CC BY-NC-ND 4.0 · Arquivos Brasileiros de Neurocirurgia: Brazilian Neurosurgery 2021; 40(02): e195-e199
DOI: 10.1055/s-0040-1719005
Case Report | Relato de Caso

Multifocal Pseudotumorous Form of Neuroparacoccidioidomycosis in an Immunocompetent Patient: A Clinicopathological Review Based on a Case Report

Neuroparacoccidioidomicose multifocal de forma pseudotumoral em paciente imunocompetente: Uma revisão clinicopatológica baseada em um relato de caso
1   Department of Pathology, Universidade Federal do Rio Grande do Sul, Porto Alegre, RS, Brazil
2   Department of Pathology, Hospital Santa Rita, Complexo Hospitalar Santa Casa, Porto Alegre, RS, Brazil
3   Department of Pathology, Grupo Hospitalar Conceição, Porto Alegre, RS, Brazil
4   Department of Pathology, Universidade Luterana do Brasil, Canoas, RS, Brazil
5   Department of Pathology, Instituto de Cardiologia, Fundação Universitária de Cardiologia, Porto Alegre, RS, Brazil
,
2   Department of Pathology, Hospital Santa Rita, Complexo Hospitalar Santa Casa, Porto Alegre, RS, Brazil
3   Department of Pathology, Grupo Hospitalar Conceição, Porto Alegre, RS, Brazil
,
6   Grupo Hospitalar Conceição, Porto Alegre, RS, Brazil
,
6   Grupo Hospitalar Conceição, Porto Alegre, RS, Brazil
,
6   Grupo Hospitalar Conceição, Porto Alegre, RS, Brazil
,
6   Grupo Hospitalar Conceição, Porto Alegre, RS, Brazil
,
7   Department of Neurosurgery, Grupo Hospitalar Conceição, Porto Alegre, RS, Brazil
,
7   Department of Neurosurgery, Grupo Hospitalar Conceição, Porto Alegre, RS, Brazil
› Author Affiliations
Funding No funding was received.

Abstract

Neuroparacoccidiodimycosis (NPDM) is an uncommon granulomatous disease, which more frequently affects immunocompromised male patients over 30 years of age in the course of chronic lung disease. Paracoccidioides brasiliensis (PB) is an endemic fungus in Brazil, and grows as thick-walled yeast (with round to oval bodies) measuring 10 µm to 60 µm in diameter. Neuroparacoccidiodimycosi may develop many years after transmission and/or primary lung involvement. The authors describe a case of NPDM affecting a male patient, 52 years of age, farmer, heavy smoker, with clinical complaint of headache, asthenia, seizures, and prostration in the previous nine months. Upon physical examination, the patient presented regular general condition, without other relevant physical alterations. Computed tomography (CT) showed multiple bilateral pulmonary nodules associated to enlargement of the mediastinal lymph node. Magnetic resonance imaging (MRI) and CT scans of the central nervous system showed six heterogeneous nodular lesions compromising the frontal and parietal lobes, the largest one measuring 3.8 × 3.2 × 3.2 cm. The hypothesis of a neoplastic process compromising the lung and brain was considered. A biopsy of the mediastinal lymph node showed epithelioid granulomas, which exhibited round, thin-walled fungal structures in Grocott silver stain. The stereotactic biopsy of the frontal lesion was constituted by necrotic tissue admixed with some round to oval, thin-walled fungi measuring 10 µm to 60 µm, compatible with PB (identified on Grocott silver stain/confirmed in culture). The diagnosis of NPDM was then established. The employed therapeutic regimen was intravenous amphotericin B, itraconazole, and sulfamethoxazole-trimetropin. After ninety days of clinical follow-up, no episodes of seizures/neurological deficits were identified, and a marked decrease in the number and size of the lung and brain lesions were found.

