Stefano Mennecozzi, Elena Spada, Riccardo Sacconi, Francesco Bandello, Giuseppe Querques
{"title":"Optical coherence tomography features of atypical congenital hypertrophy of retinal pigment epithelium in a patient with familial adenomatous polyposis.","authors":"Stefano Mennecozzi, Elena Spada, Riccardo Sacconi, Francesco Bandello, Giuseppe Querques","doi":"10.1097/ICB.0000000000001722","DOIUrl":"https://doi.org/10.1097/ICB.0000000000001722","url":null,"abstract":"<p><strong>Purpose: </strong>To describe optical coherence tomography (OCT) features of atypical congenital hypertrophy of retinal pigment epithelium (CHRPE) associated with familial adenomatous polyposis (FAP).</p><p><strong>Methods: </strong>Case report.</p><p><strong>Results: </strong>A 34-year-old patient diagnosed with FAP was referred to our clinic for fundus examination. We characterized the lesions we found using multimodal imaging (infrared, autofluorescence, OCT and widefield and ultra-widefield fundus image). We described 3 possible stages of progression of the lesions in a single patient and we found a possible evolutionary stage with a \"2 binary reflective sign\".</p><p><strong>Conclusions: </strong>CHRPE lesions can show a progression from only retinal pigment epithelium (RPE) hyperreflectivity to complete outer retinal atrophy and even intraretinal RPE migration. We hypothesize that the 2 binary reflective sign could represent an hemorrhage between RPE and outer retina which could lead, together with the reduced catabolic activity of pathological RPE, to overlying retinal atrophy.</p>","PeriodicalId":53580,"journal":{"name":"Retinal Cases and Brief Reports","volume":" ","pages":""},"PeriodicalIF":0.0,"publicationDate":"2025-02-03","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143191146","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}
{"title":"Persistent macular hole and cystoid macular edema treated with pars plana vitrectomy after failure with topical therapy.","authors":"Landon J Rohowetz, Harry W Flynn","doi":"10.1097/ICB.0000000000001721","DOIUrl":"https://doi.org/10.1097/ICB.0000000000001721","url":null,"abstract":"<p><strong>Purpose: </strong>To report a patient with a persistent macular hole and cystoid macular edema (CME) successfully treated with pars plana vitrectomy and internal limiting membrane peeling after failed topical therapy.</p><p><strong>Methods: </strong>Case report.</p><p><strong>Results: </strong>A 70-year-old male presented with a history of decreased vision in the right eye for 3-4 months. Best-corrected visual acuity at the initial examination was 20/80 in the right eye and 20/30 in the left. Optical coherence tomography demonstrated vitreomacular traction with a full-thickness macular hole and prominent CME in the right eye. He was started on topical prednisolone acetate and ketorolac. The width of the hole initially decreased but eventually returned to baseline size despite continued topical therapy. Conservative management with topical therapy was continued in accordance with the patient's preferences. He later underwent phacoemulsification 20 months after presentation without significant improvement in visual acuity. Pars plana vitrectomy with internal limiting membrane peeling and C3F8 tamponade was ultimately performed 2 years after initial presentation. Postoperatively, the macular hole was closed and visual acuity improved to 20/40 at 3 months.</p><p><strong>Conclusion: </strong>Macular holes with significant CME may close with topical therapy alone. In persistent or recalcitrant cases, delayed surgical intervention to close the macular hole can be successful even at 2 years.</p>","PeriodicalId":53580,"journal":{"name":"Retinal Cases and Brief Reports","volume":" ","pages":""},"PeriodicalIF":0.0,"publicationDate":"2025-01-31","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143191151","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}
Rola N Hamam, Mahdi M Hassoun, Abdallah G Rebeiz, Dalia El Hadi, Ziad F Bashshur
