Pub Date : 2025-11-02eCollection Date: 2025-01-01DOI: 10.1159/000546891
Alba Chiara Termite, Pasquale Viggiano, Giacomo Boscia, Giovanni Alessio, Francesco Boscia
Introduction: The aim of the study was to evaluate the long-term efficacy of eplerenone in patients with peripapillary pachychoroid syndrome (PPS), a recently described entity characterized by vascular congestion of Haller's layer vessels near the optic nerve.
Case presentations: Three male patients (mean age: 70.3 years) with PPS received oral eplerenone 50 mg daily for 12 months. Baseline and post-treatment choroidal thickness, visual acuity, and retinal fluid status were assessed. At baseline, mean choroidal thickness was 420 µm, and mean visual acuity was 70 letters. After 12 months of treatment, all patients showed a significant reduction in choroidal thickness (mean: 48 µm, p < 0.05) and near-complete resolution of intraretinal and subretinal fluid. No adverse events were reported.
Conclusions: Long-term eplerenone treatment appears effective in reducing choroidal thickness and improving visual outcomes in PPS patients. These findings suggest mineralocorticoid receptor antagonists may be valuable in managing pachychoroid spectrum disorders.
简介:本研究的目的是评估依普利酮对乳头周围厚脉络膜综合征(PPS)患者的长期疗效,PPS是最近发现的一种以视神经附近哈勒层血管充血为特征的实体。病例介绍:三名患有PPS的男性患者(平均年龄:70.3岁)每天口服依普利酮50mg,持续12个月。评估基线和治疗后脉络膜厚度、视力和视网膜液状态。基线时,平均脉络膜厚度为420µm,平均视力为70个字母。治疗12个月后,所有患者的脉膜厚度均显著减少(平均48µm, p < 0.05),视网膜内和视网膜下积液几乎完全溶解。无不良事件报告。结论:长期应用依普利酮治疗可有效降低PPS患者的脉络膜厚度,改善视力。这些发现提示矿皮质激素受体拮抗剂可能在治疗厚脉络膜谱系障碍中有价值。
{"title":"Long-Term Eplerenone Treatment in Peripapillary Pachychoroid Syndrome: A Case Series.","authors":"Alba Chiara Termite, Pasquale Viggiano, Giacomo Boscia, Giovanni Alessio, Francesco Boscia","doi":"10.1159/000546891","DOIUrl":"10.1159/000546891","url":null,"abstract":"<p><strong>Introduction: </strong>The aim of the study was to evaluate the long-term efficacy of eplerenone in patients with peripapillary pachychoroid syndrome (PPS), a recently described entity characterized by vascular congestion of Haller's layer vessels near the optic nerve.</p><p><strong>Case presentations: </strong>Three male patients (mean age: 70.3 years) with PPS received oral eplerenone 50 mg daily for 12 months. Baseline and post-treatment choroidal thickness, visual acuity, and retinal fluid status were assessed. At baseline, mean choroidal thickness was 420 µm, and mean visual acuity was 70 letters. After 12 months of treatment, all patients showed a significant reduction in choroidal thickness (mean: 48 µm, <i>p</i> < 0.05) and near-complete resolution of intraretinal and subretinal fluid. No adverse events were reported.</p><p><strong>Conclusions: </strong>Long-term eplerenone treatment appears effective in reducing choroidal thickness and improving visual outcomes in PPS patients. These findings suggest mineralocorticoid receptor antagonists may be valuable in managing pachychoroid spectrum disorders.</p>","PeriodicalId":9635,"journal":{"name":"Case Reports in Ophthalmology","volume":"16 1","pages":"888-895"},"PeriodicalIF":0.6,"publicationDate":"2025-11-02","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12674667/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"145676559","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: Optic disc pit maculopathy is a challenging retinal disease that can significantly impact the quality of life, especially in pediatric patients. We report our surgical approach in the management of an 8-year-old boy with this rare condition who was referred to our clinic.
