Ocular immunology and inflammation · 2026
Bilateral en coup de sabre with bilateral ophthalmic involvement and hearing loss: A rare case report with review of literature.
СтатьяНазвание и аннотация: Перевод пока не подготовлен
Обработка статьи · этапы, ошибки и управление
Загрузка состояния…
Ошибки и ограничения
История · было → стало
Состояние обновляется каждые 5 секунд, пока блок открыт. Процент показывается только там, где известен объём работы. Пауза останавливает продолжение этапа; текущий запрос к источнику может завершиться.
Mahal Ramneek, Verma Saurabh, Azad Shorya Vardhan, Shaikh Nawazish, Kumawat Devesh, Markan Ashish, Venkatesh Pradeep
Аннотация
PURPOSE: En coup de sabre (ECDS) is a rare craniofacial variant of linear scleroderma characterized by a band-like induration of the frontoparietal skin resembling the strike of a sabre. Bilateral involvement is exceptionally rare, and concurrent bilateral ophthalmic manifestations with mixed hearing loss (sensorineural and conductive) have not been previously described.
METHOD: We report a case of bilateral ECDS presenting with bilateral periorbital involvement including madarosis and adnexal skin atrophy, along with bilateral hearing loss and retinal ischemia resulting in extensive neovascularization.
RESULT: A structured review of reported ophthalmic manifestations in linear scleroderma and ECDS is presented. The simultaneous occurrence of bilateral ocular adnexal involvement, retinal ischemia causing tractional retinal detachment (TRD), secondary Coats-like features, and bilateral mixed hearing loss in bilateral ECDS is a previously unreported combination that widens the phenotypic spectrum of this condition.
CONCLUSION: This case underscores the need for comprehensive ophthalmic and audiological surveillance in all patients with ECDS, particularly those with bilateral or extensive craniofacial disease.
METHOD: We report a case of bilateral ECDS presenting with bilateral periorbital involvement including madarosis and adnexal skin atrophy, along with bilateral hearing loss and retinal ischemia resulting in extensive neovascularization.
RESULT: A structured review of reported ophthalmic manifestations in linear scleroderma and ECDS is presented. The simultaneous occurrence of bilateral ocular adnexal involvement, retinal ischemia causing tractional retinal detachment (TRD), secondary Coats-like features, and bilateral mixed hearing loss in bilateral ECDS is a previously unreported combination that widens the phenotypic spectrum of this condition.
CONCLUSION: This case underscores the need for comprehensive ophthalmic and audiological surveillance in all patients with ECDS, particularly those with bilateral or extensive craniofacial disease.