Zarina S. Ali, Robert L. Bailey, Lawrence B. Daniels, Venus Vakhshori, Daniel J. Lewis, Alisha T. Hossain, Karlyndsay Y. Sitterley, John Y. K. Lee, Phillip B. Storm, Gregory G. Heuer and Sherman C. Stein
No clear treatment guidelines for pediatric craniopharyngiomas exist. The authors developed a decision analytical model to evaluate outcomes of 4 surgical approaches for craniopharyngiomas in children, including attempted gross-total resection (GTR), planned subtotal removal plus radiotherapy, biopsy plus radiotherapy, and endoscopic resections of all kinds.
Pooled data, including the authors' own experience, were used to create evidence tables, from which incidence, relative risks, and summary outcomes in quality-adjusted life years (QALYs) were calculated for the 4 management strategies.
Quality-adjusted life years at the 5-year follow-up were 2.3 ± 0.1 for attempted GTR, 2.9 ± 0.2 for planned subtotal removal plus radiotherapy, 3.9 ± 0.2 for biopsy plus radiotherapy, and 3.7 ± 0.2 for endoscopic resection (F = 17,150, p < 0.001). Similarly, QALYs at 10-year follow-up were 4.5 ± 0.2 for attempted GTR, 5.7 ± 0.5 for planned subtotal removal plus radiotherapy, and 7.8 ± 0.5 for biopsy plus radiotherapy (F = 6,173, p < 0.001). On post hoc pairwise comparisons, the differences between all pairs compared were also highly significant (p < 0.001). Since follow-up data at 10 years are lacking for endoscopic cases, this category was excluded from 10-year comparisons.
Biopsy with subsequent radiotherapy is the preferred approach with respect to improved overall quality of life. While endoscopic approaches also show promise in preserving quality of life at five-year follow-up, there are not sufficient data to draw conclusions about this comparison at 10 years.
Zarina S. Ali, Dara Bakar, Yun R. Li, Alex Judd, Hiren Patel, Eric L. Zager, Gregory G. Heuer and Sherman C. Stein
Neonatal brachial plexus palsy (NBPP) represents a significant health problem with potentially devastating consequences. The most common form of NBPP involves the upper trunk roots. Currently, primary surgical repair is performed if clinical improvement is lacking. There has been increasing interest in “early” surgical repair of NBPPs, occurring within 3–6 months of life. However, early treatment recommendations ignore spontaneous recovery in cases of Erb's palsy. This study was undertaken to evaluate the optimal timing of surgical repair in this group with respect to quality of life.
The authors formulated a decision analytical model to compare 4 treatment strategies (no repair or repair at 3, 6, or 12 months of life) for infants with persistent NBPPs. The model derives data from a critical review of published studies and projects health-related quality of life and quality-adjusted life years over a lifetime.
When evaluating the quality of life of infants with NBPP, improved outcomes are seen with delayed surgical repair at 12 months, compared with no repair or repair at early and intermediate time points, at 3 and 6 months, respectively. ANOVA showed that the differences among the 4 groups are highly significant (F = 8369; p < 0.0001). Pairwise post hoc comparisons revealed that there are highly significant differences between each pair of strategies (p < 0.0001). Meta-regression showed no evidence of improved outcomes with more recent treatment dates, compared with older ones, for either nonsurgical or for surgical treatment (p = 0.767 and p = 0.865, respectively).
These data support a delayed approach of primary surgical reconstruction to optimize quality of life. Early surgery for NBPPs may be an overly aggressive strategy for infants who would otherwise demonstrate spontaneous recovery of function by 12 months. A randomized, controlled trial would be necessary to fully elucidate the natural history of NBPP and determine the optimal time point for surgical intervention.
Zarina S. Ali, Eric L. Zager, Gregory G. Heuer and Sherman C. Stein
Willem Pondaag and Martijn J. A. Malessy
Zarina S. Ali, Gregory G. Heuer, Ryan W. F. Faught, Shriya H. Kaneriya, Umar A. Sheikh, Idrees S. Syed, Sherman C. Stein and Eric L. Zager
Adult upper trunk brachial plexus injuries result in significant disability. Several surgical treatment strategies exist, including nerve grafting, nerve transfers, and a combination of both approaches. However, no existing data clearly indicate the most successful strategy for restoring elbow flexion and shoulder abduction in these patients. The authors reviewed the literature to compare outcomes of the three surgical repair techniques listed above to determine the optimal approach to traumatic injury to the upper brachial plexus in adults.
