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Acquired factor VIII inhibitor in a patient with dermatomyositis--a case study. 2008

Ayodele Ayoola, and Wajahath Mohsini, and Sein Aung
Department of Medicine, Franklin Square Hospital Center, Baltimore, MD 21237, USA. ayodeleayoola@hotmail.com

BACKGROUND Acquired factor VIII inhibitor is an antibody formed against FVIII and seen in patients without FVIII replacement therapy. This entity has been associated with both autoimmune disease and infections. METHODS We report the case of a 65-year-old man with dermatomyositis who presented with a bleeding episode following venipuncture and hematuria. His coagulation parameters were followed as well as the inhibitor level. His acquired FVIII inhibitor was managed successfully with rituximab. CONCLUSIONS Our patient's bleeding episodes resolved and the inhibitor level was undetectable after 12 months. This is the first case of acquired FVIII inhibitor in association with dermatomyositis.

UI MeSH Term Description Entries
D008297 Male Males
D003882 Dermatomyositis A subacute or chronic inflammatory disease of muscle and skin, marked by proximal muscle weakness and a characteristic skin rash. The illness occurs with approximately equal frequency in children and adults. The skin lesions usually take the form of a purplish rash (or less often an exfoliative dermatitis) involving the nose, cheeks, forehead, upper trunk, and arms. The disease is associated with a complement mediated intramuscular microangiopathy, leading to loss of capillaries, muscle ischemia, muscle-fiber necrosis, and perifascicular atrophy. The childhood form of this disease tends to evolve into a systemic vasculitis. Dermatomyositis may occur in association with malignant neoplasms. (From Adams et al., Principles of Neurology, 6th ed, pp1405-6) Polymyositis-Dermatomyositis,Dermatomyositis, Adult Type,Dermatomyositis, Childhood Type,Dermatopolymyositis,Juvenile Dermatomyositis,Juvenile Myositis,Adult Type Dermatomyositis,Childhood Type Dermatomyositis,Dermatomyositis, Juvenile,Myositis, Juvenile,Polymyositis Dermatomyositis
D005169 Factor VIII Factor VIII of blood coagulation. Antihemophilic factor that is part of the factor VIII/von Willebrand factor complex. Factor VIII is produced in the liver and acts in the intrinsic pathway of blood coagulation. It serves as a cofactor in factor X activation and this action is markedly enhanced by small amounts of thrombin. Coagulation Factor VIII,Factor VIII Clotting Antigen,Factor VIII Coagulant Antigen,Factor VIII Procoagulant Activity,Thromboplastinogen,Blood Coagulation Factor VIII,F VIII-C,Factor 8,Factor 8 C,Factor Eight,Factor VIIIC,Hyate-C,Hyatt-C,F VIII C,Hyate C,HyateC,Hyatt C,HyattC
D006801 Humans Members of the species Homo sapiens. Homo sapiens,Man (Taxonomy),Human,Man, Modern,Modern Man
D000069283 Rituximab A murine-derived monoclonal antibody and ANTINEOPLASTIC AGENT that binds specifically to the CD20 ANTIGEN and is used in the treatment of LEUKEMIA; LYMPHOMA and RHEUMATOID ARTHRITIS. CD20 Antibody, Rituximab,GP2013,IDEC-C2B8,IDEC-C2B8 Antibody,Mabthera,Rituxan,IDEC C2B8,IDEC C2B8 Antibody,Rituximab CD20 Antibody
D000368 Aged A person 65 years of age or older. For a person older than 79 years, AGED, 80 AND OVER is available. Elderly
D000911 Antibodies, Monoclonal Antibodies produced by a single clone of cells. Monoclonal Antibodies,Monoclonal Antibody,Antibody, Monoclonal
D001323 Autoantibodies Antibodies that react with self-antigens (AUTOANTIGENS) of the organism that produced them. Autoantibody
D016896 Treatment Outcome Evaluation undertaken to assess the results or consequences of management and procedures used in combating disease in order to determine the efficacy, effectiveness, safety, and practicability of these interventions in individual cases or series. Rehabilitation Outcome,Treatment Effectiveness,Clinical Effectiveness,Clinical Efficacy,Patient-Relevant Outcome,Treatment Efficacy,Effectiveness, Clinical,Effectiveness, Treatment,Efficacy, Clinical,Efficacy, Treatment,Outcome, Patient-Relevant,Outcome, Rehabilitation,Outcome, Treatment,Outcomes, Patient-Relevant,Patient Relevant Outcome,Patient-Relevant Outcomes
D058846 Antibodies, Monoclonal, Murine-Derived Antibodies obtained from a single clone of cells grown in mice or rats. Murine-Derived Monoclonal Antibodies,Antibodies, Murine-Derived Monoclonal,Monoclonal Antibodies, Murine-Derived,Murine Derived Monoclonal Antibodies

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