--- a +++ b/processing/MACCROBAT/28248858.txt @@ -0,0 +1,15 @@ +A 31-year-old man developed diabetes insipidus with urine volume up to 10 to 20 L every 24 hours in 2003. +Four years later, he complained of fatigue, anorexia, jaundice and pruritus, and a symptomatic occipital mass. +Laboratory tests showed an abnormal liver enzyme (Table 1), the patient was negative for hepatitis viruses. +As shown in Fig.1, abdominal MRI showed multiple low-density lesions in the liver on the T1-weighted image and obvious expansion of the intrahepatic bile duct on the T2-weighted image. +Magnetic resonance cholangiopancreatography revealed multifocal intrahepatic bile duct strictures and dilatation, but the common hepatic duct was normal, it was highly suggestive of SC. +The neurohypophyseal area MRI showed the thickened hypothalamic nuclei and a low-density signal of 4.9 × 5.6 mm in size in the hypothalamic-pituitary area. +A multisystem, high-risk organ LCH was confirmed after occipital mass was biopsied in the local hospital, the patient was given ursodeoxycholic acid 150 mg 3 times a day. +Then, he began to receive a course of COEP chemotherapy (cyclophosphamide, 1000 mg; vincristine, 2 mg; epirubicin, 90 mg; and prednisone, 90 mg) in 2009. +However, on the 5th day of the 1st COEP chemotherapy, the patient appeared to severe liver function injury with an obvious increase of serum bilirubin (Table 1). +He refused further chemotherapy. +One year later, the patient developed liver decompensation with bleeding esophageal varices, ascites, and splenomegaly, and he was referred to LT (Model for End Stage Liver Disease score 17). +He underwent successful orthotopic LT in November 2011 in our center, and the donor came from voluntary deceased citizen organ donation in China. +Liver histopathology after LT revealed micronodular cirrhosis with SC and positive immunostaining (CD1a and S100), suggestive of LCH involving in the liver (Fig.2). +Postoperatively, the man continued to be immunosuppressed with tacrolimus and mycofenolate mofetil. +The patient is currently well with normal liver function and no evidence of recurrence of LCH for 4 and a half years follow-up.