Indian Journal of Dental Research

CASE REPORT
Year
: 2006  |  Volume : 17  |  Issue : 2  |  Page : 82--6

Sinonasal verrucous carcinoma with oral invasion


P Karthikeya, VG Mahima, G Bhavna 
 Department of Oral Medicine and Radiology, JSS Dental College and Hospital, S.S. Nagar, Mysore-15, India

Correspondence Address:
P Karthikeya
Department of Oral Medicine and Radiology, JSS Dental College and Hospital, S.S. Nagar, Mysore-15
India

Abstract

Verrucous carcinoma is a rare warty variant of squamous cell carcinoma, most often seen in the oral cavity and larynx. Its occurrence in the sinonasal tract is rare. This tumor constitutes approximately 1% of all sinonasal neoplasms. The clinical presentation and the histopathological features of verrucous carcinoma are a subject of continuous discussion amongst diagnosticians and pathologists. A case with oral and nasal presentation of this tumor is reported here.



How to cite this article:
Karthikeya P, Mahima V G, Bhavna G. Sinonasal verrucous carcinoma with oral invasion.Indian J Dent Res 2006;17:82-6


How to cite this URL:
Karthikeya P, Mahima V G, Bhavna G. Sinonasal verrucous carcinoma with oral invasion. Indian J Dent Res [serial online] 2006 [cited 2020 Nov 30 ];17:82-6
Available from: https://www.ijdr.in/text.asp?2006/17/2/82/29885


Full Text

 INTRODUCTION



Lauren Ackerman coined the term 'verrucous carcinoma' in 1947 to describe a morphologic variant of squamous cell carcinoma with distinctive clinical and pathological features [1],[2]. It is a well-defined clinical entity, predictably slow growing, late to metastasize and usually amenable to successful local excision (1, 3). The pathogenesis is not yet fully elucidated. Leading theories include HPV infection and chemical carcinogenesis. Furthermore, lesions develop at sites of chronic irritation and inflammation [2].

The oral cavity is by far the most common location of this tumor [4]. Extraoral sites of involvement are larynx, rectal mucosa, skin from the breast, ear canal, soles of feet [5], genitalia, esophagus, nasal cavity [1] and maxillary sinus [6],[7].

 CASE REPORT



A 50-year-old male patient reported with pain and swelling in the upper front teeth region and in the nose since one and a half months. The swelling was insidious in onset and was associated with mild, intermittent pain. He gave history of extraction of the upper front teeth due to decay 3 months back. There were no other associated symptoms. He also noticed a small pea sized swelling in the nose, which gradually grew in size, to attain the present size since one and a half months. The swelling was insidious in onset, associated with mild, intermittent pain and caused him difficulty in breathing.

Personal history revealed that the patient had been using snuff intranasally 2-3 times per day for the past 12 years. He was also a chronic smoker, smoking 10-12 beedis per day for the past 15 years. The patient's past medical and family history were non-contributory.

General examination revealed that the patient was moderately built and nourished, presenting with normal gait and satisfactory vital signs.

Extra oral examination ofthepatient showed a diffuse swelling present bilaterally in the nasal region with a proliferative growth evident in both the nostrils [Figure 1].

Intranasal examination revealed an extensive, expansile exophytic proliferative growth present in the nostrils, more prominent on the right side. Colour of the mucosa over the growth varied from white to pink with no other secondary changes noted over the growth [Figure 2]. On palpation, themucosawas tender and soft to firm in consistency.

A solitary right submandibular lymph node was palpable, which was tender, firm in consistency and mobile.

Intraoral examination revealed missing 11, 12, 21 and 47. 'There was labial vestibular obliteration in the region of the missing teeth in anterior maxillary teeth region extending from 13 to 22 and a sinus tract seen in the midline surrounded by hyperplastic tissue, which was similar in colour to that of adjacent mucosa [Figure 3]. On palpation, the mucosawas tender and softin consistency.

A localized round swelling measuring approximately one and a half­cm in diameter was seen on the palatal mucosa on the right side crossing the midline to a certain extent. A sinus tract was seen over the superior most part of the swelling. The overlying mucosa was erythematous. [Figure 4]. On palpation, the swelling was tender and soft in consistency.

