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Nader Pouratian, W. Jeff Elias, John A. Jane Jr., Lawrence H. Phillips II and John A. Jane Sr.

Object

Many patients develop neurological symptoms related to spinal cord tethering after perinatal repair of myelomeningocele. This is referred to as secondary tethered cord syndrome (STCS). The authors describe their methodology and evaluate the intraoperative utility and postoperative outcomes of electrophysiologically guided untethering for STCS. In addition, the authors describe the use of electrophysiological guidance to identify an “autonomous placode” in the untethering of the cord in STCS.

Methods

The authors retrospectively identified 46 untethering procedures in 38 patients who had undergone perinatal myelomeningocele repair and in whom the index surgery was for tethered cord release at the site of the repair. In all cases, both passive (electromyography) and active (detection of compound muscle action potentials) electrophysiological monitoring was used. The proximity to neural elements was determined based on the current used; eliciting compound muscle action potentials with a ≤ 10-mA stimulation was assumed to represent direct neural stimulation. Clinical records were reviewed to evaluate the utility of electrophysiological guidance and patient outcomes.

Results

The median age at the time of untethering was 9.5 years (range 0.5–54 years). The median follow-up time was 42 months (range 3–172 months). Progressive bowel and bladder dysfunction, diagnosed either clinically or by cystometrogram, and low-back pain were the most common presenting symptoms. Intraoperative findings indicated that the most common causes of tethering were dense scar (76%) and a tethered placode (39%). Electrophysiological monitoring identified functional neural tissue near tethered elements and provided intraoperative guidance in all cases. In 41% of cases (19 cases), the untethering plan was noted to have been significantly influenced by intraoperative neurophysiological findings. Moreover, an autonomous placode was identified in 6 patients who were nonambulatory preoperatively and had presented with increasing pain and spasticity. In electrophysiologically silent areas, more aggressive dissection and untethering were possible. Symptoms of low-back pain, lower-extremity paresthesia, and lower-extremity spasticity were most likely to improve after untethering surgery (91, 88, and 82%, respectively). Sectioning above the electrophysiologically defined autonomous placode resulted in significant improvement in back pain and lower-extremity spasticity in 5 of 6 patients. There was 1 case of immediate postoperative neurological deterioration (fecal incontinence). All patients remained clinically stable or improved on long-term follow-up, except for 6 (16% of patients) who required a total of 7 additional procedures for recurrent symptoms (median time to repeat surgery 36 months). Complications were noted in 8 cases, including infections and CSF leaks.

Conclusions

Surgical untethering of STCS halts progression and often improves preoperative symptoms. Electrophysiological monitoring, using both a threshold-based interpretation system and continuous electromyography monitoring, provides an efficient, effective, and reliable method for intraoperative guidance, thereby limiting iatrogenic injury and providing a means to identify and untether autonomous placodes. Electrophysiological monitoring also allows for more aggressive dissection and untethering in functionally silent regions, possibly decreasing retethering rates.

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Han Yan, Taylor J. Abel, Naif M. Alotaibi, Melanie Anderson, Toba N. Niazi, Alexander G. Weil, Aria Fallah, John H. Phillips, Christopher R. Forrest, Abhaya V. Kulkarni, James M. Drake and George M. Ibrahim

OBJECTIVE

In this systematic review and meta-analysis the authors aimed to directly compare open surgical and endoscope-assisted techniques for the treatment of sagittal craniosynostosis, focusing on the outcomes of blood loss, transfusion rate, length of stay, operating time, complication rate, cost, and cosmetic outcome.

METHODS

A literature search was performed in compliance with the Preferred Reporting Items for Systematic Reviews and Meta-Analyses (PRISMA) guidelines. Relevant articles were identified from 3 electronic databases (MEDLINE, EMBASE, and CENTRAL [Cochrane Central Register of Controlled Trials]) from their inception to August 2017. The quality of methodology and bias risk were assessed using the Effective Public Health Practice Project Quality Assessment Tool for Quantitative Studies. Effect estimates between groups were calculated as standardized mean differences with 95% CIs. Random and fixed effects models were used to estimate the overall effect.

RESULTS

Of 316 screened records, 10 met the inclusion criteria, of which 3 were included in the meta-analysis. These studies reported on 303 patients treated endoscopically and 385 patients treated with open surgery. Endoscopic surgery was associated with lower estimated blood loss (p < 0.001), shorter length of stay (p < 0.001), and shorter operating time (p < 0.001). From the literature review of the 10 studies, transfusion rates for endoscopic procedures were consistently lower, with significant differences in 4 of 6 studies; the cost was lower, with differences ranging from $11,603 to $31,744 in 3 of 3 studies; and the cosmetic outcomes were equivocal (p > 0.05) in 3 of 3 studies. Finally, endoscopic techniques demonstrated complication rates similar to or lower than those of open surgery in 8 of 8 studies.

CONCLUSIONS

Endoscopic procedures are associated with lower estimated blood loss, operating time, and days in hospital. Future long-term prospective registries may establish advantages with respect to complications and cost, with equivalent cosmetic outcomes. Larger studies evaluating patient- or parent-reported satisfaction and optimal timing of intervention as well as heterogeneity in outcomes are indicated.

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Han Yan, Taylor J. Abel, Naif M. Alotaibi, Melanie Anderson, Toba N. Niazi, Alexander G. Weil, Aria Fallah, John H. Phillips, Christopher R. Forrest, Abhaya V. Kulkarni, James M. Drake and George M. Ibrahim

OBJECTIVE

Despite increasing adoption of endoscopic techniques for repair of nonsagittal single-suture craniosynostosis, the efficacy and safety of the procedure relative to established open approaches are unknown. In this systematic review the authors aimed to directly compare open surgical and endoscope-assisted techniques for the treatment of metopic, unilateral coronal, and lambdoid craniosynostosis, with an emphasis on quantitative reported outcomes.

METHODS

A literature search was performed in compliance with the Preferred Reporting Items for Systematic Reviews and Meta-Analyses (PRISMA) guidelines. Relevant articles were identified from 3 electronic databases (MEDLINE, EMBASE, and CENTRAL [Cochrane Central Register of Controlled Trials]) from their inception to August 2017. The quality of methodology and bias risk were assessed using the Effective Public Health Practice Project (EPHPP) Quality Assessment Tool for Quantitative Studies.

RESULTS

Of 316 screened records, 7 studies were included in a qualitative synthesis of the evidence, of which none were eligible for meta-analysis. These reported on 111 unique patients with metopic, 65 with unilateral coronal, and 12 with lambdoid craniosynostosis. For all suture types, 100 (53%) children underwent endoscope-assisted craniosynostosis surgery and 32 (47%) patients underwent open repair. These studies all suggest that blood loss, transfusion rate, operating time, and length of hospital stay were superior for endoscopically treated children. Although potentially comparable or better cosmetic outcomes are reported, the paucity of evidence and considerable variability in outcomes preclude meaningful conclusions.

CONCLUSIONS

Limited data comparing open and endoscopic treatments for metopic, unilateral coronal, and lambdoid synostosis suggest a benefit for endoscopic techniques with respect to blood loss, transfusion, length of stay, and operating time. This report highlights shortcomings in evidence and gaps in knowledge regarding endoscopic repair of nonsagittal single-suture craniosynostosis, emphasizing the need for further matched-control studies.