Idiopathic Leukoplakia- Report of a Rare Case and Review

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.

Full text

Journal of Clinical and Diagnostic Research. 2015 Mar, Vol-9(3): ZD11-ZD12 1111
DOI: 10.7860/JCDR/2015/11197.5648 Case Report
Keywords: Idiopathic leukoplakia, Keratosis, Potentially malignant, Tongue lesion, White lesion
CASE REPORT
A 78-year-old male patient reported to the outpatient department,
with a complaint of white patch on the right lateral border of the
tongue for one month. The physical examination was unremarkable.
Oral examination revealed a homogenous greyish white plaque on
the right ventrolateral border measuring 4 cm x 2.5 cm [Table/
Fig-1&2]. The surface showed cracked mud appearance and on
palpation the lesion felt as raised and rough. The lesion was non
tender and non scrapable. Lips, buccal mucosa, pharynx, and extra
oral soft tissues appeared normal. Lymphadenopathy was absent.
The patient was neither under any medication nor had any tobacco
associated habits. Dental examination revealed crowns i.r.t #11,
#12#13,#14,#15,#16,#44,#45,#46,#47,#34, and #35. No sharp
margins were evident on palpation of these crowns. A provisional
diagnosis of (idiopathic) leukoplakia was considered.
Investigations
Routine blood investigations were reported as normal. Toluidine
blue stain testing came negative. Incisional biopsy was planned and
it included normal and affected mucosa from the site.
Differential diagnosis
Considering the patient’s age, traumatic or frictional keratosis was
included. Teeth wear in the elderly can cause frictional keratosis
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.
Treatment
Complete excision of the lesion was performed and the histological
report was again consistent with leukoplakia.
Follow-up
The patient was followed up once in four months to check
recurrence. The patient did not reveal any signs of recurrence even
after one year following excision [Table/Fig-4].
DISCUSSION
Idiopathic leukoplakia (IL) being rare, must be differentiated from
tobacco associated leukoplakia [1]. Van der Waal et al., in a study
reported idiopathic leukoplakia to have an incidence of 36% [2].
Clinical manifestations more commonly appear on the tongue [3]
but can develop on the gingiva [4]. Sapna et al., reported idiopathic
leukoplakia on the gingiva in a 40-year-old male prevalent for
four months [4]. These lesions are commonly observed in female
patients, invariably with epithelial dysplasia [2]. They can recur and
have tendency towards malignant transformation. Vechio et al.,
[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,
which transformed into verrucus proliferative leukoplakia and
ultimately squamous cell carcinoma. Oral leukoplakia in general
is a potentially malignant disorder with an overall malignant
Dentistry Section
Idiopathic Leukoplakia-
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.
[Table/Fig-1]: Lateral aspect of the lesion

Shesha Prasad R et al., Idiopathic Leukoplakia- Report of a Rare Case and Review www.jcdr.net
Journal of Clinical and Diagnostic Research. 2015 Mar, Vol-9(3): ZD11-ZD12
1212
transformation of 1% worldwide. 0.3% malignant transformation
is seen in the Indian population [6]. IL shows greater tendency for
malignant transformation and studies have shown that the malignant
transformation ranges from 0.13% to 36.4% [7]. Vaander Waal and
Arduino have reported that the lesions on the lateral border of the
tongue in elderly, especially Indian men are considered at a greater
risk for carcinogenesis [6,7]. Arduino also reported that the lesions
on the ventrolateral surface of the tongue have shown greater risk
of aneuploidy and loss of heterozygosity [7] which are the earliest
changes towards malignant transformation.
The present lesion in this patient was considered as idiopathic as
patient did not have any tobacco associated habits. Other causes
like friction, trauma from sharp teeth, chemical injury, candidiasis
were ruled out. The case discussed by Vechio et al., [5], showed
signs of metamorphosis to verrucus form whereas the present
case showed no such signs of metamorphosis. As described in
numerous studies by Vaander Waal and Arduino, the present case
had all the risk factors which warranted complete excision of the
lesion. The duration of the lesion was approximately seven weeks
before excision and had showed no signs of regression. The
incisional biopsy revealed histopathological findings of hyperplastic
parakeratotic epithelium with no signs of dysplasia. However, mild
signs of dysplasia were noted in the second specimen following
complete excision. This case was followed up for one year and no
recurrence has been noted till date.
Points to be noted
IL is considered as a pre-malignant or a potentially malignant lesion
more aggressive than oral leukoplakias. As it is less prevalent, its
diagnosis poses a major challenge for practitioners. The diagnosis
of IL is mainly based on exclusion.
[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
Accurate diagnosis can be established only by detailed contributing
history such as tobacco related habits, systemic diseases,
medications and conclusively by histological studies correlating with
history.
CONCLUSION
Cases of ILs are scantily reported in literature. Further research
elaborating the cause of IL, its genetic predisposition, reason for
tongue as the common site involved, rationale behind increased
malignant transformation when compared with tobacco associated
form, and as to why the elderly population are affected more though
there is no continuous exposure to any tobacco associated habit
needs to be addressed. Extensive reporting and analysis is required
for possible answers.
REFERENCES
Isaäc van der Waal, Axell T. Oral leukoplakia: a proposal for uniform reporting. [1]
Oral Oncol. 2002;(38):521–26.
Freitas MD, Carrión AB, Vila PG,López JA, García AG,Rey JMG. Clinicopathologic [2]
aspects of oral leukoplakia in smokers and non-smokers. Oral Surg Oral Med
Oral Pathol Oral Radiol Endod. 2006;102:199-203.
Faecher RS, Thomas JE. Idiopathic leukoplakia lingualis. [3] Gen Dent.
1993;41(6):547-49.
Sapna L, Vandana KL. Idiopathic linear leukoplakia of gingiva: A rare case report. [4]
Journal of Indian Society of Periodontology. 2010;14:198-200.
Vechio AD, Nakagima E, Azevedo LH, Alves FA, Migliari DA. Rapid progression [5]
of a idiopathic leukoplakia to aproliferative verrucous leukoplakia lesion and then
squamous cell carcinoma. Quintessence Int. 2012;43:583-85.
Isaäc van der Waal. Potentially malignant disorders of the oral and oropharyngeal [6]
mucosa; terminology, classification and present concepts of management. Oral
Oncol. 2009;(45):317–23.
Arduino PG, Bagan J, El-Naggar AK,Carrozzo M. Urban Legends Series: Oral [7]
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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.
E-mail: drsheshaprasad@gmail.com
FINANCIAL OR OTHER COMPETING INTERESTS: None.
Date of Submission: Sep 10, 2014
Date of Peer Review: Oct 09, 2014
Date of Acceptance: Oct 13, 2014
Date of Publishing: Mar 01, 2015