Skip to main content

Advertisement

Log in

Intraoral Salivary Duct Cyst: Clinical and Histopathologic Features of 177 Cases

  • Original Paper
  • Published:
Head and Neck Pathology Aims and scope Submit manuscript

Abstract

The salivary duct cyst (SDC) is a reactive ductal ectasia most frequently seen in major salivary glands, and likely caused by obstruction. The aim of this study is to define the clinical and histopathologic spectrum of intraoral SDCs. Cases were retrieved from the archives of Harvard School of Dental Medicine/StrataDx, Inc. from January 2012 to August 2014. There were 177 cases of which 103 (58.2%) occurred in females, with a median age of 56 (range 2–95). Approximately half of cases (45.8%) presented in the area of the buccal mucosa, lower lip mucosa, or mandibular vestibule, and 23.2% presented in the floor of mouth. SDCs were lined at least focally by 1–2 layers of cuboidal/columnar epithelium in 85.3% of cases and showed varying degrees of metaplasia (oncocytic, mucous cell, squamous, ciliated, apocrine-like) in 68.4% of cases. Intraluminal mucous stasis was present in 41.8% of SDCs, incipient calcification was present within 4.5% of SDCs, and chronic obstructive sialadenitis was seen in 90.2% of cases. No cysts showed adenomatous ductal proliferations or true papillary structures with fibrovascular cores, although 41.2% exhibited reactive undulation of cyst lining. Thirty-nine ‘papillary oncocytic cystadenoma-like’ SDCs (22.0%) demonstrated complete oncocytic metaplasia and marked undulation. An additional seven such cysts (4.0%) had a ‘Warthin tumor-like’ lymphoplasmacytic infiltrate. Intraoral SDCs occur most commonly in the sixth decade of life in locations distinct from extravasation mucoceles, likely secondary to intraluminal obstruction. SDCs show diverse histopathology and certain phenotypic variants may be mistaken for papillary oncocytic cystadenoma or Warthin tumor.

This is a preview of subscription content, log in via an institution to check access.

Access this article

Price excludes VAT (USA)
Tax calculation will be finalised during checkout.

Instant access to the full article PDF.

Fig. 1
Fig. 2
Fig. 3
Fig. 4
Fig. 5

Similar content being viewed by others

References

  1. Work WP. Cysts and congenital lesions of the parotid gland. Otolaryngol Clin North Am. 1977;10(2):339–43.

    PubMed  Google Scholar 

  2. Batsakis JG, Raymond AK. Sialocysts of the parotid glands. Ann Otol Rhinol Laryngol. 1989;98(6):487–9.

    Article  CAS  PubMed  Google Scholar 

  3. Ellis GL, Auclair PL, American Registry of Pathology, Armed Forces Institute of Pathology(U.S.). Tumors of the salivary glands. AFIP atlas of tumor pathology Fourth series, vol fasc 9. Washington, DC: American Registry of Pathology in collaboration with the Armed Forces Institute of Pathology; 2008.

    Google Scholar 

  4. Eversole LR. Oral sialocysts. Arch Otolaryngol—Head Neck Surg. 1987;113(1):51–6.

    Article  CAS  PubMed  Google Scholar 

  5. Southam JC. Retention mucoceles of the oral mucosa. J Oral Pathol. 1974;3(4):197–202.

    Article  CAS  PubMed  Google Scholar 

  6. Chi AC, Lambert PR 3rd, Richardson MS, Neville BW. Oral mucoceles: a clinicopathologic review of 1,824 cases, including unusual variants. J Oral Maxillofac Surg. 2011;69(4):1086–93. doi:10.1016/j.joms.2010.02.052.

    Article  PubMed  Google Scholar 

  7. Cohen L. Mucoceles of the oral cavity. Oral Surg Oral Med Oral Pathol. 1965;19:365–72.

    Article  CAS  PubMed  Google Scholar 

  8. Harrison JD. Salivary mucoceles. Oral Surg Oral Med Oral Pathol. 1975;39(2):268–78.

    Article  CAS  PubMed  Google Scholar 

  9. Yamasoba T, Tayama N, Syoji M, Fukuta M. Clinicostatistical study of lower lip mucoceles. Head Neck. 1990;12(4):316–20.

    Article  CAS  PubMed  Google Scholar 

  10. Oliveira DT, Consolaro A, Freitas FJ. Histopathological spectrum of 112 cases of mucocele. Braz Dent J. 1993;4(1):29–36.

