hematopoiesis (CH). Results: Eighteen cases complied the cohort (seventeen cases from the literature and the case reported herein). Nearly half of the eighteen cases of PCMZL with transformation died of progressive disease (44%). Transformed cases were more commonly seen in patients with >2 sites at initial diagnosis. EBV was assessed in 5 patients, 3 were positive (all died of disease). Two patients with NGS studies demonstrated TET2 and DNMT3A mutations. Conclusions: Transformation of EBV positive PCMZL appears to be a poor prognostic indicator, with our reported case being the first well defined case transformed to PBL, suspected to arise from myeloid-CH.
Epstein-Barr 病毒阳性、原发性皮肤边缘区淋巴瘤,伴有转化:病例报告和文献综述
爱泼斯坦-巴氏病毒(EBV)阳性的原发性皮肤边缘区淋巴瘤(PCMZL)并不常见,随后的转化也很罕见。方法:我们报告了一名 EBV 阳性的原发性皮肤边缘区淋巴瘤(PCMZL)患者,该患者随后转变为浆液性淋巴瘤,我们还回顾了有关转变后的原发性皮肤边缘区淋巴瘤的文献,以评估其临床和病理特征。在我们描述的病例中,患者表现为多灶性 PCMZL,发生了大 B 细胞转化并伴有浆液性分化,随后发生了浆液性转化(PBL),尽管经过多线治疗,患者最终还是死于疾病进展。患者既往有银屑病关节炎病史(曾多次接受免疫调节治疗)。淋巴瘤和未受累的骨髓具有相同的体细胞DNMT3A和TET2突变,这表明该淋巴瘤与克隆性造血(CH)有关。结果18个病例组成了研究队列(17个病例来自文献,本文报告的病例来自文献)。在 18 例转化的 PCMZL 患者中,近一半(44%)死于疾病进展。转化病例多见于初诊时有>2个部位的患者。对 5 例患者进行了 EBV 评估,其中 3 例为阳性(均死于疾病)。两名患者的 NGS 研究显示其存在 TET2 和 DNMT3A 突变。结论EBV 阳性 PCMZL 的转化似乎是一个不良的预后指标,我们报告的病例是首例明确转化为 PBL 的病例,疑似来自髓系-CH。
本文章由计算机程序翻译,如有差异,请以英文原文为准。
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来源期刊
ACS Applied Bio Materials
ACS Applied Bio Materials Chemistry-Chemistry (all)
CiteScore
9.40
自引率
2.10%
发文量
464
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