Clinical challenge: fatal mucormycotic osteomyelitis caused by Rhizopus microsporus despite aggressive multimodal treatment

Abstract

BackgroundMucormycosis is an invasive mycotic disease caused by fungi in the zygomycetes class. Although ubiquitous in the environment, zygomycetes are rarely known to cause invasive disease in immunocompromised hosts with a high mortality even under aggressive antifungal and surgical therapy. Clinically, mucormycosis frequently affects the sinus occasionally showing pulmonary or cerebral involvement. However skeletal manifestation with Rhizopus microsporus (RM) osteomyelitis leading to emergency surgical proximal femoral resection with fatal outcome has not been described yet.Case presentationWe report the case of a 73-year-old male suffering from myelodysplastic syndrome with precedent bone marrow transplantation. Six months after transplantation he consulted our internal medicine department in a septic condition with a four week history of painful swelling of the right hip. Radiography, computed tomography and magnetic resonance imaging revealed multiple bone infarcts in both femurs. In the right femoral head, neck and trochanteric region a recent infarct showed massive secondary osteomyelitis, breaking through the medial cortex. Emergency surgical proximal femoral resection was performed due to extensive bone and soft tissue destruction. Microbiological and basic local alignment search tool (BLAST) analysis revealed RM. Amphotericin B and posaconazole treatment with septic revision surgery was performed. However the disease ran a rapid course and was fatal two months after hospital admission.ConclusionThis alarming result with extensive RM osteomyelitis in the proximal femur of an immunocompromised patient may hopefully warn medical staff to perform early imaging and aggressive surgical supported multimodal treatment in similar cases.Electronic supplementary materialThe online version of this article (doi:10.1186/1471-2334-14-488) contains supplementary material, which is available to authorized users.

Osteoarticular Infections Caused by Non-Aspergillus Filamentous Fungi in Adult and Pediatric Patients

“…Osteoarticular mucormycosis is a highly destructive infection with poor prognosis, if not diagnosed early. 151 …”

Therapy of Mucormycosis

“…Similarly, a study in neutropenic mice infected with R. oryzae found that amphotericin B and posaconazole dose combination did not prolong survival or decrease organ fungal burden to a greater extent than AMB monotherapy [ 79 ]. Human studies evaluating polyene-triazole combination for the treatment of mucormycosis are limited [ 76 , 80 , 81 , 82 , 83 , 84 , 85 ]. In one retrospective case series, 32 patients with hematological malignancies or aplastic anemia, and mucormycosis unresponsive to prior monotherapy (mainly with LAMB), were treated with combination of polyene and posaconazole [ 86 ].…”

A Case Series of Pulmonary Mucormycosis Caused by Rhizopus Microsporus

“…Mucormycosis is an emerging angioinvasive infection caused by ubiquitous filamentous fungi of the order Mucorales that commonly affect the sinuses (39%) and the lungs (24%) [5,6]. To establish infection, Mucorales species form spores that are small (3-11 µm) enough to reach the distal alveolar spaces and then overcome phagocytosis by macrophages and neutrophils to germinate into hyphae [3].…”