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Geographic atrophy (GA) remains a leading cause of central vision loss with no known cure. Until recently, there were no approved treatments for GA, often resulting in poor quality of life for affected patients. GA is characterized by atrophic lesions on the retina that may eventually threaten the fovea. Emerging treatments have demonstrated the ability to reduce the rate of lesion growth, potentially preserving visual function. Avacincaptad pegol (ACP; Astellas Pharma Inc), a complement component 5 inhibitor, is an FDA-approved treatment for GA that has been evaluated in numerous clinical trials. Here we review the current clinical trial landscape of ACP, including critical post hoc analyses that suggest ACP may reduce the risk of severe loss among patients with GA.
Geographic atrophy (GA) is an advanced form of eye disease age-related macular degeneration. In people with GA, light-sensitive cells at the back of the eye (the retina) start to die, forming lesions. GA lesions usually get bigger over time and can lead to blindness. New medicines are being studied that work by slowing the growth of GA lesions. Avacincaptad pegol (ACP) is one medicine that acts on the immune system and is designed to block the C5 protein, helping stop the immune system from attacking cells in the retina. Based on clinical studies, ACP was shown to slow the growth of GA over time and has been approved by the FDA. This review article summarizes research on ACP.
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PURPOSE: To evaluate the impact of reduction in geographic atrophy (GA) lesion growth on visual acuity in the GATHER trials using categorical outcome measures. DESIGN: Randomized, double-masked, sham-controlled phase 3 trials. PARTICIPANTS: Aged ≥50 years with noncenter point-involving GA and best-corrected visual acuity (BCVA) of 25 to 80 ETDRS letters in the study eye. METHODS: GATHER1 consisted of 2 parts. In part 1, 77 patients were randomized 1:1:1 to avacincaptad pegol (ACP) 1 mg, ACP 2 mg, and sham. In part 2, 209 patients were randomized 1:2:2 to ACP 2 mg, ACP 4 mg, and sham. In GATHER2, patients were randomized 1:1 to ACP 2 mg (n = 225) and sham (n = 223). A post hoc analysis of 12-month data for pooled ACP 2 mg and sham groups is reported. MAIN OUTCOME MEASURES: Proportion of study eyes that experienced ≥10-, ≥15-, or ≥20-BCVA ETDRS letter loss from baseline to month 12; time-to-event analysis of persistent vision loss of ≥10, ≥15, or≥ 20 BCVA letters from baseline at ≥2 consecutive visits over 12 months; proportion of study eyes with BCVA loss to a level below driving eligibility threshold at month 12 among those eligible to drive at baseline. RESULTS: Lower proportions of study eyes experienced ≥10-, ≥15-, or ≥20-BCVA letter loss from baseline over 12 months with ACP 2 mg (11.6%, 4.0%, and 1.6%, respectively) versus sham (14.1%, 7.6%, and 4.5%, respectively). There was a reduction in the risk of persistent loss of ≥15 BCVA ETDRS letters with ACP 2 mg (3.4%) versus sham (7.8%) through 12 months. A lower proportion of study eyes treated with ACP 2 mg reached the threshold for driving ineligibility versus sham by 12 months. CONCLUSIONS: Treatment with ACP 2 mg delayed the risk of progression to persistent vision loss (i.e., ≥10-, ≥15-, and ≥20-BCVA letter loss or BCVA loss to a level below driving eligibility threshold) versus sham over 12 months. FINANCIAL DISCLOSURE(S): Proprietary or commercial disclosure may be found in the Footnotes and Disclosures at the end of this article.
