PMID- 28593392
OWN - NLM
STAT- MEDLINE
DCOM- 20170922
LR  - 20181202
IS  - 1573-2622 (Electronic)
IS  - 0012-4486 (Linking)
VI  - 135
IP  - 1
DP  - 2017 Aug
TI  - Multimodal imaging and functional correlations identify unusual cases of macular 
      retinal pigment epithelium hypopigmentation occurring without functional loss.
PG  - 77-83
LID - 10.1007/s10633-017-9592-z [doi]
AB  - PURPOSE: Patients with unusual macular retinal pigment epithelium (RPE) 
      hypopigmentation are described and analyzed using retinal multimodal imaging. 
      METHODS: We report three cases of patients with unilateral (2) or bilateral (1) 
      macular lesions discovered incidentally on fundoscopy. A comprehensive ophthalmic 
      examination including visual acuity, fundoscopy, spectral-domain optical 
      coherence tomography (SD-OCT), short-wavelength light and near-infrared 
      autofluorescence, fluorescein angiography, microperimetry, multifocal 
      electroretinogram, adaptive optics (AO), and OCT-angiography (OCT-A) has been 
      performed. RESULTS: Visual acuity was 20/20 in both eyes of all patients. The 
      lesion appeared hyperautofluorescent on short-wavelength light and 
      hypoautofluorescent on near-infrared light. Fluorescein angiography revealed a 
      sharply demarcated macular hyperfluorescence without any leakage, suggesting a 
      window defect. Interestingly, SD-OCT revealed only a choroidal hyperreflectivity 
      in relation to the lesions without any abnormality of the outer retinal layers. 
      Microperimetry was normal except for 1 patient with bilateral lesion and subtle 
      decrease in macular sensitivity. Mf ERG was normal in all three patients. AO 
      showed a well-preserved cone mosaic, suggesting that the abnormality was 
      localized under the photoreceptor layers. OCT-A revealed hyperreflectivity just 
      below the RPE layer, corresponding to the macular lesion observed on fundoscopy 
      and the choroidal hyperreflectivity seen on SD-OCT. CONCLUSIONS: Macular RPE 
      hypopigmentation should be considered in case of an isolated macular lesion 
      without functional visual impairment or anatomical defect on SD-OCT.
FAU - Boulanger-Scemama, E
AU  - Boulanger-Scemama E
AD  - Fondation Ophtalmologique Adolphe de Rothschild, Paris, France.
FAU - Akesbi, J
AU  - Akesbi J
AD  - Centre Hospitalier National d'Ophtalmologie des Quinze-Vingts, Service II, 75012, 
      Paris, France.
FAU - Tick, S
AU  - Tick S
AD  - DHUSight Restore, INSERM-DGOS CIC 1423, Centre Hospitalier National 
      d'Ophtalmologie des Quinze-Vingts, Paris, France.
FAU - Mohand-Said, S
AU  - Mohand-Said S
AD  - DHUSight Restore, INSERM-DGOS CIC 1423, Centre Hospitalier National 
      d'Ophtalmologie des Quinze-Vingts, Paris, France.
AD  - INSERM U968, CNRS UMR_7210, Institut de la Vision, UPMC Univ Paris 06, Sorbonne 
      Universites, 17 Rue Moreau, 75012, Paris, France.
FAU - Sahel, J-A
AU  - Sahel JA
AD  - Fondation Ophtalmologique Adolphe de Rothschild, Paris, France.
AD  - DHUSight Restore, INSERM-DGOS CIC 1423, Centre Hospitalier National 
      d'Ophtalmologie des Quinze-Vingts, Paris, France.
AD  - INSERM U968, CNRS UMR_7210, Institut de la Vision, UPMC Univ Paris 06, Sorbonne 
      Universites, 17 Rue Moreau, 75012, Paris, France.
AD  - Department of Ophthalmology, The University of Pittsburgh School of Medicine, 
      Pittsburgh, PA, 15213, USA.
FAU - Audo, I
AU  - Audo I
AD  - DHUSight Restore, INSERM-DGOS CIC 1423, Centre Hospitalier National 
      d'Ophtalmologie des Quinze-Vingts, Paris, France. isabelle.audo@inserm.fr.
AD  - INSERM U968, CNRS UMR_7210, Institut de la Vision, UPMC Univ Paris 06, Sorbonne 
      Universites, 17 Rue Moreau, 75012, Paris, France. isabelle.audo@inserm.fr.
LA  - eng
PT  - Case Reports
PT  - Journal Article
DEP - 20170607
PL  - Netherlands
TA  - Doc Ophthalmol
JT  - Documenta ophthalmologica. Advances in ophthalmology
JID - 0370667
SB  - IM
MH  - Adult
MH  - Aged
MH  - Electroretinography
MH  - Female
MH  - Fluorescein Angiography
MH  - Humans
MH  - Hypopigmentation/*diagnosis/physiopathology
MH  - Male
MH  - *Multimodal Imaging
MH  - Ophthalmoscopy
MH  - Retinal Dystrophies/*diagnosis/physiopathology
MH  - Retinal Pigment Epithelium/*pathology
MH  - Tomography, Optical Coherence/methods
MH  - Vision Disorders/*physiopathology
MH  - Visual Acuity/*physiology
MH  - Visual Field Tests
OTO - NOTNLM
OT  - Depigmented retinal pigment epithelium
OT  - Hypopigmentation
OT  - Near-infrared autofluorescence
OT  - Short-wavelength autofluorescence
EDAT- 2017/06/09 06:00
MHDA- 2017/09/25 06:00
CRDT- 2017/06/09 06:00
PHST- 2017/03/16 00:00 [received]
PHST- 2017/05/23 00:00 [accepted]
PHST- 2017/06/09 06:00 [pubmed]
PHST- 2017/09/25 06:00 [medline]
PHST- 2017/06/09 06:00 [entrez]
AID - 10.1007/s10633-017-9592-z [pii]
AID - 10.1007/s10633-017-9592-z [doi]
PST - ppublish
SO  - Doc Ophthalmol. 2017 Aug;135(1):77-83. doi: 10.1007/s10633-017-9592-z. Epub 2017 
      Jun 7.