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}
Introduction: Patients with membranoproliferative glomerulonephritis (MPGN) type III may exhibit the saw-tooth pattern of cuticular drusen. This study reports a four-and-a-half-year retrospective time-dependent observation of cuticular drusen associated with immune complex-mediated MPGN type III patients.
Case presentation: A 32-year-old woman was referred for ocular evaluation due to systemic steroids used for nephrotic syndrome caused by immune complex-mediated MPGN type III. She had no ocular symptoms. The initial examination showed that her best-corrected visual acuity (BCVA) was 1.0 oculus uterque (OU). Color fundus photographs revealed pigmentary abnormalities in the macular and perimacular regions OU. Fundus autofluorescence (FAF) images demonstrated numerous tiny dots with a hypoautofluorescent center surrounded by a rim of hyperautofluorescence. Enhanced depth imaging-optical coherence tomography (OCT) revealed triangular morphologic features, represented by a saw-tooth pattern with internal hyporeflectivity. Over 4.5 years, the internal reflectivity of the drusen gradually increased. Moreover, depigmentation and yellow color changes in the macular and perimacular regions worsened, without obvious changes on FAF. Her BCVA remained at 1.0 OU, without new ocular symptoms or cataract progression during the 4.5-year follow-up period. Both eyes had a significant positive correlation between swept-source OCT-based mean internal reflectivity measured by the ImageJ software and the observation period. However, no correlation was found between the estimated glomerular filtration rate (eGFR) and the observation period or between eGFR and mean internal reflectivity.
Conclusion: The internal reflectivity of cuticular drusen associated with immune complex-mediated MPGN type III showed time-dependent changes on OCT and worsened depigmentation and yellow color changes in the macular and perimacular regions, independent of renal function.
{"title":"Morphological Changes in the Saw-Tooth Pattern of Cuticular Drusen Associated with Immune Complex-Mediated Membranous Proliferative Glomerulonephritis Type III: A Case Report.","authors":"Takayuki Tanaka, Satoru Kase, Kiriko Nishiyama-Hirooka, Michiyuki Saito, Susumu Ishida","doi":"10.1159/000547059","DOIUrl":"10.1159/000547059","url":null,"abstract":"<p><strong>Introduction: </strong>Patients with membranoproliferative glomerulonephritis (MPGN) type III may exhibit the saw-tooth pattern of cuticular drusen. This study reports a four-and-a-half-year retrospective time-dependent observation of cuticular drusen associated with immune complex-mediated MPGN type III patients.</p><p><strong>Case presentation: </strong>A 32-year-old woman was referred for ocular evaluation due to systemic steroids used for nephrotic syndrome caused by immune complex-mediated MPGN type III. She had no ocular symptoms. The initial examination showed that her best-corrected visual acuity (BCVA) was 1.0 oculus uterque (OU). Color fundus photographs revealed pigmentary abnormalities in the macular and perimacular regions OU. Fundus autofluorescence (FAF) images demonstrated numerous tiny dots with a hypoautofluorescent center surrounded by a rim of hyperautofluorescence. Enhanced depth imaging-optical coherence tomography (OCT) revealed triangular morphologic features, represented by a saw-tooth pattern with internal hyporeflectivity. Over 4.5 years, the internal reflectivity of the drusen gradually increased. Moreover, depigmentation and yellow color changes in the macular and perimacular regions worsened, without obvious changes on FAF. Her BCVA remained at 1.0 OU, without new ocular symptoms or cataract progression during the 4.5-year follow-up period. Both eyes had a significant positive correlation between swept-source OCT-based mean internal reflectivity measured by the ImageJ software and the observation period. However, no correlation was found between the estimated glomerular filtration rate (eGFR) and the observation period or between eGFR and mean internal reflectivity.</p><p><strong>Conclusion: </strong>The internal reflectivity of cuticular drusen associated with immune complex-mediated MPGN type III showed time-dependent changes on OCT and worsened depigmentation and yellow color changes in the macular and perimacular regions, independent of renal function.</p>","PeriodicalId":9635,"journal":{"name":"Case Reports in Ophthalmology","volume":"16 1","pages":"535-541"},"PeriodicalIF":0.6,"publicationDate":"2025-06-25","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12306962/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144741305","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 : 2025-06-24eCollection 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}