Sethi Sanjeev, Cuiffo Barry P, Pinkus Geraldine S, Rennke Helmut G
Division of Renal Pathology and Electron Microscopy, Department of Pathology, Brigham and Women's Hospital, Harvard Medical School, Boston, MA 02115, USA.
Am J Kidney Dis. 2002 Jan;39(1):183-8. doi: 10.1053/ajkd.2002.29914.
Intracellular crystal formation in histiocytes in multiple myeloma and other lymphoproliferative disorders is an uncommon presentation. In all cases, crystal formation is associated with accumulation of histiocytes containing light chain or immunoglobulin inclusions, and the disorder has been termed crystal-storing histiocytosis. We report a case of crystal-storing histiocytosis affecting the kidney with prominent infiltration of the mesangium by large mononuclear and multinuclear cells that contained eosinophilic crystalline material in the setting of a low-grade B-cell lymphoproliferative disorder. The interstitium did not contain similar crystal-containing histiocytes. On electron microscopy, the mononuclear and multinuclear cells in the mesangium were filled with rhomboid and needle-shaped crystals. Immunofluorescence studies showed the material to be positive for lambda light chains but negative for kappa light chains. To our knowledge, this is the first report of crystal-storing histiocytosis involving the kidney in a low-grade B-cell lymphoproliferative disorder with an immunophenotype of a marginal zone lymphoma and of exclusive expansion of the mesangium by infiltrating histiocytes containing needle-shaped and rhomboid crystals that were positive for lambda light chains. We report this case to illustrate an unusual finding of mesangial infiltration by crystal-storing histiocytes, and we review the literature of renal involvement by crystal-storing histiocytosis and crystal deposition in lymphoproliferative disorders.
在多发性骨髓瘤和其他淋巴增殖性疾病中,组织细胞内晶体形成是一种不常见的表现。在所有病例中,晶体形成与含有轻链或免疫球蛋白包涵体的组织细胞积聚有关,这种病症被称为晶体储存组织细胞增多症。我们报告一例晶体储存组织细胞增多症累及肾脏的病例,在低度B细胞淋巴增殖性疾病背景下,可见大量单核和多核细胞显著浸润肾小球系膜,这些细胞含有嗜酸性结晶物质。肾间质中未发现类似的含晶体组织细胞。电镜检查显示,肾小球系膜中的单核和多核细胞充满菱形和针状晶体。免疫荧光研究表明,该物质λ轻链呈阳性,κ轻链呈阴性。据我们所知,这是首例关于低度B细胞淋巴增殖性疾病中晶体储存组织细胞增多症累及肾脏的报道,该疾病具有边缘区淋巴瘤的免疫表型,且肾小球系膜仅被含有针状和菱形晶体且λ轻链呈阳性的浸润性组织细胞所扩张。我们报告该病例以说明晶体储存组织细胞浸润肾小球系膜这一不寻常发现,并回顾晶体储存组织细胞增多症累及肾脏及淋巴增殖性疾病中晶体沉积的相关文献。