Both PubMed and EMBASE databases were searched for English-language articles containing the MeSH topic “brachial plexus” in conjunction with the word “injury” or “trauma” in the title and “surgery” or “repair” as a MeSH subheading or in the title, excluding pediatric articles and those articles limited to avulsions. The search was also limited to articles published after 1990 and containing at least 10 operated cases involving upper brachial plexus injuries. The search was supplemented with articles obtained through the “Related Articles” feature on PubMed and the bibliographies of selected publications. From the articles was collected information on the operation performed, number of operated cases, mean subject ages, sex distribution, interval between injury and surgery, source of nerve transfers, mean duration of follow-up, year of publication, and percentage of operative success in terms of elbow flexion and shoulder abduction of the injured limb. The recovery of elbow flexion and shoulder abduction was separately analyzed. A subanalysis was also performed to assess the recovery of elbow flexion following various neurotization techniques.
As regards the restoration of elbow flexion, nerve grafting led to significantly better outcomes than either nerve transfer or the combined techniques (F = 4.71, p = 0.0097). However, separating the Oberlin procedure from other neurotization techniques revealed that the former was significantly more successful (F = 82.82, p < 0.001). Moreover, in comparing the Oberlin procedure to nerve grafting or combined procedures, again the former was significantly more successful than either of the latter two approaches (F = 53.14; p < 0.001). In the restoration of shoulder abduction, nerve transfer was significantly more successful than the combined procedure (p = 0.046), which in turn was significantly better than nerve grafting procedures (F = 5.53, p = 0.0044).
According to data in this study, in upper trunk brachial plexus injuries in adults, the Oberlin procedure and nerve transfers are the more successful approaches to restore elbow flexion and shoulder abduction, respectively, compared with nerve grafting or combined techniques. A prospective, randomized controlled trial would be necessary to fully elucidate differences in outcome among the various surgical approaches.
Robert G. Whitmore, Jill N. Curran, Zarina S. Ali, Praveen V. Mummaneni, Christopher I. Shaffrey, Robert F. Heary, Michael G. Kaiser, Anthony L. Asher, Neil R. Malhotra, Joseph S. Cheng, John Hurlbert, Justin S. Smith, Subu N. Magge, Michael P. Steinmetz, Daniel K. Resnick and Zoher Ghogawala
The authors have established a multicenter registry to assess the efficacy and costs of common lumbar spinal procedures using prospectively collected outcomes. Collection of these data requires an extensive commitment of resources from each site. The aim of this study was to determine whether outcomes data from shorter-interval follow-up could be used to accurately estimate long-term outcome following lumbar discectomy.
An observational prospective cohort study was completed at 13 academic and community sites. Patients undergoing single-level lumbar discectomy for treatment of disc herniation were included. SF-36 and Oswestry Disability Index (ODI) data were obtained preoperatively and at 1, 3, 6, and 12 months postoperatively. Quality-adjusted life year (QALY) data were calculated using SF-6D utility scores. Correlations among outcomes at each follow-up time point were tested using the Spearman rank correlation test.
One hundred forty-eight patients were enrolled over 1 year. Their mean age was 46 years (49% female). Eleven patients (7.4%) required a reoperation by 1 year postoperatively. The overall 1-year follow-up rate was 80.4%. Lumbar discectomy was associated with significant improvements in ODI and SF-36 scores (p < 0.0001) and with a gain of 0.246 QALYs over the 1-year study period. The greatest gain occurred between baseline and 3-month follow-up and was significantly greater than improvements obtained between 3 and 6 months or 6 months and 1 year(p < 0.001). Correlations between 3-month, 6-month, and 1-year outcomes were similar, suggesting that 3-month data may be used to accurately estimate 1-year outcomes for patients who do not require a reoperation. Patients who underwent reoperation had worse outcomes scores and nonsignificant correlations at all time points.
This national spine registry demonstrated successful collection of high-quality outcomes data for spinal procedures in actual practice. Three-month outcome data may be used to accurately estimate outcome at future time points and may lower costs associated with registry data collection. This registry effort provides a practical foundation for the acquisition of outcome data following lumbar discectomy.
