Trial Outcomes & Findings for Recombinant Human Insulin-Like Growth Factor (rhIGF-1) Treatment of Short Stature Associated With IGF-1 Deficiency (NCT NCT00125190)

NCT ID: NCT00125190

Last Updated: 2020-07-27

Results Overview

Height was measured standing without shoes as the average of three measurements by the same observer using identical technique with a Harpenden or other wall mounted stadiometer. The subject was repositioned between each measurement. Height velocity during an interval of time is defined as the change in height during the time interval divided by the duration of the time interval. Missing Week 34 heights were imputed using the last height SD score carried forward.

Recruitment status

COMPLETED

Study phase

PHASE2/PHASE3

Target enrollment

45 participants

Primary outcome timeframe

Pretreatment to Week 34

Results posted on

2020-07-27

Participant Flow

This was a open-label, multi-center and single-arm study conducted at 12 investigational sites (11 active) between 12 January 2005 and 14 January 2009. A total of 45 subjects were enrolled in this study.

The screening period consisted of two-staged clinic visits for up to 6 weeks, followed by an open-label treatment period of 86 weeks.

Participant milestones

Participant milestones
Measure
rhIGF-1 QD
During the treatment phase (Day 1 to Week 86), subjects received subcutaneous (SC) injections of rhIGF-1 at an initial dose of 60 microgram per kilogram (mcg/kg) once daily (QD) starting on Day 1 (Visit 3). From Week 2 (Visit 4) subsequent dose adjustments were made in order to achieve the target serum IGF-1 concentration for the subject's age and sex. The maximum dose in any circumstance was 240 mcg/kg/day.
Overall Study
STARTED
45
Overall Study
Completed 34 Weeks of Study
43
Overall Study
COMPLETED
30
Overall Study
NOT COMPLETED
15

Reasons for withdrawal

Reasons for withdrawal
Measure
rhIGF-1 QD
During the treatment phase (Day 1 to Week 86), subjects received subcutaneous (SC) injections of rhIGF-1 at an initial dose of 60 microgram per kilogram (mcg/kg) once daily (QD) starting on Day 1 (Visit 3). From Week 2 (Visit 4) subsequent dose adjustments were made in order to achieve the target serum IGF-1 concentration for the subject's age and sex. The maximum dose in any circumstance was 240 mcg/kg/day.
Overall Study
Subject/Parent Decision
7
Overall Study
Non-compliance
2
Overall Study
Adverse Event
1
Overall Study
Other
1
Overall Study
Lost to Follow-up
4

Baseline Characteristics

Recombinant Human Insulin-Like Growth Factor (rhIGF-1) Treatment of Short Stature Associated With IGF-1 Deficiency

Baseline characteristics by cohort

Baseline characteristics by cohort
Measure
rhIGF-1 QD
n=45 Participants
During the treatment phase (Day 1 to Week 86), subjects received SC injections of rhIGF-1 at an initial dose of 60 mcg/kg QD starting on Day 1 (Visit 3). From Week 2 (Visit 4) subsequent dose adjustments were made in order to achieve the target serum IGF-1 concentration for the subject's age and sex. The maximum dose in any circumstance was 240 mcg/kg/day.
Age, Continuous
8.7 years
STANDARD_DEVIATION 2.8 • n=5 Participants
Age, Customized
Children (2-11 years)
36 Participants
n=5 Participants
Age, Customized
Adolescents (12-17 years)
9 Participants
n=5 Participants
Sex: Female, Male
Female
7 Participants
n=5 Participants
Sex: Female, Male
Male
38 Participants
n=5 Participants
Race/Ethnicity, Customized
Black
1 participants
n=5 Participants
Race/Ethnicity, Customized
Hispanic
12 participants
n=5 Participants
Race/Ethnicity, Customized
White
31 participants
n=5 Participants
Race/Ethnicity, Customized
Other
1 participants
n=5 Participants
Body Mass Index Standard Deviation (SD) Score
-0.4 SDs
STANDARD_DEVIATION 0.7 • n=5 Participants
Bone Age Imputed
7.2 years
STANDARD_DEVIATION 2.6 • n=5 Participants
Height for Age SD Score
-2.7 SDs
STANDARD_DEVIATION 0.6 • n=5 Participants
IGFBP-3 SD Score
-0.7 SDs
STANDARD_DEVIATION 1.0 • n=5 Participants
IGF-1 SD Score
-2.6 SDs
STANDARD_DEVIATION 0.5 • n=5 Participants
Maximum Stimulated GH
20.5 nanogram per milliliter
STANDARD_DEVIATION 9.9 • n=5 Participants
Weight for Age SD Score
-2.3 SDs
STANDARD_DEVIATION 0.7 • n=5 Participants

