Sen Pradhnya, Ramawat Easha, Mohan Amit, Shah Chintan
Consultant and Head, Children Eye Care Center, Sadguru Netra Chikitsalaya, Chitrakoot, India.
Consultant, Pediatric Ophthalmology and Strabismus, Sadguru Netra Chikitsalaya, Chitrakoot, India.
Strabismus. 2019 Dec;27(4):211-214. doi: 10.1080/09273972.2019.1665690. Epub 2019 Sep 21.
Scleroderma, "en coup de sabre" is a rare disorder, characterized by linear depressed scarring at frontoparietal area of the face, seldom associated with ophthalmological findings. Extraocular muscle involvement and paralytic strabismus are rarely associated with linear scleroderma. Here we are reporting a case of unilateral linear scleroderma in an 8-year-old child, with features of strabismus fixus secondary to fibrosed medial rectus (MR) muscle, enophthalmos and en coup de saber (sword-like scar) on the eyebrow. MRI orbit was suggestive of the bulky MR and the inferior rectus muscle. MR recession along with partial loop myopexy resulted in cosmetically acceptable ocular alignment in this case. Histopathology of the portion of muscle and intermuscular septum showed diffuse fibrosis of the muscle. Linear scleroderma may be the causative etiology of strabismus fixus in our case, never reported before in literature.
硬皮病,“剑伤样”是一种罕见的疾病,其特征为面部额顶区域出现线状凹陷性瘢痕,很少伴有眼科表现。眼外肌受累和麻痹性斜视很少与线状硬皮病相关。在此,我们报告一例8岁儿童单侧线状硬皮病病例,其具有因内侧直肌纤维化继发的固定性斜视、眼球内陷以及眉部“剑伤样”(剑状瘢痕)特征。眼眶磁共振成像提示内侧直肌和下直肌增粗。在该病例中,内侧直肌后徙联合部分肌袢固定术使眼部外观上达到可接受的眼位矫正。肌肉和肌间隔部分的组织病理学显示肌肉弥漫性纤维化。在我们的病例中,线状硬皮病可能是固定性斜视的病因,这在文献中此前未曾报道过。