"Star of Bethlehem sign" in the analysis of the evolution of brain lesions during and after treatment for neuroparacoccidioidomycosis.

Objective: To describe the clinical and radiological evolution of lesions during and after treatment in patients diagnosed with neuroparacoccidioidomycosis (NPCM). Materials and Methods: This was a retrospective study of the medical records, computed tomography scans, and magnetic resonance imaging (MRI) scans of patients with NPCM treated between September 2013 and January 2022. Results: Of 36 cases of NPCM, eight were included in the study. One patient presented only with pachymeningeal and skull involvement, and seven presented with pseudotumors in the brain. Collectively, the eight patients presented with 52 lesions, of which 46 (88.5%) were supratentorial. There were 32 lesions with a diameter ≤ 1.2 cm, of which 27 (84.4%) disappeared during the treatment. In three cases, there were lesions > 1.2 cm that showed a characteristic pattern of evolution on MRI: an eccentric gadolinium contrast-enhanced nodule, with a subsequent decreased in the size and degree of contrast enhancement of the lesions. Conclusion: In NPCM, supratentorial lesions seem to predominate. Lesions ≤ 1.2 cm tend to disappear completely during treatment. Lesions > 1.2 cm tend to present with a similar pattern, designated the "Star of Bethlehem sign", throughout treatment.

Objetivo: Descrever a evolução clínica e radiológica das lesões durante e após o tratamento de pacientes diagnosticados com neuroparacoccidioidomicose (NPCM). Materiais e Métodos: Revisamos os prontuários médicos, estudos de tomografia computadorizada e ressonância magnética (RM) de pacientes com NPCM de nossa instituição, no período de setembro de 2013 a janeiro de 2022. Resultados: Dos 36 casos de NPCM, oito foram incluídos no presente estudo. Um caso apresentava apenas envolvimento paquimeníngeo e ósseo craniano e sete casos apresentavam lesões encefálicas pseudotumorais, totalizando 52 lesões, sendo 46 (88,5%) supratentoriais. Dentre 32 lesões com diâmetro ≤ 1,2 cm, 27 (84,4%) apresentaram resolução completa durante o tratamento. Três casos apresentaram padrão semelhante de evolução da lesão na RM em lesões > 1,2 cm, caracterizado pelo aparecimento de nódulo excêntrico com impregnação pelo gadolínio, seguido de redução das dimensões e do realce nodular pelo contraste nos estudos subsequentes. Conclusão: A NPCM apresenta-se predominantemente com lesões supratentoriais. Lesões ≤ 1,2 cm tendem a desaparecer completamente durante o tratamento. Lesões > 1,2 cm tendem a apresentar um padrão de imagem de RM característico ao longo do tratamento, descrito como o "sinal da Estrela de Belém".

Paracoccidioidomycosis cases by Paracoccidioides lutzii in southeastern Brazil.

BACKGROUND AND OBJECTIVE: Paracoccidioidomycosis (PCM) is a systemic fungal disease caused by the thermodimorphic fungi Paracoccidioides spp. Their distribution is highly variable. Paracoccidioides lutzii is predominantly found in North and Middle-West Brazil and Ecuador. This study evaluated the clinicopathological characteristics of 10 patients diagnosed with PCM caused by P. lutzii in a reference center located in southeastern Brazil. DESIGN: Double immunodiffusion assay (DID) was used to investigate 35 patients' sera with negative serology for P. brasiliensis against a P. lutzii CFA (cell-free antigen). RESULTS: Out of the 35 retested patients, 10 (28.6%) were positive for P. lutzii CFA. Four patients did not report any displacement to P. lutzii endemic areas. Our results reinforce the importance of using different antigens when testing patients with clinical manifestations of PCM and negative serological tests for P. brasiliensis, primarily in cases of the report of displacement to or former residence in P. lutzii endemic regions. CONCLUSIONS: The availability of tests for different Paracoccidioides species antigens is fundamental for reaching an adequate diagnosis, patient follow-up, and definition of prognosis.

"Star of Bethlehem sign" in the analysis of the evolution of brain lesions during and after treatment for neuroparacoccidioidomycosis / "Sinal da Estrela de Belém" na análise evolutiva das lesões durante e após o tratamento em pacientes com neuroparacoccidioidomicose

Abstract Objective: To describe the clinical and radiological evolution of lesions during and after treatment in patients diagnosed with neuroparacoccidioidomycosis (NPCM). Materials and Methods: This was a retrospective study of the medical records, computed tomography scans, and magnetic resonance imaging (MRI) scans of patients with NPCM treated between September 2013 and January 2022. Results: Of 36 cases of NPCM, eight were included in the study. One patient presented only with pachymeningeal and skull involvement, and seven presented with pseudotumors in the brain. Collectively, the eight patients presented with 52 lesions, of which 46 (88.5%) were supratentorial. There were 32 lesions with a diameter ≤ 1.2 cm, of which 27 (84.4%) disappeared during the treatment. In three cases, there were lesions > 1.2 cm that showed a characteristic pattern of evolution on MRI: an eccentric gadolinium contrast-enhanced nodule, with a subsequent decreased in the size and degree of contrast enhancement of the lesions. Conclusion: In NPCM, supratentorial lesions seem to predominate. Lesions ≤ 1.2 cm tend to disappear completely during treatment. Lesions > 1.2 cm tend to present with a similar pattern, designated the "Star of Bethlehem sign", throughout treatment.

