Case Reports in Ophthalmology最新文献

Introduction: Epibulbar choristoma is a benign congenital lesion containing histologically normal-appearing tissue in an abnormal ectopic location. An epibulbar choristoma is classified as either epibulbar dermoid, dermolipoma, or complex choristoma based on histological examination. The case presented was a presumed epibulbar dermolipoma with no signs of ossification on imaging, examination, or intraoperatively until the specimen was examined histologically, clarifying the lesion as an epibulbar complex choristoma. Reassuringly, the presence of bone in such lesions should not change management.

Case presentation: A mother noticed a small fleshy mass on her 9-year-old daughter's superotemporal bulbar conjunctiva. The suspected epibulbar dermolipoma was confirmed with MRI and initially managed conservatively. Two years later, she was referred for apparent growth and cosmetic concerns, and she underwent surgical debulking.

Conclusion: We present this case for its unusual presentation and histological findings. Orbital surgeons should be aware of the possibility of ossification of epibulbar choristomas and avoid confusion with alternative diagnoses. Clarification of the latest classification system for epibulbar choristomas is provided.

Introduction: Photodynamic therapy (PDT) has shown substantial benefit in the treatment of choroidal hemangioma (CH) in recent years. This report describes the use of PDT with overlapping spots in a patient with Sturge-Weber syndrome (SWS) and large circumscribed CH.

Case presentation: A 9-year-old girl with SWS and a history of glaucoma in her left eye was referred to a retina clinic for possible macular changes. Examination revealed decreased vision in the left eye, pigmentary changes in the macula, and choroidal thickening in the posterior pole. After being lost to follow-up for 2 years, the patient returned with further vision deterioration with best-corrected visual acuity (BCVA) of 20/150 and new subretinal fluid (SRF). Imaging findings were consistent with a diagnosis of CH and SRF. PDT with verteporfin was initiated on the entire area with multiple overlapping spots, resulting in resolution of SRF and improvement in visual acuity and choroidal contour. At 18-month post-treatment, the patient's BCVA was 20/25 with no recurrence of SRF or increased choroidal thickening. Significant pigmentary changes and subretinal hyper-reflective material were observed in the OCT of the treated area.

Conclusion: Multiple overlapping laser spots of PDT can result in longstanding regression of large circumscribed CH in a patient with SWS with excellent final visual acuity. However, significant subretinal changes may also result following this method of treatment.

Introduction: We report a case of bilateral pigment dispersion syndrome after 13 years of uncomplicated implantable collamer lens (ICL) surgery.

Case presentation: A 53-year-old woman was referred from her optometrist to our glaucoma clinic due to early superonasal visual field loss in both eyes. She was asymptomatic with no changes in visual acuity and had undergone bilateral ICL implantation 13 years ago to correct her high myopia. Clinical examination revealed pigment deposition on the corneal endothelium, iris transillumination defects, and iris vaulting at the areas of contact with the ICL. Gonioscopy showed open angles with significant pigmentation of the trabecular meshwork. The diagnosis of pigment dispersion syndrome secondary to ICL implantation was made, and subsequent follow-up visits demonstrated normal intraocular pressure IOP and stable visual fields.

Conclusion: Pigmentary dispersion syndrome can occur several years after ICL implantation. This case report emphasizes the need for long-term follow-up and monitoring after ICL surgery.

Introduction: Oculogyric crisis (OGC) is a dystonic movement disorder of varying durations that manifests as bilateral paroxysmal upward eye deviation accompanied by involuntary blinking, tongue protrusion, and autonomic symptoms. Separately, septo-optic dysplasia (SOD) is a congenital disorder involving hypoplasia of the optic nerve as well as hypothalamic and pituitary abnormalities. In the presented case, we report a case of OGC in the setting of SOD with proposed pathogenesis.

Case presentation: A 27-year-old female presented with a history of SOD (optic nerve hypoplasia and hypopituitarism) with acute, recurrent, painless, bilateral, intermittent, simultaneous tonic conjugate upward eye deviation (i.e., OGC) and dystonic body posturing. She experienced her first episode upon meeting her biological sister for the first time at a loud, crowded public restaurant with continued episodes of OGC increasing in frequency and duration over the subsequent months. She later responded well to treatment with carbidopa/levodopa.

Conclusion: Based on our current understanding of OGC, we hypothesize that acute stressful life events in the setting of prior hypothalamic-pituitary axis dysfunction secondary to SOD could lower the threshold for developing OGC. Although most cases of OGC are idiopathic, various etiologies including medications, stress, and hormonal imbalance have been postulated as possible pathogenic mechanisms. We describe a case of SOD with OGC, and based upon our review of the English language ophthalmic literature, we believe that our case is novel.

Introduction: Blau syndrome (BS) is a rare autoimmune disease. We report here an atypical case of BS.

Case presentation: We present a case of late-onset eye manifestations in a Chinese girl of 18 years old with sporadic BS, presenting with panuveitis. We performed comprehensive ocular examinations including fluorescein fundus angiography and indocyanine green angiography for her. The oral hormone plus local anti-inflammatory eye drops have well controlled the inflammation of her eyes.

Conclusion: Our case highlights the necessity of systemic medical history inquiry for every eye discomfort.

Introduction: This report describes a case of elevated intraocular pressure following the use of 0.125% atropine eye drops in a child wearing orthokeratology lenses.

Case presentation: A 9-year-old boy presented to our clinic with myopia, and he had been wearing orthokeratology lenses overnight for 23 months. He was treated previously with a once-daily administration of topical 0.125% atropine eye drops to reduce myopic progression. Three days after treatment, his intraocular pressure was 36 mm Hg in the right eye and 32 mm Hg in the left eye. Two days after the discontinuation of atropine eye drops and overnight orthokeratology lenses, the intraocular pressure was 18/20 mm Hg in both eyes.

