|Year : 2020 | Volume
| Issue : 2 | Page : 136-140
Cutaneous leishmaniasis: An uncommon finding in South-South Nigeria
Benson Uchechukwu Okwara, Edesiri Ejovwoke Ighorodje
Department of Medicine, University of Benin Teaching Hospital, Benin City, Edo State, Nigeria
|Date of Submission||16-Jul-2019|
|Date of Decision||18-Sep-2019|
|Date of Acceptance||14-Nov-2019|
|Date of Web Publication||10-Jul-2020|
Dr. Benson Uchechukwu Okwara
Department of Medicine, University of Benin Teaching Hospital, PMB 1111, Benin City, Edo State
Leishmaniasis, a neglected tropical disease, is of public health significance, especially in the North-West and North-East Nigeria,which forms the belt of the disease in the country. It is caused by flagellate protozoa of the genus Leishmania and is transmitted through the bite of infected female Phlebotomine sandfly. Leishmaniasis presents as self-limiting cutaneous lesions, a mutilating mucocutaneous disease, or a lethal systemic illness. Description of the disease in the southern parts of Nigeria is a rarity. A 28-year-old male presented at the dermatology clinic of UBTH, Benin City, Edo State, South-South Nigeria, with multiple, painless, indurated nodular plaques on the left upper limb. Histology showed features suggestive of leishmaniasis. The patient was treated with oral itraconazole. Diagnosis of leishmaniasis possesses a significant challenge in nonendemic areas; however, a very good history and a high index of suspicion are imperative in overcoming this diagnostic challenge.
Keywords: Cutaneous, leishmaniasis, Phlebotomine, tropical
|How to cite this article:|
Okwara BU, Ighorodje EE. Cutaneous leishmaniasis: An uncommon finding in South-South Nigeria. Sahel Med J 2020;23:136-40
| Introduction|| |
Leishmaniasis is caused by vector-borne protozoan parasites of the genus Leishmania and transmitted via infected female sandflies (Phlebotomus and Lutzomyia). Over 88 countries are affected by the disease with an estimated 350 million people are at risk. In most countries, the incidence numbers are probably underestimated because cases are not recognized and reporting is not mandatory.
Infection with Leishmania parasites can give rise to three clinical manifestations. A localized cutaneous leishmaniasis (CL) with single to multiple skin ulcers, satellite lesions, or nodular lymphangitis; a CL with mucosal involvement (MCL); and systemic visceral leishmaniasis (VL) with involvement of internal organs, such as the liver, spleen, and bone marrow, which is lethal if not appropriately treated.
CL is worldwide the most prevalent clinical form of leishmaniasis, and 90 % of all CL cases occur in only seven countries: Afghanistan, Algeria, Brazil, Iran, Peru, Saudi Arabia, and Syria.
| Case Report|| |
A 28-year-old male presented with a 8-month history of lesions on the left arm and elbow [Figure 1]a and b]. He had worked at a cucumber farm in Jalingo, Taraba State, Nigeria, about 12 months prior to presentation. He noticed rashes which appeared initially like insect bites but subsequently evolved into “fluid-” containing lesions and later firm, nonpainful rashes over several weeks. The lesions progressively increased in size. Physical examination showed multiple, nontender, nonulcerative indurated plaques with grouped papules aligning the margins on an erythematous base, located on the left arm and elbow. The papules were crusted at initial presentation to the clinic. There was no evidence of mucosal or gastrointestinal involvement. A skin punch biopsy was taken from the edge of the lesion for histology. Histopathological examination showed atrophic epidermis with an underlying dermis densely infiltrated by mixed inflammatory cells, predominantly lymphocytes and macrophages. Many macrophages contain eosinophilic-to-amphophilic intracytoplasmic bodies which are probably Leishmania amastigotes, and occasionally multinucleated giant cells [Figure 2]. Hemagglutination assay to rule out systemic involvement could not be done. HIV screening was negative. He was treated with tablet itraconazole 200 mg twice daily for 6 weeks. After 2 months, crusting resolved. No new lesions appeared on further examination.
|Figure 1: Group crusted and indurated papules on the elbow, (b) multiple grouped and indurated papules on the left arm|
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| Discussion|| |
Leishmaniasis is an anthroponotic and/or zoonotic infection  that is caused by the blood and tissue dwelling obligate intracellular protozoa of the genus Leishmania., Natural transmission of Leishmania parasites is carried out by sandflies of the genus Phlebotomus (Old World) or Lutzomyia (New World) [Figure 3]. The World Health Organization (WHO) classifies leishmaniasis as a category 1 disease (emerging and uncontrolled). Leishmaniasis could present as a self-resolving cutaneous lesion, a mutilating mucocutaneous disease, or a lethal systemic illness. Cutaneous leishmaniasis (CL) is the most common form of the disease.,
|Figure 3: Lifecycle of Leishmania spp. (anthroponotic and zoonotic transmission courtesy: researchgate.net)|
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Old-World CL, also known as oriental sore or tropical sore, is a chronic infectious ulcerative skin disease mainly caused by Leishmania major, Leishmania infantum, Leishmania aethiopica, and Leishmania tropica (classically dermotropic)., It is caused by the bite of hematophagous female Phlebotomine sandfly with predilection for intertropical and warm temperate zones. It feeds nocturnally on exposed areas of the body during outdoor activities while laying eggs in rodent burrows, bird's nest, and house wall fissures. Reservoir hosts are mainly rodents in the sub-Saharan Africa, with canines recognized as reservoir hosts in Nigeria. In West Africa, only L. major was reported from reservoir hosts.
