Sansal Gedik, Reyhan Nilgun Karakaya, Olgar Ocal, Hatice Deniz Ilhan, Mehmet Saim Kazan
{"title":"May Acute Appendicitis Complicate the Management of Pseudotumor Cerebri in Shunt-Dependent Patients? A Paediatric Case Report.","authors":"Sansal Gedik, Reyhan Nilgun Karakaya, Olgar Ocal, Hatice Deniz Ilhan, Mehmet Saim Kazan","doi":"10.1159/000552023","DOIUrl":"10.1159/000552023","url":null,"abstract":"<p><strong>Introduction: </strong>Shunt-dependent secondary pseudotumor cerebri may remain clinically stable for many years; however, intra-abdominal sepsis such as perforated appendicitis can necessitate abrupt shunt removal, eliminating cerebrospinal fluid (CSF) diversion and precipitating sudden, vision-threatening intracranial pressure (ICP) elevation.</p><p><strong>Case presentation: </strong>A 17-year-old girl with a long-standing ventriculoperitoneal shunt for a childhood arachnoid cyst initially developed papilledema that was successfully managed with the addition of a lumboperitoneal shunt. Nine months later, she presented with perforated appendicitis requiring emergent appendectomy and removal of both shunts because of the high risk of ascending shunt infection. Although visual acuity was initially preserved after surgery, rapidly progressive bilateral papilledema developed within days, accompanied by marked retinal nerve fibre layer thickening and enlarging visual field defects despite maximal acetazolamide therapy. As reinstitution of CSF diversion was not feasible, bilateral optic nerve sheath fenestration (ONSF) was performed as a vision-preserving intervention. Visual acuity, visual fields, and papilledema improved promptly and subsequently normalised.</p><p><strong>Conclusion: </strong>Perforated appendicitis in shunt-dependent patients may precipitate abrupt, vision-threatening ICP elevation following unavoidable shunt removal. Abdominal pain in patients with a history of CSF diversion should raise immediate suspicion for intra-abdominal sepsis. Early recognition of appendicitis may prevent perforation-related shunt complications, and timely ONSF is critical to avert permanent optic nerve damage when CSF diversion cannot be promptly restored.</p>","PeriodicalId":9635,"journal":{"name":"Case Reports in Ophthalmology","volume":"17 1","pages":"602-609"},"PeriodicalIF":0.6,"publicationDate":"2026-04-29","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC13327671/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"148374573","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":"Disseminated <i>Mycobacterium haemophilum</i> Infection Complicating Immunosuppressive Therapy in HLA-A29 Positive Birdshot Chorioretinopathy: Case Report.","authors":"Brady Hogan, Amit Reddy, Alan G Palestine","doi":"10.1159/000551676","DOIUrl":"10.1159/000551676","url":null,"abstract":"<p><strong>Introduction: </strong>This case describes the clinical course and management of birdshot chorioretinopathy in a patient who developed disseminated <i>Mycobacterium (M.) haemophilum</i> infection while on long-term systemic immunomodulatory therapy (IMT), highlighting the role of local corticosteroid therapy when systemic agents are contraindicated.</p><p><strong>Case presentation: </strong>A 74-year-old male with stable birdshot chorioretinopathy maintained on adalimumab and mycophenolate mofetil developed progressive cutaneous lesions and systemic symptoms, ultimately diagnosed as disseminated <i>M. haemophilum</i>. Systemic IMT was discontinued, resulting in reactivation of intraocular inflammation. Due to the ongoing infectious process contraindicating IMT, local corticosteroid therapies were utilized to control ocular inflammation while mitigating systemic risk. The patient also experienced herpetic epithelial keratitis and steroid-induced ocular hypertension following local corticosteroid therapy, necessitating antiviral therapy and IOP-lowering drops. With multidisciplinary care, intraocular inflammation and vision were stabilized.</p><p><strong>Conclusions: </strong>This case highlights the complexities of managing vision-threatening uveitis in the setting of systemic opportunistic infections, limiting the ability to utilize systemic IMT. Local corticosteroid delivery may offer effective control in such scenarios but carries their own risks.