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Thrombotic thrombocytopenic purpura is a thrombotic microangiopathy, caused by a deficiency of ADAMTS-13 (a desintegrin and metalloproteinase with a thrombospondin type 1 motif, member 13). It is considered a medical emergency and its standard treatment is based on plasma exchange associated with high doses of corticosteroids. Those patients refractory to standard treatment have a high risk of morbidity-mortality. Through this clinical case, a patient refractory to all the treatments is presented. She was treated whit caplacizumab that is humanized bivalent antibody acting against the domain A1 of the von Willebrand factor and prevents interaction with the glycoprotein Ib-IX-V (GPIb-IX-X). After one year from the end of treatment, the patient remains stable and asymptomatic. Caplacizumab is a good choice in those patients who do not respond to any of the standard treatments and that can lead to a decrease in mortality in patients with refractory thrombotic thrombocytopenic purpura.

Caplacizumab: First Case of Thrombotic Thrombocytopenic Purpura Refractory Treated in Spain outside Clinical Trial