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Polymorphous corneal dystrophy(PPCD)

MedGen UID:
87382
Concept ID:
C0339284
Disease or Syndrome
Synonyms: CORNEAL DYSTROPHY, HEREDITARY POLYMORPHOUS POSTERIOR; Posterior polymorphous corneal dystrophy
SNOMED CT: Posterior polymorphous corneal dystrophy (29504002); Polymorphous corneal dystrophy (29504002)
 
Related genes: GRHL2, OVOL2, ZEB1, COL8A2
 
HPO: HP:0007915
Monarch Initiative: MONDO:0020364
OMIM®: 122000
OMIM® Phenotypic series: PS122000

Definition

Posterior polymorphous corneal dystrophy (PPCD) is a rare disorder involving metaplasia and overgrowth of corneal endothelial cells (Krafchak et al., 2005). In patients with PPCD, these cells manifest in an epithelial morphology and gene expression pattern, produce an aberrant basement membrane, and, sometimes, spread over the iris and nearby structures in a way that increases the risk for glaucoma. Symptoms can range from very aggressive to asymptomatic and nonprogressive, even within the same family. The age of diagnosis is, most often, in the second or third decade of life. Clinically, PPCD is characterized by vesicles, bands, and polymorphous opacities at the level of the Descemet membrane and corneal endothelium. Peripheral anterior iris adhesions, iris atrophy, pupillary ectropion, and corectopia may also develop. Occasional severe visual disability results from secondary glaucoma or corneal edema. On ultrastructural examination, corneal endothelial cells show fibroblastic and epithelial-like transformation (summary by Liskova et al., 2012). Genetic Heterogeneity of Posterior Polymorphous Corneal Dystrophy Other forms of PPCD include PPCD2 (609140), caused by mutation in the COL8A2 gene (120252) on chromosome 1p34.3; PPCD3 (609141), caused by mutation in the ZEB1 gene (189909) on chromosome 10p; and PPCD4 (618031), caused by mutation in the GRHL2 gene (608576) on chromosome 8q22. [from OMIM]

Conditions with this feature

Posterior polymorphous corneal dystrophy 1
MedGen UID:
343836
Concept ID:
C1852555
Disease or Syndrome
A posterior polymorphous corneal dystrophy that has material basis in autosomal dominant inheritance of mutation in the OVOL2 gene on chromosome 20p11.23.

Professional guidelines

PubMed

Lechner J, Dash DP, Muszynska D, Hosseini M, Segev F, George S, Frazer DG, Moore JE, Kaye SB, Young T, Simpson DA, Churchill AJ, Héon E, Willoughby CE
Invest Ophthalmol Vis Sci 2013 May 3;54(5):3215-23. doi: 10.1167/iovs.13-11781. PMID: 23599324

Recent clinical studies

Etiology

Kannabiran C, Chaurasia S, Ramappa M, Mootha VV
Indian J Ophthalmol 2022 Jul;70(7):2239-2248. doi: 10.4103/ijo.IJO_992_22. PMID: 35791103Free PMC Article
Elhusseiny AM, Saeed HN
Cornea 2022 Jun 1;41(6):734-739. Epub 2021 Aug 30 doi: 10.1097/ICO.0000000000002847. PMID: 34469341
Davidson AE, Hafford-Tear NJ, Dudakova L, Sadan AN, Pontikos N, Hardcastle AJ, Tuft SJ, Liskova P
Eur J Hum Genet 2020 Jan;28(1):126-131. Epub 2019 Jun 14 doi: 10.1038/s41431-019-0448-8. PMID: 31201376Free PMC Article
Woreta FA, Davis GW, Bower KS
Surv Ophthalmol 2015 Mar-Apr;60(2):115-22. Epub 2014 Aug 23 doi: 10.1016/j.survophthal.2014.08.003. PMID: 25307289
Raber IM, Fintelmann R, Chhabra S, Ribeiro MP, Eagle RC Jr, Orlin SE
Cornea 2011 Oct;30(10):1120-4. doi: 10.1097/ICO.0b013e3182114452. PMID: 21760500

Diagnosis

Safran JP, Hammersmith KM, Milman T
Ophthalmology 2023 Dec;130(12):1354. Epub 2023 Apr 12 doi: 10.1016/j.ophtha.2023.03.003. PMID: 37055288
Kannabiran C, Chaurasia S, Ramappa M, Mootha VV
Indian J Ophthalmol 2022 Jul;70(7):2239-2248. doi: 10.4103/ijo.IJO_992_22. PMID: 35791103Free PMC Article
Elhusseiny AM, Saeed HN
Cornea 2022 Jun 1;41(6):734-739. Epub 2021 Aug 30 doi: 10.1097/ICO.0000000000002847. PMID: 34469341
Davidson AE, Hafford-Tear NJ, Dudakova L, Sadan AN, Pontikos N, Hardcastle AJ, Tuft SJ, Liskova P
Eur J Hum Genet 2020 Jan;28(1):126-131. Epub 2019 Jun 14 doi: 10.1038/s41431-019-0448-8. PMID: 31201376Free PMC Article
Weiss JS, Møller HU, Aldave AJ, Seitz B, Bredrup C, Kivelä T, Munier FL, Rapuano CJ, Nischal KK, Kim EK, Sutphin J, Busin M, Labbé A, Kenyon KR, Kinoshita S, Lisch W
Cornea 2015 Feb;34(2):117-59. doi: 10.1097/ICO.0000000000000307. PMID: 25564336

