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Enhancing Visual Acuity through Combined Intraocular Lens Fixation, Penetrating Keratoplasty, and Vitrectomy: The Role of Temporary Landers Wide-Field Keratoprosthesis. 通过联合固定眼内透镜、穿透性角膜移植术和玻璃体切除术提高视力:临时性 Landers 宽视野角膜假体的作用。
IF 0.5 Q4 OPHTHALMOLOGY Pub Date : 2024-08-13 eCollection Date: 2024-01-01 DOI: 10.1159/000540287
Takahiro Zusho, Takashi Ono, Yukako Taketani, Mikiko Kimakura, Tetsuya Toyono, Koichiro Sugimoto, Taku Toyama, Takashi Ueta, Makoto Aihara, Takashi Miyai

Introduction: Complex corneal conditions present surgical challenges and necessitate innovation. Here, we present two cases where we performed intraocular lens trans-scleral fixation using the double-needle Yamane technique, followed by penetrating keratoplasty and vitrectomy using a temporary Landers wide-field keratoprosthesis.

Case presentation: Case 1 involved a 70-year-old man with an aphakic eye of bullous keratopathy and corneal opacity owing to multiple penetrating and endothelial keratoplasty, endophthalmitis, and herpetic keratitis. His visual acuity was counting fingers at 20 cm before surgery. Penetrating keratoplasty with vitrectomy and intraocular lens scleral fixation was performed using the double-needle Yamane technique, and 10 months postoperatively, his best-corrected visual acuity improved to 0.6, presenting a clear cornea. Case 2 involved a 62-year-old man who underwent penetrating keratoplasty twice for corneal perforation and therapeutic penetrating keratoplasty with vitrectomy for traumatic globe rupture, resulting in the loss of the intraocular lens. The patient exhibited graft failure, and his best-corrected visual acuity was 0.03. Utilizing a temporary Landers wide-field keratoprosthesis, we performed penetrating keratoplasty and intraocular lens trans-scleral fixation without complications. His final best-corrected visual acuity improved to 0.15 with a clear cornea.

Conclusions: Trans-scleral fixation of intraocular lens with penetrating keratoplasty, using temporary Landers wide-field keratoprosthesis, yielded positive clinical outcomes without serious complications.

导言:复杂的角膜病症给手术带来了挑战,因此有必要进行创新。在这里,我们介绍了两例使用双针 Yamane 技术进行眼内晶状体经巩膜固定,然后使用临时 Landers 宽视野角膜假体进行穿透性角膜成形术和玻璃体切除术的病例:病例 1 患者是一名 70 岁的男性,因多次穿透性和内皮性角膜移植术、眼底病和疱疹性角膜炎而导致牛皮状角膜病变和角膜混浊。手术前,他的视力为 20 厘米数指。术后 10 个月,他的最佳矫正视力提高到 0.6,角膜清晰。病例 2 涉及一名 62 岁的男性,他因角膜穿孔接受了两次穿透性角膜移植术,又因外伤性眼球破裂导致眼内晶状体缺失而接受了治疗性穿透性角膜移植术和玻璃体切除术。患者出现移植失败,最佳矫正视力为 0.03。我们利用一个临时的 Landers 宽视野角膜假体,进行了穿透性角膜成形术和眼内晶状体经巩膜固定术,没有出现并发症。他的最终最佳矫正视力提高到了 0.15,角膜清晰:结论:使用临时性 Landers 宽视野角膜假体进行穿透性角膜移植手术的眼内晶状体经巩膜固定术取得了良好的临床效果,未出现严重并发症。
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引用次数: 0
Cat Scratch Disease Presenting with Right Branch Retinal Artery Occlusion and Left Neuroretinitis. 猫抓病伴有右视网膜分支动脉闭塞和左侧神经视网膜炎。
IF 0.5 Q4 OPHTHALMOLOGY Pub Date : 2024-08-12 eCollection Date: 2024-01-01 DOI: 10.1159/000540125
Samira Jafari, Jonathan A Micieli

Introduction: Bartonella henselae, the causative agent of cat scratch disease (CSD), presents with diverse ocular manifestations, posing diagnostic challenges. This study aimed to elucidate the diagnostic complexities through a unique case.

