Houston C S, Zaleski W A, Rozdilsky B
Am J Med Genet. 1982 Oct;13(2):211-23. doi: 10.1002/ajmg.1320130212.
The clinical, radiological, and neuropathological findings in early onset Cockayne syndrome are illustrated in identical twins and their brother. Their appearance of dwarfism with small head and prominent beaked nose strongly resembled that seen in the Seckel syndrome, but unlike patients with that syndrome they had a normal birth weight (for twins), thick cranial vaults, intracranial calcification, and a severe degree of mental retardation. The twins were deaf and blind, with optic atrophy and retinal pigmentation, while their brother had cataracts. Their hands and feet were large in proportion to their small trunk. They had cutaneous sensitivity to any slight exposure to ultraviolet light and severe neurologic problems with incoordination and spasticity. Radiologic findings included microcephaly, a thick cranial vault, a small pelvis, coxa valga, and "ivory epiphyses" in terminal phalanges of hands and feet. Pathologic findings included atrophy of white matter with widespread patchy demyelination, and massive siderocalcific deposits in the brain, particularly in the basal ganglia and cerebellum. While autosomal recessive inheritance is most likely, formally X-linked inheritance cannot be excluded.
早发型科凯恩综合征的临床、放射学和神经病理学表现,在一对同卵双胞胎及其兄弟身上得到了体现。他们身材矮小、头部小且喙状鼻突出,其外貌与塞克尔综合征患者极为相似,但与该综合征患者不同的是,他们出生体重正常(双胞胎而言)、颅穹隆厚、颅内钙化且智力发育迟缓程度严重。这对双胞胎失聪失明,伴有视神经萎缩和视网膜色素沉着,而他们的兄弟患有白内障。他们的手脚相对于短小的躯干来说较大。他们的皮肤对任何轻微的紫外线照射都敏感,并且存在严重的神经问题,表现为共济失调和痉挛。放射学检查结果包括小头畸形、颅穹隆增厚、骨盆小、髋外翻以及手足末端指骨的“象牙样骨骺”。病理学检查结果包括白质萎缩伴广泛散在性脱髓鞘,以及大脑中大量含铁钙沉着物,尤其是在基底神经节和小脑中。虽然最可能是常染色体隐性遗传,但也不能排除X连锁遗传的可能性。