Pub Date : 2024-05-31eCollection Date: 2024-01-01DOI: 10.1159/000539184
Elin Bohman, Rikard Linderoth, Sara Jonmarker Jaraj, Gustav Stålhammar, Eva Dafgård Kopp
Introduction: A known but uncommon complication following breast augmentation with silicone implants is the rupture of these implants and subsequent silicone migration through the lymphatic system. Exceptionally, there are sporadic instances of silicone granulomas forming in distant, non-lymphatic sites, posing diagnostic and management challenges in clinical practice.
Case presentation: A 56-year-old woman presented with slowly progressive diplopia and photosensitivity during the past 12 months. Ophthalmic examination revealed restriction of movement in all gazes in the right eye. Investigation with magnetic resonance imaging and positron emission tomography-computed tomography showed enlarged superior lateral and inferior rectus muscles in the right orbit, and a diffusely enlarged lacrimal gland in the left orbit, as well as a ruptured silicone breast implant on the right side. In addition, multiple enlarged lymph nodes were found throughout the body, as well as a mass in the internal oblique muscle of the abdominal wall. Fine-needle aspiration biopsy of the axillary lymph node and surgical biopsy of extraocular muscles confirmed a diagnosis of silicone granulomas. The patient received anti-inflammatory treatment with intravenous steroids but with no effect on symptoms.
Conclusions: This case illustrates a rare instance of silicone dissemination from a ruptured breast implant leading to granuloma formation in multiple organs, including the orbit. Notably, the spread of silicone appeared to occur through both lymphatic and hematogenous routes. This finding underscores the importance of considering silicone granulomas in the differential diagnosis of orbital lesions for patients with a history of silicone implants.
{"title":"Silicone Granulomas in the Orbit following Breast Implant Rupture: Case Report.","authors":"Elin Bohman, Rikard Linderoth, Sara Jonmarker Jaraj, Gustav Stålhammar, Eva Dafgård Kopp","doi":"10.1159/000539184","DOIUrl":"10.1159/000539184","url":null,"abstract":"<p><strong>Introduction: </strong>A known but uncommon complication following breast augmentation with silicone implants is the rupture of these implants and subsequent silicone migration through the lymphatic system. Exceptionally, there are sporadic instances of silicone granulomas forming in distant, non-lymphatic sites, posing diagnostic and management challenges in clinical practice.</p><p><strong>Case presentation: </strong>A 56-year-old woman presented with slowly progressive diplopia and photosensitivity during the past 12 months. Ophthalmic examination revealed restriction of movement in all gazes in the right eye. Investigation with magnetic resonance imaging and positron emission tomography-computed tomography showed enlarged superior lateral and inferior rectus muscles in the right orbit, and a diffusely enlarged lacrimal gland in the left orbit, as well as a ruptured silicone breast implant on the right side. In addition, multiple enlarged lymph nodes were found throughout the body, as well as a mass in the internal oblique muscle of the abdominal wall. Fine-needle aspiration biopsy of the axillary lymph node and surgical biopsy of extraocular muscles confirmed a diagnosis of silicone granulomas. The patient received anti-inflammatory treatment with intravenous steroids but with no effect on symptoms.</p><p><strong>Conclusions: </strong>This case illustrates a rare instance of silicone dissemination from a ruptured breast implant leading to granuloma formation in multiple organs, including the orbit. Notably, the spread of silicone appeared to occur through both lymphatic and hematogenous routes. This finding underscores the importance of considering silicone granulomas in the differential diagnosis of orbital lesions for patients with a history of silicone implants.</p>","PeriodicalId":9635,"journal":{"name":"Case Reports in Ophthalmology","volume":"15 1","pages":"465-471"},"PeriodicalIF":0.5,"publicationDate":"2024-05-31","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11250684/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"141626143","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}
Pub Date : 2024-05-30eCollection Date: 2024-01-01DOI: 10.1159/000539125
Daisuke Nakata, Hiroshi Okada, Koji Ueoka, Yoshiaki Shimada, Atsuhiro Tanikawa, Masayuki Horiguchi, Yasuki Ito
Introduction: We report a case of cerebral venous sinus thrombosis (CVST) that presented with bilateral optic disc swelling and diplopia in the absence of headaches.
