91 15 19 2015 1 1,2) 3) 2) 4) 2) 5) 5) 6) 6) 1,2) 2014 11 6 2014 12 1 18 2 class 19.5 MRI 21x15 calponin p63 2 3 SPA Key words: 1) 2) 3) 4) 5) 6) Masaki Suzuki 1,2), Hironori Noge 3), Michiyo Kambe 2), Takashi Kishimoto 4), Satoshi Ota 2), Hideaki Chazono 5), Yoshitaka Okamoto 5), Takuro Horikoshi 6), Takashi Uno 6) and Yukio Nakatani 1,2) : A case of sclerosing polycystic adenosis of the parotid gland. 1) Department of Diagnostic Pathology, Graduate School of Medicine, Chiba University, Chiba 260-8670. 2) Department of Pathology, Chiba University Hospital, Chiba 260-8677. 3) School of Medicine, Chiba University, Chiba 260-8670. 4) Department of Molecular Pathology, Graduate School of Medicine, Chiba University, Chiba 260-8670. 5) Department of Otorhinolaryngology, Head and Neck Surgery, Graduate School of Medicine, Chiba University, Chiba 260-8670. 6) Department of Radiology, Chiba University Hospital, Chiba 260-8677. Phone: 043-222-7171. Fax: 043-226-2180. E-mail: logical.tree@gmail.com Received November 6, 2014, Accepted December 1, 2014.
16 Ⅰ sclerosing polycystic adenosis; SPA 1996 Smith 1 67 1-12 SPA 1,2,5 18 SPA 1 3, 4, 5 21x15 Ⅱ 18 X- 1 2 MRI 2 class 10 19.5 16.0 14.3 MRI T1 T2 STIR MRI 1 X 2 2 2 1 MRI 2 A: HE x40 B: 2 HE x200
1 17 3 4 A HE x40 B HE x200 CHE x200 D HE x40 Ⅲ 5 A: calponin, B: p63 2 calponin p63 2 EBER in situ hybridization IgG4 SPA 3 sclerosing polycystic adenosis; SPA 1996 Smith 1 67 1-12 10 80 7 SPA
18 SPA SPA SPA SPA SPA SPA 5Küttner IgG4 12 SPA 5 IgG4 polycystic dysgenetic disease PDD5 PDD SPA 1,2,5 SPA 10 1,3,4,10 ductal carcinoma in situ DCIS 2,3,4,5,6,7,8 10 SPA adenosis Skalova PCR human androgen receptor: HUMARA X 6 EBER Epstein- Barr 9 PSA neoplasm Gnepp4 DCIS SPA SPA SUMMARY This 18-year-old female was incidentally found to have a left parotid gland tumor when she visited a local doctor with a complaint of pollen allergy. Aspiration cytology, performed twice, failed to give a definite diagnosis. The patient was referred to our hospital. The tumor was well-demarcated on ultrasonography and showed a heterogeneous image on MRI. Pleomorphic adenoma with hyalinized fibrosis was suspected on a core needle biopsy. The patient underwent superficial lobectomy of the left parotid gland. Gross examination of the resected specimen showed a 21mm-sized well-demarcated, solid and white tumor. Microscopically, it showed irregularly defined lobules composed of abundant hyalinized
1 19 fibrous tissue and variably sized collections of ducts with cystic change. Hyperplasia of ductal elements with a cribriform pattern and acinar cells with eosinophilic cytoplasmic granules were focally seen. Immunohistochemistry demonstrated preservation of the biphasic epithelial architecture with the basally situated cells of the ductal element positively stained for myoepithelial markers. We diagnosed the present case as sclerosing polycystic adenosis. At 3 years follow-up, the patient is free of recurrence and metastasis. Close follow-up, however, is necessary since there have been a few reported cases of SPA that recurred, harbored carcinoma within the lesion, or was shown to be a clonal process. 1 Smith BC, Ellis GL, Slater LJ, Foss RD. Sclerosing polycystic adenosis of major salivary glands. A clinicopathologic analysis of nine cases. Am J Surg Pathol 1996; 20: 161-70. 2 Skálová A, Michal M, Simpson R, Stárek I, Přádná J, Pfaltz M. Sclerosing polycystic adenosis of parotid gland with dysplasia and ductal carcinoma in situ. Virchows Arch 2002; 440: 29-35. 3 Petersson F. Sclerosing polycystic adenosis of salivary glands: a review with some emphasis on intraductal epithelial proliferations. Head Neck Pathol 2013; 7 Suppl 1: S97-106. 4 Gnepp DR. Salivary gland tumor wishes to add to the next WHO Tumor Classification: sclerosing polycystic adenosis, mammary analogue secretory carcinoma, cribriform adenocarcinoma of the tongue and other sites, and mucinous variant of myoepithelioma. Head Neck Pathol 2014; 8: 42-9. 5 Gnepp DR, Wang LJ, Brandwein-Gensler M, Slootweg P, Gill M, Hille J. Sclerosing polycystic adenosis of the salivary gland: a report of 16 cases. Am J Surg Pathol 2006; 30: 154-64. 6 Skalova A, Gnepp DR, Simpson RH, Lewis JE, Janssen D, Sima R, Vanecek T, Di Palma S, Michal M. Clonal nature of sclerosing polycystic adenosis of salivary glands demonstrated by using the polymorphism of the human androgen receptor HUMARA locus as a marker. Am J Surg Pathol 2006; 30: 939-44. 7 Bharadwaj G, Nawroz I, O Regan B. Sclerosing polycystic adenosis of the parotid gland. Br J Oral Maxillofac Surg 2007; 45: 74-76. 8 Fulciniti F, Losito NS, Ionna F, Longo F, Aversa C, Botti G, Foschini MP. Sclerosing polycystic adenosis of the parotid gland: Report of one case diagnosed by fine-needle cytology with in situ malignant transformation. Diagn Cytopathol 2010; 38: 368-73. 9 Swelam WM. The pathogenic role of Epstein- Barr virus EBV in sclerosing polycystic adenosis. Pathol Res Pract 2010; 206: 565-71. 10 Canas MR and Felix A. Invasive carcinoma arising from sclerosing polycystic adenosis of the salivary gland. Virchows Arch 2014; 464: 621-5. 11 Manojlovic S, Virag M, Milenovic A, Manojlovic L, Salek Z, Skalova A. Sclerosing polycystic adenosis of parotid gland: a unique report of two cases occurring in two sisters. Pathol Res Pract 2014; 210: 342-5. 12 Mokhtari S, Atarbashi Moghadam S, Mirafsharieh A. Sclerosing polycystic adenosis of the retromolar pad area: a case report. Case Rep Pathol 2014; 2014: 982432. 13 Kitagawa S, Zen Y, Harada K, Sasaki, M, Sato Y, Minato H, Watanabe K, Kurumaya H, Katayanagi K, Masuda S, Niwa H, Tsuneyama K, Saito K, Haratake J, Takagawa K, Nakanuma Y. Abundant IgG4-positive plasma cell infiltration characterizes chronic sclerosing sialadenitis Kuttner s tumor. Am J Surg Pathol 2005; 29: 783-91.