- ICH GCP
- Amerikanska kliniska prövningsregistret
- Klinisk prövning NCT06953583
En studie för att lära sig mer om effekterna och långsiktiga säkerheten i BIIB141 (Omaveloxolone) hos deltagare med Friedreichs ataxi i åldern 2 till 15 år gammal (BRAVE)
En fas 3, 2-delad, randomiserad, dubbelblind, placebokontrollerad studie (del 1) och öppen etikettförlängning (del 2) för att utvärdera effektiviteten, säkerheten, farmakokinetiken och farmakodynamiken för omaveloxolon (BIIB141) hos deltagare med Friedreichs ataxi Aged 2 till <16 år
I denna studie kommer forskare att lära sig mer om effekterna och säkerheten i BIIB141, även känd som Omaveloxolone eller Skyclarys®. Detta läkemedel har godkänts eller gjorts tillgängligt för läkare att förskriva för personer med Friedreichs ataxi (FA) som är minst 16 år gamla. Men det är ännu inte tillgängligt för barn och tonåringar med FA som är yngre än 16 år gamla. Huvudmålet med denna studie är att lära sig hur BIIB141 fungerar i kroppen och om dess säkerhet hos barn och tonåringar som är 2 till 15 år gamla.
De viktigaste frågorna som forskare vill svara i denna studie är:
- Hur påverkar BIIB141 deltagarnas FA -symtom balans och stabilitet?
- Hur många deltagare har medicinska problem under studien?
- Finns det några förändringar i deltagarnas allmänna hälsa under studien?
- Finns det några förändringar i deltagarnas hjärthälsa?
- Finns det några förändringar i hur deltagarna rör sig genom puberteten? Puberteten är tiden i någons liv när deras kropp förändras från ett barn till en vuxen.
Forskare kommer också att lära sig mer om:
- Hur kroppen bearbetar BIIB141 hos barn och tonåringar
Denna studie kommer att göras enligt följande:
- Deltagarna kommer att screenas för att kontrollera om de kan gå med i studien. Screeningperioden kommer att vara upp till 28 dagar, varefter deltagarna kommer att kolla in sitt studieforskningscenter.
- Det finns två delar i denna studie. Under del 1 kommer deltagarna att ta antingen BIIB141 eller en placebo en gång om dagen.
- I del 1 kommer deltagarna att ta BIIB141 eller placebo i ett studieforskningscenter på dag 1 och sedan vid personbesök vid vecka 4, vecka 12, vecka 26 och vecka 52. På alla andra dagar kommer de att ta BIIB141 eller placebo hemma. Del 1 varar upp till 52 veckor.
- Under del 2 kommer deltagarna från del 1 antingen att fortsätta ta BIIB141 eller starta det om de tog placebo. Del 2 kommer att pågå i upp till 104 veckor.
- I del 1 kommer deltagarna att ha upp till tio besök i sitt studieforskningscenter och ett telefonsamtal vid vecka 2. I del 2 kommer deltagarna att ha besök vid veckor 4, 8,12, 26 och var 26 veckor efter det tills de lämnar studien och ett telefonsamtal vid vecka 2. Det kommer att finnas ett slutligt telefonsamtal för att kontrollera deltagarna 31 dagar efter deras sista dos.
- Varje deltagare kommer att vara i studien i upp till cirka 3 år
Studieöversikt
Status
Betingelser
Intervention / Behandling
Detaljerad beskrivning
Studietyp
Inskrivning (Beräknad)
Fas
- Fas 3
Kontakter och platser
Studiekontakt
- Namn: US Biogen Clinical Trial Center
- Telefonnummer: 866-633-4636
- E-post: clinicaltrials@biogen.com
Studera Kontakt Backup
- Namn: Patient Navigator
- Telefonnummer: 57078 1-877-223-3576
- E-post: biogenBRAVE_patientnavigator@thermofisher.com
Studieorter
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New South Wales
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Randwick, New South Wales, Australien, 2031
- Har inte rekryterat ännu
- Sydney Children's Hospital
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Victoria
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Parkville, Victoria, Australien, 3052
- Rekrytering
- Murdoch Childrens Research Institute (MCRI)
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Federal District
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Brasília, Federal District, Brasilien, 70200-730
- Rekrytering
- L2 Ip - Instituto de Pesquisas Clinicas Ltda - ME
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Kontakt:
- Telefonnummer: +55 61 3445-4300
- E-post: eduardo.vasconcellos@l2ip.com.br
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Huvudutredare:
- Ingrid Faber Faber de Vasconcellos
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São Paulo
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Campinas, São Paulo, Brasilien, 13083-970
- Har inte rekryterat ännu
- University of Campinas (UNICAMP) School of Medical Sciences
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Kontakt:
- Telefonnummer: +55 19 3521-8922
- E-post: mcfrancajr@uol.com.br
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Huvudutredare:
- Marcondes França
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São Paulo, São Paulo, Brasilien, 04024-002
- Rekrytering
- PSEG Centro de Pesquisa Clinica
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Huvudutredare:
- Paulo Victor Sgobbi de Souza
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Kontakt:
- Telefonnummer: +5511972375577
- E-post: pvsgobbi@gmail.com
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Copenhagen, Danmark, 2100
- Har inte rekryterat ännu
- Rigshospitalet - Juliane Marie Centret (JMC) Copenhagen
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Kontakt:
- Telefonnummer: +45 35-45-50-93
- E-post: alfred.peter.born@regionh.dk
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Huvudutredare:
- Alfred Peter Born
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Paris, Frankrike, 75012
- Rekrytering
- AP-HP - Hôpital Armand Trousseau