Case presentation: A 54-year-old woman with no relevant medical history presented with a 2-week history of diplopia and no loss of visual acuity in each eye. Eye movements revealed bilateral abduction deficits, and fundoscopic examination revealed bilateral optic disc swelling. Non-contrast computed tomography of the head showed no abnormalities. Magnetic resonance venography revealed the absence of flow in the superior sagittal and left transverse sinuses as a consequence of thrombosis. The patient was diagnosed with intracranial hypertension associated with abducens nerve palsies secondary to CVST and was initiated on anticoagulant therapy. CVST can lead to stroke even in younger individuals.
Conclusion: CVST should be considered in differential diagnosis when bilateral papilledema and abducens nerve palsies are present, even in the absence of headache or other neurological findings.
{"title":"Cerebral Venous Sinus Thrombosis with Bilateral Optic Disc Swelling and Bilateral Sixth Nerve Palsies in the Absence of Headache: A Case Report.","authors":"Daisuke Nakata, Hiroshi Okada, Koji Ueoka, Yoshiaki Shimada, Atsuhiro Tanikawa, Masayuki Horiguchi, Yasuki Ito","doi":"10.1159/000539125","DOIUrl":"10.1159/000539125","url":null,"abstract":"<p><strong>Introduction: </strong>We report a case of cerebral venous sinus thrombosis (CVST) that presented with bilateral optic disc swelling and diplopia in the absence of headaches.</p><p><strong>Case presentation: </strong>A 54-year-old woman with no relevant medical history presented with a 2-week history of diplopia and no loss of visual acuity in each eye. Eye movements revealed bilateral abduction deficits, and fundoscopic examination revealed bilateral optic disc swelling. Non-contrast computed tomography of the head showed no abnormalities. Magnetic resonance venography revealed the absence of flow in the superior sagittal and left transverse sinuses as a consequence of thrombosis. The patient was diagnosed with intracranial hypertension associated with abducens nerve palsies secondary to CVST and was initiated on anticoagulant therapy. CVST can lead to stroke even in younger individuals.</p><p><strong>Conclusion: </strong>CVST should be considered in differential diagnosis when bilateral papilledema and abducens nerve palsies are present, even in the absence of headache or other neurological findings.</p>","PeriodicalId":9635,"journal":{"name":"Case Reports in Ophthalmology","volume":"15 1","pages":"459-464"},"PeriodicalIF":0.5,"publicationDate":"2024-05-30","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11249734/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"141626123","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}
Pub Date : 2024-05-24eCollection Date: 2024-01-01DOI: 10.1159/000538736
Munevver Duran, David Shieh, Preston Choi, Yu-Guang He, Rafael L Ufret-Vincenty
Introduction: We aimed to describe a clinical presentation of central serous retinopathy that poses a diagnostic and management dilemma.
Case presentation: A 30-year-old male patient presented with bilateral vision loss and multifocal serous retinal detachments involving the posterior pole of both eyes. Optical coherence tomography revealed prominent bilateral bacillary layer detachments. The patient complained of recent headaches and tinnitus. However, the clinical exam did not reveal overt inflammation and the patient admitted to being under significant stress. The clinical presentation raised concerns for both central serous retinopathy (CSR) and Vogt-Koyanagi-Harada (VKH). Additional findings, including white fundus spots and focal areas of retinal vascular leakage, were seen in our patient. We highlight these because, while they have been described in CSR, they are not commonly discussed and could add to the diagnostic dilemma. After a conservative approach that avoided steroids, our patient showed marked improvement over the following month, supporting a diagnosis of CSR.
