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Move Forward Together

  • POMBILITI + OPFOLDA is the only therapy in LOPD studied in a head-to-head randomized controlled trial in predominantly ERT-experienced patients1-3
  • ERT-experienced patients in the trial were treated on average >7 years with ERT prior to entering the PROPEL clinical trial3
  • Walking ability and respiratory function were evaluated over 104 weeks4
  • Demonstrated safety over 104 weeks4

Study Design and Baseline Characteristics

PROPEL: The first and only head-to-head, randomized, Phase 3 trial in LOPD with a majority ERT-experienced patient cohort1-3

Nearly 80% of the study population was ERT-experienced

PROPEL: Study design-info graphic PROPEL: Study design-info graphic
  • Primary endpoint: Change from baseline to Week 52 in 6-minute walk distance (6MWD) for comparison of superiority3‡
  • Key secondary endpoint: Change from baseline to Week 52 in forced vital capacity (FVC)3
  • POMBILITI in combination with OPFOLDA is not approved for use in ERT-naïve patients with LOPD. The ERT-naïve patient subgroup enrolled too few patients to conclusively interpret the data1,3

6MWD, 6-minute walk distance; ERT, enzyme replacement therapy; FVC, forced vital capacity; IV, intravenous; LOPD, late-onset Pompe disease; QOW, every other week.
*After database lock, 1 ERT-naïve subject was found to have intentionally underperformed at baseline. This subject was excluded from all 6MWD efficacy results; this change did not alter the statistical outcomes of the study.5
Dosage based on patient weight.2
Primary endpoint for superiority was not met.3

All ERT-experienced patients in the study had been treated for years3

Minimum treatment time for inclusion was 2 years

PROPEL: Baseline characteristics for patients on POMBILITI + OPFOLDA versus the Comparator -info graphic PROPEL: Baseline characteristics for patients on POMBILITI + OPFOLDA versus the Comparator -info graphic

PROPEL: Results at 52 Weeks

ERT-experienced patients taking POMBILITI + OPFOLDA demonstrated measurable improvement in 6MWD and PP FVC vs comparator1,2§‖

Man walking-icon ERT-subgroup analysis: prespecified

Improvement in 6-Minute Walk Distance (6MWD)1,2

PROPEL: 6-minute walk distance (6MWD) results for patients on POMBILITI + OPFOLDA versus the Comparator-info graphic PROPEL: 6-minute walk distance (6MWD) results for patients on POMBILITI + OPFOLDA versus the Comparator-info graphic

PP, percent predicted; SE, standard error.
§A US-approved alglucosidase alfa product was not used in this clinical trial. Conclusions cannot be drawn from this clinical trial regarding comparative effectiveness between a US-approved alglucosidase alfa product and POMBILITI in combination with OPFOLDA.1,2
Primary endpoint for superiority was not met.3
For the ERT-experienced group, the treatment difference of the mean was estimated by nonparametric analysis of covariance that included treatment, gender, baseline 6MWD, age, weight, and height in the model. Missing data at Week 52 was imputed using last observed values.1,2

Lungs-icon ERT-subgroup analysis: prespecified

Improvement in Percent-Predicted (PP) Forced Vital Capacity (FVC)1,2

PROPEL: Forced vital capacity (FVC) results for patients on POMBILITI + OPFOLDA versus the Comparator-info graphic PROPEL: Forced vital capacity (FVC) results for patients on POMBILITI + OPFOLDA versus the Comparator-info graphic

#For the ERT-experienced group, the treatment difference of the mean was estimated by analysis of covariance that included treatment, gender, baseline FVC, age, weight, and height in the model. Missing data at Week 52 was imputed using last observed values.1,2

Hex4 biomarker-icon ERT-subgroup analysis: prespecified

Improvement in Hex4 Biomarker1

ERT-experienced patients taking POMBILITI + OPFOLDA showed a reduction in Hex4 (a metabolite of excess glycogen in patients with LOPD), as measured by Glc4 concentration vs an increase with the Comparator.

PROPEL: Hex4 results for patients on POMBILITI + OPFOLDA versus those on the Comparator-info graphic PROPEL: Hex4 results for patients on POMBILITI + OPFOLDA versus those on the Comparator-info graphic

Glc4, glucosidase tetrasaccharide; Hex4, hexose tetrasaccharide.

