Koga Shunsuke, Yang Zhaohai, Alipour Zahra
Department of Pathology and Laboratory Medicine, Hospital of the University of Pennsylvania, Philadelphia, PA, USA.
Int J Surg Pathol. 2025 Dec;33(8):1846-1851. doi: 10.1177/10668969251343639. Epub 2025 May 29.
Crystal-storing histiocytosis is a rare condition characterized by the accumulation of crystalline immunoglobulins within histiocytes, often associated with plasma cell disorders such as multiple myeloma. We report a 70-year-old man with a history of IgG-lambda multiple myeloma, systemic amyloidosis, and end-stage renal disease managed with peritoneal dialysis and long-term lanthanum carbonate therapy for hyperphosphatemia, who presented with gastrointestinal bleeding. Endoscopy revealed nodular erythematous mucosa in the duodenum, and biopsies showed histiocytosis in the lamina propria. The histiocytes showed abundant cytoplasm containing periodic acid-Schiff-positive diastase-resistant intracellular material. Immunohistochemical staining revealed diffuse CD68 positivity and lambda light-chain restriction of the storage material in the histiocytes, supporting a diagnosis of crystal-storing histiocytosis. Differential diagnoses, including Whipple disease, mycobacterial infections, lanthanum deposition, amyloidosis, and xanthoma, were excluded based on histological findings and clinical history. This report highlights the importance of recognizing crystal-storing histiocytosis in uncommon sites such as the duodenum, especially in the context of plasma cell dyscrasias.
晶体贮积性组织细胞增多症是一种罕见疾病,其特征为组织细胞内晶体免疫球蛋白的蓄积,常与浆细胞疾病如多发性骨髓瘤相关。我们报告一名70岁男性,有IgG-λ型多发性骨髓瘤、系统性淀粉样变性病史,终末期肾病,采用腹膜透析及长期服用碳酸镧治疗高磷血症,该患者出现胃肠道出血。内镜检查显示十二指肠黏膜有结节状红斑,活检显示固有层存在组织细胞增多症。组织细胞显示丰富的胞质,含有对高碘酸-希夫染色呈阳性且耐淀粉酶的细胞内物质。免疫组化染色显示组织细胞内贮存物质弥漫性CD68阳性及λ轻链限制,支持晶体贮积性组织细胞增多症的诊断。根据组织学检查结果和临床病史排除了包括惠普尔病、分枝杆菌感染、镧沉积、淀粉样变性和黄瘤等鉴别诊断。本报告强调了在十二指肠等不常见部位识别晶体贮积性组织细胞增多症的重要性,尤其是在浆细胞发育异常的背景下。