Pub Date : 2026-01-29eCollection Date: 2026-01-01DOI: 10.1159/000550668
Yiqing Gong, Xiang Cao, Yang Bai
Introduction: Thelazia callipaeda is a parasitic nematode that infects the eyes of humans and other mammals. This case highlights its incidental discovery during ocular surgery and underscores the importance of preoperative vigilance.
Case presentation: The parasite was identified intraoperatively in a 74-year-old female with a long-standing history of diabetes who was undergoing surgery for diabetic tractional retinal detachment in her right eye. She reported poor hygiene habits but no direct pet contact. The parasite, found within the conjunctival sac, was excised and confirmed by pathological examination. Postoperative care included antibiotic eye drops. At the 3-month follow-up, her best corrected visual acuity had improved to 6/120 with no recurrence.
Conclusion: This case emphasizes the critical role of comprehensive preoperative examination for the timely detection of ocular parasites to prevent complications. It also highlights the need for clinician awareness of Thelazia infection, particularly in high-risk individuals, and the importance of appropriate postoperative follow-up to optimize outcomes and minimize recurrence risk.
{"title":"Incidental Discovery of Conjunctival <i>Thelazia callipaeda</i> during Vitrectomy for Diabetic Tractional Retinal Detachment: A Case Report.","authors":"Yiqing Gong, Xiang Cao, Yang Bai","doi":"10.1159/000550668","DOIUrl":"https://doi.org/10.1159/000550668","url":null,"abstract":"<p><strong>Introduction: </strong><i>Thelazia callipaeda</i> is a parasitic nematode that infects the eyes of humans and other mammals. This case highlights its incidental discovery during ocular surgery and underscores the importance of preoperative vigilance.</p><p><strong>Case presentation: </strong>The parasite was identified intraoperatively in a 74-year-old female with a long-standing history of diabetes who was undergoing surgery for diabetic tractional retinal detachment in her right eye. She reported poor hygiene habits but no direct pet contact. The parasite, found within the conjunctival sac, was excised and confirmed by pathological examination. Postoperative care included antibiotic eye drops. At the 3-month follow-up, her best corrected visual acuity had improved to 6/120 with no recurrence.</p><p><strong>Conclusion: </strong>This case emphasizes the critical role of comprehensive preoperative examination for the timely detection of ocular parasites to prevent complications. It also highlights the need for clinician awareness of <i>Thelazia</i> infection, particularly in high-risk individuals, and the importance of appropriate postoperative follow-up to optimize outcomes and minimize recurrence risk.</p>","PeriodicalId":9635,"journal":{"name":"Case Reports in Ophthalmology","volume":"17 1","pages":"195-201"},"PeriodicalIF":0.6,"publicationDate":"2026-01-29","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12975153/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"147431101","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 : 2026-01-29eCollection Date: 2026-01-01DOI: 10.1159/000550669
Anita Sharma, Nikita Dash
Introduction: Ocular syphilis has atypical presentation in immunocompromised individuals. We report a case of intermediate uveitis as a delayed immune-mediated relapse following the successful treatment of syphilitic retinitis in an HIV-positive individual.
Case presentation: A 30-year-old HIV-positive man presented with decreased vision in his left eye. Examination revealed left eye granulomatous anterior uveitis with vitritis, retinal vasculitis, and optic disc edema. The patient was diagnosed with syphilitic uveitis, reactive for rapid plasma reagin (RPR), and initiated on intravenous penicillin by the internist. Topical corticosteroids were initiated, and oral corticosteroids were added after 1 week. Over 2 months, the lesions resolved clinically with decreasing RPR titers. After 2 months of complete remission and tapering of therapy, the patient presented with renewed disc edema, macular edema, vitreous cells, and snowballs suggestive of intermediate uveitis. The RPR titers showed a decreasing pattern.
Conclusion: The patient responded well to oral corticosteroids, indicating a likely immune-mediated relapse rather than reinfection. This case highlights the need for continued surveillance, even after clinical resolution.
