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Idiopathic linear leukoplakia of gingiva: A rare case report

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White lesions of the oral cavity are not uncommon though majority of them are benign. This case report documents a rare case of idiopathic linear leukoplakia of gingiva with no apparent etiology. Initial examination revealed a non-scrapable linear white lesion on the marginal and papillary gingiva of upper right teeth region. Incisional biopsy was taken for pathologic evaluation. Patient was treated with routine oral hygiene procedures and excision of the lesions. The histopathological results demonstrated hyperparakeratinized/orthokeratinized hyperplastic oral epithelium with orthokeratin-filled clefts and with no dysplasia. Clinical results demonstrated no recurrence after electrosurgical intervention. This paper reports a rare case of idiopathic linear leukoplakia of gingiva which was non-dysplastic in nature. Electrosurgical treatment proved to be successful compared to surgical technique as there was no recurrence even after two years of follow-up.
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198 Journal of Indian Society of Periodontology - Vol 14, Issue 3, Jul-Sep 2010
Case Report
Address for
correspondence:
Dr. Vandana K.L.,
Department of
Periodontics, College
of Dental Sciences,
Davangere - 577 004,
Karnataka, India.
E-mail: vanrajs@gmail.com
Submission: 08-01-2010
Accepted: 18-09-2010
DOI: 10.4103/0972-
124X.75918
Department of
Periodontics, D.A.P.M
R.V. Dental College,
Bangalore, and 1College
of Dental Sciences,
Davangere, Karnataka,
India
Idiopathic linear leukoplakia of
gingiva: A rare case report
N. Sapna, K. L. Vandana1
Abstract:
White lesions of the oral cavity are not uncommon though majority of them are benign. This case report documents
a rare case of idiopathic linear leukoplakia of gingiva with no apparent etiology. Initial examination revealed a
non-scrapable linear white lesion on the marginal and papillary gingiva of upper right teeth region. Incisional
biopsy was taken for pathologic evaluation. Patient was treated with routine oral hygiene procedures and excision
of the lesions. The histopathological results demonstrated hyperparakeratinized/orthokeratinized hyperplastic
oral epithelium with orthokeratin-lled clefts and with no dysplasia. Clinical results demonstrated no recurrence
after electrosurgical intervention. This paper reports a rare case of idiopathic linear leukoplakia of gingiva which
was non-dysplastic in nature. Electrosurgical treatment proved to be successful compared to surgical technique
as there was no recurrence even after two years of follow-up.
Key words:
Gingiva, idiopathic, linear leukoplakia, non-malignant
INTRODUCTION
Any condition that increases the thickness
of the epithelium causes it to appear white
by increasing the distance to the vascular bed.
Most often lesions appear white because of a
thickening of the keratin layer, or hyperkeratosis.
Other common causes of a white appearance
include acanthosis, an increase in the amount
of edema uid in the epithelium, and reduced
vascularity in the underlying lamina propria.
Surface ulcerations covered by a brin cap can
also appear white, as would collapsed bullae.[1]
The development of oral white patches is not
uncommon, but, fortunately, the majority of
lesions are due to benign conditions. However, a
small percentage of white patches may represent
either oral cancer or have an association with the
likelihood of the development of oral cancer. The
presence of sinister lesions cannot be assessed by
clinical appearance alone and clinical diagnosis
of any persistent white patch should therefore
be conrmed histologically.[2]
Oral leukoplakia can be defined as “a
predominantly white lesion of the oral mucosa
that cannot be characterized as any other
definable lesion; some oral leukoplakia will
transform into cancer”.[3]
Leukoplakia can be subdivided according to
etiology and clinical factors and one of the types
is idiopathic leukoplakia where no etiology for
the patch can be found.[4]
This paper describes a case of idiopathic linear
leukoplakia involving the marginal and papillary
gingiva with no apparent etiology.