Case presentation: An 8-year-old male patient presented with visual acuity of 20/200 in the left eye. After complete examination and OCT imaging was performed, the diagnosis of optic disc pit maculopathy (ODPM) was obtained. ODPM is a challenging condition to treat without a gold standard approach, especially in the pediatric population. A surgical approach was decided in order to treat this condition. This involved performing a pars plana vitrectomy and an internal limiting membrane (ILM) flap over the optic disc pit. The operation was successful as the ILM flap covered the optic disc pit sufficiently enough to block communication between the vitreous cavity and the subretinal space. This led to the resolution of macular edema during the postoperative period. One year post surgery, the boy's visual acuity improved to 20/32, accompanied by the patient's overall satisfaction and the anatomical confirmation of the successful result using OCT.
Conclusion: Taking into consideration the surgical difficulties associated with this age group, we highlight the efficacy and safety of the ILM flap inversion technique as a viable surgical option for optic disc pit maculopathy in pediatric patients.
{"title":"A Case Report of Optic Disc Pit Maculopathy in an 8-Year-Old Boy: Vitrectomy with Internal Limiting Membrane Flap in a Child.","authors":"Stergios Ntikos, Spyridon Doumazos, Aikaterini Barlampa, Eirini Okoutsidou, Niki Zampogianni, Stylianos A Kandarakis, Panagiotis Stavrakas, Asli Perente, Doukas Dardabounis, Petros Petrou","doi":"10.1159/000549278","DOIUrl":"10.1159/000549278","url":null,"abstract":"<p><strong>Introduction: </strong>Optic disc pit maculopathy is a challenging retinal disease that can significantly impact the quality of life, especially in pediatric patients. We report our surgical approach in the management of an 8-year-old boy with this rare condition who was referred to our clinic.</p><p><strong>Case presentation: </strong>An 8-year-old male patient presented with visual acuity of 20/200 in the left eye. After complete examination and OCT imaging was performed, the diagnosis of optic disc pit maculopathy (ODPM) was obtained. ODPM is a challenging condition to treat without a gold standard approach, especially in the pediatric population. A surgical approach was decided in order to treat this condition. This involved performing a pars plana vitrectomy and an internal limiting membrane (ILM) flap over the optic disc pit. The operation was successful as the ILM flap covered the optic disc pit sufficiently enough to block communication between the vitreous cavity and the subretinal space. This led to the resolution of macular edema during the postoperative period. One year post surgery, the boy's visual acuity improved to 20/32, accompanied by the patient's overall satisfaction and the anatomical confirmation of the successful result using OCT.</p><p><strong>Conclusion: </strong>Taking into consideration the surgical difficulties associated with this age group, we highlight the efficacy and safety of the ILM flap inversion technique as a viable surgical option for optic disc pit maculopathy in pediatric patients.</p>","PeriodicalId":9635,"journal":{"name":"Case Reports in Ophthalmology","volume":"16 1","pages":"863-868"},"PeriodicalIF":0.6,"publicationDate":"2025-11-02","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12674666/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"145676505","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 : 2025-10-30eCollection Date: 2025-01-01DOI: 10.1159/000549279
Giulia Gregori, Lorenzo Mangoni, Alessio Muzi, Cesare Mariotti, Marco Lupidi
Introduction: Laser photobiomodulation, including retinal rejuvenation therapy (2RT), is a system which selectively targets the retinal pigment epithelium by a concise 3 ns pulse duration. The advantage of this laser system over the traditional thermal laser is that the pulsed, very short duration laser effects can be titrated as spatially confined photodisruptors without resultant conductive thermal spread and therefore collateral damage. It has been investigated primarily in age-related macular degeneration (AMD), particularly in decreasing drusen and slowing the rate of AMD progression. In this case, we have described a case of neovascular maculopathy occurring shortly after 2RT in a young myopic patient.
Case presentation: We report the case of a 28-year-old male who presented with unilateral visual impairment following laser 2RT. Two months before, he was subjected to photorefractive keratectomy for moderate myopia (-3.00 D). The baseline optical coherence tomography (OCT) imaging revealed "sharp-peaked" pigment epithelium detachment (PED) in the subfoveal area. Fluorescein angiography indicated a focal area of irregular foveal hyperfluorescence. Observation was advised, and laser 2RT was performed. However, 1 month later, the patient developed a neovascular lesion in the same eye, confirmed by OCT-angiography, requiring urgent intravitreal anti-VEGF therapy.
