Trial Outcomes & Findings for A Study to Describe the Experience of Both Patients and Their Clinicians in the Treatment of Fabry Disease With Enzyme Replacement Therapy. (NCT NCT04281537)

NCT ID: NCT04281537

Last Updated: 2026-02-05

Results Overview

Total time (minutes) spent by HCPs on all activities related to administration of a single dose of ERT in patients with Fabry Disease including pre-infusion (pre-administration patient consultation; infusion and premedication preparation), ERT (and premedication) administration, and post-administration assessment and documentation. Results are shown for all patients with Fabry Disease on ERT in study

Recruitment status

COMPLETED

Target enrollment

82 participants

Primary outcome timeframe

up to 7 weeks

Results posted on

2026-02-05

Participant Flow

76 patients with Fabry Disease on enzyme replacement therapy (ERT) and 6 caregivers of patients with Fabry Disease on ERT were enrolled into the study per protocol for a total of 82 participants. As was pre-specified in the study protocol, the Agalsidase Alfa and Agalsidase Beta arms are reported as a combined group ("Patients with Fabry Disease on ERT") and not separately.

Participant milestones

Participant milestones
Measure
Patients With Fabry Disease on ERT
Patients with Fabry Disease receiving Enzyme Replacement Therapy (agalsidase alfa or agalsidase beta) infusion every other week
Caregiver
Caregiver of patient with Fabry Disease on ERT
Overall Study
STARTED
76
6
Overall Study
COMPLETED
76
6
Overall Study
NOT COMPLETED
0
0

Reasons for withdrawal

Withdrawal data not reported

Baseline Characteristics

Age of caregivers was not reported in same manner as for patients with Fabry Disease. Caregiver ages reported as counts within a range: age 31-40 (n=2), 41-50 (n=3), and 61-70 (n=1)

Baseline characteristics by cohort

Baseline characteristics by cohort
Measure
Patients With Fabry Disease on ERT
n=76 Participants
Patients with Fabry Disease receiving Enzyme Replacement Therapy (agalsidase alfa or agalsidase beta) infusion every other week
Caregiver
n=6 Participants
Caregiver of patient with Fabry Disease on ERT
Total
n=82 Participants
Total of all reporting groups
Age, Continuous
48.9 years
n=76 Participants • Age of caregivers was not reported in same manner as for patients with Fabry Disease. Caregiver ages reported as counts within a range: age 31-40 (n=2), 41-50 (n=3), and 61-70 (n=1)
48.9 years
n=76 Participants • Age of caregivers was not reported in same manner as for patients with Fabry Disease. Caregiver ages reported as counts within a range: age 31-40 (n=2), 41-50 (n=3), and 61-70 (n=1)
Age, Customized
Caregiver age range · 31 to 40 years
0 Participants
Caregiver ages were reported as a count within a range. Data for ages of patients with Fabry Disease on ERT are reported separately on a per patient basis
2 Participants
n=6 Participants • Caregiver ages were reported as a count within a range. Data for ages of patients with Fabry Disease on ERT are reported separately on a per patient basis
2 Participants
n=6 Participants • Caregiver ages were reported as a count within a range. Data for ages of patients with Fabry Disease on ERT are reported separately on a per patient basis
Age, Customized
Caregiver age range · 41 to 50 years
0 Participants
Caregiver ages were reported as a count within a range. Data for ages of patients with Fabry Disease on ERT are reported separately on a per patient basis
3 Participants
n=6 Participants • Caregiver ages were reported as a count within a range. Data for ages of patients with Fabry Disease on ERT are reported separately on a per patient basis
3 Participants
n=6 Participants • Caregiver ages were reported as a count within a range. Data for ages of patients with Fabry Disease on ERT are reported separately on a per patient basis
Age, Customized
Caregiver age range · 61 to 70 years
0 Participants
Caregiver ages were reported as a count within a range. Data for ages of patients with Fabry Disease on ERT are reported separately on a per patient basis
1 Participants
n=6 Participants • Caregiver ages were reported as a count within a range. Data for ages of patients with Fabry Disease on ERT are reported separately on a per patient basis
1 Participants
n=6 Participants • Caregiver ages were reported as a count within a range. Data for ages of patients with Fabry Disease on ERT are reported separately on a per patient basis
Sex: Female, Male
Female
31 Participants
n=76 Participants
5 Participants
n=6 Participants
36 Participants
n=82 Participants
Sex: Female, Male
Male
45 Participants
n=76 Participants
1 Participants
n=6 Participants
46 Participants
n=82 Participants
Race and Ethnicity Not Collected
0 Participants
Race and Ethnicity were not collected from any participant.
Region of Enrollment
Turkey
19 participants
n=76 Participants
1 participants
n=6 Participants
20 participants
n=82 Participants
Region of Enrollment
Japan
4 participants
n=76 Participants
0 participants
n=6 Participants
4 participants
n=82 Participants
Region of Enrollment
Taiwan
30 participants
n=76 Participants
5 participants
n=6 Participants
35 participants
n=82 Participants
Region of Enrollment
Brazil
23 participants
n=76 Participants
0 participants
n=6 Participants
23 participants
n=82 Participants
ERT Administered to Patients
Agalsidase alfa/biosimilar
28 Participants
n=76 Participants • Type of ERT is not applicable to caregiver group
0 Participants
Type of ERT is not applicable to caregiver group
28 Participants
n=76 Participants • Type of ERT is not applicable to caregiver group
ERT Administered to Patients
Agalsidase beta/biosimilar
48 Participants
n=76 Participants • Type of ERT is not applicable to caregiver group
0 Participants
Type of ERT is not applicable to caregiver group
48 Participants
n=76 Participants • Type of ERT is not applicable to caregiver group

