Pub Date : 2024-10-18DOI: 10.1097/ICB.0000000000001684
Bruno F A Ferreira, Gustavo Sakuno, Juliana M Kato, Rodrigo H Sato, Álvaro F Ferreira, Ruy F B G Missaka, Yoshitaka Nakashima
Purpose: To use multimodal imaging to report a case of dengue fever presenting with Henle fiber layer hemorrhage (HH) and acute paracentral acute middle maculopathy (PAMM).
Methods: Fundus photography and autofluorescence, fluorescein angiography, and optical coherence tomography (OCT) were acquired over a month, and a literature review of dengue fever-associated maculopathy was performed.
Results: A 19-year-old man presented with a sudden loss of vision in both eyes 12 days after experiencing initial systemic symptoms suggestive of dengue fever. His best-corrected visual acuity (BCVA) was counting fingers in the right eye (OD) and 20/200 in the left eye (OS). Pupillary light reflex was reduced in both eyes (OU). Dilated fundoscopic examination revealed optic disc swelling and a small deep hemorrhage near the fovea in OU. OCT revealed a petaloid shape hyperreflectivity involving both outer nuclear and plexiform layers in OU and a subtle hyperreflectivity thinning the inner nuclear layer (INL) in OS, findings compatible with HH and PAMM, respectively. Immunoglobin M was positive for dengue fever. Thus, a diagnosis of dengue fever-associated maculopathy was performed and oral steroids were started. After a month, the final BCVA reached 20/25 (OD) and 20/20 (OS), the HH reabsorbed utterly, and a INL thinning in the area of the PAAM was observed.
Conclusion: Dengue fever-associated maculopathy can present with PAAM and HH. Multimodal imaging is helpful to characterize the extension of the changes in the retina and to monitor its recovery. In such cases, structural and functional damage may occur despite treatment with oral steroids.
{"title":"PARACENTRAL ACUTE MIDDLE MACULOPATHY AND HENLE FIBER LAYER HEMORRHAGE IN DENGUE FEVER: MULTIMODAL IMAGING AND REVIEW OF THE LITERATURE.","authors":"Bruno F A Ferreira, Gustavo Sakuno, Juliana M Kato, Rodrigo H Sato, Álvaro F Ferreira, Ruy F B G Missaka, Yoshitaka Nakashima","doi":"10.1097/ICB.0000000000001684","DOIUrl":"https://doi.org/10.1097/ICB.0000000000001684","url":null,"abstract":"<p><strong>Purpose: </strong>To use multimodal imaging to report a case of dengue fever presenting with Henle fiber layer hemorrhage (HH) and acute paracentral acute middle maculopathy (PAMM).</p><p><strong>Methods: </strong>Fundus photography and autofluorescence, fluorescein angiography, and optical coherence tomography (OCT) were acquired over a month, and a literature review of dengue fever-associated maculopathy was performed.</p><p><strong>Results: </strong>A 19-year-old man presented with a sudden loss of vision in both eyes 12 days after experiencing initial systemic symptoms suggestive of dengue fever. His best-corrected visual acuity (BCVA) was counting fingers in the right eye (OD) and 20/200 in the left eye (OS). Pupillary light reflex was reduced in both eyes (OU). Dilated fundoscopic examination revealed optic disc swelling and a small deep hemorrhage near the fovea in OU. OCT revealed a petaloid shape hyperreflectivity involving both outer nuclear and plexiform layers in OU and a subtle hyperreflectivity thinning the inner nuclear layer (INL) in OS, findings compatible with HH and PAMM, respectively. Immunoglobin M was positive for dengue fever. Thus, a diagnosis of dengue fever-associated maculopathy was performed and oral steroids were started. After a month, the final BCVA reached 20/25 (OD) and 20/20 (OS), the HH reabsorbed utterly, and a INL thinning in the area of the PAAM was observed.</p><p><strong>Conclusion: </strong>Dengue fever-associated maculopathy can present with PAAM and HH. Multimodal imaging is helpful to characterize the extension of the changes in the retina and to monitor its recovery. In such cases, structural and functional damage may occur despite treatment with oral steroids.</p>","PeriodicalId":53580,"journal":{"name":"Retinal Cases and Brief Reports","volume":" ","pages":""},"PeriodicalIF":0.0,"publicationDate":"2024-10-18","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"142562648","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
Pub Date : 2024-10-18DOI: 10.1097/ICB.0000000000001687
Melissa Lu, Mohammed Al Kaabi, Cynthia Xin-Ya Qian
Purpose: To report and discuss a case of brainstem anesthesia and contralateral amaurosis following a sub-Tenon block.
