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Advanced diagnostic approaches for SHAPU: Metagenomic sequencing and interleukin analysis SHAPU 的先进诊断方法:元基因组测序和白细胞介素分析
Pub Date : 2024-07-15 DOI: 10.1016/j.ajoint.2024.100056
Ranju Kharel Sitaula , Rajeev Shrestha , Pratap Karki , Haramaya Gurung , Anadi Khatri KC , Nishan Katuwal , Navin Adhikari , Rajendra Bhatta , Eliya Shrestha , Indraman Maharjan , Pursottam Joshi , Biraj Man Karmacharya , Angira Shrestha , Ananda K Sharma , Sagun Narayan Joshi , Madan Prasad Upadhaya

Purpose

Seasonal Hyperacute Panuveitis (SHAPU) is an ocular emergency where the genesis between infective and immunological is under investigation. This is the first report in the literature that attempts to explore the unidentified cause using next-generation metagenomic sequencing and immunomarker analysis.

Design

Cross-sectional quantitative study.

Methods

In this study, vitreous samples were collected from the SHAPU cases during the outbreak of 2019/2020. The vitreous samples of 10 random cases got selected for metagenomic next-generation sequencing. And 5 vitreous samples ran for immune analysis of levels of interleukin (IL) 6 and 10 using the ELISA technique. The ocular samples were extracted and sequenced on Illumina iSeq100 and analyzed through CZ ID platform. The true hits, for the suspected causative organisms were confirmed using qPCR through designed primers specific for that particular organism.

Results

A total of 30 eyes of 30 patients were included. Upon sequencing the DNA libraries, hits were observed for Streptococcus pneumoniae in 2/10 cases, confirmed by qPCR. IL-6 level was increased in 5/5 cases with IL-6/IL-10 greater than 1.

Conclusion

We report the presence of S. pneumoniae for the first time using a metagenomics analysis and increased IL-6 to IL-10 ratio via immune-analysis, indicating that SHAPU could have both infective and immunological facets on its causation.

目的季节性高急性葡萄膜炎(SHAPU)是一种眼部急症,感染性和免疫性之间的成因正在研究之中。这是文献中第一份试图利用新一代元基因组测序和免疫标志物分析来探索未确定病因的报告。设计横断面定量研究。方法本研究收集了2019/2020年疫情爆发期间SHAPU病例的玻璃体样本。随机选取 10 例病例的玻璃体样本进行元基因组下一代测序。5份玻璃体样本采用ELISA技术对白细胞介素(IL)6和10的水平进行免疫分析。眼部样本在 Illumina iSeq100 上进行提取和测序,并通过 CZ ID 平台进行分析。通过为特定生物设计的特异引物,使用 qPCR 对疑似致病生物的真正命中进行确认。对 DNA 文库进行测序后,2/10 的病例发现了肺炎链球菌,并通过 qPCR 进行了确认。结论我们首次通过元基因组学分析报告了肺炎链球菌的存在,并通过免疫分析报告了IL-6与IL-10比值的升高,这表明SHAPU可能同时具有感染和免疫两方面的致病因素。
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引用次数: 0
Solitary fibrous tumor of the eyelid 眼睑单发纤维性肿瘤
Pub Date : 2024-07-14 DOI: 10.1016/j.ajoint.2024.100050
Samantha D Butterfield , David Zlotnick , Rona Z Silkiss
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引用次数: 0
Optic cup morphology associated with glaucomatous damage: Findings from the Primary Open-Angle African American Glaucoma Genetics (POAAGG) Study 与青光眼损害相关的视杯形态:原发性开角型非裔美国人青光眼遗传学(POAAGG)研究结果
Pub Date : 2024-07-14 DOI: 10.1016/j.ajoint.2024.100053
Anusha Mamidipaka , Isabel Di Rosa , Patrick Augello , Yineng Chen , Rebecca Salowe , Yan Zhu , Victoria Addis , Prithvi Sankar , Ebenezer Daniel , Gui-Shuang Ying , Joan O'Brien

Purpose

While it has been well established that advanced glaucoma is associated with a large cup-to-disc ratio (CDR), it is not known if the shape of the cup, particularly excavated cups with expanded lower portions (bean-pot cups), play any additional role in glaucomatous damage. We investigated this among individuals of African ancestry, a population that is more vulnerable to glaucoma than any other ethnic group.

Design

Case-control study

Methods

  

Setting

Institutional (University of Pennsylvania)

Subjects

3,255 eyes from 1,734 glaucoma cases from the Primary Open-Angle African American Glaucoma Genetics (POAAGG) study.

Procedure

Two graders independently assessed quantitative and qualitative aspects of the optic cup, with any discrepancies adjudicated by an ophthalmologist. The predominant cup shape (>50 %) was chosen in cases in which features of two or more cup shapes were present in the same eye. Comparisons of demographic and ocular characteristics among three cup shape groups (conical, cylindrical, and bean-pot) performed using generalized linear models, and generalized estimated equations applied to account for inter-eye correlation.

Main Outcome Measures

Qualitative features of cup shape and phenotypic traits, in conjunction with demographic and genetic information.

Results

Of 3,255 eyes, a total of 1,339 (41.1 %) exhibit a conical cup shape, 1,470 (45.2 %) have a cylindrical cup shape, and 446 (13.7 %) display a bean-pot cup shape. Compared to other cup morphology, bean-pot cups are significantly associated with lower MD, larger CDR, higher IOP, thinner RNFL, and worse VA in logMAR (all p < 0.001). Genetic analysis does not show any association between various genetic variants and cup shape. Factors independently predictive of bean-pot cupping include younger age at diagnosis (aOR 0.96 per 1 year increase in age of enrollment, p < 0.0001), CDR (adjusted odds ratio (aOR) 1.87, p < 0.0001), and the presence of certain optic disc features, including visible pores in the LC (aOR 2.76, p < 0.0001), nasalization of vessels (aOR 2.64, p < 0.0001), and vessel bayonetting (aOR 2.94, p < 0.0001).

