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• W2894887218 abstract "SESSION TITLE: Critical Care 2 SESSION TYPE: Fellow Case Report Posters PRESENTED ON: 10/09/2018 01:15 PM - 02:15 PM INTRODUCTION: Because of its rarity, acquired hemophilia A (AHA) has limited data to support its management. Thus, consensus recommendations from available clinical trial, registry data, and clinical experience are used to guide the management of AHA. CASE PRESENTATION: A 55-year-old female with a history of hypertension, diabetes, morbid obesity, and tobacco use presented with recurrent bloody urine. Over the preceding two months, she had already undergone multiple cystoscopies with cauterizations of a bleeding artery near the left ureteral orifice. After an unsuccessful attempt to embolize the left anterior iliac artery, an explorative laparotomy was performed and revealed diffuse bleeding within the bladder concerning for coagulopathy. The prothrombin time and international normalized ratio were normal however the activated prothrombin time was elevated. The patient was ultimately found to have a factor VIII inhibitor level of 109 Bethesda Units (normal, < 5) and a factor VIII activity level of less than 1% (normal, 60-150%). The patient's bleeding was initially managed with high dose steroids, recombinant factor VIIa and VIII, prothrombin complex concentrate (PCC), desmopressin, aminocaproic acid bladder irrigation and intravenous immunoglobulin (IVIG). Because the patient continued to bleed, she was started on high-dose cyclophosphamide and received four sessions of plasma exchange followed by rituximab. Finally, porcine factor VIII and porcine factor VIII bound to von Willebrand factor was administered and the bladder irrigation resumed, after which the bleeding resolved. The patient was continued on oral cyclophosphamide when she was able to swallow pills. DISCUSSION: In patients with acquired hemophilia A and high inhibitor titers, IVIG and factor VIII replacement therapies such as desmopressin and factor VIII alone are ineffective, regardless of the dose. Instead, these patients require bypass agents that circumvent the need for FVIII (i.e. activated PCC or recombinant factor VIIa) and recombinant products such as porcine factor VIII, though there is a 3-5% risk of thrombotic events with bypassing agents and a risk of developing de-novo inhibitors against recombinant products. Studies show that the combination of cyclophosphamide and steroids may be superior to rituximab and steroids alone in achieving stable clinical remission (70% vs. 59% and 48%, respectively), but rituximab may result in fewer relapses (2%) compared to the combination therapy or steroids alone (10 and 11%, respectively). Finally, data to support the use of aminocaproic acid is limited and the role of IVIG and plasmapheresis is unclear. CONCLUSIONS: This case highlights the unique challenges of treating acquired hemophilia with high inhibitor titers. Despite limited clinical trial data, a thorough understanding of the pathophysiology of AHA is important to manage this disorder efficiently and effectively. Reference #1: Baudo F. Blood. 2012; 120(1):39-46. Reference #2: Kessler CM and Knöbl P. Eur J Haematol. 2015; 95 Suppl 81:36-44. Reference #3: Kruse-Jarres R. Am J Hematol. 2017; 92:695-705. DISCLOSURES: No relevant relationships by Amali Jayasinghe, source=Web Response No relevant relationships by Jennifer Parrish, source=Web Response No relevant relationships by Tomoko Tagawa, source=Web Response No relevant relationships by Janine Vintch, source=Web Response no disclosure on file for Showshan Yang-Ting" @default.
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• W2894887218 date "2018-10-01" @default.
• W2894887218 modified "2023-09-27" @default.
• W2894887218 title "MANAGEMENT OF PERSISTENT BLEEDING IN ACQUIRED HEMOPHILIA A WITH HIGH-TITER INHIBITOR" @default.
• W2894887218 doi "https://doi.org/10.1016/j.chest.2018.08.258" @default.
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