Department of Ophthalmology, Santa Croce e Carle Hospital, Cuneo, Italy.
Santa Croce e Carle Hospital Pharmacy, Cuneo, Italy.
Cornea. 2021 Feb 1;40(2):228-231. doi: 10.1097/ICO.0000000000002612.
This study aimed to present the efficacy and safety of cenegermin eye drop (Oxervate; Dompè Farmaceutici, Milan, Italy) treatment in a pediatric patient affected by neurotrophic keratopathy (NK) with Goldenhar syndrome.
This case reports an infant presenting ulceration and a small central opacity in the cornea of the right and left eyes, respectively. The NK bilaterally worsened despite the use of therapeutic contact lenses and temporary partial tarsorrhaphy. Magnetic resonance imaging showed absence and hypoplasia of the right and left trigeminal nerves, respectively. Cenegermin eye drops were administered 1 drop/each eye, 6 times daily for 8 weeks to promote corneal healing.
Complete healing was achieved in both eyes after treatment. During the 16-month follow-up period, no epithelial defect, recurrence, or complications were noticed, whereas corneal opacities progressively became clearer, although insignificant improvements in corneal sensitivity or in the reflex tearing were observed.
Cenegermin was effective in treating NK in an infant with Goldenhar syndrome.
本研究旨在介绍治疗神经退行性角膜病变(NK)伴 Goldenhar 综合征患儿的疗效和安全性。
本病例报告了一名婴儿,其右眼和左眼分别出现角膜溃疡和小的中央混浊。尽管使用了治疗性隐形眼镜和临时部分睑裂缝合术,双侧 NK 仍恶化。磁共振成像显示右侧和左侧三叉神经分别缺失和发育不良。给予患者 Cenegermin 滴眼剂,每只眼 1 滴,每天 6 次,持续 8 周,以促进角膜愈合。
治疗后双眼均完全愈合。在 16 个月的随访期间,未观察到上皮缺损、复发或并发症,而角膜混浊逐渐变得更清晰,尽管角膜敏感性或反射性流泪无明显改善。
Cenegermin 对治疗 Goldenhar 综合征伴 NK 的婴儿有效。