Conclusion: CSR can mimic VKH disease. A high level of suspicion is needed to avoid instituting steroid therapy that could induce a severe iatrogenic exacerbation of the disease.
{"title":"Atypical Central Serous Chorioretinopathy Masquerading as Vogt-Koyanagi-Harada Disease: A Case Report.","authors":"Munevver Duran, David Shieh, Preston Choi, Yu-Guang He, Rafael L Ufret-Vincenty","doi":"10.1159/000538736","DOIUrl":"10.1159/000538736","url":null,"abstract":"<p><strong>Introduction: </strong>We aimed to describe a clinical presentation of central serous retinopathy that poses a diagnostic and management dilemma.</p><p><strong>Case presentation: </strong>A 30-year-old male patient presented with bilateral vision loss and multifocal serous retinal detachments involving the posterior pole of both eyes. Optical coherence tomography revealed prominent bilateral bacillary layer detachments. The patient complained of recent headaches and tinnitus. However, the clinical exam did not reveal overt inflammation and the patient admitted to being under significant stress. The clinical presentation raised concerns for both central serous retinopathy (CSR) and Vogt-Koyanagi-Harada (VKH). Additional findings, including white fundus spots and focal areas of retinal vascular leakage, were seen in our patient. We highlight these because, while they have been described in CSR, they are not commonly discussed and could add to the diagnostic dilemma. After a conservative approach that avoided steroids, our patient showed marked improvement over the following month, supporting a diagnosis of CSR.</p><p><strong>Conclusion: </strong>CSR can mimic VKH disease. A high level of suspicion is needed to avoid instituting steroid therapy that could induce a severe iatrogenic exacerbation of the disease.</p>","PeriodicalId":9635,"journal":{"name":"Case Reports in Ophthalmology","volume":"15 1","pages":"448-457"},"PeriodicalIF":0.5,"publicationDate":"2024-05-24","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11250661/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"141626122","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}
Introduction: Inflammatory juvenile conjunctival nevus (IJCN) is a rare condition affecting both children and adolescents. It has misleading clinical and histopathological features; therefore, careful assessment is necessary. We present a case of IJCN with a rare pathological type and misleading histopathological features.
Case presentation: A 13-year-old girl with IJCN in the right eye was treated with antiallergic and steroid eye drops but showed no response and was referred to our hospital for excisional biopsy. Slit-lamp examination revealed a nonpigmented juxtalimbal tumor in the right eye. Histopathologically, nevus cells with mild nuclear atypia proliferated within the conjunctival epithelium. Confluent growth of junctional nests, conjunctival cysts, and prominent inflammatory infiltration were also observed. Considering the young age of the patient and immunohistochemical characteristics (HMB-45, SOX10, p16 and Ki-67), the patient was finally diagnosed with IJCN. IJCN has three pathological subtypes - compound, subepithelial, and junctional - depending on the location of the nevus cells. This case was diagnosed as a rare junctional type, as most of the examined sections only showed lesions within the epithelium; no lesions were clearly identified extending beneath the epithelium.
Conclusion: The pathological diagnosis of IJCN is difficult because some features of IJCN suggest malignancy. Detailed microscopic examination, immunohistochemical staining, and the patient's young age helped render a final diagnosis.
