Pub Date : 2025-01-30eCollection Date: 2025-01-01DOI: 10.1159/000543219
Meishar Meisel, Eran Berkowitz, Avi Schwalb, Beatrice Tiosano
Introduction: Herein, we report on the option and clinical advantage of the Preserflo MicroShunt insertion into the posterior chamber ciliary sulcus in a patient with advanced pseudoexfoliation glaucoma at a high risk of corneal decompensation.
Case presentation: We describe a 64-year-old advanced pseudoexfoliation glaucoma patient who despite maximal tolerated medical therapy and two failed glaucoma surgeries, still suffered from uncontrolled intraocular pressure (IOP) of 36 mm Hg in his left eye. The patient underwent the implantation of the Preserflo MicroShunt. The shunt was placed nasally into an area of unscarred conjunctiva, with the anterior part of the shunt inserted into the ciliary sulcus. Postoperatively, the patient's IOP dropped to 6 mm Hg on the first day and stabilized at 11 mm Hg at the 6-month mark. An elevated, posteriorly located bleb was observed, and the patient no longer required additional topical medications. Endothelial cell (EC) count remained stable with no signs of corneal edema. The patient did not experience any serious postoperative complications.
Conclusion: Implantation of the Preserflo MicroShunt into the ciliary sulcus appears to be a viable option for patients at high risk of corneal decompensation, hence, offering effective IOP control while minimizing EC loss. Further studies with larger patient groups are warranted to better evaluate the safety and efficacy of this technique.
{"title":"The Road Less Traveled: Ciliary Sulcus Implantation of the Preserflo MicroShunt.","authors":"Meishar Meisel, Eran Berkowitz, Avi Schwalb, Beatrice Tiosano","doi":"10.1159/000543219","DOIUrl":"10.1159/000543219","url":null,"abstract":"<p><strong>Introduction: </strong>Herein, we report on the option and clinical advantage of the Preserflo MicroShunt insertion into the posterior chamber ciliary sulcus in a patient with advanced pseudoexfoliation glaucoma at a high risk of corneal decompensation.</p><p><strong>Case presentation: </strong>We describe a 64-year-old advanced pseudoexfoliation glaucoma patient who despite maximal tolerated medical therapy and two failed glaucoma surgeries, still suffered from uncontrolled intraocular pressure (IOP) of 36 mm Hg in his left eye. The patient underwent the implantation of the Preserflo MicroShunt. The shunt was placed nasally into an area of unscarred conjunctiva, with the anterior part of the shunt inserted into the ciliary sulcus. Postoperatively, the patient's IOP dropped to 6 mm Hg on the first day and stabilized at 11 mm Hg at the 6-month mark. An elevated, posteriorly located bleb was observed, and the patient no longer required additional topical medications. Endothelial cell (EC) count remained stable with no signs of corneal edema. The patient did not experience any serious postoperative complications.</p><p><strong>Conclusion: </strong>Implantation of the Preserflo MicroShunt into the ciliary sulcus appears to be a viable option for patients at high risk of corneal decompensation, hence, offering effective IOP control while minimizing EC loss. Further studies with larger patient groups are warranted to better evaluate the safety and efficacy of this technique.</p>","PeriodicalId":9635,"journal":{"name":"Case Reports in Ophthalmology","volume":"16 1","pages":"68-73"},"PeriodicalIF":0.5,"publicationDate":"2025-01-30","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11842094/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143467062","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 : 2025-01-24eCollection Date: 2025-01-01DOI: 10.1159/000543760
Konstantina Bachtalia, Sotiria Palioura
Introduction: Thermal injuries associated with cosmetic eyelid surgery can significantly impair corneal integrity, potentially leading to vision loss. Conventional management, including bandage contact lens use, topical steroids, lubrication, autologous serum tears, and vitamin C, plays a crucial role in preventing long-term complications. This case report explores the potential benefits of multilayer amniotic membrane transplantation (AMT) as an adjunct to standard therapy for treating diathermy-associated corneal burns. Review of the literature advocates the beneficial effects of supplementary AMT in managing thermal corneal trauma.
