Case Reports in Ophthalmology最新文献

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An Unusual Retinal Presentation of a Novel COL11A1 Mutation: A Case Report.
IF 0.5
Case Reports in Ophthalmology Pub Date : 2025-01-10 eCollection Date: 2025-01-01 DOI: 10.1159/000542708
Tobias Peschaut, Monja Michelitsch, Martina Brandner, Sandra Kamper, Lisa Ofner-Ziegenfuss, Jasmin Blatterer, Heidelis Anna Tichy, Laura Posch-Pertl
{"title":"An Unusual Retinal Presentation of a Novel <i>COL11A1</i> Mutation: A Case Report.","authors":"Tobias Peschaut, Monja Michelitsch, Martina Brandner, Sandra Kamper, Lisa Ofner-Ziegenfuss, Jasmin Blatterer, Heidelis Anna Tichy, Laura Posch-Pertl","doi":"10.1159/000542708","DOIUrl":"10.1159/000542708","url":null,"abstract":"<p><strong>Introduction: </strong>Stickler syndrome is a rare collagenopathy, caused by mutations in various genes coding for fibrillar collagens II, IX, and XI. The disorder can be subdivided into different groups, depending on the genes affected and clinical features found in patients. Ocular symptoms, such as high myopia, retinal detachments, or anomalies in the vitreous, are present in most forms of Stickler syndrome. In this case report, we present a patient with an unusual retinal phenotype.</p><p><strong>Case presentation: </strong>Subject of this case report is a 33-year-old woman, who was examined at the Department of Ophthalmology at Medical University of Graz. A thorough ophthalmological examination was conducted, detailed medical and family history acquired, and genetic testing performed. Best corrected visual acuity was 20/20 on both eyes; however, impaired binocular vision associated with intermittent exotropia was found. Furthermore, dilated fundoscopy showed an unusual, hypopigmented spotted retinal phenotype. Fundus autofluorescence showed multiple hyperfluorescent spots corresponding with the spotted retinal appearance. Genetic testing revealed a novel variant in the gene <i>COL11A1</i>. No other ocular abnormalities which are associated with <i>COL11A1</i> were found.</p><p><strong>Conclusion: </strong>Several subtypes of Stickler syndrome have been reported in medical literature, greatly varying in clinical manifestations. Many different mutations in the gene <i>COL11A1</i> have been discovered and are typically associated with Stickler syndrome type 2. To our best knowledge, this is the first report of a patient with a mutation in the <i>COL11A1</i> gene presenting with a hypopigmented spotted retina.</p>","PeriodicalId":9635,"journal":{"name":"Case Reports in Ophthalmology","volume":"16 1","pages":"62-67"},"PeriodicalIF":0.5,"publicationDate":"2025-01-10","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11842027/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143467064","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}
引用次数: 0
Peripheral Retinal Ischemia and Secondary Neovascular Glaucoma after Cerclage for Retinal Detachment: A Case Report.
IF 0.5
Case Reports in Ophthalmology Pub Date : 2025-01-07 eCollection Date: 2025-01-01 DOI: 10.1159/000543239
Feliciana Menna, Marcel Menke, Markus Tschopp
{"title":"Peripheral Retinal Ischemia and Secondary Neovascular Glaucoma after Cerclage for Retinal Detachment: A Case Report.","authors":"Feliciana Menna, Marcel Menke, Markus Tschopp","doi":"10.1159/000543239","DOIUrl":"10.1159/000543239","url":null,"abstract":"<p><strong>Introduction: </strong>Various surgical techniques, including 360° buckling surgery with a retinal cerclage, have been employed to achieve retinal reattachment. Although retinal cerclage is generally effective, long-term complications can arise. Peripheral retinal ischemia and secondary neovascular glaucoma are rare, but serious complications can occur even years after successful retinal reattachment.</p><p><strong>Case presentation: </strong>We report a rare case of a 79-year-old woman who underwent 360° buckling surgery with cerclage for retinal detachment 10 years ago. Although the initial surgery successfully reattached the retina, she later developed a complication characterized by peripheral retinal ischemia and secondary neovascular glaucoma.</p><p><strong>Conclusion: </strong>Early detection and prompt management of such complications are crucial to prevent irreversible visual impairment.</p>","PeriodicalId":9635,"journal":{"name":"Case Reports in Ophthalmology","volume":"16 1","pages":"86-94"},"PeriodicalIF":0.5,"publicationDate":"2025-01-07","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11842065/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143466649","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}
引用次数: 0
Ultra-Widefield OCT Images of Proliferative Vitreoretinopathy under Silicone Oil.
