Fever with perinasal and tongue lesions: A diagnostic challenge.

The diagnosis may be challenging, and high suspicion index should be maintained in immunosuppressed patients with unusual mucocutaneous lesions, even in non-endemic areas for mucocutaneous leishmaniasis.

Leishmania RNA virus-1 is similarly detected among metastatic and non-metastatic phenotypes in a prospective cohort of American Tegumentary Leishmaniasis.

American Tegumentary Leishmaniasis (ATL) is an endemic and neglected disease of South America. Here, mucosal leishmaniasis (ML) disproportionately affects up to 20% of subjects with current or previous localised cutaneous leishmaniasis (LCL). Preclinical and clinical reports have implicated the Leishmania RNA virus-1 (LRV1) as a possible determinant of progression to ML and other severe manifestations such as extensive cutaneous and mucosal disease and treatment failure and relapse. However, these associations were not consistently found in other observational studies and are exclusively based on cross-sectional designs. In the present study, 56 subjects with confirmed ATL were assessed and followed out for 24-months post-treatment. Lesion biopsy specimens were processed for molecular detection and quantification of Leishmania parasites, species identification, and LRV1 detection. Among individuals presenting LRV1 positive lesions, 40% harboured metastatic phenotypes; comparatively 58.1% of patients with LRV1 negative lesions harboured metastatic phenotypes (p = 0.299). We found treatment failure (p = 0.575) and frequency of severe metastatic phenotypes (p = 0.667) to be similarly independent of the LRV1. Parasite loads did not differ according to the LRV1 status (p = 0.330), nor did Leishmanin skin induration size (p = 0.907) or histopathologic patterns (p = 0.780). This study did not find clinical, parasitological, or immunological evidence supporting the hypothesis that LRV1 is a significant determinant of the pathobiology of ATL.

Tongue Nodule as Primary Manifestation of American Cutaneous Leishmaniasis in an Immunocompetent Patient.

Leishmaniasis is a parasitic disease considered an endemic public health problem in developing countries, where it is a reportable disease. Isolated oral manifestation is rare, and its clinical manifestations are variable. In this paper we describe an unusual case of an immunocompetent patient, 57-year-old man with a painless reddish submucosal nodule located on the tongue dorsum. Microscopical analysis showed chronic inflammatory infiltrate with macrophages containing leishmania in cytoplasmic vacuoles. PCR assays confirmed the diagnosis and patient was treated with meglumine antimoniate for 30 days. Absence of the parasite was confirmed by PCR. Thirteen years after treatment, a scar fibrosis persisted on the tongue dorsum. The case reported reveals that leishmaniasis should be considered in the diagnosis of tongue nodules in immunocompetent patients.

Case Report: A Single-Center Case Series on Skin Manifestations of Leishmaniasis from a Non-Endemic State in Southern India.

Leishmaniasis is endemic in the Indian subcontinent with predominance of visceral leishmaniasis (VL) due to Leishmania donovani. Cutaneous leishmaniasis (CL) is uncommon, and mucocutaneous leishmaniasis (MCL) is rarely reported in this region. Recent reports reveal a changing epidemiology and atypical manifestations. A retrospective study of 52 suspected cases with cutaneous and mucosal involvement seen from January 2008 to December 2018 in a tertiary care setting in a non-endemic state in southern India is reported. Twelve patients were confirmed to have leishmaniasis; seven had MCL, two had CL, and three had post-kala-azar dermal leishmaniasis (PKDL). All cases were male, with a median age of 41.5 years (interquartile range, 30-55.5 years), and the median duration of the disease was 6 years (interquartile range, 1-9.5 years). Patients with MCL had mucosal involvement including destructive ulcero-proliferative lesions due to delayed diagnosis; none had a history of travel to countries endemic for MCL and all were attributable to L. donovani species. On the other hand, Leishmania major which was the causative species in both CL patients was associated with travel to the Middle East. Patients with PKDL presented with multiple plaques and hypopigmented patches; one had concomitant VL and all were from endemic areas. Hitherto uncommon MCL, caused by potentially atypical variants of L. donovani, has emerged as a new manifestation of leishmaniasis in this region. A high index of suspicion based on lesions seen and history of travel combined with PCR-based diagnostics are required to confirm diagnosis for the various skin manifestations of leishmaniasis.

