Case Reports in Ophthalmology最新文献

{"title":"Probable Arteritic Posterior Ischemic Optic Neuropathy with Atypical Recovery Secondary to Biologically Occult Giant Cell Arteritis: A Case Report.","authors":"Alexandre Balon, Thyago Machado, Mohamed-Amine Ouacha, Aurélie Ledoux","doi":"10.1159/000552018","DOIUrl":"10.1159/000552018","url":null,"abstract":"<p><strong>Introduction: </strong>Posterior ischemic optic neuropathy (PION) is a rare form of ischemic optic neuropathy, and arteritic PION secondary to giant cell arteritis (GCA) usually has a poor prognosis.</p><p><strong>Case presentation: </strong>We report a rare case of probable arteritic PION secondary to GCA with an atypically slow course and remarkable visual recovery. A 66-year-old man under mild chronic immunosuppression for autologous bone marrow transplantation presented with progressive visual loss and normal inflammatory markers (erythrocyte sedimentation rate [ESR] 14 mm/h, C-reactive protein [CRP] 1.10 mg/L). Contrast-enhanced orbital and cerebral MRI showed no vasculitic or perioptic abnormalities. Despite the atypical presentation, temporal artery biopsy performed 10 days after high-dose corticosteroid initiation confirmed healed GCA. The patient experienced major recovery of central vision (visual acuity 0.9) after 1 month of treatment.</p><p><strong>Conclusion: </strong>This case highlights the diagnostic challenge of biologically and radiologically silent GCA, the potential modifying role of chronic immunosuppression on disease course and recovery, and the importance of maintaining suspicion for arteritic etiologies and considering temporal artery biopsy even when ESR, CRP, and MRI are normal.</p>","PeriodicalId":9635,"journal":{"name":"Case Reports in Ophthalmology","volume":"17 1","pages":"570-575"},"PeriodicalIF":0.6,"publicationDate":"2026-04-18","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC13299154/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"148344020","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}

{"title":"Bilateral, Simultaneous Common Canalicular Obstruction Occurring 10 Years after Endoscopic Dacryocystorhinostomy: A Case Report.","authors":"Jai Paris, Abdullah I Almater, Dinesh Selva","doi":"10.1159/000552100","DOIUrl":"10.1159/000552100","url":null,"abstract":"<p><strong>Introduction: </strong>Dacryocystorhinostomy (DCR) is the gold standard treatment for nasolacrimal duct obstruction, with high long-term success rates. Failure most commonly occurs early, while late complications are rare and less well characterised. Common canalicular obstruction is an increasingly recognised cause of delayed failure.</p><p><strong>Case presentation: </strong>This report describes a 71-year-old female who presented 10 years after sequential bilateral endoscopic DCR with spontaneous, simultaneous recurrence of bilateral epiphora. Endoscopic examination demonstrated patent nasal ostia with bilateral common canalicular obstruction. The patient had no identifiable risk factors for canalicular disease. She underwent revision bilateral endoscopic DCR with membranotomy and silicone intubation, with intraoperative findings confirming distal membranous obstruction. Complete symptom resolution was achieved at follow-up.</p><p><strong>Conclusion: </strong>This case highlights that late DCR failure may occur even after a decade and may be attributable to common canalicular obstruction. The unusual bilateral and simultaneous presentation raises the possibility of underlying inflammatory mechanisms. Clinicians should maintain long-term awareness of this rare complication.</p>","PeriodicalId":9635,"journal":{"name":"Case Reports in Ophthalmology","volume":"17 1","pages":"687-691"},"PeriodicalIF":0.6,"publicationDate":"2026-04-18","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC13391063/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"148560883","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}