Resumo

A neuroparacoccidiodimicose (NPDM) é uma doença granulomatosa incomum, que acomete mais freqüentemente pacientes imunocomprometidos do sexo masculino com mais de 30 anos, no curso de doença pulmonar crônica. Paracoccidioides brasiliensis (PB) é um fungo endêmico no Brasil e cresce como levedura de parede espessa (com corpos arredondados a ovais) medindo 10–60 μm de diâmetro. A NPDM pode se desenvolver muitos anos após a transmissão e / ou envolvimento pulmonar primário. Os autores descrevem um caso de NPDM em paciente masculino, 52 anos, agricultor, tabagista pesado, com queixa clínica de cefaleia, astenia, convulsões e prostração nos últimos nove meses. Ao exame físico, o paciente apresentava estado geral regular, sem outras alterações físicas relevantes. A tomografia computadorizada (TC) mostrou múltiplos nódulos pulmonares bilaterais associados a linfonodomegalia mediastinal. A TC / ressonância magnética do sistema nervoso central revelou seis lesões nodulares heterogêneas comprometendo os lobos frontal e parietal, a maior delas medindo 3,8x3,2x3,2 cm. Foi considerada a hipótese do processo neoplásico comprometendo pulmão e cérebro. A biópsia de linfonodo mediastinal mostrou granulomas epitelioides, que exibiam estruturas fúngicas arredondadas e de paredes finas na coloração pela prata de Grocott. A biópsia estereotáxica da lesão frontal era constituída por tecido necrótico entremeado por algumas estruturas fúngicas redondas a ovais e de parede fina, medindo 10–60 μm, compatível com PB (identificado na coloração de prata Grocott / confirmado em cultura). O diagnóstico de NPDM foi então estabelecido. O esquema terapêutico empregado foi anfotericina B intravenosa, itraconazol e sulfametoxazol-trimetropina. Após 90 dias de acompanhamento clínico, nenhum episódio de convulsão / déficit neurológico foi identificado, e uma diminuição acentuada no número e tamanho das lesões pulmonares e cerebrais foi encontrada.



Publication History

Received: 07 January 2020

Accepted: 24 August 2020

Article published online:
26 November 2020

© 2020. Sociedade Brasileira de Neurocirurgia. This is an open access article published by Thieme under the terms of the Creative Commons Attribution-NonDerivative-NonCommercial License, permitting copying and reproduction so long as the original work is given appropriate credit. Contents may not be used for commecial purposes, or adapted, remixed, transformed or built upon. (https://creativecommons.org/licenses/by-nc-nd/4.0/)

Thieme Revinter Publicações Ltda.
Rua do Matoso 170, Rio de Janeiro, RJ, CEP 20270-135, Brazil

