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16 PAEDIATRIC Dental Tribune Middle East & Africa Edition | 4/2017 Ulcerative Lichen Planus in childhood. Case study By Dr. Chiyadu Padmini, MDS, Dr. K.Y ellamma Bai, MDS, Dr. Vinil Chait- anya, MDS, Dr. M.Shilpa Reddy, MDS Abstract Lichen planus (LP) is a chronic in- flammatory mucocutaneous condi- tion which is relatively common in adults but rarely affects children. The present study is on report an unusu- al case of ulcerative oral LP involving the dorsum of tongue in 12 year old boy. Patient complained of painful oral lesion on the tongue which was burning in nature and obstructing talking and eating spicy foods. On in- tra oral examination, a white ulcera- tive lesion on the dorsum of tongue was observed. Diagnosis was made based on clinical examination and histopathological features. We in- stituted local treatment and patient responded well to the treatment. Al- though rarely reported in childhood, lichen planus should be considered in a differential diagnosis of hyper- keratotic, reticular and ulcerative le- sions of the oral mucosa in children. Keywords: Oral lichen planus (OLP), childhood, ulcer. Introduction Lichen planus (LP) is an autoim- mune, chronic, inflammatory dis- ease that affects mucosal and cuta- neous tissues. The exact etiology of LP is unknown, but it is believed to result from an abnormal T-cell me- diated immune response in which basal epithelial cells are recognized as foreign because of changes in the antigenicity of their cell surface1. Oral lichen planus (OLP) is a com- mon disease in the middle aged and elderly population, and has a preva- lence of about 0.5% to 2%. In con- trast, oral lichen planus in childhood (OLP) is rare and it was first reported in 1920’s. Oral mucosal involvement in adult itself account for 0.5% to 19% while in children, it is very uncom- mon.2 The oral lesions are more pleomor- phic than those of their cutaneous forms and subtypes are categorized as reticular, papular, plaque-like, atrophic, erosive, and bullous3. The erosive form is extremely rare in children and few reports on this subject have been published in the literature. Herewith, we are presenting a case of 12 year old boy having erosive lichen planus without cutaneous involve- ment, which responded very well to the treatment. This article is also reviews ulcerative oral lichen planus in children and emphasizes its diag- nosis from other oral white and red lesions in children. Case Report A 12 year old boy reported to the Department of Pedodontics and Preventive dentistry, with the chief complaint of ulcer on his dorsum of the tongue which is causing burning sensation on consuming spicy foods from past 1 year. There is no significant Medical his- tory observed. On extra oral exami- nation patient was normal. On intra oral examination, a single irregular red & white ulcerative lesion meas- uring approximately 2.5x1.0 cm in size, with granulation tissue at the centre surrounded by an inflamma- tory red border on the dorsum of the tongue noticed. There was a de- papillation of filiform paillae in and around the lesion (Fig. 1). Oral hy- giene of the patient was good with- out any dental restorations. The differential diagnosis was lichen planus and lichenoid lesions. To ex- clude lichenoid reaction, we inves- tigated his medical status and there was no history of any drug intake. The patient and his parents also de- nied any habits that may potentially cause oral mucosal ulcerations. Histopathological examination showed hyperparakeratosis of strati- fied squamous epithelium and basal cell degeneration with dense band- linked lymphocytic infiltration at the epithelial-connective tissue interface (Fig. 2). Both clinical and histopatho- logical features were consistent with ulcerative oral lichen planus. Specific treatment for ulcerative OLP was topical 0.1% triamcinolone acetonide combined with 1% clotri- mazole 3-5 times per day for the duration of one week. Topical anes- thetic was given for the pain relief. First review of the patient after 15 days showed significant reduction in both symptoms and signs of the oral lesions. (Fig. 3) After 15 days, there is good prognosis in the recovery of ulcerative lichen planus. Erosive oral ulcerative oral lichen planus had completely healed at the end of 30 days. (Fig. 4) Patient was observed on periodic recall follow up. Oral lichen planus in childhood (OLPc) is rare and only a few reports are available in the literature. 