Hamano K, Tsukamoto H, Yazawa T, Yoshimura M, Takita H
Department of Pediatrics, University of Tsukuba, Japan.
Pediatr Neurol. 1994 Jun;10(4):320-4. doi: 10.1016/0887-8994(94)90130-9.
Two siblings presented with identical features of progressive peripheral paralysis of the lower motor neuron type, pyramidal signs, cranial nerve palsy which included external ocular palsy and deafness, and internal ocular palsy; both died before 1 year of age. Pathologic examination of the central nervous system in both patients revealed degeneration and loss of spinal and cranial nerve motor nuclei, including the oculomotor nucleus. In addition, there was degeneration of the Edinger-Westphal nuclei and demyelination of the corticospinal tract under the midbrain. Although spinal cord lesions were indistinguishable from those of Werdnig-Hoffmann disease, the 2 patients are not considered to have Werdnig-Hoffmann disease from the clinicopathologic findings.
两名兄弟姐妹出现了相同的症状,表现为下运动神经元型进行性周围性麻痹、锥体束征、包括外眼肌麻痹和耳聋在内的颅神经麻痹以及内眼肌麻痹;两人均在1岁前死亡。对两名患者的中枢神经系统进行病理检查发现,脊髓和颅神经运动核包括动眼神经核发生了变性和丧失。此外,动眼神经副核出现变性,中脑下方的皮质脊髓束发生脱髓鞘。尽管脊髓病变与韦尼克-霍夫曼病的病变难以区分,但根据临床病理结果,这两名患者不被认为患有韦尼克-霍夫曼病。