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ORIGINAL RESEARCH article
Front. Med.
Sec. Infectious Diseases: Pathogenesis and Therapy
Volume 11 - 2024 |
doi: 10.3389/fmed.2024.1453211
Diagnostic Challenges in Cutaneous Leishmaniasis Due to Atypical Leishmania infantum: Pathologists' Insights from Re-emergence Zones
Provisionally accepted- 1 Immunology Frontier Research Center, Institute of Medical Science, The University of Tokyo, Tokyo, Japan
- 2 Faculty of Medicine, Ege University, Bornova, İzmir, Türkiye
- 3 The University of Tokyo, Bunkyo, Tōkyō, Japan
- 4 Acıbadem University, Istanbul, Türkiye
- 5 Izmir International Biomedicine and Genome Institute, Dokuz Eylül University, Izmir, Türkiye
Background: Leishmaniasis, a parasitic infection affecting both humans and animals, is increasingly spreading across Mediterranean and European regions, largely driven by human migration and environmental changes. In countries like Turkey and across Europe, which have seen large influxes of migrants, the incidence of cutaneous leishmaniasis (CL) is rising, with cases now appearing in cities where the disease was previously undocumented. In these previously non-endemic areas, physicians unfamiliar with the characteristic lesions may misdiagnose CL, particularly in cases with only cutaneous manifestations. This study aims to evaluate the impact of re-emerging CL on the routine diagnostic practices of pathologists in Turkey, by retrospectively reviewing cases. We conducted a retrospective analysis of CL cases diagnosed between 2013 and 2022 at a single pathology center in Turkey, covering multiple provinces. Twelve cases of CL were identified and analyzed based on clinical presentation, pre-diagnosis, histopathological findings, and molecular diagnostics. DNA extraction and PCR were performed on paraffin-embedded tissue samples to identify the Leishmania species involved.Results: Out of the twelve CL cases reviewed, seven exhibited morphological findings strongly suggestive of CL (MFSS of CL), warranting further microbiological evaluation. All patients presented with non-healing skin lesions characterized by central ulceration, crater-like formations, or papulonodular lesions. Notably, CL was included in the clinical pre-diagnosis in only 58.3% of cases, while it was not considered in the remaining 41.7% of cases. Clinicians initially pre-diagnosed skin tumors in six cases (50%), four of which led to wide surgical excision. Histopathological examination in all cases revealed chronic or mixed (acute/chronic) inflammation, predominantly rich in histiocytes. To further investigate the role of Leishmania species in the pre-diagnosis, DNA extraction and PCR were performed on paraffin-embedded tissue samples, identifying L. infantum as the causative agent in ten cases and L. major in two cases. Notably, L. infantum was the causative agent in all five cases initially misdiagnosed as skin tumors, which were also associated with a granulomatous type of chronic inflammation.This study highlights the diagnostic challenges in distinguishing CL from skin tumors, particularly in non-endemic regions where L. infantum is an atypical causative agent.
Keywords: Leishmania infantum, Cutanaous leishmaniasis, Skin tumors, PCR, Dermatopathology, Surgery, Misdiagnosis, reemergence
Received: 22 Jun 2024; Accepted: 02 Sep 2024.
Copyright: © 2024 Ekemen, Nalcaci, Toz, Sanjoba, Demirkesen, Cetin, Tecimer, Yildiz, Gursel, Ince, OZBEL and Coban. This is an open-access article distributed under the terms of the Creative Commons Attribution License (CC BY). The use, distribution or reproduction in other forums is permitted, provided the original author(s) or licensor are credited and that the original publication in this journal is cited, in accordance with accepted academic practice. No use, distribution or reproduction is permitted which does not comply with these terms.
* Correspondence:
Suheyla Ekemen, Immunology Frontier Research Center, Institute of Medical Science, The University of Tokyo, Tokyo, 565-0871, Japan
Chizu Sanjoba, The University of Tokyo, Bunkyo, 113-8654, Tōkyō, Japan
Cuyan Demirkesen, Acıbadem University, Istanbul, 34662, Türkiye
Emel D. Cetin, Acıbadem University, Istanbul, 34662, Türkiye
Tulay Tecimer, Acıbadem University, Istanbul, 34662, Türkiye
Pelin Yildiz, Acıbadem University, Istanbul, 34662, Türkiye
Umit Ince, Acıbadem University, Istanbul, 34662, Türkiye
Cevayir Coban, Immunology Frontier Research Center, Institute of Medical Science, The University of Tokyo, Tokyo, 565-0871, Japan
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