Human Lobomycosis Caused by Paracoccidioides (Lacazia) loboi, Panama, 2022.

We report a patient from Panama who had lobomycosis caused by Paracoccidioides (Lacazia) loboi. We used combined clinical-epidemiologic and phylogenetic data, including a new gene sequence dataset on this fungus in Panama, for analysis. Findings contribute useful insights to limited knowledge of this fungal infection in the Mesoamerican Biologic Corridor.

Lobomycosis in a Post-Covid 19 Patient: A Case Report and Review of Literature.

AIM: To document a case of lobomycosis and to discuss its epidemiology & diagnosis. CASE REPORT: A 53-year-old male presented with a history of nasal congestion, nasal discharge, and epistaxis following Covid 19 infection. On physical examination, there was necrotic slough in the nasal vestibule near the inferior turbinate. Scrapings and punch biopsy were taken from the lesion. Hematoxylin-eosin-stained sections showed necrotic and mucoid areas with mixed inflammatory cell infiltration and numerous budding yeasts 3- 7µm diameter in singles, and small clusters with single narrow based budding as well as multiple budding including sequential budding forming "chains of yeasts". A diagnosis of Lobomycosis was made. Yeasts of lobomycosis are often confused with other yeasts such as P. brasiliensis, Candida spp., B. dermatitidis, and Cryptococci, but characteristic 'sequential budding' with a 'chain of yeasts" aid in the final diagnosis. Demonstration of yeasts with characteristic chains either in tissue sections or in potassium hydroxide (KOH) preparation of scraped material, exudate, or exfoliative cytology is the mainstay in the diagnosis as the organisms are uncultivable in vitro in culture medium.

Lacaziosis: immunohistochemical evaluation of elements of the humoral response in cutaneous lesions.

Lacaziosis is a cutaneous mycosis caused by the fungus Lacazia loboi, described in different countries of Latin America and prevalent in the Amazon region. The ineffective immune response against the agent seems to be related to a Th2 pattern of cytokines. There are few reports exploring elements of the humoral response in these lesions. Our aim was to investigate some elements focusing on B cells, plasma cells and local expression of IgG and IgM antibodies. Forty skin biopsies of lower limbs were selected. The diagnosis of lacaziosis was based on direct mycological examination and histological analysis. The visualization of fungal cells was improved by using Gridley's staining. An immunohistochemical protocol was performed to detect the expression of B cells, plasma cells, IgG and IgM. A double staining was performed to explore the presence of yeasts in the cytoplasm of keratinocytes, using an anti-AE1 AE3 antibody over Gridley's staining. The inflammatory infiltrate consisted of macrophages, multinucleated giant cells, lymphocytes, and fibrosis. Fungal cells were frequent in the stratum corneum and in both, the dermis and, in 50% of the specimens, also in the epidermis. Cells expressing IgG were more abundant when compared to cells expressing IgM. B cells and the presence of IgG might indicate that the humoral response promotes a Th2 immune response resulting in an anti-inflammatory phenotype. Our results lead us to suggest a possible role of B cells and immunoglobulins in the mechanisms of lacaziosis pathogenesis.

Case for diagnosis. Keloidal cord-like lesion on the leg

Abstract We report a 74-year-old male presented to an outpatient dermatology clinic in Manaus, Amazonas, with a one-year history of pruritic, keloidal lesions on his left lower extremity. Histopathology showed round structures in reticular dermis. Grocott methenamine silver stain revealed numerous round yeasts with thick double walls, occurring singly or in chains connected by tubular projections. The diagnosis was lobomycosis. Although the keloidal lesions presented by this patient are typical of lobomycosis, their linear distribution along the left lower limb is unusual.

What is your diagnosis? Keloidal cord-like lesion on the leg.

