Origlieri Catherine, Geddie Brooke, Karwoski Bethany, Berl Madison M, Elling Nancy, McClintock William, Alexander Janet, Bazemore Marlet, de Beaufort Heather, Hutcheson Kelly, Miller Marijean, Taylormoore Jonathan, Jaafar Mohamad S, Madigan William
Children's National Health System, Washington, District of Columbia.
Helen DeVos Children's Hospital, Grand Rapids, Michigan.
J AAPOS. 2016 Apr;20(2):136-40. doi: 10.1016/j.jaapos.2015.10.020.
The antiepileptic drug vigabatrin is known to cause permanent loss of vision. Both visual field testing and electroretinogram are used to detect retinal damage. Adult data on optical coherence tomography (OCT) shows that retinal nerve fiber layer (RNFL) thinning may be an early indicator of vigabatrin-induced retinal toxicity. The purpose of this study was to investigate whether OCT can detect early vigabatrin-induced retinal toxicity in children.
Pediatric patients (≤18 years of age) requiring vigabatrin for seizure control who were followed at our institution were invited to participate. Patients were examined according to manufacturer guidelines, with most examinations taking place under general anesthesia. RNFL thickness was measured by OCT (Stratus Model 3000, Zeiss) and compared to total cumulative dose of vigabatrin. In most cases, indirect ophthalmoscopy, fundus photography, and electroretinography were also performed.
OCT and complete dosing data was available for 19 patients. Patients with tuberous sclerosis (TS, n = 12) received higher cumulative doses (mean, 1463 g) than non-TS patients (mean, 351 g, P = 0.044). RNFL thinning was detected in the nasal (P < 0.01), superior (P < 0.01), and inferior (P < 0.05) quadrants in patients with TS, particularly once cumulative dose exceeded 1500 g.
In our study population of patients with TS, higher cumulative doses of vigabatrin were associated with RNFL thinning in the nasal, superior, and inferior quadrants. These findings were pronounced once cumulative dose exceeded 1500 g. This pattern of RNFL thinning is similar to what has been shown in adult patients taking vigabatrin.
抗癫痫药物vigabatrin已知会导致永久性视力丧失。视野测试和视网膜电图均用于检测视网膜损伤。关于光学相干断层扫描（OCT）的成人数据显示，视网膜神经纤维层（RNFL）变薄可能是vigabatrin诱导的视网膜毒性的早期指标。本研究的目的是调查OCT能否检测儿童早期vigabatrin诱导的视网膜毒性。
邀请在我们机构接受随访、需要使用vigabatrin控制癫痫发作的儿科患者（≤18岁）参与。患者按照制造商指南进行检查，大多数检查在全身麻醉下进行。通过OCT（蔡司Stratus 3000型）测量RNFL厚度，并与vigabatrin的总累积剂量进行比较。在大多数情况下，还进行了间接检眼镜检查、眼底摄影和视网膜电图检查。
19例患者有OCT和完整的给药数据。结节性硬化症（TS，n = 12）患者的累积剂量（平均1463 g）高于非TS患者（平均351 g，P = 0.044）。在TS患者的鼻侧（P < 0.01）、上方（P < 0.01）和下方（P < 0.05）象限检测到RNFL变薄，尤其是累积剂量超过1500 g时。
在我们的TS患者研究人群中，vigabatrin的累积剂量较高与鼻侧、上方和下方象限的RNFL变薄有关。当累积剂量超过1500 g时，这些发现更为明显。这种RNFL变薄模式与服用vigabatrin的成年患者中所显示的情况相似。
