Pub Date : 2025-08-04eCollection Date: 2025-01-01DOI: 10.1159/000547739
Marcelo Zas, Marcos Mendaro, Guido Bregliano, Alejandro Javier Aguilar, Maria Carolina Pozzoni, Ashish Sharma
Introduction: We report a case of spontaneous closure after early reopening of a previously successfully treated macular hole (MH).
Case presentation: A 73-year-old patient presented with visual acuity of 20/100 and complained of defective vision. Examination and investigations confirmed a full-thickness MH. Surgical intervention led to complete closure of the hole on OCT within 5 days. However, the patient reported recurrence of symptoms 2 weeks later, and reopening of the hole was diagnosed. Re-surgery was advised, but surprisingly, the hole spontaneously closed by the seventh week and remained closed at the last follow-up (6 months post-surgery), with visual acuity improving to 20/50.
Conclusions: Reopening of an MH can occur months or years after successful treatment, and the underlying reasons remain unclear. While reoperation is often indicated, spontaneous healing, although rare, is possible. This case highlights the potential for spontaneous closure of a reopened MH, suggesting that a wait-and-watch approach may be worthwhile before deciding on further surgery.
{"title":"The Hole Story: Spontaneous Closure after Reopening.","authors":"Marcelo Zas, Marcos Mendaro, Guido Bregliano, Alejandro Javier Aguilar, Maria Carolina Pozzoni, Ashish Sharma","doi":"10.1159/000547739","DOIUrl":"10.1159/000547739","url":null,"abstract":"<p><strong>Introduction: </strong>We report a case of spontaneous closure after early reopening of a previously successfully treated macular hole (MH).</p><p><strong>Case presentation: </strong>A 73-year-old patient presented with visual acuity of 20/100 and complained of defective vision. Examination and investigations confirmed a full-thickness MH. Surgical intervention led to complete closure of the hole on OCT within 5 days. However, the patient reported recurrence of symptoms 2 weeks later, and reopening of the hole was diagnosed. Re-surgery was advised, but surprisingly, the hole spontaneously closed by the seventh week and remained closed at the last follow-up (6 months post-surgery), with visual acuity improving to 20/50.</p><p><strong>Conclusions: </strong>Reopening of an MH can occur months or years after successful treatment, and the underlying reasons remain unclear. While reoperation is often indicated, spontaneous healing, although rare, is possible. This case highlights the potential for spontaneous closure of a reopened MH, suggesting that a wait-and-watch approach may be worthwhile before deciding on further surgery.</p>","PeriodicalId":9635,"journal":{"name":"Case Reports in Ophthalmology","volume":"16 1","pages":"615-620"},"PeriodicalIF":0.6,"publicationDate":"2025-08-04","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12503639/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"145249979","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 : 2025-07-28eCollection Date: 2025-01-01DOI: 10.1159/000547670
Adam Ayoub, Matthew Hartwig, Adam S Hassan
Introduction: Hydroxyapatite implants are widely used in orbital reconstruction due to their biocompatibility and capacity for osseointegration. Complications such as implant exposure or extrusion typically occur in the early postoperative period, with late-onset cases being exceedingly rare.
Case presentation: This report presents a 78-year-old female who developed hydroxyapatite implant extrusion 7 years after orbital volume augmentation for an anophthalmic socket. She presented with lower eyelid retraction, ectropion, redness, and a fistula in the inferior fornix. Surgical management included implant removal, placement of a dermis fat graft from the abdomen to restore orbital volume, and correction of eyelid deformities using a tarsal strip and full-thickness skin graft. Postoperatively, the patient achieved successful resolution of symptoms, restoration of orbital volume, and correction of eyelid position.
Conclusion: This case emphasizes the need for long-term surveillance following orbital reconstruction and highlights effective management strategies for rare delayed implant extrusion.
