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自主神经病变及脑脊液中蛋白细胞分离作为原发性淀粉样变性的首发特征:一例报告

Autonomic Neuropathy and Albuminocytologic Dissociation in Cerebrospinal Fluid As the Presenting Features of Primary Amyloidosis: A Case Report.

作者信息

Li Jingjing, Li Yi, Chen Hongbing, Xing Shihui, Feng Huiyu, Liu Dawei, Wang Dilong, Zeng Jinsheng, Fan Yuhua

机构信息

Department of Neurology and Stroke Center, The First Affiliated Hospital, Sun Yat-sen University, Guangzhou, China.

Department of Neurology, Guangzhou Red Cross Hospital, Medical College, Jinan University, Guangzhou, China.

出版信息

Front Neurol. 2017 Jul 26;8:368. doi: 10.3389/fneur.2017.00368. eCollection 2017.

Abstract

OBJECTIVE

Primary amyloidosis is a disease with a poor prognosis and multi-organ involvement. Here, we report the clinical and pathological features of a patient with primary amyloidosis featuring autonomic neuropathy as the initial symptom and albuminocytologic dissociation in the cerebrospinal fluid (CSF).

METHODS

The patient was a 60-year-old Chinese male with numbness, orthostatic hypotension, and gastrointestinal symptoms. For diagnosis, we performed an electromyogram (EMG), lumbar puncture, Bence Jones protein urine test, serum electrophoresis blood test, sural nerve and rectal membrane biopsies, transthyretin (TTR) gene sequencing, and bone marrow puncture.

RESULTS

Congo red staining of sural nerve and rectal membrane biopsies showed amyloid deposition and apple-green birefringence was visualized under polarized light microscopy. TTR gene sequencing showed no causative mutation. Following lumbar puncture, normal CSF cell counts and elevated CSF protein concentration (1,680 mg/L) were detected. Bone marrow puncture showed that out of the total number of whole blood cells, 0.56% were abnormal plasma cells and that 87.4% of the total number of plasma cells were abnormal. EMG results showed mixed peripheral nerve damage predominately in the sensory nerve fibers.

CONCLUSION

Obvious symptoms of neuropathy, particularly autonomic neuropathy, albuminocytologic dissociation, and organ function damage suggested a diagnosis of amyloidosis. In such patients, neurologists should use caution to differentiate between chronic inflammatory demyelinating polyneuropathy, primary amyloidosis, and familial amyloid neuropathy.

摘要

目的

原发性淀粉样变性是一种预后不良且多器官受累的疾病。在此,我们报告一例以自主神经病变为首发症状且脑脊液(CSF)出现蛋白细胞分离的原发性淀粉样变性患者的临床及病理特征。

方法

该患者为一名60岁的中国男性,有麻木、体位性低血压及胃肠道症状。为明确诊断,我们进行了肌电图(EMG)、腰椎穿刺、本周氏蛋白尿检、血清电泳血检、腓肠神经及直肠黏膜活检、转甲状腺素蛋白(TTR)基因测序以及骨髓穿刺。

结果

腓肠神经及直肠黏膜活检的刚果红染色显示有淀粉样沉积,在偏振光显微镜下可见苹果绿色双折射。TTR基因测序未发现致病突变。腰椎穿刺后,检测到脑脊液细胞计数正常但脑脊液蛋白浓度升高(1680mg/L)。骨髓穿刺显示,全血细胞总数中,0.56%为异常浆细胞,且浆细胞总数的87.4%为异常。肌电图结果显示主要为感觉神经纤维的混合性周围神经损伤。

结论

明显的神经病变症状,尤其是自主神经病变、蛋白细胞分离及器官功能损害提示淀粉样变性的诊断。对于此类患者,神经科医生应谨慎鉴别慢性炎症性脱髓鞘性多发性神经病、原发性淀粉样变性及家族性淀粉样神经病。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/b4c5/5526844/92f26c98d946/fneur-08-00368-g001.jpg

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