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Endogenous Aspergillus endophthalmitis complicated by florid retinal neovascularization: double trouble. 内源性曲霉菌性眼内炎并发绚丽的视网膜新生血管:双重困扰。
Q3 Medicine Pub Date : 2025-09-24 DOI: 10.1097/ICB.0000000000001823
Pooja Bansal, Shruti Bhattacharya, Khushboo Chawla, Priyadarshi Gupta, Meenakshi Thakar, Shivani Tyagi, Poonam Loomba

Purpose: To describe the presentation, clinical course, and management of a patient with endogenous Aspergillus endophthalmitis complicated by florid retinal neovascularization.

Methods: A 32-year-old immunocompetent female developed a loss of vision in the right eye one week after receiving intravenous dextrose infusions in a local healthcare setting. Ocular examination revealed dense vitreous exudates for which an urgent vitrectomy was performed, with a presumptive diagnosis of endogenous endophthalmitis. Microbiological analysis of vitreous samples revealed Aspergillus flavus.

Results: Intravitreal and oral antifungals lead to the resolution of inflammation. However, the patient soon presented with large neovascular fronds involving the disc and posterior pole, rapidly progressing to tractional retinal detachment involving the macula. The intravitreal anti-vascular endothelial growth factor (anti-VEGF) ranibizumab was injected, followed by repeat vitrectomy and membrane dissection with gas tamponade. This resulted in a satisfactory anatomical outcome but poor visual gain due to submacular scarring.

Conclusions: Endogenous Aspergillus endophthalmitis warrants early diagnosis and aggressive management to avoid complications such as ocular neovascularization, retinal detachment, and phthisis bulbi. Retinal neovascularization in endogenous endophthalmitis is a rarely observed finding, but if present, it indicates severe ongoing inflammation or underlying retinal ischemia.

目的:描述一位内源性曲霉菌性眼内炎并发绚丽的视网膜新生血管的患者的表现、临床过程和治疗。方法:一名32岁具有免疫功能的女性在当地医疗机构接受静脉葡萄糖输注一周后右眼视力丧失。眼科检查发现密集的玻璃体渗出物,进行了紧急玻璃体切除术,推定诊断为内源性眼内炎。玻璃体样品的微生物学分析显示为黄曲霉。结果:玻璃体内和口服抗真菌药物均能有效缓解炎症。然而,患者很快表现为累及椎间盘和后极的大新生血管瘤,并迅速发展为累及黄斑的牵引性视网膜脱离。玻璃体内注射抗血管内皮生长因子(anti-VEGF)雷尼珠单抗,然后重复玻璃体切除和气体填塞膜剥离。这导致了令人满意的解剖结果,但由于黄斑下疤痕导致视力下降。结论:内源性曲霉菌性眼内炎需要早期诊断和积极治疗,以避免并发症,如眼部新生血管、视网膜脱离和球炎。内源性眼内炎的视网膜新生血管很少观察到,但如果存在,则表明严重的持续炎症或潜在的视网膜缺血。
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引用次数: 0
Peripapillary retinoschisis associated with open angle glaucoma responding to dorzolamide. 乳头周围视网膜裂伴开角型青光眼对多唑胺的反应。
Q3 Medicine Pub Date : 2025-09-24 DOI: 10.1097/ICB.0000000000001822
Omar Moussa, Erin Flynn, Jedrzej Golebka, Stanley Chang

Purpose: Presenting a case of peripapillary retinoschisis in a patient with glaucomatous optic neuropathy that resolved after changing therapy from topical bimatoprost to timolol/dorzolamide.

Methods: A 70-year-old female who presented for a second opinion for peripapillary retinoschisis in the left eye.

Results: The patient's visual acuity was 20/25 and 20/30 in the right and left eyes, respectively. Dilated fundoscopy revealed posterior vitreous detachment (PVD) of the left eye, temporal cupping with a superotemporal notch of the left optic nerve, and bilateral macular drusen and pigmentary changes of the retinal pigment epithelium (RPE). Optical coherence tomography of the macula of the left eye showed a PVD, drusen, and peripapillary retinoschisis extending to and involving the fovea. Topical prostaglandin analogue eye drop was discontinued and replaced by dorzolamide/timolol. The peripapillary retinoschisis resolved after medication change over the following 48 months and has not recurred.

Conclusion: We present a case of peripapillary retinoschisis in a phakic patient that developed during treatment with a topical prostaglandin analogue. The schisis resolved after switching the prostaglandin analogue to a topical carbonic anhydrase inhibitor. We suggest that the prostaglandin may have had a role in initiating peripapillary retinoschisis associated with glaucomatous optic neuropathy and that carbonic anhydrase inhibitors are a potential treatment for this condition.

