Annular lichen planus involving the esophagus: a rare diagnosis

Abstract

A 49-year-old woman presented with a 2-year history of a sore mouth, annular, atrophic patches, and chronic ulcerative vulvovaginitis. For 6 years, she had suffered from dysphagia to solids which was increasing in severity. There was no past history of heartburn or other symptoms of esophageal reflux.Mucocutaneous examination showed white erosive patches on the buccal mucosa, cicatricial alopecia, dystrophic nails, and annular atrophic pigmented plaques localized on the trunk ( Fig. 1). Genital examination showed atrophic and sclerotic vulvovaginal lesions with synechia. Cutaneous biopsy showed an atrophic epidermis, a dense lymphocytic infiltrate in the upper dermis with degeneration of the basal epithelium, and Civatte bodies. Serologic tests for hepatitis B and C were negative. A diagnosis of cutaneous annular lichen planus with nail, scalp, oral, and genital involvement was made.Fiberoptic endoscopy, undertaken 6 years previously, revealed erosive and two ulcerative lesions in the esophagus, extending to 20 cm from the incisors. Biopsy from the affected upper esophagus revealed nonspecific inflammatory changes with lymphocytic infiltration. A barium swallow revealed a 5-mm, regular and inflammatory stricture in the upper esophagus ( Fig. 2).A diagnosis of stenosing lichen planus of the esophagus was made 6 years after the first symptoms of dysphagia. Treatment was started with oral prednisolone, 50 mg daily. DiscussionAnnular lichen planus is a relatively rare skin manifestation of lichen planus with exceptional symptomatic esophageal involvement. 1-4 It presents usually with painful dysphagia and sometimes stricture formation. Rarely, esophageal involvement is asymptomatic. 5 A case of esophageal lichen planus associated with cutaneous, genital, oral, scalp, and nail involvement is reported, illustrating the diagnostic and therapeutic difficulties of an esophageal localization.The diagnosis is difficult and is established when examination is performed to exclude other causes of esophagitis and stricture formation, such as peptic esophagitis 6-9 or any other malignant carcinoma. The diagnosis is important because of its prognostic and therapeutic distinction. 5 In our case report, the diagnosis was suspected due to the absence of a past history of symptoms of esophageal reflux, the regular and inflammatory stricture in the barium swallow, and dysphagia and odynophagia without any explanation. The associated skin, oral, genital, scalp, and nail involvement, confirmed by histology, allowed a diagnosis of esophageal stenosing lichen planus.Nevertheless, the diagnosis of lichen planus as a cause of dysphagia was considered only 6 years after the onset of esophageal symptoms because of the initial absence of skin lesions. Indeed, our patient presented with annular and atrophic plaques, considered as annular lichen planus. This annular form is an uncommon variant of lichen planus. 10,11 Steroid treatment in association with dilatation produced considerable improvement and a rapid symptomatic remission. 1,4,9 Other ...