Case presentation: A 42-year-old male presented with vision loss in the right eye, subsequent to flu-like symptoms following exposure to a stray kitten. Clinical examination revealed branch retinal artery occlusion (BRAO) in the right eye and neuroretinitis in the left, indicating concurrent ocular manifestations of CSD. Thorough investigations, including serological testing, ruled out alternative causes, highlighting the rarity of such coexisting ocular complications.

Conclusions: The coexistence of BRAO and neuroretinitis in different eyes underscores the variable presentation of CSD. Recognition of infectious etiologies, particularly Bartonella, is paramount in diagnosing ocular vasculopathies. This case emphasizes the importance of considering Bartonella infection in patients with ocular vascular occlusions, especially in the context of recent cat exposure and systemic symptoms suggestive of CSD.

导言:河南巴顿氏菌是猫抓病(CSD)的致病菌,其眼部表现多种多样,给诊断带来了挑战。本研究旨在通过一个独特的病例来阐明诊断的复杂性:一名 42 岁的男性因接触一只流浪小猫而出现流感样症状,随后右眼视力下降。临床检查发现右眼视网膜动脉分支闭塞(BRAO),左眼神经视网膜炎,表明同时存在 CSD 的眼部表现。包括血清学检测在内的全面检查排除了其他病因,突显了这种并存的眼部并发症的罕见性:结论:BRAO和神经视网膜炎在不同的眼睛中同时存在,这凸显了CSD表现的多变性。识别感染性病因,尤其是巴顿氏菌,对于诊断眼部血管疾病至关重要。本病例强调了在眼部血管闭塞患者中考虑巴顿氏菌感染的重要性,尤其是在近期接触过猫且全身症状提示 CSD 的情况下。
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引用次数: 0
Bilateral Endogenous Bacterial Endophthalmitis with Asynchrony for 14 Months due to Klebsiella pneumoniae: A Case Report. 肺炎克雷伯氏菌导致双侧内源性细菌性眼内炎,14 个月不同步:病例报告。
IF 0.5 Q4 OPHTHALMOLOGY Pub Date : 2024-08-12 eCollection Date: 2024-01-01 DOI: 10.1159/000540471
Pedro Javier Nuova

Introduction: The aim of this study was to describe a very rare case of endogenous bacterial endophthalmitis caused by Klebsiella pneumoniae in both eyes with a difference in the onset of symptoms of 14 months in an immunocompetent patient.

Case presentation: A 66-year-old immunocompetent man presented with asynchronous bilateral endogenous endophthalmitis produced by the K. pneumoniae bacterium at the starting point of a liver abscess after cholecystectomy surgery, causing endophthalmitis 1 year and 2 months apart between an eye and another. The first was diffuse anteroposterior endophthalmitis in the left eye that ended in visual loss and phthisis bulbi due to delayed initial diagnosis and established treatment, and the second was focal endophthalmitis in the right eye that preserved the organ and resulted in a vision of 20/20 due to early suspected diagnosis and rapid instituted treatment.

Conclusion: To our knowledge, this is the first published case of a long asynchronous bilateral endogenous bacterial endophthalmitis caused by K. pneumoniae with a prolonged difference of 14 months in the onset of symptoms between one eye and another. This case is a vision-threatening ophthalmologic emergency that can be associated with life-threatening systemic morbidities. The early diagnosis of infection represents a challenge for clinicians, ophthalmologists, and microbiologists.