Conclusions: In summary, this case illustrates a progressive maculopathy culminating in choroidal neovascularization triggered by laser 2RT in a young myopic patient.
{"title":"Neovascular Maculopathy after Laser Retinal Rejuvenation Therapy in a Young Myopic Patient: A Case Report.","authors":"Giulia Gregori, Lorenzo Mangoni, Alessio Muzi, Cesare Mariotti, Marco Lupidi","doi":"10.1159/000549279","DOIUrl":"10.1159/000549279","url":null,"abstract":"<p><strong>Introduction: </strong>Laser photobiomodulation, including retinal rejuvenation therapy (2RT), is a system which selectively targets the retinal pigment epithelium by a concise 3 ns pulse duration. The advantage of this laser system over the traditional thermal laser is that the pulsed, very short duration laser effects can be titrated as spatially confined photodisruptors without resultant conductive thermal spread and therefore collateral damage. It has been investigated primarily in age-related macular degeneration (AMD), particularly in decreasing drusen and slowing the rate of AMD progression. In this case, we have described a case of neovascular maculopathy occurring shortly after 2RT in a young myopic patient.</p><p><strong>Case presentation: </strong>We report the case of a 28-year-old male who presented with unilateral visual impairment following laser 2RT. Two months before, he was subjected to photorefractive keratectomy for moderate myopia (-3.00 D). The baseline optical coherence tomography (OCT) imaging revealed \"sharp-peaked\" pigment epithelium detachment (PED) in the subfoveal area. Fluorescein angiography indicated a focal area of irregular foveal hyperfluorescence. Observation was advised, and laser 2RT was performed. However, 1 month later, the patient developed a neovascular lesion in the same eye, confirmed by OCT-angiography, requiring urgent intravitreal anti-VEGF therapy.</p><p><strong>Conclusions: </strong>In summary, this case illustrates a progressive maculopathy culminating in choroidal neovascularization triggered by laser 2RT in a young myopic patient.</p>","PeriodicalId":9635,"journal":{"name":"Case Reports in Ophthalmology","volume":"16 1","pages":"841-846"},"PeriodicalIF":0.6,"publicationDate":"2025-10-30","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12659666/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"145647564","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 : 2025-10-25eCollection Date: 2025-01-01DOI: 10.1159/000549142
Simran Ohri, Iden Amiri, Gitanjali M Fleischman, David Fleischman
Introduction: Infectious mononucleosis (IM), most commonly caused by Epstein-Barr virus (EBV), classically presents with fever, pharyngitis, and cervical lymphadenopathy. However, less common manifestations such as bilateral periorbital edema, known as Hoagland sign, may precede or obscure more typical features, complicating diagnosis. Although described in the literature for decades, Hoagland sign remains underrecognized in clinical practice, especially in pediatric and young adult patients. This oversight can lead to misdiagnosis, delayed testing, unnecessary antibiotic use, and patient distress.
Case presentation: We present 2 cases of female patients, one pediatric and one young adult, who initially presented with bilateral periorbital edema and nonspecific systemic symptoms. In both cases, infectious mononucleosis was not initially suspected. One patient was treated for streptococcal pharyngitis and later developed a rash typical of antibiotic-associated IM. The second patient was denied EBV testing at an urgent care clinic despite a direct request, due to provider unfamiliarity with the association between periorbital edema and IM. Both were eventually diagnosed with EBV via serologic testing and recovered with supportive care.
Conclusion: These cases highlight a critical gap in provider awareness that, if addressed, could reduce diagnostic delays and improve patient care. Recognizing Hoagland sign as a valid early clue for IM, particularly in female patients,can help differentiate benign viral illness from more concerning differentials such as orbital cellulitis, nephrotic syndrome, or allergic reactions. This report serves as a clinical reminder to include EBV in the differential diagnosis of atraumatic, bilateral eyelid swelling and to educate frontline providers on this underappreciated presentation.