Methods: Single surgical case report of a patient who underwent an uncomplicated pars plana vitrectomy for an epiretinal membrane peel in the left eye following a sub-Tenon anesthesia technique.
Results: Post-operatively, the patient experienced symptoms of brainstem anesthesia as well as akinesia, a non-reactive pupil, and reduced visual acuity in the contralateral eye which gradually resolved within 24 hours. Imaging was within normal limits and did not reveal any anatomic abnormalities.
Conclusion: Despite the use of a blunt-ended cannula in a sub-Tenon block, there is a non-negligible risk of it penetrating surrounding structures such as the optic nerve sheath. This case emphasizes the importance of monitoring for brainstem anesthesia and contralateral eye affections in patients both intra- and post-operatively.
{"title":"Case report: A Unique Case of Brainstem Anesthesia and Contralateral Amaurosis Following a Sub-Tenon Block.","authors":"Melissa Lu, Mohammed Al Kaabi, Cynthia Xin-Ya Qian","doi":"10.1097/ICB.0000000000001687","DOIUrl":"https://doi.org/10.1097/ICB.0000000000001687","url":null,"abstract":"<p><strong>Purpose: </strong>To report and discuss a case of brainstem anesthesia and contralateral amaurosis following a sub-Tenon block.</p><p><strong>Methods: </strong>Single surgical case report of a patient who underwent an uncomplicated pars plana vitrectomy for an epiretinal membrane peel in the left eye following a sub-Tenon anesthesia technique.</p><p><strong>Results: </strong>Post-operatively, the patient experienced symptoms of brainstem anesthesia as well as akinesia, a non-reactive pupil, and reduced visual acuity in the contralateral eye which gradually resolved within 24 hours. Imaging was within normal limits and did not reveal any anatomic abnormalities.</p><p><strong>Conclusion: </strong>Despite the use of a blunt-ended cannula in a sub-Tenon block, there is a non-negligible risk of it penetrating surrounding structures such as the optic nerve sheath. This case emphasizes the importance of monitoring for brainstem anesthesia and contralateral eye affections in patients both intra- and post-operatively.</p>","PeriodicalId":53580,"journal":{"name":"Retinal Cases and Brief Reports","volume":" ","pages":""},"PeriodicalIF":0.0,"publicationDate":"2024-10-18","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"142562546","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
Pub Date : 2024-10-18DOI: 10.1097/ICB.0000000000001680
Justin Muste, Kimberly Baynes, Sunil K Srivastava
Purpose: To describe the first case of Coccidioides posadasii endophthalmitis.
Methods: A 41-year-old man referred for acute retinal necrosis (ARN) failing 3 months of outpatient treatment was referred for further management. Three consecutive pars plana vitrectomies were required to identify C. posadasii as the source of the endophthalmitis. Two amphotericin intravitreal injections were delivered in addition to three foscarnet injections, two clindamycin, and one ceftazidime. Therapeutic intervention was complicated by patient compliance.
Results: Despite repeated surgical and medical intervention, vision was lost. The patient later developed fungal meningitis.
Conclusions: Given parallels between C. posadasii and C. immitis endogenous endophthalmitis, a combined medical and surgical approach can combat C. posadasii in cases of good patient compliance with therapy.
