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 Table of Contents  
CASE REPORT
Year : 2016  |  Volume : 22  |  Issue : 1  |  Page : 59-61

Is cholesteatoma a precursor of verrucous carcinoma: A diagnostic dilemma


1 Department of ENT-HNS, Kasturba Medical College, Manipal University, Manipal, Karnataka, India
2 Department of Radiation Oncology, Kasturba Medical College, Manipal University, Manipal, Karnataka, India

Date of Web Publication16-Feb-2016

Correspondence Address:
Rohit Singh
Department of ENT.HNS, Kasturba Medical College, Manipal University, Manipal - 576 104, Karnataka
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/0971-7749.176515

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  Abstract 

Verrucous carcinoma is an unusually well-differentiated variant of squamous cell carcinoma, characterized by local invasion and cytologically benign appearance. We report a case of 52-year-old male who presented with postauricular growth and cholesteatoma in the right ear. High-resolution computed tomography temporal bone showed an enhancing soft tissue mass lesion extending into the mastoid, external auditory canal, and middle ear. Growth was histopathologically confirmed to be verrucous carcinoma extending from the cholesteatoma in mastoid cavity. Review of literature confirms the rarity of cases of cholesteatoma which could progress and present as verrucous carcinoma in the postauricular region.

Keywords: Cholesteatoma, Postauricular region, Verrucous carcinoma


How to cite this article:
Singh R, Nair DH, Mishra A, Singh A. Is cholesteatoma a precursor of verrucous carcinoma: A diagnostic dilemma. Indian J Otol 2016;22:59-61

How to cite this URL:
Singh R, Nair DH, Mishra A, Singh A. Is cholesteatoma a precursor of verrucous carcinoma: A diagnostic dilemma. Indian J Otol [serial online] 2016 [cited 2021 Jul 28];22:59-61. Available from: https://www.indianjotol.org/text.asp?2016/22/1/59/176515




  Introduction Top


Squamous epithelial lesions in middle ear include cholesteatoma, squamous carcinoma, pseudoepitheliomatous hyperplasia, and few cases of verrucous carcinoma of the middle ear.

Verrucous carcinoma was coined by Ackermann in 1948 when he was describing the mucosal lesions with a warty pappilomatous appearance in the mouth.[1] Verrucous carcinoma also commonly called as snuff dippers cancer is most commonly seen in the oral cavity. Hence, the reported cases of verrucous carcinoma are mostly present in the mouth but also have been described in the larynx, esophagus, nasopharynx, penis, vagina, vulva, scrotum, perineum, cervix, and pyriform fossa.[2]

Here we present an interesting case, wherein cholesteatoma of the middle ear and mastoid progressed to form a verrucous carcinoma in the postauricular region.


  Case Report Top


A 52-year-old male presented with a 3 years history of growth in the right postauricular region with ear discharge and hearing loss since childhood. Ear discharge was on and off, mucoid; however, in the past few years it had become foul smelling. Examination of ear revealed an ulceroproliferative growth 3 cm × 4 cm over the right postauricular region, with an irregular surface, everted margins, floor formed by postauricular muscles, and the base fixed to the underlying tissues [Figure 1]. A tract was visualized arising from the ulcer going deep into the region of mastoid. Examination of ear under a microscope revealed cholesteatoma, seen filling the middle ear with mucopurulent discharge. Pure tone audiogram revealed right profound hearing loss with left moderate mixed hearing loss.
Figure 1: Clinical picture shows ulceroproliferative growth 3 cm × 4 cm over the right postauricular region

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High-resolution computed tomography temporal bone revealed an enhancing soft tissue mass lesion in the right postauricular region extending into the mastoid, external auditory canal, and middle ear cavity [Figure 2].
Figure 2: High-resolution computed tomography temporal bone showing an enhancing soft tissue mass lesion in right postauricular region extending into the mastoid, external auditory canal, and middle ear cavity

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The patient underwent right canal wall down modified radical mastoidectomy with wide excision of postauricular lesion and chonchomeatoplasty. Intraoperatively, there was an ulceroproliferative growth in the postauricular region with a fistula into the mastoid cavity. Cholesteatoma sac was present in the middle ear and mastoid antrum. Incus and malleus were absent. Automastoidectomy cavity was present. Stapes, facial nerve, lateral semicircular canal, sinus plate, and dural plate were intact.

Histopathological examination of the postauricular growth revealed findings of verrucous exophytic and endophytic growth lined by markedly acanthotic epidermis showing hyperkeratosis with frequent keratin pearls and horn cysts, increased mitosis, bulbous elongation of rete ridges surrounded by dense lymphoplasmacytic infiltrates suggestive of verrucous carcinoma [Figure 3]. Tissue from mastoid antrum and middle ear was identified as cholesteatoma sac. The patient has been disease free on 1 year follow-up.
Figure 3: Histopathological picture showing findings of verrucous exophytic and endophytic growth lined by markedly acanthotic epidermis showing hyperkeratosis with frequent keratin pearls and horn cysts, increased mitosis, bulbous elongation of rete ridges surrounded by dense lymphoplasmacytic infiltrates

