Oral Lichen Planus in Childhood associated with Cutaneous ... · Oral Lichen Planus in Childhood Associated with Cutaneous Lichen Nitidus Phulambrikar, et al. 17 International Journal

International Journal of Advanced Health Sciences • Vol 1 Issue 9 • January 2015 16

Oral Lichen Planus in Childhood associated with Cutaneous Lichen Nitidus: Case ReportTushar Phulambrikar, Nikita Goyal, Manasi Kode, Shali P Magar1Professor & Head, Department of Oral Medicine & Radiology, Sri Aurobindo College of Dentistry & PG Institute, Indore, Madhya Pradesh, India, 2Post Graduate Student, Department of Oral Medicine & Radiology, Sri Aurobindo College of Dentistry & PG Institute, Indore, Madhya Pradesh, India, 3Associate Professor, Department of Oral Medicine & Radiology, Sri Aurobindo College of Dentistry & PG Institute, Indore, Madhya Pradesh, India, 4Reader, Department of Oral Medicine & Radiology, Sri Aurobindo College of Dentistry & PG Institute, Indore, Madhya Pradesh, India

thyroiditis, myasthenia gravis, alopecia areata, thymoma, autoimmune polyendocrinopathy.7-12

In addition to that, several studies have found an association of lichen planus with atopic dermatitis.13,14 Although the association of lichen planus and lichen nitidus is controversial, however it has been documented in the literature. In this case report, we are presenting a case of OLP in childhood with associated lichen nitidus.

CASE REPORT

A 14-year-old boy was referred to Department of Oral Medicine, and Radiology with the complaint of burning sensation in the mouth (Figure 1). He reported that burning sensation had been present for preceding 1-year, which aggravates on consuming spicy food. Because of the disability, he had not been to the school for many days. His family history and medical history were unremarkable at initial presentation. Routine blood investigation was normal. General examination did not reveal any findings.

Skin examination showed multiple tiny papules which were of skin color, shiny, non-pruritic and measuring about 0.3-0.7 mm in dimension present on extensor

INTRODUCTION

Oral lichen planus (OLP) is a common chronic immunologic inflammatory mucocutaneous disorder that varies in appearance from keratotic (reticular or plaque like) to erythematous and ulcerative forms.1

OLP is a common disease in middle aged and elderly population and has a prevalence of about 0.5-2%. OLP is rare in children with few reports available in the literature.2

The etiology of lichen planus remains uncertain, but many factors have been implicated. Such factors include the genetic predisposition, infective agents, systemic diseases, graft versus host disease, drug reactions, and hypersensitivity to dental materials, vitamin deficiencies,3 and psychological factors.4,5

Baccaglini et al. 2013 reports indicate that hepatitis C virus is associated with lichen planus and might be involved in pathogenesis of OLP.6 Lichen planus has been associated with several autoimmune diseases, including lupus erythematosus, pemphigus, Sjogren syndrome, autoimmune liver disease, autoimmune

Corresponding Author: Nikita Goyal, Department of Oral Medicine & Radiology, Sri Aurobindo College of Dentistry & PG Institute, Indore - 452 001, Madhya Pradesh, India. Phone: +91-9926517414, E-mail: [email protected]

ABSTRACT

Lichen planus is relatively common mucocutaneous disease of unknown etiology. However it is considered to be immune mediated entity. Lichen planus characteristics involving skin and oral mucosa have been well-described in the literature all over the world. The mean age of occurrence is in fourth to fifth decade considering it to be rare in children. Herewith A case report of oral lichen planus (OLP) with cutaneous lichen nitidus in a 14-year-old boy is presented. Only one case of OLP and cutaneous lichen nitidus has been reported yet. However, lichen planus in association with lichen nitidus has been documented. We had searched the available literature on OLP in children from 1970 to 2014. Analysis of the data showed the increase cases of OLP in childhood in particular geographic regions suggesting the concomitant role of environmental factors as etiology.

Keywords: Childhood lichen planus, Lichen nitidus, Oral lichen planus

Case Report

Oral Lichen Planus in Childhood Associated with Cutaneous Lichen Nitidus Phulambrikar, et al.

17 International Journal of Advanced Health Sciences • Vol 1 Issue 9 • January 2015

surface of elbow of left hand and on index finger of left hand (Figures 2 and 3). Intraoral examination revealed fine white radiating striae was present on lower labial mucosa, bilateral buccal mucosa, floor of mouth, and attach gingiva bilaterally, few erythematous

macules were present on right and left buccal mucosa (Figures 4-6). His oral hygiene was good without any dental restoration. Diagnosis of OLP and cutaneous lichen nitidus was made.