{"title":"Relapsing Intermediate Uveitis following Resolved Syphilitic Retinitis in an HIV-Positive Patient: A Case Report.","authors":"Anita Sharma, Nikita Dash","doi":"10.1159/000550669","DOIUrl":"10.1159/000550669","url":null,"abstract":"<p><strong>Introduction: </strong>Ocular syphilis has atypical presentation in immunocompromised individuals. We report a case of intermediate uveitis as a delayed immune-mediated relapse following the successful treatment of syphilitic retinitis in an HIV-positive individual.</p><p><strong>Case presentation: </strong>A 30-year-old HIV-positive man presented with decreased vision in his left eye. Examination revealed left eye granulomatous anterior uveitis with vitritis, retinal vasculitis, and optic disc edema. The patient was diagnosed with syphilitic uveitis, reactive for rapid plasma reagin (RPR), and initiated on intravenous penicillin by the internist. Topical corticosteroids were initiated, and oral corticosteroids were added after 1 week. Over 2 months, the lesions resolved clinically with decreasing RPR titers. After 2 months of complete remission and tapering of therapy, the patient presented with renewed disc edema, macular edema, vitreous cells, and snowballs suggestive of intermediate uveitis. The RPR titers showed a decreasing pattern.</p><p><strong>Conclusion: </strong>The patient responded well to oral corticosteroids, indicating a likely immune-mediated relapse rather than reinfection. This case highlights the need for continued surveillance, even after clinical resolution.</p>","PeriodicalId":9635,"journal":{"name":"Case Reports in Ophthalmology","volume":"17 1","pages":"217-222"},"PeriodicalIF":0.6,"publicationDate":"2026-01-29","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12987672/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"147462667","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 : 2026-01-29eCollection Date: 2026-01-01DOI: 10.1159/000550754
Omer Othman Abdullah
Introduction: Simultaneous branch retinal artery occlusion (BRAO) and central retinal vein occlusion (CRVO) is an exceptionally rare cause of acute vision loss, particularly in young individuals without systemic vascular risk factors. Blood transfusion has been implicated in rare thromboembolic and hyperviscosity-related complications but is infrequently associated with retinal vascular occlusions.
Case presentation: A 20-year-old man presented with sudden painless unilateral vision loss shortly after receiving a packed red blood cell transfusion for severe anemia. Ophthalmic examination revealed optic disc edema, venous engorgement, scattered retinal hemorrhages, and superior hemi-macular whitening in the affected eye. Structural optical coherence tomography demonstrated inner retinal hyperreflectivity and subsequent thinning, consistent with ischemic retinal injury. Fluorescein angiography was contraindicated due to an acute hypersensitivity reaction. Extensive systemic, hematologic, inflammatory, and thrombophilia workup was unremarkable. Despite conservative and supportive management, including hyperbaric oxygen therapy and anticoagulation, visual recovery remained limited at follow-up.
Conclusion: This case highlights a rare presentation of concurrent BRAO and CRVO occurring shortly after blood transfusion in a young patient without identifiable systemic risk factors. Although causality cannot be definitively established, the close temporal relationship and exclusion of alternative etiologies suggest transfusion-related rheologic changes as a plausible contributing factor. Clinicians should remain vigilant for acute retinal vascular events following transfusion, even in young patients, and prompt ophthalmic evaluation is essential.
{"title":"Simultaneous Branch Retinal Artery and Central Retinal Vein Occlusions following Blood Transfusion in a Young Man: A Case Report.","authors":"Omer Othman Abdullah","doi":"10.1159/000550754","DOIUrl":"10.1159/000550754","url":null,"abstract":"<p><strong>Introduction: </strong>Simultaneous branch retinal artery occlusion (BRAO) and central retinal vein occlusion (CRVO) is an exceptionally rare cause of acute vision loss, particularly in young individuals without systemic vascular risk factors. Blood transfusion has been implicated in rare thromboembolic and hyperviscosity-related complications but is infrequently associated with retinal vascular occlusions.</p><p><strong>Case presentation: </strong>A 20-year-old man presented with sudden painless unilateral vision loss shortly after receiving a packed red blood cell transfusion for severe anemia. Ophthalmic examination revealed optic disc edema, venous engorgement, scattered retinal hemorrhages, and superior hemi-macular whitening in the affected eye. Structural optical coherence tomography demonstrated inner retinal hyperreflectivity and subsequent thinning, consistent with ischemic retinal injury. Fluorescein angiography was contraindicated due to an acute hypersensitivity reaction. Extensive systemic, hematologic, inflammatory, and thrombophilia workup was unremarkable. Despite conservative and supportive management, including hyperbaric oxygen therapy and anticoagulation, visual recovery remained limited at follow-up.