|Year : 2022 | Volume
| Issue : 3 | Page : 737-740
Extraorally proliferating verrucous carcinoma of buccal mucosa mimicking squamous cell carcinoma: A rare case presentation
S Akhil1, RS Krishna Prasad2, KL Girish3, T Harinee4, RG Mridula1
1 Department of Oral Pathology, Malabar Dental College and Research Centre, Vattamkulam, Kerala, India
2 Krishna Dental and Aesthetic Centre, Melpuram, Tamil Nadu, India
3 Department of Oral Pathology, Sree Mookambika Institute of Dental Sciences, Kanniyakumari, Tamil Nadu, India
4 Department of Oral Surgery, Sree Mookambika Institute of Dental Sciences, Kanniyakumari, Tamil Nadu, India
|Date of Submission||24-Jun-2019|
|Date of Decision||30-Jan-2020|
|Date of Acceptance||03-Feb-2020|
|Date of Web Publication||2-Nov-2022|
Dr. S Akhil
Department of Oral Pathology, Malabar Dental College and Research Centre, Manoor Chekanoor Road, Mudur Post, Edappal, Malappuram - 679 578, Kerala
Source of Support: None, Conflict of Interest: None
Verrucous carcinoma (VC) (Ackerman's tumor/Snuff dipper's cancer) is a variant of oral squamous cell carcinoma (SCC) with minimum aggressive potential. The most common sites of involvement in the head-and-neck regions are oral cavity and larynx. VC of the oral cavity is a different clinicopathologic tumor distinguished from the usual SCC because of its local invasiveness, nonmetastasizing behavior, and special clinical appearance, but long-standing cases have shown transformation into SCC. An accurate pathological diagnosis is difficult because of an inadequate tumor sample for the study, and more importantly, a close collaboration is needed between a clinician and a pathologist to achieve the correct diagnosis. In this article, we discuss a case of 42-year-old male with VC of the left buccal mucosa which was proliferating extraorally.
Keywords: Extraoral proliferation, left buccal mucosa, verrucous carcinoma
|How to cite this article:|
Akhil S, Krishna Prasad R S, Girish K L, Harinee T, Mridula R G. Extraorally proliferating verrucous carcinoma of buccal mucosa mimicking squamous cell carcinoma: A rare case presentation. J Datta Meghe Inst Med Sci Univ 2022;17:737-40
| Introduction|| |
Oral verrucous carcinoma (OVC) was first described by Ackerman in 1948 through his paper which described 31 cases with similar clinical and histopathological features. This entity has been described in the literature under various names such as Buschke-Loewenstein tumor, Ackerman's tumor, oral florid papillomatosis, carcinoma cuniculatum, and epithelioma cuniculatum. It is considered as a well-differentiated variant of squamous cell carcinoma (SCC) with characteristic clinical and histopathological features., The tumor grows slowly and is locally invasive with very less chance of metastasis. OVC appears as a white thick plaque, which is painless resembling a cauliflower. Buccal mucosa is the most common site of oral mucosal involvement. Mandibular alveolar crest, gingiva, and tongue are also affected., The exact etiology of OVC is unknown; tobacco chewing and smoking are projected as the major causative factors. As it is more commonly seen in persons who use snuff and chewable tobacco, it is called as “snuff dipper's cancer.” Various treatment modalities including surgery, radiation, chemotherapy, or combination of these have been put forward but each with its own controversies. Here, we report one unusual case of verrucous carcinoma (VC) which was extending extraorally and review the etiology, pathogenesis differential diagnosis, and the management of this tumor.