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Huvudutredare:
- Florence Renaldo
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Kontakt:
- Telefonnummer: +33 1 85 34 00 29
- E-post: Florence.renaldo@aphp.fr
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Hérault
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Montpellier, Hérault, Frankrike, 34090
- Rekrytering
- CHU de Montpellier- Hôpital Gui De Chauliac
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Kontakt:
- Telefonnummer: 33 04 67 33 01 82
- E-post: a-roubertie@chu-montpellier.fr
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Huvudutredare:
- Agathe Roubertie
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California
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Los Angeles, California, Förenta staterna, 90095
- Har inte rekryterat ännu
- UCLA Neurology Outpatient Clinic at Westwood
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Kontakt:
- Telefonnummer: 310-794-1195
- E-post: sperlman@mednet.ucla.edu
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Huvudutredare:
- Susan Perlman
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Florida
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Gainesville, Florida, Förenta staterna, 32610-3010
- Rekrytering
- Norman Fixel Institute for Neurological Diseases UF Health
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Kontakt:
- Telefonnummer: 352-733-3032
- E-post: s.subramony@neurology.ufl.edu
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Huvudutredare:
- Sankarsubramoney Subramony
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Tampa, Florida, Förenta staterna, 33612
- Rekrytering
- USF Health Morsani College of Medicine Department of Neurology
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Huvudutredare:
- Theresa Zesiewicz
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Kontakt:
- Telefonnummer: 813-974-5909
- E-post: tzesiewi@hsc.usf.edu
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Pennsylvania
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Philadelphia, Pennsylvania, Förenta staterna, 19104
- Rekrytering
- Children's Hospital of Philadelphia - Buerger Center for Advanced Pediatric Care - PIN
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Kontakt:
- Telefonnummer: 215-590-2242
- E-post: lynchd@pennmedicine.upenn.edu
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Huvudutredare:
- David Robinson Lynch
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Tennessee
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Memphis, Tennessee, Förenta staterna, 38105-3678
- Rekrytering
- St. Jude Children's Research Hospital - PIN
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Huvudutredare:
- Richard Finkel
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Kontakt:
- Telefonnummer: 407-650-7250
- E-post: richard.finkel@stjude.org
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Virginia
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Norfolk, Virginia, Förenta staterna, 23507-1910
- Rekrytering
- CHKD's Health Center - South Campus - PIN
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Kontakt:
- Telefonnummer: 757-668-6981
- E-post: proud.research@chkd.org
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Huvudutredare:
- Crystal Proud
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Washington
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Seattle, Washington, Förenta staterna, 98105-3901
- Rekrytering
- Seattle Children's Hospital
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Kontakt:
- Telefonnummer: 206-987-2078
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Huvudutredare:
- Alicia Henriquez
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National Capital Territory of Delhi
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New Delhi, National Capital Territory of Delhi, Indien, 110029
- Indragen
- All India Institute of Medical Sciences (AIIMS) - New Delhi
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-
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Dublin, Irland, D01 XD99
- Rekrytering
- CHI at Temple Street
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Kontakt:
- Telefonnummer: 2 (353) 187-8472
- E-post: declan.orourke@cuh.ie
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Huvudutredare:
- Declan O'Rourke
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Milan, Italien, 20133
- Rekrytering
- Fondazione IRCCS Istituto Neurologico Carlo Besta
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Kontakt:
- Telefonnummer: +39 022394 2210
- E-post: isabella.moroni@istituto-besta.it
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Huvudutredare:
- Isabella Moroni
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Lazio
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Rome, Lazio, Italien, 165
- Har inte rekryterat ännu
- Ospedale Pediatrico Bambino Gesu Irccs
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Huvudutredare:
- Gessica Vasco
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Kontakt:
- Telefonnummer: +39 066859 3461
- E-post: gessica.vasco@opbg.net
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Veneto
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Conegliano, Veneto, Italien, 31015
- Har inte rekryterat ännu
- IRCCS Eugenio Medea - Polo. Scientifico Veneto
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Huvudutredare:
- Gabriella Paparella
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Kontakt:
- Telefonnummer: +39 3383065324
- E-post: gabriella.paparella@lanostrafamiglia.it