Case Report: Unusual Presentation of Pharyngeal Mucosal Leishmaniasis due to Leishmania (Viannia) braziliensis.

Mucosal leishmaniasis (ML) affects predominantly the nose and occurs usually weeks or months after the cure of the primary cutaneous lesion. The pathology of ML is characterized by an exaggerated inflammatory reaction with infiltration of lymphocytes, macrophages, and plasma cells. There is also a paucity of parasites and a strong delayed-type hypersensitivity reaction. Herein, we report a case of a young man who had a large ulcer in his left leg and complained of dysphagia. In nasofibrolaryngoscopy, there were nodular lesions in the oropharynx and rhinopharynx. The skin lesion biopsy showed a chronic inflammation with amastigotes inside macrophages, and DNA of Leishmania braziliensis confirmed the diagnosis of ML in tissue biopsied from the pharynx. The leishmaniasis skin test was negative. Cytokine evaluation showed lack of production of interferon (IFN)-γ, interleukin (IL)-1ß, and IL-17 with enhancement of these cytokine levels after cure.

Case Report: Mucosal Leishmaniasis in New York City.

The six previously reported civilian cases of mucosal leishmaniasis (ML) diagnosed in the United States have all represented imported New World ML. We describe two new patients with ML diagnosed in New York City-a Syrian immigrant with a nasal mass (Leishmania tropica), the first report of Old World ML in the United States, and an American ecologist who worked in Bolivia and had been treated for cutaneous infection 23 years before developing lesions (L. (Viannia) braziliensis) initially of the uvula, soft palate, and posterior pharynx and subsequently the larynx.

Cutaneous and mucocutaneous leishmaniasis: experience of a Mediterranean hospital.

BACKGROUND: Leishmaniasis, considered by the World Health Organization as one of the most important tropical diseases, is endemic in the Mediterranean Basin. The aim of this study was to evaluate epidemiological and clinical characteristics of cutaneous (CL) and mucocutaneous leishmaniasis (MCL) in La Fe University Hospital, Valencia, Spain. The particular focus was on diagnosis techniques and clinical differences according to the immunological status of the patients. METHODS: An eleven-year retrospective observational study of CL and MCL episodes at the hospital was performed. Epidemiological, clinical and therapeutic variables of each case, together with the microbiological and anatomopathological diagnosis, were analyzed. RESULTS: A total of 42 patients were included, 30 of them were male and 28 were immunocompetent. Most of the cases (36/42) were diagnosed in the last 5 years (2013-2017). The incidence of CL and MCL increased from 3.6/100,000 (2006-2012) to 13.58/100,000 (2013-2017). The majority of the patients (37/42) exhibited CL, in 30 cases as single lesions (30/37). Ulcerative lesions were more common in immunosuppressed patients (13/14) than in immunocompetent patients (20/28), (P = 0.2302). The length of lesion presence before diagnosis was 7.36 ± 6.72 months in immunocompetent patients and 8.79 ± 6.9 months in immunosuppressed patients (P = 0.1863). Leishmania DNA detection (92.3%) was the most sensitive diagnostic technique followed by Giemsa stain (65%) and histopathological examination (53.8%). Twelve patients (12/42) had close contact with dogs or were living near to kennels, and 10 of them did not present underlying conditions. Intralesional glucantime (21/42) and liposomal amphotericin B (7/42) were the most common treatments administered in monotherapy. All patients evolved successfully and no relapse was reported. CONCLUSIONS: Some interesting clinical and epidemiological differences were found in our series between immunocompetent and immunosuppressed patients. Future studies can take these results further especially by studying patients with biological therapy. Skin biopsies combining NAAT with histological techniques are the most productive techniques for CL or MCL diagnosis.

Case Report: Mucocutaneous Leishmaniasis Masquerading as Idiopathic Midline Granulomatous Disease.

Mucocutaneous leishmaniasis (MCL) is a rare infection caused by several species within the genus Leishmania. We present a patient with multifocal MCL masquerading as idiopathic midline granulomatous disease, featuring the unusual complication of ocular leishmaniasis, as a result of prolonged immunosuppressive therapy. We review clinical features, diagnosis, and treatment of this syndrome.

