Chiclero's ulcer: An unusual presentation of Leishmania tropica in Syria

Abstract

Cutaneous leishmaniasis (CL) has been an endemic disease in Syria for decades. The first reports of CL from the Syrian city Aleppo date back to the 17th century. The recent crisis has further empowered the spread of this infection not only in Syria, but also in the neighboring countries. Here, we describe a case of a 67-year-old patient with a 6-month, nonhealing ulcerative lesion of the left auricle. It was initially diagnosed as either a squamous or a basal cell carcinoma. However, the biopsy revealed an unexpected finding of Leishmania amastigotes in the histiocytes. Consequently, CL of the auricle, chiclero's ulcer, was diagnosed. Polymerase chain reaction, in turn, revealed Leishmania tropica to be the causing factor; a rather rare one for chiclero's ulcer. The lesion completely resolved after a 3-week glucantime regimen, without any recurrence after an 8-month follow-up.

Auricular leishmaniasis mimicking squamous cell carcinoma of the pinna

“…CL can occur on any exposed area of the body; however, the pinna or the auricle is a very rare site for the disease2 12 with less than 15 cases reported in existing literature to the best of our knowledge. Patients with CL of the auricle tend to present with painful crusted, erythematous and ulcerative enlargements of the pinna2 13; however, it may be also a painless lesion as reported by Goldin et al 14. The clinical course of the disease varies significantly and was reported to be anywhere between 2 and 16 months by Morente et al based on their analysis of case reports of Old World CL involving the pinna 12.…”

“…Based on this presentation, it is very easy to misdiagnose CL of the auricle, as it presents very similarly to many other pathologies. It is especially easy to mistake for a neoplastic lesion, as evidenced by Youssef et al ’s case report which describes an ulcerative, non-healing auricular lesion initially diagnosed as either squamous or basal cell carcinoma and Tarkan et al ’s case report describing a lesion presumed to be angiolymphoid hyperplasia of the auricle, both of which were eventually found to be CL of the auricle 13 15…”

The Investigation of the Association of Cutaneous Leishmaniasis in Biopsy Specimens of the Patients with Granulomatous Disease and Skin Cancer Using the Molecular Method

“…In another case study, conducted on a 22-year-old female pregnant patient carrying lesion in the ear amastigotes of Leishmania parasite was detected ( 27 ). In Syria, for a 67-year-old male patient with a case in the ear for 6 months, amastigotes of Leishmania spp was observed in hematoxylin-stained preparations ( 28 ).…”

Unusual Observations in Leishmaniasis—An Overview

“…The lesions of auricular leishmaniasis mimicking squamous cell carcinoma were described [ 128 , 129 , 130 ]. L. tropica is an atypical species causing chiclero’s ulcer ( Figure 3 b) [ 122 , 131 ]. The differential diagnosis for auricular leishmaniasis includes the symptoms of infections such as lupus vulgaris, mycobacterial disease, fungal infections and syphilis with the involvement of the ear as a site, and which may lead to the misdiagnosis of CL [ 132 ].…”