Based on the clinical manifestations, a differential diagnosis of sinonasal carcinoma namely, invasive squamous cell carcinoma or papillary squamous cell carcinoma or verrucous carcinoma was given as manifestations of a single disease entity. Considering the oral and nasal manifestations as findings of two separate disease entities, the diagnosis for oral lesion given was chronic suppurative osteomyelitis and the differential diagnosis given for nasal lesions were nasal polyp, nasopharyngeal carcinoma, inverted ductal papilloma, nasal carcinoma and squamous papilloma

The patient was subjected to the radiographic investigations. Anterior maxillary occlusal radiograph revealed irregular radiolucency present in the anterior part of the palate extending posteriorly upto 16 [Figure 5]. Orthopantomograph showed similar radiolucency in the right maxilla inferior and mesial to the right maxillary sinus [Figure 6]. Water's view showed complete haziness of the right maxillary sinus and diffuse radiopacity in right nasal cavity suggesting involvement of the maxillary sinus [Figure 7]. Axial and coronal sections of CT scan were performed. Axial sections showed an irregular soft tissue mass arising from the floor and alveolar process of maxilla and hard palate, measuring around 4.5 curs in diameter [Figure 8]. Coronal sections showed destruction of the floor of right maxillary antrum, right side of floor of nasal cavity and alveolar processes of maxilla, highly suggestive of a malignant pathology [Figure 9].

Incisional biopsy of the growth from the right nasal cavity revealed well differentiated stratified squamous epithelium overlying the connective tissue with pushing margins and intact basement membrane. Epithelial cells exhibited vacuolation and atypia. The underlying connective tissue showed dense inflammatory infiltrate [Figure 10, 11]. The findings were consistent with a histopathological diagnosis of verrucous carcinoma Based on history, clinical, radiographic and histopathological findings, a final diagnosis of sinonasal verrucous carcinoma with oral emoachment was given.

Subtotal maxillectomy was performed along with soft tissue reconstruction using forehead flap [Figure 12]. The excised specimen was subjected to histopathological examination, the findings of which were again consistent with verrucous carcinoma

 DISCUSSION



The clinicopathologic concept of verrucous carcinoma originated in 1948 when Lauren Ackerman described a locally aggressive exophytic lesion of low-grade squamous cell carcinoma with little metastatic potential as verrucous carcinoma in the oral cavity of 31 patients [8],[9].

Oral, pharyngeal, laryngeal and esophageal lesions are referred to as verrucous carcinoma or Ackerman tumor [9]. This tumor has also been known as oral florid papillomatosis, a term coined in 1960 by Rock and Fisher and subsequently Wechsler and Fisher stressed on the locally aggressive but clinically benign nature in 1962 [9],[10].

In the head and neck region, verrucous carcinoma occurs most commonly in the oral cavity [4] and less frequently in the larynx [6], [7],[11]. It is a rare tumor of the sinonasal tract [7], [11],[12],[13] and the maxillary antrum [6],[7]. Approximately 28 cases only have been reported in medical literature so far [11]. The primary sinonasal malignancies are rare, constituting 0.2-0.8% of all malignancies and 3% of head and neck malignancies [14]. Sinonasal tumors are often discovered when they are quite large. Delay in diagnosis has been attributed to the fact that these lesions are asymptomatic or cause non-specific symptoms when they are small. When pain occurs, it is an indicator of perineural extension of malignancy/ tumor infection [14]. Bone erosion occurs when the tumor spreads and involves adjacent structures causing dysfunction and resulting in symptoms like nasal obstruction, epistaxis and anosmia In case of oral involvement, it causes loose teeth, poorly fitting dentures, trismus, facial asymmetry and visible/palpable bulge in the oral cavity. The involvement of nasal cavity leads to nasal obstruction and blood stained/ purulent discharge confusing the condition with rhinosinusitis, which is a common condition [15],[16]. It is important to differentiate this tumor clinically and histologically from benign papilloma and well-differentiated non-verrucous squamous cell carcinoma [11],[13]. Our patient gave history of nasal obstruction and on inspection we found an exophytic growth in the palate.

Verrucous carcinoma in general presents in the age group between 50 and 80 years with a male predominance closely related to the habit of tobacco chewing and snuff dipping [1] which also found in our patient.

