Surgical Management of Oral Verrucous Carcinoma and Its Recurrence

International Journal of Medical and Pharmaceutical Case Reports 6(2): 1-6, 2016; Article no.IJMPCR.22662 ISSN: 2394-109X, NLM ID: 101648033

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Surgical Management of Oral Verrucous Carcinoma and Its Recurrence: A Case Report and an Update Nikhil Srivastava1, Akshay Shetty1, Vijay Apparaju2* and Rahul Dev Goswami3 1

Department of Oral and Maxillofacial Surgery, Sri Rajiv Gandhi College of Dental Sciences and Hospital, Cholanagar, Hebbal, Bangalore, Karnataka, India. 2 Department of Periodontics, Sri Rajiv Gandhi College of Dental Sciences and Hospital, Cholanagar, Hebbal, Bangalore, Karnataka, India. 3 Department of Oral Medicine and Radiology, Sri Rajiv Gandhi College of Dental Sciences and Hospital, Cholanagar, Hebbal, Bangalore, Karnataka, India. Authors’ contributions This work was carried out in collaboration between all authors. Author NS wrote the draft of the manuscript, provided the case, the figures and supervised the work. Author AS managed the literature searches. Authors VA and RDG were designed the figures, managed literature searches and contributed to the correction of the draft. All authors read and approved the final manuscript. Article Information DOI: 10.9734/IJMPCR/2016/22662 Editor(s): (1) José M. Matés, Department of Molecular Biology and Biochemistry, Faculty of Sciences, University of Málaga, Spain. Reviewers: (1) Somchai Amornyotin, Mahidol University, Bangkok, Thailand. (2) Wagih Mommtaz Ghannam, Mansoura University, Egypt. Complete Peer review History: http://sciencedomain.org/review-history/12459

th

Case Study

Received 17 October 2015 Accepted 9th November 2015 th Published 27 November 2015

ABSTRACT Aim: To enlighten and update the clinicians about the diagnosis and management of recurrent verrucous carcinoma. Presentation of Case: A single case of Oral verrucous carcinoma of the alveolar ridge is reported. The patient was treated by surgical resection of the lesion there was neither local recurrence nor distant metastasis observed. Conclusion: Oral verrucous carcinoma (OVC) is type of well differentiated squamous cell carcinoma. OVC generally is a slow grower and has a higher chance of local invasion then metastasis. Buccal mucosa and gingiva (usually mandibular) are the sites most commonly affected, and majority of the patients tend to be elderly and male. Surgical resection with sufficient safety margin is recommended. _____________________________________________________________________________________________________ *Corresponding author: E-mail: [email protected];

Srivastava et al.; IJMPCR, 6(2): 1-6, 2016; Article no.IJMPCR.22662

Keywords: Ackerman’s tumor; verrucous carcinoma; oral cavity; cancer. 44 region. It was a firm to soft lesion which was non-tender and non scrapable. The lesion 2 was grey white, elevated, patch like with irregular margin extending from the vestibular region to the alveolar ridge, and had a Crack mud appearance with shallow cracks (Fig. 2).

1. INTRODUCTION Oral verrucous Carcinoma (OVC) is a rare variant of oral squamous cell carcinoma. In the year 1948, Lauren V. Ackermann was the first person who described this neoplasm of the oral mucous membrane. This is now known as ‘‘Verrucous carcinoma of Ackermann’’ or ‘‘Ackermann’s tumor’’. OVC grows slowly and tends to be locally aggressive if not treated appropriately. However, even with local tumors there is a thought to ponder about that regional or distant metastasis is rare. This tumor is predominantly seen in males between 60-70 yrs [1]. Tobacco is not known to be associated as a known risk factor as Oral papilloma verrucous leukoplakia (OPVL) can occur both in smokers and non-smokers [1]. Human papilloma virus (HPV) and OPVL is closely associated. It has been reported that upto 89% of OPVL are positive for HPV [2], especially for type 16 and 18 [3]. Apparently, there is no unambiguous pathogenic link between HPV and OPVL [4] and also an association has been reported with Epstein-Barr virus or candida infection [2,3]. Inspite all such works, the cause for OPVL is still as mysterious as the disease itself [5,6].

