Case Reports in Ophthalmology最新文献

Introduction: The purpose of this clinical report was to describe an unprecedented case of bilateral pressure-induced stromal keratopathy (PISK) following corneal photorefractive keratectomy, associated with presumed herpetic keratitis, and to present tomographic and biomechanical findings before and after appropriate treatment.

Case presentation: A 33-year-old male patient was referred to our clinic with suspected delayed corneal epithelial healing 3 weeks after an uncomplicated PRK. A central layer of corneal opacity with a presumed fluid-filled interface area was observed upon slit lamp biomicroscopy. Scheimpflug images from the Pentacam^® revealed a hyperreflective area beneath the central cornea. Scheimpflug-based corneal tomography, biomechanical assessment using the Pentacam^® AXL Wave, and the Corvis ST^® were conducted. Goldmann applanation tonometry measured 23/13 mm Hg, while noncontact tonometry intraocular pressure measured with the Corvis ST^® (Corvis ST IOPnct) was 40.5/43.5 mm Hg. Treatment with oral valacyclovir, combined with ocular hypotensive therapy, led to a significant reduction in IOP and improved corneal deformation parameters after 1 month.

Conclusion: Surgeons should be aware of the inaccuracy of Goldmann applanation tonometry in PISK, which can occur after LASIK or surface ablation.

Introduction: We present a case of a patient with preceding vitreomacular traction (VMT) who developed a full-thickness macular hole (FTMH) following his sixth intravitreal aflibercept injection for the treatment of age-related macular degeneration and review the literature on risk factors and pathogenesis of this adverse event.

Case presentation: FTMH can occur after an extended number of repeat intravitreal injections in the setting of worsening vitreomacular adhesion or VMT. This patient's FTMH was successfully treated surgically in a timely manner, and additional injections were resumed safely.

Conclusions: Patients with an unexpected decrease in vision after intravitreal injections should be reevaluated with optical coherence tomography to rule out alternative pathology including vitreomacular interface abnormalities. FTMH, if present, should be treated promptly to allow for resumption of therapy as needed and visual optimization.

Introduction: Corneal graft detachment is a major postoperative complication of Descemet's stripping automated endothelial keratoplasty (DSAEK). When a corneal graft becomes detached, corneal endothelial function generally fails, and repeat corneal transplantation is required. Herein, we report a rare case in which a transparent cornea was maintained after the removal of a dislocated DSAEK graft.

Case presentation: A 79-year-old woman with a residual lens cortex who had undergone cataract surgery was referred to our hospital. The cortex was removed, and bullous keratopathy progressed. Six months after the initial surgery, DSAEK was performed under topical anesthesia without any complications. Although the corneal graft had attached fairly well, it detached from the host cornea 3 weeks later. Two months after DSAEK, an air tamponade was used to treat the anterior chamber with single interrupted suturing; however, the graft detached again, except for the suture site. Because the detached cornea became cloudy in the anterior chamber, it was surgically removed 8 months after DSAEK. Accordingly, the host cornea transparency improved to a best-corrected visual acuity of 0.8 with a rigid gas permeable lens and a central corneal thickness of 580 μm. The corneal endothelial cell density was 995 cells/mm².

Conclusion: Removal of the corneal graft from the dislocated cloudy graft improved the visual acuity of this patient after DSAEK. The condition of the cornea should be carefully monitored after corneal endothelial transplantation, even after the graft has been dislocated.

Introduction: Leber hereditary optic neuropathy (LHON) is a mitochondrial disorder that typically presents with painless, central visual loss, hyperaemia of the optic nerve head, and peripapillary telangiectasias. Most LHON cases are due to one of three variants, but several less common variants also exist. We describe a clinical case of LHON associated with the variant m.3866T>C, which is possibly linked to LHON.

Case presentation: A 59-year-old Caucasian woman experienced acute, bilateral, and painless visual loss. She reported cigarette smoking, and elevated phosphatidylethanol suggested harmful alcohol consumption. Her best-corrected visual acuity (BCVA) was 20/100 for the right eye and 20/50 for the left eye. She could only read the Ishihara demonstration plate, and threshold perimetry demonstrated reduced central sensitivity bilaterally. Her optic nerve heads were hyperaemic, with peripapillary telangiectasias. The visual symptoms and clinical findings suggested LHON. Magnetic resonance imaging demonstrated a tuberculum sella meningioma and two cerebral aneurysms, which we regarded as incidental findings. Genetic testing did not identify common LHON variants but a rare homoplasmic variant, m.3866T>C, which studies suggest might cause LHON or act in synergy with other variants to increase the disease penetrance. After initiating test-of-treatment with idebenone 900 mg per day, the patient's BCVA improved to 20/32 for both eyes and then stabilized.