Case description: A 75-year-old man presented with unilateral diathermy-associated corneal laceration superior to the visual axis following cosmetic blepharoplasty. Vision had deteriorated from his baseline 20/30 to 20/125 postoperatively. Intervention involved multilayer AMT alongside standard care, including topical steroids and antibiotics, oral doxycycline, and vitamin C. Postoperative course was monitored with anterior segment optical coherence tomography (AS-OCT) imaging. By 3 months, complete corneal healing and restoration of baseline visual acuity were achieved.
Conclusion: This is the first documented case of multilayer AMT as an effective supplement to conventional management of diathermy-associated corneal injury. While AMTs unique properties likely contributed to corneal healing and visual recovery, the results should be interpreted in the context of a multimodal therapeutic approach.
导言:与眼睑美容手术相关的热损伤会严重损害角膜的完整性,可能导致视力丧失。传统的治疗方法,包括使用绷带隐形眼镜、外用类固醇、润滑剂、自体血清泪液和维生素 C,在预防长期并发症方面起着至关重要的作用。本病例报告探讨了多层羊膜移植(AMT)作为标准疗法辅助治疗电热相关性角膜烧伤的潜在益处。文献综述认为,多层羊膜移植在治疗热角膜创伤方面具有辅助疗效:一名 75 岁的男子在眼睑整容术后出现单侧眼轴上方的电热相关性角膜裂伤。术后视力从基线 20/30 下降到 20/125。干预措施包括多层 AMT 以及标准护理,包括局部类固醇和抗生素、口服多西环素和维生素 C。三个月后,角膜完全愈合,视力恢复到基线水平:这是第一例记录在案的多层 AMT 作为传统治疗电疗相关角膜损伤的有效补充的病例。虽然 AMT 的独特性能可能有助于角膜愈合和视力恢复,但应结合多模式治疗方法来解释这一结果。
{"title":"Management of Diathermy-Induced Corneal Burn following Upper Eyelid Blepharoplasty with Multilayer Amniotic Membrane Transplantation: A Case Report and Review of the Literature.","authors":"Konstantina Bachtalia, Sotiria Palioura","doi":"10.1159/000543760","DOIUrl":"10.1159/000543760","url":null,"abstract":"<p><strong>Introduction: </strong>Thermal injuries associated with cosmetic eyelid surgery can significantly impair corneal integrity, potentially leading to vision loss. Conventional management, including bandage contact lens use, topical steroids, lubrication, autologous serum tears, and vitamin C, plays a crucial role in preventing long-term complications. This case report explores the potential benefits of multilayer amniotic membrane transplantation (AMT) as an adjunct to standard therapy for treating diathermy-associated corneal burns. Review of the literature advocates the beneficial effects of supplementary AMT in managing thermal corneal trauma.</p><p><strong>Case description: </strong>A 75-year-old man presented with unilateral diathermy-associated corneal laceration superior to the visual axis following cosmetic blepharoplasty. Vision had deteriorated from his baseline 20/30 to 20/125 postoperatively. Intervention involved multilayer AMT alongside standard care, including topical steroids and antibiotics, oral doxycycline, and vitamin C. Postoperative course was monitored with anterior segment optical coherence tomography (AS-OCT) imaging. By 3 months, complete corneal healing and restoration of baseline visual acuity were achieved.</p><p><strong>Conclusion: </strong>This is the first documented case of multilayer AMT as an effective supplement to conventional management of diathermy-associated corneal injury. While AMTs unique properties likely contributed to corneal healing and visual recovery, the results should be interpreted in the context of a multimodal therapeutic approach.</p>","PeriodicalId":9635,"journal":{"name":"Case Reports in Ophthalmology","volume":"16 1","pages":"133-142"},"PeriodicalIF":0.5,"publicationDate":"2025-01-24","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11842102/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143467098","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 : 2025-01-22eCollection Date: 2025-01-01DOI: 10.1159/000543240
Jeffrey A Brown, Louis Z Cai, Jesse D Sengillo, James Lin, Harry W Flynn
Introduction: Demarcation laser photocoagulation (DLP) is an infrequently utilized modality for limited retinal detachments. The current study, a retrospective consecutive case series, reviewed anatomic and visual outcomes in these patients at a single academic center.
