Pub Date : 2023-10-31DOI: 10.1080/01658107.2023.2271803
David A. Bellows, Noel C.Y. Chan, John J. Chen, Hui-Chen Cheng, Panitha Jindahra, Peter W. MacIntosh, Collin McClelland, Michael S. Vaphiades, Xiaojun Zhang
{"title":"Neuro-Ophthalmic Literature Review","authors":"David A. Bellows, Noel C.Y. Chan, John J. Chen, Hui-Chen Cheng, Panitha Jindahra, Peter W. MacIntosh, Collin McClelland, Michael S. Vaphiades, Xiaojun Zhang","doi":"10.1080/01658107.2023.2271803","DOIUrl":"https://doi.org/10.1080/01658107.2023.2271803","url":null,"abstract":"","PeriodicalId":19257,"journal":{"name":"Neuro-Ophthalmology","volume":"60 12","pages":"0"},"PeriodicalIF":0.0,"publicationDate":"2023-10-31","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"135810181","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 : 2023-10-31DOI: 10.1080/01658107.2023.2273473
Daniel Adamkiewicz, Maja Magazin, Dilip Thomas
ABSTRACTAnisocoria is a common finding in ophthalmic clinical practice. History taking and examination is critical in appropriately diagnosing and managing anisocoria, as the differential can be extensive ranging from benign to life-threatening entities. This case discusses the presentation of a 22-year-old female with a history of myopia and hyperhidrosis who presented with pharmacologic anisocoria which was presumed to be from inadvertent topical exposure to conventional glycopyrrolate tablets. To our knowledge, pharmacologic mydriasis from exposure to residue from conventional glycopyrrolate tablets has not been reported in the English literature. This case highlights the importance of medication and contact lens handling with anticholinergic agents.KEYWORDS: Anisocoriaoral glycopyrrolatemydriasisanticholinergicpharmacologic dilation Disclosure statementNo potential conflict of interest was reported by the authors.ConsentNo identifiable patient information is included. Our patient consented to the submission of this manuscript.Additional informationFundingThe authors reported there is no funding associated with the work featured in this article.
{"title":"A Case of Pharmacologic Anisocoria in Systemic Glycopyrrolate Use from Presumed Local Ocular Inoculation","authors":"Daniel Adamkiewicz, Maja Magazin, Dilip Thomas","doi":"10.1080/01658107.2023.2273473","DOIUrl":"https://doi.org/10.1080/01658107.2023.2273473","url":null,"abstract":"ABSTRACTAnisocoria is a common finding in ophthalmic clinical practice. History taking and examination is critical in appropriately diagnosing and managing anisocoria, as the differential can be extensive ranging from benign to life-threatening entities. This case discusses the presentation of a 22-year-old female with a history of myopia and hyperhidrosis who presented with pharmacologic anisocoria which was presumed to be from inadvertent topical exposure to conventional glycopyrrolate tablets. To our knowledge, pharmacologic mydriasis from exposure to residue from conventional glycopyrrolate tablets has not been reported in the English literature. This case highlights the importance of medication and contact lens handling with anticholinergic agents.KEYWORDS: Anisocoriaoral glycopyrrolatemydriasisanticholinergicpharmacologic dilation Disclosure statementNo potential conflict of interest was reported by the authors.ConsentNo identifiable patient information is included. Our patient consented to the submission of this manuscript.Additional informationFundingThe authors reported there is no funding associated with the work featured in this article.","PeriodicalId":19257,"journal":{"name":"Neuro-Ophthalmology","volume":"66 1","pages":"0"},"PeriodicalIF":0.0,"publicationDate":"2023-10-31","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"135809289","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":"Recurrent Third Nerve Paresis