Jorge Lobo's disease with malignant degeneration to squamous cell carcinoma: case report

Abstract Jorge Lobo's disease (JLD) is a chronic, granulomatous fungal infection caused by the traumatic implantation of the fungus Lacazia loboi in the cutaneous and subcutaneous tissues, with the presence of isolated nodular and coalescent keloidal lesions. Malignant degeneration is rare. This case report describes a 64-year-old male patient with JLD for 30-years who showed a change in the aspect of a lesion in the left lower limb. Histopathological examination confirmed the progression to well-differentiated squamous cell carcinoma (SSC). JLD is highly prevalent in tropical and subtropical regions, requiring monitoring concerning the transformation into SSC in long-term lesions.

Lobomycosis: exuberant presentation with malignant transformation

Abstract Lobomycosis is a chronic granulomatous infection caused by the yeast Lacazia loboi, typically found in tropical and subtropical geographical areas. Transmission occurs through traumatic inoculation into the skin, especially in exposed areas, of men who work in contact with the soil. Lesions are restricted to the skin and subcutaneous tissue, with a keloid-like appearance in most cases. The occurrence of squamous cell carcinoma on skin lesions with a long evolution is well known; however, there are scarce reports of lobomycosis that developed into squamous cell carcinoma. The authors report a patient from the Brazilian Amazon region, with lobomycosis and carcinomatous degeneration, with an unfavorable outcome, due to late diagnosis.

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.

Cirurgia plástica e o tratamento de doenças infectocontagiosas: lobomicose / Plastic surgery for the treatment of contagious diseases: lobomycosis

Introdução: Lacaziose é uma doença rara que afeta principalmente trabalhadores de áreas tropicais, sendo descritos aproximadamente 500 casos no mundo. A lacaziose é um doença parasitária causada pelo fungo saprófita Lacazia loboi, para o qual não existe um tratamento específico. A cirurgia é o tratamento mais eficiente para as deformidades causadas pela doença. Entretanto, é um tratamento temporário, uma vez que as recidivas são frequentes. Lacazia loboi acomete duas espécies de golfinhos, o Tursiops truncates e o Sotalia guianensis. A literatura aborda o tratamento cirúrgico de maneira superficial, pois não existem trabalhos específicos descrevendo o tratamento cirúrgico para essa doença. Métodos: Descrevemos aqui nossos 8 anos de experiência no Hospital de Base de Porto Velho-Rondônia com 22 casos submetidos a tratamento cirúrgico e acompanhados. Resultados: A maioria dos pacientes (91%) já se submeteram a pelo menos um tratamento cirúrgico associado ao tratamento antifúngico. Os pacientes apresentavam lesões com tempo de evolução entre 5 meses e 6 anos previamente ao tratamento cirúrgico. Apenas dois casos eram virgens de tratamento. Conclusão: Nossos pacientes foram acompanhados, mas apenas 11 dos 22 pacientes retornaram para acompanhamento. Recorrências foram observadas em 9 dos 11 pacientes, com um período de latência de 5 meses (AU)

Introduction: Lacaziosis is a rare disease that mainly affects workers in tropical areas, with approximately 500 cases reported worldwide. Lacaziosis is a parasitic disease caused by the saprophytic fungus Lacazia loboi; there is no specific treatment for this disease. Surgery is the most effective treatment for the deformities caused by the disease. However, it is a temporary treatment, since disease recurrence is frequently observed. Lacazia loboi affects two species of dolphin, Tursiops truncates and Sotalia guianensis. The available literature discusses the surgical treatment in a superficial way , because there are no specific studies describing the surgical treatment for this disease. Methods: Here, we describe our 8 years of experience with lacaziosis at the Hospital de Base de Porto Velho - Rondônia; a total of 22 patients underwent surgical treatment and were followed-up. Results: The majority of the patients (91%) had already submitted to at least one surgical treatment together with antifungal treatment. The patients presented with lesions with disease progression ranging from 5 months to 6 years prior to surgical treatment. Only two patients were treatment-naive. Conclusion: Our patients were followed-up; however, only 11 of the 22 patients returned for follow-up. Recurrences were observed in 9 of the 11 patients, with a latency period of 5 months.(AU)

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.

Jorge Lobo's disease

AbstractJorge Lobo's Disease is a rare, chronic granulomatous cutaneous mycosis, which is typical of tropical and subtropical regions. It is caused by the traumatic implantation of the fungus Lacazia loboi into the skin and subcutaneous tissue. The disease was first described in 1931 by Jorge Lobo, in Recife (PE), Brazil. It is common in Central and South America, and predominates in the Amazon region. We report a case of Jorge Lobo's Disease, which had been initially referred as being paracoccidioidomycosis. We emphasize clinical and diagnostic features of the disease.

Lacaziosis - unusual clinical presentation

Lacaziosis or Jorge Lobo's disease is a fungal, granulomatous, chronic infectious disease caused by Lacazia loboi, which usually affects the skin and subcutaneous tissue. It is characterized by slow evolution and a variety of cutaneous manifestations with the most common clinical expression being nodular keloid lesions that predominate in exposed areas. We report the case of a patient who had an unusual clinical presentation, with a single-plated lesion on the back. Histopathological examination confirmed the diagnosis of Lacaziosis.

