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Childhood lichen planus: A review of literature Research area: Oral medicine and pathology a Bassel tarakji, Alaa’ Melhemb Corresponding author: a Head of Department of oral medicine and diagnostic science Alfarabi College of dentistry, Kingdom of Saudi Arabia, Riyadh Fax: 00966-2324580 Email: [email protected] B Lecturer at alfarabi dental school Alfarabi College of dentistry, Kingdom of Saudi Arabia, Riyadh Keywords: Lichen planus, children, premalignant lesion Abstract: Oral lichen planus is a relatively common autoimmune mucocutaneousdisease affecting between 0.5% and 2.2% of the population[1]. Lichen affects the skin, mucous membranes , nails and hair and it was first described and named by the British physician Erasmus Wilson in 1869[2]. Skin lesions in lichen planus usuallyresolve within 1-2 years where in oral lichen planus it persists for 20 years or more. The majority of patients are between 30 and 50 years of age and about 60% are women .Oral lichen planus is also seen in children although rare[1,3,4]. The purpose of this article is to review the literature concerning oral lichen planus in childhood which seems to be a rare entity in this group of patients and its reports are scarce [3, 5, 7-15]. 1 For that purpose a literature search using MEDLINE ,accessed via The National Library of Medicine PubMed interface (http://www.ncbi.nlm.nih.gov/pubmed), searching for articles relating to childhood oral lichen planus in English and using the following in search : (childhood oral lichen planus , juvenile oral lichen plans ,oral lichen planus in pediatric patients , oral lichen planus in children ). These references were obtained and from their bibliographies, secondary references were also identified and acquired. The abstracted literature was reviewed. Most of the studies were case reports or case series, no randomized controlled trials were found and no statistical analysis is presented because the collected data were different and the results cannot be compared.Studies that were reviewed are shown in Table 1. Lichen planus is considered rare in children ( comprise 2-3% of reported cases[15], but it does not appear to be uncommon in India and middle east countries with a study reporting an incidence of 7.5% childhood lichen planus among all registered lichen planus patients in the clinic[2,12,16,17]. Luis Montoya et al [18] in their study registered 24 cases of childhood lichen planus out of 235 lichen planus occurrences (10.2%) over a period of 22 years. It has been hypothized that the rarity of associated autoimmune conditions, exposure to drugs and other triggers that have been known to initiate lichen planus in adults may be responsible for the overall rarity of LP in children [2, 6]. In the majority of childhood lichen planus studies no significant gender predominance was identified[18],however Sharma et al found the male: female ratio as 2:1 in their 50 childhood lichen plans case series [17]. Kumar et al reported that more girls were affected and that was convenient with Montoya et al[18], Handa et al [19]and Sanjaya et al[15]. 2 Etiopathogenesis: Lichen planus is a unique disorder of unknown exact etiology and in most lichen planus cases the precipitating factors are unknown and the disease is idiopathic ,however many factors have been implicated in its uncertain etiology. Such factors include genetic predisposition(More recent studies suggest that at least 50% of the cases reported , had familial histories of lichen planus), infective agents , systemic diseases , graft vs. host disease , drug reactions , hypersensitivity to dental materials and vitamin deficiencies .the association of HBV or HCV infection with lichen planus has been given particular focus [20]. Lichen planus has also been associated with several auto-immune diseases including lupus erythematosus,pemphigus, Sjogren’s syndrome and autoimmune liver disease [5, 6, 16]. For childhood lichen planus no precipitating factors were observed in the study by Kanwar et al[2] as well as by Handa et al[19] ,Sharma et al and Montoya el[18] but a number of childhood LP cases were reported after hepatitis B vaccination and after combined measles-mumps-rubella vaccination[2,16]. The family history of LP is more commonly positive in childhood than in adulthood patients [6, 16]. The pathogenesis of lichen planus is not completely understood but there appear to be a mechanism for activating the regional