Article

Mycopathologia

, Volume 172, Issue 1, pp 77-81

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

  • Tiago G. WoyciechowskyAffiliated withUniversidade Federal de Santa Maria Email author 
  • , Diogo C. DalcinAffiliated withUniversidade Federal de Santa Maria
  • , José Wellington A. dos SantosAffiliated withUniversidade Federal de Santa Maria
  • , Gustavo T. MichelAffiliated withUniversidade Federal de Santa Maria

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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.

Keywords

Paracoccidioidomycosis Paraccocidioides brasiliensis Immunosuppressive drugs TNF-α blocking agents Rheumatoid arthritis Bone sarcoma