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| CASE REPORT |
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| Year : 2014 | Volume
: 20
| Issue : 1 | Page : 41-43 |
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Ceruminous gland adenoma
Himanshu Varshney1, Vivek Taneja2, MK Taneja3
1 Department of ENT and Head Neck Surgery, Sri Sai Hospital, Moradabad, India
2 Department of Otorhinolaryngology, Subharti Medical College and Hospital, Meerut, Uttar Pradesh, India
3 Director, Indian Institute of Ear Diseases, Muzaffarnagar, India
| Date of Web Publication | 1-Apr-2014 |
Correspondence Address:
Himanshu Varshney
Department of ENT and Head Neck Surgery, Sri Sai Hospital, Moradabad - 244 001, Uttar Pradesh
India
 Source of Support: None, Conflict of Interest: None  | Check |
DOI: 10.4103/0971-7749.129825
Ceruminous adenoma is a rare neoplasm of the external auditory meatus (EAM) with benign clinical behavior. They demonstrate a dual cell population of basal myoepithelial-type cells and luminal ceruminous cells. Cerumen pigment, cytokeratin 7 (CK7) and p63 can help to distinguish this tumor from other neoplasms that occur in the region. Complete surgical excision results in an excellent long-term clinical outcome. We present a case of histologically confirmed ceruminous adenoma of the EAM in a surgically treated 38-year-old female. She presented with recurrent serosanguineous discharge along with flakes from the right ear along with hearing impairment. She is doing well in last 8 months follow-up. Keywords: Cerumen pigment, Ceruminous adenoma, Cholesteatoma external auditory canal, External auditory meatus
How to cite this article:
Varshney H, Taneja V, Taneja M K. Ceruminous gland adenoma. Indian J Otol 2014;20:41-3 |
| Introduction | |  |
Ceruminous adenoma is a rare neoplasm of EAM with benign clinical behavior and was first reported by Hang in 1894. The Ceruminous adenomas are located primarily in the cartilaginous portion of the external auditory canal. They demonstrate a dual cell population of basal myoepithelial-type cells and luminal ceruminous cells. The mean age of presentation is the sixth decade. The most often symptom of the ceruminous adenoma of the external auditory meatus (EAM) is the unilateral conductive hearing loss. It is treated by complete surgical excision with excellent prognosis.
| Case Report | | |
The present case report is about a 38-year-old female patient who presented with recurrent foul smelling blood-stained discharge from the right ear along with hearing impairment. In general examination, the patient was of average built and physically fit. No abnormality could be revealed in general medical and gynecological examination. Her routine ENT examination was inconclusive including left ear findings. Right ear was examined. Pinna was mildly tender with excoriation of inferior canal wall. EAM was almost blocked by shiny pea-size swelling over posterior canal wall. Auditory canal was cleaned by micro-suction under magnification. Thick cholesteatoma-like flakes removed; whole of cartilaginous and bony canal was eroded, but no puckering. Tympanic membrane was intact. On palpation mass was tense, firm and indurated with intact overlying skin. A computed tomography (CT) scan of both temporal bones was carried out, which was inconclusive except obliteration of outer end of right EAM. Tympanic cavity and mastoid cleft were normal. No intracranial involvement seen. History was negative for tinnitus, trauma or previous surgery. There was no evidence of cervical lymphadenopathy or systemic lymphadenopathy. X-ray chest (posteroanterior view) was normal. Pure-tone audiometry revealed average 40 dB conductive hearing loss in right (affected) ear.
The mass was removed in-toto with overlying skin and 3 mm skin margins all around under local anesthesia and sedation through endaural approach. Canal dressing was done with Vaseline gauze pack which changed after 7 days. Repeated dressing done at every 4 th day for 3 weeks. Patient was put on Clindamycin 300 mg three times a day with injectable amikacin 500 mg intramuscular twice a day and analgesic. Histopathological findings revealed the diagnosis of ceruminous adenoma. She is doing well in last 8 months follow-up.
| Discussion | | |
Ceruminous adenoma (ceruminoma) of the EAM is a rare neoplasm with benign clinical behavior. This tumor is mainly composed of the ceruminous gland cells (modified apocrine sweat glands). These glands are localized deep in the skin mostly in the cartilaginous part of the EAM. These glands contribute material that combines with sebaceous gland products and desquamated epithelial cells to produce a waxy substance known as cerumen (from Latin cçra "wax").[1] Ceruminous glands are composed of an inner secretory layer of cells and an outer myoepithelial layer of cells. The glands drain into larger ducts, which, in turn, drain into hair follicles that populate the external ear canal (EAC). [2]
The ultimate cause for these tumors remains unknown. Ceruminous tumors have been reported in patients as young as 12 years and as old as 92 years. [3],[4] The mean age of presentation is the sixth decade. The sex distribution for ceruminous adenomas is nearly 50-50. In our case, the patient is 38-year-old female.
The most often symptom of the ceruminous adenoma of EAM is the unilateral conductive hearing loss. Occasionally symptoms of this tumor (pain, otorrhea and rarely paralysis) can result from an otitis externa secondary to meatus obstruction. Chief complaints of our patient were serosanguineous discharge from the right ear along with decreased hearing and pain. The largest series of ceruminous adenomas to date reported average symptom duration of 16 months. [5]
The differential diagnosis are ceruminous adenoma, ceruminous adenocarcinomas, neuro-endocrine adenoma of middle ear, parotid pleomorphic adenoma, meningioma and paraganglioma, which are difficult to diagnose at the initial stage, hence histology is only diagnostic. [5]
Macroscopically these tumors are circumscribed but not encapsulated. When removed intact, they appear as polypoid masses covered by a non-ulcerated epithelium. The largest series of these tumors to date reports a mean size of 1.1 cm and a range of 0.4-2.0 cm. [6] Adenomas have also been reported as being grossly cystic. [4] In our case, it is shiny, tense, firm and indurated pea-size swelling with intact overlying skin over posterior canal wall at the outer end of EAM.
