α-2b interferon and oral clodronate for Gorham's disease
α-2b interferon and oral clodronate for Gorham's disease
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DOI：
10.1016/s0140-6736(05)63639-2
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发表时间：
1997-12-20
期刊：
影响因子：
168.9
通讯作者：
Åström, G
中科院分区：
文献类型：
--
作者：
Hagberg, F;Lamberg, K;Åström, G
An 83-year-old man was treated with 0· 2 mg moxonidine daily because of moderate hypertension. No other medication was given. The patient’s past history was uneventful. A routine laboratory check before treatment with moxonidine showed normal values for leucocytes, platelets, haemoglobin, creatinine, bilirubin, and liver enzymes. 9 months after the start of moxonidine, he developed itching and nausea. Painless jaundice then occurred, accompanied by a maculopapular rash.Ultrasound examination showed a normal-sized liver with a 2 cm diameter cyst in the left lobe, but no dilatation of the biliary ducts and no stones. Bilirubin rose to 173 µmol/L and transaminases were higher than 1000 U/L, whereas there was only a slight elevation of gamma-glutamyl transpeptidase and alkaline phosphatase (table). Hepatitis B surface antigen and hepatitis B and C antibodies were negative. Hepatitis A IgG antibodies were positive but IgM antibodies negative. Because of rising bilirubin (up to 412 µmol/L), a coagulation disorder, and hypoalbuminaemia (17 g/L) with generalised oedema the patient was admitted to the hospital 10 days after discontinuation of moxonidine. Abnormal ultrasound confirmed previous results with no dilatation of the bile ducts and no other pathological findings. Liver biopsy showed severe localised intralobular and intraportal lymphoplasmocellular infiltrates, including some neutrophils and intracytoplasmatic intracanalicular cholestasis. There was only mild proliferation of the small bile ducts but considerable localised pericellular fibrosis. Antinuclear, antimitochondrial, and antineutrophil antibodies in the serum were negative and α-1-antitrypsin, caeruloplasmin, and copper levels were normal. A skin biopsy
