Weaver Kristin J, McDowell Michael M, White Michael D, Tempel Zachary J, Zwagerman Nathan T, Deibert Christopher P, Bonfield Christopher M, Johnson Stephen, Greene Stephanie
Department of Neurosurgery, University of Mississippi Medical Center, Jackson, Mississippi, USA.
Department of Neurosurgery, Children's Hospital of Pittsburgh University of Pittsburgh Medical Center, Pittsburgh, Pennsylvania, USA.
Pediatr Neurosurg. 2021;56(3):229-238. doi: 10.1159/000515038. Epub 2021 Apr 13.
We sought to compare our large single-institution cohort of postnatal myelomeningocele closure to the 2 arms of the Management of Myelomeningocele Study (MOMS) trial at the designated trial time points, as well as assess outcomes at long-term follow-up among our postnatal cohort.
A single-institutional retrospective review of myelomeningocele cases presenting from 1995 to 2015 at Children's Hospital of Pittsburgh was performed. We compared outcomes at 12 and 30 months to both arms of the MOMS trial and compared our cohort's outcomes at those designated time points to our long-term outcomes. Univariate statistical analysis was performed as appropriate.
One-hundred sixty-three patients were included in this study. All patients had at least 2-year follow-up, with a mean follow-up of 10 years (range 2-20 years). There was no difference in the overall distribution of anatomic level of defect. Compared to our cohort, the prenatal cohort had a higher rate of tethering at 12 months of age, 8 versus 1.8%. Conversely, the Chiari II decompression rate was higher in our cohort (10.4 vs. 1.0%). At 30 months, the prenatal cohort had a higher rate of independent ambulation, but our cohort demonstrated the highest rate of ambulation with or without assistive devices among the 3 groups. When comparing our cohort at these early time points to our long-term follow-up data, our cohort's ambulatory function decreased from 84 to 66%, and the rate of detethering surgery increased almost 10-fold.
This study demonstrated that overall ambulation and anatomic-functional level were significantly better among our large postnatal cohort, as well as having significantly fewer complications to both fetus and mother, when compared to the postnatal cohort of the MOMS trial. Our finding that ambulatory ability declined significantly with age in this patient population is worrisome for the long-term outcomes of the MOMS cohorts, especially given the high rates of cord tethering at early ages within the prenatal cohort. These findings suggest that the perceived benefits of prenatal closure over postnatal closure may not be as substantial as presented in the original trial, with the durability of results still remaining a concern.
我们试图在指定的试验时间点，将我们大型单机构队列中的产后脊髓脊膜膨出闭合情况与脊髓脊膜膨出管理研究（MOMS）试验的两组进行比较，并评估我们产后队列的长期随访结果。
对1995年至2015年在匹兹堡儿童医院出现的脊髓脊膜膨出病例进行单机构回顾性研究。我们将12个月和30个月时的结果与MOMS试验的两组进行比较，并将我们队列在这些指定时间点的结果与我们的长期结果进行比较。进行了适当的单变量统计分析。
本研究纳入了163例患者。所有患者至少随访2年，平均随访10年（范围2至20年）。缺损解剖水平的总体分布没有差异。与我们的队列相比，产前队列在12个月大时的脊髓栓系发生率更高，分别为8%和1.8%。相反，我们队列中的Chiari II减压率更高（10.4%对1.0%）。在30个月时，产前队列的独立行走率更高，但在这三组中，我们的队列在使用或不使用辅助装置的情况下行走率最高。当将我们队列在这些早期时间点的数据与我们的长期随访数据进行比较时，我们队列的行走功能从84%下降到66%，脊髓栓系松解手术的发生率增加了近10倍。
本研究表明，与MOMS试验的产后队列相比，我们大型产后队列的总体行走能力和解剖功能水平明显更好，对胎儿和母亲的并发症也明显更少。我们发现该患者群体的行走能力随年龄显著下降，这对于MOMS队列的长期结果来说令人担忧，特别是考虑到产前队列中早期脊髓栓系的高发生率。这些发现表明，产前闭合相对于产后闭合的预期益处可能不如原始试验中所呈现的那么大，结果的持久性仍然是一个问题。