PROPEL: Safety Profile at 52 Weeks

Demonstrated Safety in 151 Patients Across 3 Clinical Trials1,2

PROPEL: Adverse reactions-info graphic PROPEL: Adverse reactions-info graphic

ADA, antidrug antibodies; PD, pharmacodynamics; PK, pharmacokinetics.
*Fulfilling at least one of the Sampson criteria.1

Study Design

PROPEL Open-Label Extension (OLE): Interim analysis evaluating the long-term efficacy and safety of POMBILITI + OPFOLDA for an additional 52 weeks4

  • After 52 weeks in the PROPEL study, 91 ERT-experienced patients and 28 ERT-naïve patients continued or crossed over to POMBILITI + OPFOLDA*†
  • Patients in the OLE study received POMBILITI + OPFOLDA every 2 weeks, beginning 2 weeks after the last dose of the ERT that they received in the PROPEL study
  • Baseline characteristics in PROPEL were representative of the LOPD population and generally similar between treatment groups

Select Endpoints4

  • Motor function: 6MWD (% predicted)
  • Respiratory function: FVC (sitting, % predicted)
  • Biomarkers: Hex4 (mmol/mol) and CK (U/L)

CK, creatine kinase; GSGC, Gait, Stairs, Gowers’ maneuver, Chair; Hex4, hexose tetrasaccharide; MMT, manual muscle test; OLE, open-label extension; PROMIS, Patient-Reported Outcomes Measurement Information System.
*POMBILITI in combination with OPFOLDA is not approved for use in ERT-naïve patients with LOPD. The ERT-naïve patient subgroup in PROPEL enrolled too few patients to conclusively interpret the data.1,3
ERT‑experienced patients are defined as those treated with ERT for at least 2 years prior to their participation in the PROPEL study.3
Dosage based on patient weight.1,2
§A US-approved alglucosidase alfa product was not used in this clinical trial. Conclusions cannot be drawn from this clinical trial regarding comparative effectiveness between a US-approved alglucosidase alfa product and POMBILITI in combination with OPFOLDA.1,2
Includes 1 patient who enrolled but was never dosed.4
11 patients discontinued due to withdrawn consent, adverse events, investigator decision, or because they were lost to follow-up.4

Important Study Limitations4

  • The OLE was unblinded and had no control group
  • This study includes data that are not in the FDA-approved Prescribing Informations for POMBILITI + OPFOLDA
  • Data were analyzed descriptively, with no statistical comparisons. Therefore, no conclusions regarding treatment differences should be made as results could represent chance findings
  • LOPD is a rare disease, so the sample size was relatively small
  • Given the heterogeneous nature of LOPD, spanning a wide spectrum of manifestations, disease severity, rates of progression, and responses to treatment, tailoring the individualized treatment approaches to optimize patient outcomes will become more critical
  • Creatine kinase (CK) is a nonspecific biomarker of muscle damage. The clinical relevancy of CK has not been established in LOPD6

OLE: Interim Analysis Through 104 Weeks

Hex4 biomarker-icon ERT-subgroup analysis: prespecified from PROPEL

Biomarker: Hex4 Levels

Hex4 is a metabolite of excess glycogen and is measured by Glc4 concentration1

OLE: Hex4 line graph results for patients on POMBILITI + OPFOLDA versus those on the Comparator-info graphic OLE: Hex4 line graph results for patients on POMBILITI + OPFOLDA versus those on the Comparator-info graphic

Elevated Hex4 levels are indicative of Pompe disease.7

OLE: Hex4 change from baseline to week 52 and 104 for patients on POMBILITI + OPFOLDA versus those on the Comparator-info graphic OLE: Hex4 change from baseline to week 52 and 104 for patients on POMBILITI + OPFOLDA versus those on the Comparator-info graphic

ERT-subgroup analysis: prespecified from PROPEL

Biomarker: Serum Creatine Kinase (CK) Levels

CK is a biomarker for muscle damage6

OLE: Serum CK line graph results for patients on POMBILITI + OPFOLDA versus those on the Comparator-info graphic OLE: Serum CK line graph results for patients on POMBILITI + OPFOLDA versus those on the Comparator-info graphic
OLE: Serum CK change from baseline to week 52 and 104 for patients on POMBILITI + OPFOLDA versus those on the Comparator-info graphic OLE: Serum CK change from baseline to week 52 and 104 for patients on POMBILITI + OPFOLDA versus those on the Comparator-info graphic

ERT-subgroup analysis: prespecified from PROPEL

6-Minute Walk Distance (6MWD) Results4

OLE: 6MWD line graph results for patients on POMBILITI + OPFOLDA versus those on the Comparator-info graphic OLE: 6MWD line graph results for patients on POMBILITI + OPFOLDA versus those on the Comparator-info graphic

#Primary endpoint for superiority was not met.4

OLE: 6MWD change from baseline to week 52 and 104 for patients on POMBILITI + OPFOLDA versus those on the Comparator-info graphic OLE: 6MWD change from baseline to week 52 and 104 for patients on POMBILITI + OPFOLDA versus those on the Comparator-info graphic