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PURPOSE: Disparities in clinical trials are a major problem because of significant underrepresentation of certain gender, racial, and ethnic groups. Several factors including stringent eligibility criteria and recruitment strategies hinder our understanding of retinal disease. Thus, we aimed to study the various reasons of screen failures and specific patient and study characteristics among screen failures. DESIGN: This is a cross-sectional retrospective study. METHODS: Screening data of 87 trials from 6 centers were analyzed. Study characteristics (disease studied, phase of trial, and route of drug administration) and patient demographics (age, gender, race, ethnicity, and employment status) were compared among different causes of screen failures. Screen failures were broadly classified into 6 categories: exclusion because of vision-based criteria, exclusion because of imaging findings, exclusion because of other factors, patient-related criteria, physician-related criteria, and miscellaneous. Descriptive statistics, Pearson chi-square test, and analysis of variance were used for statistical analysis. MAIN OUTCOME MEASURES: Prevalence of various reasons for screen failures in multiple trials and its trend among different study and patient characteristics. RESULTS: Among 87 trials and 962 patients, 465 (48.2%) patients were successfully randomized and 497 (51.8%) patients were classified as screen failures. The trials were conducted for various retinal diseases. Mean age was 76.50 ±10.45 years and 59.4% were females. Predominantly White patients (93.4%) and unemployed/retired patients (66.6%) were screened. Of the 497 screen failures, most were because of patients not meeting inclusion criteria of imaging findings (n = 221 [44.5%]) followed by inclusion of vision-based criteria (n = 73 [14.7%]), exclusion because of other factors (n = 75 [15.1%]), patient-related (n = 34 [6.8%]), physician-related (n = 28 [5.6%]), and miscellaneous reasons (n = 39 [7.8%]). Reason for screen failure was not available for 27 (5.4%) patients. A higher proportion of patients screened for surgical trials (15%) declined to participate in the study compared with noninvasive trials involving topical drugs and photobiomodulation (0%) (P = 0.02). CONCLUSIONS: Patients not meeting the imaging and vision-cased criteria were the most common reasons for screen failures. White patients and unemployed patients predominantly participated in clinical trials. Patients are more inclined to continue participation in noninvasive clinical trials compared with surgical trials. Better recruitment strategies and careful consideration of study criteria can aid in decreasing the rate of screen failures.
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PURPOSE: To evaluate the 24-week efficacy and safety of the dual angiopoietin-2 (Ang-2) and vascular endothelial growth factor (VEGF)-A inhibitor faricimab versus aflibercept in patients with vein occlusion. DESIGN: Phase 3, global, randomized, double-masked, active comparator-controlled trials: BALATON/COMINO (ClincalTrials.gov identifiers: NCT04740905/NCT04740931; sites: 149/192). PARTICIPANTS: Patients with treatment-naïve foveal center-involved macular edema resulting from branch (BALATON) or central or hemiretinal (COMINO) RVO. METHODS: Patients were randomized 1:1 to faricimab 6.0 mg or aflibercept 2.0 mg every 4 weeks for 24 weeks. MAIN OUTCOME MEASURES: Primary end point: change in best-corrected visual acuity (BCVA) from baseline to week 24. Efficacy analyses included patients in the intention-to-treat population. Safety analyses included patients who received ≥ 1 doses of study drug. RESULTS: Enrollment: BALATON, n = 553; COMINO, n = 729. The BCVA gains from the baseline to week 24 with faricimab were noninferior versus aflibercept in BALATON (adjusted mean change, +16.9 letters [95.03% confidence interval (CI), 15.7-18.1 letters] vs. +17.5 letters [95.03% CI, 16.3-18.6 letters]) and COMINO (+16.9 letters [95.03% CI, 15.4-18.3 letters] vs. +17.3 letters [95.03% CI, 15.9-18.8 letters]). Adjusted mean central subfield thickness reductions from the baseline were comparable for faricimab and aflibercept at week 24 in BALATON (-311.4 µm [95.03% CI, -316.4 to -306.4 µm] and -304.4 µm [95.03% CI, -309.3 to -299.4 µm]) and COMINO (-461.6 µm [95.03% CI, -471.4 to -451.9 µm] and -448.8 µm [95.03% CI, -458.6 to -439.0 µm]). A greater proportion of patients in the faricimab versus aflibercept arm achieved absence of fluorescein angiography-based macular leakage at week 24 in BALATON (33.6% vs. 21.0%; nominal P = 0.0023) and COMINO (44.4% vs. 30.0%; nominal P = 0.0002). Faricimab was well tolerated, with an acceptable safety profile comparable with aflibercept. The incidence of ocular adverse events was similar between patients receiving faricimab (16.3% [n = 45] and 23.0% [n = 84] in BALATON and COMINO, respectively) and aflibercept (20.4% [n = 56] and 27.7% [n = 100], respectively). CONCLUSIONS: These findings demonstrate the efficacy and safety of faricimab, a dual Ang-2/VEGF-A inhibitor, in patients with macular edema secondary to retinal vein occlusion. FINANCIAL DISCLOSURE(S): Proprietary or commercial disclosure may be found in the Footnotes and Disclosures at the end of this article.
