Literature DB >> 24526317

Macular optical coherence tomography findings and GPR143 mutations in patients with ocular albinism.

H E Sepúlveda-Vázquez1, C Villanueva-Mendoza, J C Zenteno, V Villegas-Ruiz, E Pelcastre-Luna, G García-Aguirre.   

Abstract

The aim of this study was to describe macular findings using spectral-domain optical coherence tomography (SD-OCT) in patients with ocular albinism (OA) and their carrier mothers, and to identify the frequency of GPR143 gene mutations in these patients. The study included five patients with a clinical diagnosis of OA. SD-OCT of the macular area was performed in both patients and their mothers. The anatomical characteristics of the macula and retinal pigment epithelium (RPE), patterns of autofluorescence and infrared imaging were analyzed. Polymerase chain reaction amplification of the complete coding sequence of GPR 143 was performed and subsequently analyzed by direct sequencing in patients and their possible carrier mothers. SD-OCT images revealed the presence of inner retinal layers in the fovea, an abnormal disposition of the Henle layer and a lack of thickening in the perifoveal area. We found increased thickness in the RPE to the outer segment and in the outer segment to the outer nuclear layer that is associated with increased visual acuity. Autofluorescence images revealed an absence of normal hipoautofluorescence in the fovea. No changes were observed in the images of their carrier mothers. Mutation screening and sequence analysis of the GPR 143 gene revealed a novel pathological mutation in two patients. Abnormalities in the macula were observed in all patients. SD-OCT is a useful tool for the assessment of patients with OA. No changes were observed in the SD-OCT of carrier mothers. Only two patients had the GPR143 gene mutation.

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Year:  2014        PMID: 24526317     DOI: 10.1007/s10792-014-9912-1

Source DB:  PubMed          Journal:  Int Ophthalmol        ISSN: 0165-5701            Impact factor:   2.031


  17 in total

1.  Abnormal foveal morphology in ocular albinism imaged with spectral-domain optical coherence tomography.

Authors:  Gabriel T Chong; Sina Farsiu; Sharon F Freedman; Neeru Sarin; Anjum F Koreishi; Joseph A Izatt; Cynthia A Toth
Journal:  Arch Ophthalmol       Date:  2009-01

2.  Spectrum of foveal development in albinism detected with optical coherence tomography.

Authors:  Patti S Harvey; Richard A King; C Gail Summers
Journal:  J AAPOS       Date:  2006-06       Impact factor: 1.220

Review 3.  Albinism: modern molecular diagnosis.

Authors:  S M Carden; R E Boissy; P J Schoettker; W V Good
Journal:  Br J Ophthalmol       Date:  1998-02       Impact factor: 4.638

4.  Foveal hypoplasia in oculocutaneous albinism demonstrated by optical coherence tomography.

Authors:  Carsten H Meyer; Deborah J Lapolice; Sharon F Freedman
Journal:  Am J Ophthalmol       Date:  2002-03       Impact factor: 5.258

5.  Correlation of visual acuity with foveal hypoplasia grading by optical coherence tomography in albinism.

Authors:  Je Hyun Seo; Young Suk Yu; Jeong Hun Kim; Ho Kyung Choung; Jang Won Heo; Seong-Joon Kim
Journal:  Ophthalmology       Date:  2007-03-06       Impact factor: 12.079

6.  Spectral domain optical coherence tomography for detection of foveal morphology in patients with nystagmus.

Authors:  Tara H Cronin; Richard W Hertle; Hiroshi Ishikawa; Joel S Schuman
Journal:  J AAPOS       Date:  2009-12       Impact factor: 1.220

7.  Involvement of OA1, an intracellular GPCR, and G alpha i3, its binding protein, in melanosomal biogenesis and optic pathway formation.

Authors:  Alejandra Young; Elisabeth B Powelson; Irene E Whitney; Mary A Raven; Steven Nusinowitz; Meisheng Jiang; Lutz Birnbaumer; Benjamin E Reese; Debora B Farber
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8.  Foveal thickness and macular volume in patients with oculocutaneous albinism.

Authors:  Natalio J Izquierdo; Andrés Emanuelli; Juan C Izquierdo; Maribel García; Carmen Cadilla; María H Berrocal
Journal:  Retina       Date:  2007 Nov-Dec       Impact factor: 4.256

9.  The ocular albinism type 1 gene product is a membrane glycoprotein localized to melanosomes.

Authors:  M V Schiaffino; C Baschirotto; G Pellegrini; S Montalti; C Tacchetti; M De Luca; A Ballabio
Journal:  Proc Natl Acad Sci U S A       Date:  1996-08-20       Impact factor: 11.205

10.  The ocular albinism type 1 protein, an intracellular G protein-coupled receptor, regulates melanosome transport in pigment cells.

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Journal:  Hum Mol Genet       Date:  2008-08-12       Impact factor: 6.150

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  2 in total

1.  The integrated analysis of RNA-seq and microRNA-seq depicts miRNA-mRNA networks involved in Japanese flounder (Paralichthys olivaceus) albinism.

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Journal:  PLoS One       Date:  2017-08-04       Impact factor: 3.240

2.  Identification of a novel GPR143 mutation in a large Chinese family with isolated foveal hypoplasia.

Authors:  Xiying Mao; Mingkang Chen; Yan Yu; Qinghuai Liu; Songtao Yuan; Wen Fan
Journal:  BMC Ophthalmol       Date:  2021-03-30       Impact factor: 2.209

  2 in total

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