Leucocytoclastic Vasculitis Presenting as Bilateral Ulcerative Keratitis: A Case Report

Abstract

Introduction: Small artery disease caused by neutrophils and immune-mediated is known as leucocytoclastic vasculitis (LCV). Clinically, it manifests as palpable, asymptomatic purpuric papules on the limbs. Ocular manifestation is rare. Here, we describe a case of peripheral ulcerative keratitis (PUK) associated with LCV.

Case presentation: A 59-year-old man was referred to the hospital with blurred vision due to corneal perforation in his left eye. He complained of itchy nodules on his hands and lower legs for 15 years and the skin biopsy of the back of his hand revealed LCV 6 years ago, which suggested erythema elevatum diutinum. The patient was under treatment with anti-inflammatory and immunosuppressive drugs and physical features of LCV seen in him included erythema on his hands and legs. After receiving conjunctival flap covering surgery, the corneal perforation was resolved. Conjunctival flaps covered cornea that limited his vision to hand motion. Six months later, he was referred to our clinic again because of pain, redness, photophobia, and tearing in the right eye, presenting with PUK. Necrotic tissue was removed during surgery, which also included a conjunctival flap covering procedure. Following surgery, the symptoms were reduced, and the postoperative eye condition remained stable.

Conclusion: To our knowledge, it is the first case of PUK secondary to LCV which was diagnosed 6 years ago. This case demonstrates that PUK associated with LCV can be successfully treated by surgical interventions.

Keywords: Leukocytoclastic vasculitis; conjunctival flap covering surgery; erythema elevatum diutinum; peripheral ulcerative keratitis.

Figure 1.

Clinical appearance of the cutaneous lesions characterized by erythematous palpable purpura and joint nodules on the dorsum of the patient’s hands.

Figure 2.

Slit-lamp photograph showing nasal conjunctival tissue were growing well in the left eye (A) and conjunctival hyperemia, marginal corneal guttering from 11 o’clock to 1 o’clock with corneal ulceration in the right eye (B). Two-month after surgery, the edges of the ulcer showed dense stromal inflammatory cell infiltrations (C). Three-month after surgery, the patient’s ocular condition remained stable (D).

Figure 3.

Skin biopsy showed massive neutrophilic infiltrate in soft tissue mainly consisted of polymorphonuclear leukocytes, leukocytoclasis, nuclear dust around the blood vessels, and perivascular fibroblastic proliferation.