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Quebec
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Montreal, Quebec, Kanada, H3H 2R9
- Rekrytering
- McGill University
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Kontakt:
- Telefonnummer: 514-412-4466
- E-post: maryam.oskoui@mcgill.ca
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Huvudutredare:
- Maryam Oskoui
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Québec, Quebec, Kanada, G1V 4G2
- Rekrytering
- CHU de Quebec -Universite Laval
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Kontakt:
- Telefonnummer: 71801 418-525-4444
- E-post: nicolas.chrestian.med@ssss.gouv.qc.ca
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Huvudutredare:
- Nicolas Chrestian
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Nijmegen, Nederländerna, 6525 GA
- Rekrytering
- Radboud Universitair Medisch Centrum
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Kontakt:
- Telefonnummer: 31 243614415
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Huvudutredare:
- Nienke van Os
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Ar Riya
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Riyadh, Ar Riya, Saudiarabien, 12875
- Indragen
- King Faisal Specialist Hospital & Research Centre
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Madrid, Spanien, 28046
- Rekrytering
- Hospital Universitario La Paz - PPDS
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Kontakt:
- Telefonnummer: +34 91 7277388
- E-post: yambee@hotmail.com
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Huvudutredare:
- Maria del Mar Garcia Romero
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Barcelona
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Espluges de Llobregat, Barcelona, Spanien, 8950
- Rekrytering
- Hospital Sant Joan de Deu - PIN
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Kontakt:
- Telefonnummer: 71465 +34 93 253 21 00
- E-post: alejandra.darling@sjd.es
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Huvudutredare:
- Alejandra Darling
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Lincolnshire
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London, Lincolnshire, Storbritannien, NW1 2BU
- Rekrytering
- University College Hospital - PPDS
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Kontakt:
- Telefonnummer: +44 773046 1357
- E-post: shpresa.pula1@nhs.net
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Huvudutredare:
- Shpresa Pula
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Oxfordshire
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Oxford, Oxfordshire, Storbritannien, OX3 9DU
- Rekrytering
- John Radcliffe Hospital
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Huvudutredare:
- Andrea Németh
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Kontakt:
- Telefonnummer: 44 1865 231556
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South Yorkshire
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Sheffield, South Yorkshire, Storbritannien, S10 5DD
- Rekrytering
- Sheffield Children's Hospital - PPDS
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Kontakt:
- Telefonnummer: 0114 226 0675
- E-post: santosh.mordekar@nhs.net
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Huvudutredare:
- Santosh Ravindra Mordekar
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Istanbul, Turkiet (Türkiye), 34093
- Indragen
- Istanbul Universitesi Istanbul Tip Fakultesi Hastanesi
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-
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Giessen, Tyskland, 35392
- Rekrytering
- UKGM - Universitätsklinikum Giessen und Marburg GmbH - Standort Gießen
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Huvudutredare:
- Andreas Hahn
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Kontakt:
- Telefonnummer: +49 641 985 43543
- E-post: andreas.hahn@paediat.med.uni-giessen.de
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Hamburg, Tyskland, 20246
- Rekrytering
- Universitatsklinikum Hamburg Eppendorf
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Kontakt:
- Telefonnummer: +49 40 7410 56126
- E-post: d.weiss@uke.de
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Huvudutredare:
- Deike Weiss
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North Rhine-Westphalia
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Aachen, North Rhine-Westphalia, Tyskland, 52074
- Rekrytering
- Universitätsklinikum Aachen
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Huvudutredare:
- Kathrin Reetz
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Kontakt:
- Telefonnummer: 492418089601
- E-post: kreetz@ukaachen.de
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-
-
-
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Innsbruck, Österrike, 6020
- Rekrytering
- Universitätsklinikum Innsbruck
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Kontakt:
- Telefonnummer: +43 5125042 3850
- E-post: sylvia.boesch@i-med.ac.at
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Huvudutredare:
- Sylvia M Boesch
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Deltagandekriterier
Urvalskriterier
Åldrar som är berättigade till studier
- Barn
Tar emot friska volontärer
Beskrivning
DEL 1 RCT: Key Inclusion Criteria:
- Diagnostiserad med genetiskt bekräftade Friedreichs ataxi (FA), dvs homozygot för guanin-adenin-adenin (GAA) upprepade expansion i intron-1 av frataxingenen, eller GAA-upprepning av expansion i 1 allel och med punktmutationer eller deletioner, eller andra icke-GAA-expansionsmutationer i den andra allelen.
