Pub Date : 2025-06-24eCollection Date: 2025-01-01DOI: 10.1159/000546567
Zainab Rustam, Sarah Aman, Nakul Singh, Rose Tan, Amir H Kashani, Peter A Campochiaro
Introduction: Alport syndrome is an inherited disease caused by mutations in COL4A5, COLA3, or COL4A4 resulting in kidney failure, hearing loss, and ocular symptoms. We report a patient with Alport syndrome who has a "stair-case/honeycomb" maculopathy, a rare but distinctive finding in this disease.
Case presentation: A 53-year-old man with Alport syndrome was referred for gradual decrease in vision. His ocular history was remarkable for intraocular lens implantation secondary to lenticonus in each eye. Fundus photography showed rare white dots in the temporal mid-periphery in each eye and fundus autofluorescence was normal. Optical coherence tomography (OCT) B-scans through the fovea showed irregular thinning of the inner retina with peaks and valleys in the macula of each eye. The ellipsoid zone was intact except for mild patchiness centrally. En face retinal structural OCT angiography (OCTA) images showed a mosaic-like honeycomb pattern in the macular region in both eyes, with hyporeflective depressions in areas of focal retinal atrophy. Retinal OCTA scans showed irregular foveal avascular zone (FAZ) areas with capillaries crossing the FAZ in the left eye, corresponding to islands of preserved retinal tissue. There was predominance of capillaries in the deeper retinal layers centrally.
Conclusion: While severe irregular thinning of the macula is not a common feature in Alport syndrome, when it is present in patients who have not been previously diagnosed, particularly in patients with renal disease, it should suggest the diagnosis of Alport syndrome. Its occurrence can be the cause of vision loss which is not commonly associated with Alport central maculopathy.
{"title":"Stair-Case/Honeycomb Maculopathy in Alport Syndrome: A Case Report.","authors":"Zainab Rustam, Sarah Aman, Nakul Singh, Rose Tan, Amir H Kashani, Peter A Campochiaro","doi":"10.1159/000546567","DOIUrl":"10.1159/000546567","url":null,"abstract":"<p><strong>Introduction: </strong>Alport syndrome is an inherited disease caused by mutations in COL4A5, COLA3, or COL4A4 resulting in kidney failure, hearing loss, and ocular symptoms. We report a patient with Alport syndrome who has a \"stair-case/honeycomb\" maculopathy, a rare but distinctive finding in this disease.</p><p><strong>Case presentation: </strong>A 53-year-old man with Alport syndrome was referred for gradual decrease in vision. His ocular history was remarkable for intraocular lens implantation secondary to lenticonus in each eye. Fundus photography showed rare white dots in the temporal mid-periphery in each eye and fundus autofluorescence was normal. Optical coherence tomography (OCT) B-scans through the fovea showed irregular thinning of the inner retina with peaks and valleys in the macula of each eye. The ellipsoid zone was intact except for mild patchiness centrally. En face retinal structural OCT angiography (OCTA) images showed a mosaic-like honeycomb pattern in the macular region in both eyes, with hyporeflective depressions in areas of focal retinal atrophy. Retinal OCTA scans showed irregular foveal avascular zone (FAZ) areas with capillaries crossing the FAZ in the left eye, corresponding to islands of preserved retinal tissue. There was predominance of capillaries in the deeper retinal layers centrally.</p><p><strong>Conclusion: </strong>While severe irregular thinning of the macula is not a common feature in Alport syndrome, when it is present in patients who have not been previously diagnosed, particularly in patients with renal disease, it should suggest the diagnosis of Alport syndrome. Its occurrence can be the cause of vision loss which is not commonly associated with Alport central maculopathy.</p>","PeriodicalId":9635,"journal":{"name":"Case Reports in Ophthalmology","volume":"16 1","pages":"496-502"},"PeriodicalIF":0.5,"publicationDate":"2025-06-24","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12263145/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144641901","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 : 2025-06-23eCollection Date: 2025-01-01DOI: 10.1159/000546622
Vignesh J Krishnan, Sam Karimaghaei, Sami H Uwaydat
Introduction: This case report describes a patient whose misdiagnosis of foveal hypoplasia was uncovered by optical coherence tomography angiography (OCTA) findings that suggested the presence of an epiretinal membrane (ERM) over foveal hypoplasia.
