- ICH GCP
- US Clinical Trials Registry
- Klinisk utprøving NCT07778836
A Study to Evaluate the Efficacy and Safety of Subcutaneous Nomlabofusp in Subjects With Friedreich's Ataxia (FORWARD-FA)
A Phase 3 Randomized, Double-Blind, Placebo-Controlled, Multicenter Study to Evaluate the Efficacy and Safety of Subcutaneous Nomlabofusp in Subjects With Friedreich's Ataxia
Studieoversikt
Status
Forhold
Intervensjon / Behandling
Detaljert beskrivelse
This is a global, randomized, multi-center, placebo-controlled study to investigate the efficacy and safety of nomlabofusp in subjects with Friedreich's ataxia
The main objectives of the study are to evaluate:
- The efficacy of subcutaneous (SC) administration of nomlabofusp compared to placebo at Week 72; 1) on the Upright Stability Score (USS) Subscale E of the Modified Friedreich's Ataxia Ratings Scale (mFARS), or 2) mFARS total score (Region specific objective)
- The efficacy of SC administration of nomlabofusp compared to placebo at Week 72 on the Clinical Global Impression of Severity (CGI-S) score
- The safety and tolerability of SC administration of nomlabofusp
Studietype
Registrering (Antatt)
Fase
- Fase 3
Kontakter og plasseringer
Studiekontakt
- Navn: Larimar Therapeutics, Inc.
- Telefonnummer: 844-511-9056
- E-post: medicalinfo@larimartx.com
Studiesteder
-
-
Illinois
-
Chicago, Illinois, Forente stater, 60611
- Rekruttering
- Northwestern Medical Group, Department of Neurology
-
Ta kontakt med:
- Rizwan S Akhtar, MD
-
Ta kontakt med:
- Cynthia Poon, PhD
- Telefonnummer: (312) 503-8216
- E-post: cynthia.poon@northwestern.edu
-
-
New Jersey
-
Eatontown, New Jersey, Forente stater, 07724
- Rekruttering
- Clinilabs
-
Ta kontakt med:
- Magdy Shenouda, MD
- Telefonnummer: (646) 215-6400
- E-post: mshenouda@clinilabs.com
-
-
Texas
-
Dallas, Texas, Forente stater, 75390
- Rekruttering
- University of Texas Southwestern Medical Center
-
Ta kontakt med:
- Vikram Shakkottai, MD, PhD
-
Ta kontakt med:
- Sharon Primeaux
- Telefonnummer: (214) 645-0563
- E-post: Sharon.Primeaux@UTSouthwestern.edu
-
-
Deltakelseskriterier
Kvalifikasjonskriterier
Alder som er kvalifisert for studier
- Barn
- Voksen
Tar imot friske frivillige
Beskrivelse
Key Inclusion Criteria:
Subjects who meet all of the following criteria are potentially eligible for study participation:
- Subject must provide genetically confirmed FRDA diagnosis report and is homozygous for GAA repeat expansions documented on the genetic diagnostic report, with repeat sizing (if available).
- Subject must complete 1 trial at Screening and 1 trial at Day -1 of the T25-FW test, using their customary assistive device (e.g., cane, 2 canes/crutches [Canadian crutches], wheeled walker/rollator or canine assistance) if needed. Each trial must be completed within 3 minutes.
Subject must have the following at Screening and Day -1 per the following upright stability items from Module E of the mFARS:
- Item #E1, Sitting Posture - no more than a maximum score of 2
- Item #E2A, Stance Feet Apart - no more than a maximum score of 2 (average of 3 attempts)
- Subject must have an mFARS score ≥ 20 and < 60 at Screening and Day -1.
- Subject must have a Functional Staging for Ataxia score of 4 or less at Screening.
- Subject demonstrates sufficient dexterity and visual acuity to prepare and self-administer SC injections of study drug daily (QD) or has an identified caregiver who will be trained and committed to prepare and administer the injections.
- Subject has a Screening HbA1c ≤ 7.0%.
- If the subject is taking permitted concomitant medication(s), subject must have been on a stable dose and frequency of medication(s) over the past 28 days prior to initiation of Screening. Subjects taking niacin and resveratrol must have been on a stable dose and frequency for 90 days prior to initiation of Screening and subjects taking omaveloxolone must have been on a stable dose and frequency for 1 years prior to initiation of Screening.
Key Exclusion Criteria
Subjects are excluded from the study if any of the following exclusion criteria are met:
- Subject who is confirmed as compound heterozygous (GAA repeat expansion on only 1 allele) for FRDA.