- Symtomatisk för FA som rapporterats av deltagaren och/eller föräldern/vårdgivaren a. Barn 7 till <16 år måste också ha en Upright Stability Score (USS) poäng på 10 till ≤ 34 vid baslinjen
Del 1 RCT: Knappsuteslutningskriterier:
- Glykosylerat hemoglobin A1C (HbA1c)> 11%
- B-typ natriuretisk peptid (BNP)> 200 picogram per milliliter (PG/ml) vid screening
- Ejektionsfraktion (EF) <40% [baserat på ekokardiogram (ECHO) som utförs vid screeningbesök]
- Kliniskt signifikant hjärtsjukdom utom mild till måttlig kardiomyopati
Del 2 OLE: Behörighetskriterier:
- Deltagarna har slutfört del 1 RCT i studien och inga avbrottskriterier har uppfyllts
Säkerhets- och tolerabilitetsdata från del 1 RCT stöder fortsättningen i utredarens bedömning
- Om alaninaminotransferas (ALT), aspartataminotransferas (AST) och/eller totalt bilirubin (TBL) är> 2 × övre gräns för normal (Uln) vid föregående besöksbedömning, del 2 dag 1 bör försenas tills ALT och AST är <1,5 × uln och TBL är <2 × Uln
- Om BNP är> 200 pg/ml vid föregående besöksbedömning, bör del 2 dag 1 försenas tills BNP är <200 pg/ml
- Om någon annan kliniskt signifikant laboratorieavvikelser finns baserat på de tidigare besöksbedömningarna, bör del 2 dag 1 försenas tills avvikelserna har lösts
- I händelse av samtidig sjukdom eller annan förändring i deltagarens hälsostatus kan ytterligare del 1 -screeningbedömningar upprepas före inledningen av del 2, baserat på utredarens bedömning i samråd med Medical Monitor
Obs: Andra protokolldefinierade inkluderings-/uteslutningskriterier kan gälla.
Studieplan
Hur är studien utformad?
Designdetaljer
- Primärt syfte: Behandling
- Tilldelning: Randomiserad
- Interventionsmodell: Parallellt uppdrag
- Maskning: Fyrdubbla
Vapen och interventioner
Deltagargrupp / Arm |
Intervention / Behandling |
|---|---|
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Experimentell: Part 1: Omaveloxolone
Participants will receive a single oral dose of omaveloxolone once a day (QD) for up to 52 weeks in Part 1 of the study.
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Administreras enligt beskrivningen i behandlingsarmen.
Andra namn:
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Placebo-jämförare: Part 1: Placebo
Participants will receive placebo, orally, QD for up to 52 weeks in Part 1 of the study.
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Administreras enligt beskrivningen i behandlingsarmen.
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Experimentell: Part 2A Continued Efficacy Evaluation: Omaveloxolone
Participants will receive a single oral dose of omaveloxolone, QD for up to 104 weeks in Part 2A of the study.
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Administreras enligt beskrivningen i behandlingsarmen.
Andra namn:
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Experimentell: Part 2B Safety: Omaveloxolone
Participants will receive a single oral dose of open-label omaveloxolone, QD for up to 104 weeks in Part 2B of the study.
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Administreras enligt beskrivningen i behandlingsarmen.
Andra namn:
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Vad mäter studien?
Primära resultatmått
Resultatmått |
Åtgärdsbeskrivning |
Tidsram |
|---|---|---|
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Part 1: Change From Baseline in Upright Stability Score (USS) Subscale E of Modified Friedreich's Ataxia Rating Scale (mFARS) at Week 52
Tidsram: Baseline, Week 52
|
The mFARS is a validated and sensitive rating scale that was developed to quantitatively assess the severity of the neurologic features of FA in adults and adolescents.
Scores on the mFARS range from 0 to 93, with lower scores indicating better neurological function.
The subscales of the mFARS assessment and maximum score for each subscale are: bulbar function (Subscale A; 2 assessments of speech and cough; maximum score = 5), upper limb coordination (Subscale B; 5 assessments of coordination of movement and function in arms and hands with each limb scored individually; maximum score = 36), lower limb coordination (Subscale C; 2 assessments of coordination of movement and function of lower limbs with each limb scored individually; maximum score = 16), and upright stability (USS, Subscale E; 9 assessments of sitting posture, stance, tandem walk, and gait assessments; maximum score = 36).
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Baseline, Week 52
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Part 2A: Change From Baseline in USS Subscale E of mFARS at Week 52
Tidsram: Baseline (Week 52 of Part 1), Week 52
|
The mFARS is a validated and sensitive rating scale that was developed to quantitatively assess the severity of the neurologic features of FA in adults and adolescents.
Scores on the mFARS range from 0 to 93, with lower scores indicating better neurological function.
The subscales of the mFARS assessment and maximum score for each subscale are: bulbar function (Subscale A; 2 assessments of speech and cough; maximum score = 5), upper limb coordination (Subscale B; 5 assessments of coordination of movement and function in arms and hands with each limb scored individually; maximum score = 36), lower limb coordination (Subscale C; 2 assessments of coordination of movement and function of lower limbs with each limb scored individually; maximum score = 16), and upright stability (USS, Subscale E; 9 assessments of sitting posture, stance, tandem walk, and gait assessments; maximum score = 36).
