Tsuruno Tomoko, Tateiwa Hiroki, Hashimoto Yuki, Katsumata Yoshifumi, Kawano Takashi
Department of Anesthesiology and Intensive Care Medicine, Kochi Medical School, Kochi, JPN.
Cureus. 2024 Aug 10;16(8):e66612. doi: 10.7759/cureus.66612. eCollection 2024 Aug.
Glutaric aciduria type I (GA-1) is a rare metabolic disorder caused by an autosomal, recessive, inherited deficiency of glutaryl-CoA dehydrogenase. Reports on the anesthetic management of patients with GA-1 are limited. It has been suggested that inhalation anesthesia is safer than propofol due to the mitochondrial dysfunction inherent in GA-1. However, inhalation anesthesia poses a risk, albeit rare, of malignant hyperthermia, which can result in severe neurological damage in GA-1 patients. Therefore, we considered that management using remimazolam might be effective and, provided a successful general anesthesia using it for a pediatric patient with GA-1. We report a case of a four-year-old girl with GA-1 who underwent a laparoscopic gastrostomy under general anesthesia. Remimazolam was used for both induction and maintenance of anesthesia. Our perioperative management also included measures to prevent a hypercatabolic condition such as adequate hydration and blood glucose control. The patient had an uneventful perioperative course and was discharged on postoperative day 7. Thus, remimazolam is proposed as a new option for anesthetic management in patients with GA-1. Additionally, tailored perioperative management that addresses the unique characteristics of GA-1 is crucial for favorable outcomes.
I型戊二酸血症(GA-1)是一种罕见的代谢紊乱疾病,由常染色体隐性遗传的戊二酰辅酶A脱氢酶缺乏引起。关于GA-1患者麻醉管理的报道有限。由于GA-1固有的线粒体功能障碍,有人认为吸入麻醉比丙泊酚更安全。然而,吸入麻醉存在恶性高热的风险,尽管罕见,但可能导致GA-1患者严重的神经损伤。因此,我们认为使用瑞马唑仑进行管理可能有效,并成功地为一名患有GA-1的儿科患者使用它实施了全身麻醉。我们报告一例四岁患有GA-1的女孩,在全身麻醉下接受腹腔镜胃造口术。瑞马唑仑用于麻醉诱导和维持。我们的围手术期管理还包括采取措施预防分解代谢亢进状态,如充分补液和控制血糖。患者围手术期过程顺利,术后第7天出院。因此,瑞马唑仑被提议作为GA-1患者麻醉管理的一种新选择。此外,针对GA-1独特特征的定制化围手术期管理对于取得良好结果至关重要。