</p><p><strong>Conclusion: </strong>This case highlights a rare presentation of concurrent BRAO and CRVO occurring shortly after blood transfusion in a young patient without identifiable systemic risk factors. Although causality cannot be definitively established, the close temporal relationship and exclusion of alternative etiologies suggest transfusion-related rheologic changes as a plausible contributing factor. Clinicians should remain vigilant for acute retinal vascular events following transfusion, even in young patients, and prompt ophthalmic evaluation is essential.</p>","PeriodicalId":9635,"journal":{"name":"Case Reports in Ophthalmology","volume":"17 1","pages":"211-216"},"PeriodicalIF":0.6,"publicationDate":"2026-01-29","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12981899/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"147462673","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 : 2026-01-22eCollection Date: 2026-01-01DOI: 10.1159/000549601
Rita Teixeira-Martins, Margarida Ribeiro, Carla Sofia Ferreira, Paulo Rocha, Pedro Louro, Amândio Rocha-Sousa, Sérgio Estrela-Silva
Introduction: Retinitis pigmentosa (RP) is a genetically and phenotypically heterogeneous inherited type of retinal dystrophy in which the RHO gene is frequently implicated. The missense variant NM_000539.3:c.545G>A, p.(Gly182Asp), has been reported in genetic databases as pathogenic, and a recent large cohort identified a single patient carrying this variant with sector RP. However, no detailed intrafamilial phenotypic characterization has been described. This study provides the first such characterization in a Portuguese family.
Case presentation: Four individuals across three generations underwent multimodal ophthalmologic evaluations, including visual acuity, fundus imaging (OCT, FAF), visual field testing, and electrophysiology. Three patients underwent genetic testing, and all carried the heterozygous RHO p.(Gly182Asp) variant. The clinical manifestations ranged from typical RP in three members to sector RP in one, with preferential inferior and nasal retinal involvement. Structural and functional severity increased with age, and all presented with nyctalopia. The oldest patient exhibited advanced degeneration with minimal central vision. This intrafamily phenotypic variability underscores the differential expression of the same genotype.
Conclusion: This is the first report linking the RHO p.(Gly182Asp) variant with both typical and sector RP, highlighting intrafamilial heterogeneity. Detailed phenotyping is crucial for genetic counseling and potential inclusion in future gene-based therapies.
{"title":"A Rare RHO Variant and Its Phenotypic Spectrum in a Portuguese Family with Retinitis Pigmentosa: A Case Series.","authors":"Rita Teixeira-Martins, Margarida Ribeiro, Carla Sofia Ferreira, Paulo Rocha, Pedro Louro, Amândio Rocha-Sousa, Sérgio Estrela-Silva","doi":"10.1159/000549601","DOIUrl":"https://doi.org/10.1159/000549601","url":null,"abstract":"<p><strong>Introduction: </strong>Retinitis pigmentosa (RP) is a genetically and phenotypically heterogeneous inherited type of retinal dystrophy in which the <i>RHO</i> gene is frequently implicated. The missense variant NM_000539.3:c.545G>A, p.(Gly182Asp), has been reported in genetic databases as pathogenic, and a recent large cohort identified a single patient carrying this variant with sector RP. However, no detailed intrafamilial phenotypic characterization has been described. This study provides the first such characterization in a Portuguese family.</p><p><strong>Case presentation: </strong>Four individuals across three generations underwent multimodal ophthalmologic evaluations, including visual acuity, fundus imaging (OCT, FAF), visual field testing, and electrophysiology. Three patients underwent genetic testing, and all carried the heterozygous RHO p.(Gly182Asp) variant. The clinical manifestations ranged from typical RP in three members to sector RP in one, with preferential inferior and nasal retinal involvement. Structural and functional severity increased with age, and all presented with nyctalopia. The oldest patient exhibited advanced degeneration with minimal central vision. This intrafamily phenotypic variability underscores the differential expression of the same genotype.</p><p><strong>Conclusion: </strong>This is the first report linking the RHO p.(Gly182Asp) variant with both typical and sector RP, highlighting intrafamilial heterogeneity. Detailed phenotyping is crucial for genetic counseling and potential inclusion in future gene-based therapies.</p>","PeriodicalId":9635,"journal":{"name":"Case Reports in Ophthalmology","volume":"17 1","pages":"171-178"},"PeriodicalIF":0.6,"publicationDate":"2026-01-22","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12959910/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"147364016","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: Reticular pseudodrusen (RPD) are an important clinical biomarker associated with progression to late age-related macular degeneration (AMD). We report polarization-sensitive optical coherence tomography (PS-OCT) findings in a case of RPD and evaluate the entropy characteristics of these lesions.
