이 페이지는 자동 번역되었으며 번역의 정확성을 보장하지 않습니다. 참조하십시오 영문판 원본 텍스트의 경우.

A Phase I/II Study of BMN053 in Subjects With Duchenne Muscular Dystrophy (DMD)

2017년 12월 6일 업데이트: BioMarin Pharmaceutical

A Phase I/II, Open-label, Dose Escalating With 48 Week Treatment Study to Assess the Safety and Tolerability, Pharmacokinetics, Pharmacodynamics and Efficacy of BMN053 (Previously Known as PRO053) in Subjects With Duchenne Muscular Dystrophy.

The purpose of the study is to see whether BMN053 is safe and effective to use as medication for Duchenne muscular dystrophy (DMD) patients with a mutation around location 53 in the DNA for the dystrophin protein.

연구 개요

상세 설명

A Phase I/II, open-label, dose escalating with 48-week treatment study to assess the safety and tolerability, pharmacokinetics, pharmacodynamics and efficacy of BMN 053 (previously known as PRO053) in subjects with Duchenne muscular dystrophy

연구 유형

중재적

등록 (실제)

9

단계

  • 2 단계
  • 1단계

연락처 및 위치

이 섹션에서는 연구를 수행하는 사람들의 연락처 정보와 이 연구가 수행되는 장소에 대한 정보를 제공합니다.

연구 장소

      • Leiden, 네덜란드, 2333ZA
        • Leids Universitair Medisch Centrum
      • Leuven, 벨기에, 3000
        • UZ Leuven, Campus Gasthuisberg
      • London, 영국, WC1N 3JH
        • Great Ormond Street Hospital for Children
      • Newcastle, 영국, NE1 3BZ
        • Institute of Genetic Medicine International Centre for Life
      • Rome, 이탈리아, 00168
        • Policlinico Universitario Agostino Gemelli
      • Paris, 프랑스, 75651
        • Institut de Myologie

참여기준

연구원은 적격성 기준이라는 특정 설명에 맞는 사람을 찾습니다. 이러한 기준의 몇 가지 예는 개인의 일반적인 건강 상태 또는 이전 치료입니다.

자격 기준

공부할 수 있는 나이

5년 (어린이, 성인)

건강한 자원 봉사자를 받아들입니다

아니

연구 대상 성별

남성

설명

Inclusion Criteria:

  1. Duchenne muscular dystrophy resulting from a mutation correctable by treatment with BMN053 confirmed by a state-of-the-art DNA diagnostic technique covering all DMD gene exons, including but not limited to MLPA (Multiplex Ligation-dependent Probe Amplification), CGH (Comparative Genomic Hybridisation), SCAIP (Single Condition Amplification/Internal Primer) or HRMCA (High-Resolution Melting Curve Analysis).
  2. Ambulant boys aged at least 5 years on the day of first dosing able to walk for at least 300 metres in the 6 minute walking distance (6MWD) test. In addition, results of the 6MWD test must be within ±30 metres of each other at 2 of 3 pre-treatment visits (screen 1, 2 and baseline) prior to first BMN053 administration.
  3. Adequate quality for biopsy (confirmed with MRI) of the lateral head of the gastrocnemius muscle. Only under exceptional circumstances will an alternative muscle (preferably brachii) be considered for biopsy and only following discussion between the Principal Investigator and the BioMarin Medical Monitor.
  4. Life expectancy of at least 3 years after inclusion in the study.
  5. Glucocorticosteroid use which is stable for at least 3 months prior to first BMN053 administration. Subjects must have been receiving glucocorticosteroids for at least 6 months prior to the first BMN053 administration.
  6. Willing and able to adhere to the study visit schedule and other protocol requirements.
  7. Written informed consent signed (by parent(s)/legal guardian and/or the subject, according to the local regulations).
  8. In France, a subject will be eligible for inclusion in this study only if either affiliated to, or a beneficiary of, a social security category.
  9. Anticipated adequate vein access for intravenous (IV) infusion.

