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The tipping point: The critical role of therapeutic apheresis in a case of refractory acquired hemophilia
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Michael Losos Scott Scrape Sarita Joshi Aaron Shmookler Jian Chen 《Journal of clinical apheresis》2017,32(6):564-566
Acquired hemophilia A (AHA) is a rare autoimmune disorder that leads to factor VIII (FVIII) deficiency via autoantibody formation. Standard treatment options include FVIII bypassing factors and immunosuppression. However, the role of therapeutic plasma exchange (TPE) is not clear in the treatment of AHA. We present a case of idiopathic AHA in a 66 year old female with severe bleeding and a FVIII inhibitor of 17.6 Bethesda units (BU). She failed to respond to standard treatment including maximum dose of recombinant FVIIa (rFVIIa), rituximab, and other immunosuppressive agents. Her FVIII inhibitor rapidly increased to 140 BU and FVIII was below 5%. TPE was initiated 3 weeks after admission and her bleeding stabilized after the first treatment and completely stopped after three treatments. Repeat testing revealed increased FVIII to 15% and FVIII inhibitor decreased to 2.0 BU. After an additional TPE treatment, her FVIII increased to 27% and FVIII inhibitor decreased to 0.6 BU and she was discharged without bleeding 40 days after admission. In this case, TPE played a critical role in reducing FVIII inhibitor, which resulted in a recovery of FVIII activity and hemostasis. Therefore, TPE should be initiated early in AHA patients with bleeding and high titer of FVIII inhibitor. 相似文献
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David N. Paglia Aaron Wey Jeremy Hreha Andrew G. Park Catherine Cunningham Linda Uko Joseph Benevenia J. Patrick O'Connor Sheldon S. Lin 《Journal of orthopaedic research》2014,32(5):727-734
This study evaluated the efficacy of using calcium sulfate (CaSO4) as a carrier for intramedullary delivery of an organic vanadium salt, vanadyl acetylacetonate (VAC) after femoral fracture. VAC can act as an insulin‐mimetic and can be used to accelerate fracture healing in rats. A heterogenous mixture of VAC and CaSO4 was delivered to the fracture site of BB Wistar rats, and mechanical testing, histomorphometry, micro‐computed tomography (micro‐CT) were performed to measure healing. At 4 weeks after fracture, maximum torque to failure, effective shear modulus, and effective shear stress were all significantly higher (p < 0.05) in rats treated with 0.25 mg/kg VAC–CaSO4 as compared to carrier control rats. Histomorphometry found a 71% increase in percent cartilage matrix (p < 0.05) and a 64% decrease in percent mineralized tissue (p < 0.05) at 2 weeks after fracture in rats treated with 0.25 mg/kg of VAC–CaSO4. Micro‐CT analyses at 4 weeks found a more organized callus structure and higher trending maximum connected z‐ray. fraction for VAC–CaSO4 groups. Evaluation of radiographs and serial histological sections at 12 weeks did not show any evidence of ectopic bone formation. As compared to previous studies, CaSO4 was an effective carrier for reducing the dose of VAC required to accelerate femoral fracture healing in rats. © 2013 Orthopaedic Research Society. Published by Wiley Periodicals, Inc. J Orthop Res 32:727–734, 2014. 相似文献
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