{"title":"Treatment of vision-threatening branch retinal artery occlusion following coronary angioplasty with surgical displacement of embolus.","authors":"Rola N Hamam, Mahdi M Hassoun, Abdallah G Rebeiz, Dalia El Hadi, Ziad F Bashshur","doi":"10.1097/ICB.0000000000001717","DOIUrl":"https://doi.org/10.1097/ICB.0000000000001717","url":null,"abstract":"<p><strong>Purpose: </strong>To report a successful case of vision restoration and macular reperfusion following branch retinal artery occlusion (BRAO) using pars plana vitrectomy with undermining the artery off the retinal bed.</p><p><strong>Methods: </strong>This case report involves a 75-year-old patient who was diagnosed immediately with BRAO following cardiac catheterization procedure. An embolus at the superior retinal artery bifurcation was noted. Initial treatments were ineffective, leading to a pars plana vitrectomy and mechanical elevation of the artery, performed six hours post-occlusion.</p><p><strong>Results: </strong>The surgical intervention successfully dislodged the embolus, resulting in significant visual improvement. Post-operative day 1, 4, and 14 assessments showed visual acuity improvement to counting fingers at 2 meters, 20/40, and 20/20, respectively. Fundoscopic examination and OCT angiography confirmed improved macular perfusion and normalization of the flow signal in the affected artery.</p><p><strong>Conclusion: </strong>This case highlights the efficacy of pars plana vitrectomy and mechanical elevation of the artery for BRAO where the embolus can be visualized. Prompt identification and referral for surgical intervention in BRAO cases with poor prognosis are crucial for favorable visual outcomes. Further research is needed to assess the efficacy of this surgical technique and other interventions for managing BRAO.</p>","PeriodicalId":53580,"journal":{"name":"Retinal Cases and Brief Reports","volume":" ","pages":""},"PeriodicalIF":0.0,"publicationDate":"2025-01-17","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143025774","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}
{"title":"Drusen Regression Following Macular Hole Surgery: A Case Report.","authors":"Shannan Berzack, Prashant K Parekh","doi":"10.1097/ICB.0000000000001720","DOIUrl":"https://doi.org/10.1097/ICB.0000000000001720","url":null,"abstract":"<p><strong>Purpose: </strong>To report a case of drusen regression following pars plana vitrectomy with internal limiting membrane peel (ILMP) in a patient with a full-thickness macular hole and dry age-related macular degeneration (AMD).</p><p><strong>Methods: </strong>A 67-year-old gentleman presented in April 2024 with a full-thickness macular hole in OS and intermediate dry AMD OU. The patient underwent pars plana vitrectomy, ILMP, and an injection of sulfur hexafluoride gas for macular hole repair in OS. The patient's macular changes were monitored through visual acuity, fundus examination and optical coherence tomography (OCT) imaging at baseline and follow-up visits.</p><p><strong>Results: </strong>The macular hole was closed and there was early stability of the macular drusen in OU. At a five-month follow-up in September 2024, he reported improved vision from 20/100 baseline to 20/40. Fundus examination as well as OCT imaging demonstrated a significant reduction of macular drusen in the operated eye, which was not observed in the fellow eye.</p><p><strong>Conclusion: </strong>This case demonstrates successful surgical management of a full-thickness macular hole in a patient with concomitant drusen regression. The regression of drusen was limited to the operated eye, which suggests that surgical interventions may influence the course of AMD in certain patients.</p>","PeriodicalId":53580,"journal":{"name":"Retinal Cases and Brief Reports","volume":" ","pages":""},"PeriodicalIF":0.0,"publicationDate":"2025-01-17","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143025772","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}
Andrea Trinco, Francesco Romano, Alessandro Invernizzi, Chiara Zaffalon, Francesca Bosello, Stefano Casati, Federico Zicarelli, Rossella D'Introno, Giovanni Staurenghi, Anna Paola Salvetti