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Inibidores da Angiogênese , Injeções Intravítreas , Edema Macular , Receptores de Fatores de Crescimento do Endotélio Vascular , Proteínas Recombinantes de Fusão , Oclusão da Veia Retiniana , Tomografia de Coerência Óptica , Fator A de Crescimento do Endotélio Vascular , Acuidade Visual , Humanos , Edema Macular/tratamento farmacológico , Edema Macular/etiologia , Edema Macular/fisiopatologia , Edema Macular/diagnóstico , Oclusão da Veia Retiniana/tratamento farmacológico , Oclusão da Veia Retiniana/complicações , Oclusão da Veia Retiniana/diagnóstico , Oclusão da Veia Retiniana/fisiopatologia , Acuidade Visual/fisiologia , Proteínas Recombinantes de Fusão/uso terapêutico , Proteínas Recombinantes de Fusão/administração & dosagem , Proteínas Recombinantes de Fusão/efeitos adversos , Receptores de Fatores de Crescimento do Endotélio Vascular/antagonistas & inibidores , Receptores de Fatores de Crescimento do Endotélio Vascular/uso terapêutico , Masculino , Feminino , Método Duplo-Cego , Inibidores da Angiogênese/uso terapêutico , Inibidores da Angiogênese/efeitos adversos , Fator A de Crescimento do Endotélio Vascular/antagonistas & inibidores , Pessoa de Meia-Idade , Idoso , Resultado do Tratamento , Angiopoietina-2/antagonistas & inibidoresRESUMO
Background: Gene therapy, successfully used in rare, monogenetic disorders, may prove to be a durable management approach for common, polygenetic conditions, including neovascular age-related macular degeneration (nAMD). Repeated injections, oftentimes monthly, and possibly for decades, of vascular endothelial growth factor antagonists (anti-VEGF), is the standard for nAMD. We hypothesised that an in-office, intravitreal administration of ixoberogene soroparvovec (ixo-vec, formerly ADVM-022), a single-dose gene therapy encoding for the proven anti-VEGF protein, aflibercept, would transform retinal cells to continually produce aflibercept to minimise treatment burden in nAMD. Methods: In this two-year, open-label, prospective, multicentre phase 1 study, patients with nAMD responding to anti-VEGF were assigned to four cohorts differing by ixo-vec dose (2 × 1011 vs 6 × 1011 vector genomes (vg/eye)) and prophylactic steroids (oral prednisone vs topical difluprednate). The primary outcome was the type, severity, and incidence of ocular and systemic adverse events (AEs); secondary endpoints included vision, central subfield thickness (CST), and the number of supplemental injections. This study was registered with ClinicalTrials.gov, NCT03748784. Findings: Thirty patients with nAMD were enrolled between November 14, 2018 and June 30, 2020 at nine study sites in the United States. No systemic ixo-vec related AEs were noted. Across both dose groups the most common adverse event was anterior chamber cell, which was reported in 11 participants in the 6 × 1011 dose group and in 7 participants in the 2 × 1011 dose group; intraocular inflammation was responsive to topical corticosteroids, with no anterior chamber cells or vitreous cells observed in 2 × 1011 vg/eye patients at the end of the study. Vision and CST remained stable throughout two years with annualised anti-VEGF injections reduced by 80% (10.0 mean annualised anti-VEGF injections to 1.9) in 2 × 1011 vg/eye and 98% (9.8 mean annualised anti-VEGF injections to 0.2) in 6 × 1011 vg/eye cohorts. Interpretation: Ixo-vec was generally well-tolerated, maintained vision, and improved anatomical outcomes in nAMD, with a substantial reduction in anti-VEGF injections. A single administration of an in-office gene therapy, with vectorised protein with an already established clinical benefit, has the potential to revolutionise the management of common ocular disorders requiring ongoing, frequent therapeutic interventions. Funding: Adverum Biotechnologies.
