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Donidalorsen

Phase 3

Hereditary Angioedema | Small molecule | Immunology |Ionis Pharmaceuticals, Inc.|Last Updated: May 28, 2026

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Trial Design
RandomizedDouble-BlindCONTROLLEDDMC
Total Trials4
Total Enrollment288
FDA Designations
No designations recorded
Clinical trial landscape

Donidalorsen · 6 trials · 3 indications

Phase 3 3Phase 2 2Phase 1 1
NCT07298447Donidalorsen Treatment in Children With Hereditary AngioedemaHereditary Angioedema (HAE)
RECRUITING20 Analytics
NCT05392114A Study to Assess the Long-Term Safety and Efficacy of Donidalorsen in the Prophylactic Treatment of Hereditary Angioedema (HAE)Hereditary Angioedema
ACTIVE NOT_RECRUITING154 Analytics
NCT05139810OASIS-HAE: A Study to Evaluate the Safety and Efficacy of Donidalorsen (ISIS 721744 or IONIS-PKK-LRx) in Participants With Hereditary Angioedema (HAE)Hereditary Angioedema
COMPLETED91 Analytics
PHASE3RECRUITING
Donidalorsen Treatment in Children With Hereditary Angioedema
Hereditary Angioedema (HAE)Unlock trial analytics
PHASE3ACTIVE NOT_RECRUITING
A Study to Assess the Long-Term Safety and Efficacy of Donidalorsen in the Prophylactic Treatment of Hereditary Angioedema (HAE)
Hereditary AngioedemaUnlock trial analytics
PHASE3COMPLETED
OASIS-HAE: A Study to Evaluate the Safety and Efficacy of Donidalorsen (ISIS 721744 or IONIS-PKK-LRx) in Participants With Hereditary Angioedema (HAE)
Hereditary AngioedemaUnlock trial analytics
Study Endpoints
Primary Endpoints
Number of Participants with Treatment Emergent Adverse Events (TEAEs)
over the period of approximately 17 months
Maximum Plasma Concentration (Cmax) of Donidalorsen
over the period of approximately 17 months
Maximum Time to Reach Cmax (Tmax) of Donidalorsen
over the period of approximately 17 months
Trough Plasma Concentration (Ctrough) of Donidalorsen
over the period of approximately 17 months
Percentage of Participants with at Least One Treatment-emergent Adverse Event (TEAE), Graded by Severity
Up to approximately 70 weeks, plus 104 weeks for Group 1; up to approximately 76 weeks, plus 104 weeks for Group 2
Time-Normalized Investigator-Confirmed (IC) HAE Attack Rate (Per Month) From Week 1 to Week 25
Week 1 to Week 25

The time-adjusted HAE attack rate was calculated as number of IC HAE attacks occurring from Week 1 to Week 25, divided by the number of days the participant contributed to the period multiplied by 28 days. An HAE attack was defined as an event with signs or symptoms consistent with an attack in at least 1 of the locations: peripheral angioedema (cutaneous swelling involving an extremity, the face, neck, torso, and/or genitourinary region), abdominal angioedema (abdominal pain, with or without abdominal distention, nausea, vomiting, or diarrhea), laryngeal angioedema (stridor, dyspnea, difficulty speaking, difficulty swallowing, throat tightening, or swelling of the tongue, palate, uvula, or larynx).

Percentage of Participants With Treatment-emergent Adverse Events (TEAEs)
Up to Week 221

An adverse event (AE) was defined as any unfavorable and unintended sign (including an abnormal laboratory finding, for example), symptom, or disease temporally associated with the use of a medicinal (investigational) product, whether or not the AE is considered related to the medicinal (investigational) product. TEAEs were defined as those AEs that either started or worsened in severity on or after the date/time of first administration of study drug in this OLE Study. TEAEs included both serious and non-serious TEAEs.

Percentage of Participants With Clinically Meaningful Changes in Clinical Laboratory Assessments
Up to Week 221

Clinical laboratory tests including clinical chemistry, hematology, coagulation, complement, inflammatory, urinalysis were performed. The percentage of participants with clinically meaningful changes were reported. Clinical meaningfulness was determined by the investigator.

Percentage of Participants With Clinically Meaningful Changes in Vital Signs
Up to Week 221

Vital sign measurements including heart rate, respiratory rate, body temperature, systolic and diastolic blood pressure and pulse pressure were assessed. Percentage of participants with clinically meaningful changes in the vital signs were reported. Clinically meaningfulness was determined by the investigator.

