Smith A C, McGavran L, Robinson J, Waldstein G, Macfarlane J, Zonona J, Reiss J, Lahr M, Allen L, Magenis E
Am J Med Genet. 1986 Jul;24(3):393-414. doi: 10.1002/ajmg.1320240303.
We describe a new and distinct syndrome involving an interstitial deletion of short arm of chromosome 17 in nine unrelated patients (six males; three females) ranging in age from 3 months to 65 years. In eight patients, a deletion of a portion of band 17p11.2 was associated with a striking similar phenotype including brachycephaly, midface hypoplasia, prognathism, hoarse voice, and speech delay with or without hearing loss, psychomotor and growth retardation, and behavior problems. The one patient with a complete deletion of band 17p11.2 was more severely affected with facial malformations, cleft palate, and major anomalies of cardiac, skeletal, and genitourinary systems; the patient died at age 6 months. Careful cytogenetic analysis including high-resolution techniques will be important for the further identification of patients with this previously unrecognized deletion syndrome.
我们描述了一种新的独特综合征,该综合征涉及9名年龄从3个月至65岁的无关患者(6名男性;3名女性)的17号染色体短臂间质性缺失。在8名患者中,17p11.2带部分缺失与一种显著相似的表型相关,包括短头畸形、面中部发育不全、凸颌、声音嘶哑、伴有或不伴有听力丧失的语言发育迟缓、精神运动和生长发育迟缓以及行为问题。1名17p11.2带完全缺失的患者受面部畸形、腭裂以及心脏、骨骼和泌尿生殖系统主要异常的影响更为严重;该患者于6个月龄时死亡。包括高分辨率技术在内的仔细细胞遗传学分析对于进一步识别患有这种先前未被认识的缺失综合征的患者很重要。