Case Reports in Ophthalmology最新文献

{"title":"Fungal Keratitis Post-FemtoLASIK: A Novel Therapeutic Approach with LASIK Flap Autograft and Penetrating Keratoplasty: A Case Report.","authors":"Jackson Chee Chea Lee, Yachana Vaibhav Shah, Hannah Kathleen Gooding, Graham David Barrett","doi":"10.1159/000538062","DOIUrl":"10.1159/000538062","url":null,"abstract":"<p><strong>Introduction: </strong>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.</p><p><strong>Case presentation: </strong>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 <i>Purpureocillium lilacinum</i> 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.</p><p><strong>Conclusion: </strong>This technique was effective and able to mitigate the progression to an open globe.</p>","PeriodicalId":9635,"journal":{"name":"Case Reports in Ophthalmology","volume":"15 1","pages":"273-278"},"PeriodicalIF":0.4,"publicationDate":"2024-03-28","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC10978041/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"140317849","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}

{"title":"Superior Oblique Muscle Extramedullary Plasmacytoma in a Patient with Multiple Myeloma and a Review of Literature.","authors":"Amirreza Veisi, Kimia Daneshvar, Sadid Hooshmandi, Maryam Najafi, Peyman Mohammadi Torbati, Kiana Hassanpour","doi":"10.1159/000538120","DOIUrl":"10.1159/000538120","url":null,"abstract":"<p><strong>Introduction: </strong>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.</p><p><strong>Case presentation: </strong>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.</p><p><strong>Conclusion: </strong>Early recognition of disease recurrence is lifesaving in MM patients; ophthalmic manifestations should be seriously considered as a sign of MM activity.</p>","PeriodicalId":9635,"journal":{"name":"Case Reports in Ophthalmology","volume":"15 1","pages":"265-272"},"PeriodicalIF":0.5,"publicationDate":"2024-03-25","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC10963055/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"140287046","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}

{"title":"Exacerbation of Diabetic Retinopathy following Hypoxia-Inducible Factor-Prolyl Hydroxylase Inhibitor Administration: A Case Report.","authors":"Nobuaki Ariyoshi, Fumiaki Higashijima, Makiko Wakuta, Tadahiko Ogata, Manami Ohta, Kazuhiro Kimura","doi":"10.1159/000537913","DOIUrl":"10.1159/000537913","url":null,"abstract":"<p><strong>Introduction: </strong>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.</p><p><strong>Case presentation: </strong>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.</p><p><strong>Conclusion: </strong>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.</p>","PeriodicalId":9635,"journal":{"name":"Case Reports in Ophthalmology","volume":"15 1","pages":"256-264"},"PeriodicalIF":0.5,"publicationDate":"2024-03-25","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC10963054/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"140287045","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}

{"title":"Giant Conjunctival Cyst of the Orbit in a Patient with Previous Enucleation.","authors":"João Ponces Ramalhão, Diogo Rodrigues, Beatriz Vieira, Miguel Gonçalves Afonso, Pedro Manuel Baptista, Nuno Jorge Lamas, Maria Araújo","doi":"10.1159/000535598","DOIUrl":"10.1159/000535598","url":null,"abstract":"<p><strong>Introduction: </strong>Conjunctival cysts of the orbit development are a rare complication in enucleated patients. They result from the entrapment of conjunctival epithelium within the orbit.</p><p><strong>Case presentation: </strong>We report the case of a 25-year-old man with a history of enucleation for a retinoblastoma of the right eye who presented with difficulty in fitting his eye prothesis. On his past medical records, there was no reference to the placement of any orbital implant at the time of the surgery. Biomicroscopy of the right eye revealed a thickened bulbar conjunctiva, an inferior symblepharon, and a translucid central area with vascularization. Imaging was remarkable for a cystic cavity filling the whole right orbit. Biopsy revealed the diagnosis of a conjunctival cyst, and drainage was performed, alleviating the patient's symptoms.</p><p><strong>Conclusion: </strong>Orbital conjunctival cysts may pose a clinical problem, and treatment modalities include surgical excision, absolute alcohol injection, and trichloroacetic acid injection.</p>","PeriodicalId":9635,"journal":{"name":"Case Reports in Ophthalmology","volume":"15 1","pages":"250-255"},"PeriodicalIF":0.4,"publicationDate":"2024-03-22","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC10959544/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"140206304","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}