Lungs-icon ERT-subgroup analysis: prespecified from PROPEL

Percent-predicted (PP) Forced Vital Capacity (FVC) Results4

OLE: FVC line graph results for patients on POMBILITI + OPFOLDA versus those on the Comparator-info graphic OLE: FVC line graph results for patients on POMBILITI + OPFOLDA versus those on the Comparator-info graphic
OLE: FVC change from baseline to week 52 and 104 for patients on POMBILITI + OPFOLDA versus those on the Comparator-info graphic OLE: FVC change from baseline to week 52 and 104 for patients on POMBILITI + OPFOLDA versus those on the Comparator-info graphic

OLE: Safety Profile Through 104 Weeks

No New Safety Signals Were Identified During the OLE4

  • Most TEAEs were mild to moderate in severity
  • The most common TEAEs were headache, diarrhea, pyrexia, fatigue, and nausea
  • Five patients withdrew from the study due to TEAEs experienced during the OLE
OLE: Adverse reactions-info graphic

IARs, infusion-associated reactions; TEAEs, treatment-emergent adverse events.
Includes data from patients treated with POMBILITI + OPFOLDA in PROPEL who may or may not have continued POMBILITI + OPFOLDA in the OLE, including data from both PROPEL and the OLE.4
Includes data from the OLE only.4

PROPEL: Safety Profile at 52 Weeks

Demonstrated Safety in 151 Patients Across 3 Clinical Trials1,2

A total of 5 patients across 3 clinical trials PERMANENTLY DISCONTINUED POMBILITI + OPFOLDA due to adverse reactions (4/5 due to serious adverse reactions)1

Life-threatening hypersensitivity reactions, including anaphylaxis, have been reported in POMBILITI-treated patients1

  • In clinical trials, 41 (27%) POMBILITI-treated patients experienced hypersensitivity reactions, including 4 (3%) reporting severe hypersensitivity reactions and 4 (3%) additional patients who experienced anaphylaxis1*

  • Infusion-associated reactions were reported in 48 (32%) patients treated with POMBILITI + OPFOLDA across three clinical trials1
  • There was no identified clinically significant effect of ADA on POMBILITI + OPFOLDA PK or PD over the treatment duration of 52 weeks in PROPEL. Due to the small number of patients with negative ADA, the effect of ADA on the effectiveness of POMBILITI + OPFOLDA is unknown1

ADA, antidrug antibodies; PD, pharmacodynamics; PK, pharmacokinetics.
*Fulfilling at least one of the Sampson criteria.1

OLE: Safety Profile Through 104 Weeks

No New Safety Signals Were Identified During the OLE4

  • Most TEAEs were mild to moderate in severity
  • The most common TEAEs were headache, diarrhea, pyrexia, fatigue, and nausea
  • Five patients withdrew from the study due to TEAEs experienced during the OLE

IARs, infusion-associated reactions; TEAEs, treatment-emergent adverse events.
Includes data from patients treated with POMBILITI + OPFOLDA in PROPEL who may or may not have continued POMBILITI + OPFOLDA in the OLE, including data from both PROPEL and the OLE.4
Includes data from the OLE only.4

Click to see additional Important Safety Information, including BOXED WARNING.

See dosing and administration

References:

  1.  POMBILITI. Prescribing information. Amicus Therapeutics US, LLC; 2024.
  2.  OPFOLDA. Prescribing information. Amicus Therapeutics US, LLC; 2024.
  3.  Schoser B, Roberts M, Byrne BJ, et al. Safety and efficacy of cipaglucosidase alfa plus miglustat versus alglucosidase alfa plus placebo in late-onset Pompe disease (PROPEL): an international, randomised, double-blind, parallel-group, phase 3 trial. Lancet Neurol. 2021;20(12):1027-1037.
  4.  Schoser B, Kishnani PS, Bratkovic D, et al. 104-week efficacy and safety of cipglucosidase alfa plus miglustat in adults with late-onset Pompe disease: a phase III open-label extension study (ATB200-07). J Neurol. 2024;271(5):2810-2823 & supplemental material.
  5.  Data on file, Amicus Therapeutics, Inc.
  6.  Burton BK, Kronn DF, Hwu WL, et al; Pompe Disease Newborn Screening Working Group. The initial evaluation of patients after positive newborn screening: recommended algorithms leading to a confirmed diagnosis of Pompe disease. Pediatrics. 2017;140(suppl 1):S14-S23.
  7.  An Y, Young SP, Kishnani PS, Millington DS, Amalfitano A, Corz D, Chen Y-T. Glucose tetrasaccharide as a biomarker for monitoring the therapeutic response to enzyme replacement therapy for Pompe disease. Mol Genet Metab. 2005;85(4):247-254.