Verrucous carcinoma: The deadly projections - A series of three cases

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1 International Journal of Medical and Dental Case Reports (2016), Article ID , 5 Pages CASE REPORT Verrucous carcinoma: The deadly projections - A series of three cases Gaurav, Vathsala Naik, Amandeep Sodhi Department of Oral Medicine and Radiology, Bangalore Institute of Dental Sciences, Bengaluru, Karnataka, India Correspondence Dr. Gaurav, Department of Oral Medicine and Radiology, Bangalore Institute of Dental Sciences, Bengaluru, Karnataka, India. dr.gaurav.sunny@gmail.com Received 10 December 2015; Accepted 16 March 2016 doi: /ins.ijmdcr.34 How to cite the article: Gaurav, Naik V, Sodhi AD. Verrucous carcinoma: The deadly projections - A series of three cases. Int J Med Dent Case Rep 2016;2:1-5. Abstract Among the numerous variants of squamous cell carcinoma (SCC) are the one which is called as verrucous carcinoma (VC). It is also called as Snuff differ s cancer, the name derived from this cancer occurring in patients who chew tobacco or snuff orally. VC is a malignant, highly differentiated, papillary growth which is diffused appearance and non-metastasizing in nature. Various subtypes of VC have been described in literature. Although surgery is considered the main treatment modality, the extent of margins and adjuvant radiotherapy are still controversial. Prognosis is mostly good, however, local recurrence is common. Prognosis is usually worse in cases of second oral SCCs which is always a risk in patients with VC. Keywords: Exophytic, papillary, prognosis, snuff dipper, verrucous Introduction Lauren V. Ackerman was the fi rst in 1988 to describe a rare variant of oral squamous cell carcinoma (SCC) called verrucous carcinoma (VC). [1,2] It is also called Ackerman s tumor. Buschke Loewenstein description of a penile lesion which although appeared benign cytologically behaved like a malignancy was the highlight of literature in 1925, thus also known as Buschke Loewenstein tumor. Among the etiological agents, tobacco chewing forms the fi rst line with lesions developing at the site of placement. This case series of ours highlights few classic presentations of VC with exophytic caulifl ower like growth and pedunculated fi nger-shaped projections which are considered the hallmarks of this type of carcinoma. [3,4] 2-3 years back, it had reached the present state for the past 2-3 months. It was also accompanied by a painful growth in the left buccal mucosa. The patient was a chronic paan chewer (added with supari about 5-6 times/day) for the past years placing the quid in her left buccal mucosa. She had, however, quit the habit for the past 6-7 months. On general physical examination, she was found to be moderately built with normal gait and posture and was well oriented to time, person and place. All her vital signs and Case Reports Case 1 A 71-year-old female patient [Figure 1] reported to the Department of Oral Medicine and Radiology complaining of severe pain, burning sensation and diffi culty in eating for the past 2-3 months. She also complained of a painful growth in her left cheek since 2 months. History of presenting illness revealed severe pain in her left buccal mucosa which was sudden in onset, gradual in progression, continuous in nature, aggravated on eating, and on opening the mouth. Although it initiated about Figure 1: Case 1: Patient s profile picture 1

