Jokiaho I, Salo A, Niemi K M, Blomstedt G C, Pihkala J
Department of Medical Genetics, Finnish Population and Family Welfare Federation, Helsinki.
Hum Genet. 1989 Oct;83(3):302-4. doi: 10.1007/BF00285180.
Deletion 3q27----3qter in an infant is described. A chromosomal abnormality was suspected because of minor facial dysmorphism and closed parietal meningocele. On the first day of life, a large exudative inflammation appeared on the skin of her back, which completely resolved after 1 week. Biopsy showed dilated sweat gland openings resembling miliaria rubra, which has not been previously reported in this age group. It is unclear if the skin change was due to the chromosomal abnormality. The meningocele was repaired at age 8 months. At age 20 months, slight neurodevelopmental delay was evident, the main features being hypertonicity and inability to walk without support. The patient has two healthy sisters, and prometaphase chromosome studies in both parents were normal. This infant represents the first example of del3q27----3qter and the first reported association of meningocele with an abnormality of chromosome 3.
本文描述了一名婴儿的3q27至3q末端缺失。由于轻微面部畸形和闭合性顶叶脑膜膨出,怀疑存在染色体异常。出生第一天,其背部皮肤出现大面积渗出性炎症,1周后完全消退。活检显示汗腺开口扩张,类似痱子,这在该年龄组中此前未见报道。尚不清楚皮肤变化是否由染色体异常所致。8个月大时进行了脑膜膨出修复手术。20个月大时,明显存在轻微神经发育迟缓,主要表现为肌张力亢进和无法独立行走。该患者有两个健康的姐妹,父母的前中期染色体研究均正常。这名婴儿是3q27至3q末端缺失的首例病例,也是首次报道的脑膜膨出与3号染色体异常相关的病例。