De Novo Renal Artery Stenosis Developed in Initially Normal Renal Arteries during the Long-Term Follow-Up of Patients with Moyamoya Disease
De Novo Renal Artery Stenosis Developed in Initially Normal Renal Arteries during the Long-Term Follow-Up of Patients with Moyamoya Disease
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DOI：
10.1016/j.jstrokecerebrovasdis.2020.104786
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发表时间：
2020-08-01
影响因子：
2.5
通讯作者：
Maehara, Taketoshi
中科院分区：
文献类型：
--
作者：
Hara, Shoko;Shimizu, Kazuhide;Maehara, Taketoshi
背景：既往血管造影证实正常的肾动脉发生肾动脉狭窄，在烟雾病患者中从未报道过。病例描述：在对儿童烟雾病患者的长期随访中，我们观察到3例患者在术后1年经血管造影证实为正常的动脉中发生肾动脉新生狭窄（平均7年，从4年到11年不等）。所有这些患者在儿童时期成功的间接搭桥手术后神经系统稳定。然而，术后10多年（平均15年，14 ~ 23年不等），患者出现高血压，并发现肾动脉新生狭窄，经血管内成形术改善。在血管成形术后的随访中，1例患者高血压复发，需要第二次和第三次血管成形术治疗再狭窄。1例患者在血管成形术2年后死于颅内出血。在2例存活患者中，亚洲人群烟雾病易感基因环指蛋白213 （RNF213; p.R4810K）点突变基因分析为杂合变异阳性。结论：在长期随访期间，最初正常的动脉可能会发生新生肾动脉狭窄，特别是在患有烟雾病的儿科患者中。考虑到烟雾病的颅外表现，临床医生应该记住，肾动脉狭窄可能在以后的生活中出现。因此，即使患者在搭桥手术后神经系统稳定，继续对这些患者进行数十年的随访也是至关重要的。监测血压和肾动脉狭窄的新发情况对于预防高血压相关的发病率和死亡率（如颅内出血）非常重要。
Background: The de novo occurrence of renal artery stenosis in renal arteries that were angiographically confirmed to be normal in the past has never been reported before in patients with moyamoya disease. Case description: During the long-term follow-up of pediatric patients with moyamoya disease, we observed 3 patients who developed de novo renal artery stenosis in arteries that had been angiographically confirmed to be normal 1 year after the surgery (7 years on average, ranging from 4 to 11 years). All of these patients were neurologically stable after successful indirect bypass surgery during childhood. However, more than 10 years after the surgery (15 years on average, ranging from 14 to 23 years), they developed hypertension and were found to have de novo renal artery stenosis, which was ameliorated by endovascular angioplasty. During the follow-up after angioplasty, 1 patient experienced a recurrence of hypertension and required a second and third angioplasty for restenosis. Another patient died of intracranial hemorrhage 2 years after angioplasty. In the 2 surviving patients, gene analysis of the ring finger protein 213 (RNF213; p.R4810K) point mutation, the susceptibility gene for moyamoya disease in the Asian population, was positive for the heterozygous variant. Conclusions: De novo renal artery stenosis might develop in initially normal arteries during long-term follow-up, particularly among pediatric patients with moyamoya disease. Considering the extracranial manifestations of moyamoya disease, clinicians should keep in mind that de novo renal artery stenosis could emerge later in their life. Thus, it is crucial to continue to follow these patients for decades, even if the patients are neurologically stable after bypass surgery. Monitoring for blood pressure and the de novo occurrence of renal artery stenosis is important to prevent hypertension-related morbidity and mortality, such as intracranial hemorrhage, in this disease population.