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ISSN: Print -2349-0977, Online - 2349-4387

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Year : 2017  |  Volume : 4  |  Issue : 3  |  Page : 154-158

Cutaneous Leishmaniasis in Nonendemic Geographic Areas: Unraveling the Itinerant Cases

1 Department of Dermatology, Command Hospital Air Force, Bengaluru, Karnataka, India
2 Department of Dermatology, Armed Forces Medical College, Pune, Maharashtra, India
3 Mehektagul Dermaclinic, New Delhi, India
4 Department of Pathology, Base Hospital, Delhi Cantt, New Delhi, India
5 Department of Dermatology, Military Hospital, Binnaguri, West Bengal, India

Date of Web Publication30-Jan-2018

Correspondence Address:
Sandeep Arora
Department of Dermatology, Command Hospital Air Force, Bengaluru - 560 007, Karnataka
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Source of Support: None, Conflict of Interest: None

DOI: 10.4103/astrocyte.astrocyte_41_17

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Background: Cutaneous leishmaniasis endemic in 70 countries worldwide and in certain regions of India is otherwise an uncommon presentation in non-endemic zones, leading to their missed diagnosis. Movement of people between these regions further exposes them to the risk of this infection. The aim of this study was to analyze the presentation of these missed cases, basis of their final diagnosis, management and follow up with the objective of highlighting cutaneous leishmaniasis as an important differential diagnosis in these cases. Methods: A prospective 4-year cohort of patients who were diagnosed as cutaneous leishmaniasis (CL) and post kala azar dermal leishmaniasis (PKDL) between 2012 and 2016 were studied. Their presentation, basis of diagnosis, treatment response to different agents and follow up was studied. Results: Of a total of 380547 patients seen in the four-year period, 15 CL and 2 PKDL symptomatic from 6 months to 7 years were diagnosed at our centre. Leishmania amastigotes were seen in smears in 10 and 4 in skin biopsy. Four were diagnosed based on polymerase chain reaction for Leishmania. Ten were managed with iv Sodium Stibogluconate (SSG), five with intralesional SSG and one with liposomal amphotericin B. All responded to treatment and were followed up for one year thereafter. Conclusion: Although CL and PKDL are rare outside their endemic zones, travelers and population exposed to newer endemic zones may result in atypical presentations which clinicians are not experienced with. This study highlights this possibility and our experience in diagnosing and treating such cases.

Keywords: Cutaneous leishmaniasis, granulomatous, leishmania donovan body, post kala azar dermal leishmaniasis, sodium stibogluconate

How to cite this article:
Arora S, Sood A, Rajeshwari, Baveja S, Arora G, Malik A, Mishra R. Cutaneous Leishmaniasis in Nonendemic Geographic Areas: Unraveling the Itinerant Cases. Astrocyte 2017;4:154-8

How to cite this URL:
Arora S, Sood A, Rajeshwari, Baveja S, Arora G, Malik A, Mishra R. Cutaneous Leishmaniasis in Nonendemic Geographic Areas: Unraveling the Itinerant Cases. Astrocyte [serial online] 2017 [cited 2021 Nov 30];4:154-8. Available from: http://www.astrocyte.in/text.asp?2017/4/3/154/224195

  Introduction Top

Cutaneous leishmaniasis (CL), a protozoal infection caused by Leishmania spp. and transmitted by sandfly, is endemic in 70 countries worldwide with majority occurring in Afghanistan, Algeria, Brazil, Pakistan, Peru, Saudi Arabia, and Syria.[1] In India, the well-known endemic zones are Rajasthan deserts and certain parts of Gangetic plains including the states of Punjab, Himachal Pradesh, National Capital Region, and parts of Uttar Pradesh; however, new endemic zones are being reported within and outside these regions.[2],[3],[4] Relative rarity of presentation in presumably nonendemic areas may lead to misdiagnosis.[5],[6]

Globally, there are an estimated 1.5–2 million new cases, 70000 deaths each year, and 350 million people are at a risk of infection and disease.[1] Morbidity because of leishmaniasis results in an estimated 2.4 million disability-adjusted life-years.

Typically, CL presents with a painless papule which increases in size over few weeks to a larger nodule or plaque, undergoes subsequent ulceration, and heals with scarring over the next few months in case of Leishmania majorand approximately a year in Leishmania tropicainfections.[7] Post kala azar dermal leishmaniasis (PKDL), a dermal sequela of visceral leishmaniasis, reportedly affects 5–15% of visceral leishmaniasis (VL) cases usually after a few years in Indian patients even in absence of documented VL.[8] Early and correct diagnosis may reduce the disease reservoir existing in untreated cases.[9] Atypical presentations and relative rarity of CL and PKDL in outpatient departments often leads to missed or delayed diagnosis.[10] We reviewed 17 cases of CL and PKDL which were managed for 6 months to 7 years as different conditions till their final diagnosis.