{"title":"Coccidioidal Endophthalmitis Secondary to Coccidoides Posadasii: A Rare Infection in Humans.","authors":"Justin Muste, Kimberly Baynes, Sunil K Srivastava","doi":"10.1097/ICB.0000000000001680","DOIUrl":"https://doi.org/10.1097/ICB.0000000000001680","url":null,"abstract":"<p><strong>Purpose: </strong>To describe the first case of Coccidioides posadasii endophthalmitis.</p><p><strong>Methods: </strong>A 41-year-old man referred for acute retinal necrosis (ARN) failing 3 months of outpatient treatment was referred for further management. Three consecutive pars plana vitrectomies were required to identify C. posadasii as the source of the endophthalmitis. Two amphotericin intravitreal injections were delivered in addition to three foscarnet injections, two clindamycin, and one ceftazidime. Therapeutic intervention was complicated by patient compliance.</p><p><strong>Results: </strong>Despite repeated surgical and medical intervention, vision was lost. The patient later developed fungal meningitis.</p><p><strong>Conclusions: </strong>Given parallels between C. posadasii and C. immitis endogenous endophthalmitis, a combined medical and surgical approach can combat C. posadasii in cases of good patient compliance with therapy.</p>","PeriodicalId":53580,"journal":{"name":"Retinal Cases and Brief Reports","volume":" ","pages":""},"PeriodicalIF":0.0,"publicationDate":"2024-10-18","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"142562592","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
Pub Date : 2024-10-18DOI: 10.1097/ICB.0000000000001678
Breno Nery, Rayssa Rocha Vieira Da Silva, Rafael Emanuel Fontes Costa Araújo, Giovana De Souza Faria, Isabela Grozza Cestari, Thayná Dantas Souto Fernandes, Ana Carolina Brandão Paganini, Laura Maria Alves De Oliveira Souza, João Pedro Ferreira Braga, Leonardo Braz De Sousa Filho, José Alencar De Sousa Segundo, Huber Martins Vasconcelos Júnior
Purpose: The objective of this article is to report a case of silicone oil migration into intraocular packing in cases of retinal detachment, while reviewing the existing literature on the various mechanisms and locations of this migration. However, it is important to highlight that the use of this material is not free from complications.
Method: This article is a case report with a literature review. The information was obtained through a review of the medical record, interview and photographic record of the patient's diagnostic methods. PubMed was used as a database to perform the search for selected articles and literature review, selecting articles published from the year 2016.
Results/discussion: The exact mechanism of migration of intraocular silicone oil to the central nervous system is still unclear, being a rare complication that may be related to several factors. We report a case in which the patient underwent vitrectomy surgery and, after several years, presented a complication related to the migration of intraocular silicone oil to the right frontal horn and third ventricle, causing headache. Conclusion: Knowledge about migration of intraocular silicone oil into the central nervous system (CNS) is extremely important, since it manifests itself in several clinical and radiological findings, which can lead to incorrect diagnoses. Thus, it is suggested the follow- up of the patient after introduction of silicone oil to avoid complications that threaten results.
{"title":"SYMPTOMATIC MIGRATION OF SILICONE OIL TO THE VENTRICULAR SYSTEM.","authors":"Breno Nery, Rayssa Rocha Vieira Da Silva, Rafael Emanuel Fontes Costa Araújo, Giovana De Souza Faria, Isabela Grozza Cestari, Thayná Dantas Souto Fernandes, Ana Carolina Brandão Paganini, Laura Maria Alves De Oliveira Souza, João Pedro Ferreira Braga, Leonardo Braz De Sousa Filho, José Alencar De Sousa Segundo, Huber Martins Vasconcelos Júnior","doi":"10.1097/ICB.0000000000001678","DOIUrl":"https://doi.org/10.1097/ICB.0000000000001678","url":null,"abstract":"<p><strong>Purpose: </strong>The objective of this article is to report a case of silicone oil migration into intraocular packing in cases of retinal detachment, while reviewing the existing literature on the various mechanisms and locations of this migration. However, it is important to highlight that the use of this material is not free from complications.</p><p><strong>Method: </strong>This article is a case report with a literature review. The information was obtained through a review of the medical record, interview and photographic record of the patient's diagnostic methods. PubMed was used as a database to perform the search for selected articles and literature review, selecting articles published from the year 2016.</p><p><strong>Results/discussion: </strong>The exact mechanism of migration of intraocular silicone oil to the central nervous system is still unclear, being a rare complication that may be related to several factors. We report a case in which the patient underwent vitrectomy surgery and, after several years, presented a complication related to the migration of intraocular silicone oil to the right frontal horn and third ventricle, causing headache. Conclusion: Knowledge about migration of intraocular silicone oil into the central nervous system (CNS) is extremely important, since it manifests itself in several clinical and radiological findings, which can lead to incorrect diagnoses. Thus, it is suggested the follow- up of the patient after introduction of silicone oil to avoid complications that threaten results.</p>","PeriodicalId":53580,"journal":{"name":"Retinal Cases and Brief Reports","volume":" ","pages":""},"PeriodicalIF":0.0,"publicationDate":"2024-10-18","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"142562651","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
Pub Date : 2024-10-18DOI: 10.1097/ICB.0000000000001682
Chloe Y Li, Erin E Flynn, Stanley Chang
Purpose: To characterize two cases of focal outer retinal atrophy and hypotony after vitrectomy.
