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Giant Cell Arteritis Presenting as Cholestatic Hepatitis. Background: Severely deranged liver function tests (LFTs) are an atypical presentation of giant cell arteritis (GCA). Atypical presentations of GCA may result in missed or delayed diagnosis. This increases the risk of visual loss, the most feared outcome of GCA. Our patient presented with significant cholestatic derangement of his LFTs with a peak alkaline phosphatase level (ALP) of 3091 IU/L, which is the highest published level for patients with GCA. Case Presentation. Our patient was investigated for abnormal LFTs associated with sinus pain, fevers, and a dry cough. Bilateral temporal artery biopsies confirmed GCA. His symptoms and LFTs improved with corticosteroids. Conclusion: This is an unusual presentation of GCA and highlights the need to consider GCA in patients with unexplained cholestatic LFT abnormalities.