However, in reports by Me Coy and Waldron, there were cases in which verrucous carcinoma arose in patients who denied the use of tobacco, suggesting there were carcinogenic factors other than those contained within tobacco for development ofverrucous carcinoma [8].

Reports have suggested that opportunistic persistent viruses mainly HPV-6 and 16, act in concert with frank carcinogenesis to promote development of verrucous carcinoma [8],[17].

In the sinonasal region, the tumor may present as a nasal polyp/ pyocele [9] or polypoid lesion with pus discharge from nasal cavity [13]. On CT and MR studies, verrucous carcinoma is difficult to differentiate from other types of squamous cell carcinoma, although an exophytic soft tissue mass displaying an irregular surface and no or minimal submucosal extension may suggest the diagnosis [18].

Histologically, verrucous carcinoma resembles avernrca by showing hyperkeratosis, parakeratosis and acanthosis in the superficial portions [19]. Low power view shows a lesion that demonstrates an exo-endophytic growth pattern. The surface is papillary with a thick parakeratinised covering that extends into deep cleft like interpapillary spaces. The rete ridges are broad andbulbous and exhibit pushing borders, which extend infiltration of connective tissue by the epithelium [20]. Parakeratin typically fills numerous clefts or crypts (parakeratin plugs) between surface projections [5].

Histopathologically, verrucous carcinoma can be differentiated from conventional squamous cell carcinoma, as there is no infiltration of the submucosa, the basement membrane remains intact. Dysplastic changes are minimal [21].

Proliferative verrucous leukoplakia and verrucous hyperplasia differ from verrucous carcinoma as they show moderate to severe dysplastic changes. Papillary squamous carcinoma has dysplasia with papilloma like structures of branching fronds with fibrous cores, but no surface keratinisations. This and rete pattern differentiate the lesion histologicallyfrom venucous carcinoma [21].

In general, verrucous carcinoma is a slow growing tumor [6] with antecedent premalignant changes that were termed verrucous hyperplasia when untreated, verrucous carcinoma can multifocally invade underlying structures including bone [19].

Metastases are usually limited to regional lymph nodes. In about 10% of the patients, the tumor evolves into a classic squamous cell carcinoma [19].

An adequate biopsy specimen is necessary for accurate diagnosis, since it is primarily the architecture of this tumor that is diagnostic. An adequate specimen is one that includes atleast one normal margin to a depth within the submucosa[20].

Surgical excision [6], [9] has been considered the treatment of choice for verrucous carcinoma The peripheral margins are most important since recurrences arise mostly from this area. Traditional excision or Mob's micrographically controlled excision can be performed [2], [9].

verrucous carcinomas of the nasopharynx and supraglottlc larynx are much more difficult to treat because of access limitations and size, which is generally large at the time of diagnosis. Management requires radiotherapy either as sole modality or combined with surgery [20].

Kraus and Perez Mesa and Perez et al [4], [22] described fear of anaplastic transformation [3], [13], [18], [22], [23]. Review of literature shows that frequency of anaplastic transformation after radiation is 30% [23]. The apparent transformation actually represents the origin of a highly aggressive new neoplasm from the cells of slow growing relatively benign lesion, due to radiation induced genetic injury [3], [23].

Irradiation can be used to treat selected patients [4], [6], [13], [18], [23]. Radiation therapy should be used in advanced verrucal type lesions in which surgical resection are difficult or not feasible. These patients require frequent and careful observation [22].

 CONCLUSION



Verrucous carcinoma is a warty variant of squamous cell carcinoma mainly occurring in the oral cavity and larynx. Its occurrence in the sinonasal region is rare and mainly attributed to snuff dipping. The presentation in the nasal cavity as an exophytic mass should alert the oral diagnostician for complete intranasal and intraoral examinations along with appropriate adjunctive radiological and histopathological examinations. Although it shows locally invasive pattern without any distantmetastasis, early diagnosis andtimely intervention improves the prognosis, decreasing the rate of anaplastic transformation.

 ACKNOWLEDGEMENT



The authors thank Dr. Saikrishna Professor, Department of Oral and Maxillofacial Surgery and Dr. Sudheendra Ex­-Assistant Professor, Department of Oral Pathology, JSS Dental College and Hospital, Mysore.

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