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Purpose: Dual inhibition of angiopoietin-2 and VEGF-A with faricimab (Vabysmo) offers excellent visual acuity gains with strong durability in patients with diabetic macular edema (ME) and neovascular age-related macular degeneration. The phase III BALATON/COMINO (NCT04740905/NCT04740931) trials will investigate the efficacy, safety, and durability of faricimab in patients with ME due to retinal vein occlusion (RVO). Design: Two identically designed global, randomized, double-masked, active comparator-controlled studies. Participants: Anti-VEGF treatment-naive patients with branch, central, or hemiretinal RVO. Methods: Patients were randomized to 6 monthly injections of faricimab 6.0 mg or aflibercept 2.0 mg. From weeks 24 to 72, all patients received faricimab 6.0 mg administered in up to 16-week intervals using an automated treatment algorithm to generate a treat-and-extend-based personalized treatment interval dosing regimen. Personalized treatment interval adjustments were based on changes in central subfield thickness (CST) and best-corrected visual acuity (BCVA). Main Outcome Measures: Primary end point was noninferiority of faricimab versus aflibercept in mean change from baseline in BCVA (week 24; noninferiority margin: 4 letters). Secondary end points (weeks 0-24) were mean change from baseline in BCVA, CST, and National Eye Institute Visual Function Questionnaire 25 composite score; proportion of patients gaining or avoiding loss of ≥ 15/≥ 10/≥ 5/> 0 letters. Secondary end points (weeks 24-72) were treatment durability (week 68); continuation of weeks 0 to 24 end points. Ocular/nonocular adverse events will be assessed. Results: In total, 1282 patients across 22 countries were enrolled (BALATON, 553 patients, 149 centers; COMINO, 729 patients, 193 centers). Conclusions: Using a novel automated interval algorithm, BALATON/COMINO will evaluate the efficacy and safety of faricimab for ME secondary to RVO and provide key insights into how to personalize treatment. Financial Disclosures: Proprietary or commercial disclosure may be found in the Footnotes and Disclosures at the end of this article.
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BACKGROUND: Geographic atrophy is an advanced form of dry age-related macular degeneration that can lead to irreversible vision loss and high burden of disease. We aimed to assess efficacy and safety of avacincaptad pegol 2 mg in reducing geographic atrophy lesion growth. METHODS: GATHER2 is a randomised, double-masked, sham-controlled, 24-month, phase 3 trial across 205 retina clinics, research hospitals, and academic institutions globally. To be eligible, patients had to be aged 50 years or older with non-centrepoint-involving geographic atrophy and best corrected visual acuity between 20/25 and 20/320 in the study eye. Eligible patients were randomly assigned (1:1) to monthly avacincaptad pegol 2 mg administered as a 100 µL intravitreal injection or sham for the first 12 months. Randomisation was performed using an interactive response technology system with stratification by factors known to be of prognostic importance in age-related macular degeneration. Patients, investigators, study centre staff, sponsor personnel, and data analysts were masked to treatment allocation. The primary endpoint was geographic atrophy lesion size measured by fundus autofluorescence at baseline, month 6, and month 12. Efficacy and safety analyses were done in the modified intention-to-treat and safety populations, respectively. This trial is registered with ClinicalTrials.gov, NCT04435366. FINDINGS: Between June 22, 2020, and July 23, 2021, 1422 patients were screened for eligibility, of whom 448 were enrolled and randomly assigned to avacincaptad pegol 2 mg (n=225) or sham (n=223). One patient in the sham group did not receive study treatment and was excluded from analyses. There were 154 (68%) female patients and 71 (32%) male patients in the avacincaptad pegol 2 mg group, and 156 (70%) female patients and 66 (30%) male patients in the sham group. From baseline to month 12, the mean rate of square-root-transformed geographic atrophy area growth was 0·336 mm/year (SE 0·032) with avacincaptad pegol 2 mg and 0·392 mm/year (0·033) with sham, a difference in growth of 0·056 mm/year (95% CI 0·016-0·096; p=0·0064), representing a 14% difference between the avacincaptad pegol 2 mg group and the sham group. Ocular treatment-emergent adverse events in the study eye occurred in 110 (49%) patients in the avacincaptad pegol 2 mg group and 83 (37%) in the sham group. There were no endophthalmitis, intraocular inflammation, or ischaemic optic neuropathy events over 12 months. To month 12, macular neovascularisation in the study eye occurred in 15 (7%) patients in the avacincaptad pegol 2 mg group and nine (4%) in the sham group, with exudative macular neovascularisation occurring in 11 (5%) in the avacincaptad pegol 2 mg group and seven (3%) in the sham group. INTERPRETATION: Monthly avacincaptad pegol 2 mg was well tolerated and showed significantly slower geographic atrophy growth over 12 months than sham treatment, suggesting that avacincaptad pegol might slow disease progression and potentially change the trajectory of disease for patients with geographic atrophy. FUNDING: Iveric Bio, An Astellas Company.
