Minn David, Christmann Dominique, De Saint-Martin Anne, Alembik Yves, Eliot Mylène, Mack Geneviève, Fischbach Michel, Flament Jacques, Veillon Francis, Dollfus Hélène
Fédération de Pédiatrie, Hôpitaux Universitaires de Strasbourg, Strasbourg, France.
Am J Med Genet. 2002 May 1;109(3):211-7. doi: 10.1002/ajmg.10348.
Schinzel-Giedion syndrome is a rare multiple congenital malformation syndrome defined by an evocative midfacial retraction, kidney and urinary malformations and multiple skeletal abnormalities associated to a recently described neurodegenerative process. Two children with SGS are reported with identical clinical findings: megacalycosis, progressive neurodegeneration with infantile spasms and hypsarrhymtic activity. Ocular investigations revealed alacrimia and corneal hypoesthesia. Computed tomography of the temporal bone showed a tuning-fork malformation of the stapes for both children. These features may contribute to further delineation of SGS as additional clinical criteria.
申泽尔-吉迪恩综合征是一种罕见的多发性先天性畸形综合征,其特征为引人注目的面中部凹陷、肾脏和泌尿系统畸形以及与最近描述的神经退行性过程相关的多种骨骼异常。报告了两名患有申泽尔-吉迪恩综合征的儿童,他们具有相同的临床表现:肾盏巨大症、伴有婴儿痉挛和高度节律失调活动的进行性神经退行性变。眼部检查发现泪液缺乏和角膜感觉减退。两名儿童的颞骨计算机断层扫描均显示镫骨呈音叉畸形。这些特征可能有助于作为额外的临床标准进一步明确申泽尔-吉迪恩综合征。