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Ability of a Molecule (Prima) to Restore Physiological Differentiation in Epithelium Expressing Gene p63

Terminated
Conditions
Ectodermal Dysplasia
Registration Number
NCT02896387
Lead Sponsor
Fondation Ophtalmologique Adolphe de Rothschild
Brief Summary

Ectodermal dysplasia associated with p63 is a rare disease which, in addition to limbic abnormalities, primarily affects the skin and cornea. The most common forms are called Ectrodactyly, Ectodermal dysplasia, palate Key for cleft lip and palate (EEC) and Ankyloblepharon, Ectodermal dysplasia, cleft lip and palate (AEC). Apart from symptomatic treatment, no cure is available. To understand the molecular defects associated with this disease and to identify therapeutic tools, a research team modelized the disease by reprograming EEC and AEC patient fibroblasts in pluripotent stem cells (iPSC), then induced iPSC differentiation in patients and controls epidermal (skin) and limbic (cornea) cells and demonstrated that the mutated cells can reproduce in vitro the abnormalities observed in patients.

P63 gene belongs to the family of p53 gene. The functions of the two proteins are very similar. Data suggest that molecule Prima could reactivate the p63 protein mutated in patients and thus alleviate skin defect healing and limbic regeneration.

Detailed Description

Not available

Recruitment & Eligibility

Status
TERMINATED
Sex
All
Target Recruitment
5
Inclusion Criteria
  • Patient with genetic pathology of the ocular surface
  • Age ≥ 7 years
Exclusion Criteria
  • Agonal glaucoma
  • low vision mostly related to retinal pathology
  • Pregnant or breast-feeding patient

Study & Design

Study Type
OBSERVATIONAL
Study Design
Not specified
Primary Outcome Measures
NameTimeMethod
the differentiation of corneal epithelium by histological observationbaseline

the differentiation of corneal epithelium by histological observation

Secondary Outcome Measures
NameTimeMethod

Trial Locations

Locations (2)

Fondation Opthalmologique A de Rothschild

🇫🇷

Paris, France

Hopital Necker

🇫🇷

Paris, France

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