Case Reports in Ophthalmology最新文献

Introduction: Although the eye is rarely the sole target of an immediate allergic-type response, ocular signs and symptoms often represent the most prominent features of systemic allergic reactions. Nonsteroidal anti-inflammatory drugs (NSAIDs), due to their cyclooxygenase inhibitory activity, can trigger nonallergic hypersensitivity responses. Ketoprofen, in particular, is one of the most potent cyclooxygenase inhibitors at therapeutic plasma concentrations and is recognized as a frequent photoallergic agent. However, localized hypersensitivity reactions following systemic administration are exceedingly rare. This report describes an unusual case of allergic conjunctivitis occurring after oral administration of ketoprofen.

Case presentation: A 13-year-old Caucasian girl presented to the pediatric emergency department of a private hospital with swelling, progressive ocular redness, burning, itching, and upper airway obstruction. These symptoms developed shortly after the oral administration of ketoprofen (20 mg/mL), prescribed to manage a low-grade fever (37.9°C) and sore throat. No additional systemic manifestations were observed.

Conclusion: An uncommon case of conjunctivitis following oral ketoprofen use highlights the need for further research on NSAID-induced hypersensitivity and the value of detailed laboratory evaluation to clarify its causes.

Introduction: This study aimed to describe the clinical characteristics of a 1-year-old male patient with Leber congenital amaurosis type 1 (LCA1) and investigate the genetic variations underlying his symptoms.

Case presentation: A comprehensive medical history of the patient was obtained with thorough examinations via mydriatic optometry, fundus photography, and flash electroretinography. To identify causative mutations, whole-exome sequencing (WES) was conducted. Potential pathogenic mutations identified with WES were further validated via Sanger sequencing, which was also performed on family members to confirm the origins of the mutations. Based on clinical and laboratory findings, the patient was diagnosed with LCA1. Two heterozygous mutations in the GUCY2D gene, c.835G>A and c.2516_2517del, were detected in the patient with WES. Both mutations were assigned as likely pathogenic according to ACMG guidelines. c.2516_2517del has not been described previously. Sanger sequencing confirmed that the unaffected father and mother carried c.835G>A and c.2516_2517del, respectively.

Conclusion: The patient was a typical case of LCA1 with two GUCY2D mutations. To the best of our knowledge, this is the first report of the allele mutation c.2516_2517del in a patient with LCA1.

Introduction: This report describes a rare case of idiopathic orbital inflammation with posterior scleritis complicated by optic perineuritis and ocular motility disorder.

Case presentation: A 22-year-old woman presented with right ocular pain, eyelid swelling, and blurring of vision for 5 days. Her best corrected visual acuity was 20/20 in the right eye, although a myopic shift was noted. An ocular motility disorder was identified on examination. Slit lamp and fundus examinations revealed diffuse scleral injection, anterior chamber inflammation, and serous retinal detachment. B-scan ultrasound showed thickening of the choroid and sclera. Gadolinium-enhanced magnetic resonance imaging revealed marked thickening and enhancement of the right optic nerve sheath. Humphrey visual field testing showed an enlarged blind spot and an inferior field defect. The systemic workup and laboratory tests were unremarkable, aside from elevated CRP and ESR. The patient was diagnosed with idiopathic orbital inflammation with posterior scleritis, optic perineuritis, and ocular motility disorder. Treatment with oral prednisolone (50 mg/day) improved all ocular findings, but a relapse occurred while tapering to 10 mg/day. The symptoms resolved after increasing the dose of prednisolone, and adalimumab was added to prevent recurrence. Adalimumab was continued while tapering prednisolone and replacing it with oral hydrocortisone for secondary adrenal insufficiency. Six months after the discontinuation of prednisolone, the patient remains in remission.

Conclusion: This rare case of idiopathic orbital inflammation with posterior scleritis, optic perineuritis, and ocular motility disorder was successfully controlled with adalimumab after relapse, suggesting its efficacy in preventing recurrence.

Introduction: Orbital T-cell lymphoma is a rare and diagnostically challenging malignancy that can closely mimic inflammatory conditions such as Tolosa-Hunt syndrome (THS), particularly due to overlapping clinical and radiological features.

