PJMS- Volume 2 Number 1: January-June 2012 Case Report Exfoliative cheilitis due to habitual lip biting and excellent response to methotrexate Gupta S1, Pande S2, Borkar M3 Abstract 1 Resident, 2Assistant Professor, 3 Professor, Department of Dermatology, NKPSIMS & RC, Digdoh Hills, Hingna Road, Nagpur-440019 [email protected] Exfoliative cheilitis is a chronic superficial inflammatory disease of the vermilion border of the lips characterized by continuous scaling. In majority of cases, cause of the disease is repetitive lip picking or biting which leads to excessive production and exfoliation of keratin and hence it is also termed as facticious cheilitis. We hereby present a rare case of a young male patient who developed exfoliative cheilitis secondary to continuous lip picking thereby resulting in severe cosmetic disfigurement. Despite intensive counseling and anxiolytic therapy, the condition was unremitting. Hence weekly methotrexate therapy (7.5mg/week) was initiated with excellent response at the end of one month. Introduction Figure 2: Exfoliative cheilitis is a rare disease which affects the vermilion border of the lips. It is characterized by unremitting production and desquamation of thick scales of keratin (1,2). Crusts may be attributed to self induced trauma such as repetitive biting, picking or licking of the lips (3). Underlying stress or psychiatric conditions may cause or exacerbate exfoliative cheilitis which regress with psychotherapy and anxiolytic-antidepressant treatment (1,3). This condition is disabling as it causes cosmetic disfigurement and also affects daily activities such as chewing and speaking (4). The lack of specific treatment makes exfoliative cheilitis a chronic disease. We present this report because of the rarity of the disease and to demonstrate excellent results to methotrexate. Marked hyperkeratosis with lymphocytic infiltration examination, there were keratotic, painless, firm yellowish white crusts over mucosa of the lower lip. Scaling and erythema was noted over the upper lip. On removal of the crusts, the underlying mucosa appeared erythematous. Oral cavity, teeth and rest of the skin was normal. There was no regional lymphadenopathy. Systemic examination revealed no abnormality. However, on psychiatric evaluation, patient admitted of repetitive lip picking. He was depressed due to severe cosmetic disfigurement of lips. Routine blood investigations were all within normal limits. An incisional biopsy that included the normal mucosa and skin was obtained. Histopathology revealed marked hyperkeratosis, parakeratosis and minimal superficial perivascular lymphoyctic infiltrate (Figure 2). No cellular atypia was observed. Based on clinical examination and histopathologic findings, patient was diagnosed as exfoliative cheilitis. Case Report A 23 year-old male presented to the Dermatology department with chief complaints of yellowish white lesions over the lower lip (Figure 1). Lesions were first noticed by the patient 9 years back and gradually increased in size to involve both lower lip and upper lip. There was history of biting and picking of the lip. Patient had received multiple treatments in the past with partial or no relief. Lesions were also refractory to topical corticosteroids but minimal improvement was noted following intralesional corticosteroids. General examination was normal. On cutaneous Treatment was started on weekly methotrexate (7.5 mg)along with folic acid 5 mg OD. After a month, significant improvement of 50-75% was noted (Figure 3). The patient is being maintained on the same therapy and is being followed up to look for remission or relapse. Discussion Exfoliative cheilitis is a rare disease of the lip characterized by visible desquamation of the lips. Although Figure 1: Prominent scaling of lower lip on the erythematous base 37 PJMS- Volume 2 Number 1: January-June 2012 Case Report Exfoliative cheilitis may resolve spontaneously, but if persistent, it is usually refractory to treatment and difficult to manage(6). Avoidance of lip sucking or biting and use of lip moisturizers has been the mainstay of therapy. Psychiatric evaluation is essential for diagnosing any emotional stress that can exacerbate lesions (9). Successful management of such patients as self-inflicted disorders of compulsivity-impulsivity spectrum mostly benefit from selective-serotonin reuptake inhibitors (7). However these agents, combined with intensive counseling and psychotherapy have not yielded desired or satisfactory results. Therefore, various therapeutic modalities like cryosurgery, topical corticosteroids and keratolytic agents have been tried in the past with variable outcomes. All of them are aimed at reducing excessive keratin and provides temporary and symptomatic relief. As our patient had severe inconvenience and cosmetic disfigurement due to the disease, we tried weekly methotrexate therapy to reduce excessive epidermal proliferation. In our patient, significant improvement in scaling and erythema was seen with methotrexate therapy without any recurrence till date. Figure 3: Reduction of scaling following methotrexate therapy definite etiology is unknown, many believe it to be secondary to compulsive or impulsive picking of the lips. As the disease is self induced in majority of cases, it is also termed as facticious cheilitis. Factitious illness behavior can be a maladaptive way of coping with stress and does not necessarily imply an ongoing factitious disorder. The disease is commonly observed in young females but can be seen in any age group and race(1,5,6). In our patient, the probable cause of the disease was habitual picking and biting of the lips. This case highlights the fact that exfoliative cheilitis is secondary to compulsive lip licking and methotrexate can be used as non-steroidal therapeutic option in severely affected patients. Atopy, actinic damage, cheilitis granulomatosa, plasma cell cheilitis, contact dermatitis, discoid lupus erythematosus and neoplasia should be considered in the differential diagnosis of crusted and ulcerated lesions of the lip (7,8). Thorough clinical history, basic laboratory tests and histopathologic evaluation are required to exclude other diseases. References 1) 2) Histologically, exfoliative cheilitis is characterized by marked hyperkeratosis and parakeratosis without significant inflammation. This could be reactive hyperplasia of the epidermis due to repetitive trauma in the form of regular picking of the lips. Trauma may induce epidermal proliferation resulting in parakeratosis and hyperkeratosis. The chances that any other factors like contact dermatitis, infectious agents, neoplasia or autoimmune disease causing excessive epidermal proliferation appear unlikely. Koilocytes or atypical cells has been consistently absent in histopathology of exfoliative cheilitis. Histopathology also helps in differentiation of this entity from discoid lupus erythmetaosus, lichenoid dermatoses, plasma cell cheiltis, cheilitis glandularis, actinic cheilitis or neoplasia (7,8). 3) 4) 5) 6) 7) 8) 9) 38 Rogers R S, 3rd, Bekic M. Diseases of the lips. SeminCutan Med Surg. 1997;16:328–336. Daley T D, Gupta A K. Exfoliative cheilitis. J Oral Pathol Med. 1995;24:177–179. Crotty C P, Dicken C H. Factitious lip crusting. Arch Dermatol. 1981;117:338–340. Mani S A, Shareef B T, Exfoliative cheilitis: Report of a case. J Can Dent Assoc. 2007 Sep;73(7):629-32 Calobrisi S D, Baselga E, Miller E S, Esterly N B. 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