{"title":"Subretinal Peripapillary Hemorrhage Suggestive of Terson Syndrome after Blunt Head Trauma: A Case Report.","authors":"Filip Slezak, Marcel N Menke, Tatjana Josifova","doi":"10.1159/000551343","DOIUrl":"10.1159/000551343","url":null,"abstract":"<p><strong>Introduction: </strong>This case report describes a rare manifestation of subretinal peripapillary hemorrhage after a \"trivial\" eye injury, suggesting Terson syndrome. The patient, a 42-year-old male, exhibited both ophthalmologic and neurologic symptoms, emphasizing the importance of interdisciplinary assessment in such cases. Despite normal initial visual acuity (VA), the patient experienced sharp retrobulbar pain, photosensitivity, and reduced subjective VA. Multimodal imaging and neurophysiological testing, including optical coherence tomography (OCT) scan, fluorescein angiography (FA), magnetic resonance imaging scan, visual evoked potential (VEP) test, retinal nerve fiber layer (RNFL), and perimetry, were utilized to diagnose the condition. This case highlights the need for prolonged monitoring as symptoms can evolve despite an initially trivial presentation.</p><p><strong>Case presentation: </strong>We report on a 42-year-old male who presented with sharp retrobulbar pain in his right eye, exacerbated by exertion and movement, along with photosensitivity and subjective reduction in VA. The patient had a history of traumatic brain injury (commotio cerebri) from an accidental impact 12 days prior. A comprehensive clinical examination was conducted, including ophthalmologic imaging (OCT, FA; RNFL) and neurophysiological testing (VEP, magnetic resonance imaging), revealing a large intraretinal hemorrhage surrounding the optic nerve. Indirect ophthalmoscopy (non-contact lens, 90-diopter lens) identified significant intraretinal bleeding in the peripapillary region of the right eye with confirmed large subretinal hematoma in the OCT Scan and with minimal optic nerve edema with venous congestion and irregular filling of the vena centralis inferior (inferonasal retinal vein), during the early phase of the FA. Initial examination revealed a Snellen VA of 1.0 in the right eye and 0.1 in the left, the latter due to severe amblyopia. Consecutive VEP testing showed normal initial VA, but the patient's neuro-ophthalmologic symptoms evolved, resulting in persistent concentration difficulties and neuro-ophthalmologic deficits.</p><p><strong>Conclusion: </strong>This case illustrates the complexity and potential severity of \"trivial\" eye injuries, particularly when associated with blunt head trauma. The interdisciplinary approach is crucial for managing such cases due to the risk of evolving neuro-ophthalmologic symptoms. Regular follow-up is recommended to monitor and address new developments in the patient's condition.</p>","PeriodicalId":9635,"journal":{"name":"Case Reports in Ophthalmology","volume":"17 1","pages":"455-463"},"PeriodicalIF":0.6,"publicationDate":"2026-04-17","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC13241079/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"148197363","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}

{"title":"Tractional Retinoschisis: A Subtle and Rare Manifestation of X-Linked Alport Syndrome - A Case Report.","authors":"Diogo Valente Fortunato, João Vasco Garrido, João Mendes","doi":"10.1159/000551894","DOIUrl":"10.1159/000551894","url":null,"abstract":"<p><strong>Introduction: </strong>Alport syndrome is a genetic condition characterized by chronic kidney disease, hearing loss, and a wide range of ophthalmological alterations. We describe the case of a patient with X-linked Alport syndrome whose main ocular manifestation was peripapillary retinoschisis.</p><p><strong>Case presentation: </strong>A 60-year-old woman was observed in our department after suspicion for Alport syndrome was raised in her son. She had no visual complaints. On observation, the patient had 20/25 Snellen best corrected visual acuity in both eyes and mild peripheral fleck retinopathy. Optical coherence tomography revealed the presence of bilateral peripapillary retinoschisis, multiple cysts within the retinal nerve fiber layer on both eyes, and vitreomacular adhesion on the left eye. Full-field electroretinogram was considered normal and autofluorescence retinography depicted mottled hyper and hypo-autofluorescent areas in the peripheral retina bilaterally. Genetic study identified a specific undescribed COL4A5 mutation in heterozygosity in the patient and in hemizygosity in her son.</p><p><strong>Conclusion: </strong>Alport syndrome is frequently associated with fleck retinopathy and lenticonus, but a wide range of ocular manifestations may be present. It is important to identify rarer and more subtle ocular phenotypes, especially among women with X-linked Alport syndrome, who tend to be underdiagnosed.</p>","PeriodicalId":9635,"journal":{"name":"Case Reports in Ophthalmology","volume":"17 1","pages":"518-525"},"PeriodicalIF":0.6,"publicationDate":"2026-04-10","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC13275058/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"148276169","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}

