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

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.11. de Almeida JN, Peçanha-Pietrobom PM, Colombo AL. Paracoccidioidomycosis in immunocompromised patients: a literature review. J Fungi. 2019;5(1):2. Available from: https://doi.org/10.3390/jof5010002
https://doi.org/10.3390/jof5010002...
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 immunosuppression22. Reis F, Collier PP, Souza TF, Lopes GP, Bronzatto E, Junior NAS, et al. Neuroparacoccidioidomycosis (NPCM): Magnetic resonance imaging (MRI) findings. Mycopathologia. 2013;175:181-6. Available from: https://doi.org/10.1007/s11046-012-9607-y.
https://doi.org/10.1007/s11046-012-9607-...
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The patient responded well to treatment with voriconazole followed by cotrimoxazole. This treatment schedule was based on standard antifungal therapy33. Shikanai-Yasuda MA, Mendes RP, Colombo AL, de Queiroz-Telles F, Kono ASG, Paniago AMM, et al. Brazilian guidelines for the clinical management of paracoccidioidomycosis. Rev Soc Bras Med Trop. 2017;50(5):715-40. Available from: https://doi.org/10.1590/0037-8682-0230-2017.
https://doi.org/10.1590/0037-8682-0230-2...
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ACKNOWLEDGMENTS

We offer our deepest thanks to the institutions that provided technical support for the development and implementation of this study.

REFERENCES

  • 1
    de Almeida JN, Peçanha-Pietrobom PM, Colombo AL. Paracoccidioidomycosis in immunocompromised patients: a literature review. J Fungi. 2019;5(1):2. Available from: https://doi.org/10.3390/jof5010002
    » https://doi.org/10.3390/jof5010002
  • 2
    Reis F, Collier PP, Souza TF, Lopes GP, Bronzatto E, Junior NAS, et al. Neuroparacoccidioidomycosis (NPCM): Magnetic resonance imaging (MRI) findings. Mycopathologia. 2013;175:181-6. Available from: https://doi.org/10.1007/s11046-012-9607-y.
    » https://doi.org/10.1007/s11046-012-9607-y
  • 3
    Shikanai-Yasuda MA, Mendes RP, Colombo AL, de Queiroz-Telles F, Kono ASG, Paniago AMM, et al. Brazilian guidelines for the clinical management of paracoccidioidomycosis. Rev Soc Bras Med Trop. 2017;50(5):715-40. Available from: https://doi.org/10.1590/0037-8682-0230-2017.
    » https://doi.org/10.1590/0037-8682-0230-2017
  • Financial Support: None.

Publication Dates

  • Publication in this collection
    06 June 2022
  • Date of issue
    2022

History

  • Received
    17 Feb 2022
  • Accepted
    30 Mar 2022
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