Bleomycin-Induced Flagellate Erythema in a Patient with Classic Kaposi’s Sarcoma: Report of a Rare Adverse Effect
Chourouq Mustapha Eid *
Dermatology Department, Ibn Sina University Hospital, Mohammed V University, Rabat, Morocco.
Fatima Zahra Sassine
Dermatology Department, Ibn Sina University Hospital, Mohammed V University, Rabat, Morocco.
Salma Moubarik
Moroccan Poison Control and Pharmacovigilance Centre (CAPM), Rabat, Morocco.
Mariame Meziane
Dermatology Department, Ibn Sina University Hospital, Mohammed V University, Rabat, Morocco.
Nadia Ismaili
Dermatology Department, Ibn Sina University Hospital, Mohammed V University, Rabat, Morocco.
Laila Benzekri
Dermatology Department, Ibn Sina University Hospital, Mohammed V University, Rabat, Morocco.
*Author to whom correspondence should be addressed.
Abstract
Aims: To report a rare, bleomycin-specific cutaneous adverse effect — flagellate erythema — occurring early in a patient treated for classic Kaposi’s sarcoma, and to review its clinical features, pathogenesis and management.
Presentation of a Case: A 61-year-old HIV-negative man with no relevant history presented with one year of slowly enlarging angiomatous nodules and infiltrated erythemato-violaceous plaques on the lower limbs and right wrist, without mucosal or nodal involvement. Skin biopsy showed irregular vascular slits dissecting collagen with a perivascular mononuclear infiltrate, and immunohistochemistry was positive for HHV-8, confirming Kaposi’s sarcoma. An extensive work-up, including HIV serology, thoraco-abdomino-pelvic computed tomography, and lymph-node ultrasonography, excluded visceral involvement. Intramuscular bleomycin monochemotherapy was initiated. Four days after the first 5-mg injection, the patient developed a pruritic maculopapular eruption on the trunk, back and proximal limbs, with linear “whip-like” streaks on the back and buttocks, typical of flagellate erythema. Pharmacovigilance causality assessment strongly implicated bleomycin (I4B4). The rash resolved completely within one week of antihistamine therapy, without residual hyperpigmentation.
Discussion: Flagellate erythema, first described in 1970, is an uncommon but characteristic reaction attributed to the low cutaneous activity of bleomycin hydrolase, with microtrauma and a heat-recall phenomenon proposed as triggers. It arises independently of dose and route, with a latency ranging from a few hours to six months, and is diagnosed clinically. Management relies on antihistamines and corticosteroids, usually without discontinuing bleomycin.
Conclusion: Flagellate erythema is a specific and potentially early complication of bleomycin that warrants close monitoring throughout treatment.
Keywords: Bleomycin, flagellate erythema, flagellate dermatitis, classic Kaposi’s sarcoma, cutaneous adverse drug reaction, intramuscular chemotherapy, pharmacovigilance, bleomycin hydrolase, post-inflammatory hyperpigmentation