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Autoimmune hepatitis overlapping with primary sclerosing cholangitis in five cases

A N McNair¹, M Moloney, B C Portmann

¹Institute of Liver Studies, King's College Hospital, London, United Kingdom.

The American Journal of Gastroenterology

|June 13, 1998

Summary

Autoimmune hepatitis/primary sclerosing cholangitis overlap syndrome can present with jaundice and elevated liver enzymes. This condition often responds well to immunosuppressive therapy, including prednisolone and azathioprine.

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Area of Science:

Hepatology
Immunology
Gastroenterology

Background:

Autoimmune hepatitis (AIH) and primary sclerosing cholangitis (PSC) are distinct liver diseases.
Overlap syndrome between AIH and PSC is rare but presents unique diagnostic and therapeutic challenges.

Observation:

Five cases of AIH/PSC overlap syndrome were identified, predominantly in young males.
Patients exhibited jaundice, elevated aminotransferases and alkaline phosphatase, hyperglobulinemia with high IgG, and autoantibodies (ANA/SMA).
Liver biopsies showed moderate to severe interface hepatitis with biliary features in most cases.

Findings:

All patients met criteria for definite AIH and responded well to immunosuppressive therapy (prednisolone, azathioprine).
Relapses occurred upon treatment reduction or withdrawal.

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Cholangiographic PSC features were present in three patients initially and developed in two others over time.

Only two patients had inflammatory bowel disease, contrasting with literature suggesting a strong association with ulcerative colitis.

Implications:

Consider AIH/PSC overlap syndrome in patients with unexplained hepatitis, autoantibodies, and biliary changes.
Immunosuppressive therapy may be beneficial for this specific overlap syndrome.
Cholangiography is recommended for diagnosis in suspected cases.