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视网膜母细胞瘤患儿单次玻璃体腔注射标准低剂量美法仑后发生的弥漫性脉络膜视网膜萎缩:病例报告

Diffuse chorioretinal atrophy after a single standard low- dose intravitreal melphalan injection in a child with retinoblastoma: a case report.

作者信息

Chao An- Ning, Kao Ling-Yuh, Liu Laura, Wang Nan-Kai

机构信息

Department of Ophthalmology, Chang Gung Memorial Hospital and Chang Gung University, College of Medicine, Fu-Hsin Street, Kweishan, Taoyuan, 3305, Taiwan.

出版信息

BMC Ophthalmol. 2016 Mar 15;16:27. doi: 10.1186/s12886-016-0204-6.

Abstract

BACKGROUND

Controlling retinoblastoma with seeding is challenging despite advances in treatment modalities. Intravitreal melphalan is an alternative to external beam radiation or enucleation for recurrent or refractory vitreous seeds. Significant ocular side effects following intravitreal melphalan injections are uncommon. Complications have been reported in eyes receiving higher concentrations of melphalan and repetitive injections. We report a case in which diffuse chorioretinal atrophy was developed at the injection site after a single, standard low-dose intravitreal melphalan injection.

CASE PRESENTATION

A 12-month-old female child without a family history of retinoblastoma presented with unilateral group C retinoblastoma in her right eye. A solitary tumour with retinal breaks on the tumour surface, and vitreous seeds overlying the tumour were observed at the 8 o'clock position of the retina. After two cycles of intra-arterial chemotherapy with melphalan, the main tumour displayed significant regression, but the vitreous seeds overlying the main tumour were still active. Because of the persistence of vitreous seeds and the inadequate response to intra-arterial melphalan treatment, intravitreal melphalan (8 μg in 0.05 mL) was injected using a 32-gauge needle 2.5 mm from the 5 o'clock position of the limbus, the meridian opposite to the vitreous seeds. After 1 month, the retina around the injection site demonstrated diffuse retinal pigment epithelium alterations with dense hard exudates. Although the main retinal mass, and vitreous seeds resolved, the hard exudates persisted for more than 2 years after the single low-dose melphalan injection.

CONCLUSIONS

Intravitreal melphalan injections should be cautiously used for eyes with refractory seeds, particularly when multiple injections are required to control retinoblastoma seeds. Dose- related retinal toxicity could occur in pre-treated eyes even when a relatively low standard dose is used. Such patients should be followed up closely to monitor the treatment response and to assess potential delayed toxicity.

摘要

背景

尽管治疗方式有所进步,但控制伴有播散的视网膜母细胞瘤仍具有挑战性。玻璃体内注射美法仑是复发性或难治性玻璃体播散替代外照射放疗或眼球摘除术的一种选择。玻璃体内注射美法仑后出现严重眼部副作用的情况并不常见。有报道称,接受较高浓度美法仑和重复注射的眼睛出现了并发症。我们报告了一例在单次标准低剂量玻璃体内注射美法仑后,注射部位出现弥漫性脉络膜视网膜萎缩的病例。

病例介绍

一名12个月大的女童,无视网膜母细胞瘤家族史,右眼患有单侧C组视网膜母细胞瘤。在视网膜8点钟位置观察到一个孤立性肿瘤,肿瘤表面有视网膜裂孔,肿瘤上方有玻璃体播散。在进行了两个周期的动脉内美法仑化疗后,主要肿瘤明显缩小,但主要肿瘤上方的玻璃体播散仍然活跃。由于玻璃体播散持续存在且对动脉内美法仑治疗反应不佳,使用32号针头在距角膜缘5点钟位置2.5毫米处(与玻璃体播散相对的子午线)注射玻璃体内美法仑(0.05毫升含8微克)。1个月后,注射部位周围的视网膜出现弥漫性视网膜色素上皮改变,并伴有致密的硬性渗出物。尽管主要视网膜肿块和玻璃体播散消退,但在单次低剂量美法仑注射后,硬性渗出物持续了两年多。

结论

对于伴有难治性播散的眼睛,应谨慎使用玻璃体内注射美法仑,尤其是在需要多次注射以控制视网膜母细胞瘤播散时。即使使用相对较低的标准剂量,在先前接受过治疗的眼睛中也可能发生剂量相关的视网膜毒性。应对此类患者进行密切随访,以监测治疗反应并评估潜在的延迟毒性。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/118c/4791807/5848f7a5e148/12886_2016_204_Fig1_HTML.jpg

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