Doença de Jorge Lobo: relato de caso e revisão de literatura / Jorge Lobo's disease: case report and literature review

Introdução e objetivos: A doença de Jorge Lobo é uma enfermidade crônica rara causada pelo fungo Lacazia loboi, caracterizada por lesões queloideanas na pele. Objetivou-se realizar revisão de literatura referente ao tema e relatar o primeiro caso de doença de Jorge Lobo atendido no Hospital Universitário Cassiano Antônio Moraes (HUCAM). Materiais e métodos: Os dados do estudo foram obtidos retrospectivamente mediante a análise e revisão sistemática do prontuário de paciente com doença de Jorge Lobo identificado no HUCAM. Relato do caso: Paciente masculino, 37 anos, encaminhado ao HUCAM em abril de 2010 apresentando lesões queloideanas e com diagnóstico prévio de lobomicose por exame anátomo-patológico. Em dezembro de 2011, foi realizada exérese parcial das lesões com enxertia; três meses depois, foi observada recidiva da lobomicose no tecido enxertado. Em junho de 2012, instituiu-se tratamento medicamentoso com clofazimina associada a fluconazol. A ausência de resposta clínica satisfatória dois meses após fez com que a doença fosse considerada fora de possibilidades terapêuticas. Discussão e revisão de literatura: A lobomicose é uma afecção endêmica na Amazônia brasileira e em outras zonas tropicais úmidas das Américas Central e do Sul. Sua transmissão ocorre pela inoculação do fungo através de soluções de continuidade na pele. O diagnóstico é feito por meio do exame clínico e do exame histopatológico ou citológico. A escolha do tipo de terapêutica varia de acordo com a extensão das lesões cutâneas. No entanto, nenhuma das terapias descritas na literatura é totalmente eficaz. Conclusão: No presente caso, falharam as duas principais propostas terapêuticas, sendo a doença considerada incurável...

Eliminação transepidérmica de parasitas na doença de Jorge Lobo / Transepidermal elimination of parasites in Jorge Lobo's disease

FUNDAMENTOS: A eliminação transepidérmica de parasitas (ETEP) tem sido pouco estudada na doença de Jorge Lobo. OBJETIVOS: Identificar aspectos morfológicos da ETEP na doença de Jorge Lobo. MÉTODOS: Recortes de biópsias de doença de Jorge Lobo emblocados em parafina foram corados pela hematoxilina-eosina e examinados. Considerou-se como ETEP, exclusivamente, a presença de parasitas em estruturas epidérmicas. RESULTADOS: Foram incluídas no estudo 40 biópsias de 37 pacientes (31 homens e seis mulheres, média de idade 51,03 anos, variação 29-80 anos) realizadas em um período de 37 anos (1967-2003), das quais foram obtidos 511 cortes (média de 12,77 cortes por caso, variação 2-39 cortes por caso). Observou-se ETEP em 110/511 (21,52 por cento) e não se observou em 401/511 cortes (78,48 por cento) (p < 0,0001). Em relação aos pacientes (37), em 15 se verificaram aspectos consistentes com ETEP (40,5 por cento), ao passo que, em 22 deles (59,5 por cento), isso não foi observado (p > 0,05). Os parasitas dispunham-se em infundíbulos hiperplásicos, formando catênulas, ou como unidades isoladas, associados ou não a células inflamatórias. CONCLUSÕES: Aspectos consistentes com ETEP, embora observados em número estatisticamente não significante de pacientes da amostra (p > 0,05), sugerem que, na doença de Jorge Lobo, o fenômeno, invariavelmente, ocorra através do epitélio infundibular. Estudos futuros serão necessários para avaliar sua eventual importância na epidemiologia da micose.

BACKGROUND: Few studies have focussed on the transepidermal elimination of parasites in Jorge Lobo's disease (lobomycosis). OBJECTIVE: To identify the morphological features of the transepidermal elimination of parasites in lobomycosis. METHODS: Sections were obtained from paraffin-embedded biopsy specimens of patients with lobomycosis and stained with hematoxylin-eosin for microscopic examination. Only the presence of parasites in epidermal structures was considered to constitute transepidermal elimination. RESULTS: Forty biopsies from 37 patients were included in the study (31 males and 6 females). The mean age of patients was 51.03 years (range 29-80 years). Biopsies performed over a period of 37 years (1967-2003) were used, from which 511 sections were obtained (a mean of 12.77 sections per case; range 2-39 sections per case). Transepidermal elimination of parasites was found in 110/511 (21.52 percent) and was absent in 401/511 sections (78.48 percent) (p<0.0001). Features consistent with the phenomenon were found in 15/37 patients (40.5 percent) and were absent in 22/37 (59.5 percent) (p>0.05). Parasites in the epidermis were detected within hyperplastic infundibula, either connected in chains or as isolated units, associated or not with inflammatory cells. CONCLUSION: Features consistent with transepidermal elimination of parasites were found in a statistically nonsignificant number of patients in the sample (p>0.05), suggesting that in Jorge Lobo's disease, this phenomenon invariably occurs through the infundibular epithelium. Future studies are required to evaluate the significance of this finding in the epidemiology of mycosis.