Mucosal leishmaniasis: A forgotten disease, description and identification of species in 50 Colombian cases / Mucosal leishmaniasis: A forgotten disease, description and identification of species in 50 Colombian cases

INTRODUCTION: Mucosal leishmaniasis has a progressive course and can cause deformity and even mutilation in the affected areas. It is endemic in the American continent and it is mainly caused by Leishmania (Viannia) braziliensis. OBJECTIVE: To describe a series of mucosal leishmaniasis cases and the infectious Leishmania species. MATERIALS AND METHODS: We included 50 patients with a clinical diagnosis of mucosal leishmaniasis and parasitological confirmation, and we described their clinical and laboratory results. We performed species typing by PCR-RFLP using the miniexon sequence and hsp70 genes; confirmation was done by sequencing. RESULTS: The median time of disease evolution was 2.9 years (range: 1 month to 16 years). The relevant clinical findings included mucosal infiltration (94%), cutaneous leishmaniasis scar (74%), total loss of the nasal septum (24%), nasal deformity (22%), and mucosal ulceration (38%). The symptoms reported included nasal obstruction (90%), epistaxis (72%), rhinorrhea (72%), dysphonia (28%), dysphagia (18%), and nasal pruritus (34%). The histopathological study revealed a pattern compatible with leishmaniasis in 86% of the biopsies, and amastigotes were identified in 14% of them. The Montenegro skin test was positive in 86% of patients, immunofluorescence in 84%, and culture in 8%. Leishmania (V.) braziliensis was identified in 88% of the samples, L. (V) panamensis in 8%, and L. (V.) guyanensis and L. (L.) amazonensis in 2% respectively. CONCLUSION: In this study, we found a severe nasal disease with destruction and deformity of the nasal septum in 25% of the cases, probably associated with late diagnosis. Leishmania (V.) braziliensis was the predominant species. We described a case of mucosal leishmaniasis in Colombia caused by L. (L.) amazonensis for the first time.

Introducción. La leishmaniasis mucosa tiene un curso progresivo y puede causar deformidad e incluso mutilación de las zonas afectadas. Es endémica en el continente americano y es causada principalmente por Leishmania (Viannia) brasiliensis. Objetivo. Describir una serie de casos de leishmaniasis mucosa y las especies de Leishmania infecciosas. Materiales y métodos. Se estudiaron 50 pacientes con diagnóstico clínico de leishmaniasis mucosa y confirmación parasitológica. Se describieron sus características clínicas y los resultados de laboratorio. La tipificación de especies se hizo mediante reacción en cadena de la polimerasa de los polimorfismos de la longitud de los fragmentos de restricción (Restriction Fragment Length Polymorphism Polymerase Chain Reaction, PCR-RFLP) en la secuencia del miniexon y el gen hsp70 y se confirmó por secuenciación. Resultados. La evolución de la enfermedad fue de un mes a dieciséis años (mediana de 2,8 años). Los hallazgos clínicos fueron los siguientes: infiltración mucosa (94 %), cicatriz de leishmaniasis cutánea (74 %), pérdida total del tabique nasal (24 %), deformidad nasal (22 %) y ulceración (38 %). Los síntomas reportados fueron: obstrucción nasal (90 %), epistaxis (72 %), rinorrea (72 %), disfonía (28 %), disfagia (18 %) y prurito nasal (34 %). La histopatología mostró un patrón compatible con leishmaniasis en 86 % de las biopsias y se identificaron amastigotes en 14 % de ellas. La prueba de Montenegro fue positiva en 86 % de los pacientes, la inmunofluorescencia en 84 %, y el cultivo en 8 %. Leishmania (V.) brasiliensis se identificó en 88 % de las muestras, L. (V) panamensis en 8 %, y L. (V.) guyanensis y L. (L.) amazonensis en 2 %, respectivamente. Conclusión. Se encontró enfermedad nasal grave con destrucción y deformidad del tabique nasal en una cuarta parte de los casos, probablemente debido a un diagnóstico tardío. Leishmania (V.) brasiliensis fue la especie predominante. Se describe por primera vez un caso de leishmaniasis mucosa causado por L. (L.) amazonensis en Colombia.

First Molecular Report of Leishmania (Leishmania) amazonensis and Leishmania (Viannia) guyanensis in Paraguayan Inhabitants Using High-Resolution Melt-PCR.