Case presentation: A 67-year-old man with no significant past medical history was referred to our retina clinic with a diagnosis of foveal hypoplasia. He had been experiencing significant vision loss for more than 1 year. OCT demonstrated absence of the foveal depression in both eyes. A subtle ERM was identified in the left eye OCT, but the presence of an ERM in the right eye OCT was equivocal. As such, it was unclear whether flattening of the fovea was attributable to hypoplasia or ERM based on OCT alone. This prompted further investigation with OCTA, which showed the presence of the FAZ in both eyes. The diagnosis of stage 2 ERM OU was made based on OCTA findings. The patient underwent cataract extraction with intraocular lens implantation, pars plana vitrectomy, and ERM peel, which resulted in improvement of visual symptoms and visual acuity. Follow-up OCT showed normalization of the foveal pit in the right eye greater than the left eye.
Conclusion: This case demonstrates the importance of utilization of OCTA in differentiating true foveal hypoplasia from this foveal "pseudo-hypoplasia" exhibited by our patient.
{"title":"A Case of Epiretinal Membrane Masquerading as Foveal Hypoplasia Uncovered by Optical Coherence Tomography Angiography.","authors":"Vignesh J Krishnan, Sam Karimaghaei, Sami H Uwaydat","doi":"10.1159/000546622","DOIUrl":"10.1159/000546622","url":null,"abstract":"<p><strong>Introduction: </strong>This case report describes a patient whose misdiagnosis of foveal hypoplasia was uncovered by optical coherence tomography angiography (OCTA) findings that suggested the presence of an epiretinal membrane (ERM) over foveal hypoplasia.</p><p><strong>Case presentation: </strong>A 67-year-old man with no significant past medical history was referred to our retina clinic with a diagnosis of foveal hypoplasia. He had been experiencing significant vision loss for more than 1 year. OCT demonstrated absence of the foveal depression in both eyes. A subtle ERM was identified in the left eye OCT, but the presence of an ERM in the right eye OCT was equivocal. As such, it was unclear whether flattening of the fovea was attributable to hypoplasia or ERM based on OCT alone. This prompted further investigation with OCTA, which showed the presence of the FAZ in both eyes. The diagnosis of stage 2 ERM OU was made based on OCTA findings. The patient underwent cataract extraction with intraocular lens implantation, pars plana vitrectomy, and ERM peel, which resulted in improvement of visual symptoms and visual acuity. Follow-up OCT showed normalization of the foveal pit in the right eye greater than the left eye.</p><p><strong>Conclusion: </strong>This case demonstrates the importance of utilization of OCTA in differentiating true foveal hypoplasia from this foveal \"pseudo-hypoplasia\" exhibited by our patient.</p>","PeriodicalId":9635,"journal":{"name":"Case Reports in Ophthalmology","volume":"16 1","pages":"503-509"},"PeriodicalIF":0.5,"publicationDate":"2025-06-23","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12263143/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144641899","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: Intrapapillary hemorrhage with adjacent peripapillary subretinal hemorrhage (IHAPSH) is a syndrome associated with intrapapillary hemorrhage and peripapillary subretinal hemorrhage that commonly occurs in myopic women. There have been no reports of patients younger than 10 years. We report a case of typical IHAPSH that occurred in a 9-year-old girl in the advanced stages of myopia.
Case presentation: A 9-year-old girl presented with gradually decreasing uncorrected visual acuity in the left eye over the past year, without complaints of floaters or blurred vision. Uncorrected visual acuity was 20/16 in the right eye and approximately 20/285 in the left eye, improving to 20/16 with -4.50 diopters sphere and -0.50 diopters cylinder at 165°. Ophthalmoscopic examination revealed intrapapillary hemorrhage and nasal peripapillary subretinal hemorrhage in her left eye. The left eye had a small, tilted papilla. Fluorescein angiography showed no leakage from the papilla. The length of the ocular axis was elongated to 24.87 mm in the left, compared to 23.15 mm in the right. We diagnosed IHAPSH and decided to follow the patient. After 8 weeks, the hemorrhage was almost completely absorbed. No recurrence has been observed since then.
Conclusion: To our knowledge, this is the youngest reported case of IHAPSH. This case underscores the potential contribution of optic disc morphology and progressive myopia to IHAPSH pathogenesis and highlights the importance of considering this condition in the differential diagnosis of optic disc hemorrhage in school-aged children.