Case presentation: We report the case of a 21-year-old male who initially fulfilled the ICHD-3 criteria for THS, demonstrating unilateral painful ophthalmoplegia and transient steroid responsiveness. However, recurrence of symptoms and subsequent histopathological evaluation confirmed the presence of extranodal orbital T-cell lymphoma. This case underscores the critical importance of maintaining a high index of suspicion for malignancy in steroid-responsive orbital syndromes, especially when atypical features or relapses occur. Early biopsy and multidisciplinary coordination enabled timely initiation of CHOEP chemotherapy, leading to slight clinical improvement. This report adds to the growing body of literature emphasizing the masquerade potential of lymphoid malignancies in orbital inflammation.

Conclusion: Orbital T-cell lymphoma can mimic benign inflammatory conditions such as orbital cellulitis. Prompt biopsy and multidisciplinary evaluation are crucial for accurate diagnosis and timely initiation of appropriate treatment.

Introduction: The FIL SSF intraocular lens (IOL) (Carlevale, Soleko, Italy) is a sutureless scleral-fixated lens designed for cases of aphakia with insufficient capsular support. While generally stable, intraoperative complications such as haptic rupture may occur. We present a novel rescue technique for scleral fixation of FIL SSF IOL following rupture of the trans-scleral plug during combined penetrating keratoplasty, three-port pars plana vitrectomy, and IOL implantation.

Case presentation: During the combined procedure, rupture of one FIL SSF IOL plug was identified. The compromised haptic was externalized into the subconjunctival space. The sclerotomy was sutured to secure the haptic, achieving both immobilization and stable positioning of the IOL. Postoperatively, the patient was followed for 6 months. Best corrected visual acuity reached 0.2 (decimal scale), intraocular pressure was stable at 14 mm Hg in the left eye, the corneal graft remained clear, and the FIL SSF IOL maintained centration without further complications.

Conclusion: This case demonstrates that intraoperative rupture of the FIL SSF IOL haptic can be effectively managed using a scleral anchoring technique. This approach provides a stable and safe alternative for salvaging IOL fixation during complex anterior and posterior segment surgeries.

Introduction: The pachychoroid spectrum refers to a group of chorioretinal disorders including pachychoroid pigment epitheliopathy (PPE), pachychoroid neovasculopathy (PNV), polypoidal choroidal vasculopathy (PCV), and central serous chorioretinopathy (CSC). These conditions are thought to represent progressive stages, beginning with subclinical retinal pigment epithelium (RPE) changes in PPE, advancing to serous retinal detachment in CSC, followed by choroidal neovascularization in PNV, and culminating in aneurysmal dilation of vessels in PCV. We present a rare case of the pachychoroid spectrum in which lesions in all four stages were simultaneously observed in a single, fovea-sparing eye.

Case presentation: An 85-year-old man presented with a 1-month history of a visual disturbance in his left eye. Examination revealed all four stages of pachychoroid disease in the same eye: choroidal neovascular membrane (CNV) with subretinal hemorrhage (pachychoroid neovascularization, PNV), multiple RPE defects (PPE), and findings consistent with CSC and PCV. He was treated with a combination of anti-VEGF injections and focal laser therapy with the lesions stabilizing after 2 years.

Conclusion: Although it has features similar to age-related macular degeneration, pachychoroid spectrum is a distinct disease entity, with a slower onset and greater response to initial therapy. It may necessitate therapies otherwise not used for other causes of neovascularization like focal laser treatment and verteporforin photodynamic therapy. It is a unique pathologic process presenting with varying stages/lesions that have distinct morphological features but are thought to be a part of the same spectrum.

Introduction: Actinic keratosis (AK) is a premalignant epithelial lesion primarily associated with chronic ultraviolet exposure. While it commonly affects sun-exposed skin, its occurrence on the ocular surface is rare. Clinically, it may resemble ocular surface squamous neoplasia (OSSN), necessitating histopathological confirmation for accurate diagnosis and appropriate management.