Case presentation: A 73-year-old man presented for evaluation of retinal angiomatous proliferation in his right eye. The left eye demonstrated multiple round, white-yellowish macular lesions consistent with RPD. Optical coherence tomography (OCT) angiography showed no macular neovascularization in the left eye. Swept-source (SS-OCT) revealed hyperreflective, spike-like deposits between the photoreceptor outer segments (PROSs) and the retinal pigment epithelium. Corresponding PS-OCT B-scans demonstrated moderately elevated polarimetric entropy values at the same locations. En face images reconstructed from the PROS layer showed circular RPD-like lesions on SS-OCT, which precisely matched high-entropy areas on PS-OCT. Merged en face maps confirmed one-to-one correspondence between RPD and regions of increased entropy.
Conclusion: PS-OCT successfully visualized RPD as moderately high-entropy lesions in the PROS layer, suggesting that these deposits are composed of non-retinal pigment epithelium material, such as lipids or inflammatory cells. These findings indicate that PS-OCT may serve as a useful complementary modality for detecting RPD and may enhance current imaging strategies for assessing RPD and monitoring AMD progression.
{"title":"Evaluation of Reticular Pseudodrusen by Polarization-Sensitive Optical Coherence Tomography: Case Report.","authors":"Ryo Terao, Chihiro Tsusu, Yuki Saeki, Kazuki Yashiro, Mami Ota, Shuichiro Aoki, Kohdai Kitamoto, Keiko Azuma","doi":"10.1159/000550439","DOIUrl":"https://doi.org/10.1159/000550439","url":null,"abstract":"<p><strong>Introduction: </strong>Reticular pseudodrusen (RPD) are an important clinical biomarker associated with progression to late age-related macular degeneration (AMD). We report polarization-sensitive optical coherence tomography (PS-OCT) findings in a case of RPD and evaluate the entropy characteristics of these lesions.</p><p><strong>Case presentation: </strong>A 73-year-old man presented for evaluation of retinal angiomatous proliferation in his right eye. The left eye demonstrated multiple round, white-yellowish macular lesions consistent with RPD. Optical coherence tomography (OCT) angiography showed no macular neovascularization in the left eye. Swept-source (SS-OCT) revealed hyperreflective, spike-like deposits between the photoreceptor outer segments (PROSs) and the retinal pigment epithelium. Corresponding PS-OCT B-scans demonstrated moderately elevated polarimetric entropy values at the same locations. <i>En face</i> images reconstructed from the PROS layer showed circular RPD-like lesions on SS-OCT, which precisely matched high-entropy areas on PS-OCT. Merged <i>en face</i> maps confirmed one-to-one correspondence between RPD and regions of increased entropy.</p><p><strong>Conclusion: </strong>PS-OCT successfully visualized RPD as moderately high-entropy lesions in the PROS layer, suggesting that these deposits are composed of non-retinal pigment epithelium material, such as lipids or inflammatory cells. These findings indicate that PS-OCT may serve as a useful complementary modality for detecting RPD and may enhance current imaging strategies for assessing RPD and monitoring AMD progression.</p>","PeriodicalId":9635,"journal":{"name":"Case Reports in Ophthalmology","volume":"17 1","pages":"171-178"},"PeriodicalIF":0.6,"publicationDate":"2026-01-22","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12956322/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"147354016","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 : 2026-01-19eCollection Date: 2026-01-01DOI: 10.1159/000549981
Ekaterina Sokolenko, Migle Lindziute, Aleksandr Martynov, Henrike Westekemper, Anke Manthey, Sami Dalbah, Nikolaos E Bechrakis, Carsten Framme, Jan Tode
Introduction: This case highlights the complexity of managing highly refractory, recurrent pterygium. Despite numerous prior surgeries by experienced anterior segment surgeons, the patient developed persistent fibrosis, symblepharon formation, and functional limitations. This underscores the need for a tailored, multidisciplinary approach, a strategy not widely documented in current literature.
