Case Reports in Ophthalmological Medicine最新文献

We report a 46-year-old male patient with retinocytoma who presented at the age of 31 asymptomatically. An intraocular retinal mass was incidentally found in his right eye, when he underwent ophthalmological assessment for refractive surgery. This tumor consisted of a calcified sessile basis partially covered by a pedunculated salmon-pink growth. Initially, the tumor was diagnosed as a retinocytoma with clinical suspicion of malignant transformation into retinoblastoma and treated by four sessions of laser photocoagulation. Six and a half years later, the tumor relapsed, and he was treated with a Ruthenium plaque. Following brachytherapy, he had two episodes of right-sided vitreous hemorrhage that spontaneously cleared up, and the remaining finding in the vitreous cavity was interpreted as asteroid hyalosis. He underwent vitrectomy about five years following brachytherapy. The analysis of the vitreous material revealed the presence of inactive vitreous seeds composed of small round blue cells, compatible with a type 2 regression.

A rare multisystemic, ciliopathic autosomal recessive disorder called Bardet-Biedl syndrome (BBS) primarily affects children of consanguineous marriages. Both men and women are affected by it. It is characterized by some major and many minor features to aid in the clinical diagnosis and management. Here, we reported two Bangladeshi patients (a 9-year-old girl and 24-year-old male) who were presented with various major and minor features of BBS. Both patients came to us with the symptoms including excessive weight gain, poor vision, and learning disabilities with polydactyly. Our case 1 presented four primary features (retinal degenerations, polydactyly, obesity, and learning deficits) and six secondary features (behavioral abnormality, delayed development, diabetes mellitus, diabetes insipidus, brachydactyly, and LVH), whereas case 2 presented five major criteria (truncal obesity, polydactyly, retinal dystrophy, learning disabilities, and hypogonadism) and six minor criteria (strabismus and cataract, delay in speech, behavioral disorder, developmental delay, brachydactyly and syndactyly, and impaired glucose tolerance test). We diagnosed the cases as BBS. Because there is no specific treatment for BBS, we highlighted the importance of diagnosing it as early as possible so that comprehensive and multidisciplinary care can be offered to prevent avoidable morbidity and mortality.

Purpose: To report a case of central retinal artery occlusion (CRAO) associated with subacute Streptococcus gordonii endocarditis secondary to a dental infection. Observations. A 27-year-old male presented with acute monocular vision loss in the setting of a stroke and seizure. A fundus exam revealed macular whitening and a cherry-red spot. Edema of the inner retinal layers was confirmed on macular optical coherence tomography, consistent with CRAO. Initial imaging (carotid Doppler, EKG, and transthoracic echocardiography) and a comprehensive laboratory workup did not reveal an etiology for the stroke or vision loss. Brain magnetic resonance imaging showed T1 hyperintensity with surrounding edema, which prompted a workup for possible septic emboli versus occult malignancy. Subsequent blood cultures led to the detection and diagnosis of Streptococcus gordonii endocarditis. It was subsequently revealed that the patient had self-extracted his molar two months prior to the onset of symptoms.

Conclusions: Endocarditis has been associated with Roth spots and inflammatory findings in the posterior segment. However, CRAO caused by vegetal septic embolism is rare. To our knowledge, this represents the first reported case of endocarditic CRAO with Streptococcus gordonii confirmed as the causative microbe. Retinal vascular occlusion in a young patient with no distinct risk factors should prompt a comprehensive dental history and infectious workup, with consideration given to early transesophageal echocardiography.

Intraocular medulloepithelioma is a rare embryonal tumor that is believed to arise from the epithelium of the medullary tube. We report a 37-year-old female with medulloepithelioma presented at the age of 17 with a one-month history of left-sided visual deterioration and visible iris lesion. Birth history and medical and family histories were insignificant. The left eye revealed a vascularized iris mass. Further examination revealed a grey-white ciliary body mass and a subluxated lens with best-corrected visual acuity (BCVA) of 0.5. The patient underwent partial lamellar corneo-sclerouvectomy. The histological and electron microscopic findings revealed medulloepithelioma. To reduce the risk of recurrence of the probable malignant tumor, she was treated with Ruthenium plaque therapy about six weeks following surgical removal. Pars plana vitrectomy and lensectomy with laser photocoagulation of the peripheral retina were performed at the removal of the brachytherapy plaque. She regained her BCVA of 1.0 in her left eye 3.5 months following pars plana vitrectomy. At 20-year follow-up, no tumor recurrence was seen and her BCVA remained 1.0.

Background: The underlying mechanisms of papilledema associated with intracranial hypertension remain unclear. A case of bilateral papillary edema in a patient with chronic idiopathic intracranial hypertension who was asymptomatic during her two pregnancies is reported. Case Presentation. A 19-year-old Caucasian female, in her third month of pregnancy, complained of difficulties with close reading. The patient's visual acuity was 20/20 on the Snellen chart and improved with a 0.50 D correction in both eyes. Near vision and slit lamp examinations revealed normal findings bilaterally. However, a fundus examination showed bilateral papillary edema without evidence of hemorrhages or neovascularization. Blood tests were unremarkable, except for a slight increase in C-reactive protein levels. The patient had a prepregnancy weight of 63 kilograms, with a BMI of 24.91 kg/m². Magnetic resonance imaging of the brain revealed features consistent with chronic idiopathic intracranial hypertension, which resolved after delivery. Two and a half years later, during a subsequent pregnancy, the patient experienced a recurrence of bilateral papillary edema due to the IIH. It was managed similarly as the first occurrence, resulting in bilateral anatomical and functional recovery. Recent research revealed that, during pregnancy, hormones interact with the central nervous system, leading to an increase in the size of neurons which could potentially result in intracranial hypertension.