{"title":"Punctate Inner Choroidopathy (PIC)-like Reaction in Stage 3 Extensive Macular Atrophy with Pseudodrusen-like Appearance (EMAP).","authors":"Andrea Trinco, Francesco Romano, Alessandro Invernizzi, Chiara Zaffalon, Francesca Bosello, Stefano Casati, Federico Zicarelli, Rossella D'Introno, Giovanni Staurenghi, Anna Paola Salvetti","doi":"10.1097/ICB.0000000000001719","DOIUrl":"https://doi.org/10.1097/ICB.0000000000001719","url":null,"abstract":"<p><strong>Purpose: </strong>To describe a rare complication in a patient with extensive macular atrophy with pseudodrusen-like appearance (EMAP), suggesting immune dysregulation in advanced stages of the disease.</p><p><strong>Methods: </strong>Case Report. Multimodal imaging -including true-color fundus photography, blue autofluorescence, high-resolution optical coherence tomography (Hi-Res OCT), swept-source OCT angiography, and dye-based angiography- was used to evaluate retinal alterations.</p><p><strong>Results: </strong>A 53-year-old woman with stage 3 EMAP presented with moderately hyperreflective subretinal material co-localizing with a large rupture of the Bruch's membrane (BrM) in the right eye. Multimodal imaging ruled out macular neovascularization, suggesting a diagnosis of punctate inner choroidopathy (PIC)-like reaction. Treatment with oral steroids led to complete regression of the subretinal inflammatory lesion.</p><p><strong>Conclusions: </strong>This case highlights a novel inflammatory complication in EMAP. Our findings emphasize a potential role of immune dysregulation in late-stage EMAP and underscore the value of multimodal imaging in the management and follow-up of these patients.</p>","PeriodicalId":53580,"journal":{"name":"Retinal Cases and Brief Reports","volume":" ","pages":""},"PeriodicalIF":0.0,"publicationDate":"2025-01-15","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143016158","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}
Afonso Murta, Catarina Mota, Bruna Cunha, Nuno Rodrigues Alves, Christopher Saunders, Sofia Pinheiro, Lívio Costa, Rita Anjos
{"title":"Multicentric Castleman's Disease presenting with bilateral panuveitis.","authors":"Afonso Murta, Catarina Mota, Bruna Cunha, Nuno Rodrigues Alves, Christopher Saunders, Sofia Pinheiro, Lívio Costa, Rita Anjos","doi":"10.1097/ICB.0000000000001715","DOIUrl":"https://doi.org/10.1097/ICB.0000000000001715","url":null,"abstract":"<p><strong>Purpose: </strong>To report a rare case of Multicentric Castleman's Disease presenting with bilateral panuveitis.</p><p><strong>Methods: </strong>Case report.</p><p><strong>Results: </strong>A 65-years-old caucasian man presented with progressive blurred vision in both eyes for two weeks, along with weight loss, polyarthralgias and reduced muscle strength persisting for about a year. Examination revealed bilateral panuveitis after intraocular lymphoma being initially considered. CT scans showed multiple lymph node enlargements suggestive of lymphoproliferative disorder. Excisional biopsy of an axillary lymph node confirmed the diagnosis of plasmacytic type Castleman's disease. Treatment was started with topical dexamethasone and tropicamide for anterior chamber inflammation, followed by oral prednisolone. After definitive diagnosis, treatment with siltuximab was initiated, which led to significant improvement in panuveitis and systemic symptoms. The patient became off corticosteroids and continued on siltuximab with stable visual acuity and low-grade vitritis.</p><p><strong>Conclusion: </strong>To the best of our knowledge we described the first case in which the diagnosis of Multicentric Castleman's Disease was established through the investigation of bilateral panuveitis, which was successfully managed with corticosteroids and siltuximab.</p>","PeriodicalId":53580,"journal":{"name":"Retinal Cases and Brief Reports","volume":" ","pages":""},"PeriodicalIF":0.0,"publicationDate":"2025-01-14","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143016152","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}