{"title":"Delayed Extrusion of Hydroxyapatite Cement Orbital Floor Implant: A Rare Case Report of Augmentation Failure.","authors":"Adam Ayoub, Matthew Hartwig, Adam S Hassan","doi":"10.1159/000547670","DOIUrl":"10.1159/000547670","url":null,"abstract":"<p><strong>Introduction: </strong>Hydroxyapatite implants are widely used in orbital reconstruction due to their biocompatibility and capacity for osseointegration. Complications such as implant exposure or extrusion typically occur in the early postoperative period, with late-onset cases being exceedingly rare.</p><p><strong>Case presentation: </strong>This report presents a 78-year-old female who developed hydroxyapatite implant extrusion 7 years after orbital volume augmentation for an anophthalmic socket. She presented with lower eyelid retraction, ectropion, redness, and a fistula in the inferior fornix. Surgical management included implant removal, placement of a dermis fat graft from the abdomen to restore orbital volume, and correction of eyelid deformities using a tarsal strip and full-thickness skin graft. Postoperatively, the patient achieved successful resolution of symptoms, restoration of orbital volume, and correction of eyelid position.</p><p><strong>Conclusion: </strong>This case emphasizes the need for long-term surveillance following orbital reconstruction and highlights effective management strategies for rare delayed implant extrusion.</p>","PeriodicalId":9635,"journal":{"name":"Case Reports in Ophthalmology","volume":"16 1","pages":"604-609"},"PeriodicalIF":0.6,"publicationDate":"2025-07-28","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12503602/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"145249797","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 : 2025-07-28eCollection Date: 2025-01-01DOI: 10.1159/000547671
Ibrahim Nagmeldin Hassan, Ghada Aljaili
Introduction: Neuro-retinitis is a rare ocular manifestation of disseminated tuberculosis (TB), often presenting with optic disc swelling and a macular star. Early diagnosis is essential to prevent permanent visual impairment, especially in TB-endemic areas where such presentations may be overlooked.
Case presentation: We report a case of a 38-year-old male who presented with a 2-week history of progressive, painless vision loss in the left eye. Fundoscopy revealed optic disc swelling and a macular star, suggestive of neuro-retinitis. Systemic evaluation, including chest imaging and immunologic testing, confirmed disseminated TB. Other potential infectious and autoimmune causes were excluded. The patient was initiated on standard anti-TB therapy (isoniazid, rifampicin, pyrazinamide, and ethambutol) along with oral corticosteroids. At 6-week follow-up, his visual acuity improved, and optic disc swelling had decreased.
Conclusion: Neuro-retinitis may be the initial presenting feature of disseminated TB. Clinicians should consider TB in the differential diagnosis of neuro-retinitis, particularly in high-prevalence settings. Early systemic evaluation and prompt initiation of anti-TB therapy can lead to favorable visual and systemic outcomes.
{"title":"Ocular Involvement in Disseminated Tuberculosis: A Case of Neuro-Retinitis.","authors":"Ibrahim Nagmeldin Hassan, Ghada Aljaili","doi":"10.1159/000547671","DOIUrl":"10.1159/000547671","url":null,"abstract":"<p><strong>Introduction: </strong>Neuro-retinitis is a rare ocular manifestation of disseminated tuberculosis (TB), often presenting with optic disc swelling and a macular star. Early diagnosis is essential to prevent permanent visual impairment, especially in TB-endemic areas where such presentations may be overlooked.</p><p><strong>Case presentation: </strong>We report a case of a 38-year-old male who presented with a 2-week history of progressive, painless vision loss in the left eye. Fundoscopy revealed optic disc swelling and a macular star, suggestive of neuro-retinitis. Systemic evaluation, including chest imaging and immunologic testing, confirmed disseminated TB. Other potential infectious and autoimmune causes were excluded. The patient was initiated on standard anti-TB therapy (isoniazid, rifampicin, pyrazinamide, and ethambutol) along with oral corticosteroids. At 6-week follow-up, his visual acuity improved, and optic disc swelling had decreased.</p><p><strong>Conclusion: </strong>Neuro-retinitis may be the initial presenting feature of disseminated TB. Clinicians should consider TB in the differential diagnosis of neuro-retinitis, particularly in high-prevalence settings. Early systemic evaluation and prompt initiation of anti-TB therapy can lead to favorable visual and systemic outcomes.</p>","PeriodicalId":9635,"journal":{"name":"Case Reports in Ophthalmology","volume":"16 1","pages":"610-614"},"PeriodicalIF":0.6,"publicationDate":"2025-07-28","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12503600/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"145249912","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 : 2025-07-28eCollection Date: 2025-01-01DOI: 10.1159/000547601
Yanting Li, Bin Chen, Yunfeng Lu
Introduction: We reported a case of late-onset opacification of a hydrophilic acrylic intraocular lens (IOL) that developed years after uneventful phacoemulsification cataract surgery. Remarkably, the calcification-related opacification was successfully reversed in vitro using ethylenediaminetetraacetic acid (EDTA) solution.
