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Objective: A unusual case of ocular toxoplasmosis with significant vitreomacular traction is reported. The patient improved significantly following pars plana vitrectomy combined with visual stimulation and occlusion therapy. Methods: The case of a 5-year-old girl with significant unilateral vision loss associated with vitreous condensation and macular traction is described. Results: Pars plana vitrectomy was carried out for vitreomacular traction release. This was followed by visual stimulation and occlusion therapy. Significant improvement was observed. Conclusion: Despite structural damage, the combination of properly indicated surgery and amblyopia management strategies allowed the achievement of maximum vision goals in this case, suggesting structural damage may be associated with functional amblyopia. RESUMO Objetivo: Relata-se um caso de apresentação atípica de toxoplasmose ocular, com importante tração vitreomacular. A paciente apresentou melhora significativa após vitrectomia via pars plana, com estimulação visual e oclusão. Métodos: Descreve-se o caso de uma menina de 5 anos, com importante perda de visão unilateral associada à condensação vítrea e à tração macular. Resultados: Foi realizada vitrectomia via pars plana para alívio da tração vitreomacular, seguida de estimulação visual e oclusão. Foi observada melhora significativa. Conclusão: Apesar dos danos estruturais, a combinação de cirurgia bem indicada com estratégias de tratamento da ambliopia permitiu alcançar o máximo do potencial visual nesta paciente, sugerindo que os danos estruturais podem estar associados à ambliopia funcional.

Objective: A unusual case of ocular toxoplasmosis with significant vitreomacular traction is reported. The patient improved significantly following pars plana vitrectomy combined with visual stimulation and occlusion therapy. Methods: The case of a 5-year-old girl with significant unilateral vision loss associated with vitreous condensation and macular traction is described. Results: Pars plana vitrectomy was carried out for vitreomacular traction release. This was followed by visual stimulation and occlusion therapy. Significant improvement was observed. Conclusion: Despite structural damage, the combination of properly indicated surgery and amblyopia management strategies allowed the achievement of maximum vision goals in this case, suggesting structural damage may be associated with functional amblyopia.