</p>","PeriodicalId":9635,"journal":{"name":"Case Reports in Ophthalmology","volume":"17 1","pages":"562-569"},"PeriodicalIF":0.6,"publicationDate":"2026-04-29","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC13286565/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"148307235","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":"Novel Compound Heterozygous Variants in <i>CDH3</i> Cause Congenital Hypotrichosis with Juvenile Macular Dystrophy: A Case Report with Longitudinal Imaging and Functional Validation.","authors":"Yuchen Lin, Ping Yu, Sunliang Cui, Sijie Xiong, Yuan Zhou, Yuhong Chen, Jijian Lin","doi":"10.1159/000551989","DOIUrl":"10.1159/000551989","url":null,"abstract":"<p><strong>Introduction: </strong>Hypotrichosis with juvenile macular dystrophy (HJMD) is a rare autosomal recessive disorder caused by biallelic <i>CDH3</i> variants. We report a Chinese patient with HJMD carrying novel compound heterozygous <i>CDH3</i> variants, providing functional validation and longitudinal imaging documentation of disease progression.</p><p><strong>Case presentation: </strong>We present an 18-year-old male with painless bilateral vision loss over 8 years and sparse scalp hair noted since 5 years of age. Multimodal imaging documented a severe maculopathy that progressed from outer retinal tubulations (ORTs) to diffuse atrophy within 2 years, accompanied by choroidal thinning and reduced choriocapillaris perfusion. The patient also exhibited the characteristic hypotrichosis. Whole exome sequencing revealed novel compound heterozygous variants in <i>CDH3</i>, including an intronic splicing variant c.391-3C>G, and a missense variant c.1625A>G (p.Asn542Ser), suggesting a diagnosis of HJMD. The minigene splice assay provided evidence that c.391-3C>G leads to aberrant splicing with intron 4 retention and exon 5 skipping. Structural modeling suggested that p.Asn542Ser disrupts a key hydrogen bond in the fourth extracellular cadherin repeat domain, potentially destabilizing the extracellular structure of P-cadherin.</p><p><strong>Conclusions: </strong>This case demonstrates that intronic <i>CDH3</i> variants require functional validation for accurate diagnosis, and that ORTs in HJMD can progress rapidly to diffuse atrophy, highlighting a potential window for future interventions.</p>","PeriodicalId":9635,"journal":{"name":"Case Reports in Ophthalmology","volume":"17 1","pages":"433-440"},"PeriodicalIF":0.6,"publicationDate":"2026-04-28","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC13200835/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"148027288","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}
Özlem Ural Fatihoglu, Murat Tunc, Ali Osman Saatci
{"title":"Place of New Anti-Vascular Endothelial Growth Factor Agents in Recalcitrant Radiation Maculopathy: Case Reports.","authors":"Özlem Ural Fatihoglu, Murat Tunc, Ali Osman Saatci","doi":"10.1159/000552133","DOIUrl":"10.1159/000552133","url":null,"abstract":"<p><strong>Introduction: </strong>Anti-vascular endothelial growth factor (VEGF) therapy is the main treatment alternative in radiation maculopathy as it reduces the macular edema and thereby improves the vision to some extent. The efficacy of newer-generation anti-VEGF agents with greater durability is less known in this entity. This report highlights the administration of faricimab and 8 mg aflibercept in the management of two recalcitrant cases.</p><p><strong>Case presentations: </strong>We report two cases with radiation retinopathy following the CyberKnife stereotactic radiotherapy for the treatment of choroidal melanoma. A 25-year-old man developed left radiation retinopathy 8 months after the treatment. Though three consecutive posterior subtenon 20 mg triamcinolone acetonide injections within 20 months, followed by two intravitreal 1.25 mg bevacizumab injections together with scatter peripheric laser treatment within 8 months, were given, there was persistent severe macular edema, peripheral retinal ischemia, and retinal neovascularizations (NVEs). He was then treated at our department with seven monthly intravitreal faricimab injections, one subsequent aflibercept 8 mg injection, and additional scatter laser photocoagulation, resulting in modest visual improvement, decreased macular edema, and disappearance of NVEs. The second case was a 35-year-old woman diagnosed with radiation retinopathy 1 year after the treatment for the right uveal melanoma. Despite two posterior subtenon 20 mg triamcinolone acetonide injections performed within 11 months, maculopathy was worsened, and she was treated sequentially with four monthly faricimab and five monthly 8 mg aflibercept injections. Macular edema improved partially, and she experienced some visual improvement.