PRIMARY outcome

Timeframe: up to 7 weeks

Population: As was pre-specified in the study protocol, the Agalsidase Alfa and Agalsidase Beta arms are reported as a combined group ("Patients with Fabry Disease on ERT") and not separately.

Total time (minutes) spent by HCPs on all activities related to administration of a single dose of ERT in patients with Fabry Disease including pre-infusion (pre-administration patient consultation; infusion and premedication preparation), ERT (and premedication) administration, and post-administration assessment and documentation. Results are shown for all patients with Fabry Disease on ERT in study

Outcome measures

Outcome measures
Measure
Patients With Fabry Disease on ERT
n=76 Participants
Patients with Fabry Disease receiving Enzyme Replacement Therapy (ERT) (agalsidase alfa or agalsidase beta) infusion every other week
Total Time Spent by Healthcare Professionals (HCPs) in the Preparation/Administration of a Single Dose of ERT in Patients With Fabry Disease
151.9 minutes
Standard Deviation 62.5

SECONDARY outcome

Timeframe: up to 7 weeks

Population: As was pre-specified in the study protocol, the Agalsidase Alfa and Agalsidase Beta arms are reported as a combined group ("Patients with Fabry Disease on ERT") and not separately.

Total time spent (in minutes) by healthcare professionals (HCP) on tasks associated with preparation and administration of a single dose of ERT (with agalsidase alfa or agalsidase beta) in patients with Fabry Disease including pre-administration activities (patient consultation, infusion/pre-medication preparation activities), administration of ERT (and pre-medications), and post-infusion activities (monitoring of patient during and after administration, completion of clinical documentation, and any other ERT-administration related activities).

Outcome measures

Outcome measures
Measure
Patients With Fabry Disease on ERT
n=76 Participants
Patients with Fabry Disease receiving Enzyme Replacement Therapy (ERT) (agalsidase alfa or agalsidase beta) infusion every other week
Total Time Spent by HCPs on Each Separate Task Associated With the Preparation and Administration of a Single Dose of ERT (With Agalsidase Alfa or Agalsidase Beta).
Time Spent by HCP on Pre-infusion activities
20.9 minutes
Standard Deviation 13.4
Total Time Spent by HCPs on Each Separate Task Associated With the Preparation and Administration of a Single Dose of ERT (With Agalsidase Alfa or Agalsidase Beta).
Time spent by HCP on ERT administration
118.2 minutes
Standard Deviation 56.3
Total Time Spent by HCPs on Each Separate Task Associated With the Preparation and Administration of a Single Dose of ERT (With Agalsidase Alfa or Agalsidase Beta).
Time spent by HCP on post-infusion activities
12.8 minutes
Standard Deviation 9.6

SECONDARY outcome

Timeframe: up to 7 weeks

Population: As was pre-specified in the study protocol, the Agalsidase Alfa and Agalsidase Beta arms are reported as a combined group ("Patients with Fabry Disease on ERT") and not separately. Data were not available for 1 patient.

