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

Study of LNP.UCD.ABE in Patients With Urea Cycle Disorders

2026년 8월 24일 업데이트: Rebecca Ahrens-Nicklas

Master Protocol for a Phase I/II Open-label Safety and Efficacy Study of LNP.UCD.ABE, a Lipid Nanoparticle-delivered Base Editing Therapy, in Patients With Urea Cycle Disorders Due to Variants Amenable to Corrective Editing by LNP.UCD.ABE

This is a single-site Phase 1/2 open-label umbrella clinical trial designed to evaluate the safety, tolerability, and efficacy of a single intravenous dose of LNP.UCD.ABE in 5 pediatric subjects with severe infantile-onset UCDs. This is a master clinical protocol in which subjects with a variant in a urea cycle disorder (UCD) gene (CPS1, OTC, ASS1, ASL, ARG, NAGS, or SLC25A15) that is demonstrated to be amenable to corrective editing by an adenine base editor (ABE) would be eligible for enrollment.

연구 개요

상세 설명

Humans ingest protein to support growth and the synthesis of a number of key macromolecules. Nitrogen waste generated from protein catabolism is converted to ammonia, which under normal physiologic conditions is converted to urea via the urea cycle. Urea is then excreted in urine to maintain whole-body nitrogen homeostasis. Loss of function of any of the six enzymes of the urea cycle-encoded by CPS1 (carbamoyl phosphate synthetase 1), OTC (ornithine transcarbamylase), ASS1 (argininosuccinate synthetase), ASL (argininosuccinate lyase), ARG (arginase), and NAGS (N-acetylglutamate synthetase)-results in a urea cycle disorder (UCD). In addition loss of the ornithine transporter, ORNT1 (encoded by SLC25A15), can also lead to disease.

Severe UCD patients typically present as neonates and have a profound decrease in function in any one of the six enzymes of the urea cycle or a lack of function of the ornithine transporter that carries urea cycle intermediates. This results in toxic accumulation of ammonia in the blood and accumulation of specific urea cycle amino acids that aid in diagnoses and therapeutic monitoring. Patients are at risk of developing extremely elevated blood ammonia levels (hyperammonemia) that can lead acutely to coma and death and chronically to profound neurologic dysfunction.

LNP.UCD.ABE is an investigational in vivo gene editing product proposed for the treatment of hyperammonemia in patients under 5 years of age with deficiencies in enzymes or a related transporter of the urea cycle who are homozygous or compound heterozygous for a pathogenic variant in any UCD gene, including CPS1, OTC, ASS1, ASL, ARG, NAGS, and SLC25A15, that can be efficiently corrected by an adenine base editor (ABE).

Each subject will have a personalized variant-specific LNP.UCD.ABE developed and evaluated during the Screening period, which may last up to 8 months. Subjects will eligible for a lead in period to establish a stable diet. After the subject's drug is developed and lead in has been completed, the subject will be administered LNP.UCD.ABE via a single intravenous infusion. After LNP.UCD.ABE administration, participants will be followed for safety and efficacy for 52 weeks.

연구 유형

중재적

등록 (추정된)

7

단계

  • 2 단계
  • 1단계

연락처 및 위치

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

연구 연락처

  • 이름: Sarah McCague
  • 전화번호: 267-426-1464
  • 이메일: cigt@chop.edu

연구 장소

    • Pennsylvania
      • Philadelphia, Pennsylvania, 미국, 19104
        • 모병
        • Children's Hospital of Philadelphia
        • 연락하다:
          • Sarah McCague
          • 전화번호: 267-426-1464
          • 이메일: cigt@chop.edu

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

  • 어린이

건강한 자원 봉사자를 받아들입니다

아니

설명

Inclusion Criteria:

  1. Diagnosis of a severe urea cycle disorder, in the judgement of the investigators.
  2. Molecular testing demonstrating homozygosity or compound heterozygosity for a disease-causing mutation in CPS1 that is targeted by a variant-specific version of the LNP.UCD.ABE drug product.
  3. Current or historical biochemical testing consistent with a urea cycle disorder
  4. At least one of the subject's alleles must be amenable to base editing by LNP.UCD.ABE, as assessed in vitro
  5. A history of an ammonia level of ≥400 μmol/L prior to age 12 months, unless a diagnosis was made prenatally and care was initiated immediately after birth

