Literature DB >> 9017054

Full-field electroretinograms in individuals with the Laurence-Mood-Bardet-Biedl syndrome.

R Riise1, S Andréasson, K Tornqvist.   

Abstract

PURPOSE: To evaluate rod and cone function in individuals with the Laurence-Moon-Bardet-Biedl syndrome.
METHODS: We obtained a full-field electroretinograms in 36 patients. If responses less than 10 microV were recorded with single white flashes a special techniques with narrow band filter and computer averaging was used.
RESULTS: No rod responses to dim blue light could be obtained in any of the patients. Residual cone flicker responses were measurable in 28 of the individuals. Those with amplitudes < 0.05 microV were significantly older than those with amplitudes > 1.00 microV. The ERG pattern was consistent within affected pairs of siblings in 8 families.
CONCLUSION: The retinal dystrophy in Laurence-Moon-Bardet-Biedl syndrome is primarily a rod-cone dystrophy, but even cone flicker amplitudes are severely reduced with further progression with age. There is no intrafamilial variability of the electroretinograms in affected siblings.

Entities:  

Mesh:

Year:  1996        PMID: 9017054     DOI: 10.1111/j.1600-0420.1996.tb00747.x

Source DB:  PubMed          Journal:  Acta Ophthalmol Scand        ISSN: 1395-3907


  7 in total

1.  Intrafamilial variation of the phenotype in Bardet-Biedl syndrome.

Authors:  R Riise; S Andréasson; M K Borgaström; A F Wright; N Tommerup; T Rosenberg; K Tornqvist
Journal:  Br J Ophthalmol       Date:  1997-05       Impact factor: 4.638

2.  Multifocal electroretinogram contributes to differentiation of various clinical pictures within a family with Bardet-Biedl syndrome.

Authors:  A Praidou; R Hagan; H Nayak; A Chandna
Journal:  Eye (Lond)       Date:  2014-07-04       Impact factor: 3.775

3.  Impaired photoreceptor protein transport and synaptic transmission in a mouse model of Bardet-Biedl syndrome.

Authors:  Muhammad M Abd-El-Barr; Kristen Sykoudis; Sara Andrabi; Erica R Eichers; Mark E Pennesi; Perciliz L Tan; John H Wilson; Nicholas Katsanis; James R Lupski; Samuel M Wu
Journal:  Vision Res       Date:  2007-12       Impact factor: 1.886

4.  Phenotypic characterization of Bbs4 null mice reveals age-dependent penetrance and variable expressivity.

Authors:  Erica R Eichers; Muhammad M Abd-El-Barr; Richard Paylor; Richard Alan Lewis; Weimin Bi; Xiaodi Lin; Thomas P Meehan; David W Stockton; Samuel M Wu; Elizabeth Lindsay; Monica J Justice; Philip L Beales; Nicholas Katsanis; James R Lupski
Journal:  Hum Genet       Date:  2006-06-23       Impact factor: 4.132

5.  Visual acuity and retinal function in patients with Bardet-Biedl syndrome.

Authors:  Adriana Berezovsky; Daniel Martins Rocha; Paula Yuri Sacai; Sung Song Watanabe; Nívea Nunes Cavascan; Solange Rios Salomão
Journal:  Clinics (Sao Paulo)       Date:  2012       Impact factor: 2.365

6.  A rod-sparing retinopathy in bardet-biedl syndrome.

Authors:  Cynthia S Chiu; Dimosthenis Mantopoulos; Simmons Lessell; Dean M Cestari
Journal:  Case Rep Ophthalmol       Date:  2015-01-27

7.  Comparative Natural History of Visual Function From Patients With Biallelic Variants in BBS1 and BBS10.

Authors:  Monika K Grudzinska Pechhacker; Samuel G Jacobson; Arlene V Drack; Matteo Di Scipio; Ine Strubbe; Wanda Pfeifer; Jacque L Duncan; Helene Dollfus; Nathalie Goetz; Jean Muller; Andrea L Vincent; Tomas S Aleman; Anupreet Tumber; Caroline Van Cauwenbergh; Elfride De Baere; Emma Bedoukian; Bart P Leroy; Jason T Maynes; Francis L Munier; Erika Tavares; Eman Saleh; Ajoy Vincent; Elise Heon
Journal:  Invest Ophthalmol Vis Sci       Date:  2021-12-01       Impact factor: 4.799

  7 in total

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