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  Discussion Top


Verrucous carcinoma is an unusually well-differentiated variant of squamous cell carcinoma, characterized by local invasion and cytologically benign appearance. Verrucous carcinoma of skin arises in moist intertrinous areas such as glans penis, labia, and gluteal fold.[3] Verrucous carcinoma has low malignant potential, but they are locally invasive and rarely metastasize. The pathologic criteria for verrucous carcinoma have been well-described.[4] This tumor is exophytic and shaggy or warty in appearance and may be associated with leukoplakia. Often there is surrounding inflammation, and regional nodes may be enlarged. Histologically, there is a proliferation of keratinizing squamous epithelium. There is a piling up of keratin on the surface, and downgrowth of “club-shaped fingers” or papillary fronds of epithelium, often without violation of basement membrane. There may be areas of cyst formation and granulomas. The lesion is cytologically benign cells are well-differentiated, with few mitosis and without atypia. Margins are described as pushing rather than infiltrating and basal membrane is not violated. Inflammatory cells may be present at the periphery of the lesion.[1],[5],[6]

A literature search reveals only 14 other reports on verrucous carcinoma of the ear. There have been some reports of verrucous carcinoma affecting head and neck but the occurrence of this tumor in ear was first described by Woodson et al. in 1981.[3] Proops et al. also showed verrucous carcinoma occurred in the middle ear and bony canal wall in chronically discharging ear.[4] Genetic studies by Albino et al. reported a 9–20-fold elevation of nuclear phosphoprotein p53 tumor suppressor gene in cholesteatoma when compared to the normal postauricular skin or tympanic membrane.

In the literature, almost all ear lesions were limited to the middle ear, external auditory canals, and mastoid cavities except one case which had extended into the posterior cranial fossa. This case draws the attention of otologists, as it is a rare case in literature wherein cholesteatoma of the middle ear and mastoid cavity has progressed and presented as a postauricular ulceroproliferative growth.

It is worthwhile mentioning some of the factors which could cause a cholesteatoma to present as postauricular verrucous growth. The bone eroding property of cholesteatoma could explain how cells migrated to postauricular skin and presented in the form of ulcerative growth. Second, the role of genetic factor like p53 gene alteration or evasion of apoptosis which could explain the continuous proliferation of cells, to form a carcinoma in the same area.

According to reports, prognosis with verrucous carcinoma seems to be related to extratemporal spread.[7],[8] None of the reported cases of verrucous carcinoma ear showed nodal metastasis. Although it shows a low rate of lymph node metastasis, many of the oral verrucous carcinoma can present at an advanced stage. In one report, lymph node metastasis in oral cavity lesions is stated as high as 30%.[9]

At present, generalization about optimal treatment and prognosis of this rare disorder are difficult to make due to paucity of clinical experience.[6] However, surgery is the best modality of treatment in both cholesteatoma and verrucous carcinoma. But prior to surgery the lesion should always be biopsied to plan a definitive treatment. The role of radiotherapy in the treatment of verrucous carcinoma is controversial.[10] It has been stated that radiotherapy is positively contraindicated because of the risk of anaplastic transformation to a more aggressive tumor.[4]

The true incidence of verrucous carcinoma of the ear may be underestimated because many of the cases may have been misdiagnosed as “aggressive” cholesteatoma or “low grade” squamous cell carcinoma. Therefore, this uncommon tumor should be considered in the differential diagnosis of squamous lesions of the ear, particularly when locally aggressive behavior contradicts a histologically benign appearance.[5]

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.

 
  References Top

1.
Ackerman LV. Verrucous carcinoma of the oral cavity. Surgery 1948;23:670-8.  Back to cited text no. 1
[PUBMED]    
2.
Proops DW, Hawke WM, van Nostrand AW, Harwood AR, Lunan M. Verrucous carcinoma of the ear. Case report. Ann Otol Rhinol Laryngol 1984;93(4 Pt 1):385-8.  Back to cited text no. 2
    
3.
Woodson GE, Jurco S 3rd, Alford BR, McGavran MH. Verrucous carcinoma of the middle ear. Arch Otolaryngol 1981;107:63-5.  Back to cited text no. 3
    
4.
Ferlito A. Diagnosis and treatment of verrucous squamous cell carcinoma of the larynx: A critical review. Ann Otol Rhinol Laryngol 1985;94(6 Pt 1):575-9.  Back to cited text no. 4
    
5.
Park CH, Park YH. Verrucous carcinoma in external auditory canal. Int Adv Otol 2011;7:122-4.  Back to cited text no. 5
    
6.
Edelstein DR, Smouha E, Sacks SH, Biller HF, Kaneko M, Parisier SC. Verrucous carcinoma of the temporal bone. Ann Otol Rhinol Laryngol 1986;95:447-53.  Back to cited text no. 6
[PUBMED]    
7.
Hagiwara H, Kanazawa T, Ishikawa K, Fujii T, Kitamura K, Noguchi Y, et al. Invasive verrucous carcinoma: A temporal bone histopathology report. Auris Nasus Larynx 2000;27:179-83.  Back to cited text no. 7
    
8.
Rinaldo A, Devaney KO, Ferlito A. Verrucous carcinoma of the ear: An uncommon and difficult lesion. Acta Otolaryngol 2004;124:228-30.  Back to cited text no. 8
[PUBMED]    
9.
Walvekar RR, Chaukar DA, Deshpande MS, Pai PS, Chaturvedi P, Kakade A, et al. Verrucous carcinoma of the oral cavity: A clinical and pathological study of 101 cases. Oral Oncol 2009;45:47-51.  Back to cited text no. 9
    
10.
Rahman M, Bhoomika B, Shahnaz A, Islam R. Verrucous carcinoma in external auditory canal: Presentation of an extremely rare case. Indian J Otolaryngol 2013;19:152-4.  Back to cited text no. 10
    


    Figures

  [Figure 1], [Figure 2], [Figure 3]


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