Spontaneous Closure of the Macular Hole in a Patient with Acquired Vitelliform Lesion

In this paper, we report an extremely rare case of spontaneous closure of a macular hole (MH) that developed in a patient in whom acquired vitelliform lesion (AVL) occurred after vitrectomy for atopic retinal detachment (ARD). A 32-year-old male developed ARD in both eyes, and retinal reattachment was achieved after vitrectomy. Five years after surgery, optical coherence tomography showed localized serous retinal detachment (SRD) and a granular lesion with a higher brightness in the subretinal fluid, thus leading to the diagnosis of AVL. One month later, an MH developed, and a follow-up examination performed 6 weeks later revealed that the MH had spontaneously closed and the SRD decreased. In the fovea, fluorescein angiography revealed a window defect due to atrophy of the retinal pigment epithelium (RPE). These findings in this present case suggest the possibility that RPE dysfunction was involved in the development of AVL and MH.

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Acute Posterior Multifocal Placoid Pigment Epitheliopathy Sharing Characteristic OCT Findings of Vogt-Koyanagi-Harada Disease

Case Reports in Ophthalmological Medicine ◽

10.1155/2019/9217656 ◽

2019 ◽

Vol 2019 ◽

pp. 1-6

Author(s):

Yuta Kitamura ◽

Toshiyuki Oshitari ◽

Masayasu Kitahashi ◽

Takayuki Baba ◽

Shuichi Yamamoto

Keyword(s):

Retinal Detachment ◽

Pigment Epithelium ◽

Retinal Pigment ◽

Late Phase ◽

Serous Retinal Detachment ◽

Subretinal Fluid ◽

Hla Typing ◽

Blurred Vision ◽

Fluid Analysis ◽

Vkh Disease

A 17-year-old male presented with acute bilateral paracentral scotomata and blurred vision. Funduscopic examination showed bilateral macular serous retinal detachment and yellow-white placoid lesions at the level of retinal pigment epithelium. OCT study showed typical VKH disease findings with marked choroidal thickening and macular serous retinal detachment partly with subretinal septa in both eyes. FA demonstrated hypofluorescence at the placoid lesions in the early phase and hyperfluorescence in the late phase. Laboratory investigation showed negative result for HLA-DR4 serotype and the patient’s cerebrospinal fluid test values were within normal range. We made the diagnosis of APMPPE from these results. At 2-month follow-up without the use of corticosteroids, OCT reexamination showed complete amelioration of subretinal fluid in both eyes. Patchy pigmentary lesions also resolved clinically with partial chorioretinal scars. The results in this case suggested OCT findings in APMPPE patients could be similar to characteristic features usually found in acute VKH disease. We recommend comprehensive assessments such as FA, cerebral spinal fluid analysis, and HLA typing which help in leading proper diagnosis.

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Multimodal imaging in retinal pigment epithelium rip with exudative bullous retinal detachment

BMJ Case Reports ◽

10.1136/bcr-2020-235731 ◽

2020 ◽

Vol 13 (9) ◽

pp. e235731

Author(s):

Padmaja Kumari Rani ◽

Aniruddh Soni

Keyword(s):

Retinal Pigment Epithelium ◽

Retinal Detachment ◽

Multimodal Imaging ◽

Pigment Epithelium ◽

Retinal Pigment ◽

Subretinal Fluid ◽

Exudative Retinal Detachment ◽

Bullous Retinal Detachment ◽

Subretinal Fluid Drainage

A middle-aged man presented with exudative retinal detachment in the left eye. He was previously diagnosed as a case of large pigment epithelial detachment in the same eye, for which he was asked to follow-up closely, citing the risk of an retinal pigment epithelium (RPE) rip. Multimodal imaging confirmed the presence of a large RPE rip with exudative retinal detachment. He is a known retrovirus patient on anti-retroviral therapy with stable CD4 counts. He was diagnosed as a possible case of bullous variant of central serous chorioretinopathy and underwent external subretinal fluid drainage. We highlight the multimodal imaging findings of RPE rip with exudative retinal detachment and its impact on the patients visual acuity.