目的:报告一例青光眼视神经病变患者的乳头周围视网膜裂,在局部治疗由比马前列素改为替马洛尔/多唑胺后消退。方法:一位70岁女性,因左眼乳头周围视网膜裂求诊。结果:患者右眼视力20/25,左眼视力20/30。眼底扩张镜检查发现左眼玻璃体后脱离(PVD),左侧视神经颞上切迹颞杯状突起,双侧黄斑水肿及视网膜色素上皮(RPE)色素改变。左眼黄斑的光学相干断层扫描显示PVD、结节和乳头周围视网膜裂延伸并累及中央窝。停用局部前列腺素类似物滴眼液,改为多唑胺/替莫洛尔。在接下来的48个月里,改变药物治疗后,乳头周围视网膜裂消失了,没有复发。结论:我们提出了一个病例乳头周围视网膜裂在一个phaic患者,发展与局部前列腺素类似物治疗期间。在将前列腺素类似物转换为局部碳酸酐酶抑制剂后,分裂解决了。我们认为前列腺素可能在引发与青光眼视神经病变相关的乳头周围视网膜裂中起作用,并且碳酸酐酶抑制剂是这种疾病的潜在治疗方法。
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引用次数: 0
Hypertensive Proliferative Choriorretinopathy. 高血压增生性脉络膜视网膜病变。
Q3 Medicine Pub Date : 2025-09-24 DOI: 10.1097/ICB.0000000000001819
Guilherme Portas-Almeida, Catarina Oliveira Rodrigues, Rita Guerra Pinto

Purpose: To report a case of hypertensive chorioretinopathy complicated by anterior segment neovascularization, refractory to PRP.

Methods: Case Report and Literature Review.

Results: A 57-year-old male with poorly controlled systemic hypertension (exceeding 200/100 mmHg) experienced progressive visual acuity loss in the left eye over two years, with worsening in the last six months. Slit-Lamp examination revealed neovascularization of the angle in the left eye. Fundoscopy disclosed pale choroidal vessels temporal to the macula of both eyes, as well as additional retinochoroidal findings, such as telangiectatic vessels and a rounded shaped pale subretinal region on the right eye and combined vascular occlusion in the left inferior hemi-retina. Fluorescein and indocyanine angiography showed delayed choroidal perfusion and marked retinal ischemia, consistent with severe Hypertensive Chorioretinopathy. Initial treatment with monthly intravitreal anti-VEGF injections and PRP led to remission of anterior segment neovascularization, but recurrence occurred after two months, requiring additional anti-VEGF therapy.

Conclusion: The insufficient response to PRP in this case suggests that choroidal ischaemia may not be as amenable to laser treatment as ischaemic retina. Highlighting the importance of regular follow-up, this report underscores the need for comprehensive hypertensive control to prevent vision-threatening outcomes.

目的:报告1例高血压性脉络膜视网膜病变合并前段新生血管,PRP难治性。方法:病例报告和文献复习。结果:一名57岁男性,患有控制不良的全身性高血压(超过200/100 mmHg),左眼视力下降超过2年,并在最近6个月恶化。裂隙灯检查显示左眼角血管新生。眼底镜检查发现双眼黄斑颞部脉络膜血管苍白,同时发现其他视网膜脉络膜病变,如右眼毛细血管扩张血管和圆形视网膜下区苍白,左侧下半视网膜合并血管闭塞。荧光素和吲哚菁血管造影显示脉络膜灌注延迟和明显的视网膜缺血,与严重的高血压脉络膜视网膜病变一致。最初每月进行玻璃体内抗vegf注射和PRP治疗可缓解前段新生血管,但两个月后复发,需要额外的抗vegf治疗。结论:本病例对PRP的反应不足,提示脉络膜缺血可能不如视网膜缺血那样适合激光治疗。该报告强调了定期随访的重要性,强调了全面控制高血压以预防威胁视力的结果的必要性。
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引用次数: 0
A Pediatric Case of Leukemia Retinopathy with Peripheral Retinal Changes Detected by Ultra-Widefield Optical Coherence Tomography. 用超宽视场光学相干断层扫描检测外周视网膜病变的儿童白血病视网膜病变1例。
Q3 Medicine Pub Date : 2025-09-23 DOI: 10.1097/ICB.0000000000001814
Yuto Uchida, Kazuma Saito, Hideo Akiyama

Purpose: We report a case of chronic myeloid leukemia (CML) diagnosed with ocular symptoms, in which dilated and tortuous retinal vessels were observed and monitored using ultra-widefield optical coherence tomography angiography (OCTA).