American tegumentary leishmaniasis is an endemic anthropozoonosis undergoing expansion on the American continent. The disease is caused by several Leishmania species and it is manifested as cutaneous and mucocutaneous leishmaniasis. In this study, we evaluate the viability of high-resolution melt polymerase chain reaction (HRM-PCR) analysis to differentiate four closely related Leishmania species as a routine tool for the diagnosis of leishmaniasis. For this purpose, biopsy specimens from cutaneous and mucocutaneous lesions were taken from 132 individuals from endemic and non-endemic areas for leishmaniasis. Each sample was processed for parasitological, histopathological, and molecular analysis. Positive biopsy samples were analyzed by HRM-PCR of a 144-bp heat-shock protein (hsp70) gene fragment, and new cases were confirmed by sequencing. Of the 132 samples analyzed, 36 (27%) were positive for Leishmania spp., of which 86% were from cutaneous lesions and 14% from mucocutaneous lesions. We identified Leishmania (Viannia) braziliensis (84%), Leishmania (Leishmania) infantum (13%), and Leishmania (Leishmania) amazonensis (3%) in cutaneous lesions, and L. (V.) braziliensis (40%), L. (L.) infantum (20%), L. (L.) amazonensis (20%), and Leishmania (Viannia) guyanensis (20%) in mucocutaneous lesions. The main purpose of this research was to report for the first time in Paraguay the presence of L. (L.) amazonensis and L. (V.) guyanensis in patients with cutaneous and mucocutaneous lesions, using the HRM-PCR technique. In addition, we report the presence of additional new cases of L. (L.) infantum in cutaneous lesions.

Mucosal leishmaniasis: A forgotten disease, description and identification of species in 50 Colombian cases / Leishmaniasis mucosa: una enfermedad olvidada, descripción e identificación de especies en 50 casos colombianos

Abstract Introduction: Mucosal leishmaniasis has a progressive course and can cause deformity and even mutilation in the affected areas. It is endemic in the American continent and it is mainly caused by Leishmania (Viannia) braziliensis. Objective: To describe a series of mucosal leishmaniasis cases and the infectious Leishmania species. Materials and methods: We included 50 patients with a clinical diagnosis of mucosal leishmaniasis and parasitological confirmation, and we described their clinical and laboratory results. We performed species typing by PCR-RFLP using the miniexon sequence and hsp70 genes; confirmation was done by sequencing. Results: The median time of disease evolution was 2.9 years (range: 1 month to 16 years). The relevant clinical findings included mucosal infiltration (94%), cutaneous leishmaniasis scar (74%), total loss of the nasal septum (24%), nasal deformity (22%), and mucosal ulceration (38%). The symptoms reported included nasal obstruction (90%), epistaxis (72%), rhinorrhea (72%), dysphonia (28%), dysphagia (18%), and nasal pruritus (34%). The histopathological study revealed a pattern compatible with leishmaniasis in 86% of the biopsies, and amastigotes were identified in 14% of them. The Montenegro skin test was positive in 86% of patients, immunofluorescence in 84%, and culture in 8%. Leishmania (V.) braziliensis was identified in 88% of the samples, L. (V) panamensis in 8%, and L. (V.) guyanensis and L. (L.) amazonensis in 2% respectively. Conclusion: In this study, we found a severe nasal disease with destruction and deformity of the nasal septum in 25% of the cases, probably associated with late diagnosis. Leishmania (V.) braziliensis was the predominant species. We described a case of mucosal leishmaniasis in Colombia caused by L. (L.) amazonensis for the first time.

