Factitious cheilitis presenting as exfoliative cheilitis: A rare case report

INTRODUCTION Exfoliative cheilitis is a rare skin condition that involves the vermillion region of lips. Among the reported cases, its prevalence is more in young individuals below the age of 30 years with a predilection for females.[1] Common clinical presentations of this condition are erythematous lesions, excessive desquamation of thick keratin scales, fissures, ulcerations, and hemorrhagic crusts of lips. Patients with exfoliative cheilitis usually experience itching, burning sensations, and dryness of lips with difficulty in chewing, smiling, and speaking. Patients also report social impairment as a result of the esthetic problems associated with the condition.[2,3] The exact pathogenesis of exfoliative cheilitis is not known. Although several causative factors like allergies, photosensitivity, and infections are associated with the condition, a factitious etiology is widely advocated. Maladaptive behaviors such as frequent lip licking, lip biting, lip sucking, and mouth breathing are considered to be the major contributing factors to a factitious etiology.[3] While factitious behaviors are consciously produced, the underlying motive behind them is considered to be unconscious. Some of the patients with factitious cheilitis were found to have comorbid personality disorders, affective disorders, anxiety disorders, and obsessive-compulsive disorders. The course of the illness is usually chronic due to a lack of specific treatment and often flares up when stress increases.[3,4] Detailed illness history, thorough clinical examinations, and relevant investigations to exclude other causes of exfoliative cheilitis are required before reaching a diagnosis of factitious cheilitis. This case report aims to describe the presentation of a case of exfoliative cheilitis that was eventually diagnosed as factitious cheilitis and was effectively managed by a novel approach based on behavioral therapy. CASE REPORT A 21-year-old female student presented to the outpatient department of Psychiatry at a tertiary care hospital with the chief complaint of recurrent desquamation and crusting of both her lips for about a year [Image 1]. History of the present illness revealed that it was 1 year before the patient had first experienced itching on her lower lip which she could not resist. Later when she examined her lips in the mirror, she noticed multiple tiny erythematous lesions over the itching area. She had applied lip balm and massaged it vigorously in an attempt to heal the lesions faster. She had repeated this act several times a day for a few days. Instead of healing of the lesions, she had developed crusting over the area. Subsequently, the condition got worsened and she had developed lesions over both her lips with crusting followed by desquamation. She had consulted a dermatologist and was diagnosed with exfoliative cheilitis. She was prescribed tablet methotrexate (15 mg/day), tablet folic acid, vitamin E, and vitamin C for oral intake and petroleum jelly for local application. The patient did not show any improvement in that treatment regimen, and the condition remained the same. Later, she had visited several dermatologists before coming to our department. She had been tried on oral immunosuppressants (cyclosporine), oral retinoids (acitretin), topical calcineurin inhibitors (tacrolimus), topical steroids (mometasone, desomide), and several moisturizing agents, with no results. All her routine investigation reports were normal, and the biopsy report of the lesion showed nonspecific inflammatory changes. The culture report also had a negative impression of fungal growth. After all pathogenic factors were ruled out, the patient was referred to the department of psychiatry, suspecting a factitious etiology.Image 1: Initial presentation of the patient with hyperkeratosis and crusting of both the lipsOn further evaluation, the patient had been self-conscious about her look since her adolescence and would spend more time in bathing and grooming herself. She revealed that 1 year before she got to know about her marriage proposal as discussed in her family, she was more concerned about her looks than before. She used to apply some herbal cosmetics over her face regularly for a few days prior to the development of the lesions over her lips. She had also a habit of frequent lip licking, lip picking, and frequent mirror gazing since her adolescence. On examination, she had multiple crusts over both her lips [Image 1]. When her lips were cleaned with normal saline, the crusts were easily removed with few bleeding points noticeable on lips. There was no atopy or exfoliation at any other part of her body. On mental status examinations, the patient was anxious and reported low self-esteem due to her illness. On psychiatric evaluation, her HAM-A[5] score was 15, suggestive of mild anxiety; the HAM-D[6] score was 10, suggestive of mild depression, whereas on Y-BOCS[7] symptom checklist, no obsessions or compulsive features were found. The patient was finally diagnosed as factitious cheilitis and hospitalized for further evaluation and management. She was started on tablet escitalopram (10 mg/day), tablet vitamin C and E supplements, and tablet clonazepam (0.5 mg) as and when required. After establishing rapport, the patient was psychoeducated regarding her illness. It was found that the patient was very much fond of taking her own picture (selfie) on her smart phone. But due to her lesions, she would feel restless, for which she would frequently lick and pick her lips. Based on Premack principle of reinforcement and Habit reversal training (HRT), behavioral therapy was individualized for the patient.