Case presentations: A search of the electronic medical record identified 10 eyes of 10 patients. Five of the 10 patients were asymptomatic at the time of initial treatment. Five patients had symptoms corresponding to retinal detachment. The asymptomatic patients remained stable without progression at the last follow-up (range 1-8 years). In 2 of 5 symptomatic patients, the retinal detachment progressed through the laser demarcation and, subsequently, underwent vitreoretinal surgery. At the last follow-up, the retina was attached in all five symptomatic patients.
Conclusion: In this small series of patients undergoing DLP, the retina remained stable in asymptomatic patients but the retinal detachment progressed through the laser demarcation in the majority of symptomatic patients.
{"title":"Demarcation Laser Photocoagulation for Rhegmatogenous Retinal Detachment: Outcomes in Symptomatic and Asymptomatic Patients.","authors":"Jeffrey A Brown, Louis Z Cai, Jesse D Sengillo, James Lin, Harry W Flynn","doi":"10.1159/000543240","DOIUrl":"10.1159/000543240","url":null,"abstract":"<p><strong>Introduction: </strong>Demarcation laser photocoagulation (DLP) is an infrequently utilized modality for limited retinal detachments. The current study, a retrospective consecutive case series, reviewed anatomic and visual outcomes in these patients at a single academic center.</p><p><strong>Case presentations: </strong>A search of the electronic medical record identified 10 eyes of 10 patients. Five of the 10 patients were asymptomatic at the time of initial treatment. Five patients had symptoms corresponding to retinal detachment. The asymptomatic patients remained stable without progression at the last follow-up (range 1-8 years). In 2 of 5 symptomatic patients, the retinal detachment progressed through the laser demarcation and, subsequently, underwent vitreoretinal surgery. At the last follow-up, the retina was attached in all five symptomatic patients.</p><p><strong>Conclusion: </strong>In this small series of patients undergoing DLP, the retina remained stable in asymptomatic patients but the retinal detachment progressed through the laser demarcation in the majority of symptomatic patients.</p>","PeriodicalId":9635,"journal":{"name":"Case Reports in Ophthalmology","volume":"16 1","pages":"114-123"},"PeriodicalIF":0.5,"publicationDate":"2025-01-22","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11842075/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143467088","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 : 2025-01-17eCollection Date: 2025-01-01DOI: 10.1159/000543084
Noémie Delaissé, Daniel Blockmans, Rita Van Ginderdeuren, Guy Missotten
Introduction: Immunoglobulin G4-related disease (IgG4-RD) is a systemic, immune-mediated disorder marked by the infiltration of IgG4-positive plasma cells and fibrosis in affected organs. This report presents a rare case of a patient with isolated nodular scleritis as an IgG4-RD (in a more precise way antineutrophil cytoplasmic antibody (ANCA)-associated vasculitis [AAV] and IgG4-RD overlap syndrome).
Case presentation: A 51-year-old woman was referred with the presumed diagnosis of conjunctival lymphoma due to a painful, salmon-colored lesion in the superior conjunctiva of the right eye. A biopsy of the conjunctiva showed a lymphoplasmacytic infiltrate with multiple IgG4-positive cells (>200 cells/high power field), elevated IgG4/IgG ratio of 66% and fibrotic tissue without obvious vasculitis, confirming the diagnosis of IgG4-related disease (IgG4-RD). ANCAs directly against myeloperoxidase were also positive, suggesting AAV. Given that the clinical signs align with both disease entities, it was concluded that the case fits in its restricted sense the newly described overlap syndrome. The scleritis was successfully treated with a tapering dose of corticosteroids and rituximab.
Conclusion: This case illustrates a rare presentation of scleritis as an IgG4-RD (in a more precise way AAV and IgG4-RD overlap syndrome) and demonstrates that rituximab and low dose of corticosteroids can lead to remission.
