DOI: 10.7860/JCDR/2015/11197.

5648
Case Report

Idiopathic Leukoplakia-
Dentistry Section

Report of a Rare Case and Review

Shesha Prasad R1, Ramakrishna T2, Anuradha pai3, Sujatha D4

ABSTRACT
Idiopathic leukoplakia is a rare potentially malignant lesion, usually found on the tongue with an increased risk of malignant transformation
as compared to the tobacco associated form. The risk of malignant transformation increases with age. Diagnosis poses a challenge to
the clinician as it is diagnosed by exclusion of other possible causes leading to hyperkeratosis. We present one such rare case in an
elderly male patient who was followed up for a year to record the course of the lesion and to report recurrences, if any.

Keywords: Idiopathic leukoplakia, Keratosis, Potentially malignant, Tongue lesion, White lesion

Case report Treatment

A 78-year-old male patient reported to the outpatient department, Complete excision of the lesion was performed and the histological
with a complaint of white patch on the right lateral border of the report was again consistent with leukoplakia.
tongue for one month. The physical examination was unremarkable.
Oral examination revealed a homogenous greyish white plaque on Follow-up
the right ventrolateral border measuring 4 cm x 2.5 cm [Table/ The patient was followed up once in four months to check
Fig-1&2]. The surface showed cracked mud appearance and on recurrence. The patient did not reveal any signs of recurrence even
palpation the lesion felt as raised and rough. The lesion was non after one year following excision [Table/Fig-4].
tender and non scrapable. Lips, buccal mucosa, pharynx, and extra
oral soft tissues appeared normal. Lymphadenopathy was absent. Discussion
The patient was neither under any medication nor had any tobacco Idiopathic leukoplakia (IL) being rare, must be differentiated from
associated habits. Dental examination revealed crowns i.r.t #11, tobacco associated leukoplakia [1]. Van der Waal et al., in a study
#12#13,#14,#15,#16,#44,#45,#46,#47,#34, and #35. No sharp reported idiopathic leukoplakia to have an incidence of 36% [2].
margins were evident on palpation of these crowns. A provisional Clinical manifestations more commonly appear on the tongue [3]
diagnosis of (idiopathic) leukoplakia was considered. but can develop on the gingiva [4]. Sapna et al., reported idiopathic
leukoplakia on the gingiva in a 40-year-old male prevalent for
Investigations four months [4]. These lesions are commonly observed in female
Routine blood investigations were reported as normal. Toluidine patients, invariably with epithelial dysplasia [2]. They can recur and
blue stain testing came negative. Incisional biopsy was planned and have tendency towards malignant transformation. Vechio et al.,
it included normal and affected mucosa from the site. [5] reported a case where idiopathic leukoplakia was persistent
for 1.2 y on the dorsal surface of tongue in a 76-year-old female,
Differential diagnosis which transformed into verrucus proliferative leukoplakia and
Considering the patient’s age, traumatic or frictional keratosis was ultimately squamous cell carcinoma. Oral leukoplakia in general
included. Teeth wear in the elderly can cause frictional keratosis is a potentially malignant disorder with an overall malignant
in the lateral aspect of the tongue. This patient had crowns in the
right posterior quadrant with no sharp margins or edges ruling out
the diagnosis of traumatic or frictional keratosis. Oral hyperplastic
candidiasis was the next differential diagnosis that was considered,
which is also commonly seen in the elderly vowing to the various
medications they consume and diabetes mellitus that commonly
causes xerostomia. This patient was neither diabetic nor was
under any medication. The mucosa appeared normal and was well
hydrated ruling out candidiasis.
Chemical injury leading to a white plaque appearance was also
considered. Usually a history of a drug or medication that is applied
locally in the area of the lesion is strongly positive. The current case
did not reveal any such history.
Incisional biopsy revealed histological features that were consistent
with hyperplastic stratified squamous epithelium with intraepithelial
inflammatory cell infiltration consistent with leukoplakia [Table/
Fig-3]. Diagnosis of idiopathic leukoplakia was derived based on
the history and clinical examination. [Table/Fig-1]: Lateral aspect of the lesion

Journal of Clinical and Diagnostic Research. 2015 Mar, Vol-9(3): ZD11-ZD12 11

 Shesha Prasad R et al., Idiopathic Leukoplakia- Report of a Rare Case and Review www.jcdr.net