Conclusion

This study shows the clinical significance of different cup shapes in glaucoma in an African ancestry population and suggests that bean-pot cups are associated with the most severe glaucomatous damage, independent of cup-disc ratio. This association should be considered when determining prognosis following glaucoma interventions.

目的虽然晚期青光眼与杯盘比(CDR)过大有关这一点已经得到证实,但是杯的形状,尤其是下部膨大的挖空杯(豆壶杯)在青光眼损害中是否起到额外的作用还不得而知。我们在非洲血统的人群中进行了调查,非洲血统的人比其他任何种族的人都更容易患青光眼。设计病例对照研究方法研究机构(宾夕法尼亚大学)研究对象来自原发性开角型非裔美国人青光眼遗传学(POAAGG)研究的 1734 例青光眼病例中的 3,255 只眼睛。如果同一只眼睛出现两种或两种以上的视杯形状,则选择最主要的视杯形状(50%)。使用广义线性模型对三组杯形(圆锥形、圆柱形和豆锅形)的人口统计学特征和眼部特征进行比较,并应用广义估计方程来考虑眼间相关性。结果 在 3255 只眼睛中,共有 1339 只(41.1%)呈现圆锥形杯状,1470 只(45.2%)呈现圆柱形杯状,446 只(13.7%)呈现豆瓣形杯状。与其他杯状形态相比,豆壶杯与较低的 MD、较大的 CDR、较高的 IOP、较薄的 RNFL 和较差的 VA(以 logMAR 表示)显著相关(所有 p < 0.001)。遗传分析未显示各种遗传变异与杯形之间存在任何关联。可独立预测豆壶形的因素包括诊断时年龄较小(入院年龄每增加 1 岁,aOR 为 0.96,p < 0.0001)、CDR(调整后的几率比(aOR)为 1.87,p < 0.0001)以及存在某些视盘特征,包括 LC 中的可见孔隙(aOR 为 2.76,p < 0.结论这项研究显示了不同杯形在非洲裔青光眼人群中的临床意义,并表明豆锅杯与最严重的青光眼损害相关,与杯盘比无关。在确定青光眼干预后的预后时,应考虑这种关联。
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引用次数: 0
3-year safety and efficacy results of PreserFlo™ Microshunt in glaucoma patients: A multicentre European cohort study 青光眼患者使用 PreserFloTM 微分流术的 3 年安全性和疗效结果:欧洲多中心队列研究
Pub Date : 2024-07-14 DOI: 10.1016/j.ajoint.2024.100054
Karl Mercieca , Raj Bhayani , Jose Maria Martinez-de-la-Casa , Michele Figus , Karsten Klabe , Alessandro Rabiolo , Constance Weber

Purpose

To report three-year outcomes of a multicenter cohort undergoing Preserflo Microshunt surgery (PFMS).

Design

Retrospective cohort study

Methods

Retrospective review of 100 consecutive eyes (91 patients) undergoing PFMS from four tertiary-referral glaucoma centers. Primary outcome were success rates. Secondary outcomes included: intraocular pressure (IOP), best-corrected visual acuity, medication, complications and postoperative interventions.

Results

100 eyes of 91 patients were included. Qualified and complete success rates (95 % CI) were 80 % (72–87 %) and 56 % (47–65 %) for Criterion A (IOP≤21 mmHg), 75 % (67–83 %) and 55 % (46–65 %) for Criterion B (IOP≤18 mmHg), 44 % (31–50 %) and 39 % (30–49 %) for Criterion C (IOP≤15 mmHg) and 10 % (5–16 %) and 10 % (5–16 %) for Criterion D (IOP≤12 mmHg) respectively. Mean IOP decreased from 23.8 mmHg to 14.1 mmHg (reduction of 35.3 %) after 36 months with a reduction of IOP-lowering agents from 2.52 (0 – 4) to 0.69 (0 – 3). 7 eyes (7 %) needed additional glaucoma surgery. Of these, 3 eyes underwent a glaucoma drainage device and 4 eyes had a trabeculectomy.36 eyes received additional procedures after PFMS implantation. Of these, 14 eyes (14 %) underwent a surgical revision and 12 eyes (12 %) a bleb needling procedure.

The complication rate was low: 1 eye developed corneal decompensation and underwent a DMEK after 24 months. 1 eye had a conjunctival erosion requiring a revision surgery. 2 eyes developed a postoperative uveitis that was treated with topical steroids and resolved without further damage. There were no hypotony-related complications.

Conclusions

PFMS surgery is a safe and effective procedure for reducing IOP and pressure-lowering therapy.

目的报告接受 Preserflo 微分流手术(PFMS)的多中心队列的三年疗效。设计回顾性队列研究方法回顾性分析四家三级青光眼转诊中心接受 Preserflo 微分流手术的 100 只连续眼球(91 名患者)。主要结果为成功率。次要结果包括:眼压(IOP)、最佳矫正视力、用药、并发症和术后干预。标准 A(眼压≤21 mmHg)的合格率和完全成功率(95 % CI)分别为 80 %(72-87 %)和 56 %(47-65 %),标准 B(眼压≤18 mmHg)的合格率和完全成功率分别为 75 %(67-83 %)和 55 %(46-65 %)、标准 C(眼压≤15 毫米汞柱)分别为 44%(31-50%)和 39%(30-49%),标准 D(眼压≤12 毫米汞柱)分别为 10%(5-16%)和 10%(5-16%)。36 个月后,平均眼压从 23.8 mmHg 降至 14.1 mmHg(降低了 35.3%),降眼压药物从 2.52(0 - 4)降至 0.69(0 - 3)。有 7 只眼睛(7%)需要进行额外的青光眼手术。其中,3 只眼睛接受了青光眼引流装置手术,4 只眼睛接受了小梁切除术。并发症发生率很低:1 只眼睛出现角膜失代偿,24 个月后接受了 DMEK。1 只眼睛出现结膜糜烂,需要进行翻修手术。两只眼睛术后出现葡萄膜炎,经局部类固醇治疗后缓解,未造成进一步损害。结论PFMS手术是一种安全有效的降低眼压和降压治疗方法。
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引用次数: 0
A randomized fellow-eye controlled trial study of 1% terpinen-4-ol vs 1% tea tree oil eyelid cleanser for the treatment of meibomian gland dysfunction 治疗睑板腺功能障碍的 1%特平宁-4-醇与 1%茶树油眼睑清洁剂的随机同眼对照试验研究
Pub Date : 2024-07-06 DOI: 10.1016/j.ajoint.2024.100052
Preamjit Saonanon , Wannit Witoonchart , Dusadee Charnvanich , Somphob Thompho , Kittichai Chaiseeda , Yuda Chongpison , Vannarut Satitpitakul

