American Journal of Ophthalmology Case Reports最新文献_第6页

Amaurosis fugax progressing to retinal artery occlusion with anterior migration of a retrobulbar embolus on orbital color Doppler imaging 眼眶彩色多普勒成像显示黑朦进展为视网膜动脉闭塞伴球后栓塞前移

American Journal of Ophthalmology Case Reports Pub Date : 2025-07-22 DOI: 10.1016/j.ajoc.2025.102395

Ari H. August , Turner D. Wibbelsman , Erik Massenzio , Sarah E. Thornton , Danijel J. Peričić

{"title":"Amaurosis fugax progressing to retinal artery occlusion with anterior migration of a retrobulbar embolus on orbital color Doppler imaging","authors":"Ari H. August , Turner D. Wibbelsman , Erik Massenzio , Sarah E. Thornton , Danijel J. Peričić","doi":"10.1016/j.ajoc.2025.102395","DOIUrl":"10.1016/j.ajoc.2025.102395","url":null,"abstract":"<div><h3>Purpose</h3><div>Orbital color Doppler imaging (CDI) is useful in the evaluation of sudden monocular vision loss, providing information on etiology which may guide management. We present two cases of amaurosis fugax progressing to retinal artery occlusion (RAO) associated with migration of a hyperechoic particle within the central retinal artery (CRA) and altered vascular dynamics found on CDI.</div></div><div><h3>Observations</h3><div>Both patients presented with amaurosis fugax, and CDI revealed a hyperechoic particle 2.8 mm from the optic nerve head in both patients. Patient 1 was found to have severe aortic stenosis and a thoracic aortic aneurysm and was managed with dual antiplatelet therapy (DAPT) while awaiting evaluation for cardiothoracic surgical repair. Ten days later, Patient 1 returned with a central RAO, and a repeat CDI showed a 1.0 mm anterior migration of the embolus with reduced CRA blood velocity and an increased resistivity index. Patient 2 was managed with DAPT and oral corticosteroids, but symptoms recurred during steroid tapering which necessitated a prolonged course of steroids. Systemic complications required reduction of steroid dosing, and the patient developed a branch RAO six months after initial presentation. Repeat CDI revealed a 0.9 mm anterior migration of the embolus, with increased CRA blood velocity and resistivity index. Systemic thrombolysis with tissue plasminogen activator and resumption of steroids did not result in visual improvement in Patient 2.</div></div><div><h3>Conclusions and importance</h3><div>The presence of a hyperechoic particle in the CRA on CDI can be seen with amaurosis fugax, and anterior migration with subsequent alterations in CDI parameters may correlate with clinical progression to embolic retinal ischemia. Visualization of an embolus may predict nonresponse to thrombolytic or anticoagulation-based treatment.</div></div>","PeriodicalId":7569,"journal":{"name":"American Journal of Ophthalmology Case Reports","volume":"39 ","pages":"Article 102395"},"PeriodicalIF":0.0,"publicationDate":"2025-07-22","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144721156","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}

引用次数: 0

Concurrent Wagner syndrome and retinopathy of prematurity 并发Wagner综合征与早产儿视网膜病变

American Journal of Ophthalmology Case Reports Pub Date : 2025-07-22 DOI: 10.1016/j.ajoc.2025.102392

Landon J. Rohowetz, David W. Redick, Kenneth C. Fan, Audina M. Berrocal

{"title":"Concurrent Wagner syndrome and retinopathy of prematurity","authors":"Landon J. Rohowetz, David W. Redick, Kenneth C. Fan, Audina M. Berrocal","doi":"10.1016/j.ajoc.2025.102392","DOIUrl":"10.1016/j.ajoc.2025.102392","url":null,"abstract":"<div><h3>Purpose</h3><div>To describe the clinical features of a patient with Wagner syndrome and a history of retinopathy of prematurity (ROP).</div></div><div><h3>Observations</h3><div>A 23-year-old Hispanic male was referred for retina evaluation. The patient had a history of regressed ROP in both eyes that was never treated. The patient was born at 26-weeks gestational age and received supplemental oxygen as a neonate. He also reported a history of strabismus treated with surgery at 6 years of age. Best-corrected visual acuity was 20/30 in both eyes. Manifest refraction revealed −8.25 diopters of myopia in both eyes. Posterior segment examination demonstrated vitreous syneresis, a regressed temporal ridge, pigmented lattice degeneration, and atrophic holes in both eyes. Fluorescein angiography revealed temporal small vessel leakage and staining in both eyes without exudation. Electroretinography demonstrated reduced a- and b-wave amplitudes in both eyes. Optical coherence tomography of the macula revealed a blunted foveal contour in both eyes. Genetic testing revealed a heterozygous versican (VCAN) mutation: c.425C > T (p.Thr142Met).</div></div><div><h3>Conclusion and importance</h3><div>The current case represents a rare combination of diseases and underscores the importance of considering concurrent retinal and vitreoretinal conditions in patients with a history of ROP.</div></div>","PeriodicalId":7569,"journal":{"name":"American Journal of Ophthalmology Case Reports","volume":"39 ","pages":"Article 102392"},"PeriodicalIF":0.0,"publicationDate":"2025-07-22","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144703769","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}