Conclusion: Low-dose atropine eye drops can cause intraocular pressure elevation in patients wearing overnight orthokeratology lenses. Although it may resolve promptly, short-term follow-up with intraocular pressure checks may be necessary for the early diagnosis and treatment of this complication.

Introduction: Rhabdomyosarcoma is a rare paediatric cancer, with the head and neck region representing a major anatomical site for rhabdomyosarcoma. In particular, orbital rhabdomyosarcoma is the most common region among children. However, rhabdomyosarcoma originating from the conjunctiva in paediatric population is a rare disease, and this knowledge is essential in order to ensure prompt treatment and early intervention.

Case presentation: We discuss a rare case of primary conjunctival rhabdomyosarcoma in an 8-year-old Caucasian girl. She presented to a paediatric ophthalmology clinic with a 5-day history of a rapidly growing conjunctival lesion in the superior fornix of the right eye. An urgent excisional biopsy was performed which yielded a large 30-mm multilobulated, vascular, and papillomatous specimen with histopathological features consistent with embryonal rhabdomyosarcoma. She was urgently referred to oncology and was treated with systemic chemotherapy.

Conclusion: Therapeutical options and prognosis of rhabdomyosarcomas are based on clinical findings, tumour staging, and grouping, combined with histopathological and molecular features. Although rare, it is important to note that in the paediatric population, rhabdomyosarcoma can originate from the conjunctiva. Knowledge of its clinical, histopathological, and imaging characteristics is essential in order to achieve early diagnosis and timely treatment.

Introduction: This case report describes a rare case of fungal keratitis following femtoLASIK. Despite targetted antifungal therapy, this case necessitated an innovative surgical approach to manage an unexpected corneal perforation.

Case presentation: A 35-year-old male presented 3 weeks post-femtoLASIK for myopic astigmatism with discomfort and reduced vision in his right eye. He was diagnosed with fungal keratitis surgery caused by Purpureocillium lilacinum and was treated with a myriad of therapy. Unfortunately, the patient developed corneal perforation during flap lift and flap bed irrigation. An innovative approach involving a tectonic autograft using a viable LASIK flap, followed by prompt penetrating keratoplasty, was utilised.

Conclusion: This technique was effective and able to mitigate the progression to an open globe.

Introduction: Multiple myeloma (MM), a plasma cell malignancy, is a systemic disease affecting various body organs. Plasmacytoma of bone and extramedullary disease (EMD) are presentations of MM. EMD is usually the sign of a more aggressive form of the disease. Herein, we report a patient with refractory MM presenting with extramedullary plasmacytoma in the superior oblique (SO) muscle.

Case presentation: A 51-year-old female presented complaining of gradual protrusion of the left eye and ocular pain from 20 days prior. She received bone marrow transplantation 1 year prior and was on a chemotherapy regimen for MM for the past 1 year. Ocular examination revealed proptosis of the left eye and mild limitations of adduction and elevation. Orbital magnetic resonance imaging demonstrated remarkable enlargement of the left SO muscle with focal contrast enhancement. The patient underwent a biopsy and mass debulking. The histopathologic exam revealed fibromuscular tissue containing a neoplasm composed of sheets of plasmacytoid cells in a varying degree of differentiation with intervening scantly vascularized stromal components. The plasmacytoid cells were diffusely positive for a cluster of differentiation 138 (CD138), leading to a diagnosis of EMD involving the EOM and soft tissue of the orbit. The patient underwent palliative radiotherapy and a systemic workup. The PET-CT scan revealed involvement of the pelvic bone and left calf. Accordingly, the chemotherapy regimen was upgraded to reflect the aggressive nature of the disease. In the last follow-up, there was no sign of tumor reactivation in the orbital soft tissues. Unfortunately, the patient succumbed to her illness 7 months following her most recent presentation.

Conclusion: Early recognition of disease recurrence is lifesaving in MM patients; ophthalmic manifestations should be seriously considered as a sign of MM activity.

Introduction: Hypoxia-inducible factor-prolyl hydroxylase (HIF-PH) inhibitors, used in the treatment of renal anemia, hold the potential to increase the production of vascular endothelial growth factors. Therefore, HIF-PH inhibitors may exacerbate retinal hemorrhage in diseases such as diabetic retinopathy. Here, we present a case involving the administration of an HIF-PH inhibitor, resulting in the exacerbation of retinal hemorrhage in a patient with diabetic retinopathy.

Case presentation: A 32-year-old man with diabetes mellitus and renal anemia caused by diabetic nephropathy was referred to our department for ophthalmic examination, revealing diabetic retinopathy with scattered retinal hemorrhages, exudates, and diabetic maculopathy in both eyes. Darbepoetin alfa was initially administered and switched to the HIF-PH inhibitor roxadustat on day 74. By day 88, fresh retinal hemorrhage was observed in the right eye. On day 132, the retinal hemorrhage had further worsened, with new preretinal hemorrhage in both eyes. Roxadustat was discontinued, replaced with darbepoetin alfa, resulting in retinal hemorrhage improvement by day 181 (49 days post-roxadustat cessation). On day 201, fundus hemorrhage further improved, optical coherence tomography showed no macular edema or subretinal fluid, and the retina was thinning. Fluorescein angiography showed neovascular vessels, active fluorescein leakage, and extensive avascular areas in both eyes, prompting pan-retinal photocoagulation. Visual acuity remained stable throughout treatment.

Conclusion: Patients with advanced diabetic retinopathy taking HIF-PH inhibitors should be aware of retinal hemorrhage exacerbations. If observed, the treatment plan, including discontinuation of the HIF-PH inhibitor or switching to another agent, should be discussed with a diabetologist, nephrologist, and ophthalmologist.