Indian Journal of Pathology and Microbiology
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Year : 2021  |  Volume : 64  |  Issue : 2  |  Page : 426-427
Syringocystadenoma Papilliferum of the middle ear: Common tumor at rare site

Department of Pathology, All India Institute of Medical Sciences, New Delhi, India

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Date of Submission02-Mar-2020
Date of Decision09-Apr-2020
Date of Acceptance15-May-2020
Date of Web Publication9-Apr-2021

How to cite this article:
Arava S, Nakhra T. Syringocystadenoma Papilliferum of the middle ear: Common tumor at rare site. Indian J Pathol Microbiol 2021;64:426-7

How to cite this URL:
Arava S, Nakhra T. Syringocystadenoma Papilliferum of the middle ear: Common tumor at rare site. Indian J Pathol Microbiol [serial online] 2021 [cited 2021 Aug 5];64:426-7. Available from: https://www.ijpmonline.org/text.asp?2021/64/2/426/313270

Dear Editor,

Syringocystadenoma papilliferum (SCAP) or ceruminoma is one of the rare begin Tumor that originates from apocrine sweat glands or ceruminous glands. The most common location is in the scalp and face, around 30%–40% is usually associated with a congenital skin lesion known as nevus sebaceous.[1],[2],[3] Nevus sebaceous is commonly seen in the early childhood and clinically presents as a linear, yellowish papules and nodules with absence of hair, especially in the scalp region. This lesion gradually increases in size till puberty and becomes papillomatous and crusted in appearance. The other less common sites of occurrence of SCAP are chest, upper arms, breast, eyelids, scrotum, and thigh region. SCAP in the external auditory canal is extremely rare and to the best of our knowledge, there are only about 11 cases reported in the literature.[2],[4] This case is of importance as it may elicit various clinical and diagnostic differentials due to its rare site and varied clinical presentation.

The present case is a 15-year-old male with a history of chronic otitis media presented to the ENT OPD with a history of ear pain and discharge. On otoscopy, a tiny mass measuring less than 0.5 cm was found to be protruding out from the right external auditory canal. It was excised and sent for histopathological confirmation with a clinical differential of chronic granulation tissue or any other infective etiology. Microscopic examination showed typical histopathological features of SCAP [Figure 1].
Figure 1: (a) Cystic invagination with papillary projections in the cavity. (b) Cyst and papillae are lined by two layers of cuboidal epithelium (arrow). (c) Stroma showing many lymphoplasmacytic cells (star) (d) Epithelial membrane antigen (EMA) immunostain highlighting the epithelium

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Commonly SCAP has varied clinical presentation and most common presentation includes raised, warty plaque or irregular, flat, gray or reddened surface. It measures from 1–3 cm in diameter and is usually solitary.[5]

Microscopically, SCAP reveal well Demarcated lesion characterized by cystic and papillary invagination from the surface epithelium. Invaginated part shows double layer of lining epithelium consisting of outer cuboidal and inner columnar epithelium with presence of apical snouts. Intervening stroma shows variable lymphoplasmacytic infiltration. The ductal structure which connects to the surface epithelium shows stratified squamous epithelial lining.[1] The main histopathological differential that should be considered is hydradenoma papilliferum in which there will be no stromal lymphoplasmacytic infiltration.[1]

Normally, the outer half of the external auditory canal contains ceruminous glands and the inner half is bone part, which is devoid of these glands. Together with modified apocrine gland and sebaceous gland, earwax is produced. Neoplastic transformation of these glands at this site is extremely rare. Occasional cases of SCAP arising from the inner bony part of the middle ear are also reported.[1],[2],[3] Because of its rare site, it elicits various clinical and radiological differentials during the evaluation. An idea of this tumor arising in the middle ear will be helpful for the pathologist for the definitive diagnosis. As this tumor is benign, complete surgical excision is the treatment of choice without any recurrences. Occasional cases of malignant transformation in SCAP have also been reported in the literature but it is extremely rare.[4]

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form the patient(s) has/have given his/her/their consent for his/her/their images and other clinical information to be reported in the journal. The patients understand that their names and initials will not be published and due efforts will be made to conceal their identity, but anonymity cannot be guaranteed.

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Conflicts of interest

There are no conflicts of interest.

   References Top

Bruschini L, Ciabotti A, De Vito A, Forli F, Cambi C, Ciancia EM, et al. Syringocystadenoma papilliferum of the external auditory canal. Am J Case Rep 2017;18:520-4.  Back to cited text no. 1
Arechvo A, Balseris S, Neverauskiene L, Arechvo I. Syringocytadenoma papilleferum of the bony external auditory canal: A rare tumor in a rare location. Case Rep Otolaryngol 2013:541679. doi: 10.1155/2013/541679.  Back to cited text no. 2
Guerra-Jiménez G, González Aguado R, Arenas Rodríguez A, Ramos Macías Á. Syringocystadenoma papilliferum of the external auditory canal. Case report and literature review. Acta Otorrinolaringol Esp 2017; 68:235-7.  Back to cited text no. 3
Brent AJ, Mota PM, Nebojsa A, Berry-Brincat A, Knapp CM. Squamous cell carcinoma arising from syringocystadenoma papilliferum of the eyelid. Can J Ophthalmol 2017;52:e235-7.  Back to cited text no. 4
Agarwal R, Kumar P, Varshney R. Syringocystadenoma papilleferum-an unusual presentation. J Clin Diagn Res 2014;8:3-4.  Back to cited text no. 5

Correspondence Address:
Sudheer Arava
Department of Pathology, All India Institute of Medical Sciences, Ansari Nagar, New Delhi - 110 029
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Source of Support: None, Conflict of Interest: None

DOI: 10.4103/IJPM.IJPM_159_20

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