Purpose

To compare the pre- and post-treatment differences in non-invasive tear breakup time (NITBUT) between a 1 % terpinen-4-ol (T4O) and a 1 % tea tree oil (TTO) eyelid cleanser

Design

A randomized controlled trial

Method

Thirty-three participants diagnosed with MGD were enrolled. All patients and the outcome evaluator were masked to the treatment allocation. One eye of each patient was randomly assigned to receive the T4O cleanser, while the fellow eye received the TTO cleanser. Baseline and eight week assessments included the collection of the ocular surface disease index (OSDI) questionnaire, NITBUT (in seconds), lipid layer thickness (in nanometers), ocular surface staining scores, and the presence of Demodex as cylindrical dandruff. The main outcome measurement was the pre- and post-treatment difference in NITBUT. Patient preference, safety, and tolerability were also assessed.

Results

There was a statistically significant increase in NITBUT in the T4O group (n = 30; 2.27 ± 1.09; 95 % confidence interval [CI]: 0.12–4.42; p = 0.038), but not in the TTO group (n = 30; 0.04 ± 1.10; 95 % CI: -2.19–2.11; p = 0.972). Both groups showed significant improvement in post-treatment ocular surface staining scores. Fifteen subjects (50 %) preferred TTO due to less cooling sensation experienced during application, nine (30 %) preferred T4O, and six (20 %) had no preference. No major adverse events were observed.

Conclusion

Eyelid cleaning with 1 % T4O in MGD for eight weeks was found to increase NITBUT with good tolerability, although it was less preferred compared to TTO.

目的比较 1 % 的松脂酸-4-醇(T4O)和 1 % 的茶树油(TTO)眼睑清洁剂在非侵入性泪液破裂时间(NITBUT)方面治疗前后的差异。所有患者和结果评估者均被蒙蔽,不知道治疗分配。每名患者的一只眼睛被随机分配接受T4O清洁剂,另一只眼睛则接受TTO清洁剂。基线和八周评估包括收集眼表疾病指数 (OSDI) 问卷、NITBUT(以秒为单位)、脂质层厚度(以纳米为单位)、眼表染色评分,以及作为圆柱形皮屑的眼球脱墨虫的存在情况。主要结果测量指标是治疗前后 NITBUT 的差异。结果T4O组(n = 30;2.27 ± 1.09;95 % 置信区间 [CI]:0.12-4.42;p = 0.038)的NITBUT有统计学意义上的显著增加,而TTO组(n = 30;0.04 ± 1.10;95 % CI:-2.19-2.11;p = 0.972)则没有。两组治疗后的眼表染色评分均有明显改善。15名受试者(50%)倾向于使用 TTO,因为在使用过程中冷却感较小,9 名受试者(30%)倾向于使用 T4O,6 名受试者(20%)没有偏好。结论在 MGD 中使用 1 % 的 T4O 进行为期八周的眼睑清洁可增加 NITBUT,且耐受性良好,但与 TTO 相比,受试者对 T4O 的偏好度较低。
{"title":"A randomized fellow-eye controlled trial study of 1% terpinen-4-ol vs 1% tea tree oil eyelid cleanser for the treatment of meibomian gland dysfunction","authors":"Preamjit Saonanon ,&nbsp;Wannit Witoonchart ,&nbsp;Dusadee Charnvanich ,&nbsp;Somphob Thompho ,&nbsp;Kittichai Chaiseeda ,&nbsp;Yuda Chongpison ,&nbsp;Vannarut Satitpitakul","doi":"10.1016/j.ajoint.2024.100052","DOIUrl":"https://doi.org/10.1016/j.ajoint.2024.100052","url":null,"abstract":"<div><h3>Purpose</h3><p>To compare the pre- and post-treatment differences in non-invasive tear breakup time (NITBUT) between a 1 % terpinen-4-ol (T4O) and a 1 % tea tree oil (TTO) eyelid cleanser</p></div><div><h3>Design</h3><p>A randomized controlled trial</p></div><div><h3>Method</h3><p>Thirty-three participants diagnosed with MGD were enrolled. All patients and the outcome evaluator were masked to the treatment allocation. One eye of each patient was randomly assigned to receive the T4O cleanser, while the fellow eye received the TTO cleanser. Baseline and eight week assessments included the collection of the ocular surface disease index (OSDI) questionnaire, NITBUT (in seconds), lipid layer thickness (in nanometers), ocular surface staining scores, and the presence of Demodex as cylindrical dandruff. The main outcome measurement was the pre- and post-treatment difference in NITBUT. Patient preference, safety, and tolerability were also assessed.</p></div><div><h3>Results</h3><p>There was a statistically significant increase in NITBUT in the T4O group (<em>n</em> = 30; 2.27 ± 1.09; 95 % confidence interval [CI]: 0.12–4.42; <em>p</em> = 0.038), but not in the TTO group (<em>n</em> = 30; 0.04 ± 1.10; 95 % CI: -2.19–2.11; <em>p</em> = 0.972). Both groups showed significant improvement in post-treatment ocular surface staining scores. Fifteen subjects (50 %) preferred TTO due to less cooling sensation experienced during application, nine (30 %) preferred T4O, and six (20 %) had no preference. No major adverse events were observed.</p></div><div><h3>Conclusion</h3><p>Eyelid cleaning with 1 % T4O in MGD for eight weeks was found to increase NITBUT with good tolerability, although it was less preferred compared to TTO.</p></div>","PeriodicalId":100071,"journal":{"name":"AJO International","volume":"1 3","pages":"Article 100052"},"PeriodicalIF":0.0,"publicationDate":"2024-07-06","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.sciencedirect.com/science/article/pii/S2950253524000522/pdfft?md5=2046fe79ed7a02ef6c7d8de683495eba&pid=1-s2.0-S2950253524000522-main.pdf","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"141605598","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 association between endophthalmitis risk following a variety of intra-ocular procedures and defensin gene profile 各种眼内手术后眼内炎风险与防御素基因图谱之间的关联
Pub Date : 2024-07-06 DOI: 10.1016/j.ajoint.2024.100034
Jonathan C. Park , Angela Zhang , Stefan DeGeus , Michael Hogden , Amanda Churchill , Niels Crama , Anneke den Hollander , Wai-Ching Lam , Peter Kertes , Robert Devenyi , Peng Yan , Efrem D Mandelcorn , Tina Felfeli , David H Steel , Richard Haynes