{"title":"Intrapapillary Hemorrhage with Adjacent Peripapillary Subretinal Hemorrhage in a 9-Year-Old Girl: A Case Report.","authors":"Chiaki Nakamura, Tadashi Matsumoto, Norihiro Watanabe, Shinichiro Kobayakawa","doi":"10.1159/000546984","DOIUrl":"10.1159/000546984","url":null,"abstract":"<p><strong>Introduction: </strong>Intrapapillary hemorrhage with adjacent peripapillary subretinal hemorrhage (IHAPSH) is a syndrome associated with intrapapillary hemorrhage and peripapillary subretinal hemorrhage that commonly occurs in myopic women. There have been no reports of patients younger than 10 years. We report a case of typical IHAPSH that occurred in a 9-year-old girl in the advanced stages of myopia.</p><p><strong>Case presentation: </strong>A 9-year-old girl presented with gradually decreasing uncorrected visual acuity in the left eye over the past year, without complaints of floaters or blurred vision. Uncorrected visual acuity was 20/16 in the right eye and approximately 20/285 in the left eye, improving to 20/16 with -4.50 diopters sphere and -0.50 diopters cylinder at 165°. Ophthalmoscopic examination revealed intrapapillary hemorrhage and nasal peripapillary subretinal hemorrhage in her left eye. The left eye had a small, tilted papilla. Fluorescein angiography showed no leakage from the papilla. The length of the ocular axis was elongated to 24.87 mm in the left, compared to 23.15 mm in the right. We diagnosed IHAPSH and decided to follow the patient. After 8 weeks, the hemorrhage was almost completely absorbed. No recurrence has been observed since then.</p><p><strong>Conclusion: </strong>To our knowledge, this is the youngest reported case of IHAPSH. This case underscores the potential contribution of optic disc morphology and progressive myopia to IHAPSH pathogenesis and highlights the importance of considering this condition in the differential diagnosis of optic disc hemorrhage in school-aged children.</p>","PeriodicalId":9635,"journal":{"name":"Case Reports in Ophthalmology","volume":"16 1","pages":"528-534"},"PeriodicalIF":0.5,"publicationDate":"2025-06-17","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12279344/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144682107","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 : 2025-06-14eCollection Date: 2025-01-01DOI: 10.1159/000546787
Megha R Kotecha, Varsha Manade, Jhimli Ta, Surbhi Chodvadiya
Introduction: Inert intraocular foreign body (IOFB) removal depends on the location, type of injury, composition, and size of IOFB and possible serious complications of intraocular surgery. Early management is crucial for better prognosis.
Case presentation: A 28-year-old male presented to the outpatient department after an alleged workplace accident. Initial assessment revealed significant diminution of vision, and on anterior segment examination with slit lamp, conjunctival congestion with no obvious entry point and no obvious scleral tear noted. Fundus examination by indirect ophthalmoscopy revealed vitreous hemorrhage, but the foreign body could not be localized due to extensive hazy media. Radiography of the orbit revealed an IOFB. The patient was managed surgically, and the intraretinal foreign body was removed using an intraocular magnet and intraocular forceps. The decision to remove the inert metal was considered because the patient had significant vision loss with vitreous hemorrhage.
Conclusion: Management of an intraretinal metallic foreign body using an intraocular magnet is a viable and effective approach. It allows precise removal with minimal retinal trauma, thus preserving visual function.