Methods: Case report of a 13-year-old girl who presented with bilateral photopsia and misty vision in the right eye.

Results: Fundus examination revealed dilated and tortuous retinal arteriovenous veins and yellowish-white exudative lesions along the retinal vessels in both eyes. A similar yellowish-white exudative lesion was observed in the macula of the right eye. A blood test showed a high white blood cell count of 588,400/µL. CML was diagnosed by hematological examination. After one month of treatment for CML, the patient was in complete hematologic remission, and the yellowish-white exudative lesions and dilatation and tortuosity of the retinal vessels improved. Ultra-widefield OCTA revealed dilated and tortuous retinal capillaries, as well as areas of non-perfusion at the initial visit; these findings improved after treatment.

Conclusion: Ultra-widefield OCTA can be used to non-invasively observe extensive retinal vascular and capillary changes in leukemic retinopathy over time.

目的:我们报告一例诊断为眼部症状的慢性髓性白血病(CML),使用超宽视场光学相干断层血管造影(OCTA)观察和监测视网膜血管扩张和弯曲。方法:报告1例13岁女童右眼双侧失光及模糊视力。结果:眼底检查示双眼视网膜动静脉扩张弯曲,视网膜血管呈黄白色渗出病变。右眼黄斑可见类似的黄白色渗出性病变。血液检查显示白细胞计数高达588,400/µL。通过血液学检查诊断CML。治疗CML 1个月后,患者血液学完全缓解,黄白色渗出病灶及视网膜血管扩张弯曲改善。超宽视场OCTA显示初次就诊时视网膜毛细血管扩张和弯曲,以及非灌注区域;这些发现在治疗后有所改善。结论:超宽视场OCTA可用于无创观察白血病视网膜病变长期广泛的视网膜血管和毛细血管变化。
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引用次数: 0
Choroidal neovascularization in AZOOR. AZOOR患者脉络膜新生血管。
Q3 Medicine Pub Date : 2025-09-23 DOI: 10.1097/ICB.0000000000001815
Nour Boungab, Adélaïde Toutee, Youssef Gourinda, Michel Paques, Sarah Mrejen

Purpose: To report mul4modal imaging features in a case of an acute zonal occult outer re4nopathy (AZOOR) complicated with choroidal neovascularization during a follow-up of 18 months.

Methods: A 53-year-old woman was referred for bilateral decreased vision and photopsia.Fundus examina4on showed a circular posterior lesion with a far-peripheral lesion in the right eye and a single peripapillary lesion in the leb eye. The lesions were depigmented in both eyes, bordered by an interrupted yellow-orange line in a beaded appearance. The right eye featured macular atrophy with pigmentary migra4on.

Results: Clinical examina4on, autofluorescence and angiography confirmed the diagnosis of AZOOR. OCT-B scan and angiography OCT confirmed the presence of an ac4ve choroidal neovascularization in the right eye. Intravitreal an4-VEGF was injected in the right eye with a favorable response.

Conclusion: AZOOR is a rare re4nal en4ty with a poorly understood pathogenic mechanism. Choroidal neovascularization is an extremely rare complica4on in AZOOR. We describe mul4modal imaging features of AZOOR with choroidal neovascularization treated by an4-VEGF.

目的:报告1例急性区域性隐匿性外视网膜病变(AZOOR)合并脉络膜新生血管的多模态影像学特征,随访18个月。方法:一名53岁女性因双侧视力下降及视力减退而就诊。眼底检查显示右眼圆形后病变伴远周病变,右眼单一乳头周围病变。双眼病变脱色,以黄橙色线为界,呈串珠状。右眼黄斑萎缩伴色素迁移。结果:临床检查、自身荧光和血管造影证实AZOOR的诊断。ct - b扫描和血管造影证实右眼存在活跃的脉络膜新生血管。右眼玻璃体内注射an4-VEGF,反应良好。结论:AZOOR是一种罕见的肾脏疾病,其发病机制尚不清楚。脉络膜新生血管是AZOOR极为罕见的并发症。我们描述了an4-VEGF治疗AZOOR伴脉络膜新生血管的多模态影像学特征。
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引用次数: 0
A 10-year-old Girl with an 8-Month History of Progressive Photophobia and Hemeralopia. 一名10岁女童,有8个月进行性畏光及近视史。
Q3 Medicine Pub Date : 2025-09-23 DOI: 10.1097/ICB.0000000000001818
Maximilian D Kong, Jin Kyun Oh, Johnathan A Bailey, Abdhel Exinor, Chanakarn Piamjitchol, Noah Heaps, Stephen H Tsang

Purpose: To report a case of nonparaneoplastic autoimmune retinopathy in a 10-year-old female with chronic granulomatous disease (CGD) due to compound heterozygous mutations in NCF1.