- Subject previously participated in a clinical trial involving nomlabofusp. Participation is defined as the subject having signed the informed consent for the study and received at least 1 dose of study drug (nomlabofusp or placebo).
- The subject has any condition, disease, or situation that could confound the results of the study or put the subject at undue risk, making participation inadvisable in the opinion of the PI.
- Women of childbearing potential who are pregnant (have a positive pregnancy test at Screening or Day -1), lactating, or planning to attempt to become pregnant during this study or within 90 days after the last dose of study drug (this includes male subjects with partners of childbearing potential who are attempting to become pregnant).
- Subject used any investigational drug or device within 90 days prior to the initiation of Screening.
- Subject previously received a gene therapy (investigational or approved) at any time in the past.
- Subject requires use of amiodarone.
- Subject used erythropoietin, etravirine, or gamma interferon within 90 days prior to the initiation of Screening.
- Subject's use of biotin supplementation exceeds 30 μg/day, either as part of a multivitamin or as a standalone supplement, within 7 days prior to the first dose of study drug. Biotin supplementation ≤ 30 μg/day is permitted if taken at a stable dose and frequency for at least 28 days prior to the initiation of Screening and there is a commitment from the subject to maintain the biotin dose throughout the study (due to interference with assays).
- Subject receives medication that requires SC injection in the abdomen or thigh.
- Subject has a Screening ECHO left ventricular ejection fraction < 45%.
Subject has a QTcF on an ECG as specified below:
- For subjects ≥ 12 and < 18 years of age, a male or female subject with a QTcF > 460 ms.
- For subjects ≥ 18 years of age, a male subject with a QTcF > 450 ms or a female subject has a QTcF > 470 ms.
- Subject has suicidal ideation as determined by a "yes" to item #2 on the C-SSRS at Screening (within the last 28 days) or at Day -1.
Studieplan
Hvordan er studiet utformet?
Designdetaljer
- Primært formål: Behandling
- Tildeling: Randomisert
- Intervensjonsmodell: Parallell tildeling
- Masking: Firemannsrom
Våpen og intervensjoner
Deltakergruppe / Arm |
Intervensjon / Behandling |
|---|---|
|
Eksperimentell: Nomlabofusp
Daily subcutaneous injection of nomlabofusp for 72 weeks
|
Nomlabofusp er et rekombinant fusjonsprotein gitt i en steril, konserveringsmiddelfri bufret løsning for subkutan injeksjon beregnet på å levere humant frataxin, proteinet som mangler i Friedreichs ataksi.
Andre navn:
|
|
Placebo komparator: Placebo
Daily subcutaneous injection of placebo for 72 weeks
|
Placebo er en steril, konserveringsmiddelfri, klar væske for subkutan injeksjon.
Andre navn:
|
Hva måler studien?
Primære resultatmål
Resultatmål |
Tiltaksbeskrivelse |
Tidsramme |
|---|---|---|
|
Change from baseline in Upright Stability Score (USS) subscale E of modified Friedreich's Ataxia Rating Scale (mFARS) at Week 72 (Region-specific)
Tidsramme: Baseline, Weeks 12, 24, 36, 48, 72
|
Number - The USS has a score range from 0 to 36.
Lower scores mean less impairment and higher scores mean greater neurological impairment.
|
Baseline, Weeks 12, 24, 36, 48, 72
|
|
Change from baseline in the total modified Friedreich's Ataxia Rating Scale (mFARS) at Week 72 (Region-specific)
Tidsramme: Baseline, Weeks 12, 24, 36, 48, 72
|
Number - The mFARS has a total score range from 0 to 93.
Lower scores mean less impairment and higher scores mean greater neurological impairment.
|
Baseline, Weeks 12, 24, 36, 48, 72
|
Sekundære resultatmål
Resultatmål |
Tiltaksbeskrivelse |
Tidsramme |
|---|---|---|
|
Change from baseline in the Clinical Global Impression-Severity (CGI-S) score at Week 72
Tidsramme: Baseline, Weeks 12, 24, 36, 48, 72
|
Number - The CGI-S is a 7-point scale.
Lower scores mean less impairment and higher scores mean greater neurological impairment.
|
Baseline, Weeks 12, 24, 36, 48, 72
|
Samarbeidspartnere og etterforskere
Sponsor
Publikasjoner og nyttige lenker
Generelle publikasjoner
- Koeppen AH. Friedreich's ataxia: pathology, pathogenesis, and molecular genetics. J Neurol Sci. 2011 Apr 15;303(1-2):1-12. doi: 10.1016/j.jns.2011.01.010.