Total patient time associated with attendance for administration of a single dose of ERT includes the total time from when the patient left home, to when they returned home and based on the reported time spent travelling, total waiting time, the total time spent during infusion episode (pre, during and post) and the other recorded activities.

Outcome measures

Outcome measures
Measure
Patients With Fabry Disease on ERT
n=75 Participants
Patients with Fabry Disease receiving Enzyme Replacement Therapy (ERT) (agalsidase alfa or agalsidase beta) infusion every other week
Total Patient Time Associated With Attendance for the Administration of a Single Dose of ERT (With Agalsidase Alfa or Agalsidase Beta).
364.1 minutes
Standard Deviation 193.9

SECONDARY outcome

Timeframe: up to 7 weeks

Population: Patients with Fabry Disease in Brazil. As was pre-specified in the study protocol, the Agalsidase Alfa and Agalsidase Beta arms are reported as a combined group ("Patients with Fabry Disease on ERT") and not separately. Data were not available for 2 patients.

Total out-of-pocket costs (eg, food, travel, internet) incurred by patients in Brazil that were directly related to attendance for the administration of a single dose of ERT.

Outcome measures

Outcome measures
Measure
Patients With Fabry Disease on ERT
n=21 Participants
Patients with Fabry Disease receiving Enzyme Replacement Therapy (ERT) (agalsidase alfa or agalsidase beta) infusion every other week
Total Costs Associated With Attendance for the Administration of a Single Dose of ERT (With Agalsidase Alfa or Agalsidase Beta) - Brazil.
166.5 R$
Standard Deviation 214.8

SECONDARY outcome

Timeframe: up to 7 weeks

Population: Patients with Fabry Disease in Taiwan. As was pre-specified in the study protocol, the Agalsidase Alfa and Agalsidase Beta arms are reported as a combined group ("Patients with Fabry Disease on ERT") and not separately. Data were not available for 1 patient.

Total out-of-pocket costs (eg, food, travel, internet) incurred by patients in Taiwan that were directly related to attendance for the administration of a single dose of ERT.

Outcome measures

Outcome measures
Measure
Patients With Fabry Disease on ERT
n=29 Participants
Patients with Fabry Disease receiving Enzyme Replacement Therapy (ERT) (agalsidase alfa or agalsidase beta) infusion every other week
Total Costs Associated With Attendance for the Administration of a Single Dose of ERT (With Agalsidase Alfa or Agalsidase Beta) - Taiwan.
709.5 NT$
Standard Deviation 855.9

SECONDARY outcome

Timeframe: up to 7 weeks

Population: Patients with Fabry Disease in Japan. As was pre-specified in the study protocol, the Agalsidase Alfa and Agalsidase Beta arms are reported as a combined group ("Patients with Fabry Disease on ERT") and not separately.

Total out-of-pocket costs (eg, food, travel, internet) incurred by patients in Japan that were directly related to attendance for the administration of a single dose of ERT.

Outcome measures

Outcome measures
Measure
Patients With Fabry Disease on ERT
n=4 Participants
Patients with Fabry Disease receiving Enzyme Replacement Therapy (ERT) (agalsidase alfa or agalsidase beta) infusion every other week
Total Costs Associated With Attendance for the Administration of a Single Dose of ERT (With Agalsidase Alfa or Agalsidase Beta) - Japan.
8781.0 ¥
Standard Deviation 15956.6

SECONDARY outcome

Timeframe: up to 7 weeks

Population: Patients with Fabry Disease in Turkey. As was pre-specified in the study protocol, the Agalsidase Alfa and Agalsidase Beta arms are reported as a combined group ("Patients with Fabry Disease on ERT") and not separately. Data were not available for 1 patient.

Total out-of-pocket costs (eg, food, travel, internet) incurred by patients in Turkey that were directly related to attendance for the administration of a single dose of ERT.

