Kawai Masataka, Inoue Tomohiro, Yonaga Takaaki, Mochizuki Kunio, Nakazawa Tadao, Masuyama Keisuke, Kondo Tetsuo
Department of Pathology, University of Yamanashi, 1110 Shimokato, Chūō, Yamanashi, Japan.
Department of Otorhinolaryngology and Head & Neck Surgery, University of Yamanashi, 1110 Shimokato, Chūō, Yamanashi, Japan.
Diagn Cytopathol. 2019 Nov;47(11):1208-1212. doi: 10.1002/dc.24283. Epub 2019 Jul 22.
Sclerosing polycystic adenosis (SPA) is a rare salivary gland disease. Histologically it resembles a low-grade ductal carcinoma in situ or sclerosing adenosis of the breast, characterized by lobular proliferation of ducts with apocrine cellular features surrounded by fibrosclerotic stroma. Although SPA is typically benign, recurrence is not uncommon, and cases with a malignant component have been documented. Thus, complete excision is desirable but preoperative diagnosis is challenging. A 12-year-old boy presented with a painless mass in the right neck. We identified a well-demarcated mass in the right parotid region measuring approximately 2 cm using cervical echography and magnetic resonance (MR) imaging. Fine-needle aspiration (FNA) revealed two cell types. There were loosely cohesive clusters of polymorphic epithelioid cells with irregular nuclei and abundant vacuolated cytoplasm containing zymogen granules. Some of these cells were binuclear. The other cell types represented normal ductal cells. The original cytological diagnosis was Warthin tumor. Right parotidectomy was performed. Histologically, we observed proliferation of ducts with granular, vacuolated, zymogen granules, and apocrine-like features in the cytoplasm with hyalinizing sclerotic stroma and some binuclear cells. Four years after parotidectomy, there has been no recurrence or malignant transformation.Cytological diagnosis of SPA is challenging on FNA specimens since SPA is a very rare entity of the salivary gland that can mimic other salivary gland neoplasms. A mixture of apocrine-like cells and sebaceous-like cells, nuclear pleomorphism, and zymogen granules can help to diagnose this rare lesion during the initial cytological diagnosis.
硬化性多囊性腺病(SPA)是一种罕见的涎腺疾病。组织学上,它类似于乳腺的低级别导管原位癌或硬化性腺病,其特征为导管小叶状增生,具有顶泌汗腺样细胞特征,周围有纤维硬化性间质。虽然SPA通常为良性,但复发并不少见,且已有出现恶性成分病例的记录。因此,理想的治疗方法是完整切除,但术前诊断具有挑战性。一名12岁男孩右侧颈部出现无痛性肿块。我们通过颈部超声检查和磁共振(MR)成像在右侧腮腺区发现一个边界清晰的肿块,大小约为2厘米。细针穿刺抽吸(FNA)显示有两种细胞类型。有一种是多形性上皮样细胞的松散聚集,细胞核不规则,胞质丰富,有空泡,含有酶原颗粒。其中一些细胞为双核。另一种细胞类型为正常导管细胞。最初的细胞学诊断为沃辛瘤。遂行右侧腮腺切除术。组织学检查发现,导管增生,细胞质内有颗粒状、空泡状、酶原颗粒以及顶泌汗腺样特征,伴有透明变性硬化性间质和一些双核细胞。腮腺切除术后四年,未见复发或恶变。由于SPA是涎腺中一种非常罕见的疾病,可模仿其他涎腺肿瘤,因此对FNA标本进行SPA的细胞学诊断具有挑战性。顶泌汗腺样细胞和皮脂腺样细胞的混合、核多形性以及酶原颗粒有助于在初次细胞学诊断时诊断这种罕见病变。