Tokunaga Naoki, Shima Hisato, Okamoto Takuya, Maekawa Masato, Minakuchi Jun
Laboratory, Kawashima Hospital, Tokushima, JPN.
Kidney Disease, Kawashima Hospital, Tokushima, JPN.
Cureus. 2024 Jan 29;16(1):e53153. doi: 10.7759/cureus.53153. eCollection 2024 Jan.
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.
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)