with Migraine: A Case Report and Review of the Literature","authors":"Sujit Kumar, Sharath Kumar Goddu Govindappa, Abdul Rawoof Bolar, Chaitra Parameshwara Adiga, Ravi Mohan Rao Basrur, Manithody Narayan Bhat Pramod, Santosh Kumar Pendyala, Jagadish Basavaraj Agadi, Rohit Shetty","doi":"10.1080/01658107.2023.2276191","DOIUrl":"https://doi.org/10.1080/01658107.2023.2276191","url":null,"abstract":"","PeriodicalId":19257,"journal":{"name":"Neuro-Ophthalmology","volume":"302 1","pages":"0"},"PeriodicalIF":0.0,"publicationDate":"2023-10-31","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"135871961","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 : 2023-10-31DOI: 10.1080/01658107.2023.2273477
Renato Correia Barbosa, Bruna Vieira, José Alberto Lemos
{"title":"Isolated Benign Unilateral Episodic Mydriasis in Relation to Anxiety: A Case Report","authors":"Renato Correia Barbosa, Bruna Vieira, José Alberto Lemos","doi":"10.1080/01658107.2023.2273477","DOIUrl":"https://doi.org/10.1080/01658107.2023.2273477","url":null,"abstract":"","PeriodicalId":19257,"journal":{"name":"Neuro-Ophthalmology","volume":"1 1","pages":"0"},"PeriodicalIF":0.0,"publicationDate":"2023-10-31","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"135870060","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}
ABSTRACTLeber’s hereditary optic neuropathy (LHON) is one of the hereditary optic neuropathies and is principally caused by three frequent mitochondria deoxyribonucleic acid (DNA) pathogenic variants (m.11778 G>A, m.3460 G>A, and m.14484T>C). These pathogenic variants account for 90% of LHON cases, with rare pathogenic variants accounting for the remaining cases. We report the first Japanese case of LHON with the m.13051 G>A pathogenic variant, which is a rare primary pathogenic variant of LHON. A 24-year-old woman developed subacute visual loss in both eyes over several months. The best corrected visual acuity (BCVA) was 6/120 in her right eye (OD) and 6/7.5 in her left eye (OS). A relative afferent pupillary defect was not detected. Humphrey visual field testing revealed a central scotoma OD and a temporal paracentral scotoma OS. Fundus examination showed the presence of a pale optic disc OD and optic disc swelling with peripapillary microangiopathy OS. Orbital magnetic resonance imaging showed no abnormal findings. As the mitochondrial DNA gene testing demonstrated the m.13051 G>A pathogenic variant, the patient was diagnosed with LHON. Subsequently, her BCVA worsened to 6/600 in each eye, followed by a nearly plateau-like progression thereafter. This mutation has been primarily reported in Europe but has not yet been confirmed in the Asian region. This case also indicates the importance of examining the whole mitochondrial DNA gene for pathogenic variants in cases where one of the three major pathogenic variants has not been not detected.KEYWORDS: Leber’s hereditary optic neuropathyoptic neuropathym.13051G>A pathogenic variantmitochondrial diseasegenetic testing AcknowledgmentsThe authors would like to thank the patient for her collaboration.Disclosure statementNo potential conflict of interest was reported by the authors.Data availability statementAll data generated or analysed during this study are included in this article. Further enquiries can be directed to the corresponding author.Statement of ethicsThe patient provided oral and written consent for publishing the data. The report does not include personal information that could identify the patient directly or indirectly. All medical interventions have been carried out according to the latest therapeutic protocols. All aspects of the present study are following the Declaration of Helsinki.Additional informationFundingThe authors reported there is no funding associated with the work featured in this article.
