Harding B N, Leonard J V, Erdohazi M
Eur J Pediatr. 1984 Feb;141(4):215-20. doi: 10.1007/BF00572763.
A detailed autopsy study of three children with ornithine carbamoyl transferase (OCT) deficiency is presented. Although variable in extent, a basic pattern of neuropathological lesions is discernible. Case 1 shows gross cerebral atrophy, cases 2 and 3 milder lesions in the basal nuclei but also multiple cerebellar heterotopias and delayed myelination. We suggest that the findings may provide evidence that OCT deficiency can have a teratogenic effect in utero and suggest that there is a need to monitor the pregnancies of carriers of this disorder.
本文对三名患有鸟氨酸氨甲酰基转移酶(OCT)缺乏症的儿童进行了详细的尸检研究。尽管病变程度各不相同,但仍可辨别出神经病理损伤的基本模式。病例1表现为严重的脑萎缩,病例2和病例3在基底核有较轻的病变,但也有多个小脑异位和髓鞘形成延迟。我们认为,这些发现可能提供证据表明OCT缺乏症在子宫内可能有致畸作用,并表明有必要监测这种疾病携带者的妊娠情况。