Case presentation: A 78-year-old male, HBsAg positive, presented with progressive diminution of vision in the right eye for 6 months and a visible mass over the right eye for the past 2 months. Best-corrected visual acuity was 6/12p in the right eye and 6/9 in the left eye. Slit-lamp examination of the right eye revealed a 4 × 1 mm keratotic lesion with surrounding hyperpigmentation on the corneal surface. Both eyes were cataractous. Fundus evaluation was within normal limits in both eyes. A clinical diagnosis of OSSN was considered. The lesion was excised and sent for histopathological analysis, which revealed features consistent with AK. The patient was treated with topical mitomycin C 0.02% and topical prednisolone acetate 1% postoperatively. No recurrence was observed on follow-up.

Conclusion: Ocular surface AK is a rare and potentially premalignant lesion that may mimic OSSN. Clinical suspicion, combined with histopathological examination, is crucial for diagnosis. Surgical excision followed by topical chemotherapy offers effective management and reduces the risk of recurrence or malignant transformation.

Introduction: The aim of the study was to report the intraoperative signs, management, and postoperative outcomes of iris hemorrhage during implantable collamer lens (ICL) surgery.

Case presentation: A 32-year-old Asian woman experienced iris bleeding via a superior incision during distal haptics delivery in ICL surgery for her right eye. The inferior iris was displaced against the ocular wall, resulting in traction-induced vertical elongation and elliptical distortion of the pupil, which indicated excessive iris traction and ultimately led to focal rupture of delicate iris vessels with intraoperative hemorrhage. Immediate injection with ophthalmic viscosurgical device into the anterior chamber aimed to tamponade the bleeding from the broken vessels; after confirming cessation of active bleeding, the surgery was proceeded and completed uneventfully. During postoperative follow-up, slit lamp examination showed persistent intraocular inflammation, increased intraocular pressure (IOP), and hyphema. Right eye was treated with anterior chamber paracentesis for the elevation of the IOP; IOP-lowering medications and topical anti-inflammation eyedrops were continued. No further interventions were performed. Slit lamp examination demonstrated progressive recovery in anterior chamber reaction. No further complications occurred during the follow-up.

Conclusion: Intraoperative iris hemorrhage is a rare but potentially serious complication for ICL surgery; timely intraoperative recognition of the pupil distortion and closely postoperative patient monitoring with medical management can avoid the irreversible damage to the eye.

Introduction: The aim of the study was to evaluate the long-term efficacy of eplerenone in patients with peripapillary pachychoroid syndrome (PPS), a recently described entity characterized by vascular congestion of Haller's layer vessels near the optic nerve.

Case presentations: Three male patients (mean age: 70.3 years) with PPS received oral eplerenone 50 mg daily for 12 months. Baseline and post-treatment choroidal thickness, visual acuity, and retinal fluid status were assessed. At baseline, mean choroidal thickness was 420 µm, and mean visual acuity was 70 letters. After 12 months of treatment, all patients showed a significant reduction in choroidal thickness (mean: 48 µm, p < 0.05) and near-complete resolution of intraretinal and subretinal fluid. No adverse events were reported.

Conclusions: Long-term eplerenone treatment appears effective in reducing choroidal thickness and improving visual outcomes in PPS patients. These findings suggest mineralocorticoid receptor antagonists may be valuable in managing pachychoroid spectrum disorders.

Introduction: Optic disc pit maculopathy is a challenging retinal disease that can significantly impact the quality of life, especially in pediatric patients. We report our surgical approach in the management of an 8-year-old boy with this rare condition who was referred to our clinic.

Case presentation: An 8-year-old male patient presented with visual acuity of 20/200 in the left eye. After complete examination and OCT imaging was performed, the diagnosis of optic disc pit maculopathy (ODPM) was obtained. ODPM is a challenging condition to treat without a gold standard approach, especially in the pediatric population. A surgical approach was decided in order to treat this condition. This involved performing a pars plana vitrectomy and an internal limiting membrane (ILM) flap over the optic disc pit. The operation was successful as the ILM flap covered the optic disc pit sufficiently enough to block communication between the vitreous cavity and the subretinal space. This led to the resolution of macular edema during the postoperative period. One year post surgery, the boy's visual acuity improved to 20/32, accompanied by the patient's overall satisfaction and the anatomical confirmation of the successful result using OCT.

Conclusion: Taking into consideration the surgical difficulties associated with this age group, we highlight the efficacy and safety of the ILM flap inversion technique as a viable surgical option for optic disc pit maculopathy in pediatric patients.