{"title":"Magnetic Extraction of an Intraretinal Foreign Body: A Case Report.","authors":"Megha R Kotecha, Varsha Manade, Jhimli Ta, Surbhi Chodvadiya","doi":"10.1159/000546787","DOIUrl":"10.1159/000546787","url":null,"abstract":"<p><strong>Introduction: </strong>Inert intraocular foreign body (IOFB) removal depends on the location, type of injury, composition, and size of IOFB and possible serious complications of intraocular surgery. Early management is crucial for better prognosis.</p><p><strong>Case presentation: </strong>A 28-year-old male presented to the outpatient department after an alleged workplace accident. Initial assessment revealed significant diminution of vision, and on anterior segment examination with slit lamp, conjunctival congestion with no obvious entry point and no obvious scleral tear noted. Fundus examination by indirect ophthalmoscopy revealed vitreous hemorrhage, but the foreign body could not be localized due to extensive hazy media. Radiography of the orbit revealed an IOFB. The patient was managed surgically, and the intraretinal foreign body was removed using an intraocular magnet and intraocular forceps. The decision to remove the inert metal was considered because the patient had significant vision loss with vitreous hemorrhage.</p><p><strong>Conclusion: </strong>Management of an intraretinal metallic foreign body using an intraocular magnet is a viable and effective approach. It allows precise removal with minimal retinal trauma, thus preserving visual function.</p>","PeriodicalId":9635,"journal":{"name":"Case Reports in Ophthalmology","volume":"16 1","pages":"515-520"},"PeriodicalIF":0.5,"publicationDate":"2025-06-14","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12266699/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144648680","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 : 2025-06-13eCollection Date: 2025-01-01DOI: 10.1159/000546935
Anny M S Cheng, Shailesh K Gupta, Tehilla Steiner, Samarth Shah, David T Y Yang, Elizabeth S Yang, Neil E Kanterman
Introduction: Low gestational birth weight is associated with increased incidence of retinopathy of prematurity (ROP). In recent years, intravitreal injection of anti-vascular endothelial growth factor (VEGF) has become more prevalent for ROP. Despite the demonstrated effectiveness following anti-VEGF injection, recurrence of ROP has been reported. A standardized treatment protocol for recurrent ROP following anti-VEGF monotherapy is still lacking, particularly for extremely low birth weight infants. This study reviews possible treatments for recurrent ROP and associated challenges.
Case presentation: We report a very low birth weight infant (500 g) with a recurrence of ROP after the initial intravitreal bevacizumab (IVB) injection, who was successfully treated with a repeat injection at a later date. No retinal detachment or recurrence was observed after a long-term follow-up of 36 months.
Conclusion: This case report highlights the complexity of managing ROP, particularly for recurrent ROP in very low birth weight infants. Premature infants with extremely low birth weight may benefit from a repeat injection of anti-VEGF after the initial IVB to treat the recurrence.
{"title":"Repeat Intravitreal Bevacizumab in a Very Low Birth Weight Infant with Recurrent ROP: 3-Year Follow-Up and Literature Review.","authors":"Anny M S Cheng, Shailesh K Gupta, Tehilla Steiner, Samarth Shah, David T Y Yang, Elizabeth S Yang, Neil E Kanterman","doi":"10.1159/000546935","DOIUrl":"10.1159/000546935","url":null,"abstract":"<p><strong>Introduction: </strong>Low gestational birth weight is associated with increased incidence of retinopathy of prematurity (ROP). In recent years, intravitreal injection of anti-vascular endothelial growth factor (VEGF) has become more prevalent for ROP. Despite the demonstrated effectiveness following anti-VEGF injection, recurrence of ROP has been reported. A standardized treatment protocol for recurrent ROP following anti-VEGF monotherapy is still lacking, particularly for extremely low birth weight infants. This study reviews possible treatments for recurrent ROP and associated challenges.</p><p><strong>Case presentation: </strong>We report a very low birth weight infant (500 g) with a recurrence of ROP after the initial intravitreal bevacizumab (IVB) injection, who was successfully treated with a repeat injection at a later date. No retinal detachment or recurrence was observed after a long-term follow-up of 36 months.</p><p><strong>Conclusion: </strong>This case report highlights the complexity of managing ROP, particularly for recurrent ROP in very low birth weight infants. Premature infants with extremely low birth weight may benefit from a repeat injection of anti-VEGF after the initial IVB to treat the recurrence.</p>","PeriodicalId":9635,"journal":{"name":"Case Reports in Ophthalmology","volume":"16 1","pages":"521-527"},"PeriodicalIF":0.5,"publicationDate":"2025-06-13","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12274061/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144673997","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 : 2025-06-12eCollection Date: 2025-01-01DOI: 10.1159/000546599
Natalie di Geronimo, Matilde Roda, Antonio Moramarco, Danilo Iannetta, Luigi Fontana
Introduction: The NewColorIris and BrightOcular implants were initially developed to address congenital iris defects. However, they found application for cosmetic purposes. Unfortunately, these implants are frequently linked to severe complications, including glaucoma, endothelial dysfunction, cataract development, and iris abnormalities. In this context, we present an unusual complication that manifested long after the implantation of the BrightOcular artificial iris.