| Case Report|| |
A 42-year-old male patient came to our department with a chief complaint of a large growth in the left side of the mouth for the past 2 years. The patient observed a small, painless growth on the left buccal mucosa 2 years back, which gradually increased to attain the present condition. The patient had a history of betel quid chewing for 40 years, 5–6 times/day, and used to keep it in the lower left buccal vestibule for 15 min after which he used to spit out the contents. On extraoral examination, a growth measuring 2 cm × 2 cm was seen protruding from the mouth [Figure 1]. Intraoral examination revealed an irregular proliferative ulcer measuring 4 cm × 6 cm seen in the left buccal mucosa with everted edges. It was extending anteriorly from commissure of the lip to the retromolar region posteriorly and superiorly from the occlusal plane of maxillary third molar to the alveolus on mandible. Lesional surface was irregular at the center with finger-like projections in the periphery. Lesion was pink in color with white areas interspersed in between [Figure 2]. On palpation, the lesion was firm, tender with indurated margins. A provisional diagnosis of SCC and verrucous hyperplasia was arrived based on the clinical observation, and for confirmatory diagnosis, an incisional biopsy was performed. Routine blood investigations were carried out, and it was found to be normal. The histopathological examination revealed parakeratinized stratified squamous epithelium, which was hyperplastic with wide and elongated rete ridges. At some areas, the epithelium showed cleft formation with parakeratin plugging [Figure 3]. The epithelium seemed to be pushing into the connective tissue [Figure 4]. The epithelium showed minimal cellular atypia. The subjacent connective tissue showed diffuse dense inflammatory cell infiltrate predominantly of lymphocytes, plasma cells, and few mast cells [Figure 5]. Based on these findings, a final diagnosis of VC was given. The patient was advised to undergo radiotherapy followed by excision of whole lesion. Currently, the patient is under radiotherapy.
|Figure 4: Epithelium showing intact basement membrane with minimal cellular atypia. Note the chronic inflammatory cell infiltrate which is present in the connective tissue subadjacent to the epithelium (H and E, ×40)|
Click here to view
|Figure 5: Broad “elephant foot-”shaped rete pegs with minimal connective tissue core (H and E, ×10)|
Click here to view
| Discussion|| |
Oral SCC (OSCC) and OVC are malignant tumors of the epithelial tissue origin. VC is a highly differentiated variety of OSCC that shows a low degree of malignancy. Various etiological factors have been put forwarded such as human papillomavirus, smoking, and smokeless tobacco in chewable forms. Shear and Pindborg reported that of 28 patients with verrucous lesions, 24 (86%) used tobacco and one was an arecanut quid user. Tobacco seems to be a major factor in causation of verrucous lesions. In our case also, the patient was a chronic tobacco user. According to a recent study, the prevalence of VC affecting oral cavity and oropharynx is 2%–12%. The male-to-female ratio of VC incidence is approximately similar. “The most affected areas are mandibular retromolar and molar area (41.6%) followed by the buccal mucosa (16.6%), the hard palate (16.6%), the floor of the mouth (16.6%), and the lip mucosa (8.3%).” Regional lymph nodes are often enlarged in VC and are tender due to inflammatory involvement, mimicking metastatic tumor. However, in our case, lymph nodes were not involved.
OVC characteristically has a heavily keratinized, or parakeratinized, irregular surface with clefting. Cleft contains parakeratin extending deep into the clefts which is like “elephant foot-” down growth, which seems to compress the connective tissue lying underneath. The prickle cell layers show bulbous hyperplasia with minimal atypia. The tumor has a well-defined lower border and basal lamina with subepithelial inflammatory infiltrate.,, Similar histopathological findings were also seen in our case.
There are various lesions which clinically resemble OVC which include papilloma, verruca vulgaris, VC, and papillary SCC.
Proliferative verrucous leukoplakia, OVC, and verrucous hyperplasia are the most difficult ones to distinguish clinically. There are characteristic histopathological differences between verrucous hyperplasia and verrucous carcinoma. Histopathology remains the golden standard for delineating these lesions [Table 1].
|Table 1: Histopathological difference between verrucous hyperplasia and verrucous carcinoma|
Click here to view
The histopathologic diagnosis of OVC needs adequate incisional biopsy since 20% of these lesions may have a routine SCC developing with in it. Due to proliferative nature of lesion, surgeon will not go deep through the tissue during biopsy causing insufficient tissue for diagnosis often requiring a second biopsy for conformation.