</p><p><strong>Conclusion: </strong>Despite administering more durable agents (faricimab and aflibercept 8 mg) even monthly, there were modest anatomical and visual improvements in both cases with recalcitrant radiation maculopathy. Our observations were preliminary clinical observations, rather than definitive evidence, regarding the efficacy of faricimab and aflibercept 8 mg in radiation maculopathy.</p>","PeriodicalId":9635,"journal":{"name":"Case Reports in Ophthalmology","volume":"17 1","pages":"583-595"},"PeriodicalIF":0.6,"publicationDate":"2026-04-28","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC13309099/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"148344010","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}
David Youhanna Mekhaiel, Alexandre Boutet, Jonathan Micieli
{"title":"Vertebral Artery Dissection Presenting as a Migrainous Visual Aura after Sexual Activity: A Case Report.","authors":"David Youhanna Mekhaiel, Alexandre Boutet, Jonathan Micieli","doi":"10.1159/000552215","DOIUrl":"10.1159/000552215","url":null,"abstract":"<p><strong>Introduction: </strong>Migrainous visual aura is characterized by gradual, fully reversible positive visual phenomena often followed by headache. However, vascular conditions such as vertebral artery dissection (VAD) may closely mimic migraine aura, particularly when atypical features are present.</p><p><strong>Case presentation: </strong>A 30-year-old previously healthy woman presented with two episodes of unilateral peripheral blurry vision with shimmering phenomena occurring minutes after sexual activity, followed by migrainous headache. She had no prior migraine history, and neurological and ophthalmologic examinations were normal. Given the side-locked symptoms and exertional trigger, vascular imaging was performed. Computed tomography angiography demonstrated severe stenosis of the left vertebral artery at the V1-V2 junction consistent with dissection, confirmed on magnetic resonance angiography. No acute infarction was identified. She was treated with dual antiplatelet therapy and remained asymptomatic at follow-up.</p><p><strong>Conclusion: </strong>VAD may present with isolated positive visual disturbances mimicking migraine aura, even in young patients without vascular risk factors. Atypical features such as first-time aura and exertional onset should prompt consideration of vascular imaging to avoid misdiagnosis.</p>","PeriodicalId":9635,"journal":{"name":"Case Reports in Ophthalmology","volume":"17 1","pages":"596-601"},"PeriodicalIF":0.6,"publicationDate":"2026-04-28","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC13322696/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"148367117","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}
Faeeqah Almhmoudi, Abdulaziz Bamusa, Abdulaziz Alrashid, Basil Khalid A Alomari, Ghaida I Alselami
{"title":"Early-Onset Retinal Dystrophy in Alpha-Mannosidosis: A Case Report.","authors":"Faeeqah Almhmoudi, Abdulaziz Bamusa, Abdulaziz Alrashid, Basil Khalid A Alomari, Ghaida I Alselami","doi":"10.1159/000551949","DOIUrl":"10.1159/000551949","url":null,"abstract":"<p><strong>Introduction: </strong>Alpha-mannosidosis is a rare inherited lysosomal storage disorder characterized by immune deficiency, intellectual disability, hearing loss, and skeletal and facial dysmorphism. It results from biallelic pathogenic variants in <i>MAN2B1</i> on chromosome 19. Ophthalmic manifestations previously described in alpha-mannosidosis include corneal opacities, lenticular changes, wheel-like cataracts, and strabismus. Although a few reports have documented retinal dystrophy in affected individuals, these cases have typically occurred later in life, between the fourth and sixth decades.</p><p><strong>Case presentation: </strong>We report a 17-year-old female with genetically confirmed alpha-mannosidosis who presented with decreased visual function. Ophthalmic evaluation revealed bilateral optic nerve atrophy, peripapillary hyperpigmentation, salt-and-pepper retinopathy, and optic coherence tomography evidence of photoreceptor disruption. Fundus imaging demonstrated tilted discs and generalized pallor. These features are consistent with early-onset retinal dystrophy.</p><p><strong>Conclusion: </strong>This case represents one of the youngest reported patients with alpha-mannosidosis and retinal dystrophy. Our findings underscore the importance of routine ophthalmologic surveillance, including optical coherence tomography, ERG, and dilated fundus examination, to facilitate early detection, guide family counseling, and optimize visual outcomes.