da Cruz Perez Danyel Elias, Silva-Sousa Yara Teresinha Corrêa, de Andrade Bruno Augusto Benevenuto, Rizo Victor Hugo Toral, Almeida Luciana Yamamoto, León Jorge Esquiche, de Almeida Oslei Paes
School of Dentistry, Oral Pathology Unit, Federal University of Pernambuco-Av. Prof. Moraes Rego, 1235, 50670-901. Recife/PE, Brazil.
Ann Diagn Pathol. 2012 Dec;16(6):527-31. doi: 10.1016/j.anndiagpath.2011.05.004. Epub 2011 Aug 16.
Crystal-storing histiocytosis is a rare manifestation of plasma cell dyscrasia/monoclonal gammopathies and lymphoproliferative disorders, characterized by cytoplasmic accumulation of crystallized immunoglobulins in histiocytes. Nevertheless, some reported cases of crystal-storing histiocytosis raise the possibility that this lesion may also be reactive. Crystal-storing histiocytosis in the oral cavity is extremely rare; only one case affecting the tongue has been reported in the English-language literature. In this report, we discuss the case of a 38-year-old man who presented a persistent periapical lesion affecting the maxillary left lateral incisor. Histopathological analysis showed numerous crystal-laden histiocytes associated with a mild plasma cell infiltrate within a fibrous stroma. The plasma cells failed to show clonal light-chain restriction, and the patient had no associated hematologic disorder or systemic disease. Thus, this lesion was probably the result of hypersecretion of immunoglobulins by polyclonal plasma cells found in the periapical lesion. Crystal-storing histiocytosis should be considered in the differential diagnosis of periapical lesions.
晶体储存性组织细胞增多症是浆细胞发育异常/单克隆丙种球蛋白病及淋巴增殖性疾病的一种罕见表现,其特征为组织细胞内出现结晶化免疫球蛋白的胞质蓄积。然而,一些已报道的晶体储存性组织细胞增多症病例提示,这种病变也可能是反应性的。口腔内的晶体储存性组织细胞增多症极为罕见;英文文献中仅报道过1例累及舌部的病例。在本报告中,我们讨论了1例38岁男性患者,其上颌左侧侧切牙出现持续性根尖周病变。组织病理学分析显示,在纤维性基质内有大量充满晶体的组织细胞,并伴有轻度浆细胞浸润。浆细胞未显示克隆性轻链限制,且该患者无相关血液系统疾病或全身性疾病。因此,这种病变可能是根尖周病变中多克隆浆细胞免疫球蛋白分泌过多的结果。在根尖周病变的鉴别诊断中应考虑晶体储存性组织细胞增多症。