Case Report

Childhood Oral Lichen Planus: Report of Two Cases

Sageena George1, Sheen Ann John2, S Anandaraj3, Jyoti Sumi Issac4, Anoop Harris5, J Reshmi6

1
Professor, Department of Pedodontics and Preventive Dentistry, PMS College of Dental Science and Research, Trivandrum, India
2
Reader, Department of Pedodontics and Preventive Dentistry, PMS College of Dental Science and Research, Trivandrum, India
3
Professor, Department of Pedodontics and Preventive Dentistry, PMS College of Dental Science and Research, Trivandrum, India
4
Reader, Department of Pedodontics and Preventive Dentistry, PMS College of Dental Science and Research, Trivandrum, India
5
Senior Lecturer, Department of Pedodontics and Preventive Dentistry, PMS College of Dental Science and Research, Trivandrum, India
6
Postgraduate Student, Department of Pedodontics and Preventive Dentistry, PMS College of Dental Science and Research, Trivandrum, India

Abstract
Corresponding
Lichen planus (LP) is a chronic mucocutaneous disease widely recognized in adults, but
author:
S. Ann John, Department of Pe- uncommon in children. The purpose of this paper is to report two cases of LP in children.
dodontics and Preventive Den- The diagnosis was made based on clinical and histopathological findings. The treatment
tistry, PMS College of Dental
Science and Research, Trivan-
consisted of antifungal and multivitamin therapy. Regression of lesions was observed. The
drum, India patients are still under follow-up. Although LP is uncommon in children, it is necessary to
have adequate knowledge about this condition for proper diagnosis and treatment.
[email protected]
Key words: Childhood; Lichen Planus; Vaccination
Received: 7 December 2014 Journal of Dentistry, Tehran University of Medical Sciences, Tehran, Iran (2015; Vol. 12, No. 5)
Accepted: 26 March 2015

INTRODUCTION precipitating factors such as stress and LP re-

Lichen Planus is an inflammatory mucocutane- lated infections in children [7,8]. Lack of
ous disease; it is a very common disease in awareness among patients and parents may also
adults but uncommon in children [1,2]. It can be a contributing factor. The purpose of this pa-
affect the skin, scalp, nails and mucous mem- per is to report two cases of childhood OLP and
branes [3]. In contrast to cutaneous LP, oral LP review the literature in this regard.
(OLP) demonstrates clinical variability. An au-
toimmune basis has been proposed; however, CASE REPORT
there is evidence suggesting that LP is not a true Case 1: An 8 year-old male patient was re-
autoimmune disease but rather a chronic, cell- ferred to the Department of Pedodontics, PMS
mediated immune disorder, involving activated College of Dental Science and Research with
lymphocytes and up-regulating cytokine pro- the complaint of pain and white discoloration
duction [1,4]. under the tongue.
Several retrospective reviews have estimated The patient had difficulty tolerating spicy food.
that only 1% -16% of LP patients are younger The medical history revealed that the boy had
than 15 years [5,6]. Factors responsible for the been vaccinated against Japanese encephalitis
rarity of juvenile OLP include a low incidence one year earlier as part of school immunization
of autoimmune diseases, systemic diseases, program.

www.jdt.tums.ac.ir May 2015; Vol. 12, No. 5 374

George et. al Childhood Oral Lichen Planus: Report of Two Cases

Fig. 1. White interlacing striae on the left buccal mucosa Fig. 2. Striae on the upper labial mucosa (case 1)

