Wu Leng, Wang Ying, Hu Chuan-Yu, Huang Chun-Ming
Department of Stomatology, Tongji Hospital of Tongji Medical College of Huazhong University of Science and Technology, Wuhan 430000, Hubei Province, China.
Department of Pathology, Tongji Hospital of Tongji Medical College of Huazhong University of Science and Technology, Wuhan 430000, Hubei Province, China.
World J Clin Cases. 2021 Apr 26;9(12):2930-2936. doi: 10.12998/wjcc.v9.i12.2930.
Sclerosing polycystic adenosis (SPA) is a rare disease of salivary glands, similar to fibrocystic disease of the breast. It occurs over a wide age range and exhibits a slight female preference. Most SPA cases have occurred in the parotid gland. The exact nature of SPA is unclear, but its tumor nature has recently been proposed. Although SPA has a good prognosis after adequate surgery, atypical lesions might occur, ranging from mild dysplasia to carcinoma in some cases. To the best of our knowledge, only five cases of SPA in the submandibular gland have been reported to date. Here, we present two new cases of SPA involving the submandibular gland.
A 50-year-old woman and a 52-year-old woman were referred to Tongji Hospital in Wuhan, China, with complaints of moderate pain, recurrent swelling, and a mass in the submandibular area. After admission, the two cases of the submandibular mass were examined physically. The boundary of the submandibular tumor was clear, and the range of motion was good. After preoperative examinations, surgery was performed on a selective basis. Postoperative histopathological examination revealed a well-defined mass with acinar structures, ducts, or cystic dilated glands of various sizes scattered in a large number of proliferative sclerosing stroma. There were flat and cuboidal cells, and eosinophils in the duct epithelium. There was also a eosinophilic substance in the lumen of dilated cysts. No atypical epithelial hyperplasia, invasive growth, or carcinoma was found. Based on the above findings, the mass was diagnosed as SPA. Both patients have remained asymptomatic and no recurrence or distant metastasis had occurred by the 7-mo and 5-year follow-up, respectively.
SPA is a rare disease of the salivary gland. Even though it has a good prognosis after adequate surgery, atypical lesions may occur from mild dysplasia to carcinoma . However, no recurrence, distant metastasis, or mortality has been reported for submandibular gland SPA. Clinicians and pathologists should be familiar with the characteristics of SPA in the submandibular gland to avoid misdiagnosis and overtreatment.
硬化性多囊性腺病（SPA）是一种罕见的涎腺疾病，类似于乳腺纤维囊性疾病。其发病年龄范围广，且略偏向女性。大多数SPA病例发生于腮腺。SPA的确切性质尚不清楚，但最近有人提出其具有肿瘤性质。尽管SPA在进行充分手术后预后良好，但在某些情况下可能会出现非典型病变，范围从轻度发育异常到癌。据我们所知，迄今为止仅报道了5例发生于下颌下腺的SPA。在此，我们报告2例累及下颌下腺的新的SPA病例。
一名50岁女性和一名52岁女性因下颌下区中度疼痛、反复肿胀及肿物而被转诊至中国武汉的同济医院。入院后，对这两例下颌下肿物进行了体格检查。下颌下肿瘤边界清晰，活动度良好。经过术前检查后，有选择地进行了手术。术后组织病理学检查显示有一个边界清楚的肿物，伴有腺泡结构、导管或大小不一的囊性扩张腺体，散在于大量增生性硬化性间质中。导管上皮中有扁平细胞、立方细胞和嗜酸性粒细胞。扩张囊肿的腔内也有一种嗜酸性物质。未发现非典型上皮增生、浸润性生长或癌。根据上述发现，该肿物被诊断为SPA。两名患者均无症状，在分别随访7个月和5年时均未出现复发或远处转移。
SPA是一种罕见的涎腺疾病。尽管在进行充分手术后预后良好，但可能会出现从轻度发育异常到癌的非典型病变。然而，尚未有关于下颌下腺SPA复发、远处转移或死亡的报道。临床医生和病理医生应熟悉下颌下腺SPA的特征，以避免误诊和过度治疗。
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