Hemophagocytic lymphohistiocytosis associated with hepatocellular carcinoma.

A man in his 60s was admitted because of abdominal pain and fatigue. Contrast enhanced computed tomography (CECT) showed a hypovascular tumor, 7 cm in size, in the left lobe of liver. He had no history of alcohol consumption. HBs antigen and HCV antibody were negative. For definitive diagnosis, biopsy of the hepatic tumor was performed. After the biopsy, the patient suddenly got high fever, and blood tests showed WBC 22,000/L, Hb 8.9 g/dL, Plts 11.6 × 104/L, AST 140 IU/L, ALT 93 IU/L, LDH 635 U/L. He died on the following day despite of supportive therapy. Autopsy revealed that the hepatic tumor was poorly differentiated hepatocellular carcinoma (HCC) and that hemophagocytic macrophages were found in the bone marrow and spleen. Based on the pathological findings of autopsy, he was finally diagnosed with hemophagocytic lymphohistiocytosis (HLH) associated with HCC. HLH is a rare and life-threaded disorder of immune overactivation. Malignancy-associated HLH is well-known; however, it is usually associated with malignant lymphoma. To our knowledge, this is the first reported case of HLH associated with HCC, which was diagnosed by autopsy. Although extremely rare, our case highlights that HLH should be considered as a differential diagnosis of unknown high fever and bicytopenia in patients with solid tumors, including HCC.

Possibility of Interstitial Lung Disease as a Phlebosclerotic Colitis Manifestation.

Phlebosclerotic colitis presents with ischemic bowels and calcification of the mesenteric veins. Owing to its rarity, we have little information on the complications of this disease. Herein, we report on a 77-year-old woman with phlebosclerotic colitis and interstitial lung disease. She was diagnosed as having phlebosclerotic colitis by CT and colonoscopy. At the same time, chest CT also showed interstitial lung disease. After 4 years, she experienced exacerbation of interstitial lung disease. She recovered without treatment. The occurrence of interstitial lung disease may have been associated with her phlebosclerotic colitis.