Literature DB >> 7754251

Pattern dystrophy and retinitis pigmentosa caused by a peripherin/RDS mutation.

S C Richards1, D J Creel.   

Abstract

PURPOSE: To describe the clinical and molecular genetic findings in members of a family with features of autosomal dominant retinitis pigmentosa (RP) and pattern dystrophy.
METHODS: Members of a four-generation family underwent ophthalmoscopic examination, electrophysiologic testing, and screening of blood samples for rhodopsin and peripherin/RDS mutations.
RESULTS: Three members of the family had clinical evidence of both RP and pattern dystrophy, and another family member had symptoms suggestive of RP. In one case of pattern dystrophy, pigment deposition in an unusual ring-like configuration was seen. All affected family members were found to have a Pro216Ser mutation in the peripherin/RDS gene on chromosome 6p, a mutation not found in unrelated (normal) spouses or in a normal control population.
CONCLUSION: In members of this family, both autosomal dominant RP and pattern dystrophy were associated with a Pro216Ser mutation in the peripherin/RDS gene.

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Year:  1995        PMID: 7754251     DOI: 10.1097/00006982-199515010-00013

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


  5 in total

1.  A novel mutation in the RDS gene in an Italian family with pattern dystrophy.

Authors:  F Testa; V Marini; S Rossi; E Interlandi; A Nesti; M Rinaldi; M Varano; C Garré; F Simonelli
Journal:  Br J Ophthalmol       Date:  2005-08       Impact factor: 4.638

2.  PRPH2 mutation update: In silico assessment of 245 reported and 7 novel variants in patients with retinal disease.

Authors:  Manon H C A Peeters; Mubeen Khan; Anoek A M B Rooijakkers; Timo Mulders; Lonneke Haer-Wigman; Camiel J F Boon; Caroline C W Klaver; L Ingeborgh van den Born; Carel B Hoyng; Frans P M Cremers; Anneke I den Hollander; Claire-Marie Dhaenens; Rob W J Collin
Journal:  Hum Mutat       Date:  2021-09-20       Impact factor: 4.700

Review 3.  Finding and interpreting genetic variations that are important to ophthalmologists.

Authors:  Edwin M Stone
Journal:  Trans Am Ophthalmol Soc       Date:  2003

4.  Mutations in the peripherin/RDS gene are an important cause of multifocal pattern dystrophy simulating STGD1/fundus flavimaculatus.

Authors:  Camiel J F Boon; Mary J van Schooneveld; Anneke I den Hollander; Janneke J C van Lith-Verhoeven; Marijke N Zonneveld-Vrieling; Thomas Theelen; Frans P M Cremers; Carel B Hoyng; B Jeroen Klevering
Journal:  Br J Ophthalmol       Date:  2007-05-15       Impact factor: 4.638

5.  Mutations in CTNNA1 cause butterfly-shaped pigment dystrophy and perturbed retinal pigment epithelium integrity.

Authors:  Nicole T M Saksens; Mark P Krebs; Frederieke E Schoenmaker-Koller; Wanda Hicks; Minzhong Yu; Lanying Shi; Lucy Rowe; Gayle B Collin; Jeremy R Charette; Stef J Letteboer; Kornelia Neveling; Tamara W van Moorsel; Sleiman Abu-Ltaif; Elfride De Baere; Sophie Walraedt; Sandro Banfi; Francesca Simonelli; Frans P M Cremers; Camiel J F Boon; Ronald Roepman; Bart P Leroy; Neal S Peachey; Carel B Hoyng; Patsy M Nishina; Anneke I den Hollander
Journal:  Nat Genet       Date:  2015-12-21       Impact factor: 38.330

  5 in total

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