We report a 74-year-old male presented to an outpatient dermatology clinic in Manaus, Amazonas, with a one-year history of pruritic, keloidal lesions on his left lower extremity. Histopathology showed round structures in reticular dermis. Grocott methenamine silver stain revealed numerous round yeasts with thick double walls, occurring singly or in chains connected by tubular projections. The diagnosis was lobomycosis. Although the keloidal lesions presented by this patient are typical of lobomycosis, their linear distribution along the left lower limb is unusual.

An atypical keloid in a Brazilian man.

Case report from French Guiana of an atypical keloid in a Brazilian man: Lobomycosis. Lobomycosis is a rare fungal skin infection, endemic to the Amazon basin. Its clinical manifestations are slow-growing keloid-like nodules, with a granulomatous reaction on histopathology. The etiological agent, Lacazia loboi, has never been isolated in the environment or cultured in a laboratory. Diagnosis is based on the typical appearance on microscopy. Treatment is disappointing, with a high recurrence rate. We report a new case in French Guiana and review the literature. Only 10 cases have been reported outside of the endemic area, 2 involving no travel; the mean time to diagnosis was 21 years. Phylogenetic analysis has recently proved that lobomycosis in dolphins is caused by a fungus more closely related to Paracoccidioides brasiliensis than to L. loboi (two very closely related species). Molecular diagnosis is possible in a few centers. Climate change may result in spreading lobomycosis to currently disease-free areas of the world.

Lobomycosis in Soldiers, Colombia.

Lobomycosis is a disease that is endemic to the Amazon rainforest and is caused by the still uncultured fungus Lacazia loboi. This disease occurs in loggers, farmers, miners, fishermen, and persons living near coastal rivers of this region. We report 6 soldiers in Colombia in whom lobomycosis developed after military service in the Amazon area. The patients had nodular and keloid-like lesions on the face, neck, trunk, and limbs. The duration of illness ranged from 2 years to 15 years. The initial diagnosis was leishmaniasis on the basis of clinical manifestations and direct smear results, but biopsies confirmed the final diagnosis of lobomycosis. Treatment with surgical excision, itraconazole and clofazimine was satisfactory. However, the follow-up time was short. Healthcare professionals responsible for the diagnosis and treatment of skin diseases need to be able to recognize the clinical signs of lobomycosis and differentiate them from those of cutaneous leishmaniasis.

Lobomycosis: a therapeutic challenge.

Lobomycosis or lacaziosis is a chronic granulomatous fungal infection caused by Lacazia loboi. Most cases are restricted to tropical regions. Transmission is believed to occur through traumatic inoculation in the skin, mainly in exposed areas. It is characterized by keloid-like nodules. There are only a few hundred cases reported. The differential diagnoses include many skin conditions, and treatment is difficult. The reported case, initially diagnosed as keloid, proved to be refractory to surgical treatment alone. It was subsequently approached with extensive surgery, cryotherapy every three months and a combination of itraconazole and clofazimine for two years. No signs of clinical and histopathological activity were detected during follow-up.

Lobomycosis: a therapeutic challenge

Abstract: Lobomycosis or lacaziosis is a chronic granulomatous fungal infection caused by Lacazia loboi. Most cases are restricted to tropical regions. Transmission is believed to occur through traumatic inoculation in the skin, mainly in exposed areas. It is characterized by keloid-like nodules. There are only a few hundred cases reported. The differential diagnoses include many skin conditions, and treatment is difficult. The reported case, initially diagnosed as keloid, proved to be refractory to surgical treatment alone. It was subsequently approached with extensive surgery, cryotherapy every three months and a combination of itraconazole and clofazimine for two years. No signs of clinical and histopathological activity were detected during follow-up.

Case Report: Molecular Confirmation of Lobomycosis in an Italian Traveler Acquired in the Amazon Region of Venezuela.