Objective

Endophthalmitis is a sight threatening complication of intra-ocular procedures. It has been suggested that a predisposition to endophthalmitis is linked to a specific genotype in the human beta-defensin 1 (HBD-1) gene when previously analysed in a small UK cohort undergoing cataract surgery. We sought to test out this hypothesis in a larger international case: control study (centres in UK, Netherlands and Canada) following a variety of intraocular procedures.

Design

International case: control study

Participants & Methods

660 individuals undergoing an intraocular procedure were recruited (165 cases of endophthalmitis and 495 controls). DNA was extracted. Single nucleotide polymorphisms, SNPs, rs11362, rs1800972 and rs2702877 in the HBD-1 gene were analysed (both individual genotypes and haplotypes were obtained).

Results

No associations were found when individual SNPs were analysed across the combined international cohort. However, analysis of the Toronto sub-cohort, showed a statistically significant association between the endophthalmitis cohort and the rs1800972 C allele (OR: 3.18, CI: 1.32 - 7.68, p = 0.01) and rs2702877 G allele (OR: 3.06, CI: 1.35 - 6.95, p = 0.017). No haplotype association was identified with endophthalmitis compared to control in both institution sub-cohorts and combined cohort analysis. A strong trend associating the rs1800972, rs11362 GG mini-haplotype with culture positive endophthalmitis groups across all groups was noted, but this did not reach statistical significance.

Conclusions

This is the first study to demonstrate a genetic link between a certain genetic profile (HBD-1 gene SNP variation) and endophthalmitis. Future pre-operative genetic testing could help identify patients at risk of endophthalmitis to guide invasive treatment options.

