Pub Date : 2024-08-23eCollection Date: 2024-01-01DOI: 10.1159/000540346
Nathan Nash Wise, Eric K Chin, David R P Almeida
Introduction: The aim of the study was to document and analyze a rare case of spontaneous retinal reattachment in a patient after successive unsuccessful vitreoretinal surgeries and to explore potential mechanisms contributing to this unexpected outcome.
Case presentation: A 61-year-old patient with a history of high myopia presented with a rhegmatogenous retinal detachment. After undergoing multiple vitreoretinal procedures, including pars plana vitrectomy with silicone oil tamponade, anatomical reattachment was not achieved, and the patient developed complex retinal detachment associated with myopic foveoschisis. Surprisingly, spontaneous reattachment of the retina was observed during routine follow-up. Comprehensive ocular examination, optical coherence tomography (OCT), and fundus autofluorescence imaging were utilized to confirm and document the retinal status. A thorough review of patient history, surgical reports, and postoperative imaging was performed to discern potential contributory factors. The patient's retina remained attached for 12 months after the last intervention without additional surgical or medical treatment. OCT images revealed normalization of retinal architecture with reestablishment of the foveal contour and partial recovery of visual acuity. No signs of proliferative vitreoretinopathy or other commonly associated complications were observed. Immunological assays and genetic testing ruled out systemic conditions that could predispose to spontaneous reattachment.
Conclusion: This case represents a remarkable instance of spontaneous retinal reattachment without surgical intervention after multiple failed procedures. The mechanisms underlying this phenomenon remain speculative; however, they may involve delayed postoperative cellular proliferation and migration, subtle intraocular pressure changes, or unrecognized vitreous traction resolution. Further investigation into similar cases may provide insights into the natural history of retinal detachment and potential self-resolving dynamics, which could inform future therapeutic strategies.
{"title":"Spontaneous Retinal Reattachment following Multiple Vitreoretinal Interventions: Case Report.","authors":"Nathan Nash Wise, Eric K Chin, David R P Almeida","doi":"10.1159/000540346","DOIUrl":"https://doi.org/10.1159/000540346","url":null,"abstract":"<p><strong>Introduction: </strong>The aim of the study was to document and analyze a rare case of spontaneous retinal reattachment in a patient after successive unsuccessful vitreoretinal surgeries and to explore potential mechanisms contributing to this unexpected outcome.</p><p><strong>Case presentation: </strong>A 61-year-old patient with a history of high myopia presented with a rhegmatogenous retinal detachment. After undergoing multiple vitreoretinal procedures, including pars plana vitrectomy with silicone oil tamponade, anatomical reattachment was not achieved, and the patient developed complex retinal detachment associated with myopic foveoschisis. Surprisingly, spontaneous reattachment of the retina was observed during routine follow-up. Comprehensive ocular examination, optical coherence tomography (OCT), and fundus autofluorescence imaging were utilized to confirm and document the retinal status. A thorough review of patient history, surgical reports, and postoperative imaging was performed to discern potential contributory factors. The patient's retina remained attached for 12 months after the last intervention without additional surgical or medical treatment. OCT images revealed normalization of retinal architecture with reestablishment of the foveal contour and partial recovery of visual acuity. No signs of proliferative vitreoretinopathy or other commonly associated complications were observed. Immunological assays and genetic testing ruled out systemic conditions that could predispose to spontaneous reattachment.</p><p><strong>Conclusion: </strong>This case represents a remarkable instance of spontaneous retinal reattachment without surgical intervention after multiple failed procedures. The mechanisms underlying this phenomenon remain speculative; however, they may involve delayed postoperative cellular proliferation and migration, subtle intraocular pressure changes, or unrecognized vitreous traction resolution. Further investigation into similar cases may provide insights into the natural history of retinal detachment and potential self-resolving dynamics, which could inform future therapeutic strategies.</p>","PeriodicalId":9635,"journal":{"name":"Case Reports in Ophthalmology","volume":"15 1","pages":"663-668"},"PeriodicalIF":0.5,"publicationDate":"2024-08-23","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11521428/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"142544007","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}
Pub Date : 2024-08-22eCollection Date: 2024-01-01DOI: 10.1159/000540603
Seonghwan Kim, Jeong Hyun Lee
Introduction: Cataract surgery is one of the most commonly performed surgical procedures worldwide. Intraoperative and postoperative complications of cataract surgery include posterior capsule rupture, retinal detachment, suprachoroidal hemorrhage, cystoid macular edema, endophthalmitis, and intraocular lens dislocation. As corneal perforation is rarely related to cataract surgery, we report a case of intraoperative iatrogenic corneal puncture that led to a full-thickness laceration that was managed without severe complications.