Globally, approximately 350 million people live in areas of active transmission of Leishmania, with 14 million people throughout Africa, Asia, Europe, and the Americas, directly affected by the disease (WHO, 2006). The global burden of leishmaniasis has been stable for some years, causing a morbidity of 2.4 million disability-adjusted life-years and a mortality of approximately 70,000 deaths, which is significantly high ranking among infectious diseases. Although a small fraction of those infected by Leishmania parasites will eventually develop the disease, it is estimated that 0.7–1 million annual cases of CL worldwide, that is, 50%–75% of cases, are attributed to CL.
Leishmaniasis is endemic in 88 countries (67 in Old World and 21 in New World). Old-World leishmaniasis is endemic in the Mediterranean Sea and the neighboring countries. In Africa, it is found in North Africa and some countries north of 10° N latitude,, with Nigeria inclusive. Nigeria's leishmaniasis belt [Figure 4]a covers states within the arid savannah zone, that is, north of 10°N. Taraba State is located in the northeastern part of Nigeria [Figure 4]b between longitude 8°00'N and latitude 10°30'E and shares borders with the States of Gombe (north), Adamawa (north-east), Plateau (west), Bauchi (north-west), and Benue (south-west), and with the Republic of Cameroon (south). In the African continent, CL due to L. major, L. tropica, and L. aethiopica is unevenly distributed from the northern to the southern areas of the continent.
|Figure 4: (a and b) Global distribution maps of the leishmaniases (courtesy: elifesciences.org) (a) Reported and predicted distribution of cutaneous leishmaniasis in the Old World. (a) Evidence consensus for the presence of the disease ranging from green (complete consensus on the absence: −100%) to purple (complete consensus on the presence of disease: +100%). The blue spots indicate occurrence points or centroids of occurrences within small polygons. (b) Predicted risk of cutaneous leishmaniasis from green (low probability of presence) to purple (high probability of presence)|
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Statistical information on the occurrence of CL in Nigeria is scarce;, with the exception of a few articles, the endemicity of leishmaniasis is not known. These few reports are domiciled in North Nigeria being a West African endemic CL foci, particularly in Bauchi, Borno, Kaduna, Katsina, Sokoto, and Zamfara states., Between 1924 and 1941, 131 cases of CL were recorded, but there is no conclusive evidence that these cases were parasitologically confirmed. In 1942, smears of 14 patients from Kano with cutaneous sores were confirmed parasitologically to be leishmaniasis;, however, an official report of the first Nigerian case of CL was made in 1944., No data have been recorded outside the Nigerian Leishmania belt, including the southern part of Nigeria, probably due to failure to recognize these lesions, inability to diagnose, poor education and awareness, or low risk due to its geographical nature as breeding grounds for the vector.
Majority of cases of CL is found to affect more males than females  aged between 20 and 40 years., The age range depicts the most active life of young males who seek academic or employment opportunities and also engage in various outdoor activities such as farming, sports, and military service. Our patient was employed on a contract basis for agricultural purposes (worked on a cucumber farmland) though he is resident in Benin City, South-South part of Nigeria.
The geographic spread is attributed to tourism, rural–urban migration, and agro–industrial developmental projects that bring nonimmune urban residents into rural areas, leading to importation of the disease into areas where it is not known. Other factors include wars, natural disasters, poor implementation of malaria control programs, global warming, and deforestation. Nigerians enlisted to have been infected with the disease in nonendemic states most likely got infected outside the region.
The presence of the parasite propagates an inflammatory response driven by T cell immunity in a bid to suppress its ability to produce leishmanicidal substances. At one extreme, localized CL (LCL) shows a vigorous immune response, with most cases resolving without intervention. This form of disease exhibits a helper T-cell subtype 1 immune response, with antibody-mediated uptake of L. major by dendritic cells and interleukin 2, interferon-gamma, and interleukin 12 as the prominent cytokines that induce disease resolution. At the other extreme, with visceral or diffuse cutaneous disease, patients exhibit relative anergy to the Leishmania organism and have a prominent helper T-cell subtype 2 cytokine profile. There is, therefore, a need to follow-up patients with LCL in order to rule out the risk of developing visceral or diffuse cutaneous disease.
