Elsevier

Acta Tropica

Volume 172, August 2017, Pages 240-254
Acta Tropica

Review
Atypical presentations of cutaneous leishmaniasis: A systematic review

https://doi.org/10.1016/j.actatropica.2017.05.022Get rights and content

Highlights

  • An overview of the clinical aspects of atypical lesions of cutaneous leishmaniasis.

  • These lesions demand a thorough investigation focused on the differential diagnosis.

  • The role of the patient’s immunological status in the development of atypical lesions.

Abstract

Cutaneous Leishmaniasis (CL) is endemic in 88 countries, showing relevant prevalences. The aim of this study was to perform a systematic review on atypical lesions of CL around the world, addressing clinico-epidemiological, immunological and therapeutic aspects. A search of the literature was conducted via electronic databases Scopus and PubMed for articles published between 2010 and 2015. The search terms browsed were “cutaneous leishmaniasis”, “atypical” and “unusual”. Based on the eligibility criteria, 34 out of 122 articles were included in the final sample. Atypical lesions may include the following forms: erythematous volcanic ulcer, lupoid, eczematous, erysipeloid, verrucous, dry, zosteriform, paronychial, sporotrichoid, chancriform and annular. In any cases, they seem to be another disease like subcutaneous and deep mycosis, cutaneous lymphoma, pseudolymphoma, basal and squamous cell carcinoma. The lesions have been reported in the face, cheeks, ears, nose, eyelid, limbs, trunk, buttocks, as well as in palmoplantar and genital regions; sometimes occurring in more than one area. The reason for clinical cutaneous leishmaniasis pleomorphism is unclear but immunosuppression seems to play an important role in some cases. There are no established guidelines for the treatment of atypical cutaneous leishmaniasis. However, pentavalent antimonials remain as first line treatment for all forms of leishmaniasis even for HIV-infected patients and atpical forms. Finally, to diagnose an atypical lesion properly, the focus has to be on the medical history and the origin of the patient, comparing them to the natural history of leishmaniasis and always reminding of possible atypical presentations, to then start searching for the best diagnostic method and treatment, reducing the misdiagnosis rate and, subsequently, controlling the disease progression. Thereby, contributing for breaking the transmission chain of the parasite, due to early correct diagnosis which, in turn, contributes to reduce the prevalence.

Introduction

Leishmaniasis collectively refers to various clinical syndromes caused by obligate intracellular protozoa of the genus Leishmania, that is transmitted by the sandfly (Ayatollahi et al., 2015, Karami et al., 2013). It is a vector-borne zoonosis, with dogs, rodents, wolves, and foxes as common reservoir hosts and humans as incidental hosts (Ayatollahi et al., 2015, da Silva et al., 2014). Approximately 1.5 million new cases are documented each year and more than 350 million people live in areas of active parasite transmission (Dassoni et al., 2013).

Cutaneous leishmaniasis (CL) is one of the four different forms of the disease and the most common form (Alhumidi, 2013); being a major public health problem in 88 countries with an endemic behavior (Dassoni et al., 2013, Oryan et al., 2013). The other three clinical forms include: visceral leishmaniasis (or Kala-azar); mucocutaneous leishmaniasis; and diffuse cutaneous leishmaniasis (DCL) (Shah et al., 2010, Talat et al., 2014). Diffuse cutaneous leishmaniasis is relatively rare and usually associated with mucous membrane involvement (Dassoni et al., 2013).

In an endemic area, it is necessary for the physician to be aware that any atypical lesion, especially chronic form, should be investigated for cutaneous leishmaniasis (Ayatollahi et al., 2015). Be attentive to the clinical examination, investigate if the patient lives in endemic area or traveled to an endemic area can facilitate the diagnosis.

There have been reports of some atypical lesions of CL around the world. These lesions can mimic many other diseases and confound the physicians, which may delay the precise diagnosis, submitting the patients to unnecessary treatments, worsening the picture, and contributing to the transmission chain of the parasite. However, there is a lack of studies addressing specifically this issue. In this scenario, we aimed to perform a systematic review of the literature on atypical lesions of cutaneous leishmaniasis, addressing the clinic-epidemiological, diagnostic and therapeutic features of these uncommon lesions.

Section snippets

Material and methods

The present study represents a systematic review of literature. The searches were conducted via the international electronic databases Scopus and PubMed, on March 20th, 2015. The search terms browsed in the databases were “cutaneous leishmaniasis ” (Medical Subject Headings – MeSH), “atypical” and “unusual”, with 5-year time limit (2010–2015).

In order to reduce the risk of bias of individual studies, where a title or abstract seemed to describe a study eligible for inclusion, the articles were

Results

Initially, the aforementioned search strategies resulted in 109 references in Scopus and 57 references in PubMed. Forty-four of them were repeated in the two databases and were counted only once. After this, there were 13 different references counted in PubMed, ending in 122 articles to be analyzed. After analyzing title, abstract and text according to the eligibility criteria, 34 articles were included in the final sample (Fig. 1). From this total, 30 (88.23%) are case reports and 4 (11.76%)

Characteristics of the atypical lesions of CL

The classic initial clinical sign of the CL is the appearance of small papules and an erythematous nodule, which may be single or multiple, usually located in an exposed region of the tegument where, after a few months, it develops into ulcers, the most common presentation (63%–91% of cases), with indurated raised outer borders, regular contours and a cross-grained background with or without a seropurulent exudate (Adriano et al., 2013, Siah et al., 2014). Commonly, CL skin lesions have a wide

Conclusions

Atypical lesions of tegumentary leishmaniasis have been reported in the majority of endemic countries. Patients’ age and gender seem to have no relation with the course or type of the lesions. Atypical lesions demonstrated aspects such as lupoid, eczematous, erysipeloid, verrucous, dry, zosteriform, paronychial, sporotrichoid, chancriform, annular and erythematous volcanic ulcer. Some cases have shown features similar to subcutaneous and deep mycosis, and malignancies such as lymphoma,

Acknowledgment

This work was supported by the Coordination for the Improvement of Higher Education Personnel, Ministry of Education of Brazil (CAPES/MEC).

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