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      Disseminated paracoccidioidomycosis with skull and mandible involvement in a heart transplant recipient

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          Abstract

          Paracoccidioidomycosis (PCM) is the most common systemic mycosis in South America, particularly in Brazil. The respiratory airways are the main route of entry and usually spread through the vascular and lymphatic systems, affecting any part of the body. Herein, we report the case of a 59-year-old woman, recipient of a heart transplant, who was being treated with mycophenolate, cyclosporine, and tacrolimus and presented intense pulsatile holocranial headache associated with nodules on the scalp and trismus. Brain computed tomography (CT) revealed skull lesions (Figures 1A and B ). Magnetic resonance imaging (MRI) of the brain was performed (Figures 1C-F). CT of the neck revealed lymph node enlargement and osteolytic lesions in the mandible. Chest CT revealed numerous small peripheral lung nodules, and abdominal CT detected an abscess in the left iliopsoas muscle (Figure 2). The fungus showed a "steering wheel" morphology detected using Grocott's methenamine staining (Figure 3). FIGURE 1: CT of the brain showing the permeative osteolytic pattern involving the left frontal and right temporal bones (A) and parietal bones (B). The permeative osteolytic pattern of the lesions is better demonstrated with MIP reconstruction. MRI of the brain showed lesions with a solid component and low signal on T2 WI (C) and on FLAIR WI (D), restricted diffusion on DWI (E), and heterogeneous enhancement on T1-WI after gadolinium in addition to pachymeningeal enhancement adjacent to the lesions (white curved arrow in F). FIGURE 2: CT and MRI of the neck showing lymph node enlargement in the right level V (white arrow in A, B, and C) and a CT of the chest showing the presence of numerous small diffuse peripheral nodules (D). A CT of the abdomen also revealed an abscess in the left iliopsoas muscle (E) and necrotic lymph nodes (F), demonstrated with a white arrow. FIGURE 3: (A) Paracoccidioidomycosis shows a prominent granulomatous inflammatory infiltrate with multinucleated giant cells containing fungal organisms (black arrow). Hematoxylin-eosin, 200x. (B) The budding yeast form of the fungus shows a classic "steering wheel" morphology (black arrows). Grocott's methenamine stain, 200x. In solid organ transplantations, chronic PCM has been described mainly in kidney transplants. Almeida et al. 1 described nine cases of PCM after kidney transplantation and one case in a liver transplant recipient. PCM in immunocompromised heart transplant patients has not been reported. PCM can develop after hematological dissemination from an active pulmonary infection or through the reactivation of a latent focus in the central nervous system after immunosuppression 2 . The patient responded well to treatment with voriconazole followed by cotrimoxazole. This treatment schedule was based on standard antifungal therapy 3 .

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          Brazilian guidelines for the clinical management of paracoccidioidomycosis

          Abstract Paracoccidioidomycosis is a systemic fungal disease occurring in Latin America that is associated with rural environments and agricultural activities. However, the incidence and prevalence of paracoccidiodomycosis is underestimated because of the lack of compulsory notification. If paracoccidiodomycosis is not diagnosed and treated early and adequately, the endemic fungal infection could result in serious sequelae. While the Paracoccidioides brasiliensis ( P. brasiliensis ) complex has been known to be the causal agent of paracoccidiodomycosis, a new species, Paracoccidioides lutzii ( P. lutzii ), has been reported in Rondônia, where the disease has reached epidemic levels, and in the Central West and Pará. Accurate diagnoses and availability of antigens that are reactive with the patients’ sera remain significant challenges. Therefore, the present guidelines aims to update the first Brazilian consensus on paracoccidioidomycosis by providing evidence-based recommendations for bedside patient management. This consensus summarizes etiological, ecoepidemiological, molecular epidemiological, and immunopathological data, with emphasis on clinical, microbiological, and serological diagnosis and management of clinical forms and sequelae, as well as in patients with comorbidities and immunosuppression. The consensus also includes discussion of outpatient treatments, severe disease forms, disease prevalence among special populations and resource-poor settings, a brief review of prevention and control measures, current challenges and recommendations.
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            Paracoccidioidomycosis in Immunocompromised Patients: A Literature Review

            Paracoccidioidomycosis (PCM) is an endemic mycosis found in Latin America that causes systemic disease mostly in immunocompetent hosts. A small percentage of PCM occurs in immunocompromised patients where low clinical suspicion of the infection, late diagnosis, and uncertainties about its management are factors that negatively impact their outcomes. We conducted a literature review searching reports on PCM associated to HIV, cancer, maligned hemopathies, solid organ transplantation, and immunotherapies, in order to check for peculiarities in terms of natural history and challenges in the clinical management of PCM in this population. HIV patients with PCM usually had low T CD4+ cell counts, pulmonary and lymph nodes involvement, and a poorer prognosis (≈50% mortality). Most of the patients with PCM and cancer had carcinoma of the respiratory tract. Among maligned hemopathies, PCM was more often related to lymphoma. In general, PCM prognosis in patients with malignant diseases was related to the cancer stage. PCM in transplant recipients was mostly associated with the late phase of kidney transplantation, with a high mortality rate (44%). Despite being uncommon, reactivation of latent PCM may take place in the setting of immunocompromised patients exhibiting clinical particularities and it carries higher mortality rates than normal hosts.
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              Neuroparacoccidioidomycosis (NPCM): Magnetic Resonance Imaging (MRI) Findings

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                Author and article information

                Journal
                Rev Soc Bras Med Trop
                Rev Soc Bras Med Trop
                rsbmt
                Revista da Sociedade Brasileira de Medicina Tropical
                Sociedade Brasileira de Medicina Tropical - SBMT
                0037-8682
                1678-9849
                06 June 2022
                2022
                : 55
                : e0110-2022
                Affiliations
                [1 ] Universidade Estadual de Campinas, Faculdade de Ciências Médicas, Departamento de Radiologia, Campinas, SP, Brasil.
                [2 ] Universidade Estadual de Campinas, Faculdade de Ciências Médicas, Departamento de Patologia, Campinas, SP, Brasil.
                Author notes
                Corresponding author: Fabiano Reis. e-mail: fabianoreis2@ 123456gmail.com

                Authors’ contribution: FVP: Conception and design of the study, Drafting the article, Acquisition of data; KPOA: Conception and design of the study, Analysis and interpretation of data; AMAMA: Conception and design of the study, Analysis and interpretation of data; FR: Conception and design of the study, Acquisition of data, Drafting the article, Final approval of the version to be submitted.

                Conflict of Interest: The authors declare that there is no conflict of interest.

                Author information
                http://orcid.org/0000-0002-0828-7806
                http://orcid.org/0000-0002-5447-5437
                http://orcid.org/0000-0002-9944-9734
                http://orcid.org/0000-0003-2256-4379
                Article
                00828
                10.1590/0037-8682-0110-2022
                9176728
                35674557
                7d6ddf14-ac2a-4a69-b7e4-84a86eb7188c

                This is an open-access article distributed under the terms of the Creative Commons Attribution License

                History
                : 17 February 2022
                : 30 March 2022
                Page count
                Figures: 3, Tables: 0, Equations: 0, References: 3
                Categories
                Images in Infectious Diseases

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