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Aniridia 2(AN2)

MedGen UID:
138010
Concept ID:
C0344543
Congenital Abnormality
Synonyms: AN2; ANIRIDIA II
SNOMED CT: AN2 (253232000); Aniridia type 2 (253232000)
 
Gene (location): ELP4 (11p13)
 
Monarch Initiative: MONDO:0014937
OMIM®: 617141

Clinical features

From HPO
Amblyopia
MedGen UID:
8009
Concept ID:
C0002418
Disease or Syndrome
Reduced visual acuity that is uncorrectable by lenses in the absence of detectable anatomic defects in the eye or visual pathways.
Congenital aniridia
MedGen UID:
1941
Concept ID:
C0003076
Congenital Abnormality
PAX6-related aniridia occurs either as an isolated ocular abnormality or as part of the Wilms tumor-aniridia-genital anomalies-retardation (WAGR) syndrome. Aniridia is a pan ocular disorder affecting the cornea, iris, intraocular pressure (resulting in glaucoma), lens (cataract and lens subluxation), fovea (foveal hypoplasia), and optic nerve (optic nerve coloboma and hypoplasia). Individuals with aniridia characteristically show nystagmus and impaired visual acuity (usually 20/100 - 20/200); however, milder forms of aniridia with subtle iris architecture changes, good vision, and normal foveal structure do occur. Other ocular involvement may include strabismus and occasionally microphthalmia. Although the severity of aniridia can vary between and within families, little variability is usually observed in the two eyes of an affected individual. WAGR syndrome. The risk for Wilms tumor is 42.5%-77%; of those who develop Wilms tumor, 90% do so by age four years and 98% by age seven years. Genital anomalies in males can include cryptorchidism and hypospadias (sometimes resulting in ambiguous genitalia), urethral strictures, ureteric abnormalities, and gonadoblastoma. While females typically have normal external genitalia, they may have uterine abnormalities and streak ovaries. Intellectual disability (defined as IQ <74) is observed in 70%; behavioral abnormalities include attention-deficit/hyperactivity disorder (ADHD), autism spectrum disorder, anxiety, depression, and obsessive-compulsive disorder. Other individuals with WAGR syndrome can have normal intellect without behavioral problems.
Lens subluxation
MedGen UID:
9718
Concept ID:
C0023316
Disease or Syndrome
Partial dislocation of the lens of the eye.
Optic atrophy
MedGen UID:
18180
Concept ID:
C0029124
Disease or Syndrome
Atrophy of the optic nerve. Optic atrophy results from the death of the retinal ganglion cell axons that comprise the optic nerve and manifesting as a pale optic nerve on fundoscopy.
Cataract
MedGen UID:
39462
Concept ID:
C0086543
Disease or Syndrome
A cataract is an opacity or clouding that develops in the crystalline lens of the eye or in its capsule.
Iris coloboma
MedGen UID:
116097
Concept ID:
C0240063
Anatomical Abnormality
A coloboma of the iris.

Professional guidelines

PubMed

Daruich A, Robert MP, Leroy C, DE Vergnes N, Beugnet C, Malan V, Valleix S, Bremond-Gignac D
Am J Ophthalmol 2022 May;237:122-129. Epub 2021 Dec 20 doi: 10.1016/j.ajo.2021.12.007. PMID: 34942114
Nischal KK
Cornea 2015 Oct;34 Suppl 10:S24-34. doi: 10.1097/ICO.0000000000000552. PMID: 26352876
Dua HS, Saini JS, Azuara-Blanco A, Gupta P
Indian J Ophthalmol 2000 Jun;48(2):83-92. PMID: 11116520

Recent clinical studies

Etiology

Li W, He XD, Yang ZT, Han DM, Sun Y, Chen YX, Han XT, Guo SC, Ma YT, Jin X, Yang HM, Gao Y, Wang ZS, Li JK, He W
Invest Ophthalmol Vis Sci 2023 Feb 1;64(2):5. doi: 10.1167/iovs.64.2.5. PMID: 36729443Free PMC Article
Karaconji T, Zagora S, Grigg JR
Clin Exp Ophthalmol 2022 Mar;50(2):232-246. Epub 2022 Jan 25 doi: 10.1111/ceo.14039. PMID: 35023613
Dumitrescu AV, Pfeifer WL, Drack AV
J AAPOS 2021 Aug;25(4):220.e1-220.e8. Epub 2021 Jul 17 doi: 10.1016/j.jaapos.2021.03.015. PMID: 34280564
Wawrocka A, Krawczynski MR
J Appl Genet 2018 May;59(2):151-159. Epub 2018 Feb 19 doi: 10.1007/s13353-017-0426-1. PMID: 29460221Free PMC Article
Nelson LB, Spaeth GL, Nowinski TS, Margo CE, Jackson L
Surv Ophthalmol 1984 May-Jun;28(6):621-42. doi: 10.1016/0039-6257(84)90184-x. PMID: 6330922