Therapy

Krolo I, Kasumović A, Radman I, Pavić P
Eur J Ophthalmol 2021 Jul;31(4):1584-1587. Epub 2021 Feb 25 doi: 10.1177/1120672121997672. PMID: 33631984
Rocha-de-Lossada C, Rachwani-Anil R, Colmenero-Reina E, Borroni D, Sánchez-González JM
J Cataract Refract Surg 2021 May 1;47(5):662-670. doi: 10.1097/j.jcrs.0000000000000468. PMID: 33149045
Bhullar PK, Venkateswaran N, Kim T
Cornea 2021 May 1;40(5):652-655. doi: 10.1097/ICO.0000000000002514. PMID: 33065714
Woreta FA, Davis GW, Bower KS
Surv Ophthalmol 2015 Mar-Apr;60(2):115-22. Epub 2014 Aug 23 doi: 10.1016/j.survophthal.2014.08.003. PMID: 25307289
Boruchoff SA, Weiner MJ, Albert DM
Am J Ophthalmol 1990 Mar 15;109(3):323-8. doi: 10.1016/s0002-9394(14)74559-3. PMID: 2309866

Prognosis

Nicklason E, Mack H, Beltz J, Jacob J, Farahani M, Colville D, Savige J
Ophthalmic Genet 2020 Feb;41(1):13-19. Epub 2020 Mar 11 doi: 10.1080/13816810.2019.1709126. PMID: 32159412
Davidson AE, Hafford-Tear NJ, Dudakova L, Sadan AN, Pontikos N, Hardcastle AJ, Tuft SJ, Liskova P
Eur J Hum Genet 2020 Jan;28(1):126-131. Epub 2019 Jun 14 doi: 10.1038/s41431-019-0448-8. PMID: 31201376Free PMC Article
Sorkin N, Einan-Lifshitz A, Boutin T, Showail M, Borovik A, Chan CC, Rootman DS
Can J Ophthalmol 2019 Apr;54(2):190-195. Epub 2018 Aug 23 doi: 10.1016/j.jcjo.2018.05.012. PMID: 30975342
Chaurasia S, Mittal R, Bichappa G, Ramappa M, Murthy SI
Int Ophthalmol 2017 Aug;37(4):945-952. Epub 2016 Sep 22 doi: 10.1007/s10792-016-0360-y. PMID: 27658681
Savige J, Sheth S, Leys A, Nicholson A, Mack HG, Colville D
Clin J Am Soc Nephrol 2015 Apr 7;10(4):703-9. Epub 2015 Feb 3 doi: 10.2215/CJN.10581014. PMID: 25649157Free PMC Article

Clinical prediction guides

Davidson AE, Hafford-Tear NJ, Dudakova L, Sadan AN, Pontikos N, Hardcastle AJ, Tuft SJ, Liskova P
Eur J Hum Genet 2020 Jan;28(1):126-131. Epub 2019 Jun 14 doi: 10.1038/s41431-019-0448-8. PMID: 31201376Free PMC Article
Savige J, Sheth S, Leys A, Nicholson A, Mack HG, Colville D
Clin J Am Soc Nephrol 2015 Apr 7;10(4):703-9. Epub 2015 Feb 3 doi: 10.2215/CJN.10581014. PMID: 25649157Free PMC Article
Del Turco C, Pierro L, Querques G, Gagliardi M, Corvi F, Manitto MP, Bandello FM
Eur J Ophthalmol 2015 Mar-Apr;25(2):177-9. Epub 2014 Oct 8 doi: 10.5301/ejo.5000526. PMID: 25363852
Nguyen DQ, Hosseini M, Billingsley G, Héon E, Churchill AJ
Acta Ophthalmol 2010 Sep;88(6):695-9. doi: 10.1111/j.1755-3768.2009.01511.x. PMID: 19432861
Cremona FA, Ghosheh FR, Laibson PR, Rapuano CJ, Cohen EJ
Cornea 2008 Apr;27(3):374-7. doi: 10.1097/ICO.0b013e31815c18fa. PMID: 18362674

Recent systematic reviews

Rocha-de-Lossada C, Rachwani-Anil R, Colmenero-Reina E, Borroni D, Sánchez-González JM
J Cataract Refract Surg 2021 May 1;47(5):662-670. doi: 10.1097/j.jcrs.0000000000000468. PMID: 33149045

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