356 Pulmonary Arterial Hypertension Associated to Antiphospholipid Syndrome in an Infant

Raul Dorbeker, Daniela Lopez Ortiz, Luisa Gamez, Alonso Gutierrez Hernandez
2012 The World Allergy Organization Journal  
Results: 32 years old female was referred because of fluctuating elevations of aminotransferases (AT) (4-to 19-fold normal) known last 6 months. She had positive HCV-RNA by RT-PCR an untypable genotype. Liver biopsy revealed chronic active hepatitis grade 2, stage 2. She also had Hashimotos thyroiditis with high titres of antithyroid antibodies. After 12 weeks of treatment with Intron A 3MU TIW and RBV 1000 mg/d, HCV RNA was negative and AT fluctuated between normal and 2-fold normal. One week
more » ... fter completing 48 weeks of treatment, AT showed 11-fold increase, but HCV remained negative. Indirect immunofluorescence became positive for ASMA (31) and ANA (41). A repeated liver biopsy showed no improvement. A therapeutic trial with RBV was started and within 11 weeks of this treatment her symptoms markedly improved and AT decreased to 2-fold. Unfortunately, cessation of RBV resulted in immediate reversion of symptoms and elevation of AT to its previous state. After restarting RBV therapy no response was observed within 2 weeks and the treatment was stopped. She was then put on prednisone 30 mg/d and azathioprine 50 mg/d. Within 6 weeks her symptoms resolved and AT and IgG became completely normal. The patient remains in remission with prednisone 5 mg and azathioprine 50 mg/ d. A re-evaluation of the primary diagnosis of hepatitis C was warranted because of unusually high AT activity and repeatedly negative anti-HCV using a third generation assay. Conclusions: This case may provide support for the hypothesis that RBV may dampen autoimmune reaction induced by IFN alpha and potentiate its anti-inflammatory effects.
doi:10.1097/01.wox.0000412119.03404.97 fatcat:odj2dofptbglhjhb5d5lkqjptu