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丙戊酸所致范科尼综合征:临床特征、危险因素、诊断与管理

Valproic-induced Fanconi syndrome: Clinical features, risk factors, diagnosis and management.

作者信息

Wang Chunjiang, Zhou Yulu, Song Liying, Deng Zhenzhen, Fang Weijin

机构信息

Department of Pharmacy, The Third Xiangya Hospital, Central South University, Changsha, Hunan, China.

Department of Pharmacy, Hunan Provincial Maternal and Child Health Care Hospital, Changsha, Hunan, China.

出版信息

Front Med (Lausanne). 2022 Sep 16;9:945244. doi: 10.3389/fmed.2022.945244. eCollection 2022.

Abstract

OBJECTIVE

Although Fanconi syndrome (FS) induced by valproate (VPA) has occasionally been reported, the detailed clinical features of the disease remain unclear. The aim of this study was to elucidate the clinical features of patients with VPA-induced FS.

METHODS

We searched Chinese and English databases for all original studies, clinical reports, and case reports on VPA-induced FS published before March 2022.

RESULTS

A total of 29 articles including 54 patients (28 males and 24 females) were included. The patients had a median age of 7 years (range 2-34 years), had severely disabled (87.0%), tube feeding (64.8%), and received an average of 1.8 medications other than VPA. The median duration of VPA treatment was 4 years (range 0.7-15.5). Pathological fractures (25.9%), unexplained fever (11.1%), muscle weakness (9.3%), and edema (9.3%) were the most common symptoms, while 18 patients were diagnosed in incidental laboratory tests. Blood tests revealed hypokalemia (69.2%), hypophosphatemia (98.0%), and hypouricemia (93.3%). Urinalysis revealed glucosuria (96.1%), proteinuria (100.0%), generalized hyperaminoaciduria (100.0 %), β2 macroglobulin (100.0%). Decreased percent total reabsorption of phosphate (%TRP) found in 94.1% of patients, and increased fractional excretion of uric acid (FEUA) were found in 100% of patients. The median time to resolution of FS after discontinuation of drug therapy was 3 months (range 0.25-18).

CONCLUSIONS

The possibility of FS needs to be considered with long-term VPA administration, especially in young, tube-fed, severely disabled patients who are co-administered with anticonvulsants. Patients receiving VPA should have regular blood and urine tests. Abnormal laboratory values returned to normal levels after VPA discontinuation.

摘要

目的

虽然偶尔有关于丙戊酸盐(VPA)诱发范科尼综合征(FS)的报道,但该病的详细临床特征仍不清楚。本研究的目的是阐明VPA诱发FS患者的临床特征。

方法

我们检索了中国和英文数据库,以查找2022年3月之前发表的所有关于VPA诱发FS的原始研究、临床报告和病例报告。

结果

共纳入29篇文章,包括54例患者(28例男性和24例女性)。患者的中位年龄为7岁(范围2 - 34岁),严重残疾(87.0%),需鼻饲(64.8%),除VPA外平均服用1.8种药物。VPA治疗的中位持续时间为4年(范围0.7 - 15.5年)。病理性骨折(25.9%)、不明原因发热(11.1%)、肌肉无力(9.3%)和水肿(9.3%)是最常见的症状,18例患者在实验室检查时偶然被诊断。血液检查显示低钾血症(69.2%)、低磷血症(98.0%)和低尿酸血症(93.3%)。尿液分析显示糖尿(96.1%)、蛋白尿(100.0%)、全身性高氨基酸尿(100.0%)、β2微球蛋白(100.0%)。94.1%的患者发现磷酸盐总重吸收率(%TRP)降低,100%的患者发现尿酸分数排泄率(FEUA)升高。停药后FS缓解的中位时间为3个月(范围0.25 - 18个月)。

结论

长期使用VPA时需要考虑发生FS的可能性,尤其是在年轻且需鼻饲、严重残疾且同时服用抗惊厥药的患者中。接受VPA治疗的患者应定期进行血液和尿液检查。停用VPA后实验室异常值恢复到正常水平。

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