CASE DISCRIPTION AND RESULTS
Clinical ndings
A 40-year-old male patient came to the
Department of Periodontics, College of Dental
Sciences, Davangere, India, with a chief
complaint of thin white lesion on the gums in the
upper front region since 4 months. He noticed
the lesion when he got his upper front teeth
removed, which was placed irregularly on the
inner side. The lesion was totally asymptomatic
except for the esthetic concern of the patient.
His medical history disclosed that he was a
well-controlled hypertensive patient since four
years. Patient did not smoke tobacco, or chew
areca nut. No history of trauma or surgery was
disclosed in that particular region. However,
patient gave history of vigorous brushing and
taking hot beverages.
Clinical examination revealed a linear white
lesion on the upper right region involving
marginal and papillary gingiva of both labial
and palatal side. It was more prominent on labial
aspect of 11, 12, 13 and 16 and palatal aspect of
11, 12, 13, 15 and 16 [Figures 1 and 2]. Lesions
were non-scrapable. The patient’s oral hygiene
was good. Radiographic examination showed
normal findings. Laboratory investigations
revealed hemoglobin level of 13.4 g%, a white
blood cell count of 5200 cells/mm3 and normal
platelet count. Random blood sugar was within
normal limits [Table 1]. There was no evidence of
skin lesions. HIV testing showed negative results.
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Journal of Indian Society of Periodontology - Vol 14, Issue 3, Jul-Sep 2010 199
Histopathological ndings
An approval from the institution’s review committee and an
informed consent from the patient were obtained. An incisional
biopsy of the lesion was performed in relation to labial gingiva
of 13 and 16 and palatal gingiva of 11 and 14 with patient under
local anesthesia. It was stained with hematoxylin and eosin
and observed under light microscope. Epithelium showed
exophytic proliferations lined by hyperparakeratinized /
orthokeratinized hyperplastic oral epithelium with few broad
and deep clefts lled with orthokeratin. Underlying connective
tissue showed lymphocytic inltration. There was no evidence
of dysplasia [Figure 3].
By correlating clinical and histopathological ndings the lesion
was diagnosed as idiopathic linear leukoplakia of the gingiva.
Treatment and follow-ups
The case was followed-up regularly for two years from August
2005 to August 2007. Scaling and oral hygiene procedures were
performed regularly. The linear white lesions were excised
using B.P. blade No. 15 under local anesthesia on the labial
aspect. Palatal lesions were not excised this time, so that they
could serve as reference lesions, if the labial lesions recurred
similar to previous lesion. At one-month reevaluation, the
labial linear white lesions had recurred. Once again the surgical
excision was done on both labial and palatal aspect and sent for
histopathological evaluation second time. Following the second
surgical intervention, linear white lesion had recurred again at
Sapna and Vandana: Leukoplakia of gingiva
Figure 1: First visit, buccal linear white lesions of marginal gingival in relation to
11, 12, 13
Figure 2: First visit, palatal linear white lesions in relation to 11, 12, 13, 15 and 16
Figure 3: H and E stain of tissue biopsy during rst visit showing hyperplastic oral
epithelium, lymphocytic inltration in connective tissue with no evidence
of dysplasia Figure 4: Recall visit, no recurrence of white lesion after two years of follow-up
Table 1: Complete hemogram
Report Normal values
Hb estimation 13.4 g% 14–18 g% (males 11.5–6.5
mg% (females)
ESR count 30 mm/m 0–15 mm (males) 0–20
mm/1 h (females)
RBC count 5.1 millus mm34.5×105–6×105/mm3
Platelet count 2.0 lacs/mm3 1×105–4×105/mm3
Total WBC count 5200 cells/mm34000–10,000/mm3
Bleeding time 1 min 45 s 1–5 min
Clotting time 3 min 15 s 4–9 min
Differential count
Neutrophils 58% 40–75%
Lymphocytes 36% 20–45%
Eosinophils 04% 01–04%
Monocytes 02% 02–08%
Basophils 00% 00–01%
Blood sugar
Random blood sugar 126.0 mg% 70 – 160 mg%
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200 Journal of Indian Society of Periodontology - Vol 14, Issue 3, Jul-Sep 2010
Sapna and Vandana: Leukoplakia of gingiva
one month’s recall. Considering the patient’s esthetic concern
and nondysplastic tissue changes in both the histopathological
report, an attempt was made to eliminate linear white lesions
using electrosurgery approximately 6 months after the rst
excision. The patient’s compliance was highly appreciable
during the maintenance phase. The regular checkup over two
years period did not reveal any recurrence of the white linear
lesion and patient was satised with the treatment and the
results [Figure 4].