Fig. 1- Frontal view of the patient Orthopantomogram revealed no involvement of bone (Fig. 2). According to the history noted and clinical examination, a provisional diagnosis of OPVL was made with homogenous leukoplakia in the right buccal mucosa. A differential diagnosis of the following was considered• • • •

Surgical excision with adequate margins of resection seems to be the clear preference for treatment. Recurrences are frequent and require additional treatments.

2. PRESENTATION OF CASE

Oral leukoplakia, Chronic hyperplastic candidiasis, Verrucous hyperplasia, and Verrucous carcinoma

The incisional biopsy was planned and carried out for both the lesions. Histopathological examination revealed soft tissue section showing epithelial proliferation with acanthosis, intracellular oedema & few areas of hyperorthokeratotic epithelium with prominent granular layer in relation to lesion 1, lesion 2 showed hyperorthokeratotic stratified squamous epithelium with prominent granular layer and few dysplastic features like prominent nucleoli, drop shaped rete ridges. This confirmed the diagnosis.

A 68 year old male patient reported to the Department of Oral and Maxillofacial Surgery with the chief complaint of growth in his lower right front tooth region since five months (Fig. 1). On taking through history, patient had undergone total extraction of lower teeth & was using removable complete denture since five years. Five months back he noticed a growth in the lower front teeth region. Patient was hypertensive and is under medication for the same. Extra-oral examination revealed no submandibular lymphadenopathy.

Surgical removal of the lesion in total was done next. Histopathological examination revealed areas of exo-endophytic growth with broad elephant –foot type rete ridges and pushing borders this confirmed the diagnosis of Verrucous Carcinoma (Fig. 3).

Intra-oral examination revealed two distinct lesions. Lesion 1 being creamy white with finger like projection, and granular texture measuring approximately 2 cm × 1 cm in size in the mandibular edentulous area in relation to 42, 43,

With a final diagnosis of Verrucous Carcinoma, considering its high recurrence rate and potential 2


(Fig. 4b and 4c). Postoperatively there was unsatisfactory wound healing with an open defect (Fig. 4d). This was managed and satisfactory healing was accomplished (Fig. 5a and 5b). The histopathological report confirmed no perineural invasion, with the underlying bone and superior, inferior, medial, lateral margins free of tumor.

for local infiltration the patient was put under follow up. The lesion recurred after a period of 1 month in the right alveolar ridge in relation to the site of verrucous lesion (Fig. 4a). So the lesion was next planned for wide local Excision with 5 mm margins in all the dimensions

Fig. 2a. Intra-oral lesions; b. Orthopantamograph before excision of lesions

Fig. 3a, b. Microscopic picture of the lesion after incisional biopsy; c, d, e. Microscopic picture after total excision of the lesion

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Fig. 4a. Intra oral picture showing the recurrent lesion; b, c. Excision of lesion with 5 mm margins; c. Open defect with unsatisfactory healing

Fig. 5a. Post Operative OPG showing the margin ; b. Optimal healing can be seen in the intraoral picture Verrucous carcinoma is a specific type of squamous cell carcinoma of the oral cavity which is a low grade variant. Its quite a rare tumor which represents only 3–4% of oral carcinomas, with an annual incidence rate of just one to three cases per one million persons [7]. The tumor is found to occur in the sixth decade and pertains to 2–8% of all squamous cell carcinoma [9]. It can be described by its slow-growth, painless nature, broad-based papillary lesion. In comparision to oral squamous cell carcinoma, verrucous carcinoma tends not to metastasize regionally or distally. There is a greater predilection in males with higher incidence in buccal mucosa sub site [5]. This has a higher chance of recurrence which can be contributed to simultaneous changes in INK4a/ARF locus along with increase of p16INK4a and homozygous deletion of [2].

