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4p-综合征和伴有唇腭裂的9号染色体四体镶嵌现象。

4p- syndrome and 9p tetrasomy mosaicism with cleft lip and palate.

作者信息

Kobayashi J, Kimijima Y, Yamada S, Amagasa T, Saito-Ohara F

机构信息

Maxillofacial Surgery, Graduate School, Tokyo Medical and Dental University, Yushima, Tokyo, Japan.

出版信息

J Craniomaxillofac Surg. 2000 Jun;28(3):165-70. doi: 10.1054/jcms.2000.0126.

Abstract

Chromosome 4p- syndrome is a multiple malformation syndrome associated with partial deletion of the short arm of chromosome 4 (4p-). It is characterized by dysmorphic features and retarded development. Cleft lip and/or palate are the major clinical manifestations. Cases of tetrasomy 9p are extremely rare; the principal clinical manifestations of this condition are characteristic craniofacial abnormalities, generalized hypotonia and severe mental retardation. We present the first case of a female infant with 4p deletion and tetrasomy 9p mosaicism, exhibiting a left-sided cleft lip, alveolus and soft palate. Karyotype analysis of lymphocytes cultured from the patient revealed that she was mosaic: 86% of the cells were 46, XX, add (4) (p15.32) and 14% were 47, XX, add (4) (p15.32), +idic (9)(q12). The G-banding pattern appeared consistent with either translocation or partial proximal deletion of 4p. In order to make a definitive cytogenetic diagnosis of isodicentric chromosome 9, fluorescence in situ hybridization (FISH) was applied. At 8 months, when the patient weighed 4.3 kg, her cleft lip was repaired. Before and after surgery there were no seizures, and the postoperative course was uneventful.

摘要

4号染色体短臂缺失综合征是一种与4号染色体短臂部分缺失(4p-)相关的多发性畸形综合征。其特征为畸形面容和发育迟缓。唇裂和/或腭裂是主要临床表现。9号染色体四体极为罕见;这种情况的主要临床表现是特征性颅面异常、全身肌张力减退和严重智力迟钝。我们报告首例患有4号染色体缺失和9号染色体四体嵌合体的女婴,表现为左侧唇裂、牙槽突裂和软腭裂。对患者培养的淋巴细胞进行核型分析显示她是嵌合体:86%的细胞为46,XX,add(4)(p15.32),14%为47,XX,add(4)(p15.32),+idic(9)(q12)。G显带模式似乎与4号染色体的易位或部分近端缺失一致。为了对9号等臂染色体做出明确的细胞遗传学诊断,应用了荧光原位杂交(FISH)技术。患儿8个月时体重4.3千克,接受了唇裂修复手术。手术前后均无癫痫发作,术后恢复顺利。

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