{"title":"Nodular Scleritis as Isolated Symptom of IgG4-Related Disease, Mimicking as Conjunctival Lymphoma: A Case Report.","authors":"Noémie Delaissé, Daniel Blockmans, Rita Van Ginderdeuren, Guy Missotten","doi":"10.1159/000543084","DOIUrl":"10.1159/000543084","url":null,"abstract":"<p><strong>Introduction: </strong>Immunoglobulin G4-related disease (IgG4-RD) is a systemic, immune-mediated disorder marked by the infiltration of IgG4-positive plasma cells and fibrosis in affected organs. This report presents a rare case of a patient with isolated nodular scleritis as an IgG4-RD (in a more precise way antineutrophil cytoplasmic antibody (ANCA)-associated vasculitis [AAV] and IgG4-RD overlap syndrome).</p><p><strong>Case presentation: </strong>A 51-year-old woman was referred with the presumed diagnosis of conjunctival lymphoma due to a painful, salmon-colored lesion in the superior conjunctiva of the right eye. A biopsy of the conjunctiva showed a lymphoplasmacytic infiltrate with multiple IgG4-positive cells (>200 cells/high power field), elevated IgG4/IgG ratio of 66% and fibrotic tissue without obvious vasculitis, confirming the diagnosis of IgG4-related disease (IgG4-RD). ANCAs directly against myeloperoxidase were also positive, suggesting AAV. Given that the clinical signs align with both disease entities, it was concluded that the case fits in its restricted sense the newly described overlap syndrome. The scleritis was successfully treated with a tapering dose of corticosteroids and rituximab.</p><p><strong>Conclusion: </strong>This case illustrates a rare presentation of scleritis as an IgG4-RD (in a more precise way AAV and IgG4-RD overlap syndrome) and demonstrates that rituximab and low dose of corticosteroids can lead to remission.</p>","PeriodicalId":9635,"journal":{"name":"Case Reports in Ophthalmology","volume":"16 1","pages":"102-106"},"PeriodicalIF":0.5,"publicationDate":"2025-01-17","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11842084/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143467102","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: Macular hole is a rare complication in patients with retinitis pigmentosa that significantly reduces visual acuity. Although vitreous surgery for macular holes generally yields favorable outcomes, postoperative macular atrophy has been reported. We report the second case of retinitis pigmentosa in a patient who developed a 13-year progressive macular atrophy after macular hole surgery.
Case presentation: A 64-year-old Japanese woman, who had been diagnosed with retinitis pigmentosa at 52 years of age, presented to our hospital with blurred vision in her left eye. Phacovitrectomy of the left eye was performed after a full-thickness macular hole was revealed by optical coherence tomography. We stained the internal limiting membrane during surgery using 0.05% indocyanine green and peeled it around the macular hole. Nevertheless, slight atrophy of the retinal pigment epithelium appeared in the left macula 17 days after surgery. The macular hole closed 1 year after surgery, and the macular atrophy gradually became more apparent and enlarged. Thirteen years later, atrophy had expanded to 2.5-disc diameters, and the left decimal best-corrected visual acuity was 0.1; no macular degeneration appeared in the right eye. Genetic examination revealed compound heterozygous variants in the EYS gene.
Conclusion: Macular atrophy can develop after dye-assisted macular hole surgery for patients with retinitis pigmentosa. Potential risk factors for the development of postoperative macular atrophy include dye toxicity, light toxicity, surgical intervention in the macula, postoperative inflammation, and genotype. However, the exact cause of atrophy remains uncertain.
{"title":"Development of Macular Atrophy after Macular Hole Surgery in an Eye with Retinitis Pigmentosa.","authors":"Yuki Goto, Kazuki Kuniyoshi, Kensuke Goto, Tomoyasu Kayazawa, Taro Kominami, Fukutaro Mano, Masuo Sakamoto, Chiharu Iwahashi, Shunji Kusaka","doi":"10.1159/000543599","DOIUrl":"10.1159/000543599","url":null,"abstract":"<p><strong>Introduction: </strong>Macular hole is a rare complication in patients with retinitis pigmentosa that significantly reduces visual acuity. Although vitreous surgery for macular holes generally yields favorable outcomes, postoperative macular atrophy has been reported. We report the second case of retinitis pigmentosa in a patient who developed a 13-year progressive macular atrophy after macular hole surgery.</p><p><strong>Case presentation: </strong>A 64-year-old Japanese woman, who had been diagnosed with retinitis pigmentosa at 52 years of age, presented to our hospital with blurred vision in her left eye. Phacovitrectomy of the left eye was performed after a full-thickness macular hole was revealed by optical coherence tomography. We stained the internal limiting membrane during surgery using 0.05% indocyanine green and peeled it around the macular hole. Nevertheless, slight atrophy of the retinal pigment epithelium appeared in the left macula 17 days after surgery. The macular hole closed 1 year after surgery, and the macular atrophy gradually became more apparent and enlarged. Thirteen years later, atrophy had expanded to 2.5-disc diameters, and the left decimal best-corrected visual acuity was 0.1; no macular degeneration appeared in the right eye. Genetic examination revealed compound heterozygous variants in the <i>EYS</i> gene.