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Spontaneous Closure of a Fully Developed Macular Hole in a Severely Myopic Eye

Case Reports in Ophthalmological Medicine ◽

10.1155/2014/182892 ◽

2014 ◽

Vol 2014 ◽

pp. 1-3

Author(s):

C. Bruè ◽

I. Rossiello ◽

J. M. Guidotti ◽

C. Mariotti

Keyword(s):

Visual Acuity ◽

Macular Hole ◽

Pigment Epithelium ◽

Retinal Pigment ◽

Spontaneous Closure ◽

Macular Holes ◽

The Right ◽

Sd Oct

Purpose.Myopic macular holes can be difficult to close with surgery and are frequently associated with retinal detachment. We report on a case of a macular hole in a severely myopic eye that underwent spontaneous closure.Methods.An observational case study.Results.A 55-year-old female was referred to Ophthalmology for a central scotoma and metamorphopsia in the right eye. Visual acuity was 1/20 in both eyes. Fundus examination showed loss of the foveal depression, with a small yellow ring in the center of the fovea in the right eye, and a tilted optic disc and peripapillary staphyloma bilaterally. Spectral domain optical coherence tomography (SD-OCT) revealed a fully developed macular hole with a rim of thickened and slightly elevated retina in the right eye. The patient refused surgery. After 4 years of follow-up, her visual acuity improved to 20/40 in the right eye, and SD-OCT revealed spontaneous sealing of the macular hole without bare retinal pigment epithelium.Conclusions.Myopic macular holes represent a challenge regarding their management, and the prognosis is often poor.

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Surgical management of a case of contractile peripapillary staphyloma with retinal detachment and macular hole

European Journal of Ophthalmology ◽

10.1177/1120672120920227 ◽

2020 ◽

pp. 112067212092022

Author(s):

Sofía H Vidal ◽

Diego Bueso Ponce ◽

Juan Esteban Unigarro ◽

Sergio Arrascue Limo ◽

Carlos Abdala Caballero

Keyword(s):

Visual Acuity ◽

Retinal Detachment ◽

Macular Hole ◽

Retinal Pigment ◽

Silicon Oil ◽

Corrected Visual Acuity ◽

Intraocular Gas ◽

Best Corrected Visual Acuity ◽

Peripapillary Staphyloma

A healthy 4-year-old male presented a fundus examination with a unilateral contractile peripapillary staphyloma surrounded by redundant retina and retinal pigment epithelium atrophy. Five years later, best-corrected visual acuity decreased to hand motion due to a retinal detachment with macular hole. One month after first vitrectomy, scleral buckle and intraocular gas, retina re-detached. Second surgery was performed with silicon oil tamponade and lensectomy without intraocular lens (IOL). Subretinal silicon oil was detected at the third month of follow-up when vitrectomy, inferior retinectomy, and laser photocoagulation of temporal border of staphyloma with silicon oil tamponade were performed. The retina remained attached and best-corrected visual acuity was 20/600 with intraocular silicon oil. A fourth surgery was performed for emulsified silicon oil extraction replaced with intraocular gas. At 6 months of follow-up, the retina re-detached again. This is a challenging vitreoretinal surgery in which re-detachments were due to retinal folds around the contractile staphyloma that raised macular hole. This is the first report of the combined presentation of contractile peripapillary staphyloma, retinal detachment and macular hole with a long-time follow-up period of years.

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Vitreoretinal Surgery for Retinal Detachment in Retinochoroidal Colobomata

European Journal of Ophthalmology ◽

10.1177/112067210801800224 ◽

2008 ◽

Vol 18 (2) ◽

pp. 304-308 ◽

Cited By ~ 4

Author(s):

S.C.B. Teoh ◽

E.J. Mayer ◽

R.J. Haynes ◽

R.H.B. Grey ◽

A.D. Dick ◽

...