导言:本研究旨在描述一例非常罕见的由肺炎克雷伯菌引起的双眼内源性细菌性眼内炎病例,该病例的免疫功能正常患者的发病时间相差 14 个月:一名66岁的免疫功能正常男子在胆囊切除手术后出现肝脓肿,在肝脓肿的起始点,肺炎克雷伯菌引起了不同步的双侧内源性眼内炎,造成双眼眼内炎的时间相差1年和2个月。第一例是左眼弥漫性前后眼内炎,由于延误了初步诊断和既定治疗,最终导致视力丧失和球虫病;第二例是右眼局灶性眼内炎,由于早期疑似诊断和迅速治疗,保住了器官,视力达到 20/20:据我们所知,这是第一例公开发表的由肺炎双球菌引起的长期不同步双侧内源性细菌性眼内炎病例,其中一只眼睛和另一只眼睛的发病时间相差14个月之久。该病例是一种危及视力的眼科急症,可能伴有危及生命的全身性疾病。感染的早期诊断是临床医生、眼科医生和微生物学家面临的一项挑战。
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引用次数: 0
Primary Vitreoretinal Lymphoma Presenting Solely with Asymptomatic Peripheral Drusenoid Lesions. 原发性玻璃体视网膜淋巴瘤仅表现为无症状的周边类色素病变
IF 0.5 Q4 OPHTHALMOLOGY Pub Date : 2024-08-08 eCollection Date: 2024-01-01 DOI: 10.1159/000540051
Brianna Lu, Jovi C Y Wong, Justin Kritzinger, David T Wong

Introduction: Primary vitreoretinal lymphoma (PVRL) is a rare malignant tumor that typically involves the retina, vitreous, or optic nerve head. PVRL often occurs concurrently with central nervous system lymphoma. Here, we present the first report of a patient with biopsy-confirmed PVRL presenting solely with asymptomatic peripheral drusenoid lesions.

Case presentation: A woman in her 70s presented with new elevated amelanotic yellow lesions with overlying pigment in both of her eyes not previously seen prior to cataract surgery. Over the next 4 months, there was waxing and waning of lesions which resolved and first appeared in the right eye and then the left. A diagnostic vitrectomy of the left eye revealed B-cell lymphoma. The patient elected for initial treatment with radiation therapy of both orbits. A new lesion was identified in her right eye nearly 18 months after starting maintenance therapy with ibrutinib, following which systemic chemotherapy with methotrexate was initiated.

Conclusion: Elevated clinical suspicion for a malignant process is needed for patients with progressive new retinal lesions in older age. Local radiation therapy to the orbits alone may not be sufficient to prevent progression despite initial presentation showing confinement of disease to the intraocular space.

简介:原发性玻璃体视网膜淋巴瘤(PVRL原发性玻璃体视网膜淋巴瘤(PVRL)是一种罕见的恶性肿瘤,通常累及视网膜、玻璃体或视神经头。原发性玻璃体视网膜淋巴瘤通常与中枢神经系统淋巴瘤同时发生。在此,我们首次报告了一名经活检确诊的 PVRL 患者,该患者仅表现为无症状的外周性类风湿性病变:一位 70 多岁的妇女在接受白内障手术之前,双眼出现了新的高出皮肤的黄色病变,并伴有色素沉着。在接下来的 4 个月中,病变逐渐消退,先出现在右眼,然后是左眼。左眼诊断性玻璃体切除术发现了 B 细胞淋巴瘤。患者选择对两个眼眶进行放射治疗。在开始接受伊布替尼维持治疗近18个月后,她的右眼发现了新的病变,随后开始接受甲氨蝶呤全身化疗:结论:对于老年视网膜进行性新病变患者,临床上需要高度怀疑恶性病变。结论:对于老年视网膜新病变进展期患者,临床上需要提高对恶性病变的怀疑。尽管最初的症状显示病变局限于眼内间隙,但仅对眼眶进行局部放射治疗可能不足以防止病变进展。
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引用次数: 0
A Temporal Association between Regression of Pachydrusen and Use of Proprotein Convertase Subtilisin Kexin 9 Inhibitor: A Case Report. 柏氏症的消退与使用蛋白转化酶亚基酶克毒素 9 抑制剂之间的时间关联:病例报告。
IF 0.5 Q4 OPHTHALMOLOGY Pub Date : 2024-08-08 eCollection Date: 2024-01-01 DOI: 10.1159/000540014
Yodpong Chantarasorn, Kriengsak Funilkul

Introduction: We aim to report the clinical course of a patient with pachychoroidopathy who experienced regression of subfoveal drusen during cholesterol treatment using PCSK9 inhibitors.

