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巴林一名患有新型突变的肉碱棕榈酰转移酶II缺乏症病例。

A Case of Carnitine Palmitoyltransferase II Deficiency in Bahrain With a Novel Mutation.

作者信息

Alsahlawi Zahra, Fadhul Zainab, Mahmood Ali, Mohamed Ali, Khalil Mohamed, Aljishi Emtithal

机构信息

Department of Pediatrics, Government Hospitals, Salmaniya Medical Complex, Manama, BHR.

Department of Pediatrics, Arabian Gulf University, Manama, BHR.

出版信息

Cureus. 2022 Jun 17;14(6):e26043. doi: 10.7759/cureus.26043. eCollection 2022 Jun.

Abstract

Carnitine palmitoyltransferase II (CPT II) deficiency is a rare genetic metabolic disorder. Three forms of the disease have been described: the lethal neonatal form, the severe infantile hepatocardiomuscular form, and the myopathic form. We report a case of the infantile form of CPT II deficiency with a novel mutation. Our patient is a seven-year-old Bahraini male who was investigated by the pediatric metabolic team following the sudden death of his twin sister in infancy. A fatty acid metabolic disorder was suspected based on his echocardiogram and tandem mass spectrometry (TMS) findings. Genetic analysis was initially inconclusive. Nonetheless, he was started on a fat-free diet, L-carnitine, and medium-chain triglycerides (MCT). At nearly two years of age, the patient had a metabolic crisis precipitated by a viral illness. TMS during this time was consistent with CPT II deficiency. Sanger sequencing then identified the presence of the variant c.161T>G (p.ille54Ser) in a homozygous state, confirming the diagnosis. Although this mutation has not been reported before in previous literature concerning CPT II deficiency, it is extremely likely that this mutation is pathogenic. Although the initial work-up of the patient was inconclusive, our clinical judgment was paramount in managing the patient.

摘要

肉碱棕榈酰转移酶II(CPT II)缺乏症是一种罕见的遗传性代谢紊乱疾病。该疾病已被描述为三种形式:致死性新生儿型、严重婴儿型肝心肌型和肌病型。我们报告一例具有新突变的婴儿型CPT II缺乏症病例。我们的患者是一名七岁的巴林男性,在其双胞胎妹妹于婴儿期突然死亡后,由儿科代谢团队对他进行了调查。基于他的超声心动图和串联质谱(TMS)检查结果,怀疑存在脂肪酸代谢紊乱。最初的基因分析结果不明确。尽管如此,还是让他开始了无脂饮食、补充左旋肉碱并服用中链甘油三酯(MCT)。在将近两岁时,该患者因病毒感染引发了代谢危机。此时的TMS检查结果与CPT II缺乏症相符。随后的桑格测序确定该患者存在纯合状态的c.161T>G(p.ille54Ser)变异,从而确诊。尽管此前关于CPT II缺乏症的文献中未曾报道过这种突变,但极有可能该突变具有致病性。虽然对该患者的初步检查结果不明确,但我们的临床判断在治疗该患者过程中至关重要。

相似文献

本文引用的文献

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Severe infantile type of carnitine palmitoyltransferase II (CPT II) deficiency due to homozygous R503C mutation.
J Inherit Metab Dis. 2007 Apr;30(2):266. doi: 10.1007/s10545-007-0536-8. Epub 2007 Feb 15.

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