</p><p><strong>Conclusion: </strong>Macular atrophy can develop after dye-assisted macular hole surgery for patients with retinitis pigmentosa. Potential risk factors for the development of postoperative macular atrophy include dye toxicity, light toxicity, surgical intervention in the macula, postoperative inflammation, and genotype. However, the exact cause of atrophy remains uncertain.</p>","PeriodicalId":9635,"journal":{"name":"Case Reports in Ophthalmology","volume":"16 1","pages":"107-113"},"PeriodicalIF":0.5,"publicationDate":"2025-01-17","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11842103/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143467094","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 : 2025-01-16eCollection Date: 2025-01-01DOI: 10.1159/000543577
Shawn Khan, Khushi Saigal, Jillene Moxam, Arash Maleki
Introduction: Acute zonal occult outer retinopathy (AZOOR) is a rare inflammatory retinal disease with rapid outer retinal function loss, photopsias, unremarkable fundus findings, and electroretinography abnormalities. AZOOR diagnosis can be challenging due to its overlap with other retinal conditions, such as acute idiopathic blind spot enlargement syndrome and autoimmune retinopathies (AIRs). Multimodal imaging, including fundus autofluorescence and optical coherence tomography, has improved detection, revealing progressive outer retinal damage. Although the etiology of AZOOR remains uncertain, autoimmune mechanisms and viral associations have been proposed. Recent studies have identified anti-retinal antibodies, complicating differentiation from other AIRs.
Case presentation: A 63-year-old male presented with photopsias, floaters, and worsening vision in his left eye. He had a prior diagnosis of AIR with serum antibodies against enolase, arrestin, and heat shock protein 27 (HSP27). Despite corticosteroid therapy, his visual acuity worsened from 20/20 to 20/60. Fundus examination showed subtle changes, and multimodal imaging revealed outer retinal damage consistent with AZOOR. He was started on mycophenolate mofetil, cyclosporine, and intravenous immunoglobulin. Over a year of follow-up, his vision improved to 20/25, and imaging showed stabilization of retinal damage.
Conclusion: This case report highlights AZOOR can be associated with secondary np-AIR. Multimodal imaging, electrophysiologic testing of retina and retinal pigment epithelial, and anti-retinal antibody may be helpful for diagnosis of these patients. A combination of conventional immunomodulatory therapy and IVIg can help with controlling AZOOR and secondary np-AIR.
{"title":"A Case of Concomitant Acute Zonal Occult Outer Retinopathy and Secondary Nonparaneoplastic Autoimmune Retinopathy.","authors":"Shawn Khan, Khushi Saigal, Jillene Moxam, Arash Maleki","doi":"10.1159/000543577","DOIUrl":"10.1159/000543577","url":null,"abstract":"<p><strong>Introduction: </strong>Acute zonal occult outer retinopathy (AZOOR) is a rare inflammatory retinal disease with rapid outer retinal function loss, photopsias, unremarkable fundus findings, and electroretinography abnormalities. AZOOR diagnosis can be challenging due to its overlap with other retinal conditions, such as acute idiopathic blind spot enlargement syndrome and autoimmune retinopathies (AIRs). Multimodal imaging, including fundus autofluorescence and optical coherence tomography, has improved detection, revealing progressive outer retinal damage. Although the etiology of AZOOR remains uncertain, autoimmune mechanisms and viral associations have been proposed. Recent studies have identified anti-retinal antibodies, complicating differentiation from other AIRs.</p><p><strong>Case presentation: </strong>A 63-year-old male presented with photopsias, floaters, and worsening vision in his left eye. He had a prior diagnosis of AIR with serum antibodies against enolase, arrestin, and heat shock protein 27 (HSP27). Despite corticosteroid therapy, his visual acuity worsened from 20/20 to 20/60. Fundus examination showed subtle changes, and multimodal imaging revealed outer retinal damage consistent with AZOOR. He was started on mycophenolate mofetil, cyclosporine, and intravenous immunoglobulin. Over a year of follow-up, his vision improved to 20/25, and imaging showed stabilization of retinal damage.</p><p><strong>Conclusion: </strong>This case report highlights AZOOR can be associated with secondary np-AIR. Multimodal imaging, electrophysiologic testing of retina and retinal pigment epithelial, and anti-retinal antibody may be helpful for diagnosis of these patients. A combination of conventional immunomodulatory therapy and IVIg can help with controlling AZOOR and secondary np-AIR.</p>","PeriodicalId":9635,"journal":{"name":"Case Reports in Ophthalmology","volume":"16 1","pages":"124-132"},"PeriodicalIF":0.5,"publicationDate":"2025-01-16","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11842096/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143467057","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 : 2025-01-13eCollection Date: 2025-01-01DOI: 10.1159/000543279
Sumer Doctor, Ayushi Agarwal, Mohammad Javed Ali, Nandini Bothra
Introduction: Dentigerous cysts (DCs) involving the orbit are extremely rare. The authors report a unique case of stand-alone orbital DC associated with ectopic canine tooth masquerading as a lacrimal sac diverticulitis with superadded preseptal cellulitis.
