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RecruitingNCT00359684Updated Sep 16, 2026

Use of Cysteamine in the Treatment of Cystinosis

An observational study in Cystinosis, sponsored by National Human Genome Research Institute (NHGRI). Recruiting at 1 site in United States. Open to participants aged 1 Week to 115 Years. Per ClinicalTrials.gov, last updated 2026-09-16.

Sponsored by National Human Genome Research Institute (NHGRI) · Observational

Study type
Observational
Model
Cohort
Time perspective
Prospective
Enrollment
330
Ages
1 Week to 115 Years
Sex
All
01

Study summary

Cystinosis is an inherited disease resulting in poor growth and kidney failure. There is no known cure for cystinosis, although kidney transplantation may help the renal failure and prolong survival. Both the kidney damage and growth failure are thought to be due to the accumulation of the amino acid cystine within the cells of the body. The cystine storage later damages other organs besides the kidneys, including the thyroid gland, pancreas, eyes, and muscle.

The drug cysteamine (Cystagon; ProCysBi) is an oral medication given to patients with cystinosis prior to kidney transplantation. The drug works by reducing the level of cystine in the white blood cells and muscle tissue. The drug may also decrease levels of cystine in the kidneys and other tissues.

This study has several goals:

  1. Long-term surveillance of cysteamine treated patients.
  2. Detection of new non-kidney complications of cystinosis.
  3. Maintenance of a patient population for genetic testing (mutational analysis) of the cystinosis gene.\<TAB>
Read the detailed description

Patients with nephropathic cystinosis have been treated with the cystine-depleting agent cysteamine since 1978. This therapy prevents or delays renal deterioration, improves growth, and depletes parenchymal tissues of cystine. Based largely upon data produced through this protocol, the Food and Drug Administration approved cysteamine bitartrate for use in cystinosis patients on August 15, 1994. Cysteamine is available as CystagonR through Mylan Pharmaceuticals in 50 mg and 150 mg capsules and as ProcysbiR in 75 mg capsules. By virtue of the current protocol, patients are admitted to the NIH Clinical Center for investigations every two years, except for cases of great interest or urgency. On each 1-3 day admission, a battery of tests is performed and the adequacy of cystine depletion by cysteamine is monitored. This protocol demonstrates the course of cystinosis patients treated with cysteamine, describes new complications of the disorder in poorly treated adults, and maintains NHGRI expertise in the field. Its monitoring and followup of patients over the course of 3 decades represents an invaluable contribution to our understanding of the natural history of this rare disease.

02

Conditions studied

  • Cystinosis

Keywords

  • Cystinosis
  • Cystine
  • Lysomal Storage Disease
  • Mutation Analysis
  • Metabolic Disease
  • Natural History
03

Who can participate

Ages eligible
1 Week to 115 Years
Sexes eligible
All
Accepts healthy volunteers
No
Sampling method
Non-probability sample

Study population

Patients with a diagnosis of cystinosis

Inclusion criteria

Diagnosis of cystinosis, whether classical or one of the variants with later onset or no renal complications.

Patients will be diagnosed as having cystinosis based upon a leucocyte cystine content greater than 1 nmol half-cystine/mg protein (normal, less than 0.2) and a typical clinical course.

Exclusion criteria

EXCLUSION CRITERIA:

Inability to travel to the NIH.

Age less than one week.

Nonviable neonates and neonates of uncertain viability will be excluded.

04

Study design

Observational model
Cohort
Time perspective
Prospective
Enrollment
330 participants (estimated)

Groups and cohorts

  • Cystinosis

    Patients with a diagnosis of cystinosis

    Drug: Cysteamine

Interventions

  • DrugCysteamine

    Cystine-depleting agent

05

What researchers measure

Primary outcomes

  1. Serve as a source of knowledge and advice for individual cystinosis patients and for the community at large

    Serve as a source of knowledge and advice for individual cystinosis patients and for the community at large

    Time frame: Follow-up can occur every two years

06

Study locations

1 of 1 sites recruiting
  • National Institutes of Health Clinical Center
    Bethesda, Maryland 20892, United States
    Recruiting
07

References and documents

Publications

  • Florenzano P, Ferreira C, Nesterova G, Roberts MS, Tella SH, de Castro LF, Brown SM, Whitaker A, Pereira RC, Bulas D, Gafni RI, Salusky IB, Gahl WA, Collins MT. Skeletal Consequences of Nephropathic Cystinosis. J Bone Miner Res. 2018 Oct;33(10):1870-1880. doi: 10.1002/jbmr.3522. Epub 2018 Jul 20. PubMed 29905968 ↗

Individual participant data

Plan to share: Yes — pending

Supporting information: Study protocol

08

Registry details

Key details

Study ID
NCT00359684
Lead sponsor
National Human Genome Research Institute (NHGRI)
Responsible party
Sponsor
First posted
Aug 2, 2006
Start date
Jan 4, 1979
Last update
Sep 16, 2026

Study contacts

William A Gahl, M.D.
Contact
gahlw@mail.nih.gov
(301) 402-2739
William A Gahl, M.D.
principal investigator · National Human Genome Research Institute (NHGRI)

Oversight

FDA-regulated drug
No
FDA-regulated device
No
View the source record on ClinicalTrials.gov ↗

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