Sebastiano Del Fabbro, Lorenzo Bianco, Alessio Antropoli, Soufiane Bousyf, Lorenza Bruno, Alessandro Arrigo, Angela Malegori, Maria Vittoria Cicinelli, Francesco Bandello, Maurizio Battaglia Parodi
{"title":"Multimodal imaging findings of the fellow eye in Stellate Nonhereditary Idiopathic Foveomacular Retinoschisis (SNIFR).","authors":"Sebastiano Del Fabbro, Lorenzo Bianco, Alessio Antropoli, Soufiane Bousyf, Lorenza Bruno, Alessandro Arrigo, Angela Malegori, Maria Vittoria Cicinelli, Francesco Bandello, Maurizio Battaglia Parodi","doi":"10.1097/ICB.0000000000001718","DOIUrl":"https://doi.org/10.1097/ICB.0000000000001718","url":null,"abstract":"<p><strong>Purpose: </strong>This study examines the multimodal imaging (MMI) findings in two cases of unilateral Stellate Nonhereditary Idiopathic Foveomacular Retinoschisis (SNIFR), including detailed findings from the unaffected fellow eye.</p><p><strong>Methods: </strong>Macular spectral domain optical coherence tomography (OCT) and 3x3 mm optical coherence tomography angiography (OCTA), microperimetry, full-field electroretinography (ff-ERG) for both the affected and the fellow eye were reviewed.</p><p><strong>Results: </strong>The MMI findings were consistent across the two cases (71-year-old female and 60-year-old female). OCT imaging of the affected eyes revealed cystoid spaces between the outer nuclear layer (ONL) and the outer plexiform layer (OPL), with the vascular network remaining intact, indicating no alterations in vessel flow. Microperimetry showed slightly lower pointwise sensitivity in both eyes compared to a normative database.</p><p><strong>Conclusion: </strong>Although SNIFR typically presents as a unilateral condition, our findings suggest that patients may have an inherent structural predisposition to macular schisis, resulting in subtle functional impairments. We hypothesize that the structural damage may involve the macular Müller cells. These observations highlight the importance of further studies to understand SNIFR's pathophysiology and inform therapeutic approaches.</p>","PeriodicalId":53580,"journal":{"name":"Retinal Cases and Brief Reports","volume":" ","pages":""},"PeriodicalIF":0.0,"publicationDate":"2025-01-14","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143016155","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}
{"title":"A 19-year-old asymptomatic woman.","authors":"William Carrera, Michelle C Liang","doi":"10.1097/ICB.0000000000001716","DOIUrl":"https://doi.org/10.1097/ICB.0000000000001716","url":null,"abstract":"<p><strong>Purpose: </strong>To describe a case of stellate multiform amelanotic choroidopathy (SMACH) with focal hyperfluorescence on indocyanine green angiography (ICGA).</p><p><strong>Methods: </strong>Case report.</p><p><strong>Results: </strong>A 19-year-old Caucasian woman was seen for an asymptomatic choroidal lesion. Visual acuity was 20/20. Fundus examination revealed an irregular, tan-colored lesion with overlying speckled hyperpigmentation in the macula of the left eye. Multimodal imaging demonstrated a thickened choroidal lesion with stellate, radially oriented projections most visible on near-infrared reflectance, red-free photography, en-face OCT, and ICGA. ICGA demonstrated early hypofluorescence outlining the borders of the lesion and a late, pinpoint area of hyperfluorescence with faint staining. The patient's fundus examination remained stable over 4 years of follow-up.</p><p><strong>Conclusion: </strong>SMACH may present with focal choroidal hyperpermeability on ICGA while lacking typical features of central serous chorioretinopathy (CSCR). This finding expands the clinical spectrum of SMACH.</p>","PeriodicalId":53580,"journal":{"name":"Retinal Cases and Brief Reports","volume":" ","pages":""},"PeriodicalIF":0.0,"publicationDate":"2025-01-14","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143016148","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}
Brenda Zhou, Curtis J Heisel, Ivy Zhu, Manjot K Gill