Case presentation: A 62-year-old woman who was visually asymptomatic complained of recent visual loss in the left eye (OS). She was diagnosed with foveal pachydrusen (OS) that had remained stable for 10 years. Three months after starting cholesterol treatment with a PCSK9 inhibitor, the latest class of lipid-lowering medication, her vision improved in parallel with gradual regression of material deposited beneath the retinal pigment epithelium (RPE). Recurrence of drusen was observed after discontinuing the drug.

Conclusions: Use of PCSK9 inhibitors may improve the retina's lipid homeostasis by increasing the number of RPE-LDL receptors and partly contribute to the improvement of ocular phenotypes associated with dysfunctional RPE in pachychoroidopathy.

导言:我们旨在报告一名患有脉络膜病变的患者在使用 PCSK9 抑制剂治疗胆固醇期间眼底色素沉着消退的临床过程:一名 62 岁的女性患者无任何视力症状,主诉最近左眼(OS)视力下降。她被诊断出患有眼窝积水症(OS),10 年来一直保持稳定。在开始使用最新一类降脂药物 PCSK9 抑制剂治疗胆固醇三个月后,她的视力随着视网膜色素上皮(RPE)下沉积物的逐渐消退而得到改善。停药后观察到色素沉着复发:结论:使用 PCSK9 抑制剂可通过增加 RPE-LDL 受体的数量来改善视网膜的脂质平衡,并在一定程度上有助于改善与脉络膜睫状体病变中 RPE 功能障碍相关的眼部表型。
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引用次数: 0
Dysthyroid Optic Neuropathy Complicated by Neuromyelitis Optica Spectrum Disorder: A Case Report. 甲状腺功能减退症视神经病变并发神经脊髓炎谱系障碍:病例报告。
IF 0.5 Q4 OPHTHALMOLOGY Pub Date : 2024-08-08 eCollection Date: 2024-01-01 DOI: 10.1159/000540496
Teruaki Tokuhisa, Takaaki Hayashi, Shusaku Omoto, Ai Kozaki, Toshu Inoue, Tadashi Nakano

Introduction: Neuromyelitis optica spectrum disorder (NMOSD) is an autoimmune disorder that principally targets the central nervous system, specifically the spinal cord and optic nerves. NMOSD is often associated with thyroid pathologies such as Graves' disease or Hashimoto's thyroiditis. Thyroid eye disease (TED) is an autoimmune condition characterized by inflammation and hypertrophy of the extraocular muscles. Dysthyroid optic neuropathy (DON), a critical complication of TED, may lead to irreversible visual loss. We report a case of DON complicated by NMOSD.

Case presentation: We report a case of an autoimmune disease presenting as DON in a 44-year-old Japanese woman with a history of Graves' disease, who experienced reduced visual acuity and orbital pain. Brain magnetic resonance imaging disclosed hypertrophy of the rectus muscles, compressing the optic nerve bilaterally. Consequently, she was diagnosed with DON and underwent three courses of steroid semi-pulse therapy and left orbital decompression surgery, alleviating optic nerve compression. Nevertheless, the visual prognosis remained poor. A subsequent serological test showed positive for aquaporin-4 antibody. Treatment with satralizumab, an interleukin-6 receptor monoclonal antibody, was initiated in conjunction with steroids to suppress the autoimmune response and reduce NMOSD relapse risk. Following this treatment, no NMOSD recurrences were reported.

