Back T, Stoltenburg-Didinger G, Ploner C J, Meisel H, Zschenderlein R
Department of Neurology, Ludwig-Maximilians University, Munich, Germany.
Neurol Res. 1997 Apr;19(2):187-91. doi: 10.1080/01616412.1997.11740794.
A 27 year-old patient developed a progressive neurological multisystem disorder. Initial signs were cerebellar ataxia and dementia, followed by rigidity and oculomotor dysfunction. Myoclonus was not present. MRI showed a marked atrophy of the spinal cord, the cerebellum, and mild (sub)cortical atrophy. CSF contained oligoclonal bands, but no anti-glutamic acid dehydrogenase antibodies. He died 33 months after onset of symptoms. Autopsy revealed widespread neuropathological alterations including perivascular lymphocytic cutting, neuronal cell loss, and micro/astrogliosis the distribution of which corresponded to the changes seen in MRI. The diagnosis of progressive encephalomyelitis with rigidity was pathohistologically confirmed. Brain samples were negative for neurotrophic viruses tested by polymerase chain reaction. A new variant of this rare disorder is described initially presenting with ataxia and dementia, but without myoclonus.
一名27岁患者患上了一种进行性神经多系统疾病。最初症状为小脑共济失调和痴呆,随后出现僵硬和动眼功能障碍。未出现肌阵挛。磁共振成像显示脊髓、小脑明显萎缩,以及轻度(皮质下)萎缩。脑脊液含有寡克隆带,但无抗谷氨酸脱氢酶抗体。患者在症状出现33个月后死亡。尸检发现广泛的神经病理学改变,包括血管周围淋巴细胞浸润、神经元细胞丢失和微/星形胶质细胞增生,其分布与磁共振成像所见变化一致。病理组织学证实为伴有僵硬的进行性脑脊髓炎。通过聚合酶链反应检测,脑样本中未发现神经营养病毒。本文描述了这种罕见疾病的一种新变体,最初表现为共济失调和痴呆,但无肌阵挛。