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Choroidal Vasculature in Bietti Crystalline Dystrophy With CYP4V2 Mutations and in Retinitis Pigmentosa With EYS Mutations.

Abstract We compare the choroidal vascular area between Bietti crystalline dystrophy (BCD) patients with CYP4V2 mutations, retinitis pigmentosa (RP) patients with EYS mutations, and normal controls, and investigate the correlation between choroidal vascular area and associated parameters.
PMID
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CHOROIDAL AND RETINAL ATROPHY OF BIETTI CRYSTALLINE DYSTROPHY PATIENTS WITH CYP4V2 MUTATIONS COMPARED TO RETINITIS PIGMENTOSA PATIENTS WITH EYS MUTATIONS.

Authors

Mayor MeshTerms

Corneal Dystrophies, Hereditary

Mutation

Retinal Diseases

Retinitis Pigmentosa

Keywords
Journal Title investigative ophthalmology & visual science
Publication Year Start




PMID- 28763560
OWN - NLM
STAT- MEDLINE
DA  - 20170801
DCOM- 20170803
LR  - 20170803
IS  - 1552-5783 (Electronic)
IS  - 0146-0404 (Linking)
VI  - 58
IP  - 10
DP  - 2017 Aug 01
TI  - Choroidal Vasculature in Bietti Crystalline Dystrophy With CYP4V2 Mutations and
      in Retinitis Pigmentosa With EYS Mutations.
PG  - 3871-3878
LID - 10.1167/iovs.17-21515 [doi]
AB  - Purpose: We compare the choroidal vascular area between Bietti crystalline
      dystrophy (BCD) patients with CYP4V2 mutations, retinitis pigmentosa (RP)
      patients with EYS mutations, and normal controls, and investigate the correlation
      between choroidal vascular area and associated parameters. Methods: This
      prospective case-series study included consecutive nine eyes of nine BCD patients
      with CYP4V2 mutations (BCD group), 16 eyes of 16 RP patients with EYS mutations
      (EYS-RP group), and 16 eyes of 16 normal volunteers matched for age and axial
      length (control group). Using swept-source optical coherence tomography, we
      obtained en face images of the choroidal vasculature at the midpoint of the
      choriocapillaris layer-Sattler's layer (inner choroid) and Haller's layer (outer 
      choroid). After binarization, we compared the inner and outer choroidal vascular 
      areas among the three groups and identified associated factors. Results: The
      outer choroidal vascular area was 43.34 +/- 5.76%, 53.73 +/- 4.92%, and 52.80 +/-
      4.10% in the BCD, EYS-RP, and control groups, respectively. This value was
      significantly smaller in the BCD group than in the EYS-RP and control groups (P <
      0.001 in both; no significant difference between the EYS-RP and control groups). 
      In the BCD group, the outer choroidal vascular area was correlated strongly with 
      the subfoveal inner choroidal thickness (P = 0.001, r = 0.91, respectively). The 
      inner choroidal vasculature could not be identified in eight of nine eyes in the 
      BCD group. Conclusions: The outer choroidal vascular narrowing might progress
      with the inner choroidal thinning in BCD, and the inner choroidal vasculature
      might be extinguished in advanced-stage BCD. Our findings may help to clarify the
      etiology of BCD.
FAU - Hirashima, Takako
AU  - Hirashima T
AD  - Department of Ophthalmology and Visual Sciences, Kyoto University Graduate School
      of Medicine, Sakyo-ku, Kyoto, Japan.
FAU - Miyata, Manabu
AU  - Miyata M
AD  - Department of Ophthalmology and Visual Sciences, Kyoto University Graduate School
      of Medicine, Sakyo-ku, Kyoto, Japan.
FAU - Ishihara, Kenji
AU  - Ishihara K
AD  - Department of Ophthalmology and Visual Sciences, Kyoto University Graduate School
      of Medicine, Sakyo-ku, Kyoto, Japan.
FAU - Hasegawa, Tomoko
AU  - Hasegawa T
AD  - Department of Ophthalmology and Visual Sciences, Kyoto University Graduate School
      of Medicine, Sakyo-ku, Kyoto, Japan.
FAU - Sugahara, Masako
AU  - Sugahara M
AD  - Department of Ophthalmology and Visual Sciences, Kyoto University Graduate School
      of Medicine, Sakyo-ku, Kyoto, Japan.
FAU - Ogino, Ken
AU  - Ogino K
AD  - Department of Ophthalmology and Visual Sciences, Kyoto University Graduate School
      of Medicine, Sakyo-ku, Kyoto, Japan.
FAU - Yoshikawa, Munemitsu
AU  - Yoshikawa M
AD  - Department of Ophthalmology and Visual Sciences, Kyoto University Graduate School
      of Medicine, Sakyo-ku, Kyoto, Japan.
FAU - Hata, Masayuki
AU  - Hata M
AD  - Department of Ophthalmology and Visual Sciences, Kyoto University Graduate School
      of Medicine, Sakyo-ku, Kyoto, Japan.
FAU - Kuroda, Yoshimasa
AU  - Kuroda Y
AD  - Department of Ophthalmology and Visual Sciences, Kyoto University Graduate School
      of Medicine, Sakyo-ku, Kyoto, Japan.
FAU - Muraoka, Yuki
AU  - Muraoka Y
AD  - Department of Ophthalmology and Visual Sciences, Kyoto University Graduate School
      of Medicine, Sakyo-ku, Kyoto, Japan.
FAU - Ooto, Sotaro
AU  - Ooto S
AD  - Department of Ophthalmology and Visual Sciences, Kyoto University Graduate School
      of Medicine, Sakyo-ku, Kyoto, Japan.
FAU - Yoshimura, Nagahisa
AU  - Yoshimura N
AD  - Department of Ophthalmology and Visual Sciences, Kyoto University Graduate School
      of Medicine, Sakyo-ku, Kyoto, Japan.
LA  - eng
PT  - Journal Article
PT  - Observational Study
PL  - United States
TA  - Invest Ophthalmol Vis Sci
JT  - Investigative ophthalmology & visual science
JID - 7703701
RN  - 0 (EYS protein, human)
RN  - 0 (Eye Proteins)
RN  - EC 1.14.13.- (CYP4V2 protein, human)
RN  - EC 1.14.14.1 (Cytochrome P450 Family 4)
RN  - Bietti Crystalline Dystrophy
SB  - IM
MH  - Adult
MH  - Aged
MH  - Blood Vessels/*pathology
MH  - Case-Control Studies
MH  - Choroid/*blood supply/pathology
MH  - *Corneal Dystrophies, Hereditary/genetics/pathology
MH  - Cytochrome P450 Family 4/*genetics
MH  - Eye Proteins/*genetics
MH  - Female
MH  - Humans
MH  - Male
MH  - Middle Aged
MH  - *Mutation
MH  - Prospective Studies
MH  - *Retinal Diseases/genetics/pathology
MH  - *Retinitis Pigmentosa/genetics/pathology
EDAT- 2017/08/02 06:00
MHDA- 2017/08/05 06:00
CRDT- 2017/08/02 06:00
AID - 2646835 [pii]
AID - 10.1167/iovs.17-21515 [doi]
PST - ppublish
SO  - Invest Ophthalmol Vis Sci. 2017 Aug 1;58(10):3871-3878. doi:
      10.1167/iovs.17-21515.