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We present 2 cases of sutureless 25-gauge pars plana vitrectomy and fluid-gas exchange, in which incorrect gas concentrations likely led to elevated intraocular pressures and retrobulbar gas. Combined removal of orbital gas with anterior orbitotomy and pars plana vitrectomy was performed in the first case to address expanding intraocular and retrobulbar gas resulting from a suspected error in gas dilution. Vitreous and orbital gas removal by needling was effective in the second case. In patients with elevated intraocular pressure and orbital gas accumulation after vitrectomy, combined intraocular and orbital decompressions were effective in optimizing clinical outcomes. There is no consensus regarding the best management of orbital gas after vitrectomy. We propose that a multidisciplinary technique should be considered, when available.
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BACKGROUND/OBJECTIVE: Investigate real-world patients receiving faricimab for the treatment of neovascular age-related macular degeneration (nAMD). SUBJECTS/METHODS: Multicenter, retrospective chart review was conducted on patients treated with faricimab for nAMD from February 2022 to September 2022. Collected data includes background demographics, treatment history, best-corrected visual acuity (BCVA), anatomic changes, and adverse events as safety markers. The main outcome measures are changes in BCVA, changes in central subfield thickness (CST) and adverse events. Secondary outcome measures included treatment intervals and presence of retinal fluid. RESULTS: After one injection of faricimab, all eyes (n = 376), previously-treated (n = 337) and treatment-naïve (n = 39) eyes demonstrated a + 1.1 letter (p = 0.035), a + 0.7 letter (p = 0.196) and a + 4.9 letter (p = 0.076) improvement in BCVA, respectively, and a - 31.3 µM (p < 0.001), a - 25.3 µM (p < 0.001) and a - 84.5 µM (p < 0.001) reduction in CST, respectively. After three injections of faricimab, all eyes (n = 94), previously-treated (n = 81) and treatment-naïve (n = 13) eyes demonstrated a + 3.4 letter (p = 0.03), a + 2.7 letter (p = 0.045) and a + 8.1 letter (p = 0.437) improvement in BCVA, and a - 43.4 µM (p < 0.001), a - 38.1 µM (p < 0.001) and a - 80.1 µM (p < 0.204) reduction in CST, respectively. One case of intraocular inflammation was observed after four injections of faricimab and resolved with topical steroids. One case of infectious endophthalmitis was treated with intravitreal antibiotics and resolved. CONCLUSIONS: Faricimab has demonstrated improvement or maintenance of visual acuity for patients with nAMD, along with rapid improvement of anatomical parameters. It has been well-tolerated with low incidence of treatable intraocular inflammation. Future data will continue to investigate faricimab for real-world patients with nAMD.
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Inibidores da Angiogênese , Degeneração Macular , Humanos , Inibidores da Angiogênese/efeitos adversos , Estudos Retrospectivos , Resultado do Tratamento , Injeções Intravítreas , Degeneração Macular/tratamento farmacológico , InflamaçãoRESUMO
Gene therapies aim to deliver a therapeutic payload to specified tissues with underlying protein deficiency. Since the 1990s, gene therapies have been explored as potential treatments for chronic conditions requiring lifetime care and medical management. Ocular gene therapies target a range of ocular disorders, but retinal diseases are of particular importance due to the prevalence of retinal disease and the current treatment burden of such diseases on affected patients, as well as the challenge of properly delivering these therapies to the target tissue. The purpose of this review is to provide an update on the most current data available for five different retinal gene therapies currently undergoing clinical trials for use against age-related macular degeneration (AMD) and the development of novel delivery routes for the administration of such therapies. Research has been performed and compiled from PubMed and the select authors of this manuscript on the treatment and effectiveness of five current retinal gene therapies: Luxturna, ADVM-022, RGX-314, GT-005, and HMR59. We present the available data of current clinical trials for the treatment of neovascular and dry age-related macular degeneration with different AAV-based gene therapies. We also present current research on the progress of developing novel routes of administration for ocular gene therapies. Retinal gene therapies offer the potential for life-changing treatment for chronic conditions like age-related macular degeneration with a single administration. In doing so, gene therapies change the landscape of treatment options for these chronic conditions for both patient and provider.