{"title":"Case Report: Periorbital Edema as an Overlooked Presentation of Epstein-Barr Virus.","authors":"Simran Ohri, Iden Amiri, Gitanjali M Fleischman, David Fleischman","doi":"10.1159/000549142","DOIUrl":"10.1159/000549142","url":null,"abstract":"<p><strong>Introduction: </strong>Infectious mononucleosis (IM), most commonly caused by Epstein-Barr virus (EBV), classically presents with fever, pharyngitis, and cervical lymphadenopathy. However, less common manifestations such as bilateral periorbital edema, known as Hoagland sign, may precede or obscure more typical features, complicating diagnosis. Although described in the literature for decades, Hoagland sign remains underrecognized in clinical practice, especially in pediatric and young adult patients. This oversight can lead to misdiagnosis, delayed testing, unnecessary antibiotic use, and patient distress.</p><p><strong>Case presentation: </strong>We present 2 cases of female patients, one pediatric and one young adult, who initially presented with bilateral periorbital edema and nonspecific systemic symptoms. In both cases, infectious mononucleosis was not initially suspected. One patient was treated for streptococcal pharyngitis and later developed a rash typical of antibiotic-associated IM. The second patient was denied EBV testing at an urgent care clinic despite a direct request, due to provider unfamiliarity with the association between periorbital edema and IM. Both were eventually diagnosed with EBV via serologic testing and recovered with supportive care.</p><p><strong>Conclusion: </strong>These cases highlight a critical gap in provider awareness that, if addressed, could reduce diagnostic delays and improve patient care. Recognizing Hoagland sign as a valid early clue for IM, particularly in female patients,can help differentiate benign viral illness from more concerning differentials such as orbital cellulitis, nephrotic syndrome, or allergic reactions. This report serves as a clinical reminder to include EBV in the differential diagnosis of atraumatic, bilateral eyelid swelling and to educate frontline providers on this underappreciated presentation.</p>","PeriodicalId":9635,"journal":{"name":"Case Reports in Ophthalmology","volume":"16 1","pages":"834-840"},"PeriodicalIF":0.6,"publicationDate":"2025-10-25","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12659628/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"145647613","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 : 2025-10-18eCollection Date: 2025-01-01DOI: 10.1159/000548958
Naina Chaturvedi, Himani Yadav, Suvarna Pandey
Introduction: Orbital lymphomas, though uncommon, are significant masquerade syndromes that can resemble inflammatory or infectious orbital conditions like orbital cellulitis.
Case presentation: We describe the case of an 80-year-old woman who experienced sudden painful vision loss, proptosis, and limited eye movement, initially treated as orbital cellulitis with compressive optic neuropathy. The absence of clinical improvement led to further imaging and a biopsy, which identified diffuse large B-cell lymphoma.
Conclusion: This case emphasizes the necessity for high clinical suspicion, particularly in older patients, and highlights the diagnostic challenges posed by steroid use and overlapping symptoms with infectious causes. We discuss the significance of a multidisciplinary approach and suggest a clinical checklist for recognizing masquerade syndromes in ophthalmology.
{"title":"Orbital Diffuse Large B-Cell Lymphoma: A Diagnostic Masquerade.","authors":"Naina Chaturvedi, Himani Yadav, Suvarna Pandey","doi":"10.1159/000548958","DOIUrl":"10.1159/000548958","url":null,"abstract":"<p><strong>Introduction: </strong>Orbital lymphomas, though uncommon, are significant masquerade syndromes that can resemble inflammatory or infectious orbital conditions like orbital cellulitis.</p><p><strong>Case presentation: </strong>We describe the case of an 80-year-old woman who experienced sudden painful vision loss, proptosis, and limited eye movement, initially treated as orbital cellulitis with compressive optic neuropathy. The absence of clinical improvement led to further imaging and a biopsy, which identified diffuse large B-cell lymphoma.</p><p><strong>Conclusion: </strong>This case emphasizes the necessity for high clinical suspicion, particularly in older patients, and highlights the diagnostic challenges posed by steroid use and overlapping symptoms with infectious causes. We discuss the significance of a multidisciplinary approach and suggest a clinical checklist for recognizing masquerade syndromes in ophthalmology.</p>","PeriodicalId":9635,"journal":{"name":"Case Reports in Ophthalmology","volume":"16 1","pages":"856-862"},"PeriodicalIF":0.6,"publicationDate":"2025-10-18","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12659678/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"145647167","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 : 2025-10-18eCollection Date: 2025-01-01DOI: 10.1159/000548728
Bar Klain, Noa Geffen, Alon Zahavi
Introduction: Glaucoma following corneal transplantation and artificial iris implantation represents a major therapeutic challenge, and data on optimal surgical management in such complex cases are scarce.
