Literature DB >> 11805522

Report of a new family with dominant congenital heredity stromal dystrophy of the cornea.

Rita Van Ginderdeuren1, Rita De Vos, Ingele Casteels, Bea Foets.   

Abstract

PURPOSE: To report a new family with the rare form of congenital and hereditary stromal dystrophy of the cornea.
METHODS: A mother and son, showing a bilateral congenital clouding of the cornea, were studied clinically and by biomicroscopy. After corneal transplantation, light microscopy and electron microscopy were performed.
RESULTS: The stroma of the cornea was bilaterally and symmetrically thickened with diffuse and homogeneous small opacities. The opacities were present at birth and slowly progressive. Visual acuity was reduced to 2/10. Electron microscopy of the excised corneas showed a thickened stroma owing to cleaving of the lamellae by alternating layers of small-diameter collagen fibrils arranged in a random fashion. The epithelium, Bowman's membrane, the endothelium, and Descemet's membrane were normal.
CONCLUSIONS: This family presents with a congenital stromal dystrophy of the cornea not linked to endothelial defects and thus differs from the more common form of congenital hereditary corneal endothelial dystrophy.

Entities:  

Mesh:

Year:  2002        PMID: 11805522     DOI: 10.1097/00003226-200201000-00025

Source DB:  PubMed          Journal:  Cornea        ISSN: 0277-3740            Impact factor:   2.651


  6 in total

1.  Bilateral cloudy cornea: is the usual suspect congenital hereditary endothelial dystrophy or stromal dystrophy?

Authors:  Banu Torun Acar; Kansu Tahir Bozkurt; Erkan Duman; Suphi Acar
Journal:  BMJ Case Rep       Date:  2016-04-22

2.  Pathophysiological mechanisms of autosomal dominant congenital stromal corneal dystrophy: C-terminal-truncated decorin results in abnormal matrix assembly and altered expression of small leucine-rich proteoglycans.

Authors:  Shoujun Chen; Mei Sun; Xianmin Meng; Renato V Iozzo; Winston W-Y Kao; David E Birk
Journal:  Am J Pathol       Date:  2011-09-03       Impact factor: 4.307

Review 3.  The IC3D classification of the corneal dystrophies.

Authors:  Jayne S Weiss; H U Møller; Walter Lisch; Shigeru Kinoshita; Anthony J Aldave; Michael W Belin; Tero Kivelä; Massimo Busin; Francis L Munier; Berthold Seitz; John Sutphin; Cecilie Bredrup; Mark J Mannis; Christopher J Rapuano; Gabriel Van Rij; Eung Kweon Kim; Gordon K Klintworth
Journal:  Cornea       Date:  2008-12       Impact factor: 2.651

4.  A novel decorin gene mutation in congenital hereditary stromal dystrophy: a Korean family.

Authors:  Jung Hye Lee; Chang-Seok Ki; Eui-Sang Chung; Tae-Young Chung
Journal:  Korean J Ophthalmol       Date:  2012-07-24

Review 5.  Pathophysiological Significance of Dermatan Sulfate Proteoglycans Revealed by Human Genetic Disorders.

Authors:  Shuji Mizumoto; Tomoki Kosho; Shuhei Yamada; Kazuyuki Sugahara
Journal:  Pharmaceuticals (Basel)       Date:  2017-03-27

6.  Role of Decorin Core Protein in Collagen Organisation in Congenital Stromal Corneal Dystrophy (CSCD).

Authors:  Christina S Kamma-Lorger; Christian Pinali; Juan Carlos Martínez; Jon Harris; Robert D Young; Cecilie Bredrup; Eva Crosas; Marc Malfois; Eyvind Rødahl; Keith M Meek; Carlo Knupp
Journal:  PLoS One       Date:  2016-02-01       Impact factor: 3.240

  6 in total

北京卡尤迪生物科技股份有限公司 © 2022-2023.