Hamidah Mahmud , Yeabsira Mesfin , Yingna Liu , Yvonne Ou , Jonathan E. Lu
{"title":"The use of trypan blue to distinguish Pseudo-Seidel sign from lacrimal ductule versus glaucoma drainage device leakage","authors":"Hamidah Mahmud , Yeabsira Mesfin , Yingna Liu , Yvonne Ou , Jonathan E. Lu","doi":"10.1016/j.ajoc.2025.102389","DOIUrl":"10.1016/j.ajoc.2025.102389","url":null,"abstract":"<div><h3>Introduction</h3><div>The superotemporal fornix location of the lacrimal gland ductule openings coincides with the typical locations of glaucoma drainage devices; as a result, a conjunctival fistula may look and behave like a lacrimal gland ductule. External morphology and Seidel test are helpful in identifying fluid flow but cannot differentiate the fluid as aqueous humor or tears. We describe a novel technique in differentiating a lacrimal gland ductule from a conjunctival fistula secondary to a glaucoma drainage device.</div></div><div><h3>Case presentation</h3><div>An 87-year-old female with a superotemporal Ahmed valve in the right eye presented with increased “tearing” over the past year, with concern for repeat tube exposure versus normal secretion from the lacrimal gland ductules. Difficulty in differentiation was further increased due to a regional conjunctival pedicled flap for previous tube exposure. Intra-operatively, trypan blue was injected into the anterior chamber of the right eye. The dye was visualized to track along the tube in the direction of the plate, and brisk flow was then observed at the conjunctival area of ambiguity. This confirmed that fluid leakage was due to a glaucoma drainage device-associated conjunctival fistula. The plate and tube were subsequently removed.</div></div><div><h3>Conclusions</h3><div>Intraocular injection of trypan blue dye was effective in identifying glaucoma drainage device leakage from tube exposure, and specifically allowing clear differentiation from physiologic lacrimal gland flow. This novel technique successfully differentiated a leaking conjunctival fistula requiring treatment from a benign physiologic finding, in a case where topical fluorescein testing was not sufficient.</div></div>","PeriodicalId":7569,"journal":{"name":"American Journal of Ophthalmology Case Reports","volume":"39 ","pages":"Article 102389"},"PeriodicalIF":0.0,"publicationDate":"2025-07-12","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144655828","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
{"title":"Therapeutic potential of allogeneic iPS cell-derived RPE transplantation for RPE65-LCA","authors":"Akiko Maeda , Daiki Sakai , Satoshi Yokota , Midori Yamamoto , Sunao Sugita , Yasuhiko Hirami , Michiko Mandai , Tadao Maeda , Masayo Takahashi , Yasuo Kurimoto","doi":"10.1016/j.ajoc.2025.102383","DOIUrl":"10.1016/j.ajoc.2025.102383","url":null,"abstract":"<div><h3>Purpose</h3><div>To evaluate the safety and therapeutic effects of induced pluripotent stem (iPS) cell-derived retinal pigment epithelium (RPE) transplantation for <em>RPE65</em>-associated Leber congenital amaurosis (<em>RPE6</em>5-LCA).</div></div><div><h3>Observations</h3><div>A 46-year-old male patient with <em>RPE6</em>5-LCA underwent allogeneic iPS cell-derived RPE transplantation. The patient's best-corrected visual acuity (VA) prior to treatment was 2.0 (logMAR). A cell suspension of iPS cell-derived RPE was transplanted into the subretinal space. On day 15 post-transplantation, intraocular pressure (IOP) increased to 46 mmHg due to local steroid treatment, resulting in a decrease in VA to light perception (LP). Retinal imaging on day 71 revealed that most transplanted cells had migrated and formed an epiretinal membrane (ERM). The ERM was surgically removed on day 112. Two years post-transplantation, the patient reported improved vision, with VA improving to 1.4 (logMAR) from LP. Full-field stimulus testing (FST) and microperimetry demonstrated increased retinal sensitivity. These improvements have been maintained for up to 4 years post-treatment.