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胎儿中脑同种异体移植治疗帕金森病后出现致命性囊肿形成。

Fatal cyst formation after fetal mesencephalic allograft transplant for Parkinson's disease.

作者信息

Mamelak A N, Eggerding F A, Oh D S, Wilson E, Davis R L, Spitzer R, Hay J A, Caton W L

机构信息

Department of Medicine, Huntington Memorial Hospital, Pasadena, California, USA.

出版信息

J Neurosurg. 1998 Oct;89(4):592-8. doi: 10.3171/jns.1998.89.4.0592.

Abstract

OBJECT

In recent years, fetal mesencephalic tissue transplant for the treatment of Parkinson's disease (PD) has been demonstrated to hold promise, but potential complications related to growth of allograft tissue have not been well described. This report explores the development and possible causation of a fatal cyst arising from a fetal transplant in the brain.

METHODS

The authors report the case of a 52-year-old woman who underwent bilateral putamenal fetal mesencephalic allograft transplant for PD at another hospital. Twenty-three months later she presented to the authors' institution in a coma. Admission computerized tomography and magnetic resonance (MR) studies revealed a contrast-enhancing mural nodule and associated large cyst arising from the left putamen and causing brainstem compression. Despite surgical decompression of the cyst, the patient did not regain consciousness. Biopsy and autopsy specimens were obtained, along with an analysis of the cyst fluid. Genotyping of the nodule and the patient's peripheral lymphocytes by using polymerase chain reaction-based microsatellite analysis was also performed. Biopsy samples and autopsy histopathological studies showed inflammatory cells, hemosiderin-laden macrophages, and astrocytosis. Scattered neurons and multiple rests of choroid plexus were also noted. The cyst had a thin wall and contained liquid that was identical in composition to cerebrospinal fluid (CSF). Genotyping demonstrated the presence of alleles in the nodule DNA that were not present in lymphocytic DNA, indicating that the nodule contained allograft tissue.

CONCLUSIONS

The authors hypothesize that the choroid plexus tissue contained in the allograft resulted in CSF production and cyst formation at the transplant site, ultimately leading to the patient's herniation syndrome. The clinical history and large size of the mural nodule indicate slow growth of this allograft site and cyst over time. This case demonstrates that unusual patterns of tissue growth can occur in the brain after fetal tissue transplant and emphasizes the need for long-term monitoring of posttransplant patients by means of MR imaging. Cell sorting should be considered to ensure transplant of pure neuronal and astroglial populations.

摘要

目的

近年来,胎儿中脑组织移植治疗帕金森病(PD)已显示出前景,但与同种异体移植组织生长相关的潜在并发症尚未得到充分描述。本报告探讨了脑内胎儿移植后致命囊肿的发生发展及可能病因。

方法

作者报告了一名52岁女性的病例,该患者在另一家医院接受了双侧壳核胎儿中脑同种异体移植治疗PD。23个月后,她昏迷状态被送至作者所在机构。入院时的计算机断层扫描和磁共振(MR)研究显示,左侧壳核有一个强化的壁结节及相关的大囊肿,导致脑干受压。尽管对囊肿进行了手术减压,但患者仍未恢复意识。获取了活检和尸检标本,并对囊液进行了分析。还通过基于聚合酶链反应的微卫星分析对结节和患者外周淋巴细胞进行了基因分型。活检样本和尸检组织病理学研究显示有炎性细胞、含铁血黄素巨噬细胞和星形细胞增生。还发现了散在的神经元和多处脉络丛遗迹。囊肿壁薄,囊液成分与脑脊液(CSF)相同。基因分型显示结节DNA中存在淋巴细胞DNA中不存在的等位基因,表明结节含有同种异体移植组织。

结论

作者推测,同种异体移植中所含的脉络丛组织导致移植部位产生脑脊液并形成囊肿,最终导致患者出现脑疝综合征。壁结节的临床病史和较大尺寸表明该同种异体移植部位和囊肿随时间缓慢生长。本病例表明,胎儿组织移植后大脑中可能出现异常的组织生长模式,并强调需要通过MR成像对移植后患者进行长期监测。应考虑细胞分选以确保移植的是纯神经元和星形胶质细胞群体。

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