Case presentation: We report the case of a 28-year-old male with uncontrolled intraocular pressure (IOP) after penetrating keratoplasty and Morcher aniridia ring implantation. Despite maximally tolerated medical therapy, IOP remained elevated. PreserFlo MicroShunt implantation was performed successfully, leading to sustained IOP reduction without medications and preservation of graft clarity at 7-month follow-up.
Conclusion: To our knowledge, this is the first reported case of PreserFlo MicroShunt implantation in a patient with combined keratoplasty and Morcher aniridia rings. This case highlights the potential role of PreserFlo as a viable option for IOP control in complex post-traumatic and postsurgical eyes. However, the findings represent a short-term (7-month) outcome, and the long-term efficacy requires further evaluation.
{"title":"PreserFlo Meets Morcher: Short-Term Outcome in Complex Post-Traumatic Eye with PKP and Aniridia Ring: A Case Report.","authors":"Bar Klain, Noa Geffen, Alon Zahavi","doi":"10.1159/000548728","DOIUrl":"10.1159/000548728","url":null,"abstract":"<p><strong>Introduction: </strong>Glaucoma following corneal transplantation and artificial iris implantation represents a major therapeutic challenge, and data on optimal surgical management in such complex cases are scarce.</p><p><strong>Case presentation: </strong>We report the case of a 28-year-old male with uncontrolled intraocular pressure (IOP) after penetrating keratoplasty and Morcher aniridia ring implantation. Despite maximally tolerated medical therapy, IOP remained elevated. PreserFlo MicroShunt implantation was performed successfully, leading to sustained IOP reduction without medications and preservation of graft clarity at 7-month follow-up.</p><p><strong>Conclusion: </strong>To our knowledge, this is the first reported case of PreserFlo MicroShunt implantation in a patient with combined keratoplasty and Morcher aniridia rings. This case highlights the potential role of PreserFlo as a viable option for IOP control in complex post-traumatic and postsurgical eyes. However, the findings represent a short-term (7-month) outcome, and the long-term efficacy requires further evaluation.</p>","PeriodicalId":9635,"journal":{"name":"Case Reports in Ophthalmology","volume":"16 1","pages":"821-828"},"PeriodicalIF":0.6,"publicationDate":"2025-10-18","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12659623/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"145647131","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 : 2025-10-18eCollection Date: 2025-01-01DOI: 10.1159/000549058
Shmuel Kagasov, Nir Gomel, Amit Biran, Asaf Friehmann, Michael Mimouni, Asaf Achiron
Introduction: Staphylococcus-related marginal keratitis is an inflammation of the corneal periphery characterized by the presence of stromal infiltrates and blepharoconjunctivitis. Conventional treatment consists of antibiotics and corticosteroid eye drops, and recurrence is prevalent upon treatment cessation. In this case report, we present a 1-year follow-up of the results of a novel therapeutic approach for recurrent marginal keratitis using a combination of chlorhexidine and vancomycin eye drops.
Case presentation: A 34-year-old male presented with an 8-year history of recurrent staphylococcus-related marginal keratitis in his right eye, consisting of 5 acute episodes, which under a conventional treatment regimen resolved within 10-14 days. During the follow-up of his right eye, an inflammation in his left eye developed as well. Treatment with a combination of chlorhexidine at 0.02% and vancomycin at 50 mg/mL eye drops brought rapid resolution of the acute inflammation with preservation of the effect for at least 1 year.
