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Rare Diseases: Natural History Studies for Drug Development: Draft Guidance for Industry

DraftOffice of the Commissioner,Office of Clinical Policy and Programs,Office of Orphan Products Development Center for Drug Evaluation and Research Center for Biologics Evaluation and Research03/25/2019

Description

The Food and Drug Administration (FDA or Agency) is announcing the availability of a draft guidance for industry entitled “Rare Diseases: Natural History Studies for Drug Development.” FDA is publishing this draft guidance to help inform the design and implementation of natural history studies that can be used to support the development of safe and effective drugs and biological products for rare diseases. A natural history study collects information about the natural history of a disease in the absence of an intervention, from the disease's onset until either its resolution or the individual's death. Although knowledge of a disease's natural history can benefit drug development for many disorders and conditions, natural history information is usually not available or is incomplete for most rare diseases; therefore, natural history information is particularly needed for these diseases.

Scope & Applicability

Product Classes

4
Drug

Defined in section 201(g) of the FD&C Act; Articles intended for use in diagnosis, cure, mitigation, treatment, or prevention of disease; Product classification based on chemical action or specific intended uses like altering pH

Biological products

development program for drug and biological products

Rare Diseases

A higher degree of uncertainty is common in drug development programs for rare diseases.

Rare Disease

drug development for a rare disease; FDA encourages the sponsor to meet with the relevant drug review division about the use of natural history data to support development of a particular drug for rare disease.

Stakeholders

4
IRB

Institutional Review Board providing study approvals

Institutional Review Board

Governs top dose in clinical studies

Sponsor

Entity responsible for submitting applications under section 524B

Statistician

Consulted by lead reviewer for checklist completion

Regulatory Context

Attributes

3
External control

potential use of natural history data as an external control in a clinical trial

Inter-rater reliability

Methods used for standardizing inter- and intra-rater reliability

Phenotypic Heterogeneity

Variation in disease presentation across patient populations

Identified Hazards

Hazards

1
Length-biased sampling

disproportionately fail to capture patients who enter and rapidly leave the database

Related CFR Sections (3)

Related Warning Letters (10)

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  • Clinical Investigator

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  • Institutional Review Board (IRB)

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    Massachusetts Institute of Technology MIT

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  • Clinical Investigator (Sponsor)

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  • Institutional Review Board (IRB)

    New York State Psychiatric Institute IRB

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See Also (8)