Percentage of Participants With Clinically Meaningful Changes in Electrocardiograms (ECGs) Parameters
Up to Week 221

The ECG parameters included ventricular rate, PR interval, QRS duration, QTc, QT corrected using the Fridericia's formula (QTcF), QT corrected using the Bazett's formula (QTcB), and overall interpretation. Percentage of participants with clinically meaningful changes in the ECG parameters were reported. Clinical meaningfulness was determined by the investigator.

Percentage of Participants Who Received At Least One Concomitant Medication
Up to Week 221

Concomitant medications include medications that participants were exposed to on or after the first dose of ISIS 721744 in the OLE study. Percentage of participants who received at least one concomitant medication were reported.

Time-normalized Number of HAE Attacks (Per Month) From Week 1 to Week 17
Week 1 to Week 17

The Week 1 to end of on-treatment period HAE attack rate was calculated for each participant as number of HAE attacks occurring from Week 1 to 28 days after the last dose date divided by the number of days the participant contributed to the period multiplied by 28 days. An HAE attack was defined as an event with signs or symptoms consistent with an attack in at least 1 of the locations: peripheral angioedema (cutaneous swelling involving an extremity, the face, neck, torso, and/or genitourinary region), abdominal angioedema (abdominal pain, with or without abdominal distention, nausea, vomiting, or diarrhea), laryngeal angioedema (stridor, dyspnea, difficulty speaking, difficulty swallowing, throat tightening, or swelling of the tongue, palate, uvula, or larynx).

Incidence and severity of adverse events that are related to treatment with Donidalorsen
Up to 176 days

The safety and tolerability of multiple doses of Donidalorsen will be assessed by determining the incidence, severity, and dose relationship of adverse events that are related to treatment with Donidalorsen.

Peak plasma Concentration (Cmax) of Donidalorsen
Up to 176 days

Maximum Donidalorsen plasma concentration, Cmax (ug/mL) will be assessed following SC administration

Time to peak plasma Concentration (Tmax) of Donidalorsen
Up to 176 days

Time to peak Donidalorsen plasma concentration, Tmax (hours) will be assessed following SC administration

Effects of Donidalorsen on plasma PKK concentration
Up to 176 days

Effects of Donidalorsen on plasma PKK concentration after multiple doses of Donidalorsen compared to baseline

