Rosai-Dorfman病(RDD)又名为窦组织细胞增生伴巨大淋巴结病(SHML),通常表现为巨大的、累及双侧的无痛性淋巴结肿大,约25%病例累及结外,而累及男性泌尿生殖道的极为罕见。作者报道2例RDD累及睾丸,同时复习其他睾丸及相关结构RDD病例(包括文献报道的4例及记录在SHML登记处的7例)的临床病理特点。13例中5例同时累及双侧睾丸,8例还累及淋巴结、肾、皮肤和肾上腺等其他部位。多数病例发生于中年人,1例发生于儿童。本组2例具 Rosai-Dorfman’s disease (RDD), also known as sinusoidal hyperplasia with giant lymph node disease (SHML), usually presents as a giant, bilateral, painless lymphadenopathy with about 25% of cases involving extranodal involvement involving men Urogenital tract is extremely rare. The authors reported two cases of RDD involving the testis and reviewing the clinicopathological features of other testicular and related structural RDD cases (including 4 reported in the literature and 7 documented in the SHML registry). Five of the 13 patients involved bilateral testes at the same time, and eight also affected lymph nodes, kidneys, skin and adrenal glands. Most cases occur in middle-aged people, one case occurred in children. The group of 2 cases with