Keyword(s):

Retinal Detachment ◽

Silicone Oil ◽

Pigment Epithelium ◽

Retinal Pigment ◽

Hand Movement ◽

Vitreoretinal Surgery ◽

Scleral Buckling ◽

Tertiary Referral Center ◽

Near Work

Purpose To report the management and outcome of retinal reattachment surgery in retinochoroidal coloboma. Methods Four patients with retinochoroidal colobomata presented to the Bristol Eye Hospital (a UK tertiary referral center for vitreoretinal surgery) with retinal detachment. INTERVENTION. All were type II colobomatous detachments (three patients with type IIB, one patient with type IID). All eyes underwent vitrectomy with endolaser and/or cryotherapy and three eyes underwent scleral buckling. Two eyes had internal tamponade with gas (SF6, C3F8) while the other two had silicone oil. Endolaser was applied over healthy retinal pigment epithelium. Results At last follow-up, all (100%) remained attached, with no recurrences. Three patients achieved visual acuity of 6/120 or better and were able to perform satisfactory near work with appropriate magnifiers. The last patient began with hand movement vision and retained similar vision but subjectively felt more navigational. Conclusions Good anatomic and functional outcomes can be achieved in this patient group with combined vitrectomy with or without scleral buckling surgery. Endolaser retinopexy is effective over healthy RPE at the margin of the coloboma combined with either gas or oil internal tamponade.

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Unusual presentation of early-onset X-linked retinoschisis: Report after 1 year of multimodal follow-up

European Journal of Ophthalmology ◽

10.1177/1120672120916722 ◽

2020 ◽

pp. 112067212091672

Author(s):

Andrea Lembo ◽

Giacomo Maria Bacci ◽

Massimiliano Serafino ◽

Stefano Lucentini ◽

Roberto Caputo ◽

...

Keyword(s):

Retinal Detachment ◽

Early Onset ◽

Pigment Epithelium ◽

Retinal Pigment ◽

Retinal Dystrophy ◽

Unusual Presentation ◽

Macular Dystrophy ◽

Wide Field ◽

The Right

Purpose: To describe the unusual presentation, diagnosis, and clinical course of an early-onset X-linked infantile retinoschisis Case report: A 6-month-old infant presented with strabismus and poor fixation. After the detection of bilateral intraretinal hemorrhage and diffuse dystrophic retinal pattern at indirect ophthalmoscopy, the patient received a complete evaluation under anesthesia. Retinal wide-field imaging, spectral domain optical coherence tomography, and electroretinogram were performed and revealed a retinoschisis involving the posterior pole and the inferior periphery in the right eye. In the left eye, an inferior retinal detachment extending to the macula was detected. Blood sample and genetic counseling were required in the strong suspicion of an inherited retinal dystrophy. Genetic tests confirmed the diagnosis of X-linked retinoschisis (RS1 gene mutation). After consultation with a pediatric vitreoretinal surgeon, a wait and see strategy was chosen. The follow up visits showed a surprisingly good natural course of the disease. Conclusion: X-linked retinoschisis is a well-known inherited retinal disease potentially affecting young children as early as 3 months old. In this case, the stunning presentation (diffuse retinal pigment epithelium dystrophic changes resembling a macular dystrophy) and the positive course of the disease (resolution of macular retinal detachment in the left eye and stability of schisis in the right eye) arise some interesting considerations about the necessity of an early surgical treatment.

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Iron is neurotoxic in retinal detachment and transferrin confers neuroprotection

Science Advances ◽

10.1126/sciadv.aau9940 ◽

2019 ◽

Vol 5 (1) ◽

pp. eaau9940 ◽

Cited By ~ 8

Author(s):

Alejandra Daruich ◽

Quentin Le Rouzic ◽

Laurent Jonet ◽

Marie-Christine Naud ◽

Laura Kowalczuk ◽

...

