Magenis R E, Brown M G, Lacy D A, Budden S, LaFranchi S
Department of Medical Genetics, University Hospitals, Portland, Oregon.
Am J Med Genet. 1987 Dec;28(4):829-38. doi: 10.1002/ajmg.1320280407.
Two unrelated females, age 15 and 5 years respectively, were studied cytogenetically because of severe mental retardation, seizures and ataxia-like incoordination. A similar deletion of the proximal long arm of chromosome 15 was found in both patients. Re-evaluation showed no voracious appetite or obesity; normal size of hands and feet, minimal to no hypotonia by history or examination and facial features not typical of the Prader-Willi syndrome. However, the facial appearance of the girls was similar to each other with mild hypertelorism. The similarity of these girls and dissimilarity to Prader-Willi syndrome suggest a different syndrome, perhaps the result of deletion of a different segment of 15q. The findings of ataxic-like movements, frequent, unprovoked and prolonged bouts of laughter and facial appearance are more compatible with the diagnosis of Angelman syndrome.
两名无血缘关系的女性,年龄分别为15岁和5岁,因严重智力发育迟缓、癫痫发作和共济失调样不协调而接受细胞遗传学研究。在两名患者中均发现15号染色体长臂近端存在类似的缺失。重新评估显示没有贪食或肥胖;手脚大小正常,根据病史或检查肌张力轻微或无异常,面部特征也不典型,不属于普拉德-威利综合征。然而,这两名女孩的面部外观彼此相似,有轻度眼距增宽。这些女孩的相似性以及与普拉德-威利综合征的不同表明存在一种不同的综合征,可能是15q不同片段缺失的结果。共济失调样运动、频繁、无端且持续的大笑发作以及面部外观等表现更符合天使综合征的诊断。