Methods: The patient was evaluated at Columbia University Irving Medical Center using a comprehensive multimodal imaging protocol that included color fundus photography, short-wave autofluorescence (SW-AF), and spectral-domain optical coherence tomography (SD-OCT). Functional assessments were conducted using Goldmann visual fields and full-field electroretinography (ERG) were completed on-site and followed the ISCEV standard protocols to ensure consistent and reproducible measurements of photoreceptor activity.

Results: A 10-year-old female presented with decreased visual acuity, light sensitivity, and decreased color vision over the past 8 months with no improvement upon refraction. Upon detection of anti-retinal antibodies, treatment was initiated with stabilization and improvement of visual acuity and ERG responses with rituximab treatment. Whole genome sequencing revealed two mutations in NCF1 causing chronic granulomatous disease.

Conclusions: We present a case of autoimmune retinopathy in a CGD patient with compound heterozygous mutations in the NCF1 gene. This novel association may suggest that autoimmunity may be involved the development of the chorioretinal lesions seen in CGD patients. It also supports the use of rituximab in the management of autoimmune retinopathy.

目的:报告一例因NCF1复合杂合突变引起的慢性肉芽肿病(CGD)的10岁女性非副肿瘤自身免疫性视网膜病变。方法:患者在哥伦比亚大学欧文医学中心使用综合多模态成像方案进行评估,包括彩色眼底摄影、短波自身荧光(SW-AF)和光谱域光学相干断层扫描(SD-OCT)。使用Goldmann视野进行功能评估,并在现场完成全视野视网膜电图(ERG),并遵循ISCEV标准方案,以确保光感受器活性测量的一致性和可重复性。结果:一名10岁女性在过去的8个月里表现为视力、光敏性和色觉下降,屈光无改善。检测到抗视网膜抗体后,开始治疗,利妥昔单抗治疗视力和ERG反应稳定和改善。全基因组测序揭示了导致慢性肉芽肿病的NCF1的两个突变。结论:我们报告了一例自身免疫性视网膜病变的CGD患者与复合杂合突变的NCF1基因。这种新的关联可能表明自身免疫可能参与了CGD患者中绒毛膜视网膜病变的发展。它还支持使用利妥昔单抗管理自身免疫性视网膜病变。
{"title":"A 10-year-old Girl with an 8-Month History of Progressive Photophobia and Hemeralopia.","authors":"Maximilian D Kong, Jin Kyun Oh, Johnathan A Bailey, Abdhel Exinor, Chanakarn Piamjitchol, Noah Heaps, Stephen H Tsang","doi":"10.1097/ICB.0000000000001818","DOIUrl":"https://doi.org/10.1097/ICB.0000000000001818","url":null,"abstract":"<p><strong>Purpose: </strong>To report a case of nonparaneoplastic autoimmune retinopathy in a 10-year-old female with chronic granulomatous disease (CGD) due to compound heterozygous mutations in NCF1.</p><p><strong>Methods: </strong>The patient was evaluated at Columbia University Irving Medical Center using a comprehensive multimodal imaging protocol that included color fundus photography, short-wave autofluorescence (SW-AF), and spectral-domain optical coherence tomography (SD-OCT). Functional assessments were conducted using Goldmann visual fields and full-field electroretinography (ERG) were completed on-site and followed the ISCEV standard protocols to ensure consistent and reproducible measurements of photoreceptor activity.</p><p><strong>Results: </strong>A 10-year-old female presented with decreased visual acuity, light sensitivity, and decreased color vision over the past 8 months with no improvement upon refraction. Upon detection of anti-retinal antibodies, treatment was initiated with stabilization and improvement of visual acuity and ERG responses with rituximab treatment. Whole genome sequencing revealed two mutations in NCF1 causing chronic granulomatous disease.</p><p><strong>Conclusions: </strong>We present a case of autoimmune retinopathy in a CGD patient with compound heterozygous mutations in the NCF1 gene. This novel association may suggest that autoimmunity may be involved the development of the chorioretinal lesions seen in CGD patients. It also supports the use of rituximab in the management of autoimmune retinopathy.</p>","PeriodicalId":53580,"journal":{"name":"Retinal Cases and Brief Reports","volume":" ","pages":""},"PeriodicalIF":0.0,"publicationDate":"2025-09-23","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"145132368","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}
引用次数: 0
The serial changes of near-infrared autofluorescence in solar retinopathy. 近红外自身荧光在太阳视网膜病变中的系列变化。
Q3 Medicine Pub Date : 2025-09-23 DOI: 10.1097/ICB.0000000000001816
Ya-Chi Huang, Jian-Sheng Wu

Purpose: To elucidate the serial changes of near-infrared autofluorescence (NIA) imaging in an adolescent male with unilateral solar retinopathy.

