Surgery and cyclosporine A in the treatment of erosive lichen planus of the feet

ARTICLE

Erosive lichen planus is characterized by painful hypertrophic areas or ulcers on palmar and plantar surfaces, loss of the toenails and cicatricial alopecia of the scalp [1, 2]. It is an unusual variant of lichen planus characterized by painful erosions or ulcers on the toes or the soles. These lesions are very disabling, they tend not to heal spontaneously and do not respond to conventional medical treatment. On one occasion systemic cyclosporine A (CyA) was successfully used, but lesions relapsed 2 months after drug withdrawal [3]. Since 1951 [4], the only treatment which has given uniformly encouraging results is surgical excision and grafting of ulcers. This technique represents the treatment of choice for erosive lichen planus.

We report a case of ulcerative lichen planus of the soles initially treated with CyA alone and later with the drug in addition to surgical treatment with excision and split-skin grafting. We compare the two treatments and we evaluate the efficacy of CyA in this rare variant of lichen planus.

Case report

The patient was 68-year-old man with a 10-year history of painful, disabling, ulcerative inflammatory lesions of the soles of both feet. All previous topical and oral treatments had proved ineffective and the disease was slowly and progressively getting worse.

Physical examination revealed large, superficial ulcers, having an irregular form, an ill-definied border and an inflammatory, erythematous, exudative base, loss of the toenails of the big and second toes of both feet, with atrophy of the nailbed. The patient also presented numerous red-violaceous, polygonal papules of 2 to 4 mm in size on the limbs and some papulo-nodules 1 to 2 cm in size on the ankles and extensor sides of shins and forearms. All the lesions were very pruriginous. No clinical manifestations were present on the oral mucosa, and no cicatricial alopecia was detected on the scalp.

Biopsy specimens from the involved area of the left foot and from lesions of the trunk and limbs were diagnostic of lichen planus.

The patient reported to have always been in good health and had not taken any drugs in the previous months. Routine laboratory parameters were normal. Markers for hepatitis B and C proved negative.

Topical preparations were applied under occlusive wet dressings and treatment with CyA was begun with a dosage of 4.5 mg/kg/day for a month followed by a progressive reduction to smaller, maintenance doses (3 mg/kg/day) for one year. The lesions showed a rapid improvement. The papules and nodules cleared, the extension of the ulcers reduced, although there was never a complete regeneration of the epidermis. With the discontinuation of the medication, the lesions recurred after about one month, in spite of the continuous application of topical antibiotic and steroid ointments. One year later the lesions were almost the same as the ones preceding the treatment (Fig. 1). The patient was in extreme pain, could not walk, and spent much of the day in an armchair. He was hospitalized. Swabs on admission were sterile. CyA administration was resumed with a dosage of 4.5 mg/kg/day and 10 days later, under local anaesthetic, the ulcer on his left sole was covered with a Thiersch split-skin graft transplanted from his left thigh, avoiding the lichen papules. The grafts took well, and no complications followed. He resumed walking 3 weeks later. To protect his foot and the skin grafts, only dry bandages were used. Healing of the graft donor sites was good, but the patient often reported a burning pain on the scar area.

CyA administration was gradually reduced to smaller maintenance doses (3 mg/kg/day). After 10 months, the reduction continued progressively until total withdrawal. By then the left foot was healed, the ulcer of his right foot was smaller (3 x 4 cm) and only a few slightly pruriginous papules were present on his elbows and ankles. He continued the treatment with oral antihistamines and wet dressing, topical antibiotics or corticosteroids on the ulcer. Now, eighteen months after the operation and ten months since the withdrawal of CyA, the condition of his left foot is stable and free from pain (Fig. 2) while his right ungrafted ulcer is painful again and increased in size. In spite of the pain, the limited walking and the time spent daily on medications, the patient has not agreed to undergo skin grafting of his right foot.

Discussion

Erosive lichen planus is considered relatively responsive to treatment. All the medical treatments used so far have given only partial and temporary benefits and could not heal large erosions on the feet [3, 5, 6]. Skin grafting has proved to be the only hope of permanent healing of the ulcerated sole, since numerous authors [1, 4-9] reported grafted cases and all were completely successful during a follow-up period of 4 to 26 years.

In the present case, we administered CyA twice. The first time, systemic CyA produced a marked, but only temporary improvement of the disease. We began the second administration of CyA some days before skin grafting. The aim was to clear the eruptions and to reduce the inflammatory ulcerations to promote the successful outcome of the skin graft. The results were very encouraging. In fact, the dermatitis improved and we were able to successfully treat a large, granulating and hypertrophic erosion of the sole using Thiersch skin grafts. All previous reports have used excision of involved areas and split-thickness graft application to the ulcerated areas. We hypothesize that CyA treatment, by clearing lesions and reducing inflammation of the ulcer in a very short time, might have contributed to the successful outcome in our case.

As the patient refuses to undergo skin grafting on his right foot, for the moment, it is possible to compare the therapeutic results obtained on the ulcers of both the left and the right foot confirming that skin grafting is surely the more effective treatment providing the only possibility for a lasting therapeutic response. In addition, surgery may prevent scarring and secondary deformities, but, especially, the potential, although unlikely development of squamous cell carcinoma reported in the course of ulcerative lichen planus [5]. CyA can be considered an alternative to the conventional treatments in the acute period of ulcerative lichen planus because it provides a drastic, although only temporary, improvement of the lesions. It may also be considered as an adjuvant therapy of skin grafting in ulcerative lichen planus.

REFERENCES

1. Cram DL, Kierland RR, Winkelmann RK. Ulcerative lichen planus of the feet: bullous variant with hair and nail lesions. Arch Dermatol 1966; 93: 692-701.

2. Frattasio A, De Francesco V, Patrone P. Lichen erosivo dei piedi. Descrizione di un caso. G Ital Dermatol Venereol 1994; 129: 387-9.

3. Cecchi R, Giomi A, Bartoli L, Tuci F. Cyclosporine A in chronic ulcerative lichen planus of the feet. Eur J Dermatol 1994; 4: 68.

4. Morgan J. Unusual case of lichen planus. Br J Dermatol 1951; 63: 370-1.

5. Male O, Azambuia R. Diagnostische und therapeutische Probleme beim Lichen ruber ulcerosus. Z Hautkr 1975; 50: 403-12.

6. Crotty CP, Su WPD, Winkelmann RK. Ulcerative lichen planus. Follow-up of surgical excision and grafting. Arch Dermatol 1980; 116: 1252-6.

7. Lendrum J. Surgical treatment of lichen planus of the soles. Br J Plast Surg 1974; 27: 171-5.

8. King D, Karkowski J, Miller SH. Plantar lichen planus ­ treatment by excision and skin grafting. Plast Reconstr Surg 1975; 56: 668-70.

9. Moss ALH, Harman RRM. Surgical treatment of painful lichen planus of the hand and foot. Br J Plast Surg 1986; 39: 402-7.