Case presentation: An 86-year-old male presented with a 2-year history of progressive visual decline in the right eye (best-corrected visual acuity: 2/200). Slit-lamp examination revealed posterior capsular opacification (PCO), and B-scan ultrasonography showed dense vitreous opacities, precluding fundus and optical coherence tomography (OCT) examinations. He had undergone cataract surgery with implantation of a Rayner monofocal IOL 10 years ago. As for the treatment of vitreous opacities, PCO, as well as investigating fundus diseases, the pars plana vitrectomy was performed. After PCO removal, fundus visualization remained poor. Intraoperative inspection revealed diffuse opacification of the IOL, which was subsequently explanted. Following removal of IOL, fundus examination revealed non-proliferative diabetic retinopathy and asteroid hyalosis. In vitro, the IOL exhibited homogeneous opacification resistant to mechanical cleaning and soaking in sterile water for 1 week. However, immersion in 4% EDTA gradually restored full optical clarity over the course of 1 month. Scanning electron microscopy (SEM) demonstrated small subsurface pits suggestive of prior crystal deposition. Energy-dispersive X-ray spectroscopy detected mainly carbon (C) and oxygen (O), without significant evidence of calcium (Ca), silicon (Si), or phosphorus (P).
Conclusions: This case highlighted the potential reversibility of hydrophilic IOL opacification through EDTA chelation, suggesting a promising direction for therapeutic management of IOL calcification in vivo in the future.
{"title":"Reversible Opacification of Intraocular Lens in vitro: Case Report.","authors":"Yanting Li, Bin Chen, Yunfeng Lu","doi":"10.1159/000547601","DOIUrl":"10.1159/000547601","url":null,"abstract":"<p><strong>Introduction: </strong>We reported a case of late-onset opacification of a hydrophilic acrylic intraocular lens (IOL) that developed years after uneventful phacoemulsification cataract surgery. Remarkably, the calcification-related opacification was successfully reversed in vitro using ethylenediaminetetraacetic acid (EDTA) solution.</p><p><strong>Case presentation: </strong>An 86-year-old male presented with a 2-year history of progressive visual decline in the right eye (best-corrected visual acuity: 2/200). Slit-lamp examination revealed posterior capsular opacification (PCO), and B-scan ultrasonography showed dense vitreous opacities, precluding fundus and optical coherence tomography (OCT) examinations. He had undergone cataract surgery with implantation of a Rayner monofocal IOL 10 years ago. As for the treatment of vitreous opacities, PCO, as well as investigating fundus diseases, the pars plana vitrectomy was performed. After PCO removal, fundus visualization remained poor. Intraoperative inspection revealed diffuse opacification of the IOL, which was subsequently explanted. Following removal of IOL, fundus examination revealed non-proliferative diabetic retinopathy and asteroid hyalosis. In vitro, the IOL exhibited homogeneous opacification resistant to mechanical cleaning and soaking in sterile water for 1 week. However, immersion in 4% EDTA gradually restored full optical clarity over the course of 1 month. Scanning electron microscopy (SEM) demonstrated small subsurface pits suggestive of prior crystal deposition. Energy-dispersive X-ray spectroscopy detected mainly carbon (C) and oxygen (O), without significant evidence of calcium (Ca), silicon (Si), or phosphorus (P).</p><p><strong>Conclusions: </strong>This case highlighted the potential reversibility of hydrophilic IOL opacification through EDTA chelation, suggesting a promising direction for therapeutic management of IOL calcification in vivo in the future.</p>","PeriodicalId":9635,"journal":{"name":"Case Reports in Ophthalmology","volume":"16 1","pages":"590-596"},"PeriodicalIF":0.6,"publicationDate":"2025-07-28","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12503548/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"145249944","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 : 2025-07-24eCollection Date: 2025-01-01DOI: 10.1159/000547429
Honggai Yan, Li Li, Yanhui Cui
Introduction: Ocular tuberculosis (OTB) can occur in any part of the eye. We present a rare case of an 11-year-old girl with tuberculosis-related neuroretinitis and central retinal vein occlusion (CRVO).