Methods: This is a retrospective case report.

Results: The patient received a course of steroids, resulting in gradual improvement in visual acuity from 20/100 to 20/50 over 12 months. NIA findings showed that the hyperautofluorescent ring dissipated, leaving a hypoautofluorescent lesion. While OCT displayed persistent photoreceptor disruption, NIA revealed subtle changes over the follow-up period. The correlation between NIA findings and structural alterations in the photoreceptors and retinal pigment epithelium (RPE) was consistent across all imaging modalities.

Conclusion: NIA imaging serves as a non-invasive tool for monitoring disease progression and evaluating the status of photoreceptors and RPE in solar retinopathy. The changes observed in NIA suggest that early intervention may positively impact the visual prognosis and disease progression. NIA may be a valuable adjunctive tool in the comprehensive evaluation of solar retinopathy.

目的:探讨青少年男性单侧太阳视网膜病变的近红外自身荧光(NIA)影像学变化。方法:回顾性分析病例。结果:患者接受了一个疗程的类固醇治疗,视力在12个月内从20/100逐渐改善到20/50。NIA结果显示高自荧光环消散,留下低自荧光病变。虽然OCT显示持续的光感受器破坏,但NIA在随访期间显示细微的变化。NIA发现与光感受器和视网膜色素上皮(RPE)结构改变之间的相关性在所有成像方式中都是一致的。结论:NIA成像是一种非侵入性的工具,可用于监测太阳视网膜病变的疾病进展,评估光感受器和RPE的状态。NIA中观察到的变化提示早期干预可能对视力预后和疾病进展有积极影响。NIA可能是一个有价值的辅助工具,在综合评价太阳视网膜病变。
{"title":"The serial changes of near-infrared autofluorescence in solar retinopathy.","authors":"Ya-Chi Huang, Jian-Sheng Wu","doi":"10.1097/ICB.0000000000001816","DOIUrl":"https://doi.org/10.1097/ICB.0000000000001816","url":null,"abstract":"<p><strong>Purpose: </strong>To elucidate the serial changes of near-infrared autofluorescence (NIA) imaging in an adolescent male with unilateral solar retinopathy.</p><p><strong>Methods: </strong>This is a retrospective case report.</p><p><strong>Results: </strong>The patient received a course of steroids, resulting in gradual improvement in visual acuity from 20/100 to 20/50 over 12 months. NIA findings showed that the hyperautofluorescent ring dissipated, leaving a hypoautofluorescent lesion. While OCT displayed persistent photoreceptor disruption, NIA revealed subtle changes over the follow-up period. The correlation between NIA findings and structural alterations in the photoreceptors and retinal pigment epithelium (RPE) was consistent across all imaging modalities.</p><p><strong>Conclusion: </strong>NIA imaging serves as a non-invasive tool for monitoring disease progression and evaluating the status of photoreceptors and RPE in solar retinopathy. The changes observed in NIA suggest that early intervention may positively impact the visual prognosis and disease progression. NIA may be a valuable adjunctive tool in the comprehensive evaluation of solar retinopathy.</p>","PeriodicalId":53580,"journal":{"name":"Retinal Cases and Brief Reports","volume":" ","pages":""},"PeriodicalIF":0.0,"publicationDate":"2025-09-23","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"145132385","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}
引用次数: 0
Retinal Vasculitis Associated with Tislelizumab in a Patient with Esophageal Squamous Cell Carcinoma. 食管鳞状细胞癌患者视网膜血管炎与Tislelizumab相关。
Q3 Medicine Pub Date : 2025-09-23 DOI: 10.1097/ICB.0000000000001817
Estel la Rojas-Pineda, Nuria Cintas, Anna Bruix, Maria Jose Capella

Purpose: To describe a case of retinal vasculitis associated with tislelizumab, an anti-PD-1 monoclonal antibody, in a patient with esophageal squamous cell carcinoma (ESCC).

Methods: Case report.

Results: A 60-year-old man undergoing treatment with tislelizumab for ESCC was referred for ophthalmologic evaluation. The patient was asymptomatic, but fundoscopic examination revealed peripheral intraretinal hemorrhages in the right eye. Fluorescein angiography revealed bilateral retinal vasculitis that was successfully controlled with systemic corticosteroids without discontinuation of tislelizumab. Four months after prednisone cessation, the patient relapsed with bilateral vision loss, and optical coherence tomography demonstrated subfoveal fluid in both eyes. Oral prednisone was reintroduced, leading to resolution of the subfoveal fluid, though visual acuity did not fully recover.