Lobomycosis is a chronic skin mycosis endemic in Amazon regions characterized by chronic nodular or keloidal lesions caused by Lacazia loboi, an uncultivable fungus. Imported cases in nonendemic countries are rare and diagnosed after years. We describe a case of lobomycosis in a healthy 55-year-old Italian traveler who had acquired the infection during 5-day-honeymoon in the Amazon region of Venezuela in 1999. Several weeks after return, he recalled pruritus and papular skin lesions on the left lower limb, subsequently evolving to a plaque-like lesion. Blastomycosis and cryptococcosis were hypothesized based on microscopic morphology of yeast-like bodies found in three consecutive biopsies, although fungal cultures were always negative. In 2016, exfoliative cytology and a biopsy specimen examination showed round yeast-like organisms (6-12 µm), isolated or in a chain, connected by short tubular projections fulfilling the morphologic diagnostic criteria of Lacazia spp. The microscopic diagnosis was confirmed by molecular identification.

Analysis of Immune Response Markers in Jorge Lobo's Disease Lesions Suggests the Occurrence of Mixed T Helper Responses with the Dominance of Regulatory T Cell Activity.

Jorge Lobo's disease (JLD) is a chronic infection that affects the skin and subcutaneous tissues. Its etiologic agent is the fungus Lacazia loboi. Lesions are classified as localized, multifocal, or disseminated, depending on their location. Early diagnosis and the surgical removal of lesions are the best therapeutic options currently available for JLD. The few studies that evaluate the immunological response of JLD patients show a predominance of Th2 response, as well as a high frequency of TGF-ß and IL-10 positive cells in the lesions; however, the overall immunological status of the lesions in terms of their T cell phenotype has yet to be determined. Therefore, the objective of this study was to evaluate the pattern of Th1, Th2, Th17 and regulatory T cell (Treg) markers mRNA in JLD patients by means of real-time PCR. Biopsies of JLD lesions (N = 102) were classified according to their clinical and histopathological features and then analyzed using real-time PCR in order to determine the expression levels of TGF-ß1, FoxP3, CTLA4, IKZF2, IL-10, T-bet, IFN-γ, GATA3, IL-4, IL-5, IL-13, IL-33, RORC, IL-17A, IL-17F, and IL-22 and to compare these levels to those of healthy control skin (N = 12). The results showed an increased expression of FoxP3, CTLA4, TGF-ß1, IL-10, T-bet, IL-17F, and IL-17A in lesions, while GATA3 and IL-4 levels were found to be lower in diseased skin than in the control group. When the clinical forms were compared, TGF-ß1 was found to be highly expressed in patients with a single localized lesion while IL-5 and IL-17A levels were higher in patients with multiple/disseminated lesions. These results demonstrate the occurrence of mixed T helper responses and suggest the dominance of regulatory T cell activity, which could inhibit Th-dependent protective responses to intracellular fungi such as L. loboi. Therefore, Tregs may play a key role in JLD pathogenesis.

Leprosy and Lobomycosis: First report from the Amazon Region.

Leprosy is still a relevant health problem in Brazil with 31 044 new cases diagnosed in 2013, of which 781 new cases diagnosed in the State of Amazonas. Lobomycosis is a cutaneous-subcutaneous mycosis caused by Lacazia loboi, an in vitro uncultivable fungus. Lobomycosis has been mainly reported in the Amazon region of Brazil and Colombia affecting mainly male farmers and workers in extraction of rubber. Lobomycosis is clinically characterised by keloid-like lesions and chronic evolution. Even if lobomycosis does not represent a major public health problem, it remains a serious condition for patients due to unsatisfactory treatment. We report a case of an old man with lepromatous leprosy diagnosed in 1983, treated with multidrug therapy until 1989 and presenting a leprosy relapse 15 years after treatment. At this time a lobomycosis was also diagnosed in a keloid-like lesion evolving for more than 30 years. This co-infection has been only rarely reported and this is the first detailed case report in the English literature.