Case presentation: Over 6 years, the patient underwent ten previous interventions, including conjunctivoplasty with ipsilateral free conjunctival flap and mitomycin C (MMC), amniotic membrane transplantation, symblepharolysis, and tenonplasty. The most recent surgery had been performed 6 months before referral to our clinic. Despite these attempts, each procedure was followed by significant fibrosis, scarring, and recurrent symblepharon formation involving the fornices. Following comprehensive multidisciplinary discussion and literature review, reconstructive surgery was undertaken. This included conjunctivoplasty with a free limbal-conjunctival autograft from the contralateral eye, adhesiolysis of the extraocular muscles, muscle belly plasty with amniotic membrane grafting, and placement of barrier sutures to reduce adhesion recurrence. During 12 months of follow-up, the patient exhibited notable clinical improvement, including enhanced ocular motility, resolution of diplopia in primary gaze, and improved visual acuity.
Conclusion: This case emphasizes the importance of individualized, innovative surgical planning for advanced recurrent pterygium. A long-term, multidisciplinary strategy can offer sustained anatomical and functional restoration in patients with extensive prior surgical failure and severe ocular surface disease.
{"title":"An Individualized Surgical Approach for the Management of Recurrent Pterygium: A Case Report.","authors":"Ekaterina Sokolenko, Migle Lindziute, Aleksandr Martynov, Henrike Westekemper, Anke Manthey, Sami Dalbah, Nikolaos E Bechrakis, Carsten Framme, Jan Tode","doi":"10.1159/000549981","DOIUrl":"10.1159/000549981","url":null,"abstract":"<p><strong>Introduction: </strong>This case highlights the complexity of managing highly refractory, recurrent pterygium. Despite numerous prior surgeries by experienced anterior segment surgeons, the patient developed persistent fibrosis, symblepharon formation, and functional limitations. This underscores the need for a tailored, multidisciplinary approach, a strategy not widely documented in current literature.</p><p><strong>Case presentation: </strong>Over 6 years, the patient underwent ten previous interventions, including conjunctivoplasty with ipsilateral free conjunctival flap and mitomycin C (MMC), amniotic membrane transplantation, symblepharolysis, and tenonplasty. The most recent surgery had been performed 6 months before referral to our clinic. Despite these attempts, each procedure was followed by significant fibrosis, scarring, and recurrent symblepharon formation involving the fornices. Following comprehensive multidisciplinary discussion and literature review, reconstructive surgery was undertaken. This included conjunctivoplasty with a free limbal-conjunctival autograft from the contralateral eye, adhesiolysis of the extraocular muscles, muscle belly plasty with amniotic membrane grafting, and placement of barrier sutures to reduce adhesion recurrence. During 12 months of follow-up, the patient exhibited notable clinical improvement, including enhanced ocular motility, resolution of diplopia in primary gaze, and improved visual acuity.</p><p><strong>Conclusion: </strong>This case emphasizes the importance of individualized, innovative surgical planning for advanced recurrent pterygium. A long-term, multidisciplinary strategy can offer sustained anatomical and functional restoration in patients with extensive prior surgical failure and severe ocular surface disease.</p>","PeriodicalId":9635,"journal":{"name":"Case Reports in Ophthalmology","volume":"17 1","pages":"162-170"},"PeriodicalIF":0.6,"publicationDate":"2026-01-19","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12935456/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"147302701","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 : 2026-01-19eCollection Date: 2026-01-01DOI: 10.1159/000550138
Luke Farson, Marina Shenouda, Agni Kakouri, Ashkan Kashanchi, Clark Sitton, Timothy McCulley, Ying Chen
Introduction: Sickle cell disease is associated with painful vaso-occlusive crises, bone infarction, avascular necrosis, and osteomyelitis. Sickle cell orbitopathy is a rare manifestation with signs similar to periorbital cellulitis and has subtle radiologic features.
Case presentation: We report a case of a five-year-old girl with sickle cell who presented with bilateral orbital infarctions and subperiosteal hematomas, notably without periorbital pain, and was treated with antibiotics and steroids.
Conclusion: Physicians should be aware of this rare manifestation, especially in the pediatric population, as it can affect subsequent management.