Conclusion: This case strengthens the evidence for m.3866T>C as a causative LHON variant. The case also raises the question as to whether this particular variant can respond favourably to treatment with idebenone.

Introduction: Perioperative visual loss (POVL) owing to hemi-retinal vein occlusion (HRVO) following prone positioning during spinal surgery is rare. Here, we report a case of HRVO with macular edema (ME) after spinal surgery that was successfully treated with intravitreal aflibercept (IVA) injections and retinal photocoagulation (RP).

Case presentation: A 63-year-old Japanese man underwent spinal surgery for lumbar spinal canal stenosis. Surgery was performed with the patient in the prone position under general anesthesia; the operation time was 305 min. No complications were associated with intraoperative anesthesia. On postoperative day 4, the patient noticed decreased visual acuity in his left eye and visited the Department of Ophthalmology on postoperative day 9. The best-corrected visual acuity (BCVA) in the left eye was 0.1. Fundus and optical coherence tomography revealed HRVO and ME in the left eye. IVA injections and RP were performed in the eye, which substantially decreased the ME and improved the patient's BCVA to 0.8.

Conclusions: HRVO can cause POVL after prone positioning during spinal surgery. This is the first case of HRVO with ME after spinal surgery, which was successfully treated with IVA injections and RP.

Introduction: We report a case of pseudoxanthoma elasticum (PXE) with an atypical phenotype likely related to a hypomorphic variant in ABCC6.

Case presentation: A 66-year-old Caucasian female with a history of a maculopathy interpreted as either age-related macular degeneration or a pattern dystrophy underwent a detailed ophthalmic evaluation. Visual acuities were 20/25, OD, and 20/20, OS. Spectral domain optical coherence and fluorescein angiography demonstrated outer retinal disruptions and breaks in retinal pigment epithelium (RPE)/Bruch's membrane bilaterally, consistent with angioid streaks. A large area of hypo- and hyperautofluorescence extending from the central retina into the peripapillary retina was documented with short-wavelength excitation autofluorescence. The area of hypoautofluorescence, which was much larger on near-infrared excitation, spared the temporal retina. Two-color dark-adapted perimetries documented severe rod sensitivity losses and less severe cone sensitivity abnormalities co-localizing with the RPE abnormalities. No obvious skin findings were observed, and initial dermatologic biopsy was negative. Gene screening identified a pathogenic ABCC6 gene variant c.1552C>T and a previously reported variant of uncertain significance c.1171A>G. A second dermatologic biopsy demonstrated positive findings consistent with PXE.

Conclusion: Although this patient had minimal skin findings, this patient had characteristic structural and functional abnormalities of a pattern dystrophy with angioid streaks and histologic evidence of PXE, suggesting compound heterozygous variants involving the hypomorphic ABCC6 c.1171A>G variant. These findings support the pathogenic role of both variants.

Introduction: We report on one case of age-related macular degeneration and one case of diabetic macular edema with concomitant retinal arterial macroaneurysm (RAM) that were treated with anti-vascular endothelial growth factor (VEGF) intravitreal injections.

Case presentation: Case 1 involved a 71-year-old woman with a 30-year history of diabetes who was undergoing dialysis. Pretreatment visual acuity in the right eye was 0.4. Fundus fluorescein angiography (FA) showed that numerous microaneurysms and RAM were located at a distance of two papillary diameters from the macular area. Diabetic macular edema was diagnosed. After 12 anti-VEGF injections, the macular edema resolved, microaneurysms decreased, and the RAM also disappeared. Visual acuity in the right eye improved to 0.7. Case 2 involved an 81-year-old woman receiving treatment for disorders including hypertension and dyslipidemia. Pretreatment visual acuity in the right eye was 0.03. And vitreous hemorrhage was present. After one ranibizumab intravitreal injection, the fundus became clearly visible. Macular subretinal hemorrhage and an RAM in the upper macula area were evident on FA and optical coherence tomography. After four intravitreal injections of ranibizumab, macular hemorrhage resolved, RAM disappeared, and visual acuity improved to 0.2. All cases were treated by intravitreal injection of anti-VEGF. After several injections, the macular hemorrhage or macular edema was resolved. RAM disappeared, and visual acuity improved.

Conclusion: Intravitreal injection of anti-VEGF appears effective for age-related macular degeneration or diabetic macular edema with concomitant RAM. Although anti-VEGF intravitreal injections are not covered by health insurance for the treatment of RAM, their effectiveness means that the expansion of indications is desirable.

