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  Table of Contents    
CASE REPORT
Year : 2013  |  Volume : 31  |  Issue : 3  |  Page : 197-200
 

Erosive oral lichen planus with cutaneous involvement in a 7-year-old girl: A rare case report


1 Department of Oral Medicine and Radiology, Hitkarini Dental College and Hospital, Jabalpur, Madhya Pradesh, India
2 Department of Periodontics, Hitkarini Dental College and Hospital, Jabalpur, Madhya Pradesh, India
3 Department of Oral Pathology, Hitkarini Dental College and Hospital, Jabalpur, Madhya Pradesh, India

Date of Web Publication11-Sep-2013

Correspondence Address:
Ganapathi Moger
Department of Oral Medicine and Radiology, Hitkarini Dental College and Hospital, Dumna Road, Jabalpur - 482 005, Madhya Pradesh
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/0970-4388.117972

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   Abstract 

Lichen planus is a chronic autoimmune, mucocutaneous disease, which can affect the oral mucosa, skin, genital mucosa, scalp and nails, and commonly seen in adults. Childhood lichen planus is a rare entity, which is characterized by skin lesions with the oral involvement being extremely uncommon. There are only a few reports on this subject in the literature. Early and correct diagnosis with adequate management is very important to avoid further complications as it is a pre-malignant condition. Herewith, we report a seven-year-old girl having erosive oral lichen planus with cutaneous involvement, which responded well to the treatment. This is to add another case of lichen planus in a child and to emphasize its consideration in the differential diagnosis of oral mucosal red and white lesions in children.


Keywords: Erosive, immunofluorescence, lichen planus, white patches


How to cite this article:
Moger G, Thippanna CK, Kenchappa M, Puttalingaiah VD. Erosive oral lichen planus with cutaneous involvement in a 7-year-old girl: A rare case report. J Indian Soc Pedod Prev Dent 2013;31:197-200

How to cite this URL:
Moger G, Thippanna CK, Kenchappa M, Puttalingaiah VD. Erosive oral lichen planus with cutaneous involvement in a 7-year-old girl: A rare case report. J Indian Soc Pedod Prev Dent [serial online] 2013 [cited 2019 Sep 15];31:197-200. Available from: http://www.jisppd.com/text.asp?2013/31/3/197/117972



   Introduction Top


Lichen planus is a common chronic inflammatory disorder that affects the skin, mucous membranes, nails, and scalp. [1] It was first described and named by British physician Erasmus Wilson in 1869. [2] The etiology of this condition remains obscure, though it appears to be immune-mediated and multifactorial. [3] Prevalence of lichen planus is about 0.5% to 2% with female to male ratio of approximately 2:1 [4],[5] and is common between the ages of 30 to 60 years, while children comprise less than 2% to 3% of the total. [1],[5]

Oral lichen planus in childhood was first described as early as in 1920s. The skin lesions mostly present as purple, scaly, flat-topped polygonal pruritic papules with wickham striae. Oral mucosal involvement in adult itself accounts to only 0.5% to 19% while in children, it is much more uncommon. [1],[5],[6] Oral lesions in children and adults show classic bilaterally symmetrical white network found on buccal mucosa, tongue, lips, and hard palate. [2],[4],[6] Erosive oral lichen planus in children is extremely rare, and only a few reports on this subject have been published in the literature. [4] Oral lichen planus is potentially malignant disorder, particularly erosive and atrophic variants. Hence, early and correct diagnosis with adequate management is very important to avoid further complications. [7],[8]

Herewith, we report a case of seven-year-old girl having erosive oral lichen planus with cutaneous involvement, which responded well to the treatment. This is to add another case of lichen planus in a child and emphasize its consideration in the differential diagnosis of oral red and white lesions, particularly, in children.


   Case Report Top


A seven-year-old girl reported to the Dental College with a complaint of burning sensation in the mouth since 2-3 months. History revealed burning present on both right and left buccal mucosae, particularly, while consuming hot and spicy food. There was intermittent mild to moderate itching on the skin of back and hands since one month. Patient's father had expired six years back due to heart attack and mother expired of cancer five years back. There was neither relevant medical nor drug history.