    CAS  PubMed  Google Scholar 

  11. Granholm C, Olsson Bergland K, Walhjalt H, Magnusson B. Oral mucoceles; extravasation cysts and retention cysts. A study of 298 cases. Swed Dent J. 2009;33(3):125–30.

    PubMed  Google Scholar 

  12. Thompson LD. Mucocele: retention and extravasation types. Ear Nose Throat J. 2013;92(3):106–8.

    PubMed  Google Scholar 

  13. Seifert G. Tumour-like lesions of the salivary glands. The new WHO classification. Pathol Res Pract. 1992;188(7):836–46.

    CAS  PubMed  Google Scholar 

  14. Elzay RP, Sweeney WT, Peters PB, Malbon BA. Oral mucoceles: a review of 161 cases. Va Dent J. 1968;45(3):15–9.

    CAS  PubMed  Google Scholar 

  15. Cataldo E, Mosadomi A. Mucoceles of the oral mucous membrane. Arch Otolaryngol. 1970;91(4):360–5.

    Article  CAS  PubMed  Google Scholar 

  16. Testa Riva F, Riva A, Puxeddu P. Ciliated cells in the main excretory duct of the submandibular gland in obstructive sialadenitis: a SEM and TEM study. Ultrastruct Pathol. 1987;11(1):1–10.

    Article  CAS  PubMed  Google Scholar 

  17. Triantafyllou A, Hunt JL, Devaney KO, Ferlito A. A perspective of comparative salivary and breast pathology. Part I: microstructural aspects, adaptations and cellular events. Eur Arch oto-rhino-laryngol. 2014;271(4):647–63. doi:10.1007/s00405-013-2488-y.

    Article  Google Scholar 

  18. Triantafyllou A, Harrison JD, Garrett JR. Microenvironmental adaptations in the parotid of ferret investigated by electron microscopy. Arch Oral Biol. 2007;52(8):768–77. doi:10.1016/j.archoralbio.2007.01.010.

    Article  PubMed  Google Scholar 

  19. Marchal F, Kurt AM, Dulguerov P, Lehmann W. Retrograde theory in sialolithiasis formation. Arch Otolaryngol—Head Neck Surg. 2001;127(1):66–8.

    Article  CAS  PubMed  Google Scholar 

  20. Jensen JL, Howell FV, Rick GM, Correll RW. Minor salivary gland calculi. A clinicopathologic study of forty-seven new cases. Oral Surg Oral Med Oral Pathol. 1979;47(1):44–50.

    Article  CAS  PubMed  Google Scholar 

  21. Anneroth G, Hansen LS. Minor salivary gland calculi. A clinical and histopathological study of 49 cases. Int J Oral Surg. 1983;12(2):80–9.

    Article  CAS  PubMed  Google Scholar 

  22. Collins EM. Papillary cystadenoma of accessory salivary gland. Am J Surg. 1958;96(6):749–50.

    Article  CAS  PubMed  Google Scholar 

  23. Chaudhry AP, Gorlin RJ, Mitchell DF. Papillary cystadenoma of minor salivary gland origin. Report of a case. Oral Surg Oral Med Oral Pathol. 1960;13:452–4.

    Article  CAS  PubMed  Google Scholar 

  24. Calhoun NR, Cerine FC, Mathews MJ. Papillary cystadenoma of the upper lip. Report of a case. Oral Surg Oral Med Oral Pathol. 1965;20(6):810–3.

    Article  CAS  PubMed  Google Scholar 

  25. Parnes EI. Papillary cystadenoma. Report of a case. Oral Surg Oral Med Oral Pathol. 1966;21(6):782–5.

    Article  CAS  PubMed  Google Scholar 

  26. Wilson DF, MacEntee MI. Papillary cystadenoma of ectopic minor salivary gland origin. Oral Surg Oral Med Oral Pathol. 1974;37(6):915–8.

    Article  CAS  PubMed  Google Scholar 

  27. Kerpel SM, Freedman PD, Lumerman H. The papillary cystadenoma of minor salivary gland origin. Oral Surg Oral Med Oral Pathol. 1978;46(6):820–6.

    Article  CAS  PubMed  Google Scholar 

  28. Kameyama Y, Okada Y, Takehana S, Mizohata M, Nishio S, Enomoto M. Papillary cystadenoma. Int J Oral Surg. 1985;14(6):556–9.

    Article  CAS  PubMed  Google Scholar 

  29. Martin RW 3rd, Corio RL, Hood AF, Katz M. Papillary cystadenoma of the lower lip mimicking hidradenoma papilliferum. J Cutan Pathol. 1993;20(6):525–7.