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Baseline (Week 52 of Part 1), Week 52
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Part 2B: Number of Participants With Treatment-Emergent Adverse Event (TEAE) and Treatment-Emergent Serious Adverse Event (TESAE)
Tidsram: From the first dose of the study drug in Part 2B up to the end of follow-up period in Part 2B (up to Week 104)
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From the first dose of the study drug in Part 2B up to the end of follow-up period in Part 2B (up to Week 104)
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Part 2B: Number of Participants With Change From Baseline in Cardiac Function Assessed by Echocardiogram (ECHO) at Weeks 52 and Week 104
Tidsram: Baseline (Week 52 of Part 1), Weeks 52 and 104
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Baseline (Week 52 of Part 1), Weeks 52 and 104
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Part 2B: Change From Baseline in Height at Weeks 52 and Week 104
Tidsram: Baseline (Week 52 of Part 1), Weeks 52 and 104
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Baseline (Week 52 of Part 1), Weeks 52 and 104
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Part 2B: Change From Baseline in Weight at Weeks 52 and Week 104
Tidsram: Baseline (Week 52 of Part 1), Weeks 52 and 104
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Baseline (Week 52 of Part 1), Weeks 52 and 104
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Part 2B: Change From Baseline in Body Mass Index (BMI) at Weeks 52 and Week 104
Tidsram: Baseline (Week 52 of Part 1), Weeks 52 and 104
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Baseline (Week 52 of Part 1), Weeks 52 and 104
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Part 2B: Change From Baseline in Columbia Suicide Severity Rating Scale (C-SSRS) at Weeks 52 and Week 104
Tidsram: Baseline (Week 52 of Part 1), Weeks 52 and 104
|
The C-SSRS is a low-burden measure of the spectrum of suicidal ideation and behavior that was developed to assess severity and track suicidal events through any treatment of individuals ≥ 6 years of age.
The C-SSRS is a clinical interview providing a summary of both ideation and behavior that can be administered by the clinician during any evaluation or risk assessment to identify the level and type of suicidality present.
The assessment includes "yes" or "no" responses for 5 questions each, related to suicidal ideation (wish to be dead, non-specific active suicidal thoughts, active suicidal ideation with any methods, active suicidal ideation with some intent, active suicidal ideation with specific plan) and suicidal behavior (preparatory acts or behavior, aborted attempt, interrupted attempt, actual attempt, suicide).
Numeric ratings are provided for severity of ideation, from 1 to 5, with 5 being the most severe.
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Baseline (Week 52 of Part 1), Weeks 52 and 104
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Part 2B: Percentage of Participants at Each Tanner Stage at Weeks 52 and Week 104
Tidsram: Baseline (Week 52 of Part 1), Weeks 52 and 104
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Assessment of Tanner stages (a scale of physical development) will be performed by a medical doctor experienced with this assessment.
Tanner score ranges from Stage 1 (childhood) to Stage 5 (full physical maturity).
Information regarding Tanner staging will be collected at baseline for all participants and will be stopped once the participant reaches Tanner Stage 5 in all gender-appropriate scales.
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Baseline (Week 52 of Part 1), Weeks 52 and 104
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Part 2B: Number of Participants at Each Tanner Stage at Weeks 52 and Week 104
Tidsram: Baseline (Week 52 of Part 1), Weeks 52 and 104
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Assessment of Tanner stages (a scale of physical development) will be performed by a medical doctor experienced with this assessment.
Tanner score ranges from Stage 1 (childhood) to Stage 5 (full physical maturity).
Information regarding Tanner staging will be collected at baseline for all participants and will be stopped once the participant reaches Tanner Stage 5 in all gender-appropriate scales.
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Baseline (Week 52 of Part 1), Weeks 52 and 104
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Sekundära resultatmått
Resultatmått |
Åtgärdsbeskrivning |
Tidsram |
|---|---|---|
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Part 1: Change From Baseline in Friedreich's Ataxia-Health Index (FA-HI) at Week 52
Tidsram: Baseline, Week 52
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The FA-HI is a participant reported survey that assesses overall disease burden on a 100-point scale, with 0 representing no disease burden and 100 representing the maximum level of disease burden containing 113 symptoms questions representing 18 symptomatic subscales.
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Baseline, Week 52
|
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Part 1: Change From Baseline in Modified Friedreich's Ataxia Rating Scale (mFARS) at Week 52
Tidsram: Baseline, Week 52
|
The mFARS is a validated and sensitive rating scale that was developed to quantitatively assess the severity of the neurologic features of FA in adults and adolescents.
Scores on the mFARS range from 0 to 93, with lower scores indicating better neurological function.
The subscales of the mFARS assessment and maximum score for each subscale are: bulbar function (Subscale A; 2 assessments of speech and cough; maximum score = 5), upper limb coordination (Subscale B; 5 assessments of coordination of movement and function in arms and hands with each limb scored individually; maximum score = 36), lower limb coordination (Subscale C; 2 assessments of coordination of movement and function of lower limbs with each limb scored individually; maximum score = 16), and upright stability (USS, Subscale E; 9 assessments of sitting posture, stance, tandem walk, and gait assessments; maximum score = 36).
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Baseline, Week 52
|
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Part 1: Change From Baseline in Patient Global Impressions-Severity (PGI-S) at Week 52
Tidsram: Baseline, Week 52
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PGI-S will be conducted for participants 7 to < 16 years of age.
These are clinically meaningful outcome measures that are participant-relevant across all age groups and disease severities for this population.
PGI -S is a 1-item questionnaire where the response is recorded on a 4-point scale scored as: 1-normal, 2-mild, 3-moderate, or 4-severe.
