Paecilomyces variotii has previously been reported as a causative pathogen for peritonitis in patients on continuous ambulatory peritoneal dialysis and shown to be usually sensitive to amphotericin B and resistant to voriconazole. We report the first case, to our knowledge, of P. variotii peritonitis in a liver transplant patient, which was unresponsive to initial liposomal amphotericin B (L-AmB) treatment and resolved dramatically after the addition of voriconazole. The present case provides evidence for the clinical and microbiological effectiveness of voriconazole combined with L-AmB in treating P. variotii peritonitis refractory to initial L-AmB treatment.