  Materials and Methods Top

All cases of CL and PKDL diagnosed at a tertiary care centre from June 2012 to Jun 2016 were analyzed for their mode of presentation, past treatment, progression of disease, and remission and relapses, if any, during their management. A detailed general, systemic, and dermatological examination was performed. All cases were investigated with a standard hemogram, biochemistry for liver and renal function, blood sugar, enzyme-linked immunosorbent assay (ELISA) for HIV, immunochromatography test for rK39, slit skin smear, and skin biopsy. Mantoux test, chest radiograph, and ultrasound abdomen were done if indicated in view of multiple clinical suspicions in past management. Polymerase chain reaction (PCR) for Leishmania was done if the skin smears and biopsies were negative for L. amastigotes. Diagnosis of CL/PKDL was made if L. amastigotes were demonstrated in skin smears or skin biopsies, rK39 positivity, or presence of a positive PCR for Leishmania.

All cases with larger lesions (>5 cm) and longer duration of disease (>6 months) were treated with intravenous inj. sodium stibogluconate (SSG) 850 mg intravenous daily for 4 weeks, rest with intralesional SSG (100 mg/ml, 50 mg/cm 2 till blanching at injection site, weekly for six doses).[11],[12] Cases of PKDL were managed with inj. SSG 850 mg intravenous daily for 20 days a month for 4 months. Liposomal amphotericin B (3 mg/kg infusion on days 1–7, 14, and 21) was used when SSG was not available. Those managed with intralesional SSG were administered topical 5% imiquimod cream thrice a week if resolution was delayed (>4 weeks). All patients underwent hematological and biochemical screening on the third day and weekly thereafter for 4 weeks and thereafter every 4 weeks after treatment while on follow-up.

  Results Top

A total of 17 cases of leishmaniasis were seen in 380547 patients examined over the 4-year period [Table 1]. Of these, 15 cases were of CL and 2 of PKDL. Of the CL cases, only 2 reported directly to our centre in absence of any treatment in the past and the other 13 cases of CL and 2 PKDL cases were either referred from other hospitals or reported on their own following a poor response to the treatment received. The youngest patient was 19 years old and oldest 50 years old with a mean age of 36 years. Males accounted for 9 cases and females 8 cases. Duration of symptoms varied from 2 months to 7 years. Place of residence of 4 cases was Rajasthan, 3 cases each from Jammu and Kashmir (Jammu district) and National Capital Region (Delhi). Only 3 occurred in active military personnel while the rest occurred in their dependants or veterans. Of these, two resided in the recently described new endemic zone in the state of Jammu and Kashmir and one at Delhi for a minimum of 1 year.
Table 1: Case profile and management

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Those who reported directly were symptomatic for a few months whereas others reported later. Among these, 4 had received 6 months of multidrug therapy for Hansen's disease and 2 were treated as discoid lupus erythematosus for their solitary crusted plaques with hydroxychloroquine and systemic steroids (one manifested on nose and other on the malar area). Two cases were managed for lupus vulgaris as well as deep fungal infection while 1 was treated for both lupus vulgaris and Hansen's disease and other 2 cases for deep fungal infections. Two cases received multiple doses of antibiotics on the presumption of having a chronic bacterial infection. One case was managed for sarcoidosis and another was referred from a surgical centre for evaluation prior to surgery.

All except one case of PKDL had lesions on face, neck, and/or scalp. This case of PKDL presented with lesions on the trunk and extremities.

Nose was affected with solitary nonresolving crusted or ulcerated plaque in 7 cases, supraorbital region in 3 cases, multiple lesions on the face, neck, and ear in 2 cases, malar area in 1 case, 1 with scalp and face lesions, and 2 cases with perioral involvement [Figure 1]. Slit skin smears were positive for L. amastigotes in 10 cases, of which 4 cases revealed them on skin biopsies too. Skin biopsies revealed dense mixed dermal inflammatory infiltrate predominantly of histiocytes, lymphocytes, and plasma cells with occasional giant cells and neutrophilic infiltrate in areas of ulceration with 10 cases showing epithelioid granulomas [Figure 2]. Three cases of epithelioid granulomas on histopathology had a few LD bodies which is uncommon. However, these were noted with great difficulty after repeated biopsies (SSS negative). Inconclusive SSS and skin biopsies in 4 cases led us to perform PCR for Leishmania, which was positive. All cases were negative for acid fast bacilli and fungal elements. Mantoux was negative in all and chest radiographs and ultrasound scan abdomen were normal. All cases were HIV negative.
Figure 1: Clinical photographs showing a varied presentation of Cutaneous Leishmaniasis on the face.

