Bilateral Nicolau syndrome after uterine artery embolization for postpartum bleeding.

Abstract

Sir We agree with the effectiveness of postoperative uterine artery embolization (UAE) for nonremoval of an abnormally invasive placenta (1). We have reported UAE complications such as arteriovenous fistula (2) and recently experienced another complication in a 26-year-old woman who arrived at the emergency room with postpartum hemorrhage after a normal spontaneous vaginal delivery. We performed bilateral UAE. Two days after a first UAE attempt, the patient presented with a hematoma in a right episiotomy site and fresh bleeding. During a second UAE attempt, she was seen to be actively bleeding from a branch of the left internal iliac artery, so we performed a second left internal iliac artery embolization. The patient had received 19 units of red blood cells and 5 units of fresh frozen plasma after delivery. One day after the second UAE, we injected one ampoule of diclofenac intramuscularly for two successive days to reduce pain. On the first day, we injected into the left buttock, and on the second day into the right side buttock. The patient experienced intense pain at both injection sites; a purulent discharge was observed and necrotic skin had denuded with the formation of a small round circular hole ulcer with 15-cm depth in the gluteal muscle area site (Figure 1). Methicillin-resistant Staphylococcus aureus was found at the ulcer site. After daily dressings and systemic antibiotics, the wound was covered with healthy granulation tissue in about 2 weeks. The diagnosis was Nicolau syndrome, a rare disease characterized by severe pain, skin rash, and soft tissue necrosis at the site of an injection (3). The typical clinical presentation is pain around the injection site soon after injection, followed by erythema, and hemorrhagic patch, and finally necrosis (4). The exact pathogenesis and etiology of Nicolau syndrome are not well understood. Following an injection, ischemia, direct damage to an end artery, vasospasm, embolization of the injected material, pressure due to the material placed around the vessel, sympathetic stimulation, and subsequent vascular spasm from peri-arterial areas are known to occur (5). The pathogenesis of Nicolau syndrome is debated, so clinicians should be aware of such rare but serious complications after intramuscular injection of common agents, such as nonsteroidal anti-inflammatory drugs, and should follow proper procedures (3). Diagnosis is clinical, based on a skin biopsy, which shows necrotic changes caused by ischemia (5). Management of Nicolau syndrome includes surgical debridement and systemic antibiotics. Use of subcutaneous rather than intramuscular injections prevents Nicolau syndrome (5). No reports are available concerning the association between bilateral injection sites and Nicolau syndrome in the gynecological field. We presumed that the UAEs caused insufficient blood supply to the buttocks. Diclofenac may have been a contributing factor at the injection site. Therefore, obstetricians and gynecologists should be aware of the successive effects of UAE on the buttocks. We suggest that our case was related to a blood supply insufficiency to the buttocks after embolization procedures.