{"title":"A Case of Inflammatory Juvenile Conjunctival Nevus with a Rare Histopathological Type.","authors":"Chihiro Iwata, Yuichi Asahina, Takashi Ono, Yukako Taketani, Mikiko Kimakura, Tetsuya Toyono, Mariko Tanaka, Makoto Aihara, Takashi Miyai","doi":"10.1159/000538593","DOIUrl":"10.1159/000538593","url":null,"abstract":"<p><strong>Introduction: </strong>Inflammatory juvenile conjunctival nevus (IJCN) is a rare condition affecting both children and adolescents. It has misleading clinical and histopathological features; therefore, careful assessment is necessary. We present a case of IJCN with a rare pathological type and misleading histopathological features.</p><p><strong>Case presentation: </strong>A 13-year-old girl with IJCN in the right eye was treated with antiallergic and steroid eye drops but showed no response and was referred to our hospital for excisional biopsy. Slit-lamp examination revealed a nonpigmented juxtalimbal tumor in the right eye. Histopathologically, nevus cells with mild nuclear atypia proliferated within the conjunctival epithelium. Confluent growth of junctional nests, conjunctival cysts, and prominent inflammatory infiltration were also observed. Considering the young age of the patient and immunohistochemical characteristics (HMB-45, SOX10, p16 and Ki-67), the patient was finally diagnosed with IJCN. IJCN has three pathological subtypes - compound, subepithelial, and junctional - depending on the location of the nevus cells. This case was diagnosed as a rare junctional type, as most of the examined sections only showed lesions within the epithelium; no lesions were clearly identified extending beneath the epithelium.</p><p><strong>Conclusion: </strong>The pathological diagnosis of IJCN is difficult because some features of IJCN suggest malignancy. Detailed microscopic examination, immunohistochemical staining, and the patient's young age helped render a final diagnosis.</p>","PeriodicalId":9635,"journal":{"name":"Case Reports in Ophthalmology","volume":"15 1","pages":"443-447"},"PeriodicalIF":0.5,"publicationDate":"2024-05-17","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11250155/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"141626120","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}
Introduction: Merkel cell carcinoma (MCC) is a rare neuroendocrine skin tumor associated with Merkel cell polyomavirus and ultraviolet light exposure. MCC typically affects older individuals, and it also influences young patients with immunosuppressive conditions. We report a case of lower eyelid MCC in a non-immunocompromised 37-year-old woman.
Case presentation: A 37-year-old woman presenting with suspected MCC on her right lower eyelid was referred to our hospital for further resection. The patient underwent wide excision with clear margins followed by reconstruction and radiation therapy. The patient has shown no signs of recurrence after 5 months of follow-up.
Conclusion: MCC needs to be considered as a possible diagnosis when examining an eyelid tumor in a young patient.
{"title":"Lower Eyelid Merkel Cell Carcinoma in a Non-Immunocompromised Young Female: A Case Report.","authors":"Yoshifumi Komatsu, Yoshiyuki Kitaguchi, Masako Kurashige, Takeshi Morimoto, Kohji Nishida","doi":"10.1159/000536098","DOIUrl":"10.1159/000536098","url":null,"abstract":"<p><strong>Introduction: </strong>Merkel cell carcinoma (MCC) is a rare neuroendocrine skin tumor associated with Merkel cell polyomavirus and ultraviolet light exposure. MCC typically affects older individuals, and it also influences young patients with immunosuppressive conditions. We report a case of lower eyelid MCC in a non-immunocompromised 37-year-old woman.</p><p><strong>Case presentation: </strong>A 37-year-old woman presenting with suspected MCC on her right lower eyelid was referred to our hospital for further resection. The patient underwent wide excision with clear margins followed by reconstruction and radiation therapy. The patient has shown no signs of recurrence after 5 months of follow-up.</p><p><strong>Conclusion: </strong>MCC needs to be considered as a possible diagnosis when examining an eyelid tumor in a young patient.</p>","PeriodicalId":9635,"journal":{"name":"Case Reports in Ophthalmology","volume":"15 1","pages":"437-442"},"PeriodicalIF":0.5,"publicationDate":"2024-05-17","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11250470/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"141626139","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}
Pub Date : 2024-05-08eCollection Date: 2024-01-01DOI: 10.1159/000538821
Amitouj S Sidhu, Charles E L Walker, Thomas D Riisfeldt, Peter J Tweedie, Natasha Gerbis, Emily J Sutherland, Neil G Simon, Lucy K Somerville, Ross Bradbury, Raymond Cook, Jonathon Parkinson, Robert Goetti, Ian C Francis
Introduction: Streptococcal meningoencephalitis (SME) is a rare, and frequently lethal, acute infection, and inflammation of the central nervous system parenchyma, with associated meningeal involvement. Bacterial meningoencephalitis is generally associated with high rates of morbidity and mortality, despite available antimicrobial and corticosteroid treatments. While Streptococcus pneumoniae is well recognised to cause bacterial meningitis, direct extension into the central nervous system parenchyma is rare.
