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PMID: 2256842 Published · ppublish English Case Reports Journal Article Research Support, Non-U.S. Gov't

Color matching and foveal densitometry in patients and carriers of an X-linked progressive cone dystrophy.

Archives of ophthalmology (Chicago, Ill. : 1960) ·Vol. 108 ·No. 12 ·1990-12-00 ·Pages 1713-9

Keunen JE, van Everdingen JA, Went LN, Oosterhuis JA, van Norren D

Abstract

We describe a family with an as yet undescribed form of X-linked progressive cone dystrophy in a five-generation pedigree, from which we report here the results of 17 male patients and 31 obligate and 13 possible female carriers. The affected males showed the characteristic picture of cone dystrophy. Foveal cone photopigment density was impaired (judged from anomaloscope settings and foveal densitometry), even at an early stage of the disease. The carriers showed no fundus abnormalities, except occasional changes due to myopia. The anomaloscope demonstrated mild pseudoprotanomaly in 27 of 31 obligate carriers and in six of 13 possible carriers. Foveal densitometry findings performed in 11 carriers always agreed with the anomaloscope findings. We conclude that the findings of pseudoprotanomaly and abnormal density differences in females of this family were the only ocular abnormalities and thus are indicative of the carrier state.

MeSH Terms
Adult Color Perception Densitometry Electrophysiology Fluorescein Angiography Fovea Centralis/physiopathology Heterozygote Humans Male Pedigree Retinitis Pigmentosa/genetics,physiopathology Visual Fields X Chromosome
Authors & Affiliations
5 authors, click to expand affiliations / ORCID
Keunen J E
E. C. Donders Institute of Ophthalmology, State University, Utrecht, the Netherlands.
van Everdingen J A
Went L N
Oosterhuis J A
van Norren D
Article Info
Journal
Archives of ophthalmology (Chicago, Ill. : 1960)
Abbr.
Arch Ophthalmol
ISSN
0003-9950
Published
1990-12-00
Pages
1713-9
Language
English
Region
United States
NLM ID
7706534
Subset
IM
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