Saal H M, Bulas D I, Allen J F, Vezina L G, Walton D, Rosenbaum K N
Division of Human Genetics, Children's Hospital Research Foundation, Children's Hospital Medical Center, Cincinatti, Ohio 45229, USA.
Am J Med Genet. 1995 Jul 17;57(4):573-8. doi: 10.1002/ajmg.1320570411.
Marfanoid phenotype with craniosynostosis (Shprintzen-Goldberg syndrome) is a rare disorder previously described in only 5 patients. We report on the sixth known patient with this condition. The findings which distinguish our patient from others reported previously are that she was ascertained prenatally as having a cloverleaf skull; this is the first female patient described with this condition. Postnatally, she presented with arachnodactyly, camptodactyly, and clover-leaf skull. Imaging studies of the brain documented microcephaly with malformed brain, hydrocephaly, and hypoplasia of the corpus callosum. She also had choanal atresia and stenosis, a clinical finding previously reported only once, in this disorder.
伴有颅缝早闭的马方样表型(施普林曾-戈德堡综合征)是一种罕见疾病,此前仅在5例患者中被描述过。我们报告了第六例已知患有这种疾病的患者。将我们的患者与之前报道的其他患者区分开来的发现是,她在产前被确定患有三叶草颅骨;这是首例被描述患有这种疾病的女性患者。出生后,她表现出蜘蛛指、屈曲指和三叶草颅骨。脑部影像学研究记录了小头畸形伴脑畸形、脑积水和胼胝体发育不全。她还患有后鼻孔闭锁和狭窄,这一临床发现在此疾病中此前仅被报道过一次。