Case presentation: A 21-year-old lady presented with left inferomedial swelling, associated with pain and redness of 1-week-duration. A lacrimal sac diverticulitis with preseptal cellulitis was suspected and imaging was requested in view of progression. Computed tomography scan of orbit revealed a well-defined heterogenous mass, with associated hyperdense lesion within the mass, prompting a possibility of a foreign body granuloma. Surgical exploration and excision confirmed the diagnosis of an ectopic canine tooth with associated DC along with excessive adhesion of the cyst to the lacrimal sac wall. Maxillary sinus did not show any abnormality. Lacrimal irrigation was patent with no recurrence noted at 6-month follow-up.
Conclusion: This report highlights the presentation of DC with associated ectopic canine tooth as an isolated orbital mass, initially manifesting as preseptal cellulitis with lacrimal sac diverticulitis. Recurrent episodes of infection can lead to synechiae formation, rendering adjacent lacrimal sac more prone to injury. Cyst excision with meticulous dissection yields good outcome.
{"title":"Dentigerous Cyst Masquerading as Lacrimal Sac Diverticulitis.","authors":"Sumer Doctor, Ayushi Agarwal, Mohammad Javed Ali, Nandini Bothra","doi":"10.1159/000543279","DOIUrl":"10.1159/000543279","url":null,"abstract":"<p><strong>Introduction: </strong>Dentigerous cysts (DCs) involving the orbit are extremely rare. The authors report a unique case of stand-alone orbital DC associated with ectopic canine tooth masquerading as a lacrimal sac diverticulitis with superadded preseptal cellulitis.</p><p><strong>Case presentation: </strong>A 21-year-old lady presented with left inferomedial swelling, associated with pain and redness of 1-week-duration. A lacrimal sac diverticulitis with preseptal cellulitis was suspected and imaging was requested in view of progression. Computed tomography scan of orbit revealed a well-defined heterogenous mass, with associated hyperdense lesion within the mass, prompting a possibility of a foreign body granuloma. Surgical exploration and excision confirmed the diagnosis of an ectopic canine tooth with associated DC along with excessive adhesion of the cyst to the lacrimal sac wall. Maxillary sinus did not show any abnormality. Lacrimal irrigation was patent with no recurrence noted at 6-month follow-up.</p><p><strong>Conclusion: </strong>This report highlights the presentation of DC with associated ectopic canine tooth as an isolated orbital mass, initially manifesting as preseptal cellulitis with lacrimal sac diverticulitis. Recurrent episodes of infection can lead to synechiae formation, rendering adjacent lacrimal sac more prone to injury. Cyst excision with meticulous dissection yields good outcome.</p>","PeriodicalId":9635,"journal":{"name":"Case Reports in Ophthalmology","volume":"16 1","pages":"95-101"},"PeriodicalIF":0.5,"publicationDate":"2025-01-13","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11842074/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143467091","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 : 2025-01-10eCollection Date: 2025-01-01DOI: 10.1159/000542708
Tobias Peschaut, Monja Michelitsch, Martina Brandner, Sandra Kamper, Lisa Ofner-Ziegenfuss, Jasmin Blatterer, Heidelis Anna Tichy, Laura Posch-Pertl
Introduction: Stickler syndrome is a rare collagenopathy, caused by mutations in various genes coding for fibrillar collagens II, IX, and XI. The disorder can be subdivided into different groups, depending on the genes affected and clinical features found in patients. Ocular symptoms, such as high myopia, retinal detachments, or anomalies in the vitreous, are present in most forms of Stickler syndrome. In this case report, we present a patient with an unusual retinal phenotype.