Histopathological examination of the oral lesion showed parakeratotic and focally atropic stratified squamous

Figure 1: Extraoral picture of 14-year-old boy

Figure 3: Multiple, tiny, skin color, shiny, papules present on index finger of left hand

Figure 2: Multiple, tiny, skin color, shiny papules present on extensor surface of elbow of left hand

Figure 4: Fine, faint, white radiating striae on left buccal mucosa

Figure 5: Fine, faint, white radiationg striae with few erythematous patches on right buccal mucosa

Figure 6: Fine faint white radiating striae extending from gingiva to buccal sulcus

Phulambrikar, et al. Oral Lichen Planus in Childhood Associated with Cutaneous Lichen Nitidus


epithelium and focal areas of basal cells liquefaction degeneration and increase intraepithelial lymphocytes. There was dense juxtaepithelial band of inflammatory cells the connective tissue shows dense collagen bundles, dilated blood vessels, muscle tissue, adipocytes. Both clinical and histopathological features were consistent with lichen planus (Figure 7).

As the patient did not respond to topical steroids, the patient was treated with a tablet betnesol forte 0.5 mg, tacrolimus ointment and isotretinoin 0.5 mg was started. Patient was followed up periodically every 15 days on commencement of treatment then after 2 months. Marked clinical Improvement followed after 6 weeks of treatment (Figures 8-11).

DISCUSSION

Lichen planus was first described in the literature by Erasmus Wilson in 1869 as predominantly a disease of middle-aged or older. OLP has been described in children in 1920s.7 The literature available

reporting the occurrences of lichen planus in pediatric age is low, and that of oral involvement is extremely rare.

Woo et al. 2007 did the literature review on childhood OLP from 1990 to 2005 and found the slight male predilection and common age of occurrence were 11 and 15 years with no ethnic predilection. Buccal mucosa was

Figure 7: Histopathologic picture

Figure 8: Left buccal mucosa

Figure 9: Right buccal mucosa

Figure 10: Lower right gingiva and buccal sulcus

Figure 11: Extensor surface of left hand elbow after 6 weeks of treatment



the commonly affected site, and most patients were with reticular pattern.15

We have further reviewed literature from 2007 to 2014 using the same criterias as used by Woo et al. and descriptive statistical analysis was performed.

We found an almost equal predilection for male (52.2%) and female (47.8%) unlike Woo et al. review who had slight male predilection. This is in contrast with lichen planus occurring in elders who have female predilection. Mean age of occurrence was 10.39 years with common age group ranging from 7-11 years. There was racial predilection, majority were of Asian origin unlike Woo et al. which showed no racial predilection. Buccal mucosa was the common site, followed by tongue, and reticular pattern was common.

Some of our findings were consistent with Chatterjee et al. 2012 who have studied 22 OLP cases, like equal predilection for male and females, buccal mucosa as common site, however the mean age reported was 15.8 years and most common form was erosive6 which is in contrast with Woo et al. 2007, Walton et al. 2010 and our study.

Majority of the case reports and studies are in Asian patients suggesting the geographic predilection. It may be due to environmental factors and/or genetic influence disease evolution15 (Table 1).

Balasubramaniam et al. 2008 studied lichen planus from outside the Indian subcontinent and found that of their 26 patients, 21 (80.8%) were from the Indian subcontinent but only 28% of the city’s general child population was from the region.17 However, Walton et al. 2010 found statistically significant greater number of African Americans with childhood lichen planus compared with the control population.18

OLP is a disease of older age as many illnesses and conditions are associated with lichen planus occur in older patients.15

Therefore, for explaining the occurrence of lichen planus in pediatric age group, various factors have been determined which includes previous hepatitis B vaccination, liver diseases such as chronic active hepatitis and genetic predisposition as in familial lichen planus.15

Woo et al. 2007 reported two cases of OLP in childhood, out of which one had a history of hepatitis B vaccination.15 Kanwar et al. 2010, studied on 100 cases of childhood lichen planus and found that 16 had been vaccinated against hepatitis B.19 Familial pattern has also been reported.20,21

However, in the current case report neither familial pattern nor hepatits B vaccination was documented.

Moreover, recently published study had also not found the association of hepatitis B and C with OLP. Chatterjee et al. 2012 conducted a retrospective study for 13 years and had not found positive serology for hepatitis B and C.16 Kumar et al. 2013 studied on correlation of hepatitis virus (B and C) and OLP and had found no serology positive concluding that other factors could be there in underlying disease.22

Childhood lichen planus commonly occurs on the skin, and oral involvement is rare.