PRIMARY outcome

Timeframe: Pretreatment to Week 34

Population: The ITT population included all treated subjects.

Height was measured standing without shoes as the average of three measurements by the same observer using identical technique with a Harpenden or other wall mounted stadiometer. The subject was repositioned between each measurement. Height velocity during an interval of time is defined as the change in height during the time interval divided by the duration of the time interval. Missing Week 34 heights were imputed using the last height SD score carried forward.

Outcome measures

Outcome measures
Measure
rhIGF-1 QD
n=45 Participants
During the treatment phase (Day 1 to Week 86), subjects received SC injections of rhIGF-1 at an initial dose of 60 mcg/kg QD starting on Day 1 (Visit 3). From Week 2 (Visit 4) subsequent dose adjustments were made in order to achieve the target serum IGF-1 concentration for the subject's age and sex. The maximum dose in any circumstance was 240 mcg/kg/day.
Height Velocity From Pretreatment (Week 0) to Week 34
7.0 centimeters per year (cm/yr)
Standard Deviation 1.5

PRIMARY outcome

Timeframe: Week 34 to 86

Population: The ITT population included all treated subjects. Only subjects in the ITT population who continued past Week 34 were included in the analysis.

Height was measured standing without shoes as the average of three measurements by the same observer using identical technique with a Harpenden or other wall mounted stadiometer. The subject was repositioned between each measurement. Height velocity during an interval of time is defined as the change in height during the time interval divided by the duration of the time interval. Missing Week 86 heights were imputed using the last height SD score carried forward.

Outcome measures

Outcome measures
Measure
rhIGF-1 QD
n=40 Participants
During the treatment phase (Day 1 to Week 86), subjects received SC injections of rhIGF-1 at an initial dose of 60 mcg/kg QD starting on Day 1 (Visit 3). From Week 2 (Visit 4) subsequent dose adjustments were made in order to achieve the target serum IGF-1 concentration for the subject's age and sex. The maximum dose in any circumstance was 240 mcg/kg/day.
Height Velocity From Week 34 to 86
6.7 cm/yr
Standard Deviation 1.8

SECONDARY outcome

Timeframe: Pretreatment and Week 34

Population: The ITT population included all treated subjects.

Height was measured standing without shoes as the average of three measurements by the same observer using identical technique with a Harpenden or other wall mounted stadiometer. The subject was repositioned between each measurement. The SD score is calculated as the subject value minus the mean divided by the standard deviation. The mean and the standard deviation vary depending on the age and sex of the child.

Outcome measures

Outcome measures
Measure
rhIGF-1 QD
n=45 Participants
During the treatment phase (Day 1 to Week 86), subjects received SC injections of rhIGF-1 at an initial dose of 60 mcg/kg QD starting on Day 1 (Visit 3). From Week 2 (Visit 4) subsequent dose adjustments were made in order to achieve the target serum IGF-1 concentration for the subject's age and sex. The maximum dose in any circumstance was 240 mcg/kg/day.
Change in Height SD Score From Pretreatment to Week 34
0.21 SDs
Standard Deviation 0.20

SECONDARY outcome

Timeframe: Pretreatment and Week 86

Population: The ITT population included all treated subjects. Only subjects who had both pretreatment and Week 86 measurements were included in the analysis.

Height was measured standing without shoes as the average of three measurements by the same observer using identical technique with a Harpenden or other wall mounted stadiometer. Subjects were repositioned between each measurement. The SD score is calculated as the patient value minus the mean divided by the standard deviation. The mean and the standard deviation vary depending on the age and sex of the child.

