An outbreak of food poisoning resulting in 13 deaths in children occurred in Malaysia during the Chinese Festival of the Nine-Emperor Gods in 1988. The offending food was a Chinese noodle called 'Loh See Fun' (LSF). The source was traced to a factory where a banned food preservative was added to make the LSF. The food poisoning was attributable to aflatoxins and boric acid. The clinical features included vomiting, pyrexia, diarrhoea, abdominal pain, anorexia, giddiness, seizures, and eventual coma. Initially, many presented with a Reye-like syndrome. Eleven post-mortem examinations were performed. The pathological findings included extensive coagulative necrosis of the liver with proliferative 'ductal/ductular metaplasia of the hepatocytes'. Giant cell formation, central vein sclerosis, bile stasis, and steatosis were also noted. There was presence of acute tubular necrosis, superficial upper gastrointestinal erosions, and ensuing encephalopathy. The eventual cause of death is acute hepatic and renal failure.
Intrahepatic cholangiocarcinoma is an uncommon disease with surgical resection as the only potentially curative modality of treatment. The pattern of failure in such tumours is recurrence in the remnant liver or metastases to lymph nodes, peritoneum and bones (1, 2). We report a case of intrahepatic cholangiocarcinoma with adrenal metastasis that was treated with simultaneous hepatic and adrenal resection. Adrenal metastasis from intrahepatic cholangiocarcinoma has not yet been reported in the published literature.A 68-year-old Indonesian gentleman was diagnosed with synchronous adrenal nodule with a large tumour in the left lateral segment of the liver. Left lateral segmentectomy with left adrenalectomy was performed on 6 November 2004. The histopathology report of the resected specimen revealed a tumour (9.0 Â 7.0 Â 6.8 cm size) in the liver with features of moderately differentiated intrahepatic cholangiocarcinoma. The nodule in the left adrenal gland was 1 cm in size and had histological features of metastatic cholangiocarcinoma ( Fig. 1).Following postoperative recovery, he was treated with four cycles of Gemcitabine and Cisplatin. In April 2006, he had recurrence of the disease in bone (right ilium), both lungs, and in the left adrenal bed. He received palliative radiotherapy for the painful lesion in the right ilium and was started on palliative chemotherapy with Gemcitabine and Xeloda. Two years following adrenalectomy, he is alive with recurrent disease.The options of treatment of a metastatic cholangiocarcinoma are limited. Such patients are generally treated with palliative chemotherapy or supportive care. Lessons from other solid malignancies like colorectal cancers and sarcomas have emphasized the value of surgical resection of metastatic disease when feasible (3,4). As our patient had a solitary adrenal metastasis, he was treated with simultaneous liver and adrenal resection. This is the first report of adrenal metastasis from intrahepatic cholangiocarcinoma. The patient developed recurrence in spite of an aggressive treatment with surgical resection and postoperative chemotherapy. The therapeutic options need to be further explored in order to provide hope to patients with metastatic cholangiocarcinoma.
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