Methods: Retrospective chart review of two patients' records between 2019 and 2023.
Results: Patient 1 underwent vitrectomy, epiretinal membrane peel, and cataract extraction for visually significant macular pucker. She developed hypotony without a wound leak and was noted to have a focal parafoveal area of ellipsoid zone disruption by 1 week post-operatively, which evolved into outer retinal and chorioretinal atrophy within 6 weeks after surgery. This area of atrophy remained stable in size, but the patient later reported a paracentral scotoma. Patient 2 had multiple previous surgeries for retinal detachment with proliferative vitreoretinopathy. Seven years later, the IOL dislocated and was exchanged with scleral fixation of a new IOL. On post-operative day 1, he had hypotony with macular folds secondary to a leaking sclerotomy wound. The sclerotomies were sutured on post-operative day 3, and his intraocular pressure normalized. However, he developed a central, focal area of chorioretinal atrophy within 1 week of the initial surgery. The size of this area of atrophy remained stable over years but resulted in reduced central vision.
Conclusion: Hypotony following vitrectomy may rarely predispose patients to the development of focal chorioretinal atrophy.
{"title":"Macular Outer Retinal Atrophy Following Vitrectomy and Hypotony: Two Cases.","authors":"Chloe Y Li, Erin E Flynn, Stanley Chang","doi":"10.1097/ICB.0000000000001682","DOIUrl":"https://doi.org/10.1097/ICB.0000000000001682","url":null,"abstract":"<p><strong>Purpose: </strong>To characterize two cases of focal outer retinal atrophy and hypotony after vitrectomy.</p><p><strong>Methods: </strong>Retrospective chart review of two patients' records between 2019 and 2023.</p><p><strong>Results: </strong>Patient 1 underwent vitrectomy, epiretinal membrane peel, and cataract extraction for visually significant macular pucker. She developed hypotony without a wound leak and was noted to have a focal parafoveal area of ellipsoid zone disruption by 1 week post-operatively, which evolved into outer retinal and chorioretinal atrophy within 6 weeks after surgery. This area of atrophy remained stable in size, but the patient later reported a paracentral scotoma. Patient 2 had multiple previous surgeries for retinal detachment with proliferative vitreoretinopathy. Seven years later, the IOL dislocated and was exchanged with scleral fixation of a new IOL. On post-operative day 1, he had hypotony with macular folds secondary to a leaking sclerotomy wound. The sclerotomies were sutured on post-operative day 3, and his intraocular pressure normalized. However, he developed a central, focal area of chorioretinal atrophy within 1 week of the initial surgery. The size of this area of atrophy remained stable over years but resulted in reduced central vision.</p><p><strong>Conclusion: </strong>Hypotony following vitrectomy may rarely predispose patients to the development of focal chorioretinal atrophy.</p>","PeriodicalId":53580,"journal":{"name":"Retinal Cases and Brief Reports","volume":" ","pages":""},"PeriodicalIF":0.0,"publicationDate":"2024-10-18","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"142562645","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
Pub Date : 2024-10-17DOI: 10.1097/ICB.0000000000001679
Fernanda Galante Dourado, Daniel de Queiroz Omote, Paula Dandara Correia de Pinho, Sérgio Luis Gianotti Pimentel
Purpose: Describe a case of symmetric bilateral macular atrophy as an ophthalmological manifestation of Mucolipidosis type 3.
Methods: Multimodal retinal imaging evaluation was performed, with color fundus photograph, fundus autofluorescence, fluorescein angiography and optical coherence tomography. Genetic testing confirmed the systemic diagnosis.