{"title":"Maxillary Ameloblastoma with Local Recurrence, Orbital Invasion, and Systemic Metastases: A Case Report and Review of the Literature.","authors":"Taylor J Linaburg, Javiera Araya, César A Briceño","doi":"10.1159/000537707","DOIUrl":"https://doi.org/10.1159/000537707","url":null,"abstract":"<p><strong>Introduction: </strong>Maxillary ameloblastoma is a rare, slow-growing odontogenic tumor that can recur after surgical excision, be locally aggressive, and rarely develop systemic metastases. We describe the course and management of a patient with recurrent maxillary ameloblastoma with orbital invasion and systemic metastases, the fourth case of its kind to be described in the literature.</p><p><strong>Case presentation: </strong>A 50-year-old female presented with left hyperglobus. A diagnosis of maxillary ameloblastoma was made based on biopsy and neuroimaging with MRI and CT. Surgical management included partial maxillectomy with orbital floor reconstruction, given the orbital invasion. Three years later, left hyperglobus recurred, and the patient was found to have orbital recurrence and lung metastases on PET imaging. The lung and orbital lesions have responded well to chemoradiation therapy without surgical intervention.</p><p><strong>Conclusion: </strong>Maxillary ameloblastoma is a rare tumor that typically arises from odontogenic tissues. Though considered benign, they can recur and in the case of our patient, metastasize. Complete surgical excision with wide surgical margins is associated with a shorter average time to recurrence and a lower incidence of metastasis. Cases of metastasis are managed with chemotherapy with or without adjuvant radiotherapy. Precision medicine may play a role in managing this entity in the future, given the discovery of differing profiles of maxillary ameloblastoma compared to mandibular. Ophthalmologists should be aware of this tumor as it can invade the orbit, resulting in significant ocular morbidity and mortality.</p>","PeriodicalId":9635,"journal":{"name":"Case Reports in Ophthalmology","volume":"15 1","pages":"238-245"},"PeriodicalIF":0.4,"publicationDate":"2024-03-19","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC10950358/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"140178670","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}

{"title":"Severe Hyperglycemia with Teprotumumab for Treatment of Thyroid Eye Disease.","authors":"Savannah Cottom, Brayden Barrientez, Andrew Melson","doi":"10.1159/000537872","DOIUrl":"https://doi.org/10.1159/000537872","url":null,"abstract":"<p><strong>Introduction: </strong>Thyroid eye disease (TED) is a rare condition involving autoimmune-mediated inflammation of the orbit and periocular structures, which can result in many debilitating symptoms. Teprotumumab, a monoclonal antibody that targets the insulin-like growth factor 1 receptor, is gaining popularity for the treatment of TED. In fact, owing to its efficacy and side effect profile, some recommend that it be considered as a first-line therapy for patients with TED. While teprotumumab is often chosen due to its efficacy and relatively favorable side effect profile compared to other treatments, there is a known risk of hyperglycemia with this mechanism of action, which is well described through clinical trials in the oncology literature. Though all cases in the clinical trial study of teprotumumab were mild, there is growing evidence that its effect on blood sugar can be more profound.</p><p><strong>Case presentation: </strong>We present a case of a well-controlled, recently diagnosed type 2 diabetic placed on teprotumumab for treatment of TED who developed life-threatening hyperglycemia. The case report provides evidence of hyperglycemic risk, as it highlights a patient's significant increase in hemoglobin A1C to 15.4 in addition to elevated serum glucose of 954 mg/dL while receiving teprotumumab.</p><p><strong>Conclusion: </strong>This case of severe hyperglycemia accentuates the need for more diligent, if not universal, glucose monitoring during teprotumumab treatment.</p>","PeriodicalId":9635,"journal":{"name":"Case Reports in Ophthalmology","volume":"15 1","pages":"246-249"},"PeriodicalIF":0.4,"publicationDate":"2024-03-19","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC10950356/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"140173832","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}

{"title":"From Hair Loss to Vision Loss: Minoxidil-Associated CRVO in a Young Female.","authors":"Lauren Pickel, Patrick Xiang Ji, Amr Abdelazim, Nirojini Sivachandran","doi":"10.1159/000537911","DOIUrl":"10.1159/000537911","url":null,"abstract":"<p><strong>Introduction: </strong>Central retinal vein occlusion (CRVO) is a common retinal vascular disorder that is most often seen in older adults and individuals with vascular risk factors.</p><p><strong>Case presentation: </strong>We report a case of CRVO with cystoid macular edema (CME) in a young, otherwise healthy patient taking minoxidil for hair loss. The patient had no known vascular risk factors, and a comprehensive coagulability workup was negative. The CRVO with CME resolved without intervention upon cessation of minoxidil.</p><p><strong>Conclusion: </strong>Possible mechanisms for minoxidil-associated retinal vascular disorders are explored. Thorough medication histories and the consideration of possible adverse drug events in patients without traditional risk factors are recommended.</p>","PeriodicalId":9635,"journal":{"name":"Case Reports in Ophthalmology","volume":"15 1","pages":"220-224"},"PeriodicalIF":0.5,"publicationDate":"2024-03-18","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC10948167/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"140157588","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}