Conclusions: The influence of hormonal fluctuations during pregnancy on the development of transient central nervous system abnormalities in individuals with chronic intracranial hypertension, leading to papillary edema, remains a matter of debate.

Purpose: We present a rare case of Citrobacter koseri culture-positive endophthalmitis in a postvitrectomy silicone oil-filled eye. Case report. A 64-year-old male patient presented to our ophthalmology emergency room with representative symptoms of acute endophthalmitis. He underwent a plana vitrectomy oil-filled tamponade previously. Preoperative and postoperative findings of the case were reported.

Results: Culture tests of aqueous, silicone oil, and vitreous taps were positive for Citrobacter koseri.

Conclusions: Culture-positive endophthalmitis in a silicone oil-filled eye has very rarely been in the literature. The described cases were caused by acute inflammatory reactions to silicone oil and were culture-negative. The postvitrectomy culture-positive endophthalmitis caused by Citrobacter is a very rare condition, and its management is not so smooth. Approaching with silicone oil removal, intraoperative intravitreal antibiotic injection, and silicone oil reinjection was performed in our case with good outcomes.

Purpose: To describe the results of an intravitreal dexamethasone implant in managing autoimmune retinopathy (AIR).

Methods: Two patients affected by AIR underwent intravitreal dexamethasone implantation and were followed by ocular coherence tomography, visual fields, and electroretinography.

Results: The patients showed an interruption of the functional losses and remained stable with semestral injections.

Conclusion: AIR is a complex condition with no standard treatment. The use of dexamethasone could be a valid option.

Background: This study is aimed at exploring a case of choroidal Langerhans cell histiocytosis (LCH) successfully treated with systemic corticosteroid and immunosuppressant. Case presentation. A 24-year-old man with known multisystem LCH developed loss of vision, ocular pain, conjunctival injection, panuveitis, and a ciliochoroidal mass. After receiving an intravenous methylprednisolone pulse, oral high-dose corticosteroids, and methotrexate, the mass resolved quickly and completely without flare-ups during 6 months of follow-up.

Conclusions: Intraocular involvement of LCH is rare and can present with or without a history of multisystemic disease. The diagnosis is challenging, and the standard treatment is not established. Systemic anti-inflammatory and immunosuppressive therapy could be an effective treatment, as the LCH itself contains an essential element of inflammation and the symptoms may be mainly inflammatory.

Purpose: Cyclic esotropia (alternate-day squint) is a poorly understood form of strabismus that mostly occurs at younger pediatric ages. It demonstrates classically a 48-hour cycle with 24 hours of manifest esotropia and 24 hours of orthotropia, which is conventionally managed by bilateral or unilateral 2-muscle surgery. We aimed to report a child with cyclic esotropia who was surgically treated by a conservative unilateral 1-muscle approach.

Methods: Case report.

Results: A 3.5-year-old girl presented to the strabismus department with an intermittent esodeviation for 2 years that became cyclic in the last 3 months. The diagnosis of primary classical cyclic esotropia was made after seeing her multiple times on different days. The girl was emmetropic bilaterally, had normal visual acuities in both eyes, and high-angle right esodeviation (45-50 ^Δ ) with normal laboratory and MRI results. Unilateral maximal single-muscle recession of the right medial rectus was performed, and the child was followed up for 9 months. The girl developed excellent alignment after the surgery both at distance and near without cyclic pattern, and near-normal stereopsis (by animals: 100 sec. of arc) with binocularity was reached. The girl did not experience under- or overcorrection nor have a recurrence postoperatively.

Conclusion: This is the first report of "one eye single-muscle" surgery for high-angle cyclic esotropia. Conservative unilateral medial rectus recession seems to be sufficient to permanently block the circadian rhythm and restore binocular fusion and stereopsis.

Background: In recent years, immune checkpoint inhibitors (ICI) have been often used for several types of cancers. Immune-related adverse events (irAEs) are autoimmune responses caused by ICI. Among the different types of irAEs, uveitis is common in ophthalmology. Moreover, there are reports on Vogt-Koyanagi-Harada (VKH) disease-like uveitis. In most cases, VKH, as in the usual VKH, is managed with intravenous methylprednisolone therapy. Case Report. A 72-year-old man was diagnosed with gastric cancer, and he was treated with nivolumab, a type of ICI. After eight cycles of nivolumab therapy, he developed fulminant type 1 diabetes mellitus and diabetic ketoacidosis. Thus, the treatment was discontinued. Subsequently, the patient was referred to our department due to bilateral blurry vision. He had decreased visual acuity in both eyes, and slit lamp examination revealed the presence of bilateral anterior chamber cells and keratic precipitates. Fundus examination showed bilateral serous retinal detachment (SRD), wavy retinal pigment epithelium (RPE), and choroidal thickening. Cerebrospinal fluid examination revealed prominent pleocytosis. Thus, we initiated eye drop therapy and subtenon injection of triamcinolone acetonide on the right eye only. After 1 month, SRD and wavy RPE disappeared, and the patient's visual acuity improved. Further, both eyes had similar improvements in visual acuity and abnormal findings. Oral prednisolone was subsequently administered for hearing loss. However, intravenous methylprednisolone was not used, and ophthalmologic findings and visual acuity did not change before and after systemic steroid therapy. One year after disease onset, SRD and wavy RPE did not relapse.

Conclusion: Nivolumab-induced VKH disease-like uveitis can have good outcomes even in a patient managed without intravenous methylprednisolone therapy.