引用次数: 0

Immediate sequential corneal allogeneic intrastromal ring segment transplantation and cataract surgery 即刻序贯角膜异体间环段移植与白内障手术

American Journal of Ophthalmology Case Reports Pub Date : 2025-07-22 DOI: 10.1016/j.ajoc.2025.102398

Gerd U. Auffarth , Victor A. Augustin , Aylin Kilic , Ramin Khoramnia , Maximilian Friedrich , Hyeck-Soo Son

{"title":"Immediate sequential corneal allogeneic intrastromal ring segment transplantation and cataract surgery","authors":"Gerd U. Auffarth , Victor A. Augustin , Aylin Kilic , Ramin Khoramnia , Maximilian Friedrich , Hyeck-Soo Son","doi":"10.1016/j.ajoc.2025.102398","DOIUrl":"10.1016/j.ajoc.2025.102398","url":null,"abstract":"<div><h3>Purpose</h3><div>To report the combined approach of corneal allogeneic intrastromal ring segment (CAIRS) transplantation and cataract surgery in a cataract patient with corneal ectasia.</div></div><div><h3>Case report</h3><div>A 75-year-old male presented with age-related corticonuclear cataract, high myopia, and corneal ectasia on his right eye. After detailed discussion of treatment options, the patient wished to undergo a combined approach of CAIRS procedure and cataract surgery on his right eye. A femtosecond-laser (Wavelight FS200, Alcon, USA) was used to create a corneal pocket at mid-stromal depth and incision at 190°. Then, cataract surgery was performed with a 2.5-mm corneal incision, manual capsulorhexis, and phacoemulsification. A 3-piece intraocular lens (IOL) with a dioptric power of 0 diopter (D) (target refraction: −3.0 D) was implanted into the capsular bag. After cataract surgery, an 8-mm CAIRS tissue with 160° arc and 500 μm thickness was inserted (KeraNatural, VisionGift, USA) into the stromal pocket. Follow-up examination at 3-months postoperatively demonstrated improved uncorrected (UDVA) and corrected distance visual acuity (CDVA), topographic parameters, as well as high satisfaction rate.</div></div><div><h3>Conclusions</h3><div>Our case report shows that an immediate sequential CAIRS transplantation and cataract surgery may be a viable option in managing cataract patients with corneal ectasia.</div></div>","PeriodicalId":7569,"journal":{"name":"American Journal of Ophthalmology Case Reports","volume":"39 ","pages":"Article 102398"},"PeriodicalIF":0.0,"publicationDate":"2025-07-22","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144711039","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}

引用次数: 0

Case report of spontaneous corneal clearance after subtotal graft detachment following combined Descemet's membrane endothelial keratoplasty and cataract surgery Descemet膜内皮角膜移植术联合白内障手术后角膜次全脱离后自发性角膜清除1例报告

American Journal of Ophthalmology Case Reports Pub Date : 2025-07-16 DOI: 10.1016/j.ajoc.2025.102385

Zehra Rizvi , Alin Megerdichian , Matthew Goldman , Frank Hwang

{"title":"Case report of spontaneous corneal clearance after subtotal graft detachment following combined Descemet's membrane endothelial keratoplasty and cataract surgery","authors":"Zehra Rizvi , Alin Megerdichian , Matthew Goldman , Frank Hwang","doi":"10.1016/j.ajoc.2025.102385","DOIUrl":"10.1016/j.ajoc.2025.102385","url":null,"abstract":"<div><h3>Purpose</h3><div>Fuchs’ Endothelial Corneal Dystrophy (FECD) is a bilateral, sporadic or autosomal dominant, non-inflammatory corneal dystrophy characterized by the progressive loss of corneal endothelial cells. While DMEK has been proven to attain better post-operative best corrected visual acuity (BCVA), it has presented the challenge of more frequent postoperative graft detachments.<sup>1,2</sup> Combined cataract surgery along with DMEK has also been reported to increase the risk of early postoperative graft detachment.<sup>3</sup>While several case reports have illustrated spontaneous corneal clearance after significant graft detachment, the question regarding how much donor tissue is required to achieve and maintain corneal clearance and to ensure long term optimal visual outcomes remains.</div></div><div><h3>Methods</h3><div>In this case report, we describe an event of spontaneous corneal clearance after near subtotal graft detachment noted in the early postoperative period in a patient with FECD who underwent combined DMEK with a 7.5 mm donor graft and intraocular lens placement.</div></div><div><h3>Results</h3><div>On postoperative week 2, the patient presented with count fingers vision, diffuse microcystic edema, and 75 % detachment of the DMEK graft. At postoperative month 6, the patient presented with vision of 20/30, a clear cornea, DMEK graft one fourth attached and folded over on itself in the anterior chamber.</div></div><div><h3>Conclusion</h3><div>Our case highlights spontaneous corneal clearance after more than three-quarters detachment of the donor graft following combined DMEK and cataract surgery. Although we cannot predict long term corneal transparency in our patient, the stability at 6 months implies that there is endothelial cell migration in patients with minimal DMEK graft attachment.</div></div>","PeriodicalId":7569,"journal":{"name":"American Journal of Ophthalmology Case Reports","volume":"39 ","pages":"Article 102385"},"PeriodicalIF":0.0,"publicationDate":"2025-07-16","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144680325","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}