leber 's遗传性视神经病变(leber 's遗传性视神经病变，LHON)是一种遗传性视神经病变，主要由三种常见的线粒体脱氧核糖核酸(DNA)致病变异(m.11778)引起m.3460 G >G>A, m.14484T>C)。这些致病性变异占LHON病例的90%，其余病例为罕见致病性变异。我们报告了日本第一例使用m.13051的LHON病例G>一种致病性变异，是一种罕见的原发性致病性LHON变异。一名24岁女性在几个月内双眼出现亚急性视力丧失。最佳矫正视力(BCVA)右眼(OD)为6/120，左眼(OS)为6/7.5。未发现相对传入瞳孔缺损。汉弗莱视野测试显示一个中央暗斑OD和一个颞旁中央暗斑OS。眼底检查显示视盘色差，视盘肿胀伴乳突周围微血管病变。眼眶磁共振未见异常。线粒体DNA基因检测表明m.13051G>一种致病变异，诊断为LHON。随后，她每只眼睛的BCVA恶化至6/600，此后几乎呈平台状进展。这种突变主要在欧洲报告，但尚未在亚洲地区得到证实。该病例还表明，在未检测到三种主要致病变异之一的情况下，检查整个线粒体DNA基因以寻找致病变异的重要性。关键词:Leber遗传性视神经病变;视神经病变;13051G>A致病性变异线粒体疾病遗传学检测感谢患者的合作。披露声明作者未报告潜在的利益冲突。数据可用性声明本研究过程中产生或分析的所有数据均包含在本文中。进一步的查询可以直接联系通讯作者。伦理声明:患者口头和书面同意公布数据。该报告不包括可以直接或间接识别患者身份的个人信息。所有医疗干预都是根据最新的治疗方案进行的。本研究的所有方面都遵循《赫尔辛基宣言》。其他信息资金:作者报告没有与本文所述工作相关的资金。
{"title":"A Japanese Case of Leber’s Hereditary Optic Neuropathy with the m.13051G>A Pathogenic Variant","authors":"Yasuyuki Takai, Mayumi Iwasa, Akiko Yamagami, Kenji Inoue, Ryoma Yasumoto, Hitoshi Ishikawa, Masato Wakakura","doi":"10.1080/01658107.2023.2273480","DOIUrl":"https://doi.org/10.1080/01658107.2023.2273480","url":null,"abstract":"ABSTRACTLeber’s hereditary optic neuropathy (LHON) is one of the hereditary optic neuropathies and is principally caused by three frequent mitochondria deoxyribonucleic acid (DNA) pathogenic variants (m.11778 G>A, m.3460 G>A, and m.14484T>C). These pathogenic variants account for 90% of LHON cases, with rare pathogenic variants accounting for the remaining cases. We report the first Japanese case of LHON with the m.13051 G>A pathogenic variant, which is a rare primary pathogenic variant of LHON. A 24-year-old woman developed subacute visual loss in both eyes over several months. The best corrected visual acuity (BCVA) was 6/120 in her right eye (OD) and 6/7.5 in her left eye (OS). A relative afferent pupillary defect was not detected. Humphrey visual field testing revealed a central scotoma OD and a temporal paracentral scotoma OS. Fundus examination showed the presence of a pale optic disc OD and optic disc swelling with peripapillary microangiopathy OS. Orbital magnetic resonance imaging showed no abnormal findings. As the mitochondrial DNA gene testing demonstrated the m.13051 G>A pathogenic variant, the patient was diagnosed with LHON. Subsequently, her BCVA worsened to 6/600 in each eye, followed by a nearly plateau-like progression thereafter. This mutation has been primarily reported in Europe but has not yet been confirmed in the Asian region. This case also indicates the importance of examining the whole mitochondrial DNA gene for pathogenic variants in cases where one of the three major pathogenic variants has not been not detected.KEYWORDS: Leber’s hereditary optic neuropathyoptic neuropathym.13051G>A pathogenic variantmitochondrial diseasegenetic testing AcknowledgmentsThe authors would like to thank the patient for her collaboration.Disclosure statementNo potential conflict of interest was reported by the authors.Data availability statementAll data generated or analysed during this study are included in this article. Further enquiries can be directed to the corresponding author.Statement of ethicsThe patient provided oral and written consent for publishing the data. The report does not include personal information that could identify the patient directly or indirectly. All medical interventions have been carried out according to the latest therapeutic protocols. All aspects of the present study are following the Declaration of Helsinki.Additional informationFundingThe authors reported there is no funding associated with the work featured in this article.","PeriodicalId":19257,"journal":{"name":"Neuro-Ophthalmology","volume":"50 1","pages":"0"},"PeriodicalIF":0.0,"publicationDate":"2023-10-31","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"135809287","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 : 2023-10-16DOI: 10.1080/01658107.2023.2255651
Akshay Badakere, Amir Ali Mir, Rahul Negi, Sampada Kulkarni, Ramesh Kekunnaya, Virender Sachdeva