The cutaneous lesions presented as polygonal, flat-topped, papules, symmetrically distributed over the skin of back and hands. Skin was dry and scaly [Figure 1] and [Figure 2]. Nails, scalp, and genitalia were normal. Intraorally, grayish-white patches with white striae on both right and left buccal mucosa on an erythematous background extending up to the retromolar regions were present [Figure 3] and [Figure 4]. Patches extended about 0.5 cm away from labial commissures to pterygomandibular regions and superioinferiorly from upper to lower buccal vestibules. Lesions were slightly tender, non-scrapable and didn't disappear on stretching. The surface was rough because of multiple papules. Melanin pigmentations were also present along the lesions.
Figure 1: Multiple papules distributed symmetrically all over the skin of hands

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Figure 2: Multiple papules involving skin of the back

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Figure 3: OLP affecting the right buccal mucosa

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Figure 4: OLP involving the left buccal mucos

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Dental hard tissue examination revealed dental caries with 63, 65, 74, and 36. Based on clinical findings and history of the patient, a provisional diagnosis of erosive oral lichen planus with cutaneous involvement was made. Clinical differential diagnosis of lichenoid reaction, contact stomatitis, and erythematous candidiasis was considered.

The incisional biopsy was done, and histopathologic features were significant with atrophic parakeratinized stratified squamous epithelium, saw teeth rete pegs and well-defined bands of chronic inflammatory cells sub-epithelially. Few degenerating keratinocytes interfaced between epithelium and connective tissue forming colloid or civatte bodies [Figure 5]. These features were suggestive of an atrophic oral lichen planus. For further confirmation, direct immunofluorescence (DIF) study was done, which showed deposition of fibrin along the basement membrane zone (BMZ) as irregular strands into superficial lamina propria [Figure 6]. Hence, after obtaining dermatologist's opinion regarding skin lesions, a final diagnosis of atrophic oral lichen planus with cutaneous involvement was made.
Figure 5: Histopathological features suggestive of Oral Lichen Planus

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Figure 6: Microphotograph of DIF shows deposition of fibrin along the BMZ as irregular strands into superficial lamina propri

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Patient was advised to undergo all required dental treatments like oral prophylaxis and restorations. General measures for management of the OLP included meticulous oral hygiene and avoidance of any form of physical injury to oral mucosa. Patient was instructed to have diet rich in fresh vegetables and fruits. Specific treatment for OLP was with topical 0.1% triamcinolone acetonide combined with 1% clotrimazole. Topical anesthetic was given for palliation. Skin lesions also were managed with corticosteroid as per dermatologist's instructions.

First review of the patient after one week showed a significant reduction of about 40-50% in both symptoms and signs of the oral lesions. At second follow-up after 15 days, there was almost 80% improvement in the condition. Erosive oral lesions had completely healed by one month of treatment, leaving asymptomatic reticular areas [Figure 7] and [Figure 8]. Even skin lesions had totally resolved by this time [Figure 9] and [Figure 10]. Patient was kept on periodic follow-up.
Figure 7: Left buccal mucosa healed after treatment

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Figure 8: Right buccal mucosa healed after treatment

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Figure 9: Skin of the hands healed after treatment

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Figure 10: Skin of the back healed after treatment

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   Discussion Top


Oral lichen planus in childhood is exceptionally rare (<2%-3% of the total), and only a few reports are available in the literature. [1],[5],[6] Most of these cases reported in the tropics, mainly from India, with some from the United Kingdom, Italy, Mexico, African America, and Kuwait. This could be due to specific genetic predisposition and environmental factors in the pathogenesis of lichen planus. [1],[4],[6]

The reasons for the rarity of this condition in children is not known, but could be attributed to the relative rarity of the factors known to play an important role in the pathogenesis of lichen planus. [1],[4],[6] Most studies on childhood oral lichen planus have shown female predilection, although in some studies, no significant gender predominance was identified. [2],[4],[6] Our case too occurred in a seven-year-old female child.

Previous cases reported with children having mainly skin and very rarely oral involvement. [1],[5],[6] Our case presented with classical pruritic skin lesions. In contrast, she had erosive oral lichen planus on both right and left buccal mucosa, which were symptomatic. Oral lichen planus typically present as a bilaterally symmetrical white network found on the buccal mucosa, tongue, lips, and hard palate. [1],[6] Occasionally, there may be erosive areas, ulcerations and very rarely, white plaque forms of oral lichen planus present. [1] In some cases, oral lichen planus may be associated with patchy brown inflammatory melanosis in the oral mucosa, although this is uncommon, [5],[6],[8] and our case showed pigmentation. The differential diagnosis of childhood LP may be quite extensive and, of course, depends on the age of the patient, the clinical variants, and severity of the lesions. [1],[4]