引用次数: 0

Corneally displaced conjunctival Melanoma: Rare presentations of recurrent melanoma 角膜移位性结膜黑色素瘤：复发性黑色素瘤的罕见表现

American Journal of Ophthalmology Case Reports Pub Date : 2025-07-15 DOI: 10.1016/j.ajoc.2025.102387

Eric Boya Lee , Ahmad Al-Moujahed , Michael D. Yu , Christopher N. Ta , Jonathan H. Lin , Prithvi Mruthyunjaya

引用次数: 0

Successful treatment of bilateral familial iris flocculi associated with aortic valve abnormalities 双侧家族性虹膜絮凝伴主动脉瓣异常的成功治疗

American Journal of Ophthalmology Case Reports Pub Date : 2025-07-12 DOI: 10.1016/j.ajoc.2025.102386

Liam D. Redden , Hesham Lakosha , Kamran M. Riaz

{"title":"Successful treatment of bilateral familial iris flocculi associated with aortic valve abnormalities","authors":"Liam D. Redden , Hesham Lakosha , Kamran M. Riaz","doi":"10.1016/j.ajoc.2025.102386","DOIUrl":"10.1016/j.ajoc.2025.102386","url":null,"abstract":"<div><h3>Purpose</h3><div>To report a case of familial iris flocculi in siblings associated with a possibly life-threatening aortic valve manifestation due to a smooth muscle alpha-actin 2 (<em>ACTA2</em>) gene variant and demonstrate successful treatment of iris flocculi with laser.</div></div><div><h3>Observations</h3><div>Two biological siblings, a 21-year-old male and a 24-year-old female, both presented with bilateral iris flocculi. The flocculi were causing visual disturbances for both patients. Treatment was provided in the form of yttrium aluminum garnet (YAG) laser to the cysts and remained stable at 2 year follow up. Cardiac investigations were launched and the female patient demonstrated a bicuspid aortic valve with a mildly dilated ascending aorta. Cardiac investigations for the male patient were normal. Gene sequencing revealed a pathogenic variant in the <em>ACTA2</em> gene.</div></div><div><h3>Conclusions and importance</h3><div>Bilateral iris flocculi is a rare ocular condition that can demonstrate a familial component. Treatment of the cysts is required only when there are visual symptoms. We demonstrate successful long term treatment using Nd: YAG laser. There is a link between iris flocculi and a variant of <em>ACTA2</em> and/or the smooth muscle (<em>MYH11</em>) genes. Patients with iris flocculi should have careful cardiac assessment looking for aortic anomalies as well as gene sequencing. Family members should be made aware and investigated.</div></div>","PeriodicalId":7569,"journal":{"name":"American Journal of Ophthalmology Case Reports","volume":"39 ","pages":"Article 102386"},"PeriodicalIF":0.0,"publicationDate":"2025-07-12","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144655830","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}

引用次数: 0

Biopsy-proven sarcoidosis lesion of the levator palpebrae superioris causing myogenic blepharoptosis 活检证实提上睑肌结节病病变引起肌源性上睑下垂

American Journal of Ophthalmology Case Reports Pub Date : 2025-07-12 DOI: 10.1016/j.ajoc.2025.102390