Outcome measures

Outcome measures
Measure
rhIGF-1 QD
n=40 Participants
During the treatment phase (Day 1 to Week 86), subjects received SC injections of rhIGF-1 at an initial dose of 60 mcg/kg QD starting on Day 1 (Visit 3). From Week 2 (Visit 4) subsequent dose adjustments were made in order to achieve the target serum IGF-1 concentration for the subject's age and sex. The maximum dose in any circumstance was 240 mcg/kg/day.
Change in Height SD Score From Pretreatment to Week 86
0.45 SDs
Standard Deviation 0.39

SECONDARY outcome

Timeframe: Pretreatment to Week 86

Population: The ITT population included all treated subjects. Only subjects who had both pretreatment and Week 86 measurements were included in the analysis.

Plain X-rays of the left hand and wrist were exposed for bone age appraisal. The films were sent to a central facility for standardized evaluation.

Outcome measures

Outcome measures
Measure
rhIGF-1 QD
n=30 Participants
During the treatment phase (Day 1 to Week 86), subjects received SC injections of rhIGF-1 at an initial dose of 60 mcg/kg QD starting on Day 1 (Visit 3). From Week 2 (Visit 4) subsequent dose adjustments were made in order to achieve the target serum IGF-1 concentration for the subject's age and sex. The maximum dose in any circumstance was 240 mcg/kg/day.
Change in Bone Age From Pretreatment to Week 86 Minus Change in Chronological Age
0.2 years
Standard Deviation 0.68

SECONDARY outcome

Timeframe: Pretreatment and Week 86

Population: The ITT population included all treated subjects. Only subjects who had both pretreatment and Week 86 measurements were included in the analysis.

Growth factor panels for measuring IGFBP-1, IGFBP-2 and IGFBP-3 were evaluated from screening and at each study visit up to Week 86. Inter-quartile range (Q1-Q3) is 10th to 90th percentile.

Outcome measures

Outcome measures
Measure
rhIGF-1 QD
n=29 Participants
During the treatment phase (Day 1 to Week 86), subjects received SC injections of rhIGF-1 at an initial dose of 60 mcg/kg QD starting on Day 1 (Visit 3). From Week 2 (Visit 4) subsequent dose adjustments were made in order to achieve the target serum IGF-1 concentration for the subject's age and sex. The maximum dose in any circumstance was 240 mcg/kg/day.
Percent Change in Serum Concentration of IGFBP-1, IGFBP-2 and IGFBP-3 From Pretreatment to Week 86
IGFBP-1
-89.6 percent change
Interval -96.0 to -13.0
Percent Change in Serum Concentration of IGFBP-1, IGFBP-2 and IGFBP-3 From Pretreatment to Week 86
IGFBP-2
37.9 percent change
Interval -25.0 to 227.0
Percent Change in Serum Concentration of IGFBP-1, IGFBP-2 and IGFBP-3 From Pretreatment to Week 86
IGFBP-3
0 percent change
Interval -27.0 to 48.0

SECONDARY outcome

Timeframe: Pretreatment and Week 86

Population: The ITT population included all treated subjects. Only subjects who had both pretreatment and Week 86 measurements were included in the analysis.

Growth factor panels for measuring ALS were evaluated from screening and at each study visit up to Week 86. Inter-quartile range (Q1-Q3) is 10th to 90th percentile.

Outcome measures

Outcome measures
Measure
rhIGF-1 QD
n=29 Participants
During the treatment phase (Day 1 to Week 86), subjects received SC injections of rhIGF-1 at an initial dose of 60 mcg/kg QD starting on Day 1 (Visit 3). From Week 2 (Visit 4) subsequent dose adjustments were made in order to achieve the target serum IGF-1 concentration for the subject's age and sex. The maximum dose in any circumstance was 240 mcg/kg/day.
Percent Change in Serum Concentration of ALS From Pretreatment to Week 86
-7.7 percent change
Interval -36.0 to 42.0

POST_HOC outcome

Timeframe: Pretreatment to Week 34

Population: Completer population included all subjects who remained on study to Week 86. Subjects who completed Week 34 were included in the analysis.