目的眼内炎是眼内手术中威胁视力的一种并发症。有人认为,眼内炎的易感性与人类 beta-defensin 1 (HBD-1) 基因的特定基因型有关。我们试图在一项更大规模的国际病例:对照研究(研究中心位于英国、荷兰和加拿大)中验证这一假设,该研究涉及各种眼内手术。提取 DNA。对 HBD-1 基因中的单核苷酸多态性(SNPs)rs11362、rs1800972 和 rs2702877 进行了分析(获得了单个基因型和单倍型)。然而,对多伦多子队列的分析表明,眼底病队列与 rs1800972 C 等位基因(OR:3.18,CI:1.32 - 7.68,p = 0.01)和 rs2702877 G 等位基因(OR:3.06,CI:1.35 - 6.95,p = 0.017)之间存在统计学意义上的显著关联。在机构子队列和合并队列分析中,与对照组相比,未发现单倍型与眼底病有关。在所有组别中,rs1800972、rs11362 GG 小型单倍型与培养阳性眼内炎组别有明显的关联趋势,但未达到统计学显著性。未来的术前基因检测可帮助识别有眼底病风险的患者,从而指导有创治疗方案的选择。
{"title":"The association between endophthalmitis risk following a variety of intra-ocular procedures and defensin gene profile","authors":"Jonathan C. Park ,&nbsp;Angela Zhang ,&nbsp;Stefan DeGeus ,&nbsp;Michael Hogden ,&nbsp;Amanda Churchill ,&nbsp;Niels Crama ,&nbsp;Anneke den Hollander ,&nbsp;Wai-Ching Lam ,&nbsp;Peter Kertes ,&nbsp;Robert Devenyi ,&nbsp;Peng Yan ,&nbsp;Efrem D Mandelcorn ,&nbsp;Tina Felfeli ,&nbsp;David H Steel ,&nbsp;Richard Haynes","doi":"10.1016/j.ajoint.2024.100034","DOIUrl":"https://doi.org/10.1016/j.ajoint.2024.100034","url":null,"abstract":"<div><h3>Objective</h3><p>Endophthalmitis is a sight threatening complication of intra-ocular procedures. It has been suggested that a predisposition to endophthalmitis is linked to a specific genotype in the human beta-defensin 1 (HBD-1) gene when previously analysed in a small UK cohort undergoing cataract surgery. We sought to test out this hypothesis in a larger international case: control study (centres in UK, Netherlands and Canada) following a variety of intraocular procedures.</p></div><div><h3>Design</h3><p>International case: control study</p></div><div><h3>Participants &amp; Methods</h3><p>660 individuals undergoing an intraocular procedure were recruited (165 cases of endophthalmitis and 495 controls). DNA was extracted. Single nucleotide polymorphisms, SNPs, rs11362, rs1800972 and rs2702877 in the HBD-1 gene were analysed (both individual genotypes and haplotypes were obtained).</p></div><div><h3>Results</h3><p>No associations were found when individual SNPs were analysed across the combined international cohort. However, analysis of the Toronto sub-cohort, showed a statistically significant association between the endophthalmitis cohort and the rs1800972 C allele (OR: 3.18, CI: 1.32 - 7.68, <em>p</em> = 0.01) and rs2702877 G allele (OR: 3.06, CI: 1.35 - 6.95, <em>p</em> = 0.017). No haplotype association was identified with endophthalmitis compared to control in both institution sub-cohorts and combined cohort analysis. A strong trend associating the rs1800972, rs11362 GG mini-haplotype with culture positive endophthalmitis groups across all groups was noted, but this did not reach statistical significance.</p></div><div><h3>Conclusions</h3><p>This is the first study to demonstrate a genetic link between a certain genetic profile (HBD-1 gene SNP variation) and endophthalmitis. Future pre-operative genetic testing could help identify patients at risk of endophthalmitis to guide invasive treatment options.</p></div>","PeriodicalId":100071,"journal":{"name":"AJO International","volume":"1 2","pages":"Article 100034"},"PeriodicalIF":0.0,"publicationDate":"2024-07-06","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.sciencedirect.com/science/article/pii/S2950253524000340/pdfft?md5=9eca9cf861048b2fccef856a3f9dba2c&pid=1-s2.0-S2950253524000340-main.pdf","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"141542906","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
Visual prognosis in retinoblastoma patients with multimodality treatments 多模式治疗视网膜母细胞瘤患者的视觉预后
Pub Date : 2024-07-06 DOI: 10.1016/j.ajoint.2024.100051
Ratima Chokchaitanasin , Nattawat Asawaworarit , Wimwipa Dieosuthichat , Suradej Hongeng , Smart Pakakasama , Usanarat Anurathapan , Duantida Songdej , Pongpak Pongphitcha , Rangsima Aroonroch , Rossukon Kaewkhaw , Ekachat Chanthanaphak , Duangnate Rojanaporn
<div><h3>Purpose</h3><div>This study evaluates factors associated with globe preservation and long-term visual outcomes according to clinical characteristics and treatment modalities in intraocular retinoblastoma patients.</div></div><div><h3>Design</h3><div>A retrospective review of medical records enrolled intraocular retinoblastoma patients between January 1, 2007, and June 30, 2020.</div></div><div><h3>Methods</h3><div><ul><li><span>•</span><span><div>Setting: Single-centered.</div></span></li><li><span>•</span><span><div>Patient or study population: Intraocular retinoblastoma patients treated at Ramathibodi Hospital, Bangkok, Thailand.</div></span></li><li><span>•</span><span><div>Main outcome measure: A statistically significant factor associated with visual prognosis corresponds to baseline characteristics, staging, anatomical involvement, and multimodal treatments.</div></span></li></ul></div></div><div><h3>Results</h3><div>86 patients (124 eyes) were included. Median age of disease bilaterality onset was significantly younger than unilaterality. Three-quarters of patients were in advanced stages (groups D or E in either ICRB or IIRC and up to cT2a from AJCC). Globe salvage rate was 54.0 % (67 eyes). ICRB Groups D-E, IIRC Groups D-E, and AJCC cT2a and more advanced stages are the globe removal-associated independent risk factors (adjusted odds ratio [AOR] [95 % CI] = 7.40 [1.36, 40.09], 8.33 [1.55, 44.73], 14.73 [1.55, 139.79], respectively). Compared to other classification, IIRC provided the highest statistical correlation from univariate analysis. IIRC Groups A-C is a good visual acuity-associated independent risk factor (AOR [95 % CI] = 4.64 [1.05, 20.43] and <em>P</em> = 0.042). Macular involvement is a worse visual acuity-associated independent risk factor (AOR [95 % CI] = 0.14 [0.02, 0.82] and <em>P</em> = 0.03). Systemic chemotherapy (86.29 %) is the mainstay treatment in our study and is the only good visual outcome-associated treatment. Subgroup analysis of all eyes receiving systemic chemotherapy reveals statistically significant preventive factors for globe removal for tumor staging with ICRB and IIRC groups A-C and AJCC stages cT1a–cT1b (odds ratio [95 % CI] = 15.75 [4.38, 56.65], 15.67 [4.34, 56.53], 9.97 [2.75, 36.18], respectively; <em>P</em>=<0.001) and prevents the worse visual outcome in these stages, (odds ratios [95 % CI] = 4.57 [1.28, 16.27], 6.61 [1.74, 25.11], and 7.50 [1.86, 30.16], respectively; <em>P</em> =< 0.05).</div></div><div><h3>Conclusions</h3><div>All recent advanced clinical stagings are globe removal outcome-associated independent risk factors. IIRC staging is the best visual results predictor. IIRC Groups A-C were associated with a good visual outcome. Macular involvement tumors are strongly associated with poor visual outcome, despite successful globe preservation from multimodality treatment. Systemic chemotherapy remains a crucial globe-saving treatment and prevents worse vision–especially in