cellular immune response and another for the T- lymphocyte response that eventuates in the destruction of the deep layered keratinocytes [1]. Clinical features: Lesions occur on both cutaneous and oral surfaces comprise about 40% of all lichen planus cases, lesions on cutaneous surfaces only comprise 35% and lesions on mucosal surfaces only are about 25% [1] 3 The clinical presentation of lichen planus is diverse .The skin lesions present as purple, polygonal, pruritic, flat topped papules. Wickham’s striae are evident on the surface of papule as a reticular network of fine white lines. Oral lesions classically present as a bilaterally symmetrical white network found on the buccal mucosa, tongue, lips and hard palate. Up to six clinical appearances of oral lichen planus have been described [16]. Reticular oral lichen planus is the most common presentation, manifesting as a lacy network of white striations. These lesions are often painless, although patients may complain of a slight roughness or dryness of the affected sites. (Fig.1) Papular oral lichen planus: manifests as small white raised areas approximately 1-2mm in diameter. These again typically arise on the buccal mucosa and dorsum of tongue, although may also present on other mucosal surfaces. Thisvariant may represent an early manifestation of the condition. Plaque-like oral lichen planus: manifests as areas of homogenous whiteness. This typically arises on the buccal mucosa or dorsum of tongue and may be more prevalent amongst those who are smokers. Atrophic oral lichen planus: There are areas of mucosal atrophy occur within the white patches. The clinical picture is one of red and white areas, but not speckled as is seen in chronic hyperplastic candidosis. Patients with this type of disease often complain of oral soreness.(Fig2.) Ulcerative lichen planus frank ulcers exist often within the hyperkeratotic areas. On occasions there may be no white striae apparent, making clinical diagnosis difficult. Patients complain of soreness, particularly with spicy or acidic foods. (Fig3). Bullous lichen planus is a rare presentation manifests as small vesicles or blister (bullae) within the white patches (Fig. 4). This may cause diagnostic confusion with mucous membrane pemphigoid or 4 pemphigus [1, 6, 15, and 25]. These variants may occur together in one patient or may transform from one to another [6]. Oral mucosal involvement in adults itself is rare [15], and in children it seems to be even more rare and very few of the children with cutaneous lesions were reported to have oral lichen planus [21] Laeijendecker et al [6] in a study of 10 years duration (1994-2003) could identify three patients out of 10000 ( with a ratio of .03%) having oral lichen planus , two of them were Asian . In the study of Kumar et al [22] oral mucosal involvement with 20-nail dystrophy was detected in only one patient among 25 children. A study of 87 patients with childhood lichen planus for Handa et al [23] reported one case with oral involvement alone, four cases with concomitant skin and mucosal involvement at the time of presentation and seven patients with mucosal involvement later during the course of the disease. Mucosal involvement was detected in three cases out of 13 children patients in a study of Nnoruka [13] and in only one patient in Luis Montoya’s study [18]. Classic lichen planus was the commonest clinical variant [13, 14, 18], and Kobner phenomenon is considered to be common in children with lichen planus [2]. Many authors contend that the clinical features of oral lichen planus and the location are essentially the same as those in adults; however, generally the prognosis of LP in childhood seems to be more favorable [6]. There have been sporadic reports in the literature that lichen planus may be responsible for the subsequent development of squamous cell carcinoma and the percentage of this transformation was estimated between 0.4% to 2% when lesions persist for more than 5 years [1, 15]. The scarcity of literature in children does not allow for objective data regarding the malignant transformation rate in 5 childhood lichen planus [6, 15].Nevertheless, the schedule of follow-up of oral lichen planus in children should be at least one or two visits per year as long as oral lichen planus in children persists and even more frequently in symptomatic childhood lichen planus, in line with oral lichen planus in adults [6]. Histopathology: In most cases of lichen planus it is not possible to make the correct diagnosis without a biopsy of the perilesional tissue. The typical histological features of striae includes hyperkeratosis or parakeratosis,saw–tooth profile of the rete ridges , liquifactive degeneration of the basal cell layer , compact band like T-cell infiltrate hugging the epithelio-mesenchymal junction and CD8 lymphocytes predominate in relation to the epithelium.