Conclusion: This case highlights the necessity of considering the possible coexistence of DON and NMOSD in patients with autoimmune diseases.

简介神经脊髓炎视神经谱系障碍(NMOSD)是一种自身免疫性疾病,主要针对中枢神经系统,特别是脊髓和视神经。NMOSD 通常与甲状腺病变有关,如巴塞杜氏病或桥本氏甲状腺炎。甲状腺眼病(TED)是一种自身免疫性疾病,其特征是眼外肌发炎和肥大。甲状腺功能减退性视神经病变(DON)是TED的一种重要并发症,可能导致不可逆的视力丧失。我们报告了一例并发 NMOSD 的 DON 病例:我们报告了一例表现为 DON 的自身免疫性疾病,患者是一名 44 岁的日本女性,曾患巴塞杜氏病,视力下降,眼眶疼痛。脑磁共振成像显示她的直肌肥大,压迫了双侧视神经。因此,她被诊断为 DON,并接受了三个疗程的类固醇半脉冲治疗和左眼眶减压手术,缓解了视神经压迫症状。然而,视力预后仍然不佳。随后的血清学检测显示,aquaporin-4 抗体呈阳性。开始使用白细胞介素-6受体单克隆抗体satralizumab与类固醇联合治疗,以抑制自身免疫反应,降低NMOSD复发风险。治疗后,未再报告NMOSD复发:本病例强调了考虑自身免疫性疾病患者可能同时患有 DON 和 NMOSD 的必要性。
{"title":"Dysthyroid Optic Neuropathy Complicated by Neuromyelitis Optica Spectrum Disorder: A Case Report.","authors":"Teruaki Tokuhisa, Takaaki Hayashi, Shusaku Omoto, Ai Kozaki, Toshu Inoue, Tadashi Nakano","doi":"10.1159/000540496","DOIUrl":"10.1159/000540496","url":null,"abstract":"<p><strong>Introduction: </strong>Neuromyelitis optica spectrum disorder (NMOSD) is an autoimmune disorder that principally targets the central nervous system, specifically the spinal cord and optic nerves. NMOSD is often associated with thyroid pathologies such as Graves' disease or Hashimoto's thyroiditis. Thyroid eye disease (TED) is an autoimmune condition characterized by inflammation and hypertrophy of the extraocular muscles. Dysthyroid optic neuropathy (DON), a critical complication of TED, may lead to irreversible visual loss. We report a case of DON complicated by NMOSD.</p><p><strong>Case presentation: </strong>We report a case of an autoimmune disease presenting as DON in a 44-year-old Japanese woman with a history of Graves' disease, who experienced reduced visual acuity and orbital pain. Brain magnetic resonance imaging disclosed hypertrophy of the rectus muscles, compressing the optic nerve bilaterally. Consequently, she was diagnosed with DON and underwent three courses of steroid semi-pulse therapy and left orbital decompression surgery, alleviating optic nerve compression. Nevertheless, the visual prognosis remained poor. A subsequent serological test showed positive for aquaporin-4 antibody. Treatment with satralizumab, an interleukin-6 receptor monoclonal antibody, was initiated in conjunction with steroids to suppress the autoimmune response and reduce NMOSD relapse risk. Following this treatment, no NMOSD recurrences were reported.</p><p><strong>Conclusion: </strong>This case highlights the necessity of considering the possible coexistence of DON and NMOSD in patients with autoimmune diseases.</p>","PeriodicalId":9635,"journal":{"name":"Case Reports in Ophthalmology","volume":null,"pages":null},"PeriodicalIF":0.5,"publicationDate":"2024-08-08","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11324271/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"141981804","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}
引用次数: 0
The "Ballerina" Sign: Posterior Capsular Rupture during Cataract Surgery. 芭蕾舞演员 "征兆:白内障手术中的后囊破裂。
IF 0.5 Q4 OPHTHALMOLOGY Pub Date : 2024-08-07 eCollection Date: 2024-01-01 DOI: 10.1159/000538439
Dan Ramon, Nadav Shemesh, Asaf Achiron

Introduction: In this case study, we present the "Ballerina" sign as a potentially valuable clinical indicator for detecting posterior capsular rupture (PCR) during cataract surgery. The purpose of this study is to highlight the significance of PCR in the context of cataract surgery and introduce this novel sign.