4 Oral lichen planus can be divided into a hyperkeratotic (white) variant, com- monly without symptoms, a reticu- lar type with Wickham striae (often symmetrical), papular, and plaque- like types. The atrophic/erythematous (red) variant and the erosive/ulcerative (yellow) variant often have persis- tent symptoms of pain or sting- ing aggravated during talking and eating spicy foods. These variants may occur together in one patient or may transform one to another. The lesions were found more com- monly on the buccal mucosa (often symmetrical), lateral margins of the tongue, gingiva and lips. Whereas cutaneous LP is self-limit- ing, ulcerative OLP is chronic, rarely undergoes spontaneous remission. The family history of LP is more commonly positive in patients with LP in childhood than in adulthood. The exact cause of ulcerative OLP remains unknown, but an immune- mediated (T cell dependant) patho- genesis has been reported. OLP in childhood was first described in 1920 and since then only few arti- cles have been published and most of studies have suggested that child- hood LP is more common in tropical countries like India.5 Sharma and Maheshwari reported 50 children with LP and with concomitant oral lesions in 15 of them and they stated that the oral mucosa seems to be less commonly involved in children with LP than in adults.6 Predisposing conditions such as graft-versus-host disease, active hep- atitis, and hepatitis B immunization are rather frequently mentioned in these reports. Kumar V and Garg BR reported only one case had oral ul- cerative lichen planus out of 25 pa- tients with cutaneous lichen planus.7 The mean interval between vaccina- tion and LP onset was three years, ranging between three months and 11 years. Handa and Sahoo reported 87 patients with childhood LP in India. Seven patients showed in- volvement of the oral mucosa and only one patient had oral ulcerative lichen planus without skin involve- ment.8 A 10 year retrospective study done by Ronald Laeijendecker et al, which comprised of 10,000 patients be- low 18 years, with a boy to girl ratio of 1:1 which have shown only 3 pa- tients (0.03%) were diagnosed with oral lichen palnus.9 A study done in United Kingdom by Alam and Ham- burger in boys aged between 6- 14 years over a period of 20 years have proved only 6 boys been diagnosed with OLP and interestingly among 6 patients, 4 were Asians.10 In 1994, Scully et al reported 3 girls with OLP, one of whom was from Asian origin.11 The difference in the prevalence of OLP in children (0.03%) versus OLP in adults (0.5–2%) is understood by less number of associated systemic diseases in children, auto-immune diseases, infections, drug usage and dental restorations in childhood, this may reduce the risk for developing OLP in childhood.12 Moreo- ver the diagnosis of OLP may be missed due to irregular den- tal check-ups, lack of symptoms and igno- rance by clinicians in diagnosing the condi- tion. The prognosis and the effect of treatment in OLP in children seem to be more favorable than in OLP in adults, which usually per- sists for many years in spite of intensive treatment and thor- ough investigation of associated factors. Malignant transfor- mation of ulcerative OLP in adults is 0.07% to 5%, however malig- nant transformation of OLP in children is not documented in the literature till now.13 Fig. 1: Dorsum of Tongue showing Ulcerative lesion Fig. 2: Photomicrograph (5x magnification) of the Lesion Conclusion Oral lichen planus in childhood is rare, especially erosive form, diag- nosis should be based on children presenting with ulcerative white le- sion in oral cavity. The schedule of follow-up of OLP in children should be 7 days, 15 days and 30 days after diagnosis to assess healing. Patient should be reviwed twice a year for regular follow ups after complete progress of the present condition. However generally, the prognosis of oral lichen planus in childhood seems to be more favorable com- pared to adults. References 1. Patel .S, Yeoman CM, Murphy .R oral lichen planus in childhood. A re- port of 3 cases. International Journal of Pediatric dentistry 2005; 15: 118-22 2. Eisen D. The clinical features, ma- lignant potential and systemic asso- ciations of oral lichen planus: a study of 723 patients. J Am Acad Dermatol 2002; 46:207–214. 3. Aguirre JM, Baga´n JV, Rodriguez C, Jimenez Y, Martı´nez Conde R, Dı´az de Rojas F. Efficacy of mometa- sone furoate microemulsion in the treatment of erosive-ulcerative oral lichen planus: pilot study. J Oral Pathol Med. 2004; 33:381e385. 4. Cottoni F, Ena P, Tedde G, et al. Lichen planus in children: a case re- port. Pediatr Dermatol 1993; 10:132– 135. 5. Kanwar AJ, De D. Lichen planus in childhood: report of 100 cases. Clin Exp Dermatol 2008;33: 423- 7 6. Sharma, R. and Maheshwari, V. (1999) Childhood lichen planus: A report of fifty cases. Pediatric Derma- tology, 16, 345-348. Editorial Note: The full reference list is available from the publisher. Dr. Chiyadu Padmini, MDS Senior Lecturer in the Department of Pedo- dontics and Preven- tive Dentistry, Malla Reddy Institute of Dental Sciences MEDCARE WOMEN & CHILDREN HOSPITAL Beside Manara Services Centre Sheikh Zayed Road Dubai, P.O Box 215565 Dr. K. Yellamma Bai, MDS Principal, Professor and Head of the De- partment of Pedodontics and Preventive dentistry, Malla Reddy College of Dental Sciences for Women Dr. Vinil Chaitanya, MDS Senior lecturer in the Department of Oral Medicine and Radiology, Malla Reddy In- stitute of Dental Sciences Dr. M. Shilpa Reddy, MDS Reader in the Department of Conserva- tive Dentistry and Endodontics, Malla Reddy Institute of Dental Sciences. Fig. 3: Mid treatment (15th day of treat- ment) showing reduction in size & heal- ing of the ulceration Fig. 4: Complete healing of ulcer on Tongue (after 30 days of treatment) Fig. 5: Pre treatment photograph Fig. 6: Mid treatment photograph Fig. 7: Post treatment photograph