- Delatycki MB, Corben LA. Clinical features of Friedreich ataxia. J Child Neurol. 2012 Sep;27(9):1133-7. doi: 10.1177/0883073812448230. Epub 2012 Jun 29.
- Goodkin DE, Hertsgaard D, Seminary J. Upper extremity function in multiple sclerosis: improving assessment sensitivity with box-and-block and nine-hole peg tests. Arch Phys Med Rehabil. 1988 Oct;69(10):850-4.
- Rudick R, Antel J, Confavreux C, Cutter G, Ellison G, Fischer J, Lublin F, Miller A, Petkau J, Rao S, Reingold S, Syndulko K, Thompson A, Wallenberg J, Weinshenker B, Willoughby E. Clinical outcomes assessment in multiple sclerosis. Ann Neurol. 1996 Sep;40(3):469-79. doi: 10.1002/ana.410400321.
- Deutsch EC, Santani AB, Perlman SL, Farmer JM, Stolle CA, Marusich MF, Lynch DR. A rapid, noninvasive immunoassay for frataxin: utility in assessment of Friedreich ataxia. Mol Genet Metab. 2010 Oct-Nov;101(2-3):238-45. doi: 10.1016/j.ymgme.2010.07.001. Epub 2010 Jul 8.
- Indelicato E, Nachbauer W, Eigentler A, Amprosi M, Matteucci Gothe R, Giunti P, Mariotti C, Arpa J, Durr A, Klopstock T, Schols L, Giordano I, Burk K, Pandolfo M, Didszdun C, Schulz JB, Boesch S; EFACTS (European Friedreich's Ataxia Consortium for Translational Studies). Onset features and time to diagnosis in Friedreich's Ataxia. Orphanet J Rare Dis. 2020 Aug 3;15(1):198. doi: 10.1186/s13023-020-01475-9.
- Lawerman TF, Brandsma R, Burger H, Burgerhof JGM, Sival DA; the Childhood Ataxia and Cerebellar Group of the European Pediatric Neurology Society. Age-related reference values for the pediatric Scale for Assessment and Rating of Ataxia: a multicentre study. Dev Med Child Neurol. 2017 Oct;59(10):1077-1082. doi: 10.1111/dmcn.13507. Epub 2017 Aug 17.
- Lazaropoulos M, Dong Y, Clark E, Greeley NR, Seyer LA, Brigatti KW, Christie C, Perlman SL, Wilmot GR, Gomez CM, Mathews KD, Yoon G, Zesiewicz T, Hoyle C, Subramony SH, Brocht AF, Farmer JM, Wilson RB, Deutsch EC, Lynch DR. Frataxin levels in peripheral tissue in Friedreich ataxia. Ann Clin Transl Neurol. 2015 Aug;2(8):831-42. doi: 10.1002/acn3.225. Epub 2015 Jul 1.
- Carpenter JR, Roger JH, Kenward MG. Analysis of longitudinal trials with protocol deviation: a framework for relevant, accessible assumptions, and inference via multiple imputation. J Biopharm Stat. 2013;23(6):1352-71. doi: 10.1080/10543406.2013.834911.
- Fisk JD, Ritvo PG, Ross L, Haase DA, Marrie TJ, Schlech WF. Measuring the functional impact of fatigue: initial validation of the fatigue impact scale. Clin Infect Dis. 1994 Jan;18 Suppl 1:S79-83. doi: 10.1093/clinids/18.supplement_1.s79.
- Bauer P, Kohne K. Evaluation of experiments with adaptive interim analyses. Biometrics. 1994 Dec;50(4):1029-41.
- Little R, Yau L. Intent-to-treat analysis for longitudinal studies with drop-outs. Biometrics. 1996 Dec;52(4):1324-33.
- Gladman D, Nash P, Goto H, Birt JA, Lin CY, Orbai AM, Kvien TK. Fatigue numeric rating scale validity, discrimination and responder definition in patients with psoriatic arthritis. RMD Open. 2020 Jan;6(1):e000928. doi: 10.1136/rmdopen-2019-000928.
- Pandolfo M. Friedreich ataxia. Arch Neurol. 2008 Oct;65(10):1296-303. doi: 10.1001/archneur.65.10.1296.
- Fahey MC, Corben L, Collins V, Churchyard AJ, Delatycki MB. How is disease progress in Friedreich's ataxia best measured? A study of four rating scales. J Neurol Neurosurg Psychiatry. 2007 Apr;78(4):411-3. doi: 10.1136/jnnp.2006.096008. Epub 2006 Oct 20.
