Ann Thorac Surg 2007;83:1197-1199
© 2007 The Society of Thoracic Surgeons
Case Reports
Oral Erosive Lichen Planus Regression After Thymoma Resection
Antonio Bobbio, MD, PhDa,*,
Paolo Vescovi, MD, PhDb,
Luca Ampollini, MDa,
Michele Rusca, MDa
a Unit of Thoracic Surgery, Department of Surgical Sciences, University of Parma, Parma, Italy
b Oral Pathology and Medicine Unit, Department of Dental-Ophthalmologic and Cervico-facial Sciences, University of Parma, Parma, Italy
Accepted for publication August 9, 2006.
* Address correspondence to Dr Bobbio, U.O. Chirurgia Toracica, Università di Parma Viale Gramsci 14, 43100 Parma, Italy (Email: antonio.bobbio{at}unipr.it).
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Abstract
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Thymomas are neoplasms known to be frequently associated with autoimmune disorders. Oral lichen planus is an immunologically based, chronic inflammatory oral mucosal disease of undetermined cause. We describe a 79-year-old patient with a 6-month history of generalized oral erosive lichen planus in whom a chest roentgenogram led to the discovery of an anterior mediastinal mass consistent with thymoma. Transsternal complete thymoma resection achieved erosive oral lichen planus regression. The clinical correlations between erosive oral lichen planus and thymoma are presented.
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Introduction
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Oral lichen planus (OLP) is a chronic inflammatory oral mucosal disease of unknown etiology that is characterized by relapses and remissions. OLP is a relatively common disorder estimated to affect 0.5% to 2% of the general population. Several types of OLP have been described, although the two main clinical presentations are the reticular and erosive forms [1–2]. Reticular OLP is usually asymptomatic and can be present as white interlacing and keratotic lines (Wickham striae), with an erythematous border involving the buccal mucosa, tongue, and gingiva. An erosive form of OLP occurs with erythema caused by inflammation or epithelial thinning, or both, with ulceration and pseudomembrane formation. Erosive OLP is always associated with burning pain and sensitivity.
OLP associated with thymoma has seldom been reported, either alone or concomitantly with other autoimmune diseases [3]. The pathogenetic and clinical relationships between OLP and thymoma encountered in this case are presented with the aim of strengthening the hypothesis that erosive OLP in the presence of a thymoma could be considered a thymoma-associated autoimmune disease.
A 79-year-old man with a history of ischemic cardiac disease and in treatment with angiotensin-converting enzyme inhibitors, amiodarone, and aspirin was seen on July 2005 at the Oral Pathology Unit of the University of Parma. An intraoral examination revealed extensive erosive OLP affecting bilaterally the buccal mucosa, the dorsal and lateral aspect of the tongue, the palate, the attached gingivae, and the upper and lower lips (Fig 1). The oral lesions were painful and impeded normal eating. The patient was partially edentulous and could not use his dental prosthesis. No extraoral abnormalities were evident. The patient did not smoke tobacco or drink alcohol. Serologic markers for hepatitis B and C virus were negative.
Two biopsy specimens, one in the buccal mucosa and one in the dorsal part of the tongue, confirmed the diagnosis of erosive OLP without signs of epithelial dysplasia. Therapeutic management consisted of successive cycles of 50 mg/day of prednisone and 0.05% clobetasol propionate or betamethasone valerate gel mixed with adhesive paste to facilitate adhesion to the oral tissue. The prophylactic use of 0.12% chlorhexidine gluconate and a nystatin rinse was prescribed to avoid fungal and bacterial infection. Medical treatment provided only transient relief of symptoms and temporary reduction in clinical manifestations; on interruption of systemic steroid, the oral erosive lesions recurred.
After 6 months, in the face of the persistent erosive OLP, a systematic chest roentgenogram was done and an enlargement of the right mediastinal shadow profile was noted. A computed tomography (CT) scan found an anterior mediastinal mass 10 cm in diameter, well circumscribed and capsulated, with a heterogeneous contrast-medium enhancement. No pleuropulmonary abnormalities were evident. Tests showed that serum anticholinergic and antismooth muscular antibody were absent and serum protein electrophoresis was normal. A cardiopulmonary exercise test found the oxygen consumption at peak of exercise to be 16.1 mL/(kg · min), which was 59% of the expected value. Cardiac ultrasound exploration measured a left ventricular ejection fraction of between 0.45 and 0.50.
At operation through a median sternotomy, a solid, gray intrathymic capsulated mass was found, and a thymectomy was performed. Definitive pathologic examination found a tumor measuring 7 x 7 x 4 cm occupying the inferior part of the thymus gland consisting of a proliferation of spindle and oval epithelial cells with rare and normal lymphocyte islets. No capsular involvement was noted, and the thymoma was classified as type A in the World Health Organization classification.
The patient was discharged from the hospital after 9 days. At the outpatient visit 1 month after surgery, the OLP had completely regressed, and the patient could now wear his dental prosthesis. Six months later, the patient was still disease free.
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Comment
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An interesting aspect of this case is that it focuses the attention on patients presenting with oral lichen planus and thymoma. The relation between thymus diseases and autoimmune disorders is well known. In 1987, a retrospective study among 172 patients with thymoma found fungal mucocutaneous disease as the most common thymoma-associated cutaneous disorder; furthermore, 2 patients in that study were noted with pemphigus and lichen planus, respectively. The incidence of the two latter diseases was no higher than that encountered in the general population [4]. Since then, paraneoplastic pemphigus encountered in patients with thymoma or other malignancies has been recognized as a well-defined humoral autoimmune syndrome characterized by autoantibody formation directed against epithelial antigens [5].
Current data on the oral localization of lichen planus suggest that this is a T-cell-mediated autoimmune disease in which autocytotoxic CD8+ T cells trigger apoptosis of the oral epithelial basal cells, and no specific auto-antibodies, as in myasthenia or paraneoplastic pemphigus, have ever been identified [6]. A recent report reviewed 16 cases of thymoma associated with lichen planus, either alone or concomitant with other immunologic diseases [3]. Of interest is that in all of those cases, an oral localization of lichen planus was always present, and in those patients in whom oral lesions were detailed, an erosive form of OLP was always described [4]. It is also interesting to note that, as in the present case, OLP regressed after complete thymectomy in at least 2 patients of this review in whom follow-up was reported [7, 8].
In only four cases has OLP previously been reported as the sole associated disease in patients with thymoma [3]. In our patient, the chronic and aggressive behavior of OLP prompted us to expand the exploration to other less-common causes implicated in its insurgency, and in this light, as suggested by others [3], a chest roentgenogram was performed to rule out a thoracic malignancy. Because thymomas are frequently discovered secondarily to the occurrence of clinical parathymic syndrome, the recognition of an erosive form of OLP as being an autoimmune disease potentially triggered by the presence of a thymoma would be of considerable importance.
In conclusion, although the association between lichen planus and thymoma is rare, the features of our case are in accordance with those previously reported in which an erosive form of OLP could occur, in at least some cases, as a parathymic syndrome. Because the mechanisms that could be implicated have still not been elucidated, it would seem appropriate to suggest that a complete thymoma resection could, in some instances, achieve definitive regression of OLP.
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References
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Abstract
Introduction