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Baseline, Week 52
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Part 1: Change From Baseline in Clinical Global Impressions-Severity (CGI-S) at Week 52
Tidsram: Baseline, Week 52
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The CGI-S will be conducted for all enrolled participants, 2 to < 16 years of age.
The CGI-S rating evaluates the severity of individual symptoms and treatment response in participants with mental disorders.
The CGI-S is a 7-point scale that requires the clinician to rate the severity of the participant's illness at the time of assessment.
A rating of 1 is considered normal, or with the least severe symptoms, a rating of 7 is extremely ill, or the worst symptoms.
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Baseline, Week 52
|
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Part 1: Change From Baseline in Friedreich's Ataxia-Activities of Daily Living (FA-ADL)
Tidsram: Baseline, Week 52
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Participants will answer the 9 questions of the FA-ADL survey in an interview style conducted by any site staff.
The FA-ADL survey assesses 9 concepts: (1) speech; (2) swallowing; (3) cutting food and handling utensils; (4) dressing; (5) personal hygiene; (6) falling; (7) walking; (8) quality of sitting position; and (9) bladder function.
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Baseline, Week 52
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Part 1: Number of Participants With Treatment-Emergent Adverse Event (TEAE) and Treatment-Emergent Serious Adverse Event (TESAE)
Tidsram: From first dose of study drug up to end of follow up period in Part 1 (up to Week 52)
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From first dose of study drug up to end of follow up period in Part 1 (up to Week 52)
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|
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Part 1: Number of Participants With Change From Baseline in Cardiac Function Assessed by ECHO at Week 52
Tidsram: Baseline, Week 52
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Baseline, Week 52
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|
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Part 1: Change From Baseline in Height at Week 52
Tidsram: Baseline, Week 52
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Baseline, Week 52
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|
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Part 1: Change From Baseline in Weight at Week 52
Tidsram: Baseline, Week 52
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Baseline, Week 52
|
|
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Part 1: Change From Baseline in Body Mass Index (BMI) at Week 52
Tidsram: Baseline, Week 52
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Baseline, Week 52
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Part 1: Change From Baseline in Columbia Suicide Severity Rating Scale (C-SSRS) at Week 52
Tidsram: Baseline, Week 52
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The C-SSRS is a low-burden measure of the spectrum of suicidal ideation and behavior that was developed to assess severity and track suicidal events through any treatment of individuals ≥ 6 years of age.
The C-SSRS is a clinical interview providing a summary of both ideation and behavior that can be administered by the clinician during any evaluation or risk assessment to identify the level and type of suicidality present.
The assessment includes "yes" or "no" responses for 5 questions each, related to suicidal ideation (wish to be dead, non-specific active suicidal thoughts, active suicidal ideation with any methods, active suicidal ideation with some intent, active suicidal ideation with specific plan) and suicidal behavior (preparatory acts or behavior, aborted attempt, interrupted attempt, actual attempt, suicide).
Numeric ratings are provided for severity of ideation, from 1 to 5, with 5 being the most severe.
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Baseline, Week 52
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Part 1: Percentage of Participants at Each Tanner Stage at Week 52
Tidsram: Baseline, Week 52
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Assessment of Tanner stages (a scale of physical development) will be performed by a medical doctor experienced with this assessment.
Tanner score ranges from Stage 1 (childhood) to Stage 5 (full physical maturity).
Information regarding Tanner staging will be collected at baseline for all participants and will be stopped once the participant reaches Tanner Stage 5 in all gender-appropriate scales.
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Baseline, Week 52
|
|
Part 1: Number of Participants at Each Tanner Stage at Week 52
Tidsram: Baseline, Week 52
|
Assessment of Tanner stages (a scale of physical development) will be performed by a medical doctor experienced with this assessment.
Tanner score ranges from Stage 1 (childhood) to Stage 5 (full physical maturity).
Information regarding Tanner staging will be collected at baseline for all participants and will be stopped once the participant reaches Tanner Stage 5 in all gender-appropriate scales.
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Baseline, Week 52
|
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Part 1: Plasma Concentrations of Omaveloxolone
Tidsram: Pre-dose and post-dose on Day 1, Weeks 4, 12 and 26
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Pre-dose and post-dose on Day 1, Weeks 4, 12 and 26
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|
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Part 2A: Change From Baseline in USS Subscale E of mFARS at Week 104
Tidsram: Baseline (Week 52 of Part 1), Week 104
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The mFARS is a validated and sensitive rating scale that was developed to quantitatively assess the severity of the neurologic features of FA in adults and adolescents.
Scores on the mFARS range from 0 to 93, with lower scores indicating better neurological function.
The subscales of the mFARS assessment and maximum score for each subscale are: bulbar function (Subscale A; 2 assessments of speech and cough; maximum score = 5), upper limb coordination (Subscale B; 5 assessments of coordination of movement and function in arms and hands with each limb scored individually; maximum score = 36), lower limb coordination (Subscale C; 2 assessments of coordination of movement and function of lower limbs with each limb scored individually; maximum score = 16), and upright stability (USS, Subscale E; 9 assessments of sitting posture, stance, tandem walk, and gait assessments; maximum score = 36).
