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Paracoccidioidomycosis Induced by Immunosuppressive Drugs in a Patient with Rheumatoid Arthritis and Bone Sarcoma: Case Report and Review of the Literature

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Abstract

Paracoccidioidomycosis (PCM) is a systemic mycosis caused by Paracoccidioides brasiliensis, which is endemic in many regions of Latin America. We describe the case of a 60-year-old man from a region endemic for PCM who presented with a long history of left hip pain. He had been treated over the past 3 years with immunosuppressive drugs (methotrexate, leflunomide, and adalimumab) for rheumatoid arthritis (RA). A hip radiograph showed lytic bone lesions, and a chest radiograph showed an expansive excavated lesion in the left lung, suggestive of a lung cancer with bone metastases. A left hip joint biopsy was inconclusive, but histological analysis of a surgical lung biopsy specimen was consistent with P. brasiliensis infection. Treatment with intravenous amphotericin B (50 mg/day) and hydrocortisone (25 mg/day) was initiated. However, increasing hip pain resulted in the amputation of the left lower limb, and the analysis of the surgical specimen revealed a diagnosis of bone sarcoma. Postoperatively, the patient developed sepsis and died approximately 1 month later. To our knowledge, this is the first report of PCM in a patient with RA who had been treated with immunosuppressive drugs, in particular TNF-α blocking agents. The atypical presentation (left hip pain alone) emphasizes the importance of considering PCM in the differential diagnosis of patients with pulmonary lesions and osteolytic lesions who live in a region endemic for PCM. This case report also demonstrates that health professionals in these regions must pay close attention to patients receiving immunosuppressive drugs because of the possibility of reactivating quiescent P. brasiliensis lesions.

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References

  1. Castón-Osorio JJ, Rivero A, Torres-Cisneros J. Epidemiology of invasive fungal infection. Int J Antimicrob Agents. 2008;32(Suppl.2):103–9.

    Article  Google Scholar 

  2. Shikanai-Yasuda MA, Telles Filho FQ, Mendes RP, Colombo AL, Moretti ML. Guidelines in paracoccidioidomycosis. Rev Soc Bras Med Trop. 2006;39(3):297–310.

    Article  PubMed  Google Scholar 

  3. Londero AT, Santos JW, Silva LA, Ramos CD. Paracoccidioidomicose induzida por droga imunssupressora em paciente com lúpus eritrematoso sistêmico. Arq Bras Med. 1988;62(3):185–7.

    Google Scholar 

  4. Santos JW, Andrade CF, Lopes TSA, Londero AT. Pseudotumoral presentation of chronic pulmonary paracoccidioidomycosis: report of a case. Mycopathologia. 1996;134:135–6.

    Article  PubMed  Google Scholar 

  5. Shikanai-Yasuda MA, Conceição YMT, Kono A, Rivitti E, Campos AF, Campos SV. Neoplasia and paracoccidioidomycosis. Mycopathologia. 2008;165:303–12.

    Article  PubMed  CAS  Google Scholar 

  6. Braun J, Rau R. An update on methotrexate. Curr Opin Rheumatol. 2009;21(3):216–23.

    Article  PubMed  CAS  Google Scholar 

  7. Bongartz T, Sutton AJ, Sweeting MJ, Buchan I, Matteson EL, Montori V. Anti-TNF antibody therapy in rheumatoid arthritis and the risk of serious infections and malignancies: a systematic review and meta-analysis of rare harmful effects in randomized controlled trials. JAMA. 2006;295(19):2275–85.

    Article  PubMed  CAS  Google Scholar 

  8. Alonso-Ruiz A, Pijoan JI, Ansuategui E, Urkaregui A, Calabozo M, Quintana A. Tumor necrosis factor alpha drugs in rheumatoid arthritis: systematic review and metaanalysis of efficacy and safety. BMC Musculoskelet Disord. 2008;9(52):1–27.

    Google Scholar 

  9. Benard G, Kavakama J, Mendes-Giannini MJ, Kono A, Duarte AJ, Shikanai-Yasuda MA. Contribution to the natural history of paracoccidioidomycosis: identification of the primary pulmonary infection in the severe acute form of the disease-a case report. Clin Infect Dis. 2005;40:1–4.

