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Coloboma, ocular, autosomal dominant(MCOPCB12)

MedGen UID:
946399
Concept ID:
CN263371
Disease or Syndrome
Synonyms: MCOPCB12; Microphthalmia/coloboma 12
 
Gene (location): PAX6 (11p13)
 
Monarch Initiative: MONDO:0007350
OMIM®: 120200

Definition

Coloboma is an ocular birth defect resulting from abnormal development of the eye during embryogenesis. It is defined as a congenital defect in any ocular tissue, typically presenting as absent tissue or a gap, at a site consistent with aberrant closure of the optic fissure. Failure of fusion can lead to coloboma of one or multiple regions of the inferior portion of the eye affecting any part of the globe traversed by the fissure, from the iris to the optic nerve, including the ciliary body, retina, and choroid. Coloboma is also frequently associated with small (microphthalmic) or absent (anophthalmic) eyes as part of an interrelated spectrum of developmental eye anomalies, and can affect either one or both eyes (summary by Kelberman et al., 2014). Microphthalmia/coloboma-12 (MCOPCB12) is characterized by inter- and intrafamilial variability. In addition to microphthalmia and coloboma, other ocular anomalies include iris hypoplasia, aphakia or small lens, lens subluxation, congenital cataract, microcornea, and sclerocornea. Some patients also exhibit neurodevelopmental anomalies (Deml et al., 2016; Williamson et al., 2020). For a discussion of genetic heterogeneity of colobomatous microphthalmia, see MCOPCB1 (300345). [from OMIM]

Professional guidelines

PubMed

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Recent clinical studies

Etiology

Dana J, Dorval G, Martin CS, Belhous K, Levy R, Marlin S, De Bie I, Mautret-Godefroy M, Rausell A, Rio M, Boucher-Brischoux E, Attié-Bitach T, Boddaert N, Pingault V
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Diagnosis

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Marszałek-Kruk BA, Wójcicki P, Dowgierd K, Śmigiel R
Genes (Basel) 2021 Sep 9;12(9) doi: 10.3390/genes12091392. PMID: 34573374Free PMC Article
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Therapy

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Prognosis

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Clinical prediction guides

Dana J, Dorval G, Martin CS, Belhous K, Levy R, Marlin S, De Bie I, Mautret-Godefroy M, Rausell A, Rio M, Boucher-Brischoux E, Attié-Bitach T, Boddaert N, Pingault V
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Recent systematic reviews

Mahmoud A, Pomar L, Lambert V, Picone O, Hcini N
Ocul Immunol Inflamm 2024 Nov;32(9):2217-2227. Epub 2024 Feb 13 doi: 10.1080/09273948.2024.2314086. PMID: 38350011
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Clin Genet 2023 Feb;103(2):146-155. Epub 2022 Oct 17 doi: 10.1111/cge.14243. PMID: 36203321
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Rooijers W, Caron CJJM, Loudon SE, Padwa BL, Dunaway DJ, Forrest CR, Koudstaal MJ
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Amirhassankhani S, Lloyd MS
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