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Biallelic pathogenic variants of the RIPPLY2 gene have been recognized to cause a subtype of autosomal recessive spondylocostal dysostosis (SCDO6), characterized by predominant cervical spine malformation with minor or absent involvement of the ribs. To date, RIPPLY2 associated SCDO6 has been described in ten patients in five studies with accompanying clinical symptoms varying from transient and recurrent torticollis to flaccid quadriplegia. Here, we describe two additional patients in one family in which the c.A238T:p.Arg80* RIPPLY2 mutation in the homozygous state, was associated with severe malformation of the posterior elements of the cervical vertebral column. In both cases neurological symptoms occurred early in life due to spinal cord compromise. These two cases, in keeping with previous reports, highlight the early and progressive natural history of cervical deformity in this rare skeletal dysplasia and the need for close neurological and radiological surveillance. Surgical decision-making needs to carefully balance the need for early intervention to protect spinal cord function on one hand, with the problem of bone malformation and skeletal immaturity on the other.

Background: The optimal treatment for odontoid fractures in older people remains debated. Odontoid fractures are increasingly relevant to clinical practice due to ageing of the population.Methods: An international prospective comparative study was conducted in fifteen European centres, involving patients aged ≥55 years with type II/III odontoid fractures. The surgeon and patient jointly decided on the applied treatment. Surgical and conservative treatments were compared. Primary outcomes were Neck Disability Index (NDI) improvement, fracture union and stability at 52 weeks. Secondary outcomes were Visual Analogue Scale neck pain, Likert patient-perceived recovery and EuroQol-5D-3L at 52 weeks. Subgroup analyses considered age, type II and displaced fractures. Multivariable regression analyses adjusted for age, gender and fracture characteristics. Results: The study included 276 patients, of which 144 (52%) were treated surgically and 132 (48%) conservatively (mean (SD) age 77.3 (9.1) vs. 76.6 (9.7), P = 0.56). NDI improvement was largely similar between surgical and conservative treatments (mean (SE) −11 (2.4) vs. −14 (1.8), P = 0.08), as were union (86% vs. 78%, aOR 2.3, 95% CI 0.97–5.7) and stability (99% vs. 98%, aOR NA). NDI improvement did not differ between patients with union and persistent non-union (mean (SE) −13 (2.0) vs. −12 (2.8), P = 0.78). There was no difference for any of the secondary outcomes or subgroups. Conclusions: Clinical outcome and fracture healing at 52 weeks were similar between treatments. Clinical outcome and fracture union were not associated. Treatments should prioritize favourable clinical over radiological outcomes.

Study Design: Randomized controlled trial Objective: Percutaneous transforaminal endoscopic discectomy (PTED) was introduced as a less invasive procedure to treat sciatica. Even though the PTED has a small scar size, it is unknown if PTED also leads to better scar-related patient-reported outcomes. Therefore, we aimed to compare scar-related outcomes between patients undergoing PTED vs open microdiscectomy. Methods: Patients with at least 6 weeks of radiating leg pain were randomized in a 1:1 ratio to PTED or open microdiscectomy. Scar-related patient-reported outcomes were measured using the Body Image Score (BIS), Cosmesis Scale (CS) and a 0-10 numeric rating scale (NRS) on scar esthetic. Results: Of the 530 included patients, 286 patients underwent PTED and 244 underwent open microdiscectomy as allocated. At 12 months of follow-up, 95% of the patients had data available. At 12 months, the BIS was 6.2 ± 1.7 in the PTED-group and 6.6 ± 1.9 in the open microdiscectomy group (between-group difference.4, 95% CI.2 to.7). CS was 21.3 ± 3.0 in the PTED-group and 18.6 ± 3.4 in the open microdiscectomy group (between-group difference −2.7, 95% CI −3.1 to −2.3). Average NRS for scar esthetic was 9.2 ± 1.3 and 7.8 ± 1.6 in the PTED and open microdiscectomy groups, respectively (between-group difference −1.4, 95% CI −1.6 to −1.2) Conclusions: PTED leads to a higher self-rated scar esthetic as compared to open microdiscectomy, while self-reported body image seems to be comparable between both groups. Therefore, from an esthetic point, PTED seems to be the preferred technique to treat sciatica.

