A 37-year-old man undergoing liver transplantation for cirrhosis caused by chronic hepatitis C and alcoholism developed large numbers of α1- antitrypsin (AAT) globules within hepatocytes of the transplanted liver during a 5-month period. This finding occurred simultaneously with severe recurrent hepatitis C. The AAT phenotype of this patient changed from MM before transplantation to MZ after transplantation; AAT levels in serum did not change significantly. After a second liver transplantation 6 months after the first, the AAT phenotype reverted to MM and globules were not detectable in the second transplanted liver. Severe recurrent hepatitis C and the appearance of AAT globules within the liver allograft of a heterozygous donor may be related. The recurrent infection may have induced accumulation of AAT protein, or, alternatively, the MZ liver may have been more susceptible to injury from recurrent viral infection.