{"title":"Sickle Cell Crisis-Induced Painless Bilateral Orbital Bone Infarction: A Case Report.","authors":"Luke Farson, Marina Shenouda, Agni Kakouri, Ashkan Kashanchi, Clark Sitton, Timothy McCulley, Ying Chen","doi":"10.1159/000550138","DOIUrl":"10.1159/000550138","url":null,"abstract":"<p><strong>Introduction: </strong>Sickle cell disease is associated with painful vaso-occlusive crises, bone infarction, avascular necrosis, and osteomyelitis. Sickle cell orbitopathy is a rare manifestation with signs similar to periorbital cellulitis and has subtle radiologic features.</p><p><strong>Case presentation: </strong>We report a case of a five-year-old girl with sickle cell who presented with bilateral orbital infarctions and subperiosteal hematomas, notably without periorbital pain, and was treated with antibiotics and steroids.</p><p><strong>Conclusion: </strong>Physicians should be aware of this rare manifestation, especially in the pediatric population, as it can affect subsequent management.</p>","PeriodicalId":9635,"journal":{"name":"Case Reports in Ophthalmology","volume":"17 1","pages":"156-161"},"PeriodicalIF":0.6,"publicationDate":"2026-01-19","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12904658/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"146200200","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 : 2026-01-19eCollection Date: 2026-01-01DOI: 10.1159/000548878
Daniel M Markowitz, Rebecca F Silverman, Lauren Colwell, Turner D Wibbelsman, Danijel Josip Pericic, Shruthi Harish, Sarah Thornton
Introduction: We report a rare case of bilateral optic disc edema in a patient found to have severe vitamin A deficiency. While vitamin A deficiency typically presents with optic disc atrophy, this case emphasizes the importance of considering vitamin A deficiency in patients with atypical optic disc edema.
Case presentation: A 39-year-old female with a history of disordered eating habits and extensive psychiatric history presented to the emergency department with acute-onset cloudiness and blurring of her vision bilaterally. Physical exam was notable for bilateral optic disc edema in the absence of increased intracranial pressure, as demonstrated by an unremarkable MRI and lumbar puncture opening pressure of 18 cm H2O. Extensive testing on serum and cerebrospinal fluid was notable only for a serum vitamin A level of less than 5 μg/dL (normal: 38-98 μg/dL). The patient underwent inpatient hospitalization and was given 100,000 units of intramuscular vitamin A for 3 days with 50,000 units per day for the following 2 weeks. A follow-up visit 1 month later revealed resolution of her optic disc edema in the right eye and only mild optic disc edema in the left eye.
Conclusion: We describe a case of bilateral optic disc edema in a patient with severe vitamin A deficiency, underscoring the potential for nutritional abnormalities to present in this fashion. We also emphasize a multidisciplinary approach to unveil the underlying causes of atypical presentations of optic disc edema and create an appropriate treatment plan for patients with nutritional deficiencies.
简介:我们报告一例罕见的双侧视盘水肿患者发现有严重的维生素a缺乏症。虽然维生素A缺乏通常表现为视盘萎缩,但本病例强调了在非典型视盘水肿患者中考虑维生素A缺乏的重要性。病例介绍:一名39岁女性,有饮食习惯紊乱史和广泛的精神病史,以双侧视力模糊和急性起病就诊于急诊科。体格检查显示,在颅内压未升高的情况下,双侧视盘水肿明显,MRI显示不明显,腰椎穿刺开口压力为18 cm H2O。对血清和脑脊液进行广泛检测,发现血清维生素a水平低于5 μg/dL(正常:38-98 μg/dL)。患者住院治疗,连续3天肌肉注射维生素A 10万单位,连续2周每天5万单位。1个月后的随访显示右眼视盘水肿消退,左眼仅轻度视盘水肿。结论:我们描述了一个严重维生素a缺乏症患者双侧视盘水肿的病例,强调了以这种方式出现营养异常的可能性。我们还强调多学科的方法来揭示视盘水肿的不典型表现的根本原因,并为营养缺乏的患者制定适当的治疗计划。
{"title":"Bilateral Optic Disc Edema Secondary to Severe Vitamin A Deficiency: A Rare Cause of Acute Vision Loss.","authors":"Daniel M Markowitz, Rebecca F Silverman, Lauren Colwell, Turner D Wibbelsman, Danijel Josip Pericic, Shruthi Harish, Sarah Thornton","doi":"10.1159/000548878","DOIUrl":"https://doi.org/10.1159/000548878","url":null,"abstract":"<p><strong>Introduction: </strong>We report a rare case of bilateral optic disc edema in a patient found to have severe vitamin A deficiency. While vitamin A deficiency typically presents with optic disc atrophy, this case emphasizes the importance of considering vitamin A deficiency in patients with atypical optic disc edema.