Virchows Archiv

, Volume 445, Issue 6, pp 641–646 | Cite as

Malignant transformation of sialadenoma papilliferum of the palate: a case report

  • Masayuki Shimoda
  • Kaori Kameyama
  • Shojiroh Morinaga
  • Youichi Tanaka
  • Kazuhiro Hashiguchi
  • Megumi Shimada
  • Yasunori Okada
Case Report


We report a case of a 79-year-old woman with an unusual salivary gland tumor that developed at the junction between the soft and hard palates. The tumor consisted of sialadenoma papilliferum (SP) with areas of an epithelial–myoepithelial carcinoma (EMC) component and a high-grade carcinoma component. There were also transitional regions among the SP, the EMC and the high-grade carcinoma components. The high-grade carcinoma component, which was similar to invasive micropapillary carcinoma of the breast, infiltrated into the right parapharyngeal space and metastasized to the lungs and cervical vertebrae. The high-grade carcinoma cells were positively immunostained for p53 protein. SP has been considered to be a benign tumor with exceptionally good prognosis, and, to the best of our knowledge, there has never been a confirmed case of malignant SP. This is the first report of SP with a definite malignant component.


Sialadenoma papilliferum Epithelial–myoepithelial carcinoma High-grade carcinoma p53 


  1. 1.
    Abrams AM, Flinck FM (1969) Sialadenoma papilliferum. A previously unreported salivary gland tumor. Cancer 24:1057–1063PubMedGoogle Scholar
  2. 2.
    Alos L, Carrilo R, Ramos J, Baez JM, Mallofre C, Fernandez PL, Cardesa A (1999) High-grade carcinoma component in epithelial-myoepithelial carcinoma of salivary glands clinicopathological, immunohistochemical and flow-cytometric study of three cases. Virchows Arch 434:291–299CrossRefPubMedGoogle Scholar
  3. 3.
    Asahina I, Abe M (1997) Sialadenoma papilliferum of the hard palate: a case report and review of literature. J Oral Maxillofac Surg 55:1000–1003CrossRefPubMedGoogle Scholar
  4. 4.
    Chau Y, Hongyo T, Aozasa K, Chan JK (2001) Dedifferentiation of adenoid cystic carcinoma: report of a case implicating p53 gene mutation. Hum Pathol 32:1403–1407CrossRefPubMedGoogle Scholar
  5. 5.
    Cheuk W, Chan J, Ngan RKC (1999) Dedifferentiation in adenoid cystic carcinoma of salivary gland. A common complication associated with an accelerated clinical course. Am J Surg Pathol 23:465–472CrossRefPubMedGoogle Scholar
  6. 6.
    Dahlin DC, Beabout JW (1971) Dedifferentiation in low-grade chondrosarcoma. Cancer 28:461–466PubMedGoogle Scholar
  7. 7.
    Di Palma S, Corletto V, Lovarino C Birindelli S, Pilotti S (1999) Unilateral aneuploid dedifferentiated acinic cell carcinoma associated with bilateral low-grade diploid acinic cell carcinoma of the parotid gland. Virchows Arch 434:361–365CrossRefPubMedGoogle Scholar
  8. 8.
    Di Palma S, Corletto V, Lovarino C, Birindelli, Pilotti S (2000) Dedifferentiation in salivary gland carcinoma. Am J Surg Pathol 24:1439–1440PubMedGoogle Scholar
  9. 9.
    Ellis GL, Auclair PL (1995) Tumors of the salivary glands. Armed Forced Institute of Pathology, Washington, DCGoogle Scholar
  10. 10.
    Evans HL (1979) Liposarcoma: a study of 55 cases with a reassessment of its classification. Am J Surg Pathol 3:507–523PubMedGoogle Scholar
  11. 11.
    Fantasia JE, Nocco CE, Lally ET (1986) Ultrastructure of sialadenoma papilliferum. Arch Pathol Lab Med 110:523–527PubMedGoogle Scholar
  12. 12.
    Fonseca I, Felix A, Soarses J (2000) Dedifferentiation in salivary gland carcinoma. Am J Surg Pathol 24:469–471CrossRefPubMedGoogle Scholar
  13. 13.
    Foschini MP, Eusebi V (1998) Carcinomas of the breast showing myoepithelial cell differentiation. A review of the literature. Virchows Arch 432:303–310CrossRefPubMedGoogle Scholar
  14. 14.