Diagnosis

Li W, He XD, Yang ZT, Han DM, Sun Y, Chen YX, Han XT, Guo SC, Ma YT, Jin X, Yang HM, Gao Y, Wang ZS, Li JK, He W
Invest Ophthalmol Vis Sci 2023 Feb 1;64(2):5. doi: 10.1167/iovs.64.2.5. PMID: 36729443Free PMC Article
Karaconji T, Zagora S, Grigg JR
Clin Exp Ophthalmol 2022 Mar;50(2):232-246. Epub 2022 Jan 25 doi: 10.1111/ceo.14039. PMID: 35023613
Shah R, Amador C, Tormanen K, Ghiam S, Saghizadeh M, Arumugaswami V, Kumar A, Kramerov AA, Ljubimov AV
Exp Eye Res 2021 Mar;204:108455. Epub 2021 Jan 21 doi: 10.1016/j.exer.2021.108455. PMID: 33485845Free PMC Article
Ihnatko R, Eden U, Fagerholm P, Lagali N
Ocul Surf 2016 Apr;14(2):196-206. Epub 2015 Dec 29 doi: 10.1016/j.jtos.2015.10.003. PMID: 26738798
Brauner SC, Walton DS, Chen TC
Int Ophthalmol Clin 2008 Spring;48(2):79-85. doi: 10.1097/IIO.0b013e318169314b. PMID: 18427263

Therapy

Magan T, Tanner A, Fajardo-Sanchez J, Lim KS, Goyal S, Rodrigues I, Amaya L, Trikha S, Kulkarni A, Hammond C, Lascaratos G, Yu-Wai-Man C
Eur J Ophthalmol 2022 Sep;32(5):2920-2927. Epub 2022 Jan 10 doi: 10.1177/11206721211073208. PMID: 35001688Free PMC Article
Nishi K, Kamei K, Ogura M, Sato M, Ishiwa S, Shioda Y, Kiyotani C, Matsumoto K, Nozu K, Ishikura K, Ito S
Pediatr Nephrol 2021 Nov;36(11):3699-3709. Epub 2021 May 14 doi: 10.1007/s00467-021-05115-7. PMID: 33988732
Angmo D, Dewan L, Behera A, Gagrani M
Eur J Ophthalmol 2021 Mar;31(2):NP116-NP118. Epub 2019 Jul 25 doi: 10.1177/1120672119866106. PMID: 31342778
Soyugelen Demirok G, Ekşioğlu Ü, Yakın M, Kaderli A, Kaderli ST, Örnek F
Turk J Ophthalmol 2019 Sep 3;49(4):183-187. doi: 10.4274/tjo.galenos.2019.07348. PMID: 31486604Free PMC Article
Young TL
Curr Opin Ophthalmol 2003 Oct;14(5):296-303. doi: 10.1097/00055735-200310000-00011. PMID: 14502058

Prognosis

Pedersen HR, Gilson SJ, Landsend ECS, Utheim ØA, Utheim TP, Baraas RC
Invest Ophthalmol Vis Sci 2023 Apr 3;64(4):18. doi: 10.1167/iovs.64.4.18. PMID: 37067366Free PMC Article
Wang P, Wu P, Wang J, Zeng Y, Jiang Y, Wang Y, Li S, Xiao X, Zhang Q
Invest Ophthalmol Vis Sci 2023 Mar 1;64(3):19. doi: 10.1167/iovs.64.3.19. PMID: 36892533Free PMC Article
Soyugelen Demirok G, Ekşioğlu Ü, Yakın M, Kaderli A, Kaderli ST, Örnek F
Turk J Ophthalmol 2019 Sep 3;49(4):183-187. doi: 10.4274/tjo.galenos.2019.07348. PMID: 31486604Free PMC Article
Adams DJ, Clark DA
Pediatr Clin North Am 2015 Apr;62(2):411-26. Epub 2015 Jan 22 doi: 10.1016/j.pcl.2014.11.005. PMID: 25836705
Lee H, Meyers K, Lanigan B, O'Keefe M
J Pediatr Ophthalmol Strabismus 2010 Jul-Aug;47(4):205-10; quiz 211-2. Epub 2009 Aug 21 doi: 10.3928/01913913-20090818-07. PMID: 20635810

Clinical prediction guides

Pedersen HR, Gilson SJ, Landsend ECS, Utheim ØA, Utheim TP, Baraas RC
Invest Ophthalmol Vis Sci 2023 Apr 3;64(4):18. doi: 10.1167/iovs.64.4.18. PMID: 37067366Free PMC Article
Wang P, Wu P, Wang J, Zeng Y, Jiang Y, Wang Y, Li S, Xiao X, Zhang Q
Invest Ophthalmol Vis Sci 2023 Mar 1;64(3):19. doi: 10.1167/iovs.64.3.19. PMID: 36892533Free PMC Article
Ihnatko R, Eden U, Fagerholm P, Lagali N
Ocul Surf 2016 Apr;14(2):196-206. Epub 2015 Dec 29 doi: 10.1016/j.jtos.2015.10.003. PMID: 26738798
Prasad S, Ram J, Sukhija J, Pandav SS, Gupta PC
Graefes Arch Clin Exp Ophthalmol 2015 May;253(5):739-43. Epub 2015 Jan 16 doi: 10.1007/s00417-014-2908-8. PMID: 25592478
Wang L, He F, Bu J, Zhen Y, Liu X, Du W, Dong J, Cooney JD, Dubey SK, Shi Y, Gong B, Li J, McBride PF, Jia Y, Lu F, Soltis KA, Lin Y, Namburi P, Liang C, Sundaresan P, Paw BH, Li W, Li DY, Phillips JD, Yang Z
Am J Hum Genet 2012 Jan 13;90(1):40-8. Epub 2012 Jan 5 doi: 10.1016/j.ajhg.2011.11.026. PMID: 22226084Free PMC Article

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