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Clinical Recommendations Draft Guidance

IND Submissions for Individualized Antisense Oligonucleotide Drug Products for Severely Debilitating or Life-Threatening Diseases: Clinical Recommendations


Draft Guidance for Industry

December 2021

Not for implementation. Contains non-binding recommendations.


Docket Number: FDA-2021-D-1140

Issued by: Center for Drug Evaluation and Research


This guidance is intended for sponsor-investigators (hereafter referred to as sponsors) developing individualized investigational antisense oligonucleotide (ASO) drug products for a severely debilitating or life-threatening (SDLT) genetic disease. Most often, individuals with such diseases will have no alternative treatment options, and their diseases will be rapidly progressing, resulting in early death and/or devastating or irreversible morbidity within a short time frame without treatment. In these situations, drug development targeted to a larger number of patients with the same disease is not anticipated because of the specificity of the mechanism of action of the ASO combined with the rarity of the treatment-amenable patient population. The gene variant or variants that are targeted by the ASO drug product should be unique to the trial participant(s) and generally only reported in a small number of patients (typically 1 to 2) in the disease population. If more than a few patients may be candidates for targeted treatment with the ASO drug product, then the ASO is no longer considered individualized, and the sponsor should discuss a drug development plan of the investigational ASO drug product for a larger patient population with the relevant review division.




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