Haug Sara J, Wong Robert W, Day Shelley, Choudhry Netan, Sneed Scott, Prasad Pradeep, Read Sarah, McDonald Richard H, Agarwal Anita, Davis Janet, Sarraf David
*West Coast Retina Medical Group, San Francisco, California; †Austin Retina Associates, Austin, Texas; ‡Herzig Eye Institute, Toronto, Ontario, Canada; §Associated Retinal Consultants P.C., Royal Oak, Michigan; ¶Retinal Disorders and Ophthalmic Genetics Division, Stein Eye Institute, University of California-Los Angeles, Los Angeles, California; **Bascom Palmer Eye Institute, University of Miami, Miami, Florida; ††Vanderbilt Eye Institute, Vanderbilt University, Nashville, Tennessee; and ‡‡Greater Los Angeles VA Healthcare Center, Los Angeles, California.
Retina. 2016 Dec;36 Suppl 1:S159-S167. doi: 10.1097/IAE.0000000000001267.
To report nine new cases of retinal degeneration secondary to didanosine toxicity and to summarize the previously reported cases in the literature.
This was a multicenter, retrospective, observational case study from seven institutions. Medical records of patients who demonstrated well-demarcated severe midperipheral chorioretinal degeneration and who were previously treated with didanosine therapy were collected and the following information was reviewed: age, gender, medical history, detailed medication history including current and previous antiretroviral use, ocular and retinal examination findings, and multimodal imaging findings with optical coherence tomography, fundus photography, wide-field fundus autofluorescence, and wide-field fluorescein angiography. When available, findings with electrophysiology testing and automated perimetry were also collected and reviewed. A literature review was also performed to collect all reported cases of chorioretinal degeneration secondary to didanosine toxicity.
Nine patients were identified who had findings consistent with peripheral retinal toxicity secondary to didanosine use. Eight of the 9 patients were men, and the median age was 54 years at the time of presentation (mean: 55 years, range, 42-71 years). Snellen distance acuity ranged from 20/20 to 20/32. At least three of the cases in the series demonstrated progression of the peripheral retinal pigment epithelium and photoreceptor atrophy despite didanosine cessation. A review of the literature revealed 10 additional cases of didanosine toxicity. Seven of the 10 cases were in men (70%), and the average age was 26 years with a wide range (2-54 years). Chorioretinal findings were very similar to this cohort.
Herein, we report the largest series of nine cases of peripheral chorioretinal degeneration secondary to didanosine toxicity in adults. When combined with the cases in the literature, 19 cases of didanosine toxicity, 4 of which occurred in children, were collected and analyzed. Three of the new cases presented showed clear progression of degeneration despite didanosine cessation. Newer nucleoside reverse transcriptase inhibitors may potentiate mitochondrial DNA damage and lead to continued chorioretinal degeneration.
报告9例继发于去羟肌苷毒性的视网膜变性新病例，并总结文献中先前报道的病例。
这是一项来自7家机构的多中心、回顾性、观察性病例研究。收集表现为界限清晰的严重中周部脉络膜视网膜变性且先前接受过去羟肌苷治疗的患者的病历，并审查以下信息：年龄、性别、病史、详细用药史（包括当前和先前的抗逆转录病毒药物使用情况）、眼部和视网膜检查结果，以及光学相干断层扫描、眼底照相、超广角眼底自发荧光和超广角荧光素血管造影的多模态成像结果。如有电生理测试和自动视野检查结果，也进行收集和审查。还进行了文献综述，以收集所有报道的继发于去羟肌苷毒性的脉络膜视网膜变性病例。
确定了9例患者，其表现与使用去羟肌苷继发的周边视网膜毒性一致。9例患者中有8例为男性，就诊时的中位年龄为54岁（平均：55岁，范围42 - 71岁）。Snellen远视力范围为20/20至20/32。该系列中至少3例病例尽管停用了去羟肌苷，但周边视网膜色素上皮和光感受器萎缩仍有进展。文献综述发现另外10例去羟肌苷毒性病例。10例病例中有7例为男性（70%），平均年龄为26岁，范围较广（2 - 54岁）。脉络膜视网膜表现与该队列非常相似。
在此，我们报告了成人中继发于去羟肌苷毒性的最大系列的9例周边脉络膜视网膜变性病例。与文献中的病例相结合，共收集并分析了19例去羟肌苷毒性病例，其中4例发生在儿童中。新出现的3例病例尽管停用了去羟肌苷，但仍显示出明显的变性进展。新型核苷类逆转录酶抑制剂可能会增强线粒体DNA损伤并导致脉络膜视网膜变性持续发展。
