Case Reports in Ophthalmology最新文献

Introduction: Ocular syphilis has atypical presentation in immunocompromised individuals. We report a case of intermediate uveitis as a delayed immune-mediated relapse following the successful treatment of syphilitic retinitis in an HIV-positive individual.

Case presentation: A 30-year-old HIV-positive man presented with decreased vision in his left eye. Examination revealed left eye granulomatous anterior uveitis with vitritis, retinal vasculitis, and optic disc edema. The patient was diagnosed with syphilitic uveitis, reactive for rapid plasma reagin (RPR), and initiated on intravenous penicillin by the internist. Topical corticosteroids were initiated, and oral corticosteroids were added after 1 week. Over 2 months, the lesions resolved clinically with decreasing RPR titers. After 2 months of complete remission and tapering of therapy, the patient presented with renewed disc edema, macular edema, vitreous cells, and snowballs suggestive of intermediate uveitis. The RPR titers showed a decreasing pattern.

Conclusion: The patient responded well to oral corticosteroids, indicating a likely immune-mediated relapse rather than reinfection. This case highlights the need for continued surveillance, even after clinical resolution.

Introduction: Simultaneous branch retinal artery occlusion (BRAO) and central retinal vein occlusion (CRVO) is an exceptionally rare cause of acute vision loss, particularly in young individuals without systemic vascular risk factors. Blood transfusion has been implicated in rare thromboembolic and hyperviscosity-related complications but is infrequently associated with retinal vascular occlusions.

Case presentation: A 20-year-old man presented with sudden painless unilateral vision loss shortly after receiving a packed red blood cell transfusion for severe anemia. Ophthalmic examination revealed optic disc edema, venous engorgement, scattered retinal hemorrhages, and superior hemi-macular whitening in the affected eye. Structural optical coherence tomography demonstrated inner retinal hyperreflectivity and subsequent thinning, consistent with ischemic retinal injury. Fluorescein angiography was contraindicated due to an acute hypersensitivity reaction. Extensive systemic, hematologic, inflammatory, and thrombophilia workup was unremarkable. Despite conservative and supportive management, including hyperbaric oxygen therapy and anticoagulation, visual recovery remained limited at follow-up.

Conclusion: This case highlights a rare presentation of concurrent BRAO and CRVO occurring shortly after blood transfusion in a young patient without identifiable systemic risk factors. Although causality cannot be definitively established, the close temporal relationship and exclusion of alternative etiologies suggest transfusion-related rheologic changes as a plausible contributing factor. Clinicians should remain vigilant for acute retinal vascular events following transfusion, even in young patients, and prompt ophthalmic evaluation is essential.

Introduction: Retinitis pigmentosa (RP) is a genetically and phenotypically heterogeneous inherited type of retinal dystrophy in which the RHO gene is frequently implicated. The missense variant NM_000539.3:c.545G>A, p.(Gly182Asp), has been reported in genetic databases as pathogenic, and a recent large cohort identified a single patient carrying this variant with sector RP. However, no detailed intrafamilial phenotypic characterization has been described. This study provides the first such characterization in a Portuguese family.

Case presentation: Four individuals across three generations underwent multimodal ophthalmologic evaluations, including visual acuity, fundus imaging (OCT, FAF), visual field testing, and electrophysiology. Three patients underwent genetic testing, and all carried the heterozygous RHO p.(Gly182Asp) variant. The clinical manifestations ranged from typical RP in three members to sector RP in one, with preferential inferior and nasal retinal involvement. Structural and functional severity increased with age, and all presented with nyctalopia. The oldest patient exhibited advanced degeneration with minimal central vision. This intrafamily phenotypic variability underscores the differential expression of the same genotype.

Conclusion: This is the first report linking the RHO p.(Gly182Asp) variant with both typical and sector RP, highlighting intrafamilial heterogeneity. Detailed phenotyping is crucial for genetic counseling and potential inclusion in future gene-based therapies.

Introduction: Reticular pseudodrusen (RPD) are an important clinical biomarker associated with progression to late age-related macular degeneration (AMD). We report polarization-sensitive optical coherence tomography (PS-OCT) findings in a case of RPD and evaluate the entropy characteristics of these lesions.

Case presentation: A 73-year-old man presented for evaluation of retinal angiomatous proliferation in his right eye. The left eye demonstrated multiple round, white-yellowish macular lesions consistent with RPD. Optical coherence tomography (OCT) angiography showed no macular neovascularization in the left eye. Swept-source (SS-OCT) revealed hyperreflective, spike-like deposits between the photoreceptor outer segments (PROSs) and the retinal pigment epithelium. Corresponding PS-OCT B-scans demonstrated moderately elevated polarimetric entropy values at the same locations. En face images reconstructed from the PROS layer showed circular RPD-like lesions on SS-OCT, which precisely matched high-entropy areas on PS-OCT. Merged en face maps confirmed one-to-one correspondence between RPD and regions of increased entropy.

Conclusion: PS-OCT successfully visualized RPD as moderately high-entropy lesions in the PROS layer, suggesting that these deposits are composed of non-retinal pigment epithelium material, such as lipids or inflammatory cells. These findings indicate that PS-OCT may serve as a useful complementary modality for detecting RPD and may enhance current imaging strategies for assessing RPD and monitoring AMD progression.

