Table 5.

Selective studies evaluating TA-TMA therapies

Published studies
DrugAuthorStudy typeNumber of patientsInclusion criteriaResponseSurvival
Eculizumab Jodele 2020 Retrospective, single institution 64 Children and young adults with hrTMA (concurrent proteinuria and elevated C5b9) and treated with eculizumab 64% response
56% CR 
OS 77% at 6 months 
 Zhang 2021 Systemic review with retrospective studies 116 (including 64 from above) Studies that included patients diagnosed with TA-TMA and eculizumab used first- or second-line 71% response
32% CR 
OS 52% at last follow-up of each study 
 Svec 2023 Retrospective, multi-institution 82 Allogeneic transplant patients <18 years with TA-TMA diagnosed by the treatment center and treated with eculizumab 48% response OS 47% at 6 months 
 Jodele 2024 Prospective, single-arm, multi-institution (NCT03518203) 21 Children and young adults, at least 5  kg, meeting criteria for hrTMA (concurrent proteinuria and elevated C5b9) 67% response
48% CR 
OS 71% at 6 months 
Narsoplimab Khaled 2022 Prospective, single-arm, multi-institution (NCT02222545) 28 Adults 18 or older with persistent TA-TMA 61% response OS 68% at 100 days 
Defibrotide Yeates 2017 Retrospective, multi-institution 39 Pediatric and adult patients who received defibrotide to treat TA-TMA 77% response OS 59%-64% at unspecified follow-up 
 Marinez-Munoz 2019 Retrospective, single institution 16 Adult patients who underwent allogeneic HCT with a diagnosis of TA-TMA 65% response OS 59% at unspecified follow-up 
Published studies
DrugAuthorStudy typeNumber of patientsInclusion criteriaResponseSurvival
Eculizumab Jodele 2020 Retrospective, single institution 64 Children and young adults with hrTMA (concurrent proteinuria and elevated C5b9) and treated with eculizumab 64% response
56% CR 
OS 77% at 6 months 
 Zhang 2021 Systemic review with retrospective studies 116 (including 64 from above) Studies that included patients diagnosed with TA-TMA and eculizumab used first- or second-line 71% response
32% CR 
OS 52% at last follow-up of each study 
 Svec 2023 Retrospective, multi-institution 82 Allogeneic transplant patients <18 years with TA-TMA diagnosed by the treatment center and treated with eculizumab 48% response OS 47% at 6 months 
 Jodele 2024 Prospective, single-arm, multi-institution (NCT03518203) 21 Children and young adults, at least 5  kg, meeting criteria for hrTMA (concurrent proteinuria and elevated C5b9) 67% response
48% CR 
OS 71% at 6 months 
Narsoplimab Khaled 2022 Prospective, single-arm, multi-institution (NCT02222545) 28 Adults 18 or older with persistent TA-TMA 61% response OS 68% at 100 days 
Defibrotide Yeates 2017 Retrospective, multi-institution 39 Pediatric and adult patients who received defibrotide to treat TA-TMA 77% response OS 59%-64% at unspecified follow-up 
 Marinez-Munoz 2019 Retrospective, single institution 16 Adult patients who underwent allogeneic HCT with a diagnosis of TA-TMA 65% response OS 59% at unspecified follow-up 
Ongoing clinical trials
DrugSponsorStudy typeNumber of patientsInclusion criteriaEnd points to collect
Ravulizumab Alexion Randomized, double-blind, placebo-controlled, multi-institution (NCT04543591) 106 estimated 12 years of age or older, HCT in the last 12 months, TMA diagnosis for at least 72 hours, weighing 30  kg or greater - TMA response at 26 weeks - OS at 26 and 52 weeks
- nonrelapse mortality at 26 and 52 weeks 
Nomacopan AKARI Therapeutics Prospective, single-arm, multi-institution (NCT04784455) 50 estimated 6 months to 18 years of age, undergone HCT with a diagnosis of TA-TMA - Transfusion independence by week 24 - rUPCR 2  mg/mg or less at 24 weeks 
Narsoplimab Omeros Prospective, single-arm, multi-institution (NCT05855083) 18 estimated Pediatric patients 28 days to <18 years of age with high-risk TMA - % clinical response - OS at 100 days after high-risk TMA diagnosis 
Pegcetacoplan Sobi Prospective, single-arm, multi-institution (NCT05148299) 12 estimated Adults 18 years or older, with a diagnosis of TA-TMA, and with rUPCR 1  mg/mg or greater - % clinical & TMA responses at 12 and 24 weeks - OS at 100 days
- OS 24 days from treatment start 
Ongoing clinical trials
DrugSponsorStudy typeNumber of patientsInclusion criteriaEnd points to collect
Ravulizumab Alexion Randomized, double-blind, placebo-controlled, multi-institution (NCT04543591) 106 estimated 12 years of age or older, HCT in the last 12 months, TMA diagnosis for at least 72 hours, weighing 30  kg or greater - TMA response at 26 weeks - OS at 26 and 52 weeks
- nonrelapse mortality at 26 and 52 weeks 
Nomacopan AKARI Therapeutics Prospective, single-arm, multi-institution (NCT04784455) 50 estimated 6 months to 18 years of age, undergone HCT with a diagnosis of TA-TMA - Transfusion independence by week 24 - rUPCR 2  mg/mg or less at 24 weeks 
Narsoplimab Omeros Prospective, single-arm, multi-institution (NCT05855083) 18 estimated Pediatric patients 28 days to <18 years of age with high-risk TMA - % clinical response - OS at 100 days after high-risk TMA diagnosis 
Pegcetacoplan Sobi Prospective, single-arm, multi-institution (NCT05148299) 12 estimated Adults 18 years or older, with a diagnosis of TA-TMA, and with rUPCR 1  mg/mg or greater - % clinical & TMA responses at 12 and 24 weeks - OS at 100 days
- OS 24 days from treatment start 

CR, complete response; HCT, hematopoietic cell transplantation; hrTMA, high-risk TMA; OS, overall survival; rUPCR, random urine protein to creatinine ratio.

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