    Article  PubMed  Google Scholar 

  30. Alexis JB, Dembrow V. Papillary cystadenoma of a minor salivary gland. J Oral Maxillofac Surg. 1995;53(1):70–2.

    Article  CAS  PubMed  Google Scholar 

  31. Guccion JG, Redman RS, Calhoun NR, Saini N. Papillary cystadenoma of the palate: a case report and ultrastructural study. J Oral Maxillofac Surg. 1997;55(7):759–64.

    Article  CAS  PubMed  Google Scholar 

  32. Mahler V, Schell H. Papillary cystadenoma: a rare tumor of the minor salivary glands. Eur J Dermatol. 1999;9(5):387–9.

    CAS  PubMed  Google Scholar 

  33. Tsurumi K, Kamiya H, Yokoi M, Kameyama Y. Papillary oncocytic cystadenoma of palatal minor salivary gland: a case report. J Oral Maxillofac Surg. 2003;61(5):631–3. doi:10.1053/joms.2003.50120.

    Article  PubMed  Google Scholar 

  34. Matsuzaka K, Kokubu E, Takeda E, Tanaka Y, Shimono M, Inoue T. Papillary cystadenoma arising from the upper lip: a case report. Bull Tokyo Dent Coll. 2003;44(4):213–6.

    Article  PubMed  Google Scholar 

  35. Ribeiro DA, Costa MR, de Assis GF. Papillary cystadenoma of the minor salivary gland of the lower lip. Dermatol Online J. 2004;10(1):14.

    PubMed  Google Scholar 

  36. Gallego L, Junquera L, Fresno MF, de Vicente JC. Papillary cystadenoma and cystadenocarcinoma of salivary glands: two unusual entities. Medicina oral, patologia oral y cirugia bucal. 2008;13(7):E460–3.

    PubMed  Google Scholar 

  37. Lim CS, Ngu I, Collins AP, McKellar GM. Papillary cystadenoma of a minor salivary gland: report of a case involving cytological analysis and review of the literature. Oral Surg Oral Med Oral Pathol Oral Radiol Endod. 2008;105(1):e28–e33. doi:10.1016/j.tripleo.2007.07.019.

    Article  PubMed  Google Scholar 

  38. Halbritter SA, Altermatt HJ, Caversaccio M, Bornstein MM. Apocrine papillary cystadenoma of a minor salivary gland on the lower lip: case presentation. Quintessence Int. 2009;40(2):167–9.

    PubMed  Google Scholar 

  39. Stathopoulos P, Gagari E. Papillary cystadenoma of the lower lip exhibiting ciliated pseudostratified columnar epithelium: report of a bizarre case and review of the literature. Oral Maxillofac Surg. 2013;17(3):161–4. doi:10.1007/s10006-012-0357-2.

    Article  PubMed  Google Scholar 

  40. Martins-Filho PR, Reinheimer DM, de Santana Santos T, Mascarenhas-Oliveira AC, Piva MR, Ferreira da Silva LC. Papillary cystadenoma of the upper lip. J Craniofac Surg. 2014;25(5):1932–3. doi:10.1097/SCS.0000000000000971.

    Article  PubMed  Google Scholar 

  41. Goto M, Ohnishi Y, Shoju Y, Wato M, Kakudo K. Papillary oncocytic cystadenoma of a palatal minor salivary gland: a case report. Oncol Lett. 2016;11(2):1220–2. doi:10.3892/ol.2015.3978.

    PubMed  Google Scholar 

  42. Tjioe KC, de Lima HG, Thompson LD, Lara VS, Damante JH, de Oliveira-Santos C. Papillary cystadenoma of minor salivary glands: report of 11 cases and review of the English literature. Head Neck Pathol. 2015;9(3):354–9. doi:10.1007/s12105-014-0602-0.

    Article  PubMed  Google Scholar 

  43. Goode RK, Auclair PL, Ellis GL. Mucoepidermoid carcinoma of the major salivary glands: clinical and histopathologic analysis of 234 cases with evaluation of grading criteria. Cancer. 1998;82(7):1217–24.

    Article  CAS  PubMed  Google Scholar 

  44. Brandwein MS, Ivanov K, Wallace DI, Hille JJ, Wang B, Fahmy A, et al. Mucoepidermoid carcinoma: a clinicopathologic study of 80 patients with special reference to histological grading. Am J Surg Pathol. 2001;25(7):835–45.

    Article  CAS  PubMed  Google Scholar 

  45. Fonseca FP, de Andrade BA, Lopes MA, Pontes HA, Vargas PA, de Almeida OP. P63 expression in papillary cystadenoma and mucoepidermoid carcinoma of minor salivary glands. Oral surgery oral medicine oral pathology oral radiology. 2013;115(1):79–86. doi:10.1016/j.oooo.2012.09.005.