Case presentation: A previously well 49-year-old man presented with sudden onset severe headache, fevers, neck stiffness, and reduced consciousness. The manifestations of SME in this patient were bilateral pupil-involving third-nerve palsies, wall-eyed bilateral internuclear ophthalmoplegia (WEBINO), bilateral blindness, bilateral deafness, a right lower motor neuron facial palsy, and upper motor neuron signs in his limbs. Initially, a partial response to high dose intravenous antibiotics occurred, but with administration of intravenous corticosteroids, further substantial resolution of the patient's neurological and neuro-ophthalmological deficits occurred.
Conclusion: This case highlights the benefit of multidisciplinary diagnostic and therapeutic interventions in a case of SME complicated by bilateral pupil-involving third-nerve palsies, WEBINO, bilateral blindness, bilateral deafness, a right lower motor neuron facial palsy, and upper motor neuron signs. It appears to be the first reported case of SME with this rare collection of neuro-ophthalmological abnormalities.
导言:链球菌脑膜脑炎(SME)是一种罕见的、经常致命的急性感染,是中枢神经系统实质的炎症,伴有脑膜受累。尽管可以使用抗菌药和皮质类固醇治疗,但细菌性脑膜脑炎的发病率和死亡率通常都很高。虽然肺炎链球菌是公认的细菌性脑膜炎的致病菌,但直接扩展到中枢神经系统实质的情况却很少见:病例介绍:一名 49 岁的男性患者因突发剧烈头痛、发热、颈部僵硬和意识减退而就诊。该患者的 SME 表现为双侧瞳孔-第三神经节麻痹、壁眼双侧核间性眼肌麻痹(WEBINO)、双侧失明、双侧耳聋、右侧下运动神经元面瘫和四肢上运动神经元体征。最初,患者对大剂量静脉注射抗生素产生了部分反应,但随着皮质类固醇的静脉注射,患者的神经和神经眼科功能障碍得到了进一步的实质性缓解:本病例强调了多学科诊断和治疗干预对并发双侧瞳孔牵涉第三神经麻痹、WEBINO、双侧失明、双侧失聪、右侧下运动神经元面瘫和上运动神经元体征的SME病例的益处。这似乎是首例报告的 SME 合并这种罕见的神经眼科异常的病例。
{"title":"Successful Management of Acute Streptococcal Meningoencephalitis Complicated by Bilateral Third-Nerve Palsies, Wall-Eyed Bilateral Internuclear Ophthalmoplegia, Blindness, and Deafness: Case Report.","authors":"Amitouj S Sidhu, Charles E L Walker, Thomas D Riisfeldt, Peter J Tweedie, Natasha Gerbis, Emily J Sutherland, Neil G Simon, Lucy K Somerville, Ross Bradbury, Raymond Cook, Jonathon Parkinson, Robert Goetti, Ian C Francis","doi":"10.1159/000538821","DOIUrl":"10.1159/000538821","url":null,"abstract":"<p><strong>Introduction: </strong>Streptococcal meningoencephalitis (SME) is a rare, and frequently lethal, acute infection, and inflammation of the central nervous system parenchyma, with associated meningeal involvement. Bacterial meningoencephalitis is generally associated with high rates of morbidity and mortality, despite available antimicrobial and corticosteroid treatments. While <i>Streptococcus pneumoniae</i> is well recognised to cause bacterial meningitis, direct extension into the central nervous system parenchyma is rare.</p><p><strong>Case presentation: </strong>A previously well 49-year-old man presented with sudden onset severe headache, fevers, neck stiffness, and reduced consciousness. The manifestations of SME in this patient were bilateral pupil-involving third-nerve palsies, wall-eyed bilateral internuclear ophthalmoplegia (WEBINO), bilateral blindness, bilateral deafness, a right lower motor neuron facial palsy, and upper motor neuron signs in his limbs. Initially, a partial response to high dose intravenous antibiotics occurred, but with administration of intravenous corticosteroids, further substantial resolution of the patient's neurological and neuro-ophthalmological deficits occurred.</p><p><strong>Conclusion: </strong>This case highlights the benefit of multidisciplinary diagnostic and therapeutic interventions in a case of SME complicated by bilateral pupil-involving third-nerve palsies, WEBINO, bilateral blindness, bilateral deafness, a right lower motor neuron facial palsy, and upper motor neuron signs. It appears to be the first reported case of SME with this rare collection of neuro-ophthalmological abnormalities.</p>","PeriodicalId":9635,"journal":{"name":"Case Reports in Ophthalmology","volume":"15 1","pages":"423-429"},"PeriodicalIF":0.4,"publicationDate":"2024-05-08","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11078538/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"140891454","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}
