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 Table of Contents  
CASE REPORT
Year : 2015  |  Volume : 2  |  Issue : 2  |  Page : 103-105

Proliferative verrucous leukoplakia: Case report and a comprehensive review of literature


1 Private Practitioner, Department of Oral Medicine and Radiology, Rishi Raj College of Dental Science and Research Centre, Bhopal, Madhya Pradesh, India
2 Department of Oral Medicine and Radiology, Rishi Raj College of Dental Science and Research Centre, Bhopal, Madhya Pradesh, India
3 Department of Oral Medicine and Radiology, Peoples Dental Academy, Bhopal, Madhya Pradesh, India

Date of Web Publication14-Jul-2015

Correspondence Address:
Kriti Shrivastava
E-114/24, Shivaji Nagar, Bhopal, Madhya Pradesh - 462 016
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/1658-6816.160779

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  Abstract 

Proliferative verrucous leukoplakia (PVL) is a rare form of oral leukoplakia, which was first described in 1985 by Hansen et al. Since then, various published case series have presented PVL as a disease with aggressive biological behavior due to its high probability of recurrence and a high rate of malignant transformation, usually higher than 70%. PVL is a long-term progressive condition, and tends to become multifocal with a progressive deterioration of the lesions, making it more and more difficult to control. According to reported literature tobacco use does not seem to have a significant influence on the appearance or progression of PVL as these lesions may occur both in smokers and nonsmokers. At present, the etiology of PVL remains unclear as well as its management and diagnosis, which is still retrospective, late and poorly defined, lacking consensus criteria.
Here we present a case of PVL and intend to discuss the key features related to etiology, diagnosis, management and prognosis of proliferative verrucous leukoplakia.

Keywords: Leukoplakia, proliferative, multifocal


How to cite this article:
Raghuvanshi V, Shrivastava K, Handa H. Proliferative verrucous leukoplakia: Case report and a comprehensive review of literature. Saudi J Oral Sci 2015;2:103-5

How to cite this URL:
Raghuvanshi V, Shrivastava K, Handa H. Proliferative verrucous leukoplakia: Case report and a comprehensive review of literature. Saudi J Oral Sci [serial online] 2015 [cited 2019 Jun 20];2:103-5. Available from: http://www.saudijos.org/text.asp?2015/2/2/103/160779


  Introduction Top


The occurrence of red and white lesions is frequent in oral cavity in the Indian sub-continent with a known prevalence of approximately 24.8% and among the various lesions; oral leukoplakia is the most common with estimated prevalence rate of 0.2-3.6%. [1] The term proliferative verrucous leukoplakia (PVL) was defined by Hansen et al. as a disease of unknown origin that clinically often begins as a single white lesion and with time tends to become multifocal, growing slowly and progressively. There is a low probability that some lesions may initially be pinkish or even red rather than white. [2] As per to the latest World Health Organization nomenclature, Oral PVL conforms to the new terminology of "potentially malignant disorders" given that it is neither a delimited lesion nor a condition. [1],[3]


  Case Report Top


A 50-year-old male patient reported to the department with a chief complaint of burning sensation in the mouth since 15 days. According to the history given by the patient, he noticed ulcers in mouth 2 years back, which gradually progressed to the current condition and are associated with burning sensation since past 15 days. There is no relevant past medical history. Patient gave a positive history of tobacco chewing 2-3 times a day since 27 years.

On intraoral examination, multifocal nonhomogenous red and white lesion was present on posterior 1/3 of right and left buccal mucosa [Figure 1]a and b and involving almost entire palate [Figure 2]. The red component was atrophic in nature. There was verrucous growth present on posterior left side of hard palate, measuring approximately 2 cm × 1 cm approximately 1 cm away from the midline [Figure 1]a and b, with gingiva associated with mandibular left 2 nd premolar and 1 st molar, edentulous left mandibular alveolus with respect to 2 nd and 3 rd molars and posterior part of left buccal mucosa. On palpation, most of the lesion was firm in consistency with a leathery texture. Lesion was non-scrapable and was slightly tender.{Figure 1}
Figure 2: Lesion involving almost entire palate

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As the lesion was multifocal and verrucous in nature, a provisional diagnosis of PVL was made differential diagnosis considered were chronic hyperplastic candidiasis, plaque type oral lichen planus, and verrucous carcinoma. Incisional biopsy was performed from two different sites, which revealed orthokeratinized stratified squamous epithelium showing nuclear and cellular pleomorphism up to two-third of epithelium, keratin pearl formation also seen in a superficial layer of epithelium. Alteration in N:C ratio along with abnormal mitosis suggestive of severe dysplasia thus indicating verrucous leukoplakia.


  Discussion Top


The term PVL was introduced by Hansen [2] and classified the pathological process of PVL into 10 grades that is, normal oral mucosa (0), homogeneous leukoplakia (2), verrucous hyperplasia (4), verrucous carcinoma (6), papillary squamous cell carcinoma (8), and poorly differentiated carcinoma (10), in which the odd scores refer to a status intermediate between those referred to by the adjacent even scores. The proliferative nature of PVL was explained on the basis of high rate of field cancerization, which was seen in PVL patients. [3] It has also been reported that there is usually a time lag between the appearances of new tumors in the same patient suggesting that it might have an infectious etiology possibly viral. Various researchers like Palefsky et al. (1995), [4] Gopalakrishnan et al. (1997) [5] and Eversole (2000). [6] However Bagαn et al. (2004) [7] detected the presence of epstein-barr virus instead of human papillomavirus in a large percent of their patient group suggesting its role in PVL.


