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Cataract 13 with adult I phenotype(CTRCT13)

MedGen UID:
811703
Concept ID:
C3805373
Disease or Syndrome
Synonyms: CATARACT 13 WITH ADULT i PHENOTYPE; CTRCT13
 
Gene (location): GCNT2 (6p24.3-24.2)
 
Monarch Initiative: MONDO:0007289
OMIM®: 116700

Definition

The i and I antigens of the I blood group system (110800) are carbohydrate structures carried on glycolipids and glycoproteins and are characterized as straight or branched glycochains composed of repeating N-acetyllactosamine (LacNAc) units, respectively. Conversion of i antigen into an I-active structure requires the activity of the I-branching enzyme, beta-1,6-N-acetylglucosaminyltransferase (GCNT2; 600429), which adds the decisive GlcNAc-beta-1-6 branch onto the straight poly-LacNAc chains. Expression of the i and I antigens on red blood cells (RBCs) is reciprocal and developmentally regulated. Adult human RBCs predominantly express I antigen, whereas fetal and neonatal RBCs predominantly express i antigen. After birth, I antigen levels increase gradually as i antigen levels fall, with the normal Ii status of adult RBCs reached after about 13 to 20 months. Mutations that specifically affect 1 of the 3 variants produced by the GCNT2 gene cause the rare adult i phenotype (see 110800), in which adult RBCs are rich in i antigen and contain low levels of I antigen. Mutations that eliminate all 3 GCNT2 variants cause the adult i phenotype with congenital cataract (review by Yu and Lin, 2011). [from OMIM]

Clinical features

From HPO
Developmental cataract
MedGen UID:
3202
Concept ID:
C0009691
Congenital Abnormality
A cataract that occurs congenitally as the result of a developmental defect, in contrast to the majority of cataracts that occur in adulthood as the result of degenerative changes of the lens.

Professional guidelines

PubMed

Iovieno A, Gore DM, Carnt N, Dart JK
Ophthalmology 2014 Dec;121(12):2340-7. Epub 2014 Aug 2 doi: 10.1016/j.ophtha.2014.06.033. PMID: 25097155
Wallace DM, Clark AF, Lipson KE, Andrews D, Crean JK, O'Brien CJ
Invest Ophthalmol Vis Sci 2013 Dec 2;54(13):7836-48. doi: 10.1167/iovs.13-12494. PMID: 24204045

Recent clinical studies

Etiology

Nguyen XT, Almushattat H, Strubbe I, Georgiou M, Li CHZ, van Schooneveld MJ, Joniau I, De Baere E, Florijn RJ, Bergen AA, Hoyng CB, Michaelides M, Leroy BP, Boon CJF
Genes (Basel) 2021 Sep 11;12(9) doi: 10.3390/genes12091404. PMID: 34573385Free PMC Article
Lipiński P, Stawiński P, Rydzanicz M, Wypchło M, Płoski R, Stradomska TJ, Jurkiewicz E, Ferdinandusse S, Wanders RJA, Vaz FM, Tylki-Szymańska A
J Appl Genet 2020 Feb;61(1):87-91. Epub 2019 Oct 18 doi: 10.1007/s13353-019-00523-w. PMID: 31628608Free PMC Article
Stelten BML, van de Warrenburg BPC, Wevers RA, Verrips A
Parkinsonism Relat Disord 2019 Jan;58:12-16. Epub 2018 Jul 19 doi: 10.1016/j.parkreldis.2018.07.006. PMID: 30054180
Lee J, Scanga HL, Dansingani KK, Taubenslag KJ, Zlotcavitch L, Chauhan BK, Sylvester CL, Morton DH, Nischal KK
Ophthalmic Genet 2018 Dec;39(6):735-740. Epub 2018 Nov 16 doi: 10.1080/13816810.2018.1547913. PMID: 30444160
Faivre L, Dollfus H, Lyonnet S, Alembik Y, Mégarbané A, Samples J, Gorlin RJ, Alswaid A, Feingold J, Le Merrer M, Munnich A, Cormier-Daire V
Am J Med Genet A 2003 Dec 1;123A(2):204-7. doi: 10.1002/ajmg.a.20289. PMID: 14598350

Diagnosis

Bell S, Malka S, Lloyd IC, Moosajee M
Genes (Basel) 2021 Jan 21;12(2) doi: 10.3390/genes12020131. PMID: 33494148Free PMC Article
Stelten BML, van de Warrenburg BPC, Wevers RA, Verrips A
Parkinsonism Relat Disord 2019 Jan;58:12-16. Epub 2018 Jul 19 doi: 10.1016/j.parkreldis.2018.07.006. PMID: 30054180
Lee J, Scanga HL, Dansingani KK, Taubenslag KJ, Zlotcavitch L, Chauhan BK, Sylvester CL, Morton DH, Nischal KK
Ophthalmic Genet 2018 Dec;39(6):735-740. Epub 2018 Nov 16 doi: 10.1080/13816810.2018.1547913. PMID: 30444160
Sekijima Y, Koyama S, Yoshinaga T, Koinuma M, Inaba Y
J Hum Genet 2018 Mar;63(3):271-280. Epub 2018 Jan 10 doi: 10.1038/s10038-017-0389-4. PMID: 29321515
Thomas S, Thomas MG, Andrews C, Chan WM, Proudlock FA, McLean RJ, Pradeep A, Engle EC, Gottlob I
Eur J Hum Genet 2014 Mar;22(3):344-9. Epub 2013 Aug 14 doi: 10.1038/ejhg.2013.162. PMID: 23942204Free PMC Article

