A woman with kidney disease on peritoneal dialysis developed an unusual fungal infection caused by Hyphopichia burtonii, a fungus typically found in spoiled food. The infection was diagnosed using advanced protein analysis technology and treated with antifungal medication after removing the dialysis catheter. Though extremely rare in humans, this case shows that unusual fungi can cause serious infections in patients with weakened immune systems and highlights the importance of considering these organisms when standard treatments don’t work.