Conclusions: A combination of chlorhexidine and vancomycin eye drops may provide an effective treatment for marginal keratitis. It demonstrated rapid improvement and maintained long-term stability, with disease control observed throughout 1-year follow-up.
{"title":"Management of Recurrent Staphylococcus-Related Marginal Keratitis with Chlorhexidine-Vancomycin Combination: A 1-Year Follow-Up Case Report.","authors":"Shmuel Kagasov, Nir Gomel, Amit Biran, Asaf Friehmann, Michael Mimouni, Asaf Achiron","doi":"10.1159/000549058","DOIUrl":"10.1159/000549058","url":null,"abstract":"<p><strong>Introduction: </strong>Staphylococcus-related marginal keratitis is an inflammation of the corneal periphery characterized by the presence of stromal infiltrates and blepharoconjunctivitis. Conventional treatment consists of antibiotics and corticosteroid eye drops, and recurrence is prevalent upon treatment cessation. In this case report, we present a 1-year follow-up of the results of a novel therapeutic approach for recurrent marginal keratitis using a combination of chlorhexidine and vancomycin eye drops.</p><p><strong>Case presentation: </strong>A 34-year-old male presented with an 8-year history of recurrent staphylococcus-related marginal keratitis in his right eye, consisting of 5 acute episodes, which under a conventional treatment regimen resolved within 10-14 days. During the follow-up of his right eye, an inflammation in his left eye developed as well. Treatment with a combination of chlorhexidine at 0.02% and vancomycin at 50 mg/mL eye drops brought rapid resolution of the acute inflammation with preservation of the effect for at least 1 year.</p><p><strong>Conclusions: </strong>A combination of chlorhexidine and vancomycin eye drops may provide an effective treatment for marginal keratitis. It demonstrated rapid improvement and maintained long-term stability, with disease control observed throughout 1-year follow-up.</p>","PeriodicalId":9635,"journal":{"name":"Case Reports in Ophthalmology","volume":"16 1","pages":"829-833"},"PeriodicalIF":0.6,"publicationDate":"2025-10-18","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12659595/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"145647596","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 : 2025-10-16eCollection Date: 2025-01-01DOI: 10.1159/000548833
Aishwarya M Angadi, Kalibo Jakhalu, Iqra Mushtaq, Saujas Shah
Introduction: Ocular cystinosis is a rare hereditary lysosomal storage disorder characterized by amino acid cystine deposition in various tissues, prominently affecting the cornea and conjunctiva. While typically diagnosed in childhood, cases in adults may present atypically or as part of the nephropathic spectrum. This report highlights the clinical presentation, diagnosis, and management of ocular cystinosis in a 37-year-old male.
Case presentation: A 37-year-old male presented with complaints of photophobia and ocular discomfort for a duration of 6 months. On slit-lamp examination, characteristic crystalline deposits were observed in the corneal stroma. Additional findings included mild conjunctival injection. Visual acuity was 6/6 in both eyes. Anterior segment optical coherence tomography demonstrated hyperreflective deposits up to corneal stroma, while specular microscopy revealed a normal endothelial cell count and morphology. Systemic evaluation revealed no signs of cystinosis. The patient was treated with topical cysteamine ophthalmic solution 0.44%, resulting in subjective improvement in photophobia and stabilization of corneal findings over a 6-month follow-up period.
Conclusion: This case underscores the importance of recognizing ocular manifestations of cystinosis in adult patients, particularly those with no history or signs of systemic involvement. Early and sustained use of topical cysteamine ophthalmic solutions can significantly mitigate symptoms and prevent progression of corneal changes. Comprehensive management necessitates a multidisciplinary approach to address systemic and ocular sequelae of the disease.
