Katedra i Oddział Kliniczny Neurochirurgii, Wojewódzki Szpital Specjalistyczny im, Św. Barbary Nr 5, ul Plac Medyków 1, 41-200 Sosnowiec, Poland.
Neurol Sci. 2011 Jun;32(3):491-6. doi: 10.1007/s10072-011-0502-y. Epub 2011 Mar 8.
The article describes paraganglioma case in woman with von Hippel-Lindau disease. She was found to be a carrier of a rare germline mutation in the VHL gene (393C>A; N131K). The patient developed large, untypical for von Hippel-Lindau disease, carotid body paraganglioma at the common carotid artery bifurcation. The carotid body paraganglioma coexisted with the haemangioblastoma situated intramedullary in region C5/C6. The haemangioblastoma reached the right-sided dorsal part of the spinal cord in section C5/C6. It produced radicular symptoms within C5/C6, followed by the later paresis of the right limbs. The haemangioblastoma was resected completely. Twelve months after the operation, the spinal symptoms receded and the carotid body paraganglioma still was asymptomatic. The current case of carotid body paraganglioma in patient with the 393C>A (N131K) missense mutation in the VHL gene, supports association of this specific mutation and VHL disease type 2, and suggests its correlation with susceptibility to paragangliomas.
本文描述了一例患有 von Hippel-Lindau 病的女性嗜铬细胞瘤病例。她被发现携带 VHL 基因(393C>A;N131K)罕见的种系突变。患者在颈总动脉分叉处形成了大的、不典型的 von Hippel-Lindau 病颈动脉体副神经节瘤。颈动脉体副神经节瘤与位于 C5/C6 髓内的血管母细胞瘤并存。血管母细胞瘤延伸至 C5/C6 段的右侧背侧。它在 C5/C6 产生神经根症状,随后是右侧肢体的迟发性瘫痪。血管母细胞瘤被完全切除。术后 12 个月,脊髓症状消退,颈动脉体副神经节瘤仍无症状。本例 VHL 基因 393C>A(N131K)错义突变的颈动脉体副神经节瘤患者,支持该特定突变与 2 型 VHL 病的关联,并提示其与嗜铬细胞瘤易感性相关。