Department of Neurology, Martin-Luther-Universität Halle-Wittenberg, Ernst-Grube-Str. 40, 06120, Halle (Saale), Germany.
Wien Klin Wochenschr. 2012 Dec;124(23-24):851-4. doi: 10.1007/s00508-012-0296-9. Epub 2012 Nov 27.
Two symptomatic patients with heterozygous carnitine palmitoyltransferase II (CPT II) deficiency are reported. Patient 1, a 21-year-old female professional tennis player, suffered from exercise-induced attacks of muscle pain, burning sensations and proximal weakness. Patient 2, a 30-year-old male amateur marathon runner developed muscle cramps and rhabdomyolysis upon extensive exercise and insolation-induced fever. In both patients, the common p.S113L mutation was found in heterozygote state. No second mutation could be found upon sequencing of all the exons of CPT2 gene including exon-intron boundaries. Biochemically, residual CPT activity in muscle homogenate upon inhibition by malonyl-CoA and Triton-X-100 was intermediate between controls and patients with mutations on both alleles. Although CPT II deficiency is an autosomal recessive disorder, the reported patients indicate that heterozygotes might also have typical attacks of myalgia, pareses or rhabdomyolysis.
报告了两名杂合子肉碱棕榈酰转移酶 II (CPT II) 缺乏症的有症状患者。患者 1 为 21 岁女性职业网球运动员,运动诱发肌肉疼痛、烧灼感和近端无力。患者 2 为 30 岁男性业余马拉松运动员,广泛运动和日晒引起发热后发生肌肉痉挛和横纹肌溶解。两名患者均发现杂合子状态下常见的 p.S113L 突变。对 CPT2 基因的所有外显子(包括外显子-内含子边界)进行测序后,未发现第二个突变。生化分析显示,经丙二酰辅酶 A 和 Triton-X-100 抑制后肌肉匀浆中的残余 CPT 活性在对照组和两个等位基因突变患者之间处于中间水平。尽管 CPT II 缺乏症是一种常染色体隐性疾病,但报告的患者表明杂合子也可能出现典型的肌痛、瘫痪或横纹肌溶解发作。