Microscopically, these tumors demonstrate variable architectures, including the formation of small closely approximated glands, areas of cystic dilatation, solid cellular proliferation and papillary arrangements. Similar to the native ceruminous glands and other apocrine-related structures, ceruminous adenomas are composed of 2 cell types. The first type consists of luminally placed cuboidal to low-columnar epithelial cells with eosinophilic cytoplasm that frequently contain small luminal tufts commonly referred to as apical caps or decapitation secretion [Figure 1]. These tufts are typical of apocrine-related glands.
The epithelial cells also contain golden-brown colored ceroid pigment that distinguishes these lesions as related to cerumen-producing glands. The epithelial cells are surrounded by a layer of myoepithelial cells (the second cell type) with less apparent cytoplasm and a smaller hyperchromatic nucleus. Cells are arranged in a glandular pattern, often with cystic dilatation. | Figure 1: Ceruminous glands are a specialized type of apocrine differentiation and include abundant eosinophilic cytoplasm and decapitation secretion
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Immunohistochemical studies differentially label these 2 cell types. Ceruminous adenomas demonstrate labeling of the luminal cells with cytokeratin 7 (CK7) and labeling of the myoepithelial cells with S100, CK 5/6 and p63 (nuclear labeling). [6] Cerumen pigment, CK7 and p63 can help to distinguish this tumor from other neoplasms that occur in the region. The few reports of immunohistochemical studies performed in ceruminous pleomorphic adenomas describe similar labeling of the epithelial and myoepithelial components. [7],[8] A single report of syringocystadenoma papilliferum with distinct epithelial (CK) and myoepithelial labeling (S100) has been published; [9] interestingly, this case was associated with a ductal carcinoma and the authors reported loss of myoepithelial labeling in the invasive component.
Ceruminous adenoma should be radical excised with adequate margins of the normal tissues. However, the anatomic constraints of the radically EAC may make complete excision difficult and residual disease or recurrences of these benign tumors has been reported. In the largest series of benign tumors to date, 4 of 40 patients had either recurrent or residual tumor in subsequent excisions, 1 of them 4 years after the initial diagnosis; none of the patients in this study had evidence of disease at the last follow-up. [6] There may be EAC stenosis associated or following radical excision of tumor which can be treated by canaloplasty. [10] The prognosis of these tumors is good. The incidence of malignancy of ceruminomas is high (up to 70%) while of cutaneous sweat gland is less than five percent. [11] Though ceruminous gland adenocarcinoma are rare, but can present with intra-cranial invasion, hence in suspected cases CT of skull and ultrasound of the abdomen should always be performed. [12] In our patient, mass was removed in-toto with overlying skin and 3 mm skin margins all around under local anesthesia and sedation through minimal endaural approach. Histopathological findings revealed the diagnosis of ceruminous adenoma. She is doing well in last 8 months follow-up.
| Conclusion | | |
Ceruminomas are rare tumors arising from the ceruminous gland, which is a modified apocrine gland in the skin of the external ear canal. Ceruminous adenoma is a benign subtype of ceruminoma. Ceruminous gland adenomas that are pedunculated rarely recur because of the ease of adequate surgical excision. Non-pedunculated ceruminous gland adenomas may be more difficult to adequately excise and are thus, more likely to recur.
| References | | |
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| 2. | Perry ET. The Human Ear Canal. Springfield, Ill: Thomas CC; 1957.
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| 3. | Hicks GW. Tumors arising from the glandular structures of the external auditory canal. Laryngoscope 1983;93:326-40.
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| 4. | Dehner LP, Chen KT. Primary tumors of the external and middle ear. Benign and malignant glandular neoplasms. Arch Otolaryngol 1980;106:13-9.
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| 5. | Giuseppe M, Serena B, Sandro B, Cristiano DN, Vincenzo S, Tommaso V, et al. Adenoma of the ceruminous gland (ceruminoma). Otol Neurotol 2011;32:e14-5.
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| 6. | Thompson LD, Nelson BL, Barnes EL. Ceruminous adenomas: A clinicopathologic study of 41 cases with a review of the literature. Am J Surg Pathol 2004;28:308-18.
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| 7. | Markou K, Karasmanis I, Vlachtsis K, Petridis D, Nikolaou A, Vital V. Primary pleomorphic adenoma of the external ear canal. Report of a case and literature review. Am J Otolaryngol 2008;29:142-6.
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| 8. | Baldus SE, Streppel M, Stennert E, Dienes HP. Pleomorphic adenoma (mixed tumor) of the external auditory canal. Differential diagnosis of the tumors of the ceruminal glands. Pathologe 1999;20:125-9.
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| 9. | Berlin L. Intracranial ceruminous adenoma. J Neurosurg 1949;6:415-8.
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| 10. | Taneja MK. Role of canaloplasty. Indian J Otolaryngol 2013;19:159-63.
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| 11. | Diaz RC, Babu SC. Ductal carcinoma arising from syringocystadenoma papilliferum in the external auditory canal. Otol Neurotol 2007;28:873-4.
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| 12. | Turner HA, Carter H, Neptune WB. Pulmonary metastases from ceruminous adenocarcinoma (cylindroma) of external auditory canal. Cancer 1971;28:775-80.
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[Figure 1]
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