Secondary Endpoints
Time-Normalized Number of Investigator-Confirmed HAE Attacks (per Month)
over the period of 12 months
Percentage of Investigator-Confirmed HAE Attack-free Participants
over the period of 12 months
Time-Normalized Number of Moderate or Severe Investigator-Confirmed HAE Attacks (per Month)
over the period of 12 months
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Study Design & Arms
AllocationNON_RANDOMIZED
MaskingNONE
ModelPARALLEL
PurposeTREATMENT
Treatment Arms
ArmTypeDescription
Donidalorsen: Group 1EXPERIMENTALParticipant weighing 9 kilograms (kg) to less than (\<)26 kg, will be administered donidalorsen over the period of one year.
Donidalorsen: Group 2EXPERIMENTALParticipant weighing greater than or equal to (≥)26 kg to \<41 kg, will be administered donidalorsen over the period of one year.
Donidalorsen: Group 3EXPERIMENTALParticipant weighing ≥41kg, will receive donidalorsen over the period of one year.
OLE ParticipantsEXPERIMENTALGroup 1 and Group 2 participants will be administered donidalorsen by SC injection for up to 157 weeks.
Pooled PlaceboPLACEBO_COMPARATORParticipants with hereditary angioedema type I/type II (HAE-1/HAE-2) received placebo subcutaneously (SC) either every 4 weeks (Weeks 1, 5, 9, 13, 17, and 21) or 8 weeks (Weeks 1, 9, and 17)
Cohort A: Donidalorsen 80 mgEXPERIMENTALParticipants with HAE-1/HAE-2 received donidalorsen, 80 mg, SC, every 4 weeks at Weeks 1, 5, 9, 13, 17, and 21.
Cohort B: Donidalorsen 80 mgEXPERIMENTALParticipants with HAE-1/HAE-2 received donidalorsen, 80 mg, SC, every 8 weeks at Weeks 1, 9, and 17.
HAE-1/HAE-2EXPERIMENTALParticipants with hereditary angioedema Type I/Type II (HAE-1/HAE-2) who received placebo or donidalorsen, 80 milligrams (mg), SC once every 4 weeks, in the previous study ISIS 721744-CS2 (NCT04030598), received donidalorsen, 80 mg, SC once every 4 weeks, from Week 1 to Week 13 (Fixed Dosing Period). Starting From Week 17 (Flexible Dosing Period), participants had 3 different dosing options as: 80 mg every 4 weeks, 80 mg every 8 weeks for participants who were attack-free for ≥12 weeks, 100 mg every 4 weeks for participants who were not attack-free for ≥12 weeks up to Week 53 based on the investigator and sponsor medical monitor recommendation. Participants continued flexible dosing from Week 53 for up to approximately 209 weeks.
HAE-nC1-INHEXPERIMENTALParticipants with hereditary angioedema with normal C1-inhibitor (HAE-nC1-INH) who received donidalorsen, 80 mg, SC, once every 4 weeks, in the previous study ISIS 721744-CS2 (NCT04030598), continued to receive donidalorsen, 80 mg, SC once every 4 weeks, from Week 1 to Week 13 (Fixed Dosing Period). Starting From Week 17 (Flexible Dosing Period), participants had 2 different dosing options as: 80 mg every 4 weeks, and 100 mg every 4 weeks for participants who were not attack-free for ≥12 weeks up to Week 53 based on the investigator and sponsor medical monitor recommendation. Participants continued flexible dosing from Week 53 for up to approximately 209 weeks.
Part A: PlaceboPLACEBO_COMPARATORParticipants with hereditary angioedema type I/type II (HAE-1/HAE-2) received placebo subcutaneously (SC) every 4 weeks at Weeks 1, 5, 9, and 13.
Part A: Donidalorsen 80 mgEXPERIMENTALParticipants with HAE-1/HAE-2 received donidalorsen, 80 mg, SC, every 4 weeks at Weeks 1, 5, 9, and 13.
Part B: Donidalorsen 80 mgEXPERIMENTALParticipants with hereditary angioedema with normal C1-inhibitor (HAE-nC1-INH) received donidalorsen, 80 mg, SC, every 4 weeks at Weeks 1, 5, 9, and 13.
DonidalorsenEXPERIMENTALAscending single and multiple doses of Donidalorsen administered subcutaneously
Placebo (sterile saline 0.9%)PLACEBO_COMPARATORCalculated volume to match active comparator
Interventions
NameTypeDescription
DonidalorsenDRUGDonidalorsen will be administered by subcutaneous (SC) injection.
PlaceboDRUGDonidalorsen-matching placebo was administered by SC injection.
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Eligibility Criteria
Age Range2 Years to 11 Years
SexALL
Healthy VolunteersNo
Study Sites10

Key Inclusion Criteria: 1. Must be between the ages of 2 and less than 12 years, inclusive, at the time of informed consent and, as applicable, assent. 2. Must weigh at least 9 kg at the time of informed consent and, as applicable, assent. 3. Documented diagnosis of HAE-1/HAE-2 based upon both of t...

Countries:United StatesItalyPolandSpainBelgiumBulgariaCanadaFranceGermanyIsraelNetherlandsPuerto RicoTurkey (Türkiye)United KingdomDenmark
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Recent Changes (Last 90 Days)
MEDIUMJun 8, 2026NCT04307381TRIAL_REMOVED: changed
MEDIUMJun 8, 2026NCT04307381TRIAL_REMOVED: changed
MEDIUMJun 8, 2026NCT04307381TRIAL_REMOVED: changed
MEDIUMJun 8, 2026NCT04307381TRIAL_REMOVED: changed
MEDIUMJun 8, 2026NCT04307381TRIAL_REMOVED: changed
LOWMay 29, 2026NCT07298447lastUpdatePostDate: changed
LOWMay 29, 2026NCT07298447lastUpdatePostDate: changed
LOWMay 29, 2026NCT07298447lastUpdatePostDate: changed
LOWMay 27, 2026NCT07298447startDate: changed
LOWMay 27, 2026NCT07298447startDate: changed
LOWMay 26, 2026NCT07298447primaryCompletionDate: changed
LOWMay 26, 2026NCT05392114primaryCompletionDate: changed
LOWMay 24, 2026NCT07298447studyFirstPostDate: changed
LOWMay 24, 2026NCT05392114studyFirstPostDate: changed