Cancer-associated retinopathy

癌症相关视网膜病变
  • 文章类型: Journal Article
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  • 文章类型: Journal Article
    这项病例对照研究比较了静脉免疫球蛋白(IVIg)治疗患者的自身免疫性视网膜病变(AIR)结局与那些没有治疗的人。在长期随访中,IVIg与视力和视网膜电图参数的保存有关。
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  • 文章类型: Case Reports
    目的:多发性骨髓瘤(MM)是导致单克隆浆细胞增殖的浆细胞发育不良。多发性骨髓瘤的眼部受累并不常见,但可能发生。MM的眼部表现可能包括角膜,uvea,和视网膜血管.我们介绍了与闷烧MM相关的自身免疫性视网膜病的罕见病例。
    方法:一名76岁女性,无明显既往病史或眼部病史,视力恶化4个月,夜间驾驶困难,和周围视力的丧失。检查可见视神经苍白和血管衰减。视野测试表明,两只眼睛都有明显的渐进性视野丧失。视网膜电图在所有条件下都消失。血清蛋白电泳显示IgG显着升高,具有M-spike,随后进行了骨髓活检,显示有12.5%的浆细胞,符合MM的诊断。CAR抗体检测抗烯醇化酶阳性,反GAPDH,和抗Rab6抗体,与自身免疫性视网膜病变一致。
    结论:与MM相关的自身免疫性视网膜病变极为罕见。这种情况的管理具有挑战性,作为基础疾病的治疗通常不会导致视觉症状的改善。最终,视力预后很差,患者和临床医生都应该意识到受到保护的视觉潜力。
    结论:自身免疫性视网膜病与多发性骨髓瘤的相关性很少见。对于医生来说,了解这些表现以确保对患者进行及时和适当的诊断和管理至关重要。
    OBJECTIVE: Multiple myeloma (MM) is a plasma cell dyscrasia leading to proliferation of monoclonal plasma cells. Ocular involvement in multiple myeloma is uncommon but can occur. The ocular manifestations of MM may include the cornea, uvea, and retinal vasculature. We present a rare case of autoimmune retinopathy associated with smoldering MM.
    METHODS: A 76-year-old female with no significant past medical or ocular history presented with four months of worsening vision, difficulty with night driving, and loss of peripheral vision. Examination was notable for pallor of the optic nerves and vascular attenuation. Visual field testing demonstrated significant and progressive field loss in both eyes. An electroretinogram was extinguished under all conditions. Serum protein electrophoresis showed a significant elevation of IgG with an M-spike, and a subsequent bone marrow biopsy was performed showing 12.5% plasma cells, consistent with the diagnosis of MM. CAR antibody testing was positive for anti-enolase, anti-GAPDH, and anti-Rab6 antibodies, consistent with autoimmune retinopathy.
    CONCLUSIONS: Autoimmune retinopathy associated with MM is exceedingly rare. Management of this condition is challenging, as treatment of the underlying disease does not often lead to improvement in visual symptoms. Ultimately, visual prognosis is very poor, and both patients and clinicians should be aware of the guarded visual potential.
    CONCLUSIONS: The association of autoimmune retinopathy with multiple myeloma is rare. It is crucial for physicians to be aware of such manifestations to ensure timely and appropriate diagnosis and management for patients.