(Fig.5) . Treatment: The aims of current oral lichen planus therapy are to eliminatemucosal erythema and ulceration, alleviate symptoms and reduce the risk of oral cancer in oral lichen planus patients. Inthis context, medical treatment is restricted currently to atrophic (erythematous), erosive (ulcerated), bullous (blistering) or symptomatic oral lichen planus lesions [24]. In asymptomatic non ulcerative lichen planus an explanation for the patient about the nature of the condition, including its etiology, clinical features, treatment, follow up and prognosis with an advice on avoidance of risk factors and reassurance are the treatment guidelines [25]. Corticosteroids are the mainstay of OLP therapybecause of their activity in dampening cell mediatedimmune activity and are administered topically, intralesionallyor systemically. The combination of systemicand topical steroid therapy is often very effective.Localized oral lesions are 6 treated with topical ointment, applied two to four times daily after meals. Thestrength and specific preparations used need to be balanced carefully with the individual patient’s needs. Generalized oral lesions are often treated effectively with a steroid mouth rinse twice daily after meals.Intralesionaland perilesional injection of steroids is useful for persistent localized erosive OLP lesions but should be used with due caution. Generalized atrophic or erosive oral lesions that do not respond to topical therapy may be treated with a short course of systemiccorticosteroids [24]. Due to the lack of controlled clinical trials, no consensus exits regarding standardized therapy regimens for LP in children [15]. In the general treatment of LP,oral antihistamines and topical corticosteroids are the main therapeutic agents. [5, 6, 17] Topical tacrolimus was applied as a treatment but several reports have shown the usefulness of it in the oral cavity [20]. A short course of systemic steroids has been used in children to control widespread disease but most protocols using topical andintralesional corticosteroids cause some adrenal suppression and clinicians need to be aware of the precise amounts of these medications being used on a regular or irregular basis[24]; adverse effects must be considered carefully[5,17]. Dapsone, griseofulvin, PUVA (Psoralen and ultraviolet A light photochemotherapy) and narrowband ultraviolet B photo therapy was also found to be safe and effective [5, 14, 17, 21]. There is neither evidence base nor consensus as to the optimumreview interval. An annual review interval of 1 year would seem reasonable formild forms of oral lichen planus, reducing to 6 monthly for more severe forms and 3monthly if dysplasia is present on biopsy[25]. This study has shown that there is a lack ofpublished evidence regarding the management of oral lichen planus at children.There is aconsensus that corticosteroids are the mainstay of oral lichen planus therapybecause of their activity in dampening cell mediatedimmune activity and are 7 administered topically, intralesionallyor systemically, although,there is dividedopinion regarding the most appropriate treatmentfor oral lichen planus at children. The rarity of the disease in children, lack of evidence and poor knowledge at general practitioners to diagnose this disease that might be increased the malignant transformation rates at children versus adults. Randomized controlledclinical trials could be used to address theseissues but are difficult to perform because of likelylow accrual rates and the need for prolongedfollow-up times to assess clinical outcomes. The guideline of the diagnosis and treatment of oral lichen planus at children should be clarified to all general practitioners in the dental clinics to detect oral lichen planus early as much as possible to prevent of the malignant transformation of that disease. Conclusion: It is recommended that although oral lichen planus is considered rare in childhood, the presence of oral lesions should alert GP’s to include childhood lichen planus in their differential diagnosis of other white lesions. A double-blind randomized and controlled studies would be necessary for a safe and effective treatment of childhood lichen planus. References: 1. J. Philip Sapp DDS MS, Lewis Roy Eversole DDS MSD MA, George W. Wysocki DDS PhD Contemporary Oral and Maxillofacial Pathology, 2004; 2e:257-262. 