Case report: Dichloroacetate-induced methaemoglobinaemia in a G6PD-deficient neonate

IF 2.4 3区 医学 Q2 HEMATOLOGY Pediatric Blood & Cancer Pub Date : 2024-10-27 DOI:10.1002/pbc.31408
Ze Lei Tan, Nicholas Beng Hui Ng, Jacqueline Soo May Ong
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Abstract

A 3-week-old neonate with glucose-6-phosphate dehydrogenase (G6PD) deficiency and primary lactic acidosis developed haemolytic jaundice and methaemoglobinaemia following treatment with dichloroacetate (DCA), a standard treatment for primary lactic acidosis. While this mechanism has been reported in the sheep model, it has not been described in humans. Our case reinforces the uncommon observation that a G6PD-deficient individual experiencing oxidative stress may develop concurrent methaemoglobinaemia. In this case, methylene blue, the standard treatment for methaemoglobinaemia, may result in further oxidative stress. The judicious use of blood transfusion to correct the oxygen-carrying capacity of our patient led to reversal of the methaemoglobinaemia.

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病例报告:一名 G6PD 缺乏症新生儿因二氯醋酸引发的高铁血红蛋白血症。
一名 3 周大的新生儿患有葡萄糖-6-磷酸脱氢酶(G6PD)缺乏症和原发性乳酸酸中毒,在接受原发性乳酸酸中毒的标准疗法二氯乙酸(DCA)治疗后,出现溶血性黄疸和高铁血红蛋白血症。虽然这种机制在绵羊模型中已有报道,但在人类中还没有描述过。我们的病例证实了一个不常见的观察结果,即 G6PD 缺乏者在经历氧化应激后可能会同时出现高铁血红蛋白血症。在这种情况下,亚甲蓝作为治疗高铁血红蛋白血症的标准疗法,可能会导致进一步的氧化应激。我们明智地使用输血来纠正患者的携氧能力,从而逆转了高铁血红蛋白血症。
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来源期刊
Pediatric Blood & Cancer
Pediatric Blood & Cancer 医学-小儿科
CiteScore
4.90
自引率
9.40%
发文量
546
审稿时长
1.5 months
期刊介绍: Pediatric Blood & Cancer publishes the highest quality manuscripts describing basic and clinical investigations of blood disorders and malignant diseases of childhood including diagnosis, treatment, epidemiology, etiology, biology, and molecular and clinical genetics of these diseases as they affect children, adolescents, and young adults. Pediatric Blood & Cancer will also include studies on such treatment options as hematopoietic stem cell transplantation, immunology, and gene therapy.
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