Analysis of immune response markers in Jorge Lobo's disease lesions suggests the occurrence of mixed T Helper responses with the dominance of regulatory T cell activity

Jorge Lobo's disease (JLD) is a chronic infection that affects the skin and subcutaneous tissues. Its etiologic agent is the fungus Lacazia loboi. Lesions are classified as localized, multifocal, or disseminated, depending on their location. Early diagnosis and the surgical removal of lesions are the best therapeutic options currently available for JLD. The few studies that evaluate the immunological response of JLD patients show a predominance of Th2 response, as well as a high frequency of TGF-ß and IL-10 positive cells in the lesions; however, the overall immunological status of the lesions in terms of their T cell phenotype has yet to be determined. Therefore, the objective of this study was to evaluate the pattern of Th1, Th2, Th17 and regulatory T cell (Treg) markers mRNA in JLD patients by means of real-time PCR. Biopsies of JLD lesions (N = 102) were classified according to their clinical and histopathological features and then analyzed using real-time PCR in order to determine the expression levels of TGF-ß1, FoxP3, CTLA4, IKZF2, IL-10, T-bet, IFN-γ, GATA3, IL-4, IL-5, IL-13, IL-33, RORC, IL-17A, IL-17F, and IL-22 and to compare these levels to those of healthy control skin (N = 12). The results showed an increased expression of FoxP3, CTLA4, TGF-ß1, IL-10, T-bet, IL-17F, and IL-17A in lesions, while GATA3 and IL-4 levels were found to be lower in diseased skin than in the control group. When the clinical forms were compared, TGF-ß1 was found to be highly expressed in patients with a single localized lesion while IL-5 and IL-17A levels were higher in patients with multiple/disseminated lesions. These results demonstrate the occurrence of mixed T helper responses and suggest the dominance of regulatory T cell activity, which could inhibit Th-dependent protective responses to intracellular fungi such as L. loboi. Therefore, Tregs may play a key role in JLD pathogenesis.

Doença de Jorge Lobo e a importância de seu estudo para a patologia animal e para a patologia humana / Jorge Lobo's disease and its study importance for human and animal medical conditions

A Doença de Jorge Lobo é uma micose subcutânea que tem como agente etiológico o fungo Paracoccidioides loboi. Este fungo pode infectar tanto humanos quanto golfinhos. O primeiro caso em humanos foi registrado no Brasil em 1931. A história natural da doença se inicia pela instalação do microorganismo nos tecidos subcutâneos e as lesões são nodulares, isoladas, com aspecto queloideano, acometendo principalmente o pavilhão auricular e os membros inferiores e superiores. Geograficamente, esta micose ocorre mais em regiões de florestas densas, quentes e úmidas. O presente artigo teve por objetivo mostrar as características da doença em homens e em golfinhos.

Jorge Lobo's disease is a subcutaneous mycosis caused by the Paracoccidioides loboi, which can infect both humans and dolphins. The first case in humans was described in Brazil, in 1931. The disease´s natural history initiates after the infection by the microorganism. The lesions are solitary nodular keloid-looking that arises, mainly, in the ear and in the upper and lower limbs. Geographically, this mycosis shows itself in dense, hot and humid forest regions. This article aimed the presentation of the disease in humans and dolphins.

Lobomycosis: an emerging disease in humans and delphinidae.

Lobomycosis, a disease caused by the uncultivable dimorphic onygenale fungi Lacazia loboi, remains to date as an enigmatic illness, both due to the impossibility of its aetiological agent to be cultured and grown in vitro, as well as because of its unresponsiveness to specific antifungal treatments. It was first described in the 1930s by Brazilian dermatologist Jorge Lobo and is known to cause cutaneous and subcutaneous localised and widespread infections in humans and dolphins. Soil and vegetation are believed to be the chief habitat of the fungus, however, increasing reports in marine mammals has shifted the attention to the aquatic environment. Infection in humans has also been associated with proximity to water, raising the hypothesis that L. loboi may be a hydrophilic microorganism that penetrates the skin by trauma. Although its occurrence was once thought to be restricted to New World tropical countries, its recent description in African patients has wrecked this belief. Antifungals noted to be effective in the empirical management of other cutaneous/subcutaneous mycoses have proven unsuccessful and unfortunately, no satisfactory therapeutic approach for this cutaneous infection currently exists.