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NCT02599961

Study to Assess the Long Term Safety and Efficacy of UX007 in Participants With Glucose Type 1 Deficiency Syndrome (Glut1 DS)

Terminated Phase 2 Results posted Last updated 11 June 2020
What this trial tests

Phase 2 trial testing UX007 in Glucose Transporter Type 1 Deficiency Syndrome in 15 participants. Terminated before completion.

Timeline
10 September 2015
Primary endpoint
22 October 2019
22 October 2019

Quick facts

Lead sponsorUltragenyx Pharmaceutical Inc
PhasePhase 2
StatusTerminated
Study typeINTERVENTIONAL
Allocationna
Designsingle group
Maskingnone
Primary purposetreatment
Enrollment15
Start date10 September 2015
Primary completion22 October 2019
Estimated completion22 October 2019
Sites9 locations across Denmark, United Kingdom, Australia, United States, Spain

Drugs / interventions tested

Conditions studied

Sponsor

Ultragenyx Pharmaceutical Inc — full company profile →

Who can join

1 and older, any sex, with Glucose Transporter Type 1 Deficiency Syndrome. Patients with the condition only — healthy volunteers not accepted.

Results — posted to ClinicalTrials.gov

Per-arm endpoint measurements with 95% confidence intervals where reported. Source: trial results section.

Number of Participants With Treatment-Emergent Adverse Events (TEAEs), Serious TEAEs, Discontinuations Due to TEAEs, and Deaths Primary · From first dose of study drug up to 36 months. The mean (SD) treatment duration was 667.9 (357) days.

An adverse event (AE) was defined as any untoward medical occurrence, whether or not considered drug related. Serious adverse events (SAEs) are AEs that at any dose, in the view of either the investigator or sponsor, results in any of the following outcomes: death; a life-threatening AE; inpatient hospitalization or prolongation of existing hospitalization; persistent or significant incapacity or disability; a congenital anomaly/birth defect; other important medical event. An AE was considered a TEAE if it occurred on or after the first dose in this study, and was not present prior to the firs

TEAEs
GroupValue95% CI
UX007 (Triheptanoin)13
Serious TEAEs
GroupValue95% CI
UX007 (Triheptanoin)2
Related TEAEs
GroupValue95% CI
UX007 (Triheptanoin)10
Serious and Related TEAEs
GroupValue95% CI
UX007 (Triheptanoin)0
Grade 3 or 4 TEAEs
GroupValue95% CI
UX007 (Triheptanoin)1
Gastrointestinal TEAEs
GroupValue95% CI
UX007 (Triheptanoin)9
TEAEs Leading to Treatment Discontinuation
GroupValue95% CI
UX007 (Triheptanoin)0
TEAEs Leading to Study Discontinuation
GroupValue95% CI
UX007 (Triheptanoin)0
Change From Baseline Over Time in Overall Seizure Frequency Per 4 Weeks Secondary · Baseline (from NCT01993186), Month 0-3, Month 4-6, Month 7-9, Month 10-12, Month 13-18, Month 19-24, Month 25-30, Month 31-36

The number of observable seizures were recorded by the subject or caregiver via diary throughout the study. Observable seizures were defined as: generalized tonic-clonic; generalized tonic; generalized clonic; generalized atonic; partial/focal with secondary generalization; myoclonic, myoclonic (astatic) atonic, myoclonic tonic; complex partial/focal; simple partial/focal motor; absence.

Change at Month 0-3
GroupValue95% CI
UX007 (Triheptanoin)-64.22± 185.564
Change at Month 4-6
GroupValue95% CI
UX007 (Triheptanoin)-64.19± 142.460
Change at Month 7-9
GroupValue95% CI
UX007 (Triheptanoin)-61.81± 164.770
Change at Month 10-12
GroupValue95% CI
UX007 (Triheptanoin)-91.93± 216.834
Change at Month 13-18
GroupValue95% CI
UX007 (Triheptanoin)-75.56± 206.406
Change at Month 19-24
GroupValue95% CI
UX007 (Triheptanoin)-110.12± 233.492
Change at Month 25-30
GroupValue95% CI
UX007 (Triheptanoin)-135.60± 249.871
Change at Month 31-36
GroupValue95% CI
UX007 (Triheptanoin)-51.88± 101.378
Change From Baseline Over Time in CNS Total Score Secondary · Baseline (from NCT01993186), Month 0, Month 6, Month 12, Month 24, Month 36

The CNS evaluates measures of neurological function and development delay, and is the sum of scores for the following domains: Weight, Height, Head Circumference, General Medical Exam, Funduscopic Exam, Cranial Nerves, Stance \& Gait, Involuntary Movements, Sensation, Cerebellar Function, Muscle Bulk, Tone \& Strength, Myotatic Reflexes, Toe Sign, Other Findings. The CNS is only scored when all domains are measured and ranges from 0 (abnormal exam) to 76 (normal exam). Higher scores are associated with higher neurological function.

