Case Reports in Ophthalmology最新文献

{"title":"Gasperini Syndrome: A Rare Brainstem Syndrome - A Case Report.","authors":"Bianca Sarah Stenitzer, Daniela Fruehwirth-Kaspar, Martina Brandner","doi":"10.1159/000551798","DOIUrl":"https://doi.org/10.1159/000551798","url":null,"abstract":"<p><strong>Introduction: </strong>This unique case demonstrates how subtle ocular motor abnormalities, even in the absence of visual complaints, can lead to the detection of an otherwise clinically silent brainstem lesion.</p><p><strong>Case presentation: </strong>We report on the incidental discovery of a brain tumor following a traffic accident. Despite the absence of ocular symptoms, abnormal oculomotor findings led to further diagnostic investigations and rapid diagnosis of a pontine lesion consistent with Gasparini syndrome.</p><p><strong>Conclusion: </strong>Our case report highlights the crucial importance of meticulous history-taking, precise neuro-ophthalmological examination, particularly the assessment of ocular motility and interdisciplinary management in patients with complex brainstem syndromes.</p>","PeriodicalId":9635,"journal":{"name":"Case Reports in Ophthalmology","volume":"17 1","pages":"402-407"},"PeriodicalIF":0.6,"publicationDate":"2026-04-03","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC13143271/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"147834186","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}

{"title":"Erratum.","authors":"","doi":"10.1159/000551159","DOIUrl":"10.1159/000551159","url":null,"abstract":"<p><p>[This corrects the article DOI: 10.1159/000550116.].</p>","PeriodicalId":9635,"journal":{"name":"Case Reports in Ophthalmology","volume":"17 1","pages":"238"},"PeriodicalIF":0.6,"publicationDate":"2026-03-19","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC13002235/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"147497713","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}

{"title":"Erratum.","authors":"","doi":"10.1159/000551160","DOIUrl":"https://doi.org/10.1159/000551160","url":null,"abstract":"<p><p>[This corrects the article DOI: 10.1159/000550003.].</p>","PeriodicalId":9635,"journal":{"name":"Case Reports in Ophthalmology","volume":"17 1","pages":"237"},"PeriodicalIF":0.6,"publicationDate":"2026-03-18","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12999192/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"147484865","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}

{"title":"Acute Retinal Necrosis with Choroidal Inflammation, Optic Perineuritis, and Early Retinal Detachment: A Case Report.","authors":"Sumayah H Banaji, Nora A Alyousif, Wael A Alsakran, Mustafa S Alhasan, Hanan A Alshalan","doi":"10.1159/000550708","DOIUrl":"https://doi.org/10.1159/000550708","url":null,"abstract":"<p><strong>Introduction: </strong>The aim of the study was to describe a rare presentation of acute retinal necrosis (ARN) in an immunocompetent adult, with choroidal, optic nerve, and orbital inflammation with exudative retinal detachment (ERD), complicated by early rhegmatogenous retinal detachment (RRD). This is a retrospective case report of atypical ARN. Clinical data, laboratory testing, and multimodal imaging were reviewed.</p><p><strong>Case presentation: </strong>Examination revealed right eye granulomatous panuveitis, including granulomatous anterior chamber inflammation, dense vitreous haze, and peripheral necrotizing hemorrhagic retinitis. Optical coherence tomography showed inner retinal hyperreflectivity, thickened choroid, and ERD. Magnetic resonance imaging confirmed inflammation of the optic nerve sheath and orbital tissues. Polymerase chain reaction (PCR) confirmed varicella-zoster virus infection. Left eye showed an old peripheral chorioretinal scar. The patient received systemic acyclovir, oral trimethoprim-sulfamethoxazole, along with systemic corticosteroids and intravitreal ganciclovir. Final resolution of ERD and improvement in orbital and intraocular inflammation were observed. The patient subsequently developed early RRD, managed by pars plana vitrectomy and silicone oil tamponade. Despite structural stabilization, visual recovery was limited due to late presentation, optic nerve, and choroidal involvement. Systemic investigations excluded alternative causes.</p><p><strong>Conclusion: </strong>ARN with concurrent panocular inflammation, choroidal, and optic nerve involvement is uncommon and may indicate a more aggressive disease course, often associated with poor visual prognosis. This case highlights that ARN with choroidal and optic nerve involvement may follow an aggressive course, requiring early PCR confirmation, intensive antiviral therapy, and timely surgical intervention to prevent severe visual loss.</p>","PeriodicalId":9635,"journal":{"name":"Case Reports in Ophthalmology","volume":"17 1","pages":"318-326"},"PeriodicalIF":0.6,"publicationDate":"2026-03-09","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC13068389/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"147670524","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}