Introduction: This case highlights the complexity of managing highly refractory, recurrent pterygium. Despite numerous prior surgeries by experienced anterior segment surgeons, the patient developed persistent fibrosis, symblepharon formation, and functional limitations. This underscores the need for a tailored, multidisciplinary approach, a strategy not widely documented in current literature.

Case presentation: Over 6 years, the patient underwent ten previous interventions, including conjunctivoplasty with ipsilateral free conjunctival flap and mitomycin C (MMC), amniotic membrane transplantation, symblepharolysis, and tenonplasty. The most recent surgery had been performed 6 months before referral to our clinic. Despite these attempts, each procedure was followed by significant fibrosis, scarring, and recurrent symblepharon formation involving the fornices. Following comprehensive multidisciplinary discussion and literature review, reconstructive surgery was undertaken. This included conjunctivoplasty with a free limbal-conjunctival autograft from the contralateral eye, adhesiolysis of the extraocular muscles, muscle belly plasty with amniotic membrane grafting, and placement of barrier sutures to reduce adhesion recurrence. During 12 months of follow-up, the patient exhibited notable clinical improvement, including enhanced ocular motility, resolution of diplopia in primary gaze, and improved visual acuity.

Conclusion: This case emphasizes the importance of individualized, innovative surgical planning for advanced recurrent pterygium. A long-term, multidisciplinary strategy can offer sustained anatomical and functional restoration in patients with extensive prior surgical failure and severe ocular surface disease.

Introduction: Sickle cell disease is associated with painful vaso-occlusive crises, bone infarction, avascular necrosis, and osteomyelitis. Sickle cell orbitopathy is a rare manifestation with signs similar to periorbital cellulitis and has subtle radiologic features.

Case presentation: We report a case of a five-year-old girl with sickle cell who presented with bilateral orbital infarctions and subperiosteal hematomas, notably without periorbital pain, and was treated with antibiotics and steroids.

Conclusion: Physicians should be aware of this rare manifestation, especially in the pediatric population, as it can affect subsequent management.

Introduction: We describe a man with severe unilateral visual loss from giant cell arteritis who had normal inflammatory markers and exhibited progression of intracranial vasculitis while on high doses of corticosteroids.

Case: A 64-year-old man presented with unilateral visual loss. He had systemic symptoms of giant cell arteritis (GCA); however, both inflammatory markers were normal. He was diagnosed with arteritic anterior ischemic optic neuropathy after temporal artery biopsy revealed classic findings of GCA and treated with high-dose corticosteroids. Despite appropriate treatment, serial imaging revealed progressive narrowing of intracranial arteries, predominantly involving anterior circulation of the brain.

Conclusion: This case highlights that GCA can rarely present with normal inflammatory markers, involve intracranial vasculature, and progress despite treatment with high doses of corticosteroids emphasizing the importance of advanced imaging and potential need for adjunctive immunosuppressive therapy in patients with refractory disease.

[This corrects the article DOI: 10.1159/000546007.].

Introduction: Neisseria gonorrhoeae is a common communicable bacterial infection in the USA. Although primarily affecting the genitourinary tract, ocular involvement - gonococcal keratoconjunctivitis - is a rare but potentially devastating manifestation that can result in corneal melt, perforation, and permanent vision loss. The management of gonococcal keratoconjunctivitis is typically medical, but surgical intervention is warranted in severe cases.

Case presentation: This case details the delayed presentation of a 49-year-old male with gonococcal keratoconjunctivitis leading to bilateral 9 × 10 mm full-thickness corneal melts, with unsuccessful corneal gluing prior to transfer. On presentation, examination showed excessive purulence, glue, disorganized anterior chamber structures, and bilaterally collapsed globes on CT. Vision was at least light perception in both eyes. Given the severity of the disease, the ability to preserve the globes was uncertain; therefore, the decision was made to undertake urgent and aggressive medical and surgical intervention with bilateral simultaneous penetrating keratoplasty, which successfully salvaged both globes. At the most recent follow-up, the exam showed a visual acuity of 20/150 with pinhole in the right and a failed graft with light perception in the left.

Conclusion: This case demonstrates the potential severity of gonococcal keratoconjunctivitis and highlights the utility of early and aggressive surgical intervention in such cases. Therapeutic keratoplasty along with amniotic membrane provided an effective method of treating gonococcal-associated corneal melt and perforation, allowing for salvage of both globes. While the severity of this case is uncommon, it provides a precedent for the role of therapeutic keratoplasty in similar situations.

Introduction: Orbital emphysema is characterized by presence of air in the soft tissues of the orbits. In severe cases, this can lead to orbital compartment syndrome, a medical emergency that can result in severe permanent vision loss without urgent intervention. Generally, the severity of presenting vision loss and longer time to intervention have been associated with worse visual outcome.

Case presentation: We present the case of a 26-year-old healthy male who developed an acute orbital compartment syndrome with orbital emphysema following nose blowing in the setting of a bacterial orbital cellulitis. Once documented to have no light perception (NLP) vision in the affected eye, he was urgently taken to the operating room for surgical drainage (approximately 2.5 h after onset of the NLP). Over the subsequent days, he gradually regained 20/20 visual acuity.

Conclusion: This case highlights that despite complete loss of vision, this patient had an excellent visual outcome.