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  • 文章类型: Systematic Review
    目的:系统文献综述以前使用的治疗方案对已证实的癌症相关性视网膜病变(CAR)患者的治疗效果。
    结论:对于非常差的CAR和视觉预后,没有普遍接受的治疗算法。我们描述了在高剂量皮质类固醇治疗后视力显着改善的CAR患者,然后进行血浆置换(PLEX),并提出了先前使用的方案在治疗已证实的CAR患者中的治疗效果的系统文献综述结果。
    方法:我们描述了一名70岁的CAR患者,他在接受高剂量的全身性皮质类固醇治疗后7次PLEX治疗后,视力显着改善。然后,我们报告了所有以前发表的英文文献的系统评价结果,这些文献讨论了用于抗体证实的CAR患者的各种治疗方案的视觉结果。
    结果:该指标患者是首例CAR患者,在接受高剂量皮质类固醇和PLEX治疗后视力持续显著改善。系统评价确定了28例抗体证实的CAR病例,其中26个用类固醇治疗,导致不同程度的视力改善58%(15/26)。从症状发作到开始治疗的时间和类固醇的剂量不影响视觉结果。除类固醇外,三名患者还接受了PLEX治疗,2/3的视力得到改善,然而,仅接受类固醇治疗的患者与接受类固醇+PLEX治疗的患者的视觉结局没有差异。
    结论:使用类固醇和/或类固醇+PLEX治疗可在60%的患者中改善视力。用PLEX去除抗恢复素抗体可以阻止对光感受器的免疫攻击,并可能改善视觉功能,因此除了类固醇外,还应该考虑它。为了建立治疗方案并进一步确定不同方法的有效性,需要对更大的队列进行进一步的研究。
    Systematic literature review of treatment efficacy of previously used protocols in treating patients with proven cancer-associated retinopathy (CAR).
    There is no universally accepted treatment algorithm for CAR and visual prognosis is very poor. We describe a patient with CAR with dramatic improvement in vision after treatment with high doses of corticosteroids followed by plasma exchange (PLEX) and present results of a systematic literature review of treatment efficacy of previously used protocols in treating patients with proven CAR.
    We describe a 70-year-old man with CAR who demonstrated dramatic improvement in vision after treatment with high doses of systemic corticosteroids followed by 7 sessions of PLEX. We then report the results of a systematic review of all previously published English literature discussing visual outcomes of various treatment regimens used for patients with antibody-proven CAR.
    The index patient is a rare case of CAR with sustained significant improvement in vision after treatment with high doses of corticosteroids followed by PLEX. The systematic review identified 28 antibody-proven cases of CAR, 27 of which were treated with steroids, which resulted in varying degrees of improvement in visual acuity in 59% (16 of 27). The time from symptom onset to initiation of treatment and the dose of steroids did not influence the visual outcome. Three patients were also treated with PLEX in addition to steroids, and 2 of 3 patients demonstrated improvement in vision; however, there was no difference in visual outcome in patients treated with steroids only versus those treated with steroids + PLEX.
    Treatment with steroids or steroids + PLEX resulted in some improvements in visual acuity in 59% of patients. Removal of antirecoverin antibodies with PLEX can arrest the immune attack on the photoreceptors and potentially improve visual function; thus, it should be considered in addition to steroids. Further studies with larger cohorts are needed to establish a treatment protocol and further determine the effectiveness of the different approaches.
    The author(s) have no proprietary or commercial interest in any materials discussed in this article.
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  • 文章类型: Review
    背景:数十种副肿瘤综合征会影响视觉系统,从结膜类天疱疮到枕叶皮质脑病。最常见的眼部综合征是双侧弥漫性葡萄膜黑素细胞增生(BDUMP)和自身免疫性视网膜病。目的:回顾这两个实体的关键特征,然后专注于过去10年中的治疗进展。研究设计:结构化数据抽象的文献综述。结果:对发病机制的主要见解一直缺乏。血浆置换似乎可以改善大部分BDUMP患者的视力。影响副肿瘤性视网膜病变的视觉结果的临床变量的数量以及各种局部和全身治疗选择使得临床有效性的解释变得困难。结论:这些疾病的罕见性使随机临床试验不太可能。临床专业组织可能是时候使用改良的Delphi方法建立共识算法来诊断和管理视网膜副肿瘤综合征,以增强临床沟通和临床试验。
    Background: Dozens of paraneoplastic syndromes affect the visual system ranging from conjunctival pemphigoid to encephalopathy of the occipital cortex. The most profiled ocular syndromes are bilateral diffuse uveal melanocytic proliferation (BDUMP) and the autoimmune retinopathies.Purpose: To review the critical features of these 2 entities then concentrate on advancements in treatment made within the last 10 years.Study Design: Literature review with structured data abstraction.Results: Major insights into pathogenesis have been wanting. Plasmapheresis appears to improve vision in a substantial proportion of patients with BDUMP. The number of clinical variables that influence visual outcome in paraneoplastic retinopathies combined with the variety of local and systemic treatment options makes interpretation of clinical effectiveness difficult.Conclusions: The rarity of these disorders makes randomized clinical trials unlikely. It may be time for a clinical professional organization to use a modified Delphi method to establish a consensus algorithm for the diagnosis and management of retinal paraneoplastic syndromes to augment clinical communications and clinical trials.
