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胱氨酸尿症导致妊娠非典型并发症的典型病程:一例病例报告及文献综述

Typical course of cystinuria leading to untypical complications in pregnancy: A case report and review of literature.

作者信息

Ivandic Ema, Maric Marjan, Elvedi-Gasparovic Vesna, Fistrek Prlic Margareta, Lamot Lovro, Jelakovic Bojan, Vukovic Brinar Ivana

机构信息

Department of Nephrology, Arterial, Hypertension, Dialysis and Transplantation, University Clinical Hospital Zagreb, Zagreb, Croatia.

Clinic of Urology, University Clinical Hospital Zagreb, Zagreb, Croatia.

出版信息

Front Med (Lausanne). 2023 Apr 6;10:1097442. doi: 10.3389/fmed.2023.1097442. eCollection 2023.

DOI:10.3389/fmed.2023.1097442
PMID:37089588
原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC10117994/
Abstract

Cystinuria is a rare genetic disorder inherited by an autosomal recessive pattern which affects the transmembrane transporter for the base amino acid cystine. It has a general prevalence of 1 in 7000 with demographic variations. Patients with cystinuria have excessive urinary excretion of cystine, which can lead to the formation of stones. Up to 70% of patients will develop chronic kidney disease that can progress even to end-stage renal disease. Symptoms usually start in the first two decades of life with a typical presentation consisting of flank pain and renal colic, usually accompanied by urinary tract infection and deterioration of kidney function. Men are typically affected twice as often as women and have a more severe clinical course. Diagnosis is made by spectrophotometric analysis of the stones that are collected after spontaneous expulsion or medical intervention. Genetic testing is not mandatory but is recommended in uncertain cases or as a part of genetic counseling. Treatment consists of diet modification, alkalization of urine, and thiol-based therapies if other measures fail to prevent stone formation. In pregnancy, cystinuria with the formation of cystine stones represents a therapeutic challenge and requires a multidisciplinary approach consisting of an uro-nephrology team and a gynecologist. We present the case of a 34-year-old woman with cystinuria on whom the diagnosis was made by analysis of the expulsed stone. While her previous pregnancies were without complications, her third pregnancy was accompanied by frequent urinary tract infections, acute worsening of kidney function, and urological interventions during pregnancy due to the formation of new stones. Despite the complicated course, the pregnancy was successfully carried to term with the delivery of a healthy female child.

摘要

胱氨酸尿症是一种罕见的遗传性疾病,以常染色体隐性模式遗传,会影响碱性氨基酸胱氨酸的跨膜转运蛋白。其总体发病率为七千分之一,存在人口统计学差异。胱氨酸尿症患者的尿液中胱氨酸排泄过多,这可能导致结石形成。高达70%的患者会发展为慢性肾病,甚至可能进展到终末期肾病。症状通常在生命的前二十年开始出现,典型表现为胁腹疼痛和肾绞痛,通常伴有尿路感染和肾功能恶化。男性受影响的频率通常是女性的两倍,临床病程也更严重。诊断通过对自然排出或经医学干预后收集的结石进行分光光度分析来进行。基因检测并非强制要求,但在不确定的情况下或作为遗传咨询的一部分时建议进行。治疗包括饮食调整、尿液碱化,如果其他措施无法预防结石形成,则采用基于硫醇的疗法。在怀孕期间,胱氨酸尿症伴胱氨酸结石形成是一个治疗挑战,需要由泌尿肾病团队和妇科医生组成的多学科方法。我们报告一例34岁患有胱氨酸尿症的女性病例,通过对排出结石的分析做出诊断。她之前的怀孕没有并发症,但第三次怀孕时伴有频繁的尿路感染、肾功能急性恶化,并且由于新结石形成在孕期进行了泌尿外科干预。尽管病程复杂,但妊娠成功足月,分娩出一名健康女婴。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/af2f/10117994/ba5bf9775949/fmed-10-1097442-g002.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/af2f/10117994/81402b8397b6/fmed-10-1097442-g001.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/af2f/10117994/ba5bf9775949/fmed-10-1097442-g002.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/af2f/10117994/81402b8397b6/fmed-10-1097442-g001.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/af2f/10117994/ba5bf9775949/fmed-10-1097442-g002.jpg

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本文引用的文献

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