The histopathological findings in our case were in accordance with essential features suggestive of OLP. [1],[2],[3],[8] Sumairi B. Ismail et al., in 2007, stated that LP shows a characteristic pattern of fibrin deposition outlining the basement membrane zone and extends irregularly into the superficial lamina propria, described as a shaggy or fibrillar pattern. [3] In some cases, deposition of IgM and less often IgA and IgG were found. [2],[3] In our case also, same characteristic pattern was present along BMZ, but test for IgM, IgA, and IgG were negative. According to the result obtained by the DIF study, we thought immune-mediated onset could be the etiology in our patient. [2]

The OLP patients are at increased risk of oral cancer, although the incidence is fairly low. Most oral malignancies arising in OLP patients are associated with erosive, atrophic, and plaque lesions. [1],[8] The scarcity of literature in children does not allow for objective data regarding the malignant transformation rate in childhood LP. [1],[4] However, this risk may be reduced by the elimination of exogenous carcinogens, effective treatment of atrophic, erosive and plaque OLP, and consumption of nutritious diets including fresh fruits and vegetables. [3],[8]

The aims of current OLP therapy are to eradicate mucosal erythema and ulceration, alleviate symptoms, and reduce the risk for oral cancer. Corticosteroids administered topically, intra-lesionally or systemically, depending on the age and condition of the patient, site, and severity of the lesions is the mainstay of the therapy. [2],[4],[5],[7],[8] Treatment of OLP with cyclosporin, azathioprine, levamisole, griseofulvin, retinoids, hydroxychloroquine sulfate, dapsone, psoralen/UVA, and Ultraviolet-B have been reported in some cases. [3],[7],[8],[9],[10] Our patient responded well to the topical corticosteroid combined with the anti-mycotic drug. Due to the lack of controlled clinical trials, no consensus exists regarding standardized therapy regimens and the prognosis for LP in children. [1],[5],[8] As there is risk of malignant transformation, particularly in erosive and atrophic oral lichen planus, early investigation with proper diagnosis and appropriate management with long-term monitoring of the patient is mandatory.

 
   References Top

1.Sanjaya PR, Hallikeri K, Angadi PV. Disseminated form of childhood lichen planus. Eastern J Med 2011;16:72-5.  Back to cited text no. 1
    
2.Anuradha CH, Chandra Sekar P, Reddy GS, Arvind Baba RS, Kumar KK, Reddy BV. Oral mucosal lichen planus in children - Report of three cases. J Orofacial Sci 2011;3:20-3.  Back to cited text no. 2
    
3.Ismail SB, Kumar SK, Zain RB. Oral lichen planus and lichenoid reactions: Etiopathogenesis, diagnosis, management and malignant transformation. J Oral Sci 2007;49:89-106.  Back to cited text no. 3
[PUBMED]    
4.Laeijendecker R, Van Joost T, Tank B, Oranje AP, Martino Neumann HA. Oral lichen planus in childhood. Pediatr Dermatol 2005;22:299-304.  Back to cited text no. 4
    
5.Oztaº P, Onder M, Ilter N, Oztaº MO. Childhood lichen planus with nail involvement: A case. Turk J Pediatr 2003;45:251-3.  Back to cited text no. 5
    
6.Luis-Montoya P, Domínguez-Soto L, Vega-Memije E. Lichen planus in 24 children with review of the literature. Pediatr Dermatol 2005;22:295-8.  Back to cited text no. 6
    
7.Edwards PC, Kelsch R. Oral lichen planus: Clinical presentation and management. J Can Dent Assoc 2002;68:494-9.  Back to cited text no. 7
[PUBMED]    
8.Sugarman PB, Savage NW. Oral lichen planus: Causes, diagnosis and management. Aust Dent J 2002;47:290-7.  Back to cited text no. 8
    
9.Pavlotsky F, Nathansohn N, Kriger G, Shpiro D, Trau H. Ultraviolet-B treatment for cutaneous lichen planus: Our experience with 50 patients. Photodermatol Photoimmunol Photomed 2008;24:83-6.  Back to cited text no. 9
[PUBMED]    
10.Basak PY, Basak K. Generalized lichen planus in childhood: Is dapsone an effective treatment modality?. Turk J Pediatr 2002;44:346-8.  Back to cited text no. 10
    


    Figures

  [Figure 1], [Figure 2], [Figure 3], [Figure 4], [Figure 5], [Figure 6], [Figure 7], [Figure 8], [Figure 9], [Figure 10]



 

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