</div></div><div><h3>Conclusions and importance</h3><div>Although this case raised safety concerns regarding the use of cell suspension for RPE transplantation, RPE transplantation may still serve as a potential therapeutic option for patients with <em>RPE65</em>-associated retinopathy, particularly those who are not eligible or older age for <em>RPE65</em> gene therapy.</div></div>","PeriodicalId":7569,"journal":{"name":"American Journal of Ophthalmology Case Reports","volume":"39 ","pages":"Article 102383"},"PeriodicalIF":0.0,"publicationDate":"2025-07-09","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144611581","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
Hashem H. Ghoraba, Chris Or, Aneesha Ahluwalia, Bethlehem Mekonnen, Cigdem Yasar, Negin Yavari, Quan Dong Nguyen
{"title":"Subconjunctival dexamethasone implant (Ozurdex) for peripheral ulcerative keratitis","authors":"Hashem H. Ghoraba, Chris Or, Aneesha Ahluwalia, Bethlehem Mekonnen, Cigdem Yasar, Negin Yavari, Quan Dong Nguyen","doi":"10.1016/j.ajoc.2025.102379","DOIUrl":"10.1016/j.ajoc.2025.102379","url":null,"abstract":"<div><h3>Purpose</h3><div>To report a case of peripheral ulcerative keratitis (PUK) successfully treated with subconjunctival dexamethasone implant (Ozurdex®) injection.</div></div><div><h3>Observations</h3><div>An 82-year-old female presented with PUK in the right eye. Systemic immunosuppressive treatment with steroids and methotrexate resulted in complete healing. The patient developed adverse events that led to discontinuation of systemic therapy. Nine months after presentation, the PUK recurred, and the patient declined to restart systemic therapy. Subconjunctival dexamethasone implant injection resulted in complete resolution. The patient received three injections over a period of 10 months. No scleral melt or ocular hypertension was noted 11 months after the first injection.</div></div><div><h3>Conclusions and importance</h3><div>Subconjunctival dexamethasone implant injection can be a well-tolerated treatment option for peripheral ulcerative keratitis.</div></div>","PeriodicalId":7569,"journal":{"name":"American Journal of Ophthalmology Case Reports","volume":"39 ","pages":"Article 102379"},"PeriodicalIF":0.0,"publicationDate":"2025-07-09","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144604093","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
Arthur S. Zupelli , Victor C.F. Bellanda , João Victor N. Arcanjo , Fernando Chahud , Raul N.G. Vianna , Rodrigo Jorge
{"title":"Macular hole closure following treatment of bilateral diffuse uveal melanocytic proliferation (BDUMP) with intravitreal methotrexate and systemic chemotherapy","authors":"Arthur S. Zupelli , Victor C.F. Bellanda , João Victor N. Arcanjo , Fernando Chahud , Raul N.G. Vianna , Rodrigo Jorge","doi":"10.1016/j.ajoc.2025.102382","DOIUrl":"10.1016/j.ajoc.2025.102382","url":null,"abstract":"<div><h3>Purpose</h3><div>This report presents an unusual case of bilateral diffuse uveal melanocytic proliferation (BDUMP) associated with a macular hole that resolved after choroidal thinning following intravitreal methotrexate and systemic chemotherapy.</div></div><div><h3>Case report</h3><div>A 78-year-old male presented with progressive visual deterioration, significant weight loss and dyspnea. Ophthalmologic examination revealed multiple elevated yellowish-white lesions and few vitreous cells in both eyes, with a macular hole in the left eye. Optical coherence tomography showed subretinal fluid, hyperreflective lesions and choroidal irregularities at posterior pole in both eyes. Systemic screening revealed a pulmonary nodule, prompting further investigation. A pleural biopsy disclosed diffuse infiltration by mature-looking lymphocytes, positive for CD20 and BCL-2, indicating a marginal zone B-cell lymphoma. The patient received systemic chemotherapy and off-label intravitreal methotrexate injections. Notably, the macular hole in the left eye spontaneously closed following treatment, leading to significant visual improvement.</div></div><div><h3>Discussion</h3><div>BDUMP poses diagnostic and therapeutic challenges necessitating a multidisciplinary approach. This case is notable for the mechanism of macular hole closure after intravitreal methotrexate and choroidal thinning. We believe that systemic chemotherapy also played an effective role in ocular symptoms management.</div></div>","PeriodicalId":7569,"journal":{"name":"American Journal of Ophthalmology Case Reports","volume":"39 ","pages":"Article 102382"},"PeriodicalIF":0.0,"publicationDate":"2025-07-08","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144632737","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