ABSTRACTOur objective was to compare the agreement between virtual reality perimetry (VRP) (order of magnitude, OM) and static automated perimetry (SAP) in various neuro-ophthalmological conditions. We carried out a retrospective analysis of visual field plots of patients with various neuro-ophthalmological conditions who underwent visual field testing using VRP and SAP and between 1 January and 31 May 2022. Two fellowship-trained neuro-ophthalmologists compared the visual field defects observed on both devices. Per cent agreement was used to compare the interpretation of the two examiners on both techniques. The study criteria were met by 160 eyes from 148 patients (mean age 44 years, range 17–74 years). The most common aetiologies were optic atrophy due to various causes, optic neuritis, ischaemic optic neuropathy, and compressive optic neuropathy. Overall, we found good agreement between VRP and SAP for bitemporal (93.8%), hemianopic (90.8%), altitudinal (79.4%), and generalised visual field defects (86.4%). The agreement was acceptable for central/centrocaecal scotomas and not acceptable for enlarged blind spots. Between the two examiners there was good agreement for bitemporal (92.3%), hemianopic (82%), altitudinal (83%), and generalised field defects (76.4%). The results of our study suggest that VRP gives overall good agreement with SAP in various neuro-ophthalmological conditions, especially those likely to produce hemianopic, altitudinal, and generalised visual field defects. This could be useful in various settings; however, future larger studies are needed to explore real-world utilisation.KEYWORDS: Agreementvirtual realityperimetrystandard automated perimetryneuro-ophthalmology AcknowledgmentsWe thank our optometry team members (namely Masuma, Fatima, Aleena Saji, Gowthami Neredimilli, Mounika Vadithya, and Saurav Ghosh, who helped us with enrolment of the patients, and collected the data), and our team at Centre for Technology Innovation, who helped with various technical aspects.Disclosure statementNo potential conflict of interest was reported by the authors.Additional informationFundingThis study is supported by the intramural funding provided by Hyderabad Eye Research Foundation.
{"title":"Agreement Between Virtual Reality Perimetry and Static Automated Perimetry in Various Neuro-Ophthalmological Conditions: A Pilot Study","authors":"Akshay Badakere, Amir Ali Mir, Rahul Negi, Sampada Kulkarni, Ramesh Kekunnaya, Virender Sachdeva","doi":"10.1080/01658107.2023.2255651","DOIUrl":"https://doi.org/10.1080/01658107.2023.2255651","url":null,"abstract":"ABSTRACTOur objective was to compare the agreement between virtual reality perimetry (VRP) (order of magnitude, OM) and static automated perimetry (SAP) in various neuro-ophthalmological conditions. We carried out a retrospective analysis of visual field plots of patients with various neuro-ophthalmological conditions who underwent visual field testing using VRP and SAP and between 1 January and 31 May 2022. Two fellowship-trained neuro-ophthalmologists compared the visual field defects observed on both devices. Per cent agreement was used to compare the interpretation of the two examiners on both techniques. The study criteria were met by 160 eyes from 148 patients (mean age 44 years, range 17–74 years). The most common aetiologies were optic atrophy due to various causes, optic neuritis, ischaemic optic neuropathy, and compressive optic neuropathy. Overall, we found good agreement between VRP and SAP for bitemporal (93.8%), hemianopic (90.8%), altitudinal (79.4%), and generalised visual field defects (86.4%). The agreement was acceptable for central/centrocaecal scotomas and not acceptable for enlarged blind spots. Between the two examiners there was good agreement for bitemporal (92.3%), hemianopic (82%), altitudinal (83%), and generalised field defects (76.4%). The results of our study suggest that VRP gives overall good agreement with SAP in various neuro-ophthalmological conditions, especially those likely to produce hemianopic, altitudinal, and generalised visual field defects. This could be useful in various settings; however, future larger studies are needed to explore real-world utilisation.KEYWORDS: Agreementvirtual