Corrosive sclerosing cholangitis is a rare postoperative complication of hepatic cystic echinococcosis. Although corrosive sclerosing cholangitis is rare, it progresses rapidly and lacks effective treatments, which usually results in a poor prognosis. This case report retrospectively analyzed the treatment and diagnosis of a case with corrosive sclerosing cholangitis following surgical treatment of hepatic cystic echinococcosis, so as to provide insights into the diagnosis and treatment of corrosive sclerosing cholangitis.