DISCUSSION
Oral white lesions reflect many different diseases and
pathological changes. Some of them relate to diseases such
as lichen planus and lupus erythematosus. Others are local
changes with a clearly dened or highly probable etiology.
Leukoplakia is the most prevalent precancerous lesion of
the oral mucosa. The label “leukoplakia” was coined by
Schwimmer.[5] Approximately 70% oral leukoplakias are found
on the lip vermillion, buccal mucosa and gingiva.[6]
Leukoplakia of the gingiva varies in appearance from a
grayish white, attened, scaly lesion to a thick, irregularly
shaped keratinous plaque.[7] The cause of leukoplakia remains
unknown though it can be associated with the use of tobacco.
The lesions where the etiology is not apparent can be termed
as idiopathic leukoplakia.[4]
In the present case, since histopathological reports did not show
any dysplastic changes, premalignant and malignant lesions
were not taken into considerations for the diagnosis.
Among reactive and inammatory white lesions, frictional
(traumatic) keratosis was not considered for the diagnosis since
an identiable source of mechanical irritation like rough or
maladjusted dentures, sharp cusps and edges of broken teeth
was not found. Though patient gave a history of hard brushing
and consuming hot beverages, lesions did not resolve even
after patient left these habits ruling out these habits as source
for the lesion.
Chemical injuries were ruled out since patient did not give any
history of usage of chemicals. Since patient did not smoke or
chew tobacco, tobacco-induced keratosis was excluded from
the diagnosis.
Infectious lesions like candidiasis, bacterial infections or viral
infections were also ruled out since patient’s history was non-
suggestive and also biopsy report did not show any changes.
The use of vigorous brushing by the patient would not lead to
occurrence of lesions on the upper right side excluding the other
quadrants especially the lower right quadrant. Considering the
patient’s history, clinical and histopathological examination,
the lesion can be termed as idiopathic linear leukoplakia of
the gingiva.
Patient has been kept under observation and till date has not
shown any recurrence of the lesion.
CONCLUSION
This case report was considered, as idiopathic leukoplakia
on the gingiva is a very rare nding. It was considered as
idiopathic lesion as no etiology could be related to the present
lesion. Further observations should be done to detect any
recurrence of the lesion.
REFERENCES
1. Bhattacharya I, Cohen DM, Silverman S. Red and white lesions of
the oral mucosa. Burket’s oral medicine diagnosis and treatment.
10th ed. New Delhi: Harcourt (India) Private Ltd; 2003. p. 85-125.
2. Lamey PJ. Oral medicine in practice: White patches. Br Dent J
1990;168:147-52.
3. Axell T. Oral white lesions with special reference to precancerous
and tobacco related lesions: Conclusion of an international
symposium held in Uppsala, Sweden in 1994. J Oral Pathol Med
1996;25:49-54.
4. Axell T. Occurrence of leukoplakia and some other oral white
lesions among 20333 adult Swedish people. Community Dent
Oral Epidemiol 1987;15:46-51.
5. Schwimmer E. Some rare clinical pictures of oral and lingual
mucosa. Arch Dermat Syph 1877;9:641-70.