Kim A. Firn, Eman Hawy, Douglas J. Van Putten

{"title":"Biopsy-proven sarcoidosis lesion of the levator palpebrae superioris causing myogenic blepharoptosis","authors":"Kim A. Firn, Eman Hawy, Douglas J. Van Putten","doi":"10.1016/j.ajoc.2025.102390","DOIUrl":"10.1016/j.ajoc.2025.102390","url":null,"abstract":"<div><h3>Purpose</h3><div>We describe a case of myogenic blepharoptosis due to sarcoidosis directly involving the levator palpebrae superioris, confirmed with biopsy.</div></div><div><h3>Observations</h3><div>A 43-year-old male with hypertension and hyperlipidemia presented with four months of progressive right blepharoptosis and two months of dry cough. Workup revealed elevated serum muramidase, hilar adenopathy, and bilateral lacrimal gland and right levator MRI enhancement. Both lacrimal gland biopsy and later levator biopsy were consistent with sarcoidosis.</div></div><div><h3>Conclusions and importance</h3><div>Most sarcoidosis-related blepharoptosis is secondary to Horner syndrome or mass effect from lacrimal gland enlargement. On literature review, we did not identify prior biopsy-confirmed direct involvement of the levator as a cause of sarcoidosis-related blepharoptosis. No cases were identified with isolated levator enhancement without involvement of other extraocular muscles. In this case, both the lacrimal gland and levator biopsies were consistent with sarcoidosis. It is possible that prior reports of sarcoidosis-related blepharoptosis secondary to lacrimal gland enlargement with positive biopsy were in fact due to direct co-involvement of the levator. The exact mechanism of sarcoidosis-related blepharoptosis impacts the proposed surgical intervention if indicated after failed medical therapy, and direct levator involvement should be considered prior to surgical planning.</div></div>","PeriodicalId":7569,"journal":{"name":"American Journal of Ophthalmology Case Reports","volume":"39 ","pages":"Article 102390"},"PeriodicalIF":0.0,"publicationDate":"2025-07-12","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144655829","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}

引用次数: 0

Removal of subretinal strands without creating an intentional retinal hole: A case report 去除视网膜下链而不造成有意的视网膜孔：1例报告

American Journal of Ophthalmology Case Reports Pub Date : 2025-07-12 DOI: 10.1016/j.ajoc.2025.102388

Sonoko Sakata , Yoshihito Sakanishi , Kenji Inoue , Nobuyuki Ebihara

{"title":"Removal of subretinal strands without creating an intentional retinal hole: A case report","authors":"Sonoko Sakata , Yoshihito Sakanishi , Kenji Inoue , Nobuyuki Ebihara","doi":"10.1016/j.ajoc.2025.102388","DOIUrl":"10.1016/j.ajoc.2025.102388","url":null,"abstract":"<div><h3>Background</h3><div>Proliferative vitreoretinopathy (PVR) is a severe vitreoretinal disease. In cases of PVR with subretinal strands (SRS), creating an intentional retinal hole is typically necessary for removing SRS during vitrectomy. Herein, we describe a novel surgical approach for managing total retinal detachment (RD) with SRS.</div></div><div><h3>Case report</h3><div>A 59-year-old man presented with vision loss in the right eye persisting for four years before the initial presentation. Visual acuity was limited to light perception. The right eye had a mature cataract, and ultrasound computed tomography indicated total retinal detachment. Consequently, combined cataract surgery and vitrectomy were performed on the right eye. After cataract surgery, vitrectomy was performed using a 27-gauge, four-port system. SRS were identified in all quadrants. A fifth port was created approximately 12 mm from the corneal limbus, facilitating the removal of SRS without an intentional retinal hole. Almost all the SRS were successfully removed through this port using vitreous forceps. Postoperatively, the fundus remained stable without complications such as choroidal hemorrhage. No retinal re-detachment was observed during 13 months of follow-up.</div></div><div><h3>Conclusion</h3><div>The subretinal port provided an effective means of removing SRS without creating an intentional retinal hole in this case of total RD with SRS. This technique could be applicable to similar cases of total RD.</div></div>","PeriodicalId":7569,"journal":{"name":"American Journal of Ophthalmology Case Reports","volume":"39 ","pages":"Article 102388"},"PeriodicalIF":0.0,"publicationDate":"2025-07-12","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144655831","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}

引用次数: 0

The use of trypan blue to distinguish Pseudo-Seidel sign from lacrimal ductule versus glaucoma drainage device leakage 台盼蓝鉴别泪道小管伪赛德尔征与青光眼引流器渗漏

American Journal of Ophthalmology Case Reports Pub Date : 2025-07-12 DOI: 10.1016/j.ajoc.2025.102389

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}

引用次数: 0

Therapeutic potential of allogeneic iPS cell-derived RPE transplantation for RPE65-LCA 异体iPS细胞来源的RPE移植治疗RPE65-LCA的潜力

American Journal of Ophthalmology Case Reports Pub Date : 2025-07-09 DOI: 10.1016/j.ajoc.2025.102383

Akiko Maeda , Daiki Sakai , Satoshi Yokota , Midori Yamamoto , Sunao Sugita , Yasuhiko Hirami , Michiko Mandai , Tadao Maeda , Masayo Takahashi , Yasuo Kurimoto

{"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}

引用次数: 0