Case Reports in Ophthalmology最新文献

Introduction: Myelin oligodendrocyte glycoprotein antibody disease (MOGAD) is a more recently characterized neuroinflammatory condition which has been implicated with optic neuritis (ON). While the potential causes of central retinal artery occlusion (CRAO) are several, neuroinflammatory causes should not be excluded from the differential diagnosis. Our unusual case presents a patient with prior history of MOGAD-ON and venous stasis retinopathy (VSR) in their right eye, who, in their current presentation, developed a CRAO in their left eye.

Case presentation: A 43-year-old female with a history of concurrent VSR and MOGAD-ON (diagnosed 6 years prior and managed with long-term immunosuppression and oral steroids) in her right eye presented with a five-hour history of pain and blurry vision in her left eye. Examination of the left eye revealed a visual acuity (VA) of 20/80, a cherry red spot (without a visualized embolus), and a new generalized visual field defect. Optical coherence tomography (OCT) revealed diffuse retinal edema in her left eye with multiple layers of retinal thinning. She was referred to stroke neurology and continued oral steroid therapy. At 6-month follow-up, her VA had markedly improved to 20/30 in her left eye, and she had stable OCT parameters.

Conclusion: This unique case demonstrates sequential VSR and CRAO in opposite eyes, which were notable for their association with recurrent MOGAD-ON. Further research is needed to better elucidate the role of MOGAD in vascular occlusive events and whether this relationship may be arteritic in etiology.

Introduction: This case report presents the clinical, histological, and genetic characteristics of a patient with ligneous conjunctivitis and to provide a long-term treatment schedule, using cryo-depleted plasma infusions in combination with topical cyclosporine as a successful therapeutic option.

Case presentation: A 12-year-old Turkish girl experienced years of ocular itching, irritation, and increased light sensitivity. Initial clinical examination revealed the presence of pseudomembranes and giant papillae on the tarsal conjunctivae of both eyes. The biopsy of a membrane from the left eye was taken for histological examination. Manifestations of severe comorbidity since infancy (congenital hydrocephalus, asthma, recurrent respiratory infections, and impaired wound healing) and parental consanguinity prompted a genetic evaluation. The pseudomembranes were composed of fibrin, granulation tissue, and inflammatory cells consistent with the diagnosis of ligneous conjunctivitis. The genetic analysis revealed homozygosity for a pathogenic missense variant in plasminogen (PLG), causing PLG deficiency. Due to severe systemic complications, the patient was successfully treated with cryo-depleted plasma infusions and cyclosporine eye drops, resulting in the complete remission of the ligneous conjunctivitis.

Conclusion: This case report highlights the importance of considering PLG deficiency in patients with recurring chronic conjunctivitis. Long-term treatment with cryo-depleted plasma infusions in combination with short-term use of topical cyclosporine may provide an efficient therapeutic option for ligneous conjunctivitis in patients with PLG deficiency.

Introduction: Mucoceles are benign, slow-growing cystic formations located within the paranasal sinuses, caused by complete ostial obstruction and accumulation of mucous secretions. Here, we report a case of a patient who initially presented with unilateral painless visual impairment and was ultimately diagnosed with bilateral sphenoid sinus mucoceles (SSMs) after two hospitalizations.

Case presentation: A 67-year-old woman presented with a 7-day history of decreased vision in her left eye. She denied any orbital pain, headache, or restricted eye movement and was diagnosed with retrobulbar ischemic optic neuropathy on the first admission. After drug treatment, the visual acuity of patient improved significantly, but 3 months after discharge, the patient was hospitalized again due to recurrent vision loss accompanied by left orbital pain and left temporal pulsatile headaches. After multiple imaging examinations, the patient was ultimately diagnosed with SSMs and her visual acuity was restored after surgical treatment.

Conclusions: The majority of SSMs are associated with ocular symptoms, with only a minority presenting solely with unilateral or bilateral vision loss, as exemplified in this case. Therefore, understanding the clinical features of visual disturbances secondary to SSMs is crucial to aiding more prompt diagnosis and treatment.

Introduction: Inflammation of the prelaminar compartment of the optic nerve involves the most anterior segment of the nerve. It can occur in isolation, in the setting of immunological or demyelinating disorders.

