Real-world data with the use of caplacizumab in the treatment of acquired thrombotic thrombocytopenic purpura: A single-center with homogeneous treatment experience.

caplacizumab plasma exchange thrombotic microangiopathy thrombotic thromobocytopenic purpura von Willebrand factor

Journal

Transfusion
ISSN: 1537-2995
Titre abrégé: Transfusion
Pays: United States
ID NLM: 0417360

Informations de publication

Date de publication:
11 2022
Historique:
revised: 29 06 2022
received: 10 05 2022
accepted: 22 07 2022
pubmed: 25 9 2022
medline: 10 11 2022
entrez: 24 9 2022
Statut: ppublish

Résumé

Recently, real-world data confirmed the effectiveness of caplacizumab in the treatment of acquired thrombotic thrombocytopenic purpura (aTTP); however, limitations as different treatment protocols from multicenter experiences and the front-line use of rituximab could overshadow the real impact of the addition of caplacizumab. We report the clinical characteristics and response to treatment of 30 consecutive cases of aTTP treated under a homogeneous therapeutic protocol with the only exception of the addition of caplacizumab in the last 10 cases (caplacizumab group), whose primary outcome we compare with the previous 20 cases (control group). Caplacizumab was started at a median of 2.5 days after diagnosis (interquartile range [IQR] 1-4) and maintained for a median of 37.5 days (IQR 32-39). Safety was in line with previous reports with 30% of patients showing mild adverse events, the most common bleeding. The caplacizumab group achieved platelet count normalization after a median of 3 (IQR 2-5) versus 4 (IQR 2-8.5) days in the control group (p = .067). The caplacizumab group required a lower median number of plasma exchanges, 10 (IQR 9-11) versus 16.5 (IQR 11-20) in the control group (p = .0053) and a shorter length of hospitalization, 12 (IQR 12-14) vs. 22 (IQR 15-27) days (p = .0025). There were no refractory cases and no deaths in the caplacizumab group compared to five refractory cases and three aTTP-related death (15%) in the control group. Our experience confirms improvement in the outcomes with a decrease in refractoriness and mortality since the introduction of caplacizumab.

Sections du résumé

BACKGROUND
Recently, real-world data confirmed the effectiveness of caplacizumab in the treatment of acquired thrombotic thrombocytopenic purpura (aTTP); however, limitations as different treatment protocols from multicenter experiences and the front-line use of rituximab could overshadow the real impact of the addition of caplacizumab.
STUDY DESIGN AND METHODS
We report the clinical characteristics and response to treatment of 30 consecutive cases of aTTP treated under a homogeneous therapeutic protocol with the only exception of the addition of caplacizumab in the last 10 cases (caplacizumab group), whose primary outcome we compare with the previous 20 cases (control group).
RESULTS
Caplacizumab was started at a median of 2.5 days after diagnosis (interquartile range [IQR] 1-4) and maintained for a median of 37.5 days (IQR 32-39). Safety was in line with previous reports with 30% of patients showing mild adverse events, the most common bleeding. The caplacizumab group achieved platelet count normalization after a median of 3 (IQR 2-5) versus 4 (IQR 2-8.5) days in the control group (p = .067). The caplacizumab group required a lower median number of plasma exchanges, 10 (IQR 9-11) versus 16.5 (IQR 11-20) in the control group (p = .0053) and a shorter length of hospitalization, 12 (IQR 12-14) vs. 22 (IQR 15-27) days (p = .0025). There were no refractory cases and no deaths in the caplacizumab group compared to five refractory cases and three aTTP-related death (15%) in the control group.
DISCUSSION
Our experience confirms improvement in the outcomes with a decrease in refractoriness and mortality since the introduction of caplacizumab.

Identifiants

pubmed: 36151933
doi: 10.1111/trf.17111
doi:

Substances chimiques

caplacizumab 2R27AB6766
Single-Domain Antibodies 0

Types de publication

Multicenter Study Journal Article

Langues

eng

Sous-ensembles de citation

IM

Pagination

2363-2369

Informations de copyright

© 2022 AABB.