Almost 10 months after this, fluid-filled blisters Case 2: An 8 year-old male patient was re-
appeared on both feet and ruptured after 2-3 ferred from a local clinic with burning sensa-
days. tion while eating spicy food. There was no rel-
These were associated with severe itching for evant medical history. They gave a history of
which he had taken Ayurvedic medicine. burning sensation since three months earlier af-
On examination, the patient had multiple cari- ter which a local dentist replaced an amalgam
ous teeth. Interlacing white striae were ob- restoration with glass ionomer in the left poste-
served on the buccal mucosa bilaterally (Fig. rior mandible. Due to no relief, the patient was
1), on the ventral tongue mucosa and also on referred to a hospital. On examination, the pa-
the labial mucosa (Fig. 2). tient had a glass ionomer restoration on tooth
On palpation, these lesions were non-tender #75, and all other teeth were caries-free. White
and non-scrapable and the buccal mucosae ap- interlacing striae were noted on the left buccal
peared rough and corrugated. Differential diag- mucosa (unilaterally) (Fig. 4). Peripheral to the
noses included OLP, lichenoid reaction and lesion, there was evidence of pigmentation. On
leukoplakia and a provisional diagnosis of LP palpation, the lesion was non-scrapable. Differ-
was made by correlating the clinical appear- ential diagnoses of lichenoid reaction and LP
ance with the age of the patient. The provisional were considered. Incisional biopsy (Fig. 5) re-
diagnosis was confirmed by histopathological vealed saw-toothed epithelial rete pegs, basal
examination following incisional biopsy from cell degeneration with subepithelial lympho-
the left buccal mucosa. The biopsy specimen cytic infiltrates and melanin incontinence,
showed typical features of LP including the which confirmed the diagnosis of LP. Routine
saw-toothed rete pegs, hyperkeratosis and lym- hematological investigations revealed nothing
phocytic infiltrate (Fig. 3). relevant. After the biopsy, the lesion and the
As the lesions were symptomatic, topical anti- symptoms subsided; thus, no treatment was
fungal treatment (clotrimazole mouth paint warranted. The patient is under follow up.
twice daily) was prescribed for the first two
weeks. DISCUSSION
On the recall session, the burning sensation had Lichen planus is a chronic inflammatory muco-
reduced considerably and the clinical appear- cutaneous disease most frequently seen in the
ance of the lesions had improved with regard to middle aged and the elderly populations with a
severity. female to male ratio of approximately 2:1 [2].

www.jdt.tums.ac.ir May 2015; Vol. 12, No. 5

375
Journal of Dentistry, Tehran University of Medical Sciences George et. al

Fig. 3. Pictomicrograph of case 1 showing saw-toothed rete pegs and lymphocytic infiltration

The first case of LP in a pediatric patient was Our first case gave a positive medical history of
reported in 1920 [3]. Childhood LP is common vaccination against Japanese encephalitis
in the tropics, and children of Asian origin may within the previous year. But this association
be more prone to this condition [1,2,5]. This cannot be confirmed conclusively because
possibly indicates that a specific genetic predis- there is no available literature regarding devel-
position (HLA-dependent) in the Asian race, in opment of LP following this particular vaccina-
spite of a negative family history, may be in- tion. Another possible etiology is the Koebner
volved in the pathogenesis of OLP [2,7]. The phenomenon. This agrees with Nnoruka, who
two cases reported here had Asian origin with reported a relationship between this phenome-
no remarkable family history. Another unusual non and the occurrence of LP [9]. In our second
finding in these cases was that both were males. case, the etiology could be the amalgam resto-
Similar cases have been reported earlier with ration but persistence of symptoms even after
predominance in Asian males than Caucasian replacement of amalgam with glass ionomer
males [2]. cement led to the diagnosis of idiopathic LP.
The etiology of LP remains obscure, but re- The clinical presentation of LP is variable. In
cently pathogenic mechanisms with an im- many cases, the onset is insidious and patients
mune-mediated basis have been proposed [7]. are unaware of this condition. Some patients re-
Causes such as allergy to dental restorative ma- port roughness of the lining of the mouth, sen-
terials, local trauma (Koebner phenomenon) [9] sitivity of the oral mucosa to hot or spicy foods,
and several infections (plaque causing microor- presence of red or white patches or oral ulcera-
ganisms)[7] have also been reported. Also, LP tions [11].
has been reported as a complication of hepatitis Mucosal lesions frequently have a bilateral,
B vaccination in both children and adults symmetrical distribution [12]. In our first case,
[1,10]. Reactions to measles-mumps-rubella symptomatic LP lesions were present bilater-
(MMR) and diphtheria-tetanus pertussis-polio ally. But in our second case, unilateral presence
(DTaP-IPV) vaccinations can also be related to of the lesion led us to a differential diagnosis of
this disease [3]. Moreover, genetic factors, lichenoid reaction. Lichenoid reactions have
stress and lifestyle can also play a role. the same clinical features as LP.

www.jdt.tums.ac.ir May 2015; Vol. 12, No. 5 376

George et. al Childhood Oral Lichen Planus: Report of Two Cases

Fig. 4. Striae on the left buccal mucosa (case 2) Fig. 5. Pictomicrograph of case 2 showing saw-toothed
rete pegs and lymphocytic infiltration

