Kim Hong-Rae, Suh Yeon-Lim, Kim Jong-Won, Lee Jung-Il
Department of Neurosurgery, Samsung Medical Center, Sungkyunkwan University School of Medicine, Seoul, Korea.
J Korean Med Sci. 2009 Aug;24(4):755-9. doi: 10.3346/jkms.2009.24.4.755. Epub 2009 Jul 30.
We report a very rare case of hemangioblastomatosis that developed after surgical removal of a solitary cerebellar hemangioblastoma (HB). A 51-yr-old man presented with back pain 10 yr after undergoing surgery for cerebellar HB. Magnetic resonance imaging showed numerous mass lesions along the entire neuraxis accompanied by prominent leptomeningeal enhancement. Genomic DNA analysis showed no mutation in the von Hippel-Lindau (VHL) genes. A surgical specimen obtained from a lesion in the cauda equina showed pathological findings identical to those of the cerebellar HB that had been resected 10 yr earlier. External beam radiation therapy and radiosurgery were subsequently performed; however, the patient succumbed one year after receiving the diagnosis of hemangioblastomatosis. The reduction of tumor cell spillage during surgery and regular long-term follow-up are recommended for patients with HBs.
我们报告了一例非常罕见的血管母细胞瘤病病例,该病例发生在孤立性小脑血管母细胞瘤(HB)手术切除之后。一名51岁男性在接受小脑HB手术10年后出现背痛。磁共振成像显示沿整个神经轴有大量肿块病变,并伴有明显的软脑膜强化。基因组DNA分析显示von Hippel-Lindau(VHL)基因无突变。从马尾病变处获取的手术标本显示出与10年前切除的小脑HB相同的病理结果。随后进行了外照射放疗和立体定向放射外科治疗;然而,该患者在被诊断为血管母细胞瘤病一年后死亡。对于HB患者,建议在手术期间减少肿瘤细胞溢出并进行定期长期随访。