Case presentation: An 81-year-old woman underwent immediate sequential bilateral cataract surgery. Cataract grading of both eyes was nuclear color grade II and cortical cataract grade III, according to the lens opacity classification system III. While performing cataract surgery on her right eye, a corneal puncture with a keratome incidentally occurred just before making the main temporal incision, resulting in a 3-mm full-thickness laceration at the peripheral cornea. One-bite of 10-0 nylon suture was placed, and the remaining procedure was performed through a superior main incision. Three-bites of 10-0 nylon suture were placed at the laceration site at the end of the procedure. The uncorrected visual acuity was 20/40 in both eyes the following day. Corneal astigmatism increased in the right eye, and the refractive error was more myopic than targeted. No leakage was observed.
Conclusion: Slight corneal contact with a keratome can lead to full-thickness laceration when the anterior chamber is filled with an ophthalmic viscosurgical device during cataract surgery. Caution should be exercised while manipulating sharp surgical instruments to avoid such complications.
简介白内障手术是全球最常见的外科手术之一。白内障手术的术中和术后并发症包括后囊破裂、视网膜脱离、脉络膜上出血、囊样黄斑水肿、眼底病和眼内晶状体脱位。由于角膜穿孔很少与白内障手术有关,我们报告了一例术中先天性角膜穿孔导致全层裂孔的病例,经过处理后未出现严重并发症:一名 81 岁的妇女立即接受了双侧顺序白内障手术。根据晶状体混浊分级系统 III,双眼白内障分级为核色 II 级和皮质白内障 III 级。在对她的右眼进行白内障手术时,就在做颞部主刀切口之前,意外地用角膜刀刺破了角膜,造成周边角膜 3 毫米全厚裂伤。缝合了一口 10-0 尼龙线,剩余的手术通过上部主切口进行。手术结束时,在裂口部位缝合了三针 10-0 尼龙线。第二天,双眼未矫正视力均为 20/40。右眼的角膜散光增加,屈光不正比目标值更近视。未发现渗漏:结论:在白内障手术中使用眼科粘弹剂手术器械填充前房时,角膜与角膜刀的轻微接触可导致全层撕裂。在操作锋利的手术器械时应小心谨慎,以避免此类并发症的发生。
{"title":"Iatrogenic Corneal Puncture at the Beginning of Cataract Surgery Which Was Managed Uneventfully.","authors":"Seonghwan Kim, Jeong Hyun Lee","doi":"10.1159/000540603","DOIUrl":"https://doi.org/10.1159/000540603","url":null,"abstract":"<p><strong>Introduction: </strong>Cataract surgery is one of the most commonly performed surgical procedures worldwide. Intraoperative and postoperative complications of cataract surgery include posterior capsule rupture, retinal detachment, suprachoroidal hemorrhage, cystoid macular edema, endophthalmitis, and intraocular lens dislocation. As corneal perforation is rarely related to cataract surgery, we report a case of intraoperative iatrogenic corneal puncture that led to a full-thickness laceration that was managed without severe complications.</p><p><strong>Case presentation: </strong>An 81-year-old woman underwent immediate sequential bilateral cataract surgery. Cataract grading of both eyes was nuclear color grade II and cortical cataract grade III, according to the lens opacity classification system III. While performing cataract surgery on her right eye, a corneal puncture with a keratome incidentally occurred just before making the main temporal incision, resulting in a 3-mm full-thickness laceration at the peripheral cornea. One-bite of 10-0 nylon suture was placed, and the remaining procedure was performed through a superior main incision. Three-bites of 10-0 nylon suture were placed at the laceration site at the end of the procedure. The uncorrected visual acuity was 20/40 in both eyes the following day. Corneal astigmatism increased in the right eye, and the refractive error was more myopic than targeted. No leakage was observed.</p><p><strong>Conclusion: </strong>Slight corneal contact with a keratome can lead to full-thickness laceration when the anterior chamber is filled with an ophthalmic viscosurgical device during cataract surgery. Caution should be exercised while manipulating sharp surgical instruments to avoid such complications.</p>","PeriodicalId":9635,"journal":{"name":"Case Reports in Ophthalmology","volume":"15 1","pages":"656-662"},"PeriodicalIF":0.5,"publicationDate":"2024-08-22","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11521449/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"142544002","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}
Pub Date : 2024-08-13eCollection Date: 2024-01-01DOI: 10.1159/000540015
Augusto Magalhães, Margarida Ribeiro, Jorge Meira, Ana Filipa Moleiro, Esmeralda Rodrigues, Elisa Leão-Teles
Introduction: Mucopolysaccharidosis type VI (MPS VI) is a rare inherited metabolic disorder, primarily attributed to the deficiency of the enzyme N-acetylgalactosamine-4-sulfatase, responsible for the degradation of dermatan sulfate and chondroitin-4-sulfate. Therefore, there is a widespread accumulation of partially degraded glycosaminoglycans. Corneal opacification is the hallmark ocular feature in the MPS. Retinal and scleral involvement in this MPS is extremely rare. The purpose of this work was to describe novel fundoscopic alterations present in patients with MPS VI.