Références

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Kremer Hovinga JA, Coppo P, Lämmle B, Moake JL, Miyata T, Vanhoorelbeke K. Thrombotic thrombocytopenic purpura. Nat Rev Dis Primers. 2017;3:17020.
Scully M, Hunt BJ, Benjamin S, Liesner R, Rose P, Peyvandi F, et al. Guidelines on the diagnosis and management of thrombotic thrombocytopenic purpura and other thrombotic microangiopathies. Br J Haematol. 2012;158(3):323-35.
Peyvandi F, Scully M, Kremer Hovinga JA, Cataland S, Knöbl P, Wu H, et al. Caplacizumab for acquired thrombotic thrombocytopenic purpura. N Engl J Med. 2016;374(6):511-22.
Scully M, Cataland SR, Peyvandi F, Coppo P, Knöbl P, Kremer Hovinga JA, et al. Caplacizumab treatment for acquired thrombotic thrombocytopenic purpura. N Engl J Med. 2019;380(4):335-46.
Völker LA, Kaufeld J, Miesbach W, Brähler S, Reinhardt M, Kühne L, et al. Real-world data confirm the effectiveness of caplacizumab in acquired thrombotic thrombocytopenic purpura. Blood Adv. 2020;4(13):3085-92.
Dutt T, Shaw RJ, Stubbs M, Yong J, Bailiff B, Cranfield T, et al. Real-world experience with caplacizumab in the management of acute TTP. Blood. 2021;137(13):1731-40.
Coppo P, Bubenheim M, Azoulay E, Galicier L, Malot S, Bigé N, et al. A regimen with caplacizumab, immunosuppression, and plasma exchange prevents unfavorable outcomes in immune-mediated TTP. Blood. 2021;137(6):733-42.
Benhamou Y, Assié C, Boelle PY, Buffet M, Grillberger R, Malot S, et al. Development and validation of a predictive model for death in acquired severe ADAMTS13 deficiency-associated idiopathic thrombotic thrombocytopenic purpura: the French TMA Reference Center experience. Haematologica. 2012;97(8):1181-6.
Scully M, Cataland S, Coppo P, Rubia J, Friedman KD, Kremer Hovinga J, et al. Consensus on the standardization of terminology in thrombotic thrombocytopenic purpura and related thrombotic microangiopathies. J Thromb Haemost. 2017;15(2):312-22.
Scully M, McDonald V, Cavenagh J, Hunt BJ, Longair I, Cohen H, et al. A phase 2 study of the safety and efficacy of rituximab with plasma exchange in acute acquired thrombotic thrombocytopenic purpura. Blood. 2011;118(7):1746-53.
Page EE, Kremer Hovinga JA, Terrell DR, Vesely SK, George JN. Rituximab reduces risk for relapse in patients with thrombotic thrombocytopenic purpura. Blood. 2016;127(24):3092-4.

Auteurs

Moraima Jiménez (M)

Servei d'Hematologia, Vall d'Hebron Hospital Universitari, Experimental Hematology, Vall d'Hebron Institute of Oncology (VHIO), Vall d'Hebron Barcelona Hospital Campus, Barcelona, Spain.
Department of Medicine, Universitat Autònoma de Barcelona, Barcelona, Spain.

Sabela Bobillo (S)

Servei d'Hematologia, Vall d'Hebron Hospital Universitari, Experimental Hematology, Vall d'Hebron Institute of Oncology (VHIO), Vall d'Hebron Barcelona Hospital Campus, Barcelona, Spain.
Department of Medicine, Universitat Autònoma de Barcelona, Barcelona, Spain.

Verónica Pons (V)

Banc de Sang y Teixits, Vall d'Hebron Barcelona Hospital Campus, Barcelona, Spain.

Carla Sánchez (C)

Servei d'Hematologia, Vall d'Hebron Hospital Universitari, Experimental Hematology, Vall d'Hebron Institute of Oncology (VHIO), Vall d'Hebron Barcelona Hospital Campus, Barcelona, Spain.

Ana Pérez (A)

Servei d'Hematologia, Vall d'Hebron Hospital Universitari, Experimental Hematology, Vall d'Hebron Institute of Oncology (VHIO), Vall d'Hebron Barcelona Hospital Campus, Barcelona, Spain.
Department of Medicine, Universitat Autònoma de Barcelona, Barcelona, Spain.

Antonieta Molero (A)

Servei d'Hematologia, Vall d'Hebron Hospital Universitari, Experimental Hematology, Vall d'Hebron Institute of Oncology (VHIO), Vall d'Hebron Barcelona Hospital Campus, Barcelona, Spain.
Department of Medicine, Universitat Autònoma de Barcelona, Barcelona, Spain.

Nayibe Miranda (N)

Servei d'Hematologia, Vall d'Hebron Hospital Universitari, Experimental Hematology, Vall d'Hebron Institute of Oncology (VHIO), Vall d'Hebron Barcelona Hospital Campus, Barcelona, Spain.

Ángela Sánchez (Á)

Servei d'Hematologia, Vall d'Hebron Hospital Universitari, Experimental Hematology, Vall d'Hebron Institute of Oncology (VHIO), Vall d'Hebron Barcelona Hospital Campus, Barcelona, Spain.

Elizabeth Tabares (E)

Servei d'Hematologia, Vall d'Hebron Hospital Universitari, Experimental Hematology, Vall d'Hebron Institute of Oncology (VHIO), Vall d'Hebron Barcelona Hospital Campus, Barcelona, Spain.

Francesc Bosch (F)

Servei d'Hematologia, Vall d'Hebron Hospital Universitari, Experimental Hematology, Vall d'Hebron Institute of Oncology (VHIO), Vall d'Hebron Barcelona Hospital Campus, Barcelona, Spain.
Department of Medicine, Universitat Autònoma de Barcelona, Barcelona, Spain.

David Valcárcel (D)

Servei d'Hematologia, Vall d'Hebron Hospital Universitari, Experimental Hematology, Vall d'Hebron Institute of Oncology (VHIO), Vall d'Hebron Barcelona Hospital Campus, Barcelona, Spain.
Department of Medicine, Universitat Autònoma de Barcelona, Barcelona, Spain.

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