PMID- 30195891
OWN - NLM
STAT- MEDLINE
DCOM- 20190906
LR  - 20190906
IS  - 1879-1891 (Electronic)
IS  - 0002-9394 (Linking)
VI  - 196
DP  - 2018 Dec
TI  - Clinical Features of Japanese Patients With Anti-alpha-enolase Antibody-Positive 
      Autoimmune Retinopathy: Novel Subtype of Multiple Drusen.
PG  - 181-196
LID - S0002-9394(18)30505-1 [pii]
LID - 10.1016/j.ajo.2018.08.044 [doi]
AB  - PURPOSE: To evaluate clinical features of Japanese patients with anti-alpha-enolase 
      antibody-positive autoimmune retinopathy (anti-enolase AIR). DESIGN: Multicenter 
      retrospective observational case series. METHODS: Forty-nine eyes of 25 Japanese 
      anti-enolase AIR patients (16 female and 9 male; mean age at first visit, 60.8 
      years) were included. Fundus characteristics, perimetry, spectral-domain optical 
      coherence tomography (SD-OCT), electroretinography (ERG), best-corrected visual 
      acuity (BCVA), and complicating systemic tumors were assessed. Protein 
      localization of alpha-enolase was examined by immunohistochemistry in an enucleated 
      eye of 1 patient. RESULTS: Patients were classified into 3 groups: multiple 
      drusen (48%), retinal degeneration (36%), and normal fundus (16%). Drusen varied 
      in size from small deposits to vitelliform-like lesions. Images on SD-OCT 
      revealed dome-shaped hyperreflectivity beneath the retinal pigment epithelium 
      (RPE), corresponding to drusen. Perimetry showed that ring scotoma was the most 
      frequent (39%). Rod-system and/or single-flash cone responses revealed decreased 
      responses in 81% of the eyes. Combined rod and cone system responses demonstrated 
      significantly lower a-wave amplitudes in the degeneration group than in the 
      drusen group (P = .005). BCVA was improved or maintained in 80% of the eyes 
      during follow-up. Malignant or benign tumors were detected in 30% of patients. 
      The RPE and photoreceptor layers were immunopositive for alpha-enolase. CONCLUSIONS: 
      The drusen subtype, scarcely described in the literature, is suggested to 
      characterize Japanese patients with anti-enolase AIR. The different funduscopic 
      features with different functional severities may have resulted from 
      antibody-mediated damage to RPE as well as photoreceptor cells.
CI  - Copyright (c) 2018 Elsevier Inc. All rights reserved.
FAU - Ando, Ryo
AU  - Ando R
AD  - Department of Ophthalmology, Faculty of Medicine and Graduate School of Medicine, 
      Hokkaido University, Sapporo, Japan.
FAU - Saito, Wataru
AU  - Saito W
AD  - Department of Ophthalmology, Faculty of Medicine and Graduate School of Medicine, 
      Hokkaido University, Sapporo, Japan. Electronic address: 
      wsaito@med.hokudai.ac.jp.
FAU - Kanda, Atsuhiro
AU  - Kanda A
AD  - Department of Ophthalmology, Faculty of Medicine and Graduate School of Medicine, 
      Hokkaido University, Sapporo, Japan.
FAU - Kase, Satoru
AU  - Kase S
AD  - Department of Ophthalmology, Faculty of Medicine and Graduate School of Medicine, 
      Hokkaido University, Sapporo, Japan.
FAU - Fujinami, Kaoru
AU  - Fujinami K
AD  - Laboratory of Visual Physiology, Division of Vision Research, National Institute 
      of Sensory Organs, National Hospital Organization, Tokyo Medical Center, Tokyo, 
      Japan.
FAU - Sugahara, Michitaka
AU  - Sugahara M
AD  - Inoue Eye Hospital, Tokyo, Japan.
FAU - Nakamura, Yosuke
AU  - Nakamura Y
AD  - Department of Ophthalmology and Visual Science, Chiba University Graduate School 
      of Medicine, Chiba, Japan.
FAU - Eguchi, Shuichiro
AU  - Eguchi S
AD  - Eguchi Eye Hospital, Hakodate, Japan.
FAU - Mori, Shohei
AU  - Mori S
AD  - Department of Ophthalmology, Faculty of Medicine and Graduate School of Medicine, 
      Hokkaido University, Sapporo, Japan.
FAU - Noda, Kousuke
AU  - Noda K
AD  - Department of Ophthalmology, Faculty of Medicine and Graduate School of Medicine, 
      Hokkaido University, Sapporo, Japan.
FAU - Shinoda, Kei
AU  - Shinoda K
AD  - Department of Ophthalmology, Teikyo University School of Medicine, Tokyo, Japan.
FAU - Ishida, Susumu
AU  - Ishida S
AD  - Department of Ophthalmology, Faculty of Medicine and Graduate School of Medicine, 
      Hokkaido University, Sapporo, Japan.
LA  - eng
PT  - Journal Article
PT  - Multicenter Study
PT  - Observational Study
DEP - 20180907
PL  - United States
TA  - Am J Ophthalmol
JT  - American journal of ophthalmology
JID - 0370500
RN  - 135844-11-0 (Recoverin)
RN  - EC 4.2.1.- (Carbonic Anhydrase II)
RN  - EC 4.2.1.11 (Phosphopyruvate Hydratase)
SB  - IM
MH  - Adolescent
MH  - Adult
MH  - Aged
MH  - Aged, 80 and over
MH  - Autoimmune Diseases/*pathology
MH  - Carbonic Anhydrase II/immunology
MH  - Electroretinography
MH  - Female
MH  - Humans
MH  - Immunohistochemistry
MH  - Japan
MH  - Male
MH  - Middle Aged
MH  - Phosphopyruvate Hydratase/*immunology/metabolism
MH  - Recoverin/immunology
MH  - Retinal Drusen/immunology/*pathology
MH  - Retrospective Studies
MH  - Tomography, Optical Coherence
MH  - Young Adult
EDAT- 2018/09/10 06:00
MHDA- 2019/09/07 06:00
CRDT- 2018/09/10 06:00
PHST- 2018/05/14 00:00 [received]
PHST- 2018/08/10 00:00 [revised]
PHST- 2018/08/29 00:00 [accepted]
PHST- 2018/09/10 06:00 [pubmed]
PHST- 2019/09/07 06:00 [medline]
PHST- 2018/09/10 06:00 [entrez]
AID - S0002-9394(18)30505-1 [pii]
AID - 10.1016/j.ajo.2018.08.044 [doi]
PST - ppublish
SO  - Am J Ophthalmol. 2018 Dec;196:181-196. doi: 10.1016/j.ajo.2018.08.044. Epub 2018 
      Sep 7.