Outcome measures

Outcome measures
Measure
Patients With Fabry Disease on ERT
n=18 Participants
Patients with Fabry Disease receiving Enzyme Replacement Therapy (ERT) (agalsidase alfa or agalsidase beta) infusion every other week
Total Costs Associated With Attendance for the Administration of a Single Dose of ERT (With Agalsidase Alfa or Agalsidase Beta) - Turkey
470.8 ₺
Standard Deviation 1005.5

SECONDARY outcome

Timeframe: up to 7 weeks

Population: As was pre-specified in the study protocol, the Agalsidase Alfa and Agalsidase Beta arms are reported as a combined group ("Patients with Fabry Disease on ERT") and not separately. Note: data were missing for 54% \[41/76\] of patients.

The percentage of patients known to have taken time off from work during their ERT episode

Outcome measures

Outcome measures
Measure
Patients With Fabry Disease on ERT
n=35 Participants
Patients with Fabry Disease receiving Enzyme Replacement Therapy (ERT) (agalsidase alfa or agalsidase beta) infusion every other week
Percentage of Patients With Work Absence Due to Attendance for This ERT Episode.
16 Participants

SECONDARY outcome

Timeframe: up to 7 weeks

Population: As was pre-specified in the study protocol, the Agalsidase Alfa and Agalsidase Beta arms are reported as a combined group ("Patients with Fabry Disease on ERT") and not separately. Note: data were missing for 54% \[41/76\] of patients.

The number of paid and unpaid hours that patients were absent from work during their ERT episode

Outcome measures

Outcome measures
Measure
Patients With Fabry Disease on ERT
n=35 Participants
Patients with Fabry Disease receiving Enzyme Replacement Therapy (ERT) (agalsidase alfa or agalsidase beta) infusion every other week
Number of Hours of Patient Work Absence Due to Attendance for ERT Episode.
Number of Paid Hours Absent from Work
6.6 hours
Standard Deviation 5.4
Number of Hours of Patient Work Absence Due to Attendance for ERT Episode.
Number of Unpaid Hours Absent from Work
5.3 hours
Standard Deviation 5.4

SECONDARY outcome

Timeframe: up to 7 weeks

Total caregiver time (in minutes) associated with accompanying a patient with Fabry Disease for the administration of a single dose of ERT (with agalsidase alfa or agalsidase beta).

Outcome measures

Outcome measures
Measure
Patients With Fabry Disease on ERT
n=6 Participants
Patients with Fabry Disease receiving Enzyme Replacement Therapy (ERT) (agalsidase alfa or agalsidase beta) infusion every other week
Total Caregiver Time Associated With Accompanying a Patient With Fabry Disease for the Administration of a Single Dose of ERT
160 minutes
Standard Deviation 70.43

SECONDARY outcome

Timeframe: up to 7 weeks

Population: Data presented are from Taiwan in New Taiwan dollars (NT$). No data were reported for the 1 caregiver in Turkey for this outcome measure.

Total costs associated with accompanying a patient with Fabry Disease for the administration of a single dose of ERT (with agalsidase alfa or agalsidase beta).

Outcome measures

Outcome measures
Measure
Patients With Fabry Disease on ERT
n=5 Participants
Patients with Fabry Disease receiving Enzyme Replacement Therapy (ERT) (agalsidase alfa or agalsidase beta) infusion every other week
Total Costs Associated With Accompanying a Patient With Fabry Disease for the Administration of a Single Dose of ERT
2085 NT$
Standard Deviation 1728.6

SECONDARY outcome

Timeframe: up to 7 weeks

Percentage of caregivers with work absence due to accompanying the patient for this episode of ERT administration.

Outcome measures

Outcome measures
Measure
Patients With Fabry Disease on ERT
n=6 Participants
Patients with Fabry Disease receiving Enzyme Replacement Therapy (ERT) (agalsidase alfa or agalsidase beta) infusion every other week
Percentage of Caregivers With Work Absence Due to Accompanying the Patient for ERT Administration.
3 Participants

SECONDARY outcome

Timeframe: up to 7 weeks

Population: Data are available from 3 caregivers in Taiwan.

Number of hours of caregivers work absence due to accompanying the patient for this ERT administration episode.

