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Year : 2021  |  Volume : 64  |  Issue : 2  |  Page : 424-426
Ceruminous adenocarcinoma of the external auditory canal- report of a rare case with a discussion of its differential diagnoses

Department of Pathology, All India Institute of Medical Sciences, Raipur, India

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Date of Submission06-Apr-2020
Date of Decision14-May-2020
Date of Acceptance05-Jul-2020
Date of Web Publication9-Apr-2021

How to cite this article:
Singh VY, Sarangi S, Agrawal K. Ceruminous adenocarcinoma of the external auditory canal- report of a rare case with a discussion of its differential diagnoses. Indian J Pathol Microbiol 2021;64:424-6

How to cite this URL:
Singh VY, Sarangi S, Agrawal K. Ceruminous adenocarcinoma of the external auditory canal- report of a rare case with a discussion of its differential diagnoses. Indian J Pathol Microbiol [serial online] 2021 [cited 2021 May 8];64:424-6. Available from: https://www.ijpmonline.org/text.asp?2021/64/2/424/313281

Dear Editor,

Ceruminous gland adenocarcinomas are extremely rare malignancies having an incidence of 2-4% among ear neoplasms.[1] These tumours are a diagnostic dilemma due to their unusual sites. The case in discussion is a 44 year female who presented with a tender, firm swelling in the cartilagenous portion of external auditory canal (EAC). A clinical diagnosis of chondroma was made. Histopathologic examination revealed the tumour to be a ceruminous gland adenocarcinoma. Ceruminous adenocarcinomas, especially the adenoid cystic variant has a prolonged course with metastatic disease after recurrences in approximately 50% of patients.[2]The purpose of this case report is to highlight the importance of an accuratediagnosis of these tumours, which might prevent incomplete management and recurrences.

A 44 year old female presented to the outdoor complaining of pain and reduced hearing in the left ear. A contrast enhanced CT scanof the head revealed a heterogenously enhancing soft tissue attenuation in cartilaginous part of left EAC in its medial most part. A fine needle aspiration was attempted from the mass but yielded a bloody aspirate. The lesion was operated on and submitted for histopathological examination. The gross examination revealed a single soft tissue piece measuring 1 x 1 x 0.4 cm.

Microscopic examination revealed a tissue lined by stratified squamous epithelium with an underlying invasive tumour arranged in glandular pattern with focal cribriforming [Figure 1]. The individual cells were round to oval and uniform with high nucleo-cytoplasmic ratio and inconspicuous nucleoli [Figure 2]. There was no evidence of any perineural or lymphovascular invasion. The tumour was infiltrating in nature. Immunohistochemistry for p63 was found to be focal in the cribriforming areas, highlighting the myoepithelial cell layer [Figure 3]. A diagnosis of ceruminous gland adenocarcinoma was rendered and the patient was summoned for a revised margin clearance surgery. The patient is currently under close follow up.
Figure 1: Photomicrograph shows tissue lined by stratified squamous epithelium with underlying tumour arranged in nested, acinar and focal cribriform pattern. (40X, HE)

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Figure 2: Photomicrograph shows tumour arranged in nested, acinar and focal cribriform pattern along with inset showing individual tumour cells (100X, HE; inset 400X, HE)

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Figure 3: Photomicrograph shows myoepithelial cell layer immunoreactive for p63 in the cribriforming area.(400X, IHC, p63)

