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未分化结缔组织病患者乳腺晶体储存组织细胞增多症1例罕见病例

A Rare Case of Crystal-Storing Histiocytosis of the Breast in a Patient With Undifferentiated Connective Tissue Disease.

作者信息

Bedell Mariel, Naous Rana

机构信息

Pathology, University of Pittsburgh Medical Center, Pittsburgh, USA.

出版信息

Cureus. 2025 Jun 9;17(6):e85620. doi: 10.7759/cureus.85620. eCollection 2025 Jun.

Abstract

Crystal-storing histiocytosis (CSH) is a rare entity usually presenting microscopically as sheets of histiocytes with intralysosomal accumulation of refractile, crystalline substances. CSH is most often the result of monoclonal immunoglobulin light chain deposition in the setting of a plasma cell neoplasm or lymphoma with plasma cell differentiation. However, this condition may arise in association with autoimmune, inflammatory, infectious, and drug-related etiologies and can harbor polyclonal immunoglobulins or other crystalline materials. It usually presents in a localized fashion, with the bone marrow being the most frequent site, but may also involve multiple anatomical sites. CSH is typically identified incidentally without clinical suspicion and can be challenging to detect histologically. However, in rare cases, it may be the sole presenting sign in an undiagnosed malignancy or immune disorder. Here, we describe an unusual case of CSH, occurring as an isolated breast mass in association with an undifferentiated connective tissue disease (UCTD) with inflammatory arthritis, features of Sjögren's (Sicca) syndrome, and subsequent laboratory findings concerning for antiphospholipid syndrome.

摘要

晶体储存性组织细胞增多症(CSH)是一种罕见疾病,在显微镜下通常表现为成片的组织细胞,伴有溶酶体内折光性晶体物质的蓄积。CSH最常见于浆细胞瘤或伴有浆细胞分化的淋巴瘤背景下的单克隆免疫球蛋白轻链沉积。然而,这种情况也可能与自身免疫、炎症、感染及药物相关病因有关,并且可能含有多克隆免疫球蛋白或其他晶体物质。它通常以局限性方式出现,骨髓是最常见的部位,但也可能累及多个解剖部位。CSH通常在无临床怀疑的情况下偶然发现,在组织学上难以检测。然而,在罕见情况下,它可能是未诊断恶性肿瘤或免疫疾病的唯一表现体征。在此,我们描述了一例不寻常的CSH病例,表现为孤立性乳腺肿块,与伴有炎性关节炎的未分化结缔组织病(UCTD)、干燥综合征特征以及随后提示抗磷脂综合征的实验室检查结果相关。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/397c/12239935/c0c454cb48d2/cureus-0017-00000085620-i01.jpg

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