目的本研究根据眼内视网膜母细胞瘤患者的临床特征和治疗方式,评估与球体保存和长期视觉结果相关的因素。设计回顾性审查2007年1月1日至2020年6月30日期间入组的眼内视网膜母细胞瘤患者的病历:患者或研究人群:患者或研究人群:在泰国曼谷拉玛提博迪医院接受治疗的眼内视网膜母细胞瘤患者:结果86名患者(124只眼)被纳入研究。双侧发病的中位年龄明显小于单侧。四分之三的患者处于晚期(ICRB 或 IIRC 的 D 组或 E 组,AJCC 的 cT2a 以下)。全球救治率为 54.0%(67 眼)。ICRB的D-E组、IIRC的D-E组以及AJCC的cT2a和更晚期是与摘除眼球相关的独立风险因素(调整后的几率比[AOR] [95 % CI] = 7.40 [1.36, 40.09]、8.33 [1.55, 44.73]、14.73 [1.55, 139.79])。与其他分类相比,IIRC 在单变量分析中提供了最高的统计相关性。IIRC A-C 组是一个良好的视觉盲点相关独立风险因素(AOR [95 % CI] = 4.64 [1.05, 20.43],P = 0.042)。黄斑受累是一个较差的视觉连续性相关独立危险因素(AOR [95 % CI] = 0.14 [0.02, 0.82],P = 0.03)。在我们的研究中,全身化疗(86.29%)是主要的治疗方法,也是唯一视觉效果较好的相关治疗方法。对所有接受全身化疗的眼睛进行亚组分析发现,ICRB 和 IIRC A-C 组以及 AJCC cT1a-cT1b 期的肿瘤分期对球囊摘除有统计学意义的预防因素(几率比 [95 % CI] = 15.75 [4.38, 56.65],15.67 [4.34, 56.53],9.97 [2.75, 36.18],分别;P=<0.001),并在这些分期中防止较差的视觉结果,(几率比[95 % CI] = 4.57 [1.28,16.27],6.61 [1.74,25.11],7.50 [1.86,30.16],分别;P=<0.05).结论所有最近的晚期临床分期都是全球清除结果相关的独立危险因素。IIRC分期是预测视觉结果的最佳指标。IIRC分期A-C组与良好的视觉结果相关。尽管多模式治疗成功地保留了眼球,但黄斑受累肿瘤与不良视觉预后密切相关。全身化疗仍是挽救眼球的关键治疗方法,可防止视力恶化--尤其是在ICRB和IRC A-C组以及AJCC cT1a-cT1b期。
{"title":"Visual prognosis in retinoblastoma patients with multimodality treatments","authors":"Ratima Chokchaitanasin ,&nbsp;Nattawat Asawaworarit ,&nbsp;Wimwipa Dieosuthichat ,&nbsp;Suradej Hongeng ,&nbsp;Smart Pakakasama ,&nbsp;Usanarat Anurathapan ,&nbsp;Duantida Songdej ,&nbsp;Pongpak Pongphitcha ,&nbsp;Rangsima Aroonroch ,&nbsp;Rossukon Kaewkhaw ,&nbsp;Ekachat Chanthanaphak ,&nbsp;Duangnate Rojanaporn","doi":"10.1016/j.ajoint.2024.100051","DOIUrl":"10.1016/j.ajoint.2024.100051","url":null,"abstract":"&lt;div&gt;&lt;h3&gt;Purpose&lt;/h3&gt;&lt;div&gt;This study evaluates factors associated with globe preservation and long-term visual outcomes according to clinical characteristics and treatment modalities in intraocular retinoblastoma patients.&lt;/div&gt;&lt;/div&gt;&lt;div&gt;&lt;h3&gt;Design&lt;/h3&gt;&lt;div&gt;A retrospective review of medical records enrolled intraocular retinoblastoma patients between January 1, 2007, and June 30, 2020.&lt;/div&gt;&lt;/div&gt;&lt;div&gt;&lt;h3&gt;Methods&lt;/h3&gt;&lt;div&gt;&lt;ul&gt;&lt;li&gt;&lt;span&gt;•&lt;/span&gt;&lt;span&gt;&lt;div&gt;Setting: Single-centered.&lt;/div&gt;&lt;/span&gt;&lt;/li&gt;&lt;li&gt;&lt;span&gt;•&lt;/span&gt;&lt;span&gt;&lt;div&gt;Patient or study population: Intraocular retinoblastoma patients treated at Ramathibodi Hospital, Bangkok, Thailand.&lt;/div&gt;&lt;/span&gt;&lt;/li&gt;&lt;li&gt;&lt;span&gt;•&lt;/span&gt;&lt;span&gt;&lt;div&gt;Main outcome measure: A statistically significant factor associated with visual prognosis corresponds to baseline characteristics, staging, anatomical involvement, and multimodal treatments.&lt;/div&gt;&lt;/span&gt;&lt;/li&gt;&lt;/ul&gt;&lt;/div&gt;&lt;/div&gt;&lt;div&gt;&lt;h3&gt;Results&lt;/h3&gt;&lt;div&gt;86 patients (124 eyes) were included. Median age of disease bilaterality onset was significantly younger than unilaterality. Three-quarters of patients were in advanced stages (groups D or E in either ICRB or IIRC and up to cT2a from AJCC). Globe salvage rate was 54.0 % (67 eyes). ICRB Groups D-E, IIRC Groups D-E, and AJCC cT2a and more advanced stages are the globe removal-associated independent risk factors (adjusted odds ratio [AOR] [95 % CI] = 7.40 [1.36, 40.09], 8.33 [1.55, 44.73], 14.73 [1.55, 139.79], respectively). Compared to other classification, IIRC provided the highest statistical correlation from univariate analysis. IIRC Groups A-C is a good visual acuity-associated independent risk factor (AOR [95 % CI] = 4.64 [1.05, 20.43] and &lt;em&gt;P&lt;/em&gt; = 0.042). Macular involvement is a worse visual acuity-associated independent risk factor (AOR [95 % CI] = 0.14 [0.02, 0.82] and &lt;em&gt;P&lt;/em&gt; = 0.03). Systemic chemotherapy (86.29 %) is the mainstay treatment in our study and is the only good visual outcome-associated treatment. Subgroup analysis of all eyes receiving systemic chemotherapy reveals statistically significant preventive factors for globe removal for tumor staging with ICRB and IIRC groups A-C and AJCC stages cT1a–cT1b (odds ratio [95 % CI] = 15.75 [4.38, 56.65], 15.67 [4.34, 56.53], 9.97 [2.75, 36.18], respectively; &lt;em&gt;P&lt;/em&gt;=&lt;0.001) and prevents the worse visual outcome in these stages, (odds ratios [95 % CI] = 4.57 [1.28, 16.27], 6.61 [1.74, 25.11], and 7.50 [1.86, 30.16], respectively; &lt;em&gt;P&lt;/em&gt; =&lt; 0.05).&lt;/div&gt;&lt;/div&gt;&lt;div&gt;&lt;h3&gt;Conclusions&lt;/h3&gt;&lt;div&gt;All recent advanced clinical stagings are globe removal outcome-associated independent risk factors. IIRC staging is the best visual results predictor. IIRC Groups A-C were associated with a good visual outcome. Macular involvement tumors are strongly associated with poor visual outcome, despite successful globe preservation from multimodality treatment. Systemic chemotherapy remains a crucial globe-saving treatment and prevents worse vision–especially in","PeriodicalId":100071,"journal":{"name":"AJO International","volume":"1 4","pages":"Article 100051"},"PeriodicalIF":0.0,"publicationDate":"2024-07-06","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"141707199","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
Short-term results with faricimab in neovascular age-related macular degeneration resistant to both ranibizumab and aflibercept 法尼单抗治疗对雷尼珠单抗和阿弗利贝赛普均耐药的新生血管性老年性黄斑变性的短期疗效
Pub Date : 2024-07-05 DOI: 10.1016/j.ajoint.2024.100049
Miklos Schneider , Yousif Subhi , Jakob Bjerager , Delila Hodzic-Hadzibegovic , Oliver Niels Klefter , Javad Nouri Hajari

