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
調査の概要
詳細な説明
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.
研究の種類
入学 (推定)
段階
- フェーズ2
- フェーズ 1
連絡先と場所
研究連絡先
- 名前:Sarah McCague
- 電話番号:267-426-1464
- メール:cigt@chop.edu
研究場所
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Pennsylvania
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Philadelphia、Pennsylvania、アメリカ、19104
- 募集
- Children's Hospital of Philadelphia
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コンタクト:
- Sarah McCague
- 電話番号:267-426-1464
- メール:cigt@chop.edu
-
-
参加基準
適格基準
就学可能な年齢
- 子
健康ボランティアの受け入れ
説明
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
研究計画
研究はどのように設計されていますか?
デザインの詳細
- 主な目的:処理
- 割り当て:なし
- 介入モデル:単一グループの割り当て
- マスキング:なし(オープンラベル)
武器と介入
参加者グループ / アーム |
介入・治療 |
|---|---|
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実験的:実験的
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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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この研究は何を測定していますか?
主要な結果の測定
結果測定 |
メジャーの説明 |
時間枠 |
|---|---|---|
|
Safety and tolerability of a single intravenous dose of LNP.UCD.ABE
時間枠: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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二次結果の測定
結果測定 |
メジャーの説明 |
時間枠 |
|---|---|---|
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Clinical efficacy of a single intravenous dose of LNP.UCD.ABE
時間枠: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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協力者と研究者
スポンサー
協力者
捜査官
- 主任研究者:Rebecca Ahrens-Nicklas, M.D., Ph.D.、Children's Hospital of Philadelphia
研究記録日
主要日程の研究
研究開始 (実際)
一次修了 (推定)
研究の完了 (推定)
試験登録日
最初に提出
QC基準を満たした最初の提出物
最初の投稿 (実際)
学習記録の更新
投稿された最後の更新 (実際)
QC基準を満たした最後の更新が送信されました
最終確認日
詳しくは
本研究に関する用語
追加の関連 MeSH 用語
その他の研究ID番号
- UCD-101
個々の参加者データ (IPD) の計画
個々の参加者データ (IPD) を共有する予定はありますか?
医薬品およびデバイス情報、研究文書
米国FDA規制医薬品の研究
米国FDA規制機器製品の研究
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