Introduction: Cytomegalovirus (CMV) retinitis in the setting of pediatric retinoblastoma is exceedingly unusual. Here, we present the first reported case of CMV retinitis in an enucleated eye with retinoblastoma after chemotherapy in the western hemisphere.

Case presentation: A 2-year-old Hispanic male without a family history of retinoblastoma presented with a 3-month history of right eye exotropia and squinting. Clinical examination revealed dense white vitreous opacities in the right eye. Ocular oncology evaluation unveiled an exudative retinal detachment with vitreous seeds, subretinal seeding, and a tumor emanating from the retina in the superonasal quadrant of the right eye. The patient was diagnosed with unilateral Group D retinoblastoma, and RB1 sequencing revealed a pathogenic variant with mosaicism. Treatment involved systemic chemotherapy, intravitreal chemotherapy, and cryotherapy. However, the patient developed a rhegmatogenous retinal detachment with diffuse vitreous hemorrhage and ultimately underwent right eye enucleation. Interestingly, histopathological analysis of the enucleated eye revealed concomitant CMV retinitis alongside retinoblastoma. After consultation with infectious disease, antiviral treatment was not initiated as the patient remained asymptomatic and maintained a recovered immune system. Repeat CMV PCR confirmed viral clearance. The patient received a prosthetic eye and continues to be monitored for retinoblastoma recurrence.

Conclusion: Clinicians should be aware of the potential for CMV retinitis to develop in retinoblastoma patients receiving chemotherapy, which may complicate clinical decision-making and management. Timely identification of CMV retinitis in this setting may improve patient ocular outcomes and overall prognosis.

Introduction: Anterior ischemic optic neuropathy (AION) is an ischemic disorder of the optic nerve and a common cause of acute, painless, permanent vision loss. It is divided into two types: arteritic AION (AAION) and non-arteritic anterior ischemic optic neuropathy (NAION). Although subretinal fluid associated with optic disc edema has been reported in cases of NAION, it is rarely described in AAION.

Case presentation: An 86-year-old female with a history of polymyalgia rheumatica presented with sudden vision loss in the left eye. Initial examination revealed left pallid optic disc edema with peripapillary hemorrhages. Optical coherence tomography (OCT) of the left macula showed intraretinal and submacular fluid. The patient was started on 50 mg of oral prednisone daily. The diagnosis of giant cell arteritis (GCA) was later confirmed with a positive temporal artery biopsy. Three weeks after presentation, an OCT was completed which demonstrated complete resolution of the intraretinal and submacular fluid. Although the presence of both intraretinal and subretinal fluid has been previously documented in cases of NAION, it is rarely reported in a patient with GCA.

Conclusion: This is a newly described case of subretinal and intraretinal fluid in a patient with AAION. We postulate that the pathophysiology behind this is mediated by associated choroidal ischemia leading to altered permeability. OCT is an important imaging modality allowing for signs of GCA to be better characterized.

Introduction: Posterior subtenon injection of triamcinolone acetonide (PSTA) is commonly done to treat refractory cases of macular edema. Complications may arise from the procedure as well as from the use of the periocular steroid medications. Side effects include subconjunctival hemorrhage, progression of cataract, scleral perforation (resulting in subretinal, subhyaloid, or intravitreal injection of the drug), retinal detachment, ptosis, orbital fat prolapse, orbital abscess, infectious scleritis, ocular hypertension, and scleral abscess. Here we describe a case of inadvertent subretinal triamcinolone acetonide (TA) deposition from a PSTA procedure without any adverse vision-threatening outcomes.

Case presentation: We report a patient who presented with a history of superior temporal left eye macula-off rhegmatogenous retinal detachment, which was successfully repaired with a scleral buckle (SB), pars plana vitrectomy, and gas placement. Due to persistent diplopia, the SB was removed after 1 year post-operatively. Due to the development of cystoid macular edema, a PSTA was performed after the patient failed topical steroids and NSAIDs. The procedure was halted early due to unexpected resistance during the injection. A dilated fundus exam showed the presence of subretinal triamcinolone acetonide. The patient was observed and found to have no complications with almost complete resolution of the triamcinolone acetonide after 3 months.

Conclusion: In previous SB patients, it is important to highlight the risk of globe penetration, subretinal deposition of TA, formation of retinal breaks, or reopening of prior retinal breaks with posterior subtenon injection, which could have adverse effects on the local retina as well as the risk of retinal detachment.