Sinusitis caused by Exserohilum rostratum after cord blood transplantation for myelodysplastic syndrome: A case report and literature review

Abstract

Invasive fungal disease is a serious infectious complication of allogeneic hematopoietic stem cell transplantation (HSCT). Exserohilum rostratum is a species causing phaeohyphomycosis, which rarely causes invasive disease in humans. We treated a case of sinusitis caused by E. rostratum after cord blood transplantation (CBT). A 60-year-old man with myelodysplastic syndrome, who had a medical history of an operation to correct deviation of the nasal septum, developed sinusitis caused by E. rostratum under prolonged profound neutropenia after a second CBT because of the graft rejection of the first transplantation. Liposomal amphotericin B improved the sinusitis. A literature review revealed nine reported cases of sinusitis caused by E. rostratum, including our case. Although five cases had severe neutropenia at onset (HSCT recipients, n = 2; aplastic anemia, n = 3), the remaining four had no preexisting immunosuppressive conditions. However, three of the four patients had preexisting nasal diseases with or without a history of surgery, as in our case. Excluding our case, the outcome was fatal in five neutropenic patients, whereas the four patients without neutropenia recovered. Although sinusitis caused by E. rostratum is rare, E. rostratum should be recognized as a possible pathogen causing sinusitis in highly immunosuppressed patients such as HSCT recipients. Preexisting nasal disease and/or nasal surgery could be risks for this infection.

Diagnosis, Prognosticators, and Management of Acute Invasive Fungal Rhinosinusitis: Multidisciplinary Consensus Statement and Evidence‐Based Review with Recommendations

“…In contrast, solid‐organ transplant susceptibility is largely due to the postoperative immunosuppressive regimen 245 . Cohort studies have reported overall mortality for patients with AIFS after receiving SCT or BMT to be 33% to 100% 52,246,247 . In a case–control study of 46 patients with AIFS, Chen et al.…”

“…245 Cohort studies have reported overall mortality for patients with AIFS after receiving SCT or BMT to be 33% to 100%. 52,246,247 In a case-control study of 46 patients with AIFS, Chen et al concluded that SCT was not an independent prognostic factor of AIFS (p = 0.68). 68 Kennedy et al similarly reported no statistically significant association between BMT and AIFS on univariate analysis (p = 0.68).…”

Invasive Fungal Rhinosinusitis Due to Co-infection with Mucormycosis and Exserohilum rostratum in a Patient with Acute Lymphoblastic Leukemia

“…The members of the heat-tolerant genus Exserohilum are ubiquitous in tropical and subtropical regions. However, less than ten cases of invasive non-allergic sinusitis due to Exserohilum have been described in the literature in haematological patients, who were typically pediatric and from warm areas [ 7 – 11 ]. In 2012, this fungus was the cause of an outbreak of meningitis associated with contaminated methylprednisolone injections [ 12 ].…”

Effect of Monocerin, a Fungal Secondary Metabolite, on Endothelial Cells

“…Among these species, E. rostratum has been described as a phytopathogen; however, an outbreak of fungal meningitis associated with contaminated methylprednisolone injections was reported by the Center of Diseases Control (CDC) in the United States in 2012 [ 9 ], culminating in the death of 8% of patients after 12 months of the diagnostic [ 10 ]. Cases of sinusitis [ 11 ] and keratitis [ 12 ] were also reported. Lately, this species has been successfully used as a model of rabbit meningoencephalitis [ 13 ].…”