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The skin of the cartilaginous part of the external auditory canal is characterized by the presence of hair follicles, sebaceous glands and ceruminous glands.[3] Ceruminous glands are modified apocrine glands and the their neoplasms are classified into benign and malignant subtypes, which comprises of ceruminous adenoma, ceruminous pleomorphic adenoma, ceruminous syringocystadenoma papilliferum and ceruminous adenocarcinoma, not otherwise specified (NOS), ceruminous adenoid cystic carcinoma and ceruminous mucoepidermoid carcinoma respectively.[2] All the known cases studied till now have been reported in adults with the mean age group being 33-82 years. A slight female preponderance has been recorded in some studies.[4] Our case concurred with respect to age and gender. It has been observed that malignancy is noted more in the younger age group than in adults. The patients generally present with complains of pain, tinnitus, diminished hearing, discharge, and infection.[4] The case under study presented with pain and diminished hearing. Aggressive tumours tend to erode the brain parenchyma leading to additional symptoms like syncope, paresthesias, paralysis or visual changes. Given the rarity of these tumours, it might be mistaken for chondromas or lesions of infective origin. These tumours generally present as a small nodule[4] in the deep dermis, but since the thickness of the skin of external auditory canal is not much, the tumour tends to be quite superficial. The nodule ranges in size from 1 to 3 cm. In some cases, these tumours are associated with cholesteatoma.

Microscopically these tumours are infiltrative and show high degree of cellular pleomorphism which helps in delineating malignant tumours from the benign ones. The tumour can show varied architectural patterns like back-to-back glandular arrangement along with focal papillary patterns. The glands might show decapitation secretion and the individual cells may show prominent nucleoli. There might be associated tumour necrosis which may aid in the diagnosis of malignancy. Despite the above features, it may be difficult to distinguish adenomas from carcinomas[4] until the margin invasion is demonstrated. These tumours can be classified as tumours of uncertain malignant potential unless proved otherwise. It has been seen that the ceruminous glands show dual cell differentiation with intact myoepithelial cell component demonstrated by P 63.[1] Our case had glandular arrangement with focal cribriforming and showed focal immunoreactivity for p63.

Ceruminous gland adenocarcinomas have high propensity for local recurrence, especially the adenoid cystic variant. A radical surgery is always recommended though for a lesion so small, radical procedures are never the first choice.[5] Frequent diagnostic dilemma leads to incomplete resection and hence higher chances of recurrence and metastasis later. In majority of the cases, the patient has to be kept on close follow up or operated on again after the biopsy report so as to ensure a radical excision.[5] The most common differential diagnoses of this condition are pleomorphic adenoma, middle ear adenoma, adnexal tumours, neuroendocrine carcinomas, chondromas and low grade adenocarcinoma. Ceruminal adenocarcinomas can be distinguished from these differentials by the decapitation secretion of these tumours along with their features of fibrosis, infiltration and dual cell differentiation.[1] Prognosis of such tumours is difficult to predict, but complete surgical excision coupled with radiation yields the best long-term results.

The above case helps to emphasize the fact that external auditory canal malignancies can be mistaken for benign tumours due to their bland appearance, small size and dual cell differentiation. Hence utmost care should be taken during excision and histopathological examination of the tumour so as to avoid incomplete management due to a missed diagnosis.

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Conflicts of interest

There are no conflicts of interest.

   References Top

Crain N, Nelson BL, Bames EL, Thompson LD. Ceruminous gland carcinomas: A clinicopathologic and immunophenotypic study of 17 cases. Head Neck Pathol 2009;3:1–17.  Back to cited text no. 1
El-Naggar AK. World Health Organization Classification of Head and Neck Tumours. 4th ed. Lyon: IARC press; 2017.  Back to cited text no. 2
Schwarz D, Drebber U, Ortmann M, Anagiotos A. Benign cylindroma: a rare differential diagnosis of external ear canal tumour. BMJ Case Rep 2016;2016:bcr2015212035.  Back to cited text no. 3
Nagarajan P. Ceruminous Neoplasms of the Ear. Head Neck Pathol 2018;12:350–61.  Back to cited text no. 4
Chung JH, Lee SH, Park CW, Tae K. Mucoepidermoid carcinoma in the external auditory canal: a case report. Cancer Res Treat 2012;44:275–8.  Back to cited text no. 5

Correspondence Address:
Sujata Sarangi
Department of Pathology, All India Institute of Medical Sciences, Raipur
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Source of Support: None, Conflict of Interest: None

DOI: 10.4103/IJPM.IJPM_326_20

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  [Figure 1], [Figure 2], [Figure 3]


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