Outcome measures

Outcome measures
Measure
Patients With Fabry Disease on ERT
n=3 Participants
Patients with Fabry Disease receiving Enzyme Replacement Therapy (ERT) (agalsidase alfa or agalsidase beta) infusion every other week
Number of Hours of Caregivers Work Absence Due to Accompanying the Patient for ERT Administration.
Number of Paid Hours Absent from Work
6.0 hours
Interval 6.0 to 6.0
Number of Hours of Caregivers Work Absence Due to Accompanying the Patient for ERT Administration.
Number of Unpaid Hours Absent from Work
3.0 hours
Interval 1.5 to 4.5

SECONDARY outcome

Timeframe: HRQoL was completed once (during the ERT infusion visit) prior to leaving the clinic

Population: As was pre-specified in the study protocol, the Agalsidase Alfa and Agalsidase Beta arms are reported as a combined group ("Patients with Fabry Disease on ERT") and not separately. The HRQoL was completed only one time per patient.

Health Related Quality of Life (HRQoL) measured by the 12 Item Short Form Survey \[SF-12\]). In the SF-12, patients were asked to rate aspects of their health across 8 domains; all domains have a maximum value of 5 (range 0-5) except physical functioning, for which the maximum score is 3 (range 0-3). Higher scores indicate better HRQoL while low scores indicate worse HRQoL.

Outcome measures

Outcome measures
Measure
Patients With Fabry Disease on ERT
n=76 Participants
Patients with Fabry Disease receiving Enzyme Replacement Therapy (ERT) (agalsidase alfa or agalsidase beta) infusion every other week
Health Related Quality of Life (HRQoL) (12 Item Short Form Survey [SF-12] Scores / Responses)
General Health
2.9 score on a scale
Standard Deviation 1.1
Health Related Quality of Life (HRQoL) (12 Item Short Form Survey [SF-12] Scores / Responses)
Physical Functioning
2.2 score on a scale
Standard Deviation 0.7
Health Related Quality of Life (HRQoL) (12 Item Short Form Survey [SF-12] Scores / Responses)
Role Physical
3.3 score on a scale
Standard Deviation 1.1
Health Related Quality of Life (HRQoL) (12 Item Short Form Survey [SF-12] Scores / Responses)
Role Emotional
3.6 score on a scale
Standard Deviation 1.1
Health Related Quality of Life (HRQoL) (12 Item Short Form Survey [SF-12] Scores / Responses)
Bodily Pain
3.6 score on a scale
Standard Deviation 1.2
Health Related Quality of Life (HRQoL) (12 Item Short Form Survey [SF-12] Scores / Responses)
Mental Health
3.5 score on a scale
Standard Deviation 0.9
Health Related Quality of Life (HRQoL) (12 Item Short Form Survey [SF-12] Scores / Responses)
Vitality
3.1 score on a scale
Standard Deviation 1.1
Health Related Quality of Life (HRQoL) (12 Item Short Form Survey [SF-12] Scores / Responses)
Social Function
3.6 score on a scale
Standard Deviation 1.2

SECONDARY outcome

Timeframe: WHO-5 was completed once (during the ERT infusion visit) prior to leaving the clinic

Population: As was pre-specified in the study protocol, the Agalsidase Alfa and Agalsidase Beta arms are reported as a combined group ("Patients with Fabry Disease on ERT") and not separately.

World Health Organization-5 Wellbeing Index \[WHO-5\] consists of 5 items and the timeframe for responses is based on the previous 2 weeks. The 5 items relate to feeling cheerful, calm, active, rested and being interested in life. For each question, patients rated the extent to which the statement applies between 0 (none of the time) and 5 (all of the time), with higher scores indicating better wellbeing. The WHO-5 total score is calculated by totaling the values of each of the five questions. The total scores range from 0 to 25, where 0 represents the worst possible quality of life and 25 represents the best possible quality of life.