Case presentation: This is a case series involving three non-related patients referred to our department from the Unit of Inherited Metabolic Diseases. Multimodal imaging was performed in every patient. Fundus photography and enhanced depth imaging optical coherence tomography (EDI-OCT) were performed. Multiple areas of yellow/orange patches were observed on fundus photography, corresponding to areas in which deposits of intermediate reflectivity in the EDI-OCT could be seen at the scleral level with associated choroidal thinning. This finding suggested the presence of scleral deposits of glycosaminoglycans.
Conclusion: To our knowledge, this is the first case series in the literature encompassing patients with MPS VI with suspected deposits of glycosaminoglycans in the sclera. The better control of the systemic comorbidities, the increase in life expectancy, and the timely management of corneal disease have allowed the identification of new, late-onset ocular manifestations in MPS patients. In addition, new imaging techniques have introduced the possibility of better characterizing and understanding these manifestations.
导言粘多糖病 VI 型(MPS VI)是一种罕见的遗传代谢性疾病,主要是由于缺乏负责降解硫酸皮质素和硫酸软骨素的 N-乙酰半乳糖胺-4-硫酸酯酶。因此,部分降解的糖胺聚糖会广泛积聚。角膜混浊是 MPS 的标志性眼部特征。视网膜和巩膜受累在这种 MPS 中极为罕见。本研究旨在描述 MPS VI 患者眼底镜下的新变化:这是一个病例系列,涉及从遗传代谢病科转诊到我科的三名非相关患者。每位患者都进行了多模态成像。进行了眼底摄影和增强深度成像光学相干断层扫描(EDI-OCT)。在眼底照片上观察到多个黄色/橙色斑块区域,这些区域在EDI-OCT中与巩膜水平的中间反射率沉积物相对应,并伴有脉络膜变薄。这一发现表明巩膜上存在糖胺聚糖沉积物:据我们所知,这是文献中第一例疑似糖胺聚糖在巩膜沉积的 MPS VI 患者。随着全身性并发症得到更好的控制、预期寿命的延长以及角膜疾病的及时治疗,MPS 患者中新的、晚发的眼部表现得以发现。此外,新的成像技术也为更好地描述和理解这些表现提供了可能。
{"title":"Novel Fundoscopic Features in Mucopolysaccharidosis Type VI: Multimodal Evaluation of Scleral Deposits.","authors":"Augusto Magalhães, Margarida Ribeiro, Jorge Meira, Ana Filipa Moleiro, Esmeralda Rodrigues, Elisa Leão-Teles","doi":"10.1159/000540015","DOIUrl":"https://doi.org/10.1159/000540015","url":null,"abstract":"<p><strong>Introduction: </strong>Mucopolysaccharidosis type VI (MPS VI) is a rare inherited metabolic disorder, primarily attributed to the deficiency of the enzyme N-acetylgalactosamine-4-sulfatase, responsible for the degradation of dermatan sulfate and chondroitin-4-sulfate. Therefore, there is a widespread accumulation of partially degraded glycosaminoglycans. Corneal opacification is the hallmark ocular feature in the MPS. Retinal and scleral involvement in this MPS is extremely rare. The purpose of this work was to describe novel fundoscopic alterations present in patients with MPS VI.</p><p><strong>Case presentation: </strong>This is a case series involving three non-related patients referred to our department from the Unit of Inherited Metabolic Diseases. Multimodal imaging was performed in every patient. Fundus photography and enhanced depth imaging optical coherence tomography (EDI-OCT) were performed. Multiple areas of yellow/orange patches were observed on fundus photography, corresponding to areas in which deposits of intermediate reflectivity in the EDI-OCT could be seen at the scleral level with associated choroidal thinning. This finding suggested the presence of scleral deposits of glycosaminoglycans.</p><p><strong>Conclusion: </strong>To our knowledge, this is the first case series in the literature encompassing patients with MPS VI with suspected deposits of glycosaminoglycans in the sclera. The better control of the systemic comorbidities, the increase in life expectancy, and the timely management of corneal disease have allowed the identification of new, late-onset ocular manifestations in MPS patients. In addition, new imaging techniques have introduced the possibility of better characterizing and understanding these manifestations.</p>","PeriodicalId":9635,"journal":{"name":"Case Reports in Ophthalmology","volume":"15 1","pages":"642-647"},"PeriodicalIF":0.5,"publicationDate":"2024-08-13","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11521405/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"142544004","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}
Introduction: Complex corneal conditions present surgical challenges and necessitate innovation. Here, we present two cases where we performed intraocular lens trans-scleral fixation using the double-needle Yamane technique, followed by penetrating keratoplasty and vitrectomy using a temporary Landers wide-field keratoprosthesis.
