氨己烯酸治疗婴儿痉挛症致头颅磁共振成像异常1例

Vigabatrin-associated brain abnormalities on magnetic resonance imaging in the treatment of infantile spasms:a case report

氨己烯酸相关的头颅磁共振成像异常(VABAM)是氨己烯酸较为罕见的不良反应,多数不伴随临床症状,但在极少情况下,会合并多动运动障碍、肌阵挛、震颤和急性脑病。VABAM真正的发病率尚不清楚,尚未被临床医师所熟知和重视。文中报道1例女性儿童,诊断为婴儿痉挛症,先后服用多种抗癫痫药物和促肾上腺皮质激素冲击治疗,在服用氨己烯酸后逐渐出现痉挛发作增多,精神萎靡;头颅磁共振成像结果提示双侧中脑背部、丘脑、苍白球前部异常信号。逐渐减停氨己烯酸后,发作频率逐渐减少,患儿精神好转,磁共振成像异常病灶消失。本文检索了相关文献报道,探讨VABAM可能的发病机制和危险因素,以期提高临床医生对该病的认识,以免漏诊、误诊,做出正确决策。

Vigabatrin-associated brain abnormalities on magnetic resonance imaging(VABAM)is a relatively rare side effect of vigabatrin,most of which are asymptomatic.However,there will be extremely rare cases with hyperkinetic disorders,myoclonus,tremor,and acute encephalopathy under certain circumstances.VABAM is often underappreciated by physicians and its accurate incidence remains unclear.A female infant who was diagnosed with infantile spasms and required adrenocorticotropic hormone therapy accompanied by various antiseizure medicines was reported.Unfortunately,she became lethargic and her spasm deteriorated gradually after vigabatrin exposure.Her brain magnetic resonance imaging revealed abnormal signals bilaterally in the dorsal midbrain,thalamus,and rostral part of the pallidum.She had a seizure amelioration and became lively as a result of vigabatrin withdrawal.In the meanwhile,magnetic resonance imaging returned to normal.Attempts were made to discover the risk factors of VABAM and potential pathogenesis.Further understanding of the disease should contribute to decreasing misdiagnosis and making precise decisions.