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Atrofia Geográfica , Degeneração Macular , Terapia Genética , Humanos , Degeneração Macular/tratamento farmacológico , Degeneração Macular/terapiaRESUMO
The Ang/Tie2 pathway complements VEGF-mediated activity in retinal vascular diseases such as DME, AMD, and RVO by decreasing vascular integrity, increasing neovascularization, and increasing inflammatory signaling. Faricimab is a bispecific antibody that has been developed as an inhibitor of both VEGF and Ang2 that has shown positive results in phase I, II and III trials. Recent Year 1 data from phase III clinical trials YOSEMITE, RHINE, TENAYA, and LUCERNE have confirmed the efficacy, safety, durability, and superiority of faricimab in patients with DME and nAMD. Faricimab, if approved, may significantly decrease treatment burden in patients with retinal vascular diseases to a greater extent than would current standard of care anti-VEGF injections.
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INTRODUCTION: Retinal disease treatment delivery is mostly limited to intravitreal injections and slow-release injectable implants due to structural barriers in the eye, and carry associated adverse effects and relatively high treatment burden. The Port Delivery System with ranibizumab (PDS) is a novel drug delivery device that is surgically implanted into the vitreous cavity and allows for continuous release of the anti-vascular endothelial growth factor (anti-VEGF) ranibizumab, eliminating the need for frequent intravitreal injections while maintaining therapeutic intraocular drug levels to control disease activity. Investigations of PDS are summarized in this review. AREAS COVERED: The most recent reported findings from preliminary studies and phase I-III trials are reviewed. We discuss the ramifications of these studies and the future potential for PDS in the treatment of retinal diseases. EXPERT OPINION: PDS is a novel drug delivery platform for the treatment of retinal diseases. Currently, the data from the PDS has shown promising efficacy and ability to substantially mitigate treatment burden while effectively generating visual and anatomic outcomes similar to those in patients receiving the standard monthly ranibizumab for neovascular age-related macular degeneration. Further studies are ongoing to investigate this novel drug delivery system in other disease states.
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Preparações Farmacêuticas , Doenças Retinianas , Inibidores da Angiogênese , Sistemas de Liberação de Medicamentos , Humanos , Injeções Intravítreas , Ranibizumab , Doenças Retinianas/tratamento farmacológicoAssuntos
Tumor Carcinoide/secundário , Neoplasias da Coroide/secundário , Neoplasias Pulmonares/patologia , Idoso , Antígenos de Neoplasias/análise , Biomarcadores Tumorais/análise , Biópsia por Agulha , Braquiterapia , Tumor Carcinoide/diagnóstico por imagem , Tumor Carcinoide/radioterapia , Neoplasias da Coroide/diagnóstico por imagem , Neoplasias da Coroide/radioterapia , Enucleação Ocular , Evolução Fatal , Feminino , Humanos , Neoplasias Hepáticas/secundário , Neoplasias Pulmonares/diagnóstico por imagem , Neoplasias Pulmonares/radioterapia , Pessoa de Meia-Idade , Proteínas de Neoplasias/análise , UltrassonografiaRESUMO
PURPOSE: To report the effect of massive intraocular hemorrhage on magnetic resonance imaging signal. METHODS: Case report. RESULTS: A 34-year-old power weightlifter developed sudden, nontraumatic loss of vision to no light perception in his right eye. Magnetic resonance imaging (MRI) revealed homogeneous bright signal on T1-weighted images and dark signal on T2-weighted images, opposite the signal of his normal left eye. These findings were suggestive of subacute massive intraocular hemorrhage. The patient elected enucleation for the blind, painful eye. The diagnosis made by MRI signal interpretation was confirmed by subsequent histopathology analysis. CONCLUSION: Based on hemoglobin degradation products, MRI can assist in judging the stage of blood breakdown and diagnosing intraocular pathology.
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A 3-year-old boy was referred because of the presence of a non-pigmented, highly vascular mass with tapioca appearance involving the superior portion of the left iris. Iris fluorescein angiography revealed early hyperfluorescence of the iris tumor with diffuse, intense late leakage of dye throughout the entire iris, not just in the region of the tumor. Cytopathologic examination revealed Touton giant cells and the presence of histiocytes, confirming the diagnosis of iris juvenile xanthogranuloma. The patient was prescribed topical prednisolone acetate, and the lesion resolved during the next 3 months. Fluorescein angiography may be useful in differentiating iris juvenile xanthogranuloma from malignant or non-inflammatory benign iris lesions.