Outcome measures

Outcome measures
Measure
Patients With Fabry Disease on ERT
n=76 Participants
Patients with Fabry Disease receiving Enzyme Replacement Therapy (ERT) (agalsidase alfa or agalsidase beta) infusion every other week
Patients General Wellbeing Measured by World Health Organization-5 Wellbeing Index (WHO-5) Scores / Responses During ERT Infusion Visit
Item 1: felt cheerful and in good spirts
3.1 score on a scale
Standard Deviation 1.3
Patients General Wellbeing Measured by World Health Organization-5 Wellbeing Index (WHO-5) Scores / Responses During ERT Infusion Visit
Item 2: felt calm and relaxed
3.2 score on a scale
Standard Deviation 1.3
Patients General Wellbeing Measured by World Health Organization-5 Wellbeing Index (WHO-5) Scores / Responses During ERT Infusion Visit
Item 3: felt active and vigorous
2.8 score on a scale
Standard Deviation 1.4
Patients General Wellbeing Measured by World Health Organization-5 Wellbeing Index (WHO-5) Scores / Responses During ERT Infusion Visit
Item 4: wake up feeling fresh and restful
2.6 score on a scale
Standard Deviation 1.5
Patients General Wellbeing Measured by World Health Organization-5 Wellbeing Index (WHO-5) Scores / Responses During ERT Infusion Visit
Item 5: daily life filled with things that interest me
3.4 score on a scale
Standard Deviation 1.2
Patients General Wellbeing Measured by World Health Organization-5 Wellbeing Index (WHO-5) Scores / Responses During ERT Infusion Visit
WHO-5 Total Score
15.1 score on a scale
Standard Deviation 5.6

SECONDARY outcome

Timeframe: Questionnaire completed during ERT infusion (baseline), the same evening as date of ERT infusion, and once during the period of 1 to 7 days post-infusion

Population: As was pre-specified in the study protocol, the Agalsidase Alfa and Agalsidase Beta arms are reported as a combined group ("Patients with Fabry Disease on ERT") and not separately.

Questionnaire measuring Fatigue. Possible scores range from 1 (not at all tired) to 5 (extremely tired); higher scores therefore indicating worse fatigue. Negative value in change from during ERT infusion to evening of ERT infusion indicates more fatigue in the evening of infusion. Negative value in change from baseline (during ERT infusion) to 1-7 days after ERT infusion indicates more fatigue during the period occurring 1-7 days after ERT infusion.

Outcome measures

Outcome measures
Measure
Patients With Fabry Disease on ERT
n=76 Participants
Patients with Fabry Disease receiving Enzyme Replacement Therapy (ERT) (agalsidase alfa or agalsidase beta) infusion every other week
Patients Level of Fatigue Measured by Fatigue Likert Scale
Level of fatigue during infusion
2.4 score on a scale
Standard Deviation 1.4
Patients Level of Fatigue Measured by Fatigue Likert Scale
Change in level of fatigue from during ERT infusion to evening of ERT infusion
-0.2 score on a scale
Standard Deviation 1.4
Patients Level of Fatigue Measured by Fatigue Likert Scale
Change in level of fatigue from during ERT infusion to 1-7 days after ERT infusion
0.1 score on a scale
Standard Deviation 1.3

SECONDARY outcome

Timeframe: WPAI completed once by patients during the period of 1 to 7 Days After ERT Infusion

Population: As was pre-specified in the study protocol, the Agalsidase Alfa and Agalsidase Beta arms are reported as a combined group ("Patients with Fabry Disease on ERT") and not separately.

Work Productivity and Activity Index (WPAI) consists of 6 items on the effect of health problems on ability to work and perform regular activities: 1=currently employed; 2=hours missed due to health problems; 3=hours missed other reasons; 4=hours actually worked; 5=degree health affected productivity while working; 6=degree health affected regular activities Score calculation (multiplied by 100 for percent): * Percent work time missed due to health: Q2/(Q2+Q4) * Percent impairment while working due to health: Q5/10 * Percent overall work impairment due to health: Q2/(Q2+Q4) + \[(1-(Q2/(Q2+Q4))) x (Q5/10)\] * Percent activity impairment due to health: Q6/10 Final scores are expressed as impairment percentages (range: 0-100%), with higher numbers indicating greater impairment/less productivity (ie, worse outcomes) and lower numbers indicating less impairment/greater productivity (ie, better outcomes). Data were collected 1 to 7 days after ERT infusion.