Case presentation: During the cataract operation on a 70-year-old patient, we observed a spiraling fragment of the nucleus attached to a vitreous string. Subsequently, an anterior vitrectomy was successfully performed without further intraoperative complications, followed by the insertion of a 3-piece lens into the sulcus. Positive visual outcomes were observed during postoperative follow-up appointments at 1 day, 2 weeks, and 3 months.

Conclusion: The documentation of the "Ballerina" sign in the medical literature is innovative, offering ophthalmic surgeons a valuable tool for early PCR detection during cataract surgeries.

导言:在本病例研究中,我们介绍了 "芭蕾舞女 "体征,它是在白内障手术中检测后囊破裂(PCR)的一个潜在的有价值的临床指标。本研究的目的是强调 PCR 在白内障手术中的重要性,并介绍这一新型体征:在为一名 70 岁的患者进行白内障手术时,我们观察到一个螺旋状的晶体核碎片附着在玻璃体串上。随后,我们成功进行了前部玻璃体切除术,术中未出现其他并发症,随后在沟内植入了一个三片式晶状体。在术后 1 天、2 周和 3 个月的随访中,观察到了积极的视觉效果:在医学文献中记录 "芭蕾舞女 "征象是一项创新,为眼科医生在白内障手术中早期发现 PCR 提供了宝贵的工具。
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引用次数: 0
Infected Inclusion Cyst of a Conjunctival Nevus Treated with a Mini-Incision: A Case Report. 结膜痣感染性包涵囊肿的小切口治疗:病例报告
IF 0.5 Q4 OPHTHALMOLOGY Pub Date : 2024-08-05 eCollection Date: 2024-01-01 DOI: 10.1159/000539846
Emmanouil Blavakis, Mateusz Kecik, Gabriele Thumann, Horace Massa

Introduction: Conjunctival cysts are usually asymptomatic but they can cause foreign body sensation and contribute to dry eye disease. The purpose of this case report is to describe the presentation and treatment of an infected inclusion cyst of a conjunctival nevus in a healthy 36-year-old patient.

Case presentation: A healthy 36-year-old man presented to the emergency department for redness and pain in his left eye for 1 day. Slit-lamp examination revealed a conjunctival hyperemia and a conjunctival nevus with 4 inclusion cysts, one of which was filled with purulent material. Fluorescein staining of the conjunctival epithelium was negative. Α mini-incision of the white cyst was performed using a 30 G needle, followed by bimanual drainage and topical treatment with tobramycin and moxifloxacin drops every 3 h for a week. A swab of the purulent drainage was positive for gram-positive flora. One week after the drainage of the cyst, the patient was asymptomatic and on slit-lamp examination, the 4 inclusion cysts were filled with a transparent liquid, there was not any vessel dilation and fluorescein staining was negative.

Conclusion: Conjunctival inclusion cysts, although considered benign, can become infected and form a conjunctival abscess. A mini-incision on the slit lamp combined with bimanual drainage and followed by topical antibiotic drops seems to be a safe and effective treatment.