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Figure 2: Photomicrograph showing diffuse dermal infiltrate with macrophages, polymorphs and LD bodies, H&E Original magnification 1000X (left), Early granulomatous infiltrate with isolated LD bodies, H&E Original magnification 1000X (right).

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Ten cases were managed with intravenous SSG, 5 with intralesional SSG, and 1 with liposomal amphotericin B. Two cases who had a partial response to intralesional SSG were further treated with topical imiquimod daily application with resolution over the next 6 weeks.

Those managed with intravenous SSG were in addition investigated for levels of serum amylase and creatinine phosphokinase. Mild intolerance with muscle pains and mild elevation of renal parameters were observed in all those treated with SSG. Pain was the main side effect with intralesional SSG. All treated cases had complete resolution of the disease and were followed up for a minimum 6 months post treatment.

  Discussion Top

CL is endemic in tropics.[2],[3],[4] Series presented here are cases from known, new, as well as nonendemic regions in India.

Face and extremities are most commonly involved as noted in this study.[13] This series presents cases reporting as early as a few months up to 7 years after onset. Forme fruste of leishmaniasis not only confuses the clinical picture but its histopathological variability may also result in delayed diagnosis.[14],[15] The possibility of missed diagnosis in CL rises to one-third if the patient reports to a non-dermatologist, especially so in areas of low endemicity as reported by Hajj et al.[16] Our cases' diagnosis was missed in their primary management even when they resided in endemic zones but reported to primary physicians. Aara et al. in a large Indian study in the endemic area of Bikaner reported a mean duration of 4 months till a patient of CL reported to the dermatologist.[17] Nearly one-fourth cases of PKDL as reported by Ramesh et al. are missed when they initially report to primary health centres and some have been diagnosed as late as 17 years after becoming symptomatic.[18],[19] One of our PKDL case was diagnosed after 7 years. Military personnel owing to their nature of job may acquire the disease in an endemic zone and report to a primary physician or dermatologist in a nonendemic zone.[20],[21] In a series of CL in Sri Lanka, one-third cases belonged to the army.[22] In this series, only 3 were personnel on active military duty, 2 serving in Jammu district and 1 at Delhi.

Diagnosis of CL/PKDL is based on clinical suspicion, and supported by demonstration of Leishmania parasite on skin smears, skin biopsies, or PCR. Early lesions show features of diffuse inflammatory infiltrate whereas well-defined epithelioid granulomas are seen later in the disease.[23] We had a large number of cases with epithelioid granulomas which only confirms the variability in histopathological presentation of this infection. Finding LD bodies with epithelioid granulomas is rare but in 3 of our cases it formed the basis of diagnosis despite negative SSS.[23] Facilities for immunochromatography for rk39 and PCR for this parasite are constrained by their availability at limited number of research centres. Higher chances of demonstration of parasite in smears versus biopsy specimens has been reported variably in literature with a greater number of studies favoring histopathology.[24],[25]

Guidelines for treatment favor species dependent treatment and systemic management with SSG as firstline therapy despite increasing incidence of resistance.[11],[26] Paromomycin cream or intralesional SSG in smaller lesions presenting early preferably not on the face and in absence of scarring are preferred options. For the rest, intravenous SSG or liposomal amphotericin B is used. Intolerance in a patient or availability of a drug may lead to an institutional preference deciding between the two. Azoles have been reported to be an effective alternative, however two of our cases were treated with itraconazole on suspicion of a deep fungal infection for 4 months with no response. Miltefosine in PKDL may not be as effective as initially reported.[27] Topical Imiquimod is reportedly more effective when used in conjunction with intralesional SSG.[28] Our two cases were placed on topical imiquimod after their initial management with intralesional SSG with a favorable response, proving it to be a good adjuvant.

This study represents young adults/individuals residing in rural or semiurban areas and military personnel possibly exposed to the parasite in their surroundings which were variably diagnosed prior to their final diagnosis of CL/PKDL. Case history revealed either an absence of consideration of this diagnosis or an inability to demonstrate the parasite. None had been investigated with PCR for Leishmania or rk39, possibly because of its nonavailability in their rural and semiurban healthcare setting. Hence, clinical suspicion is paramount even if the condition is otherwise considered rare, followed by a search for the parasite. In a majority of cases (13 of 17 cases), we could demonstrate the parasite. PCR for Leishmania which is otherwise not available in peripheral healthcare as well as many urban centres confirmed the diagnosis in 4 of our cases. Species identification reports are not available with us.

This article aims to highlight the importance of clinical suspicion in diagnosing CL. Dermatologists and primary care physicians must be alert regarding this diagnosis as even though CL is rarely fatal it may cause considerable morbidity with widespread involvement, disfigurement, and scarring.

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Conflicts of interest

There are no conflicts of interest.

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  [Figure 1], [Figure 2]

  [Table 1]

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[Pubmed] | [DOI]


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