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A Phase III Double-blind Study With Idebenone in Patients With Duchenne Muscular Dystrophy (DMD) Taking Glucocorticoid Steroids

Phase 3
Terminated
Conditions
Duchenne Muscular Dystrophy (DMD)
Interventions
Registration Number
NCT02814019
Lead Sponsor
Santhera Pharmaceuticals
Brief Summary

The purpose of the study is to assess the efficacy of idebenone in delaying the loss of respiratory function in patients with DMD receiving concomitant glucocorticoid steroids

Detailed Description

The SIDEROS trial is a randomized, placebo controlled, parallel group study of the efficacy of idebenone in delaying the loss of respiratory function, whilst also monitoring safety and tolerability of idebenone in at least 266 DMD patients taking stable dose of concomitant glucocorticoid steroids.

The study treatment period will be 18 months/ 78 weeks and the idebenone dose will be 900 mg/day. Participants can use deflazacort or prednisolone and be on any dose regimen.

Since glucocorticoid steroids are widely used in ambulant boys from an early age until late into teenage and even adult years, this study will not take age and ambulatory status into account and will only exclude patients that need daytime ventilator assistance.

The schedule of assessments will include a Screening Visit and up to 9 protocol visits, including a Follow-up Visit.

A Screening Visit will take place a maximum of 4 weeks prior to the Baseline Visit (Visit 1, study day -1). Beginning at Baseline, the patient will receive study medication to be taken at home, and will undergo regular assessments in the clinic throughout the study period until Visit 8 at Week 78 at which time the study will be completed and medication discontinued.

All patients completing Visit 8/Week 78, and considered eligible by the Investigator will be able to participate in an open-label extension study (SIDEROS-E) and will continue to receive idebenone until idebenone is commercially available for patients included in the study or SIDEROS-E is terminated by the Sponsor, whichever occurs first. The duration of the SIDEROS-E study will be defined in a separate protocol.

For all patients not participating in the extension study (SIDEROS-E), a Follow-up Visit (Visit 9/Follow-up Visit) will take place 4 weeks after end of Treatment at Visit 8/Week 78 or after premature discontinuation of study medication.

Each hospital visit will include efficacy assessments (respiratory function assessed by hospital-based spirometry, oxygen saturation, end-tidal CO2) and safety assessments (adverse events, concomitant medication, physical examination, vital signs, safety laboratory evaluations). In addition, respiratory function will be assessed weekly at home with a hand-held device in order to closely monitor respiratory function between hospital visits.

The study medication, all medical procedures and laboratory testing, and the visits to the study centre are free of charge. In addition the patients will receive a travel allowance to cover reasonable expenses to and from the study centre. Participants will not otherwise be compensated for this study.

Recruitment & Eligibility

Status
TERMINATED
Sex
Male
Target Recruitment
255
Inclusion Criteria
  1. Male patients with a 35% ≤ FVC ≤ 80% of predicted value at Screening and at Baseline and who, in the opinion of the investigator are in the respiratory function decline phase.
  2. Minimum 10 years old at Screening.
  3. Signed and dated Informed Consent Form.
  4. Documented diagnosis of DMD (severe dystrophinopathy) and clinical features consistent of typical DMD at diagnosis (i.e. documented delayed motor skills and muscle weakness by age 5 years). DMD should be confirmed by mutation analysis in the dystrophin gene or by substantially reduced levels of dystrophin protein (i.e. absent or <5% of normal) on Western blot or immunostaining.
  5. Chronic use of systemic glucocorticoid steroids for DMD related conditions continuously for at least 12 months prior to Baseline without any dose adjustments on a mg/kg basis in the last 6 months (only dose adjustments determined by weight changes are allowed).
  6. Ability to provide reliable FVC values at Screening and Baseline, and reproducible within 15% (relative change) at Baseline compared to Screening.
  7. Patients assessed by the Investigator as willing and able to comply with the requirements of the study, possess the required cognitive abilities and are able to swallow study medication.
  8. Patients who prior to Screening have been immunized with 23-valent pneumococcal polysaccharide vaccine or any other pneumococcal polysaccharide vaccine as per national recommendations, as well as annually immunized with inactivated influenza vaccine.
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Exclusion Criteria
  1. Symptomatic heart failure (defined as patients with structural heart disease, dyspnea, fatigue and impaired tolerance to exercise; Stage C by the ACCF/AHA guideline or NYHA Classes III-IV) and/or symptomatic ventricular arrhythmias.
  2. Ongoing participation in any other therapeutic trial and/or intake of any investigational drug within 90 days prior to Baseline (only exception allowed is use of Deflazacort in the US as part of the Expanded Access Program, or any approved corticosteroid product in trial for regimen optimization, for which the patient met the inclusion criterion 5).
  3. Ongoing exon-skipping therapy or read-through gene therapy for DMD; previous exon-skipping or read-through gene therapy is allowed if the stop date was more than 6 months prior to Screening.
  4. Planned or expected spinal fusion surgery during the study period (as judged by the Investigator; i.e. due to rapidly progressing scoliosis), previous spinal fusion surgery is allowed if it took place more than 6 months prior to Screening.
  5. Asthma, bronchitis/COPD, bronchiectasis, emphysema, pneumonia or presence of any other non-DMD respiratory illness that affects respiratory function.
  6. Chronic use of beta2-agonists or any use of other bronchodilating/bronchoconstricting medication (inhaled steroids, sympathomimetics, anticholinergics, antihistamines); chronic use is defined as a daily intake for more than 14 days.
  7. Any bronchopulmonary illness that required treatment with antibiotics within 3 months prior to Screening.
  8. Moderate or severe hepatic impairment (use as guidance Child-Pugh class B [7 to 9 points] or Child-Pugh class C [10 to 15 points] - see Appendix B) or severe renal impairment (eGFR <30 mL/min/1.73 m2).
  9. Prior or ongoing medical condition or laboratory abnormality that in the Investigator's opinion may put the patient at significant risk may confound the study results or may interfere significantly with the patient's participation in the study.
  10. Relevant history of or current drug or alcohol abuse, or use of any tobacco or marijuana products/smoking.
  11. Known individual hypersensitivity to idebenone or to any of the ingredients or excipients of the study medication.
  12. Daytime ventilator assistance (defined as use of any assisted ventilation while awake).