Case presentation: We present two 32- and 41-year-old female patients with sequential bilateral isolated prelaminar optic neuritis with atypical features. In both instances, plasma exchange (PLEX) was initiated for the second affected eye after corticosteroids proved ineffective, resulting in excellent visual recovery. Both patients lost vision in the fellow eye due to a similar attack that was also refractory to steroids. Our cases did not show any associated demyelinating lesions on imaging and did not show biomarkers such as aquaporin-4 or myelin oligodendrocyte glycoprotein (MOG) antibody in serum workup. They were followed for more than 5 years with stable vision in the only eye.

Conclusion: In cases of isolated severe progressive prelaminar optic nerve inflammation refractory to steroid treatment, PLEX can be helpful, even if workup is unremarkable for neuromyelitis optica or other systemic and neurologic conditions.

Introduction: Orbital myositis (OM) is an inflammatory condition of the extraocular muscles, often idiopathic but occasionally associated with systemic diseases or malignancies. Secondary OM due to metastatic disease is rare. We report a case of OM as the initial manifestation of metastatic renal cell carcinoma, emphasizing the need for comprehensive evaluation of atypical ocular presentations.

Case presentation: An 81-year-old male with a history of age-related macular degeneration presented with acute onset of pain and restricted movement in his left eye. Computed tomography imaging revealed an osteolytic lesion in the left sphenoid bone, causing reactive myositis. Further systemic evaluation identified a left renal mass with evidence of pulmonary and skeletal metastases. A core needle biopsy confirmed the diagnosis of metastatic renal cell carcinoma. Given the advanced disease stage, the patient was managed with palliative treatment. Despite medical interventions, he succumbed to the disease 6 months after symptom onset.

Conclusion: This case underscores the significance of considering malignancy in the differential diagnosis of OM, particularly in elderly patients with atypical ocular symptoms. Early recognition and systemic evaluation are crucial for timely diagnosis and management of underlying malignancies presenting with orbital involvement.

Introduction: Patients with membranoproliferative glomerulonephritis (MPGN) type III may exhibit the saw-tooth pattern of cuticular drusen. This study reports a four-and-a-half-year retrospective time-dependent observation of cuticular drusen associated with immune complex-mediated MPGN type III patients.

Case presentation: A 32-year-old woman was referred for ocular evaluation due to systemic steroids used for nephrotic syndrome caused by immune complex-mediated MPGN type III. She had no ocular symptoms. The initial examination showed that her best-corrected visual acuity (BCVA) was 1.0 oculus uterque (OU). Color fundus photographs revealed pigmentary abnormalities in the macular and perimacular regions OU. Fundus autofluorescence (FAF) images demonstrated numerous tiny dots with a hypoautofluorescent center surrounded by a rim of hyperautofluorescence. Enhanced depth imaging-optical coherence tomography (OCT) revealed triangular morphologic features, represented by a saw-tooth pattern with internal hyporeflectivity. Over 4.5 years, the internal reflectivity of the drusen gradually increased. Moreover, depigmentation and yellow color changes in the macular and perimacular regions worsened, without obvious changes on FAF. Her BCVA remained at 1.0 OU, without new ocular symptoms or cataract progression during the 4.5-year follow-up period. Both eyes had a significant positive correlation between swept-source OCT-based mean internal reflectivity measured by the ImageJ software and the observation period. However, no correlation was found between the estimated glomerular filtration rate (eGFR) and the observation period or between eGFR and mean internal reflectivity.

Conclusion: The internal reflectivity of cuticular drusen associated with immune complex-mediated MPGN type III showed time-dependent changes on OCT and worsened depigmentation and yellow color changes in the macular and perimacular regions, independent of renal function.

Introduction: Alport syndrome is an inherited disease caused by mutations in COL4A5, COLA3, or COL4A4 resulting in kidney failure, hearing loss, and ocular symptoms. We report a patient with Alport syndrome who has a "stair-case/honeycomb" maculopathy, a rare but distinctive finding in this disease.

Case presentation: A 53-year-old man with Alport syndrome was referred for gradual decrease in vision. His ocular history was remarkable for intraocular lens implantation secondary to lenticonus in each eye. Fundus photography showed rare white dots in the temporal mid-periphery in each eye and fundus autofluorescence was normal. Optical coherence tomography (OCT) B-scans through the fovea showed irregular thinning of the inner retina with peaks and valleys in the macula of each eye. The ellipsoid zone was intact except for mild patchiness centrally. En face retinal structural OCT angiography (OCTA) images showed a mosaic-like honeycomb pattern in the macular region in both eyes, with hyporeflective depressions in areas of focal retinal atrophy. Retinal OCTA scans showed irregular foveal avascular zone (FAZ) areas with capillaries crossing the FAZ in the left eye, corresponding to islands of preserved retinal tissue. There was predominance of capillaries in the deeper retinal layers centrally.