Case presentation: Case 1 involved a 70-year-old man with an aphakic eye of bullous keratopathy and corneal opacity owing to multiple penetrating and endothelial keratoplasty, endophthalmitis, and herpetic keratitis. His visual acuity was counting fingers at 20 cm before surgery. Penetrating keratoplasty with vitrectomy and intraocular lens scleral fixation was performed using the double-needle Yamane technique, and 10 months postoperatively, his best-corrected visual acuity improved to 0.6, presenting a clear cornea. Case 2 involved a 62-year-old man who underwent penetrating keratoplasty twice for corneal perforation and therapeutic penetrating keratoplasty with vitrectomy for traumatic globe rupture, resulting in the loss of the intraocular lens. The patient exhibited graft failure, and his best-corrected visual acuity was 0.03. Utilizing a temporary Landers wide-field keratoprosthesis, we performed penetrating keratoplasty and intraocular lens trans-scleral fixation without complications. His final best-corrected visual acuity improved to 0.15 with a clear cornea.
Conclusions: Trans-scleral fixation of intraocular lens with penetrating keratoplasty, using temporary Landers wide-field keratoprosthesis, yielded positive clinical outcomes without serious complications.
{"title":"Enhancing Visual Acuity through Combined Intraocular Lens Fixation, Penetrating Keratoplasty, and Vitrectomy: The Role of Temporary Landers Wide-Field Keratoprosthesis.","authors":"Takahiro Zusho, Takashi Ono, Yukako Taketani, Mikiko Kimakura, Tetsuya Toyono, Koichiro Sugimoto, Taku Toyama, Takashi Ueta, Makoto Aihara, Takashi Miyai","doi":"10.1159/000540287","DOIUrl":"10.1159/000540287","url":null,"abstract":"<p><strong>Introduction: </strong>Complex corneal conditions present surgical challenges and necessitate innovation. Here, we present two cases where we performed intraocular lens trans-scleral fixation using the double-needle Yamane technique, followed by penetrating keratoplasty and vitrectomy using a temporary Landers wide-field keratoprosthesis.</p><p><strong>Case presentation: </strong>Case 1 involved a 70-year-old man with an aphakic eye of bullous keratopathy and corneal opacity owing to multiple penetrating and endothelial keratoplasty, endophthalmitis, and herpetic keratitis. His visual acuity was counting fingers at 20 cm before surgery. Penetrating keratoplasty with vitrectomy and intraocular lens scleral fixation was performed using the double-needle Yamane technique, and 10 months postoperatively, his best-corrected visual acuity improved to 0.6, presenting a clear cornea. Case 2 involved a 62-year-old man who underwent penetrating keratoplasty twice for corneal perforation and therapeutic penetrating keratoplasty with vitrectomy for traumatic globe rupture, resulting in the loss of the intraocular lens. The patient exhibited graft failure, and his best-corrected visual acuity was 0.03. Utilizing a temporary Landers wide-field keratoprosthesis, we performed penetrating keratoplasty and intraocular lens trans-scleral fixation without complications. His final best-corrected visual acuity improved to 0.15 with a clear cornea.</p><p><strong>Conclusions: </strong>Trans-scleral fixation of intraocular lens with penetrating keratoplasty, using temporary Landers wide-field keratoprosthesis, yielded positive clinical outcomes without serious complications.</p>","PeriodicalId":9635,"journal":{"name":"Case Reports in Ophthalmology","volume":"15 1","pages":"648-655"},"PeriodicalIF":0.5,"publicationDate":"2024-08-13","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11324278/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"141981805","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}
Pub Date : 2024-08-12eCollection Date: 2024-01-01DOI: 10.1159/000540125
Samira Jafari, Jonathan A Micieli
Introduction: Bartonella henselae, the causative agent of cat scratch disease (CSD), presents with diverse ocular manifestations, posing diagnostic challenges. This study aimed to elucidate the diagnostic complexities through a unique case.
Case presentation: A 42-year-old male presented with vision loss in the right eye, subsequent to flu-like symptoms following exposure to a stray kitten. Clinical examination revealed branch retinal artery occlusion (BRAO) in the right eye and neuroretinitis in the left, indicating concurrent ocular manifestations of CSD. Thorough investigations, including serological testing, ruled out alternative causes, highlighting the rarity of such coexisting ocular complications.
Conclusions: The coexistence of BRAO and neuroretinitis in different eyes underscores the variable presentation of CSD. Recognition of infectious etiologies, particularly Bartonella, is paramount in diagnosing ocular vasculopathies. This case emphasizes the importance of considering Bartonella infection in patients with ocular vascular occlusions, especially in the context of recent cat exposure and systemic symptoms suggestive of CSD.