Results: Genetic testing confirmed the diagnosis of Mucolipidosis type 3 and macular bilateral atrophy was considered a rare manifestation of the systemic disease.
Discussion: Ophthalmological manifestations in mucolipidosis are rare, with corneal opacities being most reported. Our patient presented bilateral macular atrophy, which had not been previously described in association with the disease, and is important to consider different diagnosis.
Conclusion: It is important to consider different diagnosis, such as retinal dystrophies and conditions causing macular atrophy, and this case highlights the importance of considering rare ophthalmological manifestations in syndromic diseases.
{"title":"Symmetric Bilateral Macular Atrophy in Mucolipidosis type 3: A Rare Manifestation.","authors":"Fernanda Galante Dourado, Daniel de Queiroz Omote, Paula Dandara Correia de Pinho, Sérgio Luis Gianotti Pimentel","doi":"10.1097/ICB.0000000000001679","DOIUrl":"https://doi.org/10.1097/ICB.0000000000001679","url":null,"abstract":"<p><strong>Purpose: </strong>Describe a case of symmetric bilateral macular atrophy as an ophthalmological manifestation of Mucolipidosis type 3.</p><p><strong>Methods: </strong>Multimodal retinal imaging evaluation was performed, with color fundus photograph, fundus autofluorescence, fluorescein angiography and optical coherence tomography. Genetic testing confirmed the systemic diagnosis.</p><p><strong>Results: </strong>Genetic testing confirmed the diagnosis of Mucolipidosis type 3 and macular bilateral atrophy was considered a rare manifestation of the systemic disease.</p><p><strong>Discussion: </strong>Ophthalmological manifestations in mucolipidosis are rare, with corneal opacities being most reported. Our patient presented bilateral macular atrophy, which had not been previously described in association with the disease, and is important to consider different diagnosis.</p><p><strong>Conclusion: </strong>It is important to consider different diagnosis, such as retinal dystrophies and conditions causing macular atrophy, and this case highlights the importance of considering rare ophthalmological manifestations in syndromic diseases.</p>","PeriodicalId":53580,"journal":{"name":"Retinal Cases and Brief Reports","volume":" ","pages":""},"PeriodicalIF":0.0,"publicationDate":"2024-10-17","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"142562649","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
Pub Date : 2024-10-16DOI: 10.1097/ICB.0000000000001674
Christopher Sun, Tien-En Tan, Yasmin Bylstra, Nathalie P Y Chiam, Ranjana S Mathur, Choi Mun Chan, Li Yu Chen, Joey S Z Poh, Rachael W C Tang, Chui Ming Gemmy Cheung, Beau J Fenner
Purpose: We describe an atypical presentation of an 11-year-old female with enhanced S-cone syndrome (ESCS).
Methods: Case report. The patient underwent a thorough ophthalmic examination and investigations such as colour fundus photography, optical coherence tomography, fundus autofluorescence, fluorescein and indocyanine angiography, an electroretinogram and genetic testing. After determining the presence of secondary choroidal neovascularization, we treated her with intravitreal Ranibizumab injections. We present her progress and a brief literature review about ESCS.
Results: An 11-year-old hyperopic female with no known family history of retinal disease or nyctalopia presented with bilateral reduced visual acuity (20/100 OD, 20/200 OS). Examination disclosed bilateral macular choroid neovascularization (CNV) with retinochoroidal anastomosis on the left eye, bilateral nummular deposits at the superior macular arcades. All three of her siblings, aged 7-14, were asymptomatic but were also hyperopic and had intraretinal schisis and focal loss or attenuation of the ellipsoid zone on optical coherence tomography (OCT). Electrophysiology showed a reduced scotopic response and a dramatically enhanced response to full field blue light stimuli meant to elicit a response primarily from S-cones. Genetic testing confirmed the presence of biallelic NR2E3 variants. Treatment with monthly intravitreal anti-VEGF resulted in improved visual acuity of 20/30 on the right eye, while the left eye had a persistent nodular scar and visual acuity remained at 20/200.
Conclusion: We describe a case of bilateral CNV in a young patient with enhanced S-cone syndrome, discuss differentials and treatment approaches. This case highlights the risk of CNV in ESCS and the importance of family screening and follow-up in affected relatives.
