Long-Term Follow-Up of Congenital Simple Hamartoma of the Retinal Pigment Epithelium: A Case Report

Background: Congenital simple hamartoma of the retinal pigment epithelium (CSHRPE) is a rare benign tumor of the retinal pigment epithelium characterized by a focal, darkly pigmented nodule in the macular lesion in healthy persons. We report a case of CSHRPE with long-term follow-up. Case: A 41-year-old Japanese woman was incidentally discovered to have a dark lesion on the fundus of the left eye. We evaluated the patient by measuring her best-corrected visual acuity (BCVA) and by slit-lamp biomicroscopy, fundus color photography, and optical coherence tomography (OCT) over a 10-year period. The BCVA gradually declined during the early follow-up period, having decreased from 1.2 to 0.8 in the left eye 3 years after the initial examination, and then has been maintained for the following 7 years. The lesion did not show a change in OCT 10 years after the first examination. Conclusion: It is important to follow a CSHRPE carefully over the long term because visual acuity might decrease.

Download Full-text

LONG-TERM FOLLOW-UP OF RETINAL PIGMENT EPITHELIUM RESTORATION AFTER A TRIPLE TEAR

Retinal Cases & Brief Reports ◽

10.1097/icb.0000000000000530 ◽

2019 ◽

Vol 13 (1) ◽

pp. 5-9 ◽

Cited By ~ 2

Author(s):

Sarwar Zahid ◽

Rosa Dolz Marco ◽

K. Bailey Freund

Keyword(s):

Retinal Pigment Epithelium ◽

Pigment Epithelium ◽

Retinal Pigment ◽

Long Term Follow Up

Download Full-text

Long-Term Follow-Up of Spontaneous Retinal Pigment Epithelium Tears in Age-Related Macular Degeneration Treated with Anti-VEGF Therapy

European Journal of Ophthalmology ◽

10.5301/ejo.2010.2285 ◽

2011 ◽

Vol 21 (1) ◽

pp. 73-76 ◽

Cited By ~ 10

Author(s):

Sebastian P. Lesniak ◽

Howard F. Fine ◽

Jonathan L. Prenner ◽

Daniel B. Roth

Keyword(s):

Retinal Pigment Epithelium ◽

Macular Degeneration ◽

Pigment Epithelium ◽

Retinal Pigment ◽

Age Related Macular Degeneration ◽

Anti Vegf ◽

Age Related ◽

Long Term Follow Up

Download Full-text

Long-term follow-up Study of Nonsenile Detachment of the Retinal Pigment Epithelium

European Journal of Ophthalmology ◽

10.1177/112067219100100205 ◽

1991 ◽

Vol 1 (2) ◽

pp. 79-84 ◽

Cited By ~ 13

Author(s):

L. Laatikainen ◽

M. Hoffren

Keyword(s):

Retinal Pigment Epithelium ◽

Pigment Epithelium ◽

Retinal Pigment ◽

Follow Up Study ◽

Long Term Follow Up

Download Full-text

Combined Hamartoma of the Retina and Retinal Pigment Epithelium in a Patient with Gorlin Syndrome: Spontaneous Partial Resolution of Traction Caused by Epiretinal Membrane

Case Reports in Ophthalmological Medicine ◽

10.1155/2016/2312196 ◽

2016 ◽

Vol 2016 ◽

pp. 1-5 ◽

Cited By ~ 1

Author(s):

José L. Sánchez-Vicente ◽

Miguel Contreras-Díaz ◽

Trinidad Rueda ◽

Enrique Rodríguez de la Rúa-Franch ◽

Fredy E. Molina-Socola ◽

...

Keyword(s):

Visual Acuity ◽

Retinal Pigment Epithelium ◽

Epiretinal Membrane ◽

Pigment Epithelium ◽

Retinal Pigment ◽

Spontaneous Resolution ◽

Fundus Examination ◽

Gorlin Syndrome ◽

Clinical Context

Purpose. To describe the case of spontaneous resolution of epiretinal membrane in a patient with Combined Hamartoma of the Retina and Retinal Pigment Epithelium (CHR-RPE), in the clinical context of Gorlin Syndrome (GS).Methods. Observational case report of a 12-year-old female patient is presented. The diagnosis of CHRRPE was made by OCT and fundus examination, which showed a mound of disorganized tissue originating from retina and retinal pigment epithelium. Epiretinal membrane (EM) was also detected. Genetic study was performed to confirm the diagnosis of GS.Results. The patient was observed for 39 months, showing spontaneous resolution of the traction caused by the EM and improvement in visual acuity (VA), which was 20/80 at initial presentation, rising to 20/40 after follow-up period.Conclusions. The presence of EM in CHR-REP is a cause of reduction of visual acuity. Management of this condition is controversial; however, we would like to highlight that spontaneous resolution of the traction caused by EM is possible, resulting in recovery of VA.