Outcome measures

Outcome measures
Measure
Patients With Fabry Disease on ERT
n=76 Participants
Patients with Fabry Disease receiving Enzyme Replacement Therapy (ERT) (agalsidase alfa or agalsidase beta) infusion every other week
Patients Levels of Work Impairment (Work Productivity and Activity Index [WPAI] Scores) - 1 to 7 Days After ERT Infusion
% Activity Impairment Due to Health
30.8 Percent
Standard Deviation 29.3
Patients Levels of Work Impairment (Work Productivity and Activity Index [WPAI] Scores) - 1 to 7 Days After ERT Infusion
% Work Time Missed Due to Health (Absenteeism)
5.0 Percent
Standard Deviation 9.5
Patients Levels of Work Impairment (Work Productivity and Activity Index [WPAI] Scores) - 1 to 7 Days After ERT Infusion
% Impairment While Working Due to Health (Presenteeism)
28.6 Percent
Standard Deviation 29.5
Patients Levels of Work Impairment (Work Productivity and Activity Index [WPAI] Scores) - 1 to 7 Days After ERT Infusion
% Overall Work Impairment Due to Health
28.9 Percent
Standard Deviation 27.7

SECONDARY outcome

Timeframe: WPAI completed once by patients on the day of the next ERT infusion (approximately 15 days after the prior infusion)

Population: As was pre-specified in the study protocol, the Agalsidase Alfa and Agalsidase Beta arms are reported as a combined group ("Patients with Fabry Disease on ERT") and not separately.

Work Productivity and Activity Index (WPAI) consists of 6 items on the effect of health problems on ability to work and perform regular activities: 1=currently employed; 2=hours missed due to health problems; 3=hours missed other reasons; 4=hours actually worked; 5=degree health affected productivity while working; 6=degree health affected regular activities Score calculation (multiplied by 100 for percent): * Percent work time missed due to health: Q2/(Q2+Q4) * Percent impairment while working due to health: Q5/10 * Percent overall work impairment due to health: Q2/(Q2+Q4) + \[(1-(Q2/(Q2+Q4))) x (Q5/10)\] * Percent activity impairment due to health: Q6/10 Final scores are expressed as impairment percentages (range: 0-100%), with higher numbers indicating greater impairment/less productivity (ie, worse outcomes) and lower numbers indicating less impairment/greater productivity (ie, better outcomes). Data were collected on day of next ERT infusion.

Outcome measures

Outcome measures
Measure
Patients With Fabry Disease on ERT
n=76 Participants
Patients with Fabry Disease receiving Enzyme Replacement Therapy (ERT) (agalsidase alfa or agalsidase beta) infusion every other week
Patients Levels of Work Impairment (Work Productivity and Activity Index [WPAI] Scores) - Day of Next ERT Infusion
% Work Time Missed Due to Health (Absenteeism)
3.7 Percent
Standard Deviation 7.3
Patients Levels of Work Impairment (Work Productivity and Activity Index [WPAI] Scores) - Day of Next ERT Infusion
% Impairment While Working Due to Health (Presenteeism)
22.6 Percent
Standard Deviation 24.9
Patients Levels of Work Impairment (Work Productivity and Activity Index [WPAI] Scores) - Day of Next ERT Infusion
% Overall Work Impairment Due to Health
25.7 Percent
Standard Deviation 26.4
Patients Levels of Work Impairment (Work Productivity and Activity Index [WPAI] Scores) - Day of Next ERT Infusion
% Activity Impairment Due to Health
32.3 Percent
Standard Deviation 30.4

SECONDARY outcome

Timeframe: WPAI-CG completed once by caregivers during the period of 1 to 7 days after ERT infusion

Work Productivity and Activity Index (WPAI) consists of 6 items on the effect of health problems on ability to work and perform regular activities: 1=currently employed; 2=hours missed due to health problems; 3=hours missed other reasons; 4=hours actually worked; 5=degree health affected productivity while working; 6=degree health affected regular activities Score calculation (multiplied by 100 for percent): * Percent work time missed due to health: Q2/(Q2+Q4) * Percent impairment while working due to health: Q5/10 * Percent overall work impairment due to health: Q2/(Q2+Q4) + \[(1-(Q2/(Q2+Q4))) x (Q5/10)\] * Percent activity impairment due to health: Q6/10 Final scores are expressed as impairment percentages (range: 0-100%), with higher numbers indicating greater impairment/less productivity (ie, worse outcomes) and lower numbers indicating less impairment/greater productivity (ie, better outcomes). Data were collected 1 to 7 days after ERT infusion.