Height was measured standing without shoes as the average of three measurements by the same observer using identical technique with a Harpenden or other wall mounted stadiometer. The subject was repositioned between each measurement. Height velocity during an interval of time is defined as the change in height during the time interval divided by the duration of the time interval. Missing Week 34 heights were imputed using the last height SD score carried forward.

Outcome measures

Outcome measures
Measure
rhIGF-1 QD
n=43 Participants
During the treatment phase (Day 1 to Week 86), subjects received SC injections of rhIGF-1 at an initial dose of 60 mcg/kg QD starting on Day 1 (Visit 3). From Week 2 (Visit 4) subsequent dose adjustments were made in order to achieve the target serum IGF-1 concentration for the subject's age and sex. The maximum dose in any circumstance was 240 mcg/kg/day.
Increase in Height Velocity From Pretreatment to Week 34
1.3 cm/yr
Standard Deviation 3.56

POST_HOC outcome

Timeframe: Pretreatment to Week 86

Population: Completer population included all subjects who remained on study to Week 86.

Height was measured standing without shoes as the average of three measurements by the same observer using identical technique with a Harpenden or other wall mounted stadiometer. The subject was repositioned between each measurement. Height velocity during an interval of time is defined as the change in height during the time interval divided by the duration of the time interval. Missing Week 86 heights were imputed using the last height SD score carried forward.

Outcome measures

Outcome measures
Measure
rhIGF-1 QD
n=30 Participants
During the treatment phase (Day 1 to Week 86), subjects received SC injections of rhIGF-1 at an initial dose of 60 mcg/kg QD starting on Day 1 (Visit 3). From Week 2 (Visit 4) subsequent dose adjustments were made in order to achieve the target serum IGF-1 concentration for the subject's age and sex. The maximum dose in any circumstance was 240 mcg/kg/day.
Increase in Height Velocity From Pretreatment to Week 86
1.3 cm/yr
Standard Deviation 3.58

Adverse Events

rhIGF-1 QD

Serious events: 2 serious events
Other events: 41 other events
Deaths: 0 deaths

Serious adverse events

Serious adverse events
Measure
rhIGF-1 QD
n=45 participants at risk
During the treatment phase (Day 1 to Week 86), subjects received SC injections of rhIGF-1 at an initial dose of 60 mcg/kg QD starting on Day 1 (Visit 3). From Week 2 (Visit 4) subsequent dose adjustments were made in order to achieve the target serum IGF-1 concentration for the subject's age and sex. The maximum dose in any circumstance was 240 mcg/kg/day.
Infections and infestations
Gastroenteritis
2.2%
1/45 • Number of events 1 • Treatment emergent adverse events were collected from Day 1 to Week 86 (approximately 21 months).
Congenital, familial and genetic disorders
Arnold-Chiari Malformation
2.2%
1/45 • Number of events 1 • Treatment emergent adverse events were collected from Day 1 to Week 86 (approximately 21 months).
Nervous system disorders
Syringomyelia
2.2%
1/45 • Number of events 1 • Treatment emergent adverse events were collected from Day 1 to Week 86 (approximately 21 months).

