Division of Neurology, 2518Cincinnati Children's Hospital, Cincinnati, OH, USA.
Division of Human Genetics, 2518Cincinnati Children's Hospital, Cincinnati, OH, USA.
J Child Neurol. 2021 Apr;36(5):357-364. doi: 10.1177/0883073820972290. Epub 2020 Nov 23.
Holoprosencephaly is the most common malformation of forebrain development and includes a wide spectrum of severity. The objective of this retrospective study was to evaluate fetal magnetic resonance imaging (MRI) associations with outcome. Of the 63 cases identified on antenatal ultrasonography, 28 cases were confirmed on fetal MRI. There were 17 live births; 9 patients died within the first month of life. There were 7 survivors. The vast majority were nonambulatory and required feeding support; none required respiratory support. We found that presence and number of non-holoprosencephaly-associated malformations was also associated with survival. Of 5 patients with 3 or more systemic anomalies, 4 died regardless of holoprosencephaly subtype and 1 was lost to follow-up. Patients with suspected holoprosencephaly on ultrasonography should have full body fetal MRI and echocardiogram to better evaluate systemic anomalies. Counseling should involve pediatric palliative care services to prepare families in caring for babies with limited life span.
无脑回畸形是最常见的前脑发育畸形,其严重程度差异较大。本回顾性研究的目的是评估胎儿磁共振成像(MRI)与结局的相关性。在产前超声检查中确定的 63 例病例中,28 例在胎儿 MRI 上得到证实。有 17 例活产;9 例在出生后第一个月内死亡。有 7 例幸存者。绝大多数患儿不能行走,需要喂养支持;无一例需要呼吸支持。我们发现,非无脑回畸形相关畸形的存在和数量也与存活相关。在 5 例有 3 种或更多全身异常的患者中,4 例死亡,无论无脑回畸形亚型如何,1 例失访。超声检查怀疑无脑回畸形的患者应行全身胎儿 MRI 和超声心动图检查,以更好地评估全身异常。咨询应包括儿科姑息治疗服务,以便为照顾预期寿命有限的婴儿的家庭做好准备。