ArticleBlood advances2026
Delayed ADAMTS13 normalization in caplacizumab/PEX-treated patients associated with anti-ADAMTS13 IgG antibody boosting.
Article in Blood advances, 2026. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Not yet cited in PubMed.
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Abstract
abstractDelayed ADAMTS13 normalization was previously reported in a subgroup of caplacizumab/plasma exchange (PEX) cases. To investigate this, anti-ADAMTS13 immunoglobulin G (IgG) levels were analysed at multiple timepointsduring an acute thrombotic thrombocytopenic purpura episode in 59 caplacizumab/PEX and 50 non-caplacizumab/PEXcases. Caplacizumab/PEX cases required fewer days of PEX, taking longer to achieve ADAMTS13 activity of >20%compared to non-caplacizumab/PEX cases (median, 33 days vs 17.5 days; P< .0001). More caplacizumab/PEX casescompared with non-caplacizumab/PEX showed increased anti-ADAMTS13 IgG levels after PEX (40% vs 25.5%; P =.14). Cases requiring >30 days of caplacizumab had higher presenting anti-ADAMTS13 IgG levels compared with <30days (63.5% vs 25%; P = .02). Half the cases requiring >30 days of caplacizumab increased anti-ADAMTS13 IgG levelsafter PEX; median 101 days to ADAMTS13 activity of >20% compared to 28 days (P< .0001) in cases without anantibody increase. 54% of caplacizumab cases had presenting anti-ADAMTS13 IgG levels of >40%; 47% had increasedantibody levels after PEX, taking longer to achieve ADAMTS13 activity of >20%. 33% of patients with presenting anti-ADAMTS13 IgG levels of <40% increased their antibody levels after PEX and took 97.5 days to achieve ADAMTS13activity of >20%. Cases with presenting anti-ADAMTS13 IgG levels <40% and no increase in antibody levels after PEXtook a median of 14 days (P = .0005). Therefore, the subgroup of caplacizumab/PEX cases with delayed ADAMTS13normalization was associated with antibody boosting, more common with a higher presenting anti-ADAMTS13 IgG and,subsequently, longer caplacizumab use. These findings were subsequently confirmed in 93 cases that were treated withcaplacizumab/PEX over 3 years.
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