Case presentation: The patient presented due to vision loss in the right eye. An examination detected edema of the optic disc with peripapillary and scattered retinal hemorrhages around the tortuous and dilated retinal veins in the right eye. Optical coherence tomography showed macular edema and subretinal fluid in the right eye. Fundus fluorescein angiography indicated hyper-fluorescence staining of the optic disc and tortuous and dilated retinal veins with wall staining and minor leakage in the right eye. Tuberculin skin test and interferon-gamma release assay were positive. The patient's vision was improved after anti-tuberculosis and systemic glucocorticoid therapy. A literature search found reports on only 5 adult patients with a similar presentation.
Conclusion: Tuberculosis-related neuroretinitis and CRVO in children are rare. Early diagnosis and treatment can somewhat restore the lost vision.
{"title":"Tuberculosis-Related Neuroretinitis and Central Retinal Vein Occlusion in a Child, and a Literature Review.","authors":"Honggai Yan, Li Li, Yanhui Cui","doi":"10.1159/000547429","DOIUrl":"10.1159/000547429","url":null,"abstract":"<p><strong>Introduction: </strong>Ocular tuberculosis (OTB) can occur in any part of the eye. We present a rare case of an 11-year-old girl with tuberculosis-related neuroretinitis and central retinal vein occlusion (CRVO).</p><p><strong>Case presentation: </strong>The patient presented due to vision loss in the right eye. An examination detected edema of the optic disc with peripapillary and scattered retinal hemorrhages around the tortuous and dilated retinal veins in the right eye. Optical coherence tomography showed macular edema and subretinal fluid in the right eye. Fundus fluorescein angiography indicated hyper-fluorescence staining of the optic disc and tortuous and dilated retinal veins with wall staining and minor leakage in the right eye. Tuberculin skin test and interferon-gamma release assay were positive. The patient's vision was improved after anti-tuberculosis and systemic glucocorticoid therapy. A literature search found reports on only 5 adult patients with a similar presentation.</p><p><strong>Conclusion: </strong>Tuberculosis-related neuroretinitis and CRVO in children are rare. Early diagnosis and treatment can somewhat restore the lost vision.</p>","PeriodicalId":9635,"journal":{"name":"Case Reports in Ophthalmology","volume":"16 1","pages":"597-603"},"PeriodicalIF":0.6,"publicationDate":"2025-07-24","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12503614/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"145249899","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 : 2025-07-18eCollection Date: 2025-01-01DOI: 10.1159/000547495
Brendan K Tao, Jonathan Micieli
Introduction: Myelin oligodendrocyte glycoprotein antibody disease (MOGAD) is a more recently characterized neuroinflammatory condition which has been implicated with optic neuritis (ON). While the potential causes of central retinal artery occlusion (CRAO) are several, neuroinflammatory causes should not be excluded from the differential diagnosis. Our unusual case presents a patient with prior history of MOGAD-ON and venous stasis retinopathy (VSR) in their right eye, who, in their current presentation, developed a CRAO in their left eye.