The prognosis of OVC is better than various other life-threating malignant tumors. Primary modality treatment for VC has been considered as to be that of surgery. When the tumor extends to the retromolar area and also when the size of lesion is bigger, combined therapy has been advocated as useful. In our case, combination treatment was selected due to the above reasons. As chances of anaplastic transformation from OVC to more aggressive SCC is possible, radiation therapy alone has been contraindicated.,, When surgery is not an option, various other treatment methods such as cytostatic drugs may be preferred. Alpha-interferon, although cannot be used as a substitute for surgical treatment, seems to support the therapy by delaying the growth of the tumor. Local recurrence of 38.5% has been reported in OVC. Using surgical therapy, the 5-year disease-free survival rate was found to be 77.6%.
| Conclusion|| |
OVC is a distinct clinicopathologic entity distinguished from the usual OSCC because of its nonmetastasizing behavior, local invasiveness, and special clinical appearance. In most of the cases, VC and verrucous hyperplasia are indistinguishable from each other clinically. Hence, histopathological conformation is mandatory to give an appropriate diagnosis.
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.
Financial support and sponsorship
Conflicts of interest
There are no conflicts of interest.
| References|| |
Ackerman LV. Verrucous carcinoma of the oral cavity. Surgery 1948;23:670-8.
Asha ML, Vini K, Chatterjee I, Patil P. Verrucous carcinoma of buccal mucosa: A case report. Int J Adv Health Sci 2014;1:19-23.
Alkan A, Bulut E, Gunhan O, Ozden B. Oral verrucous carcinoma: A study of 12 cases. Eur J Dent 2010;4:202-7.
Schwartz RA. Verrucous carcinoma of the skin and mucosa. J Am Acad Dermatol 1995;32:1-21.
Neville BW, Damm DD, Allen CM, Bouquot JE. Epithelial pathology. In: Oral and Maxillofacial Pathology. 3rd
ed. Philadelphia: Elsevier publication; 2009. p. 422.
Yoshimura Y, Mishima K, Obara S, Nariai Y, Yoshimura H, Mikami T. Treatment modalities for oral verrucous carcinomas and their outcomes: Contribution of radiotherapy and chemotherapy. Int J Clin Oncol 2001;6:192-200.
Shear M, Pindborg JJ. Verrucous hyperplasia of the oral mucosa. Cancer 1980;46:1855-62.
Rekha KP, Angadi PV. Verrucous carcinoma of the oral cavity: A clinico-pathologic appraisal of 133 cases in Indians. Oral Maxillofac Surg 2010;14:211-8.
Rajendran R. Benign and malignant tumors of the oral cavity. In: Rajendran R, Sivapathasundharam B, editors. Shafer's Textbook of Oral Pathology. 7th
ed. India: Elsevier Publication; 2012. p. 122.
Swetha P, Supriya NA, Kumar GR. Characterization of different verrucous mucosal lesions. Indian J Dent Res 2013;24:642-4.
] [Full text]
Kallarakkal TG, Ramanathan A, Zain RB. Verrucous papillary lesions: Dilemmas in diagnosis and terminology. Int J Dent 2013;2013:298249.
Koch BB, Trask DK, Hoffman HT, Karnell LH, Robinson RA, Zhen W, et al
. National survey of head and neck verrucous carcinoma: Patterns of presentation, care, and outcome. Cancer 2001;92:110-20.
Mallya S, Miqdad SM, Nayak S, Jose M. Papillary–verrucous lesion of the oral mucosa: A need for detailed histopathological examination. JIMD 2015;2:16367. http://dx.doi.org/10.18320/JIMD/201502.03163
. [Last accessed on 2019 Feb 10].
Passi D, Singh G, Gupta C, Patra D. Verrucous carcinoma – A diagnositic dilemma: Case series, differential diagnosis, therapy and literature review. J Adv Med Dent Sci 2014;2:141-46.
[Figure 1], [Figure 2], [Figure 3], [Figure 4], [Figure 5]