{"title":"Atypical Cogan's Syndrome Presenting as Anterior Scleritis: Case Report.","authors":"João Mendes, Francisco Mendes, Diogo Valente Fortunato, João Vasco Garrido, Rita Condesso, Augusto Candeias","doi":"10.1159/000551227","DOIUrl":"https://doi.org/10.1159/000551227","url":null,"abstract":"&lt;p&gt;&lt;strong&gt;Introduction: &lt;/strong&gt;Cogan's syndrome is a rare autoimmune vasculitis characterized by ocular inflammation and audiovestibular dysfunction, with the atypical form presenting with inflammatory ocular disease other than interstitial keratitis. Typical Cogan's syndrome manifests with interstitial keratitis and audiovestibular symptoms occurring within a 2-year interval, whereas atypical Cogan's syndrome involves inflammatory ocular disease without interstitial keratitis (such as scleritis, episcleritis, uveitis, or choroiditis), audiovestibular impairment, and systemic manifestations with delays longer than 2 years between different organ involvements. Atypical Cogan's syndrome usually presents with scleritis and choroiditis and is more frequently associated with systemic inflammation compared to typical forms, with approximately 70% of patients developing systemic manifestations. The diagnosis remains one of exclusion, requiring meticulous investigation to rule out infectious and other autoimmune etiologies, as no specific diagnostic criteria or biomarkers exist.&lt;/p&gt;&lt;p&gt;&lt;strong&gt;Case presentation: &lt;/strong&gt;A 40-year-old White female presented with recurrent episodes of ocular inflammation and sensorineural hearing loss over a 10-year period, initially treated as isolated conditions. She was evaluated for systemic autoimmune and infectious causes, all of which were excluded through serologic and imaging studies. Anterior scleritis was confirmed on ophthalmic examination with incomplete blanching on topical phenylephrine testing. Audiometry revealed bilateral sensorineural hearing loss. A diagnosis of atypical Cogan's syndrome was made based on clinical history and exclusion of other etiologies. High-dose oral corticosteroids (prednisolone 40 mg daily) led to initial improvement. However, the patient experienced disease flares following intolerance or inadequate response to several steroid-sparing agents, including methotrexate (discontinued due to alopecia after 3 months), azathioprine (150 mg daily for 6 months), and mycophenolate mofetil (3,000 mg daily for 6 months). A subconjunctival triamcinolone injection (40 mg) was administered during a relapse, and adalimumab (40 mg subcutaneously every 2 weeks) was subsequently initiated, resulting in clinical stability with no further ocular or auditory deterioration at 12-month follow-up. This therapeutic escalation represents an evidence-based approach for refractory disease, as infliximab has been shown to be the only significant predictor of vestibulo-auditory improvement in Cogan's syndrome, with other TNF-α antagonists including adalimumab demonstrating efficacy in case reports.&lt;/p&gt;&lt;p&gt;&lt;strong&gt;Conclusion: &lt;/strong&gt;Cogan's syndrome is a rare autoimmune vasculitis that can present with various ocular and auditory manifestations. While interstitial keratitis is classically described, anterior scleritis represents a less common but recognized presentation. This case underscores the importance ","PeriodicalId":9635,"journal":{"name":"Case Reports in Ophthalmology","volume":"17 1","pages":"371-379"},"PeriodicalIF":0.6,"publicationDate":"2026-03-09","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC13105469/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"147763499","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}

