Rees H, Ang L C, Casey R, George D H
Department of Pathology, Vancouver General Hospital, University of British Columbia, Canada.
Pediatr Neurosurg. 1995;22(6):321-7. doi: 10.1159/000120923.
The association of neuroaxonal dystrophy and osteopetrosis is reported in 2 siblings born to non-consanguineous parents. The 1st child was diagnosed as having infantile osteopetrosis shortly after delivery. A computed tomography scan of the head revealed agenesis of the corpus callosum. She died at the age of 9 months. Post-mortem examination showed pneumonia and bony sclerosis. Neuropathological examination revealed cerebral atrophy, ventricular dilation, absence of the corpus callosum, and a small hippocampus. Neuroaxonal spheroids were found in hippocampus, basal ganglia, pons, medulla, spinal cord, cranial nerves, cerebellum, and peripheral nerves. Ultrastructural examination revealed membranous cytoplasmic bodies and electron-dense granular deposits within the neuroaxonal spheroids as well as the soma of neurons. The 2nd child was delivered at 36 weeks of gestation because of intrauterine fetal distress. The diagnosis of osteopetrosis and partial agenesis of the corpus callosum was made shortly after delivery. The child died at 1 month without an autopsy. There are rare cases reported previously with the association of neuroaxonal dystrophy and osteopetrosis. We review these cases and compare them with ours.
据报道,在一对非近亲父母所生的两兄弟中,出现了神经轴索性营养不良与骨硬化症的关联。第一个孩子在出生后不久被诊断为婴儿型骨硬化症。头部计算机断层扫描显示胼胝体发育不全。她在9个月大时死亡。尸检显示有肺炎和骨质硬化。神经病理学检查发现脑萎缩、脑室扩张、胼胝体缺失以及海马体较小。在海马体、基底神经节、脑桥、延髓、脊髓、颅神经、小脑和周围神经中发现了神经轴索球体。超微结构检查显示,神经轴索球体内以及神经元胞体中有膜性细胞质体和电子致密颗粒沉积物。第二个孩子因宫内胎儿窘迫在妊娠36周时出生。出生后不久被诊断为骨硬化症和胼胝体部分发育不全。这个孩子在1个月时死亡,未进行尸检。此前有罕见病例报道过神经轴索性营养不良与骨硬化症的关联。我们回顾这些病例并与我们的病例进行比较。