    Article  Google Scholar 

  46. Lanzel EA, Pourian A, Sousa Melo SL, Brogden KA, Hellstein JW. Expression of membrane-bound mucins and p63 in distinguishing mucoepidermoid carcinoma from papillary cystadenoma. Head Neck Pathol. 2016. doi:10.1007/s12105-016-0735-4.

    PubMed  PubMed Central  Google Scholar 

  47. Molyneux EM, Rochford R, Griffin B, Newton R, Jackson G, Menon G, et al. Burkitt’s lymphoma. The Lancet. 2012;379(9822):1234–44. doi:10.1016/S0140-6736(11)61177-X.

    Article  Google Scholar 

  48. Iwai T, Baba J, Murata S, Mitsudo K, Maegawa J, Nagahama K, et al. Warthin tumor arising from the minor salivary gland. J Craniofac Surg. 2012;23(5):e374–e6. doi:10.1097/SCS.0b013e318254359f.

    Article  PubMed  Google Scholar 

  49. Takezawa K, Jackson C, Gnepp DR, King TC. Molecular characterization of Warthin tumor. Oral Surg Oral Med Oral Pathol Oral Radiol Endod. 1998;85(5):569–75.

    Article  CAS  PubMed  Google Scholar 

  50. Honda K, Kashima K, Daa T, Yokoyama S, Nakayama I. Clonal analysis of the epithelial component of Warthin’s tumor. Hum Pathol. 2000;31(11):1377–80.

    Article  CAS  PubMed  Google Scholar 

  51. Patel DK, Morton RP. Demographics of benign parotid tumours: Warthin’s tumour versus other benign salivary tumours. Acta Otolaryngol. 2016;136(1):83–6. doi:10.3109/00016489.2015.1081276.

    Article  PubMed  Google Scholar 

  52. Teymoortash A, Werner JA. Tissue that has lost its track: Warthin’s tumour. Virchows Arch. 2005;446(6):585–8. doi:10.1007/s00428-005-1276-5.

    Article  CAS  PubMed  Google Scholar 

  53. Seifert G, Bull HG, Donath K. Histologic subclassification of the cystadenolymphoma of the parotid gland. Analysis of 275 cases. Virchows Arch. 1980;388(1):13–38.

    Article  CAS  Google Scholar 

  54. Park CK, Manning JT Jr, Battifora H, Medeiros LJ. Follicle center lymphoma and Warthin tumor involving the same anatomic site. Report of two cases and review of the literature. Am J Clin Pathol. 2000;113(1):113–9. doi:10.1309/MJH0-RQGX-U128-VFC6.

    Article  CAS  PubMed  Google Scholar 

  55. Hwang JY, Kim SW, Yang SC, Kim CD. Extraparotid Warthin tumor in upper cervical lymph node accompanied by primary cervical tuberculosis. Otolaryngol Head Neck Surg. 2011;144(4):646–7. doi:10.1177/0194599811398187.

    Article  PubMed  Google Scholar 

  56. Fantasia JE, Miller AS. Papillary cystadenoma lymphomatosum arising in minor salivary glands. Oral Surg Oral Med Oral Pathol. 1981;52(4):411–6.

    Article  CAS  PubMed  Google Scholar 

Download references

Funding

No funding was received during the study or in preparation of the manuscript.

Author information

Authors and Affiliations

Authors

Corresponding author

Correspondence to Ivan J. Stojanov.

Ethics declarations

Conflict of interest

The authors declare that they have no conflict of interest.

Ethical Approval

This article does not contain any studies with human participants or animals performed by any of the authors.

Informed Consent

Informed consent was not obtained as this study consists of secondary research performed on excess tissue of specimens used for diagnostic purposes. Research was conducted using deidentified samples and private patient information was not accessed at any point during the design of the study, data collection/interpretation, or manuscript preparation.

Rights and permissions

Reprints and permissions

About this article

Check for updates. Verify currency and authenticity via CrossMark

Cite this article

Stojanov, I.J., Malik, U.A. & Woo, SB. Intraoral Salivary Duct Cyst: Clinical and Histopathologic Features of 177 Cases. Head and Neck Pathol 11, 469–476 (2017). https://doi.org/10.1007/s12105-017-0810-5

Download citation

  • Received:

  • Accepted:

  • Published:

  • Issue Date:

  • DOI: https://doi.org/10.1007/s12105-017-0810-5

Keywords

Navigation