Note: Patients who suffer from a severe, unstable condition including (but not limited to) cancer, auto-immune diseases, hematological diseases, metabolic disorders or immunodeficiencies, and who are at risk of an aggravation unrelated to the study condition, can only be included in the study if accepted in writing by the Sponsor's Senior Clinical Research Physician.

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Study & Design

Study Type
INTERVENTIONAL
Study Design
PARALLEL
Arm && Interventions
GroupInterventionDescription
placeboplacebomatching placebo tablets
idebenone 150 mg film-coated tabletsIdebenone 150 mg film-coated tablets900 mg idebenone/day (2 tablets to be taken 3 times a day with meals)
Primary Outcome Measures
NameTimeMethod
Change From Baseline in Forced Vital Capacity percent predicted (FVC %p) at Week 7878 weeks

Delaying the loss of respiratory function in patients with DMD receiving glucocorticoid steroids as measured by changes in FVC %p from Baseline to Week 78 using hospital based spirometry.

Secondary Outcome Measures
NameTimeMethod
Change From Baseline in Percent Predicted Peak Expiratory Flow (PEF %p) at Week 7878 weeks

Delaying the loss of respiratory function in patients with DMD receiving glucocorticoid steroids as measured by:

•The change from Baseline to Week 78 in PEF %p assessed by hospital-based spirometry measurements

Change from Baseline in Inspiratory Flow Reserve (IFR) at Week 7878 weeks

Delaying the loss of respiratory function in patients with DMD receiving glucocorticoid steroids as measured by:

•The change from Baseline to Week 78 in IFR assessed by hospital-based spirometry measurements

Change From Baseline in Forced Vital Capacity (FVC) at Week 7878 weeks

Delaying the loss of respiratory function in patients with DMD receiving glucocorticoid steroids as measured by:

•The time to first 10% decline in FVC (L) during the 78-week treatment period, assessed by hospital-based spirometry measurements

Trial Locations

Locations (63)