{"title":"Gasperini Syndrome: A Rare Brainstem Syndrome - A Case Report.","authors":"Bianca Sarah Stenitzer, Daniela Fruehwirth-Kaspar, Martina Brandner","doi":"10.1159/000551798","DOIUrl":"10.1159/000551798","url":null,"abstract":"<p><strong>Introduction: </strong>This unique case demonstrates how subtle ocular motor abnormalities, even in the absence of visual complaints, can lead to the detection of an otherwise clinically silent brainstem lesion.</p><p><strong>Case presentation: </strong>We report on the incidental discovery of a brain tumor following a traffic accident. Despite the absence of ocular symptoms, abnormal oculomotor findings led to further diagnostic investigations and rapid diagnosis of a pontine lesion consistent with Gasparini syndrome.</p><p><strong>Conclusion: </strong>Our case report highlights the crucial importance of meticulous history-taking, precise neuro-ophthalmological examination, particularly the assessment of ocular motility and interdisciplinary management in patients with complex brainstem syndromes.</p>","PeriodicalId":9635,"journal":{"name":"Case Reports in Ophthalmology","volume":"17 1","pages":"402-407"},"PeriodicalIF":0.6,"publicationDate":"2026-04-03","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC13143271/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"147834186","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}

{"title":"Bromfenac with Sodium Bisulfite Inducing Contact Dermatitis and Corneal Ring Infiltrate: A Case Report.","authors":"Christopher D Gelston, John J Fowler, Brittany M Perzia","doi":"10.1159/000551524","DOIUrl":"10.1159/000551524","url":null,"abstract":"<p><strong>Introduction: </strong>The aim of the study was to report a case of bromfenac with sodium bisulfite preservative induced contact dermatitis and a secondary sterile corneal ring infiltrate.</p><p><strong>Case presentation: </strong>A 44-year-old male presented with a 1-week unilateral periocular dermatitis and eye redness. Initially the dermatitis responded to oral prednisone 80 mg and subsequent taper over 2 weeks. Upon tapering the prednisone, the patient returned with a recurrent dermatitis a week later. Dermatological workup included a skin biopsy demonstrating spongiotic dermatitis and a skin panel allergy test positive for sodium metabisulfite. The patient later mentioned self-administering bromfenac ophthalmic eye drops that contained sodium bisulfite. After the prolonged bromfenac use, the patient also developed a sterile corneal ring ulcer. The dermatitis finally resolved after discontinuing the bromfenac ophthalmic drops; however, the ring ulcer persisted after 3 months.</p><p><strong>Conclusion: </strong>Topical bromfenac ophthalmic drops may induce a severe contact dermatitis related to the inactive ingredient sodium bisulfite within the solution. Chronic use of the bromfenac may also cause a sterile ring infiltrate. Caution should be given to patients in using eye drops with sulfite sensitivity or allergy.</p>","PeriodicalId":9635,"journal":{"name":"Case Reports in Ophthalmology","volume":"17 1","pages":"479-484"},"PeriodicalIF":0.6,"publicationDate":"2026-04-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC13258266/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"148249381","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}

{"title":"A Case Report of Presumed West Nile Virus Chorioretinitis in a Non-Endemic Area: Atypical Serology and Imaging Features of Old and New Lesions.","authors":"Matteo Belletti, Yanny Perez Jimenez, Piergiorgio Neri, Francesco Pichi","doi":"10.1159/000551782","DOIUrl":"10.1159/000551782","url":null,"abstract":"<p><strong>Introduction: </strong>West Nile virus (WNV) infection is a recognized cause of neuroinvasive disease and chorioretinitis, typically reported in endemic regions and often associated with positive IgM serology. However, atypical presentations with discordant serology can occur, particularly in non-endemic settings, posing diagnostic challenges.</p><p><strong>Case presentation: </strong>A 58-year-old woman developed acute encephalitis and bilateral panuveitis shortly after travel to a non-endemic region. Serum and cerebrospinal fluid testing showed positive WNV IgG and negative IgM. Multimodal retinal imaging revealed the coexistence of inactive-appearing chorioretinal scars and active full-thickness retinitis, with characteristic wedge-shaped and linear lesion patterns. Fundus autofluorescence, fluorescein angiography, indocyanine green angiography, and optical coherence tomography allowed differentiation of lesions with varying morphology and stages of activity. Although these findings do not confirm viral relapse or reactivation, they suggest prior infection with possible delayed or recurrent ocular and central nervous system involvement. Systemic and topical corticosteroid therapy resulted in rapid clinical and imaging improvement.</p><p><strong>Conclusion: </strong>This case highlights the diagnostic value of multimodal imaging in suspected WNV chorioretinitis and underscores the limitations of serology alone. Atypical serological profiles in non-endemic settings require careful interpretation, and multimodal imaging may provide critical insights into disease activity and staging.</p>","PeriodicalId":9635,"journal":{"name":"Case Reports in Ophthalmology","volume":"17 1","pages":"498-510"},"PeriodicalIF":0.6,"publicationDate":"2026-03-30","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC13268619/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"148263373","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}