Therapy

Amudhavalli SM, Hanson R, Angle B, Bontempo K, Gripp KW
Am J Med Genet A 2018 Jul;176(7):1648-1656. doi: 10.1002/ajmg.a.38832. PMID: 30160832
Chiquet C, Musson C, Aptel F, Boisset S, Maurin M
Am J Ophthalmol 2018 Jul;191:76-82. Epub 2018 Apr 11 doi: 10.1016/j.ajo.2018.03.042. PMID: 29654738
Iovieno A, Gore DM, Carnt N, Dart JK
Ophthalmology 2014 Dec;121(12):2340-7. Epub 2014 Aug 2 doi: 10.1016/j.ophtha.2014.06.033. PMID: 25097155
Wallace DM, Clark AF, Lipson KE, Andrews D, Crean JK, O'Brien CJ
Invest Ophthalmol Vis Sci 2013 Dec 2;54(13):7836-48. doi: 10.1167/iovs.13-12494. PMID: 24204045
Sundaresan P, Ravindran RD, Vashist P, Shanker A, Nitsch D, Talwar B, Maraini G, Camparini M, Nonyane BA, Smeeth L, Chakravarthy U, Hejtmancik JF, Fletcher AE
PLoS One 2012;7(3):e33001. Epub 2012 Mar 8 doi: 10.1371/journal.pone.0033001. PMID: 22412971Free PMC Article

Prognosis

Hernández-Da Mota S, Lima-Gómez V, Rodríguez-Ayala E, Fromow-Guerra JJ, Melo-Granados EAR
Gac Med Mex 2019;155(5):493-498. doi: 10.24875/GMM.19004992. PMID: 31695225
Jin H, Zhu B, Liu X, Jin J, Zou H
J Pharm Biomed Anal 2019 Sep 10;174:414-421. Epub 2019 Jun 10 doi: 10.1016/j.jpba.2019.06.013. PMID: 31212142
Sekijima Y, Koyama S, Yoshinaga T, Koinuma M, Inaba Y
J Hum Genet 2018 Mar;63(3):271-280. Epub 2018 Jan 10 doi: 10.1038/s10038-017-0389-4. PMID: 29321515
Thomas S, Thomas MG, Andrews C, Chan WM, Proudlock FA, McLean RJ, Pradeep A, Engle EC, Gottlob I
Eur J Hum Genet 2014 Mar;22(3):344-9. Epub 2013 Aug 14 doi: 10.1038/ejhg.2013.162. PMID: 23942204Free PMC Article
Yu LC, Twu YC, Chang CY, Lin M
Blood 2001 Dec 15;98(13):3840-5. doi: 10.1182/blood.v98.13.3840. PMID: 11739194

Clinical prediction guides

Nguyen XT, Almushattat H, Strubbe I, Georgiou M, Li CHZ, van Schooneveld MJ, Joniau I, De Baere E, Florijn RJ, Bergen AA, Hoyng CB, Michaelides M, Leroy BP, Boon CJF
Genes (Basel) 2021 Sep 11;12(9) doi: 10.3390/genes12091404. PMID: 34573385Free PMC Article
Chen JW, Zhao L, Zhang F, Li L, Gu YH, Zhou JY, Zhang H, Meng M, Zhang KH, Le WD, Dong CB
Chin Med J (Engl) 2015 Jul 5;128(13):1714-23. doi: 10.4103/0366-6999.159340. PMID: 26112709Free PMC Article
Thomas S, Thomas MG, Andrews C, Chan WM, Proudlock FA, McLean RJ, Pradeep A, Engle EC, Gottlob I
Eur J Hum Genet 2014 Mar;22(3):344-9. Epub 2013 Aug 14 doi: 10.1038/ejhg.2013.162. PMID: 23942204Free PMC Article
Zhang Q, Guo X, Xiao X, Yi J, Jia X, Hejtmancik JF
Mol Vis 2004 Nov 17;10:890-900. PMID: 15570218
Toutain A, Ayrault AD, Moraine C
Am J Med Genet 1997 Aug 22;71(3):305-14. doi: 10.1002/(sici)1096-8628(19970822)71:3<305::aid-ajmg11>3.0.co;2-o. PMID: 9268101

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