Case presentation: A 43-year-old female with a history of concurrent VSR and MOGAD-ON (diagnosed 6 years prior and managed with long-term immunosuppression and oral steroids) in her right eye presented with a five-hour history of pain and blurry vision in her left eye. Examination of the left eye revealed a visual acuity (VA) of 20/80, a cherry red spot (without a visualized embolus), and a new generalized visual field defect. Optical coherence tomography (OCT) revealed diffuse retinal edema in her left eye with multiple layers of retinal thinning. She was referred to stroke neurology and continued oral steroid therapy. At 6-month follow-up, her VA had markedly improved to 20/30 in her left eye, and she had stable OCT parameters.
Conclusion: This unique case demonstrates sequential VSR and CRAO in opposite eyes, which were notable for their association with recurrent MOGAD-ON. Further research is needed to better elucidate the role of MOGAD in vascular occlusive events and whether this relationship may be arteritic in etiology.
{"title":"Central Retinal Artery Occlusion Associated with Myelin Oligodendrocyte Glycoprotein Antibody Disease: A Case Report.","authors":"Brendan K Tao, Jonathan Micieli","doi":"10.1159/000547495","DOIUrl":"10.1159/000547495","url":null,"abstract":"<p><strong>Introduction: </strong>Myelin oligodendrocyte glycoprotein antibody disease (MOGAD) is a more recently characterized neuroinflammatory condition which has been implicated with optic neuritis (ON). While the potential causes of central retinal artery occlusion (CRAO) are several, neuroinflammatory causes should not be excluded from the differential diagnosis. Our unusual case presents a patient with prior history of MOGAD-ON and venous stasis retinopathy (VSR) in their right eye, who, in their current presentation, developed a CRAO in their left eye.</p><p><strong>Case presentation: </strong>A 43-year-old female with a history of concurrent VSR and MOGAD-ON (diagnosed 6 years prior and managed with long-term immunosuppression and oral steroids) in her right eye presented with a five-hour history of pain and blurry vision in her left eye. Examination of the left eye revealed a visual acuity (VA) of 20/80, a cherry red spot (without a visualized embolus), and a new generalized visual field defect. Optical coherence tomography (OCT) revealed diffuse retinal edema in her left eye with multiple layers of retinal thinning. She was referred to stroke neurology and continued oral steroid therapy. At 6-month follow-up, her VA had markedly improved to 20/30 in her left eye, and she had stable OCT parameters.</p><p><strong>Conclusion: </strong>This unique case demonstrates sequential VSR and CRAO in opposite eyes, which were notable for their association with recurrent MOGAD-ON. Further research is needed to better elucidate the role of MOGAD in vascular occlusive events and whether this relationship may be arteritic in etiology.</p>","PeriodicalId":9635,"journal":{"name":"Case Reports in Ophthalmology","volume":"16 1","pages":"575-581"},"PeriodicalIF":0.6,"publicationDate":"2025-07-18","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12503543/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"145250011","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 : 2025-07-07eCollection Date: 2025-01-01DOI: 10.1159/000546953
Anna Bonde Scheel-Bech, Marie Louise Roed Rasmussen, Steffen Heegaard, Ida Maria Schmidt, Jakob Stensballe
Introduction: This case report presents the clinical, histological, and genetic characteristics of a patient with ligneous conjunctivitis and to provide a long-term treatment schedule, using cryo-depleted plasma infusions in combination with topical cyclosporine as a successful therapeutic option.
Case presentation: A 12-year-old Turkish girl experienced years of ocular itching, irritation, and increased light sensitivity. Initial clinical examination revealed the presence of pseudomembranes and giant papillae on the tarsal conjunctivae of both eyes. The biopsy of a membrane from the left eye was taken for histological examination. Manifestations of severe comorbidity since infancy (congenital hydrocephalus, asthma, recurrent respiratory infections, and impaired wound healing) and parental consanguinity prompted a genetic evaluation. The pseudomembranes were composed of fibrin, granulation tissue, and inflammatory cells consistent with the diagnosis of ligneous conjunctivitis. The genetic analysis revealed homozygosity for a pathogenic missense variant in plasminogen (PLG), causing PLG deficiency. Due to severe systemic complications, the patient was successfully treated with cryo-depleted plasma infusions and cyclosporine eye drops, resulting in the complete remission of the ligneous conjunctivitis.