{"title":"VITREORETINAL ABNORMALITIES DURING MACULAR HOLE REPAIR IN ALPORT SYNDROME.","authors":"Brenda Zhou, Curtis J Heisel, Ivy Zhu, Manjot K Gill","doi":"10.1097/ICB.0000000000001498","DOIUrl":"10.1097/ICB.0000000000001498","url":null,"abstract":"<p><strong>Purpose: </strong>The purpose of the study was to describe abnormal vitreoretinal findings during macular hole repair in Alport syndrome.</p><p><strong>Methods: </strong>A case report of preoperative, intraoperative, and postoperative findings related to macular hole surgery in a patient with Alport syndrome was discussed.</p><p><strong>Results: </strong>A 50-year-old woman with Alport syndrome was found to have bilateral full-thickness macular holes. Surgery was recommended for her left eye given recent onset of vision loss and smaller hole size (313 μ m). Intraoperatively, the vitreous was found to have fine fibrillar strands and to be abnormally adherent to the retinal surface. There was little to no internal limiting membrane present. Vitrectomy was performed with posterior cortical hyaloid peeling and 15% C3F8 gas tamponade. Two months after surgery, the macular hole was successfully closed.</p><p><strong>Conclusion: </strong>Abnormal vitreous composition and adherence to the retinal surface may contribute to macular hole formation in patients with Alport syndrome. Standard surgical approaches including internal limiting membrane peeling may not be feasible, although thorough removal of the posterior cortical hyaloid may be sufficient to achieve macular hole closure.</p>","PeriodicalId":53580,"journal":{"name":"Retinal Cases and Brief Reports","volume":" ","pages":"80-83"},"PeriodicalIF":0.0,"publicationDate":"2025-01-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11649179/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"41219923","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
{"title":"RESOLUTION OF CHOROIDAL METASTASIS AND ASSOCIATED SUBRETINAL EXUDATION OF METASTATIC CUTANEOUS MELANOMA 15 DAYS AFTER TREATMENT WITH COMBINED TARGETED THERAPY.","authors":"Victor Wanten, Eva Vanhonsebrouck, Julie Jacob","doi":"10.1097/ICB.0000000000001495","DOIUrl":"10.1097/ICB.0000000000001495","url":null,"abstract":"<p><strong>Purpose: </strong>The purpose of this study was to report a case of metastatic cutaneous melanoma to the choroid with rapid and complete resolution of associated choroidal elevation and subretinal fluid after the initiation of combined targeted therapy.</p><p><strong>Methods: </strong>We describe a case of a 41-year-old man diagnosed with a metastatic cutaneous melanoma to the choroid of his right eye, for which treatment with dabrafenib/trametinib was initiated.</p><p><strong>Results: </strong>A 41-year-old man with a medical history of a BRAF V600E/V600E2/V600D-mutated invasive superficial spreading cutaneous melanoma presented with acute metamorphopsia and blurred vision in his right eye. Examination revealed a best-corrected visual acuity (BCVA) of 20/22 and two elevated choroidal lesions temporal to the fovea with subretinal exudation to the fovea on fundoscopy. On repeat examination 3 days later, his vision had further decreased to 20/50 with an increase of subretinal fluid. Treatment with BRAF/MEK inhibitor dabrafenib/trametinib was initiated, with complete resolution of the choroidal masses and subretinal exudation and improvement of the BCVA to 20/22 after only 15 days. An 8-week follow-up after the start of therapy showed stable fundoscopic and tomographic findings, with further improvement of BCVA to 20/17 and no ocular side effects.</p><p><strong>Conclusion: </strong>A case of metastatic cutaneous melanoma to the choroid with choroidal elevation and subretinal exudation to the fovea, for which treatment with dabrafenib/trametinib was initiated. Rapid and complete resolution of choroidal metastasis and the associated subretinal exudation after the initiation of combined targeted therapy was seen, without any ocular side effects.</p>","PeriodicalId":53580,"journal":{"name":"Retinal Cases and Brief Reports","volume":" ","pages":"50-53"},"PeriodicalIF":0.0,"publicationDate":"2025-01-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"41177469","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}