Conclusions: To our knowledge, retinal vasculitis associated with tislelizumab has not been previously reported. This case highlights the need for close ophthalmologic monitoring in patients receiving immune checkpoint inhibitors, as ocular immune-related adverse events can develop or recur at any point during treatment and even be asymptomatic.

目的:描述一例食管鳞状细胞癌(ESCC)患者与抗pd -1单克隆抗体tislelizumab相关的视网膜血管炎。方法:病例报告。结果:一名60岁的男性接受tislelizumab治疗ESCC,被转介进行眼科评估。患者无症状,但眼底镜检查发现右眼视网膜内出血。荧光素血管造影显示双侧视网膜血管炎,通过全身皮质类固醇成功控制,没有停止tislelizumab。强的松停用4个月后,患者复发双侧视力丧失,光学相干断层扫描显示双眼中央凹下积液。再次引入口服强的松,导致中央凹下积液的溶解,但视力没有完全恢复。结论:据我们所知,与tislelizumab相关的视网膜血管炎以前没有报道过。该病例强调了接受免疫检查点抑制剂的患者需要密切的眼科监测,因为眼部免疫相关不良事件可能在治疗期间的任何时候发生或复发,甚至是无症状的。
{"title":"Retinal Vasculitis Associated with Tislelizumab in a Patient with Esophageal Squamous Cell Carcinoma.","authors":"Estel la Rojas-Pineda, Nuria Cintas, Anna Bruix, Maria Jose Capella","doi":"10.1097/ICB.0000000000001817","DOIUrl":"https://doi.org/10.1097/ICB.0000000000001817","url":null,"abstract":"<p><strong>Purpose: </strong>To describe a case of retinal vasculitis associated with tislelizumab, an anti-PD-1 monoclonal antibody, in a patient with esophageal squamous cell carcinoma (ESCC).</p><p><strong>Methods: </strong>Case report.</p><p><strong>Results: </strong>A 60-year-old man undergoing treatment with tislelizumab for ESCC was referred for ophthalmologic evaluation. The patient was asymptomatic, but fundoscopic examination revealed peripheral intraretinal hemorrhages in the right eye. Fluorescein angiography revealed bilateral retinal vasculitis that was successfully controlled with systemic corticosteroids without discontinuation of tislelizumab. Four months after prednisone cessation, the patient relapsed with bilateral vision loss, and optical coherence tomography demonstrated subfoveal fluid in both eyes. Oral prednisone was reintroduced, leading to resolution of the subfoveal fluid, though visual acuity did not fully recover.</p><p><strong>Conclusions: </strong>To our knowledge, retinal vasculitis associated with tislelizumab has not been previously reported. This case highlights the need for close ophthalmologic monitoring in patients receiving immune checkpoint inhibitors, as ocular immune-related adverse events can develop or recur at any point during treatment and even be asymptomatic.</p>","PeriodicalId":53580,"journal":{"name":"Retinal Cases and Brief Reports","volume":" ","pages":""},"PeriodicalIF":0.0,"publicationDate":"2025-09-23","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"145132397","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}
引用次数: 0
12-Year follow-up of a Benign Yellow Dot Maculopathy case 12-Year Follow-Up of BYDM. 1例良性黄斑病变12年随访
Q3 Medicine Pub Date : 2025-09-22 DOI: 10.1097/ICB.0000000000001810
Annamaria Nunziata, Sebastiano Del Fabbro, Alessio Antropoli, Lorenzo Bianco, Alessandro Arrigo, Francesco Bandello, Ahmad M Mansour, Maurizio Battaglia Parodi

Purpose: Benign Yellow Dot Maculopathy (BYDM) is a rare macular phenotype first described in 2017, characterized by yellow macular dots with preserved visual function. Although is considered a non-progressive condition, long-term follow-up data are limited. This case report presents a 12-year follow-up of a patient with BYDM, offering insights into its long-term stability over time.

Methods: The patient underwent comprehensive multimodal imaging, including optical coherence tomography (OCT), fundus autofluorescence (FAF), electro-oculography (EOG), and microperimetry. Genetic analysis via next-generation sequencing (NGS) was also performed to explore potential genetic associations.

Results: At baseline, FAF revealed characteristic hyperautofluorescent yellow dots in the macular and mid-peripheral retina, while OCT showed no structural abnormalities. EOG demonstrated subnormal responses, suggesting retinal pigment epithelium (RPE) dysfunction, and microperimetry indicated a global reduction in retinal sensitivity. Genetic testing identified two heterozygous variants of uncertain significance (ALMS1 and GPR179), but no pathogenic mutations associated with macular dystrophies. Over the 12-year follow-up, no progression of structural or functional abnormalities was observed, supporting the non-progressive nature of BYDM.