realityperimetrystandard automated perimetryneuro-ophthalmology AcknowledgmentsWe thank our optometry team members (namely Masuma, Fatima, Aleena Saji, Gowthami Neredimilli, Mounika Vadithya, and Saurav Ghosh, who helped us with enrolment of the patients, and collected the data), and our team at Centre for Technology Innovation, who helped with various technical aspects.Disclosure statementNo potential conflict of interest was reported by the authors.Additional informationFundingThis study is supported by the intramural funding provided by Hyderabad Eye Research Foundation.","PeriodicalId":19257,"journal":{"name":"Neuro-Ophthalmology","volume":"76 1","pages":"0"},"PeriodicalIF":0.0,"publicationDate":"2023-10-16","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"136116355","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 : 2023-09-15DOI: 10.1080/01658107.2023.2257311
Jorge Cárdenas-Belaunzarán, Karen A. Cerrillo-Avila
ABSTRACTVision specialists will benefit from increased awareness of posterior cortical atrophy (PCA) syndrome. Failure to adequately identify the chief complaint as a visual symptom may lead to incorrect diagnosis or diagnostic delay. A previously healthy, 59-year-old woman presented with a 5-year history of ‘losing her stuff’. Upon psychiatric and neuro-ophthalmological evaluation, this symptom was better recognised as a feature of visual agnosia and simultanagnosia. She also presented with multiple previously unrecognised symptoms indicative of higher visual processing dysfunction, such as alexia without agraphia, ocular motor apraxia, optic ataxia, prosopagnosia, akinetopsia and topographagnosia, so further assessment to investigate for PCA was carried out. After a work-up including cognitive assessment, brain structural/functional imaging, and laboratory tests she was diagnosed with visual-variant Alzheimer’s disease. Patients with PCA merit a detailed review of their symptoms, as well as the use of office tests such as cognitive evaluation tools, different types of perimetry, colour vision tests, and non-delayed psychiatric consultation for correct management and assessment. This report will emphasise five key aspects to be considered when evaluating patients with PCAKEYWORDS: Posterior cortical atrophyAlzheimer’s diseaseAlzheimer’s visual variantvisual agnosiamemory deficitAtypical alzheimer’scase report Authors’ contributionsJCB and KCA drafted the manuscript and collected patient information, JCB critically revised the manuscript for intellectual content and supervised the project. All authors have read and approved the final manuscript.Disclosure statementNo potential conflict of interest was reported by the authors.Consent for publicationWritten informed consent was obtained from the patient.Ethics approval and consent to participateThe study was approved by the internal review board of Asociación para Evitar la Ceguera en México, I. A. P.Additional informationFundingThe authors reported there is no funding associated with the work featured in this article.
视觉专家将受益于对后皮质萎缩(PCA)综合征意识的提高。未能充分识别主诉作为视觉症状可能导致诊断错误或诊断延误。一名健康的59岁女性出现了5年的“失去她的东西”的历史。经精神病学和神经眼科评估，这种症状被更好地认为是视觉失认症和同时失认症的特征。她还表现出多种先前未被认识到的症状，表明有更高的视觉加工功能障碍，如无失写症的失读症、眼运动失用症、视觉共济失调、面孔失认症、动位失认和地形失认，因此进行了进一步的评估以研究PCA。在进行了包括认知评估、大脑结构/功能成像和实验室检查在内的检查后，她被诊断出患有视觉变异性阿尔茨海默病。PCA患者需要对他们的症状进行详细的检查，并使用办公室测试，如认知评估工具、不同类型的视野检查、色觉测试和非延迟的精神病学咨询，以正确的管理和评估。本报告将强调评估pcak患者时应考虑的五个关键方面关键词:后皮质萎缩阿尔茨海默病阿尔茨海默病视觉变化视觉失智记忆缺陷非典型阿尔茨海默病病例报告作者的贡献sjcb和KCA起草了手稿并收集了患者信息，JCB对手稿的智力内容进行了批判性修改并监督了该项目。所有作者都阅读并批准了最终稿件。披露声明作者未报告潜在的利益冲突。发表同意获得患者的书面知情同意。伦理批准和同意参与该研究是由内部审查委员会Asociación para Evitar la Ceguera en macimxico, i.a.p.p批准的。附加信息:资金来源作者报告没有与本文所述工作相关的资金来源。
