Erysipeloid Cutaneous Leishmaniasis in the Old World: A Systematic Review

Erysipeloid cutaneous leishmaniasis (ECL) is a rare presentation of cutaneous leishmaniasis that classically presents as erythematous, infiltrated lesions on the centrofacial region. We conducted a systematic review on the demographic characteristics, clinical presentations, laboratory findings, and treatment of ECL in the Old World. A systematic literature search was performed using the terms ("erysipeloid" OR "erysipelas") AND "leishmaniasis" across Web of Science, PubMed, Scopus, Embase, and Google Scholar. Only studies reporting cases from the Old World were included. Based on the anatomic location of lesions, we defined three clinical distributions of ECL: (1) centrofacial involvement, (2) non-centrofacial lesions involving the face and/or scalp, and (3) lesions on the neck, trunk, and extremities. Our search yielded 3290 publications, of which 24 met the inclusion criteria. In total, 103 patients with ECL, including 73 females (70.9%) and 30 males (29.1%) with a mean age ± standard deviation of 52.1 ± 12.3 years, were included. Seventy-two patients (57 females, 15 males) had centrofacial lesions. Females comprised 83.8% of this subgroup, a difference that was statistically significant (p < 0.005). ECL was more common in postmenopausal females. ECL lesions on the trunk and extremities were reported in five patients. Sixty-five patients (63.1%) were treated with pentavalent antimonials alone or in combination with other drugs. In conclusion, ECL is a rare presentation of cutaneous leishmaniasis, most often affecting elderly women and primarily localizing to the mid-face. Old age and menopause might predispose to ECL development. Clinical suspicion is necessary for the diagnosis of lesions suggestive of ECL in endemic regions.