    Article  Google Scholar 

  10. Trad HS, Trad CS, Junior JE, Muglia VF. Revisão Radiológica de 173 Casos Consecutivos de Paracoccidioidomicose. Radiol Bras. 2006;39(3):175–9.

    Article  Google Scholar 

  11. Severo LC, Palombini BC, Utz E, Braun SN. Paracoccidioidomicose pulmonar resultante de reativação de lesão quiescente, em paciente imunossuprimido. J Pneumolog. 1980;6(1):21–2.

    Google Scholar 

  12. Borgia G, Reynaud L, Cerini R, Ciampi R, Schioppa O, Dello Russo M, Gentile I, Piazza M. A case of paracoccidioidomycosis: experience with long-term therapy. Infection. 2000;28(2):119–20.

    Article  PubMed  CAS  Google Scholar 

  13. Marchiori E. Aspectos radiológicos das lesões osteoarticulares na paracoccidioidomicose—Considerações Sobre 20 Casos. Radiol Bras. 1989;22:5–16.

    Google Scholar 

  14. Wanke B. Paracoccidioidomicose: Inquérito Intradérmico com Paracoccidioidina em Zona Urbana do Município do Rio de Janeiro. Tese à Faculdade de Medicina, Universidade Federal do Rio de Janeiro. 1976:16–17.

  15. Severo LC, Londero AT, Geyer GR, Porto NS. Acute pulmonary paracoccidioidomycosis in an immunosuppressed patient. Mycopathologia. 1979;68(3):171–4.

    Article  PubMed  CAS  Google Scholar 

  16. Sugar AM, Restrepo A, Stevens DA. Paracoccidioidomycosis in the immunosuppressed host: report of a case and review of the literature. Am Rev Respir Dis. 1984;129:340–2.

    PubMed  CAS  Google Scholar 

  17. Shikanai-Yasuda MA, Duarte MI, Nunes DF, Lacaz CS, Sabagga E, Abdala E, Duarte AJ, Lopes MH. Paracoccidioidomycosis in a renal transplant recipient. J Med Vet Mycol. 1995;33:411–4.

    Article  PubMed  CAS  Google Scholar 

  18. Zavascki AP, Bienardt JC, Severo LC. Paracoccidioidomycosis in organ transplant recipient: case report. Rev Inst Med Trop Sao Paulo. 2004;46(5):279–81.

    Article  PubMed  Google Scholar 

  19. Wallis SR. Infectious complications of tumor necrosis factor blockade. Curr Opin Infec Dis. 2009;22:402–9.

    Google Scholar 

  20. Smith JA, Kauffman CA. Endemic fungal infections in patients receiving tumor necrosis factor-α inhibitor therapy. Drugs. 2009;69(11):1403–15.

    Article  PubMed  CAS  Google Scholar 

  21. Tsiodras S, Samonis G, Boumpas DT, Kontoyiannis DP. Fungal infections complicanting tumor necrosis factor α blockade therapy. Mayo Clin Proc. 2008;83(2):181–94.

    Article  PubMed  CAS  Google Scholar 

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Authors and Affiliations

  1. Universidade Federal de Santa Maria, Santa Maria, RS, Brazil

    Tiago G. Woyciechowsky, Diogo C. Dalcin, José Wellington A. dos Santos & Gustavo T. Michel

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  1. Tiago G. Woyciechowsky
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  2. Diogo C. Dalcin
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  3. José Wellington A. dos Santos
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  4. Gustavo T. Michel
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Correspondence to Tiago G. Woyciechowsky.

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Woyciechowsky, T.G., Dalcin, D.C., dos Santos, J.W.A. et al. Paracoccidioidomycosis Induced by Immunosuppressive Drugs in a Patient with Rheumatoid Arthritis and Bone Sarcoma: Case Report and Review of the Literature. Mycopathologia 172, 77–81 (2011). https://doi.org/10.1007/s11046-011-9403-0

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