    Gallo O, Franchi A, Bianchi S, Boddi V, Giannelli E, Alajmo E (1995) p53 oncoprotein expression in parotid gland carcinoma is associated with clinical outcome. Cancer 75:2037–2044PubMedGoogle Scholar
  15. 15.
    Henley JD, Geary WA, Jackson CL, Wu CD, Gnepp DR (1997) Dedifferentiated acinic cell carcinoma of the parotid gland: a distinct rarely described entity. Hum Pathol 28:869–873CrossRefPubMedGoogle Scholar
  16. 16.
    Maiorano E, Favia G, Ricco R (1996) Sialadenoma papilliferum: an immunohistochemical study of five cases. J Oral Pathol Med 25:336–342PubMedGoogle Scholar
  17. 17.
    Meis JM (1991) “Dedifferentiation” in bone and soft tissue tumors: a histological indicator of tumor progression. Pathol Annu 26:37–62PubMedGoogle Scholar
  18. 18.
    Michal M, Skalova A, Mukensnabl P (2000) Micropapillary carcinoma of the parotid gland arising in mucinous cystadenoma. Virchows Arch 437:465–468CrossRefPubMedGoogle Scholar
  19. 19.
    Mills SE, Garland TA, Allen MS (1984) Low-grade papillary adenocarcinoma of palatal salivary gland origin. Am J Surg Pathol 8:367–374PubMedGoogle Scholar
  20. 20.
    Mostofi R, Wood RS, Christison W, Talerman A (1992) Low-grade papillary adenocarcinoma of minor salivary glands. Case report and literature review. Oral Surg Oral Med Oral Pathol 73:591–595PubMedGoogle Scholar
  21. 21.
    Nakahata A, Deguchi H, Yanagawa T, Yoshida H, Sato M, Hayashi Y (1990) Coexpression of intermediate-sized filaments in sialadenoma papilliferum and other salivary gland neoplasms. J Oral Pathol Med 19:313–318PubMedGoogle Scholar
  22. 22.
    Piana S, Cavazza A, Pedroni C, Scotti R, Serra L, Gardini G (2002) Dedifferentiated acinic cell carcinoma of the parotid gland with myoepithelial features. Arch Pathol Lab Med 126:1104–1105PubMedGoogle Scholar
  23. 23.
    Pimentel MT, Lopez Amado M, Garcia Sarandeses (1995) A Recurrent sialadenoma papilliferum of the buccal mucosa. J Laryngol Otol 109:787–790PubMedGoogle Scholar
  24. 24.
    Rennie JS, MacDonald DG, Critchlow HA (1984) Sialadenoma papilliferum. A case report and review of the literature. Int J Oral Surg 13:452–454PubMedGoogle Scholar
  25. 25.
    Shirasuna K, Watatani K, Miyazaki T (1984) Ultrastructure of a sialadenoma papilliferum. Cancer 53:468–474PubMedGoogle Scholar
  26. 26.
    Simpson RH, Prasad AR, Lewis JE, Skalova A, David L (2003) Mucin-rich variant of salivary duct carcinoma: a clinicopathologic and immunohistochemical study of four cases. Am J Surg Pathol 27:1070–1079Google Scholar
  27. 27.
    Solomon MP, Rosen Y, Alfonso A (1978) Intraoral papillary squamous cell tumor of the soft palate with features of sialadenoma papilliferum-? malignant sialadenoma papilliferum. Cancer 42:1859–1869PubMedGoogle Scholar
  28. 28.
    Stanley RJ, Weiland LH, Olsen KD, Pearson BW (1988) Dedifferentiated acinic cell (acinous) carcinoma of the parotid gland. Otolaryngol Head Neck Surg 98:155–161PubMedGoogle Scholar
  29. 29.
    Ubaidat MA, Robinson RA, Belding PJ, Merryman DJ (2001) Sialadenoma papilliferum of the hard palate: report of 2 cases and immunohistochemical evaluation. Arch Pathol Lab Med 125:1595–1597PubMedGoogle Scholar

Copyright information

© Springer-Verlag 2004

Authors and Affiliations

  • Masayuki Shimoda
    • 1
  • Kaori Kameyama
    • 2
  • Shojiroh Morinaga
    • 3
  • Youichi Tanaka
    • 4
  • Kazuhiro Hashiguchi
    • 5
  • Megumi Shimada
    • 6
  • Yasunori Okada
    • 1
  1. 1.Department of PathologySchool of Medicine, Keio UniversityTokyoJapan
  2. 2.Division of Diagnostic PathologyKeio University HospitalTokyoJapan
  3. 3.Division of Diagnostic PathologyKitasato Institute HospitalTokyoJapan
  4. 4.Division of Surgical PathologyTokyo Dental College Ichikawa General HospitalChibaJapan
  5. 5.Division of OtorhinolaryngologyKitasato Institute HospitalTokyoJapan
  6. 6.Division of Internal MedicineKitasato Institute HospitalTokyoJapan

Personalised recommendations