Literature DB >> 7180914

Ocular and systemic manifestations of the Bardet-Biedl syndrome.

R V Campo, T M Aaberg.   

Abstract

Four women with the Bardet-Biedl syndrome had ophthalmoscopic findings compatible with a severe rod-cone degeneration. The patients were legally blind (visual acuity, 20/200 or worse) in one or both eyes before the age of 30 years. Two patients with early involvement had macular bull's-eye pigment epithelial changes. Two other patients had more advanced disease with geographic atrophy of the macular pigment epithelium and underlying choriocapillaris. Bone spicule formation was variable. Electrophysiologic findings were consistent with severe derangement of both the rod and cone systems. All four patients had intraretinal capillary leakage along the vascular arcades and from the optic nerve without cystoid macular edema. Extensive endocrinologic evaluation showed no objective evidence of hypogenitalism in the three patients tested. Three patients had renal disease, secondary to vesicoureteral reflux, or hypertension, or both.

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Year:  1982        PMID: 7180914     DOI: 10.1016/0002-9394(82)90299-9

Source DB:  PubMed          Journal:  Am J Ophthalmol        ISSN: 0002-9394            Impact factor:   5.258


  8 in total

1.  The PROM1 mutation p.R373C causes an autosomal dominant bull's eye maculopathy associated with rod, rod-cone, and macular dystrophy.

Authors:  Michel Michaelides; Marie-Claire Gaillard; Pascal Escher; Leila Tiab; Matthew Bedell; François-Xavier Borruat; Daniel Barthelmes; Ruben Carmona; Kang Zhang; Edward White; Michelle McClements; Anthony G Robson; Graham E Holder; Keith Bradshaw; David M Hunt; Andrew R Webster; Anthony T Moore; Daniel F Schorderet; Francis L Munier
Journal:  Invest Ophthalmol Vis Sci       Date:  2010-04-14       Impact factor: 4.799

2.  Clinical pathology and retinal vascular structure in the Bardet-Biedl syndrome.

Authors:  T Bek; T Rosenberg
Journal:  Br J Ophthalmol       Date:  1995-01       Impact factor: 4.638

3.  Rod-cone interactions in the ERG of a patient with Bardet-Biedl syndrome.

Authors:  J M Harrison; W A Van Heuven
Journal:  Doc Ophthalmol       Date:  1985-08-30       Impact factor: 2.379

4.  Genome-wide suppressor screen identifies USP35/USP38 as therapeutic candidates for ciliopathies.

Authors:  I-Chun Tsai; Kevin A Adams; Joyce A Tzeng; Omar Shennib; Perciliz L Tan; Nicholas Katsanis
Journal:  JCI Insight       Date:  2019-11-14

5.  Novel clinical findings in autosomal recessive NR2E3-related retinal dystrophy.

Authors:  Vittoria Murro; Dario Pasquale Mucciolo; Andrea Sodi; Ilaria Passerini; Dario Giorgio; Gianni Virgili; Stanislao Rizzo
Journal:  Graefes Arch Clin Exp Ophthalmol       Date:  2018-10-15       Impact factor: 3.117

Review 6.  Retinitis pigmentosa, pigmentary retinopathies, and neurologic diseases.

Authors:  M Tariq Bhatti
Journal:  Curr Neurol Neurosci Rep       Date:  2006-09       Impact factor: 5.081

7.  Hydrometrocolpos and polydactyly: a common neonatal presentation of Bardet-Biedl and McKusick-Kaufman syndromes.

Authors:  A David; P Bitoun; D Lacombe; J C Lambert; A Nivelon; J Vigneron; A Verloes
Journal:  J Med Genet       Date:  1999-08       Impact factor: 6.318

8.  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

  8 in total

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