{"title":"Shimmering Clarity: A Rare Case Report of Ocular Cystinosis.","authors":"Aishwarya M Angadi, Kalibo Jakhalu, Iqra Mushtaq, Saujas Shah","doi":"10.1159/000548833","DOIUrl":"10.1159/000548833","url":null,"abstract":"<p><strong>Introduction: </strong>Ocular cystinosis is a rare hereditary lysosomal storage disorder characterized by amino acid cystine deposition in various tissues, prominently affecting the cornea and conjunctiva. While typically diagnosed in childhood, cases in adults may present atypically or as part of the nephropathic spectrum. This report highlights the clinical presentation, diagnosis, and management of ocular cystinosis in a 37-year-old male.</p><p><strong>Case presentation: </strong>A 37-year-old male presented with complaints of photophobia and ocular discomfort for a duration of 6 months. On slit-lamp examination, characteristic crystalline deposits were observed in the corneal stroma. Additional findings included mild conjunctival injection. Visual acuity was 6/6 in both eyes. Anterior segment optical coherence tomography demonstrated hyperreflective deposits up to corneal stroma, while specular microscopy revealed a normal endothelial cell count and morphology. Systemic evaluation revealed no signs of cystinosis. The patient was treated with topical cysteamine ophthalmic solution 0.44%, resulting in subjective improvement in photophobia and stabilization of corneal findings over a 6-month follow-up period.</p><p><strong>Conclusion: </strong>This case underscores the importance of recognizing ocular manifestations of cystinosis in adult patients, particularly those with no history or signs of systemic involvement. Early and sustained use of topical cysteamine ophthalmic solutions can significantly mitigate symptoms and prevent progression of corneal changes. Comprehensive management necessitates a multidisciplinary approach to address systemic and ocular sequelae of the disease.</p>","PeriodicalId":9635,"journal":{"name":"Case Reports in Ophthalmology","volume":"16 1","pages":"847-855"},"PeriodicalIF":0.6,"publicationDate":"2025-10-16","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12659670/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"145647413","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: Sildenafil, a widely prescribed phosphodiesterase-5 (PDE5) inhibitor for erectile dysfunction, is generally well tolerated. However, ocular vascular complications - including non-arteritic anterior ischemic optic neuropathy, central serous chorioretinopathy, and retinal vein occlusion (RVO) - have been reported. Though rare, such events can occur even in individuals without conventional systemic risk factors.
Case presentation: A 55-year-old man with no prior medical history developed sudden, painless visual blurring in his right eye 48 h after ingesting a single 100 mg dose of sildenafil. Clinical evaluation revealed a superotemporal branch RVO with macular edema. A comprehensive systemic and hematologic workup, including thrombophilia screening and cardiac evaluation, yielded normal results. The patient was treated with an intravitreal injection of bevacizumab, leading to anatomical and visual improvement.
Conclusion: This case draws attention to a possible association between sildenafil use and retinal vascular events, particularly BRVO, even in healthy individuals. The temporal proximity and absence of systemic risk factors suggest a potential dose-independent vascular susceptibility. Clinicians should be aware of this rare but potentially vision-threatening complication.
{"title":"When Erectile Dysfunction Therapy Clouds the Vision: ST-BRVO after Sildenafil in a Healthy Male.","authors":"Nilesh Giri, Deepaswi Bhavsar, Renu Magdum, Harshita Kashyap, Himani Yadav","doi":"10.1159/000548573","DOIUrl":"10.1159/000548573","url":null,"abstract":"<p><strong>Introduction: </strong>Sildenafil, a widely prescribed phosphodiesterase-5 (PDE5) inhibitor for erectile dysfunction, is generally well tolerated. However, ocular vascular complications - including non-arteritic anterior ischemic optic neuropathy, central serous chorioretinopathy, and retinal vein occlusion (RVO) - have been reported. Though rare, such events can occur even in individuals without conventional systemic risk factors.</p><p><strong>Case presentation: </strong>A 55-year-old man with no prior medical history developed sudden, painless visual blurring in his right eye 48 h after ingesting a single 100 mg dose of sildenafil. Clinical evaluation revealed a superotemporal branch RVO with macular edema. A comprehensive systemic and hematologic workup, including thrombophilia screening and cardiac evaluation, yielded normal results. The patient was treated with an intravitreal injection of bevacizumab, leading to anatomical and visual improvement.</p><p><strong>Conclusion: </strong>This case draws attention to a possible association between sildenafil use and retinal vascular events, particularly BRVO, even in healthy individuals. The temporal proximity and absence of systemic risk factors suggest a potential dose-independent vascular susceptibility. Clinicians should be aware of this rare but potentially vision-threatening complication.</p>","PeriodicalId":9635,"journal":{"name":"Case Reports in Ophthalmology","volume":"16 1","pages":"813-820"},"PeriodicalIF":0.6,"publicationDate":"2025-10-16","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12659608/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"145647362","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: Compared with sub-Tenon's anesthesia (STA), trans-Tenon's capsule retrobulbar anesthesia (TTRBA) provides rapid and complete akinesia. However, excessive anesthetic volume may cause complications such as choroidal detachment (CD).