[Table/Fig-2]: Ventrolateral aspect of the lesion [Table/Fig-3]: 10X view: Hyperplastic epithelium showing keratin pearl formation and individual cell keratinisation
[Table/Fig-4]: No recurrence after one year follow up

transformation of 1% worldwide. 0.3% malignant transformation Accurate diagnosis can be established only by detailed contributing
is seen in the Indian population [6]. IL shows greater tendency for history such as tobacco related habits, systemic diseases,
malignant transformation and studies have shown that the malignant medications and conclusively by histological studies correlating with
transformation ranges from 0.13% to 36.4% [7]. Vaander Waal and history.
Arduino have reported that the lesions on the lateral border of the
tongue in elderly, especially Indian men are considered at a greater Conclusion
risk for carcinogenesis [6,7]. Arduino also reported that the lesions Cases of ILs are scantily reported in literature. Further research
on the ventrolateral surface of the tongue have shown greater risk elaborating the cause of IL, its genetic predisposition, reason for
of aneuploidy and loss of heterozygosity [7] which are the earliest tongue as the common site involved, rationale behind increased
changes towards malignant transformation. malignant transformation when compared with tobacco associated
The present lesion in this patient was considered as idiopathic as form, and as to why the elderly population are affected more though
patient did not have any tobacco associated habits. Other causes there is no continuous exposure to any tobacco associated habit
like friction, trauma from sharp teeth, chemical injury, candidiasis needs to be addressed. Extensive reporting and analysis is required
were ruled out. The case discussed by Vechio et al., [5], showed for possible answers.
signs of metamorphosis to verrucus form whereas the present
case showed no such signs of metamorphosis. As described in References
[1] Isaäc van der Waal, Axell T. Oral leukoplakia: a proposal for uniform reporting.
numerous studies by Vaander Waal and Arduino, the present case
Oral Oncol. 2002;(38):521–26.
had all the risk factors which warranted complete excision of the [2] Freitas MD, Carrión AB, Vila PG,López JA, García AG,Rey JMG. Clinicopathologic
lesion. The duration of the lesion was approximately seven weeks aspects of oral leukoplakia in smokers and non-smokers. Oral Surg Oral Med
before excision and had showed no signs of regression. The Oral Pathol Oral Radiol Endod. 2006;102:199-203.
[3] Faecher RS, Thomas JE. Idiopathic leukoplakia lingualis. Gen Dent.
incisional biopsy revealed histopathological findings of hyperplastic 1993;41(6):547-49.
parakeratotic epithelium with no signs of dysplasia. However, mild [4] Sapna L, Vandana KL. Idiopathic linear leukoplakia of gingiva: A rare case report.
signs of dysplasia were noted in the second specimen following Journal of Indian Society of Periodontology. 2010;14:198-200.
complete excision. This case was followed up for one year and no [5] Vechio AD, Nakagima E, Azevedo LH, Alves FA, Migliari DA. Rapid progression
of a idiopathic leukoplakia to aproliferative verrucous leukoplakia lesion and then
recurrence has been noted till date. squamous cell carcinoma. Quintessence Int. 2012;43:583-85.
[6] Isaäc van der Waal. Potentially malignant disorders of the oral and oropharyngeal
Points to be noted mucosa; terminology, classification and present concepts of management. Oral
Oncol. 2009;(45):317–23.
IL is considered as a pre-malignant or a potentially malignant lesion [7] Arduino PG, Bagan J, El-Naggar AK,Carrozzo M. Urban Legends Series: Oral
more aggressive than oral leukoplakias. As it is less prevalent, its Leukoplakia. Oral Dis. 2013;19(7):642-59.
diagnosis poses a major challenge for practitioners. The diagnosis
of IL is mainly based on exclusion.

PARTICULARS OF CONTRIBUTORS:
1. Lecturer, Department of Oral Medicine & Radiology, The Oxford Dental College, Bommanahalli, Hosur Road, Bangalore, Karnataka, India.
2. Professor, department of Oral & Maxillofacial Surgery, The Oxford Dental College, Hospital, Bommanahalli, Hosur Road, Bangalore, Karnataka, India.
3. Professor & Head, Department of Oral Medicine & Radiology, The Oxford Dental College, Hospital & Research Centre,
Bommanahalli, Hosur Road, Bangalore, Karnataka, India.
4. Professor, Department of Oral Medicine & Radiology, The Oxford Dental College, Hospital & Research Centre, Bommanahalli,
Hosur Road, Bangalore, Karnataka, India.

NAME, ADDRESS, E-MAIL ID OF THE CORRESPONDING AUTHOR:

Dr. Shesha Prasad R,
The Oxford Dental College Bommanahalli, Hosur Road Bengaluru-560061, Karnataka, India. Date of Submission: Sep 10, 2014
E-mail: [email protected] Date of Peer Review: Oct 09, 2014
Date of Acceptance: Oct 13, 2014
Financial OR OTHER COMPETING INTERESTS: None. Date of Publishing: Mar 01, 2015

12 Journal of Clinical and Diagnostic Research. 2015 Mar, Vol-9(3): ZD11-ZD12