Case presentation: A 28-year-old woman presented to our emergency room with blurred vision in both eyes. She had undergone bilateral cosmetic iris implantation (BrightOcular, Stellar Devices, New York, NY, USA) 6 years earlier in Tunisia. At the first examination, her best corrected visual acuity was hand motion in the right eye and 20/100 in the left eye, and intraocular pressure (IOP) was 45 mm Hg and 30 mm Hg, respectively. Despite the maximum-tolerated glaucoma medical treatment, the elevated IOP persisted, leading to the decision to perform bilateral sequential Baerveldt glaucoma drainage device implantation. However, she subsequently developed combined central retinal artery and vein occlusion in the right eye and hypotensive maculopathy in the left eye; the latter resolving within 1 month. Two months post-surgery, extensive neovascularization above the implant of the right eye was observed, and this was successfully treated with three sequential injections of bevacizumab.
Conclusion: Cosmetic iris implantation is associated with severe, sight-threatening complications. Herein, we describe a previously unreported case of angle neovascularization with new vessels growing over the artificial iris implant. The condition regressed after intravitreal anti-vascular endothelial growth factor injections.
介绍:NewColorIris和brightoular植入物最初是为了解决先天性虹膜缺陷而开发的。然而,他们发现了用于美容目的的应用。不幸的是,这些植入物经常与严重的并发症有关,包括青光眼、内皮功能障碍、白内障发展和虹膜异常。在这种情况下,我们提出了一个不寻常的并发症,表现在植入术后很长一段时间亮眼人工虹膜。病例介绍:一位28岁的女性因双眼视力模糊来到我们的急诊室。6年前,她在突尼斯接受了双侧美容虹膜植入手术(brightoular, Stellar Devices, New York, NY, USA)。首次检查时,其最佳矫正视力为右眼手部运动,左眼20/100,眼压(IOP)分别为45 mm Hg和30 mm Hg。尽管进行了最大耐受性的青光眼药物治疗,但IOP持续升高,导致决定进行双侧顺序Baerveldt青光眼引流装置植入术。然而,她随后出现右眼视网膜中央动脉和静脉合并闭塞和左眼低血压黄斑病变;后者在1个月内解决。术后两个月,观察到右眼植入物上方有广泛的新生血管形成,并通过连续三次注射贝伐单抗成功治疗。结论:美容性虹膜植入术存在严重的、威胁视力的并发症。在此,我们描述了一个以前未报道的角度新生血管与新血管生长在人工虹膜植入物。玻璃体内注射抗血管内皮生长因子后病情有所好转。
{"title":"Neovascular Proliferation over a Cosmetic Artificial Iris Implant.","authors":"Natalie di Geronimo, Matilde Roda, Antonio Moramarco, Danilo Iannetta, Luigi Fontana","doi":"10.1159/000546599","DOIUrl":"10.1159/000546599","url":null,"abstract":"<p><strong>Introduction: </strong>The NewColorIris and BrightOcular implants were initially developed to address congenital iris defects. However, they found application for cosmetic purposes. Unfortunately, these implants are frequently linked to severe complications, including glaucoma, endothelial dysfunction, cataract development, and iris abnormalities. In this context, we present an unusual complication that manifested long after the implantation of the BrightOcular artificial iris.</p><p><strong>Case presentation: </strong>A 28-year-old woman presented to our emergency room with blurred vision in both eyes. She had undergone bilateral cosmetic iris implantation (BrightOcular, Stellar Devices, New York, NY, USA) 6 years earlier in Tunisia. At the first examination, her best corrected visual acuity was hand motion in the right eye and 20/100 in the left eye, and intraocular pressure (IOP) was 45 mm Hg and 30 mm Hg, respectively. Despite the maximum-tolerated glaucoma medical treatment, the elevated IOP persisted, leading to the decision to perform bilateral sequential Baerveldt glaucoma drainage device implantation. However, she subsequently developed combined central retinal artery and vein occlusion in the right eye and hypotensive maculopathy in the left eye; the latter resolving within 1 month. Two months post-surgery, extensive neovascularization above the implant of the right eye was observed, and this was successfully treated with three sequential injections of bevacizumab.</p><p><strong>Conclusion: </strong>Cosmetic iris implantation is associated with severe, sight-threatening complications. Herein, we describe a previously unreported case of angle neovascularization with new vessels growing over the artificial iris implant. The condition regressed after intravitreal anti-vascular endothelial growth factor injections.</p>","PeriodicalId":9635,"journal":{"name":"Case Reports in Ophthalmology","volume":"16 1","pages":"482-488"},"PeriodicalIF":0.5,"publicationDate":"2025-06-12","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12240572/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144599548","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 : 2025-06-10eCollection Date: 2025-01-01DOI: 10.1159/000546662
Kasey M Cooper, Nina S Boal, Karolyn A Wanat, Laila Mossa-Basha, Kartikey Acharya, Natnaelle E Admassu, Gregory J Griepentrog
Introduction: Eyelid ulceration can result from malignancy, trauma, infection, or inflammation. Immunosuppressed patients, such as those on methotrexate (MTX) therapy, are at higher risk for opportunistic infections like Purpureocillium lilacinum and Epstein-Barr virus (EBV)-associated mucocutaneous ulcers (EBVMCU). We report a rare case of eyelid ulceration due to concurrent EBVMCU and P. lilacinum infection.