[8] She was advised to take 'selfie' of her face every hour on her smart phone while awake and save at least four best 'selfies' on her smart phone every day for further evaluation. Before taking 'selfie,' she was instructed to clean her lips with normal saline with the help of cotton and then to apply petroleum jelly to her lips. Whenever she would feel any urge to pick or lick her lips, she was instructed to cross both her index fingers tightly (making a 'kattii') until the urge persisted. She was also trained with breathing exercises for relaxation. The patient was very cooperative to behavioral therapy. During her stay in the hospital, the patient had shown significant improvement within 2 weeks [Image 2]. At the time of her discharge, she was prescribed vitamin C and E supplements and petroleum jelly for local application, and tablet escitalopram was reduced to 5 mg/day up to her next follow-up. She was advised to practice the behavioral techniques at home and to maintain an activity scheduling chart. After 2 weeks during her first follow-up, the patient's lips were looking apparently normal and she was not worried about her illness anymore [Image 3]. Her medications were stopped, and she was advised to practice breathing exercises whenever she would feel restless.Image 2: Series of images taken during hospital stay as a part of behavioral therapyImage 3: Patient reporting for the first follow up post dischargeDISCUSSION Although the presentation of psychocutaneous disorders varies widely, the underlying cause is psychological stress in which the skin is the target. The maladaptive coping response to psychological stress leads to self-mutilating behaviors that may cause factitious skin lesions.[9] Factitious cheilitis may be one of the presentations in which maladaptive behaviors such as frequent lip licking, lip biting, and lip sucking are commonly associated. Laboratory and pathological test findings are usually inconclusive. If any microorganism colonies are found on histological examinations, they appear to be secondary findings rather than being pathogenic. Due to diagnostic confusion, most of the patients initially tried topical calcineurin inhibitors (tacrolimus and pimecrolimus), systemic steroids, and moisturizing agents, with poor results.[3] There is no empirical treatment guideline to deal with the condition, Further comorbid psychiatric disorders and personality disorders make the prognosis worst.[4] Exfoliative cheilitis typically manifests at young age, as evidenced by the literature, and this was also observed in our instance.[1] Maladaptive behaviors like frequent lip licking and lip picking behaviors can be considered as the causative factor of exfoliation in our patient. In any case of crusted or ulcerative conditions of lips, atopy, actinic damage, contact dermatitis, photosensitivity, and neoplasia should be considered in the differential diagnosis to exfoliative cheilitis. For this, appropriate laboratory investigations are essential to rule out organic causes.[4] A series of 13 patients from Brazil reported with complain of pain and crusting over lips and expressed great concern showing improvement with antidepressants. All the cases were associated with attached material and lack of inflammation, and a name "ointment pseudocheilitis" was proposed for these cases.[10] In the patient described above, she had mild depression and mild anxiety on a psychological evaluation. Her marriage proposal might be considered as her perceived stress toward the illness. Similarly, another reported case of a suspected immunodeficiency disease was later found to be diagnosed with exfoliative cheilitis associated with underlying nontreated anxiety and psychological trauma.[11] Most of the patients suffering from factitious illness however need frequent medical attention but resistive toward psychological interventions.[4] For this reason, the patient should be properly psychoeducated about the illness before starting any intervention. Also the psychological interventions should be individualized for each patient. For repetitive maladaptive behaviors like lip picking, HRT can be applied successfully.[12] In our case, a "competing response" to the repetitive behavior developed in the patient in the form of involving both hands in making gesture "katti" to extinct the repetitive maladaptive behaviors of lip picking. As per Premack principle of reinforcement, a less desirable behavior can be reinforced by the opportunity to engage in a more desirable behavior.[8] Based on this principle, we reinforce our patient to take care of her lips in order to have a beautiful 'selfie' of her face as a 'differential reinforcement' technique based on Premack's principle. CONCLUSION Factitious cheilitis is a rare condition with a varied clinical presentation. After excluding other etiologies of cheilitis, factitious behaviors remain a major contributory factor of exfoliative cheilitis. Although the maladaptive behaviors are consciously produced, the motive behind them is mostly unconscious. Most of the patients waste their precious time and resources by seeking frequent medical attention with poor response to medications but are reluctant toward any psychological interventions. They can be helped by creating a flexible nonjudgmental therapeutic relationship to make them comfortable with psychological interventions. With behavioral therapies like HRT and reinforcement schedules, dramatic response can be seen within a short period. For this, a proper illness history is required for individualizing the interventions. Declaration of patient consent The authors certify that they have obtained all appropriate patient consent forms. In the form the patient(s) has/have given his/her/their consent for his/her/their images and other clinical information to be reported in the journal. The patients understand that their names and initials will not be published and due efforts will be made to conceal their identity, but anonymity cannot be guaranteed. Financial support and sponsorship Nil. Conflicts of interest There are no conflicts of interest.