6. Neville BW, Damm DD, Allen CM, Bouquot JE. Epithelial
pathology. Oral Pathology and Maxillofacial Pathology. 2nd ed.
Philadelphia: Saunders; 2004.
7. Carranza FA, Hogan EL. Gingival enlargement. Carranza’s
clinical periodontology. 9th ed. Philadelphia: Saunders; 2003. p.
279-96.
Source of Support: Nil, Conict of Interest: None declared.
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... It is the most prevalent precancerous lesion of the oral mucosa. Some oral leukoplakia will transform into cancer [15]. The annual risk of malignant transformation of leukoplakia, if not malignant already at the first visit, is approximately 2-3% [16]. ...
... The annual risk of malignant transformation of leukoplakia, if not malignant already at the first visit, is approximately 2-3% [16]. Approximately 70% of oral leukoplakias are found on the lip vermillion, buccal mucosa and gingiva [15]. In the study by Cebeci et al., white lesions were observed in 2.2% of patients [17]. ...
... In the study by Cebeci et al., white lesions were observed in 2.2% of patients [17]. Gender, age and oral distribu- tion of leukoplakia in our study were confirmed in other studies [15][16][17]. In our opinion, clinical characteristics and the risk of malignant transformation in predominantly white lesions of the alveolar ridge ("alveolar ridge keratosis") and the buccal gingiva ("frictional keratosis") is lower than for similar lesions located on the borders of the tongue or the floor of the mouth. ...
Article
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Introduction: Oral lesions are divided into non-neoplastic lesions, potentially malignant lesions and neoplastic lesions. More clinical data are needed to determine their helpful clinical pattern. Aim: To present the epidemiological, clinical and histopathological characteristics of the oral lesions. Material and methods: The retrospective study group comprised records of 208 patients which were reviewed according to selected epidemiological and clinical features. All the biopsy specimens were classified into: reactive lesions, precancerous lesions/potentially malignant lesions, salivary gland pathologies, benign and malignant tumours. Results: The lower lip was the most common site involved followed by buccal and vestibular mucosa. The most frequent diagnoses were fibroma, mucocele and papilloma. The predominant pathomorphological forms were nodule and bulla. The most frequent salivary gland pathology was mucocele. Fibroma was the most frequent pathomorphological diagnosis, followed by mucocele and reactive lesions such as irritation fibroma (IF) and granuloma. Conclusions: In cases of oral mucosal lesions, we propose the following algorithm: the exclusion of all odontogenic and iatrogenic causes; the detection and elimination of harmful habits, parafunctions and irritants from the oral cavity especially from the vestibule of the oral cavity and from the lips; all surgical treatment should be performed only after the proper detection and elimination of causative factors to decrease the risk of recurrence; excisional biopsy or in more diffuse lesions incisional biopsy is recommended to confirm clinical diagnosis; and consideration of other factors that can modify the clinical pattern of oral lesions, such as oral hygiene, systemic diseases, and drugs.
... 15 Likewise, in a case of "idiopathic linear leukoplakia of gingiva", the patient presented with linear white lesions on the maxillary facial and palatal gingiva of multiple teeth. 19 Incisional biopsy showed hyperplastic oral epithelium with para-/ortho-keratosis, with a lymphocytic band and no dysplasia. The patient had two recurrences following excisional biopsies, but no recurrence was noted at two year follow-up post electrosurgery. ...
... The presence of white lesions on marginal gingiva of multiple teeth that showed rapid progression, recurrence and benign histopathology fit the proposed criteria for PVL. 19 Another study reported two cases of isolated white lesions on the marginal gingiva of teeth, one provisionally diagnosed as frictional keratosis. 13 The final diagnoses of the biopsied lesions were epithelial hyperplasia consistent with KUS, and verrucous hyperplasia with no features of epithelial dysplasia. ...
