RESUMO
Invasive fungal sinusitis causes painful orbital apex syndrome with ophthalmoplegia and visual loss; the mechanism is unclear. We report an immunocompromised patient with invasive fungal sinusitis in whom the visual loss was due to posterior ischaemic optic neuropathy, shown on diffusion-weighted MRI, presumably from fungal invasion of small meningeal-based arteries at the orbital apex. After intensive antifungal drugs, orbital exenteration and immune reconstitution, the patient survived, but we were uncertain if the exenteration helped. We suggest that evidence of acute posterior ischaemic optic neuropathy should be a contra-indication to the need for orbital exenteration in invasive fungal sinusitis.
Assuntos
Fendas de Ciclodiálise/cirurgia , Oftalmia Simpática/etiologia , Procedimentos Cirúrgicos Oftalmológicos/efeitos adversos , Adulto , Fendas de Ciclodiálise/diagnóstico por imagem , Angiofluoresceinografia , Glucocorticoides/uso terapêutico , Humanos , Masculino , Microscopia Acústica , Oftalmia Simpática/diagnóstico , Oftalmia Simpática/tratamento farmacológico , Prednisolona/uso terapêutico , Tomografia de Coerência ÓpticaRESUMO
PURPOSE: To present the first reported case of presumptive intraocular recurrence of lymphoma following Chimeric Antigen Receptor (CAR) T-cell therapy despite systemic control by CD19-CAR T cells. METHODS: Observational case report. RESULTS: A 59-year-old man with diffuse, large, B-cell lymphoma subsequently developed secondary central nervous system disease despite chemotherapy. He underwent stem cell transplantation but relapsed again and was scheduled to receive CAR T-cell therapy. He developed vitritis several weeks before treatment, with vitreous biopsy showing non-Hodgkin B-cell lymphoma. He received CAR T-cell therapy following the vitrectomy. He presented 3 months following CAR T-cell therapy with nonspecific right eye floaters and discomfort, with the optical coherence tomography revealing subretinal saw-tooth deposits in the right eye, highly suggestive of lymphoma. This is despite having good systemic control with no other disease elsewhere in the body. He received intravitreal methotrexate to good effect. CONCLUSION: To our knowledge, this is the first case of a vitreoretinal lymphoma nonresponsive to CAR T-cell therapy, despite good central nervous system and systemic control. This is suggestive of anti-CD19 CAR T cells not trafficking into the eye in sufficient numbers to eliminate CD19-expressing neoplastic B cells. We suggest regular ophthalmic follow-up after CAR-T-cell therapy for patients where there is evidence of ocular involvement.
Assuntos
Neoplasias do Sistema Nervoso Central , Neoplasias Oculares , Linfoma Intraocular , Linfoma Difuso de Grandes Células B , Receptores de Antígenos Quiméricos , Neoplasias da Retina , Masculino , Humanos , Pessoa de Meia-Idade , Linfoma Intraocular/diagnóstico , Linfoma Intraocular/terapia , Receptores de Antígenos Quiméricos/uso terapêutico , Imunoterapia Adotiva , Neoplasias da Retina/diagnóstico , Neoplasias da Retina/terapia , Corpo Vítreo , Neoplasias Oculares/terapia , Proteínas Adaptadoras de Transdução de Sinal , Terapia Baseada em Transplante de Células e TecidosAssuntos
Clozapina/efeitos adversos , Degeneração Macular/induzido quimicamente , Doenças Retinianas/induzido quimicamente , Esquizofrenia/tratamento farmacológico , Assistência ao Convalescente , Antipsicóticos/efeitos adversos , Antipsicóticos/uso terapêutico , Clozapina/administração & dosagem , Clozapina/uso terapêutico , Eletrorretinografia/métodos , Humanos , Degeneração Macular/diagnóstico por imagem , Degeneração Macular/patologia , Masculino , Pessoa de Meia-Idade , Doenças Retinianas/diagnóstico por imagem , Doenças Retinianas/patologia , Esquizofrenia/complicações , Tomografia de Coerência Óptica/métodosRESUMO
BACKGROUND: To report a case of a subretinal, unilateral, peripapillary granuloma that was diagnosed as sarcoidosis by a 27-gauge pars plana vitrectomy subretinal biopsy. Sarcoidosis is a chronic idiopathic granulomatous inflammatory disease, that has ocular involvement in 10-80% of patients. It is often mistaken for many other primary ocular diseases because the condition can involve any structure in or around the eye. Previous case reports of peripapillary sarcoidosis have either been limited to the choroid or presented with additional ocular and systemic signs, hence have not required an intraocular biopsy. CASE PRESENTATION: A 54-year-old Filipino male presented with a 6-month history of painless blurred vision in his right eye. Fundus examination revealed a large white peripapillary lesion. Enhanced-depth imaging optical coherence tomography confirmed the subretinal location of the mass. Indocyanine green angiography demonstrated absolute hypofluorescent blockage with satellite lesions. A whole-body positron emission tomography scan demonstrated widespread lymphadenopathy, but investigations including an inguinal lymph node biopsy were inconclusive. Following growth of the peripapillary lesion and worsening vision, a 27-gauge pars plana vitrectomy subretinal biopsy was performed which confirmed sarcoidosis. He was treated with oral corticosteroids and transitioned to long term immunotherapy with methotrexate. CONCLUSIONS: Sarcoidosis can present in the subretinal space, around the optic nerve without other ocular findings.
RESUMO
BACKGROUND/AIMS: Human grading of digital images from diabetic retinopathy (DR) screening programmes represents a significant challenge, due to the increasing prevalence of diabetes. We evaluate the performance of an automated artificial intelligence (AI) algorithm to triage retinal images from the English Diabetic Eye Screening Programme (DESP) into test-positive/technical failure versus test-negative, using human grading following a standard national protocol as the reference standard. METHODS: Retinal images from 30 405 consecutive screening episodes from three English DESPs were manually graded following a standard national protocol and by an automated process with machine learning enabled software, EyeArt v2.1. Screening performance (sensitivity, specificity) and diagnostic accuracy (95% CIs) were determined using human grades as the reference standard. RESULTS: Sensitivity (95% CIs) of EyeArt was 95.7% (94.8% to 96.5%) for referable retinopathy (human graded ungradable, referable maculopathy, moderate-to-severe non-proliferative or proliferative). This comprises sensitivities of 98.3% (97.3% to 98.9%) for mild-to-moderate non-proliferative retinopathy with referable maculopathy, 100% (98.7%,100%) for moderate-to-severe non-proliferative retinopathy and 100% (97.9%,100%) for proliferative disease. EyeArt agreed with the human grade of no retinopathy (specificity) in 68% (67% to 69%), with a specificity of 54.0% (53.4% to 54.5%) when combined with non-referable retinopathy. CONCLUSION: The algorithm demonstrated safe levels of sensitivity for high-risk retinopathy in a real-world screening service, with specificity that could halve the workload for human graders. AI machine learning and deep learning algorithms such as this can provide clinically equivalent, rapid detection of retinopathy, particularly in settings where a trained workforce is unavailable or where large-scale and rapid results are needed.