Durán-Ferreras E, Mir-Mercader J, Morales-Martínez M D, Martínez-Parra C
Servicio de Neurología, Hospital Universitario Virgen Macarena, Sevilla, Spain.
Rev Neurol. 2004;38(12):1136-8.
Anticonvulsant hypersensitivity syndrome (AHS) is characterised by fever, skin rashes and involvement of the internal organs. Owing to the low frequency with which it appears and its high clinical heterogeneity, it is not always suspected. Moreover, the symptoms often overlap with those of a vasculitis or of an infection. The most commonly associated antiepileptic drugs (AED) are the aromatic agents. We report the case of a female patient who developed AHS with several different AED and presented an especially severe kidney and skin disorder due to carbamazepine (CBZ).
We describe the case of a 26-year-old woman who, after being diagnosed as suffering from secondarily generalised partial seizures, began treatment with 200 mg/12 hours CBZ. A few weeks later, she developed itchy skins lesions compatible with exanthematic pustulosis, together with acute kidney failure requiring haemodialysis. A biopsy study of the kidney revealed immunoallergic tubulointerstitial nephropathy, which is a lesion that has only very occasionally been reported in relation to CBZ therapy. The patient also presented a moderate rise in the level of transaminases and leukocytosis with eosinophilia. She was discharged from hospital without AED but suffered new seizures and was treated with phenytoin and, later, with valproic acid, both as monotherapy. With these drugs she developed AHS consisting in fever, rashes, eosinophilia and subclinical hepatitis. In epicutaneous tests with anticonvulsants, the three AED presented a positive reading, as well as others. The patient was treated with tiagabine, and there were no further hypersensitivity phenomena and a good control of seizures was achieved.
AHS is an infrequent, but potentially serious, clinical entity and must therefore be suspected in patients taking AED who develop fever, rashes or disorders affecting the internal organs.
抗惊厥药物超敏反应综合征(AHS)的特征为发热、皮疹及内脏受累。因其发病率低且临床异质性高,故并非总能被怀疑。此外,其症状常与血管炎或感染的症状重叠。最常与之相关的抗癫痫药物(AED)为芳香族药物。我们报告一例女性患者,其使用多种不同的AED后发生AHS,并因卡马西平(CBZ)出现了特别严重的肾脏和皮肤疾病。
我们描述了一名26岁女性的病例,该患者被诊断为继发性全身性部分性癫痫发作后,开始服用200mg/12小时的CBZ进行治疗。几周后,她出现了与疹性脓疱病相符的瘙痒性皮肤病变,同时伴有需要血液透析的急性肾衰竭。肾脏活检显示为免疫过敏性肾小管间质性肾炎,这是一种与CBZ治疗相关的极为罕见的病变。该患者还出现转氨酶水平中度升高及伴有嗜酸性粒细胞增多的白细胞增多症。她出院时未服用AED,但再次发作癫痫,先后接受苯妥英钠单药治疗及随后的丙戊酸单药治疗。使用这些药物后,她出现了由发热、皮疹、嗜酸性粒细胞增多及亚临床肝炎组成的AHS。在对抗惊厥药物进行的皮肤试验中,这三种AED以及其他一些药物的检测结果均为阳性。该患者接受了噻加宾治疗,未再出现超敏反应现象,癫痫发作得到了良好控制。
AHS是一种罕见但可能严重的临床病症,因此对于服用AED后出现发热、皮疹或影响内脏的疾病的患者必须予以怀疑。