Luke Macyszyn, Mark Attiah, Tracy S. Ma, Zarina Ali, Ryan Faught, Alisha Hossain, Karen Man, Hiren Patel, Rosanna Sobota, Eric L. Zager and Sherman C. Stein
Moyamoya disease (MMD) is a chronic cerebrovascular disease that can lead to devastating neurological outcomes. Surgical intervention is the definitive treatment, with direct, indirect, and combined revascularization procedures currently employed by surgeons. The optimal surgical approach, however, remains unclear. In this decision analysis, the authors compared the effectiveness of revascularization procedures in both adult and pediatric patients with MMD.
A comprehensive literature search was performed for studies of MMD. Using complication and success rates from the literature, the authors constructed a decision analysis model for treatment using a direct and indirect revascularization technique. Utility values for the various outcomes and complications were extracted from the literature examining preferences in similar clinical conditions. Sensitivity analysis was performed.
A structured literature search yielded 33 studies involving 4197 cases. Cases were divided into adult and pediatric populations. These were further subdivided into 3 different treatment groups: indirect, direct, and combined revascularization procedures. In the pediatric population at 5- and 10-year follow-up, there was no significant difference between indirect and combination procedures, but both were superior to direct revascularization. In adults at 4-year follow-up, indirect was superior to direct revascularization.
In the absence of factors that dictate a specific approach, the present decision analysis suggests that direct revascularization procedures are inferior in terms of quality-adjusted life years in both adults at 4 years and children at 5 and 10 years postoperatively, respectively. These findings were statistically significant (p < 0.001 in all cases), suggesting that indirect and combination procedures may offer optimal results at long-term follow-up.
Jennifer Hong, Jared M. Pisapia, Zarina S. Ali, Austin J. Heuer, Erin Alexander, Gregory G. Heuer and Eric L. Zager
Neurogenic thoracic outlet syndrome (nTOS) is an uncommon compression syndrome of the brachial plexus that presents with pain, sensory changes, and motor weakness in the affected limb. The authors reviewed the clinical presentations and outcomes in their series of pediatric patients with surgically treated nTOS over a 6-year period.
Cases of nTOS in patients age 18 years or younger were extracted for analysis from a prospective database of peripheral nerve operations. Baseline patient characteristics, imaging and neurophysiological data, operative findings, and outcomes and complications were assessed.
Twelve patients with 14 cases of nTOS surgically treated between April 2010 and December 2016 were identified. One-third of the patients were male, and 2 male patients underwent staged, bilateral procedures. Disabling pain (both local and radiating) was the most common presenting symptom (100%), followed by numbness (35.7%), then tingling (28.6%). The mean duration of symptoms prior to surgery was 15.8 ± 6.6 months (mean ± SD). Sports-related onset of symptoms was seen in 78.6% of cases. Imaging revealed cervical ribs in 4 cases, prominent C-7 transverse processes in 4 cases, abnormal first thoracic ribs in 2 cases, and absence of bony anomalies in 4 cases. Neurophysiological testing results were normal in 85.7% of cases. Conservative management failed in all patients, with 5 patients reporting minimal improvement in symptoms with physical therapy. With a mean follow-up after surgery of 22 ± 18.3 months (mean ± SD), pain relief was excellent (> 90%) in 8 cases (57.1%), and good (improved > 50%) in 6 cases (42.9%). On univariate analysis, patients who reported excellent pain resolution following surgery at long-term follow-up were found to be significantly younger, and to have suffered a shorter duration of preoperative symptoms than patients who had worse outcomes. Lack of significant trauma or previous surgery to the affected arm was also associated with excellent outcomes. There were 4 minor complications in 3 patients within 30 days of surgery: 1 patient developed a small pneumothorax that resolved spontaneously; 1 patient suffered a transient increase in pain requiring consultation, followed by hiccups for a period of 3 hours that resolved spontaneously; and 1 patient fell at home, with transient increased pain in the surgically treated extremity. There were no new neurological deficits, wound infections, deep vein thromboses, or readmissions.
Pediatric nTOS commonly presents with disabling pain and is more frequently associated with bony anomalies compared with adult nTOS. In carefully selected patients, surgical decompression of the brachial plexus results in excellent pain relief, which is more likely to be seen in younger patients who present for early surgical evaluation.