{"title":"Industrial Chlorine Gas Leak: Combined Ocular and Respiratory Injury in 2 Patients - A Case Report.","authors":"Vishaka T Vatkar, Harshita Kashyap, Saujas Rashmin Shah, Himani Yadav, Krutika Jeevan Ladi","doi":"10.1159/000551439","DOIUrl":"10.1159/000551439","url":null,"abstract":"&lt;p&gt;&lt;strong&gt;Introduction: &lt;/strong&gt;Even though chlorine gas accidents are rarely seen, when an individual comes into contact with the gas, it can still cause considerable injury, particularly to the eyes and the airways. Chlorine is a strongly irritating and toxic gas, instantly recognizable by its sharp, suffocating odor and yellow-green color. It is widely used in industrial work and water purification, and has also been employed in certain conflict situations [Proc Am Thorac Soc. 2010;7(4):257-63 and Ann Occup Environ Med. 2014;26:29]. The extent of harm caused by chlorine inhalation is largely determined by its moderate water solubility, which allows the gas to reach and irritate both the upper and lower parts of the airway. Once chlorine comes into contact with moist epithelial surfaces, it reacts with the mucus layer to produce hydrochloric acid and hypochlorous acid (HOCl) [Environ Health Perspect. 2010;118(10):1343-7 and Eur Respir J. 2001;17(3):436-41]. These substances can inflame or injure the conjunctiva and, though uncommon, may result in chemical burns or corneal epithelial defects. This process also triggers the generation of reactive oxygen species, which further amplify tissue damage [Chest. 1997;112(3):853-8]. In this report, we present 2 cases in which patients developed corneal injury, respiratory distress, and even third-degree periocular burns following acute chlorine gas exposure.&lt;/p&gt;&lt;p&gt;&lt;strong&gt;Case presentation: &lt;/strong&gt;We report the cases of two male laborers, aged 22 and 43 years, who arrived at our centre within an hour after accidental chlorine gas exposure at a rubber manufacturing facility. Both experienced an abrupt decline in vision in both eyes, breathlessness, and third-degree burns affecting the periocular region. Ocular examination revealed grade 1 corneal burns as per the Roper-Hall classification and grade 1 injuries according to the Dua classification, with visible epithelial defects and patches of limbal ischemia. Chest imaging indicated chemical pneumonitis in both individuals, with the younger patient displaying more severe lung involvement. Treatment was initiated immediately and involved extensive irrigation with a balanced salt solution, followed by the application of preservative-free topical antibiotics, lubricants, cycloplegics, a short course of topical corticosteroids, and fortified insulin eye drops. Systemic management included intravenous dexamethasone, oral doxycycline, and vitamin C. Both required supplemental oxygen and close respiratory observation due to their breathing difficulties. By the end of 2 weeks, both patients showed marked clinical improvement, with clear corneas, complete epithelial healing, better visual acuity, and progressive relief of respiratory symptoms.&lt;/p&gt;&lt;p&gt;&lt;strong&gt;Conclusion: &lt;/strong&gt;Chlorine gas exposure can lead to a combination of ocular surface burns and chemical pneumonitis. Even when the initial symptoms seem mild, patients may later develop notable eye surface","PeriodicalId":9635,"journal":{"name":"Case Reports in Ophthalmology","volume":"17 1","pages":"485-497"},"PeriodicalIF":0.6,"publicationDate":"2026-03-30","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC13263007/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"148249400","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}