Conclusions: This case represents one of the longest follow-ups reported for BYDM, confirming its long-term stability. Despite subtle functional alterations, the absence of disease progression supports its benign nature. Further research is needed to refine the clinical spectrum of BYDM and distinguish it from other macular disorders.

目的:良性黄斑病变(BYDM)是一种罕见的黄斑表型,于2017年首次被描述,其特征是黄斑点保留视觉功能。虽然被认为是一种非进展性疾病,但长期随访数据有限。本病例报告对一名BYDM患者进行了12年的随访,为其长期稳定性提供了见解。方法:对患者进行光学相干断层扫描(OCT)、眼底自体荧光(FAF)、眼电成像(EOG)和显微视野检查等综合多模态影像学检查。通过下一代测序(NGS)进行遗传分析,探索潜在的遗传关联。结果:在基线时,FAF显示黄斑和中周视网膜的特征性高自荧光黄点,而OCT未显示结构异常。EOG显示亚正常反应,提示视网膜色素上皮(RPE)功能障碍,显微检查显示视网膜敏感性整体降低。基因检测发现了两个不确定意义的杂合变异(ALMS1和GPR179),但没有与黄斑营养不良相关的致病性突变。在12年的随访中,未观察到结构或功能异常的进展,支持BYDM的非进行性。结论:该病例是BYDM随访时间最长的病例之一,证实了其长期稳定性。尽管有细微的功能改变,但没有疾病进展支持其良性性质。需要进一步的研究来细化BYDM的临床谱,并将其与其他黄斑疾病区分开来。
{"title":"12-Year follow-up of a Benign Yellow Dot Maculopathy case 12-Year Follow-Up of BYDM.","authors":"Annamaria Nunziata, Sebastiano Del Fabbro, Alessio Antropoli, Lorenzo Bianco, Alessandro Arrigo, Francesco Bandello, Ahmad M Mansour, Maurizio Battaglia Parodi","doi":"10.1097/ICB.0000000000001810","DOIUrl":"https://doi.org/10.1097/ICB.0000000000001810","url":null,"abstract":"<p><strong>Purpose: </strong>Benign Yellow Dot Maculopathy (BYDM) is a rare macular phenotype first described in 2017, characterized by yellow macular dots with preserved visual function. Although is considered a non-progressive condition, long-term follow-up data are limited. This case report presents a 12-year follow-up of a patient with BYDM, offering insights into its long-term stability over time.</p><p><strong>Methods: </strong>The patient underwent comprehensive multimodal imaging, including optical coherence tomography (OCT), fundus autofluorescence (FAF), electro-oculography (EOG), and microperimetry. Genetic analysis via next-generation sequencing (NGS) was also performed to explore potential genetic associations.</p><p><strong>Results: </strong>At baseline, FAF revealed characteristic hyperautofluorescent yellow dots in the macular and mid-peripheral retina, while OCT showed no structural abnormalities. EOG demonstrated subnormal responses, suggesting retinal pigment epithelium (RPE) dysfunction, and microperimetry indicated a global reduction in retinal sensitivity. Genetic testing identified two heterozygous variants of uncertain significance (ALMS1 and GPR179), but no pathogenic mutations associated with macular dystrophies. Over the 12-year follow-up, no progression of structural or functional abnormalities was observed, supporting the non-progressive nature of BYDM.</p><p><strong>Conclusions: </strong>This case represents one of the longest follow-ups reported for BYDM, confirming its long-term stability. Despite subtle functional alterations, the absence of disease progression supports its benign nature. Further research is needed to refine the clinical spectrum of BYDM and distinguish it from other macular disorders.</p>","PeriodicalId":53580,"journal":{"name":"Retinal Cases and Brief Reports","volume":" ","pages":""},"PeriodicalIF":0.0,"publicationDate":"2025-09-22","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"145132389","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}
引用次数: 0
Vitreous metastasis of laryngeal carcinoma presenting as a white mass: A case report. 喉癌玻璃体转移表现为白色肿块1例。
Q3 Medicine Pub Date : 2025-09-22 DOI: 10.1097/ICB.0000000000001805
Takako Ohnishi, Mariko Horinaka, Akira Ohno, Tadayoshi Koda, Kenta Uto, Harumasa Yokota, Taiji Nagaoka, Satoru Yamagami

Purpose: We report a case of a vitreous opacity identified as a white inferiorly located vitreous mass. Vitreous surgical biopsy revealed that it was vitreous metastasis of laryngeal carcinoma.