 
  • References

  • 1 de Almeida SM, Salvador GLO, Roza TH. et al. Geographical evaluation of Neuroparacoccidioidomycosis and Paracoccidioidomycosis in Southern Brazil. Mycoses 2018; 61 (08) 587-593
  • 2 Carvalho de Medeiros F, Curcio de Moraes A, Buzelin Nunes M, Dellaretti M. Neuroparacoccidioidomycosis mimicking brain tumor in an immunocompetent patient. Rev Neurol (Paris) 2018; 174 (04) 268-271
  • 3 Silva-Vergara ML, Rocha IH, Vasconcelos RR. et al. Central nervous system paracoccidioidomycosis in an AIDS patient: case report. Mycopathologia 2014; 177 (1-2): 137-141
  • 4 Kontoyiannis DP, Marr KA, Park BJ. et al. Prospective surveillance for invasive fungal infections in hematopoietic stem cell transplant recipients, 2001-2006: overview of the Transplant-Associated Infection Surveillance Network (TRANSNET) Database. Clin Infect Dis 2010; 50 (08) 1091-1100
  • 5 dos Santos VM, Xavier RM, Cortes JA, Osterne EM, de Worisch Ferreira Lopes M. Pseudotumoral neuroparacoccidioidomycosis: one case report. Mycopathologia 2008; 166 (03) 155-158
  • 6 Reis F, Collier PP, Souza TF. et al. Neuroparacoccidioidomycosis (NPCM): magnetic resonance imaging (MRI) findings. Mycopathologia 2013; 175 (1-2): 181-186
  • 7 Gomes Carvalho Neto E, Coletto A, Biazus PG, Pereira Dos Santos I, Rieder CRM, de Castro Ribeiro M. Neuroparacoccidioidomycosis. Neurol Neuroimmunol Neuroinflamm 2018; 6 (01) e519
  • 8 de Almeida SM, Roza TH, Salvador GLO. et al. Neurological and multiple organ involvement due to Paracoccidioides brasiliensis and HIV co-infection diagnosed at autopsy. J Neurovirol 2017; 23 (06) 913-918
  • 9 Fagundes-Pereyra WJ, Carvalho GT, Góes AM, das Chagas Lima e Silva F, de Sousa AA. [Central nervous system paracoccidioidomycosis: analysis of 13 cases]. Arq Neuropsiquiatr 2006; 64 (2A) 269-276
  • 10 de Almeida SM, Roza TH, Salvador GLO. et al. Autopsy and biopsy study of paracoccidioidomycosis and neuroparacoccidioidomycosis with and without HIV co-infection. Mycoses 2018; 61 (04) 237-244
  • 11 Francesconi F, da Silva MT, Costa RL. et al. Long-term outcome of neuroparacoccidioidomycosis treatment. Rev Soc Bras Med Trop 2011; 44 (01) 22-25
  • 12 Dall Bello AG, Severo CB, Oliveira FdeM, Severo LC. Disseminated paracoccidioidomycosis (simulating metastatic lung cancer) and Strongyloides stercoralis hyperinfestation in a steroid-treated patient. J Clin Microbiol 2011; 49 (05) 2054-2055
  • 13 Dias AB, Giugno CS, Scapineli JO, Tarso GP, Curtis RM, Lenhardt R. Neuroparacoccidioidomycosis with concomitant pulmonary and vocal cord lesions. Arq Neuropsiquiatr 2018; 76 (11) 802-804
  • 14 Elias Jr J, dos Santos AC, Carlotti Jr CG. et al. Central nervous system paracoccidioidomycosis: diagnosis and treatment. Surg Neurol 2005; 63 (Suppl. 01) S13 –S21, discussion S21
  • 15 Alves GR, Lauda Filho JD, Haygert CJ. Neuroparacoccidioidomycosis. Rev Soc Bras Med Trop 2011; 44 (05) 654
  • 16 de Almeida SM, Queiroz-Telles F, Teive HA, Ribeiro CE, Werneck LC. Central nervous system paracoccidioidomycosis: clinical features and laboratorial findings. J Infect 2004; 48 (02) 193-198
  • 17 Paniago AM, de Oliveira PA, Aguiar ES. et al. Neuroparacoccidioidomycosis: analysis of 13 cases observed in an endemic area in Brazil. Trans R Soc Trop Med Hyg 2007; 101 (04) 414-420
  • 18 Marques da Silva SH, Colombo AL, Blotta MH, Lopes JD, Queiroz-Telles F, Pires de Camargo Z. Detection of circulating gp43 antigen in serum, cerebrospinal fluid, and bronchoalveolar lavage fluid of patients with paracoccidioidomycosis. J Clin Microbiol 2003; 41 (08) 3675-3680
  • 19 Jorge Jr LA, Yamashita S, Trindade AP. et al. Pseudotumoral neuroparacoccidioidomycosis of the posterior fossa: A case report and review of the literature. Surg Neurol Int 2017; 8: 76
  • 20 Rosa Júnior M, Amorim AC, Baldon IV. et al. Paracoccidioidomycosis of the central nervous system: CT and MR imaging findings. AJNR Am J Neuroradiol 2019; 40 (10) 1681-1688
  • 21 Corti M, Trione N, Risso D. et al. Disseminated paracoccidioidomycosis with a single brainstem lesion. A case report and literature review. Neuroradiol J 2010; 23 (04) 454-458
  • 22 Buccheri R, Benard G. Opinion: Paracoccidioidomycosis and HIV immune recovery inflammatory syndrome. Mycopathologia 2018; 183 (03) 495-498
  • 23 de Almeida SM, Roza TH. HIV immune recovery inflammatory syndrome and central nervous system paracoccidioidomycosis. Mycopathologia 2017; 182 (3-4): 393-396
  • 24 Pedroso VS, Vilela MC, Santos PC, Cisalpino PS, Rachid MA, Teixeira AL. Traffic of leukocytes and cytokine up-regulation in the central nervous system in a murine model of neuroparacoccidioidomycosis. Mycopathologia 2013; 176 (3-4): 191-199