Other adverse events

Other adverse events
Measure
rhIGF-1 QD
n=45 participants at risk
During the treatment phase (Day 1 to Week 86), subjects received SC injections of rhIGF-1 at an initial dose of 60 mcg/kg QD starting on Day 1 (Visit 3). From Week 2 (Visit 4) subsequent dose adjustments were made in order to achieve the target serum IGF-1 concentration for the subject's age and sex. The maximum dose in any circumstance was 240 mcg/kg/day.
Gastrointestinal disorders
Abdominal Pain
6.7%
3/45 • Number of events 3 • Treatment emergent adverse events were collected from Day 1 to Week 86 (approximately 21 months).
Gastrointestinal disorders
Abdominal Pain Upper
6.7%
3/45 • Number of events 4 • Treatment emergent adverse events were collected from Day 1 to Week 86 (approximately 21 months).
Gastrointestinal disorders
Diarrhoea
11.1%
5/45 • Number of events 6 • Treatment emergent adverse events were collected from Day 1 to Week 86 (approximately 21 months).
Gastrointestinal disorders
Stomach Discomfort
6.7%
3/45 • Number of events 3 • Treatment emergent adverse events were collected from Day 1 to Week 86 (approximately 21 months).
Gastrointestinal disorders
Vomiting
40.0%
18/45 • Number of events 21 • Treatment emergent adverse events were collected from Day 1 to Week 86 (approximately 21 months).
General disorders
Injection Site Bruising
6.7%
3/45 • Number of events 5 • Treatment emergent adverse events were collected from Day 1 to Week 86 (approximately 21 months).
General disorders
Pyrexia
31.1%
14/45 • Number of events 18 • Treatment emergent adverse events were collected from Day 1 to Week 86 (approximately 21 months).
Infections and infestations
Gastroenteritis
13.3%
6/45 • Number of events 8 • Treatment emergent adverse events were collected from Day 1 to Week 86 (approximately 21 months).
Infections and infestations
Gastroenteritis Viral
6.7%
3/45 • Number of events 4 • Treatment emergent adverse events were collected from Day 1 to Week 86 (approximately 21 months).
Infections and infestations
Influenza
11.1%
5/45 • Number of events 6 • Treatment emergent adverse events were collected from Day 1 to Week 86 (approximately 21 months).
Infections and infestations
Nasopharyngitis
6.7%
3/45 • Number of events 5 • Treatment emergent adverse events were collected from Day 1 to Week 86 (approximately 21 months).
Infections and infestations
Otitis Media
11.1%
5/45 • Number of events 7 • Treatment emergent adverse events were collected from Day 1 to Week 86 (approximately 21 months).
Infections and infestations
Sinusitis
8.9%
4/45 • Number of events 4 • Treatment emergent adverse events were collected from Day 1 to Week 86 (approximately 21 months).
Infections and infestations
Upper Respiratory Tract Infection
28.9%
13/45 • Number of events 24 • Treatment emergent adverse events were collected from Day 1 to Week 86 (approximately 21 months).
Infections and infestations
Viral Infection
15.6%
7/45 • Number of events 13 • Treatment emergent adverse events were collected from Day 1 to Week 86 (approximately 21 months).
Metabolism and nutrition disorders
Hypoglycaemia
11.1%
5/45 • Number of events 14 • Treatment emergent adverse events were collected from Day 1 to Week 86 (approximately 21 months).
Musculoskeletal and connective tissue disorders
Arthralgia
13.3%
6/45 • Number of events 8 • Treatment emergent adverse events were collected from Day 1 to Week 86 (approximately 21 months).
Nervous system disorders
Headache
40.0%
18/45 • Number of events 32 • Treatment emergent adverse events were collected from Day 1 to Week 86 (approximately 21 months).
Respiratory, thoracic and mediastinal disorders
Cough
24.4%
11/45 • Number of events 13 • Treatment emergent adverse events were collected from Day 1 to Week 86 (approximately 21 months).
Respiratory, thoracic and mediastinal disorders
Nasal Congestion
13.3%
6/45 • Number of events 9 • Treatment emergent adverse events were collected from Day 1 to Week 86 (approximately 21 months).
Respiratory, thoracic and mediastinal disorders
Pharyngolaryngeal Pain
8.9%
4/45 • Number of events 5 • Treatment emergent adverse events were collected from Day 1 to Week 86 (approximately 21 months).
Respiratory, thoracic and mediastinal disorders
Rhinorrhoea
6.7%
3/45 • Number of events 3 • Treatment emergent adverse events were collected from Day 1 to Week 86 (approximately 21 months).
Skin and subcutaneous tissue disorders
Dermatitis Contact
6.7%
3/45 • Number of events 3 • Treatment emergent adverse events were collected from Day 1 to Week 86 (approximately 21 months).
Skin and subcutaneous tissue disorders
Rash
11.1%
5/45 • Number of events 7 • Treatment emergent adverse events were collected from Day 1 to Week 86 (approximately 21 months).

Additional Information

Medical Director

Ipsen

Phone: see email

Results disclosure agreements

  • Principal investigator is a sponsor employee
  • Publication restrictions are in place

Restriction type: OTHER