    • If the patient is taking a nitrogen scavenger medication, their ammonia level may currently be in the normal range
    • If the patient is diagnosed prenatally, then personal history, family history, or analysis of mutations should indicate a high likelihood of a severe UCD.
  6. Subjects more than 8 weeks from the initial diagnosis of a UCD must have demonstrated:

    • a persistent need for dietary protein restriction and chronic administration of a nitrogen scavenger medication, AND / OR
    • a recurrent hyperammonemic event AND / OR
    • a history of a hyperammonemia-induced seizure
  7. Weight >3.5 kg at the time of screening
  8. Legal guardian(s) capable of giving signed informed consent, which includes compliance with the requirements and restrictions listed in the informed consent form (ICF) and in this protocol.

Exclusion Criteria:

  1. Abnormal liver function, electrolyte, coagulation, or blood count laboratory values thought not attributable to the underlying urea cycle disorder;
  2. Demonstrated need for urgent liver transplantation due to liver failure, in the opinion of the investigators;
  3. Participation in a prior gene therapy trial or participation in a trial of an investigational product in the last 12 months;
  4. History of liver transplantation;
  5. Any other diseases or conditions that the investigators would consider to pose unacceptable risk to the subject;
  6. Inability or unwillingness to comply with the visit schedule and study assessments;
  7. Any genetic variation in the causative urea cycle disorder gene that, in the opinion of the investigators, may decrease the potential efficacy of the drug product;
  8. History of severe hypersensitivity or anaphylaxis to polyethylene glycol (PEG)-containing products, such as PEG-containing vaccines or laxatives

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: 해당 없음
  • 중재 모델: 단일 그룹 할당
  • 마스킹: 없음(오픈 라벨)

무기와 개입

참가자 그룹 / 팔
개입 / 치료
실험적: 실험적
Each subject will have a personalized variant-specific LNP.UCD.ABE developed and evaluated in real time. Each member of the LNP.UCD.ABE drug product (DP) family is a lipid nanoparticle (LNP)-based editing therapeutic comprising lipid excipients, a messenger RNA (mRNA) drug substance (DS) encoding an adenine base editor (ABE), and a single guide RNA (gRNA) DS.

연구는 무엇을 측정합니까?

주요 결과 측정

결과 측정
측정값 설명
기간
Safety and tolerability of a single intravenous dose of LNP.UCD.ABE
기간: 52 weeks
Incidence of treatment-emergent adverse events as assessed by CTCAE version 6.0 criteria at 52 weeks after LNP.UCD.ABE administration.
52 weeks

2차 결과 측정

결과 측정
측정값 설명
기간
Clinical efficacy of a single intravenous dose of LNP.UCD.ABE
기간: 16 weeks
The proportion of the population that can tolerate: 1. an increase in protein intake to 100% of the recommended dietary allowance (RDA) for age and/or at least a 50% reduction in the nitrogen scavenger medication dose, without an associated hyperammonemic event [defined as an ammonia level ≥ 2.5× upper limit of normal (ULN) on a non-hemolyzed specimen and the presence of symptoms of hyperammonemia], by 16 weeks after administration of LNP.UCD.ABE.
16 weeks

공동 작업자 및 조사자

여기에서 이 연구와 관련된 사람과 조직을 찾을 수 있습니다.

수사관

  • 수석 연구원: Rebecca Ahrens-Nicklas, M.D., Ph.D., Children's Hospital of Philadelphia

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)

2026년 8월 7일

기본 완료 (추정된)

2028년 9월 1일

연구 완료 (추정된)

2028년 9월 1일

연구 등록 날짜

최초 제출

2026년 6월 18일

QC 기준을 충족하는 최초 제출

2026년 6월 18일

처음 게시됨 (실제)

2026년 6월 25일

연구 기록 업데이트

마지막 업데이트 게시됨 (실제)

2026년 8월 26일

QC 기준을 충족하는 마지막 업데이트 제출

2026년 8월 24일

마지막으로 확인됨

2026년 8월 1일

추가 정보

이 연구와 관련된 용어

개별 참가자 데이터(IPD) 계획

개별 참가자 데이터(IPD)를 공유할 계획입니까?

아니요

약물 및 장치 정보, 연구 문서

미국 FDA 규제 의약품 연구

예

미국 FDA 규제 기기 제품 연구

아니

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