2023.08.03.23

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Cutaneous Leishmaniasis. A case report.

Ramos Escobedo Lhianella. ¹, Cando Endara Karen Lisbeth ², Barrazueta Serrano Wendy Karina³, Jaramillo Amaya Silvio David⁴, Cabrera Rodriguez Hermes Miguel.⁵

¹ Universidad Central del Ecuador / Quito / Ecuador); [email protected] .

²Universidad Central del Ecuador / Quito / Ecuador); [email protected] .

³ Universidad Central del Ecuador / Quito / Ecuador); [email protected] .

⁴ Universidad Central del Ecuador/ Quito / Ecuador); [email protected] .

⁵ Hospital Luis G. Dávila / Tulcán / Ecuador); [email protected] . ORCID (0009-0004-1231-2869);

* Correspondence: Ramos L.; [email protected]; Tel.: +593 97 869 7025

Available from: http://dx.doi.org/10.21931/RB/2023.08.03.23

ABSTRACT

This article's uniqueness focuses on diagnosing Leishmaniasis, with particular attention to clinical manifestations and demographic factors when skin smear microscopy reveals a negative result and molecular tests are unavailable to the professional. The clinical case concerns an adult patient presenting a hyperkeratotic lesion in the right temporomalar region and another ulcerative lesion in the right atrial pavilion. A smear of the lesion was requested, resulting in a negative. However, two months later, new ulcerative lesions appeared, and a new smear was carried out with a positive result. Treatment with intramuscular Glucantime was started once daily for 21 days. When no improvement of ulcerated lesions in the temporomalar area and pinna was observed, the route of administration was modified by adding intralesional Glucantime, with evident improvement in the clinical state. It is relevant to consider that identifying demographic risk factors and the natural history of the lesions can be determining aspects for the diagnosis, even when the cytological study is negative. It is essential to ensure an adequate technique of taking the test to avoid false negatives and the correct administration of the chosen medication.

Keywords: Leishmaniasis; cutaneous, glucantime.

INTRODUCTION

Leishmaniasis is a parasitic disease transmitted by the bite of infected female sandflies. Its reservoir is animals and sometimes man. It is a neglected tropical disease in about 98 countries, mainly in tropical and subtropical regions. It is estimated that between 700,000 and 1 million new leishmaniasis cases occur worldwide each year. Ecuador is an endemic region of Leishmaniasis, especially in the coastal and Amazon provinces. Different species of Leishmania parasites have been identified in the country, such as L. tropica, L. amazonensis and L. panamensis. It can present in various clinical forms, including cutaneous, mucocutaneous and visceral Leishmaniasis, with cutaneous Leishmaniasis being the most common form in Ecuador. The National Directorate of Epidemiological Surveillance of Ecuador in the Gazette of Vector-Borne Diseases in 2018 reported 1336 cases throughout the country, of which 1302 are cutaneous and 34 are mucocutaneous. During the year 2019 (epidemiological weeks 1 to 32) 733 cases were reported, where 722 were cutaneous and 11 mucocutaneous type. ¹

The disease begins as localized areas of erythema where the bite occurred, which later evolves into a papule and progressively increases in size until forming dry or exudative central ulcerations with raised and defined borders, which they usually have. Hyperkeratosis may also sometimes form. These lesions are painless and are located mainly at the level of exposed regions. ²

The control of Leishmaniasis in Ecuador has been a challenge since the characteristics of the vectors associated with several socioeconomic, environmental and ecological factors, as well as the lack of resources, the difficulty in early diagnosis, the lack of access to effective treatments and the presence of animal reservoirs. However, prevention and control strategies, such as timely detection and treatment, vector control and health education, have been implemented to reduce the incidence of the disease.

CASE REPORT

A 34-year-old male patient self-identified as mestizo with no relevant personal and family clinical or surgical history. After a trip to a rural region of San Lorenzo on the Ecuadorian coast, he experiences an insect bite that triggers erythema and itchy papules at the right hemiface and pinna level. After 21 days, these lesions evolve into an ulcerative form associated with hyperkeratosis. He goes for medical attention for the first time, where the presumptive diagnosis is a bacterial infection of the skin and soft tissues, and they prescribe antibiotic therapy. However, the symptoms worsened, and he sought a new medical evaluation.

During the physical examination, it was observed that the patient was in good general condition and with an adequate nutritional status. Two lesions were identified: a hyperkeratotic lesion of approximately 2x2 cm in the right temporomalar region and the second lesion, of ulcerative type, in the helix of the ipsilateral pinna, with about 1x2 cm in size, painless, with irregular and raised borders, a deep base and granulomatous and exudative appearance associated with fetidity, areas of necrosis and hyperkeratosis. (Figure 1.) The remainder of the physical examination showed no significant findings.