IF 0.5
Case Reports in Ophthalmology Pub Date : 2025-01-07 eCollection Date: 2025-01-01 DOI: 10.1159/000542391
Masaki Kinoshita, Susumu Sakimoto, Masaki Suzue, Masanori Kanai, Nobuhiko Shiraki, Kohji Nishida
{"title":"Ultra-Widefield OCT Images of Proliferative Vitreoretinopathy under Silicone Oil.","authors":"Masaki Kinoshita, Susumu Sakimoto, Masaki Suzue, Masanori Kanai, Nobuhiko Shiraki, Kohji Nishida","doi":"10.1159/000542391","DOIUrl":"10.1159/000542391","url":null,"abstract":"<p><strong>Introduction: </strong>Proliferative vitreoretinopathy (PVR) is a condition in which proliferation forms after rhegmatogenous retinal detachment (RRD) surgery or old RRD, and sometimes, PVR can occur under silicone oil (SO). Here, we report 2 cases of PVR in which we evaluated preoperative B-scan images of proliferation under SO using ultra-widefield swept-source OCT (UWF-SS-OCT).</p><p><strong>Case presentation: </strong>The first case was a 61-year-old male who presented with PVR under SO tamponade after multiple pars plana vitrectomies (PPVs) at the previous hospital, and the second case was a 23-year-old man patient who was diagnosed with Coats disease in childhood and presented with PVR under SO after PPV for total RRD at our hospital. B-scan images of UWF-SS-OCT provided findings of epiretinal two-layered proliferation or emulsified SO in the peripheral area.</p><p><strong>Conclusion: </strong>UWF-SS-OCT is useful for preoperative assessment of the proliferation of PVR before SO removal. In addition, peculiar proliferation may occur in the eye under SO.</p>","PeriodicalId":9635,"journal":{"name":"Case Reports in Ophthalmology","volume":"16 1","pages":"57-61"},"PeriodicalIF":0.5,"publicationDate":"2025-01-07","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11842007/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143467099","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}
引用次数: 0
Novel Von Hippel-Lindau Germline Variants in Iranian Patients with Retinal Capillary Hemangioblastoma. 伊朗视网膜毛细血管母细胞瘤患者的新型 Von Hippel-Lindau 基因变异。
IF 0.5
Case Reports in Ophthalmology Pub Date : 2025-01-03 eCollection Date: 2025-01-01 DOI: 10.1159/000543119
Fatemeh Azimi, Kowsar Bagherzadeh, Golnaz Khakpoor, Saeed Talebi, Ahad Sedaghat, Reza Mirshahi, Hengameh Kasraei, Reza Afshar Kiaee, Masood Naseripour
{"title":"Novel Von Hippel-Lindau Germline Variants in Iranian Patients with Retinal Capillary Hemangioblastoma.","authors":"Fatemeh Azimi, Kowsar Bagherzadeh, Golnaz Khakpoor, Saeed Talebi, Ahad Sedaghat, Reza Mirshahi, Hengameh Kasraei, Reza Afshar Kiaee, Masood Naseripour","doi":"10.1159/000543119","DOIUrl":"10.1159/000543119","url":null,"abstract":"<p><strong>Introduction: </strong>The aim of this study was to determine the potential genetic cause of retinal capillary hemangioblastoma (RCH) with symptoms of Von Hippel-Lindau (VHL) disease.</p><p><strong>Case presentations: </strong>Three Iranian families (5 RCH patients) with novel variants are included in this study. The <i>VHL</i> variant analysis was performed by the Sanger sequencing technique. Molecular dynamics (MDs) simulations were conducted to analyze conformational changes resulting from variants in VHL protein structure and were compared with that of the native structure. Novel variant sites, including c.511A>C, c.511A>T, and c.514C>T in exon 3 of the <i>VHL</i> gene were identified. According to the American College of Medical Genetics (ACMG) classifications, c.514C>T (p.P172S) and c.511A>C (p.K171Q) are classified as variants of uncertain significance (VUS), and c.511A>T (p.K171*) is classified as a likely pathogenic variant. MD simulations demonstrated overall fluctuations of the proteins structure and a significantly lower degree of flexibility in the α-domain for the variant-encoded VHL protein structure compared to that of the native form.</p><p><strong>Conclusion: </strong>The structural information and computational analysis of the identified variants are predicted to induce conformational changes that limit the flexibility of protein VHL interaction interface with Elongin B/C, Elongin C/B, and Cullin-2, which are necessary for hypoxia-inducible factor 1-α binding. The genetic variants identified in Iranian patients with RCH may aid in the molecular confirmation of other patients diagnosed with VHL and their at-risk family members. These pioneering results that include detailed structural and functional analysis of a variant's effect on the VHL protein may serve as a model for future studies.</p>","PeriodicalId":9635,"journal":{"name":"Case Reports in Ophthalmology","volume":"16 1","pages":"74-85"},"PeriodicalIF":0.5,"publicationDate":"2025-01-03","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11842073/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143466447","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}
引用次数: 0
Vasoproliferative Retinal Tumors: Manifestations, Management, and Outcomes in a Case Series.
