Aniridia: A Panocular Developmental Malformation

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Introduction:
Aniridia (OMIM 106210) is a Panocular developmental malformation with complete or partial Iris hypoplasia being major feature of spectrum.(1) The prevalence of Aniridia (AN) is 1:40,000 to 1:100,000 without any known predilections for sex or gender.(1-3) The spectrum involves not only iris, but also the cornea, lens, optic nerve and fovea.(4)
AN can be familial with Autosomal Dominant Inheritance (AD) in two third of cases, while sporadic in rest of the patients. It can occur as an Isolated Ocular malformation without any systemic involvement or as part of Wilms tumor, Aniridia, Genital Anomalies, Retardation (WAGR) syndrome.(4, 5) AN results from mutations in the Paired Box gene-6 (PAX-6) located on chromosome 11p13, which encodes a highly conserved transcriptional regulator with two DNA binding and a transcriptional trans-activation domain.(6, 7) It is essential for oculogenesis, Central Nervous System (CNS), olfactory system(7) and endocrine glands.(8) Further, it plays multiple roles in development of the cornea, iris, lens and retina during ocular morphogenesis(7, 8) by regulating PAX6 itself, PAX2 , SRY -box 2 (SOX2) gene and a series of retinal transcription factors and structural proteins including crystallins of lens.(9-13) The PAX6 database currently documents 826 total variants and 357 unique DNA sequence changes.(14) Documented mutations in PAX6 include Premature Termination Codon (PTC), C-terminal extensions (CTE) and missense mutations.(14, 15)
Aniridia may be the initial manifestation of WAGR syndrome where deletion of (Wilms tumor 1) WT1 gene deletion in chromosome 11p13 gives rise to the phenotype.(16) WAGR syndrome results from a constitutional complete or partial deletion of 11p13 leading to conc...

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30. Defreyn A, Maugery J, Chabrier S, Coullet J. [Gillespie syndrome: an uncommon presentation of congenital aniridia]. Journal francais d'ophtalmologie. 2007;30(1):e1. Epub 2007/02/09. Syndrome de Gillespie: un cas rare d'aniridie congenitale.
31. Roy FH. Aniridia, Cerebellar Ataxia, and Mental Deficiency (Gillespie Syndrome). Ocular Syndromes and Systemic Diseases. 4th ed. USA: MedRounds; 2007. p. 18.

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