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In brief, plated cells were incubated for 30 min at 37 C, 5% CO 2 in the staining solution (HBSS containing 1 g ml 1 Hoechst 33342 (62247, Invitrogen, Thermo Fisher Scientific) and either 5 M CellROXTM, 1 M FerroOrange or 1 M BODIPY 581/591 C11)

Methods: Patients within the Childhood Liver Disease Research Network (ChiLDReN) enrolled in the Longitudinal Study of Genetic Causes of Intrahepatic Cholestasis (LOGIC) study were identified with BASD by local centers via biochemical evidence based on mass spectrometry analysis of urine/serum or from biallelic pathologic mutations in genes encoding enzymes in the bile acid synthetic pathway