老年黑猩猩(类人猿)多葡聚糖体疾病的研究。

IF 1.3 4区 医学 Q4 CLINICAL NEUROLOGY Neuropathology Pub Date : 2023-12-01 Epub Date: 2023-04-21 DOI:10.1111/neup.12906
Sanjeev Gumber, Fawn Connor-Stroud, Dustin Howard, Xiaodong Zhang, Brenda J Bradley, Chet C Sherwood, Lary C Walker
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引用次数: 0

摘要

一只57岁的雌性黑猩猩出现了短暂的嗜睡史和迅速进行性下肢无力,最终丧失使用能力。死后检查未发现神经系统或其他器官系统的明显损伤。组织学分析显示,在脊髓的颈、胸、腰椎和尾椎区域的白质和灰质中存在圆形的、嗜碱性到嗜两性的葡聚糖小体(PGBs)。仅在前脑样本中观察到罕见的PGBs。脊髓内病变呈多形性,苏木精染色、周期性酸希夫染色、阿利新蓝染色、甲苯胺蓝染色、Bielschowsky银染色、grocotto - gomori甲胺银染色呈阳性,von Kossa染色、刚果红染色呈阴性。免疫组化评价显示泛素抗体反应性,但胶质纤维酸性蛋白、神经元特异性烯醇化酶、神经丝、tau蛋白和Aβ蛋白均阴性。电镜显示,在轴突内和细胞外空间有由密集堆积的细丝组成的非膜结合沉积物。轴突内PGBs与轴突精细结构的破坏和周围髓鞘的解体有关。这些发现是对与黑猩猩神经功能障碍有关的PGBs的首次描述。临床病理上,该疾病类似于人类成人PGB病。
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Polyglucosan body disease in an aged chimpanzee (Pan troglodytes).

A 57-year-old female chimpanzee presented with a brief history of increasing lethargy and rapidly progressive lower-limb weakness that culminated in loss of use. Postmortem examination revealed no significant gross lesions in the nervous system or other organ systems. Histological analysis revealed round, basophilic to amphophilic polyglucosan bodies (PGBs) in the white and gray matter of the cervical, thoracic, lumbar, and coccygeal regions of spinal cord. Only rare PGBs were observed in forebrain samples. The lesions in the spinal cord were polymorphic, and they were positively stained with hematoxylin, periodic acid Schiff, Alcian blue, toluidine blue, Bielschowsky silver, and Grocott-Gomori methenamine-silver methods, and they were negative for von Kossa and Congo Red stains. Immunohistochemical evaluation revealed reactivity with antibodies to ubiquitin, but they were negative for glial fibrillary acidic protein, neuron-specific enolase, neurofilaments, tau protein, and Aβ protein. Electron microscopy revealed non-membrane-bound deposits composed of densely packed filaments within axons and in the extracellular space. Intra-axonal PGBs were associated with disruption of the axonal fine structure and disintegration of the surrounding myelin sheath. These findings are the first description of PGBs linked to neurological dysfunction in a chimpanzee. Clinicopathologically, the disorder resembled adult PGB disease in humans.

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来源期刊
Neuropathology
Neuropathology 医学-病理学
CiteScore
4.10
自引率
4.30%
发文量
105
审稿时长
6-12 weeks
期刊介绍: Neuropathology is an international journal sponsored by the Japanese Society of Neuropathology and publishes peer-reviewed original papers dealing with all aspects of human and experimental neuropathology and related fields of research. The Journal aims to promote the international exchange of results and encourages authors from all countries to submit papers in the following categories: Original Articles, Case Reports, Short Communications, Occasional Reviews, Editorials and Letters to the Editor. All articles are peer-reviewed by at least two researchers expert in the field of the submitted paper.
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