Kole Matthew J, Fridley Jared S, Jea Andrew, Bollo Robert J
Department of Neurosurgery, Baylor College of Medicine;
J Neurosurg Pediatr. 2014 Jun;13(6):685-9. doi: 10.3171/2014.3.PEDS13534. Epub 2014 Apr 18.
Currarino syndrome is a rare constellation of congenital anomalies characterized by the triad of sacral dysgenesis, presacral mass, and anorectal malformation. It is frequently associated with other congenital anomalies, often including occult spinal dysraphism. Mutations in the MNX1 gene are identified in the majority of cases. The authors report a rare case of Currarino syndrome in an infant with tethered cord syndrome and a dorsal lipomyelomeningocele continuous with a presacral intradural spinal lipoma, in addition to an imperforate anus and a scimitar sacrum. They review the literature to highlight patterns of occult spinal dysraphism in patients with Currarino syndrome and their relationship to tethered cord syndrome. Approximately 60% of the patients with Currarino syndrome reported in the literature have an occult spinal dysraphism. Published studies suggest that the risk of tethered cord syndrome may be higher among patients with a lipoma and lower among those with a teratoma or anterior meningocele.
库拉里诺综合征是一种罕见的先天性异常组合,其特征为骶骨发育不全、骶前肿块和肛门直肠畸形三联征。它常与其他先天性异常相关,通常包括隐性脊柱裂。大多数病例中可检测到MNX1基因突变。作者报告了1例罕见的库拉里诺综合征婴儿病例,该婴儿除肛门闭锁和弯刀状骶骨外,还伴有脊髓拴系综合征以及与骶前硬脊膜内脊髓脂肪瘤相连的背侧脂肪脊髓脊膜膨出。他们回顾文献以强调库拉里诺综合征患者隐性脊柱裂的模式及其与脊髓拴系综合征的关系。文献报道的库拉里诺综合征患者中约60%患有隐性脊柱裂。已发表的研究表明,脂肪瘤患者发生脊髓拴系综合征的风险可能较高,而畸胎瘤或前脑膨出患者的风险较低。