Case presentation: Subject of this case report is a 33-year-old woman, who was examined at the Department of Ophthalmology at Medical University of Graz. A thorough ophthalmological examination was conducted, detailed medical and family history acquired, and genetic testing performed. Best corrected visual acuity was 20/20 on both eyes; however, impaired binocular vision associated with intermittent exotropia was found. Furthermore, dilated fundoscopy showed an unusual, hypopigmented spotted retinal phenotype. Fundus autofluorescence showed multiple hyperfluorescent spots corresponding with the spotted retinal appearance. Genetic testing revealed a novel variant in the gene COL11A1. No other ocular abnormalities which are associated with COL11A1 were found.
Conclusion: Several subtypes of Stickler syndrome have been reported in medical literature, greatly varying in clinical manifestations. Many different mutations in the gene COL11A1 have been discovered and are typically associated with Stickler syndrome type 2. To our best knowledge, this is the first report of a patient with a mutation in the COL11A1 gene presenting with a hypopigmented spotted retina.
{"title":"An Unusual Retinal Presentation of a Novel <i>COL11A1</i> Mutation: A Case Report.","authors":"Tobias Peschaut, Monja Michelitsch, Martina Brandner, Sandra Kamper, Lisa Ofner-Ziegenfuss, Jasmin Blatterer, Heidelis Anna Tichy, Laura Posch-Pertl","doi":"10.1159/000542708","DOIUrl":"10.1159/000542708","url":null,"abstract":"<p><strong>Introduction: </strong>Stickler syndrome is a rare collagenopathy, caused by mutations in various genes coding for fibrillar collagens II, IX, and XI. The disorder can be subdivided into different groups, depending on the genes affected and clinical features found in patients. Ocular symptoms, such as high myopia, retinal detachments, or anomalies in the vitreous, are present in most forms of Stickler syndrome. In this case report, we present a patient with an unusual retinal phenotype.</p><p><strong>Case presentation: </strong>Subject of this case report is a 33-year-old woman, who was examined at the Department of Ophthalmology at Medical University of Graz. A thorough ophthalmological examination was conducted, detailed medical and family history acquired, and genetic testing performed. Best corrected visual acuity was 20/20 on both eyes; however, impaired binocular vision associated with intermittent exotropia was found. Furthermore, dilated fundoscopy showed an unusual, hypopigmented spotted retinal phenotype. Fundus autofluorescence showed multiple hyperfluorescent spots corresponding with the spotted retinal appearance. Genetic testing revealed a novel variant in the gene <i>COL11A1</i>. No other ocular abnormalities which are associated with <i>COL11A1</i> were found.</p><p><strong>Conclusion: </strong>Several subtypes of Stickler syndrome have been reported in medical literature, greatly varying in clinical manifestations. Many different mutations in the gene <i>COL11A1</i> have been discovered and are typically associated with Stickler syndrome type 2. To our best knowledge, this is the first report of a patient with a mutation in the <i>COL11A1</i> gene presenting with a hypopigmented spotted retina.</p>","PeriodicalId":9635,"journal":{"name":"Case Reports in Ophthalmology","volume":"16 1","pages":"62-67"},"PeriodicalIF":0.5,"publicationDate":"2025-01-10","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11842027/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143467064","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 : 2025-01-07eCollection Date: 2025-01-01DOI: 10.1159/000543239
Feliciana Menna, Marcel Menke, Markus Tschopp
Introduction: Various surgical techniques, including 360° buckling surgery with a retinal cerclage, have been employed to achieve retinal reattachment. Although retinal cerclage is generally effective, long-term complications can arise. Peripheral retinal ischemia and secondary neovascular glaucoma are rare, but serious complications can occur even years after successful retinal reattachment.