{"title":"Cat Scratch Disease Presenting with Right Branch Retinal Artery Occlusion and Left Neuroretinitis.","authors":"Samira Jafari, Jonathan A Micieli","doi":"10.1159/000540125","DOIUrl":"10.1159/000540125","url":null,"abstract":"<p><strong>Introduction: </strong><i>Bartonella henselae</i>, the causative agent of cat scratch disease (CSD), presents with diverse ocular manifestations, posing diagnostic challenges. This study aimed to elucidate the diagnostic complexities through a unique case.</p><p><strong>Case presentation: </strong>A 42-year-old male presented with vision loss in the right eye, subsequent to flu-like symptoms following exposure to a stray kitten. Clinical examination revealed branch retinal artery occlusion (BRAO) in the right eye and neuroretinitis in the left, indicating concurrent ocular manifestations of CSD. Thorough investigations, including serological testing, ruled out alternative causes, highlighting the rarity of such coexisting ocular complications.</p><p><strong>Conclusions: </strong>The coexistence of BRAO and neuroretinitis in different eyes underscores the variable presentation of CSD. Recognition of infectious etiologies, particularly <i>Bartonella</i>, is paramount in diagnosing ocular vasculopathies. This case emphasizes the importance of considering <i>Bartonella</i> infection in patients with ocular vascular occlusions, especially in the context of recent cat exposure and systemic symptoms suggestive of CSD.</p>","PeriodicalId":9635,"journal":{"name":"Case Reports in Ophthalmology","volume":"15 1","pages":"628-632"},"PeriodicalIF":0.5,"publicationDate":"2024-08-12","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11324260/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"141981803","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}
Pub Date : 2024-08-12eCollection Date: 2024-01-01DOI: 10.1159/000540471
Pedro Javier Nuova
Introduction: The aim of this study was to describe a very rare case of endogenous bacterial endophthalmitis caused by Klebsiella pneumoniae in both eyes with a difference in the onset of symptoms of 14 months in an immunocompetent patient.
Case presentation: A 66-year-old immunocompetent man presented with asynchronous bilateral endogenous endophthalmitis produced by the K. pneumoniae bacterium at the starting point of a liver abscess after cholecystectomy surgery, causing endophthalmitis 1 year and 2 months apart between an eye and another. The first was diffuse anteroposterior endophthalmitis in the left eye that ended in visual loss and phthisis bulbi due to delayed initial diagnosis and established treatment, and the second was focal endophthalmitis in the right eye that preserved the organ and resulted in a vision of 20/20 due to early suspected diagnosis and rapid instituted treatment.
Conclusion: To our knowledge, this is the first published case of a long asynchronous bilateral endogenous bacterial endophthalmitis caused by K. pneumoniae with a prolonged difference of 14 months in the onset of symptoms between one eye and another. This case is a vision-threatening ophthalmologic emergency that can be associated with life-threatening systemic morbidities. The early diagnosis of infection represents a challenge for clinicians, ophthalmologists, and microbiologists.
{"title":"Bilateral Endogenous Bacterial Endophthalmitis with Asynchrony for 14 Months due to <i>Klebsiella pneumoniae</i>: A Case Report.","authors":"Pedro Javier Nuova","doi":"10.1159/000540471","DOIUrl":"10.1159/000540471","url":null,"abstract":"<p><strong>Introduction: </strong>The aim of this study was to describe a very rare case of endogenous bacterial endophthalmitis caused by <i>Klebsiella pneumoniae</i> in both eyes with a difference in the onset of symptoms of 14 months in an immunocompetent patient.</p><p><strong>Case presentation: </strong>A 66-year-old immunocompetent man presented with asynchronous bilateral endogenous endophthalmitis produced by the <i>K. pneumoniae</i> bacterium at the starting point of a liver abscess after cholecystectomy surgery, causing endophthalmitis 1 year and 2 months apart between an eye and another. The first was diffuse anteroposterior endophthalmitis in the left eye that ended in visual loss and phthisis bulbi due to delayed initial diagnosis and established treatment, and the second was focal endophthalmitis in the right eye that preserved the organ and resulted in a vision of 20/20 due to early suspected diagnosis and rapid instituted treatment.</p><p><strong>Conclusion: </strong>To our knowledge, this is the first published case of a long asynchronous bilateral endogenous bacterial endophthalmitis caused by <i>K. pneumoniae</i> with a prolonged difference of 14 months in the onset of symptoms between one eye and another. This case is a vision-threatening ophthalmologic emergency that can be associated with life-threatening systemic morbidities. The early diagnosis of infection represents a challenge for clinicians, ophthalmologists, and microbiologists.</p>","PeriodicalId":9635,"journal":{"name":"Case Reports in Ophthalmology","volume":"15 1","pages":"633-641"},"PeriodicalIF":0.5,"publicationDate":"2024-08-12","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11324282/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"141981802","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}
Pub Date : 2024-08-08eCollection Date: 2024-01-01DOI: 10.1159/000540051
Brianna Lu, Jovi C Y Wong, Justin Kritzinger, David T Wong
Introduction: Primary vitreoretinal lymphoma (PVRL) is a rare malignant tumor that typically involves the retina, vitreous, or optic nerve head. PVRL often occurs concurrently with central nervous system lymphoma. Here, we present the first report of a patient with biopsy-confirmed PVRL presenting solely with asymptomatic peripheral drusenoid lesions.
