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史密斯-利姆利-奥皮茨(RSH)综合征的产前表现:30例回顾性调查

Antenatal manifestations of Smith-Lemli-Opitz (RSH) syndrome: a retrospective survey of 30 cases.

作者信息

Goldenberg Alice, Wolf Claude, Chevy Françoise, Benachi Alexandra, Dumez Yves, Munnich Arnold, Cormier-Daire Valérie

机构信息

Department of Medical Genetics, Hôpital Necker-Enfants-Malades, 149 rue de Sèvres, 75043 Paris cedex 15, France.

出版信息

Am J Med Genet A. 2004 Feb 1;124A(4):423-6. doi: 10.1002/ajmg.a.20448.

Abstract

Smith-Lemli-Opitz (SLO) syndrome or RSH syndrome is an autosomal recessive multiple malformation, and mental retardation syndrome ascribed to 7-dehydrocholesterol reductase deficiency, and usually diagnosed in the early postnatal period. Reviewing a series of 30 cases of SLO, we have investigated the variable antenatal expression of the disorder. Intrauterine growth retardation (IUGR) was the most frequent detectable trait (20/30). IUGR was either isolated (9/20) or associated with at least one other anomaly (11/20), including nuchal edema, renal, cardiac, cerebral malformations, genital anomalies, or polydactyly. In this last group, 3/11 presented with multiple malformations (> or =3 anomalies). In 5/30 cases, isolated nuchal edema (3/30), and isolated cardiac (1/30) or renal malformations (1/30) were the only detectable anomalies. Ultrasound findings were considered normal in 5/30 cases and were abnormal in 25/30 cases (83%), but early detection of multiple malformations was rare (3/30, 10%). We suggest giving consideration to a more systematic sterol analysis when dealing with IUGR, especially when associated anomalies are detected.

摘要

史密斯-利姆利-奥皮茨(SLO)综合征或RSH综合征是一种常染色体隐性遗传的多发性畸形和智力发育迟缓综合征,归因于7-脱氢胆固醇还原酶缺乏,通常在出生后早期被诊断出来。回顾30例SLO病例系列,我们研究了该疾病不同的产前表现。宫内生长迟缓(IUGR)是最常见的可检测特征(20/30)。IUGR要么是孤立的(9/20),要么与至少一种其他异常相关(11/20),包括颈部水肿、肾脏、心脏、脑部畸形、生殖器异常或多指畸形。在最后一组中,3/11表现为多发性畸形(≥3种异常)。在5/30的病例中,孤立的颈部水肿(3/30)、孤立的心脏(1/30)或肾脏畸形(1/30)是唯一可检测到的异常。30例中有5例超声检查结果被认为正常,25例(83%)异常,但早期发现多发性畸形很少见(3/30,10%)。我们建议在处理IUGR时,尤其是检测到相关异常时,考虑进行更系统的甾醇分析。

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