Purpose

Some patients with neovascular age-related macular degeneration (nAMD) do not respond to anti-VEGF treatment or develop tolerance or tachyphylaxis. If optimal treatment response is lacking with a given drug, switching to another is a common practice. The aim of this study was to report short-term outcomes of treatment switch to faricimab in real-world patients with nAMD resistant to both aflibercept and ranibizumab.

Design

Single-center, retrospective real-world cohort study.

Methods

Chart-review of 13 eyes of 13 multi-switch patients using electronic injection database, electronic medical records, and optical coherence tomography (OCT) data from May–September 2023.

Results

Faricimab treatment led to absence of fluid (subretinal, or both subretinal and intraretinal fluid) in 31% of the eyes and a reduction of fluid in 77% of the eyes at 4 weeks following the switch. The best-corrected visual acuity did not change significantly. No ocular or systemic safety events were observed.

Conclusions

Switching to faricimab seems a reasonable option in patients with nAMD who were previously resistant to both aflibercept and ranibizumab treatment. We noted a high rate of morphological response and improvement in anatomical parameters, with nearly one-third of the patients achieving dry macula after just one injection. No change in BCVA was seen. No safety issues were noted. The durability of these results warrants additional investigations.

Trial registration

ClinicalTrials.gov, registration number: NCT06231121, date of registration: 21/01/2024, retrospectively registered.

目的一些新生血管性老年黄斑变性(nAMD)患者对抗血管内皮生长因子治疗无反应,或出现耐受性或过敏性反应。如果某种药物缺乏最佳治疗反应,换用另一种药物是常见的做法。本研究旨在报告对阿弗利贝西和雷尼珠单抗均耐药的 nAMD 患者改用法尼单抗治疗的短期疗效。方法使用电子注射数据库、电子病历和 2023 年 5 月至 9 月的光学相干断层扫描 (OCT) 数据,对 13 名多次换药患者的 13 只眼睛进行回顾性分析。结果换药 4 周后,31% 的眼睛没有积液(视网膜下积液或视网膜下积液和视网膜内积液),77% 的眼睛积液减少。最佳矫正视力没有明显变化。结论对于既往对阿夫利韦齐和雷尼单抗治疗耐药的nAMD患者来说,改用法尼单抗似乎是一个合理的选择。我们注意到形态学反应和解剖学参数的改善率很高,近三分之一的患者仅注射一次后就出现了干性黄斑。BCVA没有变化。未发现安全性问题。试验注册ClinicalTrials.gov,注册号:NCT06231121:NCT06231121,注册日期:2024 年 1 月 21 日,回顾性注册。
{"title":"Short-term results with faricimab in neovascular age-related macular degeneration resistant to both ranibizumab and aflibercept","authors":"Miklos Schneider ,&nbsp;Yousif Subhi ,&nbsp;Jakob Bjerager ,&nbsp;Delila Hodzic-Hadzibegovic ,&nbsp;Oliver Niels Klefter ,&nbsp;Javad Nouri Hajari","doi":"10.1016/j.ajoint.2024.100049","DOIUrl":"https://doi.org/10.1016/j.ajoint.2024.100049","url":null,"abstract":"<div><h3>Purpose</h3><p>Some patients with neovascular age-related macular degeneration (nAMD) do not respond to anti-VEGF treatment or develop tolerance or tachyphylaxis. If optimal treatment response is lacking with a given drug, switching to another is a common practice. The aim of this study was to report short-term outcomes of treatment switch to faricimab in real-world patients with nAMD resistant to both aflibercept and ranibizumab.</p></div><div><h3>Design</h3><p>Single-center, retrospective real-world cohort study.</p></div><div><h3>Methods</h3><p>Chart-review of 13 eyes of 13 multi-switch patients using electronic injection database, electronic medical records, and optical coherence tomography (OCT) data from May–September 2023.</p></div><div><h3>Results</h3><p>Faricimab treatment led to absence of fluid (subretinal, or both subretinal and intraretinal fluid) in 31% of the eyes and a reduction of fluid in 77% of the eyes at 4 weeks following the switch. The best-corrected visual acuity did not change significantly. No ocular or systemic safety events were observed.</p></div><div><h3>Conclusions</h3><p>Switching to faricimab seems a reasonable option in patients with nAMD who were previously resistant to both aflibercept and ranibizumab treatment. We noted a high rate of morphological response and improvement in anatomical parameters, with nearly one-third of the patients achieving dry macula after just one injection. No change in BCVA was seen. No safety issues were noted. The durability of these results warrants additional investigations.</p></div><div><h3>Trial registration</h3><p>ClinicalTrials.gov, registration number: NCT06231121, date of registration: 21/01/2024, retrospectively registered.</p></div>","PeriodicalId":100071,"journal":{"name":"AJO International","volume":"1 3","pages":"Article 100049"},"PeriodicalIF":0.0,"publicationDate":"2024-07-05","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.sciencedirect.com/science/article/pii/S2950253524000492/pdfft?md5=d028d60b60e49757e831eb6ae9af7a20&pid=1-s2.0-S2950253524000492-main.pdf","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"141605599","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
Rapid assessment of avoidable blindness (RAAB): A 15-year follow-up in Gegharkunik province of Armenia 可避免盲症快速评估 (RAAB):亚美尼亚盖加尔库尼克省的 15 年跟踪调查
Pub Date : 2024-06-28 DOI: 10.1016/j.ajoint.2024.100048
Aida Giloyan , Vahe Khachadourian , Tsovinar Harutyunyan , Naira Khachatryan , Varduhi Petrosyan

Purpose

This study assessed the changes in the prevalence of visual impairment (VI)/blindness, cataract surgical coverage (CSC), visual outcome after cataract surgery, and main barriers to cataract surgery among the population aged 50 years and older in Gegharkunik province of Armenia from 2003 - 4 to 2019.