Case presentation: A woman in her 70s presented with new elevated amelanotic yellow lesions with overlying pigment in both of her eyes not previously seen prior to cataract surgery. Over the next 4 months, there was waxing and waning of lesions which resolved and first appeared in the right eye and then the left. A diagnostic vitrectomy of the left eye revealed B-cell lymphoma. The patient elected for initial treatment with radiation therapy of both orbits. A new lesion was identified in her right eye nearly 18 months after starting maintenance therapy with ibrutinib, following which systemic chemotherapy with methotrexate was initiated.
Conclusion: Elevated clinical suspicion for a malignant process is needed for patients with progressive new retinal lesions in older age. Local radiation therapy to the orbits alone may not be sufficient to prevent progression despite initial presentation showing confinement of disease to the intraocular space.
简介:原发性玻璃体视网膜淋巴瘤(PVRL原发性玻璃体视网膜淋巴瘤(PVRL)是一种罕见的恶性肿瘤,通常累及视网膜、玻璃体或视神经头。原发性玻璃体视网膜淋巴瘤通常与中枢神经系统淋巴瘤同时发生。在此,我们首次报告了一名经活检确诊的 PVRL 患者,该患者仅表现为无症状的外周性类风湿性病变:一位 70 多岁的妇女在接受白内障手术之前,双眼出现了新的高出皮肤的黄色病变,并伴有色素沉着。在接下来的 4 个月中,病变逐渐消退,先出现在右眼,然后是左眼。左眼诊断性玻璃体切除术发现了 B 细胞淋巴瘤。患者选择对两个眼眶进行放射治疗。在开始接受伊布替尼维持治疗近18个月后,她的右眼发现了新的病变,随后开始接受甲氨蝶呤全身化疗:结论:对于老年视网膜进行性新病变患者,临床上需要高度怀疑恶性病变。结论:对于老年视网膜新病变进展期患者,临床上需要提高对恶性病变的怀疑。尽管最初的症状显示病变局限于眼内间隙,但仅对眼眶进行局部放射治疗可能不足以防止病变进展。
{"title":"Primary Vitreoretinal Lymphoma Presenting Solely with Asymptomatic Peripheral Drusenoid Lesions.","authors":"Brianna Lu, Jovi C Y Wong, Justin Kritzinger, David T Wong","doi":"10.1159/000540051","DOIUrl":"10.1159/000540051","url":null,"abstract":"<p><strong>Introduction: </strong>Primary vitreoretinal lymphoma (PVRL) is a rare malignant tumor that typically involves the retina, vitreous, or optic nerve head. PVRL often occurs concurrently with central nervous system lymphoma. Here, we present the first report of a patient with biopsy-confirmed PVRL presenting solely with asymptomatic peripheral drusenoid lesions.</p><p><strong>Case presentation: </strong>A woman in her 70s presented with new elevated amelanotic yellow lesions with overlying pigment in both of her eyes not previously seen prior to cataract surgery. Over the next 4 months, there was waxing and waning of lesions which resolved and first appeared in the right eye and then the left. A diagnostic vitrectomy of the left eye revealed B-cell lymphoma. The patient elected for initial treatment with radiation therapy of both orbits. A new lesion was identified in her right eye nearly 18 months after starting maintenance therapy with ibrutinib, following which systemic chemotherapy with methotrexate was initiated.</p><p><strong>Conclusion: </strong>Elevated clinical suspicion for a malignant process is needed for patients with progressive new retinal lesions in older age. Local radiation therapy to the orbits alone may not be sufficient to prevent progression despite initial presentation showing confinement of disease to the intraocular space.</p>","PeriodicalId":9635,"journal":{"name":"Case Reports in Ophthalmology","volume":"15 1","pages":"607-613"},"PeriodicalIF":0.5,"publicationDate":"2024-08-08","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11324244/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"141981809","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}
Pub Date : 2024-08-08eCollection Date: 2024-01-01DOI: 10.1159/000540014
Yodpong Chantarasorn, Kriengsak Funilkul
Introduction: We aim to report the clinical course of a patient with pachychoroidopathy who experienced regression of subfoveal drusen during cholesterol treatment using PCSK9 inhibitors.