Andreas Arnold-Vangsted , Tomas Ilginis , Jens Folke Kiilgaard , Steffen Heegaard , Carsten Faber
{"title":"Vitreoretinal lymphoma presenting as amaurosis fugax","authors":"Andreas Arnold-Vangsted , Tomas Ilginis , Jens Folke Kiilgaard , Steffen Heegaard , Carsten Faber","doi":"10.1016/j.ajoc.2025.102380","DOIUrl":"10.1016/j.ajoc.2025.102380","url":null,"abstract":"<div><h3>Purpose</h3><div>To describe a case of vitreoretinal lymphoma with unilateral vascular involvement.</div></div><div><h3>Observations</h3><div>A 80-year-old male with recurrent amaurosis fugax (AF) in the left eye was seen in the ophthalmological department. During a period of seven months the patient developed central vein occlusion (CRVO) and later the panuveitis suspected to be acute retinal necrosis (ARN) before being diagnosed with vitreoretinal lymphoma (VRL). The patient preferred enucleation as treatment which allowed thorough histological examination revealing Epstein Barr virus-associated vitreoretinal lymphoma.</div></div><div><h3>Conclusions and importance</h3><div>Vascular involvement with AF and CRVO is yet to be described in relation to VRL. This case-study underlines the critical importance of considering VRL when the clinical course deviates from the expected.</div></div>","PeriodicalId":7569,"journal":{"name":"American Journal of Ophthalmology Case Reports","volume":"39 ","pages":"Article 102380"},"PeriodicalIF":0.0,"publicationDate":"2025-07-08","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144611579","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
Prabakaran Ganesh , Roxana A. Hojjatie , Sruthi Arepalli
{"title":"Bilateral acute retinal necrosis after ofatumumab masquerading as intermediate uveitis in a patient with multiple sclerosis","authors":"Prabakaran Ganesh , Roxana A. Hojjatie , Sruthi Arepalli","doi":"10.1016/j.ajoc.2025.102377","DOIUrl":"10.1016/j.ajoc.2025.102377","url":null,"abstract":"<div><h3>Purpose</h3><div>To report an unique case of bilateral acute retinal necrosis (ARN) associated with ofatumumab use masquerading as intermediate uveitis in a patient with multiple sclerosis.</div></div><div><h3>Observation</h3><div>A 47-year-old female with multiple sclerosis was transitioned to ofatumumab (Arzerra/Kesimpta, Novartis, Cambridge, MA, USA) for better systemic control of her disease. Two months later, she developed floaters with a scotoma in her right eye. Initially, she was diagnosed with intermediate uveitis secondary to a presumed multiple sclerosis flare. However, her symptoms worsened and she was referred to Emory University. Fundus examination revealed bilateral ARN. The patient discontinued the medication and was treated with systemic and intravitreal antivirals with improvement; however, she developed a retinal detachment in her right eye that required surgical repair.</div></div><div><h3>Conclusions and importance</h3><div>This is a unique case of bilateral ARN after starting ofatumumab medication for multiple sclerosis. This case also highlights the importance of discerning between intermediate uveitis associated with multiple sclerosis and infectious uveitis in the setting of immunocompromise.</div></div>","PeriodicalId":7569,"journal":{"name":"American Journal of Ophthalmology Case Reports","volume":"39 ","pages":"Article 102377"},"PeriodicalIF":0.0,"publicationDate":"2025-07-08","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144694671","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