Exclusion Criteria:

  1. Current or history of liver disease or impairment.
  2. Current or history of renal disease or impairment.
  3. At least two aPTT above upper limit of normal (ULN) within the last month prior to first dose of BMN053.
  4. Screening platelet count below the lower limit of normal (LLN).
  5. Acute illness within 4 weeks prior to first dose of BMN053 which may interfere with the study assessments.
  6. Severe mental retardation and/or behavioural problems which, in the opinion of the Investigator, prohibit participation in this study.
  7. Severe cardiomyopathy which, in the opinion of the Investigator prohibits participation in this study. If a subject has a left ventricular ejection fraction <45% at screening, the Investigator should discuss inclusion of the subject with the Medical Monitor.
  8. Expected need for daytime mechanical ventilation within the next year.
  9. Use of anticoagulants, antithrombotics or antiplatelet agents.
  10. Use of idebenone or other forms of coenzyme Q10 within 1 month prior to the start of the screening for the study.
  11. Use of nutritional or herbal supplements which, in the opinion of the Investigator, may influence muscle performance within 1 month prior to first dose of BMN053.
  12. Use of any other investigational product or participation in another trial with an investigational product, within 6 months prior to the start of the screening for the study.

공부 계획

이 섹션에서는 연구 설계 방법과 연구가 측정하는 내용을 포함하여 연구 계획에 대한 세부 정보를 제공합니다.

연구는 어떻게 설계됩니까?

디자인 세부사항

  • 주 목적: 치료
  • 할당: 무작위화되지 않음
  • 중재 모델: 병렬 할당
  • 마스킹: 없음(오픈 라벨)

무기와 개입

참가자 그룹 / 팔
개입 / 치료
실험적: Dose escalation phase
In the dose-escalation phase, following screening assessment, two cohorts of three subjects each receive two single doses of BMN 053 in two study periods (i.e., four single doses in total per subject). In each study period they will receive BMN 053 by IV infusion and by SC injection (separated by one week). The proposed doses are 1 mg/kg (Cohort 1, study period 1), 3 mg/kg (Cohort 2, study period 1), 6 mg/kg (Cohort 1, study period 2) and 9 mg/kg (Cohort 2, study period 2). The actual doses may be amended or repeated based on emerging data from previous doses.
All doses of BMN053 have been administered as subcutaneous injections.
실험적: Regimen selection
After completion of the dose-escalation period of Cohort 1, the safety data of the subjects will be reviewed by a DSMB and if no safety concerns these subjects will continue to receive 6 mg/kg BMN053 weekly by SC injection for 48 weeks. 3 more treatment-naïve subjects will be entered into this Group. These 6 subjects will form Group 1 of the Regimen Selection phase who received 6 mg/kg SC. At the time of this amendment (4) this part of the study has been completed. Following completion of the dose-escalation study period of Cohort 2 (9 mg/kg), the planned review of the preliminary plasma PK data from the dose-escalation phase showed a relative bioavailability of 50% for BMN053 with SC dosing (50% lower plasma AUC after SC dosing compared to IV dosing). Taking into consideration the risk of injection site reactions noted with similar compounds when administered SC over longer term, the planned 9 mg/kg BMN053 weekly by SC injection will be discontinued to be replaced by an IV regimen.

All doses of BMN053 will be administered as IV infusions. The proposed doses are as follows:

• 3 mg/kg

All doses of BMN053 will be administered as IV infusions. The proposed doses are as follows:

• 4-6 mg/kg

실험적: 48-week Treatment Phase

Thirty additional treatment-naïve subjects will be recruited for the primary evaluation and will receive treatment at the recommended regimen for a total of 48 weeks. Subjects dosed initially in the dose escalation phase and/or the regimen selection phase of the study will not be included in the primary analysis.

Following completion of the 2nd study period for Cohort 2, the safety data will be reviewed by the DSMB and in the absence of safety concerns the subjects may enter the 48 week treatment phase and receive 9 mg/kg PRO053 once weekly by SC injection. Three new subjects will enter cohort 2 (i.e. 6 subjects in total at this dose level).

After the initial 12 subjects have completed 12 weeks of dosing the dose for the Treatment group (30 new subjects) will be selected based on the totality of the 12-week data from those initial 12 subjects. The initial 12 subjects will also be dosed on the selected dose (i.e. continue on their dose or [down-]titrate).