CL is classified based on its clinical appearance into moist or dry type; localized, diffuse, mucocutaneous; and post-kala-azar dermal leishmaniasis  and on the duration of symptoms into acute (<1 year) and chronic (>1 year). The incubation period is highly variable  (1–3 weeks for moist type; longer for dry type). The lesions are usually in noncovered regions of the body, mainly the face, neck, or arms. Following the bite, LCL typically presents as a tiny, reddish, and often itchy papule that gradually enlarges and bursts to an ulcer, and the edges of the ulcer become raised and firm with the surrounding skin exhibiting a dusky red color. Most infected individuals are not aware of the time and place of the sandfly bite, and they described the initial lesion as a “boil” or an ulcer refusing to heal after they have tried all forms of traditional remedies. In general, cutaneous lesions resolve spontaneously after 2 months to 2 years, leaving a disfiguring, mottled, depressed scar, with altered pigment, which persists throughout life. Depending on the level of immunity and species involved, disease could evolve into mucocutaneous lesions, diffuse lesions, or involve the viscera.
CL has its peculiarities in HIV coinfected patients; lesions tend to be more severe, multiple, and atypical with more than one clinical form. Healing is protracted, and they have poor response to treatment.
Diagnosis of CL depends on the clinical signs, epidemiological information, polymerase chain reaction (PCR), and culture of infected tissue, or finding the intracellular parasite in biopsy material from the periphery of the lesion. A history of a visit to an endemic area is of diagnostic importance because a bite could occur from the sandfly. The diagnosis of leishmaniasis is confirmed by the detection of parasites in the Giemsa-stained smear specimen  of cutaneous lesions, lymph node, bone marrow, spleen, or of the spinal fluid. Parasites are detected in 10%–20% of cases. In addition, other microorganisms from the wound may infect the culture material and stop Leishmania growth. Histopathological findings are amastigotes (Leishman–Donovan bodies) with features of granulomas consisting of lymphocytes, histiocytes, and plasma cells with epithelioid cells. Frequently, Leishmania can be found within the histiocytes and cultivated in Nicolle–Novy–MacNeal medium for up to 40% of cases.
Leishmania can also be detected by serological tests (indirect immunofluorescence and enzyme-linked immunosorbent assay). The highest sensitivity and specificity can be reached by PCR using the isolated DNA from the lesion and validated genus-specific PCR primers. In developing countries like Nigeria, there is difficulty in accessing a few of the diagnostic tools.
Previously, treatment was recommended for every case of CL, but this is no longer the conventional practice as treatment must be individualized. Old-World disease tends to be self-limiting and heal spontaneously. With low morbidity and almost negligible mortality rate of CL, patients in our setting would usually present late after self-medicating and application of traditional remedies. Leishmaniasis caused by this species does not necessarily need to be treated unless the lesion is in a cosmetically or functionally sensitive site.
Multiple treatment options are used throughout the world for cutaneous disease. Because antileishmanial drugs are potentially toxic, the WHO  recommends simple wound care or local therapy as first-line treatment, followed or replaced by systemic therapy if local therapy fails or cannot be performed. The azoles are majorly used in Nigeria with good response. This could be attributed to unavailability, side effect profile, and cost implication of majority of the therapies outlined such as the antimonials and sodium stibogluconate. Cure rates vary among topicals alone or with systemic therapy. In a bid to reduce exposure to adverse effects of the antimonials, a study done in Sokoto (published in 2012) demonstrated a single-dose treatment with topical silver nitrate to be more effective.
Complications include scarring, disfigurement, and consequently leading to social stigma. Reactivation of dormant infection (after 15 years) accounts for most re-infection, but re-infection with different strains may occur.
Prevention strategies include avoiding outdoor activities from dusk to dawn; presoak/spray outer layer or gear with permethrin; use of repellants containing 20%–30% N, N-diethyl-meta-toluamide or 20% picaridin on exposed skin; and sleeping under a permethrin-treated bed net or in an air-conditioned room. No vaccine or prophylactic medications (for those living in endemic areas) is available yet for the prevention of leishmaniasis.
New Zealand, Antarctica, and the Pacific islands have been documented to be Leishmania-free.
| Conclusion|| |
CL is rarely reported in the southern part of Nigeria; previously documented cases were in the 1990s. The increasing rate of migration to and from endemic regions of leishmaniasis may potentially lead to more cases of leishmaniasis in nonendemic areas. Awareness and education, good history, high index of suspicion, and laboratory investigations will significantly aid the diagnosis of leishmaniasis, particularly in nonendemic regions.
Ethical approval and declaration of patient consent
Ethical approval (ADM/22/A/VOL.VII/14743031) was obtained from University of Benin Teaching Hospital (UBTH) Human Research Ethics Committee on 3rd July 2019. The authors certify that they have obtained all appropriate patient consent forms. In the form, the patient has given his consent for his images and other clinical information to be reported in the journal. The patient understands that his name and initial will not be published, and due efforts will be made to conceal identity, but anonymity cannot be guaranteed.
Financial support and sponsorship
Conflicts of interest
There are no conflicts of interest.
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[Figure 1], [Figure 2], [Figure 3], [Figure 4]