{"title":"Preserflo™ microshunt for the treatment of refractory childhood glaucoma: A case series study","authors":"Yuri Tanaka , Shiho Kasahara , Ryo Tomita , Norie Nonobe , Kazuhide Kawase , Kenya Yuki","doi":"10.1016/j.ajoc.2025.102384","DOIUrl":"10.1016/j.ajoc.2025.102384","url":null,"abstract":"<div><h3>Purpose</h3><div>We report the outcomes of five patients with pediatric glaucoma, who were treated with Preserflo™ microshunt surgery.</div></div><div><h3>Methods</h3><div>This prospective study included five consecutive patients who underwent Preserflo™ microshunt surgery at the Nagoya University Hospital. The main outcome measures evaluated in this study were mean intraocular pressure (IOP), number of glaucoma medications, and best corrected visual acuity (BCVA). Paired t-tests with Bonferroni correction were used to compare the baseline and follow-up periods, where p < 0.05 was considered statistically significant.</div></div><div><h3>Results</h3><div>Average age at the time of surgery was 10 ± 4 years. Three patients had primary congenital glaucoma, one had Sturge-Weber syndrome, and one had juvenile open-angle glaucoma. Preoperative average IOP was 37.6 ± 4.7 mmHg. After microshunt surgery, IOP significantly decreased to 6.3 ± 1.2 mmHg in 1 day, 6.6 ± 1.7 mmHg in 1 week, 9.6 ± 6.1 mmHg in 1 month, 12.0 ± 7.4 mmHg in 3 months, 10.0 ± 5.2 mmHg in 6 months, 14.2 ± 8.7 mmHg in 9 months, and 10.5 ± 6.5 mmHg in 1 year (p < 0.01 for each paired <em>t</em>-test). One year postoperatively, we placed a Baerveldt glaucoma implant in case 1. No significant change was observed between the pre- and post-operative periods in BCVA. No postoperative complications, including choroidal detachment, shallow or flat anterior chamber, hypotony maculopathy, or conjunctival leak, were observed in this case series.</div></div><div><h3>Conclusion</h3><div>Microshunt surgery may be a useful option for treating glaucoma in pediatric patients, in whom trabeculotomy failed.</div></div>","PeriodicalId":7569,"journal":{"name":"American Journal of Ophthalmology Case Reports","volume":"39 ","pages":"Article 102384"},"PeriodicalIF":0.0,"publicationDate":"2025-07-08","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144604094","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
{"title":"Tay-Sachs disease in a child of indigenous Guatemalan-Mayan origin with macular brown spots and perifoveal whitening","authors":"Mya Abousy , Lauren Hucko , Audina Berrocal , Erick Vinicio Saenz","doi":"10.1016/j.ajoc.2025.102381","DOIUrl":"10.1016/j.ajoc.2025.102381","url":null,"abstract":"<div><h3>Purpose</h3><div>This report describes an unusual macular presentation of Tay-Sachs disease in a two-year-old female of Guatemalan-Mayan origin. This case serves to build upon the very limited literature regarding ophthalmic manifestations of Tay-Sachs disease in the non-Caucasian population.</div></div><div><h3>Observations</h3><div>The patient and family presented with concern for poor visual acuity and hyperacusis. A brown macular spot with perifoveal whitening was identified in both eyes on fundus examination. Laboratory studies revealed low hexosaminidase A levels, which confirmed the diagnosis of Tay-Sachs disease.</div></div><div><h3>Conclusions and importance</h3><div>This is an extremely rare case of Tay-Sachs disease in a patient of Guatemalan-Mayan origin diagnosed by enzyme studies and fundus examination. Further, it is one of the few documented cases of “brown spots” appreciated in association with Tay-Sachs disease, as opposed to the classic “cherry red spot”. This case presentation challenges the nomenclature of the “cherry red spot” to account for the variations in the fundus that may occur in different racial and ethnic groups that present with Tay-Sachs disease.</div></div>","PeriodicalId":7569,"journal":{"name":"American Journal of Ophthalmology Case Reports","volume":"39 ","pages":"Article 102381"},"PeriodicalIF":0.0,"publicationDate":"2025-07-08","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144611927","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
