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围产期(胎儿和新生儿)结节性硬化症:综述。

Perinatal (fetal and neonatal) tuberous sclerosis: a review.

机构信息

Department of Pathology, Rady Children's Hospital San Diego, San Diego, California 92123, USA.

出版信息

Am J Perinatol. 2009 Nov;26(10):755-60. doi: 10.1055/s-0029-1223267. Epub 2009 May 18.

Abstract

Tuberous sclerosis (TSC) is an autosomal-dominant disorder that presents with highly variable clinical manifestations including seizures, mental retardation, skin lesions, and hamartomas affecting multiple organ systems such as the heart, brain, eye, and kidney. A 42-year retrospective review of 70 patients consisting of 43 fetuses and 27 neonates with TSC were analyzed. There was a 16% positive family history for the disease. Obvious signs are present in the perinatal period. Cardiac rhabdomyoma(s) detected on routine antenatal sonography, arrhythmias, cerebral lesions, hydrops, and stillbirth are the major presenting findings in the fetus, whereas respiratory distress, arrhythmias, murmurs, and cardiomegaly are the main signs initially in the neonate. Cardiac rhabdomyomas comprised 32.8% of the TSC pathological findings; those of central nervous system origin, 47.5%; and renal cystic disease, 13.2%. Skin lesions and retinal hamartomas were noted rarely at birth. Of the 15 survivors, 87% developed a seizure disorder; 33%, residual cardiac rhabdomyomas; and 27%, mental retardation. The survival rates of patients diagnosed antenatally was practically the same as for those after birth, 21% and 22%, respectively. The overall survival was low, 15/70 or 21%. When TSC occurs in the perinatal period, it is associated with a high incidence of morbidity and mortality.

摘要

结节性硬化症(TSC)是一种常染色体显性遗传病,临床表现高度可变,包括癫痫、智力障碍、皮肤损伤以及影响心脏、大脑、眼睛和肾脏等多个器官系统的错构瘤。对 70 例 TSC 患者(包括 43 例胎儿和 27 例新生儿)的 42 年回顾性研究结果进行了分析。该病的阳性家族史发生率为 16%。围产期存在明显的体征。常规产前超声检查发现心脏横纹肌瘤、心律失常、脑损伤、水肿和死胎是胎儿的主要表现,而呼吸窘迫、心律失常、杂音和心脏增大是新生儿的主要初始体征。心脏横纹肌瘤占 TSC 病理发现的 32.8%,中枢神经系统起源的占 47.5%,囊性肾病占 13.2%。皮肤损伤和视网膜错构瘤很少在出生时出现。15 例幸存者中,87%发生癫痫发作;33%有残留的心脏横纹肌瘤;27%有智力障碍。产前诊断和产后诊断的患者生存率分别为 21%和 22%,基本相同。总的生存率较低,为 15/70 或 21%。当 TSC 发生在围产期时,其发病率和死亡率较高。

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