Ceruminous Adenoma: Rare Neoplasm in Human being. Case Presentation

Introduction

The external auditory canal contains both sebaceous and modified sweat or apocrine (ceruminous) glands located in the deep stroma.

Tumors originating in these latter glands are common in canines and felines and very rare in humans. These tumors have been labeled with different terminologies since the 1950´s. It is still a neoplasm of difficult differential diagnosis with malignant neoplasms. There is much controversy regarding the nomenclature, origin, histology, classification and pathology of these lesions. Due to the above, this clinical case is presented and a recount of the pathology is made for its dissemination among health professionals.

Clinical Case

A 54-year-old male, with no relevant history, who reported right hearing loss, aural fullness, mild otalgia and occasional ipsilateral otorrhea of ​months of evolution with no apparent triggering cause.

On physical examination, a solid, firm, pinkish, non- painful, non-ulcerated lesion was observed that obstructed

90% of the lumen of the right external auditory canal, preventing visualization of the tympanic membrane, the rest without alterations.

Computed tomography of the ears is requested, where a lesion with a pedicle in the roof of the right external auditory canal is visualized, homogeneous, without bone or cartilage erosion, limited to the canal, without extension to the tympanic cavity and with the presence of the entire tympanic membrane. .

Surgical resection was performed, obtaining a report from the tumor pathology service with histopathological changes compatible with ceruminous adenoma, with no data of malignancy.

The patient is currently under follow-up and asymptomatic.

Discussion

A recount of the most significant information reported in specialized search engines was made, showing that there is very little information in this regard, due to the rarity of the pathology.

These tumors have been labeled with different terminologies since the 1950’s: hidradenomas, myoepitheliomas, cylindromas, mixed skin tumors, pleomorphic ceruminous adenoma, papilliferous ceruminous syringocystoadenoma, ceruminoma, ceruminous adenoma, the latter being the one that persists [1, 2]. Thompson, et al. [3] defined it in a review and unified all existing terms in the literature and described it as a well-differentiated and localized benign neoplasm with the presence of an occasional cystic component and glandular papillary proliferation with histology similar to ceruminous glands. Currently, the World Health Organization recognizes two categories of ceruminous neoplasms: malignant and benign.

It is still a neoplasm with a difficult differential diagnosis with malignant neoplasms, so it is essential to search for microscopic data suggestive of malignancy: perineural invasion, mitotic activity, deep nuclear pleomorphism and tumor necrosis [4], which is why it is beneficial to perform immunohistochemical analysis adding the measurement of antibodies and membrane markers such as: cytokeratin, protein S-100 (basal cells), CD117 and epithelial membrane antigen [5].

Regarding the origin, three theories are proposed: embryonic alterations, ectopic remnants of salivary glands or aberrations of the ceruminous gland per se [6].

The clinical presentation is very non-specific and the symptoms can last for years, which is related to the size of the lesion and the degree of obstruction of the external auditory canal, presenting hearing loss, otalgia, otorrhoea, otorrachia, etc. [7].

In addition to all this controversy, treatment is added, suggesting local resection with close follow-up, local resection with free margins, radical resection and even the addition of radiotherapy, depending on the atypia present and the histological aggressiveness reported by the pathologist [7].

The differential diagnosis includes: ceruminous adenocarcinoma with infiltrative characteristics and atypical mitoses and neuroendocrine adenoma of the middle ear, among others [8].

Conclusions

As previously mentioned, there is very little information in the literature and there are no management guidelines for the pathology due to its low prevalence.

Fortunately, in this clinical case, no data suggestive of microscopic malignancy were found, so a good long-term prognosis is expected.

The patient will be given timely follow-up in the outpatient clinic for at least 5 years. Today, he is asymptomatic It is considered an interesting case due to its very low frequency of presentation in human beings, for the disclosure of the pathology within the specialty.