Conclusion: This case report highlights the importance of considering PLG deficiency in patients with recurring chronic conjunctivitis. Long-term treatment with cryo-depleted plasma infusions in combination with short-term use of topical cyclosporine may provide an efficient therapeutic option for ligneous conjunctivitis in patients with PLG deficiency.
{"title":"Cryo-Depleted Plasma Infusions for Ligneous Conjunctivitis.","authors":"Anna Bonde Scheel-Bech, Marie Louise Roed Rasmussen, Steffen Heegaard, Ida Maria Schmidt, Jakob Stensballe","doi":"10.1159/000546953","DOIUrl":"10.1159/000546953","url":null,"abstract":"<p><strong>Introduction: </strong>This case report presents the clinical, histological, and genetic characteristics of a patient with ligneous conjunctivitis and to provide a long-term treatment schedule, using cryo-depleted plasma infusions in combination with topical cyclosporine as a successful therapeutic option.</p><p><strong>Case presentation: </strong>A 12-year-old Turkish girl experienced years of ocular itching, irritation, and increased light sensitivity. Initial clinical examination revealed the presence of pseudomembranes and giant papillae on the tarsal conjunctivae of both eyes. The biopsy of a membrane from the left eye was taken for histological examination. Manifestations of severe comorbidity since infancy (congenital hydrocephalus, asthma, recurrent respiratory infections, and impaired wound healing) and parental consanguinity prompted a genetic evaluation. The pseudomembranes were composed of fibrin, granulation tissue, and inflammatory cells consistent with the diagnosis of ligneous conjunctivitis. The genetic analysis revealed homozygosity for a pathogenic missense variant in plasminogen (PLG), causing PLG deficiency. Due to severe systemic complications, the patient was successfully treated with cryo-depleted plasma infusions and cyclosporine eye drops, resulting in the complete remission of the ligneous conjunctivitis.</p><p><strong>Conclusion: </strong>This case report highlights the importance of considering PLG deficiency in patients with recurring chronic conjunctivitis. Long-term treatment with cryo-depleted plasma infusions in combination with short-term use of topical cyclosporine may provide an efficient therapeutic option for ligneous conjunctivitis in patients with PLG deficiency.</p>","PeriodicalId":9635,"journal":{"name":"Case Reports in Ophthalmology","volume":"16 1","pages":"542-550"},"PeriodicalIF":0.6,"publicationDate":"2025-07-07","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12331225/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144798294","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 : 2025-07-07eCollection Date: 2025-01-01DOI: 10.1159/000546758
Li Wang, Yingqian Tan, Fang Li, Yue Liang, Guangui Chen, Amy Michelle Huang, Lina Li, Junming Wang, Zhiping Liu
Introduction: Mucoceles are benign, slow-growing cystic formations located within the paranasal sinuses, caused by complete ostial obstruction and accumulation of mucous secretions. Here, we report a case of a patient who initially presented with unilateral painless visual impairment and was ultimately diagnosed with bilateral sphenoid sinus mucoceles (SSMs) after two hospitalizations.
Case presentation: A 67-year-old woman presented with a 7-day history of decreased vision in her left eye. She denied any orbital pain, headache, or restricted eye movement and was diagnosed with retrobulbar ischemic optic neuropathy on the first admission. After drug treatment, the visual acuity of patient improved significantly, but 3 months after discharge, the patient was hospitalized again due to recurrent vision loss accompanied by left orbital pain and left temporal pulsatile headaches. After multiple imaging examinations, the patient was ultimately diagnosed with SSMs and her visual acuity was restored after surgical treatment.