简介结膜囊肿通常无症状,但会引起异物感并导致干眼症。本病例报告旨在描述一名 36 岁健康患者结膜痣感染性包涵囊肿的表现和治疗:一名 36 岁的健康男子因左眼红肿、疼痛 1 天来急诊就诊。裂隙灯检查发现结膜充血,结膜痣上有 4 个包涵囊肿,其中一个充满了脓性物质。结膜上皮的荧光素染色呈阴性。使用 30 G 的针头对白色囊肿进行了 Α 迷你切口,随后进行了双侧引流,并使用妥布霉素和莫西沙星滴眼液进行局部治疗,每 3 小时一次,持续一周。脓性引流液的拭子显示革兰氏阳性菌群呈阳性。囊肿引流一周后,患者已无症状,经裂隙灯检查,4 个包涵囊肿内充满透明液体,无任何血管扩张,荧光素染色阴性:结论:结膜包涵囊肿虽然被认为是良性的,但也可能发生感染并形成结膜脓肿。在裂隙灯上做一个小切口,结合双侧引流术,然后局部滴用抗生素,似乎是一种安全有效的治疗方法。
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引用次数: 0
Bevacizumab and External Beam Radiation Therapy for Diffuse Choroidal Hemangioma in Sturge-Weber Syndrome: A Case Report. 贝伐单抗和体外放射疗法治疗 Sturge-Weber 综合征弥漫性脉络膜血管瘤:病例报告。
IF 0.5 Q4 OPHTHALMOLOGY Pub Date : 2024-08-05 eCollection Date: 2024-01-01 DOI: 10.1159/000539655
Jeffrey Yu, Dena Ballouz, Yannis M Paulus

Introduction: Sturge-Weber syndrome, a congenital vascular disorder, is associated with diffuse choroidal hemangiomas in which the current mainstay of treatment is radiation therapy, including external beam radiation therapy (EBRT). The purpose of this case report was to present a novel combination of treatments for diffuse choroidal hemangioma.

Case presentation: A 37-year-old man with a history of Sturge-Weber-associated glaucoma presented with an acute-onset decrease in vision in the right eye. Best-corrected visual acuity (BCVA) at the presentation was 20/400 in the right eye. Examination revealed a total macula-off, bullous, folded exudative retinal detachment and findings consistent with diffuse choroidal hemangioma. The patient was treated with a single injection of intravitreal anti-vascular endothelial growth factor (anti-VEGF) agent bevacizumab and 10 fractions of EBRT of the right eye. Follow-up examination at 17 months demonstrated complete resolution of subretinal fluid and no evidence of choroidal elevation on B-scan. Final BCVA in the right eye was 20/1,000.

Conclusion: This case uses simultaneous treatment with EBRT and bevacizumab in the treatment of diffuse choroidal hemangioma and associated exudative retinal detachment. Clinicians may use anti-VEGF agents early in the course of the disease in determining whether they may assist in preventing visual decline.

