Labno-Kirszniok Katarzyna, Nieszporek Teresa, Wiecek Andrzej, Helbig Grzegorz, Lubinski Jan
Department of Nephrology, Endocrinology and Metabolic Diseases, Medical University of Silesia, Francuska Street 20/24, Katowice 40-027, Poland.
Hered Cancer Clin Pract. 2013 Aug 22;11(1):11. doi: 10.1186/1897-4287-11-11.
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.
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。