{"title":"Visual Agnosia Mimicking Memory Impairment: A Case Report of Posterior Cortical Atrophy","authors":"Jorge Cárdenas-Belaunzarán, Karen A. Cerrillo-Avila","doi":"10.1080/01658107.2023.2257311","DOIUrl":"https://doi.org/10.1080/01658107.2023.2257311","url":null,"abstract":"ABSTRACTVision specialists will benefit from increased awareness of posterior cortical atrophy (PCA) syndrome. Failure to adequately identify the chief complaint as a visual symptom may lead to incorrect diagnosis or diagnostic delay. A previously healthy, 59-year-old woman presented with a 5-year history of ‘losing her stuff’. Upon psychiatric and neuro-ophthalmological evaluation, this symptom was better recognised as a feature of visual agnosia and simultanagnosia. She also presented with multiple previously unrecognised symptoms indicative of higher visual processing dysfunction, such as alexia without agraphia, ocular motor apraxia, optic ataxia, prosopagnosia, akinetopsia and topographagnosia, so further assessment to investigate for PCA was carried out. After a work-up including cognitive assessment, brain structural/functional imaging, and laboratory tests she was diagnosed with visual-variant Alzheimer’s disease. Patients with PCA merit a detailed review of their symptoms, as well as the use of office tests such as cognitive evaluation tools, different types of perimetry, colour vision tests, and non-delayed psychiatric consultation for correct management and assessment. This report will emphasise five key aspects to be considered when evaluating patients with PCAKEYWORDS: Posterior cortical atrophyAlzheimer’s diseaseAlzheimer’s visual variantvisual agnosiamemory deficitAtypical alzheimer’scase report Authors’ contributionsJCB and KCA drafted the manuscript and collected patient information, JCB critically revised the manuscript for intellectual content and supervised the project. All authors have read and approved the final manuscript.Disclosure statementNo potential conflict of interest was reported by the authors.Consent for publicationWritten informed consent was obtained from the patient.Ethics approval and consent to participateThe study was approved by the internal review board of Asociación para Evitar la Ceguera en México, I. A. P.Additional informationFundingThe authors reported there is no funding associated with the work featured in this article.","PeriodicalId":19257,"journal":{"name":"Neuro-Ophthalmology","volume":"205 1","pages":"0"},"PeriodicalIF":0.0,"publicationDate":"2023-09-15","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"135395567","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 : 2023-09-15DOI: 10.1080/01658107.2023.2255664
Minjun Hur, Nanthaya Tisavipat, Collin M. McClelland, John Chen
{"title":"Proceedings of the 45th Annual Upper Midwest Neuro-Ophthalmology Group Meeting, 21 July 2023","authors":"Minjun Hur, Nanthaya Tisavipat, Collin M. McClelland, John Chen","doi":"10.1080/01658107.2023.2255664","DOIUrl":"https://doi.org/10.1080/01658107.2023.2255664","url":null,"abstract":"","PeriodicalId":19257,"journal":{"name":"Neuro-Ophthalmology","volume":"75 1","pages":"0"},"PeriodicalIF":0.0,"publicationDate":"2023-09-15","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"135397266","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 : 2023-09-14DOI: 10.1080/01658107.2023.2251575
Katharina Valentin, Thomas Georgi, Regina Riedl, Haleh Aminfar, Christoph Singer, Thomas Klopstock, Andreas Wedrich, Mona Schneider
ABSTRACTThe aim of this study was to evaluate the therapeutic effect of idebenone in patients with OPA1-dominant optic atrophy (DOA). Sixteen patients with genetically confirmed OPA1-DOA were treated with 900 mg idebenone daily for 12 months. The primary endpoint was the best recovery/least deterioration of visual acuity. Secondary endpoints were the changes of visual acuity, colour vision, contrast sensitivity, visual field, peripapillary retinal nerve fibre layer thickness (pRNFLT), and visual-related quality of life. For the primary endpoint, a significant increase was observed for the right eye (p = .0027), for the left eye (p = .0111) and for the better-seeing eye (p = .0152). For visual fields, a significant improvement was observed for the left eye between baseline and 9 months (p = .0038). Regarding pRNFLT, a significant decrease was found for the left eye between baseline and 3 months (p = .0413) and between baseline and 6 months (p = .0448). In the visual function questionnaire, a significant improvement was observed in the subscale