Cantú综合征的进一步病例:排除隐性亚端粒染色体畸变。

Hartmut Engels, Kristin Bosse, Antje Ehrbrecht, Susanne Zahn, Alexander Hoischen, Peter Propping, Lutz Bindl, Heiko Reutter
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引用次数: 26

摘要

Cantú综合征包括多毛症、骨软骨发育不良和心脏肥大,迄今已有18例患者报道。我们报告一个患有Cantú综合征的婴儿。除了典型的表现外,他有相对轻微的放射学和心脏学表现。先前未描述的发现包括幽门狭窄和碱性磷酸酶水平升高。脑部扫描显示双侧丘脑纹状动脉钙化,外腔和侧脑室变宽。由于该患者是迄今为止报道的最年轻的患者,我们的研究结果完善了新生儿和年幼婴儿Cantú综合征的临床谱。Cantú综合征的病因和遗传方式尚不清楚。大多数病例是散发的。微缺失被认为是Cantú综合征的可能原因。近年来,一些多发性先天性异常和智力低下综合征已被证明是由亚端粒染色体畸变引起的。我们通过荧光原位杂交(FISH)筛选基因座特异性探针,排除了患者隐性亚端粒染色体异常的存在。
本文章由计算机程序翻译,如有差异,请以英文原文为准。
Further case of Cantú syndrome: exclusion of cryptic subtelomeric chromosome aberrations.

Cantú syndrome consists of hypertrichosis, osteochondrodysplasia, and cardiomegaly, and has been reported in 18 patients to date. We report an infant with Cantú syndrome. In addition to typical findings, he had relatively mild radiological and cardiological manifestations. Previously undescribed findings included pyloric stenosis and elevated alkaline phosphatase levels. Brain scans showed bilateral calcification of the Arteriae thalamostriatae and widening of the outer liquor spaces and lateral ventricles. Because the propositus is the youngest patient reported to date, our findings refine the clinical spectrum of Cantú syndrome in neonates and young infants. The etiology and mode of inheritance of Cantú syndrome are unknown. Most cases are sporadic. Microdeletions have been discussed as a possible cause of Cantú syndrome. Recently, several syndromes with multiple congenital anomalies and mental retardation have been shown to be caused by subtelomeric chromosome aberrations. We excluded the presence of a cryptic subtelomeric chromosome anomaly in our patient by fluorescence in situ hybridization (FISH) screening with locus-specific probes.

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