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G综合征的进一步描述:婴儿吞咽困难的一个可控制的遗传病因。

Further delineation of the G syndrome: a manageable genetic cause of infantile dysphagia.

作者信息

Wilson G N, Oliver W J

机构信息

Department of Pediatrics, Montreal Children's Hospital, McGill University, Quebec, Canada.

出版信息

J Med Genet. 1988 Mar;25(3):157-63. doi: 10.1136/jmg.25.3.157.

Abstract

Three families including five subjects with the G or Opitz-Frias syndrome are added to 23 published cases who had dysphagia; characteristics of the two affected relatives were added to 19 well documented published reports. The data from index cases support the concept of the G syndrome as a constellation of midline defects, which include hypertelorism or telecanthus (89%), oesophageal dysmotility (69%), laryngotracheal clefts (44%), cleft palate or bifid uvula (34%), heart defects (29%), hypospadias (100% of males), renal or ureteral anomalies (42%), and mental retardation (38%). Affected relatives, often identified by hypertelorism, dysphagia, or hypospadias, had a much lower incidence of associated defects and mental retardation. They provide a more rounded but still biased view of a syndrome compatible with normal intelligence and life span. The data do not support a highly characteristic face in the G syndrome, which discriminates it from the phenotypically similar BBB syndrome. The variable expressivity and five cases of male to male transmission observed in 18 families are consistent with autosomal dominant inheritance. Vigilance for the morphological characteristics of G syndrome in patients with dysphagia is underscored by the potential for normal development with appropriate intervention.

摘要

三个家族(共五名患有G综合征或Opitz-Frias综合征的患者)被纳入了已发表的23例吞咽困难病例中;两个受影响亲属的特征被添加到19篇记录详尽的已发表报告中。索引病例的数据支持将G综合征视为一组中线缺陷的概念,这些缺陷包括眼距过宽或内眦距过宽(89%)、食管运动障碍(69%)、喉气管裂(44%)、腭裂或悬雍垂裂(34%)、心脏缺陷(29%)、尿道下裂(男性为100%)、肾脏或输尿管异常(42%)以及智力发育迟缓(38%)。受影响的亲属通常通过眼距过宽、吞咽困难或尿道下裂得以识别,其相关缺陷和智力发育迟缓的发生率要低得多。他们提供了一个更全面但仍有偏差的关于一种与正常智力和寿命相符的综合征的观点。数据不支持G综合征有高度特征性面容的说法,该面容可将其与表型相似的BBB综合征区分开来。在18个家族中观察到的可变表达性以及5例男性向男性的传递与常染色体显性遗传一致。吞咽困难患者对G综合征形态特征的警惕因适当干预后正常发育的可能性而得到强调。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/628e/1015479/303acf0dc47a/jmedgene00065-0014-a.jpg

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