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Background: Potentially malignant lesions of the gingiva may frequently present as well-demarcated white lesions confined to the marginal gingiva. These lesions often become thick and verrucoid and spread along the marginal gingiva to encircle the tooth. Some cases of marginal gingival leukoplakia, over time, progress to extensively involve the gingiva fulfilling the criteria for proliferative verrucous leukoplakia (PVL). The objective of this study is to raise awareness of this pattern of leukoplakia by reporting a series of cases of marginal gingival leukoplakia. Methods: An IRB approved retrospective search of UF and UNMC oral biopsy services was performed for all gingival biopsies. Inclusion criteria included cases (a) exhibiting marginal gingival leukoplakia, and (b) with accompanying clinical images. Results: A total of 30 cases of marginal gingival leukoplakia were included. All cases presented as well-demarcated leukoplakias, either on the buccal or lingual gingival margin, or circumferentially forming a "ring around the collar" of single or multiple teeth. Eight patients had recurrent lesions and twelve had multifocal involvement. Six of the 12 patients with multifocal involvement presented with a "ring around the collar". The histopathologic diagnoses were representative of benign lesions in 7 cases, premalignant in 13, and malignant or suggestive of malignancy in 10 cases. Seven patients had carcinoma at the time of first biopsy, whereas 6 cases showed progression at time of follow-up. Conclusion: This study aims to raise awareness that marginal gingival leukoplakia may represent potentially malignant lesions, and if circumferential and/or thick, may be the first manifestation of PVL. This article is protected by copyright. All rights reserved.
... As the lesion described in the present report is a rare occurrence, difficulties were encountered when naming the lesion, especially with references to the terms "keratosis" and "leukoplakia." According to literature, similar lesions have been named as linear gingival keratosis [3] as well as linear gingival leukoplakia [9]. When linear gingival keratosis/leukoplakia reported in literature [10] was compared with the present lesion, it did not show bone erosion. ...
... When linear gingival keratosis/leukoplakia reported in literature [10] was compared with the present lesion, it did not show bone erosion. In contrast to lesions reported in literature [9,10], the present lesion showed dysplasia and p16 positivity. However, in this case, the lesion was diagnosed as an early stage of PVL rather than linear gingival keratosis, due to the fact that it occurred in a female in the gingiva and also as it was refractory to surgical management. ...
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This report describes a case of proliferative verrucous leukoplakia (PVL) of the gingiva with no discernible aetiology, which presented in a 36-year-old female. The initial nonscrapable gingival lesion was treated with CO 2 laser ablation, and the histopathological evaluation was carried out. The presence of koilocytic cells in the superficial epithelium led to immunohistochemical investigations with p16 antibody, which showed strong nuclear positivity and slight cytoplasmic positivity in >50% of the cells with >25% confluency. However, it was not possible to confirm the presence of HPV infection with further investigations due to logistic reasons. The lesion recurred twice within a short time despite the surgical resection following the first recurrence. Thus, this paper presents a case of proliferative verrucous leukoplakia, which demonstrated a significant resistance to routine treatment protocols recommended in the management of such lesions.
... Oral leukoplakia may be located on lip vermillion, gingival, tongue and floor of mouth, and it is on these latter regions that there is a higher risk of malignancy (around 43%) [38,40]. Oral leukoplakia distribution may be local or disseminated [41] (Figure 6 -A, F). Histological examination reveals a range of epithelium changes varying from innocuous hyperplasia to dysplasia of varying degrees [18]. ...
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Introduction Leukoplakia of the marginal gingivae is uncommon and in most cases reported up to date represents a manifestation of proliferative verrucous leukoplakia. The clinical and pathologic features of two cases of leukoplakia confined to the marginal gingiva are described and their biologic significance is discussed. Case Presentation The cases involved two female patients, non‐smokers, aged 82 and 57. The lesions clinically appeared as small, well‐demarcated white plaques on the marginal gingiva of posterior teeth. After being totally excised, microscopic examination showed keratosis of unknown significance in the first patient and verrucous hyperplasia in the latter, while immunohistochemistry for p16 INK4A was negative for both. There was no recurrence in 7 months and 5 months after excision, respectively. Conclusion A white plaque on the marginal gingiva may be overlooked due to its small size or may be misdiagnosed as frictional keratosis. However, it may represent leukoplakia, a potentially malignant disorder. Therefore, diagnosis and management should follow the established guidelines for leukoplakia.