Design

A cross-sectional study.

Methods

A multi-stage cluster sampling technique was used to conduct Rapid Assessment of Cataract Surgical Services (RACSS) and Rapid Assessment of Avoidable Blindness (RAAB) surveys in 2003–4 and 2019, respectively. Overall, 56 clusters (40 people in each cluster) were selected for RACSS, and 24 clusters (50 people in each cluster) for RAAB. Both surveys collected data on participants’ demographics, presenting/pinhole visual acuity (PVA/PinVA), the principal cause of VI, CSC, and barriers to cataract surgery.

Results

Overall, 1,787 people participated in RACSS and 1,099 people in RAAB. Between 2003–4 and 2019, age- and sex-adjusted prevalence of bilateral blindness decreased from 2.0 % to 1.4 % while severe VI decreased from 5.2 % to 2.2 % and moderate VI from 16.0 % to 7.3 %. Untreated cataract contributed to 71.9 % and 40.9 % of all blindness cases in 2003–4 and 2019, respectively. The CSC at the threshold <6/18 improved from 23.7 % in 2003–4 to 64.8 % in 2019. Both surveys mentioned the cost as the most common barrier to cataract surgery.

Conclusion

Despite substantial improvements, cataract remained the main cause of blindness in the province. Increasing financial coverage of cataract surgeries and improving the quality of surgeries could increase their utilization of services and help improve visual outcomes in the population.

目的本研究评估了2003-2004年至2019年期间亚美尼亚盖加尔库尼克省50岁及以上人口视力损伤(VI)/失明患病率、白内障手术覆盖率(CSC)、白内障手术后视觉效果以及白内障手术主要障碍的变化情况。方法 采用多阶段群组抽样技术,分别在 2003-4 年和 2019 年进行了白内障手术服务快速评估(RACSS)和可避免失明快速评估(RAAB)调查。白内障手术服务快速评估调查共选取了 56 个群组(每个群组 40 人),可避免盲疾快速评估调查共选取了 24 个群组(每个群组 50 人)。这两项调查均收集了参与者的人口统计数据、现视/针孔视力(PVA/PinVA)、VI 的主要原因、CSC 和白内障手术障碍等方面的数据。结果共有 1787 人参加了 RACSS,1099 人参加了 RAAB。2003-4 年至 2019 年期间,经年龄和性别调整后,双侧失明率从 2.0% 降至 1.4%,重度视力障碍从 5.2% 降至 2.2%,中度视力障碍从 16.0% 降至 7.3%。在 2003-4 年和 2019 年,未经治疗的白内障分别占所有失明病例的 71.9 % 和 40.9 %。达到 <6/18 临界值的 CSC 从 2003-4 年的 23.7% 提高到 2019 年的 64.8%。两项调查均提到,费用是白内障手术最常见的障碍。扩大白内障手术的财政覆盖面并提高手术质量,可以提高白内障手术的利用率,有助于改善居民的视觉效果。
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引用次数: 0
Teprotumumab-associated aural fullness and ageusia: A case report of persistent complications following thyroid eye disease treatment 特普鲁单抗相关的耳廓饱满和老年性耳聋:甲状腺眼病治疗后持续并发症的病例报告
Pub Date : 2024-06-27 DOI: 10.1016/j.ajoint.2024.100047
David A. Hsiou, Jesse A. Terrell, Ryan P. Nolan, Lucy I. Mudie, Michael T. Yen MD

Purpose

As an inhibitor of the insulin-like growth factor-1 (IGF-1) receptor, teprotumumab is utilized in the treatment of thyroid eye disease (TED) in which there is overexpression of IGF-1 receptors by orbital fibroblasts. Clinical trials and additional studies have brought the otologic side effects of the drug to attention. Discontinuing the drug leads to resolution in some patients, whereas others report chronic symptoms, including hearing loss. Gustatory side effects have not been elucidated to the same degree, and there are no specific recommendations on how to screen for or address otologic or gustatory complications of teprotumumab. This case report details the course of a patient with prolonged aural fullness and ageusia following teprotumumab treatment.

Design and Methods

Case report.

Results

A 71-year-old woman presenting with thyroid eye disease underwent seven cycles of teprotumumab infusion therapy. Her symptom profile, including aural fullness and ageusia, led to treatment cessation prior to completion of her final infusion. Following this treatment course, her thyroid eye disease clinical activity score improved and almost all her medication-associated adverse effects resolved.

Conclusion

This report highlights the first documented case of ageusia, and an unusually protracted course of aural fullness, with teprotumumab use. The case affirms the value of otologic and gustatory screening and continued evaluations before, during, and after receiving teprotumumab, and further characterizes aural fullness and ageusia as potential lasting consequences of therapy.

目的 作为胰岛素样生长因子-1(IGF-1)受体的抑制剂,特普鲁单抗被用于治疗眼眶成纤维细胞过度表达 IGF-1 受体的甲状腺眼病(TED)。临床试验和其他研究使人们开始关注该药物对耳部的副作用。一些患者停药后症状缓解,而另一些患者则出现慢性症状,包括听力下降。食欲方面的副作用尚未得到同样程度的阐明,目前也没有关于如何筛查或解决特普鲁单抗耳科或食欲方面并发症的具体建议。本病例报告详细描述了一名患者在接受替普鲁单抗治疗后出现长期耳部饱胀和老年性耳聋的病程。她的症状包括耳部饱胀和老年痴呆,因此在完成最后一次输注前停止了治疗。本报告重点介绍了第一例有记录的老年性耳鸣病例,以及使用替普鲁单抗后异常持久的耳部饱胀病程。该病例肯定了耳科和味觉筛查的价值,以及在接受泰普单抗治疗前、治疗期间和治疗后持续进行评估的价值,并进一步说明了听力饱胀和老年性耳鸣是治疗的潜在持久后果。
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引用次数: 0
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