Krzystolik K, Cybulski C, Lubiński J
Zakład Genetyki i Patomorfologii, Pomorskiej Akademii Medycznej w Szczecinie.
Neurol Neurochir Pol. 1998 Sep-Oct;32(5):1119-33.
Hippel-Lindau disease is one of inherited tumour susceptibility syndromes. The most common lesions are located in central nervous system, retina and visceral organs. In Poland the disease was rarely diagnosed although the prevalence is much higher than it was supposed and is estimated as 1: 30-50,000. It is inherited in an autosomal dominant manner with age related penetrance reaching almost 98% penetrance at the age of 60 and variable expression. The VHL gene is located near the tip of the short arm of chromosome 3 (3p25-26). Classical lesions in VHL patients are: haemangioblastomas of CNS, retina, cysts and clear cell carcinoma of kidney, cysts and tumours of pancreas, phaeochromocytoma and paraganglioma, papillary cystadenoma of epididymis and endolymphatic sac tumours. Multifocal, often bilateral lesions in form of benign cysts, vascular tumours or carcinomas occur. Management of the lesions often differs from that in sporadic cases of the tumours. Non-symptomatic lesions of CNS need no treatment, neither do non-symptomatic tumours of epididymis and some of phaeochromocytomas. Kidney carcinoma is treated when it reaches a certain size preferably by nephron-sparing surgery. Special care should be provided to pregnant VHL patients. Available DNA testing enables to identify VHL carriers. Although the mean age of death in VHL patients is 41 at the moment a proper prophylactic, diagnostic and treatment management can probably prolong survival of the patients and limit complications of the disease. The coordination between genetic consultants and clinicians is crucial in the management of the patients. The authors coordinate work of Polish VHL Registry and Polish VHL Association.
希佩尔-林道病是一种遗传性肿瘤易感性综合征。最常见的病变位于中枢神经系统、视网膜和内脏器官。在波兰,这种疾病很少被诊断出来,尽管其患病率比预期的要高得多,估计为1:30000 - 50000。它以常染色体显性方式遗传,与年龄相关的外显率在60岁时几乎达到98%,且表现多样。VHL基因位于3号染色体短臂末端附近(3p25 - 26)。VHL患者的典型病变有:中枢神经系统、视网膜的血管母细胞瘤,肾囊肿和透明细胞癌,胰腺囊肿和肿瘤,嗜铬细胞瘤和副神经节瘤,附睾乳头状囊腺瘤和内淋巴囊肿瘤。会出现多灶性、通常为双侧的病变,表现为良性囊肿、血管肿瘤或癌。这些病变的处理方式通常与散发性肿瘤病例不同。中枢神经系统无症状病变无需治疗,附睾无症状肿瘤和一些嗜铬细胞瘤也无需治疗。肾癌在达到一定大小时进行治疗,最好采用保留肾单位手术。应特别关注VHL孕妇患者。现有的DNA检测能够识别VHL携带者。尽管目前VHL患者的平均死亡年龄为41岁,但适当的预防、诊断和治疗管理可能会延长患者的生存期并限制疾病的并发症。遗传咨询顾问和临床医生之间的协调对于患者的管理至关重要。作者协调波兰VHL登记处和波兰VHL协会的工作。