Vasoconstriction caused by paradoxical vascular responses is thought to underlie systemic sclerosis (SSc) related digital ulcer (DU). Recently, botulinum toxin (BTX) has emerged as a chemical sympathectomy. Updated guidelines indicate that BTX injection is a treatment option for severe and refractory Raynaud phenomenon and DU.
Case 1: A 62-year-old female presented with painful DU. She was diagnosed with diffuse cutaneous SSc 10 months prior. As the DU pain was uncontrolled, we injected BTX-type A (10 IU of Meditoxin [Medytox Inc., Seoul, Korea] in 0.1 mL saline) subcutaneously into the palm, just proximal to the A1 pulley of the left second finger (Fig. 1A). Initially, the fingertip ulcer was 5 × 4 mm in area, with dark pigmentation (Fig. 1B). After additional injections 4 and 5 weeks later, significant improvements were evident (Fig. 1C and 1D).
Case 2: A 50-year-old SSc female was taking endothelin receptor antagonist to treat the recurrent active DU. At the first visit, a deep ulceration 3 × 3 mm in area was evident (Fig. 2A). We injected BTX-type A as described above for case 1. Four days later, the ulcer had improved, but another ulcer had developed (Fig. 2B). We administered further BTX injections 7 and 8 weeks after the first injection. Although there was transient exacerbation of the DU caused by wound infection, gradual improvement was evident (Fig. 2C and 2D).
BTX injection is considered as a possible alternative therapy for SSc-related DUs that inhibits the release of acetylcholine and blocks the transmission of the norepinephrine vesicle at the sympathetic nerve fiber, preventing vascular smooth muscle vasoconstriction. BTX may also inhibit endothelial exocytosis of endothelin-1 and increase the activity of nitric oxide. Our patients exhibited improvements in refractory DU lesions after serial BTX injections, but more data are required to verify efficacy and to determine the optimal dose and injection frequency.
This study was approved by the Institutional Review Board of Soonchunhyang University Seoul Hospital (2017-12-015). Written informed consents were obtained.
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