Case presentation: We report a rare case of a 79-year-old woman who underwent 360° buckling surgery with cerclage for retinal detachment 10 years ago. Although the initial surgery successfully reattached the retina, she later developed a complication characterized by peripheral retinal ischemia and secondary neovascular glaucoma.
Conclusion: Early detection and prompt management of such complications are crucial to prevent irreversible visual impairment.
{"title":"Peripheral Retinal Ischemia and Secondary Neovascular Glaucoma after Cerclage for Retinal Detachment: A Case Report.","authors":"Feliciana Menna, Marcel Menke, Markus Tschopp","doi":"10.1159/000543239","DOIUrl":"10.1159/000543239","url":null,"abstract":"<p><strong>Introduction: </strong>Various surgical techniques, including 360° buckling surgery with a retinal cerclage, have been employed to achieve retinal reattachment. Although retinal cerclage is generally effective, long-term complications can arise. Peripheral retinal ischemia and secondary neovascular glaucoma are rare, but serious complications can occur even years after successful retinal reattachment.</p><p><strong>Case presentation: </strong>We report a rare case of a 79-year-old woman who underwent 360° buckling surgery with cerclage for retinal detachment 10 years ago. Although the initial surgery successfully reattached the retina, she later developed a complication characterized by peripheral retinal ischemia and secondary neovascular glaucoma.</p><p><strong>Conclusion: </strong>Early detection and prompt management of such complications are crucial to prevent irreversible visual impairment.</p>","PeriodicalId":9635,"journal":{"name":"Case Reports in Ophthalmology","volume":"16 1","pages":"86-94"},"PeriodicalIF":0.5,"publicationDate":"2025-01-07","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11842065/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143466649","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: Proliferative vitreoretinopathy (PVR) is a condition in which proliferation forms after rhegmatogenous retinal detachment (RRD) surgery or old RRD, and sometimes, PVR can occur under silicone oil (SO). Here, we report 2 cases of PVR in which we evaluated preoperative B-scan images of proliferation under SO using ultra-widefield swept-source OCT (UWF-SS-OCT).
Case presentation: The first case was a 61-year-old male who presented with PVR under SO tamponade after multiple pars plana vitrectomies (PPVs) at the previous hospital, and the second case was a 23-year-old man patient who was diagnosed with Coats disease in childhood and presented with PVR under SO after PPV for total RRD at our hospital. B-scan images of UWF-SS-OCT provided findings of epiretinal two-layered proliferation or emulsified SO in the peripheral area.
Conclusion: UWF-SS-OCT is useful for preoperative assessment of the proliferation of PVR before SO removal. In addition, peculiar proliferation may occur in the eye under SO.
{"title":"Ultra-Widefield OCT Images of Proliferative Vitreoretinopathy under Silicone Oil.","authors":"Masaki Kinoshita, Susumu Sakimoto, Masaki Suzue, Masanori Kanai, Nobuhiko Shiraki, Kohji Nishida","doi":"10.1159/000542391","DOIUrl":"10.1159/000542391","url":null,"abstract":"<p><strong>Introduction: </strong>Proliferative vitreoretinopathy (PVR) is a condition in which proliferation forms after rhegmatogenous retinal detachment (RRD) surgery or old RRD, and sometimes, PVR can occur under silicone oil (SO). Here, we report 2 cases of PVR in which we evaluated preoperative B-scan images of proliferation under SO using ultra-widefield swept-source OCT (UWF-SS-OCT).</p><p><strong>Case presentation: </strong>The first case was a 61-year-old male who presented with PVR under SO tamponade after multiple pars plana vitrectomies (PPVs) at the previous hospital, and the second case was a 23-year-old man patient who was diagnosed with Coats disease in childhood and presented with PVR under SO after PPV for total RRD at our hospital. B-scan images of UWF-SS-OCT provided findings of epiretinal two-layered proliferation or emulsified SO in the peripheral area.</p><p><strong>Conclusion: </strong>UWF-SS-OCT is useful for preoperative assessment of the proliferation of PVR before SO removal. In addition, peculiar proliferation may occur in the eye under SO.</p>","PeriodicalId":9635,"journal":{"name":"Case Reports in Ophthalmology","volume":"16 1","pages":"57-61"},"PeriodicalIF":0.5,"publicationDate":"2025-01-07","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11842007/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143467099","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}