Keyword(s):

Retinal Detachment ◽

Vision Loss ◽

Ex Vivo ◽

Pigment Epithelium ◽

Retinal Pigment ◽

Subretinal Fluid ◽

Predictive Marker ◽

Visual Recovery ◽

Neuroprotective Effects

In retinal detachment (RD), photoreceptor death and permanent vision loss are caused by neurosensory retina separating from the retinal pigment epithelium because of subretinal fluid (SRF), and successful surgical reattachment is not predictive of total visual recovery. As retinal iron overload exacerbates cell death in retinal diseases, we assessed iron as a predictive marker and therapeutic target for RD. In the vitreous and SRF from patients with RD, we measured increased iron and transferrin (TF) saturation that is correlated with poor visual recovery. In ex vivo and in vivo RD models, iron induces immediate necrosis and delayed apoptosis. We demonstrate that TF decreases both apoptosis and necroptosis induced by RD, and using RNA sequencing, pathways mediating the neuroprotective effects of TF are identified. Since toxic iron accumulates in RD, we propose TF supplementation as an adjunctive therapy to surgery for improving the visual outcomes of patients with RD.

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Fingolimod-associated central serous chorioretinopathy in a young girl

BMJ Case Reports ◽

10.1136/bcr-2021-243207 ◽

2021 ◽

Vol 14 (8) ◽

pp. e243207

Author(s):

Gajanan Chavhan Pratima ◽

Doris Benita ◽

Sandip Sarkar ◽

Amit Kumar Deb

Keyword(s):

Central Serous Chorioretinopathy ◽

Complete Resolution ◽

Pigment Epithelium ◽

Retinal Pigment ◽

Subretinal Fluid ◽

Corrected Visual Acuity ◽

Sphingosine 1 Phosphate ◽

Eye Examination ◽

The Right

Fingolimod is a sphingosine-1-phosphate analogue used for the treatment of multiple sclerosis. We, hereby, report a rare case of fingolimod-associated central serous chorioretinopathy (CSCR) in a 21-year-old woman who presented with blurring of vision in the right eye 3 weeks after initiation of oral fingolimod. On examination, best-corrected visual acuity was 20/20 in both the eyes. Fundus examination revealed shallow, serous macular neurosensory detachment in the right eye, and it was confirmed with spectral domain optical coherence tomography. Left eye fundus was normal. Fluorescein angiography showed focal retinal pigment epithelium leak inferior to the fovea. A diagnosis of fingolimod-associated CSCR was made. Oral fingolimod was discontinued. Subsequent follow-up visits showed partial resolution of CSCR at 2 weeks and at 1 month and complete resolution of the subretinal fluid at 2 months. CSCR is, therefore, a rare adverse effect of oral fingolimod treatment. Baseline eye examination and subsequent follow-up at regular intervals are recommended for patients on fingolimod.

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Retinal pigment epithelium aperture in acute central serous chorioretinopathy: Another novel possible pathological mechanism

European Journal of Ophthalmology ◽

10.1177/11206721211002444 ◽

2021 ◽

pp. 112067212110024

Author(s):

Chunyan Lei ◽

Rui Hua ◽

Jianan Duan ◽

Meixia Zhang

Keyword(s):

