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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

Anticonvulsant hypersensitivity syndrome after phenytoin administration in an adolescent patient: a case report and review of literature.

作者信息

Ghannam Malik, Mansour Shaden, Nabulsi Aya, Abdoh Qusay

机构信息

AN-Najah National University Teaching Hospital, Asira, Nablus, West bank Palestine.

AN-Najah National University, Nablus, West bank Palestine.

出版信息

Clin Mol Allergy. 2017 Jun 15;15:14. doi: 10.1186/s12948-017-0069-0. eCollection 2017.

Abstract

BACKGROUND

Hypersensitivity is a rare adverse drug reaction (ADR) associated with anti-epileptic medications. Phenytoin is one of the commonly used drugs for treatment of epilepsy that encounters a hypersensitivity reaction. This reaction can be ranged from mild cutaneous rash to anticonvulsant hypersensitivity syndrome (AHS) or drug reaction with eosinophilia and systemic symptoms (DRESS) that includes fever, rash, eosinophilia and involvement of multiple internal organs.

CASE PRESENTATION

A 15 year old middle eastern female patient from Gaza strip with free past medical and allergic history. She presented to An-Najah National University Hospital (NNUH) in Nablus with intermittent high grade fever, jaundice, rash and skin peeling. On examination, she had axillary and inguinal lymphadenopathy, moderate splenomegaly and diffuse maculopapular rash. The patient was on phenytoin which started 1 month prior to her presentation as a seizure prophylaxis due to previous head injury. Eventually, the patient was diagnosed with AHS/DRESS.

CONCLUSIONS

AHS is a diagnosis of exclusion and it is significantly underreported that requires a high index of suspicion. We liked to share this case and shed the light in more details on AHS/DRESS. Our goal was to help making AHS more reported in the literature in adolescent patients, as well as to make physicians more alert of this condition's seriousness when they prescribe antiepileptic medications in particular. In this report, we included the first case of AHS which was reported in an adolescent patient in Palestine. Moreover, we reviewed the available literature for a better understanding of the pathophysiology and management of AHS. We still believe that the full understanding of the pathogenesis of AHS is lacking, and also we are lacking a clinical tool or scoring system to determine the severity of AHS/DRESS.

摘要

背景

超敏反应是一种与抗癫痫药物相关的罕见药物不良反应(ADR)。苯妥英钠是治疗癫痫常用药物之一,会引发超敏反应。这种反应范围从轻度皮疹到抗惊厥药超敏综合征(AHS)或伴有嗜酸性粒细胞增多和全身症状的药物反应(DRESS),后者包括发热、皮疹、嗜酸性粒细胞增多以及多个内脏器官受累。

病例介绍

一名来自加沙地带的15岁中东女性患者,既往无病史及过敏史。她因间歇性高热、黄疸、皮疹和皮肤脱皮就诊于纳布卢斯的纳贾赫国立大学医院(NNUH)。检查发现,她有腋窝和腹股沟淋巴结肿大、中度脾肿大以及弥漫性斑丘疹。该患者因之前头部受伤,在就诊前1个月开始服用苯妥英钠以预防癫痫发作。最终,患者被诊断为AHS/DRESS。

结论

AHS是一种排除性诊断,其报告严重不足,需要高度怀疑。我们希望分享此病例,并更详细地阐述AHS/DRESS。我们的目标是促使AHS在青少年患者的文献中得到更多报道,同时让医生在开具抗癫痫药物时更加警惕这种疾病的严重性。在本报告中,我们纳入了巴勒斯坦青少年患者中首例AHS病例。此外,我们回顾了现有文献以更好地了解AHS的病理生理学和管理。我们仍然认为,对AHS发病机制缺乏全面了解,而且我们也缺乏确定AHS/DRESS严重程度的临床工具或评分系统。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/12a1/5472963/4565c4bf7266/12948_2017_69_Fig1_HTML.jpg

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