Study of LNP.UCD.ABE in Patients With Urea Cycle Disorders
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
研究概览
详细说明
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.
研究类型
注册 (估计的)
阶段
- 阶段2
- 阶段1
联系人和位置
学习联系方式
- 姓名:Sarah McCague
- 电话号码:267-426-1464
- 邮箱:cigt@chop.edu
学习地点
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Pennsylvania
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Philadelphia、Pennsylvania、美国、19104
- 招聘中
- Children's Hospital of Philadelphia
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接触:
- Sarah McCague
- 电话号码:267-426-1464
- 邮箱:cigt@chop.edu
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-
参与标准
资格标准
适合学习的年龄
- 孩子
接受健康志愿者
描述
Inclusion Criteria:
- Diagnosis of a severe urea cycle disorder, in the judgement of the investigators.
- 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.
- Current or historical biochemical testing consistent with a urea cycle disorder
- At least one of the subject's alleles must be amenable to base editing by LNP.UCD.ABE, as assessed in vitro
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.
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
- Weight >3.5 kg at the time of screening
- 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:
- Abnormal liver function, electrolyte, coagulation, or blood count laboratory values thought not attributable to the underlying urea cycle disorder;
- Demonstrated need for urgent liver transplantation due to liver failure, in the opinion of the investigators;
- Participation in a prior gene therapy trial or participation in a trial of an investigational product in the last 12 months;
- History of liver transplantation;
- Any other diseases or conditions that the investigators would consider to pose unacceptable risk to the subject;
- Inability or unwillingness to comply with the visit schedule and study assessments;
- 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;
- History of severe hypersensitivity or anaphylaxis to polyethylene glycol (PEG)-containing products, such as PEG-containing vaccines or laxatives
学习计划
研究是如何设计的?
设计细节
- 主要用途:治疗
- 分配:不适用
- 介入模型:单组作业
- 屏蔽:无(打开标签)
武器和干预
参与者组/臂 |
干预/治疗 |
|---|---|
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实验性的:实验性的
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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.
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研究衡量的是什么?
主要结果指标
结果测量 |
措施说明 |
大体时间 |
|---|---|---|
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Safety and tolerability of a single intravenous dose of LNP.UCD.ABE
大体时间:52 weeks
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Incidence of treatment-emergent adverse events as assessed by CTCAE version 6.0 criteria at 52 weeks after LNP.UCD.ABE administration.
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52 weeks
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次要结果测量
结果测量 |
措施说明 |
大体时间 |
|---|---|---|
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Clinical efficacy of a single intravenous dose of LNP.UCD.ABE
大体时间:16 weeks
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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.
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16 weeks
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合作者和调查者
合作者
调查人员
- 首席研究员:Rebecca Ahrens-Nicklas, M.D., Ph.D.、Children's Hospital of Philadelphia
研究记录日期
研究主要日期
学习开始 (实际的)
初级完成 (估计的)
研究完成 (估计的)
研究注册日期
首次提交
首先提交符合 QC 标准的
首次发布 (实际的)
研究记录更新
最后更新发布 (实际的)
上次提交的符合 QC 标准的更新
最后验证
更多信息
与本研究相关的术语
其他相关的 MeSH 术语
其他研究编号
- UCD-101
计划个人参与者数据 (IPD)
计划共享个人参与者数据 (IPD)?
药物和器械信息、研究文件
研究美国 FDA 监管的药品
研究美国 FDA 监管的设备产品
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