Retinal Pigment Epithelium ◽

Central Serous Chorioretinopathy ◽

Pigment Epithelium ◽

Retinal Pigment ◽

Subretinal Fluid ◽

Near Infrared Reflectance ◽

Pathological Mechanism ◽

Acute Central Serous Chorioretinopathy ◽

Sd Oct

Purpose: To present retinal pigment epithelium (RPE) aperture related to an avascular pigment epithelium detachment (PED) secondary to acute central serous chorioretinopathy (CSC). Methods: Case report. Results: A 47-year-old man diagnosed as acute CSC presented with RPE aperture in the superonasal area of the macula in his left eye during follow-up. At 2-week follow-up, his decimal best-corrected visual acuity (BCVA) was improved from 0.08 to 0.6 and subretinal fluid was partially absorbed. However, the near-infrared reflectance demonstrated a round mild hyperreflective lesion on the superonasal area of the macula. On spectral-domain optical coherence tomography (SD-OCT), RPE band of the round lesion discontinued but RPE fractured edges without shrinkage and curling. Fundus autofluorescence (FAF) showed RPE aperture appeared as round hypoautofluorescence and hyperautofluorescence outlined its borderline. OCT angiography demonstrated that no evidence of neovascularization within the sub-RPE space. En Face OCT confirmed that the RPE aperture developed at the edge of the PED lesion. At 8-month follow-up, his decimal BCVA was improved to 1.0 and SD-OCT demonstrated spontaneous resolution of subretinal fluid and restoration of RPE structure, with complete flattening of PED. However, FAF revealed hypoautofluorescence mingled with slight hyperautofluorescence within the lesion. Conclusions: To the best of our knowledge, this is the first report of an RPE aperture secondary to acute CSC. Our case indicated another novel possible pathological mechanism that in the relatively healthy RPE, increased hydrostatic pressure simply itself could contribute to RPE aperture.

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Acute exudative polymorphous vitelliform maculopathy during pembrolizumab treatment for metastatic melanoma: a case report

BMC Ophthalmology ◽

10.1186/s12886-021-02011-4 ◽

2021 ◽

Vol 21 (1) ◽

Author(s):

Ine Lambert ◽

Giuseppe Fasolino ◽

Gil Awada ◽

Robert Kuijpers ◽

Marcel ten Tusscher ◽

...

Keyword(s):

Retinal Detachment ◽

Checkpoint Inhibitors ◽

Pigment Epithelium ◽

Retinal Pigment ◽

Anterior Segment ◽

Subretinal Fluid ◽

Additional Treatment ◽

Central Visual Field ◽

Immunomodulating Therapy ◽

Neurosensory Retinal Detachment

Abstract Background The use of immunomodulating therapy to treat various cancers has been on the rise and these immune checkpoint inhibitors are known to cause ocular side effects. In this article a case of acute exudative polymorphous vitelliform maculopathy (AEPVM) is reported which developed during a first line treatment with pembrolizumab. Case presentation A 54-year-old woman was referred because of blurry vision in both eyes with a yellow spot in the central visual field of the left eye. These symptoms started after four treatments with pembrolizumab (a monoclonal antibody against the programmed cell death receptor-1) for a metastatic recurrent vaginal mucosal melanoma. Her best corrected visual acuity was 10/10 in both eyes with a correction of + 2.00 bilaterally. There were no inflammatory findings in the anterior segment or the vitreous. Fundoscopy revealed an attenuation of the foveal reflex with subtle yellow-white subretinal macular deposits (vitelliform lesions) in both eyes. Fluorescein angiography did not show staining or leakage in the mid-phase, neither a late staining. Spectral-domain optical coherence tomography of the macula illustrated bilateral neurosensory retinal detachment with a thick, highly reflective band at the outer photoreceptor segment. En face structural OCT at the level of the photoreceptors showed focal areas of increased signal corresponding to hyperreflective vitelliform material. The treatment with pembrolizumab was ceased immediately. During the following visits we slowly saw an improvement of the neurosensory retinal detachment. After almost four months a total resolution of the subretinal fluid was visualized in both eyes without the use of additional treatment, though the vitelliform deposits persisted. Conclusions The development of AEPVM in melanoma patients could be triggered by treatment with Pembrolizumab. Pembrolizumab has the potential to disturb indirectly the retinal pigment epithelium homeostasis with accumulation of lipofuscin deposits and subretinal fluid, both signs of AEPVM.

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