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Sean N. Neifert, Lauren K. Grant, Jonathan J. Rasouli, Ian Thomas McNeill, Samuel K. Cho, and John M. Caridi

This report describes a 42-year-old man who presented with an α-type spinal deformity with a Cobb angle of 224.9° and associated spinal cord rotation greater than 90°. Preoperative imaging revealed extensive spinal deformity, and 3D modeling confirmed the α-type nature of his deformity. Intraoperative photography demonstrated spinal cord rotation greater than 90°, which likely contributed to the patient’s poor neurological status. Reports of patients with Cobb angles ≥ 100° are rare, and to the authors’ knowledge, there have been no published cases of adult α-type spinal deformity. Furthermore, very few cases or case series of spinal cord rotation have been published previously, with no single patient having rotation greater than 90° to the authors’ knowledge. Given these two rarities presenting in the same patient, this report can provide important insights into the operative management of this difficult form of spinal deformity.

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Sean N. Neifert, Hammad A. Khan, David B. Kurland, Nora C. Kim, Kaleb Yohay, Devorah Segal, Amer Samdani, Steven Hwang, and Darryl Lau

OBJECTIVE

Neurofibromatosis type 1 (NF1) dystrophic scoliosis is an early-onset, rapidly progressive multiplanar deformity. There are few studies on the surgical management of this patient population. Specifically, perioperative morbidity, instrument-related complications, and quality-of-life outcomes associated with surgical management have not been systematically evaluated. In this study, the authors aimed to perform a systematic review on the natural history, management options, and surgical outcomes in patients who underwent NF1 dystrophic scoliosis surgery.

METHODS

A PubMed search for articles with “neurofibromatosis” and either “dystrophic” or “scoliosis” in the title or abstract was performed. Articles with 10 or more patients undergoing surgery for NF1 dystrophic scoliosis were included. Data regarding indications, treatment details, morbidity, and outcomes were summarized and analyzed with descriptive statistics.

RESULTS

A total of 310 articles were identified, 48 of which were selected for full-text review; 30 studies describing 761 patients met the inclusion criteria. The mean age ranged from 7 to 22 years, and 99.7% of patients were younger than 18 years. The mean preoperative coronal Cobb angle was 75.2°, and the average correction achieved was 40.3°. The mean clinical follow-up in each study was at least 2 years (range 2.2–19 years). All patients underwent surgery with the intent of deformity correction. The scoliosis regions addressed were thoracic curves (69.6%) and thoracolumbar (11.1%) and lumbar (14.3%) regions. The authors reported on a variety of approaches: posterior-only, combined anterior-posterior, and growth-friendly surgery. For fixation techniques, 42.5% of patients were treated with hybrid constructs, 51.5% with pedicle screw–only constructs, and 6.0% with hook-based constructs. Only 0.9% of patients underwent a vertebral column resection. The nonneurological complication rate was 14.0%, primarily dural tears and wound infections. The immediate postoperative neurological deficit rate was 2.1%, and the permanent neurological deficit rate was 1.2%. Ultimately, 21.5% required revision surgery, most commonly for implant-related complications. Loss of correction in both the sagittal and coronal planes commonly occurred at follow-up. Five papers supplied validated patient-reported outcome measures, showing improvement in the mental health, self-image, and activity domains.

CONCLUSIONS

Data on the surgical outcomes of dystrophic scoliosis correction are heterogeneous and sparse. The perioperative complication rate appears to be high, although reported rates of neurological deficits appear to be lower than clinically observed and may be underreported. The incidence of implant-related failures requiring revision surgery is high. There is a great need for multicenter prospective studies of this complex type of deformity.

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Michael L. Martini, Sean N. Neifert, William H. Shuman, Emily K. Chapman, Alexander J. Schüpper, Eric K. Oermann, J Mocco, Michael Todd, James C. Torner, Andrew Molyneux, Stephan Mayer, Peter Le Roux, Mervyn D. I. Vergouwen, Gabriel J. E. Rinkel, George K. C. Wong, Peter Kirkpatrick, Audrey Quinn, Daniel Hänggi, Nima Etminan, Walter M. van den Bergh, Blessing N. R. Jaja, Michael Cusimano, Tom A. Schweizer, Jose I. Suarez, Hitoshi Fukuda, Sen Yamagata, Benjamin Lo, Airton Leonardo de Oliveira Manoel, Hieronymus D. Boogaarts, R. Loch Macdonald, and

OBJECTIVE

Rescue therapies have been recommended for patients with angiographic vasospasm (aVSP) and delayed cerebral ischemia (DCI) following subarachnoid hemorrhage (SAH). However, there is little evidence from randomized clinical trials that these therapies are safe and effective. The primary aim of this study was to apply game theory–based methods in explainable machine learning (ML) and propensity score matching to determine if rescue therapy was associated with better 3-month outcomes following post-SAH aVSP and DCI. The authors also sought to use these explainable ML methods to identify patient populations that were more likely to receive rescue therapy and factors associated with better outcomes after rescue therapy.

METHODS

Data for patients with aVSP or DCI after SAH were obtained from 8 clinical trials and 1 observational study in the Subarachnoid Hemorrhage International Trialists repository. Gradient boosting ML models were constructed for each patient to predict the probability of receiving rescue therapy and the 3-month Glasgow Outcome Scale (GOS) score. Favorable outcome was defined as a 3-month GOS score of 4 or 5. Shapley Additive Explanation (SHAP) values were calculated for each patient-derived model to quantify feature importance and interaction effects. Variables with high SHAP importance in predicting rescue therapy administration were used in a propensity score–matched analysis of rescue therapy and 3-month GOS scores.

RESULTS

The authors identified 1532 patients with aVSP or DCI. Predictive, explainable ML models revealed that aneurysm characteristics and neurological complications, but not admission neurological scores, carried the highest relative importance rankings in predicting whether rescue therapy was administered. Younger age and absence of cerebral ischemia/infarction were invariably linked to better rescue outcomes, whereas the other important predictors of outcome varied by rescue type (interventional or noninterventional). In a propensity score–matched analysis guided by SHAP-based variable selection, rescue therapy was associated with higher odds of 3-month GOS scores of 4–5 (OR 1.63, 95% CI 1.22–2.17).

CONCLUSIONS

Rescue therapy may increase the odds of good outcome in patients with aVSP or DCI after SAH. Given the strong association between cerebral ischemia/infarction and poor outcome, trials focusing on preventative or therapeutic interventions in these patients may be most able to demonstrate improvements in clinical outcomes. Insights developed from these models may be helpful for improving patient selection and trial design.