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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.
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Abstract Among 20 333 people aged 15 yr or above, the prevalences of oral white lesions were calculated based on a partly new classification. The total prevalences were: cheek and lip biting 5.1%, smoker's palate 1.1%, frictional white lesion 5.5%, snuff dipper's lesion 8.0%, preleukoplakia 6.4% and leukoplakia 3.6%. If all these lesions were pooled, the prevalence was 24.8% and if only the entities cheek and lip biting and smoker's palate were excluded it became 20.1%. If weak “preleukoplakic” lesions were excluded from the latter figure (he prevalence for marked whitish lesions was 13.8%. Etiologic and clinical subgroups of leukoplakia showed the following prevalences: using the etiologic subgroups, idiopathic leukoplakia 0.7% and tobacco-associated leukoplakia 2.9%; using the clinical subgroups, homogeneous leukoplakia 3.5% and non-homogeneous leukoplakia 0.3%. The intraoral location pattern of leukoplakias was preponderant in the commissural and buccal areas. However, the idiopathic leukoplakias showed a somewhat more even distribution and thus a more similar distribution to that of oral cancer.
Article
The seventh article in this series deals with conditions of the oral mucosa which present as white patches. Although the majority of white patches are benign, certain lesions are associated with premalignancy or malignancy. Unfortunately, the presence of any sinister lesion cannot be assessed by clinical appearance and therefore accurate diagnosis (involving biopsy) is mandatory whenever there is uncertainty about the clinical diagnosis of an oral white patch.
Article
An international group of epidemiologists, clinicians and pathologists with a special interest in oral white lesions and their precancerous significance has reviewed earlier work on this topic and identified some of the problems associated with previous definitions, descriptions and classifications. Modifications to these definitions, descriptions and classifications have been proposed, accompanied by explanations of the reasons for identifying the need for changes to be made. Leukoplakia may be a provisional or definitive diagnosis dependent upon the circumstances of oral examination and the availability of other information. Guidelines are provided to assist in the application of the definitions of oral leukoplakia and illustrations depict the homogeneous and non-homogeneous clinical variants. Consideration is also given to the importance of a red component in a white lesion, or a lesion that is entirely red (erythroplakia). A new clinical staging procedure for oral leukoplakia is also proposed.
Some rare clinical pictures of oral and lingual mucosa
  • E Schwimmer
Schwimmer E. Some rare clinical pictures of oral and lingual mucosa. Arch Dermat Syph 1877;9:641-70.
Carranza’s clinical periodontology
  • FA Carranza
  • EL Hogan
Red and white lesions of the oral mucosa. Burket's oral medicine diagnosis and treatment. 10 th ed. New Delhi: Harcourt (India) Private Ltd
  • I Bhattacharya
  • D M Cohen
  • S Silverman
Bhattacharya I, Cohen DM, Silverman S. Red and white lesions of the oral mucosa. Burket's oral medicine diagnosis and treatment. 10 th ed. New Delhi: Harcourt (India) Private Ltd; 2003. p. 85-125.
Epithelial pathology. Oral Pathology and Maxillofacial Pathology. 2 nd ed. Philadelphia: Saunders
  • Bw Neville
  • Dd Damm
  • Cm Allen
  • Je Bouquot
Neville BW, Damm DD, Allen CM, Bouquot JE. Epithelial pathology. Oral Pathology and Maxillofacial Pathology. 2 nd ed. Philadelphia: Saunders; 2004.