3. DISCUSSION White lesions are frequently present in the oral cavity with an occurrence of approximately 24.8%. Amongst them oral leukoplakia (OL) has an incidence of 0.2-3.6% [7]. In a retrospective study, Hansen et al. [8], reported that 26 of the 30 cases which were diagnosed to be OL converted to oral carcinomas in patients followed up for 1-20 years (average, 6.1 years). After this study, these lesions were renamed to oral proliferative verrucous leukoplakia (OPVL). According to the latest World Health Organization nomenclature, As OPVL is neither a limiting lesion or a condition it is included in the new terminology of “potentially malignant disorders”. In a spectrum it can be assessed as a continuum of oral disease with hyperkeratosis and verrucous carcinoma or squamous cell carcinoma being at the two ends [9]. 4


The microscopic findings associated with OPVL depend on the stage of the disease and the extent of the biopsy. Hansen et al. [8], had suggested certain histologic stages in the spectrum of OPVL with intermediates. Grade 0 Grade 2 Grade 4 Grade 6 Grade 8 Grade 10

In order to make the diagnosis of PVL, it was suggested that one of the two following combinations of the criteria mentioned before has to be met. 1. Three major criteria (E being among them). or 2. Two major criteria (E being among them) + two minor criteria.

: Normal mucosa : Hyperkeratosis (clinical leukoplakia) : Verrucous hyperplasia : Verrucous carcinoma : Papillary squamous cell carcinoma :Less well-differentiated squamous cell carcinoma

The surgical excision has been considered to be the gold standard treatment for such lesions. Walvekar et al. in a study of 101 cases of oral verrucous carcinoma concluded that there is excellent prognosis with surgical management. They further emphasized the surgical excision with adequate margins and need for close follow up [10]. In our case the incisional biopsy had shown the features of verrucous leukoplakia so an excisional biopsy was considered, subsequently a diagnosis of verrucous carcinoma was reached which required resection with adequate margin.

Despite these criteria it is pertaininent to note that the initial phase of these lesions generally exhibits an interface of lymphocytic infiltrate that has an eminent lichenoid pattern associated with basal vacuolar degeneration consisting of cells undergoing apoptosis and eosinophilic bodies, similar to types of oral lichen planus. Therefore, OPVL cannot be defined by a single histological feature in the initial stages. Because of the lack of definite histological criteria, the diagnosis of OPVL is based on both clinical and histopathologic evidence of progression [3]. There are few studies which provides a set diagnostic criteria – Cerero-Lapiedra et al. [7], established the following major and minor criteria:

Apart from this surgical modality, in some cases in which tumor extends till retromolar area we can give the combined therapy of radiation and surgery. Cytostatic drugs can also be used in cases where surgery is not indicated. αinterferon can be used as supportive therapy as it slows the growth of tumor [10]. It is suggested that any such suspicious lesions should first be undertaken for biopsy and only after proper diagnosis, the optimal treatment should be carried out.

3.1 Major Criteria a. A leukoplakia lesion which occursat more than two different oral sites, found most frequently in the gingiva, alveolar processes and palate. b. The presence of a verrucous area. c. That the lesions have spread during progression of the disease. d. That there has been a recurrence in an area previously treated. e. Histopathologically, they can present from simple epithelial hyperkeratosis to verrucous hyperplasia, verrucous carcinoma or OSCC, whether in situ or infiltrating.

4. CONCLUSION Verrucous lesions of the oral cavity are a specific clinical entity with varied histopathology. Surgical resection with wide margins followed by optimal reconstruction is of paramount importance to optimize the disease and functional outcome.

CONSENT All authors declare that ‘written informed consent was obtained from the patient for publication of this case report and accompanying images.

3.2 Minor Criteria ETHICAL APPROVAL a. An OL lesion is of at least 3 cm dimension when adding all the affected areas. b. Female patient. c. That patient being a non-smoker. d. A disease history of higher than 5 years.

It is not applicable.

COMPETING INTERESTS Authors have interests exist. 5

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REFERENCES

© 2016 Srivastava et al.; This is an Open Access article distributed under the terms of the Creative Commons Attribution License (http://creativecommons.org/licenses/by/4.0), which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited.

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