The demographic factors identified in the patient, in addition to the lack of response to antibiotic therapy, led to the diagnostic suspicion of Leishmaniasis, so a smear of the lesion was requested for cytological study. However, the result was negative. After that, the patient interrupted the medical follow-up.

After approximately three months, new ulcerative lesions appeared in the right scapular and left mammary regions (Figure 2), so a new smear of the most recent lesion was requested and analyzed in a different laboratory, resulting in a positive for Leishmaniasis. Besides, a culture of the lesion was also carried out, isolating Kliebsella oxytoca, a bacteria sensitive to cephalexin, amikacin, cefazolin, ceftazidime, ceftriaxone, tigecycline; and ampicillin-resistant sulbactam, ciprofloxacin and gentamicin.

Figure 1. A) Hyperkeratotic lesion with irregular erythematous borders with necrotic tissue at the central level. B) Ulcerative lesion with clean base with raised and irregular edges. Necrotic tissue at the level of the lower border.

Figure 2. Ulcerative lesion with a clean base and defined edges not raised (white arrow)

In this way, the lack of follow-up of the patient, the presence of hyperkeratotic lesions, which are not frequently the classic lesions, the false-negative result reported by the laboratory at the onset of symptoms, and the lack of access to complementary molecular tests became a diagnostic challenge.

The differential diagnoses of Leishmaniasis included a bacterial infection, such as impetigo. However, the hyperkeratotic lesions did not present the characteristic of myelaceous crust typical of such a condition. The possibility of cutaneous malignancy, specifically squamous cell carcinoma, was also considered. Still, the lesions' evolution was short, and some appeared in regions without sun exposure. Another consideration was lichen simplex chronicus, but no history of friction or mechanical trauma was found at the site of the lesions. Clinical syndromes of cutaneous Leishmaniasis have a wide range of presentation, determined by clinical course, pharmacological options and response to treatment, so the prognosis is highly variable. Although many infections can resolve clinically without treatment, often treated patients do not experience clearance of the parasitic infection. In this case study, the benefits of treatment included accelerated healing of skin lesions and reduced risk of further spread of disease, with the long-term hope of reducing the likelihood of recurrence.

In the first medical contact, the patient was treated with amoxicillin + clavulanic acid without improvement of the lesions. However, once the diagnosis of cutaneous Leishmaniasis was confirmed, and after renal and hepatic function tests( Cr:0.80 mg/dL, CDK EPI 100.13 mL/min/1.73m², Urea: 20 mg/dL, , AST: 32 ALT: 30, Indirect Bilirubin: 0.2 mg/dl, Total Bilirubin: 0.7 mg/dl, GGT:10 U/L, Alkaline Phosphatase: 121 U/L, as well as an electrocardiogram(sinuses rhythm, heart rate 78 beats per minute, no ekg alterations was found), all with results within normal parameters: treatment was started with Glucantime 1500 mg / 5ml, administered intramuscularly at a dose of 5ml once a day for 21 days. When no improvement of the face and pinna ulcerative lesions was observed, the drug administration route was modified, adding intralesional Glucantime at a dose of 0.5 ml distributed by each lesion. Thus, after one month of intralesional treatment with Glucantime, a progressive remission of all lesions was observed, leaving only atrophic scars and post-inflammatory hyperpigmentation (Figure 3). Post-treatment renal and liver function tests were performed to ensure that meglumine antimoniate poisoning did not occur. The patient tolerated the treatment and demonstrated adequate adherence to it, no adverse reactions were reported until one month after finishing the treatment

Figure 3. Post-inflammatory lesions after 21 days of Glucantime administration (white arrow).

DISCUSION

Leishmaniasis in Ecuador is a public health problem that has persisted over the years, which allowed accumulating experience in diagnosing and treating the disease, so it was one of the first diagnoses to consider in this patient.

Regarding the weaknesses identified, it is essential to emphasize that a negative histological diagnosis does not exclude the diagnosis of Leishmania since it is essential to remember the variable sensitivity of this study and the influence of an inadequate technique of sample taking. In our study case a second sample of the patient analysed in in a different laboratory was needed, to establish the final diagnosis of cutaneous Leishmaniasis. Another factor to mention is that limited resources often prevent access to advanced diagnostic tests or optimal pharmacological treatments, limiting the ability to provide adequate care to patients with Leishmaniasis.