Conclusion: While severe irregular thinning of the macula is not a common feature in Alport syndrome, when it is present in patients who have not been previously diagnosed, particularly in patients with renal disease, it should suggest the diagnosis of Alport syndrome. Its occurrence can be the cause of vision loss which is not commonly associated with Alport central maculopathy.

Introduction: This case report describes a patient whose misdiagnosis of foveal hypoplasia was uncovered by optical coherence tomography angiography (OCTA) findings that suggested the presence of an epiretinal membrane (ERM) over foveal hypoplasia.

Case presentation: A 67-year-old man with no significant past medical history was referred to our retina clinic with a diagnosis of foveal hypoplasia. He had been experiencing significant vision loss for more than 1 year. OCT demonstrated absence of the foveal depression in both eyes. A subtle ERM was identified in the left eye OCT, but the presence of an ERM in the right eye OCT was equivocal. As such, it was unclear whether flattening of the fovea was attributable to hypoplasia or ERM based on OCT alone. This prompted further investigation with OCTA, which showed the presence of the FAZ in both eyes. The diagnosis of stage 2 ERM OU was made based on OCTA findings. The patient underwent cataract extraction with intraocular lens implantation, pars plana vitrectomy, and ERM peel, which resulted in improvement of visual symptoms and visual acuity. Follow-up OCT showed normalization of the foveal pit in the right eye greater than the left eye.

Conclusion: This case demonstrates the importance of utilization of OCTA in differentiating true foveal hypoplasia from this foveal "pseudo-hypoplasia" exhibited by our patient.

Introduction: Intrapapillary hemorrhage with adjacent peripapillary subretinal hemorrhage (IHAPSH) is a syndrome associated with intrapapillary hemorrhage and peripapillary subretinal hemorrhage that commonly occurs in myopic women. There have been no reports of patients younger than 10 years. We report a case of typical IHAPSH that occurred in a 9-year-old girl in the advanced stages of myopia.

Case presentation: A 9-year-old girl presented with gradually decreasing uncorrected visual acuity in the left eye over the past year, without complaints of floaters or blurred vision. Uncorrected visual acuity was 20/16 in the right eye and approximately 20/285 in the left eye, improving to 20/16 with -4.50 diopters sphere and -0.50 diopters cylinder at 165°. Ophthalmoscopic examination revealed intrapapillary hemorrhage and nasal peripapillary subretinal hemorrhage in her left eye. The left eye had a small, tilted papilla. Fluorescein angiography showed no leakage from the papilla. The length of the ocular axis was elongated to 24.87 mm in the left, compared to 23.15 mm in the right. We diagnosed IHAPSH and decided to follow the patient. After 8 weeks, the hemorrhage was almost completely absorbed. No recurrence has been observed since then.

Conclusion: To our knowledge, this is the youngest reported case of IHAPSH. This case underscores the potential contribution of optic disc morphology and progressive myopia to IHAPSH pathogenesis and highlights the importance of considering this condition in the differential diagnosis of optic disc hemorrhage in school-aged children.

Introduction: Inert intraocular foreign body (IOFB) removal depends on the location, type of injury, composition, and size of IOFB and possible serious complications of intraocular surgery. Early management is crucial for better prognosis.

Case presentation: A 28-year-old male presented to the outpatient department after an alleged workplace accident. Initial assessment revealed significant diminution of vision, and on anterior segment examination with slit lamp, conjunctival congestion with no obvious entry point and no obvious scleral tear noted. Fundus examination by indirect ophthalmoscopy revealed vitreous hemorrhage, but the foreign body could not be localized due to extensive hazy media. Radiography of the orbit revealed an IOFB. The patient was managed surgically, and the intraretinal foreign body was removed using an intraocular magnet and intraocular forceps. The decision to remove the inert metal was considered because the patient had significant vision loss with vitreous hemorrhage.

Conclusion: Management of an intraretinal metallic foreign body using an intraocular magnet is a viable and effective approach. It allows precise removal with minimal retinal trauma, thus preserving visual function.