Surgical resection of spinal cord hemangioblastomas remains a challenging endeavor: the neurosurgeon's aim to reach total tumor resections directly endangers their aim to minimize post-operative neurological deficits. The currently available tools to guide the neurosurgeon's intra-operative decision-making consist mostly of pre-operative imaging techniques such as MRI or MRA, which cannot cater to intra-operative changes in field of view. For a while now, spinal cord surgeons have adopted ultrasound and its submodalities such as Doppler and CEUS as intra-operative techniques, given their many benefits such as real-time feedback, mobility and ease of use. However, for highly vascularized lesions such as hemangioblastomas, which contain up to capillary-level microvasculature, having access to higher-resolution intra-operative vascular imaging could potentially be highly beneficial. µDoppler-imaging is a new imaging modality especially fit for high-resolution hemodynamic imaging. Over the last decade, µDoppler-imaging has emerged as a high-resolution, contrast-free sonography-based technique which relies on High-Frame-Rate (HFR)-ultrasound and subsequent Doppler processing. In contrast to conventional millimeter-scale (Doppler) ultrasound, the µDoppler technique has a higher sensitivity to detect slow flow in the entire field-of-view which allows for unprecedented visualization of blood flow down to sub-millimeter resolution. In contrast to CEUS, µDoppler is able to image high-resolution details continuously, without being contrast bolus-dependent. Previously, our team has demonstrated the use of this technique in the context of functional brain mapping during awake brain tumor resections and surgical resections of cerebral arteriovenous malformations (AVM). However, the application of µDoppler-imaging in the context of the spinal cord has remained restricted to a handful of mostly pre-clinical animal studies. Here we describe the first application of µDoppler-imagi

Surgical resection of spinal cord hemangioblastomas remains a challenging endeavor: the neurosurgeon’s aim to reach total tumor resections directly endangers their aim to minimize post-operative neurological deficits. The currently available tools to guide the neurosurgeon’s intra-operative decision-making consist mostly of pre-operative imaging techniques such as MRI or MRA, which cannot cater to intra-operative changes in field of view. For a while now, spinal cord surgeons have adopted ultrasound and its submodalities such as Doppler and CEUS as intra-operative techniques, given their many benefits such as real-time feedback, mobility and ease of use. However, for highly vascularized lesions such as hemangioblastomas, which contain up to capillary-level microvasculature, having access to higher-resolution intra-operative vascular imaging could potentially be highly beneficial. µDoppler-imaging is a new imaging modality especially fit for high-resolution hemodynamic imaging. Over the last decade, µDoppler-imaging has emerged as a high-resolution, contrast-free sonography-based technique which relies on High-Frame-Rate (HFR)-ultrasound and subsequent Doppler processing. In contrast to conventional millimeter-scale (Doppler) ultrasound, the µDoppler technique has a higher sensitivity to detect slow flow in the entire field-of-view which allows for unprecedented visualization of blood flow down to sub-millimeter resolution. In contrast to CEUS, µDoppler is able to image high-resolution details continuously, without being contrast bolus-dependent. Previously, our team has demonstrated the use of this technique in the context of functional brain mapping during awake brain tumor resections and surgical resections of cerebral arteriovenous malformations (AVM). However, the application of µDoppler-imaging in the context of the spinal cord has remained restricted to a handful of mostly pre-clinical animal studies. Here we describe the first application of µDoppler-imagi

