Cutaneous Leishmaniasis: A Rare Cause of Perichondritis

Abstract

Dear Editor, Cutaneous leishmaniasis (CL) can present in various clinical manifestations and mimic many dermatological diseases. Here, we present a case of CL involving the perichondrium of the ear, characterised solely by erythema and edema. A 49-year-old male patient presented with erythema, warmth, and pain on his left ear for 4 months. Previously, he had several oral antibiotics, topical tacrolimus, and topical nadifloxacin with no response. The patient’s medical history was notable for type II diabetes mellitus as the only existing condition. Physical examination revealed erythema, edema, and tenderness on the left auricula [Figure 1a]. Laboratory investigations indicated a normal complete blood count, renal and liver function tests, C-reactive protein, and sedimentation levels. Serology for hepatitis and human immunodeficiency virus was negative. Temporal magnetic resonance imaging (MRI) showed edema consistent with perichondritis in the left auricle. Bacteriological, mycological, and mycobacteriological tissue cultures from the periauricular skin and edematous cartilage were negative. Despite a 4-week course of parenteral antibiotic treatments, which included ampicillin-sulbactam, ceftazidime, linezolid, metronidazole, piperacillin-tazobactam, tigecycline, and meropenem, there was no observed improvement. Histopathological examination of the skin biopsy from the periauricular region revealed granuloma structures including multinuclear giant cells in the dermis, inflammatory cell infiltration involving lymphocytes and plasma cells, and infrequent eosinophil leukocytes. Histochemical analysis with PAS, Orsein, Acid Fast, and GROCOTT’s GMS did not identify any specific microorganisms [Figure 2]. Histopathology of cartilage biopsy revealed an inflammatory cell infiltration including plasma cells and a distinct area marked by necrosis and necrobiotic changes [Figure 3]. Further investigations were performed due to the presence of granuloma structures in the skin biopsy. The tuberculin skin test was found to be 16 mm, and the interferon-gamma (Quantiferon) test was negative. No leishmania parasites were found in the smear specimen, and no growth was observed in the NNN (Novy-MacNeal-Nicolle) medium. Upon further examination, polymerase chain reaction (PCR) revealed Leishmania infantum in the tissue sample. Systemic meglumine antimoniate was administered at a dose of 20 mg/kg/day, and intralesional meglumine antimoniate treatment was applied twice a week. Remarkably, no side effects were reported throughout the treatment period. The parenteral meglumine antimoniate regimen was completed over 21 days, leading to a complete regression of the patient’s clinical condition [Figure 1b]. Ear involvement is rare with Old World CL agents (L. major, L. tropica, L. infantum, and L. aethiopica).[1] There are only a few CL cases presented with solely erythema and edema of the ear, lacking the typical papulonodules, prominent ulceration, and crusts.[1-3] These cases typically resemble perichondritis. Additionally, there is a documented case of CL with ear involvement classified as perichondritis; however, the text does not specify cartilage involvement.[4] The main methods used for the diagnosis of CL are smear, culture, PCR, and histopathological examination. Among these methods, PCR is the most sensitive one.[5] Two previous reports made the diagnosis by Giemsa staining, and PCR was not performed.[1,2] In two reports, PCR was performed, and L. infantum[3] and L. major[4] were found to be causative agents. CL was not identified in the smear, culture, and biopsy samples; however, PCR proved conclusive in our case. The histopathological findings of CL include diffuse and densely packed inflammatory infiltration, composed of lymphocytes, histiocytes, and plasma cells, which mainly occupies the superficial and mid-dermis and may extend to the deep dermis.[6] A Grenz zone is typically not observed. The mostly observed type granulomas are non-necrotizing granulomas with or without amastigotes. However, sarcoidal granulomas and, more rarely, necrobiotic palisading granulomas and foreign-body granulomata may also be seen.[6]Figure 1: (a) Erythema and edema seen on left auricula; (b) complete healing after the treatmentFigure 2: Granuloma structures including multinuclear giant cells in the dermis, inflammatory cell infiltration involving lymphocytes and plasma cells, and infrequent eosinophil leukocytes (H and E X200)Figure 3: Inflammatory cell infiltration including plasma cells and an area characterised by necrosis and necrobiotic changes (H and E X 100)Therefore, in patients with perichondritis exhibiting granulomas in the histopathological examination such as our case, CL should be remembered, especially in those who have a history of travel to or residence in an endemic region. However, our patient is a Turkish patient who lives in western Turkey, which is not an endemic region for leishmaniasis, and has no history of travelling to an endemic region. Even when smear or culture results show no evidence of parasites, PCR analysis should be conducted if clinically suggestive of CL. Declaration of patient consent The authors certify that they have obtained all appropriate patient consent forms. In the form, the patient(s) has/have given his/her/their consent for his/her/their images and other clinical information to be reported in the journal. The patients understand that their names and initials will not be published and due efforts will be made to conceal their identity, but anonymity cannot be guaranteed. Financial support and sponsorship Nil. Conflicts of interest There are no conflicts of interest.