IF 0.5
Case Reports in Ophthalmology Pub Date : 2024-12-16 eCollection Date: 2025-01-01 DOI: 10.1159/000542956
Ana Faria Pereira, Rita Teixeira-Martins, Amandio Rocha-Sousa, Susana Penas
{"title":"Vasoproliferative Retinal Tumors: Manifestations, Management, and Outcomes in a Case Series.","authors":"Ana Faria Pereira, Rita Teixeira-Martins, Amandio Rocha-Sousa, Susana Penas","doi":"10.1159/000542956","DOIUrl":"10.1159/000542956","url":null,"abstract":"<p><strong>Introduction: </strong>Vasoproliferative retinal tumors (VPRTs) are rare, benign retinal lesions characterized by abnormal vascular proliferation. They may appear sporadically or with systemic conditions like von Hippel-Lindau disease, often resembling other retinal lesions and posing diagnostic challenges. VPRTs can lead to vision loss if not adequately managed. Current management ranges from observation to different treatment strategies, from laser photocoagulation, photodynamic therapy, cryotherapy to intravitreal injections and vitrectomy. However, optimal approach is still debated due to VPRTs' rarity and varied presentations.</p><p><strong>Case presentations: </strong>This series presents three VPRT cases. The first, a 73-year-old man who presented with sudden vision loss due to a VPRT-related vitreous hemorrhage, required multiple interventions, including vitrectomies and anti-VEGF injections, but suffered recurrent macular edema and glaucoma, requiring ongoing management. The second, a 42-year-old asymptomatic man, showed lipid exudation and peripheral retinal detachment, needing photodynamic therapy, laser, and cryotherapy. The third, a 56-year-old woman with decreased visual acuity and epiretinal membrane, achieved stability and good visual outcomes after vitrectomy with cryotherapy.</p><p><strong>Conclusions: </strong>VPRTs pose diagnostic and therapeutic challenges given their heterogeneous presentations and potential for complications. Our cases illustrate that a multimodal management strategy - incorporating tailored combinations of therapies such as laser photocoagulation, cryotherapy, anti-VEGF injections, and surgery - is essential to address individual characteristics of each patient's disease. Personalized treatment approaches allow for precise targeting of the lesions while minimizing risk of complications, contributing to better disease stabilization and visual outcomes. Early detection and tailored management are essential, though further research is needed to establish standardized treatment protocols.</p>","PeriodicalId":9635,"journal":{"name":"Case Reports in Ophthalmology","volume":"16 1","pages":"27-36"},"PeriodicalIF":0.5,"publicationDate":"2024-12-16","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11842064/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143467106","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}
引用次数: 0
Bilateral Serous Retinal Detachment as an Initial Manifestation of Systemic Lupus Erythematosus due to Hypoalbuminemia: A Case Report.