Purpose: Multiple surgical techniques are practiced to treat sciatica caused by lumbar disk herniation. It is unknown which factors surgeons find important when offering certain surgical techniques. The objective of this study is threefold: 1) determine the relative weight surgeons place on various characteristics of sciatica treatment, 2) determine the trade-offs surgeons make between these characteristics and 3) identify preference heterogeneity for sciatica treatment. Methods: A discrete choice experiment was conducted among members of two international neurosurgical organizations. Surgeons were asked on their preferences for surgical techniques using specific scenarios based on five characteristics: effectiveness on leg pain, risk of recurrent disk herniation, duration of postoperative back pain, risk of complications and recovery period. Results: Six-hundred and forty-one questionnaires were filled in, the majority by neurosurgeons. All characteristics significantly influenced the preferences of the respondents. Overall, the risk of complications was the most important characteristic in the decision to opt-in or opt-out for surgery (35.7%). Risk of recurrent disk herniation (19.6%), effectiveness on leg pain (18.8%), postoperative back pain duration (13.5%) and length of recovery period (12.4%) followed. Four latent classes were identified, which was partly explained by the tenure of the surgeon. Surgeons were willing to trade-off 57.8% of effectiveness on leg pain to offer a treatment that has a 1% complication risk instead of 10%. Conclusion: In the context of this discrete choice experiment, it is shown that neurosurgeons consider the risk of complications as most important when a surgical technique is offered to treat sciatica, while the risk of recurrent disk herniation and effectiveness are also important factors. Neurosurgeons were prepared to trade off substantial amounts of effectiveness to achieve lower complication rates.

OBJECTIVE: Lumbar discectomy is a frequently performed procedure to treat sciatica caused by lumbar disc herniation. Multiple surgical techniques are available, and the popularity of minimally invasive surgical techniques is increasing worldwide. Clinical outcomes between these techniques may not show any substantial differences. As lumbar discectomy is an elective procedure, patients' own preferences play an important role in determining the procedure they will undergo. The aims of the current study were to determine the relative preference weights patients apply to various attributes of lumbar discectomy, determine if patient preferences change after surgery, identify preference heterogeneity for choosing surgery for sciatica, and calculate patient willingness to pay for other attributes.METHODS: A discrete choice experiment (DCE) was conducted among patients with sciatica caused by lumbar disc herniation. A questionnaire was administered to patients before they underwent surgery and to an independent sample of patients who had already undergone surgery. The DCE required patients to choose between two surgical techniques or to opt out from 12 choice sets with alternating characteristic levels: waiting time for surgery, out-of-pocket costs, size of the scar, need of general anesthesia, need for hospitalization, effect on leg pain, and duration of the recovery period.RESULTS: A total of 287 patients were included in the DCE analysis. All attributes, except scar size, had a significant influence on the overall preferences of patients. The effect on leg pain was the most important characteristic in the decision for a surgical procedure (by 44.8%). The potential out-of-pocket costs for the procedure (28.8%), the wait time (12.8%), need for general anesthesia (7.5%), need for hospitalization (4.3%), and the recovery period (1.8%) followed. Preferences were independent of the scores on patient-reported outcome measures and baseline characteristics. Three

Objective: Percutaneous transforaminal endoscopic discectomy (PTED) is gaining popular-ity by both surgeons and patients as a less invasive treatment option for sciatica. Concerns, however, exist for its learning curve. No previous study has assessed the learning process of PTED. Hereby we present the learning process of 3 surgeons learning PTED. Methods: This analysis was conducted alongside a multicenter randomized controlled trial. After attending a cadaveric workshop, 3 spine-dedicated surgeons started performing PTED, initially under the supervision of a senior surgeon. After each 5 cases, and up to case 20, the learning process was evaluated using the validated questionnaires (objective structured assessment of technical skills [OSATS], global operative assessment of laparoscopic skills [GOALS]) and a 10-step checklist specifically developed for PTED. Results: In total, 3 learning curve surgeons performed a total of 161 cases. Based on self-assessment, surgeons improved mostly in the domains “time and motion,” “respect for tis-sue,” and “knowledge and handling of instruments.” Learning curve surgeons were more able to detect differences in performances on the OSATS than the senior surgeon. Based on the GOALS, the biggest improvements could be seen in “depth-perception” and “autono-my.” Based on the 10-item specific checklist, all surgeons performed all 10 steps by case 10, while only 1 surgeon performed all steps adequately by case 15. Conclusion: Based on these study results, PTED appears to be successfully adopted step-wise by 3 spine-dedicated surgeons. From 15 cases on, most steps are performed adequate-ly. However, more cases might be necessary to achieve good clinical results. Validated tools are needed to determine the cutoff when a surgeon should be able to perform PTED inde-pendently.