</p><p><strong>Case presentation: </strong>A 39-year-old female with a history of disordered eating habits and extensive psychiatric history presented to the emergency department with acute-onset cloudiness and blurring of her vision bilaterally. Physical exam was notable for bilateral optic disc edema in the absence of increased intracranial pressure, as demonstrated by an unremarkable MRI and lumbar puncture opening pressure of 18 cm H<sub>2</sub>O. Extensive testing on serum and cerebrospinal fluid was notable only for a serum vitamin A level of less than 5 μg/dL (normal: 38-98 μg/dL). The patient underwent inpatient hospitalization and was given 100,000 units of intramuscular vitamin A for 3 days with 50,000 units per day for the following 2 weeks. A follow-up visit 1 month later revealed resolution of her optic disc edema in the right eye and only mild optic disc edema in the left eye.</p><p><strong>Conclusion: </strong>We describe a case of bilateral optic disc edema in a patient with severe vitamin A deficiency, underscoring the potential for nutritional abnormalities to present in this fashion. We also emphasize a multidisciplinary approach to unveil the underlying causes of atypical presentations of optic disc edema and create an appropriate treatment plan for patients with nutritional deficiencies.</p>","PeriodicalId":9635,"journal":{"name":"Case Reports in Ophthalmology","volume":"17 1","pages":"263-274"},"PeriodicalIF":0.6,"publicationDate":"2026-01-19","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC13008406/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"147509825","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 : 2026-01-15eCollection Date: 2026-01-01DOI: 10.1159/000550515
Niv Levi, Alexandre Boutet, Edward Margolin
Introduction: We describe a man with severe unilateral visual loss from giant cell arteritis who had normal inflammatory markers and exhibited progression of intracranial vasculitis while on high doses of corticosteroids.
Case: A 64-year-old man presented with unilateral visual loss. He had systemic symptoms of giant cell arteritis (GCA); however, both inflammatory markers were normal. He was diagnosed with arteritic anterior ischemic optic neuropathy after temporal artery biopsy revealed classic findings of GCA and treated with high-dose corticosteroids. Despite appropriate treatment, serial imaging revealed progressive narrowing of intracranial arteries, predominantly involving anterior circulation of the brain.
Conclusion: This case highlights that GCA can rarely present with normal inflammatory markers, involve intracranial vasculature, and progress despite treatment with high doses of corticosteroids emphasizing the importance of advanced imaging and potential need for adjunctive immunosuppressive therapy in patients with refractory disease.
{"title":"Intracranial Giant Cell Arteritis Progressing despite Treatment with High Doses of Corticosteroids: Case Report.","authors":"Niv Levi, Alexandre Boutet, Edward Margolin","doi":"10.1159/000550515","DOIUrl":"https://doi.org/10.1159/000550515","url":null,"abstract":"<p><strong>Introduction: </strong>We describe a man with severe unilateral visual loss from giant cell arteritis who had normal inflammatory markers and exhibited progression of intracranial vasculitis while on high doses of corticosteroids.</p><p><strong>Case: </strong>A 64-year-old man presented with unilateral visual loss. He had systemic symptoms of giant cell arteritis (GCA); however, both inflammatory markers were normal. He was diagnosed with arteritic anterior ischemic optic neuropathy after temporal artery biopsy revealed classic findings of GCA and treated with high-dose corticosteroids. Despite appropriate treatment, serial imaging revealed progressive narrowing of intracranial arteries, predominantly involving anterior circulation of the brain.</p><p><strong>Conclusion: </strong>This case highlights that GCA can rarely present with normal inflammatory markers, involve intracranial vasculature, and progress despite treatment with high doses of corticosteroids emphasizing the importance of advanced imaging and potential need for adjunctive immunosuppressive therapy in patients with refractory disease.</p>","PeriodicalId":9635,"journal":{"name":"Case Reports in Ophthalmology","volume":"17 1","pages":"202-210"},"PeriodicalIF":0.6,"publicationDate":"2026-01-15","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12975152/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"147430316","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}