IF 0.5
Case Reports in Ophthalmology Pub Date : 2024-12-11 eCollection Date: 2025-01-01 DOI: 10.1159/000543005
Zikang Xu, Anyi Liang, Dan Cao, Liang Zhang
{"title":"Bilateral Serous Retinal Detachment as an Initial Manifestation of Systemic Lupus Erythematosus due to Hypoalbuminemia: A Case Report.","authors":"Zikang Xu, Anyi Liang, Dan Cao, Liang Zhang","doi":"10.1159/000543005","DOIUrl":"10.1159/000543005","url":null,"abstract":"<p><strong>Introduction: </strong>There are diverse etiologies of serous retinal detachment (SRD), and hypoalbuminemia is one of the rare causes of it. We report a case of bilateral SRD attributed to decreased serum albumin level caused by systemic lupus erythematosus (SLE).</p><p><strong>Case presentation: </strong>A 28-year-old female with no significant past medical history presented with bilateral palpebral edema for 10 days and blurred vision for 3 days. Optical coherence tomography demonstrated subretinal fluid in both eyes; fluorescein angiography revealed multiple hyperfluorescent spots with late leakage and pooling. Extensive serological and systemic examinations showed a slight decrease in serum albumin level with no other notable abnormalities. With the suspected diagnosis of Vogt-Koyanagi-Harada disease, the patient received high-dose intravenous corticosteroid therapy without any improvement. However, significant clinical improvement was achieved after albumin supplementation and corticosteroid dose tapering. The decrease in albumin levels in this patient was ultimately found to be due to protein-losing enteropathy (PLE) caused by SLE.</p><p><strong>Conclusions: </strong>This case presented a rare cause of bilateral SRD associated with hypoalbuminemia induced by SLE-related PLE. The report revealed that ocular involvements such as palpebral edema and SRD can be the initial manifestations of SLE rather than common lupus retinopathy. It emphasized the values of ocular manifestations as a clue for prompt diagnosis and management of systemic conditions.</p>","PeriodicalId":9635,"journal":{"name":"Case Reports in Ophthalmology","volume":"16 1","pages":"48-56"},"PeriodicalIF":0.5,"publicationDate":"2024-12-11","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11842062/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143467074","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}
引用次数: 0
Retinal Vasculitis in the Setting of Mycobacterium tuberculosis Exposure: Clinical Course and Management of Three Cases.
IF 0.5
Case Reports in Ophthalmology Pub Date : 2024-12-10 eCollection Date: 2025-01-01 DOI: 10.1159/000542075
Lauren C Kiryakoza, Jesse D Sengillo, Maria Paula Fernandez, Avinash Pathengay, Thomas A Albini, Harry W Flynn
{"title":"Retinal Vasculitis in the Setting of <i>Mycobacterium tuberculosis</i> Exposure: Clinical Course and Management of Three Cases.","authors":"Lauren C Kiryakoza, Jesse D Sengillo, Maria Paula Fernandez, Avinash Pathengay, Thomas A Albini, Harry W Flynn","doi":"10.1159/000542075","DOIUrl":"10.1159/000542075","url":null,"abstract":"<p><strong>Introduction: </strong>Retinal vasculitis in the setting of positive <i>Mycobacterium tuberculosis</i> (mTB) exposure represents an important diagnostic dilemma. Tubercular retinal vasculitis (TRV) is a recognized clinical entity, but it previously lacked diagnostic consensus. It shares clinical features with cases diagnosed as Eales disease. A precise distinction between the entities is lacking. Historically, Eales disease represented an idiopathic, obliterative retinal vasculitis seen in young males. Research has identified a potential relationship of Eales to mTB exposure, but the exact role of the organism is unclear, and the label \"Eales disease\" is reserved for truly idiopathic cases. We present the clinical course of 3 patients with retinal vasculitis and mTB exposure who were managed prior to group consensus guidelines for management of TRV. We discuss the evolving clinical meaning of Eales disease and review consensus TRV management guidelines.</p><p><strong>Case presentation: </strong>The clinical course of 3 patients with retinal vasculitis and mTB exposure were reviewed. Two patients were male, and 1 was female. Mean age at presentation was 56.8 years. Mean follow-up was 119.5 months. Mean presenting visual acuity was Snellen 20/25 in the right eye and 20/30 in the left eye. Mean visual acuity at final visit was 20/30 in both eyes. All 3 patients received antitubercular therapy (ATT). Two patients underwent retinal laser photocoagulation in one or both eyes. Two patients experienced multiple episodes of vitreous hemorrhage. One patient underwent pars plana vitrectomy for non-clearing vitreous hemorrhage in both eyes.</p><p><strong>Conclusion: </strong>The cases demonstrate the potential diagnostic and management variability created by the overlapping features of Eales disease and TRV. All 3 cases had some form of positive mTB testing (radiologic, immunologic, or both) and received different ATT regimens. Case 1 was labeled Eales disease, and cases 2 and 3 were labeled TRV. All 3 had favorable outcomes but only with control of the inflammation and neovascular complications. Recent consensus guidelines now provide guidance on ATT initiation in cases of TRV.</p>","PeriodicalId":9635,"journal":{"name":"Case Reports in Ophthalmology","volume":"16 1","pages":"18-26"},"PeriodicalIF":0.5,"publicationDate":"2024-12-10","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11842010/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143466882","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}
引用次数: 0
Atypical COVID-19-Related Acute Macular Neuroretinopathy with Progression Resulting in Severe Vision Loss.
