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Posterior polymorphous corneal dystrophy 3(PPCD3)

MedGen UID:
322978
Concept ID:
C1836724
Disease or Syndrome
Synonym: PPCD3
 
Gene (location): ZEB1 (10p11.22)
 
Monarch Initiative: MONDO:0012200
OMIM®: 609141

Definition

Posterior polymorphous corneal dystrophy-3 (PPCD3) 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 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. PPCD3 is often associated with corneal steepening, and some patients may be diagnosed with keratoconus before PPCD (Fernandez-Gutierrez et al., 2023). Retrocorneal membranes have been reported, sometimes extending onto the lens (Moroi et al., 2003). For a discussion of genetic heterogeneity of posterior polymorphous corneal dystrophy, see PPCD1 (122000). [from OMIM]

Clinical features

From HPO
Inguinal hernia
MedGen UID:
6817
Concept ID:
C0019294
Finding
Protrusion of the contents of the abdominal cavity through the inguinal canal.
Corneal dystrophy
MedGen UID:
3619
Concept ID:
C0010036
Disease or Syndrome
The term corneal dystrophy embraces a heterogenous group of bilateral genetically determined non-inflammatory corneal diseases that are restricted to the cornea.
Keratoconus
MedGen UID:
44015
Concept ID:
C0022578
Disease or Syndrome
A cone-shaped deformity of the cornea characterized by the presence of corneal distortion secondary to thinning of the apex.
Reduced visual acuity
MedGen UID:
65889
Concept ID:
C0234632
Finding
Diminished clarity of vision.
Corneal guttata
MedGen UID:
488833
Concept ID:
C0271288
Finding
Corneal guttata are droplet-like accumulations of non-banded collagen on the posterior surface of Descemet's membrane. The presence of focal thickenings of Descemet's membrane histologically named guttae. Cornea guttata can be easily diagnosed in vivo and ex vivo by means of specular microscopy as it gives dark areas where no endothelial cells are visible.
Congenital ectopic pupil
MedGen UID:
224790
Concept ID:
C1271219
Congenital Abnormality
Ectopia pupillae is a congenital eye malformation in which the pupils are displaced from their normal central position.

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

Diagnosis

Jang MS, Roldan AN, Frausto RF, Aldave AJ
Vision Res 2014 Jul;100:88-92. Epub 2014 Apr 26 doi: 10.1016/j.visres.2014.04.007. PMID: 24780443Free PMC Article
Liskova P, Palos M, Hardcastle AJ, Vincent AL
JAMA Ophthalmol 2013 Oct;131(10):1296-303. doi: 10.1001/jamaophthalmol.2013.405. PMID: 23807282
Mehta JS, Vithana EN, Tan DT, Yong VH, Yam GH, Law RW, Chong WG, Pang CP, Aung T
Invest Ophthalmol Vis Sci 2008 Jan;49(1):184-8. doi: 10.1167/iovs.07-0847. PMID: 18172091

Therapy

Zakharevich M, Kattan JM, Chen JL, Lin BR, Cervantes AE, Chung DD, Frausto RF, Aldave AJ
Mol Vis 2017;23:740-752. Epub 2017 Oct 14 PMID: 29046608Free PMC Article

Prognosis

Chung DW, Frausto RF, Ann LB, Jang MS, Aldave AJ
Invest Ophthalmol Vis Sci 2014 Sep 4;55(10):6159-66. doi: 10.1167/iovs.14-15247. PMID: 25190660Free PMC Article
Yellore VS, Papp JC, Sobel E, Khan MA, Rayner SA, Farber DB, Aldave AJ
Genet Med 2007 Apr;9(4):228-34. doi: 10.1097/gim.0b013e31803c4dc2. PMID: 17438387

Clinical prediction guides

Zakharevich M, Kattan JM, Chen JL, Lin BR, Cervantes AE, Chung DD, Frausto RF, Aldave AJ
Mol Vis 2017;23:740-752. Epub 2017 Oct 14 PMID: 29046608Free PMC Article
Chung DW, Frausto RF, Chiu S, Lin BR, Aldave AJ
Invest Ophthalmol Vis Sci 2016 Aug 1;57(10):4136-43. doi: 10.1167/iovs.16-19533. PMID: 27537263Free PMC Article
Evans CJ, Liskova P, Dudakova L, Hrabcikova P, Horinek A, Jirsova K, Filipec M, Hardcastle AJ, Davidson AE, Tuft SJ
Ann Hum Genet 2015 Jan;79(1):1-9. Epub 2014 Dec 1 doi: 10.1111/ahg.12090. PMID: 25441224
Chung DW, Frausto RF, Ann LB, Jang MS, Aldave AJ
Invest Ophthalmol Vis Sci 2014 Sep 4;55(10):6159-66. doi: 10.1167/iovs.14-15247. PMID: 25190660Free PMC Article
Yellore VS, Papp JC, Sobel E, Khan MA, Rayner SA, Farber DB, Aldave AJ
Genet Med 2007 Apr;9(4):228-34. doi: 10.1097/gim.0b013e31803c4dc2. PMID: 17438387

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