Outcome measures

Outcome measures
Measure
Patients With Fabry Disease on ERT
n=6 Participants
Patients with Fabry Disease receiving Enzyme Replacement Therapy (ERT) (agalsidase alfa or agalsidase beta) infusion every other week
Caregiver's Levels of Work Impairment (Work Productivity and Activity Index [WPAI]-CG Scores) - 1 to 7 Days After ERT Infusion.
% Work Time Missed Due to Health (Absenteeism)
0 Percent
Standard Deviation 0
Caregiver's Levels of Work Impairment (Work Productivity and Activity Index [WPAI]-CG Scores) - 1 to 7 Days After ERT Infusion.
% Impairment While Working Due to Health (Presenteeism)
17.5 Percent
Standard Deviation 20.6
Caregiver's Levels of Work Impairment (Work Productivity and Activity Index [WPAI]-CG Scores) - 1 to 7 Days After ERT Infusion.
% Overall Work Impairment Due to Health
17.5 Percent
Standard Deviation 20.6
Caregiver's Levels of Work Impairment (Work Productivity and Activity Index [WPAI]-CG Scores) - 1 to 7 Days After ERT Infusion.
% Activity Impairment Due to Health
18.3 Percent
Standard Deviation 18.3

SECONDARY outcome

Timeframe: WPAI-CG completed once by caregivers on day of the next ERT infusion (approximately 15 days after prior infusion)

Work Productivity and Activity Index (WPAI) consists of 6 items on the effect of health problems on ability to work and perform regular activities: 1=currently employed; 2=hours missed due to health problems; 3=hours missed other reasons; 4=hours actually worked; 5=degree health affected productivity while working; 6=degree health affected regular activities Score calculation (multiplied by 100 for percent): * Percent work time missed due to health: Q2/(Q2+Q4) * Percent impairment while working due to health: Q5/10 * Percent overall work impairment due to health: Q2/(Q2+Q4) + \[(1-(Q2/(Q2+Q4))) x (Q5/10)\] * Percent activity impairment due to health: Q6/10 Final scores are expressed as impairment percentages (range: 0-100%), with higher numbers indicating greater impairment/less productivity (ie, worse outcomes) and lower numbers indicating less impairment/greater productivity (ie, better outcomes). WPAI was completed by caregiver on day of next ERT infusion.

Outcome measures

Outcome measures
Measure
Patients With Fabry Disease on ERT
n=6 Participants
Patients with Fabry Disease receiving Enzyme Replacement Therapy (ERT) (agalsidase alfa or agalsidase beta) infusion every other week
Caregiver's Levels of Work Impairment (Work Productivity and Activity Index [WPAI]-CG Scores) - Day of Next ERT Infusion.
% Work Time Missed Due to Health (Absenteeism)
5.0 Percent
Standard Deviation 10.0
Caregiver's Levels of Work Impairment (Work Productivity and Activity Index [WPAI]-CG Scores) - Day of Next ERT Infusion.
% Impairment While Working Due to Health (Presenteeism)
17.5 Percent
Standard Deviation 20.6
Caregiver's Levels of Work Impairment (Work Productivity and Activity Index [WPAI]-CG Scores) - Day of Next ERT Infusion.
% Overall Work Impairment Due to Health
21.0 Percent
Standard Deviation 24.3
Caregiver's Levels of Work Impairment (Work Productivity and Activity Index [WPAI]-CG Scores) - Day of Next ERT Infusion.
% Activity Impairment Due to Health
16.7 Percent
Standard Deviation 18.6

SECONDARY outcome

Timeframe: up to 7 weeks

The Caregiver Strain Index (CSI) consists of 12 questions with Yes/No answer and measures the strain of care provision in five domains (financial, physical, psychological, social and personal). Yes = 1 and No = 0. The range is from 0-12. Total scores are the sum of all responses. A score of 7 or higher indicates a high level of stress.

Outcome measures

Outcome measures
Measure
Patients With Fabry Disease on ERT
n=6 Participants
Patients with Fabry Disease receiving Enzyme Replacement Therapy (ERT) (agalsidase alfa or agalsidase beta) infusion every other week
Level of Strain in Providing Care for a Patient With Fabry Disease (Caregiver Strain Index [CSI] Scores / Responses).
4.0 score on a scale
Standard Deviation 2.3

Adverse Events

Patient With Fabry Disease on ERT

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

Caregiver

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

Serious adverse events

Adverse event data not reported

Other adverse events

Adverse event data not reported

Additional Information

Patient Advocacy

Amicus Therapeutics, Inc.

Phone: 001 6096622000

Results disclosure agreements

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