8 2. Kanwar AJ, DeD.Lichenplanus in children. Indian J DermatolVenerolLeprol 2010; 76:366372. 3. Sumairi B. Ismail, Satish K. S. Kumar and Rosnah B. Zain. Oral lichen planus and lichenoid reactions: etiopathogenes diagnosis, management and malignant transformation Journal of Oral Science 2007; 49:89-106 4. Mona Soliman MD, Ahmed EL Kharbotly MD and Ali Saafan Ph.D. Management of oral lichen planus using diode laser (980nm). A clinical study Egyptian Dermatology Online Journal 2005; 1:3. 5. Mihn Sook Jue, M.D., Jung Woo Lee, M.D., JooYeonKo, M.D., KwangYeoll Yeo, M.D., JoungSoo Kim, M.D., HeeJoon Yu, M.D. Childhood Lichen Planus with PalmoplantarInvolvement. Ann Dermatol, 2010.22:51-53. 6. Ronald Laeijendecker M.D., Theodoor Van Joost M.D., Ph.D, Bhupendra Tank Ph.D., Arnold P. Oranje M.D., Ph.D., H. A. Martino Neumann M.D., Ph.D. ,Oral Lichen Planus in Childhood.Pediatric Dermatology,2005.22.299-304 7. De Moraes PC, Teixeira RG, Tacchelli DP, Bönecker M, Junqueira JL, Oliveira LB,Atypical case of oral lichen planus in a pediatric patient: clinical presentation and management.Pediatr Dent,2011,33: 445-7. 8. M GunaShekhar, Reddy Sudhakar, Mohammad Shahul, John Tenny, ManyamRavikanth, N Manikyakumar. Dermatol Online, 2010, 16 (8): 9. 9. Woo VL, Manchanda-Gera A, Park DS, Yoon AJ, Zegarelli DJ. Juvenile oral lichen planus: a report of 2 cases. Pediatr Dent. 2007, 29(6):525-30. 10. S. Patel, c. M. Yeoman, R. Murphy. Oral lichen planus in childhood: a report of three cases. International Journal of Paediatric Dentistry 2005, 15:118–122. 9 11. Alam F, Hamburger J. Oral mucosal lichen planus in children. Int J Paed Dent 2001;11:209– 214. 12. Ch Anuradha, G Sridhar Reddy, S R K Nandan, Shamala Ravi Kumar and B VenkatRamana Reddy N Y State Dent J2011; 77(6):28-30. 13. Edith N Nnoruka. PediatrDermato l2007 24(5):495-8 14. Nanda A, Al-Ajmi HS, Al-Sabah H, Al-Hasawi F, Alsaleh QA, Asad Al-Hamad. PediatrDermatol, 2001; 18:1-4. 15. Sanjaya PR, KaveriHallikerib, Punnya V Angadic. Disseminated form of childhood lichen planus. Eastern Journal of Medicine 2011. 16: 72-75. 16. Usha Mohan Das, Beena JP. Oral Lichen Planus in Children. International Journal of Clinical Pediatric Dentistry, 2009; 2(1):49-51. 17. PýnarÖztas, MeltemÖnder, NilselÝlter, Murat OrhanÖztas. Childhood lichen planus with nail involvement: a case. The Turkish Journal of Pediatrics 2003; 45: 251-253. 18. Pilar Luis-Montoya, M.D., Luciano Dominguez-Soto, M.D., and Elisa Vega-Memije, M.D. Lichen Planus in 24 Children with Review of the Literature. Pediatric Dermatology 2005; 22:295–298. 19. Handa S, Sahoo B. Childhood lichen planus: a study of 87 cases. Int J Dermatol 2002; 41:423–427. 20. Yohei Hamade, Ken Arita, Ellen Toyonaga, Nao Saito, Daisuke Inokuma, KokichiHamasaka and Hiroshi Shimizu.ActaDermVenerol 2012;92:339-409. 21. Pinar Y Başak, Kayhan Başak, Generalized lichen planus in childhood: is dapsone an effective treatment modality? The Turkish journal of pediatrics 2002;44:346-348 10 22. Kumar FV, Garg BR, Baruah MC, et al. Childhood lichen planus. J Dermatol 1993; 20:175– 177. 23. Handa S, Sahoo B. Childhood lichen planus: a study of 87 cases. Int J Dermatol 2002; 41:423–427. 24. PB Sugerman, NW Savage.Oral lichen planus: Causes, diagnosis and managementAustralian Dental Journal 2002; 47 :( 4):290-297. 25. The British Society for Oral Medicine. Guidelines for the Management of Oral Lichen Planus in Secondary Care 2010. 26. Sharma R, Maheshwari V.Childhood lichen planus: a report of fifty cases1999;16(5):345-8 Legends of figures: Fig1: Shows white lesion on the buccal mucosa which represent reticular form of oral lichen planus Fig2: Shows white lesion with areas of red color within it on the lateral surface of the tongue and diagnosed as atrophic oral lichen planus. Fig3: Ulcer exists within a white area on the buccal mucosa and sulcus and diagnosed as ulcerative type of oral lichen planus. Fig4: Bulla within a white patch on the buccal mucosa diagnosed as bullous oral lichen planus Fig5 : Histological features of lichen planus includes hyperkeratosis,saw–tooth profile of the rete ridges , band like T-cell infiltrate hugging the epithelio-mesenchymal junction . 11