Kanwar et al. 1991 presented report of seventeen patients involving the legs, arms, trunk, neck, nails, scalp, and face. Oral involvement was seen in one patient.23 Kumar 1993 et al. reported 1 out of 25 (4%) patient with OLP.24 Sharma and Maheswari 1999 reported OLP in 15 of 50 (30%) children.14 Nanda et al. 2001 reported 9 of 23 (39%) children with oral lesions.25 Handa and Sahoo 2002 reported 87 patients with childhood lichen planus in India 8 (9%) patients showed involvement of oral mucosa.26 Montoya et al. 2005 reported 1 of 16 (6%) children with oral lesions.13 Laeijendecker et al. 2005 conducted a 10 years retrospective study comprised of 10,000 patients below 18 years, showed the prevalence of 0.03% (3 patients) with oral involvement.27 Recently, two large studies have been published reporting the incidence of OLP in childhood as 17% and 18% out of 100 and 316 children respectively.19,28

There is greater variability in prevalence of OLP in childhood ranging from 0.03% to 39% maximum depending upon the region involved. This strongly indicates the geographic or racial predilection suggesting the genetic influence.

In addition, lichen planus has been found to be in association with other systemic diseases. In our case, we found it to be associated with cutaneous lichen nitidus. To our best knowledge, only single case report has been found which showed a correlation of lichen nitidus and OLP.29

CONCLUSION

Lichen planus in childhood is uncommon, and the oral involvement is extremely rare. However, there is evidence of increased documentation of case reports and studies in recent years suggesting increase in the number of cases of childhood OLP indicating the rise of environmental factors.

Phulambrikar, et al. Oral Lichen Planus in Childhood Associated with Cutaneous Lichen Nitidus


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Table 1: Summary of juvenile oral lichen planus (1990‑2014)No. of cases

Age/sex

Ethnicity Site Skin involvement

Clinical form

Case reportsKhandelwal et al. 201330 1 10/f Asian NS NS ReticularMoger et al. 201331 1 7/f Asian BM Yes ErosivePadmini et al. 20132 1 12/f Asian Tongue No UlcerativeSanjaya et al. 201132 1 9/f Asian BM, tongue, hard palate, lips Yes ReticularDemoraes et al. 201133 1 7/f Caucasian Lip No ReticularAnuradha et al. 201134 3 12/f Asian BM No Reticular

9/f Asian BM No Reticular7/f Asian BM, lip Yes Papular

Gunashekhar et al. 201035 1 7/m Asian BM, FOM, tongue No ReticularDas et al. 200936 1 12/f Asian BM No ReticularWoo et al. 200715 2 9/f Caucasian Tongue No Erosive

11/f Caucasian Tongue, BM No ReticularLaeijendecker et al. 200527 3 11/f Asian BM No Reticular

14/f Caucasian BM, tongue No Reticular16/m Asian BM No Erosive

Patel et al. 200537 2 6/m Caucasian Tongue No Erosive15/f Caucasian Tongue, FOM No Plaque

Singhal 200521# 1 11/m NS Tongue No AtrophicSandhu et al. 200320 1 12/f NS NS No ReticularAlam and Hamburger 20018 6 6/m Asian BM Tongue No Reticular, Erosive

7/m Asian BM Atrophic8/m Caucasian Gingiva Reticular11/m Caucasian BM, Tongue Reticular, Papular14/m Asian BM, Tongue Reticular, Plaque14/m Asian BM Atrophic

Scully et al. 19947 2 10/f Caucasian FOM Erosive11/f Caucasian Tongue Erosive

Review studiesPandhi et al. 201428 57/316 <14a Asian NS Yes NSZheng-Yu Shen et al. 201238 5/518 <20 Asian NS NS NSChattergee et al. 201216 22 <18b

M: f-1:1Asian BM>tongue Yesc Erosive

Walton et al. 201018# 8/36 <18d

M: f-2:1African american and other races

BM Yes Reticular>erosive

Xue et al. 200539 4/674 10-13 NS NS NS Reticular (2), Erosive (2)Eisen 200240 5/723 <15 NS NS Yes Erosive, atrophicHanda et al. 200226 12/87 7-11.5 NS BM (5), Lips (3) Yes ReticularSharma and Maheshwari 199914 15/50 <14 NS BM Yes Reticular (skin)

#Familiar lichen planus, aMean age 10.28 years, bMean age-15.18 years, cTwo patient had skin involvement, dMean age 11.8 years, FOM: Floor of mouth, BM: Buccal mucosa, NS: Not specified



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How to cite this article: Phulambrikar T, Goyal N, Kode M, Magar SP. Oral Lichen Planus in Childhood Associated with Cutaneous Lichen Nitidus: Case Report. Int J Adv Health Sci 2015;1(9):16-21.

Source of Support: Nil, Conflict of Interest: None declared.

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Oral Lichen Planus in Childhood associated with Cutaneous ... · Oral Lichen Planus in Childhood Associated with Cutaneous Lichen Nitidus Phulambrikar, et al. 17 International Journal