{"title":"Hidradenitis Suppurativa Presenting with Ocular Manifestations in a 44-Year-Old Male: A Case Report.","authors":"Eleftherios Chatzimichail, Peter Meyer, Géraldine Peters, Zisis Gatzioufas, Alexandra Steinemann","doi":"10.1159/000551644","DOIUrl":"10.1159/000551644","url":null,"abstract":"<p><strong>Introduction: </strong>Οcular manifestations of hidradenitis suppurativa (HS) are uncommon and typically include episcleritis, keratitis, and uveitis. We report a rare case of posterior scleritis and peripheral ulcerative keratitis (PUK) in a patient with HS.</p><p><strong>Case presentation: </strong>A 44-year-old male with no significant systemic or ocular history presented with a painful corneal ulcer in the left eye, which was diagnosed as PUK. External examination showed facial scarring and only in the course and only during subsequent visits did he reveal his axillary lesions. Initial eye treatment with topical antibiotics and steroids led to marked improvement. Two weeks later, the patient developed pain in the right eye and was diagnosed based on clinical findings of posterior scleritis, which was confirmed by B-scan ultrasonography. Concurrent worsening of HS and the presence of a conjunctival granuloma. A conjunctival biopsy was performed alongside a comprehensive systemic work-up, both of which excluded infectious etiologies and systemic rheumatological autoimmune disease. The patient responded well to systemic corticosteroids and continues to demonstrate stable vision and ocular health during follow-up. Over thchatzimie course of his disease, he was diagnosed with peripheral ulcerative keratitis (PUK) and anterior scleritis in both eyes, with the addition of posterior scleritis in the left eye. Treatment at this stage included topical dexamethasone, tobramycin, and systemic corticosteroids. Marked improvement in ocular symptoms and visual acuity was achieved. Histopathological analysis of the conjunctival biopsy revealed a benign histiocytic granuloma without evidence of malignancy or Splendore-Hoeppli phenomenon. No systemic rheumatologic autoimmune disease, including sarcoidosis, was identified.</p><p><strong>Conclusion: </strong>This case highlights the importance of recognizing ocular involvement in HS, particularly posterior scleritis and PUK, which may indicate underlying autoinflammatory dysregulation. Interdisciplinary management is essential for preserving vision. To our knowledge, this is the first report of posterior scleritis, PUK and conjunctival granuloma in the context of HS.</p>","PeriodicalId":9635,"journal":{"name":"Case Reports in Ophthalmology","volume":"17 1","pages":"511-517"},"PeriodicalIF":0.6,"publicationDate":"2026-03-27","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC13271692/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"148270337","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}

{"title":"Case Report: A Peculiar Pinguecula? A Surprising IgG4-Related Conjunctival Lesion.","authors":"Naama Lippin, Carol L Karp, Asaf Friehmann","doi":"10.1159/000551674","DOIUrl":"10.1159/000551674","url":null,"abstract":"<p><strong>Introduction: </strong>IgG4-related disease (IgG4-RD) is a chronic immune-mediated fibroinflammatory condition that may involve multiple organs but rarely presents as an isolated conjunctival lesion. Ocular surface involvement can mimic benign inflammatory conditions, leading to diagnostic delay. We report a rare case of isolated conjunctival IgG4-RD initially misdiagnosed as recurrent pingueculitis.</p><p><strong>Case presentation: </strong>A 37-year-old female presented with a gelatinous, cystic mass on the bulbar conjunctiva. Comprehensive ophthalmic examination and anterior segment optical coherence tomography (OCT) revealed a well-demarcated subconjunctival lesion without scleral or intraocular invasion. Differential diagnoses included conjunctival lymphoma, ocular surface squamous neoplasia, and nonspecific orbital inflammation. An excisional biopsy was performed, and histopathological examination confirmed IgG4-RD, demonstrating dense lymphoplasmacytic infiltration with CD138- and IgG4-positive plasma cells, storiform fibrosis, and abundant collagen fibers. Postoperatively, a suspected local recurrence was successfully treated with topical dexamethasone. At 6-month follow-up, complete clinical remission was achieved without additional local or systemic therapy.</p><p><strong>Conclusion: </strong>This case highlights the importance of considering IgG4-RD in the differential diagnosis of atypical conjunctival lesions. Tissue biopsy remains essential for definitive diagnosis, while adjunctive imaging modalities such as anterior-segment OCT may aid in lesion characterization, even in the absence of systemic disease.</p>","PeriodicalId":9635,"journal":{"name":"Case Reports in Ophthalmology","volume":"17 1","pages":"539-544"},"PeriodicalIF":0.6,"publicationDate":"2026-03-26","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC13286556/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"148307321","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}