{"title":"Erratum.","authors":"","doi":"10.1159/000550782","DOIUrl":"10.1159/000550782","url":null,"abstract":"<p><p>[This corrects the article DOI: 10.1159/000549864.].</p>","PeriodicalId":9635,"journal":{"name":"Case Reports in Ophthalmology","volume":"17 1","pages":"187"},"PeriodicalIF":0.6,"publicationDate":"2026-03-06","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12965735/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"147376165","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}

{"title":"Benign Yellow Dot Maculopathy in a Palestinian Child: A Case Report.","authors":"Yahya Alswaiti, Othman Mustafa","doi":"10.1159/000551354","DOIUrl":"https://doi.org/10.1159/000551354","url":null,"abstract":"<p><strong>Introduction: </strong>Benign yellow dot maculopathy (BYDM) is a recently recognized macular phenotype, first described in 2017. It may occur sporadically or follow a familial pattern. The condition is characterized by a rare, asymptomatic, early-onset, nonprogressive maculopathy, with multiple yellow dots at the macula and minimal or no functional impairment. Owing to its benign and frequently asymptomatic course, this condition is likely underdiagnosed and underreported.</p><p><strong>Case presentation: </strong>We report a 6-year-old Palestinian patient who was incidentally found to have bilateral, discrete yellow dots confined to the macular region, associated with reduced visual acuity. A comprehensive ophthalmic evaluation was performed. Multimodal imaging, including optical coherence tomography, demonstrated preserved retinal architecture, while electrophysiological testing showed normal global retinal function, supporting a benign, nonprogressive phenotype. Following careful exclusion of other inherited and acquired macular disorders, a diagnosis of BYDM was established.</p><p><strong>Conclusion: </strong>This report documents a case of BYDM in a Palestinian patient and contributes to the limited existing literature on this rare macular phenotype. To our knowledge, this is the first reported case from a Palestinian patient. Recognition of this entity is important to avoid misdiagnosis, unnecessary investigations, and patient anxiety.</p>","PeriodicalId":9635,"journal":{"name":"Case Reports in Ophthalmology","volume":"17 1","pages":"408-414"},"PeriodicalIF":0.6,"publicationDate":"2026-03-05","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC13143270/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"147834204","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}

{"title":"Acetazolamide-Induced Bilateral Uveal Effusions and Secondary Angle Closure: A Case Report.","authors":"Kaela Braue, Keith Martin, Ortal Fogel Tempelhof","doi":"10.1159/000551289","DOIUrl":"https://doi.org/10.1159/000551289","url":null,"abstract":"<p><strong>Introduction: </strong>Acetazolamide-induced uveal effusion and secondary angle closure is a rarely reported but clinically significant phenomenon. The reaction is idiosyncratic and independent of dosage or prior exposure.</p><p><strong>Case presentation: </strong>A 52-year-old patient developed severe bilateral uveal effusions, acute angle closure, and myopic shift 24 h after commencing acetazolamide for altitude sickness prophylaxis. The case demonstrates unusually severe posterior involvement after a single prophylactic dose, with dramatic recovery following initiation of intravenous methylprednisolone.</p><p><strong>Conclusion: </strong>Given the widespread use of acetazolamide in ophthalmology and other medical disciplines, clinicians must be aware of potentially sight-threatening drug reactions. Prescription of acetazolamide for altitude sickness prophylaxis represents a unique risk, as patients are likely to be travelling to remote destinations. First-time dosing should occur where urgent medical assessment is readily accessible.</p>","PeriodicalId":9635,"journal":{"name":"Case Reports in Ophthalmology","volume":"17 1","pages":"396-401"},"PeriodicalIF":0.6,"publicationDate":"2026-03-03","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC13134845/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"147811434","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}