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Baseline (Week 52 of Part 1), Week 104
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Part 2A: Change from baseline in mFARS at Weeks 52 and Week 104
Tidsram: Baseline (Week 52 of Part 1), Weeks 52 and 104
|
The mFARS is a validated and sensitive rating scale that was developed to quantitatively assess the severity of the neurologic features of FA in adults and adolescents.
Scores on the mFARS range from 0 to 93, with lower scores indicating better neurological function.
The subscales of the mFARS assessment and maximum score for each subscale are: bulbar function (Subscale A; 2 assessments of speech and cough; maximum score = 5), upper limb coordination (Subscale B; 5 assessments of coordination of movement and function in arms and hands with each limb scored individually; maximum score = 36), lower limb coordination (Subscale C; 2 assessments of coordination of movement and function of lower limbs with each limb scored individually; maximum score = 16), and upright stability (USS, Subscale E; 9 assessments of sitting posture, stance, tandem walk, and gait assessments; maximum score = 36).
|
Baseline (Week 52 of Part 1), Weeks 52 and 104
|
|
Part 2A: Number of Participants With TEAE and TESAE
Tidsram: From the first dose of the study drug in Part 2A up to the end of follow-up period in Part 2A (up to Week 104)
|
From the first dose of the study drug in Part 2A up to the end of follow-up period in Part 2A (up to Week 104)
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|
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Part 2A: Number of Participants With Change From Baseline in Cardiac Function Assessed by ECHO at Weeks 52 and Week 104
Tidsram: Baseline (Week 52 of Part 1), Weeks 52 and 104
|
Baseline (Week 52 of Part 1), Weeks 52 and 104
|
|
|
Part 2A: Change From Baseline in Height at Weeks 52 and Week 104
Tidsram: Baseline (Week 52 of Part 1), Weeks 52 and 104
|
Baseline (Week 52 of Part 1), Weeks 52 and 104
|
|
|
Part 2A: Change From Baseline in Weight at Weeks 52 and Week 104
Tidsram: Baseline (Week 52 of Part 1), Weeks 52 and 104
|
Baseline (Week 52 of Part 1), Weeks 52 and 104
|
|
|
Part 2A: Change From Baseline in BMI at Weeks 52 and Week 104
Tidsram: Baseline (Week 52 of Part 1), Weeks 52 and 104
|
Baseline (Week 52 of Part 1), Weeks 52 and 104
|
|
|
Part 2A: Change From Baseline in C-SSRS at Weeks 52 and Week 104
Tidsram: Baseline (Week 52 of Part 1), Weeks 52 and 104
|
The C-SSRS is a low-burden measure of the spectrum of suicidal ideation and behavior that was developed to assess severity and track suicidal events through any treatment of individuals ≥ 6 years of age.
The C-SSRS is a clinical interview providing a summary of both ideation and behavior that can be administered by the clinician during any evaluation or risk assessment to identify the level and type of suicidality present.
The assessment includes "yes" or "no" responses for 5 questions each, related to suicidal ideation (wish to be dead, non-specific active suicidal thoughts, active suicidal ideation with any methods, active suicidal ideation with some intent, active suicidal ideation with specific plan) and suicidal behavior (preparatory acts or behavior, aborted attempt, interrupted attempt, actual attempt, suicide).
Numeric ratings are provided for severity of ideation, from 1 to 5, with 5 being the most severe.
|
Baseline (Week 52 of Part 1), Weeks 52 and 104
|
|
Part 2A: Percentage of Participants at Each Tanner Stage at Weeks 52 and Week 104
Tidsram: Baseline (Week 52 of Part 1), Weeks 52 and 104
|
Assessment of Tanner stages (a scale of physical development) will be performed by a medical doctor experienced with this assessment.
Tanner score ranges from Stage 1 (childhood) to Stage 5 (full physical maturity).
Information regarding Tanner staging will be collected at baseline for all participants and will be stopped once the participant reaches Tanner Stage 5 in all gender-appropriate scales.
|
Baseline (Week 52 of Part 1), Weeks 52 and 104
|
|
Part 2A: Number of Participants at Each Tanner Stage at Weeks 52 and Week 104
Tidsram: Baseline (Week 52 of Part 1), Weeks 52 and 104
|
Assessment of Tanner stages (a scale of physical development) will be performed by a medical doctor experienced with this assessment.
Tanner score ranges from Stage 1 (childhood) to Stage 5 (full physical maturity).
Information regarding Tanner staging will be collected at baseline for all participants and will be stopped once the participant reaches Tanner Stage 5 in all gender-appropriate scales.
|
Baseline (Week 52 of Part 1), Weeks 52 and 104
|
|
Part 2A: Plasma Concentrations of Omaveloxolone
Tidsram: Pre-dose and post-dose on Day 1, Weeks 4, 12 and 26
|
Pre-dose and post-dose on Day 1, Weeks 4, 12 and 26
|
|
|
Part 2B: Change From Baseline in mFARS Including USS at Weeks 52 and Week 104
Tidsram: Baseline (Week 52 of Part 1), Weeks 52 and 104
|
The mFARS is a validated and sensitive rating scale that was developed to quantitatively assess the severity of the neurologic features of FA in adults and adolescents.
