Cytomegalovirus reactivation in autoimmune liver diseases. Can we diagnose it earlier?: A case report

Abstract

Rationale: Clinical evidence showing that biliary inflammation predisposes individuals to cytomegalovirus (CMV) disease has been growing, and some studies even indicate that this risk may surpass that associated with immunosuppression alone. Autoimmune hepatitis (AIH) can be challenging to manage and often necessitates lifelong use of immunosuppressive regimens. Additionally, AIH can be associated with chronic biliary diseases in conditions known as overlap syndromes. In one such syndrome, AIH is associated with primary sclerosing cholangitis, a well-established cause of chronic biliary inflammation that results in ductal destruction and fibrosis, thus combining 2 significant risk factors for CMV disease.

Patient concerns: We report a case of a young woman with AIH and primary sclerosing cholangitis since her childhood. She was taking prednisone, azathioprine, and tacrolimus and experienced jaundice. Azathioprine and tacrolimus serum levels were adequate and AIH was well controlled, whereas total bilirubin, alkaline phosphatase, and gamma-glutamyl transferase were elevated. After the virus reactivation, the patient has been concerned about the immunosuppressive drugs she has been taken.

Diagnoses: CMV reactivation was diagnosed only after hospital admission when serum polymerase chain reaction was 3153 copies/mL.

Intervention: Intravenous ganciclovir resulted in rapid improvement.

Outcomes: The patient achieved full recovery and no further hospitalization was needed.

Lessons: Patients with similar conditions need careful vigilance for biliary CMV reactivation. Even if polymerase chain reaction is not available, CMV serology should be performed as soon as the symptoms are detected, avoiding delays in diagnosis and treatment.

Keywords: autoimmune diseases; biliary tract diseases; cholestasis; cytomegalovirus; immunosuppression therapy; intrahepatic.

Figure 1.

Liver biopsies showing findings of autoimmune hepatitis and primary sclerosing cholangitis. (A) Septal inflammation with spillover by lymphocytes (arrow). (B) Portal tract with lymphocytic inflammation centered on a bile duct (arrow). (C) Portal septal fibrosis (arrow). (A) Hematoxylin and Eosin stain (2004). (B) Masson trichrome stain (2007). (C) Reticulin stain (2020).

Figure 2.

Timeline of the main events. AIH = autoimmune hepatitis, CMV = cytomegalovirus, CT = computed tomography, MRI = magnetic resonance imaging.