Bleomycin‐induced flagellate erythema after venous malformation sclerosis—Case report and brief review

Abstract

Bleomycin-induced flagellate erythema is a rare but typical skin toxicity of bleomycin. We report the case of a boy with a left foot venous malformation who developed this skin rash after two sessions of bleomycin intralesional injection. We discuss the mechanism and characteristics of this reaction to bleomycin, which is usually benign and self-limited. We conclude that, although rare, flagellate pigmentation can occur when bleomycin is used as a sclerosant in children.Bleomycin is an antibiotic with cytotoxic properties, commonly used in combination regimens for the treatment of Hodgkin's and non-Hodgkin's lymphoma; squamous cell carcinoma of the head and neck; and germ cell, gynecologic, and skin tumors.1 In addition to its antitumor activity, bleomycin is used as a sclerosant in the treatment of vascular malformations and in recurrent malignant pleural effusions.1 Bleomycin-induced toxicities are more common in the lungs and skin because of a lower activity of bleomycin hydrolase in these organs. 2The reported dermatologic side effects of bleomycin include alopecia, Raynaud's syndrome, hyperkeratosis, nail bed changes, palmar and plantar desquamation, eczematous changes, digital gangrene, and pigmentary alterations. 3,4 Less commonly, skin toxicity presents as flagellate erythema, a unique drug rash that appears as "whiplike" linear streaks. It may affect the face, trunk, or limbs. 5Moulin first described it in 1970. 6Although the use of bleomycin as an antineoplastic agent has been decreasing, 5 there has been growing interest in its use as a sclerosant because of the low risk of side effects and its low cost. In addition, patients recover quickly, with good outcomes. 7We report a case of bleomycin-associated flagellate erythema after intralesional injection of a venous malformation and discuss the mechanism and characteristics of this reaction. | CASE REPOR TWe report the case of a 14-year-old boy with a venous malformation (VM) in the left lower limb. He underwent partial excision of the lesion at the age of 9 years. The postoperative period was complicated by a hematoma and skin ulceration, which was managed using local wound care. There was no other significant past medical or surgical history and no dermatologic disorders or allergies. A skin biopsy of a lesion on his left flank was obtained. Pathologic examination showed lymphocytic and histiocytic infiltration around

Intraoperative Anaphylaxis Following Injection of a Bleomycin-Gelatin Solution for Sclerotherapy

“…There have also been anecdotal reports of acute adverse effects including hyperpyrexia, hypotension, altered mental status, and respiratory distress during the administration of bleomycin for chemotherapy in oncology patients [11,14]. Although bleomycin was not determined to be the cause of the allergic reaction in our patient, other anecdotal reports have suggested its potential role in an allergic reaction known as flagellate erythema, a unique drug rash that appears as whip-like or linear streaks following local injection or application [15][16][17]. It may affect the face, trunk, or limbs.…”

Cutaneous reactions to pediatric cancer treatment: Part I. Conventional chemotherapy

“…38 One common skin reaction to bleomycin is flagellate erythema, which has been reported in both adults and children. 37, [39][40][41] This presents with pruritus and an erythematous or hyperpigmented linear eruption on the trunk and limbs, appearing within a few hours or up to 6 months after administration ( Figure 6). Dermal sclerosis can also result from bleomycin in adults and children, sometimes appearing within the lesions of flagellate erythema.…”

Sclerotherapy with Adjunctive Stasis of Efflux (STASE) in Venous Malformations: Techniques and Strategies

“…39 One must be wary of rarely reported but devasting complications of flagellate erythema, acute lung toxicity or pulmonary fibrosis. [40][41][42][43] There is a report of one infant with acute pulmonary toxicity after one session of 1.2 mg/kg bleomycin and a case of pulmonary hypersensitivity reaction occurring in one patient after a second session of only 0.28 mg/kg or 0.4U/kg. 41,42 As such, there is ongoing academic debate as to safe per session and cumulative lifetime doses.…”

“…Bleomycin specific complications include fever, flu-like symptoms, skin tingling, hyperpigmentation, flagellate rash pulmonary hypersensitivity and pulmonary toxicity/fibrosis. 40 Hyperpigmentation can occur in a few hours or as late as 2 months and may occur in regions of skin scratching or adhesive placement and removal, however, may also be idiosyncratic and due to hypersensitivity. Judicious use of adhesives perprocedurally may be helpful in reducing local hyperpigmentation.…”