{"title":"Laser Iris Depigmentation Resulting in Markedly Elevated Intraocular Pressure and Herpes Simplex Keratitis with Corneal Scarring: Case Report.","authors":"Wasim Jaber, Malachy Nemet, Michael Waisbourd","doi":"10.1159/000551226","DOIUrl":"https://doi.org/10.1159/000551226","url":null,"abstract":"<p><strong>Introduction: </strong>Laser iris depigmentation (LID) is a cosmetic procedure designed to alter eye color by reducing iris pigment using an Nd:YAG laser. While this procedure is increasingly popular, it carries risk of serious, sight-threatening complications. This report highlights two cases that emphasize these potential complications.</p><p><strong>Case presentation: </strong>Two patients presented after undergoing multiple sessions of LID performed abroad. They exhibited similar symptoms, including blurry vision, nausea, and eye pain. Case 1 had intraocular pressure (IOP) of 34 mm Hg in both eyes and later developed herpes simplex keratitis, which resulted in corneal scarring. Case 2 presented with IOP of 60 mm Hg in one eye. Both patients were admitted for topical IOP-lowering treatment and received systemic administration of acetazolamide and mannitol, resulting in normalized IOP levels. However, in the first patient, corneal scarring led to a marked decline in visual acuity.</p><p><strong>Conclusions: </strong>LID may be associated with serious, sight-threatening complications, including significant increases in IOP and corneal scarring. It is crucial for both patients and healthcare providers to be fully aware of these potential risks, which may outweigh the potential esthetic benefits of the procedure, and to actively monitor postoperatively for any signs of these adverse effects.</p>","PeriodicalId":9635,"journal":{"name":"Case Reports in Ophthalmology","volume":"17 1","pages":"312-317"},"PeriodicalIF":0.6,"publicationDate":"2026-02-27","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC13075871/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"147688544","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}

{"title":"Combined Hamartoma of the Retina and Retinal Pigment Epithelium: The Contribution of Optical Coherence Tomography in Redefining Its Development and Natural History - A Case Series.","authors":"Andrea Lembo, Francesco Pichi, Alessandra Cernuschi, Luca Marelli, Doris Hadjstilanou, Paolo Nucci","doi":"10.1159/000551030","DOIUrl":"https://doi.org/10.1159/000551030","url":null,"abstract":"<p><strong>Introduction: </strong>Combined hamartoma of the retina and retinal pigment epithelium (CHRRPE) is a rare, benign retinal tumor typically diagnosed in childhood and traditionally believed to involve the full thickness of the retina and the retinal pigment epithelium (RPE) from early stages. With the advent of optical coherence tomography (OCT), increasing evidence suggests that the primary origin of these lesions may lie within the inner retinal layers, with progressive outward extension over time. However, reports documenting very early-stage CHRRPE confined to the inner retina remain scarce.</p><p><strong>Case presentation: </strong>We report two pediatric cases of early-stage CHRRPE diagnosed at a tertiary referral center. A 4-year-old and a 10-year-old boy presented with unexplained reduced visual acuity associated with anisometropic amblyopia or strabismus. Fundoscopic findings were subtle or barely detectable, with no evident pigmentation, fibrosis, or exudation. Multimodal imaging, particularly OCT, revealed increased retinal thickness, loss of normal foveal depression, and disorganization of the inner retinal layers up to the outer plexiform layer, while the outer retina and RPE were preserved. OCT angiography showed distortion of the superficial and deep capillary plexuses in one case. These tomographic features were consistent with CHRRPE and allowed exclusion of other differential diagnoses, including foveal hypoplasia and malignant retinal tumors.</p><p><strong>Conclusion: </strong>These cases illustrate the earliest OCT-detectable phenotype of CHRRPE, supporting the concept that the lesion originates within the inner retina and progressively extends toward the outer retinal layers. Our findings highlight the critical role of OCT in identifying subtle CHRRPE variants in children with otherwise unexplained amblyopia or strabismus and contribute to a better understanding of the developmental origin and natural history of this rare entity.</p>","PeriodicalId":9635,"journal":{"name":"Case Reports in Ophthalmology","volume":"17 1","pages":"345-356"},"PeriodicalIF":0.6,"publicationDate":"2026-02-27","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC13089993/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"147721730","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}