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Drug‐induced vanishing bile duct syndrome (VBDS) is a rare complication of severe cholestatic hepatitis characterized by progressive ductopenia despite withdrawal of the offending agent. Prognosis ranges from gradual recovery to liver failure requiring transplantation. We report the first known pediatric case of biopsy‐confirmed VBDS following atovaquone–proguanil exposure. An 11‐year‐old girl developed severe cholestatic hepatitis approximately 2 months after a 21‐day course of malaria prophylaxis. Liver biopsy demonstrated marked ductopenia, with bile ducts identified in only 2 of 22 portal tracts. Despite a severe presentation, she achieved complete biochemical and clinical recovery within 6 months. This case expands the spectrum of drug‐induced VBDS in children and highlights the potential for favorable outcomes despite significant duct loss.
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Kaihlanen et al. (2026) studied this question.
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