Case presentation: A 59-year-old man who had previously undergone cataract surgery presented with rapidly progressive visual field constriction in the right eye. The best corrected visual acuity (BCVA) was 20/16; intraocular pressure, 13 mm Hg; and axial length, 29.99 mm. Multiple retinal breaks were detected in the superior and nasal retina, with retinal detachment extending from the superior to the inferior retina. Vitrectomy was performed under local anesthesia. STA (2.5 mL lidocaine) was followed by TTRBA (3.0 mL initially, plus an additional total of 4.5 mL administered in two injections) and 7.5 mg intravenous pentazocine. The cumulative TTRBA volume reached 7.5 mL within 50 min. Immediately after the final injection, an orange choroidal elevation developed across the entire retina, including the macula, without vascular whitening. BCVA recovered to 20/20 at 2 weeks postoperatively, with retinal reattachment.
Conclusion: This case suggests that repeated intraconal anesthetic injections totaling >7 mL in a short time may induce macula-involving serous CD via sustained intraorbital pressure elevation. Thus, limiting volume and considering lower volume alternatives may reduce risk.
{"title":"Macula-Involving Serous Choroidal Detachment following Multiple Short-Interval Trans-Tenon's Capsule Retrobulbar Anesthesia Injections during Vitreoretinal Surgery: A Case Report.","authors":"Ayako Sadahide, Ayaka Kato, Chika Yokota, Hirokazu Sakaguchi","doi":"10.1159/000548928","DOIUrl":"10.1159/000548928","url":null,"abstract":"<p><strong>Introduction: </strong>Compared with sub-Tenon's anesthesia (STA), trans-Tenon's capsule retrobulbar anesthesia (TTRBA) provides rapid and complete akinesia. However, excessive anesthetic volume may cause complications such as choroidal detachment (CD).</p><p><strong>Case presentation: </strong>A 59-year-old man who had previously undergone cataract surgery presented with rapidly progressive visual field constriction in the right eye. The best corrected visual acuity (BCVA) was 20/16; intraocular pressure, 13 mm Hg; and axial length, 29.99 mm. Multiple retinal breaks were detected in the superior and nasal retina, with retinal detachment extending from the superior to the inferior retina. Vitrectomy was performed under local anesthesia. STA (2.5 mL lidocaine) was followed by TTRBA (3.0 mL initially, plus an additional total of 4.5 mL administered in two injections) and 7.5 mg intravenous pentazocine. The cumulative TTRBA volume reached 7.5 mL within 50 min. Immediately after the final injection, an orange choroidal elevation developed across the entire retina, including the macula, without vascular whitening. BCVA recovered to 20/20 at 2 weeks postoperatively, with retinal reattachment.</p><p><strong>Conclusion: </strong>This case suggests that repeated intraconal anesthetic injections totaling >7 mL in a short time may induce macula-involving serous CD via sustained intraorbital pressure elevation. Thus, limiting volume and considering lower volume alternatives may reduce risk.</p>","PeriodicalId":9635,"journal":{"name":"Case Reports in Ophthalmology","volume":"16 1","pages":"807-812"},"PeriodicalIF":0.6,"publicationDate":"2025-10-13","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12659597/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"145647518","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}