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  • 文章类型: Journal Article
    副肿瘤综合征是在全身恶性肿瘤状态下发生的一组临床症状。神经系统的副肿瘤综合征可以影响中枢和周围神经系统的任何部分,也可能影响眼睛。在神经眼科,副肿瘤综合征有多种表现,可影响传入和传出视觉系统。传入系统可能涉及视神经,视网膜和葡萄膜;传出系统可能涉及眼球运动,神经肌肉关节或不自主的眼球运动和瞳孔异常,也可能有视觉系统外的其他神经系统症状。本文讨论了临床表现,病理机制,神经眼科副肿瘤综合征的检测方法和治疗方法。副肿瘤综合征的表现多种多样,诊断是困难的,应该系统地考虑治疗。鉴别诊断,这些表现的最佳评估和管理不仅是治疗的关键,也是一个挑战。
    Paraneoplastic syndrome is a group of clinical symptoms that occur in the state of systemic malignant tumors. Paraneoplastic syndrome of the nervous system can affect any part of the central and peripheral nervous system and may also affect the eyes. In neuroophthalmology, paraneoplastic syndrome has a variety of manifestations that can affect both the afferent and efferent visual systems. The afferent system may involve the optic nerve, retina and uvea; the efferent system may involve eye movement, neuromuscular joints or involuntary eye movements and pupil abnormalities and may also have other neurological symptoms outside the visual system. This article discusses the clinical manifestations, pathological mechanisms, detection methods and treatment methods of paraneoplastic syndrome in neuroophthalmology. The performance of paraneoplastic syndrome is diverse, the diagnosis is difficult, and the treatment should be considered systematically. Differential diagnosis, optimal evaluation and management of these manifestations is not only the key to treatment but also a challenge.
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  • 文章类型: Journal Article
    BACKGROUND: Paraneoplastic neurological syndromes (PNS) are rare neurological conditions and they are mostly triggered by autoimmune mechanisms. Cancer-related retinopathies (CAR) are even rarer and commonly related with breast tumor in woman. This limits our knowledge about pathophysiology of CAR. In this study, we question the association between histopathological findings and onconeural antibodies in breast cancer.
    METHODS: Thirty-two patients with newly diagnosed breast cancer admitted to the oncology outpatient clinic were included in the study. None of the participants have visual complaints. After the neurological examination of the patients, two tubes of 5 cc venous blood were obtained by screening onconeuronal antibodies. Samples were investigated in ASDETAE (İstanbul University Experimental Medicine Research Institute).
    RESULTS: Patients included in the study included one patinet (3.1%) with grade 1, 14 patients (43.8%) with grade 2 and 17 patients (53.1%) with grade 3 invasive breast cancer. Perineural invasion was detected in 5 (15.6%) patients. Progesterone receptor positivity was found in 26 (81.2%) patients and estrogen receptor positivity was found in 27 (84.4%) patients. In 7 (21.9%) patients, CERBB2 was positive and in 25 (78.1%) patients, Ki 67 was positive. A total of 12 (37.5%) patients had onconeuroneal antibody positivity. Antibody positivity was significantly higher in patients with high grade tumor (p=0.008).
    CONCLUSIONS: There may be a relationship between tumor grade and the presence of onconeuronal antibodies in breast cancer patients. By the detection of new biochemical markers, significant contribution can be made to the early diagnosis and treatment of underlying cancer.