Change at Month 0
GroupValue95% CI
UX007 (Triheptanoin)11.85± 20.331
Change at Month 6
GroupValue95% CI
UX007 (Triheptanoin)9.36± 18.495
Change at Month 12
GroupValue95% CI
UX007 (Triheptanoin)13.64± 21.371
Change at Month 24
GroupValue95% CI
UX007 (Triheptanoin)3.38± 3.092
Change at Month 36
GroupValue95% CI
UX007 (Triheptanoin)0.00± NA
Change From Baseline Over Time in SF-10 Health Survey for Children Physical Summary Score Secondary · Baseline (from NCT01993186), Month 0, Month 6, Month 12, Month 18, Month 24, Month 30

The SF-10 Health Survey for Children was administered to caregivers of participants aged 5-17 years. Responses are used to generate 2 component summary scores: Physical Summary Score and the Psychosocial Summary Score. The T-score based scale scores were centered so that a score of 50 corresponds to the average score in a comprehensive 2006 sample (a combination of general population and supplemental disability and chronic condition samples). Higher scores are associated with better quality of life.

Change at Month 0
GroupValue95% CI
UX007 (Triheptanoin)0.25± 16.917
Change at Month 6
GroupValue95% CI
UX007 (Triheptanoin)9.56± 21.522
Change at Month 12
GroupValue95% CI
UX007 (Triheptanoin)-2.67± 9.475
Change at Month 18
GroupValue95% CI
UX007 (Triheptanoin)-9.35± 12.702
Change at Month 24
GroupValue95% CI
UX007 (Triheptanoin)-8.96± 20.075
Change at Month 30
GroupValue95% CI
UX007 (Triheptanoin)7.74± 2.273
Change From Baseline Over Time in SF-10 Health Survey for Children Psychosocial Summary Score Secondary · Baseline (from NCT01993186), Month 0, Month 6, Month 12, Month 18, Month 24, Month 30

The SF-10 Health Survey for Children was administered to caregivers of participants aged 5-17 years. Responses are used to generate 2 component summary scores: Physical Summary Score and the Psychosocial Summary Score. The T-score based scale scores were centered so that a score of 50 corresponds to the average score in a comprehensive 2006 sample (a combination of general population and supplemental disability and chronic condition samples). Higher scores are associated with better quality of life.

Change at Month 0
GroupValue95% CI
UX007 (Triheptanoin)0.59± 11.049
Change at Month 6
GroupValue95% CI
UX007 (Triheptanoin)-2.29± 12.366
Change at Month 12
GroupValue95% CI
UX007 (Triheptanoin)-7.43± 15.514
Change at Month 18
GroupValue95% CI
UX007 (Triheptanoin)-6.60± 14.987
Change at Month 24
GroupValue95% CI
UX007 (Triheptanoin)-8.25± 15.316
Change at Month 30
GroupValue95% CI
UX007 (Triheptanoin)8.91± 12.599
Change From Baseline Over Time in SF-12v2 Health Survey PCS Score Secondary · Baseline (from NCT01993186), Month 0, Month 6, Month 12, Month 18

SF-12v2 was assessed for adults 18 years of age and older. Eight domain scores were used to generate 2 component summary scores: physical health (PCS) and mental health (MCS). The PCS and MCS scores have mean of 50 and SD of 10. The T-score based scoring method scores the data in relation to U.S. general population T-scores. Therefore, all scores obtained that are below 50 can be interpreted as below the U.S. general population T-score and scores above 50 can be interpreted as above the U.S. general population T-score. Higher global scores are associated with better quality of life.

Change at Month 0
GroupValue95% CI
UX007 (Triheptanoin)10.25± 5.077
Change at Month 6
GroupValue95% CI
UX007 (Triheptanoin)12.37± 5.367
Change at Month 12
GroupValue95% CI
UX007 (Triheptanoin)5.09± 1.619
Change at Month 18
GroupValue95% CI
UX007 (Triheptanoin)-0.35± NA
Change From Baseline Over Time in SF-12v2 Health Survey MCS Score Secondary · Baseline (from NCT01993186), Month 0, Month 6, Month 12, Month 18

SF-12v2 was assessed for adults 18 years of age and older. Eight domain scores were used to generate 2 component summary scores: physical health (PCS) and mental health (MCS). The PCS and MCS scores have mean of 50 and SD of 10. The T-score based scoring method scores the data in relation to U.S. general population T-scores. Therefore, all scores obtained that are below 50 can be interpreted as below the U.S. general population T-score and scores above 50 can be interpreted as above the U.S. general population T-score. Higher global scores are associated with better quality of life.

Change at Month 0
GroupValue95% CI
UX007 (Triheptanoin)8.63± 2.249
Change at Month 6
GroupValue95% CI
UX007 (Triheptanoin)5.84± 3.316
Change at Month 12
GroupValue95% CI
UX007 (Triheptanoin)6.90± 5.706
Change at Month 18
GroupValue95% CI
UX007 (Triheptanoin)16.96± NA

Adverse events — posted to ClinicalTrials.gov

Time frame: From first dose of study drug up to 36 months. The mean (SD) treatment duration was 667.9 (357) days.. Reporting threshold: 5%. Adverse-event reports describe events observed during the trial — not all are caused by the drug.

UX007 (Triheptanoin)
Serious: 2/15 (13%)
Deaths: 0/15

Serious adverse events (5 terms)

ReactionSystemUX007 (Triheptanoin)
Intestinal ObstructionGastrointestinal disorders
Croup InfectiousInfections and infestations
Ear InfectionInfections and infestations
Otitis MediaInfections and infestations
Otitis Media AcuteInfections and infestations
Other adverse events (52 terms — click to expand)

ReactionSystemUX007 (Triheptanoin)
DiarrhoeaGastrointestinal disorders
VomitingGastrointestinal disorders
HeadacheNervous system disorders
Abdominal PainGastrointestinal disorders
NauseaGastrointestinal disorders
Upper Respiratory Tract InfectionInfections and infestations
Viral Upper Respiratory Tract InfectionInfections and infestations
LethargyNervous system disorders
Back PainMusculoskeletal and connective tissue disorders
Petit Mal EpilepsyNervous system disorders
TachycardiaCardiac disorders
Abdominal Pain UpperGastrointestinal disorders
Breath OdourGastrointestinal disorders
ConstipationGastrointestinal disorders
DysphagiaGastrointestinal disorders
FlatulenceGastrointestinal disorders
HaematocheziaGastrointestinal disorders
PyrexiaGeneral disorders
ThirstGeneral disorders
Seasonal AllergyImmune system disorders
Ear InfectionInfections and infestations
InfluenzaInfections and infestations
Otitis MediaInfections and infestations
Otitis Media AcuteInfections and infestations
Pharyngitis StreptococcalInfections and infestations
ContusionInjury, poisoning and procedural complications
Head InjuryInjury, poisoning and procedural complications
Ligament SprainInjury, poisoning and procedural complications
Procedural PainInjury, poisoning and procedural complications
Alanine Aminotransferase IncreasedInvestigations
Blood Glucose IncreasedInvestigations
Weight IncreasedInvestigations
HypoglycaemiaMetabolism and nutrition disorders
HyponatraemiaMetabolism and nutrition disorders
Muscle SpasmsMusculoskeletal and connective tissue disorders
Muscular WeaknessMusculoskeletal and connective tissue disorders
Musculoskeletal PainMusculoskeletal and connective tissue disorders
Pain In ExtremityMusculoskeletal and connective tissue disorders
Disturbance In AttentionNervous system disorders
DysarthriaNervous system disorders

Most-reported serious reactions: Intestinal Obstruction, Croup Infectious, Ear Infection, Otitis Media, Otitis Media Acute.

Data from ClinicalTrials.gov NCT02599961 adverse events section.

Sponsor's own description

The primary objective of the study is to evaluate the long-term safety of UX007 in Glut1 DS participants.

Publications & conference data

1 peer-reviewed publication reference this trial (live from Europe PMC):

  1. Triheptanoin: First Approval.
    Shirley M. · · 2020 · cited 26× · PMID 32897506 · DOI 10.1007/s40265-020-01399-5

Verify or expand the search:

Other trials of UX007

Trials testing the same drug.

Other recruiting trials for Glucose Transporter Type 1 Deficiency Syndrome

Currently open trials in the same condition.

Other Ultragenyx Pharmaceutical Inc trials

Trials by the same sponsor.

Verify against primary sources

Data sources for this page

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