Sonomura Kazuhiro, Ehara Hozue, Adachi Hiroya, Yamane Yusuke, Kawata Eri, Mori Yasukiyo
Deparment of Nephrology, Matsushita Memorial Hospital, 5-55 Sotojima-Cho Moriguchi-Shi, Osaka, 570-8540, Japan.
Department of Hematology, Matsushita Memorial Hospital, 5-55 Sotojima-Cho Moriguchi-Shi, Osaka, 570-8540, Japan.
CEN Case Rep. 2025 Jun;14(3):428-433. doi: 10.1007/s13730-025-00977-4. Epub 2025 Feb 13.
Lymphoplasmacytic lymphoma (LPL) is a type of low-grade B-cell lymphoma, with 90-95% of cases associated with Waldenström macroglobulinemia, characterized by the presence of IgM-type M-protein. We report, for the first time, a case of LPL-producing Bence Jones (BJ) protein kappa. The patient was a 78-year-old woman admitted to our department due to general fatigue and proteinuria that had persisted for 2 months. No M-protein was detected by blood immunofixation, but kappa-type BJ protein was detected in the urine. Light microscopy of a kidney biopsy sample revealed infiltration of lymphocytes and plasma cells into the perirenal adipose tissue and renal interstitium. The infiltrating cells exhibited kappa light chain restriction. Bone marrow examination revealed clusters of immature plasmacytoid lymphocytes that were CD20 positive, CD5 negative, and exhibited light chain restriction. Genetic analysis detected a MYD88 mutation, leading to the diagnosis of LPL in the patient. Six months after starting treatment with tirabrutinib, urinary protein levels improved to 0.2 g/gCr. Renal infiltration was identified due to urinary protein, and currently, no extramedullary lesions outside the kidneys are observed. Tirabrutinib has been extremely effective, but careful follow-up is still required.
淋巴浆细胞淋巴瘤(LPL)是一种低度恶性B细胞淋巴瘤,90%至95%的病例与华氏巨球蛋白血症相关,其特征为存在IgM型M蛋白。我们首次报告了一例产生本斯·琼斯(BJ)蛋白κ的LPL病例。该患者为一名78岁女性,因全身乏力和蛋白尿持续2个月入院。血液免疫固定电泳未检测到M蛋白,但尿液中检测到κ型BJ蛋白。肾脏活检样本的光镜检查显示淋巴细胞和浆细胞浸润至肾周脂肪组织和肾间质。浸润细胞表现出κ轻链限制。骨髓检查发现不成熟浆细胞样淋巴细胞簇,其CD20阳性、CD5阴性且表现出轻链限制。基因分析检测到MYD88突变,从而确诊该患者为LPL。开始使用替拉布替尼治疗6个月后,尿蛋白水平改善至0.2 g/gCr。因尿蛋白发现肾脏浸润,目前未观察到肾脏以外的髓外病变。替拉布替尼极为有效,但仍需密切随访。