{"title":"Pigmented perivascular epithelioid cell tumors of the orbit with NONO::TFE3 fusion: Molecular evaluation and literature review","authors":"Naoko Takeda-Miyata , Ken-ichi Yoshida , Miho Shirono , Akihide Watanabe , Yoji Kukita , Chie Sotozono , Eiichi Konishi","doi":"10.1016/j.ajoc.2025.102378","DOIUrl":"10.1016/j.ajoc.2025.102378","url":null,"abstract":"<div><h3>Purpose</h3><div>To elucidate the clinical characteristics of rare orbital pigmented perivascular epithelioid cell tumors (PEComa) with <em>transcription factor E3</em> (<em>TFE3</em>) rearrangement through a literature review.</div></div><div><h3>Observations</h3><div>A 49-year-old female was referred to our hospital with a mass measuring 16 × 14 mm in the extraconal space of the right orbit. Suspecting hemangioma or distended hematoma clinically, the tumor was completely excised through the right nasal conjunctiva. Although initial histological evaluation raised the suspicion of malignant melanoma, subsequent immunohistochemistry and molecular analyses led to a diagnosis of <em>TFE3</em>-rearranged PEComa with the <em>NONO</em>::<em>TFE3</em> fusion gene. The patient's visual function was preserved 14 months postoperatively, with no evidence of recurrence.</div></div><div><h3>Conclusions and importance</h3><div>Although rare, <em>TFE3-</em>rearranged PEComa should be considered in the differential diagnosis of pigmented orbital tumors.</div></div>","PeriodicalId":7569,"journal":{"name":"American Journal of Ophthalmology Case Reports","volume":"39 ","pages":"Article 102378"},"PeriodicalIF":0.0,"publicationDate":"2025-07-08","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144611580","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
{"title":"Incidental detection of retinal nerve fiber layer thinning with slowed progression following optic nerve-carotid artery decompression: A case report","authors":"Hidenori Takahashi , Keisuke Ohtani , Takeshi Hara , Kensuke Kawai","doi":"10.1016/j.ajoc.2025.102376","DOIUrl":"10.1016/j.ajoc.2025.102376","url":null,"abstract":"<div><h3>Purpose</h3><div>This report aims to present a case of the retinal nerve fiber layer (NFL) thinning, in which the progression of thinning was slowed following optic nerve-carotid artery decompression surgery.</div></div><div><h3>Observations</h3><div>A 41-year-old ophthalmologist incidentally noticed a generalized thinning of NFL in his left eye using optical coherence tomography (OCT). Five years earlier, the OCT showed only mild thinning. While visual acuity and visual fields were normal, the patient reported a blurriness. The MRI revealed an ectatic and tortuous left internal carotid artery compressing the left optic nerve. Due to the lack of reports on the correlation between NFL thickness and visual acuity, an internal review was conducted. We searched for cases with a progression from healthy visual acuity to blindness, and in the only case identified, we investigated the correlation between NFL thickness and visual acuity. Applying this correlation, it was predicted that the patient would lose vision by age 48. As a result, the patient decided to receive decompression surgery. Following the surgery, the rate of NFL thinning significantly slowed from −1.9 μm/year to −0.57 μm/year (p < .0001). At the current age of 48, the patient's corrected visual acuity is 20/16, and visual field remains normal. The predicted onset of blindness has been postponed by 10 years.</div></div><div><h3>Conclusions and importance</h3><div>We report a case in which compression of the optic nerve by the internal carotid artery was observed in a patient with retinal nerve fiber layer (NFL) thinning. A slowing of NFL thinning was noted following surgical decompression.</div></div>","PeriodicalId":7569,"journal":{"name":"American Journal of Ophthalmology Case Reports","volume":"39 ","pages":"Article 102376"},"PeriodicalIF":0.0,"publicationDate":"2025-07-08","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144632826","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}