IF 0.5
Case Reports in Ophthalmology Pub Date : 2024-12-10 eCollection Date: 2025-01-01 DOI: 10.1159/000542802
Eva Magdalene Weinberger, Loka Thangamathesvaran, J Fernando Arevalo
{"title":"Atypical COVID-19-Related Acute Macular Neuroretinopathy with Progression Resulting in Severe Vision Loss.","authors":"Eva Magdalene Weinberger, Loka Thangamathesvaran, J Fernando Arevalo","doi":"10.1159/000542802","DOIUrl":"10.1159/000542802","url":null,"abstract":"<p><strong>Introduction: </strong>We review the clinical course of a patient with decreased vision in the setting of COVID-19 infection consistent with an atypical presentation of acute macular neuroretinopathy (AMN).</p><p><strong>Case presentation: </strong>A 56-year-old Caucasian woman developed a scotoma in the right eye 3 days after COVID-19 diagnosis and in her left eye on day 5. Baseline exam showed significantly reduced visual acuity bilaterally best corrected visual acuity of 20/60 in the right eye and 20/40 in the left eye from a patient-reported baseline of 20/20 in each eye. Examination of the fundus was remarkable for small flame-shaped hemorrhages in the superior arcade of both eyes. Near-infrared reflectance imaging revealed a singular wedge-shaped lesion in each eye close to the fovea and spectral domain-optical coherence tomography confirmed disruption of the photoreceptor layer and ellipsoid zones. Our leading diagnosis given the presentation was COVID-19-associated AMN. Given no evidence of a clear treatment, observation was selected. Three weeks later, visual acuity deteriorated further to 20/100 OD and 20/80 OS, with persistence of the wedge-shaped lesions. At 3-month follow-up, fundus photographs remained unremarkable; however, visual acuity had dropped further to 20/300 bilaterally, with persistence of the scotomata and outer retinal layer disruptions. 6 months later, treatment with a dexamethasone implant improved vision to 20/125 OD and 20/150 OS.</p><p><strong>Conclusion: </strong>Among COVID-19-induced AMN, our case is remarkable for the severe progression of visual impairment over 3 months of follow-up and improvement with a dexamethasone implant. Furthermore, absence of classical AMN lesions on fundus photography raises the question whether COVID-induced AMN may lead to a clinically distinct, potentially more severe picture than AMN arising from previously identified causes.</p>","PeriodicalId":9635,"journal":{"name":"Case Reports in Ophthalmology","volume":"16 1","pages":"41-47"},"PeriodicalIF":0.5,"publicationDate":"2024-12-10","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11842036/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143467070","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}
引用次数: 0
Candy Cane Hypopyon Secondary to Intraocular Mantle Cell Lymphoma: A Case Report. 继发于眼内套细胞淋巴瘤1例。
IF 0.5
Case Reports in Ophthalmology Pub Date : 2024-12-06 eCollection Date: 2024-01-01 DOI: 10.1159/000541413
Fatima Babiker, Avery Zhou, Ploysai Rujkorakarn, Andrew M Philip, Tate Valerio, Yasmin Massoudi, Stephen D Anesi, C Stephen Foster
{"title":"Candy Cane Hypopyon Secondary to Intraocular Mantle Cell Lymphoma: A Case Report.","authors":"Fatima Babiker, Avery Zhou, Ploysai Rujkorakarn, Andrew M Philip, Tate Valerio, Yasmin Massoudi, Stephen D Anesi, C Stephen Foster","doi":"10.1159/000541413","DOIUrl":"10.1159/000541413","url":null,"abstract":"<p><strong>Introduction: </strong>Bilateral hemorrhagic hypopyon, also known as candy cane hypopyon, is an extremely rare presentation which we report as a unique case in association with intraocular mantle cell lymphoma (MCL).