{"title":"NR2E3-Associated Retinopathy Presenting with Bilateral Choroidal Neovascularization.","authors":"Christopher Sun, Tien-En Tan, Yasmin Bylstra, Nathalie P Y Chiam, Ranjana S Mathur, Choi Mun Chan, Li Yu Chen, Joey S Z Poh, Rachael W C Tang, Chui Ming Gemmy Cheung, Beau J Fenner","doi":"10.1097/ICB.0000000000001674","DOIUrl":"https://doi.org/10.1097/ICB.0000000000001674","url":null,"abstract":"<p><strong>Purpose: </strong>We describe an atypical presentation of an 11-year-old female with enhanced S-cone syndrome (ESCS).</p><p><strong>Methods: </strong>Case report. The patient underwent a thorough ophthalmic examination and investigations such as colour fundus photography, optical coherence tomography, fundus autofluorescence, fluorescein and indocyanine angiography, an electroretinogram and genetic testing. After determining the presence of secondary choroidal neovascularization, we treated her with intravitreal Ranibizumab injections. We present her progress and a brief literature review about ESCS.</p><p><strong>Results: </strong>An 11-year-old hyperopic female with no known family history of retinal disease or nyctalopia presented with bilateral reduced visual acuity (20/100 OD, 20/200 OS). Examination disclosed bilateral macular choroid neovascularization (CNV) with retinochoroidal anastomosis on the left eye, bilateral nummular deposits at the superior macular arcades. All three of her siblings, aged 7-14, were asymptomatic but were also hyperopic and had intraretinal schisis and focal loss or attenuation of the ellipsoid zone on optical coherence tomography (OCT). Electrophysiology showed a reduced scotopic response and a dramatically enhanced response to full field blue light stimuli meant to elicit a response primarily from S-cones. Genetic testing confirmed the presence of biallelic NR2E3 variants. Treatment with monthly intravitreal anti-VEGF resulted in improved visual acuity of 20/30 on the right eye, while the left eye had a persistent nodular scar and visual acuity remained at 20/200.</p><p><strong>Conclusion: </strong>We describe a case of bilateral CNV in a young patient with enhanced S-cone syndrome, discuss differentials and treatment approaches. This case highlights the risk of CNV in ESCS and the importance of family screening and follow-up in affected relatives.</p>","PeriodicalId":53580,"journal":{"name":"Retinal Cases and Brief Reports","volume":" ","pages":""},"PeriodicalIF":0.0,"publicationDate":"2024-10-16","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"142958890","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
Pub Date : 2024-10-16DOI: 10.1097/ICB.0000000000001671
Vichar Trivedi, Qisheng You, Rao Me, Patrick Sy Lee, Kim Le, David Tran, Xihui Lin
Purpose: To report two patient cases demonstrating the management of refractory macular holes through the application of temporary thin amniotic membrane grafts, followed by subsequent graft removal upon achieving hole closure.
Methods: Comprehensive chart and literature review was conducted utilizing the PubMed database.
Results: We describe two patients who underwent repeat pars plana vitrectomy for treatment of refractory macular holes. In both cases, the epi-retinal placement of a thin amniotic membrane graft (AMG) was done to achieve hole closure. Following a period of retinal stabilization, the amniotic membranes were removed due to the healthy appearance of the outer retinal layers and the ellipsoid zone, ultimately resulting in an improved final visual acuity in both patients.
Conclusion: This case series demonstrates a new approach of using a temporary AMG to close refractory macular holes. After graft removal, both patients reported enhanced visual acuity and subjective visual improvement, accompanied by the stable closure of macular holes on serial OCT scans.
