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首例生前活检证实的坏死性黄色肉芽肿伴单克隆丙种球蛋白病肾脏受累的病例报告。

First case report of pre-mortem biopsy-proven kidney involvement by necrobiotic xanthogranuloma with monoclonal gammopathy.

机构信息

Department of Renal Medicine, Concord Repatriation General Hospital, Hospital Rd, Concord, NSW, Australia.

Department of Anatomical Pathology, Concord Repatriation General Hospital, Hospital Rd, Concord, NSW, Australia.

出版信息

J Nephrol. 2022 Jul;35(6):1753-1757. doi: 10.1007/s40620-021-01186-9. Epub 2021 Nov 5.

Abstract

Necrobiotic xanthogranuloma (NXG) is a rare non-Langerhans cell histiocytosis with characteristic cutaneous features and rare visceral involvement. More than 80% of individuals with this disease have a detectable paraprotein but the precise pathogenesis remains obscure. A 68-year-old man with known cutaneous necrobiotic xanthogranuloma presented with acute kidney injury and imaging suggestive of bilateral perinephric infiltration. Renal biopsy showed a prominent histiocytic infiltration of renal capsule and cortex with necrobiosis and characteristic 'Touton-type' giant cells suggestive of necrobiotic xanthogranuloma involvement. Kidney function returned to normal and cutaneous lesions improved with a combination of corticosteroid, chlorambucil and rituximab. This case represents only the second reported incidence of kidney involvement by necrobiotic xanthogranuloma and the first with acute kidney injury and pre-mortem histopathology. This report adds to a small body of literature on the diagnosis and management of visceral involvement by this rare disease.

摘要

坏死性黄色肉芽肿(NXG)是一种罕见的非朗格汉斯细胞组织细胞增生症,具有特征性的皮肤表现和罕见的内脏受累。超过 80%的该病患者可检测到单克隆丙种球蛋白,但确切的发病机制仍不清楚。一名 68 岁男性患有已知的皮肤坏死性黄色肉芽肿,出现急性肾损伤和影像学提示双侧肾周浸润。肾活检显示肾包膜和皮质有明显的组织细胞浸润,伴有坏死和特征性的“Touton 样”巨细胞,提示坏死性黄色肉芽肿受累。肾功能恢复正常,皮肤病变在皮质类固醇、苯丁酸氮芥和利妥昔单抗联合治疗后得到改善。本例是坏死性黄色肉芽肿肾脏受累的第二例报道,也是首例伴有急性肾损伤和生前组织病理学的报道。本报告增加了关于这种罕见疾病内脏受累的诊断和治疗的少量文献。

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