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拉莫三嗪所致抗惊厥药超敏反应综合征的延迟表现

Delayed Presentation of Anticonvulsant Hypersensitivity Syndrome Secondary to Lamotrigine.

作者信息

Jarwan Rafeef O, Ali Mohsin

机构信息

Preventive Medicine, King Abdulaziz University Hospital, Jeddah, SAU.

Family Medicine, King Saud Bin Abdulaziz University for Health Sciences, Jeddah, SAU.

出版信息

Cureus. 2022 Jul 28;14(7):e27399. doi: 10.7759/cureus.27399. eCollection 2022 Jul.

DOI:10.7759/cureus.27399
PMID:36046292
原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC9418624/
Abstract

Anticonvulsant hypersensitivity syndrome (AHS) is a rare condition that needs to be seriously recognized and diagnosed. However, it is difficult to diagnose it since its clinical manifestation mimics other common infectious and neoplastic diseases. AHS manifests as skin rash that is preceded by fever accompanying internal organ involvement, with the liver being mostly affected. AHS is a condition that develops secondary to anticonvulsant exposure like phenytoin, phenobarbitone, carbamazepine, and lamotrigine. A defect in epoxide hydroxylase leading to the accumulation of toxic metabolites of aromatic anticonvulsant is hypothesized to play a role in developing AHS. This report presents a case of a 49-year-old epileptic Asian female, who complained of persistent high-grade fever followed by generalized maculopapular rash, high liver enzymes, and pancytopenia. The patient had unremarkable past history and systematic review. The patient went through various investigations to rule out infections and systematic diseases and all investigations were normal. After excluding every possible cause, the patient was diagnosed with AHS secondary to lamotrigine usage for one month.

摘要

抗惊厥药物超敏反应综合征(AHS)是一种罕见疾病,需要得到认真识别和诊断。然而,由于其临床表现与其他常见的感染性和肿瘤性疾病相似,因此难以诊断。AHS表现为皮疹,皮疹出现前伴有发热并累及内脏器官,其中肝脏受累最为常见。AHS是一种因接触苯妥英钠、苯巴比妥、卡马西平和拉莫三嗪等抗惊厥药物而继发的疾病。据推测,环氧化物水解酶缺陷导致芳香族抗惊厥药物的有毒代谢产物积累,在AHS的发生中起作用。本报告介绍了一名49岁的亚洲癫痫女性病例,该患者主诉持续高热,随后出现全身性斑丘疹、肝酶升高和全血细胞减少。患者既往病史及系统回顾均无异常。患者接受了各种检查以排除感染和系统性疾病,所有检查结果均正常。在排除所有可能的病因后,该患者被诊断为因使用拉莫三嗪一个月继发的AHS。

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本文引用的文献

1
Anticonvulsant hypersensitivity syndrome: clinic case and literature review.抗惊厥药物超敏反应综合征:临床病例及文献综述
Rev Fac Cien Med Univ Nac Cordoba. 2020 Aug 21;77(3):211-213. doi: 10.31053/1853.0605.v77.n3.27857.
2
Anticonvulsant hypersensitivity syndrome after phenytoin administration in an adolescent patient: a case report and review of literature.青少年患者服用苯妥英钠后出现抗惊厥药物超敏反应综合征:一例报告及文献复习
Clin Mol Allergy. 2017 Jun 15;15:14. doi: 10.1186/s12948-017-0069-0. eCollection 2017.
3
Anticonvulsant hypersensitivity syndrome secondary to carbamazepine.卡马西平继发的抗惊厥药超敏反应综合征
Proc (Bayl Univ Med Cent). 2017 Jan;30(1):94-96. doi: 10.1080/08998280.2017.11929545.
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Anticonvulsant hypersensitivity syndrome: a rare and serious complication.抗惊厥药超敏反应综合征:一种罕见且严重的并发症。
Med J Aust. 2011 Jun 6;194(11):609-10. doi: 10.5694/j.1326-5377.2011.tb03118.x.