目的:报告两例通过应用临时薄羊膜移植物治疗难治性黄斑孔,并在孔闭合后移除移植物的病例:方法:利用 PubMed 数据库进行全面的病历和文献回顾:结果:我们描述了两名为治疗难治性黄斑孔而再次接受玻璃体旁切除术的患者。在这两个病例中,都在视网膜外放置了薄羊膜移植片(AMG),以达到封闭孔洞的目的。视网膜稳定一段时间后,由于视网膜外层和椭圆体区外观健康,羊膜被移除,最终两名患者的最终视力都得到了改善:本系列病例展示了一种使用临时 AMG 封闭难治性黄斑孔的新方法。移植物移除后,两名患者的视力均有所提高,主观视力也有所改善,同时在连续的 OCT 扫描中,黄斑孔也稳定闭合。
{"title":"TEMPORARY AMNIOTIC MEMBRANE GRAFT PLACEMENT FOR TREATMENT OF REFRACTORY MACULAR HOLES.","authors":"Vichar Trivedi, Qisheng You, Rao Me, Patrick Sy Lee, Kim Le, David Tran, Xihui Lin","doi":"10.1097/ICB.0000000000001671","DOIUrl":"https://doi.org/10.1097/ICB.0000000000001671","url":null,"abstract":"<p><strong>Purpose: </strong>To report two patient cases demonstrating the management of refractory macular holes through the application of temporary thin amniotic membrane grafts, followed by subsequent graft removal upon achieving hole closure.</p><p><strong>Methods: </strong>Comprehensive chart and literature review was conducted utilizing the PubMed database.</p><p><strong>Results: </strong>We describe two patients who underwent repeat pars plana vitrectomy for treatment of refractory macular holes. In both cases, the epi-retinal placement of a thin amniotic membrane graft (AMG) was done to achieve hole closure. Following a period of retinal stabilization, the amniotic membranes were removed due to the healthy appearance of the outer retinal layers and the ellipsoid zone, ultimately resulting in an improved final visual acuity in both patients.</p><p><strong>Conclusion: </strong>This case series demonstrates a new approach of using a temporary AMG to close refractory macular holes. After graft removal, both patients reported enhanced visual acuity and subjective visual improvement, accompanied by the stable closure of macular holes on serial OCT scans.</p>","PeriodicalId":53580,"journal":{"name":"Retinal Cases and Brief Reports","volume":" ","pages":""},"PeriodicalIF":0.0,"publicationDate":"2024-10-16","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"142512823","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
Purpose: This report describes the case of a 13-month-old boy diagnosed with neurofibromatosis type 1, who presented with retinal vascular abnormalities including extensive non-perfusion and neovascularization. We also discuss the observed changes following photocoagulation treatment.
Methods: A 13-month-old boy presented to the Department of Ophthalmology at Peking University People's Hospital with a reduction in the width of the left palpebral fissure for the past 6 months.
Results: The boy exhibited more than six café-au-lait spots larger than 5 mm in diameter on his trunk and legs. Fundus examination of the left eye revealed significant neovascularization in the temporal periphery of the retina, with late leakage and non-perfusion also noted temporally in fluorescein angiography (FA). Magnetic resonance imaging of the brain and orbits showed an enlarged left sphenoid body, a widened left cavernous sinus, and a large plexiform neurofibroma. Laser treatment was performed on the left eye. Five months later, the neovascularization was controlled.
Conclusion: Careful fundus examinations and systemic reviews, especially FA, are essential. Timely laser treatment is crucial for controlling disease progression and preventing retinal detachment.
{"title":"Retinal vascular abnormality of non-perfusion and neovascularization in a toddler with neurofibromatosis type 1.","authors":"Qiaozhu Zeng, Yusheng Zhong, Tong Qian, Yong Cheng","doi":"10.1097/ICB.0000000000001673","DOIUrl":"https://doi.org/10.1097/ICB.0000000000001673","url":null,"abstract":"<p><strong>Purpose: </strong>This report describes the case of a 13-month-old boy diagnosed with neurofibromatosis type 1, who presented with retinal vascular abnormalities including extensive non-perfusion and neovascularization. We also discuss the observed changes following photocoagulation treatment.</p><p><strong>Methods: </strong>A 13-month-old boy presented to the Department of Ophthalmology at Peking University People's Hospital with a reduction in the width of the left palpebral fissure for the past 6 months.</p><p><strong>Results: </strong>The boy exhibited more than six café-au-lait spots larger than 5 mm in diameter on his trunk and legs. Fundus examination of the left eye revealed significant neovascularization in the temporal periphery of the retina, with late leakage and non-perfusion also noted temporally in fluorescein angiography (FA). Magnetic resonance imaging of the brain and orbits showed an enlarged left sphenoid body, a widened left cavernous sinus, and a large plexiform neurofibroma. Laser treatment was performed on the left eye. Five months later, the neovascularization was controlled.</p><p><strong>Conclusion: </strong>Careful fundus examinations and systemic reviews, especially FA, are essential. Timely laser treatment is crucial for controlling disease progression and preventing retinal detachment.</p>","PeriodicalId":53580,"journal":{"name":"Retinal Cases and Brief Reports","volume":" ","pages":""},"PeriodicalIF":0.0,"publicationDate":"2024-10-10","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"142480601","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
Pub Date : 2024-10-08DOI: 10.1097/ICB.0000000000001665
Claudio Iovino, Luciana Damiano, Valerio Piccirillo, Francesco Testa, Giuseppe Giannaccare, Francesca Simonelli
Purpose: To report a case of giant pigment epithelium detachment (PED) secondary to chronic central serous chorioretinopathy (cCSC) successfully treated with photobiomodulation (PBM).