Case presentation: A 62-year-old woman who was visually asymptomatic complained of recent visual loss in the left eye (OS). She was diagnosed with foveal pachydrusen (OS) that had remained stable for 10 years. Three months after starting cholesterol treatment with a PCSK9 inhibitor, the latest class of lipid-lowering medication, her vision improved in parallel with gradual regression of material deposited beneath the retinal pigment epithelium (RPE). Recurrence of drusen was observed after discontinuing the drug.
Conclusions: Use of PCSK9 inhibitors may improve the retina's lipid homeostasis by increasing the number of RPE-LDL receptors and partly contribute to the improvement of ocular phenotypes associated with dysfunctional RPE in pachychoroidopathy.
{"title":"A Temporal Association between Regression of Pachydrusen and Use of Proprotein Convertase Subtilisin Kexin 9 Inhibitor: A Case Report.","authors":"Yodpong Chantarasorn, Kriengsak Funilkul","doi":"10.1159/000540014","DOIUrl":"10.1159/000540014","url":null,"abstract":"<p><strong>Introduction: </strong>We aim to report the clinical course of a patient with pachychoroidopathy who experienced regression of subfoveal drusen during cholesterol treatment using PCSK9 inhibitors.</p><p><strong>Case presentation: </strong>A 62-year-old woman who was visually asymptomatic complained of recent visual loss in the left eye (OS). She was diagnosed with foveal pachydrusen (OS) that had remained stable for 10 years. Three months after starting cholesterol treatment with a PCSK9 inhibitor, the latest class of lipid-lowering medication, her vision improved in parallel with gradual regression of material deposited beneath the retinal pigment epithelium (RPE). Recurrence of drusen was observed after discontinuing the drug.</p><p><strong>Conclusions: </strong>Use of PCSK9 inhibitors may improve the retina's lipid homeostasis by increasing the number of RPE-LDL receptors and partly contribute to the improvement of ocular phenotypes associated with dysfunctional RPE in pachychoroidopathy.</p>","PeriodicalId":9635,"journal":{"name":"Case Reports in Ophthalmology","volume":"15 1","pages":"614-620"},"PeriodicalIF":0.5,"publicationDate":"2024-08-08","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11324243/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"141981788","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}
Introduction: Neuromyelitis optica spectrum disorder (NMOSD) is an autoimmune disorder that principally targets the central nervous system, specifically the spinal cord and optic nerves. NMOSD is often associated with thyroid pathologies such as Graves' disease or Hashimoto's thyroiditis. Thyroid eye disease (TED) is an autoimmune condition characterized by inflammation and hypertrophy of the extraocular muscles. Dysthyroid optic neuropathy (DON), a critical complication of TED, may lead to irreversible visual loss. We report a case of DON complicated by NMOSD.
Case presentation: We report a case of an autoimmune disease presenting as DON in a 44-year-old Japanese woman with a history of Graves' disease, who experienced reduced visual acuity and orbital pain. Brain magnetic resonance imaging disclosed hypertrophy of the rectus muscles, compressing the optic nerve bilaterally. Consequently, she was diagnosed with DON and underwent three courses of steroid semi-pulse therapy and left orbital decompression surgery, alleviating optic nerve compression. Nevertheless, the visual prognosis remained poor. A subsequent serological test showed positive for aquaporin-4 antibody. Treatment with satralizumab, an interleukin-6 receptor monoclonal antibody, was initiated in conjunction with steroids to suppress the autoimmune response and reduce NMOSD relapse risk. Following this treatment, no NMOSD recurrences were reported.
Conclusion: This case highlights the necessity of considering the possible coexistence of DON and NMOSD in patients with autoimmune diseases.