Johns Hopkins University

🇺🇸

Baltimore, Maryland, United States

Rare Disease Research

🇺🇸

Atlanta, Georgia, United States

Childrens Hospital of Los Angeles

🇺🇸

Los Angeles, California, United States

University of Iowa

🇺🇸

Iowa City, Iowa, United States

Shriners Hospitals for Children-Tampa

🇺🇸

Tampa, Florida, United States

Gillette Children's Specialty Healthcare

🇺🇸

Saint Paul, Minnesota, United States

Children's Hospital of Philadelphia

🇺🇸

Philadelphia, Pennsylvania, United States

Hôpital des enfants

🇫🇷

Toulouse, France

Cook Children's Medical Center

🇺🇸

Fort Worth, Texas, United States

Universitätsklinikum Hamburg-Eppendorf, Klinik für Kinder- und Jugendmedizin

🇩🇪

Hamburg, Germany

Universitätsklinikum Essen

🇩🇪

Essen, Germany

Universitätsklinik Freiburg Zentrum für Kinderheilkunde und Jugendmedizin

🇩🇪

Freiburg, Germany

Loma Linda University Healthcare

🇺🇸

San Bernardino, California, United States

CHRU de Montpellier - Hôpital Gui de Chauliac

🇫🇷

Montpellier, France

University of Kansas

🇺🇸

Fairway, Kansas, United States

Uniklinik Köln

🇩🇪

Köln, Germany

Fondazione IRCCS Eugenio Medea

🇮🇹

Bosisio Parini, Italy

University of Alabama

🇺🇸

Birmingham, Alabama, United States

Universitätsmedizin Berlin Campus Virchow-Klinikum

🇩🇪

Berlin, Germany

Hospital Sant Joan de Déu Neuropediatra

🇪🇸

Barcelona, Spain

University Hospital Leuven

🇧🇪

Leuven, Belgium

Centro Clinico NEMO (NEuroMuscular Omnicentre), Niguarda Hospital

🇮🇹

Milano, Italy

Cincinnati Children's Hospital

🇺🇸

Cincinnati, Ohio, United States

UCL, National Hospital for Neurology and Neurosurgery

🇬🇧

London, United Kingdom

Hospital La Fe de Valencia

🇪🇸

Valencia, Spain

Sahlgrenska University Hospital

🇸🇪

Gothenburg, Sweden

Universitätsklinikum Heidelberg Zentrum für Kinder- und Jugendmedizin

🇩🇪

Heidelberg, Germany

LUMC

🇳🇱

Leiden, Netherlands

Istituto Giannina Gaslini

🇮🇹

Genova, Italy

Leeds Teaching Hospital NHS Trust

🇬🇧

Leeds, United Kingdom

Zentrum für neuromuskuläre Erkrankungen

🇩🇪

München, Germany

Hospital Universitari Vall D' Hebron

🇪🇸

Barcelona, Spain

Hospital Universitario La Paz

🇪🇸

Madrid, Spain

Children's University Hospital

🇮🇪

Dublin, Ireland

Institute of Neurology at Schneider Children's Medical Center of Israel

🇮🇱

Petah Tiqva, Israel

Dipartimento di Clinica Neurologica e Psichiatrica dell'Età Evolutiva della Fondazione IRCCS "C. Mondino" di Pavia

🇮🇹

Pavia, Italy

Hospital Universitario Virgen del Rocio

🇪🇸

Sevilla, Spain

Banner University of Arizona Medical Center

🇺🇸

Tucson, Arizona, United States

David Geffen School of Medicine at UCLA

🇺🇸

Los Angeles, California, United States

UC Davis Department of Physical Medicine and Rehabilitation

🇺🇸

Sacramento, California, United States

Children's Hospital Boston

🇺🇸

Boston, Massachusetts, United States

Neurosciences Institute, Neurology - Charlotte Carolinas Healthcare System

🇺🇸

Charlotte, North Carolina, United States

I-Motion - Plateforme d'essais cliniques pédiatriques Hôpital Armand Trousseau bâtiment Lemariey porte 20

🇫🇷

Paris, France

Hôpital Hôtel Dieu

🇫🇷

Nantes, France

Servizio di Cardiomiologia e Genetica Medica, AOU Università degli Studi della Campania "Luigi Vanvitelli"

🇮🇹

Napoli, Italy

Robert Jones and Agnes Hunt Orthopaedic Hospital

🇬🇧

Oswestry, United Kingdom

Oxford University hospitals NHS Foundation Trust

🇬🇧

Oxford, United Kingdom

Service de neuropédiatrie Pôle Pédiatrie CHRU de Lille - Hôpital Jeanne de Flandre

🇫🇷

Lille, France

Gottfried von Preyer'sches Kinderspital

🇦🇹

Wien, Austria

Centre de Référence Neuromusculaire, CHR Citadelle

🇧🇪

Liège, Belgium

Phoenix Children's Hospital

🇺🇸

Phoenix, Arizona, United States

Sofia Medical University

🇧🇬

Sofia, Bulgaria

Center for neuromuscular disorders, Universitäts-Kinderspital beider Basel (UKBB)

🇨🇭

Basel, Switzerland

MetroHealth Medical Center

🇺🇸

Cleveland, Ohio, United States

Semmelweis University 2nd Department of Paediatrics

🇭🇺

Budapest, Hungary

Radboud university medical centre

🇳🇱

Nijmegen, Netherlands

U.O.C. Neuropsichiatria Infantile

🇮🇹

Roma, Italy

Scientific Coordinator Nemo Sud Clinical Center

🇮🇹

Messina, Italy

Reparto Di Neurologia dell'Osperdale Di Padova

🇮🇹

Padova, Italy

Great Ormond Street Hospital for Children

🇬🇧

London, United Kingdom

John Walton Muscular Dystrophy Research Centre

🇬🇧

Newcastle, United Kingdom

Royal Hallamshire Hospital

🇬🇧

Sheffield, United Kingdom

University of Rochester Medical Center

🇺🇸

Rochester, New York, United States

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