general vision (p = .0156) and in the composite score (p = .0256). In conclusion, best recovery of visual acuity improved, even though the amount of improvement was small. Furthermore, a maintenance of visual function after 12 months of idebenone intake could be observed as well as a significant improvement in vision-related quality of life.Whether this effect is due to idebenone treatment, the placebo effect, or is explainable by the natural progression of DOA, remains unclear.Trial registration: EU Clinical Trials Register, EudraCT Number: 2019-001493-28KEYWORDS: Dominant optic atrophyOPA1idebenoneoptic neuropathyvisual function AcknowledgementsThe authors thank all patients for their participation in this study.Disclosure statementK. Valentin received travel reimbursements from Chiesi Pharmaceuticals GmbH, H. Aminfar received travel reimbursements from Santhera Pharmaceuticals and Chiesi Pharmaceuticals GmbH. T. Klopstock received travel reimbursements and speaker honoraria from Santhera Pharmaceuticals and Chiesi Pharmaceuticals GmbH and M. Schneider received speaker honoraria from Santhera Pharmaceuticals. T. Georgi, R. Riedl, C. Singer, and A. Wedrich report no competing interests.Additional informationFundingThis work was supported by Chiesi Pharmaceuticals GmbH by a project-related grant for monitoring, pregnancy tests, registration fees, and patients’ insurance, as well as provision of study medication free of charge.
{"title":"Idebenone Treatment in Patients with OPA1-Dominant Optic Atrophy: A Prospective Phase 2 Trial","authors":"Katharina Valentin, Thomas Georgi, Regina Riedl, Haleh Aminfar, Christoph Singer, Thomas Klopstock, Andreas Wedrich, Mona Schneider","doi":"10.1080/01658107.2023.2251575","DOIUrl":"https://doi.org/10.1080/01658107.2023.2251575","url":null,"abstract":"ABSTRACTThe aim of this study was to evaluate the therapeutic effect of idebenone in patients with OPA1-dominant optic atrophy (DOA). Sixteen patients with genetically confirmed OPA1-DOA were treated with 900 mg idebenone daily for 12 months. The primary endpoint was the best recovery/least deterioration of visual acuity. Secondary endpoints were the changes of visual acuity, colour vision, contrast sensitivity, visual field, peripapillary retinal nerve fibre layer thickness (pRNFLT), and visual-related quality of life. For the primary endpoint, a significant increase was observed for the right eye (p = .0027), for the left eye (p = .0111) and for the better-seeing eye (p = .0152). For visual fields, a significant improvement was observed for the left eye between baseline and 9 months (p = .0038). Regarding pRNFLT, a significant decrease was found for the left eye between baseline and 3 months (p = .0413) and between baseline and 6 months (p = .0448). In the visual function questionnaire, a significant improvement was observed in the subscale general vision (p = .0156) and in the composite score (p = .0256). In conclusion, best recovery of visual acuity improved, even though the amount of improvement was small. Furthermore, a maintenance of visual function after 12 months of idebenone intake could be observed as well as a significant improvement in vision-related quality of life.Whether this effect is due to idebenone treatment, the placebo effect, or is explainable by the natural progression of DOA, remains unclear.Trial registration: EU Clinical Trials Register, EudraCT Number: 2019-001493-28KEYWORDS: Dominant optic atrophyOPA1idebenoneoptic neuropathyvisual function AcknowledgementsThe authors thank all patients for their participation in this study.Disclosure statementK. Valentin received travel reimbursements from Chiesi Pharmaceuticals GmbH, H. Aminfar received travel reimbursements from Santhera Pharmaceuticals and Chiesi Pharmaceuticals GmbH. T. Klopstock received travel reimbursements and speaker honoraria from Santhera Pharmaceuticals and Chiesi Pharmaceuticals GmbH and M. Schneider received speaker honoraria from Santhera Pharmaceuticals. T. Georgi, R. Riedl, C. Singer, and A. Wedrich report no competing interests.Additional informationFundingThis work was supported by Chiesi Pharmaceuticals GmbH by a project-related grant for monitoring, pregnancy tests, registration fees, and patients’ insurance, as well as provision of study medication free of charge.","PeriodicalId":19257,"journal":{"name":"Neuro-Ophthalmology","volume":"14 1","pages":"0"},"PeriodicalIF":0.0,"publicationDate":"2023-09-14","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"134910768","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 : 2023-09-14DOI: 10.1080/01658107.2023.2251580