</p>","PeriodicalId":9635,"journal":{"name":"Case Reports in Ophthalmology","volume":"17 1","pages":"471-478"},"PeriodicalIF":0.6,"publicationDate":"2026-04-28","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC13252936/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"148223423","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":"Presumed Sterile Cluster Endophthalmitis following Intravitreal Triamcinolone Acetonide: A Rare Case Series.","authors":"Bhumika Rath, Aarwin Joshua, Sweta Mohapatra, Rajkumar Patra, Pratistha Rath, Nandhakumar Chinnaiyan","doi":"10.1159/000552110","DOIUrl":"10.1159/000552110","url":null,"abstract":"<p><strong>Introduction: </strong>Presumed sterile endophthalmitis (PSE) is a noninfectious inflammatory reaction that mimics infectious endophthalmitis following intravitreal injections. It is a rare but clinically significant complication of intravitreal triamcinolone acetonide (IVTA). This case report describes the clinical presentation, diagnosis, and management of PSE following combined IVTA and anti-vascular endothelial growth factor (VEGF) therapy in patients with chronic diabetic macular edema.</p><p><strong>Case presentation: </strong>In a single operative theater setting, 8 patients received intravitreal anti-VEGF injections and 3 patients received a combination of anti-VEGF (ranibizumab) with triamcinolone acetonide (Aurocort). All injections were performed under sterile precautions. All 3 patients who received combined therapy developed sudden-onset of symptoms within 24 h post-injection, presenting with severe pain, redness, and marked vision loss. Initial visual acuity ranged from hand motion to perception of light. Clinical findings included lid and periorbital edema, severe conjunctival congestion, corneal edema with Descemet membrane folds, anterior chamber cells and flare, sluggish pupils, and dense vitreous inflammation. Vitreous tap and core vitrectomy with intravitreal vancomycin, ceftazidime, and dexamethasone were performed in 2 patients. Cultures from the vitreous, triamcinolone acetonide vial, and ranibizumab vial were negative for organisms. Final visual acuity improved to 6/9-6/12 in treated patients; 1 patient was lost to follow-up.</p><p><strong>Conclusions: </strong>PSE may occur after combined IVTA. Early diagnosis, prompt vitrectomy, and intravitreal antibiotics with steroids are critical for favorable outcomes. The visual prognosis is generally good and depends largely on timely intervention and the status of the underlying retinal disease.</p>","PeriodicalId":9635,"journal":{"name":"Case Reports in Ophthalmology","volume":"17 1","pages":"576-582"},"PeriodicalIF":0.6,"publicationDate":"2026-04-28","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC13309095/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"148343950","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}
Justin Chen, Michael Ying Zhao, Landon James Rohowetz, Harry W Flynn
{"title":"Giant Retinal Hole and Retinal Detachment following Exercise Band Injury: A Case Report.","authors":"Justin Chen, Michael Ying Zhao, Landon James Rohowetz, Harry W Flynn","doi":"10.1159/000551603","DOIUrl":"10.1159/000551603","url":null,"abstract":"<p><strong>Introduction: </strong>Elastic resistance bands have been widely adopted in exercise programs for all age-groups. However, increased use of these resistance bands has been associated with a rise in blunt facial and ocular injuries, of which severe ones may require surgical intervention.</p><p><strong>Case presentation: </strong>A 15-year-old male with high myopia presented 2 weeks after being struck in the left eye by an exercise resistance band. Examination revealed a large 12 mm × 15 mm retinal hole with rolled edges in the temporal periphery and a macula-involving retinal detachment with early proliferative vitreoretinopathy in the left eye. Following 3 vitreoretinal surgeries, retina reattachment was accomplished. At last follow-up at postoperative year 1 from the 3rd surgery, best-corrected visual acuity was 20/40 with a gas-permeable contact lens.</p><p><strong>Conclusion: </strong>This case report demonstrates that favorable visual outcomes may be achievable despite macula-involving rhegmatogenous retinal detachment and early proliferative vitreoretinopathy from an elastic band injury.