导言:Sturge-Weber综合征是一种先天性血管疾病,与弥漫性脉络膜血管瘤有关,目前的主要治疗方法是放射治疗,包括体外放射治疗(EBRT)。本病例报告旨在介绍一种治疗弥漫性脉络膜血管瘤的新型综合疗法:病例介绍:一名 37 岁的男子,曾患斯特格-韦伯相关性青光眼,急性发作时右眼视力下降。就诊时右眼最佳矫正视力(BCVA)为 20/400。检查发现黄斑完全脱落,呈牛皮状,褶皱渗出性视网膜脱离,检查结果与弥漫性脉络膜血管瘤一致。患者接受了一次玻璃体内注射抗血管内皮生长因子(anti-VEGF)药物贝伐单抗和 10 次右眼 EBRT 治疗。17 个月的随访检查显示,视网膜下积液完全消退,B 型扫描也未发现脉络膜隆起。右眼的最终BCVA为20/1,000:本病例采用 EBRT 和贝伐单抗同时治疗弥漫性脉络膜血管瘤和相关渗出性视网膜脱离。临床医生可在病程早期使用抗血管内皮生长因子药物,以确定它们是否有助于防止视力下降。
{"title":"Bevacizumab and External Beam Radiation Therapy for Diffuse Choroidal Hemangioma in Sturge-Weber Syndrome: A Case Report.","authors":"Jeffrey Yu, Dena Ballouz, Yannis M Paulus","doi":"10.1159/000539655","DOIUrl":"10.1159/000539655","url":null,"abstract":"<p><strong>Introduction: </strong>Sturge-Weber syndrome, a congenital vascular disorder, is associated with diffuse choroidal hemangiomas in which the current mainstay of treatment is radiation therapy, including external beam radiation therapy (EBRT). The purpose of this case report was to present a novel combination of treatments for diffuse choroidal hemangioma.</p><p><strong>Case presentation: </strong>A 37-year-old man with a history of Sturge-Weber-associated glaucoma presented with an acute-onset decrease in vision in the right eye. Best-corrected visual acuity (BCVA) at the presentation was 20/400 in the right eye. Examination revealed a total macula-off, bullous, folded exudative retinal detachment and findings consistent with diffuse choroidal hemangioma. The patient was treated with a single injection of intravitreal anti-vascular endothelial growth factor (anti-VEGF) agent bevacizumab and 10 fractions of EBRT of the right eye. Follow-up examination at 17 months demonstrated complete resolution of subretinal fluid and no evidence of choroidal elevation on B-scan. Final BCVA in the right eye was 20/1,000.</p><p><strong>Conclusion: </strong>This case uses simultaneous treatment with EBRT and bevacizumab in the treatment of diffuse choroidal hemangioma and associated exudative retinal detachment. Clinicians may use anti-VEGF agents early in the course of the disease in determining whether they may assist in preventing visual decline.</p>","PeriodicalId":9635,"journal":{"name":"Case Reports in Ophthalmology","volume":null,"pages":null},"PeriodicalIF":0.5,"publicationDate":"2024-08-05","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11324206/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"141981801","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}
引用次数: 0
A Simple Orbital Lymphatic Malformation Clinically Mimics Primary Conjunctival Cyst. 临床上与原发性结膜囊肿相似的单纯眼眶淋巴畸形
IF 0.5 Q4 OPHTHALMOLOGY Pub Date : 2024-07-25 eCollection Date: 2024-01-01 DOI: 10.1159/000540126
Sasikant Leelawongs, Luksanaporn Krungkraipetch

Introduction: A simple orbital lymphatic malformation usually presents with acute proptosis with ophthalmoplegia after an upper respiratory tract infection. Various treatments have been described but the lesions often recur after treatment due to their infiltrative nature.

Case presentations: A 6-year-old girl presented with a 5 × 10 mm translucent mass in the left inferior fornix for 3 years but she denied diplopia and visual disturbance. Orbital computed tomography with contrast emphasized the clinical findings, a nonseptated cystic lesion without enhancement in the anterior orbit. The lesion was surgically totally excised. Histopathological findings revealed an endothelial lining cyst without intraluminal cells consistent with a lymphatic malformation. In a 1-year follow-up, the lesion does not recur.

Conclusion: The simple orbital lymphatic malformation can present as a single nonseptate cystic lesion. Pathological study should be done for the definitive diagnosis. Surgical excision can achieve a favorable outcome.

简介单纯性眼眶淋巴畸形通常在上呼吸道感染后表现为急性突眼并伴有眼肌麻痹。已有多种治疗方法,但由于其浸润性,病变往往在治疗后复发:一名 6 岁女孩因左侧下穹窿有一个 5 × 10 毫米的半透明肿块就诊 3 年,但她否认有复视和视力障碍。眼眶计算机断层扫描造影证实了临床发现,即眼眶前部无强化的非化脓性囊性病变。病灶经手术完全切除。组织病理结果显示,病变为内皮衬里囊肿,无腔内细胞,与淋巴畸形一致。随访1年,病变未再复发:结论:单纯性眼眶淋巴畸形可表现为单发无隔膜的囊性病变。结论:单纯性眼眶淋巴畸形可表现为单发无隔膜囊性病变,应进行病理检查以明确诊断。手术切除可获得良好的疗效。
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引用次数: 0
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