The only difference is in the site of involvement CONCLUSION

like the palate, which is atypical of LP. In most Although OLP is considered rare in children,
cases, the cause for lichenoid reactions cannot presence of a non-scrapable white lesion should
be identified; hence the diagnosis by exclusion alert the clinicians to consider LP in the list of
is idiopathic LP. Although the differential diag- differential diagnoses. Timely diagnosis with
noses were considered, histopathological fea- proper treatment oriented towards the etiologi-
tures confirmed the diagnosis of LP in both cal factor can prevent further complications es-
cases. Use of topical corticosteroids is the most pecially in young children.
widely accepted treatment for OLP because it
reduces patients pain and inflammation. Sev- REFERENCES
eral therapies, including intra-lesional injec- 1- Scully C, Beyli M, Ferreiro MC, Ficarra G,
tion, retinoids, dapsone, tacrolimus, and ultra- Gill Y, Griffiths M, et al. Update on oral lichen
violet light have been tested with varying re- planus: etiopathogensis and management. Crit
sults [2,3]. Surgical excision, cryosurgery and Rev Oral Biol Med. 1998;9(1):86-122.
laser therapy have also been recommended 2- Alam F, Hamburger J. Oral mucosal lichen
[13]. Candida albicans is present in about 37% planus in children. Int J Paediatr Dent. 2001
of OLP lesions. Clinical improvement with re- May; 11(3):209-14.
lief of symptoms has been reported following 3- De Moraes PC, Teixeira RG, Tacchelli DP,
the use of amphotericin-B, nystatin and azole Bnecker M, Junqueira JL, Oliveira LB. Atyp-
antifungals. ical case of oral lichen planus in a pediatric pa-
Miconazole gel is useful as an adjunct to topical tient: Clinical presentation and management.
steroids [13]. In our first case, since the patient Pediatr Dent. 2011 Sep-Oct;33(5):445-7.
complained of severe burning sensation, imme- 4- Eisen D, Carrozzo M, Bagan Sebastian JV,
diate treatment with topical antifungals was Thongprasom K. Number V Oral lichen planus:
done and there was marked reduction in the clinical features and management. Oral Dis.
symptoms. While in our second case, since the 2005 Nov;11(6):338-49.
lesion subsided spontaneously after the inci- 5- Kumar V, Garg BR, Baruah MC, Vasireddi
sional biopsy, no treatment was warranted. SS. Childhood lichen planus (LP). J Dermatol.
There are a number of studies on OLP with re- 1993 Mar;20(3):175-7.
gard to its malignant transformation in the past 6- Luis-Montoya P, Domnguez-Soto L, Vega-
few decades [14]. However, there is considera- Memije E. Lichen planus in 24 children with
ble controversy regarding the malignant poten- review of the literature. Pediatr Dermatol. 2005
tial of OLP. Jul-Aug;22(4):295-8.

www.jdt.tums.ac.ir May 2015; Vol. 12, No. 5

377
Journal of Dentistry, Tehran University of Medical Sciences George et. al

7- Laeijendecker R, Van JT, Tank B, Oranje (2):89-106.

AP, Neumann HA. Oral lichen planus in child- 12- Boorghani M, Gholizadeh N, Taghavi Ze-
hood. Pediatr Dermatol. 2005 Jul-Aug;22(4): nouz A, Vatankhah M, Mehdipour M. Oral li-
299-304. chen planus: clinical features, etiology, treat-
8- Eisen D. The clinical features, malignant po- ment and management; a review of literature. J
tential, and systemic associations of oral lichen Dent Res Dent Clin Dent Prospects. 2010 Win-
planus: A study of 723 patients. J Am Acad ter;4(1):3-9.
Dermatol. 2002 Feb;46(2):207-14. 13- Lodi G, Scully C, Carrozzo M, Griffiths M,
9- Nnoruka EN. Lichen planus in African chil- Sugerman PB, Thongprasom K. Current con-
dren: A study of 13 patients. Pediatr Dermatol. troversies in oral lichen planus: report of an in-
2007 Sep-Oct;24(5):495-8. ternational consensus meeting. Part 2. Clinical
10- Woo VL, Manchanda-Gera A, Park DS, management and malignant transformation.
Yoon AJ, Zegarelli DJ. Juvenile oral lichen Oral Surg Oral Med Oral Pathol Oral Radiol
planus: a report of 2 cases. Pediatr Dent. 2007 Endod. 2005 Aug;100(2):164-78.
Nov-Dec;29(6):525-30. 14- Silverman S Jr, Gorsky M, Lozada NF. A
11- Ismail SB, Kumar SK, Zain RB. Oral li- prospective follow up of 570 patients with oral
chen planus and lichenoid reactions: Etiopath- lichen planus: persistence, remission and ma-
ogenesis, diagnosis, management and malig- lignant association. Oral Surg Oral Med Oral
nant transformation. J Oral Sci. 2007 Jun;49 Pathol. 1985 Jul;60(1):30-4.

www.jdt.tums.ac.ir May 2015; Vol. 12, No. 5 378