Richard N. Sather, Michael S. Lee
ABSTRACTMercury has been described as been in daily household items such as soaps, skin-lightening creams (SLC), and topical disinfectants. Mercury exposure can reportedly cause damage to the optic nerve and retina. A 30-year-old Somali woman presented with decreased vision and was found to have bilateral optic atrophy. Neuroimaging and laboratory work-up for nutritional deficiencies, heavy metals, and syphilis were performed. Evaluation revealed normal neuroimaging and laboratory work-up except for elevated serum and urine mercury levels. Mercury levels at the initial blood test was 11.1 ug/L (normal limits < 10.0 ug/L) and was 15.7 ug/L on repeat testing. A 24-h urine test showed elevated mercury at 16 ug/24 h (normal limits < 2 ug/24 h). Evaluation of an unlabelled SLC that she was using showed the presence of mercury. It is worth testing for heavy metals in the work-up of bilateral optic atrophy. Clinicians should consider cosmetic products as a potential source of mercury exposure and recommend discontinuation if mercury is present.KEYWORDS: Toxic optic neuropathymercury toxicityoptical coherence tomographyretinal nerve fibre layercosmetic products Disclosure statementNo potential conflict of interest was reported by the authors.Supplementary materialSupplemental data for this article can be accessed online at https://doi.org/10.1080/01658107.2023.2251580.Additional informationFundingThe authors reported there is no funding associated with the work featured in this article.
{"title":"Toxic Optic Neuropathy Due to Mercury in Skin Lightening Products","authors":"Richard N. Sather, Michael S. Lee","doi":"10.1080/01658107.2023.2251580","DOIUrl":"https://doi.org/10.1080/01658107.2023.2251580","url":null,"abstract":"ABSTRACTMercury has been described as been in daily household items such as soaps, skin-lightening creams (SLC), and topical disinfectants. Mercury exposure can reportedly cause damage to the optic nerve and retina. A 30-year-old Somali woman presented with decreased vision and was found to have bilateral optic atrophy. Neuroimaging and laboratory work-up for nutritional deficiencies, heavy metals, and syphilis were performed. Evaluation revealed normal neuroimaging and laboratory work-up except for elevated serum and urine mercury levels. Mercury levels at the initial blood test was 11.1 ug/L (normal limits < 10.0 ug/L) and was 15.7 ug/L on repeat testing. A 24-h urine test showed elevated mercury at 16 ug/24 h (normal limits < 2 ug/24 h). Evaluation of an unlabelled SLC that she was using showed the presence of mercury. It is worth testing for heavy metals in the work-up of bilateral optic atrophy. Clinicians should consider cosmetic products as a potential source of mercury exposure and recommend discontinuation if mercury is present.KEYWORDS: Toxic optic neuropathymercury toxicityoptical coherence tomographyretinal nerve fibre layercosmetic products Disclosure statementNo potential conflict of interest was reported by the authors.Supplementary materialSupplemental data for this article can be accessed online at https://doi.org/10.1080/01658107.2023.2251580.Additional informationFundingThe authors reported there is no funding associated with the work featured in this article.","PeriodicalId":19257,"journal":{"name":"Neuro-Ophthalmology","volume":"111 1","pages":"0"},"PeriodicalIF":0.0,"publicationDate":"2023-09-14","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"134911341","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}
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