{"title":"GNB1-Related Rod-Cone Dystrophy: A Case Report.","authors":"Giovanni Marco Conti, Francesca Cancellieri, Mathieu Quinodoz, Karolina Kaminska, Veronika Vaclavik, Carlo Rivolta, Hoai Viet Tran","doi":"10.1159/000537997","DOIUrl":"10.1159/000537997","url":null,"abstract":"<p><strong>Introduction: </strong>The <i>GNB1</i> (guanine nucleotide-binding protein, β1) gene encodes for the ubiquitous β1 subunit of heterotrimeric G proteins, which are associated with G-protein-coupled receptors (GPCRs). <i>GNB1</i> mutations cause a neurodevelopmental disorder characterized by a broad clinical spectrum. A novel variant has recently been confirmed in a case of rod-cone dystrophy.</p><p><strong>Case presentation: </strong>We describe the second confirmed case of a classical rod-cone dystrophy associated with a mutation located in exon 6 of <i>GNB1</i> [NM_002074.5:c.217G>C, p.(Ala73Pro)] in a 56-year-old patient also presenting mild intellectual disability, attention deficit/hyperactivity disorder, and truncal obesity.</p><p><strong>Conclusion: </strong>This paper confirms the role of <i>GNB1</i> in the pathogenesis of a classic rod-cone dystrophy and highlights the importance of including this gene in the genetic analysis panel for inherited retinal diseases.</p>","PeriodicalId":9635,"journal":{"name":"Case Reports in Ophthalmology","volume":"15 1","pages":"230-237"},"PeriodicalIF":0.4,"publicationDate":"2024-03-18","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC10948171/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"140157589","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}

{"title":"Melanocytic Nevus of the Superior Conjunctival Fornix: A Case Report.","authors":"Modupe O Adetunji, Thomas J Cummings, Miguel A Materin, Arpita S Maniar","doi":"10.1159/000537876","DOIUrl":"10.1159/000537876","url":null,"abstract":"<p><strong>Introduction: </strong>Conjunctival nevi are benign tumors that are commonly located at the nasal or temporal limbus and rarely in the fornix or tarsus. We report a case of a patient presenting with a solitary compound cystic nevus of the conjunctival fornix in the background of bilateral complexion-associated melanosis.</p><p><strong>Case presentation: </strong>A 71-year-old African-American female was referred for evaluation of an incidentally noted melanocytic lesion of the right conjunctival fornix. The patient underwent an excisional biopsy, revealing histological features consistent with a compound cystic nevus.</p><p><strong>Conclusion: </strong>This finding is noteworthy due to the rarity of conjunctival nevi originating in the fornix. The case underscores the importance of excisional biopsy in evaluating conjunctival forniceal melanocytic lesions to exclude malignant melanoma, a critical consideration for prognosis.</p>","PeriodicalId":9635,"journal":{"name":"Case Reports in Ophthalmology","volume":"15 1","pages":"225-229"},"PeriodicalIF":0.4,"publicationDate":"2024-03-18","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC10948165/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"140157590","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}

{"title":"Combining Surgery, Radiotherapy, and Topical Chemotherapy to Prevent Primary Orbital Exenteration for Atypical Caruncular Melanoma: A Case Report.","authors":"Cédric De Landsheer, Valentien Merlevede, Celine Jacobs, Jo Van Dorpe, Julie De Zaeytijd, Virginie G S Ninclaus, Dimitri Roels","doi":"10.1159/000536590","DOIUrl":"10.1159/000536590","url":null,"abstract":"<p><strong>Introduction: </strong>This case report demonstrates the possibility of successful eye and vision-sparing therapy for caruncular melanoma.</p><p><strong>Case presentation: </strong>We present an atypical presentation of a caruncular melanoma. After excisional biopsy, residual flat conjunctival melanosis resolved using topical chemotherapy (5-fluorouracil), which was well tolerated. Relapse of the melanoma was treated with external beam radiotherapy, but the tumor grew despite treatment. Eighteen months after complete excision of the relapsed melanoma, the patient remains tumor-free while the eye and its function remain preserved.</p><p><strong>Conclusion: </strong>This case report suggests that aggressive eye-sparing therapy for caruncular melanoma combining surgery, adjuvant topical chemotherapy, and external beam radiotherapy, can be an alternative for primary orbital exenteration.</p>","PeriodicalId":9635,"journal":{"name":"Case Reports in Ophthalmology","volume":"15 1","pages":"212-219"},"PeriodicalIF":0.4,"publicationDate":"2024-03-15","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC10942793/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"140142827","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}