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  • 文章类型: Journal Article
    眼部累及副肿瘤综合征表现出多种临床症状。了解背景病理生理和免疫病理因素可以帮助做出与患者表现出的体征和症状相一致的更精细的鉴别诊断。有两个主要的病理生理学分支:(1)自身免疫病理机制,与癌症相关的视网膜病变(CAR),黑色素瘤相关视网膜病变(MAR),癌症相关性视锥功能障碍(CACD),副肿瘤卵黄样黄斑病变(PVM),副肿瘤性视神经炎(PON),和(2)异位肽,通常由肿瘤表达的生长因子(T-exGF)引起,并伴有双侧弥漫性葡萄膜黑素细胞增生(BDMP)。对患者症状进行细致的系统分析是关键的诊断步骤,辅以多模态成像,其中包括眼底摄影,光学相干层析成像,眼底自发荧光,荧光素眼底血管造影,电生理检查,如果由临床医生开处方,有时会出现眼底吲哚美辛绿色血管造影。诊断需要评估循环抗体的存在。抗视网膜自身抗体与视觉副肿瘤综合征高度相关,除了监测治疗外,还可以通过对临床表现进行分类来指导诊断。
    Ocular-involving paraneoplastic syndromes present a wide variety of clinical symptoms. Understanding the background pathophysiological and immunopathological factors can help make a more refined differential diagnosis consistent with the signs and symptoms presented by patients. There are two main pathophysiology arms: (1) autoimmune pathomechanism, which is presented with cancer-associated retinopathy (CAR), melanoma-associated retinopathy (MAR), cancer-associated cone dysfunction (CACD), paraneoplastic vitelliform maculopathy (PVM), and paraneoplastic optic neuritis (PON), and (2) ectopic peptides, which is often caused by tumor-expressed growth factors (T-exGF) and presented with bilateral diffuse uveal melanocytic proliferation (BDUMP). Meticulous systematic analysis of patient symptoms is a critical diagnostic step, complemented by multimodal imaging, which includes fundus photography, optical coherent tomography, fundus autofluorescence, fundus fluorescein angiography, electrophysiological examination, and sometimes fundus indocyjanin green angiography if prescribed by the clinician. Assessment of the presence of circulating antibodies is required for diagnosis. Antiretinal autoantibodies are highly associated with visual paraneoplastic syndromes and may guide diagnosis by classifying clinical manifestations in addition to monitoring treatment.
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  • 文章类型: Journal Article
    To determine if immunological markers (1) are significantly different between autoimmune retinopathy (AIR) patients and controls and (2) correlate with disease progression in AIR patients.
    We enrolled patients with a possible AIR diagnosis, as well as control participants without eye disease, autoimmunity, or cancer. Immunological markers were tested in all participants. In addition, AIR patients had up to three blood draws for testing over their disease course. For AIR patients, clinical measures, including visual acuity (VA) and Goldmann visual field (GVF) area, were recorded at each draw. We used the Mann-Whitney U test to compare the immunological markers between AIR patients and controls. We used multilevel mixed-effect regression to investigate the correlation between markers and clinical parameters over time in AIR patients.
    Seventeen patients with AIR and 14 controls were included. AIR patients had a higher percent of monocytes (Z = 3.076, P = 0.002). An increase in immunoglobulin G against recoverin was correlated with a VA decrease (β = 0.0044, P < 0.0001). An increase in monocyte proportion was correlated with a decrease in GVF area (β = -7.27, P = 0.0021). Several markers of B-cell depletion were correlated with GVF improvement.
    Monocytes may play a role in AIR pathophysiology and be a disease activity marker. B-cell depletion markers correlated with clinical parameter improvement, particularly GVF.
    This work elucidates immunologic markers that may improve the accuracy of diagnosis and treatment of AIR.
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  • 文章类型: Journal Article
    Autoimmune retinopathy (AIR) refers to a group of rare autoimmune retinal degenerative diseases presumably caused by cross-reactivity of serum autoantibodies against retinal antigens. The pathogenesis of AIR remains largely presumptive and there are a significant number of antiretinal antibodies that have been detected in association with AIR. The diagnosis of AIR is largely based on the demonstration of antiretinal antibodies in the serum along with suggestive clinical features and ancillary investigations. A high index of suspicion along with early diagnosis and treatment may play a critical role to lower the risk of irreversible immunological damage to the retinal cells in these patients. A multi-disciplinary approach for complete management and evaluation is helpful in such conditions. Various therapeutic options have been described for the treatment of AIR, though there is no consensus on standard treatment protocol.
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