Background: The overlap syndrome of primary biliary cholangitis (formerly called primary biliary cirrhosis) and primary sclerosing cholangitis is an extremely rare condition that has never been described in association with other immune-mediated diseases, including psoriatic arthritis. While treatment with anti-Tumour Necrosis Factor-alpha (TNF-alpha) agents has proved to be effective in inflammatory arthropathies such as psoriatic arthritis, they have been employed in only a limited number of patients with autoimmune hepatitis, and their effectiveness is unclear. Case presentation: We report the case of a 51-year-old female affected with psoriatic arthritis concomitant to overlapping primary biliary cholangitis and primary sclerosing cholangitis in whom 28 months of adalimumab treatment improved the symptoms of the inflammatory arthropathy as well as those of both cholangiopathies. Conclusion: Our results suggest that further studies examining the therapeutic role of this particular TNF-a blocker are warranted in cholestatic autoimmune hepatitis patients, and in particular in those individuals in whom the disease is associated with inflammatory arthropathies.