Methods: A 56-year-old man on immune-chemotherapy for laryngeal carcinoma presented to our hospital with photopsia in his left eye (OS) for two weeks.

Results: Slit-lamp examination revealed 0.5+ cells in the anterior chamber and 0.5+ cells in the vitreous humor OS. The optic nerve papilla was normal in both eyes, although slight macular edema was present OS. A white mass formed by a vitreous opacity precipitated at the inferior of the left fundus. Due to ongoing systemic treatment, the patient was unable to make frequent visits to ophthalmology, and the second visit occurred one month after the initial examination. Fundus angiography revealed retinal vasculitis OS. Two months after the initial visit, tractional retinal detachment with retinal hemorrhage occurred. Therefore, a 25-gauge lens vitrectomy was performed immediately. Histopathological examination of the vitreous obtained intraoperatively revealed atypical squamous cells identified as a vitreous metastasis of laryngeal carcinoma.

Conclusions: Vitreous metastasis of laryngeal carcinoma had not been previously reported. On the initial examination, there were few findings other than a white friable mass presenting as a vitreous opacity precipitating at the inferior of the fundus. Vitreous opacity in patients with carcinoma should be considered a potential indicator of metastatic tumors.

目的:我们报告一例玻璃体混浊,确认为位于玻璃体下方的白色肿块。玻璃体手术活检显示为喉癌的玻璃体转移。方法:56岁喉癌免疫化疗患者左眼失光2周。结果:裂隙灯检查显示前房0.5+细胞,玻璃体内可见0.5+细胞。双眼视神经乳头正常,但有轻微黄斑水肿。左侧眼底下方由玻璃体混浊形成的白色肿块。由于正在进行全身治疗,患者无法频繁去眼科就诊,第二次就诊是在初次检查后一个月。眼底血管造影显示视网膜血管炎。初次就诊后2个月,出现牵引性视网膜脱离伴视网膜出血。因此,立即进行了25号晶状体切除术。术中玻璃体的组织病理学检查显示非典型鳞状细胞被确定为喉癌的玻璃体转移。结论:喉癌的玻璃体转移未见报道。在最初的检查中,除了在眼底下方沉淀的玻璃体混浊样的白色易碎肿块外,几乎没有其他发现。癌患者的玻璃体混浊应被视为转移性肿瘤的潜在指标。
{"title":"Vitreous metastasis of laryngeal carcinoma presenting as a white mass: A case report.","authors":"Takako Ohnishi, Mariko Horinaka, Akira Ohno, Tadayoshi Koda, Kenta Uto, Harumasa Yokota, Taiji Nagaoka, Satoru Yamagami","doi":"10.1097/ICB.0000000000001805","DOIUrl":"https://doi.org/10.1097/ICB.0000000000001805","url":null,"abstract":"<p><strong>Purpose: </strong>We report a case of a vitreous opacity identified as a white inferiorly located vitreous mass. Vitreous surgical biopsy revealed that it was vitreous metastasis of laryngeal carcinoma.</p><p><strong>Methods: </strong>A 56-year-old man on immune-chemotherapy for laryngeal carcinoma presented to our hospital with photopsia in his left eye (OS) for two weeks.</p><p><strong>Results: </strong>Slit-lamp examination revealed 0.5+ cells in the anterior chamber and 0.5+ cells in the vitreous humor OS. The optic nerve papilla was normal in both eyes, although slight macular edema was present OS. A white mass formed by a vitreous opacity precipitated at the inferior of the left fundus. Due to ongoing systemic treatment, the patient was unable to make frequent visits to ophthalmology, and the second visit occurred one month after the initial examination. Fundus angiography revealed retinal vasculitis OS. Two months after the initial visit, tractional retinal detachment with retinal hemorrhage occurred. Therefore, a 25-gauge lens vitrectomy was performed immediately. Histopathological examination of the vitreous obtained intraoperatively revealed atypical squamous cells identified as a vitreous metastasis of laryngeal carcinoma.</p><p><strong>Conclusions: </strong>Vitreous metastasis of laryngeal carcinoma had not been previously reported. On the initial examination, there were few findings other than a white friable mass presenting as a vitreous opacity precipitating at the inferior of the fundus. Vitreous opacity in patients with carcinoma should be considered a potential indicator of metastatic tumors.</p>","PeriodicalId":53580,"journal":{"name":"Retinal Cases and Brief Reports","volume":" ","pages":""},"PeriodicalIF":0.0,"publicationDate":"2025-09-22","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"145139298","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}
引用次数: 0
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Retinal Cases and Brief Reports
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