- Liu, C. (1993). "Bartlett's Decomposition of the Posterior Distribution of the Covariance for Normal Monotone Ignorable Missing Data." Journal of Multivariate Analysis 46:198-206.
- Dmitrienko, A., Yuan, Y. (2017). Interim data monitoring. Analysis of Clinical Trials Using SAS: A Practical Guide (Second Edition). Dmitrienko, A., Koch, G.G. (editors). SAS Press: Cary, NC.
- Lawrence, J. and Hung, H.M.J. (2003), Estimation and Confidence Intervals after Adjusting the Maximum Information. Biom. J., 45: 143-152. https://doi.org/10.1002/bimj.200390001
- Li KH. Imputation using Markov chains. J Stat Comput Simul 1988;30:57-79.
- Lynch DR, Chin MP, Delatycki MB, Subramony SH, Corti M, Hoyle JC, Boesch S, Nachbauer W, Mariotti C, Mathews KD, Giunti P, Wilmot G, Zesiewicz T, Perlman S, Goldsberry A, O'Grady M, Meyer CJ. Safety and Efficacy of Omaveloxolone in Friedreich Ataxia (MOXIe Study). Ann Neurol. 2021 Feb;89(2):212-225. doi: 10.1002/ana.25934. Epub 2020 Nov 5.
- Meyer RD, Ratitch B, Wolbers M, Marchenko O, Quan H, Li D, Fletcher C, Li X, Wright D, Shentu Y, Englert S, Shen W, Dey J, Liu T, Zhou M, Bohidar N, Zhao PL, Hale M. Statistical Issues and Recommendations for Clinical Trials Conducted During the COVID-19 Pandemic. Stat Biopharm Res. 2020 Jul 6;12(4):399-411. doi: 10.1080/19466315.2020.1779122.
- MSCfCP Guidelines. Fatigue and multiple sclerosis: evidence-based management strategies for fatigue in multiple sclerosis. 1998
- nQuery (2017). Sample Size and Power Calculation. "Statsols" (Statistical Solutions Ltd), Cork, Ireland.
- Qu Y, Dai B. Return-to-baseline multiple imputation for missing values in clinical trials. Pharm Stat. 2022 May;21(3):641-653. doi: 10.1002/pst.2191. Epub 2022 Jan 5.
- Ratitch, B., O'Kelly, M. (2011). Implementation of pattern-mixture models using standard SAS/STAT procedures
- Rubin, D.B. (1987). Multiple Imputation for Nonresponse in Surveys. New York: John Wiley and Sons. DOI:10.1002/9780470316696
Studierekorddatoer
Studer hoveddatoer
Studiestart (Antatt)
Primær fullføring (Antatt)
Studiet fullført (Antatt)
Datoer for studieregistrering
Først innsendt
Først innsendt som oppfylte QC-kriteriene
Først lagt ut (Faktiske)
Oppdateringer av studieposter
Sist oppdatering lagt ut (Faktiske)
Siste oppdatering sendt inn som oppfylte QC-kriteriene
Sist bekreftet
Mer informasjon
Begreper knyttet til denne studien
Ytterligere relevante MeSH-vilkår
- Hjernesykdommer
- Sykdommer i sentralnervesystemet
- Sykdommer i nervesystemet
- Genetiske sykdommer, medfødte
- Metabolske sykdommer
- Nevrodegenerative sykdommer
- Heredodegenerative lidelser, nervesystemet
- Ryggmargssykdommer
- Mitokondrielle sykdommer
- Cerebellare sykdommer
- Spinocerebellare degenerasjoner
- Medfødte, arvelige og neonatale sykdommer og abnormiteter
- Ernæringsmessige og metabolske sykdommer
- Friedreich Ataxia
Andre studie-ID-numre
- CLIN-1601-301
- 2025 (U.S. NIH-stipend/kontrakt: Faculty of Social Sciences Scientific Grant at the University of Gdańsk)
- 2025-521628-31-00 (Ctis)
Plan for individuelle deltakerdata (IPD)
Planlegger du å dele individuelle deltakerdata (IPD)?
Legemiddel- og utstyrsinformasjon, studiedokumenter
Studerer et amerikansk FDA-regulert medikamentprodukt
Studerer et amerikansk FDA-regulert enhetsprodukt
Denne informasjonen ble hentet direkte fra nettstedet clinicaltrials.gov uten noen endringer. Hvis du har noen forespørsler om å endre, fjerne eller oppdatere studiedetaljene dine, vennligst kontakt register@clinicaltrials.gov. Så snart en endring er implementert på clinicaltrials.gov, vil denne også bli oppdatert automatisk på nettstedet vårt. .