Suppr超能文献

一名患有冯·希佩尔-林道病的38岁接受血液透析患者的急性髓系白血病。

Acute myeloid leukemia in a 38-year-old hemodialyzed patient with von Hippel-Lindau disease.

作者信息

Labno-Kirszniok Katarzyna, Nieszporek Teresa, Wiecek Andrzej, Helbig Grzegorz, Lubinski Jan

机构信息

Department of Nephrology, Endocrinology and Metabolic Diseases, Medical University of Silesia, Francuska Street 20/24, Katowice 40-027, Poland.

出版信息

Hered Cancer Clin Pract. 2013 Aug 22;11(1):11. doi: 10.1186/1897-4287-11-11.

Abstract

Von Hippel-Lindau disease (VHL disease) is a hereditary cancer predisposition syndrome caused by mutations of the von Hippel-Lindau tumor suppressor gene. The gene product, pVHL, regulates the level of proteins that play a central role in protecting cells against hypoxia. Clinical hallmarks of von Hippel-Lindau disease are the development of central nervous system hemangioblastomas, renal cell carcinoma, pheochromocytoma, neuroendocrine tumors and endolymphatic sac tumors.In this article the case of a 38-year old hemodialyzed patient who became ill with acute myeloid leukemia (AML) three years after being diagnosed with von Hippel-Lindau disease is presented.After cytostatic treatment the patient went into complete hematologic remission but there was still residual disease at the genetic level. After consolidation therapy patient developed bone marrow aplasia and severe pneumonia. Despite intensive treatment the patient died from acute respiratory failure.In this paper we present for the first time a case of von Hippel-Lindau disease associated with acute myeloid leukemia. No evidence of relationship between VHL disease and blood cancers has been demonstrated so far. Despite the fact that there is an increased risk of cancer development in hemodialyzed patients, cancer is a relatively rare cause of death in the dialysed population, and the most common malignancies are genitourinary cancers. It seems likely that development of acute myeloid leukemia in patient with VHL disease can be related to epigenetic alterations of the VHL gene, but further studies are needed.

摘要

冯·希佩尔-林道病(VHL病)是一种遗传性癌症易感综合征,由冯·希佩尔-林道肿瘤抑制基因突变引起。基因产物pVHL调节在保护细胞免受缺氧中起核心作用的蛋白质水平。冯·希佩尔-林道病的临床特征是中枢神经系统血管母细胞瘤、肾细胞癌、嗜铬细胞瘤、神经内分泌肿瘤和内淋巴囊肿瘤的发生。本文介绍了一例38岁的血液透析患者,该患者在被诊断为冯·希佩尔-林道病三年后患上急性髓系白血病(AML)。在进行细胞抑制治疗后,患者实现了完全血液学缓解,但在基因水平上仍有残留疾病。巩固治疗后,患者出现骨髓再生障碍和严重肺炎。尽管进行了强化治疗,患者仍死于急性呼吸衰竭。在本文中,我们首次报告了一例与急性髓系白血病相关的冯·希佩尔-林道病病例。迄今为止,尚未证实VHL病与血癌之间存在关联。尽管血液透析患者患癌症的风险增加,但癌症在透析人群中是相对罕见的死亡原因,最常见的恶性肿瘤是泌尿生殖系统癌症。VHL病患者发生急性髓系白血病似乎可能与VHL基因的表观遗传改变有关,但还需要进一步研究。

相似文献

5
Von Hippel-Lindau disease: a single gene, several hereditary tumors.冯·希佩尔-林道病:一个基因,多种遗传性肿瘤。
J Endocrinol Invest. 2018 Jan;41(1):21-31. doi: 10.1007/s40618-017-0683-1. Epub 2017 Jun 6.

本文引用的文献

2
von Hippel-Lindau disease: a clinical and scientific review.血管母细胞瘤病:临床与科学综述。
Eur J Hum Genet. 2011 Jun;19(6):617-23. doi: 10.1038/ejhg.2010.175. Epub 2011 Mar 9.
3
Regulation of erythropoietin production.促红细胞生成素的产生调节。
J Physiol. 2011 Mar 15;589(Pt 6):1251-8. doi: 10.1113/jphysiol.2010.195057. Epub 2010 Nov 15.
6
The pattern of excess cancer in dialysis and transplantation.透析和移植中癌症过多的模式。
Nephrol Dial Transplant. 2009 Oct;24(10):3225-31. doi: 10.1093/ndt/gfp331. Epub 2009 Jul 8.
8
Methylation analysis of the von Hippel-Lindau gene in acute myeloid leukaemia and myelodysplastic syndromes.
Leukemia. 2008 Jun;22(6):1293-5. doi: 10.1038/sj.leu.2405053. Epub 2007 Dec 20.
10
Disturbances of acquired immunity in hemodialysis patients.血液透析患者获得性免疫紊乱
Semin Dial. 2007 Sep-Oct;20(5):440-51. doi: 10.1111/j.1525-139X.2007.00283.x.

文献检索

告别复杂PubMed语法,用中文像聊天一样搜索,搜遍4000万医学文献。AI智能推荐,让科研检索更轻松。

立即免费搜索

文件翻译

保留排版,准确专业,支持PDF/Word/PPT等文件格式,支持 12+语言互译。

免费翻译文档

深度研究

AI帮你快速写综述,25分钟生成高质量综述,智能提取关键信息,辅助科研写作。

立即免费体验