All doses of PRO053 will be administered as IV infusions. The proposed doses will be decided upon completion of the Regimen Selection Phase of Groups 2 and 3
실험적: Dosing extension
All subjects who have completed the dose escalation and regimen selection phase of the study (N=15), and subjects who have complete the treatment phase of the study who have tolerated the treatment will be offered to continue dosing in the dosing extension with ongoing assessment of efficacy, safety, and tolerability of BMN 053. Safety, efficacy, PK/PD and biomarker assessments will be performed at scheduled visits; adverse events (AEs) and concomitant medications and therapies will be continuously monitored. The dose extension phase will provide BMN 053 treatment for 48 weeks.
All doses of PRO053 will be administered as IV infusions. The proposed doses will be decided upon completion of the Regimen Selection Phase of Groups 2 and 3 and the Treatment Phase Group 4.

연구는 무엇을 측정합니까?

주요 결과 측정

결과 측정
기간
6분 걷기 테스트에서 기준선에서 변경
기간: 치료 단계 48주 후
치료 단계 48주 후

2차 결과 측정

결과 측정
기간
근력
기간: 48주 치료 단계 후
48주 치료 단계 후
Muscle function
기간: after 48 weeks treatment phase
after 48 weeks treatment phase
Pulmonary function
기간: after 48 weeks treatment phase
after 48 weeks treatment phase
Functional outcomes questionnaire
기간: after 48 weeks treatment phase
after 48 weeks treatment phase
Adverse Events
기간: after single intravenous and subcutaneous doses, and after 48 weeks of treatment phase
after single intravenous and subcutaneous doses, and after 48 weeks of treatment phase
Safety Laboratory
기간: after single intravenous and subcutaneous doses, and after 48 weeks of treatment phase
after single intravenous and subcutaneous doses, and after 48 weeks of treatment phase
Cardiac function
기간: after single intravenous and subcutaneous doses, and after 48 weeks of treatment phase
after single intravenous and subcutaneous doses, and after 48 weeks of treatment phase
Pharmacokinetic parameters at different dose levels
기간: after single intravenous and subcutaneous doses, and after 48 weeks of treatment phase
after single intravenous and subcutaneous doses, and after 48 weeks of treatment phase
Presence of (BMD-like) dystrophin expression in muscle biopsy
기간: after 48 weeks treatment phase
after 48 weeks treatment phase
Production of exon skip 53 mRNA in muscle biopsy
기간: after 48 weeks treatment phase
after 48 weeks treatment phase

공동 작업자 및 조사자

여기에서 이 연구와 관련된 사람과 조직을 찾을 수 있습니다.

수사관

  • 수석 연구원: V. Straub, Prof., Institute of Genetic Medicine, Newcastle University, UK

간행물 및 유용한 링크

연구에 대한 정보 입력을 담당하는 사람이 자발적으로 이러한 간행물을 제공합니다. 이것은 연구와 관련된 모든 것에 관한 것일 수 있습니다.

유용한 링크

연구 기록 날짜

이 날짜는 ClinicalTrials.gov에 대한 연구 기록 및 요약 결과 제출의 진행 상황을 추적합니다. 연구 기록 및 보고된 결과는 공개 웹사이트에 게시되기 전에 특정 품질 관리 기준을 충족하는지 확인하기 위해 국립 의학 도서관(NLM)에서 검토합니다.

연구 주요 날짜

연구 시작 (실제)

2013년 6월 1일

기본 완료 (실제)

2016년 8월 3일

연구 완료 (실제)

2016년 8월 3일

연구 등록 날짜

최초 제출

2013년 7월 2일

QC 기준을 충족하는 최초 제출

2013년 10월 7일

처음 게시됨 (추정)

2013년 10월 8일

연구 기록 업데이트

마지막 업데이트 게시됨 (실제)

2017년 12월 8일

QC 기준을 충족하는 마지막 업데이트 제출

2017년 12월 6일

마지막으로 확인됨

2017년 12월 1일

추가 정보

이 정보는 변경 없이 clinicaltrials.gov 웹사이트에서 직접 가져온 것입니다. 귀하의 연구 세부 정보를 변경, 제거 또는 업데이트하도록 요청하는 경우 register@clinicaltrials.gov. 문의하십시오. 변경 사항이 clinicaltrials.gov에 구현되는 즉시 저희 웹사이트에도 자동으로 업데이트됩니다. .

구독하다