Resumen Introducción. La leishmaniasis mucosa tiene un curso progresivo y puede causar deformidad e incluso mutilación de las zonas afectadas. Es endémica en el continente americano y es causada principalmente por Leishmania (Viannia) brasiliensis. Objetivo. Describir una serie de casos de leishmaniasis mucosa y las especies de Leishmania infecciosas. Materiales y métodos. Se estudiaron 50 pacientes con diagnóstico clínico de leishmaniasis mucosa y confirmación parasitológica. Se describieron sus características clínicas y los resultados de laboratorio. La tipificación de especies se hizo mediante reacción en cadena de la polimerasa de los polimorfismos de la longitud de los fragmentos de restricción (Restriction Fragment Length Polymorphism Polymerase Chain Reaction, PCR-RFLP) en la secuencia del miniexon y el gen hsp70 y se confirmó por secuenciación. Resultados. La evolución de la enfermedad fue de un mes a dieciséis años (mediana de 2,8 años). Los hallazgos clínicos fueron los siguientes: infiltración mucosa (94 %), cicatriz de leishmaniasis cutánea (74 %), pérdida total del tabique nasal (24 %), deformidad nasal (22 %) y ulceración (38 %). Los síntomas reportados fueron: obstrucción nasal (90 %), epistaxis (72 %), rinorrea (72 %), disfonía (28 %), disfagia (18 %) y prurito nasal (34 %). La histopatología mostró un patrón compatible con leishmaniasis en 86 % de las biopsias y se identificaron amastigotes en 14 % de ellas. La prueba de Montenegro fue positiva en 86 % de los pacientes, la inmunofluorescencia en 84 %, y el cultivo en 8 %. Leishmania (V.) brasiliensis se identificó en 88 % de las muestras, L. (V) panamensis en 8 %, y L. (V.) guyanensis y L. (L.) amazonensis en 2 %, respectivamente. Conclusión. Se encontró enfermedad nasal grave con destrucción y deformidad del tabique nasal en una cuarta parte de los casos, probablemente debido a un diagnóstico tardío. Leishmania (V.) brasiliensis fue la especie predominante. Se describe por primera vez un caso de leishmaniasis mucosa causado por L. (L.) amazonensis en Colombia.

Differentiation of mucosal lesions in mucocutaneous leishmaniasis and paracoccidioidomycosis.

Mucocutaneous leishmaniasis and paracoccidioidomycosis are infectious diseases with similar epidemiological and clinical aspects. Cases of both diseases may manifest similar lesions in the mucosa. Therefore, the determination of distinguishing characteristics for the purpose of differential diagnosis is critical for better management of the diseases. The present study evaluated factors that assist in the differentiation of mucosal lesions between these diseases. This cross-sectional study included data from medical records of 122 cases of mucocutaneous leishmaniasis and 83 cases of paracoccidioidomycosis attended at the university hospital Cassiano Antonio Moraes, located in Vitória, Espírito Santo State, Brazil. Comparison between the diseases included the following variables: sex, age, time of disease evolution, location of the lesion and symptoms. Adults and males were affected by both diseases at higher rates. Lesions in the nasal region (95.1%; p-value = 0.000) and the pharynx (20.5%; p-value = 0.009) and nasal obstruction (34.4%; p-value = 0.000) were associated with leishmaniasis. Paracoccidioidomycosis was associated with lesions in the oral region (90.4%; p-value = 0.000), oral pain (16.9%; p-value = 0.000), and hoarseness (14.5%; p-value = 0.008). In leishmaniasis, lesions in oral regions were not associated with oral pain and were frequently located close to the nasal area. The manifestations cited above could improve the differential diagnosis of leishmaniasis and paracoccidioidomycosis, and thereby potentially aid in the choice of appropriate confirmatory diagnostic testing.

Raro compromiso lingual de leishmaniasis mucocutánea por Leishmania perteneciente al subgénero Viannia / Rare Tongue Compromising of Mucocutaneous Leishmaniasis by Leishmania Subgenus Viannia

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[Atypical mycocutaneous manifestation of Leishmania tropica in a refugee].

In this case report a male 19-year-old Syrian refugee presented with a sore throat. A biopsy from larynx detected Leishmania tropica compatible with leishmaniasis, although L. tropica does not normally cause mucosal leishmaniasis (CL). An immunodeficiency was detected, and the patient was treated for hypogammaglobulinaemia and CL three times, before the symptoms disappeared. Leishmaniasis is a disease, which should be taken into consideration, when refugees present with atypical clinical manifestations, especially in immunosuppressed patients.

Leprosy and American cutaneous leishmaniasis coinfection.

Brazil is a country with a high prevalence of infectious diseases such as leprosy and leishmaniasis. However, coinfection of these diseases is still poorly understood. We report a case of a patient who presented with lepromatous leprosy and cutaneous-mucosal leishmaniasis at the same period. After clinical, laboratory, and histopathological diagnosis, the treatment was introduced and the patient showed important clinical improvement. He was followed in our outpatient clinic. Both pathologies play an important role in the immune system. Depending on the immune response profile of the host, diseases may present themselves in different ways. In this case, the patient showed a divergent immune response for each disease. We hypothesized that this response is specific for each pathogen.

Leishmaniasis mucocutánea en paciente inmunocompetente: a propósito de un caso / Mucocutaneous leishmaniasis in an immunocompetent patient: a case report

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