</p>","PeriodicalId":9635,"journal":{"name":"Case Reports in Ophthalmology","volume":"17 1","pages":"534-538"},"PeriodicalIF":0.6,"publicationDate":"2026-04-28","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC13282106/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"148293571","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":"Preseptal Cellulitis as the Presenting Sign of Systemic Inflammatory Disease Secondary to Chikungunya Virus: A Case Report.","authors":"Merve Kulbay, Stuti M Tanya, Feyza Sule Aslan, Raheem Remtulla, Marcele Fonseca Falcao","doi":"10.1159/000552091","DOIUrl":"10.1159/000552091","url":null,"abstract":"<p><strong>Introduction: </strong>Preseptal cellulitis in children, a leading cause of pediatric orbital inflammation, often masquerades as more severe systemic inflammatory conditions. Chikungunya virus is an arbovirus with a wide spectrum of systemic manifestations that can lead to potentially harmful complications within the nervous, cardiovascular, and renal systems if left undetected. A diagnostic challenge resides with its rare ophthalmic presenting feature of preseptal cellulitis. Herein, we described a case of Chikungunya virus (CHIKV) masquerading as a preseptal cellulitis that led to a systemic inflammatory disease in a pediatric patient.</p><p><strong>Case presentation: </strong>A 12-year-old male presented with unilateral preseptal cellulitis, with mildly elevated intraocular pressure, following a recent trip to Egypt. Visual acuity and color vision were preserved. Left knee effusion, cervical and inguinal adenopathy, and abdominal tenderness were noted. Imaging demonstrated bilateral preseptal cellulitis with an associated dacryoadenitis. Extensive multidisciplinary investigations determined the presence of positive CHIKV IgM titers with a diagnosis of inflammatory pancreatitis and biopsy-proven acute kidney tubular injury. Clinical improvement was noted with supportive treatment.</p><p><strong>Conclusion: </strong>CHIKV can masquerade as a preseptal cellulitis in pediatric patients and lead to a multisystem inflammatory disease. Prompt clinical suspicion, diagnosis, and treatment can prevent harmful nervous, cardiovascular, and renal system complications.</p>","PeriodicalId":9635,"journal":{"name":"Case Reports in Ophthalmology","volume":"17 1","pages":"545-552"},"PeriodicalIF":0.6,"publicationDate":"2026-04-18","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC13286566/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"148307295","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}
Jedrzej Golebka, Omar Moussa, Dvir Koenigstein, Royce W S Chen, Noga Harizman
{"title":"Delayed-Onset <i>Staphylococcus epidermidis</i> Endophthalmitis Masquerading as Chronic Anterior Uveitis 15 Years after Cataract Surgery: A Case Report.","authors":"Jedrzej Golebka, Omar Moussa, Dvir Koenigstein, Royce W S Chen, Noga Harizman","doi":"10.1159/000552089","DOIUrl":"10.1159/000552089","url":null,"abstract":"<p><strong>Introduction: </strong>The aim of the study was to report a case of chronic postoperative endophthalmitis that presented 15 years after cataract surgery.</p><p><strong>Case presentation: </strong>A 73-year-old man presented with ocular inflammation and blurred vision. He was diagnosed with bilateral chronic uveitis by his previous providers. The patient's ocular inflammation persisted despite multiple immunosuppressive modalities, both topical and systemic. An infectious etiology was suspected, and an intraocular lens (IOL)/Soemmering's ring explanation surgery was performed. Specimens were sent to pathology and microbiology. Pathology and microbiology reports came back positive for <i>Staphylococcus epidermidis</i> from both the IOL and the capsular bag.</p><p><strong>Conclusion: </strong>To the best of our knowledge, this is a rare and unique case of culturally proven <i>S. epidermidis</i> postoperative endophthalmitis diagnosed 20 years after the inoculating surgery. This case demonstrates the challenges of managing a severe case of ocular inflammation and the resulting increased intraocular pressure not responding to multiple modalities of topical and systemic immunosuppressive medications.</p>","PeriodicalId":9635,"journal":{"name":"Case Reports in Ophthalmology","volume":"17 1","pages":"526-533"},"PeriodicalIF":0.6,"publicationDate":"2026-04-18","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC13278730/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"148293514","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}