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Angiofluoresceinografia , Doenças da Íris/diagnóstico , Xantogranuloma Juvenil/diagnóstico , Pré-Escolar , Diagnóstico Diferencial , Glucocorticoides/uso terapêutico , Humanos , Iris/irrigação sanguínea , Doenças da Íris/tratamento farmacológico , Masculino , Prednisolona/análogos & derivados , Prednisolona/uso terapêutico , Xantogranuloma Juvenil/tratamento farmacológicoRESUMO
CASE REPORT: A 58-year-old white female noted a lesion on her left lower eyelid, present for 3 months. Examination disclosed a dark-blue, subepithelial, smooth mass along the cilia near the punctum. The lesion did not transmit light and was thought to be a solid mass, such as a deep-blue nevus or melanoma versus a cystic mass with internal debris. Ultrasound biomicroscopy showed a fairly circumscribed echolucent mass measuring 3.2 x 3.5 mm suggestive of a cyst with numerous internal echo impedances. Following resection, histopathologic evaluation disclosed a cyst lined by a dual layer of eccrine sweat ductal epithelium, consistent with eccrine hidrocystoma. COMMENTS: Eyelid hidrocystomas can be pigmented and can be confused clinically with melanocytic tumors. Ultrasound biomicroscopy can provide helpful diagnostic information about the internal architecture of such lesions.
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Glândulas Écrinas/diagnóstico por imagem , Neoplasias Palpebrais/diagnóstico por imagem , Hidrocistoma/diagnóstico por imagem , Microscopia Acústica/métodos , Neoplasias das Glândulas Sudoríparas/diagnóstico por imagem , Diagnóstico Diferencial , Glândulas Écrinas/patologia , Glândulas Écrinas/cirurgia , Neoplasias Palpebrais/patologia , Neoplasias Palpebrais/cirurgia , Feminino , Seguimentos , Hidrocistoma/patologia , Hidrocistoma/cirurgia , Humanos , Pessoa de Meia-Idade , Neoplasias das Glândulas Sudoríparas/patologia , Neoplasias das Glândulas Sudoríparas/cirurgiaRESUMO
PURPOSE: To describe an intraocular lacrimal gland choristoma confirmed with fine-needle aspiration biopsy in a child. METHODS: Case report. RESULTS: A 6-year-old Asian girl developed a pink, vascularized, cerebriform mass with marked corectopia in the inferonasal iris of the left eye. The differential diagnosis included juvenile xanthogranuloma, nevus, melanoma, medulloepithelioma, lacrimal gland choristoma, and atypical retinoblastoma. Fluorescein angiography was suggestive of a noninflammatory, nonleaking solid mass. Fine-needle aspiration biopsy showed benign epithelial cells consistent with lacrimal tissue. CONCLUSION: Lacrimal gland choristoma shows notable clinical features and fine-needle aspiration biopsy can be confirmatory.
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Doenças da Coroide/patologia , Doenças da Íris/patologia , Aparelho Lacrimal , Biópsia por Agulha Fina , Criança , Diagnóstico Diferencial , Feminino , Angiofluoresceinografia , HumanosRESUMO
OBJECTIVE: To evaluate the technique and complications of fine-needle aspiration biopsy (FNAB) for iris tumors. DESIGN: Retrospective, nonrandomized, single-center case series. PARTICIPANTS: One hundred eyes of 100 patients with diagnostically challenging iris tumors evaluated with FNAB. MAIN OUTCOME MEASURES: Biopsy technique and complications. RESULTS: Of more than 1400 patients referred with an iris tumor over a 24-year period, 100 underwent FNAB for diagnostic purposes. The median patient age was 44 years. The median basal dimension of the lesion was 9.0 mm and median thickness was 2.5 mm. A limbal entry with transaqueous approach parallel to the iris was used in 100% of patients. Entry was from the superotemporal (52%) or inferotemporal (42%) direction. Needle gauge size was 22 (9%), 25 (55%), 27 (25%), or 30 (9%), depending on the friability and vascularity of the tumor. Adequate tumor sample was achieved in 99 eyes (99%). At the time of FNAB, the only immediate complication was partial hyphema (34%). Two weeks after FNAB, persistent hyphema was found in 6 eyes (6%), requiring surgical washout in 1 patient. There were no cases of recurrent hyphema, vitreous hemorrhage, prolonged hypotony, lens damage, endophthalmitis, or extraocular tumor seeding. At mean follow-up of 5 years, there was no incident of tumor recurrence on the iris, along the needle tract, or on the corneal or epibulbar surface. CONCLUSIONS: Using our technique, FNAB for diagnostically challenging iris tumors can achieve high yield with relatively few complications.