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首例已知的透析患者遗传性杂合性丁酰胆碱酯酶缺乏症病例。

The First-Known Case of Hereditary Heterozygous Butyrylcholinesterase Deficiency in a Patient on Dialysis.

作者信息

Tokunaga Naoki, Shima Hisato, Okamoto Takuya, Maekawa Masato, Minakuchi Jun

机构信息

Laboratory, Kawashima Hospital, Tokushima, JPN.

Kidney Disease, Kawashima Hospital, Tokushima, JPN.

出版信息

Cureus. 2024 Jan 29;16(1):e53153. doi: 10.7759/cureus.53153. eCollection 2024 Jan.

DOI:10.7759/cureus.53153
PMID:38420074
原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC10900173/
Abstract

Serum levels of butyrylcholinesterase (BChE) are commonly used to assess liver function. Its levels have been reported to be significantly lower in patients undergoing dialysis. To the best of our knowledge, this is the first report of hereditary heterozygous BChE deficiency in a patient undergoing dialysis. Medical staff involved in the care of patients with BChE deficiency should be aware of anesthetic usage, because prolonged neuromuscular paralysis following the administration of succinylcholine or mivacurium may occur. However, in the heterozygotes, BChE activity is not completely absent. Therefore, differentiating patients undergoing dialysis is challenging. A 52-year-old man underwent living-related kidney transplantation for focal segmental glomerulosclerosis at 22 years of age. As the renal function gradually worsened, the patient began to receive combined hemodialysis and peritoneal dialysis therapy. No problems with anesthesia were observed in past surgeries. The patient's BChE levels fluctuated between 76 and 170 U/L (reference range: 198-495 U/L); however, they had never been previously investigated. We suspected hereditary heterozygous BChE deficiency because the patient's sister was also diagnosed with it. DNA sequencing revealed a heterozygous missense mutation (Gly365Arg) and a K-variant (Ala539Thr). Patients on dialysis with low serum BChE levels often present with low albumin levels which may be overlooked as malnutrition. Thus, BChE deficiency should be suspected in patients on dialysis with unexplained low serum BChE levels. In the case of heterozygous BChE deficiency, the reference value is low, and continuous monitoring is crucial.

摘要

血清丁酰胆碱酯酶(BChE)水平常用于评估肝功能。据报道,透析患者的BChE水平显著降低。据我们所知,这是首例关于透析患者遗传性杂合性BChE缺乏症的报告。参与BChE缺乏症患者护理的医务人员应了解麻醉药物的使用情况,因为使用琥珀酰胆碱或米库氯铵后可能会出现长时间的神经肌肉麻痹。然而,在杂合子中,BChE活性并未完全缺失。因此,鉴别透析患者具有挑战性。一名52岁男性在22岁时因局灶节段性肾小球硬化接受了亲属活体肾移植。随着肾功能逐渐恶化,患者开始接受血液透析和腹膜透析联合治疗。过去的手术中未观察到麻醉问题。患者的BChE水平在76至170 U/L之间波动(参考范围:198 - 495 U/L);然而,此前从未对其进行过检测。我们怀疑该患者患有遗传性杂合性BChE缺乏症,因为其妹妹也被诊断出患有此病。DNA测序显示存在杂合错义突变(Gly365Arg)和K变异体(Ala539Thr)。血清BChE水平低的透析患者常伴有低白蛋白水平,这可能被误诊为营养不良而被忽视。因此,对于血清BChE水平 unexplained 降低的透析患者,应怀疑BChE缺乏症。在杂合性BChE缺乏症的情况下,参考值较低,持续监测至关重要。 (注:原文中“unexplained”疑有误,可能是“unexpected”之类的词,这里按原文翻译)

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本文引用的文献

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Cureus. 2023 Apr 28;15(4):e38234. doi: 10.7759/cureus.38234. eCollection 2023 Apr.
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Butyrylcholinesterase level as an independent prognostic factor for overall survival in patients on maintenance hemodialysis: a single-center retrospective study.丁酰胆碱酯酶水平作为维持性血液透析患者总生存的独立预后因素:一项单中心回顾性研究。
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Butyrylcholinesterase deficiency and its clinical importance in anaesthesia: a systematic review.
丁酰胆碱酯酶缺乏及其在麻醉中的临床意义:系统评价。
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4
Low serum butyrylcholinesterase is independently related to low fetuin-A in patients on hemodialysis: a cross-sectional study.血液透析患者血清丁酰胆碱酯酶水平低与胎球蛋白-A水平低独立相关:一项横断面研究
Int Urol Nephrol. 2018 Sep;50(9):1713-1720. doi: 10.1007/s11255-018-1957-z. Epub 2018 Aug 20.
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Protein Energy Wasting and Sarcopenia in Dialysis Patients.透析患者的蛋白质能量消耗与肌肉减少症
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6
Butyrylcholinesterase level as an independent factor of erythropoiesis-stimulating agent resistance in patients on maintenance hemodialysis: a single-center cross-sectional study.维持性血液透析患者中丁酰胆碱酯酶水平作为促红细胞生成素抵抗的独立因素:一项单中心横断面研究
Clin Exp Nephrol. 2018 Oct;22(5):1174-1181. doi: 10.1007/s10157-018-1569-z. Epub 2018 Mar 29.
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Significance of preoperative butyrylcholinesterase level as an independent predictor of survival in patients with upper urinary tract urothelial carcinoma treated with nephroureterectomy.术前丁酰胆碱酯酶水平作为接受肾输尿管切除术治疗的上尿路尿路上皮癌患者生存独立预测指标的意义
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Assessment of the value of serum cholinesterase as a liver function test for cirrhotic patients.评估血清胆碱酯酶作为肝硬化患者肝功能检测指标的价值。
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