Methods: Case report.
Results: A 55-year-old man complained a worsening of vision in the left eye (LE) over the last 18 months.A complete ophthalmological evaluation encompassing best corrected visual acuity (BCVA) measurement, spectral-domain optical coherence tomography (SD-OCT), fundus autofluorescence (FAF), fluorescein angiography (FA) and microperimetry (MP) was performed.SD-OCT showed several small PEDs around the macula in the right eye and a giant macular serous PED with subretinal fluid (SRF) in the LE. FA disclosed multiple hyperfluorescent roundish areas in the posterior pole bilaterally, with a macular pooling in the LE.A diagnosis of cCSC was made, and patient underwent PBM with one session for week for four weeks, followed by one session bi-weekly for two months.SD-OCT of the LE showed a gradual flattening of the macular PED at 3 months, with a complete regression of the latter and of the SRF at 6- and 12-month follow up with no residual signs of chorioretinal atrophy on FAF. BCVA improved from 20/80 at baseline to 20/25 at the last follow up, and this functional improvement was further confirmed by MP.
Conclusions: PBM can be considered a safe and effective treatment strategy for the management of cCSC with SRF and serous PED.
{"title":"Photobiomodulation Therapy for Serous Pigment Epithelium Detachment in Chronic Central Serous Chorioretinopathy.","authors":"Claudio Iovino, Luciana Damiano, Valerio Piccirillo, Francesco Testa, Giuseppe Giannaccare, Francesca Simonelli","doi":"10.1097/ICB.0000000000001665","DOIUrl":"https://doi.org/10.1097/ICB.0000000000001665","url":null,"abstract":"<p><strong>Purpose: </strong>To report a case of giant pigment epithelium detachment (PED) secondary to chronic central serous chorioretinopathy (cCSC) successfully treated with photobiomodulation (PBM).</p><p><strong>Methods: </strong>Case report.</p><p><strong>Results: </strong>A 55-year-old man complained a worsening of vision in the left eye (LE) over the last 18 months.A complete ophthalmological evaluation encompassing best corrected visual acuity (BCVA) measurement, spectral-domain optical coherence tomography (SD-OCT), fundus autofluorescence (FAF), fluorescein angiography (FA) and microperimetry (MP) was performed.SD-OCT showed several small PEDs around the macula in the right eye and a giant macular serous PED with subretinal fluid (SRF) in the LE. FA disclosed multiple hyperfluorescent roundish areas in the posterior pole bilaterally, with a macular pooling in the LE.A diagnosis of cCSC was made, and patient underwent PBM with one session for week for four weeks, followed by one session bi-weekly for two months.SD-OCT of the LE showed a gradual flattening of the macular PED at 3 months, with a complete regression of the latter and of the SRF at 6- and 12-month follow up with no residual signs of chorioretinal atrophy on FAF. BCVA improved from 20/80 at baseline to 20/25 at the last follow up, and this functional improvement was further confirmed by MP.</p><p><strong>Conclusions: </strong>PBM can be considered a safe and effective treatment strategy for the management of cCSC with SRF and serous PED.</p>","PeriodicalId":53580,"journal":{"name":"Retinal Cases and Brief Reports","volume":" ","pages":""},"PeriodicalIF":0.0,"publicationDate":"2024-10-08","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"142904107","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}