Zhao Yijun,Zhu Linfeng,Yang Fan,et al.Treatment of renal artery aneurysm related to neurofibromatosis type 1 with renal autotransplantation in children:one case report with a literature review[J].Journal of Clinical Pediatric Surgery,,():781-785.[doi:10.3760/cma.j.cn101785-202502006-014]
Treatment of renal artery aneurysm related to neurofibromatosis type 1 with renal autotransplantation in children:one case report with a literature review
- Keywords:
- Neurofibromatosis Type 1; Renal Artery Aneurysm; Kidney Transplantation; Surgical Procedures; Operative; Treatment Outcome; Child
- Abstract:
- Objective To explore the clinical characteristics,diagnostics and treatments of renal artery aneurysm related to neurofibromatosis type 1,as well as the prognosis of pediatric renal autotransplantation. Methods Retrospective analysis was performed for clinical data of a child with renal artery aneurysm related to neurofibromatosis type 1,who underwent renal autotransplantation.The databases of PubMed,MEDLINE,Web of Science,Wanfang,China National Knowledge Infrastructure (CNKI) and VIP were searched with such keywords as "neurofibromatosis type 1","renal artery aneurysm" and "kidney autotransplantation".The searching started from January 1980 to December 2024.Duplicate literatures were excluded and clinical features,diagnosis,treatment and prognosis summarized. Results A 167-month-old boy was admitted with a 15-day history of elevated blood pressure.Upon admission,blood pressure measurements were 146/96 mmHg (left arm),168/93 mmHg (right arm),177/99 mmHg (left leg) and 172/96 mmHg (right leg).Multiple scattered café-au-lait dermal spots were noted.Computed tomography angiography (CTA) revealed fusiform dilation at renal hilum near left renal artery.And renal Doppler ultrasound identified a segmental branch of left renal artery with fusiform dilation in its midportion.The response to oral antihypertensive medication was poor.Ultimately,left nephrectomy,resection of left renal artery aneurysm,reconstruction of left renal artery and renal autotransplantation were performed.Postoperatively,transplant kidney developed occlusion of renal artery branches.Over a 3-month follow-up period,blood pressure normalized with oral antihypertensive agents.CTA hinted at transplant kidney slightly smaller with normal blood supply.Four cases were collected from literature searching.Among a total of 5 eligible cases,there were 2 males and 3 females with an average age of 21(13-28) year and an average diameter of (1.0-2.4) cm.Four of them presented with hypertension.Renal autotransplantation with renal artery reconstruction was performed.In two hypertensive cases,blood pressure was controlled after additional use of oral antihypertensive agents. Conclusions Renal artery aneurysm related to neurofibromatosis type is rare in children.And hypertension is a common initial symptom.Pediatric renal autotransplantation is both safe and effective.
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Memo
收稿日期:2025-2-5。
基金项目:浙江省科技厅公益技术应用研究项目(LGF22H050004)
通讯作者:陈光杰,Email:dr.chenguangjie@zju.edu.cn