2 Gaurav, et al. Verrucous Carcinoma Case Series parameters were within the normal limits. On extra oral examination, no gross facial asymmetry was detected. The mouth opening was reduced to 32 mm. The regional lymph nodes were non-tender and non-palpable, and the salivary fl ow was adequate. On intraoral examination, the soft tissues such as lips, labial mucosa, palate, tonsils, oropharynx, and the fl oor of the mouth were apparently normal. The tongue was found to be thickly coated, dry and fi ssured. On examining the buccal mucosa, an exophytic plaque like growth with fi nger-like projections was seen on the left buccal mucosa, irregular in shape and measuring approximately 5 cm 6 cm extending anteroposteriorly from the retrocomissures involving the lower labial mucosa to the retromolar pad area and superioinferiorly from the upper gingivobuccal sulcus (GBS) to the lower GBS. The exophytic growth was more pronounced in the anterior region from the retrocomissures to the canine region, however, the latter part of the lesion manifested as white plaque. The surface appeared wrinkled with no signs of sloughing, bleeding or discharge of any kind. Margins of the lesion appeared raised and everted with irregular borders. The laterotrusive tongue movement was restricted toward the left side, but the protrusive movement was normal. On palpation, the lesion was tender and soft to fi rm in consistency. Surface over the lesion appeared rough, granular, wrinkled and indurated with no signs of bleeding or discharge of any kind [Figure 2]. On hard tissue examination, all complements of teeth were present in all the four quadrants with generalized attrition and a thick band of local deposits of plaque and calculus. Mobility was present with respect to few teeth viz., 15, 16, 24 (Grade I); 31, 41 (Grade II); 25, 36, 37 (Grade III). Based on the patient s history and clinical fi ndings, the lesion was provisionally diagnosed as VC on the left buccal mucosa. Differential diagnosis for this lesion: Verrucous hyperplasia and squamous papilloma are few of the malignant conditions mimicking this lesion clinically. Following investigations were carried out: 1. Panoramic radiograph [Figure 3] 2. Incisional biopsy [Figure 4]. The panoramic radiograph showed no intraosseous involvement. The histopathological examination [Figure 4] showed the presence of various characteristic dysplastic features revealed these cases to be VCs based on examination of the biopsied tissues. Eosin and hematoxylin stained sections showed connective tissue and epithelium which was parakeratinized and hyperplastic stratifi ed squammous type in nature. The section also showed broad rete ridges with blunt margins and intact basement membrane. A dense infl ammatory infi ltrate consisting of lymphocytes and plasma cells were also seen. On analyzing the investigation results, fi nal diagnosis of VC of the left buccal mucosa (Stage IV [T 4 N X ]) was given. Case 2 A case of 56-year-old female patient [Figure 5] reported to the department with a complaint of painful extraoral swelling associated with a growth intraorally. The patient was a known diabetic, hypertensive for the past 20 years and had undergone cardiac bypass surgery 2 years back. Local examination of the patient s chief complaint revealed a diff use extraoral swelling [Figure 6] measuring approximately 1.5 cm 2 cm on left lower third of the face extending anteroposteriorly from 1 cm behind the corner of the mouth to 2 cm in front of the angle of the mandible and superioinferiorly from 1.5 cm below the ala tragal line to 0.5 cm above the inferior border of the mandible. Surface over the swelling appears smooth with no signs of any discharge and color resembling that of adjacent normal structures. On palpation, there was no local rise in temperature and was tender. It was fi rm in consistency. Skin Figure 2: Case 1: The lesion on the left buccal mucosa with its extensions and finger- like projections Figure 3: Case 1: The panoramic radiograph showing no bony involvement Figure 4: Case 1: The photomicrograph of the lesion 2

3 Verrucous Carcinoma Case Series Gaurav, et al. over the swelling was pinchable and there was no alteration in sensory fi ndings like paresthesia or numbness. On palpation, the inspectory fi ndings were confi rmed. The intraoral lesion [Figure 7] presented as an exophytic growth with fi ngerlike projections on the left buccal mucosa, irregular in shape and measuring approximately 4 cm 3 cm with a wrinkled surface and a characteristic caulifl ower appearance. After the histopathological, investigations were carried out, it was diagnosed as VC of the left buccal mucosa (Stage IV [T 4 N X ]). No evidence of intraosseous involvement was elicited from the panoramic radiograph [Figure 8]. Case 3 A 75-year-old male patient [Figure 9] presented with an extensive intraoral growth on his left buccal mucosa associated with an extraoral swelling with a sinus opening and lymph node involvement which is unique for a VC. The extraoral swelling (measuring approximately 1.2 cm 2.5 cm) with the sinus opening [Figure 10] was seen in the middle third of the face measuring approximately 1.5 cm 2.5 cm. There were signs of pus discharge. The extensive intraoral growth [Figure 11] measured 4 cm 5 cm and extended anteroposteriorly from the retrocommisures to the retromolar pad region on the left buccal mucosa and superioinferiorly from the upper to the lower GBS on the left side. Left submandibular lymph nodes (Level IA) and the upper jugular lymph nodes (Level II) were tender and palpable (solitary, soft to fi rm in consistency, measuring <2 cm, mobile and superfi cial). After investigations (panoramic radiograph [Figure 12], chest X-ray and incisional biopsy) were carried out, fi nal diagnosis of VC of the left buccal mucosa (Stage IV [T 4 N 2 ]) was given. This case, however, was quite unique in its presentation with lymph node involvement which was highly unlikely for a VC where normally the regional lymph nodes are not involved. Chest X-rays were also taken to rule out metastasis which showed no such signs of metastasis. Thus, this series of three cases of VC projects three varying presentations of the same lesion. Discussion and Review of Literature Is believed to have the same biological potential. About 4:1 female/male ratio has been shown in Hansen et al. s study and an approximately equal sex distribution in other studies Figure 5: Case 2: Patient s profile picture with extraoral swelling on left side Figure 7: Case 2: Extensive growth see on the left buccal mucosa Figure 6: Case 2: Extraoral swelling on left lower third of the face Figure 8: Case 2: No bony involvement elicited on the panoramic radiograph 3