Conclusions: The majority of SSMs are associated with ocular symptoms, with only a minority presenting solely with unilateral or bilateral vision loss, as exemplified in this case. Therefore, understanding the clinical features of visual disturbances secondary to SSMs is crucial to aiding more prompt diagnosis and treatment.
{"title":"Unilateral Painless Visual Loss in Sphenoid Mucoceles with Optic Neuropathy.","authors":"Li Wang, Yingqian Tan, Fang Li, Yue Liang, Guangui Chen, Amy Michelle Huang, Lina Li, Junming Wang, Zhiping Liu","doi":"10.1159/000546758","DOIUrl":"10.1159/000546758","url":null,"abstract":"<p><strong>Introduction: </strong>Mucoceles are benign, slow-growing cystic formations located within the paranasal sinuses, caused by complete ostial obstruction and accumulation of mucous secretions. Here, we report a case of a patient who initially presented with unilateral painless visual impairment and was ultimately diagnosed with bilateral sphenoid sinus mucoceles (SSMs) after two hospitalizations.</p><p><strong>Case presentation: </strong>A 67-year-old woman presented with a 7-day history of decreased vision in her left eye. She denied any orbital pain, headache, or restricted eye movement and was diagnosed with retrobulbar ischemic optic neuropathy on the first admission. After drug treatment, the visual acuity of patient improved significantly, but 3 months after discharge, the patient was hospitalized again due to recurrent vision loss accompanied by left orbital pain and left temporal pulsatile headaches. After multiple imaging examinations, the patient was ultimately diagnosed with SSMs and her visual acuity was restored after surgical treatment.</p><p><strong>Conclusions: </strong>The majority of SSMs are associated with ocular symptoms, with only a minority presenting solely with unilateral or bilateral vision loss, as exemplified in this case. Therefore, understanding the clinical features of visual disturbances secondary to SSMs is crucial to aiding more prompt diagnosis and treatment.</p>","PeriodicalId":9635,"journal":{"name":"Case Reports in Ophthalmology","volume":"16 1","pages":"559-566"},"PeriodicalIF":0.6,"publicationDate":"2025-07-07","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12342703/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144834083","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: Inflammation of the prelaminar compartment of the optic nerve involves the most anterior segment of the nerve. It can occur in isolation, in the setting of immunological or demyelinating disorders.
Case presentation: We present two 32- and 41-year-old female patients with sequential bilateral isolated prelaminar optic neuritis with atypical features. In both instances, plasma exchange (PLEX) was initiated for the second affected eye after corticosteroids proved ineffective, resulting in excellent visual recovery. Both patients lost vision in the fellow eye due to a similar attack that was also refractory to steroids. Our cases did not show any associated demyelinating lesions on imaging and did not show biomarkers such as aquaporin-4 or myelin oligodendrocyte glycoprotein (MOG) antibody in serum workup. They were followed for more than 5 years with stable vision in the only eye.
Conclusion: In cases of isolated severe progressive prelaminar optic nerve inflammation refractory to steroid treatment, PLEX can be helpful, even if workup is unremarkable for neuromyelitis optica or other systemic and neurologic conditions.