AbstractObjective To assess whether percutaneous transforaminal endoscopic discectomy (PTED) is non-inferior to conventional open microdiscectomy in reduction of leg pain caused by lumbar disc herniation. Design Multicentre randomised controlled trial with non-inferiority design. Setting Four hospitals in the Netherlands. Participants 613 patients aged 18-70 years with at least six weeks of radiating leg pain caused by lumbar disc herniation. The trial included a predetermined set of 125 patients receiving PTED who were the learning curve cases performed by surgeons who did not do PTED before the trial. Interventions PTED (n=179) compared with open microdiscectomy (n=309). Main outcome measures The primary outcome was self-reported leg pain measured by a 0-100 visual analogue scale at 12 months, assuming a non-inferiority margin of 5.0. Secondary outcomes included complications, reoperations, self-reported functional status as measured with the Oswestry Disability Index, visual analogue scale for back pain, health related quality of life, and self-perceived recovery. Outcomes were measured until one year after surgery and were longitudinally analysed according to the intention-to-treat principle. Patients belonging to the PTED learning curve were omitted from the primary analyses. Results At 12 months, patients who were randomised to PTED had a statistically significantly lower visual analogue scale score for leg pain (median 7.0, interquartile range 1.0-30.0) compared with patients randomised to open microdiscectomy (16.0, 2.0-53.5) (between group difference of 7.1, 95% confidence interval 2.8 to 11.3). Blood loss was less, length of hospital admission was shorter, and timing of postoperative mobilisation was earlier in the PTED group than in the open microdiscectomy group. Secondary patient reported outcomes such as the Oswestry Disability Index, visual analogue scale for back pain, health related quality of life, and self-perceived recovery, were similarly in

OBJECTIVE Lumbar discectomy is a frequently performed procedure to treat sciatica caused by lumbar disc herniation. Multiple surgical techniques are available, and the popularity of minimally invasive surgical techniques is increasing worldwide. Clinical outcomes between these techniques may not show any substantial differences. As lumbar discectomy is an elective procedure, patients’ own preferences play an important role in determining the procedure they will undergo. The aims of the current study were to determine the relative preference weights patients apply to various attributes of lumbar discectomy, determine if patient preferences change after surgery, identify preference heterogeneity for choosing surgery for sciatica, and calculate patient willingness to pay for other attributes. METHODS A discrete choice experiment (DCE) was conducted among patients with sciatica caused by lumbar disc herniation. A questionnaire was administered to patients before they underwent surgery and to an independent sample of patients who had already undergone surgery. The DCE required patients to choose between two surgical techniques or to opt out from 12 choice sets with alternating characteristic levels: waiting time for surgery, out-of-pocket costs, size of the scar, need of general anesthesia, need for hospitalization, effect on leg pain, and duration of the recovery period. RESULTS A total of 287 patients were included in the DCE analysis. All attributes, except scar size, had a significant influence on the overall preferences of patients. The effect on leg pain was the most important characteristic in the decision for a surgical procedure (by 44.8%). The potential out-of-pocket costs for the procedure (28.8%), the wait time (12.8%), need for general anesthesia (7.5%), need for hospitalization (4.3%), and the recovery period (1.8%) followed. Preferences were independent of the scores on patient-reported outcome measures and baseline characteri