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Introduction: Acquired hemophilia A is very rare autoimmune disorder caused by an autoantibody to factor VIII that interferes with its coagulant function. It may be associated with a number of conditions such as lymphoproliferative disorders, drugs and solid malignancies. Hemarthrosis in Acquired hemophilia A is very rare clinical presentation and was never reported in cases induced by penicillin.

Case presentation: We report a 65-year-old male, born and living in Brazil, who presented hemorrhagic episodes complicated with joint hemarthrosis due to acquired hemophilia A caused by a commonly used drug, Benzathine penicillin.Disease management was focused on controlling bleeding, primarily with the use of factor VIII inhibition bypassing activity and eradicate the autoantibody using various immunosuppressants. The challenges in manage this case included delayed diagnosis, durable remissions, difficulty achieving hemostasis. Clinical and laboratory coagulation response has improved eight weeks after only with the administration of double immunosuppression medication: corticosteroid and cyclophosphamide.

Conclusion: This case gives us the lesson that prompt diagnosis and treatment achieved by hematologists may improve the prognosis and prevent severe bleeding. Early detection is vital to improving outcomes.