{"title":"Vision Restoration of Steroid-Refractory Prelaminar Optic Nerve Inflammation with Plasmapheresis: Report of Two Cases and Literature Review.","authors":"Nazanin Ebrahimiadib, Ghazal Ghochani, Samira Yadegari, Bryce E Buchowicz, Sahel Soltani Shahgholi, Afsaneh Azarkish, Hanieh Niktinat, Hamid Riazi-Esfahani, Narges Hassanpoor","doi":"10.1159/000547225","DOIUrl":"10.1159/000547225","url":null,"abstract":"<p><strong>Introduction: </strong>Inflammation of the prelaminar compartment of the optic nerve involves the most anterior segment of the nerve. It can occur in isolation, in the setting of immunological or demyelinating disorders.</p><p><strong>Case presentation: </strong>We present two 32- and 41-year-old female patients with sequential bilateral isolated prelaminar optic neuritis with atypical features. In both instances, plasma exchange (PLEX) was initiated for the second affected eye after corticosteroids proved ineffective, resulting in excellent visual recovery. Both patients lost vision in the fellow eye due to a similar attack that was also refractory to steroids. Our cases did not show any associated demyelinating lesions on imaging and did not show biomarkers such as aquaporin-4 or myelin oligodendrocyte glycoprotein (MOG) antibody in serum workup. They were followed for more than 5 years with stable vision in the only eye.</p><p><strong>Conclusion: </strong>In cases of isolated severe progressive prelaminar optic nerve inflammation refractory to steroid treatment, PLEX can be helpful, even if workup is unremarkable for neuromyelitis optica or other systemic and neurologic conditions.</p>","PeriodicalId":9635,"journal":{"name":"Case Reports in Ophthalmology","volume":"16 1","pages":"567-574"},"PeriodicalIF":0.6,"publicationDate":"2025-07-04","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12503469/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"145249962","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 : 2025-07-02eCollection Date: 2025-01-01DOI: 10.1159/000545489
Alba Gómez-Benlloch, Julia N Widmer-Pintos, Consuelo Arnaldos-López
Introduction: Orbital myositis (OM) is an inflammatory condition of the extraocular muscles, often idiopathic but occasionally associated with systemic diseases or malignancies. Secondary OM due to metastatic disease is rare. We report a case of OM as the initial manifestation of metastatic renal cell carcinoma, emphasizing the need for comprehensive evaluation of atypical ocular presentations.
Case presentation: An 81-year-old male with a history of age-related macular degeneration presented with acute onset of pain and restricted movement in his left eye. Computed tomography imaging revealed an osteolytic lesion in the left sphenoid bone, causing reactive myositis. Further systemic evaluation identified a left renal mass with evidence of pulmonary and skeletal metastases. A core needle biopsy confirmed the diagnosis of metastatic renal cell carcinoma. Given the advanced disease stage, the patient was managed with palliative treatment. Despite medical interventions, he succumbed to the disease 6 months after symptom onset.
Conclusion: This case underscores the significance of considering malignancy in the differential diagnosis of OM, particularly in elderly patients with atypical ocular symptoms. Early recognition and systemic evaluation are crucial for timely diagnosis and management of underlying malignancies presenting with orbital involvement.
{"title":"Reactive Orbital Myositis as a First Manifestation of Renal Cell Carcinoma.","authors":"Alba Gómez-Benlloch, Julia N Widmer-Pintos, Consuelo Arnaldos-López","doi":"10.1159/000545489","DOIUrl":"10.1159/000545489","url":null,"abstract":"<p><strong>Introduction: </strong>Orbital myositis (OM) is an inflammatory condition of the extraocular muscles, often idiopathic but occasionally associated with systemic diseases or malignancies. Secondary OM due to metastatic disease is rare. We report a case of OM as the initial manifestation of metastatic renal cell carcinoma, emphasizing the need for comprehensive evaluation of atypical ocular presentations.</p><p><strong>Case presentation: </strong>An 81-year-old male with a history of age-related macular degeneration presented with acute onset of pain and restricted movement in his left eye. Computed tomography imaging revealed an osteolytic lesion in the left sphenoid bone, causing reactive myositis. Further systemic evaluation identified a left renal mass with evidence of pulmonary and skeletal metastases. A core needle biopsy confirmed the diagnosis of metastatic renal cell carcinoma. Given the advanced disease stage, the patient was managed with palliative treatment. Despite medical interventions, he succumbed to the disease 6 months after symptom onset.</p><p><strong>Conclusion: </strong>This case underscores the significance of considering malignancy in the differential diagnosis of OM, particularly in elderly patients with atypical ocular symptoms. Early recognition and systemic evaluation are crucial for timely diagnosis and management of underlying malignancies presenting with orbital involvement.</p>","PeriodicalId":9635,"journal":{"name":"Case Reports in Ophthalmology","volume":"16 1","pages":"551-558"},"PeriodicalIF":0.6,"publicationDate":"2025-07-02","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12338982/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144820575","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}