Resumo Objetivo: Descrever a evolução clínica e radiológica das lesões durante e após o tratamento de pacientes diagnosticados com neuroparacoccidioidomicose (NPCM). Materiais e Métodos: Revisamos os prontuários médicos, estudos de tomografia computadorizada e ressonância magnética (RM) de pacientes com NPCM de nossa instituição, no período de setembro de 2013 a janeiro de 2022. Resultados: Dos 36 casos de NPCM, oito foram incluídos no presente estudo. Um caso apresentava apenas envolvimento paquimeníngeo e ósseo craniano e sete casos apresentavam lesões encefálicas pseudotumorais, totalizando 52 lesões, sendo 46 (88,5%) supratentoriais. Dentre 32 lesões com diâmetro ≤ 1,2 cm, 27 (84,4%) apresentaram resolução completa durante o tratamento. Três casos apresentaram padrão semelhante de evolução da lesão na RM em lesões > 1,2 cm, caracterizado pelo aparecimento de nódulo excêntrico com impregnação pelo gadolínio, seguido de redução das dimensões e do realce nodular pelo contraste nos estudos subsequentes. Conclusão: A NPCM apresenta-se predominantemente com lesões supratentoriais. Lesões ≤ 1,2 cm tendem a desaparecer completamente durante o tratamento. Lesões > 1,2 cm tendem a apresentar um padrão de imagem de RM característico ao longo do tratamento, descrito como o "sinal da Estrela de Belém".

Paracoccidioidomycosis: What We Know and What Is New in Epidemiology, Diagnosis, and Treatment.

Paracoccidioidomycosis (PCM) is a systemic mycosis endemic to Latin America caused by thermodimorphic fungi of the genus Paracoccidioides. In the last two decades, enhanced understanding of the phylogenetic species concept and molecular variations has led to changes in this genus' taxonomic classification. Although the impact of the new species on clinical presentation and treatment remains unclear, they can influence diagnosis when serological methods are employed. Further, although the infection is usually acquired in rural areas, the symptoms may manifest years or decades later when the patient might be living in the city or even in another country outside the endemic region. Brazil accounts for 80% of PCM cases worldwide, and its incidence is rising in the northern part of the country (Amazon region), owing to new settlements and deforestation, whereas it is decreasing in the south, owing to agriculture mechanization and urbanization. Clusters of the acute/subacute form are also emerging in areas with major human intervention and climate change. Advances in diagnostic methods (molecular and immunological techniques and biomarkers) remain scarce, and even the reference center's diagnostics are based mainly on direct microscopic examination. Classical imaging findings in the lungs include interstitial bilateral infiltrates, and eventually, enlargement or calcification of adrenals and intraparenchymal central nervous system lesions are also present. Besides itraconazole, cotrimoxazole, and amphotericin B, new azoles may be an alternative when the previous ones are not tolerated, although few studies have investigated their use in treating PCM.

Trichosporon asahii causing subcutaneous mycoses in an immunocompetent patient: case report and a minireview.

Trichosporon spp. are a constituent of the normal flora of humans that can cause both superficial and invasive infections, mainly in immunocompromised and immunocompetent hosts, respectively. Herein, we a report of Trichosporon asahii causing subcutaneous fungal infection (SFI) in an immunocompetent patient after carpal tunnel surgery. Although susceptible to fluconazole, the treatment of SFI failed even using high doses of this azole. The skin lesion improved following the administration of voriconazole. We conducted a literature minireview searching reports on SFI in immunocompetent patients to check for epidemiological, diagnostic, therapeutic, and outcome characteristics. A total of 32 cases were reported. Despite being uncommon, the clinical suspicion and early diagnosis of SFI in immunocompetent patients undergoing previous surgery are important. Our study indicated that the azoles are the most active antifungal agents against Trichosporon spp., except for fluconazole, and voriconazole can be considered the first therapeutic option.

Screening paracoccidioidomycosis by double immunodiffusion test in a referred diagnostic center in Brazilian southeastern: an accessible tool.

