McIntire Patrick J, Kilic Ayse Irem, Chen Hannah H, Atieh Mohammed, Wojcik Eva M, Pambuccian Stefan E
Loyola University Medical Center, Department of Pathology and Laboratory Medicine, Maywood, Illinois.
Loyola University Medical Center, Department of Pathology and Laboratory Medicine, Maywood, Illinois.
J Am Soc Cytopathol. 2019 Jul-Aug;8(4):190-205. doi: 10.1016/j.jasc.2019.01.008. Epub 2019 Feb 5.
Rosai-Dorfman disease (RDD) is a rare usually self-limited non-Langerhans cell histiocytosis of unknown etiology. Nodal and extranodal RDD appear to represent distinct conditions with different molecular alterations and prognosis. They also pose different diagnostic challenges on biopsies and fine-needle aspiration (FNA) cytology. The aim of this study was to report on 3 cases of intra-abdominal RDD and perform an extensive review of the literature on FNA findings of RDD.
We reviewed FNA specimens from cases diagnosed histologically or cytologically as RDD during the past 10 years. We searched the PubMed and Google Scholar databases for cases of RDD sampled by FNA.
We identified 3 cases of intra-abdominal RDD, involving the kidney, periportal lymph node, and pancreas. FNA of the latter was hypocellular with fibrosis and was nondiagnostic. FNA of the first 2 yielded hypercellular smears that were diagnosed as RDD due to the identification of emperipolesis occurring in large uni- or binucleated histiocytes with large nuclei, fine chromatin, and prominent nucleoli in smears and cell-block sections. Immunohistochemistry showed positive staining for S100 and CD68 and negative staining for CD1a. The large histiocytes with emperipolesis were more difficult to identify histologically and their demonstration required immunohistochemical stains.
Our experience and an extensive review of the literature suggest that extranodal RDD can be diagnosed on FNA, and that the recognition of histiocytes with emperipolesis may be less challenging cytologically than histologically. The fibrosis frequently seen in extranodal RDD may lead to nondiagnostic aspirates, however.
罗萨伊-多夫曼病（RDD）是一种罕见的、通常自限性的病因不明的非朗格汉斯细胞组织细胞增生症。淋巴结型和结外型RDD似乎代表了具有不同分子改变和预后的不同病症。它们在活检和细针穿刺（FNA）细胞学检查中也带来不同的诊断挑战。本研究的目的是报告3例腹腔内RDD病例，并对RDD的FNA检查结果的文献进行广泛综述。
我们回顾了过去10年中经组织学或细胞学诊断为RDD的病例的FNA标本。我们在PubMed和谷歌学术数据库中搜索了通过FNA采样的RDD病例。
我们确定了3例腹腔内RDD，累及肾脏、肝门周围淋巴结和胰腺。后者的FNA细胞少且有纤维化，诊断不明确。前两例的FNA涂片细胞丰富，由于在涂片和细胞块切片中发现大的单核或双核组织细胞内有吞噬现象，这些组织细胞核大、染色质细、核仁明显，被诊断为RDD。免疫组织化学显示S100和CD68染色阳性，CD1a染色阴性。有吞噬现象的大组织细胞在组织学上更难识别，其显示需要免疫组织化学染色。
我们的经验和对文献的广泛综述表明，结外型RDD可通过FNA诊断，并且在细胞学上识别有吞噬现象的组织细胞可能比在组织学上更具挑战性。然而，结外型RDD中常见的纤维化可能导致穿刺抽吸物诊断不明确。
