Abdolahi Mitra, Padilla Osvaldo, Dave Sandeep S, Thakkar Devang, Nejati Reza
Department of Pathology, Fox Chase Cancer Center, Temple University Health System, Philadelphia, PA, 19111, USA.
Department of Pathology, PL Foster School of Medicine, Texas Tech University Health Sciences Center, 4625 Alberta Ave, El Paso, TX, 79905, USA.
J Hematop. 2025 Jul 24;18(1):35. doi: 10.1007/s12308-025-00651-9.
Plasma cell neoplasms encompass a spectrum of disorders characterized by the clonal proliferation of plasma cells. Plasmablastic transformation in these neoplasms poses diagnostic and clinical challenges due to its aggressive nature and morphological overlap with other malignancies, including plasmablastic lymphoma (PBL). We report a case of discordant extramedullary plasmablastic transformation in a 55-year-old HIV-negative female presenting with an oral lesion diagnosed as plasmablastic plasmacytoma. Initial imaging indicated localized disease without systemic involvement. Despite undergoing chemotherapy and achieving a partial response, the patient developed osteolytic lesions 2 years later. Subsequent pathology evaluation confirmed mature plasma cell myeloma (PCM) morphology. Whole exome sequencing (WES) and whole RNA expression analysis revealed shared mutations (PTPN13, KRAS, LTK) and a MYC::BMP6 translocation in both lesions, supporting a clonal relationship. Additionally, the oral plasmablastic lesion revealed a KLHL6 mutation and an extra MYC::IGL translocation, which were absent in the tibial lesion. The KLHL6 mutation has not been previously reported in studies of discordant extramedullary plasmacytoma with plasmablastic transformation. This case highlights the diagnostic complexity of plasmablastic plasmacytoma presenting as the initial manifestation of plasma cell myeloma. It underscores the necessity of a thorough evaluation, including bone marrow biopsy, to accurately differentiate plasmablastic transformation from PBL and ensure accurate diagnosis and appropriate management.
浆细胞肿瘤包括一系列以浆细胞克隆性增殖为特征的疾病。这些肿瘤中的浆母细胞转化因其侵袭性本质以及与其他恶性肿瘤(包括浆母细胞淋巴瘤(PBL))的形态学重叠而带来诊断和临床挑战。我们报告一例55岁HIV阴性女性发生的不一致性髓外浆母细胞转化病例,该患者出现口腔病变,诊断为浆母细胞性浆细胞瘤。初始影像学检查显示为局限性疾病,无全身受累。尽管接受了化疗并取得部分缓解,但患者在2年后出现溶骨性病变。随后的病理评估证实为成熟浆细胞骨髓瘤(PCM)形态。全外显子测序(WES)和全RNA表达分析显示两个病变均存在共同突变(PTPN13、KRAS、LTK)以及MYC::BMP6易位,支持克隆关系。此外,口腔浆母细胞病变显示存在KLHL6突变和额外的MYC::IGL易位,而胫骨病变中不存在这些情况。KLHL6突变在先前关于伴有浆母细胞转化的不一致性髓外浆细胞瘤的研究中尚未见报道。该病例突出了以浆细胞骨髓瘤初始表现形式出现的浆母细胞性浆细胞瘤的诊断复杂性。它强调了进行全面评估(包括骨髓活检)的必要性,以准确区分浆母细胞转化与PBL,并确保准确诊断和恰当管理。