PURPOSE: This article shows reports of the clinical-epidemiological characteristics and serological screening in patients assisted by a reference center for PCM care, University Hospital Cassiano Antonio Moraes, Federal University of Espirito Santo, Brazil. METHODS: The patient's sera with PCM were analyzed by DID test at the beginning and the end treatment. Clinical and demographic data were also collected to characterize the sample. RESULTS: One hundred patients with a suspected diagnosis of PCM were evaluated. Serology by DID test was used as a screen in all patients. The test was positive for 79 patients (72 for Paracoccidioides brasiliensis and 7 for Paracoccidioides lutzii). Serology was negative in 21 sera, although all of them were diagnosed PCM by histopathologic or direct exam. Serological follow-up was performed during the treatment of all patients. After treatment, 58(58%) had negative serology and 33(33%) low levels of antibodies (≤ 1:16). CONCLUSION: Our results indicate the importance of the DID test for the screening and monitoring of PCM and that the incidence of P. lutzii might be greater than expected in areas where it is not the predominant PCM species. Therefore, this article may contribute to improving the knowledge and clinical management about this disease.

Paracoccidioidomycosis due to Paracoccidioides brasiliensis S1 associated with acquired immunodeficiency syndrome: A case report.

BACKGROUND: Paracoccidioidomycosis (PCM) is an endemic disease in Latin America. In immunocompetent hosts, PCM occurs in two main clinical forms: acute and chronic. However, in HIV-infected patients PCM may show up simultaneous manifestations of acute and chronic forms. CASE REPORT: We present the case of a patient diagnosed with HIV who had disseminated skin lesions and generalized lymphadenopathy, as well as respiratory and central nervous system involvement. The PCM diagnosis was confirmed by direct KOH examination, double immunodiffusion and the isolation of the fungus in samples of an abscess in the subcostal region. The isolate was identified as Paracoccidioides brasiliensis S1 by species-specific PCR using primers for protein-coding gene GP43 (exon 2) followed by PCR-RFLP of the alpha-tubulin gene. CONCLUSIONS: There are few data in literature reporting species-specific molecular identification of Paracoccidioides in HIV/PCM patients. Therefore, this case report may contribute to improve the knowledge about this severe disease, its causative cryptic species, and its consequences to patients.

New Insights on Pulmonary Paracoccidioidomycosis.

Paracoccidioidomycosis (PCM) is one of the most relevant systemic endemic mycoses in Latin American countries, especially in South American countries, with endemic and hyperendemic areas. The real burden of PCM may be underestimated because of a lack of compulsory case notification. Recent phylogenetic data revealed that Paracoccidioides brasiliensis comprises several cryptic species including P. brasiliensis and P. lutzii. However, the genetic biodiversity of Paracoccidioides does not affect the clinical manifestations or therapeutic response to therapy. Lung involvement is a common finding, especially in patients experiencing the chronic form of the disease, and, because of its similarities with tuberculosis, clinicians must be alert to the possibility of PCM in patients with chronic respiratory manifestations and epidemiological risk factors for this fungal disease.

Case Report: Paracoccidioidomycosis in Solid Organ Transplantation: Disseminated Disease in a Liver Recipient and Literature Review.

Paracoccidioidomycosis (PCM) is an endemic systemic mycosis that is of great importance in Latin America. Its occurrence in solid organ transplantation (SOT) is rare, but with high mortality rate. In this report, we describe a case of PCM in a liver transplant recipient 19 months after transplantation. The patient presented with multiple skin abscesses, arthritis, osteolytic lesions, and pulmonary and adrenal involvement. Despite the presence of disseminated disease and the patient's immunosuppressed condition, the patient responded well to prolonged antifungal treatment with no sequelae, thus suggesting that early diagnosis and correct treatment may lead to favorable outcomes in SOT recipients with PCM.

Pulmonary paracoccidioidomycosis: a case report of reactivation in a patient receiving biological therapy.

Paracoccidioidomycosis is an endemic disease in Latin America that is rarely associated with immunosuppression and biological therapy. Herein, we report for the first time a case of pulmonary paracoccidioidomycosis reactivation after infliximab treatment. A 47-year-old man from Brazil received infliximab to treat psoriatic spondyloarthropathy and presented with cough, dyspnea, weight loss, and fever. Chest computed tomography revealed a pulmonary nodule and biopsy confirmed paracoccidioidomycosis. Treatment with sulfamethoxazole and trimethoprim was initiated for fungal infection and infliximab was reintroduced two months later. Considering his clinical improvement and favorable radiologic evolution, antifungal therapy was discontinued after 29 months.