Case presentation: An 82-year-old female with rheumatoid arthritis on chronic MTX therapy presented with a 6-month history of an ulcerated left upper eyelid lesion. Cultures confirmed P. lilacinum, and histopathology revealed EBV-positive atypical B cells. The ulcer resolved after discontinuing MTX and treating with oral voriconazole.
Conclusion: This case highlights the need to consider both fungal and viral infections in immunosuppressed patients with unusual eyelid lesions. Correct diagnosis and appropriate treatment led to a successful outcome in this patient.
{"title":"Eyelid Ulcer in an Immunocompromised Patient.","authors":"Kasey M Cooper, Nina S Boal, Karolyn A Wanat, Laila Mossa-Basha, Kartikey Acharya, Natnaelle E Admassu, Gregory J Griepentrog","doi":"10.1159/000546662","DOIUrl":"10.1159/000546662","url":null,"abstract":"<p><strong>Introduction: </strong>Eyelid ulceration can result from malignancy, trauma, infection, or inflammation. Immunosuppressed patients, such as those on methotrexate (MTX) therapy, are at higher risk for opportunistic infections like <i>Purpureocillium lilacinum</i> and Epstein-Barr virus (EBV)-associated mucocutaneous ulcers (EBVMCU). We report a rare case of eyelid ulceration due to concurrent EBVMCU and <i>P. lilacinum</i> infection.</p><p><strong>Case presentation: </strong>An 82-year-old female with rheumatoid arthritis on chronic MTX therapy presented with a 6-month history of an ulcerated left upper eyelid lesion. Cultures confirmed <i>P. lilacinum</i>, and histopathology revealed EBV-positive atypical B cells. The ulcer resolved after discontinuing MTX and treating with oral voriconazole.</p><p><strong>Conclusion: </strong>This case highlights the need to consider both fungal and viral infections in immunosuppressed patients with unusual eyelid lesions. Correct diagnosis and appropriate treatment led to a successful outcome in this patient.</p>","PeriodicalId":9635,"journal":{"name":"Case Reports in Ophthalmology","volume":"16 1","pages":"510-514"},"PeriodicalIF":0.5,"publicationDate":"2025-06-10","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12263146/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144641900","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: Pyoderma gangrenosum is a rare form of neutrophilic dermatosis, with ocular involvement being atypical. We present a rare case of ulcerative colitis (UC) complicated by nodular scleritis and pyoderma gangrenosum, both occurring almost simultaneously.
Case presentation: A 55-year-old man with active UC initially presented to our hospital with anterior diffuse scleritis and a peripheral corneal ulcer in his left eye. The condition rapidly progressed to bilateral nodular scleritis. Concurrently, multiple painful abscesses developed on his trunk and head. A skin biopsy confirmed the diagnosis of pyoderma gangrenosum. Laboratory tests revealed elevated C-reactive protein and erythrocyte sedimentation rates, along with the increased proteinase-3 antineutrophil cytoplasmic antibody (PR3-ANCA). A comprehensive systemic evaluation ruled out vasculitis, and an elevated PR3-ANCA level was attributed to active UC. After excluding other potential causes of scleritis, the patient was diagnosed with bilateral nodular scleritis and pyoderma gangrenosum associated with active UC. As the scleritis did not respond to 0.1% betamethasone eye drops and prednisolone ophthalmic ointment, oral glucocorticoids were initiated, leading to significant improvement in skin and ocular inflammation as well as clinical remission of UC.
Conclusions: UC and pyoderma gangrenosum are rare causes of nodular scleritis. This rare case underscores the importance of reviewing a patient's systemic disease history and recognizing systemic symptoms to identify the underlying cause of scleritis and initiate appropriate treatment in a timely manner.
