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丙酸血症伴新型突变患儿反复发作性胰腺炎

Propionic Acidemia with Novel Mutation Presenting as Recurrent Pancreatitis in a Child.

机构信息

Department of Emergency Medicine, School of Medicine, Kyungpook National University, Daegu, Korea.

Department of Pediatrics, Yeungnam University College of Medicine, Yeungnam University Hospital, Daegu, Korea.

出版信息

J Korean Med Sci. 2019 Dec 9;34(47):e303. doi: 10.3346/jkms.2019.34.e303.

Abstract

Propionic acidemia (PA) is a rare organic acidemia resulting from a deficiency of the mitochondrial enzyme propionyl-coenzyme A carboxylase. Most cases are diagnosed after the detection of metabolic abnormalities-such as hyperammonemia, metabolic acidosis, and ketosis-associated with complaints of vomiting, feeding difficulties, and hypotonia during the neonatal period. However, in rare late-onset cases, mild or vague symptoms make the diagnosis more challenging. Even though acute pancreatitis is relatively uncommon in children, it can occur in association with PA. We present the case of a 4-year-old child who was admitted owing to the complaint of recurrent pancreatitis and had not previously been diagnosed with having metabolic disease. During inpatient treatment for acute pancreatitis, convulsions occurred with concomitant hyperammonemia, metabolic acidosis, coagulopathy, and shock 1 week after the administration of total parenteral nutrition. He was diagnosed to have PA after a metabolic work-up and confirmed to have novel mutation by molecular genetic analysis. Because children with PA may have acute pancreatitis, although rare, vomiting and abdominal pain should raise a suspicion of acute pancreatitis. On the contrary, even among children who have never been diagnosed with a metabolic disease, if a child has recurrent pancreatitis, metabolic pancreatitis caused by organic acidemia should be considered.

摘要

丙酸血症(PA)是一种罕见的有机酸血症,由线粒体酶丙酰辅酶 A 羧化酶缺乏引起。大多数病例是在新生儿期出现代谢异常(如高氨血症、代谢性酸中毒和酮症)相关的呕吐、喂养困难和低张力等症状后诊断出来的。然而,在罕见的迟发性病例中,症状轻微或模糊,使诊断更加具有挑战性。尽管儿童急性胰腺炎相对少见,但它可能与 PA 有关。我们报告了一例 4 岁儿童,因反复胰腺炎发作入院,此前未被诊断为代谢疾病。在接受急性胰腺炎住院治疗期间,在接受全肠外营养 1 周后出现抽搐,伴有高氨血症、代谢性酸中毒、凝血功能障碍和休克。在代谢检查后诊断为 PA,并通过分子遗传学分析证实存在新的突变。由于 PA 患儿可能会发生急性胰腺炎,尽管罕见,但呕吐和腹痛应引起对急性胰腺炎的怀疑。相反,即使是那些从未被诊断为代谢疾病的儿童,如果孩子反复发生胰腺炎,也应考虑由有机酸血症引起的代谢性胰腺炎。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/b74e/6900407/5b87d67ed296/jkms-34-e303-g001.jpg

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