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Hepatic involvement in Wegener's granulomatosis: a case report.

Goritsas C, Paissios NP, Trigidou R, Delladetsima J - J Med Case Rep (2010)

Bottom Line: A liver biopsy demonstrated the presence of mild non-specific lobular hepatitis and periodic acid-Schiff positive Lafora-like inclusions in a large number of his liver cells.The patient was treated with prednisone and cyclophosphamide, which was followed by subsequent remissions of chest X-ray findings and liver function studies.We favor the hypothesis that hepatic vasculitis may be the cause of acute hepatocellular necrosis.

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Affiliation: Department of Internal Medicine, Sotiria General Hospital, 152 Mesogeion Avenue, Athens, 11527, Greece.

ABSTRACT

Introduction: We report the case of a 58-year-old Caucasian Greek man who presented with dry cough, fever, bilateral alveolar infiltrates and acute hepatitis.

Case presentation: After a lung biopsy, the patient was diagnosed with Wegener's granulomatosis. The diagnosis was supported by the presence of anti-proteinase-3 anti-neutrophil cytoplasmic antibodies. A liver biopsy demonstrated the presence of mild non-specific lobular hepatitis and periodic acid-Schiff positive Lafora-like inclusions in a large number of his liver cells. The patient was treated with prednisone and cyclophosphamide, which was followed by subsequent remissions of chest X-ray findings and liver function studies.

Conclusion: What makes this case worth reporting is the coexistence of liver inflammation with a biochemical profile of severe anicteric non-viral, non-drug induced hepatitis coinciding with the diagnosis of Wegener's granulomatosis. Our paper may be the first report of hepatic involvement in a patient diagnosed with Wegener's granulomatosis. The aetiological link between the two diseases is supported by the reversion of hepatitis after the immunosuppression of Wegener's granulomatosis. We favor the hypothesis that hepatic vasculitis may be the cause of acute hepatocellular necrosis.

No MeSH data available.


Related in: MedlinePlus

Liver function tests in the course of time (0 on the X axis stands for the first day of admission to our department).
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Figure 4: Liver function tests in the course of time (0 on the X axis stands for the first day of admission to our department).

Mentions: Our patient later received a combined treatment of 1 mg/kg/day prednisone and 2 mg/kg/day of cyclophosphamide. Our patient's status gradually improved with a remission of both the chest X-ray findings and the liver function studies 25 days after the initiation of treatment (Figure 4). After all signs of active disease had disappeared, a gradual tapering of corticosteroid and cyclophosphamide therapy was initiated. Eighteen months after his hospitalization, the patient is free of symptoms, with normal results for liver function tests and without any sign of relapse.


Hepatic involvement in Wegener's granulomatosis: a case report.

Goritsas C, Paissios NP, Trigidou R, Delladetsima J - J Med Case Rep (2010)

Liver function tests in the course of time (0 on the X axis stands for the first day of admission to our department).
© Copyright Policy - open-access
Related In: Results  -  Collection

License
Show All Figures
getmorefigures.php?uid=PMC2821396&req=5

Figure 4: Liver function tests in the course of time (0 on the X axis stands for the first day of admission to our department).
Mentions: Our patient later received a combined treatment of 1 mg/kg/day prednisone and 2 mg/kg/day of cyclophosphamide. Our patient's status gradually improved with a remission of both the chest X-ray findings and the liver function studies 25 days after the initiation of treatment (Figure 4). After all signs of active disease had disappeared, a gradual tapering of corticosteroid and cyclophosphamide therapy was initiated. Eighteen months after his hospitalization, the patient is free of symptoms, with normal results for liver function tests and without any sign of relapse.

Bottom Line: A liver biopsy demonstrated the presence of mild non-specific lobular hepatitis and periodic acid-Schiff positive Lafora-like inclusions in a large number of his liver cells.The patient was treated with prednisone and cyclophosphamide, which was followed by subsequent remissions of chest X-ray findings and liver function studies.We favor the hypothesis that hepatic vasculitis may be the cause of acute hepatocellular necrosis.

View Article: PubMed Central - HTML - PubMed

Affiliation: Department of Internal Medicine, Sotiria General Hospital, 152 Mesogeion Avenue, Athens, 11527, Greece.

ABSTRACT

Introduction: We report the case of a 58-year-old Caucasian Greek man who presented with dry cough, fever, bilateral alveolar infiltrates and acute hepatitis.

Case presentation: After a lung biopsy, the patient was diagnosed with Wegener's granulomatosis. The diagnosis was supported by the presence of anti-proteinase-3 anti-neutrophil cytoplasmic antibodies. A liver biopsy demonstrated the presence of mild non-specific lobular hepatitis and periodic acid-Schiff positive Lafora-like inclusions in a large number of his liver cells. The patient was treated with prednisone and cyclophosphamide, which was followed by subsequent remissions of chest X-ray findings and liver function studies.

Conclusion: What makes this case worth reporting is the coexistence of liver inflammation with a biochemical profile of severe anicteric non-viral, non-drug induced hepatitis coinciding with the diagnosis of Wegener's granulomatosis. Our paper may be the first report of hepatic involvement in a patient diagnosed with Wegener's granulomatosis. The aetiological link between the two diseases is supported by the reversion of hepatitis after the immunosuppression of Wegener's granulomatosis. We favor the hypothesis that hepatic vasculitis may be the cause of acute hepatocellular necrosis.

No MeSH data available.


Related in: MedlinePlus