4 Gaurav, et al. Verrucous Carcinoma Case Series Figure 9: Case 3: Patient s profile picture Figure 11: Case 3: Severe extensive growth on the left buccal mucosa Figure 12: Case 3: No bony changes elicited on the panoramic radiograph Figure 10: Case 3: Extraoral sinus opening on left side of the face with respect to this carcinoma. VC refers to the group of exophytic lesions or tumors (mucosal/cutaneous) that appear pedunculated or above the epithelial surface with micronodular surface and blunt margins of pushing kind. Synonyms for oral VC (OVC) include epithelioma cuniculatum, carcinoma cuniculatum, and fl orid oral papillomatosis. Various factors determining its macroscopic appearance include degree of keratinization, changes in surrounding mucosa and duration of the lesion. White, warty appearance clinically is the presence of keratin or irregular moist mucosal surfaces. Lesions show well defi ned lower border and blunt rete ridges. A variant named verrucous hyperplasia was coined by Shear and Pindborg, in 1980, which is now considered as an early or antecedent stage of VC. 10 histological stages of VC have been described by Walvekar et al. [6] starting from slow growing persistent benign unifocal lesion to poorly or less differentiated SCCs. These stages were later modifi ed and reduced to four by Batsakis et al. which included: Clinically fl at leukoplakia without dysplasia Verrucous hyperplasia VC Conventional SCC. [7,8] Infl ammatory infi ltrates in histopathological sections from the biopsied tissues consist of histiocytes, lymphocytes, and plasma cells. [5] Batsakis et al. [6] proposed another terminology called hybrid VC which is a non-verrucous SCC arising synchronously with VC in the same macroscopic fi eld. Various treatment modalities have been tried, but surgical management is the treatment of choice until date. [9-11] When the tumor extends to the retromolar area, combined therapy is useful. Cytostatic drugs such as 5-Fu and colchicine form the main line of treatment in medical management where surgery is not indicated. [1,12] Conclusion Surgical management has brought about excellent prognosis in various cases of OVC. Positive margins require surgical resection. Following up of the patients becomes mandatory owing to enhanced incidence of local recurrences reported in literature 4

5 Verrucous Carcinoma Case Series Gaurav, et al. associated with propensity to developing second primary cancers. Most common sites include the GBS followed by hard palate and maxillary alveolus. The presence of other potentially malignant disorders in association with OVC predisposes it for multicentricity and strengthens the argument for a close follow-up. References 1. Prioleau PG, Santa Cruz DJ, Meyer JS, Bauer WC. Verrucous carcinoma: A light and electron microscopic, autoradiographic, and immunofluorescence study. Cancer 1980;45: 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: Singh K, Kalsotra P, Khajuria R. Manhas M. Verrucous carcinoma (Ackerman s tumor) of mobile tongue. JK Sci 2004;6: Depprich RA, Handschel JG, Fritzemeier CU, Engers R, Kubler NR. Hybrid verrucous carcinoma of the oral cavity: A challenge for the clinician and the pathologists. Oral Oncol Extra 2006;42: Regezi AJ, Sciuba JJ, Jordan RC. Oral Pathology Clinical Pathological Correlations. 5 th ed. St. Louis, Missouri: Saunders Elsevier; Rajendran R. Benign and malignant tumors of the oral cavity. In: Rajendran R, Sivapathasundaram B, editors. Shafer s Textbook of Oral Pathology. 5 th ed. New Delhi: Elsevier; p Varshney S, Singh J, Saxena RK, Kaushal A, Pathak VP. Verrucous carcinoma of larynx. Indian J Otolaryngol Head Neck Surg 2004;56: Cardesa A, Slootweg PJ. Pathology of the Head and Neck. Germany, Berlin, Heidelberg: Springer Verlag; Cawson RA, Odell EW, Porter S. Cawson s Essentials of Oral Pathology and Oral Medicine. 7 th ed. Edinburgh: Churchill Livingstone; Wenig BM, Cohan JM. General principles of management of head and neck cancer. In: Harrison LB, Sessions RB, Hong WK, editors. Head and Neck Cancer: A Multidisciplinary Approach. 3 rd ed. Philadelphia: Lippincott William and Wilkins; p Neville BW, Damm DD, Allen CM, Bouquot JE. Oral and Maxillofacial Pathology. 2 nd ed. Philadelphia: W.B. Saunders; Stokes SM, Castle CJ. Oral cancer: Part II. Clin Update 2004;26: