Trial Outcomes & Findings for A 2-Period Crossover Study of BPN14770 in Adults Males With Fragile X Syndrome (NCT NCT03569631)

NCT ID: NCT03569631

Last Updated: 2024-12-19

Results Overview

A TEAE was any untoward medical occurrence associated with the use of a drug in humans, whether or not considered drug related. A Serious Adverse Event (SAE) was an AE that met at least 1 of the following criteria: resulted in death, was life-threatening, required inpatient hospitalization or prolongation of existing hospitalization for the AE, persistent or significant disability/incapacity or substantial disruption of the ability to conduct normal life functions, congenital anomaly/birth defect (in the child of a participant who was exposed to the study drug), important medical event or reaction. A summary of all Serious Adverse Events and Other Adverse Events (nonserious) regardless of causality is located in the 'Reported Adverse Events' Section.

Recruitment status

COMPLETED

Study phase

PHASE2

Target enrollment

30 participants

Primary outcome timeframe

Up to 24 weeks

Results posted on

2024-12-19

Participant Flow

Participant milestones

Participant milestones
Measure
Sequence A-B
Participants received 25 mg BPN1470 capsules twice daily in Period 1 followed by matching placebo in Period 2.
Sequence B-A
Participants received placebo in Period 1 followed by 25 mg BPN1477 capsules twice daily in Period 2.
Period 1 (12 Weeks)
STARTED
15
15
Period 1 (12 Weeks)
Received At Least 1 Dose of Study Drug
15
15
Period 1 (12 Weeks)
COMPLETED
15
15
Period 1 (12 Weeks)
NOT COMPLETED
0
0
Period 2 (12 Weeks)
STARTED
15
15
Period 2 (12 Weeks)
Received At Least 1 Dose of Study Drug
15
15
Period 2 (12 Weeks)
COMPLETED
15
15
Period 2 (12 Weeks)
NOT COMPLETED
0
0

Reasons for withdrawal

Withdrawal data not reported

Baseline Characteristics

A 2-Period Crossover Study of BPN14770 in Adults Males With Fragile X Syndrome

Baseline characteristics by cohort

Baseline characteristics by cohort
Measure
Sequence A-B
n=15 Participants
Participants received 25 mg BPN1470 capsules twice daily in Period 1 followed by matching placebo in Period 2.
Sequence B-A
n=15 Participants
Participants received placebo in Period 1 followed by 25 mg BPN1477 capsules twice daily in Period 2.
Total
n=30 Participants
Total of all reporting groups
Age, Continuous
31.2 years
STANDARD_DEVIATION 6.82 • n=5 Participants
32.1 years
STANDARD_DEVIATION 8.00 • n=7 Participants
31.6 years
STANDARD_DEVIATION 7.32 • n=5 Participants
Sex: Female, Male
Female
0 Participants
n=5 Participants
0 Participants
n=7 Participants
0 Participants
n=5 Participants
Sex: Female, Male
Male
15 Participants
n=5 Participants
15 Participants
n=7 Participants
30 Participants
n=5 Participants
Ethnicity (NIH/OMB)
Hispanic or Latino
1 Participants
n=5 Participants
1 Participants
n=7 Participants
2 Participants
n=5 Participants
Ethnicity (NIH/OMB)
Not Hispanic or Latino
14 Participants
n=5 Participants
14 Participants
n=7 Participants
28 Participants
n=5 Participants
Ethnicity (NIH/OMB)
Unknown or Not Reported
0 Participants
n=5 Participants
0 Participants
n=7 Participants
0 Participants
n=5 Participants
Race (NIH/OMB)
American Indian or Alaska Native
0 Participants
n=5 Participants
1 Participants
n=7 Participants
1 Participants
n=5 Participants
Race (NIH/OMB)
Asian
0 Participants
n=5 Participants
0 Participants
n=7 Participants
0 Participants
n=5 Participants
Race (NIH/OMB)
Native Hawaiian or Other Pacific Islander
1 Participants
n=5 Participants
2 Participants
n=7 Participants
3 Participants
n=5 Participants
Race (NIH/OMB)
Black or African American
0 Participants
n=5 Participants
0 Participants
n=7 Participants
0 Participants
n=5 Participants
Race (NIH/OMB)
White
14 Participants
n=5 Participants
12 Participants
n=7 Participants
26 Participants
n=5 Participants
Race (NIH/OMB)
More than one race
0 Participants
n=5 Participants
0 Participants
n=7 Participants
0 Participants
n=5 Participants
Race (NIH/OMB)
Unknown or Not Reported
0 Participants
n=5 Participants
0 Participants
n=7 Participants
0 Participants
n=5 Participants

PRIMARY outcome

Timeframe: Up to 24 weeks

Population: Safety Population included all participants who were randomized and received at least one dose of study drug. Here, 'Overall number of participants analyzed' = participants evaluable for this outcome measure.

A TEAE was any untoward medical occurrence associated with the use of a drug in humans, whether or not considered drug related. A Serious Adverse Event (SAE) was an AE that met at least 1 of the following criteria: resulted in death, was life-threatening, required inpatient hospitalization or prolongation of existing hospitalization for the AE, persistent or significant disability/incapacity or substantial disruption of the ability to conduct normal life functions, congenital anomaly/birth defect (in the child of a participant who was exposed to the study drug), important medical event or reaction. A summary of all Serious Adverse Events and Other Adverse Events (nonserious) regardless of causality is located in the 'Reported Adverse Events' Section.

Outcome measures

Outcome measures
Measure
BPN14770
n=30 Participants
Participants received 25 mg BPN14770 capsules twice daily for 12 weeks either in Period 1 or Period 2.
Placebo
n=30 Participants
Participants received placebo to match 25 mg BPN14770 capsules twice daily for 12 weeks either in Period 1 or Period 2.
Number of Participants With Treatment-Emergent Adverse Events (TEAEs)
11 Participants
8 Participants

SECONDARY outcome

Timeframe: Baseline to Week 12

Population: Completers population included all randomized participants who completed both periods of treatment with no significant protocol violations.

The NIH-TCB is a battery of extensively validated computer-administered cognitive tests administered on an iPad. The Crystallized Composite score assessment includes Picture Vocabulary and Oral Reading Recognition tests. The NIH toolbox standard score has a mean of 100 and standard deviation (SD) of 15. Higher scores indicate better intellectual abilities.

Outcome measures

Outcome measures
Measure
BPN14770
n=29 Participants
Participants received 25 mg BPN14770 capsules twice daily for 12 weeks either in Period 1 or Period 2.
Placebo
n=30 Participants
Participants received placebo to match 25 mg BPN14770 capsules twice daily for 12 weeks either in Period 1 or Period 2.
Change From Baseline in National Institute of Health Toolbox Cognitive Battery Modified for Intellectual Disabilities Crystallized Cognition Composite (NIH-TCB CCC) Score
0.1 units on a scale
Standard Deviation 3.45
-0.9 units on a scale
Standard Deviation 7.18

SECONDARY outcome

Timeframe: Baseline to Week 12

Population: Intent-to-Treat (ITT) population included all randomized participants who received at least one dose of treatment and returned for at least one follow-up visit.

The VAS was used to measure the severity of Daily Functioning skills, which is a specific behavioral symptom targeted in this study. Parents or caregivers marked this behavior on a visual line on a score from 0 to 100, with higher scores indicating better quality of life.

Outcome measures

Outcome measures
Measure
BPN14770
n=30 Participants
Participants received 25 mg BPN14770 capsules twice daily for 12 weeks either in Period 1 or Period 2.
Placebo
n=30 Participants
Participants received placebo to match 25 mg BPN14770 capsules twice daily for 12 weeks either in Period 1 or Period 2.
Change From Baseline Between BPN14770 and Placebo Arm in Visual Analog Scale (VAS) Daily Functioning
9.4 millimeters (mm)
Standard Deviation 27.91
6.8 millimeters (mm)
Standard Deviation 22.77

SECONDARY outcome

Timeframe: Baseline to Week 12

Population: Intent-to-Treat (ITT) population included all randomized participants who received at least one dose of treatment and returned for at least one follow-up visit.

The VAS was used to measure the severity of Language skills, which is a specific behavioral symptom targeted in this study. Parents or caregivers marked this behavior on a visual line on a score from 0 to 100, with higher scores indicating better quality of life.

Outcome measures

Outcome measures
Measure
BPN14770
n=30 Participants
Participants received 25 mg BPN14770 capsules twice daily for 12 weeks either in Period 1 or Period 2.
Placebo
n=30 Participants
Participants received placebo to match 25 mg BPN14770 capsules twice daily for 12 weeks either in Period 1 or Period 2.
Change From Baseline Between BPN14770 and Placebo Arm in VAS Language
18.2 millimeters (mm)
Standard Deviation 21.47
15.4 millimeters (mm)
Standard Deviation 22.07

Adverse Events

BPN14770

Serious events: 1 serious events
Other events: 5 other events
Deaths: 0 deaths

Placebo

Serious events: 0 serious events
Other events: 6 other events
Deaths: 0 deaths

Serious adverse events

Serious adverse events
Measure
BPN14770
n=30 participants at risk
Participants received 25 mg BPN14770 capsules twice daily for 12 weeks either in Period 1 or Period 2.
Placebo
n=30 participants at risk
Participants received placebo to match 25 mg BPN14770 capsules twice daily for 12 weeks either in Period 1 or Period 2.
Infections and infestations
Bursitis infective
3.3%
1/30 • First dose of study drug up to 24 weeks
Safety Population included all participants who were randomized and received at least one dose of study drug.
0.00%
0/30 • First dose of study drug up to 24 weeks
Safety Population included all participants who were randomized and received at least one dose of study drug.

Other adverse events

Other adverse events
Measure
BPN14770
n=30 participants at risk
Participants received 25 mg BPN14770 capsules twice daily for 12 weeks either in Period 1 or Period 2.
Placebo
n=30 participants at risk
Participants received placebo to match 25 mg BPN14770 capsules twice daily for 12 weeks either in Period 1 or Period 2.
Gastrointestinal disorders
Diarrhoea
0.00%
0/30 • First dose of study drug up to 24 weeks
Safety Population included all participants who were randomized and received at least one dose of study drug.
6.7%
2/30 • First dose of study drug up to 24 weeks
Safety Population included all participants who were randomized and received at least one dose of study drug.
Gastrointestinal disorders
Vomiting
10.0%
3/30 • First dose of study drug up to 24 weeks
Safety Population included all participants who were randomized and received at least one dose of study drug.
6.7%
2/30 • First dose of study drug up to 24 weeks
Safety Population included all participants who were randomized and received at least one dose of study drug.
Infections and infestations
Upper respiratory tract infection
6.7%
2/30 • First dose of study drug up to 24 weeks
Safety Population included all participants who were randomized and received at least one dose of study drug.
10.0%
3/30 • First dose of study drug up to 24 weeks
Safety Population included all participants who were randomized and received at least one dose of study drug.

Additional Information

Shionogi Clinical Trials Administrator Clinical Support Help Line

Shionogi

Phone: 1-800-849-9707

Results disclosure agreements

  • Principal investigator is a sponsor employee The sponsor can embargo results from a PI's center until the combined results from the completed study have been published in full or the sponsor confirms there will be no multicenter study publication. Results communications must be provided to the sponsor for review at least 60 days before submission for publication. By written request, the sponsor can extend the embargo up to an additional 60 days. The sponsor cannot require changes to scientific content and cannot further extend the embargo.
  • Publication restrictions are in place

Restriction type: OTHER