</p><p><strong>Case presentation: </strong>A 63-year-old white male presented with a 3-week history of conjunctival injection OS that was unresponsive to erythromycin ointment and topical steroids, in the setting of recently discovered diffuse lymphadenopathy and malaise. On presentation, he was found to have bilateral hemorrhagic hypopyon. Lymph node biopsy was diagnostic of MCL, and subsequent anterior chamber paracentesis confirmed intraocular MCL. The patient was put into remission with intravitreal rituximab injections, systemic chemotherapy, and external beam radiation.</p><p><strong>Conclusion: </strong>Cases of MCL with ocular involvement typically involve ocular adnexal structures, and seldom involve the uvea. Furthermore, this patient represents an extremely unusual case in his presentation with a hemorrhagic hypopyon.</p>","PeriodicalId":9635,"journal":{"name":"Case Reports in Ophthalmology","volume":"15 1","pages":"833-840"},"PeriodicalIF":0.5,"publicationDate":"2024-12-06","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11623969/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"142794511","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}
引用次数: 0
Acute Myelin Oligodendrocyte Glycoprotein-IgG Optic Neuritis without Optic Nerve Enhancement.
IF 0.5
Case Reports in Ophthalmology Pub Date : 2024-12-03 eCollection Date: 2025-01-01 DOI: 10.1159/000542889
Bava Jeyanathan, Jonathan Micieli
{"title":"Acute Myelin Oligodendrocyte Glycoprotein-IgG Optic Neuritis without Optic Nerve Enhancement.","authors":"Bava Jeyanathan, Jonathan Micieli","doi":"10.1159/000542889","DOIUrl":"10.1159/000542889","url":null,"abstract":"<p><strong>Introduction: </strong>Optic neuritis (ON) is a common neuro-ophthalmological condition and remains a significant indicator of inflammatory conditions affecting the central nervous system. Varying etiologies exist for ON including multiple sclerosis, NMOSD, and MOGAD. Differential diagnosis is achieved using both radiological and serological testing. MRI characteristics of MOG-ON include T2 hypersensitive lesions, nerve swelling, and gadolinium enhancement of the affected optic nerve. While acute MOG-ON usually presents with optic nerve enhancement, recognizing atypical presentations is critical in accurate diagnosis and effective management.</p><p><strong>Case presentation: </strong>We herein present a case of a 67-year-old woman presenting with sudden decrease in vision in the right eye. The patient underwent a 3T MRI of the orbits and brain 5 days post vision loss which returned normal right optic nerve appearance at presentation (no edema, enhancement or increased T2 signal). Further serological testing of the MOG antibody returned positive (1:100) while APQ4 antibodies were negative. This yielded a diagnosis of MOG-IgG-ON. Subsequently, the patient was treated with IV methylprednisolone 1 g daily for 5 days followed by prednisone 1 mg/kg, resulting in marked improvement in vision.</p><p><strong>Conclusion: </strong>This case highlights the complexity involved in diagnosing ON, especially in the context of MOGAD. Absence of optic nerve enhancement in this patient calls attention to the possibility of subclinical inflammation and/or detectable enhancement later in clinical course. Our findings, along with existing literature, highlight the need for clinicians to consider atypical MRI presentations in MOG-ON cases. Recognizing that normal MRI does not exclude MOG-ON is important for optimizing diagnostic accuracy and effective treatment interventions.</p>","PeriodicalId":9635,"journal":{"name":"Case Reports in Ophthalmology","volume":"16 1","pages":"7-11"},"PeriodicalIF":0.5,"publicationDate":"2024-12-03","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11703545/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143467060","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}
引用次数: 0
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