Pulmonary paracoccidioidomycosis: a case report of reactivation in a patient receiving biological therapy

Abstract Paracoccidioidomycosis is an endemic disease in Latin America that is rarely associated with immunosuppression and biological therapy. Herein, we report for the first time a case of pulmonary paracoccidioidomycosis reactivation after infliximab treatment. A 47-year-old man from Brazil received infliximab to treat psoriatic spondyloarthropathy and presented with cough, dyspnea, weight loss, and fever. Chest computed tomography revealed a pulmonary nodule and biopsy confirmed paracoccidioidomycosis. Treatment with sulfamethoxazole and trimethoprim was initiated for fungal infection and infliximab was reintroduced two months later. Considering his clinical improvement and favorable radiologic evolution, antifungal therapy was discontinued after 29 months.

Paracoccidioidomycosis: Epidemiological and Clinical Aspects in 546 Cases Studied in the State of Espírito Santo, Brazil.

Paracoccidioidomycosis (PCM) is an endemic disease in the southeast region of Brazil, which includes the state of Espírito Santo (ES). This historic case series analyses 546 patients treated in this state from 1978 to 2012. Patients aged from 7 to 83 years, 509 males and 37 females, yielding a ratio of 13.7:1. Most of the patients (81.4%) originated from rural areas in ES, 71.0% being farmers. A higher concentration of cases was observed in municipalities located along the western range of the state. Sixty patients (11.0%) had an acute/subacute form of the disease, 485 (88.9%) had a chronic form, and one had a subclinical form. The most affected organs included the lungs, oropharyngeal mucosa, lymph nodes, skin, and larynx. The diagnosis was confirmed by histopathology in 252 (46.2%) cases, direct examination in 168 (30.7%), both exams in 111 (20.3%) and serology in 15 (2.8%). Tuberculosis, acquired immune deficiency syndrome, leishmaniasis, and intestinal parasites were the most frequently associated infectious diseases. From 328 patients followed up, total regression of the lesions was observed in 17.4%, partial regression in 77.4%, and no regression in 17 (5.2%) cases. Regarding the number of cases in this series, ES emerges as an important endemic area for PCM in Brazil.

Amphotericin B lipid complex in the treatment of severe paracoccidioidomycosis: a case series.

Amphotericin B deoxycholate is the main option for intravenous (i.v.) treatment of severe paracoccidioidomycosis (PCM). This is the first report of amphotericin B lipid complex (ABLC) in the treatment of PCM. Among 28 patients, cure was achieved in all patients (100%) using ABLC. Mean and median daily doses of ABLC were 3.39 mg/kg/day and 3.35 mg/kg/day, respectively. ABLC may be a choice in the treatment of severe forms of PCM or when i.v. options are required.

Osteomielite de punho em paciente com paracoccidioidomicose disseminada: uma rara apresentação / Osteomyelitis of the wrist in a patient with disseminated paracoccidioidomycosis: a rare presentation

Paracoccidioidomicose é a micose sistêmica endêmica mais frequente no Brasil. No início, o paciente não desenvolve sintomas. Com a progressão da doença, o indivíduo pode apresentar envolvimento disseminado, sendo que o acometimento ósseo é extremamente raro. O objetivo deste artigo é avaliar as alterações ósseas encontradas em estudos de imagem em um paciente com osteomielite de punho decorrente de paracoccidioidomicose disseminada.

Paraccocidioidomycosis is the most frequently found endemic systemic mycosis in Brazil. No symptoms are observed in the early phases of the disease. As the disease progresses, the patient may present disseminated involvement, but bone involvement is extremely rare. The present report is aimed at evaluating bone changes found on imaging studies in a patient with osteomyelitis of the wrist as a result of disseminated paracoccidioidomycosis.

[First description of human bronchial syngamosis in the State of Espírito Santo]. / Primeira descrição de singamose brônquica ocorrida no Estado do Espírito Santo.

We report the case of a 48-year-old man who presented progressive dry cough associated with chest pain over a four-month period. After ruling out the most probable etiologies, bronchofibroscopy was performed and showed a pair of Syngamus laringeus inside the left upper lobe bronchus. The patient became asymptomatic after the nematodes had been removed.

Primeira descrição de singamose brônquica ocorrida no Estado do Espírito Santo / First description of human bronchial syngamosis in the State of Espírito Santo

Relatamos o caso de um homem de 48 anos, que evoluiu durante 4 meses com quadro de tosse seca associada à dor torácica. Após serem descartadas as etiologias mais prováveis, realizou-se uma broncofibroscopia que revelou a presença de um casal de Syngamus laringeus no brônquio do lobo superior esquerdo. O paciente tornou-se assintomático após a retirada do nematóide.

We report the case of a 48-year-old man who presented progressive dry cough associated with chest pain over a four-month period. After ruling out the most probable etiologies, bronchofibroscopy was performed and showed a pair of Syngamus laringeus inside the left upper lobe bronchus. The patient became asymptomatic after the nematodes had been removed.