Pub Date : 2024-05-08eCollection Date: 2024-01-01DOI: 10.1159/000538824
Zachary George Angus, Penelope McKelvie, Thomas G Hardy
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.
{"title":"Epibulbar Complex Choristoma Containing Bone: A Case Report and Closer Look at Classifications.","authors":"Zachary George Angus, Penelope McKelvie, Thomas G Hardy","doi":"10.1159/000538824","DOIUrl":"10.1159/000538824","url":null,"abstract":"<p><strong>Introduction: </strong>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.</p><p><strong>Case presentation: </strong>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.</p><p><strong>Conclusion: </strong>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.</p>","PeriodicalId":9635,"journal":{"name":"Case Reports in Ophthalmology","volume":"15 1","pages":"418-422"},"PeriodicalIF":0.4,"publicationDate":"2024-05-08","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11078539/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"140891447","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}
Pub Date : 2024-04-30eCollection Date: 2024-01-01DOI: 10.1159/000535948
Alex Y Pai, Michael M Han, Sameh Mosaed, Mohammad Riazi Esfahani
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.
{"title":"Overlapping Spots of Photodynamic Therapy for Treatment of Choroidal Hemangioma in Sturge-Weber Syndrome: A Case Report.","authors":"Alex Y Pai, Michael M Han, Sameh Mosaed, Mohammad Riazi Esfahani","doi":"10.1159/000535948","DOIUrl":"https://doi.org/10.1159/000535948","url":null,"abstract":"<p><strong>Introduction: </strong>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.</p><p><strong>Case presentation: </strong>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.</p><p><strong>Conclusion: </strong>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.</p>","PeriodicalId":9635,"journal":{"name":"Case Reports in Ophthalmology","volume":"15 1","pages":"411-417"},"PeriodicalIF":0.4,"publicationDate":"2024-04-30","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11060702/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"140847413","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}
Pub Date : 2024-04-26eCollection Date: 2024-01-01DOI: 10.1159/000538547
Andreas Katsimpris, Manjula Kumarasamy
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.
{"title":"Bilateral Late-Onset Pigment Dispersion Syndrome following Implantable Collamer Lens Surgery: A Case Report.","authors":"Andreas Katsimpris, Manjula Kumarasamy","doi":"10.1159/000538547","DOIUrl":"https://doi.org/10.1159/000538547","url":null,"abstract":"<p><strong>Introduction: </strong>We report a case of bilateral pigment dispersion syndrome after 13 years of uncomplicated implantable collamer lens (ICL) surgery.</p><p><strong>Case presentation: </strong>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.</p><p><strong>Conclusion: </strong>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.</p>","PeriodicalId":9635,"journal":{"name":"Case Reports in Ophthalmology","volume":"15 1","pages":"399-405"},"PeriodicalIF":0.4,"publicationDate":"2024-04-26","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11052560/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"140856039","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}