Scores on the mFARS range from 0 to 93, with lower scores indicating better neurological function.
The subscales of the mFARS assessment and maximum score for each subscale are: bulbar function (Subscale A; 2 assessments of speech and cough; maximum score = 5), upper limb coordination (Subscale B; 5 assessments of coordination of movement and function in arms and hands with each limb scored individually; maximum score = 36), lower limb coordination (Subscale C; 2 assessments of coordination of movement and function of lower limbs with each limb scored individually; maximum score = 16), and upright stability (USS, Subscale E; 9 assessments of sitting posture, stance, tandem walk, and gait assessments; maximum score = 36).
|
Baseline (Week 52 of Part 1), Weeks 52 and 104
|
|
Parts 2A and 2B: Change From Baseline in FA-HI at Weeks 52 and Week 104
Tidsram: Baseline (Week 52 of Part 1), Weeks 52 and 104
|
The FA-HI is a participant reported survey that assesses overall disease burden on a 100-point scale, with 0 representing no disease burden and 100 representing the maximum level of disease burden containing 113 symptoms questions representing 18 symptomatic subscales.
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Baseline (Week 52 of Part 1), Weeks 52 and 104
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Parts 2A and 2B: Change From Baseline in PGI-S at Weeks 52 and Week 104
Tidsram: Baseline (Week 52 of Part 1), Weeks 52 and 104
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PGI-S will be conducted for participants 7 to < 16 years of age.
These are clinically meaningful outcome measures that are participant-relevant across all age groups and disease severities for this population.
PGI -S is a 1-item questionnaire where the response is recorded on a 4-point scale scored as: 1-normal, 2-mild, 3-moderate, or 4-severe.
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Baseline (Week 52 of Part 1), Weeks 52 and 104
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Parts 2A and 2B: Change From Baseline in CGI-S at Weeks 52 and Week 104
Tidsram: Baseline (Week 52 of Part 1), Weeks 52 and 104
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The CGI-S will be conducted for all enrolled participants, 2 to < 16 years of age.
The CGI-S rating evaluates the severity of individual symptoms and treatment response in participants with mental disorders.
The CGI-S is a 7-point scale that requires the clinician to rate the severity of the participant's illness at the time of assessment.
A rating of 1 is considered normal, or with the least severe symptoms, a rating of 7 is extremely ill, or the worst symptoms.
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Baseline (Week 52 of Part 1), Weeks 52 and 104
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Parts 2A and 2B: Change from baseline in FA-ADL at Part 2A Weeks 52 and Week 104
Tidsram: Baseline (Week 52 of Part 1), Weeks 52 and 104
|
Participants will answer the 9 questions of the FA-ADL survey in an interview style conducted by any site staff.
The FA-ADL survey assesses 9 concepts: (1) speech; (2) swallowing; (3) cutting food and handling utensils; (4) dressing; (5) personal hygiene; (6) falling; (7) walking; (8) quality of sitting position; and (9) bladder function.
|
Baseline (Week 52 of Part 1), Weeks 52 and 104
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Samarbetspartners och utredare
Sponsor
Utredare
- Studierektor: Medical Director, Biogen
Publikationer och användbara länkar
Användbara länkar
Studieavstämningsdatum
Studera stora datum
Studiestart (Faktisk)
Primärt slutförande (Beräknad)
Avslutad studie (Beräknad)
Studieregistreringsdatum
Först inskickad
Först inskickad som uppfyllde QC-kriterierna
Första postat (Faktisk)
Uppdateringar av studier
Senaste uppdatering publicerad (Faktisk)
Senaste inskickade uppdateringen som uppfyllde QC-kriterierna
Senast verifierad
Mer information
Termer relaterade till denna studie
Ytterligare relevanta MeSH-villkor
- Hjärnsjukdomar
- Sjukdomar i centrala nervsystemet
- Sjukdomar i nervsystemet
- Genetiska sjukdomar, medfödda
- Metaboliska sjukdomar
- Neurodegenerativa sjukdomar
- Heredodegenerativa störningar, nervsystemet
- Ryggmärgssjukdomar
- Mitokondriella sjukdomar
- Cerebellära sjukdomar
- Spinocerebellära degenerationer
- Medfödda, ärftliga och neonatala sjukdomar och abnormiteter
- Närings- och metabola sjukdomar
- Friedreich Ataxi
- Undermåliga droger
- Farmaceutiska förberedelser
- omeloxolon
Andra studie-ID-nummer
- 296FA301
- 2025-520896-13 (Annan identifierare: EU CT Number)
Plan för individuella deltagardata (IPD)
Planerar du att dela individuella deltagardata (IPD)?
IPD-planbeskrivning
Läkemedels- och apparatinformation, studiedokument
Studerar en amerikansk FDA-reglerad läkemedelsprodukt
Studerar en amerikansk FDA-reglerad produktprodukt
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