  Diagnosis Top


There are only two studies, one by Ghazali et al. [8] and another by Gandolfo et al. [9] which apply a set of diagnostic criteria to their respective cases, although these are just a transcription of Hansen's definition. Thus, Ghazali et al. [8] established the following criteria [Table 1].
FIgure 1:

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In order to make the diagnosis of PVL, they proposed that one of the two following combinations of criteria mentioned in [Table 2] should be met:

  1. Three major criteria (being E among them) or
  2. Two major criteria (being E among them) + two minor criteria.
Table 2: Major and minor diagnostic criteria for PVL

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  Management Top


Randomized controlled studies on PVL are lacking, and hence the published data are from series of retrospective cases or case reports only. There is no effective management reported, and recurrences have been seen even after treatment. Schoelch et al. reported laser treatment using CO2 and Nd:YAG lasers, but found a high rate of recurrences (83%). [10] Fettig et al. [11] also found that the lesions recurred after conservative scalpel or laser excision, and many developed into verrucous or oral squamous cell carcinoma.

Femiano et al. [12] reported an open trial of surgery in 25 cases with PVL compared with another 25 cases treated with surgery and the antiviral methisoprinol when the methisoprinol appeared to offer a significant benefit, but these results have yet to be confirmed in other studies. Thus, the lesions can be managed with surgery, carbon dioxide laser, and photodynamic therapy.

 
  References Top

1.
Cerero-Lapiedra R, Baladé-Martínez D, Moreno-López LA, Esparza-Gómez G, Bagán JV. Proliferative verrucous leukoplakia: A proposal for diagnostic criteria. Med Oral Patol Oral Cir Bucal 2010;15: e839-45.  Back to cited text no. 1
    
2.
Hansen LS, Olson JA, Silverman S Jr. Proliferative verrucous leukoplakia. A long-term study of thirty patients. Oral Surg Oral Med Oral Pathol 1985;60:285-98.  Back to cited text no. 2
    
3.
Scheifele C, Reichart PA. Is there a natural limit of the transformation rate of oral leukoplakia? Oral Oncol 2003;39:470-5.  Back to cited text no. 3
    
4.
Palefsky JM, Silverman S Jr, Abdel-Salaam M, Daniels TE, Greenspan JS. Association between proliferative verrucous leukoplakia and infection with human papillomavirus type 16. J Oral Pathol Med 1995;24:193-7.  Back to cited text no. 4
    
5.
Gopalakrishnan R, Weghorst CM, Lehman TA, Calvert RJ, Bijur G, Sabourin CL, et al. Mutated and wild-type p53 expression and HPV integration in proliferative verrucous leukoplakia and oral squamous cell carcinoma. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 1997;83:471-7.  Back to cited text no. 5
    
6.
Eversole LR. Papillary lesions of the oral cavity: Relationship to human papillomaviruses. J Calif Dent Assoc 2000;28:922-7.  Back to cited text no. 6
    
7.
Bagán JV, Murillo J, Poveda R, Gavaldá C, Jiménez Y, Scully C. Proliferative verrucous leukoplakia: Unusual locations of oral squamous cell carcinomas, and field cancerization as shown by the appearance of multiple OSCCs. Oral Oncol 2004;40:440-3.  Back to cited text no. 7
    
8.
Ghazali N, Bakri MM, Zain RB. Aggressive, multifocal oral verrucous leukoplakia: Proliferative verrucous leukoplakia or not? J Oral Pathol Med 2003;32: 383-92.  Back to cited text no. 8
    
9.
Gandolfo S, Castellani R, Pentenero M. Proliferative verrucous leukoplakia: A potentially malignant disorder involving periodontal sites. J Periodontol 2009;80:274-81.  Back to cited text no. 9
    
10.
Schoelch ML, Sekandari N, Regezi JA, Silverman S Jr. Laser management of oral leukoplakias: A follow-up study of 70 patients. Laryngoscope 1999;109: 949-53.  Back to cited text no. 10
    
11.
Fettig A, Pogrel MA, Silverman S Jr, Bramanti TE, Da Costa M, Regezi JA. Proliferative verrucous leukoplakia of the gingiva. Oral Surg Oral Med Oral Pathol Oral Radiol Endod. 2000;90:723-30.  Back to cited text no. 11
    
12.
Femiano F, Gombos F, Scully C. Oral proliferative verrucous leukoplakia (PVL); open trial of surgery compared with combined therapy using surgery and methisoprinol in papillomavirus-related PVL. Int J Oral Maxillofac Surg 2001;30:318-22.  Back to cited text no. 12
    


    Figures

  [Table 1], [Figure 2]
 
 
    Tables

  [Table 1], [Table 2]



 

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Abstract
Introduction
Case Report
Discussion
Diagnosis
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References
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