Three new PAX6 mutations including one causing an unusual ophthalmic phenotype associated with neurodevelopmental abnormalities
2007

New PAX6 Mutations Linked to Eye and Brain Abnormalities

Sample size: 78 publication Evidence: moderate

Author Information

Author(s): Dansault Anouk, David Gabriel, Schwartz Claire, Jaliffa Carolina, Vieira Véronique, de la Houssaye Guillaume, Bigot Karine, Catin Françise, Tattu Laurent, Chopin Catherine, Halimi Philippe, Roche Olivier, Van Regemorter Nicole, Munier Francis, Schorderet Daniel, Dufier Jean-Louis, Marsac Cécile, Ricquier Daniel, Menasche Maurice, Penfornis Alfred, Abitbol Marc

Primary Institution: Université René Descartes-Paris V

Hypothesis

PAX6 mutations may cause a range of ocular and neurodevelopmental abnormalities.

Conclusion

The study identified three novel PAX6 mutations associated with various ocular phenotypes and neurodevelopmental issues.

Supporting Evidence

  • Three novel heterozygous mutations were identified in unrelated families.
  • The L46P mutation was linked to bilateral microphthalmia and cataracts.
  • The S74G mutation was associated with cognitive impairments and neurological issues.

Takeaway

Scientists found new changes in a gene called PAX6 that can cause eye problems and affect how the brain develops.

Methodology

Patients were phenotyped and the PAX6 gene was screened by direct sequencing.

Limitations

The study may have overlooked mutations outside the studied genomic region.

Participant Demographics

Participants included 78 individuals aged 0 to 73, with various ocular and neurological conditions.

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