Background. Unique characteristics, impact of therapy with antifungal agents, and outcome of infections with Scedosporium species were assessed in transplant recipients. Methods. The patients comprised a total of 80 transplant recipients with Scedosporium infections, including 13 patients from our institutions ( University of Pittsburgh Medical Center [ Pittsburgh, PA], University of Maryland [ Baltimore], Duke University Medical Center [ Durham, NC], Emory University [ Atlanta, GA], and Hospital Gregorio Maranon [ Madrid, Spain]) and 67 reported in the literature. The transplant recipients were compared with 190 non - transplant recipients with scedosporiosis who were described in the literature. Results. Overall, 69% of the infections in hematopoietic stem cell transplant ( HSCT) recipients and 53% of the infections in organ transplant recipients were disseminated. HSCT recipients, compared with organ transplant recipients, were more likely to have infections caused by Scedosporium prolificans (P= .045), to have an earlier onset of infection (P= .007), to be neutropenic (P< .001), and to have fungemia (P= .04). Time elapsed from transplantation to Scedosporium infection in transplant recipients has increased in recent years (P= .002). The mortality rate among transplant recipients with scedosporiosis was 58%. In a logistic regression model using amphotericin B as comparison treatment, voriconazole was associated with a trend towards better survival ( odds ratio [ OR], 10.40; P= .08). Presence of disseminated infection ( OR, 0.20; P= .03) predicted lower survival, and receipt of adjunctive surgery as treatment ( OR, 5.52; P= .02) independently predicted a better survival in this model. Conclusions. Scedosporium infections in transplant recipients were associated with a high rate of dissemination and a poor outcome overall. The use of newer triazole agents warrants consideration as a therapeutic modality for these infections.