Literature DB >> 9801043

Macular function testing in a German pedigree with North Carolina macular dystrophy.

K Rohrschneider1, A Blankenagel, F E Kruse, T Fendrich, H E Völcker.   

Abstract

BACKGROUND: Our purpose was to investigate central visual function in North Carolina macular dystrophy (MCDR1). A German family with genetically proven MCDR1 was followed for more than 20 years.
METHODS: In addition to routine clinical examinations, static and kinetic fundus perimetry was performed with the scanning laser ophthalmoscope. The center of fixation was determined.
RESULTS: Visual acuity was 0.6 or better in all but one eye of seven members of this family and did not markedly change during follow-up. Fundus examination confirmed that the fixation was at the nasal edge of the central scar in five affected eyes with reduced stability of fixation. One eye had a prominent gliotic membrane, and fixation was within this area.
CONCLUSIONS: Patients had nearly normal visual acuity even though they presented with deep central scotomas and a shift of the center of fixation away from the fovea. Because MCDR1 has good long-term functional prognosis, there might be an early transdifferentiation of the new locus of fixation.

Entities:  

Mesh:

Year:  1998        PMID: 9801043     DOI: 10.1097/00006982-199805000-00013

Source DB:  PubMed          Journal:  Retina        ISSN: 0275-004X            Impact factor:   4.256


  6 in total

Review 1.  Genotype-phenotype correlations and differential diagnosis in autosomal dominant macular disease.

Authors:  A Iannaccone
Journal:  Doc Ophthalmol       Date:  2001-05       Impact factor: 2.379

2.  North Carolina Macular Dystrophy Is Caused by Dysregulation of the Retinal Transcription Factor PRDM13.

Authors:  Kent W Small; Adam P DeLuca; S Scott Whitmore; Thomas Rosenberg; Rosemary Silva-Garcia; Nitin Udar; Bernard Puech; Charles A Garcia; Thomas A Rice; Gerald A Fishman; Elise Héon; James C Folk; Luan M Streb; Christine M Haas; Luke A Wiley; Todd E Scheetz; John H Fingert; Robert F Mullins; Budd A Tucker; Edwin M Stone
Journal:  Ophthalmology       Date:  2015-10-24       Impact factor: 12.079

3.  Clinical and genetic characterization of a Danish family with North Carolina macular dystrophy.

Authors:  Thomas Rosenberg; Ben Roos; Thorkild Johnsen; Niels Bech; Todd E Scheetz; Michael Larsen; Edwin M Stone; John H Fingert
Journal:  Mol Vis       Date:  2010-12-09       Impact factor: 2.367

4.  Duplication events downstream of IRX1 cause North Carolina macular dystrophy at the MCDR3 locus.

Authors:  Valentina Cipriani; Raquel S Silva; Gavin Arno; Nikolas Pontikos; Ambreen Kalhoro; Sandra Valeina; Inna Inashkina; Mareta Audere; Katrina Rutka; Bernard Puech; Michel Michaelides; Veronica van Heyningen; Baiba Lace; Andrew R Webster; Anthony T Moore
Journal:  Sci Rep       Date:  2017-08-08       Impact factor: 4.379

5.  Fixation behavior in macular dystrophy assessed by microperimetry.

Authors:  Wei-Yu Chiang; Jong-Jer Lee; Yi-Hao Chen; Chih-Hsin Chen; Yung-Jen Chen; Pei-Chang Wu; Po-Chiung Fang; Hsi-Kung Kuo
Journal:  Graefes Arch Clin Exp Ophthalmol       Date:  2018-06-09       Impact factor: 3.117

6.  A novel duplication involving PRDM13 in a Turkish family supports its role in North Carolina macular dystrophy (NCMD/MCDR1).

Authors:  Kent W Small; Stijn Van de Sompele; Karen Nuytemans; Andrea Vincent; Ozge Ozalp Yuregir; Emine Ciloglu; Cahfer Sariyildiz; Toon Rosseel; Jessica Avetisjan; Nitin Udar; Jeffery M Vance; Margaret A Pericak-Vance; Elfride De Baere; Fadi S Shaya
Journal:  Mol Vis       Date:  2021-09-01       Impact factor: 2.367

  6 in total

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