Osteomyelitis and aortic arteritis with thrombosis as primary manifestations of severe paracoccidioidomycosis: a case report

Abstract

Paracoccidioidomycosis (PCM) configures a deep mycosis caused by Paracoccidioides spp., a neglected tropical disease. We describe a fatal case of PCM that manifested itself as osteomyelitis with thrombosis in the iliac artery in a man with frequent contact with an endemic region in Sao Paulo, Brazil. A 67-year-old man living in an endemic area presented with osteomyelitis of the femur and iliac artery thrombosis on admission to the hospital. Computed tomography of the chest showed multiple cavitated pulmonary nodules. The patient rapidly progressed to irreversible respiratory failure. The autopsy showed disseminated PCM and iliac artery thrombosis. Laboratory investigation confirmed P. brasiliensis infection with a strain identified in Sao Paulo as P. brasiliensis complex by phylogenetic analysis. Atypical PCM remains a diagnostic challenge. Increased awareness of the sites of infection and its clinical presentations will improve patient management.

Figure 1. Severe paracoccidioidomycosis with arteritis and thrombosis of the iliac artery: (A) Radiography of the right femur showing multiple lytic lesions in the right femur, with periosteal reaction (arrows); (B) Nuclear magnetic resonance showing deep abscess in the right thigh associated with femoral lytic lesions; (C) Ischemia of the right thigh; (D) Fistula on the lateral area the right thigh associated with osteomyelitis, soft tissue abscess, and cutaneous infection by Paracoccidioides; (E) Right toe with ischemia and distal gangrene; (F) Atherosclerotic aorta with thrombus occluding right iliac artery (arrow); (G-I) Organized mycotic thrombus within the right iliac artery, with numerous yeasts in the medial layer (blue arrow) and in the thrombus (black arrow) (HE, 50×) compatible with Paracoccidioides spp (H: arrow, HE, 400×, and insets, by Grocott stain, 400×). The yeasts are round, with wide size range, and multiple budding (H, insets); (I) Immunohistochemistry reaction staining in brown immune cells expressing IL-17 in the aortic granuloma with Paracoccidioides spp. forms (Peroxydase, 400×). HE: hematoxylin-eosin.

Figure 2. Phylogenetic analysis of P. brasiliensis associated with a fatal human case. The maximum likelihood phylogenetic tree of the genus Paracoccidioides was based on partial gp43 gene sequences (n = 75). The black circles represent the bootstrap support values, and their sizes vary according to the support value (0 to 100). The black triangles highlight the samples characterized here from the atypical case of PCM, in which the two samples cluster with sequences characterized a P. brasiliensis complex.