Most episodes of fungal sinusitis in immunocompromised patients are caused by Aspergillus species. To treat such infections, surgical debridement and anti-fungal therapy have been recommended; it is also clear however that an adequate neutrophil count is important in controlling such infections. We report a case in which fungal sinusitis was shown to be due to P. boydii and in which the infection recurred over a period of 2 years during episodes of neutropenia in spite of vigorous surgical and medical therapy.