{"title":"Nodular Scleritis and Pyoderma Gangrenosum Associated with Active Ulcerative Colitis: A Case Report.","authors":"Kazuki Yashiro, Sozaburo Ihara, Hikari Boki, Amane Yamamoto, Rie Tanaka","doi":"10.1159/000546828","DOIUrl":"10.1159/000546828","url":null,"abstract":"<p><strong>Introduction: </strong>Pyoderma gangrenosum is a rare form of neutrophilic dermatosis, with ocular involvement being atypical. We present a rare case of ulcerative colitis (UC) complicated by nodular scleritis and pyoderma gangrenosum, both occurring almost simultaneously.</p><p><strong>Case presentation: </strong>A 55-year-old man with active UC initially presented to our hospital with anterior diffuse scleritis and a peripheral corneal ulcer in his left eye. The condition rapidly progressed to bilateral nodular scleritis. Concurrently, multiple painful abscesses developed on his trunk and head. A skin biopsy confirmed the diagnosis of pyoderma gangrenosum. Laboratory tests revealed elevated C-reactive protein and erythrocyte sedimentation rates, along with the increased proteinase-3 antineutrophil cytoplasmic antibody (PR3-ANCA). A comprehensive systemic evaluation ruled out vasculitis, and an elevated PR3-ANCA level was attributed to active UC. After excluding other potential causes of scleritis, the patient was diagnosed with bilateral nodular scleritis and pyoderma gangrenosum associated with active UC. As the scleritis did not respond to 0.1% betamethasone eye drops and prednisolone ophthalmic ointment, oral glucocorticoids were initiated, leading to significant improvement in skin and ocular inflammation as well as clinical remission of UC.</p><p><strong>Conclusions: </strong>UC and pyoderma gangrenosum are rare causes of nodular scleritis. This rare case underscores the importance of reviewing a patient's systemic disease history and recognizing systemic symptoms to identify the underlying cause of scleritis and initiate appropriate treatment in a timely manner.</p>","PeriodicalId":9635,"journal":{"name":"Case Reports in Ophthalmology","volume":"16 1","pages":"475-481"},"PeriodicalIF":0.5,"publicationDate":"2025-06-10","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12237287/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144590553","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 : 2025-06-05eCollection Date: 2025-01-01DOI: 10.1159/000546659
Fatma Kassem Mohamed, Mohamed S I Mohamed, Maha M El Shafei, Anant Pai, Hashem Abu Serhan
Introduction: The objective of this study was to report successful management of multidrug-resistant Pseudomonas aeruginosa endophthalmitis in a patient with a complex ophthalmic history using intracameral povidone-iodine (PI).
Case presentation: A 59-year-old male with a history of cataract surgery and multiple retinal detachment repairs presented with acute left eye pain, chemosis, redness, and vision loss. Diagnosis of exogenous endophthalmitis was made. The patient had anterior chamber washout, along with aqueous and vitreous tapping with intravitreal vancomycin, ceftazidime, and intravenous ciprofloxacin. Since infection persisted despite antibiotic therapies, a repeated anterior chamber washout followed by intracameral injection of PI 0.1% was done. Vision improved to 6/60 with no bacterial regrowth or inflammatory membranes on discharge and follow-up.
Conclusions: Intracameral PI may offer a solution for endophthalmitis cases where standard antibiotics are ineffective. This case supports the potential role of PI in managing resistant intraocular infections.