It is also important to highlight that a detailed demographic and epidemiological data history is relevant, including the patient's place of origin or travel history to endemic areas. The clinical disease manifestations are also transcendental for the diagnosis of cutaneous Leishmaniasis, which should be considered with evidence of an ulcer of evolution more fantastic than four weeks, macroscopically with violet edges, indurated and raised, not painful, based on granulomatous content. ³

The anamnesis and clinical findings should be complemented with laboratory tests. Direct smear of the lesion is usually the most frequently implemented laboratory method, especially in endemic regions, because it is an inexpensive and uncomplex test. Smear is a direct microscopic analysis of the specimen with Giemsa stain, in which amastigotes can be observed with positive results. The sample can be obtained by making an incision and scraping the active edge of the ulcer or by scraping the inner edge of it. Other diagnostic methods, such as biopsy or culture, may be considered. Due to the limited sensitivity of these tests, a combination of them is usually recommended. This must be associated with a correct sampling technique. ³^,⁴ Leishmaniasis can be genotyped by Polymerase Chain Reaction (PCR) techniques, a rapid diagnostic modality with high sensitivity and specificity, providing a specific treatment by identifying the Leishmania species.³

The patient treatment was based on the current recommendations of the Pan American Health Organization, which prescribed pentavalent antimonials intralesionally in patients with localized cutaneous Leishmaniasis caused by L. braziliensis or L. amazonensis. It is not mandatory to determine the species of Leishmania to establish pharmacological treatment, so choosing the most prevalent species at the regional level can guide the therapy while considering the patient's clinical status, the availability of the drug and the risk-benefit balance.⁵

In its latest report, the Pan American Health Organization establishes that in immunocompetent adult patients who can maintain follow-up, local treatments with pentavalent antimonials can be recommended when there are 1-3 lesions with dimensions up to 900 mm² located in any region except the head and periarticular areas, performing 3-5 infiltrations of 1-5 ml per lesion, covering it completely, at an interval between sessions of 3-7 days, with a maximum total volume of 15 ml per day. ⁶

On the other hand, systemic treatments are indicated in single or multiple lesions of more than 900 mm² in any body region, injuries of any size to the head or periarticular areas, or in patients previously treated locally with no response to treatment or with recurrences. Miltefosine 2.5 mg/kg/day, with a maximum dose of 150 mg/day, is recommended for 28 days. Splitting the doses and taking the drug after meals is suggested to reduce gastrointestinal side effects. Pentavalent antimonials may also be considered intramuscularly or intravenously at 20 mg/kg/day once daily for 20 days. ⁶

CONCLUSIONS

Identifying demographic factors and the lesson features are critical factors for diagnosing cutaneous Leishmaniasis, and in this case, prevailed against a negative cytological study. Before taking the sample, it is important to reinforce the appropriate technique to reduce the probability of a false negative, which allows this pathology to be underdiagnosed.

Our patient was unambiguously diagnosed with Leishmaniasis after the smear using Giemsa stain.

As there are various treatment alternatives, criteria such as the location and surface of the lesion and the response to the established treatment should be considered. In addition, it should be remembered that Leishmaniasis could be associated with overlapping bacterial infections; therefore, the joint performance of a culture should be considered.

Supplementary Materials: The following are available online at www.revistabionatura.com/xxx/s1, Image 1-3: Evolution of lesions pre and post-treatment

Author Contributions: Each author actively contributed to the preparation and writing of the paper and all authors have read and agreed to the published

Funding: This research received no external funding.

Institutional Review Board Statement: The study was conducted according to the guidelines of the Declaration of Helsinki.

Informed Consent Statement: Written informed consent has been obtained from the patient to publish this paper.

Data Availability Statement: Data Availability Statements in the "Bionatura Research Data Policies" section at https://www.revistabionatura.com/policies.html.

Conflicts of Interest: The authors declare no conflict of interest.

REFERENCES

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2. Obaldía A, Delgado E, Rocha S. Approach to cutaneous Leishmaniasis. Sinergia [Internet]. 2023; Available from: https://revistamedicasinergia.com/index.php/rms/article/view/985/2142

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Received: 28 May 2023/ Accepted: 15 July 2023 / Published:15 September 2023

Citation: Ramos L, Cando K, Barrazueta W, Jaramillo S, Cabrera H. Cutaneous Leishmaniasis. A case report. Revis Bionatura 2023;8 (3) 23. http://dx.doi.org/10.21931/RB/2023.08.03.23