Recurrence of autoimmune hepatitis after orthotopic liver transplantation and after reduction in immunosuppressive treatment is reported in a 43-year-old man. Diagnosis was based on biological, serological and morphological data, as well as on a complete biological recovery following steroid resumption. Recurrence was observed only 4 months after transplantation, when an abrupt increase in liver function tests was observed in association with a mainly lobular hepatitis in liver biopsy. Such a histological appearance has been previously described in the case of recurrence after immunosuppressive treatment weaning in non-transplanted patients. In contrast, this histological feature has not been reported in the rare transplanted patients in whom such recurrence has been observed. Thereafter, the evolution of the disease in our patient remained slow, likely dampened by the administration of cyclosporine and azathioprine. Morphological features are detailed, and importance of immunosuppressive therapy in patients transplanted for end-stage autoimmune hepatitis is emphasized.