Washimi Kota, Kasajima Rika, Sato Shinya, Nezu Yutaka, Takahashi Hiroyuki, Sakai Rika, Nakamura Naoya, Takagi Masayuki, Hasegawa Chie, Yoshioka Emi, Okubo Yoichiro, Katayama Kotoe, Imoto Seiya, Yokose Tomoyuki, Miyagi Yohei
Department of Pathology, Kanagawa Cancer Center, Yokohama, Japan.
Molecular Pathology and Genetics Division, Kanagawa Cancer Center Research Institute, Yokohama, Japan.
Cancer Rep (Hoboken). 2025 Feb;8(2):e70129. doi: 10.1002/cnr2.70129.
Liposarcoma and lymphoma are very rare tumors, and their combination is extremely rare. Moreover, there have been no reports of liposarcoma and lymphoma occurring in the same region.
A 58-year-old man presented to Kanagawa Cancer Center with a mass in his left thigh and underwent a needle biopsy. Histological analysis showed an increase in the number of small lymphocytes and plasma cells; immunohistochemical analysis showed an increase in CD20-positive cells with Lambda light-chain restriction; therefore, the diagnosis of B-cell malignancy with plasma cell differentiation was made. A bone marrow biopsy specimen showed infiltration of atypical cells of the same phenotype and increased serum IgM-M levels; therefore, a diagnosis of Waldenström macroglobulinemia/lymphoplasmacytic lymphoma (LPL) was made. The needle biopsy specimen showed scattered CDK4-positive cells in the background of the lymphoma cells and sporadic MDM2 signal amplification on fluorescence in situ hybridization, suggesting mixed well-differentiated liposarcoma (WDL). Tumor resection was performed. The tumor contained a mixture of WDL and LPL areas. RNA sequencing revealed upregulated expression of chemokine genes, including CCL5, CCL18, and CCL19, in WDL and that of the corresponding chemokine receptor genes CCR4, CCR6, and CCR7 in the lymphoma cells.
Chemokine-chemokine receptor axes may be involved in the pathogenesis of LPL cell-infiltrating WDL. This is an extremely rare case, and we have reported some considerations regarding the tumorigenesis of LPL cell-infiltrating WDL.
脂肪肉瘤和淋巴瘤是非常罕见的肿瘤，它们同时出现极为罕见。此外，尚无同一区域发生脂肪肉瘤和淋巴瘤的报道。
一名58岁男性因左大腿肿块就诊于神奈川癌症中心，并接受了针吸活检。组织学分析显示小淋巴细胞和浆细胞数量增加；免疫组化分析显示CD20阳性细胞增加且伴有λ轻链限制；因此，诊断为具有浆细胞分化的B细胞恶性肿瘤。骨髓活检标本显示相同表型的非典型细胞浸润且血清IgM-M水平升高；因此，诊断为华氏巨球蛋白血症/淋巴浆细胞性淋巴瘤（LPL）。针吸活检标本显示在淋巴瘤细胞背景中有散在的CDK4阳性细胞，荧光原位杂交显示偶发MDM2信号扩增，提示混合性高分化脂肪肉瘤（WDL）。进行了肿瘤切除。肿瘤包含WDL和LPL区域的混合物。RNA测序显示WDL中趋化因子基因（包括CCL5、CCL18和CCL19）表达上调，淋巴瘤细胞中相应趋化因子受体基因CCR4、CCR6和CCR7表达上调。
趋化因子-趋化因子受体轴可能参与LPL细胞浸润WDL的发病机制。这是一例极其罕见的病例，我们报告了一些关于LPL细胞浸润WDL肿瘤发生的思考。