Download Full-text

Long-Term Effect of Half-Fluence Photodynamic Therapy on Fundus Autofluorescence in Acute Central Serous Chorioretinopathy

Journal of Ophthalmology ◽

10.1155/2020/8491712 ◽

2020 ◽

Vol 2020 ◽

pp. 1-7

Author(s):

Martin Stattin ◽

Stefan Hagen ◽

Daniel Ahmed ◽

Eva Smretschnig ◽

Florian Frommlet ◽

...

Keyword(s):

Photodynamic Therapy ◽

Central Serous Chorioretinopathy ◽

Pigment Epithelium ◽

Fundus Autofluorescence ◽

Retinal Pigment ◽

Long Term Follow Up ◽

Acute Central Serous Chorioretinopathy ◽

Mean Differences

Purpose. To evaluate normalized short-wavelength fundus autofluorescence (SW-FAF) imaging changes over time as a predictive parameter for the retinal pigment epithelium (RPE) function in eyes compromised by acute central serous chorioretinopathy (CSCR) after indocyanine green angiography-guided verteporfin (Visudyne®, Novartis Pharma, Basel, Switzerland) photodynamic therapy (PDT) with a half-fluence rate (25 J/cm2). Methods. Quantitative data of SW-FAF grey values (SW-FAF GV) from a 350 μm (SW-350) and 1200 μm (SW-1200) diameter circle centered on the fovea and normalized with the level of SW-FAF GV in a 30° image of 20 eyes in 11 patients initially treated for unilateral acute symptomatic CSCR were collected and retrospectively analyzed after 7 years. A 2-sided t-test was calculated to explore the differences of SW-350 and SW-1200 between one month and the long-term follow-up. Results. Mean differences (95% CI) in SW-FAF GV between 1 month and 7 years after half-fluence PDT were 0.07 ± 0.11 for SW-350 ([95% CI: −0.002; 0.14], p=0.06) and 0.11 ± 0.15 for SW-1200 ([95% CI: 0.01; 0.21], p=0.03). Mean differences in SW-FAF GV of the contralateral untreated eye were 0.06 ± 0.14 for SW-350 ([95% CI: −0.04; 0.17], p=0.22) and 0.05 ± 0.13 for SW-1200 ([95% CI: −0.04; 0.15], p=0.22). Conclusion. After 7 years, normalized SW-FAF GV were significantly lower in eyes with resolved acute CSCR treated with reduced-fluence PDT compared to the follow-up after 1 month without correlation to explicit pattern changes or structural damages. Half-fluence PDT remains a safe and considerable treatment option in acute CSCR.

Download Full-text

On the problem of the classification of congenital glaucoma

Russian Pediatric Ophthalmology ◽

10.18821/1993-1859-2016-11-4-179-183 ◽

2016 ◽

Vol 11 (4) ◽

pp. 179-183 ◽

Cited By ~ 1

Author(s):

L. A Katargina ◽

Andrei Olegovich Tarasenkov ◽

E. V Mazanova

Keyword(s):

Visual Acuity ◽

Age And Growth ◽

Congenital Glaucoma ◽

Initial Examination ◽

Anatomical Changes ◽

Long Term Follow Up ◽

Objective State

Objective. To monitor congenital glaucoma and to determine the proportion of the exposed stage of the disease revealed during the initial examination and the long-term follow up. Materials and methods. The present study included 27 children (47 eyes) at the age ranging from 8 to 17 years presenting with various forms of compensated congenital glaucoma at the initial, advanced, and terminal stages of the disease. Results. The clinical and functional characteristics of the eyes at each stage of congenital glaucoma were analyzed during the initial examination and the long-term follow-up period. It was found that the currently universally accepted classification of congenital glaucoma that subdivides the disease into stages based on the structural anatomical changes in the eyes ceases to adequately reflect the objective state of the visual system as the child grows. Conclusion. The study has demonstrated that the stage of congenital glaucoma needs to be corrected in accordance with the age and growth of the child and the possibility of its examination with the use of the psychophysical methods and the evaluation of visual acuity.