简介神经脊髓炎视神经谱系障碍(NMOSD)是一种自身免疫性疾病,主要针对中枢神经系统,特别是脊髓和视神经。NMOSD 通常与甲状腺病变有关,如巴塞杜氏病或桥本氏甲状腺炎。甲状腺眼病(TED)是一种自身免疫性疾病,其特征是眼外肌发炎和肥大。甲状腺功能减退性视神经病变(DON)是TED的一种重要并发症,可能导致不可逆的视力丧失。我们报告了一例并发 NMOSD 的 DON 病例:我们报告了一例表现为 DON 的自身免疫性疾病,患者是一名 44 岁的日本女性,曾患巴塞杜氏病,视力下降,眼眶疼痛。脑磁共振成像显示她的直肌肥大,压迫了双侧视神经。因此,她被诊断为 DON,并接受了三个疗程的类固醇半脉冲治疗和左眼眶减压手术,缓解了视神经压迫症状。然而,视力预后仍然不佳。随后的血清学检测显示,aquaporin-4 抗体呈阳性。开始使用白细胞介素-6受体单克隆抗体satralizumab与类固醇联合治疗,以抑制自身免疫反应,降低NMOSD复发风险。治疗后,未再报告NMOSD复发:本病例强调了考虑自身免疫性疾病患者可能同时患有 DON 和 NMOSD 的必要性。
{"title":"Dysthyroid Optic Neuropathy Complicated by Neuromyelitis Optica Spectrum Disorder: A Case Report.","authors":"Teruaki Tokuhisa, Takaaki Hayashi, Shusaku Omoto, Ai Kozaki, Toshu Inoue, Tadashi Nakano","doi":"10.1159/000540496","DOIUrl":"10.1159/000540496","url":null,"abstract":"<p><strong>Introduction: </strong>Neuromyelitis optica spectrum disorder (NMOSD) is an autoimmune disorder that principally targets the central nervous system, specifically the spinal cord and optic nerves. NMOSD is often associated with thyroid pathologies such as Graves' disease or Hashimoto's thyroiditis. Thyroid eye disease (TED) is an autoimmune condition characterized by inflammation and hypertrophy of the extraocular muscles. Dysthyroid optic neuropathy (DON), a critical complication of TED, may lead to irreversible visual loss. We report a case of DON complicated by NMOSD.</p><p><strong>Case presentation: </strong>We report a case of an autoimmune disease presenting as DON in a 44-year-old Japanese woman with a history of Graves' disease, who experienced reduced visual acuity and orbital pain. Brain magnetic resonance imaging disclosed hypertrophy of the rectus muscles, compressing the optic nerve bilaterally. Consequently, she was diagnosed with DON and underwent three courses of steroid semi-pulse therapy and left orbital decompression surgery, alleviating optic nerve compression. Nevertheless, the visual prognosis remained poor. A subsequent serological test showed positive for aquaporin-4 antibody. Treatment with satralizumab, an interleukin-6 receptor monoclonal antibody, was initiated in conjunction with steroids to suppress the autoimmune response and reduce NMOSD relapse risk. Following this treatment, no NMOSD recurrences were reported.</p><p><strong>Conclusion: </strong>This case highlights the necessity of considering the possible coexistence of DON and NMOSD in patients with autoimmune diseases.</p>","PeriodicalId":9635,"journal":{"name":"Case Reports in Ophthalmology","volume":"15 1","pages":"621-627"},"PeriodicalIF":0.5,"publicationDate":"2024-08-08","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11324271/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"141981804","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}
Pub Date : 2024-08-07eCollection Date: 2024-01-01DOI: 10.1159/000538439
Dan Ramon, Nadav Shemesh, Asaf Achiron
Introduction: In this case study, we present the "Ballerina" sign as a potentially valuable clinical indicator for detecting posterior capsular rupture (PCR) during cataract surgery. The purpose of this study is to highlight the significance of PCR in the context of cataract surgery and introduce this novel sign.
Case presentation: During the cataract operation on a 70-year-old patient, we observed a spiraling fragment of the nucleus attached to a vitreous string. Subsequently, an anterior vitrectomy was successfully performed without further intraoperative complications, followed by the insertion of a 3-piece lens into the sulcus. Positive visual outcomes were observed during postoperative follow-up appointments at 1 day, 2 weeks, and 3 months.
Conclusion: The documentation of the "Ballerina" sign in the medical literature is innovative, offering ophthalmic surgeons a valuable tool for early PCR detection during cataract surgeries.
{"title":"The \"Ballerina\" Sign: Posterior Capsular Rupture during Cataract Surgery.","authors":"Dan Ramon, Nadav Shemesh, Asaf Achiron","doi":"10.1159/000538439","DOIUrl":"10.1159/000538439","url":null,"abstract":"<p><strong>Introduction: </strong>In this case study, we present the \"Ballerina\" sign as a potentially valuable clinical indicator for detecting posterior capsular rupture (PCR) during cataract surgery. The purpose of this study is to highlight the significance of PCR in the context of cataract surgery and introduce this novel sign.</p><p><strong>Case presentation: </strong>During the cataract operation on a 70-year-old patient, we observed a spiraling fragment of the nucleus attached to a vitreous string. Subsequently, an anterior vitrectomy was successfully performed without further intraoperative complications, followed by the insertion of a 3-piece lens into the sulcus. Positive visual outcomes were observed during postoperative follow-up appointments at 1 day, 2 weeks, and 3 months.</p><p><strong>Conclusion: </strong>The documentation of the \"Ballerina\" sign in the medical literature is innovative, offering ophthalmic surgeons a valuable tool for early PCR detection during cataract surgeries.</p>","PeriodicalId":9635,"journal":{"name":"Case Reports in Ophthalmology","volume":"15 1","pages":"602-606"},"PeriodicalIF":0.5,"publicationDate":"2024-08-07","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11324203/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"141981810","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}