{"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}
João Ponces Ramalhão, Diogo Rodrigues, Beatriz Vieira, Miguel Gonçalves Afonso, Pedro Manuel Baptista, Nuno Jorge Lamas, Maria Araújo
{"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}
Savannah Cottom, Brayden Barrientez, Andrew Melson
{"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}
Lauren Pickel, Patrick Xiang Ji, Amr Abdelazim, Nirojini Sivachandran
{"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}
Giovanni Marco Conti, Francesca Cancellieri, Mathieu Quinodoz, Karolina Kaminska, Veronika Vaclavik, Carlo Rivolta, Hoai Viet Tran
{"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}
Modupe O Adetunji, Thomas J Cummings, Miguel A Materin, Arpita S Maniar
{"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}
Cédric De Landsheer, Valentien Merlevede, Celine Jacobs, Jo Van Dorpe, Julie De Zaeytijd, Virginie G S Ninclaus, Dimitri Roels
{"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}
Leire Olazaran, Ana Jiménez, Pablo González de Los Mártires, Gonzalo Guerrero, Nerea Gangoitia, Iñigo Salmeron, Ane Galarza, Ana Sofía Argüelles, Beatriz Elso, Irene Reyzabal, Esther Compains, Henar Heras, Santiago López
{"title":"White Dot Syndromes: Report of Three Cases.","authors":"Leire Olazaran, Ana Jiménez, Pablo González de Los Mártires, Gonzalo Guerrero, Nerea Gangoitia, Iñigo Salmeron, Ane Galarza, Ana Sofía Argüelles, Beatriz Elso, Irene Reyzabal, Esther Compains, Henar Heras, Santiago López","doi":"10.1159/000536336","DOIUrl":"10.1159/000536336","url":null,"abstract":"<p><strong>Introduction: </strong>The term white dot syndromes has been used to refer conditions that differ in their morphology and prognosis. We report three cases of different pathologies encompassed within the white dot syndromes.</p><p><strong>Case presentations: </strong>Case 1: A 26-year-old female presented with scotoma in her right eye. Fundus examination revealed multiple white dots that demonstrated early hyperfluorescence with late staining on FA. OCT showed discontinuities in inner segment-outer segment junction associated with columnar-shaped outer retinal hyperreflective bands. AF revealed multiple hyperautofluorescent dots around the posterior pole, compatible with multiple evanescent white dot syndrome. The symptoms improved without treatment. Case 2: A 16-year-old male presented with retinal lesions compatible with punctate inner choroidopathy in his right eye. OCT showed lesion in the outer retinal layer. FAF revealed parafoveal hypoautofluorescent dots with early hyperfluorescence and late staining on FFA. After oral corticotherapy, they progress to atrophic scars. Case 3: A 65-year-old male presented with scotoma and decreased vision in his right eye. OCT showed hyperreflectivity in the outer layer that progresses to a large atrophic plaque with foveal affectation. FAF demonstrated hyperautofluorescent placoid lesion occupying macular area, compatible with acute posterior multifocal placoid pigment epitheliopathy. Retinal lesions improved with systemic corticosteroids.</p><p><strong>Conclusion: </strong>The FAF pattern helps know the distribution of the lesions. It represents a noninvasive method that has been shown to be useful in the diagnosis and monitoring of white dot syndromes.</p>","PeriodicalId":9635,"journal":{"name":"Case Reports in Ophthalmology","volume":"15 1","pages":"202-211"},"PeriodicalIF":0.4,"publicationDate":"2024-03-14","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC10939511/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"140130801","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":"Capsular Block Syndrome after an Intravitreal Injection of Ranibizumab: A Case Report.","authors":"Jae Suk Kim","doi":"10.1159/000537755","DOIUrl":"10.1159/000537755","url":null,"abstract":"<p><strong>Introduction: </strong>We present a case of capsular block syndrome that occurred after intravitreal injection of ranibizumab in a patient with age-related macular degeneration, which has not been reported in the literature.</p><p><strong>Case presentation: </strong>A 78-year-old male presented with decreased visual acuity in the right eye. Slit-lamp examination findings were unremarkable; however, AMD was diagnosed based on fundus examination, fluorescein angiography, and optical coherence tomography (OCT). Subsequently, the patient was administered an intravitreal injection of ranibizumab. A slit-lamp examination revealed residual cortical material, numerous inflammatory cells, and posterior capsular distension 1 week after the injection. OCT showed an adhesion of the intraocular lens to the continuous curvilinear capsulorhexis site. The patient's vision improved following Nd:YAG laser posterior capsulotomy.</p><p><strong>Conclusion: </strong>Meticulous cortical removal is crucial during phacoemulsification to prevent capsular block syndrome. In patients with a history of cataract surgery, verifying the absence of residual cortical material before administering an intravitreal injection of ranibizumab is important.</p>","PeriodicalId":9635,"journal":{"name":"Case Reports in Ophthalmology","volume":"15 1","pages":"196-201"},"PeriodicalIF":0.4,"publicationDate":"2024-03-12","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC10932552/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"140109293","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}