Download Full-text

Long-Term Follow-Up in Bietti Crystalline Dystrophy

European Journal of Ophthalmology ◽

10.1177/112067210701700434 ◽

2007 ◽

Vol 17 (4) ◽

pp. 680-682 ◽

Cited By ~ 23

Author(s):

A.M. Mansour ◽

S.H. Uwaydat ◽

C.-C. Chan

Keyword(s):

Electron Microscopy ◽

Pigment Epithelium ◽

Retinal Pigment ◽

Visual Fields ◽

Clinical Findings ◽

Transmission Electron ◽

Bietti Crystalline Dystrophy ◽

Long Term Follow Up

Purpose To present a long-term follow-up of Bietti crystalline dystrophy. Methods Two brothers are presented including the clinical findings, fluorescein angiography, electrophysiology (electroretinography [ERG], electrooculography [EOG], adaptometry), optical coherence tomography (OCT), and transmission electron microscopy of bulbar conjunctiva and peripheral blood lymphocytes. The clinical findings were documented over a period of 25 years in one brother and 5 years in the other. Results The most striking features were deposits in the retina that were formed de novo with old ones replaced by choroidal atrophy in advanced stage of the disease. The light rise (EOG), rod- and cone-driven responses (ERG), and visual fields were affected progressively during the course. These changes of the retinal pigment epithelium and choriocapillaris were observed in the second decade and worsened gradually. OCT demonstrated preferential crystal accumulation in the inner retina. Cytoplasmic lipid crystalline inclusions were found in lymphocytes and conjunctival fibroblasts by transmission electron microscopy. Conclusions Bietti crystalline retinopathy is a progressive retinal disease characterized by retinal crystals gradually replaced by atrophy of the retinal pigment epithelium and gradual constriction of visual fields.

Download Full-text

Long-term safety and tolerability of subretinal transplantation of embryonic stem cell-derived retinal pigment epithelium in Asian Stargardt disease patients

British Journal of Ophthalmology ◽

10.1136/bjophthalmol-2020-316225 ◽

2020 ◽

pp. bjophthalmol-2020-316225

Author(s):

Youngje Sung ◽

Min Ji Lee ◽

Jinjung Choi ◽

Sang Yoon Jung ◽

So Young Chong ◽

...

Keyword(s):

Clinical Trial ◽

Visual Acuity ◽

Retinal Pigment Epithelium ◽

Pigment Epithelium ◽

Retinal Pigment ◽

Embryonic Stem ◽

Rpe Cells ◽

Corrected Visual Acuity ◽

Best Corrected Visual Acuity

BackgroundAlthough human embryonic stem cells (hESCs) have been considered a potential therapeutic option for regenerative medicine, there are some concerns regarding tumorigenicity, immunogenicity and ethical considerations. Stargardt macular dystrophy (SMD) is the most common form of juvenile macular degeneration that causes early onset blindness. Therapeutic options for SMD remain limited, although several treatment strategies are currently under investigation. Here, we report a 3-year assessment of a phase I clinical trial involving subretinal transplantation of hESC-retinal pigment epithelium (RPE) cells in patients with SMD.MethodsThis prospective, non-randomised clinical trial included three patients with SMD. All transplant recipients had central visual acuity no better than 20/400. Trans-pars plana vitrectomy was performed in the eye with poorer vision. RPE cells were reconstituted in balanced salt solution plus, then injected into the subretinal space using a semi-automated subretinal injection method.ResultsNo serious adverse events occurred throughout the 3-year period following the injection of hESC-RPE cells. The functional and anatomical results were favourable, compared with the natural course of SMD reported in the ProgStar study. One patient showed best-corrected visual acuity improvement, while the other patients had stable best-corrected visual acuity during the 3-year follow-up period.ConclusionThese results suggest the long-term safety, tolerability, and feasibility of subretinal hESC-derived RPE cell transplantation in regenerative medicine.Trial registration numberNCT01625559.

Download Full-text