Skip to main content

Syringocystadenoma papilliferum in the middle ear

Abstract

Syringocystadenoma papilliferum is a rare adnexal tumor of controversial origin that often occurs as a solitary tumor derived from apocrine and eccrine glands. It is more common in children and adolescents and affects the head and neck regions, although occurrences on other anatomical locations have been described. Routine H&E-stained sections revealed cystic epidermal invaginations with papillary projections. The superficial portions of the cyst were lined by stratified keratinizing epithelium, whereas the deeper papillated portion exhibited a double layer of basal-like cells and luminal eosinophilic columnar cells with focal decapitation secretions. A female patient, aged 29 years, presented to us in the outpatient clinic suffering from left earache and hearing loss since the past 3 months, with gradual onset and a slowly progressive course. On examination, we found a left aural polyp, which was firm and grayish in color, but extremely tender on touch. Biopsy for histopathological examination was consistent with a diagnosis of syringocystadenoma papilliferum. The patient received regular follow-up and is doing well with regard to her earache with marked hearing improvement. To conclude, solitary lesions seen in unusual locations generate multiple differential diagnoses and should be confirmed by histopathology in order to exclude other pathologies and define the appropriate therapeutic approach.

References

  1. 1

    Malhotra P, Singh A, Ramesh V. Syringocystadenoma papilliferum on the thigh: an unusual location. Indian J Dermatol Venereol Leprol 2009; 75:170–172.

    Article  Google Scholar 

  2. 2

    Sood A, Khaitan BK, Khanna N, Kumar R, Singh MK. Syringocystadenoma papilliferum at unusual sites. Indian J Dermatol Venereol Leprol 2000; 66:328–329.

    CAS  PubMed  Google Scholar 

  3. 3

    Yap FB, Lee BR, Baba R. Syringocystadenoma papilliferum in an unusual location beyond the head and neck region: a case report and review of literature. Dermatol Online J 2010; 16:4.

    PubMed  Google Scholar 

  4. 4

    Ghosh SK, Bandyopadhyay D, Chatterjee G, Bar C. Syringocystadenoma papilliferum: an unusual presentation. Pediatr Dermatol 2009; 26:758–759.

    Article  Google Scholar 

  5. 5

    Pahwa P, Kaushal S, Gupta S, Khaitan BK, Sharma VK, Sethuraman G. Linear syringocystadenoma papilliferum: an unusual location. Pediatr Dermatol 2011; 28:61–62.

    Article  Google Scholar 

  6. 6

    Hyams VJ, Michaels L. Benign adenomatous neoplasm (adenoma) of the middle ear. Clin Otolaryngol Allied Sci 1976; 1:17–26.

    CAS  Article  Google Scholar 

  7. 7

    Quaranta A, Mininni F, Resta L. Salivary gland choristoma of the middle ear: a case report. J Laryngol Otol 1981; 95:953–956.

    CAS  Article  Google Scholar 

  8. 8

    Derlacki EL, Barney PL. Adenomatous tumors of the middle ear and mastoid. Laryngoscope 1976; 86:1123–1135.

    CAS  Article  Google Scholar 

  9. 9

    Jahrdoerfer RA, Fechner RE, Selman JW, Moon CNJr, Powell JB. Adenoma of the middle ear. Laryngoscope 1983; 93:1041–1044.

    Google Scholar 

  10. 10

    Diamandopolous GTh, Meissner VA. Neoplasia. In: Kissane JH, editor. Andersen’s pathology. 8th ed. St Louis: CV Mosby; 1985. 378–382.

    Google Scholar 

  11. 11

    Mills SE, Fechner RE. Middle ear adenoma. A cytologically uniform neoplasm displaying a variety of architectural patterns. Am J Surg Pathol 1984; 8:677–685.

    CAS  PubMed  Google Scholar 

  12. 12

    Kar M, Kar JK, Maiti S. Giant linear syringocystadenoma papilliferum of the back. Indian J Dermatol Venereol Leprol 2012; 78:123.

    Article  Google Scholar 

  13. 13

    Yoshii N, Kanekura T, Setoyama M, Kanzaki T. Syringocystadenoma papilliferum: report of the first case on the lower leg. J Dermatol 2004; 31:939–942.

    Article  Google Scholar 

  14. 14

    Sangma MM, Dasiah SD, Bhat V R. Syringocystadenoma papilliferum of the scalp in an adult male - a case report. J Clin Diagn Res 2013; 7:742–743.

    PubMed  PubMed Central  Google Scholar 

  15. 15

    Rammeh-Rommani S, Fezaa B, Chelbi E, Kammoun MR, Ben Jilani SB, Zermani R. Syringocystadenoma papilliferum: report of 8 cases. Pathologica 2006; 98:178–180.

    CAS  PubMed  Google Scholar 

  16. 16

    Dufrechou L, Acosta A, Beltramo P, Pomies V, Caruso R, Salmenton GM, Alvarez M. Syringocystadenoma papilliferum arising on the scrotum. Pediatr Dermatol 2013; 30:e12–e13.

    Article  Google Scholar 

  17. 17

    Kim MS, Lee JH, Lee WM, Son SJ. A case of tubular apocrine adenoma with syringocystadenoma papilliferum that developed in a nevus sebaceous. Ann Dermatol 2010; 22:319–322.

    Article  Google Scholar 

  18. 18

    Bruno CB, Cordeiro FN, Soares Fdo E, Takano GH, Mendes LS. Dermoscopic aspects of syringocystadenoma papilliferum associated with nevus sebaceus. An Bras Dermatol 2011; 86:1213–1216.

    Article  Google Scholar 

  19. 19

    Rao VA, Kamath GG, Kumar A. An unusual case of syringocystadenoma papilliferum of the eyelid. Indian J Ophthalmol 1996; 44:168–169.

    CAS  PubMed  Google Scholar 

  20. 20

    Michaels L, Wells M. Squamous cell carcinoma of the middle ear. Clin Otolaryngol Allied Sci 1980; 5:235–248.

    CAS  Article  Google Scholar 

  21. 21

    Hill BA, Kohut RI. Metastatic adenocarcinomas of the temporal bone. Arch Otolaryngol 1976; 102:568–571.

    CAS  Article  Google Scholar 

Download references

Author information

Affiliations

Authors

Corresponding author

Correspondence to Wael A. Alzamil.

Additional information

This is an open access article distributed under the terms of the Creative Commons Attribution-NonCommercial-ShareAlike 3.0 License, which allows others to remix, tweak, and build upon the work non-commercially, as long as the author is credited and the new creations are licensed under the identical terms.

Rights and permissions

This article is licensed under a Creative Commons Attribution 4.0 International License, which permits use, sharing, adaptation, distribution and reproduction in any medium or format, as long as you give appropriate credit to the original author(s) and the source, provide a link to the Creative Commons licence, and indicate if changes were made. The images or other third party material in this article are included in the article's Creative Commons licence, unless indicated otherwise in a credit line to the material. If material is not included in the article's Creative Commons licence and your intended use is not permitted by statutory regulation or exceeds the permitted use, you will need to obtain permission directly from the copyright holder. To view a copy of this licence, visit http://creativecommons.org/licenses/by/4.0/.

Reprints and Permissions

About this article

Verify currency and authenticity via CrossMark

Cite this article

Alzamil, W.A. Syringocystadenoma papilliferum in the middle ear. Egypt J Otolaryngol 33, 124–127 (2017). https://doi.org/10.4103/1012-5574.199403

Download citation

Keywords

  • adnexal tumor of the middle ear
  • middle-ear adenoma
  • syringocystadenoma papilliferum in the middle ear