{"title":"Intracameral Povidone-Iodine for Multidrug-Resistant <i>Pseudomonas aeruginosa</i> Exogenous Endophthalmitis: A Case Report and Literature Review.","authors":"Fatma Kassem Mohamed, Mohamed S I Mohamed, Maha M El Shafei, Anant Pai, Hashem Abu Serhan","doi":"10.1159/000546659","DOIUrl":"10.1159/000546659","url":null,"abstract":"<p><strong>Introduction: </strong>The objective of this study was to report successful management of multidrug-resistant <i>Pseudomonas aeruginosa</i> endophthalmitis in a patient with a complex ophthalmic history using intracameral povidone-iodine (PI).</p><p><strong>Case presentation: </strong>A 59-year-old male with a history of cataract surgery and multiple retinal detachment repairs presented with acute left eye pain, chemosis, redness, and vision loss. Diagnosis of exogenous endophthalmitis was made. The patient had anterior chamber washout, along with aqueous and vitreous tapping with intravitreal vancomycin, ceftazidime, and intravenous ciprofloxacin. Since infection persisted despite antibiotic therapies, a repeated anterior chamber washout followed by intracameral injection of PI 0.1% was done. Vision improved to 6/60 with no bacterial regrowth or inflammatory membranes on discharge and follow-up.</p><p><strong>Conclusions: </strong>Intracameral PI may offer a solution for endophthalmitis cases where standard antibiotics are ineffective. This case supports the potential role of PI in managing resistant intraocular infections.</p>","PeriodicalId":9635,"journal":{"name":"Case Reports in Ophthalmology","volume":"16 1","pages":"468-474"},"PeriodicalIF":0.5,"publicationDate":"2025-06-05","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12215200/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144552438","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: The co-occurrence of traumatic optic neuropathy (TON) and central retinal artery occlusion (CRAO) presents a significant clinical challenge due to their complex interactions and potential for profound visual impairment. This case report details a rare instance of both conditions following blunt ocular trauma, aiming to enhance understanding of their interplay and the necessity for timely intervention.
Case report: A 45-year-old woman presented with sudden vision loss in her right eye 3 h after sustaining blunt trauma from a wood fragment while chopping firewood. Initial examination revealed no light perception (NLP) in the affected eye and a relative afferent pupillary defect. Imaging revealed hyperdensity and thickening of the posterior sclera, the intraocular and anterior intraorbital optic nerve. Despite administration of intravenous steroids and a neurosurgical consultation for TON, retinal evaluation 1 day post-trauma revealed retinal edema, pallor, multiple vascular occlusions, and a cherry-red spot, leading to a diagnosis of CRAO associated with TON. Ongoing follow-ups showed persistent NLP in the right eye.
Conclusion: This case underscores the potential for concurrent TON and CRAO following ocular trauma, leading to a complex visual outcome, while also examining the underlying mechanisms that may contribute to this phenomenon. The report highlights the need for further investigation into effective therapeutic strategies and underscores the complexities involved in managing traumatic ocular conditions. Future studies should focus on optimizing medical or surgical interventions to improve visual outcomes for patients facing this challenging combination of injuries.
{"title":"Double Jeopardy: Uncommon Concurrent Traumatic Optic Neuropathy and Central Retinal Artery Occlusion - A Case Report.","authors":"Narges Karrabi, Maryam Najafi, Amirreza Veisi, Tara Bakhshian, Omid Emadi, Shayan Heshmati","doi":"10.1159/000546759","DOIUrl":"10.1159/000546759","url":null,"abstract":"<p><strong>Introduction: </strong>The co-occurrence of traumatic optic neuropathy (TON) and central retinal artery occlusion (CRAO) presents a significant clinical challenge due to their complex interactions and potential for profound visual impairment. This case report details a rare instance of both conditions following blunt ocular trauma, aiming to enhance understanding of their interplay and the necessity for timely intervention.</p><p><strong>Case report: </strong>A 45-year-old woman presented with sudden vision loss in her right eye 3 h after sustaining blunt trauma from a wood fragment while chopping firewood. Initial examination revealed no light perception (NLP) in the affected eye and a relative afferent pupillary defect. Imaging revealed hyperdensity and thickening of the posterior sclera, the intraocular and anterior intraorbital optic nerve. Despite administration of intravenous steroids and a neurosurgical consultation for TON, retinal evaluation 1 day post-trauma revealed retinal edema, pallor, multiple vascular occlusions, and a cherry-red spot, leading to a diagnosis of CRAO associated with TON. Ongoing follow-ups showed persistent NLP in the right eye.</p><p><strong>Conclusion: </strong>This case underscores the potential for concurrent TON and CRAO following ocular trauma, leading to a complex visual outcome, while also examining the underlying mechanisms that may contribute to this phenomenon. The report highlights the need for further investigation into effective therapeutic strategies and underscores the complexities involved in managing traumatic ocular conditions. Future studies should focus on optimizing medical or surgical interventions to improve visual outcomes for patients facing this challenging combination of injuries.</p>","PeriodicalId":9635,"journal":{"name":"Case Reports in Ophthalmology","volume":"16 1","pages":"489-495"},"PeriodicalIF":0.5,"publicationDate":"2025-06-05","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12258874/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144636234","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}