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新生儿筛查时代遗传性酪氨酸血症患者发生肝细胞癌的病例。

Case of hepatocellular carcinoma in a patient with hereditary tyrosinemia in the post-newborn screening era.

作者信息

Imseis Essam M, Bynon John S, Thornhill Chad

机构信息

Essam M Imseis, Chad Thornhill, Department of Pediatrics, Gastroenterology Division, the University of Texas Health Science Center at Houston, Houston McGovern Medical School, Houston, TX 77030, United States.

出版信息

World J Hepatol. 2017 Mar 28;9(9):487-490. doi: 10.4254/wjh.v9.i9.487.

Abstract

Hereditary tyrosinemia type 1 (HT-1) is a metabolic disorder caused by a defect in tyrosine degradation. Without treatment, symptoms of hepatomegaly, renal tubular dysfunction, growth failure, neurologic crises resembling porphyrias, rickets and possible hepatocellular carcinoma can develop. The use of 2-(2-nitro-4-trifluoromethylbenzoyl)-1,3-cyclohexanedione and early diagnosis through newborn screening initiatives have resulted in a sharp decline in morbidity and mortality associated with this disease. We present a case report of a 7-year-old patient with HT-1 who was born prior to the addition of tyrosinemia to the newborn screening in her birth area. At her time of diagnosis, the patient had developed many of the symptoms associated with her disease, including chronic kidney disease, rickets, and myopathy that left her non-ambulatory. During her initial evaluation, she was also noted to have hepatocellular carcinoma. With cadaveric liver transplantation and nutritional support, her symptoms all either resolved or stabilized. Her case illustrates the severity of the disease if left untreated, the need for vigilance in populations who do not routinely receive newborn screens, and the markedly improved outcomes in patients following transplant.

摘要

1型遗传性酪氨酸血症(HT - 1)是一种由酪氨酸降解缺陷引起的代谢紊乱疾病。若不进行治疗,可出现肝肿大、肾小管功能障碍、生长发育迟缓、类似卟啉病的神经危机、佝偻病以及可能的肝细胞癌等症状。使用2 -(2 - 硝基 - 4 - 三氟甲基苯甲酰基)- 1,3 - 环己二酮以及通过新生儿筛查项目进行早期诊断,已使该疾病相关的发病率和死亡率大幅下降。我们报告一例7岁HT - 1患者的病例,该患者出生时其出生地尚未将酪氨酸血症纳入新生儿筛查项目。在诊断时,患者已出现许多与该疾病相关的症状,包括慢性肾病、佝偻病和肌病,导致她无法行走。在初次评估时,还发现她患有肝细胞癌。通过尸体肝移植和营养支持,她的症状全部得到缓解或稳定。她的病例说明了若不治疗该疾病的严重性、对未常规接受新生儿筛查人群保持警惕的必要性,以及移植后患者的预后显著改善。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/39bb/5368626/efd760258df1/WJH-9-487-g001.jpg

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